DEFINING AND DIAGNOSING BURNING MOUTH SYNDROME.pdf

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2013;144(10):1135-1142  JADA Czyscon, Carl K. Cramer and Joel Epstein Gary D. Klasser, Andres Pinto, Jonathan M. orofacial pain programs American postgraduate oral medicine and syndrome: Perceptions of directors of North Defining and diagnosing burning mouth October 7, 2013):  online at jada.ada.org ( this information is curr ent as of The following resources related to this article are available http://jada.ada.org/content/144/10/1135 can be found in the online version of this article at: including high-resolution figures, Updated information and services http://jada.ada.org/content/144/10/1135/#BIBL , 6 of which can be accessed free: 63 articles This article cites http://www.ada.org/990.aspx at: permission to reproduce this article in whole or in part can be found of this article or about reprints Information about obtaining Association. republication strictly prohibited without prior written permission of the American Dental Copyright © 2013 American Dental Association. All rights reserved. Reproduction or   o n  O  c  t   o  b  e r  7  , 2  0 1  3  j   a  d  a  .  a  d  a  .  o r  g D  o  w n l   o  a  d  e  d f  r  o m   o n  O  c  t   o  b  e r  7  , 2  0 1  3  j   a  d  a  .  a  d  a  .  o r  g D  o  w n l   o  a  d  e  d f  r  o m   o n  O  c  t   o  b  e r  7  , 2  0 1  3  j   a  d  a  .  a  d  a  .  o r  g D  o  w n l   o  a  d  e  d f  r  o m   o n  O  c  t   o  b  e r  7  , 2  0 1  3  j   a  d  a  .  a  d  a  .  o r  g D  o  w n l   o  a  d  e  d f  r  o m   o n  O  c  t   o  b  e r  7  , 2  0 1  3  j   a  d  a  .  a  d  a  .  o r  g D  o  w n l   o  a  d  e  d f  r  o m   o n  O  c  t   o  b  e r  7  , 2  0 1  3  j   a  d  a  .  a  d  a  .  o r  g D  o  w n l   o  a  d  e  d f  r  o m   o n  O  c  t   o  b  e r  7  , 2  0 1  3  j   a  d  a  .  a  d  a  .  o r  g D  o  w n l   o  a  d  e  d f  r  o m   o n  O  c  t   o  b  e r  7  , 2  0 1  3  j   a  d  a  .  a  d  a  .  o r  g D  o  w n l   o  a  d  e  d f  r  o m   o n  O  c  t   o  b  e r  7  , 2  0 1  3  j   a  d  a  .  a  d  a  .  o r  g D  o  w n l   o  a  d  e  d f  r  o m  

Transcript of DEFINING AND DIAGNOSING BURNING MOUTH SYNDROME.pdf

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2013;144(10):1135-1142 JADA

Czyscon, Carl K. Cramer and Joel EpsteinGary D. Klasser, Andres Pinto, Jonathan M.

orofacial pain programsAmerican postgraduate oral medicine andsyndrome: Perceptions of directors of NorthDefining and diagnosing burning mouth

October 7, 2013): online at jada.ada.org ( this information is current as of The following resources related to this article are available

http://jada.ada.org/content/144/10/1135can be found in the online version of this article at:

including high-resolution figures,Updated information and services

http://jada.ada.org/content/144/10/1135/#BIBL, 6 of which can be accessed free:63 articlesThis article cites

http://www.ada.org/990.aspxat:permission to reproduce this article in whole or in part can be found

of this article or aboutreprintsInformation about obtaining

Association.republication strictly prohibited without prior written permission of the American Dental

Copyright © 2013 American Dental Association. All rights reserved. Reproduction or

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ABSTRACTBackgrond. The authors conducted a study to ascertain par-

ticipants’ perceptions o and confdence in their responses

regarding the defnition and diagnosis o burning mouth

syndrome (BMS).

Mehods. The authors developed an eight-question questionaire

with input rom several experienced clinicians in the felds o oral

medicine (OM) and oroacial pain (OFP) and sent it to directors

(n = 20; OM = 10; OFP = 10) o accredited postgraduate training 

programs in North America. They used descriptive statistics to

analyze the results.Resls. The response rate was 65 percent (n = 13; OM = 6;

OFP = 7). Participants reported a mean o 7.3 cases o BMS in

any given three-month period, with 89 percent o these cases

managed within the programs. They identifed, with a high

degree o confdence, overall criteria or establishing a defnition

and defnitive diagnosis o BMS.

Conclsions. There were multiple similarities among partici-

pants’ responses regarding the elements to be included in the

defnition and diagnosis o BMS.

Pracical Implicaions. These data provide inormation on

current status o defnitions and diagnostic guidelines and may

assist in development o uture consensus statements on BMSthat incorporate additional geographical representation and ap-

propriate methodology.

Key Words. Burning mouth syndrome; diagnosis; respondents’

perceptions; confdence rating scale.

 JADA 2013;144(10):1135-1142.

Dr. Klasser is an associate professor, Division of Diagnostic Sciences, School of Dentistry, Louisiana State University Health Sciences Center, 1100 FloridaAve., Box 140, New Orleans, La. 70119, e-mail [email protected]. Address reprint requests to Dr. Klasser.Dr. Pinto is an associate professor, University Hospitals Case Medical Center and Department of Oral and Maxillofacial Medicine and Diagnostic Sciences,School of Dental Medicine, Case Western Reserve University, Cleveland.Mr. Czyscon is a third-year student, College of Dentistry, University of Illinois at Chicago.Dr. Cramer was a clinical assistant professor, Department of Oral Biological and Medical Sciences, Faculty of Dentistry, University of British Columbia,Vancouver, British Columbia, Canada, when this article was written. He now is deceased.Dr. Epstein is the director, Oral Medicine, and an adjunct professor, Division of Otolaryngology and Head and Neck Surgery, City of Hope, Duarte, Calif.

The International Associa-tion or the Study o Pain1 (IASP) deines burning mouth syndrome (BMS) as a

burning pain in the tongue or otheroral mucous membrane persisting or at least our months and asso-ciated with normal oral mucosaand normal laboratory indings.2,3 The IASP diagnostic criteria are asollows: burning sensation in the

tongue or other parts o the oral mu-cosa, usually bilateral and associa-ted with dysgeusia, dry mouth anddenture intolerance. The Interna-tional Headache Society4 (IHS) de-scribed BMS as an intraoral burn-ing sensation or which no medicalor dental cause can be ound. TheIHS urther noted that pain maybe conined to the tongue (gloss-odynia) with associated symptomsthat include subjective dryness o the mouth (xerostomia), paresthesiaand altered taste. The IHS providedthe ollowing diagnostic criteria orBMS: pain in the mouth presentdaily and persisting or most o theday, oral mucosa o normal appear-ance and exclusion o local and

Defining and diagnosing burning mouthsyndrome

Perceptions of directors of North American postgraduateoral medicine and orofacial pain programs

Gary D. Klasser, DMD, Cer. Orofacial Pain; Andres Pino, DMD, MPH; Jonahan M. Czyscon,BSc; Carl K. Cramer, DMD, MSc; Joel Epsein, DMD, MSD, Dip ABOM, FRCD(C), FDS RCS(Edin)

Copyright © 2013 American Dental Association. All Rights Reserved.

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 1136 JADA 144(10) http://jada.ada.org October 2013

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tions such as anemia, diabetes, thyroid diseaseor gastroesophageal relux disorder. Thereore,the clinician bases a diagnosis o BMS on clini-cal presentation and on the exclusion o localand systemic actors.

To date, there is a lack o qualitative andquantitative analyses regarding clinicians’

understanding o the diagnosis o BMS. We ap-proached these limitations by soliciting opin-ions (via a conidence rating scale [CRS]) romexperienced health care practitioners who treatBMS. This technique engages the resourceso all participants and results in an enhanceddecision-making ability among members o thegroup with regard to resolution o the clinicalproblem addressed.29 This process has beenused with an array o oral health issues, includ-ing decision support or diagnosis, and has ledto improved outcomes or the conditions under

investigation (such as outcomes assessmentor periodontal therapy, reerral criteria inpediatric dentistry and indications or use o radiography).30

Our aim in this study was to gather dataabout the perceptions o a group o oral medicineand oroacial pain training program directorsrom the United States and Canada in terms o the deinition o BMS and the various actorsand variables used in, and assisting with, thedetermination o its deinitive diagnosis.

MEtHODS

We designed a structured questionnaire withinput rom our experienced clinicians in oralmedicine and oroacial pain (two rom eachield) who did not participate in the studydirectly. (The study protocol was approved byan accredited institutional review board.) Mostquestions were open-ended to acilitate variabil-ity o responses. This broad approach capturedthe most inormation regarding the respondent’sopinion without limiting answers or leading him or her. The only question with designatedresponse categories involved speciic diagnostictesting or conditions associated with oral burn-ing sensation that excluded BMS. We derivedthese categories rom the current literature onBMS and the knowledge o the our experiencedclinicians.

The questionnaire (Figure) contained eightquestions pertaining to the diagnosis o BMS,

systemic diseases. In light o these somewhatimprecise deinitions and descriptions, it is easyto understand the challenge acing health carepractitioners when evaluating patients withBMS and the barriers to achieving an accurateand reliable diagnosis.

The prevalence o BMS is reported to be be-

tween 0.7 percent and 5.0 percent o the generalpopulation, depending on the methodology (sur-vey or clinical assessment) being used in andthe geographical setting o the study.5-8 BMS isreported most commonly by women in the ithto seventh decade3,9,10 and usually maniests in aperiod between three years beore and 12 yearsater the onset o menopause.3 It rarely mani-ests beore the age o 30 years.11,12 Investigatorsreport emale-to-male ratios ranging rom 3:1 to16:1.3,8,13-15 The pain commonly occurs bilaterally,involving the anterior two-thirds o the tongue,

ollowed by the dorsum and lateral borderso the tongue, the anterior aspect o the hardpalate and the labial mucosa o the lips. Theburning pain also may occur simultaneously atmultiple sites.3,11,14-18 Other symptoms that mani-est with the burning complaint include tastealterations,3,19 oten described as the presenceo a constant oul, bitter or metallic taste sensa-tion, which may be equally as disturbing as ormore disturbing than the oral burning pain it-sel.5 Intriguingly, there are conlicting objectivedata regarding decreased salivary low rates in

people with BMS.20-23

Nevertheless, indings insome studies have shown qualitative changes insalivary composition.21,23,24

Several classiication schemes have beenproposed to assist in the diagnosis o BMS. Onesuch classiication, proposed by Lamey25 andLamey and Lewis,26 contains three subtypes ac-cording to variations in pain intensity over 24hours. This classiication has not been validatedand does not appear to be widely accepted bythe scientiic community. Jaaskelainen27 pro-posed three distinct subclasses that were basedon neurophysiological, psychophysical and unc-tional imaging studies. Gremeau-Richard andcolleagues,28 using a double-masked crossoverdesign, reported there to be two distinct groupso people with BMS, who could be classiied onthe basis o the location o neuropathic changes(that is, mediated by the peripheral or the cen-tral nervous system). A more pragmatic clinicalapproach is to separate BMS into two distinctcategories: primary (essential or idiopathic)BMS, in which there is a lack o evidence o anyother disease, and secondary BMS, in which anoral burning sensation is identiied secondary to

other clinical abnormalities or to systemic condi-

ABBREVIATION KEY. BMS: Burning mouth syndrome.CRS: Conidence rating scale. IASP: International Association or the Study o Pain. IHS: InternationalHeadache Society. OFP: Oroacial pain. OM: Oral

medicine.

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time o questionnaire distribution, there were20 accredited postgraduate OM and OFP train-ing programs in the United States and Canada(n = 20; OM = 10; OFP = 10). The questionnairewas delivered either electronically or via postalservice to all training program directors in theUnited States and Canada. We made a secondattempt to deliver the questionnaire to thosewho did not respond to the initial inquiry. Wecalculated and recorded descriptive statistics or

the CRS ratings.

and the responses to thesequestions were analyzed in thisstudy. We addressed the ollow-ing items: assessment o therespondents’ clinical experienceas it pertained to BMS; the mostcommon characteristics to be

used in a deinition o BMS; thecriteria necessary (including signs and symptoms) to makea deinitive diagnosis o BMSwhile addressing local, systemicand psychological actors thatneed to be ruled out; diagnostictests used to support a diag-nosis o BMS; and perceptionregarding the etiopathogenesiso BMS. Because the majority o questions were in an open-ended

ormat, thereby allowing mul-tiple responses rom each par-ticipant, we grouped responsesaccording to consensus intobroader, more inclusive catego-ries or discussion purposes. Allresponses, with the exception o those related tothe participants’ clin-ical experience, weremeasured according to a CRS. The CRS is

a Likert-like numericrating scale with an-chors o 1 (meaning “Iam very uncertain”)and 7 (“I am very cer-tain”), and we includ-ed it to enhance thecertainty o (strengtho belie in) responsesand reduce ambigu-ity (imprecision inthe judged probabili-ties).30-32 Respondentsused this categoricalscale to rate their lev-el o conidence in their selection o each option,and we averaged all answers to produce a meanscore with appropriate conidence intervals andstandard deviations. We considered a high coni-dence rating as a 6.0 or greater.

We obtained inormation about directors o postgraduate oral medicine (OM) and oroacialpain (OFP) programs through our personalknowledge, as well as through consulting withthe American Academy o Oral Medicine and

the American Academy o Oroacial Pain. At the

tABLE 1

Characteristics respondents most commonlychose to be used in a definition of burning mouthsyndrome.CHARACtERIStIC NO. OF

RESPONDENtS(PERCENtAGE OF

tHE tOtAL*)

MEAN (SD†)CRS‡ SCORE

95 PERCENtCONFIDENCEINtERVAL

Brning in he Oral Mcosa 10 (77) 6.1 (1.10) 5.42-6.78

 Absence of Clinical Findings 7 (54) 6.1 (1.07) 5.35-6.93

Brning in he tonge 4 (31) 6.3 (0.96) 5.31-7.19

Chronic Pain (Brning PainLasing Longer than threeMonhs)

4 (31) 6.8 (0.50) 6.26-7.24

* Total number o respondents = 13.† SD: Standard deviation.

‡ CRS: Confdence rating scale.

Please list the mean number of patients with burning mouth syndrome

(BMS) seen every three months.

Please list the most common characteristic(s) to be used in a definition

of BMS.

Please list the criteria (including signs and symptoms) necessary for a

definitive diagnosis of BMS.

Please list the local factors needing to be ruled out before a definitive

diagnosis of BMS can be made.

Please list the systemic factors needing to be ruled out before a definitive

diagnosis of BMS can be made.

Please list the psychological factors needing to be ruled out before a

definitive diagnosis of BMS can be made.

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

Please describe the etiopathogenesis of BMS.

1

2

3

4

5

6

7

8

Figre. Questionnaire used in the study.

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6.6 [0.55]; 95 percent CI, 6.12-7.08), sot-tissuebiopsy (n = 3; mean [SD] CRS score, 6.3 [0.58];95 percent CI, 5.68-6.98) and taste testing (n =2; mean [SD] CRS score, 6.0 [0.0]). Table 4 il-lustrates that the majority o respondents (54.5percent) associated BMS with a neuropathicetiopathogenesis (Figure, question 8). Interest-ingly, participants displayed uncertainty intheir responses when deining the etiopathogen-esis or BMS (Figure, question 8) as idiopathicor unknown or psychological or psychosocial(Table 4).

DISCuSSION

To our knowledge, this is the irst study inwhich investigators have garnered clinicians’perceptions and described their degree o conidence in their responses regarding thediagnostic criteria or BMS across emerging diagnostic disciplines such as oral medicineand oroacial pain. The results o our studysupport the assumption that the 13 postgradu-ate program directors (OM = 6; OFP = 7) whoresponded to our survey had experience in theclinical management o BMS (or instance, amean o 7.3 cases o BMS had been diagnosedin their clinics in any given three-month pe-riod), thereby justiying their participation asa sample o clinicians qualiied to render per-ceptions regarding this condition. Participantsdisplayed overall conidence in their responsesand consistency with the current literature1-4 with respect to the most common characteris-tics to be used in a deinition o BMS (Table 1).Notably, only our o the 13 program directorsreported a burning sensation in the tongue andchronic pain as characteristics that should be

used in a deinition o BMS. Perhaps many o 

RESuLtSThirteen (OM = 6; OFP = 7) o the 20 postgradu-ate directors responded, representing a responserate o 65 percent. The survey results showedthat a mean o 7.3 cases o BMS had beendiagnosed in each postgraduate program in anygiven three-month period (Figure, question 1),and approximately 89 percent o those cases hadbeen managed within the postgraduate clin-ics. Table 1 describes the characteristics thatrespondents most commonly indicated shouldbe used in a deinition o BMS (Figure, question

2). Table 2 describes the criteria respondents re-ported most requently as being necessary or adeinitive diagnosis o BMS (Figure, question 3).The CRS results (Tables 1 and 2) demonstratedan overall elevated degree o conidence (6.0 orgreater) among participants in their responsesto the question about the most common charac-teristics to be used in a deinition o BMS (Fig-ure, question 2) and to the question regarding the criteria necessary or a deinitive diagnosiso BMS (Figure, question 3), with the exceptiono xerostomia (Table 2).

Respondents reported various actors (Figure,questions 4-6) as needing to be ruled out beorethey could provide a deinitive diagnosis o BMS(Table 3). The majority o respondents indicatedmeasurement o salivary low rates (n = 11;mean [standard deviation {SD}] CRS score, 6.2[0.60], 95 percent conidence interval [CI], 5.82-6.54) as a diagnostic test to rule out a diagnosiso BMS (Figure, question 7). This was ollowedby serologic studies (n = 8; mean [SD] CRSscore, 6.1 [0.64]; 95 percent CI, 5.69-6.57), medi-cation substitution (n = 7; mean [SD] CRS score,6.1 [0.69]; 95 percent CI, 5.63-6.65), microbio-

logical cultures (n = 5; mean [SD] CRS score,

tABLE 2

Criteria respondents reported most frequently as necessaryfor a definitive diagnosis of burning mouth syndrome.CRItERION NO. OF

RESPONDENtS*MEAN (SD†)CRS‡ SCORE

95 PERCENtCONFIDENCEINtERVAL

Sbjecive Sensaion of Inraoral Alered Sensory Sympoms 10 6.2 (0.63) 5.81-6.59

 Absence of Mcosal Pahology or Abnormaliies 7 6.1 (0.69) 5.63-6.65

 Absence of Denal Eiology  4 6.0 (0.82) 5.20-6.80

Presence of Brning Pain 4 6.0 (0.82) 5.20-6.80

 Xerosomia (Dryness of he Moh) 4 5.3 (1.70)§ 3.58-6.92

 Absence of Salivary Gland Hypofncion, Dysfncionor Boh (Objecive Lack of Salivary Flow Measred

 According o Weigh or Volme)

3 6.3 (0.58) 5.68-6.98

* Total number o respondents = 13.† SD: Standard deviation.‡ CRS: Confdence rating scale.§ Criterion with a mean CRS score < 6.0.

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oral mucosa as compared with that only in thetongue region is more consistent with the clini-cal indings described in the literature.3,14,16-18 Itis possible the majority o respondents did not

endorse the inclusion o chronic pain (n = 4) in

the respondents believed the deinition o BMSshould be more encompassing, including theentire oral mucosa (n = 10) rather than being restricted to the tongue region (n = 4). Certainly,

the presence o a burning sensation in the entire

tABLE 3

Factors respondents indicated as needing to be ruled out before adefinitive diagnosis of burning mouth syndrome could be made.*FACtOR (tOtAL NO. OF RESPONSES) NO. OF RESPONSES

(PERCENtAGE OFtOtAL RESPONSES)

MEAN (SD†)CRS‡ SCORE

95 PERCENtCONFIDENCEINtERVAL

Local (n = 55)Oral mucosal lesions (lichen planus, benign migratoryglossitis, benign mucous membrane pemphigoid,pemphigus, oral ulcers, oral cancer, leukoplakia)

11 (20.0) 6.4 (0.67) 5.96-6.76

Microbial infection (fungal,§ viral or bacterial) 10 (18.2) 6.2 (0.63) 5.81-6.59

Dental issues (poorly fitting dental prostheses, mechanicalirritations, dental caries, periodontal or pulpal disease)

9 (16.4) 6.2 (0.44) 5.93-6.51

Salivary gland hypofunction/dysfunction 8 (14.5) 6.3 (0.71) 5.76-6.74

Contact allergy/mucosal contact irritants (contact allergyor allergy to dental materials, mouthwash, spices, foodingredients, additives or preservatives)

8 (14.5) 6.1 (0.35) 5.89-6.37

Parafunctional habits of tongue or lips 4 (7.3) 6.5 (0.58) 5.93-7.07

Other (nerve trauma or neuropathy, toxic environmentalexposure, taste disorder)

5 (9.1) 6.4 (0.55) 5.92-6.88

Sysemic (n = 31)

Autoimmune disorders (Sjögren syndrome, connectivetissue disease, systemic lupus erythematosus) 8 (25.8) 6.4 (0.52) 6.02-6.74

Vitamin or nutritional deficiencies 6 (19.4) 6.3 (0.52) 5.92-6.74

Endocrine disorders (diabetes, metabolic diseases) 6 (19.4) 6.3 (0.52) 5.92-6.74

Medication adverse effects 4 (12.9) 6.8 (0.50) 6.26-7.24

Other (hematologic disorder, gastrointestinal condition,cancer, psychopathology)

7 (22.6) 6.4 (0.79) 5.85-7.01

Psychological (n = 28)

Anxiety 11 (39.3) 6.0 (0.77) 5.54-6.46

Depression 9 (32.1) 5.9 (0.78) 5.38-6.40

Somatoform disorder 4 (14.3) 6.0 (0.82) 5.20-6.80

Other (psychosis, obsessive-compulsive disorder) 4 (14.3) 6.5 (0.58) 5.93-7.07

* Multiple responses were allowed rom a single participant.† SD: Standard deviation.‡ CRS: Confdence rating scale.§ There were six responses (each with a CRS score = 6.0) endorsing ungal inection as a local actor, which composed 60 percent

o the responses in the “microbial inection” category.

tABLE 4

Etiopathogenetic theories for burning mouth syndrome.NAtuRE OF HYPOtHEtICAL EtIOPAtHOGENESIS* NO. OF RESPONSES

(PERCENtAGE OFtOtAL RESPONSES

[N = 33])

MEAN (SD†)CRS‡ SCORE

95 PERCENtCONFIDENCEINtERVAL

Neropahic (Peripheral or Cenral Neropahiesor Sensiizaion)

18 (54.5) 6.1 (0.87) 5.66-6.46

Idiopahic or unknown 5 (15.2) 5.6 (1.10)§ 4.60-6.60

Psychological or Psychosocial 3 (9.1) 5.3 (0.58)§ 4.68-5.98

Hormonal 2 (6.1) 6.0 (0.0) NA¶

Oher (tase Dysfncion or Dysreglaion, NriionalDeficiency, Chronic ulceraion)

5 (15.2) 6.0 (0.0) NA

* Multiple responses were allowed rom a single participant.† SD: Standard deviation.‡ CRS: Confdence rating scale.§ Hypotheses with a mean CRS score < 6.0.¶ NA: Not applicable.

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symptoms managed accordingly or an inectiono this type.

Beore diagnosing BMS, the health care prac-titioner would be prudent to obtain a thoroughhistory and perorm a comprehensive exami-nation involving the use o adjunctive tests,imaging or both when deemed necessary. The

use o certain diagnostic tests assists in ruling out actors that may be responsible or the oralburning symptoms, thus distinguishing primaryrom secondary BMS.

Eleven respondents endorsed the measure-ment o salivary low rates as an importantdiagnostic test to determine salivary glandhypounction or dysunction. Although there iscontroversy among clinicians regarding the roleo salivary low in BMS, our data suggest thathealth care practitioners should incorporate intotheir diagnostic armamentarium and decision-

making processes a means o objectively meas-uring salivary low rates and methods o ruling out salivary conditions beore they provide a de-initive diagnosis o BMS. Participants did notindicate the need or use o imaging (dental ormedical) in the diagnosis o BMS. This was mostlikely because we did not provide participantswith the option o endorsing this diagnostic test,having deliberately excluded it rom the desig-nated response categories (Figure, question 7).There may have been additional diagnostic test-ing alternatives that we overlooked and did not

include in the questionnaire. Furthermore, ow-ing to the nature o the methodology, by whichwe orced respondents to choose a response roma prescribed menu, we were not able to elicitthese other possibilities (we provided no spacein which they could record diagnostic tests notpresent in the menu supplied), thereby intro-ducing the potential or bias to the responses. Itis interesting that three participants endorsedthe use o sot-tissue biopsy as an importantdiagnostic test to rule out a diagnosis o BMS. Although there is no established consensus re-garding the sampling o sot tissue (or example,to rule out mucosal disease or to observe small-iber axonal degeneration in the tongue) or adeinitive diagnosis o BMS, it is possible theseparticipants responded in this manner becausethey suspected other sot-tissue diseases’ (Table3) causing oral burning sensations that could bemisconstrued as BMS.

 Although the precise etiology and patho-physiology o BMS still is elusive (Table 4), theresults o this survey are representative o thecurrent published literature regarding BMSinvolving both central50-53 and peripheral10,54-56 

neuropathic mechanisms. More than one-hal 

the deinition o BMS because they consideredthat an acute onset o an oral burning sensationwould be an acceptable criterion to be includedin a deinition o BMS. Alternatively, they mayhave believed that the concept o chronic pain,as deined in our study (pain lasting longer thanthree months), was not consistent with their

perception o chronic pain, which possibly in-volved a longer period (or instance, six months).The nature o these responses may relect, inpart, the nature o the reerral network andthe delay in patients’ reerral to OM and OFPclinics and programs.33 We did not investigateurther the issue o the temporal componentregarding chronic pain in this study.

To assist the health care practitioner in pro-viding a deinitive diagnosis and to assist withconsistency among clinical trials, it is importantto standardize diagnostic criteria. Overall, par-

ticipants in this study were conident in theirresponses in this regard in identiying the keydiagnostic criteria, and they reported criteriasimilar to those published in the literature(Table 2) as being necessary or a deinitive di-agnosis o BMS.5,34-39

It is o paramount importance or the healthcare practitioner to understand that BMS is adiagnosis supported by the nature o the symp-tomatic complaint and the exclusion o variouslocal and systemic actors (Table 3). A detaileddiscussion o these actors is beyond the scope

o this article; however, reviews regarding themappear in the literature.36-41

The results o our study (Table 3) indicatethat participants’ responses were consistentwith the current literature regarding the ac-tors that need to be ruled out in the diagnosiso BMS. These were reported with a moderateto high level o conidence (range o mean CRSscores, 5.9-6.8; 95 percent CI, 5.20-7.24). Theemphasis on the need to rule out ungal inec-tion may be due to the oten associated elevatedprevalence o Candida species reported inpeople with BMS.42-47 The presence o a ungalinection—oten associated with a bitter or me-tallic taste (a symptom also commonly reportedby patients with BMS) and clinical indings o erythema (erythematous candidiasis) or pseudo-membranes (pseudomembranous candidiasis)on the oral mucosa—oten represents the truesource o oral burning pain.45 Patients withthese symptoms may report increased pain oneating, likely because o irritation o the muco-sa.48,49 The importance o ruling out the presenceo a ungal inection cannot be understated—and i such an inection is identiied, a diagnosis

o secondary BMS would be appropriate, with

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and management o BMS. We observed similari-ties among respondents who had a high degreeo conidence regarding variables associatedwith the diagnosis o BMS, such as neuropathicetiopathogenesis and objective assessment o salivary low. Global standardization o the de-ining criteria or BMS and necessary diagnostic

algorithms can be ormulated by convening leaders in the ield rom dierent geographicalregions. This worthwhile exercise, in addition toollowing strict methods or the development o a comprehensive consensus statement that ex-pands the current deinitions o BMS describedearlier in this article, certainly would aid in theadvancement o our knowledge o this conditionto the beneit o both health care practitionersand the patients they serve. n

Disclosure. None o the authors reported any disclosures.

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4. Lance JW, Olesen J, eds; International Headache Society. TheInternational Classiication o Headache Disorders. 2nd ed. Cepha-lalgia 2004;24(suppl 1):9-160.

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o the total number (n = 33) o responses (54.5percent) supported the concept o BMS’ having a neuropathic etiopathogenesis. Additionally,theories involving psychological and psycho-social issues25,57-63 and hormonal actors3,8,64,65 reported in the literature also were reported bythe respondents. Generally, in comparison with

other responses regarding the various topicsaddressed in the study, participants displayeda lower level o conidence in their responses inthis category. This may be explained by the lacko strong scientiic evidence supporting any oneparticular theory or the etiopathogenesis o BMS (perhaps with the exception o the neuro-pathic component).Limitations. This study was limited by be-

ing based on sel-reports gathered via a mainlyopen-ended survey. The open-ended ormatrequired categorization o responses by the

investigators, which may have introduced mis-classiication bias. Furthermore, because thenumber o people surveyed was limited by thenumber o accredited programs (that is, 20 pro-grams), the actual number o participants (n =13, providing a response rate o 65 percent) wassmall. Although we did not conduct a ormalassessment o each participant’s knowledge re-garding BMS and did not control or variabilityin participants’ education, these individuals hadconsiderable clinical education and experiencein the diagnosis and management o BMS and

were active in caring or patients with thesesymptoms. The CRS technique allowed us togauge their level o conidence in their respons-es. Involving a greater number o participantsby expanding the survey beyond North Americaor recruiting a greater number o experiencedclinicians in the disciplines o OM and OFPand applying nominal techniques or consensusbuilding would be o great beneit and shouldbe a consideration in uture studies. We did notperorm a ormal sample-size calculation, as ourintent in this study was to capture the universeo all training program directors in North Amer-ica. Furthermore, the reliability o the CRStechnique, owing to its cross-sectional nature,could not be demonstrated in this study.

CONCLuSIONS

The indings in this study present an initialexploration o the perceptions o program direc-tors o OM and OFP postgraduate programs inNorth America regarding diagnostic paradigms,clinical presentations and etiologic and patho-physiological theories regarding BMS. The par-ticipants were postgraduate program directors

who were clinicians experienced in the diagnosis

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