Pediatrics and Neonatology (2013) 54, 56e59

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CASE REPORT

Eosinophilic Meningitis Caused by

Angiostrongylus cantonensis in an
Adolescent With Mental Retardation and Pica
Disorder
Chang-Wei Hsueh a, Huan-Sheng Chen b, Chen-Hua Li c,d, Yu-Wei Chen c,d,*

a
Department of Pediatrics, Landseed Hospital, Taoyuan, Taiwan
b
Division of Infection, Department of Internal Medicine, Landseed Hospital, Taoyuan, Taiwan
c
Department of Neurology, Landseed Hospital, Taoyuan, Taiwan
d
Department of Neurology, National Taiwan University Hospital, Taipei, Taiwan

Received Mar 9, 2011; received in revised form Aug 29, 2011; accepted Sep 14, 2011

Key Words Eosinophilic meningitis or encephalitis is a rare disorder and is most commonly caused by An-
acute urinary giostrongylus cantonensis. Humans are accidentally infected when they ingest raw snails or
retention; vegetables contaminated with the parasite larvae. Because of the improvement in sanitary
Angiostrongylus food handling practices, the occurrence of A. cantonensis eosinophilic meningitis has been
cantonensis; decreasing in Taiwan in recent decades. The common symptoms and signs of eosinophilic
eosinophilic meningitis are severe headache, neck stiffness, paresthesia, vomiting, nausea, and fever.
meningitis; Acute urinary retention is a rare presentation. We report a case of A. cantonensis eosinophilic
pica meningitis in an intellectually disabled patient who presented with acute urinary retention
without any other meningeal signs. The patient received supportive treatment with corticoste-
roid therapy and was discharged and received urinary rehabilitation at home.
Copyright 2012, Taiwan Pediatric Association. Published by Elsevier Taiwan LLC. All rights
reserved.

1. Introduction

Eosinophilic meningitis or myelitis can be found in various

* Corresponding author. Landseed Hospital, Number 77, Guangtai parasitic infestations of the central nervous system (CNS),
Road, Pingzhen City, Taoyuan County 324, Taiwan. in inflammatory diseases such as tuberculous meningitis,
E-mail address: chanyw@landseed.com.tw (Y.-W. Chen). cerebrospinal syphilis, viral and fungal meningitis, in drug

1875-9572/$36 Copyright 2012, Taiwan Pediatric Association. Published by Elsevier Taiwan LLC. All rights reserved.
http://dx.doi.org/10.1016/j.pedneo.2012.11.005
Eosinophilic meningitis with A. cantonensis
allergies, in multiple sclerosis, and in neoplasms such as
malignant lymphoma, Hodgkin disease, and leukemia.1
Among the parasitic agents, Angiostrongylus cantonensis
and Gnathostoma spinigerum are the most common path-
ogens in eosinophilic meningitis. G. spinigerum occurs in
Southeast Asian countries, mainly in rural communities
where the ingestion of raw snails or insufficiently cooked
poultry is the main source of this type of infection. Eosin-
ophilic meningitis caused by G. spinigerum has never been
reported in Taiwan.2e4
A. cantonensis is the most common parasitic cause of
eosinophilic meningitis in Taiwan.5 A. cantonensis is
a nematode that inhabits the pulmonary arteries and heart
of rodents. Humans may be accidentally infected by the
parasite. The common initial presentations of eosinophilic
meningitis are headache, neck stiffness, paresthesia,
vomiting, nausea, and fever. Acute urinary retention is
a symptom rarely associated with the parasitic infection.
We report here a case of a patient who showed few
meningeal signs but was finally confirmed to have A. can-
tonensis eosinophilic meningitis.
2. Case Report
A 15-year-old boy with moderate intellectual disability was
brought to the emergency department (ED) because of
progressive conscious disturbance for 2 weeks, reported by
the attending teachers in the nursing home where he resided.
The associated symptoms included paucity of speech and
decreased engagement in playing and other daily activity. He
had visited the pediatrics clinic and ED for irritability and
abdominal pain six times within the preceding 2 weeks.
Physical examination at the first visit to the ED revealed
normal body temperature (36.2 C), elevated blood pressure
(152/100 mmHg), and mildly distended abdomen. No neck
stiffness or increased chin-to-chest distance was present. A
laboratory study showed leukocytosis (white blood cell
count: 16.75  109/L) with mild eosinophilia (5.6%).
Nephrotic syndrome had been diagnosed 8 years previously,
and the patient was taking regular oral steroids (predniso-
lone, 45 mg/day) before admission. A radiograph of the
patients abdomen showed a normal bowel-gas pattern. He
was then discharged from the ED.
In the following week, this patient came to the hospital
twice because of abdominal pain and shaking of the
shoulders. A follow-up laboratory study 1 week later
showed progression of leukocytosis (19.9  109/L) and
eosinophilia (16%).
Difficulty in urination had been noted on Day 8 of the
illness and was associated with decreased speech. The
patient came to our ED again, and abdominal plain film
showed a pelvic mass suggesting urinary bladder distension
with bowel displacement. Transurethral catheterization
revealed 1650 mL of residual urine (Figure 1).
On Day 17, the patient became drowsy all day long and
was not able to stand up. He was admitted for further
management. Neither fever nor convulsion had been
observed by his family and attending teacher since his first
visit to our ED. However, the patients family and attending
teacher reported that he had pica disorder, whereby he
consumed nonfood items outside the house.
57
Figure 1 Radiograph of the kidneys, ureters, and bladder
showing a pelvic mass with bowel displacement.
On admission, the patient was afebrile but irritable,
with E3V2M5 on the Glasgow Coma Scale. His general
physical examination was unremarkable, except for
tachycardia (111/min) and elevated blood pressure (140/
97 mmHg). His neck was supple. Neurologic examination
showed isocoria with retained light reflex. Other examina-
tion with limited compression and cooperation showed
weakness of all four limbs (2e3/5 on the Medical Research
Council Scale), which was greater in the lower extremities,
characterized by difficulty bending the knees. Increased
deep tendon reflex of knee jerks was recorded, without
obvious Babinski sign bilaterally. There was no edema in his
extremities, face, or scrotum.
The patients leukocyte counts in peripheral blood kept
elevating, from 16.75  109/L at presentation to
21.06  109/L at admission. The percentages of bandform
neutrophils increased simultaneously from 5.6% to 26%.
Serum albumin concentration was 3.7 g/dL. Routine
urinalysis yielded negative results.
We suspected that the patients clinical symptoms may
have been caused by a central nervous system infection. A
lumbar puncture was performed with opening pressure of
210 mmH2O. The color of cerebrospinal fluid (CSF) was
whitish turbid, and the analysis showed a white blood cell
count of 0.837  109/L, with 86% eosinophils and 14%
lymphocytes. The CSF chemistry showed a glucose level of
2.5 mmol/L, and protein 106 mg/dL. Culture for CSF
showed all negative results for virus, fungi, bacteria, and
mycobacteria. Results of CSF polymerase chain reaction for
herpes simplex virus-1 and -2 were also negative. Magnetic
resonance imaging (MRI) study of the cervical spine showed
no compressive lesion (Figure 2), and MRI of the brain
showed no enhancing or space-occupying lesions (Figure 3).
58
Figure 2 Sagittal T2-weighted MRI showing neither
compression nor intramedullary lesions.
Based on the aforementioned clinical, laboratory, and
radiologic results, eosinophilic meningitis was demon-
strated. The patients serum and CSF specimens were sent
to the Department of Parasitology, College of Medicine,
Kaohsiung Medical University. The antibody levels in the
serum and CSF of this patient were detected by enzyme-
linked immunosorbent assay by using an antigen with
a molecular weight of 204 kDa from fifth-stage worms
of A. cantonensis; that antigen had been purified using
monoclonal antibodies.6
After admission, we started empirical treatment with
ceftriaxone, and 4 mg of dexamethasone was administered
intravenously every 6 hours. A second lumbar puncture was
performed 4 days after the first, and the follow-up CSF
analysis showed a white blood cell count of 0.171  109/L
with 25% eosinophils. After 10 days of dexamethasone
administration, treatment was interrupted because of
a urinary tract infection. The patients activities improved
and his sleep quality was better during hospitalization, with
sequelae of urine retention. He was discharged with a Foley
catheter inserted and received urinary rehabilitation at the
nursing home for the disabled where he resided.
3. Discussion
Eosinophilic meningitis is a rare neurologic disorder that
can be caused by infectious or noninfectious etiology.
Noninfectious causes include malignancy, intracranial
foreign body, and medication.7 In eosinophilic meningitis
caused by infection, A. cantonensis and G. spinigerum are
the most common agents.8 Eosinophilic meningitis is most
commonly caused by the rat lungworm A. cantonensis.9 It is
endemic in Taiwan, Southeast Asia, the Pacific islands,
Australia, and the Caribbean islands.10e13 The life cycle of
A. cantonensis involves snails, slugs, or fish as intermediate
C.-W. Hsueh et al
hosts and rodents as definitive hosts. Human beings are
accidentally infected when they eat poorly cooked or raw
fish, slugs, or snails, or vegetables contaminated by infec-
ted rats. Most of the cases in Taiwan were reported before
the 1970s.12 Because of increasingly improved sanitary food
handling practices, only scattered cases have been re-
ported during the past several decades. In Taiwan, the
cases with eosinophilic meningitis caused by A. cantonensis
comprised children in the past three to four decades, and
adults in the past two decades. The decreased case
numbers in children may be related to the urbanization and
improvement of public health, leading to less frequent
exposure of children to snails and other intermediate host
animals.5
Institutionalized intellectually disabled children have
been reported to be prone to parasitic infection.14 Pica
disorder, defined as consumption of nonfood items, is one
of the most frequently observed eating disorders in people
with a developmental disability.15 The prevalence of pica in
institutionalized intellectually disabled people was re-
ported to be 25.8%.16 Patients with such an eating disorder
are prone to intake of the raw intermediate hosts of
A. cantonensis. Such patients require special supervision by
staff, and several behavior interventions have been
reported.17
Diagnosis of A. cantonensis eosinophilic meningitis is
confirmed by recovery of larvae from the central nervous
system, but the frequency of detecting these worms in
patients is very low.12 Presumptive diagnosis may be based
on laboratory findings in the blood and CSF, a history of
eating intermediate hosts, serologic tests, and clinical
symptoms.6 The common symptoms and signs of A. canto-
nensis eosinophilic meningitis are headache, neck stiffness,
paresthesia, vomiting, nausea, and fever. Abdominal pain
and weakness of the extremities are less common.18 Our
patient presented with acute urine retention, which is
rarely reported.19,20
Acute urinary retention, which refers to the inability to
void urine by oneself, is prevalent in older people and rare
in children and adolescents.21 Here, we report a case of
acute urinary retention that accompanied eosinophilic
meningitis in an adolescent with mental retardation who
lived in a nursing home. The more common causes of acute
urinary retention in children include neurologic disorders,
severe voiding dysfunction, urinary tract infection, con-
stipation, adverse drug effects, local inflammatory causes,
locally invasive neoplasms, and obstructive lesions.22,23 The
neurologic disorders that accompany acute urinary reten-
tion include peripheral nervous diseases such as sacral
herpes and central nervous diseases such as aseptic
meningitis and eosinophilic meningitis.19,20,23 However, the
typical symptoms and neurologic signs of meningitis may be
mild or even absent initially. As in our patient, there may
be no vomiting, fever, or neck stiffness initially, and the
CNS infection can be difficult to diagnose early. Most cases
of A. cantonensis eosinophilic meningitis are mild and self-
limiting. Lumbar puncture should be performed to relieve
the headache caused by increasing intracranial pressure.
Treatment is supportive and symptomatic and is based on
the use of analgesics with or without corticosteroids.12
The diagnosis could be difficult if the patient presents
few meningeal irritative symptoms, as in our case. Because
Eosinophilic meningitis with A. cantonensis
Figure 3 Enhanced brain MRI showing negative findings.
of our patients mental retardation, he was unable to
describe his symptoms, such as headache, clearly and in
detail, yet he still exhibited no vomiting, fever, nuchal
rigidity, or Brudzinski or Kernig signs. A. cantonensis
eosinophilic meningitis is not commonly known to present
with acute urinary retention. Acute urinary retention is
a urologic emergency, and urethral catheterization is the
primary treatment to avoid bladder injury. Because acute
urinary retention is associated with a variety of causes, the
definite cause should be determined carefully. Although
A. cantonensis eosinophilic meningitis is a usually mild and
self-limiting disease, death can occur in severe cases.12
In conclusion, an institutionalized mentally retarded
adolescent with pica disorder could be at high risk for
eosinophilic meningitis caused by A. cantonensis. Further
work-up, including neuroimaging and CSF study, should be
performed to confirm the possible causes of eosinophilic
meningitis and start appropriate treatment promptly.
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