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Acta Psychiatr Scand 2013: 127 (Suppl. 441): 147 2012 John Wiley & Sons A/S.

& Sons A/S. Published by Blackwell Publishing Ltd


All rights reserved ACTA PSYCHIATRICA SCANDINAVICA
DOI: 10.1111/acps.12038

Rediscovering catatonia: the biography of a


treatable syndrome
Fink Max. Rediscovering catatonia: the biography of a treatable Max Fink
syndrome. Department of Psychiatry and Neurology Emeritus, Stony
Brook University, Long Island, NY, USA
Objective: Catatonia, a disorder of movement and mood, was described
and named in 1874. Other observers quickly made the same recognition.
By the turn of the century, however, catatonia was incorporated as a
type within a conjured syndrome of schizophrenia. There, catatonia has
lain in the psychiatric classication for more than a century.
Method: We review the history of catatonia and its present status. In
the 1970s, the tie was questioned when catatonia was recognized among
those with mood disorders. The recognition of catatonia within the
neuroleptic malignant syndrome oered eective treatments of high
doses of benzodiazepines and electroconvulsive therapy (ECT), again
questioning the tie. A verifying test for catatonia (the lorazepam
sedation test) was developed. Soon the syndromes of delirious mania,
toxic serotonin syndrome, and the repetitive behaviors in adolescents
with autism were recognized as treatable variations of catatonia.
Results: Ongoing studies now recognize catatonia among patients
labeled as suering from the Gilles de la Tourettes syndrome, anti- Key words: schizophrenia; treatment; diagnosis;
classification; catatonia; tonic immobility
NMDAR encephalitis, obsessivecompulsive disease, and various
mutisms. Max Fink, 23 Spring Hollow Road, PO Box 457, St.
Conclusion: Applying the treatments for catatonia to patients with these James, NY 11780, USA.
E-mail: mfink@attglobal.net or max.fink@stonybrook.edu
syndromes oers opportunities for clinical relief. Catatonia is a
recognizable and eectively treatable neuropsychiatric syndrome. It has
many faces. It warrants recognition outside schizophrenia in the
psychiatric disease classication. Accepted for publication October 4, 2012

Introduction: liberating catatonia from


schizophrenia
chiatrist, Emil Kraepelin also recognized
The ultimate court of appeal is observation and experi-
catatonia. But Kraepelin included catatonia and
ment, and not authority. Thomas H. Huxley (1)
two other syndromes, hebephrenia and paranoia,
For an illness to be eectively treated, it has rst as markers within a grand scheme that he labeled
to be recognized; only then can physicians apply dementia praecox (later, schizophrenia).
their knowledge and skills. Catatonia is an identi- Kraepelin was a prolic writer of textbooks
able syndrome of abnormal movement and emo- widely regarded as gospel truth. As a result, for
tion rst described by the German psychiatrist more than a century, catatonia was buried within
Karl Kahlbaum in 1874 among the patients in his schizophrenia as one of many types. To this day,
psychiatric asylum. His description was soon when clinicians recognize catatonia, they immedi-
armed by other physicians and broadly accepted ately see schizophrenia and reexively prescribe
as an identiable syndrome among psychiatric dis- neuroleptic drugs even when the signs of psychosis
orders. Within two decades, another German psy- are absent. The Kraepelin equation of catato-

1
Max Fink

nia = schizophrenia dominates clinical thought Neuroleptic drugs such as chlorpromazine and
and writing. Such prescription is not helpful; haloperidol, the drugs used to relieve psychosis,
indeed, it is often harmful as neuroleptic drugs aect the bodys motor functions. They induce
often precipitate a toxic syndrome in patients with tremors, peculiar rhythmic movements of mouth
catatonia that is recognized as the neuroleptic and tongue (dyskinesia), and stien and slow body
malignant syndrome. More egregious, however, is movement (rigidity). They induce acute toxic states
the failure to prescribe the specic and eective of fever and delirium that are accompanied by the
treatments for catatonia as they are not recognized catatonia signs of mutism, repetitive acts, and
as treatments for schizophrenia. This mistaken rigidities. These toxic signs were increasingly rec-
conception has plagued patients for more than a ognized during the 1970s as the reports of fatalities
century and has distorted the classication of psy- grew; by 1980, the toxic consequences were named
chiatric diseases. the neuroleptic malignant syndrome (NMS).
These chapters of Rediscovering Catatonia trace By 1984 at Stony Brook, we had identied three
the biography of catatonia, relate how it is identi- patients with NMS, and each exhibited signs of
ed, describe its many forms, look forward to catatonia. We applied the promoted treatments of
identifying other forms of catatonia that are now the time; one slowly recovered but two did not.
not recognized and inadequately treated, discuss The lifesaving benets of ECT in these conditions
the fear hypothesis that explains catatonias origin, had been described in 1952 and repeatedly con-
and discuss catatonias history within psychiatric rmed (2). When the two patients who had not
classications with the present promise of relief in responded to medications were treated with ECT,
the DSM-5 iteration. they recovered quickly (3).
Catatonia is a syndrome of body movement and Although we lacked understanding of the causes
speech related to disorders of emotion. Mutism of malignant catatonia, we did know that ECT
(not speaking), negativism (refusing commands), relieved it. Relatively few hospitals had ECT treat-
posturing and rigidity (holding unusual bodily pos- ment units, but all could prescribe agents like bro-
tures), and repetitive speech and acts (repeating mocriptine that increased the brain levels of the
meaningless words, phrases, or motor movements, dopamine neurotransmitter that is directly inhib-
often self-injurious) are its main signs. The charac- ited by neuroleptic drugs. A national debate
teristic delusions, hallucinations, and disturbances ensued, carried on for more than two decades,
in thought that are central to the image of schizo- whether the neuroleptic-induced malignant syn-
phrenia are not features of catatonia. Catatonia is a drome was an abnormality of dopamine metabo-
distinct syndrome that in the past half century has lism and best treated with dopamine agonists or an
been increasingly reported outside schizophrenia. example of malignant catatonia that was best trea-
Eective treatments for catatonia were devel- ted with ECT. The debate focused my interest in
oped in the 1930s rst the barbiturates and then catatonia (46).
electroconvulsive therapy (ECT). In the 1980s, the Two former students, Michael A. Taylor and
barbiturates were replaced by the benzodiazepines Richard Abrams, had been instrumental in report-
for clinical use. Neither the sedative drugs nor ing catatonia in patients with manicdepressive ill-
ECT are considered eective in schizophrenia and nesses and in 1976 had questioned the teaching
are therefore not applied in cases of catatonia that catatonia was necessarily an aspect of schizo-
when it is presumed to be a manifestation of phrenia (7, 8). In 1990, I began to collaborate with
schizophrenia. Inadequate treatment and pro- Taylor, and in our rst essay, we argued that cata-
longed illness result when this error of diagnosis is tonia was best seen as a distinct syndrome indepen-
made. The neuroleptic drugs that are promoted as dent of schizophrenia, arguing for their separation
treatments for schizophrenia are ineective for cat- (9). At the time, the Diagnostic and Statistical
atonia; indeed, they increase toxicity and occasion- Manual of psychiatric disorders of the American
ally lead to death. Psychiatric Association was undergoing its fourth
I was trained as a neurologist and psychiatrist revision, and we urged that catatonia be separated
and began my academic career as a researcher in from schizophrenia. To accommodate our view,
1954. Like many of my peers, only occasionally although still beholden to the dominant perspec-
did I recognize catatonia in practice. I was not tive of catatonia as a form of schizophrenia, the
awakened to the extent of catatonia until the 1980s DSM-IV committees added a class of Catatonia
when as an Attending Physician at the University secondary to a medical condition with a distinct
Hospital of Stony Brook University on Long code of 293.89 (10).
Island I supervised the care of hospitalized patients Our interest in catatonia its main features,
and its ECT treatment unit. how it is found, in what forms, and how best trea-

2
Rediscovering catatonia

As the connection to schizophrenia has been


loosened, catatonia is increasingly recognized
among the childhood disorders of Gilles de la Tou-
rettes syndrome, obsessivecompulsive disorder,
and diverse mutisms the selective or elective
and the pervasive refusal syndrome recognized in
the classication of diseases. Catatonia is also seen
in patients with post-traumatic stress disorders,
passive acceptance of rape assault, and fear-
induced freezing in urban violence and military
disasters. These connections support a hypothesis
of catatonia as a residual response to fear of immi-
nent doom from the time when humans feared
Fig. 1. Average annual number of citations marked as cata- predators (15).
tonia in each 5-year period. Most psychiatric disorders are identied by
the presence of symptoms from symptom check-
ted led us to publish the textbook Catatonia: lists. No laboratory tests, common in clinical
A Clinicians Guide to Diagnosis and Treatment in medicine, are used. But catatonia is among the
2003 (11) and to urge again that catatonia war- few psychiatric diagnoses that are operationally
ranted a home of its own in the classication (12). dened. In the chapters that follow, catatonia is
This publication was the rst text on catatonia dened as published by Michael Taylor and
since the original publication by Karl Kahlbaum myself in 2003: Patients who exhibit two or more
130 years earlier (13). motor signs of catatonia (recognized in Catatonia
Much has changed since then catatonia is Rating Scales) for more than 24 h and that
more often recognized, eective treatments are quickly improve with test doses of lorazepam are
commonly applied, favorable outcomes are presumptively diagnosed as suering from catato-
increasingly reported, and death rates for medica- nia. Recovery of the illness with relief of the
tion-induced neurotoxic syndromes have fallen. motor and vegetative signs after treatment with
Increasing numbers of citations to catatonia in benzodiazepines and ECT validates the diagnosis.
the medical literature are reected in the NIH Pub- An operational diagnostic procedure assures
Med indices (Fig. 1). recognition of the syndrome and oers patients
Two new forms of catatonia have been recog- typically eective and prompt relief. For those
nized since our publication. In 2007 the syndrome who are misidentied as catatonic when they are
of anti-NMDAR encephalitis was described as a not, the principal hazard is the delay in instituting
neuropsychiatric disorder of diverse manifestations a more appropriate treatment, if there is one. The
connected to an autoimmune etiology. More than benzodiazepines are not hazardous, and the test
70% of the diagnosed patients exhibit catatonia doses are well within clinical experience for safety.
that is neither recognized nor properly treated. Although ECT is widely stigmatized as brain dam-
Instead, patients are subjected to intensive searches aging and capable of erasing memories, these haz-
for hidden tumors, oered surgery if a silent tumor ards pale before the risks to life inherent in the
is recognized, and prescribed potent steroids, more malignant forms of catatonia and the pro-
immunotherapy, and plasma exchange with longed illness that follows inappropriate diagnosis
minimal benet. and ineective treatment.
A more dramatic impetus to tracing this history The American Psychiatric Association DSM-5
has been the recognition that the self-injurious Psychosis Work Group presented its recommenda-
behaviors of children and adolescents suering tions on catatonia online in May 2012, listing
from autism spectrum disorders may be signs of many proposed changes. When these are published
catatonia that are responsive to catatonia treat- in DSM-5, the much needed separation of catato-
ments. The recognition of catatonia in these disor- nia from schizophrenia will be accomplished.
ders and demonstrations of the ecacy of catatonia Much will then need to be done in professional
treatments are largely the eorts of Drs. Lee Wach- and public education to assure recognition and
tel at Johns Hopkins Medical Center and Dirk treatment of catatonia as an independent psychiat-
Dhossche at the University of Mississippi (14). ric syndrome.

3
Max Fink

Chapter 1: catatonia is discovered


In some ways disease does not exist until we have agreed mania and melancholia, the stupidities (mental
that it does by perceiving, naming, and responding retardation), epilepsies, and the dementias of
to it. CE Rosenberg 1986 (16) aging, as a result of head trauma or alcoholism.
Among the patients were some who postured
Catatonia is a syndrome of dysfunctions in
and stared, frequently mute but occasionally repet-
movement and emotion. It was rst culled in the
itive in speech and movement, occasionally
1870s from the welter of behaviors that are com-
appearing in stupor as if dead, or in delirious furor
mon in every asylum. Unlike diseases of a single
and unresponsive to painful stimuli. In 1874, Karl
body organ and narrowly dened functions,
Kahlbaum, the 46-year-old director of a private
abnormal behaviors encompass the whole organ-
psychiatric sanitarium in the German town of
ism. What did practitioners see when they entered
Gorlitz, described his experience with such patients
an asylum in the mid-1800s and tried to make
in a small book of 104 pages that he labeled Die
sense of what they encountered?
Katatonie. It was his third attempt to classify the
The entrance door is locked, and one of a ring of
behaviors of his patients (17).
large keys is used to enter. The moment the door
From an extensive clinical practice, Kahlbaum
opens, the entrance is lled with a cacophony of
described 26 case histories that included disabling
cries and the pungent odors of urine, sweat, and
disorders of movement and speech among their
the medicine paraldehyde (a liquid sedative with
symptoms. The patients were immobile and postur-
the odor of vinegar). Loud voices, some sing-song
ing, remaining in one position for hours; or moving
and some cackling, and occasional screams ll the
continually, pacing, or hitting a wall or themselves;
entranceway. Some patients are tied down on a
or standing and staring, unblinking, repeating
pallet, while others sit motionless seemingly una-
words or phrases over and over again, sometimes
ware of the activities around them, unresponsive to
in whispers or in repetitive shouts. They had other
questions and even to painful stimuli; and yet, oth-
signs of mental illness melancholia (depression,
ers are rocking, posturing Christ-like, staring, and
withdrawal to stupor, and suicide risk), mania
pacing. When they do respond, many are sad and
(continuous movement, grandiosity, delirium), psy-
despondent, or frightened, or grandiose, or angry.
chosis (peculiar thoughts that brought forth unu-
Speech may be normal, but more often it is repeti-
sual speech), and dementia (poor memory,
tive and meaningless. Occasionally a patient has an
disorientation, poor self-care). Many were also suf-
epileptic seizure.
fering the systemic diseases of syphilis, tuberculo-
Trying to distinguish causes and predict the
sis, or epilepsy. Onsets and courses of illness
course of an illness in an individual patient was chal-
uctuated with sudden changes in retardation and
lenging. Over time, the behaviors of individual
withdrawal, or mania and excitement, or delusional
patients may remain the same day after day but more
and suicidal thoughts, so much so that one school
often the symptoms evolve, sadness and despon-
of professional thought described the behaviors as
dency alternating with excitement and mania, for
expressions of a single disease of many causes.
example, deteriorating to ever poorer self-care, until
Before specic syndromes were described, the vari-
dementia and a vegetative state ensues.
ations in symptoms seemed to appear and disap-
How was one to extract a recognizable disease
pear but no single pattern was discerned. The term
from this welter of dysfunctions? No help was
Einheitspsychose unitary psychosis was in
likely to be gleaned from studies of the brain after
common use until Kahlbaum and his colleague
death, either in the search for abnormal tissues in
Heckers description of hebephrenia and catatonia
the sliced brain or cellular changes in stained brain
and Kraepelins images of dementia praecox and
tissues, even when examined with the nest micro-
manicdepressive insanity were accepted.
scopes. At the time, dementia paralytica had been
Kahlbaum describes a young man admitted to
described as the neurological consequence of syph-
the sanitarium who becomes intensely fearful when
ilis. It was recognized during life by progressive
he is told that he has syphilis. (This case record is
paralyses accompanied by grandiose thoughts,
paraphrased from Case #2 of the English transla-
impaired memory, and silly comments. After
tion of the Kahlbaum text by George Mora on the
death, thickened brain coverings were found as
publications 100th anniversary. Mora was a pedi-
evidence of the fatal disease. But such direct patho-
atric psychiatrist lecturing at Yale and New York
logical evidence was the exception. Also demar-
Universities.)
cated and generally accepted at the time were

4
Rediscovering catatonia

A 33-year-old peasant terminates a love aair, becomes Attempts at classication were being made in
depressed and is reluctant to talk or participate in social other branches of medicine, especially in bacteriol-
activity. His depressed moods become constant after he ogy. Unique infectious diseases were identied by
develops a syphilitic infection. Muscular twitching in the the patterns of clinical symptoms and the course of
face and extremities and severe tonic contractions of the the illness. Pathogens were identied in body u-
back muscles follow. He retires to his bed without inter- ids, and illnesses were labeled by the name of the
est in his surroundings; his eyes remain shut, refuses to invading pathogen, oering opportunities to test
reply when addressed or even to speak. He eats little. On treatments in homogeneous samples of one illness
admission to the sanitarium he is inactive, lies prone in type. These eorts in clinical medicine were models
bed, giving no answer to questions put to him. He main- for Kahlbaum. He saw his eorts as similar to
tains any posture he is made to assume for long periods. those that described the motor disorders of tabes
It is possible to place his extremities in the most extrava- dorsalis, the loss of motor control of the limbs
gant postures that he changes gradually to an original when syphilis aected the spinal cord and the para-
passive position. When induced to walk, he moves for- noia and dementia of dementia paralytica. Kahl-
ward slowly, leaning to the right and extending his right baums descriptions and demarcations of catatonia
leg stiy. Sensory responses are greatly reduced over the were so well dened that others soon recognized
entire body with little reaction to needle pricking. similar cases.
Three months later he suddenly begins to speak inces-
santly, expressing a few monotonous phrases over and Kahlbaums first classification of psychiatric disorders
over as Love is God, Love, Love, Love is God, Love is
God with permutations, adding short phrases as Is it Born in eastern Germany on December 28, 1828,
so? or I say, spoken softly or at times shouting loudly. Kahlbaum had been schooled at the universities of
While voicing these phrases he sometimes lies at on his Konigsberg, Wurzburg, and Leipzig. He was
back; at times the upper part of his body is raised and awarded an MD in Berlin with a thesis on the
head bent stiy back. He becomes hoarse, speaking anatomy and histology of the intestinal tract of
softly with intermittent shouting. This condition per- birds in 1854. After joining the faculty at Konigs-
sisted with brief interruptions. He died a year later after berg, he established training courses in psychiatry
contracting tuberculosis (13). for medical students and published a monograph
on sensory delusions. By 1863, he had developed a
A detailed analysis of Kahlbaums cases was complex glossary of mental disorders that he pub-
undertaken by Daniel Rogers who nds 25 case lished as Die Gruppierung der psychischen Krankhe-
vignettes (18). Rogers analyzes 21 vignettes for iten und die Einteilung der Seelenstorungen (20).
motor signs characterized as signs of catatonia. He listed many peculiar behaviors and clustered
Kahlbaum recognized 17 motor signs. Since then, the symptoms into disorders according to their
additional signs, altogether about 40, have been principal features (depression, mania, psychosis)
listed in Catatonia Rating Scales. The most recent and course of illness (continuous, recurrent, and
addition to the recognized catatonia signs is the episodic). In an obsessively detailed organization,
repetitive self-injuries seen in children and adoles- he gave each form a unique name. Vesania typica
cents suering from autism spectrum disorders anticipated the manicdepressive illness of Kraepe-
(19). lin and vesania progressiva described a deteriora-
Consider the eort entailed by Kahlbaums tion in mood to delirium and dementia. Vecordia
attempt at classication. No catalog of psychiatric were disorders beginning in puberty, including
diseases existed nor had criteria been described paraphrenia hebetica, a disorder that later was
with which to label an illness or to predict a per- labeled hebephrenia. A disorder in mood labeled
sons fate. No eective treatments were known, dysthymia exhibited a retarded form dysthymia
and persons recovered, remained ill, or died inde- atra and an agitated form, dysthymia elata. In
pendent of physicians eorts. Some patients sud- that burst of creativity, he had not yet described
denly recovered after a severe systemic infection or the concept of catatonia.
after an epileptic seizure and could leave the asy- Kahlbaums rst classication was lled with
lum. At best, physical restraints and sedation with obsessive details and not based on any logical prin-
opiates were prescribed for the unruly, and feeding ciple that could be explained to others, so it is no
was forced to maintain life in the stuporous surprise that this creation was deemed too complex
and negativistic. By providing a clean, protected to be understood and was not accepted by his col-
home, the physicians hoped that recovery would leagues. In similar fashion, the iterations of the
be spontaneous. DSM classication describe illnesses without a

5
Max Fink

basis in biological or medical principles. The class The melancholia expresses itself rst as a sadness and
names and descriptions are committee-driven, and depression of mood that gradually consolidates into
depending on the skills and enthusiasm of vocal delusions ending in a brooding persecutory mania or
leaders, a diagnosis is either included or eliminated a delusion of injury. A shallowness of aect appears
from the accepted list. The criteria are descriptive Side by side with his morbid lamentations of his terrible
based on the presence of symptoms, with some misery [the patient] frequently cannot suppress an
attention to course. The few biological criteria that impulse to laughter and silly jokes.
have been developed are excluded.
Kahlbaum left the university faculty in 1866 to Of special importance are disturbances of form [of
become a sta physician at the Reimer Sanitarium, speech] First there is a peculiar deviation from logical
a private sanitarium in Gorlitz, a village in eastern sentence formation [with] a noticeable tendency to
Germany that bordered Poland. A year later, he remain stuck on a topic once he has taken it up
bought the hospital, became its director, and main- The mode of speech and expression sinks well below
tained it until his death in 1899. his level of education [and] we see a propensity to gush,
In his personal life, Kahlbaum was liberal in a preference for sentimental descriptions, a seemingly
politics, ascetic in not using tobacco or alcohol, poetic diction, and consequently an overow of empty
regular in church attendance, and active in the misshapen phrases.
church choir. He traveled frequently to meetings
and to art museums. In 1866, he married the cou- On etiology, Hecker wrote:
sin of his colleague Ewald Hecker. The union pro- the rst striking fact is that we are mostly (although not
duced three sons and one daughter. Kahlbaums always) dealing with individuals who have been slightly
wife died in 1884 and he remarried in 1888, pro- retarded in their physical and in their mental develop-
ducing two more sons. After his death, his son ment from early on a certain weakness of intellect,
Siegfried, who had trained in psychiatry with Carl laziness, and inability to do mental work is already
Wernicke, directed the sanitarium until 1943 when noticed in childhood, but this is not as extreme or as
it was taken over by the government for military noticeable as in idiotism.
uses.
The condition was commonly recognized among
patients in the asylums. A few decades later, hebe-
Hebephrenia phrenia, like catatonia, was co-opted by the Ger-
In 1866, the same year that he became hospital man psychiatrist Emil Kraepelin within his
director, Kahlbaum was joined by Ewald Hecker conjured disorder of dementia praecox. Hebe-
as a sta physician. Together, they continued an phrenia was the principal form of the syndrome
interest in classication. In Die Gruppierung later labeled schizophrenia. In successive psychiat-
Kahlbaum had described a syndrome of para- ric disease classications, hebephrenia has often
phrenia hebetica, and together, they described been relabeled. The present term disorganized
the syndrome that became a model for the schizophrenia is one of the ve recognized types
description of catatonia. It began as a disorder of schizophrenia (22, 23).
in thought and speech with an onset in adoles- At one time, hebephrenia was frequently seen in
cence. By early adulthood, it had progressed to chronic care institutions. The illness was consid-
dementia. Child-like, often imbecilic behaviors ered progressive, poorly responsive to treatments,
limited the ability of the patients to take part in often ending in silly behaviors and dementia.
normal schooling and even in adequate self-care. Patients became lifelong wards of society as they
In 1871, Ewald Hecker described these condi- were unable to care for themselves. The processes
tions in seven cases in the 35-page essay Die of deinstitutionalization that closed the sanitaria in
Hebephrenie (21). the last quarter of the twentieth century shifted
The illness begins its progressive course with patients with hebephrenia to adult homes and
melancholia and mania between the ages of 18 and chronic disease institutions.
22 years. Over time, thinking becomes confused
and speech becomes silly and repetitive, a course Catatonia
that ends in dementia. Hecker said:
As Hecker and Kahlbaum were collaborating at
In most cases the disease apparently begins as a sequel
Gorlitz, we can picture their discussions of each
to a profound alteration in emotions with decidedly mel-
patient and their search for other examples of
ancholic symptoms.
hebephrenia. And just as Hecker was riveted by

6
Rediscovering catatonia

hebephrenia, Kahlbaum focused his attention after very severe physical or mental stress such as a
upon what is now recognized as catatonia. His terrifying experience. The patient remains motionless,
rst public discussion of catatonia was before an without speaking, and with a rigid masklike facies, the
audience at the University of Konigsberg in eyes focused at a distance . The general impression
1866 (24). At Innsbruck 3 years later, he conveyed is one of profound mental anguish, or an
described two cases of melancholia with stupor immobility induced by severe mental shock (13).
that exhibited the signs of catatonia. The discus-
sion after his presentation was sparse, but a Citing fear as central to the syndrome, Kahl-
published comment noted that enlarging the baum titled his book Die Katatonie, oder das Span-
classication with a new diagnosis focused on a nungs-Irresein, translated as Catatonia, the
muscular tension psychosis would not be help- tension insanity.
ful. (25) Other observers wrote that a subject suering
In 1874, 3 years after Heckers 1871 article on Kahlbaums disease might lay like a log of wood,
hebephrenia, Kahlbaum formally introduced stretched out, able to behold with his eyes but
catatonia to psychiatry. His description is valid unable to speak a word for several days. The
today. malady occurs suddenly and he stops as if he were
frozen, his eyes open, xed and motionless (26).
Catatonia is a brain disease with a cyclic, alternating On recovery, patients say that they desired to
course, in which the mental symptoms are, consecu- speak but were unable to do so, sensing impending
tively, melancholy, mania, stupor, confusion, and even- doom. Persons in catatonic stupor are frozen,
tually dementia. One or more of these symptoms may be petried, and experience extreme fear (27).
absent from the complete series of psychic symptom Recent authors also describe patients as giving the
complexes. In addition to the mental symptoms, loco- appearance of intense anxiety (2830).
motor neural processes with the general character of Kahlbaum compared his image of catatonia to
convulsions occur as typical symptoms. what at that time had recently been demarcated as
Lacking eective treatment, Kahlbaum observed dementia paralytica, the general paralysis of the
the full course that marked the disease: insane associated with syphilis:
In this newly dened group of disorders, similar to gen-
The typical signs of the condition termed atonic melan-
eral paralysis of the insane (GPI) with or without
cholia [a depressive illness with the characteristic motor
delusions of grandeur clinical changes in the locomotor
signs of catatonia] may be described as a state in which
apparatus form the main and typical features of the dis-
the patient remains entirely motionless, without speak-
ease; in addition, each disease (GPI and this disease)
ing, and with a rigid, masklike facies; the eyes focused
exhibits manifold patterns of symptoms. In GPI the par-
at a distance; he seems devoid of any will to move or
alytic components are of many varying grades of severity
react to any stimuli; there may be fully developed
and type; one or other may be absent in any particular
waxen exibility, as in cataleptic states, or only
case. In the same way as in GPI, the spastic signs in
indications, distinct, nevertheless, of this striking
the newly described clinical form of the disease are also
phenomenon.
manifold and varied These muscular symptoms dis-
The motor-inhibited form of the illness is distin- play alterations in muscular tone and I would like to
guishable from its excited forms, often resolving name this disease entity the tonic mental disorder (Span-
dramatically and suddenly, a resolution that nungs-Irresein) or vesania katatonica (catatonia) (13).
clearly distinguished catatonia from an association
In this progressive understanding, we see Kahl-
with schizophrenia.
baum identifying the principal signs of the disor-
The general impression conveyed by such patients is one der, the emotional basis, and the progressive
of profound mental anguish, or an immobility induced course. His model was the developing understand-
by severe mental shock; it has been classied either ing of neurosyphilis. Although no connection to an
among the states of depression (which explains the term infection had been made, a search for brain lesions
atonic melancholia) or among the conditions of feeble- in anatomy and histology for each psychiatric dis-
mindedness (stupor or dementia stupida); others have order was ongoing. New techniques for staining
regarded it as a combination of the two. brain tissues identied dierent cellular forms
(e.g., neurons and glia), and many authors
Here Kahlbaum oers a clue to his thinking of searched for brain lesions and cell forms to associ-
the cause of catatonia. His patients appeared to be ate with behaviors. Kahlbaum was not enthusiastic
astonished or thunderstruck. The syndrome about this search:
appeared:

7
Max Fink

This anatomicopathological work produced much valu- behavior is self-injurious, when the withdrawal is
able material but contributed nothing to the basic views to stupor or to delirious mania, and when the
on the origin of mental illness or on the anatomical locus changes in autonomic activity are severe, catato-
of their diverse and signicant manifestations; the view nia becomes life-threatening. In most instances,
is now spreading that only comprehensive clinical obser- the signs are well tolerated, and with the eective
vation of cases can bring order and clarity into the mate- treatments known today, the subject returns to
rial by using the method of clinical pathology. It is the state of functioning before the onset of the
futile to search for an anatomy of melancholy or mania, illness.
etc. because each of these forms occurs under the most
varied relationships and combinations with other states,
and they are just as little the expressions of an inner Kahlbaum after catatonia
pathological process as the complex of symptoms called As a hospital director, Kahlbaum shared the belief
fever. How wrong it inevitably was to expect patho- in moral treatment for the psychiatric ill the
logical anatomy alone to reform the obsolete psychiatric benevolent, supportive, non-punitive, socially
framework. encouraging environment that emphasized rehabil-
In the nal chapter, Kahlbaum, nding no bene- itation measures. By 1887, his hospital consisted of
cial treatments, argued for supportive and non- 18 buildings with 130 patient rooms; large dining
punitive hospital care at a time when the debilitat- and recreation halls; theater, library, and reading
ing treatments of bleeding, purging, and dunking rooms; tennis and croquet courts, and an indoor-
sped many persons to untimely deaths. An interest heated gymnasium, all in a park-like setting with
in electricity as a source of treatments for psy- gardens and a farm. He developed a special child
chiatric diseases both the symptoms of the treatment unit, the Padagogicum, with school-
patients and the characteristics of electricity were rooms, employed trained teachers, and specialists
ephemeral led to many imaginative experimental in handicrafts and educational programs. His
applications. Kahlbaum ends his discussion of asylum became a model for the care of children
treatments, possibly anticipating the role of and adolescents.
electricity: Kahlbaums interest in the psychiatric care of
children was enduring. In 1883, he published an
On the other hand it seems we have methods of very essay on child psychiatry, rening the concept of
great therapeutic importance in faradic and galvanic hebephrenia and arguing for special treatment for
electricity, although for the time being the indications juveniles, much as he had developed in his Pada-
have not yet been worked out, and in many cases the gogicum. His rened description of the condition
action might be explained more on the basis of mental [hebephrenia or pubertal neurosis], wrote Ernest
than of physiopathological eects. Harms in 1962,
The catatonia that we envision today is not the must be designated the rst realistically established con-
result of structural tissue changes (as in strokes, cept of child psychiatry. (31)
tumors, traumatic injury) nor infections or singu-
What is Kahlbaums place in the history of
lar systemic disorders, but as extensions of physio-
psychiatry? In celebrating the centenary of the
logical functions. The signs of catatonia are not
publication of Die Katatonie, Rafael Katzenstein
novel behaviors but exaggerations of every
cited Kahlbaum as
humans repertoire of tics, mannerisms, compul-
sions, rituals, speech interjections, and postures. the rst German psychiatrist to systematically elaborate
So long as these habits do not interfere with nor- forms of mental diseases from the pure clinical view-
mal living, the subjects and the observers adjust, point. He identied the defects in earlier formulations
considering the peculiarities as part of the individ- [ie, the Einheitspsychose] and sought to organize psychi-
uals personality. When the changes in behavior atric states according to observations of onset, course,
are acute and by their severity impede normal and outcome (25).
living, an illness is perceived and relief is sought.
Late in life, Emil Kraepelin assessed Kahl-
Many patients, including some severely ill, recover
baums role in clinical medicine in this way:
normal functions and do so rapidly, without per-
manent eects, an indication that the catatonia He [Kahlbaum] was the rst to stress the necessity of
motor signs result from variations in physiology juxtaposing the condition of the patient, his transitory
and not from changes in structures or tissues. We symptoms and the basic pattern underlying his disease.
do not view catatonia as similar to neurosyphilis The condition of a patient may change often and in
as Kahlbaum conjectured. But when the repetitive diverse ways, with the result that in the absence of other

8
Rediscovering catatonia

clues any attempt to rescue him from his plight is oneself on the total picture of the psychosis as obtained
doomed to failure. Moreover, identical or remarkably by comprehensive clinical observation. In emphasizing
similar symptoms may accompany dierent diseases. the course of the illness, he added a new viewpoint (33).
Their inner nature may only be revealed through their
By the end of the twentieth century, Kahlbaums
progress and termination and, in some instances, at post
contribution in extracting catatonia from the
mortem.
welter of altered behaviors was increasingly appre-
On the basis of such considerations Kahlbaum sought to ciated and biographies appeared (3436).
delineate a second pattern of illness similar to that of In my view, as a clinician, Kahlbaum made the
[general] paralysis and, like it, including both mental dis- best of the knowledge of his time and extracted
orders and physical concomitants: catatonia, in which two clinical entities that are still appreciated by cli-
muscular tension provided a basis for comparison with nicians today. He had the misfortune, however, to
[general] paralysis. Although his interpretation is open have his work co-opted by a fellow German psy-
to criticism, Kahlbaum deserves credit for having chiatrist whose skill as a textbook writer (that went
suggested the right approach. Careful attention to the through eight editions) hid Kahlbaums work
progress and termination of mental disorders, informa- within the shadow of a poorly dened abnormal
tion gleaned in some instances from autopsies, and behavior syndrome that even today, after more
insight into underlying causes have made possible the than a century, lacks any measurable and veriable
juxtaposition of a vast array of evidence and often diag- characteristics and also lacks eective treatment.
nosis on the basis of symptom pattern. We can today In the past few decades, a few intrepid clinicians
formulate in specic terms what earlier doctors could have resurrected Kahlbaums image of catatonia
surmise but not prove (32). and rened its characteristics in a way that are
both objectively veriable and eminently treatable.
Kahlbaums work was praised by Karl Jaspers,
Present-day clinical skills indicate that many
a German psychopathologist and author of the
syndromes that are actually forms of catatonia are
denitive text of psychiatry after Kraepelin:
hidden in plain sight. As these are reframed as
Kahlbaum formulated two fundamental requirements: treatable aspects of catatonia, the place of Karl
rstly, the entire course of the mental illness must be Kahlbaum as a father of modern clinical psychia-
taken as basically the most important thing for any for- try will be assured.
mulation of disease entities and, secondly, one must base

9
Max Fink

Chapter 2: catatonia disappears


It is true that man is still, as he has always been, subject baums catatonia, and a paranoid psychosis (a
to error; his judgments are often incorrect, his beliefs delusional fear of impending harm) as one disease,
false, his opinions changeable from age to age. But expe- a position that under the later name of schizophre-
rience of error is his best guide to truth, often dearly nia has persisted in psychiatric classications until
bought, and therefore, the more to be relied upon. the present.
Alfred Russel Wallace, 1905 (37) Manicdepressive insanity began later in life,
Kraepelin said, with uctuations into relative
Kahlbaums 1874 account of catatonia was
health and recurrent illness without progressing to
delineated so well that others quickly recognized
dementia. Although he found instances of catatonia
the disorder that he described. Within 3 years, cat-
in both the dementia praecox and manicdepressive
atonia as Kahlbaum delineated was reported in
populations, he marked catatonia as a principal
four patients with depression and mania (38).
sign of dementia praecox. He established a cate-
From New York Citys Wards Island in 1883, cat-
gorical rewall in his system between these two
atonia was recognized in patients with melancholia
disorders, a rewall that persists to this day in the
(39). Four years later, a German report cited cata-
worlds principal psychiatric classications.
tonia in manic patients (40). By 1898, six subtypes
For neither illness did Kraepelin oer dening
of catatonia had been described (41), and catatonia
characteristics. Nor were his descriptions improved
had been distinguished from dementia paralytica
upon in the succeeding century to develop a well-
(neurosyphilis) in a study of 227 psychiatric
dened disease description, or to dene specic
patients (42).
tests, or to identify eective treatments. In psychi-
An active academic industry commenting on
atric manuals, schizophrenia remains a heteroge-
catatonia quickly followed among German,
neous conglomeration of disturbances of speech,
French, and American authors (11). Each eort, in
thought, and aect. Manicdepressive illness has
samples of 112 patients, conrmed Kahlbaums
undergone greater development into two major
descriptions and took a position on whether the
syndromes, one characterized by depressive mood
syndrome was best explained by systemic medical
disorder and one by mania. Each has been divided
or by psychologic mechanisms. As each recogni-
into non-specic overlapping types without den-
tion sharpened the image of the syndrome, catato-
ing characteristics, without eective treatments,
nia became more strongly established as an
and ignoring the extensive evidence that the two
identiable psychiatric entity, making the eventual
syndromes appear in the same subjects as their
development of treatments increasingly likely.
illnesses progress (44).
Then catatonia was recognized by the German
Kraepelin regarded catatonia only as a type
psychiatrist Emil Kraepelin (18561926), the head
within his concept of dementia praecox. The demo-
of psychiatry at Heidelberg and then at Munich.
tion of catatonia from an individual syndrome to
As an asylum physician, Kraepelin cared for
that of a subtype, a marker, and in the present
chronically ill psychiatric patients whose prognosis
classications as a specier of dementia praecox
was poor. In the absence of eective treatments,
has hidden catatonia from recognition as a unique
the illness of many patients ended in dementia. In
syndrome for more than a century. This is in no
the nineteenth-century tradition of the study of
small part a tribute to the enormous inuence
diseases, Kraepelin sought to classify his patients
Kraepelin has had in the history of psychiatry.
illnesses. He adopted Kahlbaums course of
A prolic writer of psychiatric textbooks, Kraepe-
illness as a dening characteristic for his own diag-
lin was the most prominent German psychiatrist
nostic formulations (43). He periodically organized
up to World War I. He published his rst textbook
and re-organized his case records into clusters of
in 1883 (45) with revisions through an 8th edition,
similar symptoms and outcome. After much shuf-
which appeared in four volumes between 1909 and
ing of the le cards describing the observations in
1915. It is little wonder that Kraepelins image of
his patients, by 1899, he described two main
catatonia overwhelmed that of Kahlbaum. Kahl-
groups with dierent courses of illness, dementia
baum published his small book of 104 pages in
praecox and manicdepressive insanity.
1874, but the voices of the many authors who
Dementia praecox, as he described it, was a dis-
endorsed his work were faint compared with the
order in thought, speech, and aect that began in
booming of Kraepelin, Eugen Bleuler, and Adolf
adolescence and progressed to dementia. Kraepelin
Meyer, among many others. It was not until 1973
lumped together Heckers hebephrenia, Kahl-

10
Rediscovering catatonia

that the English translation of Kahlbaums book nia as an independent syndrome was buried. Until
became available and sparked the studies that the end of the twentieth century, Kahlbaums
found catatonia outside schizophrenia. catatonia was viewed within Kraepelins conjured
Kraepelin received his MD in 1878 and begun formulation of dementia praecox and subsequently
his academic travels rst as Professor at Dorpat within Bleulers concept of schizophrenia.
(Estonia) in 1886, then at Heidelberg in 1890. He
moved to Munich in 1903 where he founded a
Conflict of paradigms in psychiatry
German Institute for Psychiatric Research with
extensive funds from American philanthropists, As catatonia was incorporated as a feature within
the banker James Loeb, and the Rockefeller more severe chronic psychoses, a new inuence in
Foundation. The Institute established Kraepelin its recognition was the worldwide inuenza pan-
and Munich as a world center for biological psy- demic of 19171919. Many who survived the infec-
chiatric research. tions developed chronic illnesses. Stupors were
Kraepelin was nurtured in a world that saw common with active periods of slow movements,
the brain as the seat of psychiatric illness. At the shuing gait, rigid posturing, xed facies,
opposite end of the psychopathology spectrum decreased eye blinking, and extended staring. (In
stood Sigmund Freud with an emphasis on individ- retrospect, these signs are better seen as examples
ual psychology. Psychiatric symptoms typically of infection-induced catatonia.)
arose from the mental experience of the patient Labeled encephalitis lethargica, the syndrome
rather than as a result of a brain disorder, a seduc- was classied and treated as a form of Parkinsons
tive paradigm that captivated clinicians interest disease that is commonly found in the elderly
especially in the United States. An image of (47). But these signs were also identical to those
dementia praecox as a brain disease was replaced of catatonia, which by this time was accepted as
by an image of disorganization induced by child- an aspect of schizophrenia, leading the Viennese
hood experiences and memories, best relieved by psychiatrist Cosimo von Economo to describe the
individual psychoanalysis, a philosophy enthusias- patients as suering the same schizophrenia illness
tically adopted by Paul Eugen Bleuler (1857 that Kraepelin had described (48). For many
1939). patients, the illness was progressive, leading to
Bleuler obtained his MD in Zurich in 1881 and, long-term hospitalization either in neurologic cen-
after various positions in psychiatric hospitals, ters or in psychiatric sanitaria. Whether the label
became a professor in Zurich and head of the Bur- of encephalitis lethargica or of catatonic schizo-
gholzli Sanitarium in 1898. He studied hypnotism phrenia was applied in an individual patient
with Sigmund Freud, published a textbook on depended not on any distinguishing ndings but
schizophrenia in 1911 and a general psychiatry text on the attitude and experience of the observer and
in 1918. the setting.
Bleuler restructured Kraepelins image of The lesson that catatonia is a treatable syn-
dementia praecox using psychological theory and drome in postencephalitic states is still ignored. In
terms. It was he who created the name schizophre- the successful book and lm Awakenings, the neu-
nia, the favored name that reies the syndrome to rologist Oliver Sacks describes the patients exhibit-
this day. He described mutism, negativism, and ing prolonged and severe motor inhibition,
rigidity as generalised and persistent blocking posturing, rigidity, and mutism that began during
an exaggeration of the phenomenon seen in the encephalitis epidemic (49). The patients resided
healthy individuals when they are overwhelmed in a home for the elderly chronic ill. Their syn-
by emotional disturbance. (46). The patient with drome was labeled parkinsonism, and the newly
catatonia was suppressing unpleasant memories heralded treatment of high-dose l-dopa was
by silence (mutism), by tenseness and rigidity applied. Limited relief occurred but was hailed in
(holds back acts that are compelled by memories), the book and lm as a revolutionary discovery. At
by refusal to obey commands, and by displacing a meeting of the Society of Biological Psychiatry in
rising emotions and tension into motor acts that 1996, I asked Dr. Sacks whether he had considered
shut out reality (posturing, grimacing, staring, the patients to have catatonia. He averred that
stereotypes). they did not, nor had he considered using the treat-
By the end of World War I, the writings of ments for catatonia for these patients (50).
Kraepelin and Bleuler dominated psychiatric The conict of paradigms between clinical neu-
thinking until the philosophy of Sigmund Freud rology and psychiatry is ongoing, to the detriment
achieved dominance in America during and after of the clinical sciences and the welfare of the
World War II. Kahlbaums perception of catato- patients. (51) Examples are discussed in the assess-

11
Max Fink

ment and treatment of the neuroleptic malignant Kraepelin/Bleuler incorporation within schizo-
syndrome (Chapter 3) and anti-NMDAR encepha- phrenia. Interest in catatonia as an identiable
litis (Chapter 5). independent syndrome waned so much that when
Daniel Rogers, in an excellent recent review of the American Psychiatric Association presented its
motor disorders in psychiatry, presents portraits of classications of psychiatric illnesses in 1952, cata-
patients with dementia praecox by Kraepelin and tonia was only recognized as a type of schizophre-
comparable portraits of postencephalitic motor nia and remained so in the succeeding revisions in
signs. The pictures are indistinguishable (18). The 1968, 1980, and 1994.
label of schizophrenia or of a neurological disease
again depends not on objective ndings but on the
Psychiatry leaves the asylum
accident whether the referral was to the neurologic
or the psychiatric clinic. And the outcomes are Catatonia was further pushed into the background
determined by the treatments associated with each by changes in psychiatry. First, the enthusiasm for
diagnosis parkinsonism with l-dopa and similar the psychodynamic philosophy that blossomed
medications and schizophrenia with neuroleptic during World War II decreased interest in systemic
drugs. medical illnesses and moved the focus of psychiat-
A retrospective note on the increasing domi- ric practice from the asylum to the private oce
nance of psychology during the rst half of the and clinic. A principal inuence was the rapid
twentieth century stated The fact that an expansion of the numbers of physicians trained in
undoubted neurological disease, encephalitis psychiatry.
lethargica, could produce a wide spectrum of Involuntary military conscription of millions
psychiatric illness challenged the central idea of the of men brought many who were not t for such
so-called functional psychoses [i.e., not derived service, leading to an expansion of the need for
from brain pathology] in an era that had come to physicians trained in psychiatry to evaluate and
be dominated by the teachings of psychoanalysis treat those who became ill. Many physicians
(52). entering military service without prior psychiatric
The identication of catatonia within and out- experience, myself included, were trained in
side schizophrenia varied in the twentieth cen- schools of military neuropsychiatry. That
tury. In 1912, catatonia was described in 30 training was dominated by the psychodynamic
patients with infectious diseases, toxic states, philosophy.
depression, mania, and delirium, encouraging the William Menninger, a conrmed Freudian ana-
belief that catatonia occurred outside schizophre- lyst, was the head of the US Armys Department
nia (53). The prognoses varied, being good when of Psychiatry. Schools for military neuropsychia-
episodes were few and worsening as the episodes try were established in San Antonio, Texas, and
increased in number. In a 1922 study of 200 in Topeka, Kansas. I was a eld medical ocer
patients who met Kraepelins constructs, catato- in the US Army in 1946 when I was assigned to
nia was found more often among the manic the school in Texas. For 4 months, I attended
depressive patients than among those with daily classes in psychoanalysis, clinical psychia-
dementia praecox (54). try, and clinical neurology. Diagnosis and treat-
August Hochs 1921 monograph Benign Stupors ments were almost wholly psychodynamic. Lip
described 25 psychiatric patients treated at the service was played to the teaching of the biologi-
Wards Island Hospital in New York City. Their cal treatments of ECT, insulin coma, and lobot-
behaviors, motor signs, course, and outcomes are omy. In that prepsychopharmacology era,
similar to the patients described by Kahlbaum. pharmacology was focused on the used of sedat-
Classifying the patients using the Kraepelin/Bleul- ing drugs. When I left military service, I was
er distinctions, Hoch reported the prognoses were helped by the Servicemans Readjustment Act,
favorable with remission of symptoms and return known as the GI Bill, to pay tuition to attend
to the community in 13 manicdepressive patients. the William Alanson White Institute of Psycho-
The outcomes were poor in the 12 patients with analysis in New York City during my postgradu-
general medical illnesses or schizophrenia (55). ate residency training in neurology and
(Hoch lacked the eective treatments for catatonia psychiatry at Monteore, Bellevue, and Hillside
developed a decade later.) Hospitals. When the war ended, many physicians
Reviewing the twentieth-century psychiatric used government education benets to extend
literature nds many articles both in Europe and their training in psychoanalysis and then to open
the United States that describe catatonia as an private oces separate from hospitals for their
independent entity, but many more accept the clinical practices.

12
Rediscovering catatonia

Few connected with the large sanitaria where were not responding rapidly, electric-induced sei-
the more severely ill were housed, and psychiatry zures were added during the coma.
increasingly became an out-patient oce practice Seizures were rst induced chemically using
focused on psychotherapy. A few clinicians applied intramuscular injections of camphor, later with
their skills among those who were in hospital care, intravenous pentylenetetrazol (metrazol). Other
but it was soon clear that the severely ill were no chemical methods of inducing seizures were
longer of interest to most practitioners as their examined. In the 1950s, the inhalant urothyl
attention was focused on the young, alert, intelli- (Indoklon) was tested in random assignment
gent, attractive, emotionally ill who did not require studies vis a vis ECT. Flurothyl, an ether with
institutional care. The acute illnesses of catatonia the ether aroma and volatility, was presented to
did not lend themselves to oce care, and as inter- the patient by way of a mask that covered
est shifted from the psychoses to the less ill, interest mouth and nose. Within three to four breaths,
in catatonia dissipated. the patient was asleep and a seizure quickly fol-
A second factor in the virtual disappearance of lowed. These treatments were as eective as
catatonia from the psychiatric radar screen was the ECT, but the complexity of the administration
development in the 1930s of treatments that could and chemical cost limited the interest, and the
eectively and quickly relieve it. In 1930, William method was discarded. These chemical induc-
Bleckwenn demonstrated the immediate relief of tions were replaced by electricity in what is now
catatonia by injections of sodium amobarbital, a electroconvulsive treatment (ECT). In each treat-
medication that decreases anxiety and induces ment, the benet accrues to the number and
sleep (56, 57). The eects ended mutism, relieved frequency of seizures induced.
posturing and staring, and encouraged speech, Frontal leucotomy was the surgical procedure
feeding, drinking, and self-care. For some, relief that severed the connections between the frontal
was immediate and complete. For others, the bene- lobes and the rest of the brain, inducing changes in
ts were transient and treatments had to be contin- thought, reducing obsessions, and often inducing
ued. The ease with which a retarded and excited seizures. Leucotomy was introduced as a full
patient with catatonia could be relieved by a single neurosurgical procedure under sterile operating
chemical injection made the sodium amytal glass room conditions. But one of the proponents, Wal-
vials in wooden cylinders ubiquitous features of ter Freeman, introduced a quick method using an
clinical care in psychiatric hospitals and emergency ECT-induced seizure as anesthetic and cutting the
rooms. brain via an ice-pick-like device introduced
During my medical school training in psychia- through the thin plate over the eyeball. (58) Public
try at Bellevue Psychiatric Hospital in 1945, and and professional distaste for this procedure is illus-
again during neurology and psychiatry residency trated in the book and lm One Flew Over the
training between 1948 and 1951, I was equipped Cuckoos Nest by Ken Kesey (59). Interest in
with vials of sodium amytal and sterile water lobotomy ended when chlorpromazine and other
and was frequently called upon to treat mutism neuroleptic drugs were introduced and shown to
and negativism for feeding and to relieve excited be safer and more eective.
catatonic states by these injections. In retrospect, These treatments typically relieved the more
the relief of catatonia by amobarbital was the severe and malignant catatonias. The physicians
rst sign of the approaching era of psychophar- prescribing the treatments most often labeled the
macology. patients as suering from schizophrenia and only
Treatments for the most seriously ill psychiatric occasionally focused interest on catatonia. While
patients appeared next insulin coma therapy in continuing treatments were needed to sustain the
1933, chemically induced seizures in 1934, frontal benets, the prognosis for full relief became excel-
leucotomy in 1936, and electrically induced lent, making catatonia among the most success-
seizures in 1938. fully treatable conditions in psychiatry. Interest
Insulin coma patients were injected with large remained weak as the patients were quickly
doses of the hormone insulin that reduced blood returned to their homes and no longer were clinical
sugar to minimal survival levels, inducing coma challenges.
until blood sugar levels were normalized by intra- In the discovery that induced seizures relieved
venous injections or tube-feeding into the stomach schizophrenia, it was most fortuitous that Ladislas
of high concentrations of glucose sugars. In treat- Meduna selected patients with catatonia for his
ment programs, comas were induced daily for rst experiments. Nine of the rst 11 patients were
4050 comas over 3 months. Seizures occurred in catatonic requiring nursing care and naso-gastric
up to 20% of the treatments, and for those who feedings. Had he selected subjects from among

13
Max Fink

those with hebephrenia, his experiences would not An example is the 1969 study published by B.
have been encouraging (60). Pauleikho based on 35 years of experience with
Were these treatments, lobotomy, insulin coma, 552 hospitalized psychiatric patients (24). Pauleik-
and ECT eective in relieving schizophrenia? The ho identied stupor in 100 patients and excite-
answer depends on the populations selected and ment in 51 patients with catatonia. In 26 patients,
the settings from which they came. For the more catatonia was malignant, that is, life-threatening
acutely ill patients, mainly from out-patient set- and severe. Presenting his data in 5-year incre-
tings that included those with catatonia and those ments, he reported a drop in the frequency of the
with the mixed syndromes of depression and diagnoses after 1953, ascribing the reduced
psychosis (labeled schizoaective illness), the treat- numbers to changes in clinic administration and
ments oered dramatic relief. For the more chroni- diagnostic styles and not to changes in the inci-
cally ill, especially those with long-standing illness dence of catatonia. In 12 patient histories, he
diagnosed as the paranoid, hebephrenic, and non- illustrated catatonia variants that are readily rec-
catatonic forms of schizophrenia, neither ECT nor ognizable today as a syndrome distinct from
the other biological treatments were benecial. The schizophrenia.
site of the assessment, whether in a long-stay In the 1970s, James Morrison at the University
hospital population or in a general hospital of Iowa examined hospital chart records and
acute treatment center, inuenced the evaluation reported an increased incidence of catatonia in
(61, 62). patients with mood disorders, chiey those with
In 1981, the neurologist B. Mahendra posed the mania (64, 65). An examination of patients admit-
question, Where have all the catatonics gone? ted to a New York City hospital psychiatric unit
(63). Noting that the psychiatric texts had com- in the same period found a higher prevalence of
mented on the decline in the recognition of cata- catatonia among the patients with mania and
tonic schizophrenia and that the decline had depression than in those with schizophrenia (7,
preceded the inuence of neuroleptic drugs, 66). The same higher concentration of catatonia
Mahendra doubted that the disorder of catatonic among manic patients than in those with schizo-
schizophrenia ever existed. The frequency of asso- phrenia was reported from Germany (67). These
ciation of catatonia and schizophrenia depended reappraisals had little immediate eect but set the
on the incidence of the many ways in which catato- stage for liberating catatonia from schizophrenia.
nia was elicited. He cited examples of catatonia As we will see, they made it possible for the
secondary to infections (as in encephalitis) and in appearance of an acute, often lethal syndrome
the excitement and retardation of the mood disor- associated with high-potency neuroleptic drugs to
ders. As an analogy, he noted that tuberculosis rekindle interest in catatonia as independent of
had been frequently found among severely ill psy- schizophrenia.
chiatric patients but had not been reied as a
tuberculous schizophrenia. Catatonia had not
disappeared, but the connection to psychosis had
been prematurely reied.

14
Rediscovering catatonia

Chapter 3: the neuroleptic malignant


syndrome
With all these disadvantages of re-examination, I can for these eects were prescribed, but with little
only expect to dig my new species [of clinical case] from relief. Despite the increasingly severe impacts on
the mass of rubbish of confused, irregular conglomera- movement and speech, the balance between the
tions of amorphous appearance, to separate it from the reduction in expressed delusions and hallucina-
encumbrance of incidental matters, and so present it, tions, aggressive acts, screaming behaviors, and
that others may be able to satisfy themselves of its frightening thoughts was considered favorably by
individuality, and then enter upon the critical and exact administrators, so that continued prescription was
analysis of its true and essential essence; or else to decide widely endorsed.
that it is only an old variety with some accidental and The benets were also considered sucient to
illusive peculiarities. Luther V. Bell, 1849 (68) replace insulin coma (ICT), a more dangerous and
unpleasant treatment then in use. In 1933, the Aus-
Catatonia faded from psychiatrys awareness
trian psychiatrist Manfred Sakel proposed treating
until the hazards of the neuroleptic malignant syn-
schizophrenia by daily injection of increasing high
drome brought it to professional attention. Fre-
doses of the hormone insulin to lower blood glu-
quently fatal, the stupor, rigid posturing, mutism,
cose levels to states of coma (69). After an hour of
and repetitive speech of critically ill delirious
observed coma, an intravenous injection or naso-
patients renewed interest in catatonia, especially
gastric gavage of concentrated glucose quickly
when the treatments for catatonia were found to
returned the patient to consciousness. Within an
relieve the neurotoxic syndrome.
hour, patients were alert having showered, toileted,
Chlorpromazine (CPZ), the rst of the neurolep-
and been fed, able to return to their treatment
tic drugs, was introduced in 1954 as a sedating
ward. Psychotic thoughts and aggressive behaviors
agent for excited psychotic patients. The aggres-
were inhibited so that about one-third of the
sion, screaming, re setting, breaking furniture,
patients left the hospital and returned to their
and physical injuries that were common in the
families after courses of up to 50 comas.
sanitaria were quickly reduced, benets that were
In the 1940s and 1950s, about 40 patients were
recognized in every psychiatric center where it was
treated with ICT each year at Hillside Hospital. In
used. As the director of psychiatric research at
the 5 years, I treated about 200 patients. Pro-
Hillside Hospital in Long Island, New York, a
longed coma, the failure of the patient to recover
200-bed sanitarium, I also studied its eects.
consciousness as glucose was administered,
Within a few weeks, the nurses who rst saw the
occurred in 3% of the treatments. The patients
benets eagerly referred their most disturbed
required intensive nursing care and careful man-
patients for the experimental treatment. By 1957,
agement of uids and blood electrolytes. We tried
the antidepressant imipramine was introduced
many medications, including the newly introduced
followed by a ood of new chemicals for the relief
steroids, with none proving useful. Three patients
of anxiety, depression, and psychosis. The enthusi-
died. Spontaneous seizures occurred in up to 20%
asms of psychopharmacology soon dominated
of the treatments, and when no benets were
both the profession and the public media and have
apparent after a month of comas, we added ECT
done so to the present.
to the comas. ICT was a laborious and riskful
But patients paid heavily for these benets.
treatment of marginal benet.
Motor movements slowed and gait became
We soon recognized that the patients who
ungainly. Sti and rigid posturing was accompa-
improved with ICT were also relieved by CPZ.
nied by tremors. Drooling slathered their cloth-
Beginning in 1957, the patients referred for ICT
ing. Their faces were emotionally at, without
were randomly assigned to either a 50-coma treat-
smiles or laughter. Some became jaundiced,
ment course of ICT or to daily oral dosing with
itched, and scratched leaving long red trails on
CPZ. In the prior 3 years of experience, we had
their arms and legs. Patients refused their medi-
learned that about 1200 mg/day of CPZ was the
cines, and in that era of paternalism and invol-
eective dosing schedule. In the study, patients
untary treatment, the new drugs were often
received doses beginning at 300 mg/day rising
administered forcibly.
weekly to 1200 mg/day. The median dosage was
We had no knowledge of how to prevent these
800 mg/day for the 10-week course.
eects. Many chemicals thought to be prophylactic

15
Max Fink

Within 15 months, we had treated 50 patients, sures, especially in older patients, voices became
half with CPZ and half with ICT. A seminal 1958 husky. Feeding and talking were made dicult by
paper in the Journal of the American Medical worm-like writhing mouth movements. Labeled
Association reported similar reductions in psycho- tardive dyskinesia and tardive dystonia, the move-
sis and aggression with both treatments (70). But ments were uncontrollable, becoming progressively
the risks for prolonged coma and death, weight more severe and unsightly. No eective treatment
gain, and nursing care costs of ICT were greater or prevention was known as patients risked the
than the risks and costs of CPZ. By the end of Scylla of psychosis or the Charybdis of dyskinesia.
1958, the hospital discontinued its use, and soon The sanitaria were soon lled by slow, shuing,
ICT had been widely abandoned throughout the trembling, shadows of human beings, occasionally
nation. laughing or giggling uncontrollably (72). The
An interesting footnote to the ICT history is motor eects of the neuroleptic drugs were so com-
described by the author Sylvia Nasar in the biogra- monly recognized that measuring the severity of
phy and lm A Beautiful Mind, the story of the tremors in handwriting was suggested as a criterion
1994 Nobel Economics Prize winner John Nash. to identify an active clinical agent (73).
He had become psychotic soon after completing The enthusiasm for prescribing these agents
his degree at Princeton and had begun a teaching would be severely lessened if the eects on move-
career. By 1961, he required hospital care for a ment were widely discussed. The pharmaceutical
paranoid psychosis and was admitted to the Tren- industry, frightened that their sales would be
ton State Hospital where ICT was still available. impeded, encouraged leading psychiatrists
His treatment with ICT relieved the principal (known opinion leaders) to publicly opine that
symptoms, but he remained chronically ill. (71) the motor changes were inconsequential and toler-
able. The movement eects were widely denied
and pushed out of professional and public aware-
Suppression of interest in movement disorders ness. In hospital settings where physicians exam-
More potent neuroleptic drugs, such as uphen- ined patients at their bedsides, the measuring and
azine, triuoperazine, and haloperidol, were next recording of the motor signs were feasible. But in
introduced to the psychiatric community and oce settings, physicians sat behind their desks,
widely prescribed. Hospital wards became quieter, and physical examinations were not carried out,
and increasing numbers of patients moved from in- further minimizing the recognition of motor signs
patient to out-patient care. The rapid discharge of of illness.
patients and the closing of the asylums, the process When newly introduced the second-generation
soon labeled deinstitutionalization, began as a or atypical neuroleptic drugs clozapine, risperi-
trickle and soon became a ood, closing the major- done, olanzapine, and quetiapine were marketed
ity of the sanitaria in the United States and in the 1990s, the banner argument for their use was
throughout the world by the end of the century. their lesser motor eects. It took more than two
Neuroleptic drugs suppress psychosis but do not decades after the rst appreciation of the motor
cure. Their eects are more like that of steroids toxicity of neuroleptic drugs for these eects to be
that suppress symptoms than like those of an anti- publicly acknowledged and then mainly as an
biotic in relieving an infection by destroying the advertising strategy to encourage prescription of
invading agent. Neuroleptic drugs require contin- the second-generation neuroleptics. It took
ued prescription to maintain health. As the dos- another two decades to recognize that the lesser
ages of neuroleptic drugs are reduced or movement eects were tied to the lesser antipsy-
withdrawn, symptoms are. We did not know chotic potency of the new agents: their benets
then, nor do we know now, for how long treat- were only slightly better than those of placebos
ments should be sustained nor did we understand (74).
the risks. Medication treatments, month after The active suppression of the recognition of
month and year after year are accepted clinical abnormal movements with neuroleptic drugs also
practice. suppressed the recognition of catatonia. The belief
As more patients exhibited peculiar motor that catatonia had disappeared was further
movements, the association with neuroleptic drugs encouraged by the segregation of the severe psychi-
was increasingly recognized. Tremors interfered atrically ill outside the experience of community
with writing and speaking, gait became slow and physicians and the many psychiatrists now active
shuing, and as eyelids did not blink, patients only in their oce settings. The full range of motor
appeared to be constantly staring. After long expo- signs of parkinsonism, tardive dyskinesias, and

16
Rediscovering catatonia

catatonia was only identied in long-stay facilities schizophrenia, including the cases labeled as the
(75). catatonic form of schizophrenia, treatment options
became confused. When patients exhibited signs of
psychosis, physicians prescribed neuroleptic drugs.
The neuroleptic malignant syndrome enables But these also increased neurotoxicity. It took
recognition of catatonia some time to realize that discontinuation of
The recognition of catatonia was energized by the neuroleptics was essential to relieve the toxic
awareness that the more potent neuroleptic drugs syndrome.
risked inducing acute febrile catatonic syndromes. Matters were not helped by an assertion that
Described within the decade after CPZ was increased motor rigidity and fever were signs of
introduced and labeled, the syndrom malin des malignant hyperthermia (MH), a syndrome of
neuroleptiques an acute neuroleptic toxic syn- muscle weakness and fever that follows the use of
drome patients suddenly became feverish, stu- inhalation anesthetics (83). This syndrome is
porous, and delirious (76, 77). Heart rate, blood restricted to a few patients with an identiable
pressure, breathing rate, and sweating increased genetic fault. For symptomatic relief of MH, the
without evidence of infection. Catatonic signs muscle relaxant dantrolene is prescribed. As a
became prominent. This life-threatening syndrome result of this confused association, dantrolene was
appeared without warning, often following a tran- added to the recommended prescriptions for NMS.
sient inuenza-like syndrome of malaise, fever, and In 1997, the newly established NMS Internet Hot-
weakness (7880). line made prescription of both bromocriptine and
As agents with greater neuroleptic potency than dantrolene and withdrawal of the oending
CPZ were marketed, severity of the toxicity neuroleptic the cornerstones of the recommended
increased. Evidence of the syndrome in eight management.
patients treated with high-potency neuroleptic Neuroleptic malignant syndrome was soon
drugs described by Alan Gelenberg of Bostons sensed as clinically identical to malignant (that is,
Massachusetts General Hospital compelled atten- lethal) catatonia (MC) that had been described
tion, and clinicians became frantic to understand before the neuroleptic drugs were introduced. Many
these phenomena (81, 82). forms of MC had been recognized, and the ecacy
The cause was puzzling. Were these acute symp- of ECT in its relief was reported by 1952 (2).
toms evidence of the blockade of dopamine neuro- By 1984, we had recognized NMS in three
transmission that was reported in laboratory patients at the University Hospital at Stony Brook,
studies and considered the basis for the relief of each with prominent signs of catatonia (3). We rst
thought disorders? Dopamine was one of the summarized the prior reports of 17 similar cases;
brains principal intercellular communication sys- 11 had been precipitated by haloperidol, a nding
tems impaired by the new drugs. Perhaps prescrip- that since has been repeatedly conrmed. Two
tion of dopamine agonists, drugs that increased patients had died with the others slowly recovering
brain dopamine levels, would reverse toxicity? after extended nursing care. Dierent psychoactive
A 1980 review of more than 60 published cases agents had been prescribed benztropine, dantro-
brought particular attention to the syndrome that lene, amantadine, bromocriptine, and diphenhy-
was labeled the neuroleptic malignant syndrome dramine but none were eective.
(NMS), a name that was widely accepted (83). The Among our patients, a 30-year-old woman had
syndrome was labeled malignant in recognition been treated with the neuroleptic uphenazine and
that many patients died in an acute autonomic then admitted to the hospital in an acute state of
storm of high fever, elevated blood pressure, rigidity, posturing, mask-like facies, profuse sweat-
increased heart rate, irregular heart rhythms, and ing, and fever. She was treated with the antihista-
sweating. Each case report recognized rigidity, mine diphenhydramine, the dopamine agonist
dyskinesia (abnormal motor movements), postur- bromocriptine, the antimanic lithium, and the anti-
ing, mutism, and negativism, the classic signs of psychotic mesoridazine, each selected to relieve
catatonia. This publication cited dopamine block- one of her symptoms. She received little benet,
ade as the cause of NMS and endorsed treatment and only after all medications were discontinued
with the dopamine agonists bromocriptine and and weeks of nursing care did the toxic syndrome
amantadine. slowly resolve and she was able to return home.
When it was realized that the same neuroleptic In a 45-year-old disorganized and psychotic
drugs that inadvertently led to the appearance of woman with depressed mood, the neuroleptic
these catatonic signs were used to relieve haloperidol and then uphenazine had been pre-

17
Max Fink

scribed. NMS was precipitated. A 5 mg diazepam Michael Taylor and I summarized the extensive evi-
dose administered intravenously as a sedative dence in the textbook Catatonia: A clinicians guide
resolved the motor syndrome quickly but tran- to diagnosis and treatment (11). But ECT is much
siently. Continued treatment with bromocriptine stigmatized, and many institutions lacked the phys-
and lorazepam oered little benet. In another ical facility or the trained personnel for its use lead-
patient, a 26-year-old man with bizarre thinking, ing to ethically troubling reports of prolonged
slow speech and movement, and the catatonic signs hospital care and unnecessary deaths in patients
of posturing and rigidity had been transferred with NMS. Fortunately, the benzodiazepines, a less
from a state hospital for treatment with ECT. An controversial and less invasive treatment than
intramuscular injection of the long-acting neuro- ECT, were also available to relieve NMS.
leptic uphenazine decanoate was given and within
72 h NMS was recognized.
Catatonia and benzodiazepines
Electroconvulsive therapy was prescribed for
both patients, and within 1 day, the toxic syn- Catatonia had rst been successfully treated with
drome had resolved dramatically for each. Their intravenous injections of the barbiturate amobar-
ECT treatment courses were continued until the bital in 1930 by William Bleckwenn in Wisconsin
toxic states had fully resolved without residual (56). Within 4 years, relief by inducing grand mal
motor or toxic signs. We concluded that NMS was seizures was described by the Hungarian neuropsy-
a physician-induced form of malignant catatonia chiatrist Ladislas Meduna (89). Both treatments
treatable by ECT. were widely adopted when catatonia was recog-
Over the next few years, other patients with nized as such. Once NMS was pictured as a form
NMS were successfully treated. We described our of catatonia, the question was raised whether bar-
awareness that the neuroleptic drugs, particularly biturates would relieve NMS. Ever since its rst
the high-potency drugs, were also toxic in eliciting description, amobarbital had been the accepted
syndromes of catatonia and fever and that the tox- immediate treatment of catatonia in emergency
icity resolved with neuroleptic drug withdrawal; room settings. By the 1980s, concerns about the
and if withdrawal alone was not sucient, ECT toxicity and safety of the barbiturates led to their
was a denitive treatment. replacement by benzodiazepines that were used as
This awareness was documented hesitantly as sedatives, soporics, and anticonvulsants.
ECT was the most stigmatized treatment in psychi- In 1983, Gregory Fricchione and his colleagues
atry. Yet, by 1990, the recognition of catatonia at Massachusetts General Hospital described the
warranted immediate treatment with ECT even rapid relief of NMS by intravenous lorazepam, a
before medication trials (4). By 1991, Denise White commonly used benzodiazepine (90). Each of four
reported that catatonia was a risk factor for neuro- patients they treated exhibited catatonia, fever,
toxic development (84) and then catatonia was agitation, and delirium, with elevated blood pres-
seen as integral to the diagnosis (85). These reports sures, rapid heart rates, and increased sweating.
crystallized the integral role of catatonia in NMS. Haloperidol had been the toxic agent in three
That same year, collaborating with Michael Tay- instances and triuoperazine in one. After various
lor, we suggested to the American Psychiatric prescribed medications and nursing care had failed
Association DSM-IV commission, then revising to relieve the syndrome, intravenous lorazepam in
the classication criteria, that catatonia warranted 2-mg doses was administered. In each patient,
a place distinct from its role as a type of schizo- NMS resolved within a day, with some symptoms
phrenia (9). Increasingly, the argument was made resolving within hours. This dramatic experience
that NMS was best regarded and treated as a form established lorazepam as an eective treatment for
of malignant catatonia and that eective relief was catatonia.
assured when treatments for catatonia were applied In discussing this experience, Greg Fricchione
rather than those considered useful for an aberra- relates: I had treated a patient in postcardiotomy
tion of neurotransmitter activity (5, 86). By 1996, a delirium with intramuscular and then intravenous
rating scale was developed for catatonia and a sys- haloperidol and he developed neuroleptic induced
tematic study established the lorazepam sedative malignant catatonia. I brought my mentor Ned
test as a diagnostic verication of catatonia. Treat- Cassem the Director of the MGH Consultation
ment with high doses of lorazepam was shown to Psychiatry Service and both a physician and
be eective for 80% of the patients with catatonia Jesuit priest with me around 8 pm to see the
and ECT eective for the remainder (87, 88). patient who had not responded to diphenhydra-
In the next decade, numerous reports found mine or benztropine. Ned chose lorazepam and I
NMS rapidly responsive to ECT, and in 2003, injected 2 mg intravenously in the presence of

18
Rediscovering catatonia

several of the nurses. Catatonia lysed [disap- phrenia was so intimate that the prior recognition
peared] dramatically in about 1 min; the nurses of schizophrenia was obligatory before catatonia
who all knew Ned asked in wonderment: What could be considered. The recognition of catatonia
did you give him Dr Cassem? And as he turned outside schizophrenia reported in the 1970s had
to leave, Ned said nonchalantly, why holy water yet to become part of psychiatrys teaching. In the
of course era of psychopharmacology, psychiatric disorders
By 1990, Patricia Rosebush and her colleagues are perceived as errors in neurotransmitter physiol-
at McMaster University in Hamilton, Canada, ogy, favoring the belief that NMS was the result of
also described complete and dramatic relief of a decrease in brain dopamine activity and making
NMS by lorazepam in a prospectively open trial in the prescription of dopamine agonists a logical
1 mg or 2 mg doses in 12 episodes (29). Other treatment. The same beliefs would not support
reports of the ecacy of lorazepam (91, 92) were treatment with benzodiazepines (or ECT) as no
followed by descriptions of the ecacy of other theory of neurotransmitter action had been pro-
benzodiazepines, clonazepam (93), diazepam (94), moted for catatonia or malignant catatonia. Beliefs
and zolpidem (95). in dopamine aberration in NMS were very strongly
At Stony Brook University Hospital in 1994, 28 held, and intense public debates were frequent for
patients with catatonia were systematically treated more than a decade.
with injected and/or oral lorazepam for up to
5 days and with ECT when lorazepam failed. Out-
Recognizing catatonia today
comes were monitored quantitatively during the
treatment phase with a Catatonia Rating Scale With the renewed interest in catatonia and the
(87). In 16 of the 21 patients (76%) who received a appreciation that catatonia often occurs indepen-
treatment trial of lorazepam, catatonia resolved dent of schizophrenia, it became important to
within 1 day. Three cases were examples of NMS. develop criteria to dene its many variations. In
In 11 patients to whom we had administered an clinical medicine, a diagnosis is made, and treat-
intravenous dose of lorazepam, immediate relief of ment is oered after the patient relates his symp-
catatonia was the result, the harbinger of the full toms and recent history; the physician examines
relief that followed treatment with high oral doses for signs of dysfunctions; laboratory tests are
of lorazepam. Four patients who did not have an requested and assessed; and based on these nd-
immediate response to intravenous lorazepam were ings, the clinician considers a diagnosis from his
successfully treated with ECT (88). glossary of disorders. After identifying the most
By 1990, two competing treatment protocols likely t, treatment is prescribed. When a specic
were prescribed for NMS the bromocriptine treatment leads to recovery, the diagnosis is con-
dantrolene model based on the dopamine/malig- sidered validated. Patients with catatonia rarely
nant hyperthermia association and the benzodiaze- complain of symptoms; the behaviors are observed
pineECT model based on the recognition that or reported, and examination identies the
NMS is the form of malignant catatonia. Although presence of identiable catatonia signs. When
no direct comparisons between the two treatments motor signs are prominent, a checklist of catatonia
were made, the ecacy of the benzodiazepine signs sorted in a Catatonia Rating Scale becomes a
ECT model assured its widespread use and accep- useful guide to the examination (11).
tance. By 1991, doubts as to the merit of dopamine Catatonia is an all-or-none phenomenon, so the
augmentation as the eective treatment for NMS presence of two or more catatonia signs for 24 h or
were expressed (96). By 1995, the hypothesized longer is sucient to consider it to be present. Sur-
connection of NMS to malignant hyperthermia veys of populations in psychiatric hospital wards
was also found wanting, and treatment with dan- and emergency rooms nd that 715% exhibit
trolene was no longer recommended (97). Thereaf- catatonia (11, 12).
ter, whether NMS was relieved or a fatality ensued Once the diagnosis is entertained, the clinician
depended not on the administration of dopamine can conrm the diagnosis by employing a sedative
agonists or dantrolene but whether the neuroleptic relaxation test (lorazepam test). William Bleck-
administration had been promptly discontinued wenns description of immediate relief of catatonia
and uid and metabolic integrity re-established. by the intravenous injection of amobarbital
Well-documented texts describing the neuroleptic sodium in his article The production of sleep and
malignant syndrome as a variant of malignant rest in psychotic cases presented both an eective
catatonia appeared in 2003 (11) and 2004 (98). treatment and a diagnostic test (56). For patients
What delayed the recognition of NMS as drug- who are mute, posturing, rigid, or with other cata-
induced catatonia? The tie of catatonia to schizo- tonia signs, the return of speech and relaxation of

19
Max Fink

posture and tension are prompt, usually within pam were successfully tested in catatonia treat-
10 min after an injection of sodium amobarbital ment. Numerous anecdotes cite the patient with
or, more commonly today, after 1 mg or 2 mg mute catatonia being sent to a hospital laboratory
lorazepam. The change may be transient or may for a procedure after an injection of lorazepam to
persist, but the immediate relaxation conrms the assure cooperation, only for the ward nurse to be
presence of catatonia. called with a complaint that the patient is overly
For many decades, amobarbital and other bar- talkative and uncooperative in the procedure
biturates were essential features of hospital emer- room. Intravenous dosing in a mute, posturing, or
gency room care. As a medical student and stuporous patient typically relieves the syndrome
resident in the 1940s, I was usually in the hospi- within a few minutes.
tal carrying a small wooden cigar case with a How assured is the response to an intravenous
glass vial of 500 mg amobarbital powder, syr- injection of 1 mg lorazepam or 2.5 mg diazepam
inge, needle, sterile water, and tourniquet as verication of catatonia? Almost all patients
always prepared and often used. In his clinical with positive scores on the Catatonia Rating Scale
review of the diagnosis and management of are relieved. Occasionally, a second dose is
catatonia in 1975, James Morrison studied 250 required and applied within 10 min. It is not
patients with catatonia of whom 97 were exam- stretching the comparison too far to say that a
ined by a barbiturate interview, the intravenous sedative drug acts with the same dose-related speed
injection of up to 5 g of amobarbital (99). He as a concentrated acid or base reverses the pH bal-
reported that 71 (73%) responded favorably, ance of a chemical solution.
meaning a quick relief of mutism, tension, pos- Once the presence of catatonia is veried, treat-
turing, and stereotypy. He gives the example of a ment today is remarkably eective. More than
23-year-old single man with a 2-year history of 80% of the patients are relieved by barbiturates or
persecutory and grandiose delusions who exhib- benzodiazepines, and when these fail, the others
ited waxy exibility and posturing of the psycho- are relieved by ECT (11).
logical pillow (head held elevated as if on a The availability of both a reliable and quick
pillow) and not responding to antipsychotic treat- diagnosis method and eective treatment has
ments. After the intravenous administration of enabled the discovery of additional forms of cata-
four grams of sodium amobarbital, the rigidity, tonia (14, 100). Once catatonia was divorced from
tension, and posturing lessened and the patient schizophrenia and the main motor signs were
began to speak of the terror he had felt when he appreciated, other forms of catatonia, such as
realized that the television was bugged and the delirious mania, toxic serotonin syndrome, and
FBI was gathering information about him. pediatric catatonia, were recognized and their
In the early 1980s, as discussed earlier, ben- diagnostic criteria and treatment algorithms
zodiazepines were recommended as safer sedatives established.
than the barbiturates, and lorazepam and diaze-

20
Rediscovering catatonia

Chapter 4: new forms are recognized


According to an old story, there are three types of base- lupus erythematosus (a highly varied systemic,
ball umpires. One says: I call them [balls and strikes] as often fatal autoimmune disease) was referred for
they are. The second says I call them as I see them. ECT by Dr. Gregory Fricchione, the head of the
And the third says What I call them is what they hospitals Consultation & Liaison Service (103).
become. Frederick Grinnell, 1992 (101) Her agitated manic behavior, facial grimacing,
mutism, and rigidity were poorly controlled. She
Recognizing the neuroleptic malignant syn-
required high doses of sedative drugs and four-
drome (NMS) as a form of malignant catatonia
limb restraints to sustain her life in intensive
showed that catatonia was found outside schizo-
nursing care. With the consent of her husband, her
phrenia. It also oered eective treatment for the
mother, and her physicians, the rst ECT was
patients and justied the challenge to the hypothe-
given on the 27th day of hospital care.
sis that NMS was the result of dopamine blockade.
Mania and delirium rapidly disappeared, speech
Breaking the association with schizophrenia freed
and self-care improved, posturing, mutism, and
clinicians to see catatonia in broader samples of
screaming were allayed, so much so that after
patients. It encouraged the development of Catato-
seven treatments the family and her physicians
nia Rating Scales and exploration of an injection
withdrew consent for further treatment. But treat-
of lorazepam as a verifying diagnostic test. It stim-
ment was incomplete and symptoms soon
ulated the search for catatonia in other guises.
returned. The ECT course continued, and she
Among patients admitted to the hospital in a
received 10 more treatments. Her psychiatric and
hyperactive agitated delirium, the uctuating signs
medical conditions were relieved, and she left the
of catatonia were commonly recognized. The risk
hospital in psychiatric and systemic remission.
of death led to many descriptions of excited deliri-
On admission, she had exhibited the systemic
ous states with prominent signs of catatonia, vari-
signs of lupus erythematosus supported by positive
ously labeled malignant, pernicious, lethal, fatal, or
antinuclear antibody tests. Treatments with intra-
mortal catatonia; or delirium becomes the focus as
venous methyl prednisolone and three plasmaphe-
in delirious mania or manic delirium; or the eponym
resis exchanges did little to improve her condition.
of Bells mania or Stauders lethal catatonia was
Treatment with cyclophosphamide was begun but
used.
her delirium and excitement led to the referral for
Acute signs of NMS were also identied in sub-
ECT. With ECT, her behavior normalized and
jects who had not been treated with neuroleptics
she returned to her home. Cyclophosphamide
but with psychoactive drugs that aected the
continued monthly, and she was well on 1-year
serotonin neurotransmitter system. These patients
re-examination.
suered what became known as the toxic serotonin
Similar cases of delirium are commonly found in
syndrome, a parallel disorder to NMS.
medical, surgical, and emergency room settings.
Increasingly, the signs of catatonia were found
For more than two centuries, clinicians have
among adolescents admitted to our facility, at
described patients with the acute onset of an
rst among those with peculiar behaviors associ-
excited delirious state, dangerous to themselves
ated with mental retardation and then those with
and to others, frequently so severe as to result in
delirious mania (102). Many patients showed
death. In 1849, Luther Bell, in a 13-year review of
repetitive self-injurious behaviors (SIB), but we
1700 admissions to Bostons McLean Hospital,
did not realize that this behavior was a form of
described 40 patients with delirium, fever, and
catatonia until we read reports from other medi-
uncontrollable excitement that alternated with stu-
cal centers among their patients with neurobe-
por. Three-quarters had died. The syndrome
havioral disorders. That the patients improved
became known as Bells mania (68).
with ECT validated the association (19). Such
In 1934, Karl Heinz Stauder described the
repetitive behaviors are now recognized within
sudden onset of intense excitement, delirium, high
the catatonia spectrum.
fever, and catalepsy in three young adults with a
family history of psychiatric illness but who previ-
Delirious mania as malignant catatonia ously had been in good medical health. In a
matter of minutes, each went from well to
Our experience associating catatonia with delirium extreme excitement and psychosis. During the
increased when an intermittently mute and scream- illness, each injured himself by slamming repeat-
ing 25-year-old woman, ill with a 3-year history of

21
Max Fink

edly into the ground or walls. All three died. Stau- After the second ECT 2 days later, he became better ori-
der termed the syndrome todliche katatonie (fatal ented and cooperative. The next day he came voluntarily
catatonia) (104). for the third treatment. Delirium, catatonia signs, and
Similar descriptions by many authors conrm excitement were fully relieved by the sixth treatment,
the characteristics of delirious mania as a form of and he was discharged to the care of his family. Loraze-
malignant catatonia (2, 105114). The principal pam was discontinued, and lithium therapy was pre-
feature is the acute onset of a nightmarish, dream- scribed for persisting mania. Follow-up 2 months later
like, overactive state that develops within a few showed him to be continuing his medical treatment with-
hours or a few days. Voices, noises, and sights are out any persisting signs of psychiatric illness.
misperceived, encouraging frightening thoughts. In this patient and in additional similar cases
Patients hit out and thrash about, harming them- described in texts on ECT, the risks of precipitat-
selves and others. Grimacing, posturing, echolalia ing a neuroleptic malignant syndrome precluded
(repeating words), and echopraxia (repeating the prescription of haloperidol and other potent
movements) are prominent. Negativism (refusing neuroleptic drugs to control behavior (70). Ben-
to obey commands) and automatic obedience zodiazepines were preferred. While doses of loraze-
(obeying commands that they are told not to obey) pam to 10 mg/day reduced manic and aggressive
are almost always present. Patients sleep poorly behavior, these did not relieve delirium. ECT,
and are unable to recall their recent experiences or however, eectively resolved the delirious syn-
the names of objects or numbers given to them. drome in each case. We had been encouraged to
They are disoriented and they confabulate. apply ECT in delirious patients by reports pub-
Patients hide in small spaces, close the doors and lished in 1952 by the Austrian clinicians Otto
blinds on windows, and remove their clothes run- Arnold and H. Stepan (115). They were faced with
ning nude in and from their homes. Garrulousness, patients with the life-threatening condition of
ights of ideas, and rambling speech alternate with malignant catatonia, and lacking any eective
mutism. Fever, rapid heart rate, elevated blood treatment risked the use of ECT. To induce grand
pressure, and rapid breathing are common. An mal seizures in acutely ill moribund patients was
example is abstracted from my review of delirious counterintuitive. Yet, 16 of 19 delirious patients
mania (109). survived when they were treated with ECT within
A 29-year-old man with a positive history for HIV had 5 days of the onset of the illness; of the 14 whose
been found wandering on a beach, naked, confused, and treatment had been delayed, none survived. Daily
unable to rationally answer questions. He was admitted ECT and up to three seizures a day were necessary
to the psychiatric emergency room of Stony Brook Uni- to relieve the more severe conditions.
versity Hospital. Despondent about his illness, he Although such published experiences were few,
stopped taking the prescribed medicines, and said that ECT became the accepted treatment. Not all the
he wished to die. The day before admission, he had been cases were associated with neuroleptic drugs,
slashing pictures of his friends and walking about the although these were the more common forms of
house with a bloody knife, muttering incoherently. physician-induced deliria (116118).
In the French literature, a dreamy, stuporous
On admission, he was excited, screaming, confused, syndrome is described as onirisme (translated as
dehydrated, unkempt, and dirty. He required four-limb oneiroid state, oneiroid syndrome, or oneirophre-
restraints and 0.5 mg of the antipsychotic chemical nia) (119). In a short delightful 1950 text, Ladislas
restraint haloperidol was administered intramuscularly. Meduna, the developer of convulsive therapy,
Within an hour he was rigid and mute, febrile, dehy- described such patients in sucient detail to
drated, and urine showed evidence of cannabinoids. identify their syndrome as a form of malignant
An intravenous injection of 1 mg lorazepam quickly catatonia (120). He distinguished the syndrome
reduced the catatonia signs. Mutism, rigidity, staring, from schizophrenia and reported 64% full remis-
and periodic excitement persisted despite daily loraze- sion with ECT.
pam dosages to 10 mg per day. Neuroleptic medication The cause of delirious mania is unknown, but
was precluded by his reaction to haloperidol. the syndrome is often preceded by an acute infec-
tion, a traumatic incident or poisoning (121123).
Surrogate consent was obtained for ECT. On day 11, the As delirium dominates the story, the connection to
rst seizure was induced and within 24 h he no longer a prior illness is usually overlooked. A connection
required protection. Mutism, rigidity, and staring disap- to bipolar disorder or manicdepressive illness is
peared. Within 2 h his temperature rose to 102F and often assumed, but is rarely conrmed by the
resolved to normal over the next 12 h. patients history. Considering delirious mania as a

22
Rediscovering catatonia

form of catatonia rather than as a form of bipolar attended unit activities. Three additional treatments
disorder oers a bridge to quick, eective relief resolved the psychosis, depressed mood, agitation, and
with catatonia treatment. motor and vegetative signs (126).
The sudden appearance of delirious mania in a Her condition met the diagnostic criteria for the
public place has been hazardous. One example, as toxic serotonin syndrome (TSS) (127). Serotonergic
described by Edward Shorter, is that of a non- drugs are used widely to relieve depressed mood
English-speaking immigrant from Poland upon his and may precipitate an acute febrile syndrome of
arrival at a Canadian airport. Confused by the twitching muscles, tremors, diarrhea, and mental
requirements of customs control, he could not nd changes. Such states follow a rapid increase in dos-
his mother whom he expected to meet. He became ing or when a selective serotonin reuptake inhibi-
increasingly agitated, sweating, dehydrated, and tor (SSRI) is combined with a monoamine oxidase
aggressive. Police were called and sought to inhibitor (MAOI).
control his behavior by electric taser applied Patients become restless and sleep poorly, their
up to four times; he fell to the ground scream- sensorium is altered, the skin is ushed, and they
ing and convulsing, and minutes later was dead complain of sweating, tremors, shivering, lethargy,
(124). Similar experiences dot the literature (125). salivation, nausea, diarrhea, and abdominal pain.
Laboratory tests of body functions show signs of
Toxic serotonin syndrome acute toxicity. Temperature and blood pressure are
elevated, tendon reexes are hyperactive, move-
Occasionally, an acute febrile and toxic reaction ments ataxic, and myoclonus appears. The big
mimicking NMS follows the administration of picture is of a malignant catatonia/neuroleptic
drugs that aect the brains serotonin system. In malignant syndrome with gastrointestinal
1994, a woman was admitted to University Hospital symptoms (128133).
at Stony Brook with the motor and vegetative signs Although most patients respond rapidly to with-
of NMS. She had not been exposed to neuroleptics drawal of the oending medicines and supportive
but had been treated with serotonergic drugs. care, ECT has been used successfully (134, 135).
A 59-year-old married woman with a 21-year history of The overlap in diagnosis of TSS and NMS is
psychiatric disorder and three prior hospitalizations for shown in a report of the calls to the NMS Informa-
agitation, mood swings, and psychosis was admitted to tion Service, an expert Internet referral service
hospital following the addition of a single 25 mg dose of (136). Of 29 calls requesting information about
nortriptyline to a regimen of l-dopa and trazodone. The NMS, the symptoms of 22 (79%) met criteria for
l-dopa was prescribed for signs of parkinsonism after TSS and 25 (89%) met criteria for NMS (137). The
many years of neuroleptic treatment. Trazodone was authors concluded that NMS and TSS are syn-
prescribed as an antidepressant of the serotonin antago- drome variants of malignant catatonia, a conclu-
nist and reuptake inhibitor class. sion supported by their rapid relief by
benzodiazepines and by ECT.
Within 5 h she became fearful, tremulous, sweating, and
incontinent of urine. The systemic signs persisted with
waxing and waning of motor restlessness, rigidity, and The pediatric catatonias
tremors. Four days later, she became confused and
For many years, catatonia was not considered to
reported episodes of explosive diarrhea.
exist in children and adolescents. Now we know
At the psychiatric emergency room, she was mute, rigid, dierently. It was hidden under the global diagno-
sweating, and tremulous with rapid heart rate and ele- sis of schizophrenia and the labels of stupor and
vated blood pressure. A single 1 mg intravenous dose of catalepsy (muscular rigidity and xity of posture
lorazepam was administered, without benet. On admis- regardless of external stimuli and decreased sensi-
sion to the psychiatric unit, she was afebrile, her posture tivity to pain). The authoritative incorporation of
was rigid and she did not obey simple commands. In the catatonia as schizophrenia by Emil Kraepelin and
absence of exposure to neuroleptics a toxic serotonin Eugen Bleuler, as we have seen, blocked consider-
syndrome (TSS) was diagnosed. Treatment with loraze- ation of catatonia in other conditions. A detailed
pam did not oer relief. ECT was begun and a day after history by Edward Shorter of the vagaries of
the rst treatment, motor rigidity, mutism, and negativ- catatonia in children relates the various ways that
ism became more prominent, and additional lorazepam catatonia has been regarded by psychiatrists.
was prescribed. The afternoon of her second treatment, Often, catatonia has been hidden under diagnoses
motor rigidity and mutism were gone, she was more of encephalitis lethargica, hysteria, hebephrenia,
interactive and responsive, denied hallucinations, and schizophrenia, and, lately, autism (138).

23
Max Fink

Few childhood disorders were recognized in pain. All are signs of catatonia. Two formal sur-
early psychiatric classications and are usually veys nd catatonia in 1217% of adolescents
dened as schizophrenia, mental retardation, and otherwise labeled with autism (143, 144). Although
tics and mutisms. In recent classications, conduct we lack systematic studies identifying and treating
and attention decit disorders have been added. In catatonia in autism, case reports now dot the liter-
DSM-IV, what had been known as mutism ature indicating instances of relief for even the
became elective mutism and then pervasive refu- most severely ill (139, 145). Children who are
sal syndrome; echophenomena and stereotypy bound to their home, unable to participate in
were relabeled Tourettes syndrome or obsessive regular schooling, are relieved of their repetitive
compulsive disorder (OCD); posturing and nega- behaviors, especially those who are injurious to self
tivism were associated with autism or autism and aggressive to their families. The behaviors are
spectrum disorders; and tantrums and excited self re-enforcing and result in bodily harm with
states labeled emotional behavior disorders or bone fractures or the loss of eyes, teeth, and n-
conduct disorders. No one of these behaviors is gers. Here is one of many case reports from the
well dened, and for none has either an eective Stony Brook University Hospital:
treatment or an understanding of the underlying
A 14-year-old intellectually disadvantaged boy was
pathophysiology been developed. When catatonia
admitted for persistent head-banging, which required
has been recognized, however, it has been possible
him to wear a protective helmet and be restrained most
to relieve the symptoms with treatments for catato-
of the day. Donalds mental age was measured as
nia (139). Here we discuss the experience with chil-
4.3 years. He communicated by guttural sounds. The
dren suering autism spectrum disorders and
unusual behaviors began at age 10 and persisted despite
mental retardation in whom the connection to
various medications and both positive and negative rein-
catatonia is well dened; in the next chapter, we
forcement treatments. He wore large gloves in addition
discuss other pediatric syndromes that meet crite-
to the helmet, and was sedated to control his high-
ria for catatonia and warrant further study.
pitched wailing and prolonged screaming. After 4 years
of failed treatment, Donald was evaluated for ECT. On
Autism spectrum disorders admission to the hospital, examination found no bodily
disorders to preclude further treatment. Consent was
Autism, a disorder of socialization and speech with obtained from his legal guardian.
an onset in early childhood, was brought to profes-
sional attention by Leo Kanner in 1943 (140) and After the sixth of twice-weekly ECT, screaming, scratch-
by Hans Asperger in 1944 (141). They described ing, and head-banging were reduced. By the 10th treat-
children with delayed maturation, poor socializa- ment, Donald no longer needed the helmet, gloves, or
tion, and a plethora of repetitive behaviors. Much restraints. Treatments were reduced to once weekly, and
debate ensued as to the distinction between child- after 16 treatments he was returned to his residence. For
hood autism, mental retardation, and childhood another 2 months, treatments were given once every
schizophrenia. Mainstreaming the education of 2 weeks and then stopped, because the psychiatrists were
these children, increased information on the Inter- satised with his progress. Anticonvulsants were pre-
net, and the rise of active parent organizations scribed and he participated in group activities without
have established autism as a diagnosis for which restraints or life-threatening behaviors (146).
special support is provided by many American Other reports cite the successful relief of catato-
school services. Children are oered aides in nia in a child with Down syndrome (147), in
classrooms and psychological assistance and autistic twins (148), and in malignant catatonia in
speech therapy at home. In communities with ben- an adolescent with a cerebellar malformation from
ets for the children and their families, the number birth (149). Another report cites two adolescent
of children labeled autistic has risen rapidly with patients with tics successfully treated with ECT
recent estimates running as high as one of every (150). The successful relief of self-inicted injuries
eight children (142). Severity of illness ranges from in youth with autism spectrum disorders is increas-
children who can be maintained at home and in ingly recognized (151154). While some relief
community schools to those who require highly occurs with high doses of lorazepam, ECT is
specialized institutional care. more eective (155, 156). The immediate
Autism is marked by mutism, echolalia, echopr- changes are dramatic, and the benets become
axia, odd hand postures, freezing of ongoing persistent when treatments are continued over
movements, rigidities, forced vocalizations, stereo- many weeks.
typy, self-injurious behaviors, and insensitivity to

24
Rediscovering catatonia

Another example of successful treatment is 30 mg daily (a neuroleptic) was prescribed leading to


related by Dr. Lee Wachtel, the medical director of further deterioration. Lorazepam 3 mg/day yielded mild
the Neurobehavioral Unit at the Kennedy Krieger improvement. Self-injury escalated to the frank loss of
Institute in Baltimore, Maryland. oral tissue and tongue bleeding.

Robert was the product of a full-term pregnancy and Robert was admitted to hospital in January, 2012 with a
spontaneous vaginal delivery. Developmental mile- diagnosis of delirious mania/agitated catatonia, and
stones were within normal limits for the rst commenced ECT 2 days after admission. ECT thrice
15 months of life, when he stopped interacting with weekly was pursued with standard methohexital
others and lost acquired speech. A pervasive develop- sedation, succinylcholine muscle relaxation, and bilat-
mental disorder was diagnosed and he began intensive eral electrode placement. Within 2 weeks self-injury,
early intervention services at a private clinic, lived at aggression and agitation ceased, (with the exception of a
home, and attended a specialized school for the next few episodes of slapping when redirected from mastur-
15 years. He was estimated to have attained upper ele- bation) but other motor, vocal and behavioral symptoms
mentary school skills; at his best, he was learning the of catatonia persisted. Tissue wounds healed.
names of European capitals and completing multi-digit
ECT frequency was increased to 45 times weekly for
multiplication.
the next 2 weeks, which led to overall symptom resolu-
At age 14, Robert evinced grunting and other abrupt tion. Lithium monotherapy was begun with ECT thrice
vocalizations, progressing to repetitive rocking, lunging, weekly and Robert showed consistent improvement
head shaking and nger apping, multiple stereotypies despite waxing and waning of symptoms.
and hair-combing echopraxia. The symptoms waxed and
On evaluation after ECT #26, Robert independently
waned for the next 3 years. He continued to attend
walked into the room at a normal pace, waved to the
school.
doctor, greeted her by name, and then began to read out
Three years and 5 months after symptom onset, Robert loud from a book. He answered multiple questions with
suddenly stopped eating and required manual feeding by minimal hesitation in the same sing-song cadence he had
others. Food spooned onto his tongue would remain for demonstrated from an early age. He interacted appropri-
several minutes until extensive prompting evoked swal- ately with his mother and caregiver and independently
lowing. He lost about 50 lbs in weight in the next few selected multiple songs and videos on his IPad, deftly
months. Rigid posturing occurred intermittently using the touch screen. The only abnormal physical
throughout the day, particularly evident at mealtimes movements observed were brief episodes of nger ap-
when utensils would remain suspended in his hand in the ping. His father said that ECT had released Robert
air, or he would clamp down so forcefully that sta from the shackles of Hell.
members feared breaking his jaw to pry out the utensils.
At the time of writing Robert had received 44 treatments
Periods of stupor, slow gait and shuing steps became
and was in the maintenance ECT phase with gradual
frequent. Aggression and self-injury ensued. Unpro-
tapering of the frequency. Medications included lithium
voked physical attacks of his family and sta members
600 mg BID with a serum level of 0.8 meq/l, lorazepam
necessitated physical holds frequently using canvas belts
2 mg TID, and memantine 5 mg BID. Robert was still
for limb immobilization. Self-injury included frenetic
unable to transition to home for lack of appropriate
twisting movements of ngers deep into the nares, self-
ECT services in his home area.
biting of hands, arms and tongue with wound formation.
He was no longer able to attend school or to leave his An interesting feature of this boys history and
home. that of numerous other similar reports is the
gradual onset of catatonia in childhood and then
For lack of local inpatient services, Robert was only
a rapid escalation during adolescence. Because
hospitalized 7 months after these symptoms began.
ECT carries a heavy burden of stigma, however,
Prior to hospitalization his days were spent sleeping
families are extremely reluctant to consider ECT,
from 4 AM to 10 AM, then lying or crouching unre-
despite the persistence of abnormal behaviors,
sponsive in his bed requiring full care in toileting,
poor maturation, and poor socialization. Parents
bathing and feeding, until early evening when agita-
who agree to treatment insist that only a
tion, self-injury, aggressive episodes, leaping, lunging,
minimum number of treatments be applied, with-
jumping, screeching and nonsensical vocalizations
drawing consent at the earliest favorable change.
would commence.
We now know that successful relief requires ECT
Local psychiatric and neurological assessments oered continuation treatments. With eective care,
no diagnosis or treatment. Extensive laboratory and adolescents are able to remain at home as a
imaging studies were within normal limits. Olanzapine participating family member, manage their daily

25
Max Fink

self-care, and participate in schooling and social The catatonic signs were accompanied by screaming and
activities. aggressive outbursts; she often required physical
restraint. She appeared depressed, refused to eat, and
was incontinent. Her speech was not intelligible to us
Mental retardation but was interpretable by her mother.
Before the recent enthusiasm for the autism diag- High doses of lorazepam diminished the catatonia signs,
nosis, patients with the same symptoms and signs but depressed mood, excitement, and incontinence con-
of delayed speech and learning, poor socialization, tinued as before. When ECT was recommended, both
and peculiar movements were labeled as suering parents signed the consent form. After two treatments,
from mental retardation. Labels are sensitive to Claudias mood and sleep improved, her appetite
social acceptance and undergo change when they returned, she was tractable, and she responded to ques-
are deemed oensive. Intellectual disability, tions and directions. After the third ECT, catatonia was
intellectually challenged, mental handicap, and gone and her behavior was easily manageable. She was
learning disability are alternative terms. For a discharged to her parents home for continuation treat-
time, the scores under 70 on intelligence quotient ment with lorazepam and weekly ECT. She took part in
tests were a measure of severity. The labels of aut- a day treatment programme for patients with mental
ism or autism spectrum disorders include many of handicaps and required no additional medications. After
these subjects as well as a cluster of patients with 5 months, ECT treatments were discontinued and Clau-
high intellectual capacities that are seen as having dia remained in the community with lorazepam as her
Asperger syndrome. principal medication. (146)
But like normal persons, they develop mood dis-
orders, acute excitements, deliria, and catatonia. A recent literature review in patients with intel-
They were often prescribed neuroleptic drugs to lectual disability treated with ECT found 72
control aggressive behaviors, and from time to reports with positive outcomes in 79% of the cases
time, the neuroleptic malignant syndrome was (158). The patients include disabilities from mild to
induced. But even when these signs would have profound. Transient complications were cited in
recommended treatment with ECT, the recommen- 13% of the reports; these included memory impair-
dation was precluded by the belief that these chil- ment, drowsiness, delirium, and spontaneous sei-
dren were already suering brain damage and that zures. Relapse occurred in 32% despite continuing
ECT would worsen the condition. From time to treatment with medications. The reviewers discuss
time, clinicians would treat such patients with the under-use of ECT because of diagnostic over-
ECT, often successfully and without a worsening shadowing the presence of the label of intellec-
of the subjects communication or language skills. tual disability prejudiced caretakers against
In 1999, we reviewed chart records for the diagno- consideration of ECT as riskful and brain damag-
sis of mental retardation of the patients admitted ing. A second hurdle was that of informed consent
to Stony Brook University Hospital. We had trea- as the disability calls into play legal issues of guard-
ted ve patients from ages 1864: two with depres- ianship, surrogate consent, and judicial consent.
sive mood, two with NMS, and one with a toxic At this writing, delirious mania (malignant
response to hallucinogens. All responded well catatonia) in all its variations, toxic serotonin syn-
to ECT without worsening of their intellectual drome, and many forms of pediatric catatonia are
abilities (157). widely considered among the diagnosable and
The treatment for catatonia in a woman with a treatable catatonia disorders. These recent recogni-
life history of mental retardation was also safe and tions are positive outcomes of our appreciation
successful. that catatonia is readily found outside schizophre-
nia and that we now are able to verify the diagno-
On admission Claudia appeared her stated age of 23, not sis and have eective treatments to validate the
speaking for long periods. She would assume postures diagnosis. The next challenge is to consider other
imitating others or after the examiner moved her limbs. disorders as possible forms of catatonia.

26
Rediscovering catatonia

Chapter 5: are these forms of catatonia?


That a new disease should be manifested at this (repeating words), and coprolalia (repeating
advanced day, in the calendar of medical history, is not scatological words), a cluster of behaviors that is
without many precedents. That such should occur in the now known as the Gilles de la Tourettes syndrome
great family of maladies involving the nervous system, (GTS). (159). It was distinguished around the same
would be the least improbable, when we reect how less time that Karl Kahlbaum described catatonia and
than all others these have been studied, comprehended was one of many attempts to identify psycho-
and treated, until within a comparatively recent period. pathological conditions with predictable outcomes.
Luther V. Bell, 1849 (68) GTS has a childhood age-of-onset and persists
to adulthood. Repetitive vocalizations, antisocial
The recognition that catatonia is the treatable
and inappropriate sexual acts, self-injury, and
core of the neuroleptic malignant syndrome
sleep disturbances are the principal features. In a
released catatonia from its steel-bound tie to
systematic examination of 55 adults with GTS,
schizophrenia and encouraged questions whether
87% exhibited echophenomena, perseveration,
other psychiatric syndromes were better considered
staring, grimacing, posturing, mannerisms, and
catatonia in disguise and be brought under the
stereotypy (160). The signs of autism and GTS
same treatment umbrella. The life-threatening syn-
overlap, and in many cases, the choice of diagnosis
dromes of delirious mania and the toxic serotonin
is in the eyes of the beholder and not in any
syndrome were the rst to be appreciated as forms
distinguishing criteria (161).
of catatonia. Next, the self-injurious behaviors that
No fully eective treatment is known. For mild
are commonly described in children and adoles-
tics, the behavior is tolerated and treatment is con-
cents with autism spectrum disorders were found
sidered unnecessary. For more severe forms, both
to be part of a catatonia syndrome that responded
negative and positive behavior re-enforcement ritu-
to ECT. But that was just the beginning.
als are prescribed, but success is limited (162, 163).
Within the DSM-IV classication of psychiatric
Reductions in the number of verbalizations are
disorders are many entities that readily meet cata-
reported with treatment with the neuroleptics
tonia criteria selective (or elective) mutism; Gilles
haloperidol and pimozide (164). Recent industry-
de la Tourettes syndrome, vocal, and transient tic
sponsored studies of olanzapine nd decreases in
disorders; the neuroleptic-induced Parkinson syn-
symptoms with greater reduction of tics compared
drome; stereotypic movement disorders; and
with placebo treatment (165). For the more
obsessivecompulsive disorder. For none are
severely debilitating forms of the illness, experi-
dened biological markers nor eective treatments
mental deep brain stimulation has been tried with
known. These mutisms and stereotypical behaviors
teasingly favorable reports. The studies lack
bear sucient characteristics of catatonia to war-
proper controls, however, so are considered
rant re-examination as catatonia variants.
exploratory (166).
In the past decade, a new diagnostic entity of
From time to time, tics have been so severe as to
anti-NMDAR encephalitis has been enthusiasti-
warrant trials with ECT. A PubMed review found
cally promoted as a unique neuropsychiatric disor-
10 reports of modest relief. In an 18-year-old man
der. Although catatonia is recognized in 70% of
with depressed mood, facial tics, grimacing, repeti-
the subjects so diagnosed, the connection to cata-
tive hand-washing, and self-injurious behavior,
tonia is not credited in the disorders evaluation
ECT resolved both the mood and the tic disorders,
nor in its treatment.
a benet that was sustained over the next year.
And, among neurologic syndromes, akinetic
When treatments stopped, he relapsed, but when
mutism, the prolonged state of stupor and paraly-
ECT was re-instated, the tics did not return and he
sis with apparent vigilance, has many characteris-
continued symptom-free while attending college.
tics of catatonia, such that exploring its relation to
In the same review, the tics and self-injurious
catatonia would be useful.
behavior of a 19-year-old autistic man were
reduced but not fully relieved with ECT (150).
Gilles de la Tourettes syndrome (GTS) Another example comes from my own experi-
ence. A 64-year-old widowed woman with recur-
In 1885, the French neuropsychiatrist Georges rent episodes of depressive illness relapsed with
Gilles de la Tourette described nine patients with repetitive scatological expressions in response to
multiple tics (sudden, repetitive, non-rhythmic any query. The tics disappeared quickly with ECT
motor movements or vocalizations), echolalia

27
Max Fink

(167). Individual case reports of the relief of GTS describes children and adolescents diagnosed with
with ECT are frequent (168172). childhood disintegrative disorder, Kleine-Levin
For all the similarity of GTS and catatonia syndrome (a disorder of excessive eating and sleep-
many descriptions of GTS meet the criteria for ing), PraderWilli syndrome (a disorder of low
catatonia reports of treatment with benzodiaze- muscular tone, small stature, and incomplete sex-
pines are surprisingly sparse. When the benzodiaz- ual development), tic disorder, and autoimmune
epine clonazepam was introduced to clinical trials, encephalitis as examples of catatonia (182). In each
reductions in symptoms were reported (173175). case, the illness resolved safely once it was recog-
The lack of interest in such experimentation is par- nized and treated with benzodiazepines and ECT.
ticularly surprising considering the persistence of The frequency of catatonia in such a wide range of
GTS and the failure of recommended treatments. child and adolescent disorders indicates that pedi-
Given the safety and ecacy of benzodiazepines, atric catatonias are not rare, particularly in epony-
further study of GTS as a form of catatonia mous disorders that are a principal part of
certainly is warranted. pediatric medicine. Dhossche also cites depriva-
tion, abuse, and trauma in the etiology of pediatric
catatonia (183) and presents examples of catatonia
Elective mutism and pervasive refusal syndrome among asylum-seeking children in Sweden (184,
In 1991, four young British girls between 9 and 14 185).
years of age were described as suering a poten- As with other pediatric syndromes that are
tially life-threatening condition manifested by pro- poorly dened and dicult to treat, it behooves
found and pervasive refusal to eat, drink, walk, clinicians responsible for patients with persistent
talk, or care of themselves in any way over a period and severe forms of mutism to consider the
of several months. The children required nasogas- connection to catatonia. But too few do so.
tric tube-feeding and spent such prolonged periods
in bed that they were occasionally requiring
Anti-N-methyl-D-aspartate receptor encephalitis
manipulations of the joints under general anesthetic
to prevent contractures (176). They eventually Limbic encephalitis, an acute neurological
recovered after extended periods of hospital care. disorder, was rst described in the 1960s as a
The cases were labeled pervasive refusal syn- paraneoplastic condition one resulting from
drome (177). Since then <30 additional cases are tissue changes induced by tumors that stimulate a
cited in the literature, with a 3 : 1 ratio of girls to form of self-poisoning. It is considered an autoim-
boys (178180). None of the reported cases had a mune disorder, a condition in which the immune
fatal outcome, although each required prolonged system mistakenly attacks and destroys healthy
hospital care. In the few patients who have been body tissue. More than 80 dierent autoimmune
the subject of long-term follow-up, the recovery disorders are described in the medical literature.
has been complete. The pathophysiology is poorly understood, and
In discussing the four cases, Brian Lask and his the treatments are empiric and of limited ecacy.
colleagues specically consider the diagnoses of The presumed connection to tumors sends
anorexia nervosa, elective mutism, post-traumatic patients to whole-body searches, but the syn-
stress disorder, and stupor, but they do not list cat- drome is also accepted as a diagnosis without
atonia (176). Yet, the children exhibit mutism, neg- nding a tumor. In normal life, various tissue
ativism, posturing, and rigidity and require receptors, one of which is the n-methyl-D-
parenteral feeding for survival. These are cardinal aspartate receptor, are part of the bodys natural
signs of catatonia, but no catatonia treatments had physiology. In the words of enthusiasts, an anti-
apparently been tried. NMDAR encephalitis reaction is present in
Separately, in one patient in Ireland, a diagnosis patients who develop:
of catatonia was formally considered in an
an acute multistage illness that progresses from
11-year-old girl. Video images of her behavior
psychosis, memory decits, seizures and language disin-
clearly met the criteria for catatonia, but her
tegration into a state of unresponsiveness with catatonic
parents refused a benzodiazepine treatment trial.
features often associated with abnormal movements and
She recovered after 18 months of nursing care in
autonomic and breathing instability.
hospital (181).
Dirk Dhossche, a child psychiatrist in Jackson, About 70% of patients have prodromal [early onset]
Mississippi, has looked further into similar cases, symptoms consisting of headache, fever, nausea, vomit-
applying the present criteria for catatonia. He ing, diarrhea or upper respiratory symptoms. [The

28
Rediscovering catatonia

psychiatric syndrome] follows within a few days or a The diagnosis of anti-NMDAR encephalitis
few weeks with a rapid disintegration of language, from depends on a specic positive serum or cerebrospi-
reduction of verbal output and echolalia [and echopraxia] nal uid antibody test (186). When the diagnosis is
to frank mutism. considered, much interest is paid to the coexistence
The initial phase of the illness is usually followed by
of encapsulated teratoma tumors. The recom-
decreased responsiveness that can alternate between
mended treatments are tumor resection and non-
periods of agitation and catatonia [with] autonomic
specic immunotherapy (corticosteroids, intrave-
movements and autonomic instability (186).
nous immunoglobulin, or plasma exchange) or
immunotherapy medications (rituximab or cyclo-
The descriptions readily meet the criteria for cat- phosphamide).
atonia disturbances of behavior marked by A report of 100 cases of this syndrome exqui-
unresponsiveness with catatonic features, a febrile sitely describes the signs of catatonia in the
course, followed by reduction in verbal output patients, but neither catatonia nor its treatments
and echolalia to frank mutism, followed by peri- are discussed (190). Case reports now dot the
ods of agitation and catatonia, in which patients literature with most patients being female and with
resist eye opening but show no response to pain- resolution after resection of ovarian teratomas
ful stimuli. Although experience with the when found. But the syndrome is also reported in
syndrome is limited, the descriptions clearly t males and among many patients without evidence
within the present conception of catatonia as a of tumors (191).
systemic syndrome unrelated to psychosis and The heightened enthusiasm for this diagnosis is
schizophrenia. reected in an Editorial in the British Journal of
A 2008 case report in the New England Journal Psychiatry in April 2012 calling for laboratory
of Medicine describes a 26-year-old woman admit- tests for anti-NMDAR encephalitis in all individ-
ted for headache, behavioral changes, abnormal uals with a rst presentation of psychosis, or peo-
movements, and mutism of 7 weeks duration. ple with psychosis and features of autonomic
After extensive laboratory test examinations, an disturbance, movement disorder, disorientation,
anti-NMDAR encephalitis was diagnosed. seizures, hyponatremia, or rapid deterioration
Throughout her illness, signs of catatonia were with the possibility of antibody-mediated encepha-
recorded but neither recognized as such nor trea- litis in mind. The recommendation continues:
ted. An ovarian teratoma was found, surgically This assessment should include, as a minimum, a
removed under anesthesia, and the syndrome neurological and cognitive examination and early
resolved within a day. Was the removal of the serum testing for antibodies against the NMDA
tumor or the anesthesia the therapeutic agent? receptor and voltage-gated potassium channel. All
The rapidity of the resolution and her course patients testing positive for these serum antibodies
favor the relief of catatonia from the sedation should be referred to neurological centers with
(187). expertise in managing these cases (192).
Another report, this one in the American Journal Support for the suggestion followed quickly with
of Psychiatry, described a 16-year-old boy with the unproven assertion that NMDA receptor anti-
protracted stupor, psychomotor retardation, mut- body encephalitis is a condition that responds to
ism, posturing, stereotypical movement, refusal to immunotherapy (193). Such a recommendation is
eat and drink, and episodic agitation (188). A posi- inconsistent with good medical practice, however,
tive blood test supported an anti-NMDAR diag- as the diagnosis directs attention to a treatment
nosis. No clear medical or toxic cause was found, that has not been proven eective.
nor were any of the prescribed psychotropic medi- The popular enthusiasm for this diagnosis is
cations or immune therapies benecial. The pres- illustrated by the rapidly increasing case report lit-
ence of catatonia was not recognized; as a erature. The initial references to anti-NMDAR
consequence, no consideration was given to its encephalitis cited in PubMed are dated 2007. By
treatments. Instead, after treatment with haloperi- September 2011, 68 reports are listed with 2 coau-
dol and other antipsychotic agents, the symptoms thored by Josef Dalmau; by February 2012, the
worsened. The syndrome eventually abated with number has increased to 126 with 32 by Dalmau;
nursing care with the patient slowly returning to and by October 24, 2012 the number of reports had
baseline function 7 months after the onset of increased to 166 with 40 co-authored by Dalmau.
symptoms. The failure to recognize catatonia or to Considering these cases as examples of catatonia
consider its treatment was criticized as a disservice is veried by the reported ecacy of ECT, cited in
to the patient as well as unnecessary trumpeting of reports in ve patients (194197). One example is
a poor clinical lesson (189). described (198):

29
Max Fink

An 11-year-old girl became uncooperative, confused, taken, using a standard ECT protocol. After 2 weeks,
and agitated with slurred speech and hallucinations the checking was reduced and he was able to leave his
after an upper respiratory tract infection. She was room, feed and care for himself. After 3 weeks, he left
admitted to the hospital with a diagnosis of acute psy- the ward and went to a meal at a restaurant accompa-
chosis and was prescribed a low dose of the atypical nied by his mother. After 5 weeks he was discharged
neuroleptic risperidone with little eect. After adminis- with a prescription for the selective serotonin reuptake
tration of chlorpromazine for agitation, she became inhibitor clomipramine. He was able to care for himself
febrile, with rapid pulse rates and body rigidity. Malig- for 2 months and then the checking returned. A second
nant catatonia was diagnosed, and a course of ECT ECT trial was less eective.
was begun. After eight treatments she resumed normal
An additional seven patients I treated showed
functioning. Ongoing abdominal complaints prompted
short-term benets. Because we had not developed
ultrasound and abdominal CT that revealed an ovarian
a protocol for out-patient ECT, we did not
tumor. Surgical removal of the ovarian teratoma may
examine the merit of continuation ECT in these
have contributed to the full recovery of the patient.
cases.
Absent controlled studies, however, proof for this con-
Considering the inecacy of present treatments
clusion is lacking as the patient had already achieved
and the lack of understanding of the pathophysiol-
near remission after an intensive 2-week course of
ogy of obsessivecompulsive disorder, greater
ECT.
attention to the role of catatonia and its treatments
Clinical accounts of patients suering from is surely warranted.
autoimmune limbic encephalitis commonly
describe catatonia, but the syndrome is seldom so
labeled nor are the eective treatments attempted. Akinetic mutism
As catatonia is present in a majority of patients Neurologic texts describe patients who lie inert
and we lack evidence of a specic treatable patho- and mute, appear alert, and regarding the clinician
physiology, it is prudent to consider catatonia in by only moving their eyes. They may follow the
the dierential diagnosis and oer its tests and movement of objects or be diverted by sound. Nei-
treatments. ther voluntary movements nor restlessness or nega-
tivism is seen. A painful stimulus may produce
reex withdrawal or no movement. Patients have
Obsessivecompulsive disorders
to be fed and are incontinent of urine and feces
Obsessivecompulsive disorder is characterized by (209). They appear lifeless and may be mistaken
repetitive thoughts and persistent motor acts that for dead (124). Initially labeled akinetic mutism,
interfere with normal life. The behaviors are simi- this condition has since been described under such
lar to those of GTS except that the expressions are names as coma vigil, apallic syndrome, sti-
not scatological and onset occurs later in life. It is man syndrome, locked-in syndrome, encephale
a persistent disorder for which no eective treat- isole, and stupor of unknown etiology. The terms
ment is known. Suggested treatments, which vary vegetative state and minimally conscious state
with the severity of the symptoms, ll the full range are now used frequently. The symptoms dier little
of psychotherapy, negative and positive behavioral from those of the more severe forms of inhibited
conditioning, medications, deep brain stimulation, catatonia. Whether the subject is seen as a form of
and surgical ablation of brain regions. Historically, akinetic mutism on a neurologic medical service or
OCD has been interpreted as an anxiety disorder, as a form of catatonia on a psychiatric service is in
and the syndrome is classied in the DSM under the eyes of the beholder and not in any specic
this umbrella. But the treatments for anxiety disor- characteristic behavior (210).
ders are not eective in OCD, which throws the Akinetic mutism was described in 1941 in a
association into question. 14-year-old girl whose illness had lasted for
Patients diagnosed with OCD exhibit signs of 8 years. She experienced headaches localized over
catatonia (199, 200), and when these patients have the left eye, disturbances in vision, and vomiting.
been treated with clonazepam (201203) and with A cyst had distended the third ventricle of the
ECT (204208,) benet has been reported. My per- brain, and relief followed the removal of its uid;
sonal experience is similar. permanent relief followed its excision (211). Simi-
lar descriptions dot the medical literature in
Faced with a severely incapacitated 24-year-old man
patients with tumors and blood vessel abnormali-
with inability to leave his room, feed or toilet himself
ties in and around the brains third ventricle and
because of checking actions, a trial of ECT was under-

30
Rediscovering catatonia

the basilar artery (212218). The stuporous state is diagnosis of record of stupor of unknown etiology.
thought to result from the direct structural involve- Plans for transfer to a long stay at a neurologic center
ment of the base of the brain (209). States of were underway.
stupor with the same neurologic signs are also A psychiatric consultant suggested an intravenous lo-
found in the acute toxic states associated with razepam test to rule out catatonia. Within a few minutes
hallucinogenic and other toxic drugs and in of injection she opened her eyes, verbalized hoarsely,
chronic infections (219221). and then moved her hands to pick up an animal gure
Patients in chronic stupors or catatonia-like and to smile. Over the next few days increasing dosages
states form a portion of every chronic neurological of lorazepam resulted in some speech, and aided sitting,
medical service. When rst seen, each patient is standing, and walking. With family consent, ECT was
thoroughly examined for brain stem lesions, and begun and after four treatments the patient was fully
when found, may be relieved medically or surgi- responsive, able to feed and care for herself, talking
cally. But in many cases, lesions are not found, and responsively. Within 2 weeks she was discharged to her
it is in such instances that the question arises home as recovered.
whether the signs meet the criteria for catatonia.
One description of akinetic mutism describes the Early in the literature of this syndrome, relief
state as similar to that of catatonic schizophrenia. was described by inducing phenobarbital coma
In those catatonic schizophrenic states that simu- (222). Zolpidem brought back to life the motor
late akinetic mutism, there is awareness, as these movements and speech of a 48-year-old woman
patients do have memory of the events happening who developed akinetic mutism after a suicide
during the catatonic state (213). Another writer, attempt by hanging (223). Failure to properly iden-
discussing the range of etiologies, some with identi- tify the locked-in syndrome as a form of catatonia
ed brain lesions and some without, noted: Varia- in one instance resulted in a subject remaining in a
tions occur: the patient may be almost totally paralyzed state for 16 years (224). The tragedy of
immobile and mute, or may have only a suggestion motor paralysis in a conscious state is not readily
of hampering of speech and movement (208). In describable (225).
each description, the signs of catatonia are among Descriptions of catatonia in the historical litera-
the physical signs noted in the examination, but ture nd many subjects at rst believed to be dead
because the accompanying behaviors, thoughts, or and their sudden revival fortuitously preventing
course are not those of schizophrenia, catatonia is their burial (124). When a brain stem lesion is iden-
not identied. The author of a detailed 1962 tied and is successfully treated surgically or medi-
review of akinetic mutism and its symptoms from cally, the outcome is determined by the site and
New York State Psychiatric Institute writes that pathology of the lesion and the ecacy of the
these simulate, in many respects, catatonic treatments. But patients without an identiable
schizophrenia (214). lesion who are residing on neurologic and medical
The following personal experience reects the hospital services in chronic care under any of the
overlap between catatonia and one of the descrip- cluster of labels designating akinetic mutism war-
tive terms for akinetic mutism, that of a stupor of rant examination for catatonia.
unknown etiology. We have explored the merits of separating cata-
tonia from schizophrenia, seen its many clinical
A 22-year-old woman, lying mute in stupor, not moving
forms, and questioned whether other illnesses can
to sound or pinprick is seen in a bed lled with furry ani-
be included under the catatonia umbrella. It is now
mal gures on the Neurology Service of an academic
useful to ask how the recognition of catatonia
medical center. Her illness began a month earlier with
faltered and look at the recent developments that
acute malaise and somnolence followed by headaches
identify catatonia as an independent syndrome in
and abnormal movements, and then a seizure. Medical
the classication of psychiatric diseases. The pro-
and extensive neurologic examinations identied no
cess, while not complete, is well underway.
cause, treatments for epilepsy were prescribed, with the

31
Max Fink

Chapter 6: classifying catatonia


The modern system of elevating every minor group, When Eugen Bleuler relabeled Kraepelins image
however triing the characters by which it is distin- of dementia praecox as schizophrenia, he retained
guished, to the rank of genus, evinces, we think, a want catatonia as its marker.
of appreciation of the true value of classication. And there catatonia has lain, a marker for
Sir Joseph Dalton Hooker, 1855 (226) schizophrenia for a 100 years, in repeated editions
of the ocial diagnostic classications until the lat-
How is catatonia recognized in psychiatric texts
est revision, the DSM-5, oers some relief.
today? For a syndrome to be recognized, it must
The rst Diagnostic and Statistical Manual of
be identied in the classications by a name, a
the American Psychiatric Association, published
description, and a coding number. Only then is it
in 1952, designated catatonia simply as schizo-
established as a disorder of professional interest.
phrenic reactions, catatonic type. These reac-
Two ocial classications, the International Classi-
tions are characterized by conspicuous motor
cation of Diseases (ICD) by the World Health
behavior, exhibiting either generalized inhibition
Organisation (227) and the Diagnostic Statistical
(stupor, mutism, negativism, and waxy exibil-
Manual (DSM) by the American Psychiatric Asso-
ity) or excessive motor activity and excitement.
ciation (228), are mainstays of clinical recognition.
The individual may regress to a state of vegeta-
The rst classications agreed on disease names
tion (228).
and characteristics for statistical purposes of
The revision in 1968 (DSM-II) described two
admissions, discharges, and deaths, but in the past
types of catatonia, both within the category of
half century, the labels have been increasingly
schizophrenia: It is frequently possible and useful
used for selecting treatment, research studies, and
to distinguish two sub-types of catatonic schizo-
physician and hospital payments. Guidelines for
phrenia. One is marked by excessive and sometime
the prescription of treatments are tied to specic
violent motor activity and excitement and the other
diagnoses. In evaluating the ecacy and safety of
by generalized inhibition manifested by stupor,
treatments, each subject is selected with narrow
mutism, negativism, or waxy exibility. In time,
diagnostic criteria to assure homogeneity of the
some cases deteriorate to a vegetative state (229).
samples. As treatments are better dened, the pay-
The third revision of 1980 mentioned catatonia
ments for physician and hospital care are tied to
under the classication catatonic type of
specic procedures. Durations of hospital care can
schizophrenia dominated by catatonic stupor,
be matched against standard care experiences,
negativism, rigidity, excitement, or posturing
allowing insurers and government agencies to eval-
(230). By the time of this formulation, however,
uate the eciency of treatment procedures and
researchers had recognized many instances of
treatment teams. Indeed, the quality and applica-
catatonia outside schizophrenia, especially among
bility of the classication system is central to the
the aectively ill.
concept of governmental control and insurance for
For half a century, it was also known that cata-
health care.
tonic patients responded well to barbiturates and
When Karl Kahlbaum described catatonia, he
to ECT, while patients identied as schizophrenic
saw many variations in the signs of the syndrome
but without evidence of catatonia did not.
and in the associated disorders. He presented 26
Although members of the DSM-III committees
cases that spanned a variety of illnesses, some that
were acquainted with this literature, they chose to
meet the present criteria for psychosis and mood
ignore it (231).
disorder and some ill with the systemic diseases of
Largely through the writings of intrepid catato-
tuberculosis and syphilis. As we have described,
nia scholars, the revision of 1994 (DSM-IV) added
two decades later, Emil Kraepelin recognized cata-
catatonia secondary to a medical disorder, a sin-
tonia just as Kahlbaum had described it. As Krae-
gular recognition outside schizophrenia (10). For
pelin shued his case record cards seeking
the mood disorders, a catatonia specier was
patterns in the main signs, he saw catatonia in
added to encourage awareness of its possible asso-
many instances, particularly in those who became
ciation with mood disorders. The absence of a cod-
models of dementia praecox. He also perceived
ing number, however, discouraged the use of a
catatonia among his other described diseases, in
specier term in diagnosis or in research studies.
patients with manicdepressive insanity and with
Literature searches now nd few citations to a cat-
paranoia, but his belief that catatonia was a mar-
atonia specier.
ker of dementia praecox dominated his writings.

32
Rediscovering catatonia

These changes did not inuence clinical practice. editor of the Schizophrenia Bulletin, invited
Catatonia was regarded as a sign of schizophrenia, Michael Taylor, Edward Shorter, and me to argue
and whenever catatonia was recognized, neurolep- the case for catatonia as an independent class
tic drugs were routinely prescribed even in patients (235). He solicited comments from others (236,
showing no evidence of disturbed thought or 237), and with these in hand, he published the
speech. responses and an overall critique by three members
Perhaps, the failure to recognize catatonia out- of the Psychosis Work Group (238).
side schizophrenia could be excused as the argu- In our submission, we reiterated the arguments
ment was lost in the growing cacophony of pleas for catatonia as an independent syndrome that we
by dierent constituencies. Each DSM revision had documented in our writings of 2003 and
expanded the numbers of principal diagnoses: throughout these essays (11, 12). Patricia Rose-
DSM-I: 106; DSM-II: 182; DSM-III: 265; DSM- bush and Michael Mazurek of the McMaster Uni-
III-R: 292; and DSM-IV: 297 (232). versity in Canada had often written on the ecacy
Recognition that the neuroleptic malignant of benzodiazepines in malignant catatonia. They
syndrome (NMS) was more eectively relieved reported catatonia in about 10% of acutely ill
by benzodiazepines and by ECT, as we have psychiatric patients, only a minority having met
seen, established catatonia as a well-dened syn- criteria for schizophrenia. Among those with
drome outside schizophrenia. By 2003, Michael aective disorders, who comprise the largest sub-
Taylor and I summarized the accumulated 130- group of patients with catatonia, the catatonia
year experience in Catatonia: A clinicians guide signs typically resolve dramatically and completely
to diagnosis and treatment (11). We argued that with benzodiazepine therapy. Rosebush and
catatonia met the criteria for a distinct clinical Mazurek warned that failure to treat catatonia
syndrome; that the singular linkage to schizo- properly before administering neuroleptic medica-
phrenia was not justied; that assigning catatonia tions increased the risk of inducing a malignant
to a separate DSM class in a home of its own reaction. Establishing catatonia as a separate
was consistent with the known experience; and diagnostic entity, the researchers argued, would
that such a designation would optimize recogni- increase recognition of a treatable neuropsychiatric
tion and treatment (12). syndrome (236).
Gabor Ungvari, Stanley Caro, and Jozsef
Gerevich opined that catatonia was historically
Catatonia in DSM-5
established, universally accepted as integral to
In 2008, the American Psychiatric Association schizophrenia, and the form should be retained.
designated Work Groups of clinicians to examine They cited the failure of benzodiazepines to relieve
the experience with the existing classications catatonia among chronic psychotic patients in
and to recommend changes for a revision to be Hong Kong. (ECT was not tried.) While these
published as DSM-5 in 2013. Catatonia was authors accepted that patients in stuporous catato-
assigned to the members of the Psychosis Work nia did respond well to benzodiazepines and ECT,
Group, still tying catatonia to the Kraepelin and and could be distinguished from those with
Bleuler conceptual tradition, despite the extensive schizophrenia, the failure of patients with
literature nding catatonia more often outside chronic catatonia to do so justied retention of
schizophrenia. As none of the Work Group catatonic schizophrenia as a unique entity in the
members had been associated with the new learn- classication (237)
ing on catatonia, any changes would necessarily Stephan Heckers, Tajiv Tandon, and Juan
be based on the eects of special pleadings from Bustillo, members of the DSM-5 Work Group,
outside groups. responded that a prominent position for catatonia
In 2010, 35 clinicianscholars, each with catato- would pave the way for better recognition of the
nia experience, joined together to urge simplica- syndrome, facilitate eective treatment, and cata-
tion of catatonia as a unique, single diagnostic lyze studies of the neural and genetic mechanisms
class and to discard the connection to schizophre- of catatonia. Although they encouraged better
nia (233). The same-year child psychiatrists who labeling of catatonia in the classication, they did
had clinical experience with catatonia, especially not support the single designation. They see cata-
among those patients with autism spectrum disor- tonia as a marker of other syndromes, of many
ders, published their evidence in support of giving entities in the psychiatric disease classication.
catatonia a classicatory home of its own (234). They also noted that a single designation for cata-
Responding to these submissions, William Car- tonia would be beyond the limits of the charge
penter, the Psychosis Work Group chairman and given the Work Group (238).

33
Max Fink

These authors also oered the jarring note that will determine the relationship between these two
If such a consolidation does occur, it should likely diagnoses.
be in the section on Psychotic Disorders because The designation of catatonia speciers for 10
catatonia is a dimension of psychosis (238) primary diagnoses sustains the secondary position
Rosebush and Mazurek expressed the same con- of catatonia in the prior classications. Whether
viction: The majority of patients with catatonia such is necessary is unclear because as we describe
have concurrent psychosis (236). These assertions in these essays, the recognition of catatonia identi-
are beliefs, holdovers that catatonia is integral to es a syndrome for which eective treatments are
schizophrenia for which the primary treatment is known. Adopting a specier model creates treat-
the prescription of neuroleptic drugs. Such associa- ment dilemmas. For example, the present recom-
tion is unfortunate because many forms of mendations sustain a problematic inconsistency
catatonia are not accompanied by the delusions, with a class of Schizophrenia with catatonia speci-
hallucinations, and disorders of thought that er. Is the prescription of a neuroleptic agent (the
characterize schizophrenia. Also, such patients standard prescription for schizophrenia) to precede
respond to the specic treatments of catato- that of the benzodiazepine or are both to be pre-
nia without resort to those used in the relief of scribed simultaneously? We know that catatonia
psychosis. sets the stage for a malignant response when a
patient is treated with neuroleptic drugs. Treat-
ment consistent with the specier label will subject
The proposed recommendations (Spring 2012) patients to unnecessary risks.
Following consideration of various revisions, the For an excited and delirious patient with signs
latest proposal published on the American Psychi- of catatonia, categorized as Bipolar disorder with
atric Associations DSM-5 Development website catatonia specier is the treatment to be the com-
in May 2012 recommended (239): monly prescribed high-potency neuroleptic drug
haloperidol for restraint, thereby risking a toxic
i) Catatonia as a type of schizophrenia is to be response? Or is the eective prescription to be high
deleted. doses of benzodiazepines and ECT, the treatments
ii) The class of Catatonia secondary to a general for catatonia? Or are both drugs to be prescribed
medical condition created in 1994 is to be simultaneously? Similar conicts follow each desig-
retained. nation of a catatonia specier.
iii) A new class of Catatonia Not Elsewhere Clas- Despite these questions, the DSM-5 recommen-
sied is to be created. dation for catatonia as an independent syndrome
iv) A catatonia specier is to be added for 10 pri- not tied to schizophrenia corrects a long-standing
mary diagnoses with the coding of xxx.x5. error that has plagued patient care and clinical
v) A list of catatonia signs is adopted from research for more than a century (235, 240). The
DSM-IV. present recommendations are suciently broad to
These recommendations were reviewed by the allow experience to rene the place of catatonia in
informal group of catatonia scholars in June 2012 clinical care. It may encourage clinicians to appre-
and were endorsed as consistent with the ongoing ciate catatonia in the many syndromes cited in
experience. The elimination of the designation of these pages, to do so early, and to oer the
schizophrenia, catatonic type, was hailed as nec- eective treatments that assure catatonia its unique
essary. The inclusion of a single class of Catato- place in medicine.
nia Not Elsewhere Classied is responsive to the The changes recommended in the DSM-5 are
experience and publications repeatedly expressed impressive and are sucient to signicantly
by the catatonia scholars. It will serve the improve clinical diagnosis. With the known treat-
requests for a single catatonia term and do ments, patient care will surely be improved. The
much to cut the tie between schizophrenia and main issue for these recommended changes will be
catatonia. their acceptance in guidelines for treatment and
The designation of Catatonia Secondary to a forthcoming textbooks. The known experience
Medical Disorder introduced in 1994 is to be nds about 80% of the patients with catatonia
maintained. The distinction between this label and respond to the benzodiazepines, but for the
that of Catatonia NEC is unclear, but the experi- remainder, the consideration of ECT brings out
ence was considered insucient to justify elimina- the broad prejudice against its use, a prejudice that
tion. It is expected that the professions experience often blocks eective treatment.

34
Rediscovering catatonia

Chapter 7: the fear response


Mans mind stretched to a new idea never goes back to but unable to speak a word for several days. The
its original dimension. malady occurs suddenly and he stops as if he were
Oliver Wendell Holmes, Sr., 1859 (241) frozen, his eyes open, xed and motionless (26).
On recovery, patients say that they desired to
How is catatonia related to other medical and
speak but were unable to do so, sensing impending
psychiatric disorders? Catatonia usually has an
doom. Persons in catatonic stupor are frozen,
acute onset and once expressed, persists for days,
petried, and experience extreme fear (27). They
months, even years if not treated. It is as common
give the appearance of intense anxiety (2830).
in children and adolescents as it is in adults. Many
In discussing catatonia in children, the historian
forms are now recognized, some not ordinarily
Edward Shorter cites an eighteenth-century French
destructive but lethal when accompanied by fever
physician who delayed the funeral services of a girl
and systemic distress. The behaviors are seen either
from the lower classes because her color had not
as inhibited and retarded and regarded as stupor
changed at all. And she recovered a few hours
or as excited and aggressive and seen as delirium
later. The girl would already have been buried sev-
or delirious mania. Catatonia is mainly a disorder
eral times, if we had not been familiar with her
of body posture, movement, and speech, although
attacks of hysterical vapors (138).
some patients report intense anxiety and fear.
In a history of malignant catatonia, Shorter
Catatonia has not been traced to a defect in a
describes the progressively severe stages of catato-
single-body organ nor to a specic physiologic
nia, malignant catatonia, neuroleptic malignant
dysfunction. After catatonia is relieved, we see no
syndrome, and delirious mania, the latter some-
residuals; it is as if the blackboard has been erased
times prompted by the horrors of war: As the ter-
with a few smudges left at the corners. It is a
ried civilian population of northern France ed
behavior of the whole organism, more like waking
the advancing German armies in 1940 there was
and sleeping or like a childs crying.
massive fear. Some of the worse aicted found
The phenomenology is distinct from that of
their way to the psychiatric hospital at Auxerre,
other psychiatric diagnoses. Some authors see
where there were 17 cases of delire aigu [delirious
catatonia as a common end-state response to
mania], almost all with a fever and ending fatally
the feeling of imminent doom inherited from
(124).
ancestral encounters with carnivores, an adapta-
In 1982, a perceptive Australian psychiatrist
tion that has changed over the millennia of
described four patients with systemic physical ill-
human development but remains a common fea-
ness who exhibited catatonia in association with
ture of life (15). In this fear hypothesis, catato-
intense fear (242). Although the patients were pre-
nia is a whole-body phenomenon, and support
scribed eective treatments for catatonia, recovery
for the hypothesis is found in the quick relief
depended, he believed, on attention to and treat-
aorded by the anxiolytic qualities of drugs used
ment of the psychological stressors. He regarded
to relieve its symptoms.
catatonia as regression to a primitive expression
When Kahlbaum envisioned catatonia as a syn-
elicited by overwhelming fear.
drome, he described his patients as astonished or
In a systematic inquiry, Georg Northo and his
thunderstruck. The syndrome appeared after
colleagues in Frankfurt, Germany, assessed the
very severe physical or mental stress such as a
experiences 3 weeks after recovery of 24 patients
terrifying experience. He said that the patient
(15 excited, 9 inhibited) who met the criteria of
remains motionless, without speaking, and with a
four or more catatonia signs (28). The patients
rigid masklike facies, the eyes focused at a distance
greatest fears were their inability to control their
devoid of any will to move or to react to any
intense anxiety. They felt threatened and feared
stimulus. He continues: The general impression
dying. They were less concerned about their lack
conveyed is one of profound mental anguish, or
of control of body movement or of self-care.
an immobility induced by severe mental shock
The association of the fear response with catato-
(13). Citing fear as the central theme of the syn-
nia among children and adolescents with autism
drome, Kahlbaum titled his book Die Katatonie,
and with tics has recently been detailed by Dirk
oder das Spannungs-Irresein, translated as Catato-
Dhossche. He reports deprivation, abuse, and
nia, The Tension Insanity.
trauma to be frequent in the expression of catato-
A writer nds subjects who lay like a log of
nia among them (148).
wood, stretched out, able to behold with his eyes

35
Max Fink

After cardiac surgery, immobilization reactions Tonic Immobility


with the characteristics of catatonia have been
encountered. One patient was immobilized and These diverse human experiences are found in ani-
almost like a statue; another was frozen and mals described as tonic immobility and negative
expressionless. She spoke barely audibly in a conditioning. Tonic immobility is the elicitation of
monotone with long pauses and made no sponta- a xed posture, usually rigid, that is elicited by qui-
neous comments. The postoperative reaction has etly holding an animal down, slowly stroking, and
been described as experiencing a catastrophe, the then gradually releasing the hold. The animal
patients resembling the photographed faces of sur- remains immobile with limbs in the unusual pos-
vivors of civil disasters, and the countenances pres- tures that they are placed. The phenomenon is
ent staring and vacant expressions of seeming demonstrated in chickens and other fowl, frogs,
frozen terror. Immobile, apathetic, and completely snakes, guinea pigs, and rabbits (247). A tradition
indierent to their fate, they respond to inquiries of pretending to be dead is described as the behav-
in monosyllables devoid of aect (243). ior of the Virginia opossum playing possum, as
A one-person experimental study demonstrated it is described in childhood play (248).
the relation of catatonia to fear. A 42-year-old A loud noise terminates the imposed posture.
woman with a long history of recurring depressions The duration of the pose is lengthened when pre-
was admitted to hospital for a recurrent episode. ceded by a frightening stimulus such as loud noise,
Treatment with the antidepressant maprotiline was electric shock, adrenalin injections, or simulated
discontinued, and the next day she was mute and predation as when a chicken is held under the head
stuporous. A single oral dose of 2.5 mg lorazepam of a model of a Coopers Hawk for 15 s before
relieved the stupor and the depressed mood, and immobilization. The duration is shortened in a
she asked to be discharged. With her consent, an dose-related fashion by pretreatment with tran-
experiment was undertaken to verify the diagnosis quilizers that sedate and inhibit fear (247).
of catatonia. She was given an intravenous bolus Intense fear is pictured as the evolutionary basis
of 0.7 mg umazenil, a benzodiazepine antagonist for both tonic immobilization and for catatonia.
that blocks the receptor and reverses the actions of Recognition that catatonia is present in 10% of
lorazepam. Almost immediately she became dizzy, acutely ill psychiatric inpatients, that it is relieved
nauseous, and anxious and feared impending acci- by the potent anxiolytic drugs, and that patients
dents to family members. The fears quickly con- give the appearance of intense anxiety, led the New
densed to certainty, and within 2 min, she was Zealand psychologist Andrew Moskowitz to pro-
again mute and stuporous and remained so for pose that catatonia is a relic of ancient defensive
2 h. She was treated with lorazepam and recov- strategies, developed during an extended period of
ered. After the rst treatment and reversal, the evolution in which humans had to face predators
experiment was repeated with the same results. The in much the same way many animals do today and
patient was discharged from hospital with prescrip- designed to maximize an individuals chances of
tions for lorazepam and carbamazepine and surviving a potentially lethal attack (15).
remained well (244). Among the defenses of prey animals are ight,
Seizures, whether naturally or experimentally ght, and dissimulation. Flight is stimulated when
induced, may immediately be followed by immo- the predator is seen at a distance; ght is an
bility and stupor. The sudden loss of conscious- option when escape is not possible; and hiding,
ness, posture, and spontaneous motor acts with dissimulation, and lack of movement are encour-
tongue biting and urinary wetting is a severely aged when the predator is at a distance that would
frightening experience. Consequences are often permit not seeing an immobile prey. The core cata-
described as either rigid-catatonic or accid- tonic symptoms of stupor, mutism, and immobility
cataleptic states that bear the characteristics of can be linked to tonic immobilization.
catatonia (245, 246). Fear conditioning, a form of emotional learning
Barbiturates and benzodiazepines that relieve that connects a neutral stimulus to one eliciting
anxious depression also relieve catatonia, an expe- fear, also suggests a link to catatonia. Repeated
rience that, as previously noted, is consonant with exposure to a neutral stimulus (e.g., ringing of a
the fear hypothesis. The rapid eect of these bell) with a painful shock stimulus ties the stimulus
agents is demonstrated by reports that administer- and response together so that ringing the bell elicits
ing parenteral lorazepam to mute and negativistic physiologic autonomic changes (e.g., catechol-
patients en route to MRI or EEG laboratories amine release from the adrenal medulla and
makes them cooperative and talkative for the sympathetic nerves) and endocrine release (e.g.,
procedure. glucocorticoids of the adrenal cortex) (249).

36
Rediscovering catatonia

The most compelling daily life images of ism are behaviors that are analogous to tonic
immobility in the face of intense fear are immobilization. Repetitive words and acts, postur-
reported in accounts of rape assault. The actress ing, and grimacing are dissimulations attempts to
Kelly McGillis presented a detailed description appear other than oneself. A childs fear and dis-
of being assaulted and raped by two men in an comfort stimulates crying that brings nursing, cud-
article on the lm The Accused, noting that, dling, and relief. Older children develop a
although she was conscious, crying, and scream- repertoire of calls, screams, cries, postures, hiding,
ing, she was unable to move (250). Studies of throwing, and breaking of objects to bring similar
tonic immobility during rape and assault describe relief. Being mute and not responding brings
intense fear, inability to move, and perceived desired attention. The behaviors may be active or
physical restraint (251, 252). Fear-induced freez- passive, and for each, the caretakers and other
ing is also reported in military disasters and in children respond, and soon postures, grimaces,
urban violence (253). repetitive acts, repetitive speech, and withholding
Cyber bullying, the sending of hostile denigrating of speech become learned behaviors that elicit
messages through social media, is an increasing reassuring responses.
stressor among adolescents, severe enough to lead to Catatonia may be regarded as a relic of the per-
withdrawal from school, hospital care, and suicide. iod in human history in which being prey for
A 14-year-old girl without prior psychiatric history aggressive animals led to the same defenses that
was admitted to hospital with anxiety, tearfulness, are seen in prey animals. Such imagery is consis-
and insomnia, which matured to an oneiroid state tent with the ecacy of the anxiolytic treatments
with mutism, immobility, waxy exibility, postur- of barbiturates and benzodiazepines. The ecacy
ing, and negativism. Treatment with daily ECT of ECT in relieving catatonia is a puzzlement,
relieved the condition in 2 weeks (254). however. We do not understand the mechanism
The experiences with tonic immobility are rele- by which induced seizures alter behavior,
vant to our understanding of post-traumatic stress although a viable hypothesis considers the brains
disorders (PTSD). In an experiment, both normal release of neuroendocrine hormones in the course
and PTSD subjects related their most disturbing of the seizure as instrumental (257). Such hor-
experiences, and the stories were recorded and edi- mone studies have not been of interest to catato-
ted into 100-word scripts. A script was then read nia scholars. It would be timely to study the
to the subject standing on a platform that mea- neuroendocrine action of the eective treatments
sures body sway. The record of the sway is broad now that catatonia is appreciated as a unique
in normal subjects but very narrow in PTSD behavior syndrome.
subjects reecting increased immobility (255). Catatonia is an abnormal behavior that is identi-
If tonic immobility is a human phenomenon, we ed by observation alone. Changes in movement,
would expect the signs to occur in patients with posture, and speech are sucient to identify the
panic disorders. In a questionnaire study of 1118 syndrome. No damage to the body remains after
community-based subjects with a history of panic recovery, indicating that the abnormal behavior is
attacks, 18% reported being immobilized always, an exaggeration of the normal state. Such observa-
36% sometimes, and 16% rarely during their tions allow the view of catatonia outside the
attacks (256). conventional causes of the bodys systemic
These many diverse experiences and descriptions disorders and encourage its consideration as an
encourage attention to the role of fear in our image inherited adaptive syndrome.
of catatonia. Stupor, rigidity, posturing, and mut-

37
Max Fink

Chapter 8: Lenvoi
The art of drawing conclusions from experiments and relieved catatonia as eectively as do the treat-
observations consists in evaluating probabilities and ments for neurosyphilis today.
estimating if they are large and numerous enough to We see immediate benets in identifying catato-
constitute proofs. The type of calculation is more com- nia as a unique syndrome outside schizophrenia.
plicated and more dicult than we think; it demands Ever since its creation, schizophrenia has been pic-
great sagacity. Antoine Lavoisier (258) tured as an illness of disorganized thought and
speech associated with delusions, hallucinations,
The birth of catatonia as a concept in 1874 was
and emotional attening. When catatonia was
followed by a stormy childhood. Within two
incorporated within schizophrenia, its recognition
decades, it was abducted into the concept of
led physicians to interpret their patients peculiar
dementia praecox and was hidden, like Cinderella,
movements and speech as psychotic even when
for almost a century. Despite the miraculous tting
thought disorder, emotional attening, and the
slippers of sodium amobarbital and induced sei-
other signs of psychosis were not present. The phy-
zures, the eective treatments devised in the 1930s
sicians reexively prescribed neuroleptic drugs as
that should have been liberating, the sequestration
treatment. Little merit has been found in this asso-
of catatonia within schizophrenia remained all but
ciation; indeed, as we have seen, such prescription
complete until the 1970s when catatonia was again
is fraught with the risk of precipitating a malignant
reported in patients with mood disorders and in
syndrome and even death.
neurotoxic states. The recognition that the neuro-
Patients meeting the criteria of catatonic schizo-
leptic malignant syndrome was a form of catatonia
phrenia respond to ECT with relief of catatonia. In
that could be eectively relieved by its treatments
most instances, the relief is accompanied with a
opened clinicians eyes to catatonia outside Krae-
disappearance of the signs that were interpreted as
pelins schizophrenia.
evidence of psychosis. But patients with other
Recognizing catatonia in NMS revived our inter-
forms of schizophrenia, the paranoid, disorga-
est in the unique syndrome and taught us its signs.
nized, undierentiated, and residual forms,
When we perceived the signs of catatonia among
respond poorly to ECT. The benet in the relief of
our delirious manic patients, we were able to treat
catatonia clearly accrues to the signs of catatonia
them eectively and save lives. Cases of NMS in
and not to those of schizophrenia.
patients who had not been exposed to neuroleptics
In the rst half of the twentieth century, catato-
led to recognition of the toxic serotonin syndrome
nia was interpreted in psychologic terms, often
as a treatable example of catatonia. The associa-
labeled as hysteria. Once the syndrome was
tion of catatonia in patients in excited delirious
relieved by amobarbital and by ECT, however, a
states and the remarkable ecacy of induced sei-
biological image took precedence. The ecacy of
zures and benzodiazepines for rapid relief brought
these treatments is now established, an experience
delirious mania within the catatonia tent.
that would be the envy of the clinicians who rst
The recognition that self-injurious behavior in
recognized catatonia but whose knowledge
patients with autism and neurodevelopmental
provided no relief for their patients.
defects were signs of catatonia argued for treat-
The appreciation of catatonia as an indepen-
ment trials that were successful. Catatonia was
dent syndrome in clinical medicine is an example
next appreciated in the repetitive behaviors of
of the successful application of the medical model
patients with Gilles de la Tourette and other tic
of diagnosis the established method of disease
disorders, obsessivecompulsive disorder, anti-
identication by history, physical examination,
NMDAR encephalitis, elective mutism, pervasive
laboratory tests, and validation by response to
refusal syndrome, and akinetic mutism. The evi-
treatment (259). Unfortunately, this medical diag-
dence for viewing these disorders as forms of cata-
nostic model is rejected for psychiatric classica-
tonia is suciently compelling to revisit their
tion and is replaced by an image match-up based
classication, clarify their diagnostic criteria, and
on the main symptoms alone. After listening to
test alternate treatment possibilities.
the patients history, the physician examines
The immediate relief of catatonia by intravenous
symptom checklists to nd a comparable classied
injections of benzodiazepines oered a useful bio-
symptom pattern. No physical examination is
logical marker that veried the clinical diagnosis.
made. No laboratory tests are applied. No refer-
Treatment with high doses of benzodiazepines
ence to prior responses to treatments is consid-
(followed by ECT when the medications failed)

38
Rediscovering catatonia

ered. As a result, the DSM classication is a mel- reected in its strong response to tricyclic antide-
ange of arbitrary and ill-dened labels, and each pressants and to ECT, and the inecacy of SSRI
individual diagnosis is almost exclusively in the and SNRI antidepressants, various psychothera-
eyes of the beholder. pies, and placebos (44, 262).
This error in the classication of melancholia
parallels the error that plagued catatonia. Despite
The melancholia error
extensive writings justifying melancholia as a dis-
Like the arbitrary classication of catatonia as a tinct medical syndrome (44, 261264) and even
form of schizophrenia, melancholia is rejected in pleadings (265), the members of the present DSM-
the DSM classication. Melancholia is a psycho- 5 Work Group on mood disorders have rejected
pathological syndrome that was well described any consideration of melancholia as a distinct
throughout history, beginning in ancient Greek entity. The present proposals for the mood disor-
and Roman medical texts. In the 1850s, a circular ders continue the classication much as developed
insanity of severe depressions alternating with for DSM-IV.
manic episodes was delineated by the French The position in the classication and treatment
psychopathologists Jules-Gabriel Baillerger and of the depressive mood disorders is reminiscent of
Jean-Pierre Falret. Other authors conrmed their the arguments that developed in the 1980s regard-
description, but it was the formulation by Emil ing the recommended treatments of the neuroleptic
Kraepelin of manicdepressive insanity that took malignant syndrome. Once the syndrome was rec-
hold and was appreciated throughout the twentieth ognized as a toxic eect of neuroleptic drugs, the
century. In 1980, the DSM-III discarded the con- connection to the dopamine neurotransmitters was
cept of a single illness of alternating phases and invoked and treatments with dopamine agonists
divided the mood disorders into major depression recommended. It took two decades to replace the
and bipolar disorder. In splitting the disease neurotransmitter model and treatment with that of
entity by polarity, the classiers sought to accom- catatonia and its specic treatment. Splitting the
modate reports that the family patterns and the mood disorders according to the unipolar or bipo-
new treatments with anticonvulsants seemed to lar model has yielded little benet in either the
distinguish the entities. The label of manicdepres- understanding of mood disorders or eective treat-
sive reaction described in DSM-II was discarded, ments. The alternative model, that of melancholia
and melancholia was denigrated to a specier of as a dened entity with specic symptoms and
mood disorders without a code number, thereby signs, laboratory test evidence of cortisol abnor-
inhibiting its use (260). mality as a diagnosis verier, and rapidity of
Melancholia is a distinct identiable syndrome treatment response to tricyclic antidepressants and
with four consistent features (44). The mood is of ECT as validating criteria, warrants better consid-
severe depression or mania or both. Suicide risk is eration in the classication.
high. Motor behavior is always altered with
agitation and restlessness or retardation and stu-
The challenges
por (261). The vegetative signs of anorexia, weight
loss, and insomnia are always present. And, cogni- The relations of melancholia and catatonia is a
tive inability to concentrate thoughts or recall challenge. The early reports of catatonia recog-
memories is a common feature. nized its appearance in patients with disorders in
Extensive studies of cortisol, the adrenal gland mood, and the ndings of catatonia among
hormone, nd hypercortisolemia elevated levels patients with manic and depressive mood disorders
of cortisol in the blood at the height of the illness were the rst awareness that the identity of catato-
and a return to normal levels with eective treat- nia with schizophrenia was false. As we have
ment and clinical relief. With relapse, the abnor- shown, patients with catatonia suer intense emo-
mality returns. Because systemic hormones exhibit tions, often as severe as those with melancholia for
diurnal rhythms, the abnormality is best measured which suicide is the major risk. Both catatonia and
by the dexamethasone suppression test, a measure melancholia are rapidly resolved with ECT, sug-
of serum cortisol over 24 h. An abnormal test is gesting an inherent commonality that is not under-
considered a marker of melancholia and is used to stood and warrants exploration. Our lack of
verify the diagnosis (44). experimental evidence makes us believe that mel-
Physiologic abnormality in melancholia is also ancholia does not resolve with benzodiazepines or
reected in abnormal sleep EEG studies and in barbiturates and that catatonia does not resolve
dysfunction of thyroid metabolism. That melan- with tricyclic antidepressant drugs. But these
cholia is a specic form of mood disorder is assumptions are not secured by clinical trials; they

39
Max Fink

may be wrong and testing is warranted. The neuro- ment for patients with catatonia. Patients who are
endocrine abnormality in melancholia is well docu- mute, negativistic, and in stupor, delirium, or furor
mented, but that in catatonia is not. These may be unable to give verbal or written consent for
questions need attention to clarify the relationship their care. In many treatment venues, the use of
between melancholia and catatonia. intravenous sedatives, and more so, the application
The connection of catatonia with seizures is of ECT is severely restricted; in some US states, it
another challenge. We know how to induce seizures is even proscribed by law.
and can oer eective treatment. We know that the Two cases illustrate how legislative obstacles to
benet for the patient resides in the seizure and not appropriate treatment of catatonia can adversely
in any aspect of the inducing agent whether the aect individual patients and their families:
stimulus is chemical, electrical, or magnetic is irrel-
A 22-year-old woman, without any prior psychiatric
evant. The eects are immediate, being appreciated
illness, developed malignant catatonia following a
within a day, but repeated seizures are required to
Caesarean delivery. The legal hurdles to obtaining
sustain relief. Each seizure aects the neuroendo-
consent under the highly restrictive rules of California
crine system, releasing a massive discharge of hor-
severely impeded her treatment for 33 days, risking her
mones into the brain and throughout the body.
life and causing unnecessary additional expense for her
Some hormone eects are persistent and seem cor-
and for society. Within 36 h of treatment she no longer
related with recovery and sustained benet (257,
required special nursing care and proceeded to full
264). While we have some information on the hor-
recovery (266).
monal eects of induced seizures, we have very lit-
tle on the hormone changes in catatonia. The A 16-year-old adolescent suering from autism spectrum
relationships between seizures and catatonia war- disorder developed life-threatening self-injurious behav-
rant greater attention. ior. After the failure of extensive psychological and medi-
Another intriguing aspect of catatonia is its simi- cal treatment trials a course of ECT in Baltimore
larity to the animal condition of tonic immobility, resolved the injurious behavior and allowed him to return
a reex that can be experimentally induced in prey to his home to be maintained by periodic continuation
animals under conditions of acute threat. Many ECT. But the family lived in Los Angeles where ECT in
authors, beginning with Kahlbaum, noted the fear children and adolescents under age 18 is legislatively
and tension of patients with catatonia. The evi- interdicted. For his treatments the family had to y every
dence for a relationship between fear and catatonia 7 to 10 days to Tucson, Arizona in order for the boy and
encourages more detailed study. the family to experience a more healthy home life.
But while we have made much progress in iden-
Kahlbaum envisioned catatonia as a disorder of
tifying the patients who have catatonia and in our
movement and mood, and in the more than a
ability to help them, the position of patients with
century since his description, we have learned to
catatonia in our society is perilous. First, the
identify the syndrome as he saw it. We have added
recognition of catatonia and its teaching are poor.
veriable tests and developed eective and safe
Catatonia is typically hidden in textbooks as a type
treatments. We are now able to detect many forms
of schizophrenia, nothing more, and its eective
that catatonia takes in the clinic. The recognition
treatments are disregarded. Published case reports
of catatonia assures not only advances in wellbeing
describe prolonged illness and even fatalities
of patients but also oers personal gratication for
because catatonia was not appreciated and not
clinicians as they restore a sick person, often mori-
appropriately treated.
bund, to full health. In liberating catatonia as an
The stigmatization of ECT is widely encouraged
independent treatable syndrome in the dictionary
by the failure of medical school faculties and psy-
of psychiatric illnesses, we honor the vision of Karl
chiatric and neurologic residency training programs
Kahlbaum and bring his image of catatonia out of
to consider the treatment a necessary part of the
the shadows.
education of their students. The failure of many
psychiatric hospitals and medical schools to equip
and maintain ECT treatment facilities is evidence of Acknowledgements
the stigma. Such failures are unethical in the princi-
ple of justice, the necessity for society to make Michael Taylor brought catatonia out of the shadows of
known eective treatments available to all citizens schizophrenia by his diagnostic studies in the 1970s. When my
interest in catatonia was aroused in the 1980s, Mickey
regardless of their illness and social status (266). explained the many variations of the syndrome and together
Further, while the state has a duty to protect the we challenged the DSM classication. We went on to collabo-
interests of minors and the psychiatrically ill, spe- rate in expanded texts on Catatonia (2003) and Melancholia
cial regulations have come to inhibit eective treat- (2006).

40
Rediscovering catatonia

Edward Shorter dramatically extended my education in the 11. Fink M, Taylor MA. Catatonia. A clinicians guide to
history of psychiatry. His 1997 History of Psychiatry is the diagnosis and treatment. Cambridge UK: Cambridge
classic modern text. In 2007, with David Healy, he wrote University Press, 2003.
Shock Therapy: A History of Electroconvulsive Treatment in 12. Taylor MA, Fink M. Catatonia in psychiatric classica-
Mental Illness. He went on to write Endocrine Psychiatry tion: a home of its own. Am J Psychiatry 2003;160:
(Oxford 2010) and asked me to add a clinical chapter. Since 12331241.
then, he has written extensively on the history of malignant 13. Kahlbaum KL. Catatonia. Levi Y, Pridon T (trans).
catatonia and delirious mania. He stimulated much of my Baltimore: Johns Hopkins University Press, 1973.
enthusiasm for the stories of the clinicians who went before 14. Dhossche DM, Wachtel LE. Catatonia is hidden in plain
us, on whose eyes we depend for our images and on whose sight among dierent pediatric disorders: a review arti-
shoulders we stand. cle. Pediatr Neurol 2010;43:307315.
Dirk Dhossche and Lee Wachtel are intrepid child and ado- 15. Moskowitz A. Scared Sti: catatonia as an evolution-
lescent psychiatrists who broadened our concepts of catatonia ary-based fear response. Psycholog Rev 2004;111:984
to include many childhood disorders now labeled by epony- 1002.
mous names. The recognition of these behaviors as forms of 16. Rosenberg , CE . Disease and social order in America:
catatonia has improved the lives of many disadvantaged and perceptions and expectations. Milbank Q 1986;64(Suppl.
disabled youth. 1):3455.
When Taylor and I rst described catatonia as a common 17. Kahlbaum KL. Die Katatonie oder das Spannungsirre-
medical syndrome, David Healy challenged our interpretation sein: eine klinische form psychischer Krankheit. Berlin:
that catatonia was a frequently diagnosable illness. He sent out Verlag August Hirshwald, 1874.
registrars to hospitals in Wales and India, used our rating 18. Rogers D. Motor disorder in psychiatry. Chichester UK:
scale, and found patients with the syndrome, as predicted. He John Wiley & Sons, 1992.
also challenged us to defend catatonia as an independent 19. Wachtel LE, Dhossche D. Self-injury in autism as an
identiable and treatable syndrome. alternate sign of catatonia: implications for electrocon-
Edward Shorter, David Healy, Jan-Otto Ottosson, Tom vulsive therapy. Med Hypothes 2010;75:111114.
Bolwig, and Kennedy Cosgrove were readers and critics of 20. Kahlbaum KL. Die Gruppierung der psychischen Kran-
early drafts of this biography. I am indebted to my wife kheiten und die Einteilung der Seelenstorungen [The
Martha and the editor Jonathan Cobb who read each revision arrangement of psychiatric disorders and the classica-
demanding increasing clarity and simplication. The text was tion of disorders of the mind]. Danzig: AW Kafemann,
ably copyedited by Susan Belanger. The index was assured by 1863.
Kristin Nyitray and Lynn Toscano, archivists at the Stony 21. Hecker E. Die hebephrenie. Virchows Archiv Patholog
Brook University Library. Anat Physiolog klin Med 1871;52:394429.
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Foundation and the predecessor International Association thology: a sourcebook. New York: John Wiley & Sons,
for Psychiatric Research who supported my studies of 1976.
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