JADA 2013;144(10):1135-1142

Defining and diagnosing burning mouth

syndrome: Perceptions of directors of North
American postgraduate oral medicine and
orofacial pain programs
What is BMS??

- burning pain in the tongue or other oral
mucous membrane persisting for at least four
months and associated with normal oral
mucosa and normal laboratory findings.

- IASP definition

Diagnostic criteria??
Burning sensation in tongue or other parts of
the oral mucosa,
usually bilateral
associated with
dysgeusia,
dry mouth
denture intolerance.

IASP diagnostic criteria
Definition by IHS
An intraoral burning sensation for which no
medical or dental cause can be found.

IHS further noted that pain may be confined
to the tongue with associated xerostomia,
paresthesia and altered taste.
IHS diagnostic criteria
Pain in the
mouth
Oral mucosa of
normal appearance
Exclusion of local
and systemic
diseases.
Persisting for
most of the day
These somewhat imprecise
definitions and descriptions
lead to challenges for the health care
practitioners when evaluating patients
with BMS
and the barriers to achieving an accurate
and reliable diagnosis.
BMS prevalence ?
0.7-5.0 % (of general population)

Prevalent in women in the 5
th
to 7
th

decade

Depends upon the methodology (clinical
assessment) and geographical setting of
the study
Usually manifests in a period between 3 yrs
before and 12 yrs after the onset of
menopause.

Rarely manifests before the age of 30 yrs.

Female:male = 3:1 - 16:1
Commonly occurs bilaterally,
May occur simultaneously at multiple sites.
Anterior
two-
thirds of
the
tongue,
Dorsum
and
lateral
borders
of
tongue
Anterior
aspect of
the hard
palate
Labial
mucosa
of the
lips.
Other symptoms!
Taste alterations
Described as a constant foul, bitter
or metallic taste sensation

which may be equally as disturbing
as or more disturbing than burning
pain itself.

Other symptoms!
Conflicting data regarding xerostomia in
BMS.

Nevertheless, qualitative changes in salivary
composition seen.
classification schemes

Classification by Lamey and Lewis (1996)
contains three subtypes according to
variations in pain intensity over 24 hours.
-Br Dent J 1989;167(11):384-389.

- Pain 2010;149(1): 27-32.
Gremeau-Richard reported two distinct
group based on the location of
neuropathic changes
Mediated by
peripheral or
central nervous system
Clin Neurophysiol 2012;123(1):71-77
Jaaskelainen proposed three distinct
subclasses based on
neuro-physiological,
psychophysical and
functional imaging studies.
A more pragmatic clinical approach is to
separate BMS into 2 distinct categories:

Primary BMS- lack of evidence of any other
disease

Secondary BMS- secondary to systemic
conditions such as anemia, diabetes, thyroid
disease or gastroesophageal reflux disorder.


Question??
What is clinicians understanding
of the diagnosis of BMS.

answer!
Opinions (via a confidence rating
scale [CRS]) from experienced health
care practitioners who treat BMS.

Answers rated with (CRS?)
CRS is numeric rating scale with anchors of
1 ( I am very uncertain) and
7 (I am very certain),
high confidence rating 6.0.

[enhance the certainty of responses and reduce
imprecision in the judged probabilities]

All responses measured according to a CRS
except those related to the participants clinical
experience.

respondents
rated their
confidence in
selection of
answer
answered
the
questions
answers
averaged to-
mean score
with CR and
SD
Same process used with an array of
oral health issues, and has led to
improved outcomes for those
conditions

such as outcomes assessment for
periodontal therapy,
referral criteria in pediatric dentistry
indications for use of radiography
AIM
Gather data
-about perceptions of a group of oral medicine
and orofacial pain training program directors
(United States and Canada)

-In terms of
definition of BMS
and the various factors and variables used in, and
assisting with, the determination of its definitive
diagnosis.
Methods

A structured questionnaire was designed
with input from four experienced clinicians in OM
and OFP (2 from each)
did not participate directly.

Most questions were open-ended to facilitate
variability of responses.
Broad approach captured the most information
without limiting answers
or leading him or her.

1 question- not open ended
The only question with
designated response categories
involved
- specific diagnostic testing for
conditions that excluded BMS.


Items addressed :

Respondents clinical experience as
it pertained to BMS

The most common characteristics
to be used in a definition of BMS

Items addressed :

Criteria necessary to make definitive
diagnosis
while addressing local, systemic and psychological
factors to be ruled out

Diagnostic tests used to support diagnosis

Perception regarding etiopathogenesis.
n = 20; OM = 10; OFP = 10.


Questionnare
1. Please list the mean number of patients with burning mouth
syndrom (BMS) seen every three months.

2. Please list the most common characteristic(s) to be used in a
definition of BMS.

3. Please list the criteria (including signs and symptoms)
necessary for a definitive diagnosis of BMS.

4. Please list the local factors needing to be ruled out before a
definitive diagnosis of BMS can be made.

5. Please list the systemic factors needing to be ruled out before
a definitive diagnosis of BMS can be made.
6. Please list the psychological factors needing to be ruled out
before a definitive diagnosis of BMS can be made.

7. What diagnostic tests are used to rule out local factors,
systemic factors or both to support a definitive diagnosis of
BMS? Please circle appropriate letter(s).
a. salivary flow rates
b. taste testing
c. serologic studies
d. soft-tissue biopsy
e. microbiological cultures
f. medication substitution

8. Please describe the etiopathogenesis of BMS
Open ended questions
Multiple responses from
each participant
Responses grouped into
broader categories
Discussion
Answer to question 1
A mean of 7.3 cases diagnosed in
each postgraduate program in
any given three-month period

And approx. 89% of those had been
managed.

Answer to question 2

Answer to question 3

Answer to questions 4,5,6

The majority of respondents indicated
measurement of salivary flow rates (n = 11)
as a diagnostic test to rule out a diagnosis of
BMS

mean [SD] CRS score, 6.2 [0.60],
95 percent confidence interval [CI], 5.82-6.54

Answer to question 7






n = 8; mean
[SD] CRS
score, 6.1
[0.64]; 95
percent CI,
5.69-6.57
Serologic
studies
n = 7; mean [SD]
CRS score, 6.1
[0.69]; 95 percent
CI, 5.63-6.65
Medication
substitution
n = 5; mean
[SD] CRS
score, 6.6
[0.55]; 95
percent CI,
6.12-7.08
Microbiologi-
cal cultures
n = 3; mean
[SD] CRS
score, 6.3
[0.58];
95 percent
CI, 5.68-6.98
Soft-tissue
biopsy
n =2;
mean
[SD] CRS
score,
6.0 [0.0]
taste
testing
Answer to question 8
Participants displayed uncertainty in their responses when
defining the etiopathogenesis for BMS as idiopathic or unknown
or psychological or psychosocial
DISCUSSION

Participants displayed overall confidence in
their responses and consistency

with the current literature

with respect to the most common characteristics
to be used in a definition of BMS
Notably, only 4 of the 13 program
directors
reported
burning sensation in the tongue and
chronic pain
as characteristics that should be used
in a definition of BMS.
Most of the respondents believed the
definition of BMS
should be more encompassing, including the
entire oral mucosa (n = 10) rather than being
restricted to the tongue region (n = 4).

Certainly, the presence of a burning
sensation in the entire oral mucosa as
compared with that only in the tongue is
more consistent with the literature.
Majority of respondents did not support the
inclusion of chronic pain (n = 4) in the definition
of BMS
because they considered that an acute onset of an
oral burning sensation would be an acceptable
criterion to be included in a definition of BMS.

Alternatively, the concept of chronic pain (pain
lasting longer than three months), was not
consistent with their perception of chronic pain,
which possibly involved a longer period (ex: 6
months).

It is of paramount importance for the health
care practitioner to understand that
BMS is a diagnosis supported by the nature of
the symptomatic complaint and
the exclusion of various local and systemic
factors.

Factors that need to be ruled out in the
diagnosis of BMS.

were reported with a moderate to high level
of confidence (range of mean CRS scores, 5.9-
6.8; 95 percent CI, 5.20-7.24).

The emphasis on the need to rule out fungal
infection may be due to the often associated
elevated prevalence of Candida species
reported in people with BMS
Fungal infection often associated with a
bitter or metallic taste (a symptom also
commonly reported by patients with BMS)
and clinical findings of erythema or
pseudomembranes often represents the
true source of burning pain.

Patients with these symptoms may report
increased pain on eating, likely because of
irritation of the mucosa.
The importance of ruling out the presence of
a fungal infection cannot be understated

and if such an infection is identified, a
diagnosis of secondary BMS would be
appropriate.
Before diagnosing BMS, thorough history and
examination is required involving the use of
adjunctive tests, imaging or both when
deemed necessary.

Certain diagnostic tests assist in ruling out
factors that may be responsible for burning
symptoms.
Eleven respondents endorsed the measurement of
salivary flow rates as an important diagnostic test to
determine salivary gland hypofunction or
dysfunction.

Although there is controversy among clinicians
regarding the role of salivary flow in BMS, this study
suggests that health care practitioners should
incorporate into their diagnostic armamentarium
and decision making processes a means of
objectively measuring salivary flow rates and
methods of ruling out salivary conditions before they
provide a definitive diagnosis of BMS.

Participants did not indicate the need for use
of imaging (dental or medical) in the diagnosis
of BMS.
This was most likely because participants were not
provided with the option of endorsing this
diagnostic test, having deliberately excluded it
from the designated response categories.

Three participants endorsed the use of soft-tissue
biopsy as an important diagnostic test to rule out a
diagnosis of BMS.

Although there is no established consensus
regarding the sampling of soft tissue (for example,
to rule out mucosal disease or to observe small fiber
axonal degeneration in the tongue) for a definitive
diagnosis of BMS,

It is possible these participants responded in this
manner because they suspected other soft-tissue
diseases causing burning sensation that could be
misconstrued as BMS.


Although the precise etio-pathology of BMS
still is elusive, the results are representative of
the current published literature regarding
BMS involving both central and peripheral
neuropathic mechanisms.

More than one-half of the total number (n =
33) of responses (54.5 %) supported the
concept of BMS having a neuropathic
etiopathogenesis.

Additionally, theories involving psychological
and psychosocial issues, and hormonal
factors reported in the literature also were
reported by the respondents.

Participants displayed a lower level of
confidence in their responses in this category.

This may be explained by the lack of strong
scientific evidence supporting any one
particular theory for the etiopathogenesis of
BMS (with the exception of the neuropathic
component).


Limitations
This study was limited by being based on self-reports
gathered via a mainly open-ended survey.

Open-ended format required categorization of
responses, which may have introduced
misclassification bias.

Number of people surveyed was limited

A formal assessment of each participants
knowledge regarding BMS was not
conducted and variability in participants
education was not controlled,
However these individuals had considerable clinical
education and experience in the diagnosis and
management of BMS and were active in caring for
patients with these symptoms.

Limitations
There may have been additional diagnostic
testing alternatives that were overlooked
and not included in the questionnaire.

Respondents were forced to choose a
response from a prescribed menu, other
possibilities were not elicited (no space
provided to record diagnostic tests not
present in the menu).

Limitations
Limitations
Temporal component regarding chronic pain
not investigated.
Formal sample-size calculation was not
performed.
Furthermore, the reliability of the CRS
technique, owing to its cross-sectional
nature, could not be demonstrated in this
study.
Conclusions

The findings in this study present an initial
exploration of the perceptions of program
directors of OM and OFP postgraduate
programs in North America regarding
diagnostic paradigms, clinical presentations
and etiologic and pathophysiological theories
regarding BMS.

Similarities were observed among
respondents who had a high degree of
confidence regarding variables associated
with the diagnosis of BMS, such as
neuropathic etiopathogenesis and
objective assessment of salivary flow.
Conclusions


This exercise will lead to development of a
comprehensive consensus statement that
expands the current definitions of BMS
described earlier
Conclusions

J Cutan Med Surg. 2014 May-Jun;18(3):174-9.

OBJECTIVE: To determine the clinical utility of patch testing in
patients with BMS.

METHODS: Retrospectively reviewed the charts of patients
diagnosed with BMS who had patch testing performed between
January 1, 2008, and July 31, 2012.

RESULTS: 132 consented to patch testing; 89 (67%) had allergic
patch test reactions. Of the patients with positive results, 66 (74%)
had results that were deemed to have possible relevance. The most
common allergens detected were nickel sulfate 2.5%, dodecyl gallate
0.3%, octyl gallate 0.3%, fragrance mix 8%, benzoyl peroxide 1%, and
cinnamic alcohol 1%.

CONCLUSIONS: Contact allergy may be an etiologic factor in some
patients with BMS. Patch testing is a useful investigation for BMS
patients.

Arq Neuropsiquiatr. 2014 Feb;72(2):91-8. doi: 10.1590/0004-282X20130218.

OBJECTIVE:To assess the efficacy of anti-xerostomic topical medication
(urea 10%) in patients with burning mouth syndrome (BMS).

METHOD:T 38 subjects diagnosed with BMS according to the IASP guidelines
were randomized to either placebo (5% sodium carboxymethylcellulose,
0.15% methyl paraben, and 10% glycerol in distilled water ) or treatment
(urea 10%) to be applied to the oral cavity 3-4 times per day for 3 months. The
patients were evaluated before and after treatment with the following
instruments: the EDOF-HC protocol (Orofacial Pain Clinic - Hospital das
Clnicas), a xerostomia questionnaire, and quantitative sensory testing.

RESULTS: There were no differences in salivary flow or gustative, olfactory,
or sensory thresholds (P>0.05). Fifteen (60%) patients reported improvement
with the treatments (P=0.336).

CONCLUSION: there were no differences between groups, and both
exhibited an association between reported improvement and salivation.

J Orofac Pain. 2013 Fall;27(4):304-13. doi: 10.11607/jop.1109.
AIM: To examine sleep complaints in patients with burning mouth
syndrome (BMS) and the relationships between these disturbances, negative
mood, and pain.

METHODS: Fifty BMS patients were compared with an equal number of healthy
controls matched for age, sex, and educational level. The Pittsburgh Sleep
Quality Index (PSQI), the Epworth Sleepiness Scale (ESS), the Hamilton Rating
Scales for Depression (HAM-D) and Anxiety (HAM-A) were administered.
Descriptive statistics, including the Mann-Whitney U test and hierarchical
multiple linear regression analyses were used.

RESULTS: BMS patients had higher scores in all items of the PSQI and ESS than
the healthy controls (P < .001). In the BMS patients, a depressed mood and
anxiety correlated positively with sleep disturbances. The Pearson correlations
were 0.68 for PSQI vs HAM-D (P < .001) and 0.63 for PSQI vs HAM-A (P < .001).

CONCLUSION: BMS patients reported a greater degree of sleep disorders,
anxiety, and depression as compared with controls. Sleep disorders could
influence quality of life of BMS patients and could be a possible treatment
target.

Int J Med Sci. 2013 Oct 29;10(12):1784-9. doi: 10.7150/ijms.6327. eCollection
2013.

OBJECTIVE: To estimate signs and symptoms of Temporomandibular
Disorders (TMD) in patients with BMS and to investigate for the existence of
an association between BMS and TMD.

MATERIALS AND METHODS: Forty-four BMS patients were enrolled; BMS
subtype was established according to the classification of Lamey. After a
gnathological evaluation, according to the protocol of the European Academy
of Craniomandibular Disorders, patients were classified by TMD criteria. The
data were compared and analyzed using a chi-square test to describe the
existence of an association between BMS and TMD.

RESULTS: 65.9% BMS patients showed disorders classified as primary signs
and symptoms of TMD according to TMD criteria, and 72.7% showed
parafunctional habits. The chi-square test revealed a statistically significant
association (p = 0.035) between BMS and TMD.

CONCLUSION: The data suggest that there is a possible relationship not yet
well understood between BMS and TMD, may be for neurophatic alterations
assumed for BMS that could be also engaged in TMD pathogenesis.

Mayo Clin Proc. 2014 Aug 28. pii: S0025-
6196(14)00540-0

OBJECTIVE: To calculate the incidence of BMS in
Olmsted County, Minnesota, from 2000
through 2010.

PATIENTS AND METHODS: By using the medical
record linkage system of the Rochester
Epidemiology Project, newly diagnosed cases of
BMS from January 1, 2000, through December 31,
2010 were identified. Diagnoses were confirmed
through the presence of burning pain symptoms
without associated clinical signs.
RESULTS: In total, 169 incident cases were identified,
representing an annual incidence of BMS of 11.4 per
100,000. Postmenopausal women aged 50 to 89 years had
the highest incidence of the disease, with the maximal
rate observed in women aged 70 to 79 years (70.3 per
100,000 ).

Limitations: participants were predominantly white,
which is a study limitation. In addition, diagnostic criteria
for identifying BMS may not apply for all situations
because no diagnostic criteria are universally recognized
for identifying BMS.

CONCLUSION: First population-based incidence study of
BMS. BMS is an uncommon disease highly associated
with female sex and advancing age.

J Formos Med Assoc. 2013 Jun;112(6):319-25. doi: 10.1016/j.jfma.2012.02.022. Epub 2012 Jun 12.

OBJECTIVE: whether there is an intimate association of the deficiency of hemoglobin
(Hb), iron, vitamin B12, or folic acid; high blood homocysteine level; and serum gastric
parietal cell antibody (GPCA) positivity with BMS.

METHODS: Blood Hb, iron, vitamin B12, folic acid, and homocysteine concentrations
and the serum GPCA level were measured in 399 BMS patients and compared with the
corresponding levels in 399 age- and sex-matched healthy control individuals.

RESULTS: 89 (22.3%), 81 (20.3%), 10 (2.5%), and six (1.5%) BMS patients had
deficiencies of Hb (men: <13 g/dL, women: <12 g/dL), iron (<60 g/dL), vitamin B12
(<200 pg/mL), and folic acid (<4 ng/mL), respectively. Moreover, 89 (22.3%) BMS patients
had abnormally high blood homocysteine level and 53 (13.3%) had serum GPCA
positivity. BMS patients had a significantly higher frequency of Hb, iron, or vitamin B12
deficiency; of abnormally elevated blood homocysteine level; or of serum GPCA
positivity than the healthy control group (all p < 0.001 except for vitamin B12 deficiency,
for which p = 0.004). However, no significant difference in frequency of folic acid
deficiency (p = 0.129) was found between BMS patients and healthy control individuals.
CONCLUSION: there is a significant association of deficiency of Hb, iron, and vitamin
B12; abnormally high blood homocysteine level; and serum GPCA positivity with BMS.


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