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BURNING MOUTH SYNDROME


Philip-John Lamey, BSc, BDS, MBChB

When researching orofacial diseases in a t u r e ~ . ’Occasionally


~~ such conditions are
scientific manner, it is fundamentally im- present coincidentally in patients with BMS,
portant that there is agreement on what con- but the diagnosis of BMS should be confined
stitutes the condition and what features are to patients with an apparently normal oral
necessary for diagnosis. In the case of the mucosa.
condition that we shall call burning mouth Some discussion has taken place about the
syndrome (BMS), the oral mucosa appears nomenclature of BMS and in particular the
clinically normal. Patients, however, say that inclusion of the word syndrome.” By defini-
the areas affected feel burning or hot or occa- tion, a syndrome is a collection of signs and
sionally scalded. symptoms, which in BMS is a normal-looking
Historically this condition was described as mucosa and a burning sensation. We increas-
glossodynia,1-7, 21, 31. 55. 83. 110. 117, 174 glossa- ingly recognize that BMS also can be associ-
pyrosis,2O, 34, 91 stomatodynia,122 stomato- ated with burning at other bodily sites and
~ ~ tongue,52*58, 131 burning
p y r o s i ~ , ~sore therefore may be as much a systemic condi-
38, 47, I3O, 174 oral dysesthesia,66,140 tion as a localized oral disorder. Personally, I
burning mouth condition,” and burning favor the retention of syndrome.
mouth syndrome.60, 95, 136 It is not always clear
from these descriptions, however, if the oral
mucosa appeared normal in all cases, and SITES AFFECTED
therefore articles reporting burning mouth
are not referring necessarily to burning The tongue is the most common site to be
mouth syndrome (BMS). When terms other reported by patients with BMS followed by
than BMS are used they often include patients the upper alveolar region, palate, lips, and
who have an obvious clinical abnormality lower alveolar region.89The burning is almost
such as geographic erosive lichen always bilateral and symmetrical. Rarely,
p l a n ~ sor
, ~oral
~ c a n d i d o s i ~In . ~the
~ latter con- other sites such as the buccal mucosa, floor
ditions, patients also may complain of a burn- of the mouth, and throat may be affected.
ing sensation but clearly do not have BMS. The precise site of burning is important and
The history of burning reported by patients may help identify precipitating factors. For
with these conditions is different and usually example, burning at the tip of the tongue
only occurs on eating hot or spicy foods.66 in a dentate subject may suggest a tongue
Furthermore, the histopathologic features of thrusting habit. The same complaint in a com-
these conditions are characteristic, whereas in plete denture wearer may suggest faulty den-
BMS there are no specific histologic fea- ture design, leading to tongue restriction.

From the Department of Oral Medicine, School of Clinical Dentistry, The Queen’s University of Belfast, Belfast,
Northern Ireland

DERMATOLOGIC CLINICS

VOLUME 14 * NUMBER 2 * APRIL 1996 339


340 LAMEY

Burning on the dorsum of the tongue in a women than men. The female to male ratio is
denture wearer may suggest a tongue postur- about 7:1, but this varies with site.89Burning
ing habit possibly to help stabilize a nonreten- of the lips alone is even more common in
tive denture. Even if sites such as the lips women, with a female to male ratio of 12:1.88
alone are involved in BMS, the same precipi- The reason for this sex prediliction has not
tating factors pertain when more typical in- been explained fully, but several other orofa-
traoral sites are affected.88 cia1 pain syndromes such as atypical facial
If patients are asked about burning at other pain and temporomandibular disorders are
body sites they frequently also report burning also more common in women. The exception
in the anogenital region.50Male clinicians are to this is periodic migrainous neuralgia,
less likely to elucidate this information than which is more common in men.87
their female counterparts. The significance of
burning at these other sites is presently un-
clear, but BMS may be a systemic syndrome.

There is broad agreement in the literature


EPIDEMIOLOGY that BMS is a condition particularly affecting
the middle-aged and elderly p ~ p u l a t i o n . ~ ~
There are no global figures available for the The youngest patient in one study was 28
prevalence of BMS. In selected populations in years old. The condition has never been re-
the United States attempts recently have been ported in children, which is odd because sev-
made to estimate the prevalence and distribu- eral of the recognized precipitating factors in
tion of a variety of orofacial pain disorders BMS also can affect children.6
including BMS.'O' The mean age of BMS patients is approxi-
In Finland, one study of 431 predominantly mately 62 years, and several studies have re-
female subjects reported that 15% had experi- ported roughly the same figure. Because
enced a prolonged oral burning sensation.60 women of this age would be postmenopausal,
These patients also underwent a thorough early reports highlighted the significance of
clinical examination and about half of the the climacteric in the etiology of BMS.'jOAs is
15% who complained of burning had some discussed further, however, such hormonal
demonstrable mucosal abnormality such as changes are probably of no significance.
geographic tongue, lichen planus, or oral can-
didosis. It would appear therefore that in a
Finnish adult population the prevalence of SUBTYPES
BMS is around 7%. Women were more fre-
quently affected by BMS than men. Several years ago, my colleagues and I pro-
Another study investigated 145 oophorec- posed a classification of BMS into three sub-
tomized women by means of a postal ques- t y p e (Table
~ ~ ~ 1).
~ Originally we observed that
t i ~ n n a i r eNo
. ~ ~individual was examined clin- some patients had burning only occasionally,
ically but approximately 18% reported a
burning sensation affecting the tongue or lips.
It was not possible to determine the true Table 1. SUBTYPES OF BURNING MOUTH
prevalence of BMS, but the survey did show SYNDROME
no temporal reduction in the frequency of Relative
oral symptoms after oophorectomy. The in- Frequency
troduction of estrogen therapy did not im- Type ("/) Symptoms
prove the patients' oral symptoms, including 1 35 Burning present every day, not
burning, leading the authors to conclude that present on waking but
estrogen deficiency following the climacteric developing as the day goes
produced vasomotor changes that may lead on, being maximal in the
evening
to neurosis. 2 55 Burning present all day, every
day
3 10 Burning only on some days
GENDER EFFECTS and tending to affect
unusual sites such as
buccal mucosa, floor of
All published studies of patients with BMS mouth, and throat
agree that the condition is more common in
BURNING MOUTH SYNDROME 341

others had it every day, but only as the day neous condition. If subtyping helps us manage
went on, and still others had burning all day, our patients better, then it is useful.
every day. We presumed, incorrectly as it
transpired, that their different history repre-
sented an evolving process, but we subse- PATIENT EVALUATION
quently realized this is not the case.89
For many BMS patients an oral medicine
specialist is not their first port of call. Many
Type 1 Burning Mouth Syndrome have been to their doctor, dentist, general
physician, general surgeon, and specialists
In this subtype patients have burning every such as otorhinolaryngologists, to no avail.
day. The burning, however, is not present on Of the 700 or so BMS patients who have
waking, but comes on as the day goes on, consulted me, not one has ever heard of any
being maximal in the evening. Around 35% other person with the same complaint. This
of BMS patients give this history. circuit of referral and feeling that they are the
only person in the world with the complaint
leads to increasing isolation and in some
cases feelings of hopelessness. The complaint
Type 2 Burning Mouth Syndrome itself, that is, ”my tongue is burning or feels
on fire,” also sounds (even to the patient)
In this subtype patients have burning every odd, and they don’t want to be labeled as
day, which is present all day from waking. having a psychiatric problem. When numer-
Approximately 55% of BMS patients give ous generalists and specialists have pro-
this history. nounced that their tongue looks normal, this
adds to the feeling that they must be imagin-
ing it, which of course they are not. In addi-
Type 3 Burning Mouth Syndrome tion, fear that the burning will develop into
cancer heightens their concern to seek treat-
In this subtype burning is only present on ment.
some days, and on others patients are asymp- It is clear that BMS patients must be treated
tomatic. In contrast to type 1 and type 2 pa- in a sympathetic manner. An unhurried clini-
tients, the burning tends to affect unusual cal environment is helpful, and privacy is
sites such as floor of mouth, buccal mucosa, essential. All our BMS clinics are run jointly
and throat. Around 10% of BMS patients give by an oral medicine specialist, a prosthodon-
this history, which previously was reported tic specialist, and a clinical psychologist. One
as atypical burning mouth of the clinicians is a woman. Having more
Recent demographic data comparing UK than one specialist is helpful because, as with
and US BMS populations have given almost all pain clinics, some patients do not take to
identical prevalence rates for the subtypes in one clinician, and trust is never established.
these two populations.81 By having three specialists, including one of
The subtypes have merit because they are their own sex, the patient is likely to trust
of prognostic significance and also indicate one of us and feel at ease to discuss causes of
the necessity for specialist investigations such stress, tension, or marital disharmony.
as patch testing to identify allergic compo- Having reassured the patient that he or she
nents to the burning. A recent study of type is one of many sufferers, it is important to go
3 BMS patients showed that their main pre- into the history in detail and subtype the
cipitating factors were emotional instability BMS. All patients are asked to quantify the
and allergic factors.97It is, however, inappro- burning using a 0 to 10 visual analog scale in
priate to patch test all BMS patients. Simi- which 0 is no burning and 10 is the worst
larly, in evaluation of treatment outcomes, burning imaginable. Typical pretreatment
type 2 patients are the most difficult to treat values are shown in Figure 1. This quantifi-
successfully because a high proportion have cation also allows the response to treatment
chronic anxiety, which is the most recalcitrant to be measured prospectively. Because social
obstacle to cure.86 circumstances also can influence pain percep-
It has taken many years to establish BMS as tion, all patients are asked to quantify what
a distinct clinical entity and having done so we we call their home circumstances, that is, how
are now recognizing that it is not a homoge- they get along with their family and friends
342 LAMEY

50 1
40 -

&
30 -
n
5
= 20-

10 -
0-
Score Known

Figure 1. Reported severity of BMS patients pretreatment.

and their job and financial considerations. A The general medical and dental history also
linear analog scale also is used, but in this is assessed, and at the end of the first inter-
scale, 10 means everything is perfect and 0 view the clinician has a good grasp of the
means that things could not be worse. When problem, its duration, its subtype, and its se-
the patient chooses a score of 6, for instance, verity, and a knowledge of the social circum-
he or she is then asked what would have to stances.
happen to take their 6 score to 10. I have Subsequently, many investigations are un-
never had a patient refuse to do this and the dertaken, all based on our knowledge of pre-
results are illuminating, for it identifies exact cipitating factors in BMS from the literature.
problems. A typical social circumstances pro- The plethora of recent reviews on BMS in
file is shown in Figure 2. The clinician has several languages has not added anything to
not been seen to pry or ask lengthy irrelevant our knowledge.* It is the relative importance
questions about possible stress, but the pa- of each individual precipitating factor that is
tient has chosen the score, and the patient
has been asked to justify the score including *References 1, 7, 15, 25, 48, 49, 61, 100, 108, 116, 132,
identification of social problems if any. 139, 148.

0 1 2 3 4 5 6 7 8 9 10
Visual Analog Score

Figure 2. Reported home and social circumstances scores by BMS patients at


initial presentation.
BURNING MOUTH SYNDROME 343

important. To date, this has been the case in 6). In half of these patients, the deficiency
only one prospective state was deemed as the sole cause of BMS
because the condition resolved following ap-
propriate replacement therapy. In a subse-
PRECIPITATING FACTORS quent report by the same 40% of 37
patients again were identified as being vita-
The following factors listed are thought to min B12, folic acid, and iron deficient. Again,
be important in precipitating BMS. They are hematinic deficiency was thought to be pri-
not presented in any particular order. marily responsible for the BMS as judged by
response to treatment.
Hematinic deficiency states
In an American study, only 1 patient of 57
Undiagnosed maturity onset diabetes mel-
was found to be hematinically defi~ient.~ That
litus study was not of BMS patients alone, how-
Oral candidal infection
ever, but included patients with mucosal dis-
A degree of xerostomia ease such as lichen planus.
Denture design faults
The rationale for investigating vitamin B12,
Parafunctional habits
folic acid, or iron deficiency in BMS patients
Fear of cancer in some ways reflects what investigations are
Allergy to a variety of substances and ma-
routinely available. Such data do not exclude,
terials of course, other deficiency states, although
Psychological states, particularly chronic assay of those may not be routinely available.
anxiety
One group of vitamins of importance are
Drug-induced disease
those of the B complex group. Biskindl6
thought that there was an intimate relation-
ship between vitamin B complex status and
Hematinic Deficiency States levels of estrogen but lacked quantitative
data. Nevertheless, one study of 86 post-
It has been known for many years that menopausal patients who presented with oral
deficiency states such as sideropenia, iron de- symptoms including BMS reported one third
ficiency anemia, pernicious anemia, and folic to respond promptly to vitamin B complex
acid deficiency could result in oral mucosal
discomfort described as a burning sensa-
tion.12,22, 42, 65, 73, lo5, 172 Occasionally, the defi-
ciency states reported were accompanied by Table 2. THE RELATIVE IMPORTANCE OF
visible changes in the affected oral tissues, PRECIPITATING FACTORS IN BURNING MOUTH
SYNDROME
but the complaint of a burning sensation
could be present when no obvious mucosal Factor Percentage of Patients
changes were evident. Hematinic deficiency states
The exact mechanism by which a variety of Anemia (1
deficiency states can lead to a burning sensa- Ferritin 6
tion is uncertain. In the case of sideropenia Folic acid Nil in our clinic
Vitamin B,, 8
and iron-deficiency anemia, depletion of iron- Vitamin B,,BPI and B6 37
related compounds such as cytochrome oxi- Undiagnosed maturity onset 3-5
dase leads to functional epithelial changes. diabetes
In vitamin B,, and folic acid deficiency oral Oral candidal infection 32% isolation but only 5%
mucosal morphologic changes similar to respond to antifungal
therapy
those described in bone marrow and red cell A degree of xerostomia Stimulated parotid flow rate
precursors have been r e p 0 ~ t e d . I ~ ~ reduced in 12%
The incidence of deficiency states reported Denture design faults Variable but only 25% are
in BMS varies (Table 2). Personally, I believe improved by new
dentures of good design
the incidence data are less important than the Parafunctional habits 20-61
fact that deficiency states can precipitate BMS Fear of cancer 20
and therefore need to be sought in all cases. Allergy 5
In one study of 55 BMS patients,lo553% were Psychological state Variable and complex but
sideropenic and 4% were low in folic acid. In approximately 36% have
marked chronic anxiety
another study,12 7 of 21 BMS patients were Drug induced Rare
low in vitamin B,, (n = 1) or folic acid (n =
344 LAMEY

replacement therapy."O Others also suggested oral sympt0ms.5~Other studies have reported
a link between glossodynia and vitamin B more encouraging results.53Of 43 previously
complex defi~iency.~, undiagnosed noninsulin-dependent diabetics
A fairly recent study quantified vitamin B1, (NIDD) 16 had BMS, and all resolved follow-
B2, and B, levels in BMS patients and age- ing glycemic therapy. A similar result was
and sex-matched control subject^.^, Of 70 pa- found in a study in which 4 of 150 BMS
tients, 28 were deficient in one or more vita- patients were found by glucose tolerance test
mins. Replacement therapy with vitamin Bl (75 g load) to be NIDD. In that study BMS
(300 mg daily), vitamin B, (20 mg daily), or resolved entirely on instituting glycemic con-
vitamin B, (150 mg daily) was given to 28 Another study found 1 NIDD patient
t1-01.~~
deficient patients and 27 patients with BMS in 20 BMS patients, so an overall figure of
who had normal levels of vitamins B1, B2, about 5% of BMS patients being NIDD seems
and B,. Eighty percent of BMS patients with reasonable.'osIt should be appreciated that in
proven vitamin deficiency were asymptom- a UK population 5% is a high figure consider-
atic at 3 months compared with none of the ing that 1%of the population are known dia-
nondeficient group, although 7% of the latter betic and 1%undiagnosed. Indeed, a patient
group reported improvement. Others recently is twice as likely to be NIDD when presenting
have repeated similar studies with disap- with a variety of oral complaints. In practice,
pointing results, but the doses of vitamins all patients with BMS need diabetes mellitus
prescribed were not e q ~ i v a l e n t . ~ ~ excluded, preferably by fasting blood sugar
Finally, other deficiency states have been estimation.
investigated in BMS patients. Zinc levels are The relationship between BMS and diabetes
equivalent in BMS patients compared with mellitus also has been investigated by study-
age- and sex-matched control subjects.'06 ing diabetic patients for symptoms of BMS.
BMS was found in 10% of patients in one
study (n = 110) and was the second most
Undiagnosed Maturity Onset common oral complaint after xerostomia in
Diabetes Mellitus another.12 Various reasons have been pro-
posed to explain the relationship between
One of the first to relate oral symptoms and BMS and diabetes mellitus, including meta-
diabetes mellitus was Sheppard, who re- bolic alterations in the oral mucosa, accompa-
viewed the literature prior to 1942.'33He con- nying xerostomia, and oral candidosis.
sidered the link largely a reflection of the
preinsulin era and with treatment the oral
cavity of diabetics should differ little from Denture Aspects
nondiabetic subjects.
Other researchers studied the link and sug- Denture design faults that increase the level
gested the oral cavity to be a sensitive indica- of functional stress or restrict normal function
tor of abnormal glucose tolerance.134, 13s Of 26 of the lingual musculature can lead to BMS.12
patients with abnormal glucose tolerance test Denture-bearing areas of the oral mucosa are
results, 9 reported occasional oral burning. In subjected to stress for which they were not
another report, 12 of 45 patients with a vari- designed and assume the role of the peri-
ety of oral complaints including burning had odontal membrane on transferring functional
abnormal glucose tolerance test results con- forces to the underlying bone.138, 147 His-
sistent with laboratory criteria at that time for tologically, such loads produce variable
diabetes mellitus.2yThe technical quality of changes in the oral mucosa as little as 1 year
the latter report was reviewed and urinalysis after denture insertion.*
questioned as a sensitive means of detecting In a study of 33 patients with BMS, 50%
diabetes mellitus or prediabetes.Il7 There are had an error in denture design that was
anecdotal reports of "sore tongue" in patients thought to be related causally to their BMS.*05
with diabetes mellitus resolving following the In a much larger study of 150 patients of
institution of glycemic whom 120 were edentulous, 60% had denture
Unselected patients with various oral com- design faults, but replacing dentures alone
plaints have been subjected to glucose toler- only helped 25% of BMS patientse9 Thus,
ance tests.I9 In one study, 30% had abnormal
results, but it was thought that early small "References 3, 26, 68, 76, 79, 109, 111, 114, 118, 156,
blood vessel disease precluded resolution of 157, 161, 162.
BURNING MOUTH SYNDROME 345

even if the patient's BMS is coincidental with using identical sampling techniques. Salivary
complete denture provision, the dentures gland output does appear related to gland
alone account for BMS only in a quarter of size, and this varies between i n d i v i d ~ a l s . ~ ~
patients. This of course reflects the multifacto- Because BMS occurs predominantly in
rial nature of BMS. Good dentures on a com- postmenopausal women, a reduction in sali-
promised mucosa can produce BMS, good vary flow in this group has been suggested
dentures on a healthy mucosa can produce as a possible cause of the burning, but good
BMS secondary to parafunction, and so on. data are lacking.38In one study, parotid sali-
As well as being of good fit, occlusal errors vary flow rate (resting and stimulated) was
also can produce a burning sensation,"j5and reduced in postmenopausal women, thus
the dentures themselves have to be made cor- lending support to a link with BMS.71Another
rectly to maintain the health of the underly- study, however, showed no differences in
ing mucous membrane.166 resting mixed saliva between postmeno-
Excess loading on dentures was reported pausal women who had burning tongues and
by Thomson145to produce a burning sensa- those who did and others have shown
tion principally in the palate, but also on the similar results.8oXerostomia itself is multifac-
tongue and lips. Symptoms of burning could torial, and factors contributing to xerostomia
appear within minutes of denture insertion also may influence BMS.30In one study, 40%
and be relieved by removing the dentures. of BMS patients complained of a degree of
Systemic factors such as deficiency states also xerostomia, but only 12% had reduced stimu-
may have been relevant to his observations, lated parotid gland Of clinical interest
and treatment should aim to deal with exces- is that the 12% of patients in whom reduced
sive load, such as by correcting the vertical salivary function could be shown said that
dimensions2,lUa or improving the supporting the burning and dryness were "the same
tissues. Parafunctional activity also leads to thing." If this is the case, then saliva substi-
excessive occlusal loading99and is probably tutes may have a role to play in the manage-
more common than was realized previously ment of some patients with BMS. Other
in dentate and edentulous individual^.^'^, 120 changes in saliva rather than just the volume
Patients who clench or grind their teeth also of saliva could be important, namely compo-
often thrust their tongue against their teeth, sitional changes. Some authors have claimed
and this can give rise to BMS. Such activity elevated protein, potassium, and phosphate
in BMS patients appears to be related to anxi- levels in women with BMS and a relationship
ety as detected by the Hospital Anxiety and to hormonal status.14Recent studies have sug-
Depression Scale.9oOthers also have linked gested no change in protein profiles between
stress to increased muscle activity and BMS patients and control Our own
clenching.167, Parafunction may be uncon- studies have, however, identified a novel pro-
scious,17 and if the patients wear dentures tein in the parotid saliva of BMS patients.126
with acrylic teeth, wear facets indicate this There is no evidence that BMS is associated
habit.lU with histopathologic changes at least in minor
If BMS patients wear dentures, they may salivary glands.l15 In a study of minor pala-
be contributing to their complaint. Careful tine glands in patients with or without the
assessment of the vertical dimension should complaint of burning, no histologic changes
ensure an adequate freeway space.", 98, were found. It was, however, unclear from
Io4, lUa Similarly, tongue movements should that study if patients did indeed have BMS.
not be restricted. The denture bases them-
selves should ensure maximal load distribu- Microbiologic Aspects
tion.14 Underextended denture bases are the
most common design and the area There are few good studies on the oral
available for support can be increased by up microflora of BMS patients. What is funda-
to 50% with fully extended bases.145 mentally important is that patients can harbor
pathogenic numbers of candidal species in-
traorally, yet have no clinical mucosal
Salivary Aspects changes.57
In health, about 40% of patients harbor
It is debatable whether salivary gland func- candidal species intraorally as assessed by
tion reduces with age.13,30, 41, 69, Few studies culture of mixed saliva.loThe techniques for
have been undertaken in a logical manner candidal isolation are important. Various
346 LAMEY

techniques are in use including 153 als have been in patients who complain of a
imprint cultures: epithelial smears, impres- burning mouth, but do not have BMS because
sion cultures? and the oral rinse technique.125 they had clinically obvious oral change^.^ At-
The oral rinse technique is simpler to perform tempts to reduce residual monomer levels by
and is quantifiable, as well as allowing detec- various means, such as boiling, are largely
tion of Staphylococcus aureus and coliforms. unsuccessful.9 There are, however, rare cases
Application of the oral rinse technique to of true allergy to polymethyl methacrylate
BMS patients showed a higher prevalence of in BMS patients who have responded to the
Candida species and coliforms in BMS patients provision of alternative denture base materi-
than control The Candida species als such as nylon.35It would appear that such
most frequently isolated was Candida albicans, patients give a type 3 BMS history, and that
and Enterobacter and Klebsiella were the most other allergens, such as the constituents of
prevalent coliforms. It is likely that the coli- foodstuffs, are more important.97A recent de-
forms were transferred to the mouth via the tailed study of type 3 BMS patients suggested
patients’ fingers, perhaps when investigating that about 50% have allergy as a major pre-
the mouth for disease. cipitating factor, and the other 50% have emo-
Treating BMS patients in which candidal tional instability as a major factor. Appro-
species were isolated (37% of 150 patients) priate patch testing by a dermatologist
improved symptoms in only 6%.89 Other experienced in the technique and professional
studies using various isolation techniques advice on dietary evidence largely resolved
claim figures of 10% candidal isolation in their condition. Numerous other substances
glossodynia patients? 13% in burning mouth have been reported as having an association
patients, and 3.5% in a heterogenous group.’” with BMS by being allergenic in susceptible
Overall, it would appear that candidal spe- individuals. These substances have included
cies play a minor role in BMS. Nevertheless, sorbic nicotinic acid epoxy
in one study 5% of patients had sustained resin (and bisphenol A),155 pyrethroid
improvement following antifungal therapy (an in~ecticide):~palladium? octyl gallate (an
and confirmed elimination of candid^.^' Can- anti~xidant),”~ benzoylperoxide, 4-tolyl dietha-
didal species still should be sought in BMS nolamine, N,N-dimethyl-4-t0luidine?~ peanut
patients, preferably by using the oral rinse extract,163 cinnamon adel~de,2~ and nickel
technique. 159

Allergic Aspects Cancerphobia

It previously has been emphasized that About 20% of patients with BMS have a
even if a patient’s symptoms of BMS began strong fear of cancer.89This fear almost is
when new dentures were provided, it is not never volunteered by the patient but should
likely that the dentures alone were responsi- be asked about by the clinician.38Reassurance
ble. Nevertheless, this apparent association that BMS is in no way related to oral cancer
led to several publications linking BMS to can do much to put a patient’s mind at ease.
allergy to polymethyl methacrylate on the as- Indeed, the outcome of treatment of BMS pa-
sumption that allergy to the acrylic resin or tients who are slightly anxious is even better
metal component of the denture base was than patients who are not anxious, and this
present. is almost certainly because cancerphobia is
It is generally accepted that true allergy to producing additional anxiety.86In type 1 and
polymethyl methacrylate in acrylic is rare.33 2 BMS patients, the presence of symptoms on
Many reported claims for allergy were dubi- a daily basis and the possible lack of success
ous both in the clinical complaint and the in the early consultation period also contrib-
way in which allergy testing was undertaken. ute to the feeling that BMS represents a seri-
Indeed, contact allergic reactions in the ous disorder about which the clinician is not
mouth themselves are rare.7oThis rarity has telling the patient. Patients who say that they
been attributed to rapid absorption and dis- think that part of their mouth looks abnormal
persal of potential allergens through the mu- have almost certainly been examining it in
cosa and to dilution and removal of potential the mirror and think they notice pathology.
allergens by saliva.45 Repeated self-examination is almost pathog-
Some reports of allergy to denture materi- nomonic of cancerphobia.
BURNING MOUTH SYNDROME 347

The Climacteric when assessing patients’ psychological status


to be objective, and many psychological pro-
In 1946, Ziskin and M o ~ l t o n ’described
~~ formas are available to allow the clinician or
the typical BMS patient as a postmenopausal psychologist to do this. Some proformas, such
woman unduly emotionally disturbed by her as the Hospital Anxiety and Depression scale,
oral condition and complaining of ner- are quick and simple to complete and have
vousness, depression, hopelessness, and in- been validated and applied to BMS patients2O
somnia, or alluding to significant cancerpho- One such study emphasized that anxiety was
bia. They also stated that younger female much more common than depression. Others
patients rendered sterile by oophorectomy also have noted hypochondriacal tendencies
also could be affected. It was claimed that (i.e., anxiety) in their 179 The
lack of female sex hormones produced histo- sources of anxiety are many and various, but
logic changes (progressive atrophy) in the include ”dissatisfaction with life.” It is for
oral and vaginal epithelium.lZ2Primate stud- this reason that, when evaluating patients
ies reported reversal of histologic changes in with BMS, a home circumstances score is
the oral mucosa of oophorectomized mon- used because it is a nonconfrontational means
k e y ~ ; and
~ ~ Ziskin’” repeated these studies of allowing patients to verbalize their con-
in women. The same group and others cerns and qualify them.
showed that although estrogen administra- Personality traits in BMS patients have
tion produced histologic changes in the oral been assessed using Cattell’s 16PF Question-
mucosa, it did not relieve symptoms of BMS. naire.86 This questionnaire is fairly lengthy
Other studies claimed benefit from hor- and requires specialist interpretation. It can,
mone replacement therapy alone or in combi- however, highlight personality traits in BMS
nation with vitamin B complex therapy.”O as shown in Figures 3 and 4. Such individuals
Some authors considered that, although the tend to be anxious, somewhat introverted and
oral symptoms may be related to the climac- self-reliant, but with low self-esteem. Others
teric, they were psychological in origin.71 have claimed that although BMS patients ap-
Menopause clinics have been used to inves- pear superficially kind and sensitive, they ac-
tigate oral symptoms that such patients may tually show suppressed hostility.18
have. In one study, 20% of patients had some Schoenberg128considered depression to be
oral symptoms, but not all had BMS.12 The a feature of BMS, but patients in that study
prevalence of oral symptoms at the climac- were excluded if any organic factor was
teric, including a burning sensation, is found. A degree of dependency and ”symp-
high.150,170 Hormone replacement therapy was tom formation” can develop in BMS pa-
of doubtful benefit, suggesting that estrogen tient~:~and in managing them, such patients
deficiency is an uncommon cause of BMS.& should receive attentive listening. Verbal ex-
Some authors have attempted to identify oral pression was encouraged, as well as offering
nuclear estrogen receptors immunohisto- appropriate explanations to social difficul-
chemically in an attempt to identify patients ties.31,83 Additional studies, this time using a
likely to benefit from hormone replacement psychological proforma, highlighted a preoc-
The present situation is that there cupation with ~ o s s . ~ ~ ~
is no proven benefit of hormone replacement This range of psychological problems that
therapy in BMS. some BMS patients have emphasizes the need
for the clinician to adopt a sympathetic ap-
proach with reassurance and avoidance of
Psychological Factors excessive treatment.12,56, 78, lo2,lo3 It is still not
clear if psychological aspects of BMS are
Psychological factors of importance in BMS cause or effect, and indeed, some patients
include simple fear of personality may have a chronic pain personality.6oState
disorders,s6 uncommon depre~sion,’~~ and and trait anxiety are elevated in BMS pa-
most significantly chronic anxietyg0or emo- t i e n t ~ with
, ~ ~ figures
~ varying between 44YOZ4
tional in~tability.~~ Emotional instability is and 62%.86 In the latter study, type 2 BMS
present in approximately 50% of type 3 BMS patients were more severely psychologically
patients.97Such patients can respond quickly disabled than type 1 BMS patients, and anxi-
to a stressful situation by developing symp- ety seemed the most recalcitrant obstacle to
toms of BMS, which then resolve when the cure. Overall, in management there is a re-
situation reverts to normal. It is important quirement for an objective assessment of psy-
348 LAMEY

A
-
X

-
5.1
B 4.9
C 3.9* Unstable

E 4.3

F 4.3' > Serious


G 6.6* * Precise
H 5.2

6 1
c 4.7
L!L 4.3' 4 Trusting

M 4.7
N 6.y * Shrewd
0 6.3
Q1 4.3* * Conservative
Q2 7.3 * Independent
Q3 5.2
6 .*5* * Tense
Q4
-
3 4 5 6 7 8
Stens

Figure 3. Mean profile recorded by Cattell's 16PF Questionnaire


Form C for BMS patients (n = 47) compared with age general
population. = P <0.001, ** = P <0.01

dormal Abnorma
4.9 5.2

5.3 4.5
5.2 3.1 Unstable

5.3 5.0
4.7 4.0

6.3 6.7
5.4 5.1
4.5 4.8
4.3 4.2

4.5 4.8
6.1 6.8
5.2 7.0 -Apprehensive
4.1 4.4
7.1 7.8
6.2 4.5 ICasual

4.6 7.7 - Tense

Stens

Figure 4. Mean profile recorded by Cattell's 16PF Questionnaire Form


C for BMS patients with (0-c [n = 281) and without (0-0 [n = 191)
an abnormal psychological profile.
BURNING MOUTH SYNDROME 349

chological status as well as a regimen to cater a perceptual deficit unrelated to pathophysio-


to psychological as well as medical and den- logic mechanisms in BMS. The authors con-
tal needs. Clearly life events are important in cluded that this alteration in sensory function
BMS 86, 149 but it has been claimed may be more plausible than a psychological
recently that BMS symptoms do not necessar- explanation of BMS.
ily correlate with stressful life events.86 When taste accompany BMS,6I
it has been stated that treatment is unsatisfac-
tory, but that depression frequently accompa-
Drug-related Burning Mouth nies chemosensory distortion.l12It is not clear
Syndrome from these studies if candidal carriage had
been sought or treated. It is common for taste
The only drugs of significance in appar- abnormalities to be due to candidal species,
ently precipitating BMS are angiotensin-con- even in the absence of clinical changes.
verting enzyme (ACE) inhibitor^.'^^ Reported Sensory changes of BMS have themselves
cases refer to scalded mouth . syndrome formed the basis of attempts to objectively
(SMS), but they almost certainly mean BMS. diagnose the condition.27Thermoesthesiome-
The three drugs involved are all ACE inhibi- try, which, it is claimed, accurately detects
tors and are lisinopril, captopril, and enala- surface temperature, showed that in BMS the
pril. Although the mechanism of BMS induc- surface mucosal temperature was lower than
tion by ACE inhibitors is not known, it does in control subjects, and this was attributed to
appear dose related and subsides on reduc- reduced blood circulation in the area. The
tion or discontinuation of therapy. diagnostic benefit of this test is still to be
subject to independent study.

Relationship to Systemic Disease


NOVEL TREATMENT OPTIONS IN
BMS may involve the complaint of burning BURNING MOUTH SYNDROME
at other body sites and as such may be a
Two recent publications have suggested
systemic disorder.50It has also, however, to be
novel ways of treating BMS. These reports are
borne in mind that accompanying psychiatric
clearly only initial observations but warrant
problems can produce somatization and re-
inclusion in any free-thinking academic com-
sult in multiple perceived somatic problems.
munity.
Only rarely is BMS a manifestation of un-
One study evaluated the use of soft laser
derlying organic disease. Two recent reports,
therapy in patients with BMS.27 Forty
however, have associated the complaint of
”stomatopyrosis” and ”stomatodiniae” pa-
BMS with acoustic n e ~ r o m aand ~ ~ temporal
tients underwent 5 days of therapy for 3 min-
arteritis51 (also known as giant cell arteritis).
utes daily on 1 cm2 of oral tissue. The re-
In the case of temporal arteritis, which affects
ported response, was complete healing by
the same age group as BMSY4 recognition of
which one assumes the patients were asymp-
the underlying disease is important because
tomatic. No follow-up data were presented.
permanent blindness is a serious complica-
The second novel treatment involves the
t i ~ n A. ~useful
~ clinical clue to the potential
use of c a ~ s a i c i n Capsaicin
.~~ is a neurotoxin
for a patient having giant cell arteritis is that
that destroys a major class of nociceptors,
they usually feel systemically unwell at tlie
primarily C polymodal and some A delta noc-
time of presentation.
iceptors. Although a mixed group of ”oral
neuropathic pain” patients were described,
some would appear to have BMS. Further
Associated Sensory Changes
data on this treatment are required as is de-
tailed information concerning patient investi-
Several groups of investigators have stud-
gation.
ied sensory changes including taste in BMS
patients. In one study, sensory and pain
thresholds to brief argon laser stimulation MANAGEMENT PROTOCOL
showed abnormal prepain perception and
disturbances in the perception of nonnocicep- This protocol is based on scientific evalua-
tive and nociceptive thermal stimuli in pain- tion of the known precipitating factors in
affected and normal regions.*41This suggests BMS and is what I follow routinely.
350 LAMEY

1. Hematinic assay of hemoglobin, ferri- such as tongue thrusting or tooth


tin, vitamin BIZ, and corrected whole clenching. It can be difficult for patients
blood folate. Deficiencies detected need to refrain from such habits. Sometimes
investigation for cause prior to supple- sugar-free chewing gum allows pa-
mentation. tients to break their habit.
2. Assay of vitamin B, and vitamin B,. If 10. In type 3 patients who are psychologi-
these are not available, then empirical cally normal, patch testing is appro-
treatment with vitamin B, (300 mg priate. Once identified, avoidance of
daily) and vitamin B6 (50 mg every 8 the relevant allergen(s) usually resolves
hours). Both are prescribed for 4 weeks the complaint, but the patient should
or rarely for 8 weeks. Patients should realize that this may take several
discontinue all self-medication with vi- months.
tamins. In our hands this management protocol
3. Assay of blood glucose on a morning renders approximately 7@/0 of patients
fasted sample. If results are equivocal, asymptomatic or greatly reduces their burn-
a glucose tolerance test (75 g load) is ing score. This protocol fails 28% of patients,
performed with referral to a physician however, and indeed made 2% of patients
with an interest in diabetes. worse. The reason for this may lie in the
4. Measurement of stimulated parotid patients' home circumstances and chronic
flow rate using a Carlsson-Crittenden anxiety. We also should bear in mind that
cup and stimulated with 1 mL of 10% other factors,, possibly organic, which we do
citric acid. Flow of less than 0.7 mL/ not yet know about, also may be important.
min is low, and the patient may benefit
from saliva substitutes.
5. An oral rinse for Candida species. The PROGNOSIS
distinction between carriage and infec-
tion (without clinical signs) is vague, It has been stated that there is not a predict-
but in practice all BMS patients in able endpoint in the outcome of treating BMS
whom Candida is isolated should re- patients.57This is certainly the case if not all
ceive 4 weeks of topical antifungal ther- relevant investigations are undertaken. There
apy. Advice on a low carbohydrate diet can be few conditions more multifactorial
and denture hygiene also should be than BMS, and we all owe it to our patients
given, if appropriate. to manage them to the best of our ability
6. Reassure about cancerphobia. This may and take cognizance of our current scientific
need to be repeated at subsequent vis- knowledge. If all investigations are under-
its. taken, it would seem that approximately 70%
7. Objective psychological assessment. of BMS patients can be rendered asymptom-
For screening purposes the Hospital atic or nearly so by treatment.89Initially, the
Anxiety and Depression scale is useful follow-up period in that study was 18
and can be supplemented with others months, and longer follow-up data are
if required. Involvement of a clinical awaited. In a mixed group of patients re-
psychologist also can be helpful. If viewed 8 to 10 years after treatment, over
there is a clear psychological problem, 50% of patients still thought they needed
then appropriate therapy is needed. treatment and continued to be high consum-
Prothiaden (dothiepin), 75 mg nocte, is ers of healthcare resource^.'^^
our drug of choice because it combines In our present state of knowledge, we do
anxiolytic and antidepressant proper- not know the long-term prognosis for BMS
ties and is not addictive. The drug patients. Spontaneous remission has been
should be avoided in patients with car- claimed>12but this is not my experience. The
diac arrhythmias. application of statistic methods to predict out-
8. Assessment of denture status if rele- come has not proved usefu1.'07,lZ1
vant. The main features of note are re-
stricted tongue space, lack of freeway
space, and underextended denture CONCLUSION
bases. Obvious design features need to
be corrected. Our knowledge of BMS has advanced enor-
9. Inquire about parafunctional habits mously in the last 10 years. These advances
BURNING MOUTH SYNDROME 351

have arisen from carefully controlled pro- 19. Brody HA, Prendergast JJ, Silverman S The rela-
spective studies aimed at addressing outcome tionship between oral symptoms, insulin release,
and glucose intolerance. Oral Surg Oral Med Oral
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sued. It is also important for our patients to 203511, 1969
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dentifrice: Report of a case. Journal of the Greater
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