716 J Neurol Neurosurg Psychiatry 2001;71:716–719 J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.71.6.716 on 1 December 2001. Downloaded from

NOSOLOGICAL ENTITIES?

Chronic facial : a multidisciplinary problem

G Madland, C Feinmann

Abstract to diVerent specialists providing diVerent treat- is an unrecognised ments, including dentists, neurologists, otorhi- and unhelpful diagnosis but one which nologists, osteopaths, chiroporacters, and psy- describes chronic that do not fit the chiatrists, with little collaboration. present classification system. Due to the There are additional important problems site of the pain, patients may seek and, concerning the recognition and definition of indeed, receive treatment from dental underlying psychiatric disturbances. Emotional practitioners and specialists, but the pain disturbance, when present, is often mild and of is often unresponsive and may have more brief duration and psychiatric classification has in common with unexplained medical proved an inadequate measure. symptoms aVecting other areas of the body, than with other dental symptoms. Chronic symptoms This review suggests a need for a diagnos- Chronic symptoms and syndromes pose a tic category of “chronic facial pain”, major challenge to medicine: they are com- which demands a multidisciplinary ap- mon, often persistent, and are associated with proach to diagnosis and management. significant distress, disability, and unnecessary (J Neurol Neurosurg Psychiatry 2001;71:716–719) expenditure of medical resources. In United Kingdom primary care, somatic symptoms and Keywords: chronic pain; facial pain syndromes account for 20% of consultations. copyright. Among medical outpatients, somatic com- plaints accounted for 35% of new referrals in a Chronic idiopathic is an ill United Kingdom study.2 Even among medical understood group of conditions, which may inpatients, a substantial proportion have com- involve the whole of the mouth and face. plaints that are found to be functional. The Unfortunately descriptions of disorders and prevalence of emotional distress and disorder treatment tend to be influenced by the in patients who attend hospital with unex- background of the specialist assessing the plained syndromes (such as irritable bowel patient. Thus patients who see maxillofacial syndrome) is higher than in patients with com- surgeons have symptoms described in terms of parable medical conditions (such as inflamma- clicking, sticking, and locking of the temporo- Behavioural Sciences tory bowel disease) and many such patients are and Dentistry, mandibular joint and pain in the associated severely disabled.3 http://jnnp.bmj.com/ Eastman Dental musculature. Ear, nose, and throat surgeons Wessely et al3 suggest that each medical spe- Institute and Hospital, may retain Costen’s outdated notion that the cialty has defined its own syndrome or 256 Gray’s Inn Road, pain is due to missing molar teeth and may syndromes in terms of symptoms that relate to London WC1X 8LD, refer on to maxillofacial surgeons or restorative UK their organ or interest, despite similarities in G Madland dental specialists. Despite advice from the case definition, reported symptoms, sex, out- C Feinmann National Institute of Health that “there is no look, and response to treatment. Patients seek evidence linking occlusal abnormalities with help from doctors for symptoms, and doctors Department of pain”, patients’ occlusions continue to be diagnose diseases to explain them. Symptoms on September 26, 2021 by guest. Protected Psychiatry and adjusted by ill informed practitioners, often are the patient’s subjective experience of Behavioural Sciences, leading to more problems for patients.1 University College changes in his or her body, diseases are objec- London, UK The NIH conference in 1996 reviewed the tively observable abnormalities in the body. G Madland issues on the management of orofacial pain, DiYculties arise when the doctor can find no C Feinmann concluding that major problems hampered objective changes to explain the patient’s present diagnostic classifications and treat- subjective experience. The symptoms are then 48 Riding House ment. Five years later, there is no greater clar- referred to as medically unexplained or func- Street, London ity in classification. W1N 8AA, UK tional. Atypical facial pain is relegated to the G Madland Current diagnoses include tension , dental rather than neurological domain. C Feinmann , neckache, temporomandibular disor- Wessely et al postulate that “the existence of der (temporomandibular joint pain dysfunction specific somatic syndromes is largely an Correspondence to: syndrome, facial arthromyalgia), and atypical artefact of medical specialisation. That is to say G Madland [email protected] facial pain. These pains seem to arise from blood that the diVerentiation of specific syndromes vessels, muscles, and joint capsules rather than reflects the tendency of specialists to focus on Received 19 December 2000 conforming to the distribution of sensory nerve only those symptoms pertinent to their and in revised form 5 March 2001 branches, as in trigeminal . Artificial specialty, rather than any real diVerences Accepted 7 March 2001 distinctions in clinical presentation lead patients between patients” (p 936).

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Symptoms and signs activity), in response to both heat and nocicep- There are four recognisable symptom com- tive stimuli, in patients with atypical facial pain plexes of chronic orofacial pain, which may, relative to controls.9 This is suggestive of an however, coexist: temporomandibular disorder exaggerated perception of pain in response to (myofascial face pain); atypical facial pain peripheral stimuli but how this might develop (atypical facial neuralgia); atypical odontalgia remains a matter for speculation. The authors (phantom tooth pain); and burning mouth thought it likely that the mechanism for the syndrome (oral dysaesthesia, glossodynia, glos- diVerences found is related to and sopyrosis). They may be considered as medi- attention and therefore that the pain might be cally unexplained symptoms aVecting four brought under conscious control. However, the regions of the mouth and face. cross sectional nature of this evidence pre- Atypical facial pain is no longer included in cludes any conclusions as to whether such cen- the International Association for the Study of tral changes are primary or secondary to pain. Pain’s “classification of chronic pain”.4 The term originated to distinguish the condition Epidemiological considerations from “typical” , as the pain Although the prevalence of joint related and neither follows the distribution of the periph- muscle related symptoms has been studied, the eral nerve, nor responds to antiepileptic agents. epidemiology of chronic facial pain remains However, the categorisation of patients with unclear. An obvious explanation for why some similar pain histories into a diagnostic pigeon people choose to consult whereas others do not hole labelled “atypical” is self contradictory, is the severity of their symptoms. Sadly, epide- and the definition of a condition by what it is miological studies of facial pain have tended to not, rather than what it is, is unsatisfactory. A ignore intensity and frequency of symptoms better term might be “chronic facial pain”, as and recorded only their presence or absence. Orofacial pain is a common problem a ect- the defining characteristics are longevity and V ing at least 10% of the adult population and site, as distinct from temporomandibular 50% of the elderly population, and its inad- disorder which aVects the jaw rather than the equate recognition and management present (mid-)face, and intraoral pains. an enormous problem to the health service. The pain is usually a continuous dull ache The sex distribution is thought to be equal in with intermittent severe episodes, primarily the general population, but four times as many aVecting areas of the face other than joints and women as men seek help.10 Barksy and Borus muscles of mastication, such as the zygomatic have described how changes in social attitudes copyright. maxilla. Pain may be bilateral and will often have reduced the public tolerance to mild have been present for several years. symptoms and benign infirmities, physiological are ineVective. responses to anxiety are also misinterpreted as Atypical odontalgia has a similar character illness. Isolated symptoms are then identified but is localised to one or more premolar or as disease by patients.11 molar teeth, simulating .5 There may be a history of inappropriate dental treatment, Associated personality and psychiatric including extraction, and subsequent recur- problems rence of symptoms apparently from another There is a long standing association in the tooth. literature between chronic facial pain and psy- Patients often attribute their pain to an ante- chological distress, particularly .12 cedent event such as a dental procedure, or This is consistent with other chronic pain http://jnnp.bmj.com/ minor trauma to the face. Despite the notori- groups, in which the prevalence of depressive ous unreliability of such retrospective reports, symptoms is consistently higher than in the these attributions have led to the suggestion general population and many other medical that chronic facial pain may be a deaVerenta- populations.13 Studies comparing patients with tion syndrome (compare phantom tooth organic lesions and patients with indeterminate pain).6 chronic pain, report considerably higher preva- Similarities to postamputation pains include lence of depression among the second.14 This burning quality, description as severe despite suggests that beliefs and attributions about the on September 26, 2021 by guest. Protected lack of sleep disturbance, poor localisation, and cause of their symptoms may aVect psychologi- delay between injury and incidence. The lack cal wellbeing in patients with chronic pain. of response to nerve block is also a common A “diathesis-stress” framework has been feature. Divergence from a classic picture may proposed to explain the high comorbidity well betray the influence of factors beyond between chronic pain and depression.15 This identifiable pathology. Depression is a common approach encourages identification of vulner- feature but is more likely to be a consequence ability factors in the individual person as well of living with pain than a precursor to it.7 as investigation into the nature of the stressor, Ninety five per cent of patients with atypical and may be a useful theoretical basis from facial pain complain of other symptoms, which to advance the study of depression in including headache, neck and backache, der- chronic pain. matitis or pruritis, irritable bowel, and dysfunc- tional uterine bleeding. This prevalence is Management much greater than in the normal population.8 The treatment oVered to a patient with chronic Positron emission tomography has demon- facial pain will be determined by the specialist strated an increased contralateral cingulate understanding of the clinician to whom he or cortex activity (and decreased prefrontal cortex she is referred.16 A multidisciplinary approach

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is therefore preferable and should be adopted with chronic facial pain.24 EVective communi- for diagnostic and prognostic assessment. Pos- cation during the early phases of facial pain sible dental or neurological causes, including may prevent the development of long term trigeminal or craniocervical neuropathy, must problems. Clinicians should be aware of the be eliminated with clinical and radiographic importance of listening to the patient’s beliefs investigation by a specialist, but extensive about their pain, and of trying to address their investigation can lead to patients being oV concerns without resorting to outdated psy- loaded from specialist to specialist in search of chogenic models of pain. Patients gain enor- a diagnosis, and feeling ill understood, overin- mous benefit from having their symptoms vestigated, and dissatisfied. Patients want to acknowledged as part of a group of conditions know what is wrong with them rather than with which clinicians are familiar. An infor- what is not wrong. Recent evidence suggests mation booklet is also likely to be helpful that it may be more helpful to assess patients in (available from the authors on request). terms of disability and coping strategies, rather Reassurance about the non-malignant nature than pain intensity itself.17 There is a real need of chronic pain is also important, but empty to make diagnosis realistic in terms of what promises that “things will get better” are patients are told and what they understand unhelpful. The message is that talking to about their problems.18 19 Patients must feel patients is often more useful, albeit sometimes that the physician believes in their pain and that more demanding, than operating on them. the patient’s understanding or attribution of the pain is accepted. It is crucial that physicians Conclusions and patients reach an agreement about how to (1) “Chronic facial pain” is a more descriptive manage the pain. Thereafter, the mainstays of term than “atypical facial pain” and should be treatment are counselling and adopted for continuous, dull pain in the face, of greater than 6 months duration, with intermit- medication. There is no evidence from ran- tent severe episodes. It diVers from temporo- domised trials of benefit from surgical inter- mandibular disorder and burning mouth syn- vention, which should in itself be enough to drome, in terms of site (mid-face), and discourage such an intervention, even in the longevity (lacking a responsive acute form— face of patient entreaty. Patients with chronic compare temporomandibular disorder), but, pain are often desirous of a concrete cause and once established, all have features in common treatment, and will therefore request surgery, with other medically unexplained conditions, but there is reason to think that surgery may in and require a multidisciplinary approach. copyright. fact worsen the problem. (2) The management of this heterogeneous Ina4yearreview of a mixed group of 71 group of patients with pain is complicated by patients with “idiopathic” facial pain who had the area of the body involved, and by the inter- initially participated in a double blind trial of action of organic and psychological factors in dothiepin, and received further courses of the the somatising process. No single clinical drug and counselling sessions, 43% of the specialty receives the training required for the patients with chronic facial pain were pain diVerential diagnosis and management of these 8 free. The “atypical” patients were the least pain disorders and, hence, there is both a need responsive. for multidisciplinary clinics, and for specific Pain relief with tricyclic agents seems to be training programmes in dental and medical independent of the antidepressant eVect of the undergraduate curricula. 20–22 drugs. These drugs are considered to act by (3) Although there is an obvious need for http://jnnp.bmj.com/ altering the sensory discriminative component prompt elimination of possible organic disease of pain. The possibility of interference with such as tumour, there is also the danger of serotonin reuptake in the brain stem has been overzealous investigation and provision of proposed.22 In two short term studies, the sero- inappropriate and irreversible dental treat- tonin antagonist iprazochrome had equivocal ment. General dental and medical practitioners eVects on a group of 30 patients with chronic must appreciate the risk of exacerbation of facial pain23 but fluoxetine improved pain associated psychological distress and the severity and distress in a group of 178 importance of psychological assessment and on September 26, 2021 by guest. Protected non-depressed patients with chronic facial counselling at an early stage. pain.24 (4) Drugs such as dothiepin and fluoxetine Unfortunately, dental and medical practi- are of proved, if modest, benefit in the manage- tioners are often reluctant to prescribe antide- ment of chronic idiopathic orofacial pain, pressant agents, mistakenly considering them although originally developed as anti- to be addictive or to have overwhelming side depressant drugs. Provision of information, eVects. In addition, inadequate dosage and drug treatment, and counselling, are comple- duration, perhaps in combination with ex- mented by cognitive therapy specifically aimed pressed lack of confidence in their eYcacy, pre- at reducing interference with life. vent the drugs from having any beneficial eVect. The liaison psychiatrist is currently the 1 National Institute of Health: technology assessment state- only specialist appropriately trained in drug ment. Management of temporomandibular disorders. Washing- ton, DC: NIH, 1996. treatments. 2 Hamilton J, Campos R, Creed F. Anxiety, depression and Cognitive behavioural therapy, in combina- management of medically unexplained symptoms in medi- cal clinics. J R Coll Physicians Lond 1996;30:18–21. tion with drug treatment, has been found to 3 Wessely S, Nimnuan C, Sharpe M. Functional somatic reduce the pain’s interference with life and to symptoms: one or many? Lancet 1999;354:936–9. 4 Merskey H, Bogduk N. Classification of chronic pain. 2nd ed. increase perceived control over life, in patients Seattle: IASP Press, 1994:59–60.

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5GraV-Radford S, Solberg W. Atypical odontalgia. Journal of 16 Raphael K, Marbach JJ. Evidence-based care of musculo- Craniomandibular Disorders and Oral Facial Pain 1992;6: skeletal facial pain: implications for the clinical science of 260–6. dentistry. J Am Dent Assoc 1997;128:73–9. 6 Marbach JJ. Is phantom tooth pain a deaVerentation 17 Madland G, Feinmann C, Newman S. Factors associated (neuropathic) syndrome? Part I: evidence derived from with anxiety and depression facial arthromyalgia. Pain pathophysiology and treatment. Oral Surgery Oral Medicine 2000;84:225–32. and Oral Pathology 1993;75:95–105. 18 Donovan JL, Black DR. Qualitative study of interpretation 7 Dohrenwend BP, Raphael KG, Marbach JJ, et al.Whyis depression comorbid with chronic myofascial face pain? A of reassurance among patients attending rheumatology clinics: “just a touch of arthritis, doctor”. 2000; : family study test of alternative hypotheses. Pain 1999;83: BMJ 320 183–92. 541–4. 8 Feinmann C. The long-term outcome of facial pain 19 Clark NM, Gong M. Management of chronic disease by treatment. J Psychosom Res 1993;37:381–7. practitioners and patients: are we teaching the wrong 9 Derbyshire SW, Jones AK, Devani P, et al. Cerebral things? BMJ 2000;320:572–5. responses to pain in patients with atypical facial pain 20 Feinmann C, Harris M. Psychogenic facial pain. Part 2: measured by positron emission tomography. J Neurol Neu- management and prognosis. Br Dent J 1984;156:205–12. rosurg Psychiatry 1994;57:1166–72. 21 Sharav Y, Singer E, Schmidt E, et al. The eVect of 10 Feinmann C. Idiopathic orofacial pain: a multidisciplinary on chronic facial pain. Pain 1987;31:199– problem. Pain: an updated review. Seattle: IASP Press, 1996: 209. 397–402. 22 Pettengill C, Reisner-Keller L. The use of tricyclic 11 Barsky AJ, Borus JF. Somatization and medicalization in the for the control of chronic orofacial pain. era of managed care. JAMA 1995;274:1931–4. Journal of Craniomandibular Practice 1997;15:53–6. 12 Mock D, Frydman W, Gordon A. Atypical facial pain: a ret- 23 Hampf G. EVect of serotonin antagonists on patients with rospective study. Oral Surgery Oral Medicine Oral Pathology atypical facial pain. 1985;59:472–4. Journal of Craniomandibular Disor- 13 Romano J, Turner J. Chronic pain and depression: does the dorders and Facial Oral Pain 1989;3:211–12. evidence support a relationship? Psychol Bull 1985;97:18–34. 24 Harrison S, Glover L, Maslin L, et al. A comparison of 14 Magni G. On the relationship between chronic pain and antidepressant medication alone and in conjunction with depression when there is no organic lesion. Pain 1987;31: cognitive behavioral therapy for chronic idiopathic facial 1–21. pain. In: Jensen T, Turner J, Weisenfeld-Hallin Z, eds. 15 Banks S, Kerns R. Explaining high rates of depression in Proceedings of the 8th World Congress on Pain: progress in chronic pain: a diathesis-stress framework. Psychol Bull pain research and management. Vol 8. Seattle: IASP Press, 1996;119:95–110. 1997.

7th European Forum on Quality Improvement in Health Care

21–23 March 2002 copyright. Edinburgh, Scotland

We are delighted to announce this forthcoming conference in Edinburgh. Authors are invited to submit papers (call for papers closes on Friday 5 October 2001) and delegate enquiries are welcome. The themes of the Forum are: x Leadership, culture change, and change management x Achieving radical improvement by redesigning care x Health policy for lasting improvement in health care systems x Patient safety http://jnnp.bmj.com/ x Measurement for improvement, learning, and accountability x Partnership with patients x Professional quality: the foundation for improvement x Continuous improvement in education and training x People and improvement. Presented to you by the BMJ Publishing Group (London, UK) and Institute for Healthcare

Improvement (Boston, USA). For more information contact: [email protected] or look at on September 26, 2021 by guest. Protected the website www.quality.bmjpg.com. Tel: +44 (0)20 7383 6409; fax: +44 (0)20 7373 6869.

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