FOCUS ARTICLE A Unified Concept of Idiopathic Orofacial : Clinical Features

Alain Woda, DDS. D es S The main features of , stomatodynia, atypical Professor odontalgia, and masticatory muscle and temporomandibular joint (TMJ) disorders are compared in this article, which included a Paul Pionchon, DDS, PhD search of articles indexed in MEDLINE. The fact tbat their termi- Assistant Professor nology has been the subject of many debates can be considered a consequence of taxonomic difficulties and uncertainties. Laboratoire de Physiologie Oro-faciale Epidemiologic studies indicate marked female predominance for University of Auvergne U.F.R. d'Odontologie all types of idiopathic . There is also a difference in Clermont-Ferrand, France tbe age of maximal prevalence between masticatory muscie and TMJ disorders and the otber entities. The clinical presentations Correspondence to: display several symptoms in common. PaÍ7i is oral, perioral, or Prof. A. Woda facial and does not follow a nervous pathway. It has been present Laboratoire de Physiologie Oro-faciale for the last 4 to 6 months or has returned periodically in tbe same University of Auvergne form over a period of several months or years. The pain is contin- U.F.R. d'Odontologie uous, has no major paroxysmal character, and is present through- 1 I, Bd Charles de Caulle out all or part of the day. It is generally abseiet during sleep. 63000 Clermont-Ferrand, France Clinical, radiographie, or laboratory examination does not reveal Faxi04 73 43 64 12 any obvious organic cause of pain. There is also a frequent pres- E-mail: [email protected] ence of certain psychologic factors, personality traits, or life events. Based on these shared characteristics, a unified concept is proposed. Each of these entities belongs to a group of idiopathic orofacial pain and could be expressed in either the jaws, the buc- cal mucosa, the teeth, tbe masticatory muscles, or tbe TMJ. I ÜRÜI-AC PAIN 1999;13:172-184.

Key words: atypical facial pain, atypical odontalgia, , epidemiology, clinical presentation

ne of the most important trends in recent research in tem- poromandibular disorders has been Co attempt to define Oaccurately che different disease processes and to standard- ize a classification scheme. Mucb clinical research bas been under- taken to define eacb subgroup of masticatory muscle and tempo- romandibular joint (TMJ) disorders. This approach bas led to a new classification of 12 disease processes, whicb form tbe group of masticatory muscle and TMJ disorders. Each may be present independently or simultaneously in a single patienC. Diagnostic cri- teria for each subgroup of tbis classification have been developed and published, witb the primary goal Co standardize research efforts in tbis area.'-- As a result, knowledge of pain due to masti- catory muscie and TMJ disorders has greatly increased, but many related orofacial pain syndromes have been left unclassified. Tt is possible that an equal amount of knowledge could be gained by looking beyond the masticatory muscle and TMJ disorders to a definition of the other types of idiopathic orofacial pain.

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Atypical facial pain, atypical odontalgia, stoma- witb systemic disease or trauma, and several types todynia, and masticatory muscle and TMJ pain of or tension-type headacbes. Tbe term disorders display common clinical characteristics. is tbus employed as a "wastebasket" definition, Partly for tbis reason, a unified concept of idio- wbich can only be applied by elimination. What is pathic orofacial pain has already been suggested.^-'' more, certain otber subgroups that had not been In addition, several of tbe proposed etiologic identified at the time, such as TMJ disorders or mecbanisms could also be sbared; for example, cluster beadacbe, could also be included in tbis alteration in tbe balance of female steroid hor- group.'** The second diagnostic concept of atypical mones, modification by lesion and/or sensitization facial pain is of that of a defined set of characteris- of nerve fibers, or involvement of tbe sympathetic tics that aim to describe a relatively homogeneous nervous system or psycbologic factors, such as life subgroup of facial pain. Diagnosis thus becomes a events. Evidently, clinical and patbopbysiologic positive procedure rather than one accomplished differences exist, but tbey could result from the by ehmination. This article will consider atypical obvious but possibly misleading fact that tbey arise facial pain as a distinct group. from 5 different tissues: bone, rootb, , Atypical facial pain can be described as a TMJ, and masticatory muscles. It can be postu- chronic pain of unknown etiology that is felt con- lated that a unique disease would be differently tinuously throughout all or part of tbe day witbin expressed in different tissues. Reeently, the priority the bone or deep tissues of tbe orofacial region. given to anatomic criteria in tbe current pain clas- Otber expressions that are no longer used as syn- sification systems^"' bas been questioned** because onyms for atypical facial pain include atypical tbis may reclassify pain entities tbat sbare common facial ,'^ dental causalgia,'- or phantom clinical features and/or mecbanisms purely on an orofacial pain.'^ organ or tissue basis. Ir is useful therefore to review tbe clinical features and mechanisms of Epidemiology and Demography rhese types of persistent orofacial pain with regard ro classification for purposes of botb research and No evaluation has been made of rhe prevalence of treatment. atypical facial pain in the general population. Tbis article aims to bring togetber tbe terminol- Fxisting figures are drawn from clinical impres- ogy, epidemiology, and for sions or from studies of clinical populations. Its eacb of the idiopathic orofacial pain subgroups prevalence would seem ro fall between tbat for and to propose diagnostic criteria tbat will allow , which is very low, and that for comparison between each entity. for masticatory muscle and TMJ disorders, whicb is bigh. All the studies^''"^^ indicate a very higb preponderance of female sufferers in these popula- tions—between 3 and 10 females for every male Atypical Facial Pain affected. This may be tbe result of a truly bigher prevalence among women or perhaps a more fre- Definition and Terminology quently expressed treatment need in the female population. It is possible tbat men may be more Despite tbe position taken by botb the readily able to assimilate tbe pain into tbeir way of International Association for the Study of Pain life (see references in Fillingim and Maixner^^). (IASP)' and tbe International Headacbe Society Tbe average age affected is that of tbe post- (MS),* tbe term "atypical facial pain" contmues to menopausal period or above. The reported average be used by almost all autbors. Tbere is no defini- age on consultation was 52 years, witb extremes of tion for atypical facial pain, precisely because the 24 and S2 years.'^-'^ Tbe average age of onset was term was coined to cover all unexplained cases of 45 years.'' Thus, atypical facial pain concerns pain. In the Uterature tbis term is used to describe chiefly menopausal or older women. 2 totally different concepts. Tbe first is that described by the lASP^ and tbe IHS,^-^ wbich sug- gest that the term should be abandoned in favor of Description "otber and unspecified pain in the jaws" or by "facial pain not fulfilling otber crireria." Tbese As the definition of atypical facial pain was origi- terms would regroup all tbe intermediate clinical nally made only by elimination, its characteristics situations tbat do not fall into one of tbe well- were first described in compatison with tbose of defined categories, sucb as cluster headacbe, trigeminal neuralgia.'^'-""^^ Both are marked by trigeminal neuralgia, associated extremely intense pain tbat is usually localized to

Joumal of Orofacial Pain 173 Woda/Pionchon the mouth, jaws, or face. In reality, the pain char- dysfunction may also be presenr. Subjective acteristics are not comparable, and a differential impressions of warmtb or swelling of rbe mucosa diagnosis between them is rarely a problem.^^ The or soft tissues are frequent and are sometimes con- pain location, for example, is different. Atypical firmed clinically by the presence of erytbema or facial pain often is of a fluctuant nature, and it is edema.-^ These signs are usually discrete, and they felt deep within the tissues,^"* in the bone of the may be constant, or more often, undergo periods maxilla or mandible. Trigeminal neuralgia is most of spontaneous remission. Cbange, or an impres- often felt in the superficial tissues only and always sion of change, in salivary flow may also be noted. in the same locus. Recently, an area over the apices of the last 2 max- The timing of episodes of pain LS also different. illary molars has been described as being bypersen- Although in borh cases tbe pain is experienced sitive to pressure and bas been shown to display a only while the parient is awake, it is continuous in temperature rise.-^'^^ give little or no atypical facial pain.-' In atypical facial pam there relief. A transient but complete relief follows the is no true trigger zone, although the pain may be administration of to the affected intensified, or more rarely decreased, by functional edentulous area.'^ movement (for example by mastication or speech). A large proportion of patients report associated Episodes of increased pain are never paroxysmal, general symptoms, such as chronic, cervical, or as they are for trigeminal neuralgia or certain lumbar spinal pain; migraines; cutaneous pruritis; other neuropathic .^' irritable bowe! syndrome; or dysmenorrbea.' At the onset of the disease, pain is often felt Psychologic signs are acknowledged by all within a defined zone of the oral cavity or midface autbors, but debate surrounds the type of pathol- that has undergone some sort of trauma. The ogy concerned. , , intense stress, trauma may be accidental or therapeutic in nature or a distressing life event in tbe 6 months preced- (eg, endodontic treatment, , or ing the onset of pain have all been impli- sinus surgery)"^ and the initial recovery may be cated.^•''•'^'-'* It is also important to compare the difficult.'* The pain is described either as having incidence of psychologic problems within this pop- been aggravated by the injury or as having been ulation and that of the general public (see refer- initiated by it.'"*-'^ Sometimes the pain reappears ences in Feinmann and Harris-^). Most important, only after a period of months or years following the existence of a causal link between psychologic the original pain experience. With time, rhe pain factors and the disease is highly contentious.^'^*" It spreads out over a larger territory. It does not should also be noted that these patients are often respect any particular nervous pathway. In one cancerophobic. third of cases, pain becomes bilateral.'*--' Often These patients express a high level of demand many different dental or surgical treatments are for invasive treatment, "* which leads to the consul- attempted, with no results other than an increase tation of many health care professionals. in symptoms'^ and partial or total edentu- Pfaffenrath et al showed that patients had con- I 14 sulted an average of 7.5 professionals, in the fol- lousness. lowing descending order: dentist; general medical Pain intensity is described as moderate to intense practitioner; neurologist; ear, nose and throat sur- and is equivalent to that described for trigémina! geon; orthognathic and maxillofacial surgeon; psy- neuralgia. Recently experienced pain is described chiatrist; ophthalmologist; and dermatologist.'^ as the worst possible pain 3 or 4 times more often The edentulous region, which is so often the result than that of pain of masticatory muscles and TMJ of these consultations, then presents a ftinctional disorders.^*' To describe the qualitative aspect of problem for the patient, who is unable to cope their pain, patients often use rhe emotional terms with removable prostbeses and who complains of of the McGill Pain Questionnaire,-"^ such as problems with chewing. The impact on other "vicious" or "excruciating." Strangely, they often aspects of quality of life has not been studied, but evoke mechanical descriptors such as "tugging," it seems that capacity to work is only relatively "constriction," "tightening," "piercing," or slightly decreased.'^ "movement" inside the underlying bone,'^ The pain IS also often described as "burning."'^ Neurologic signs are not obvious, but there may Diagnostic Criteria be dysesthesia, , and/or .''^''^'^' Paresthesia is described in diverse terms, such as The first diagnostic criteria that were proposed in "numbness" or "pins and needles."'^'^' Signs and the literature probably lack specificity.''^ The crite- symptoms due to sympathetic or vascular ria suggested by the IHS^'^ have been questioned

174 Volume 13, Number3, 1999 Wods/Pionchon

by Pfaffenrath et al,'^ who noted that if these crite- "neurovascular odontalgia,"^'•^^•^^ and more ria are applied strictly then they must lead to a recently "neuropathic orofacial pain."^^ number of false negarives,'^ and they therefore proposed certain changes to improve the rare of Epidemiology and Demography inclusion and the sensitivity. Unfortunately, since the choice of their population of sufferers of atypi- The prevalence of atypical odontalgia in the gen- cal facial pain is not fully described, it is difficult eral population is unknown. However, knowledge to judge rhe validity of their conclusions. The pro- of the frequenr association between endodontic posed criteria for atypical facial pain, presented treatment and the onset of pain inspired 2 retro- here, are derived from those of the IHS.^>' They spective studies'^-'"' of patients who had undergone have nor been validated. endodontic treatment. At least S patients m the first study and 3 in the second study presented • The pain is initially well-circumscribed and is with atypical odontalgia; this represented a mini- limired to a zone of the face or oral cavity. The mal prevalence of 2.5% and 3%, respectively, for pain may then spread over a larger area and the 2 populations studied.'^'"'*' Recent figures give become diffuse. !t is experienced deep within the a slightly higher estimate."" Women constituted 68 tissues. to 100% of the populations studied,-'-"-''^'"'"'-'''^ • The pain has been presenr for 4 to 6 months or with an average age hetween 40 and 51 more (remission is sometimes possible). • Pam is continuous throughout all or almost all the day except during sleep. • The pain does not have a major paroxysmal Description component. • There is no definitive etiology, and a diagnosis Pain is localized to a tooth that shows no dis- cannor be confirmed by any one clinical, radio- cernible pathology to cause such symptoms. logic, or laborator>' examinarion. Premolars and molars are most often affected, and " There is no associated . those in the maxilla are more often affected than those in the mandible.'' The term "phantom The IHS adds that rhe pain may be iniriated by a tooth"^'*''"' refers to the situation when a tooth surgical intervention or trauma to the face, teeth, that has heen extracted seems to be the source of or oral mucosa. There is a high female predomi- pain. It is difficult to tell whether the pain is expe- nance. rienced in rhe dental tissues or in the bone rhat has replaced the roots of the tooth. Pain is continuous throughout the day, or part of it. Sleep is not dis- Atypical Odontalgia turbed.-'"* Symptoms may be present over several months or may come and go periodically over the Definition and Terminology years, but the pain is always of the same nature.-'"' The tooth or teeth concerned may be hypersensi- Atypical odontalgia may be defined as pain of den- tive to thermal stimulation but not to percussion. tal origin wirhour a definitive organic cause. Forms The overlying alveolar bone may be extremely sen- of oral pain that are not of dental origin musr be sitive to moderate, sustained pressure.^^ The pain eliminated. Atypical odontalgia probably repre- is moderate to intense""^ and presents little^* or sents a clinical form of atypical facial pain--^•''•'- no^^ paroxysmal characteristic. Description of the whose separate classification is justified by its pain is very varied"'^ but is often "throbbing" or localization to 1 or more teeth and by the precise "aching. "^^ description of its characteristics that has been Symptoms begin in adulthood and are reported given by Graff-Radford and Solberg.^^ The term by authors ro be usually^'''''^•'''' or always^^ initi- "phantom roorh pain" has heen proposed to ated by dental treatment, although the treatment describe a similar phenomenon that is intermediate may be as noninvasive as the preparation of an hetween or that resembles both atypical facial pain occlusal rest.^^ The patient often demands treat- and atypical odontalgia.^''••'•' Other rerms have ment that leads to roor rreatments and to extrac- been used that describe a disease approaching the tions, which are all the worse for the fact that they clinical picture of atypical odontalgia or that of tend to increase the intensity and the spatial extent atypical facial pain. These include "idiopathic of the pain suffeted." Following extractions, the odontalgia," "idiopathic periodonralgia," "vascu- pain may be transferred to a tooth adjacent to the lar ," "migrainous neuralgia," edentulous space.'"' The center of the pain is oiten

Joumal of Orofacial Pain 175 Wod s/PÍO ne h on localized over the neck of the tooth. It is possible as a separare entity. When stomatodynia is that following successive extractions, the course of defined as above, tben it can be classified apart the atypical odontalgia hecomes atypical orofacial and not merely as a symptom, Tbis explains why pain. tbe authors prefer this definition to that of the The success of local or rcgiotial anesthesia is IASP,* "burning pain of the tongue or mucosa," equivocal,^^ Anesthesia may be achieved withotit a which describes a symptom. Tbe term "stomato- change in the intensity of pain, which would sitg- dynia" is preferable to that of "burning mouth gest a neuropathic origin. This sign has been pro- syndrome" for the same reasons. Many other posed by Graff-Radford and Solberg as a diagnos- terms have been proposed tbat empbasize one tic criterion.^^'*'' Allodynia is frequently aspect or anotber of the disease, eg, glossodynia, experienced.^^•^'' idiopathic glossodynia, sore mouth, burning tongue, orai dysestbesia, stomatopyresis, or glos- The hnk between atypical odontalgia and psy- 47—4Q chologic signs is even more strongly disputed than sopyrests. for the other subgroups of atypical facial pain. The few discrete psychologic problems observed tn a Epidemiology and Demography large number of cases may be explained by the nature of the pain itself.''^ The possibility of a pre- An important variable between tbe epidemiologic disposing psychologic factor is, however, consid- studies presented in the literature is the choice of the sample, which is rarely representative of the general populatton. This ts emphasized in a study Diagnostic Criteria in which 3 different populations were sampled for comparison. It was found that tbe prevalence of The criteria presented here are similar to those oral pain among patients attending a menopause, proposed hy Graff-Radford and Solberg,^^ They diabetic, or general dental clinic was 26%, 10%, have not been validated. and 2.6%, respectively,™ Another difficulty arises from the tmprecision of the definitions used to • Pain is localized to a tooth that is present in the reach the diagnosis of stomatodynia, A very high mouth or has recently been extracted. prevalence tnay be reported by studies that take • Paiti has been present for tbe last 4 to 6 tnonths into account al! forms of pain or burning sensation or has returned periodically in the same form in the oral mucosa. This is particularly true for over the last period of months or years, epidemiologic studies undertaken with postal ques- • Paiti is cotitinuous throughout all or part of the tionnaires,'^-"*"'- These factors could explain why day except during sleep. a prevalence as high as 15% has been reported in • The pain has no major paroxysrnal character. the general population.^' The studies of the • Chnical or radiographie examination does not Toronto group demonstrate tbis well. They found reveal any obvious cause of pain. an initial prevalence of 4,5% among the general Other factors that are often associated: population of Toronto following a postal ques- tionnaire.'^ When these results were followed up • Allodynia (termed by Graff- by a telephone interview, a more precise diagnosis Radford and Solberg^^) was made, and the prevalence fell to 1.5%.*'' Even • Unreliable effect of local anesthesia according to the authors tbis figure is probably unreaiistically high.'' The National Health Interview Survey is a carefully developed study Stomatodynia that was applied to tbe whole of the United States. It found 0.7% positive replies to the question Definition and Terminology "During the past 6 months, did you have more than 1 prolonged, unexplained burning sensation Stomatodynia ts characterized by pain tn the oral in your tongue or any other part of your mucosa that cannot be attributed to any known mouth?"'^ Since a positive reply to this question organic cause. This definition excludes pain from does not give a definite diagnosis of stomatodynia, the lingual mucosa or from the oral mucosa that it can be assumed tbat the true prevalence is in fact could be explained by local or systemic pathology. below 0.7%. This figure still seems high, and the When pain or burning of the mucosa are caused fact that another study found a totally different by a known disease process, it reflects only 1 estimate (less than 0.01%)'''' suggests that current symptom of this pathology and cannot be classed data are unreliable.

176 Volume 13. Number 3, 1999 Woda/Pionchon

Women are much more frequently affected than Reported changes in diet linked to this disease men. The relative proporción is hetween 3 and 20 could be a result of the aggravation of pain females for each male, depending on the intensity that can be experienced wich certain siudy.''^'''*-'''''^""^^ These figures may be biased by foods in certain patients." the fact that women may be more likely tban men to seek medical assistance.'^ Women affected are Physiopathogenic Hypotheses Limited to menopausal or postmenopausal'*^'''^"^^'''^'''"^^ and Stomatodynia have an average age of approximately 60 Tbe face that the disease affecCs primarily posC- menopausal women suggests thaC hormonal or Description degenerative factors may be implicated in the eti- ology of the disease. It is, bowever, essential to Many descriprions can be found in tbe medical lit- exclude burning sensation of tbe oral mucosa due erature.^'•'^''''•^'-^"''-5'*-"-^-' Tbe patient describes Co nonspecific causes, such as the sequelae of pain, or occasionally dysesthesia, localized Co the radiotherapy, mucositis associated with chemo- buccopharyngeal mucosa, thac shows no organic therapy, or induced by psychoCropic sign of pathology. Tbe most frequently affected medicarion. Other factors that can cause pain areas are the tongue, the palate and gingivae, tbe identical co that of stomatodynia are iron defi- lips, and che pharynx.^^''^'^^ Pain is generally bilat- ciency anemia or Candida albicans , buC eral and symmetrical'^ and is always independent these will not be considered here. It seems tbat of a nervous pathway. tbese causes of burning oral pain occur less fre- Pain is continuous rhroughour all or part of the quently tban burning pain of idiopathic origin." day and tends to viJorsen over tbe daytime.^ '^^ It Otbet specific local etiologic factors that have usually occurs daily.^'' Sleep disturbance may be been proposed for stomatodynia include allergy, recorded as a parallel pbenomenon,"* buc there is electrogalvanism, presence of a partial denture, parafunction, or salivary gland dysfunction. None not a causal relationship, as loss of sleep is rarely of tbese proposals have been scientifically con- due to tbe presence of pain.'"'^^ Stomacodynia is firmed.''^•'"''•''•^^•^''•^^ Similarly, certain systemic generally present over a number of years^'''^" but disorders have been evoked but tbeir implication cbere may be periods of remission.'•* never proven.^- These include Gougeraud-Sjögren Pain is generally spontaneous, but in certain syndrome, diabetes, and vitamin deficiency, but patients it may be triggered by certain foods, par- any link is unlikely to be causal.''^•'"''•'^ ticularly spicy or acidic foods.'''^^'^^ In other patients, food or drink may alleviate pain.'^'" Otber daily activities may alter the intensity of the Diagnostic Criteria pain sensation.'' Pain intensity vaties greatly berween patients, ranging from a simple irritación The criteria proposed here are not validared and to the worst pain imaginable. There is no major e.xclude oral pain that is a result of defined local or paroxysmal component. The usual term found ro systemic disease. descrihe the pain is "burning."^' • Pain or dyseschesia in the buccopharyngeal Subjective impressions of xerostomia (dry mucosa. mouth),^^'^''^^ thirst,^'' or (alteration in • Pain has been present for tbe last 4 to 6 months caste perception)-^^'^^'-'^'^*'^^ often accompany che or has returned periodically in tbe same form pain. Certain psychologic disordets are often over the last period of montbs or years. noted, most frequently depression or anxi- • Pain is continuous rhrougbout all or part of tbe gjy_i8,65-fi-69 Rojo et ai^* found tbat their patients day except during sleep. with stomacodynia were divided into 2 different • Tbere is no major paroxysmal character. groups of equal size. The first presenred more • Clinical or radiographie examination does not symptoms of anxiety, depression, obsession, soma- reveal any obvious cause of pain. tizacion, and hostility than the controi group, while the second group was not distinguishable The following characteristics are noted: from the control. This shows that although anxiety • Higb female prevalence and depression are frequently encountered in tbis • Presence of a depressive, anxious, or somatic population, they are by no means present in all psychologic factor cases63,6? In addition, many pacients with stoma- todynia

Journal of Orofacial Pain 177 Woda/Pionchon

Idiopathic Facial Arthromyalgia stated^^: "Tbere is no way of knowing wbether tbe cbaracteristics tbat constitute tbe proposed cate- Many original articles and reviews bave been pub- gories reported actually exist or are artificial con- lisbed on masticatory muscle and TMJ disorders. structions based primarily on clinical experience." It is out of rbe scope of this paper to exhaustively Moreover, tbe extensive overlap of signs and review all tbe classifications, epidemiologic data, symptoms could be more tban a "disturbing trap clinical features, and proposals for diagnostic crite- for scientists and clinicians" and could indicate ria. Tbe reader is referred to the relevant rbat patients are scattered in a continuum, with no reviews.^•"•^^"''^ Tbe present chapter is aimed at isolated group of patients corresponding to a chni- presenting the arguments in favor of inclusion of cally well-defined illness. what are generally called rhe remporomandibular Anotber point of view is to distinguish 2 groups disorders within the general concept of idiopatbic of patients within the category of TMJ disorders. facial pain. The first grotip consists of patients suffering from pain with an identified somatic cause, either Definition and Terminology related to general disease, such as neoplasia or rheumatoid artbritis, or to a degenerative TMJ "Temporomandibular disorders" is rbe interna- problem. Tbe second group, much larger, consists tionally established term^''-" for these conditions. of patients suffering from pain that cannot be eas- Up to tbis point tbey bave been called "mastica- ily explained by a somatic origin, at least with the tory muscle and remporomandibular joint disor- presently available knowledge. In tbis latter case, ders." Tbese are collective terms tbat embrace a unexplained pain related to tbe masticatory mus- number of clinical problems involving the muscles cles and/or TMJ is the main sign, and we propose, of mastication, tbe TMJ, and associated after Feinmann et al,-'''^^ to term tbese conditions structures.^'''^•''*''''^ Tbe TMJ disorders are consid- "idiopathic facial artbromyalgia." The term "dys- ered a subclass of musculoskeletal disorders,•'^•^^ function" can easily be discarded given tbe fact and tbe similarities between some of their parbo- tbat almost all patients ¡97%) seek treatment pbysiologic features witb otber diseases, such as because of pain.''^ , tension beadacbe, or low , Many recent systems of classification differenti- bave recently been stressed.^" ate between 3 groups of TMJ disorders, ie, In rhe past, TMJ disorders were considered a myofascial pam, intracapsular or disc disorders, single homogeneous syndrome. Tbis belief can be and degenerative disorders (see Clark et al"). It is seen in the many different terms tbat were used, tempting to distinguisb idiopatbic facial myalgia sucb as Costen syndrome,^' TMJ dysfunction,^' from idiopathic facial artbralgia, the former corre- pain dysfunction syndrome,'*'-*''^ or myofascial sponding to the facia! form of myofascial pain syn- pain-dysfunction syndrome.^' Modern tbinking drome and the latter to the disc derangemenr dis- supports the view that tbe TMJ disorders represent orders. There are several reasons not to do so. a cluster of related diseases tbat bave many clinical True idiopathic facial myalgia syndrome fre- features m common and wbose signs and symp- queiirly presents witbout joint signs. In tbis group, toms are locahzed to the TMJ and/or muscles of tberefore, pain is tbe principal feature and appears mastication.^'^^ As a consequence, a large amount as rbe first diagnostic criterion, even if otber usual of work has been dedicated to the division of this signs of TMJ disorders are present.' On tbe other group into several subgroups whose signs and band, tbe leading chnical feature of disc derange- symptoms overlap.^ In tbis classification system, ment disorders is related to tbe dysfunctional joint multiple diagnoses are possible, meaning tbat a and not to tbe pain itself. In tbe case of a pure disc single patient can be categorized into several dif- disorder witbout pain, tbe need for treatment ferent divisions. This approacb bas proved useful, should be carefully considered, since aggravation as it has deepened knowledge of the clinical sub- of the condition is uncommon.'-^^'^^ In addition, groups of TMJ disorders and bas given clinicians a there is now much data that clearly sbows a poot better basis from wbich to choose a suitable treat- correlation between disc dysfunction and noises ment plan. It has also given the researcher a nomi- from rbe joint,^''"^- whicb are commonly sought nally homogeneous group of patients. Tbis classifi- during clinical examination as a basic sign for cation may, bowever, be misleading from a diagnosis. Therefore rhe primary indication for taxonomic and pathopbysiologic point of view. treatment of tbe disc derangement disorders is No data indicates that different mechanisms are joint pain ratber than dysfunction, tbe former acting in eacb of tbe different subgroups. As Turk being also frequently associated witb muscle pain.

178 Volume 13, Number 3, 1999 Woda/Pionchon

Table 1 Summary of the Principal Signs and Symptoms of the Different Types of Idiopathic Orofacial Pain* ^

Idiopathic facial Atypical facial Atypical odontalgia Scomatodynia arthromyalgia Sign/symptom pain (bone) (toudi) (niucosa) ¡muscle, articulation) Independent of a nervous Independent of a nervous Independent of a nervous Independent of a pathway. Unilateral initially, pathway. A single tootii pathway. Often bilateral nervous patiiway. becomesbilateral'^-^'-'" initially but may spread^ and symmetrical^^ Unilateral or bilateral Time period of pain Continuous^' ^^ Continuous witii possible Continuous Continuous with remission-^'' remissions Paroxysmal No No No Pain dunng sieep Infrequent*"'"" Uncommon'"^ but disturbed sleep'"^ Intensity Moderate to intense^ Moderate to intense''^ Weak to intense^ Weak to intense Descnptors Emotional.™ mechanical,'^ Vaned"^ Buming«^ Spontaneous (dull, buming'^'™ aching) or during function or voiuntary movements Initiating factor Frequantiy trauma'^'^ Frequently dentai Stressful life event Possible direct may be implicatet)^^ trauma'"^; whiplash, possibie Effect of local At ieast temporary Ambiguous effect Temporary reiief anestiietios relief'^ (for tngger points) Neurologic signs Frequently dysesthesia, Frequently allodyr Aliodynia (trigger aliodynia, paresthesia''''^" points in myofascial painÜ Effect of analgesics None or weak^ None or weak' None or weak^ Sometimes effective Psychologic factors Debatable^""'''« Frequent Sympathetic signs No but possibly Not descnbed Implication suggested or implication implicated"^ for myofasciai pain Other associated Boneoavity,'"-"^-'" None Xerostomia,^''^ Limited range of iocal signs osteoporosis? mandibuiar motion, thirst^' masticatory and TMJ palpation tenderness, TMJ sounds (nonspe- cific),"^ bruxism and oral habits 32120 Mean no. of previous 7.5 ° consultations Prevalence Unknown 2,5 to 3% of 4 to 5%^'' endodontio ca Mean age (y) 5216 .a 55 to 6 Second to fourtii decades of age'^ Mate:female ratio 1:3to 1:i 1:3 to 1:3 to 1:9 The descnpUon of idiopathic aniirornyaigja JS tiiat typicaiiy used lor the TMJ dis rders (from Okeson' oi Lipton and Di if not othe altiiough the two terms do rot cover eKsctiy the same subgroups.

Therefore, this is in line with the more general fication it allows for multiple diagnoses and a list concept of arthromyalgia of the face (Table 1 ¡. of Research Diagnostic Criteria, which are intended to fit vahdatioti processes,'•- Epidemiology and Demography Epidemiologic studies should also take the natu- ral history of the disease into account. Longi- There are many sources of confusion in the epi- tudinal studies have shown that the semiology of demiologic studies that have examined the preva- this group of diseases varies with age in a non-ran- lence of masticatory muscle and TMJ disorders. dom fashion. Marked variation is noted in the The wide variety of clinical presentations and the inrensit}' of pain and in the presenting of signs and ahsence of objective diagnostic criteria have driven symptoms over time.^-^'^'' The prevalence of idio- a research group to define both a system of classi- pathic arthromyaigia is greatest among young

Journal of Orofacial Pain 179 Woda/Pionchon adults and lessens from 40 years of age.^' The tra- It frequendy radiates to other parts of the face but ditional belief in an inevitable aggravation of prob- does nor follow a nerve trajectory. Pain is continu- lems is not valid. When the results of epidemiologic ous but can he triggered or exacerbated by move- studies are interpreted, the difference must also be ment or function. Intensity may vary over each 24- noted between an isolated or weak sign and hour period'"^ and is usually experienced only intense, lasting pain that induces the patient to seek during the day,^''* even if patients often complain treatment. of disturbed sleep.'"^ Variety also exists in the Despite these reservations, the prevalence of length of episodes of pain and remission between masticatory muscle and TMJ disorders has begun episodes. The pain is generally weak to moderate to be better recognized and can be summarized as and is often described as dull or aching. There is follows. Presence of an isolated sign is extremely no major paroxysmal character and few neuro- common, with 76% of a sample of North logic signs. In myofascial pain, however, some American students presenting with at least 1 sign, kind of allodynia is present, as trigger points can males and females alike.'-^ A recent study^^ be identified. reported a prevalence of at least 1 ob|ective sign Limitation or asymmetry of mandibular move- among 44% of subjects. This study was under- ment; noise in the joint described as clicking, pop- taken on a large representative sample of the ping, or crepitus; and association with bruxism are Dutch population. The same group confirmed frequent. The specificity or the lack of specificity these figures but showed hy meta-analysis that of these signs has been rhe subject of many recent there is a great range of prevalence of signs (between 0 and 93%). The prevalence of pain is studies.^^'^""'^ Some other nonspecific and unex- reported as being between 5 and 30%, depending plained complaints are tinnitus, ear fullness, and on the criteria used for definition of intensit)' and perceived hearing loss. Psychologic factors are fre- frequency of symptoms. In North America, a con- quently present and may predispose or perpetuate sensus value of 12% has been estabhshed for pain the condition. suffered in the 6 months preceding study.'^"^''*' Despite this, the need for treatment for this type of pain only approaches 4 or 5%, and only 2% of Diagnostic Criteria for All Orofacial Pain parients had sought treatment in the 9 months pre- Entities ceding study.^^ Partly validated diagnostic criteria are available for Of those patients seeking treatment, the major- the different subgroups of TMJ disorders.^•^•^"^ No ity are female (3 to 9 females for each male^-^*'^^). criteria have been proposed for the different types The pain is of greater severity for women, both of idiopathic arthromyalgia. The description of physical and psychologic, among those seeking pain found in the literature does, however, allow treatment."" diagnostic criteria common for the whole group of idiopathic orofacial pain to be proposed. Description • Pain is oral, perioral, or facial and does not fol- Only a very short description of the signs and low a nervous pathway. symptoms based on the guidelines edited by the • Pain has been present for the last 4 to 6 months American Academy of Orofacial Pain (AAOP)^ or has returned periodically in the same form and on the National Institutes of Health (NIH) over a period of several months or years. technology assessment conference on management • Pain is continuous and is present throughout all of temporomandibular disorders^"^ will be pre- or part of the day and is infrequent during sleep. sented. Detailed descriptions of the different sub- • There is no major paroxysmal character. groups can be found in many books and • Clinical, radiographie, or laboratory examina- re vie WS.'•'^"^'^'^"^ A comparison of the signs and tion does not reveal any organic cause of pain. symptoms of idiopathic orofacial arthromyalgia The following characteristics are noted: with those of the other types of idiopathic orofa- cial pain is presented in Table 1. • A marked female predominance Pain is by far the most frequent symptom of • The frequent presence of certain psychologic TMJ disorders*^ and is by definition the basic one factors, personalit}' traits, or life events when idiopathic facial arthromyalgia is considered. Pain is located in the muscles of mastication, preauricular area, or TMJ^' on one ot both sides.

180 Volume 13, Mumber3, 1999 Woda/Pionclion

Taxonomy and Research Perspectives signs and symptoms sbould allow tbe definition of distinct entities on a scientific basis. Epidemiologic Glass i tica tion of pain in the head atid neck region characteristics also need to be studied. Wbile the into specific diseases, syndromes, or pain entities prevalence, evolution without treatment, and tbe relies largely upon work undertaken by tbe IASP'' populations at risk are beginning to be elucidated or by the IHS^'^ and later completed by the for muscular and TMJ disorders, this is not the AAOP." Difficulty in classifying the subgroups of case for the other potential subgroups of idio- idiopatbic facial pain entities is illustrated pathic orofacial pain. In particular, the prevalence markedly in these reference texts. The 4 distinct of atypical facial pain in the general population is groups of atypical orofacial pain are never totally unknown, despite its impact on patients' grouped together and are not even always lives. Its prevalence among populations at risk, for described individually. Furtbermore, in the intro- example in menopausal women, would have duction of a classification for all types of pain, the important clinical implications. Also, it could be experts of the IASP' cite atypical facial pain under presumed that the prevalence of stomatodynia the title of "some controversial issues." For would be, in tbese groups, higher than the current estimate of less than 1% in the general population. Merksey and Bogduk,^ the term "atypical facial Although longitudinal studies are not feasible, pro- pain" is excluded from the classification because it jects studying the natural history of atypical facial does not tepresent a well-defined entity and could pain or stomatodynia would be clinically useful. correspond to different pathologic situations, This type of data could influence the politics of depending on the case (such as TMJ disorders, public health. Finally, a better knowledge of tax- atypical odontalgia, or ). In this classifica- onomy and epidemiologs' of these diseases would tion, the 2 subgroups of muscle and |omt pain be helpful in defining inclusion criteria of clinical (myofascial pain and arthralgia) are not separated. trials aimed at the improvement of treatments. Tbe IHS,^-' for its part, differentiates neither stomatodynia nor atypical odontalgia. Myofascial pain of the face is classified, but the myofascial subgroup is linked to tension-type . The Acknowledgments term "atypical facial pain" is also absent from this classification and is replaced by a group entitled We are indebted to D. Faulks for English corrections and to "facial pain not corresponding to any of tbe pre- A.M. Gaj'dier and M. Chalus for rbeir excellent secretarial ser- vices. This work hab been supported by a grant from European ceding groups." This phrase is used interchange- Cotïimunity BlO4.9S,0076. ably with the tetm "atypical facial pain" by tbe AAOP," which associates it with atypical odontal- gia and with sympathetically maintained pain. References Stomatodynia and the different groups of muscle and TMJ pain are described but are not linked 1. Truelove EL, Sommers EE, LeResche L, Dworkin SF, Vun together or with the other idiopathic facial pains. Korff M. Clinical diagnostic criteria for TMD, new classi- This brief review of the best-known classifica- fication permits multiple diagnoses. J Am Dent Assoc 1992; 123:47-54. tions emphasizes the confusion that reigns in tbis 2, Dworkin SF, LeResche L. Research diagnostic criteria for area. Tbe principal criterion used in the classifica- temporotnandibulat disorders: Review, criteria, examitia- tion of pain is that of localization.' Thts principle rions and specifications, critique. 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