- Email: [email protected]
Burning mouth syndrome
Mouth Syndrome Successfulc Treatment with Combined ~sy~huthe~a~y and Psychopharmacotherapy Boudewijn
Van Houdenhove,
M.D. and Peter Joostens, M.D.
Abstract: A case is presented that illustrates the possible role of significant life events and depression in the etiology of the ~r~iffg mouth s~drome. Furt~r~re, successful tr~tme~t with a com~~ationof a selective serotoninye-uptake b~ocki#g antidepressantf’sertraline)and psychodynamicallyoriented psychotherapyis described.
The burning mouth syndrome (BMS) is a clinical entity characterized by a painful, burning sensation in the oral area, with no visual mucosal abno~alities on clinical examination. Mostly the tongue region is involved, hence, the older term “glossodynia.” However, in half the patients, other locations are involved, e.g., the frontal twothirds of the upper palate or the lower lip. During the last decade, BMS has received a great deal of attention in the stomatological and odontological literature, and Tourne and Fricton [l] and Mott et al. [2] have critically reviewed the present state of knowledge on the syndrome. The prevalence of BMS is still uncertain, as there are no epidemiological studies on the syndrome in general population groups in different parts of the world. However, BMS does not appear to be uncommon (e.g., according to some authors, it may be seen in 5% of a general dental population), and there is a consensus among clinicians and researchers that the syndrome is most prevalent in postmenopausal females [ 1,2]. Many etiological theories have been propounded, e.g., Candida glossitis, vitamin B defiDepartment of Psychiatry, Katholielce Universiteit Leuven, Leuven, Belgium Address reprint requests to: Prof. B. Van Houdenhove, Afdelmg Liaison-psychiatric, U.Z. Pellenberg, B-3212 Lubbeek, Belgium.
General Hospital Psychiatry 17, 385-388, 1995 0 1995 Elsevier Science Inc. 655 Avenue of the Americas, New York, NY 10010
ciencies, diabetes mellitus, estrogen deficiency at menopause, local factors such as inadequate dental bungs, denture wear, pasa~c~onal oral habits, sahvary disturbances, galvanism, and so forth. In spite of intense research efforts, no concmsive organic explanation for the cause of BMS has been found, and the condition is courtly considered as “idiopathic” by most authors [1,2]. Psychopathological factors have been frequently cited as associated with BMS. Lamb et al. [3] showed an occurrence of emotional instability, anxiety, and depression in 60% of a cohort of 47 patients with BMS. Van der Ploeg et al. [4] obtained similar results, with clearly higher ratings for depression, anxiety, and neuroticism. Recently, Rojo et al. [5,6] also found controlled evidence for more affective and neurotic d~~~a~~s (depression/anxiety symptoms, obsessive symptoms, somatization tendencies, and hostility) in a subgroup of BMS patients “with psychiatric disease.” Comparable findings were recently reported by Eli et al. [7J who found more premorbid psychiatric/psychological treatment in BMS patients as well; however, no differences between patients and controls were found on a questionnaire measuring associated stressful life events [S], This was in contrast to an earlier report of Hammaren and Hugoson [9] which showed the high prevalence of distressing life events in a selected group of BMS patients, using a semis~c~red psychiatric interview. From a therapeutic perspective, several forms of local oral treatment (e.g., denture adaptation, habit control) and systemic treatment (e.g., hormone or vitamin replacements) have been tried out, with disparate results (for a review see 11,211. Some authors found that nonresponders were psy385 ISSN 0143~8343J95/$9.50 ssxx 0163-8343(95)mo61-u
B. Van Houdenhove
and P. Joostens
chologically more disturbed than those responsive to treatment [3]. Other studies demonstrated a beneficial effect of tricyclic antidepressants and benzodiazepines on BMS, and this alleviation was shown to be present in both depressed and nondepressed subjects [U&12]. According to Eli et al. [7], the individual response to pain determines whether cure is possible (by either pharmacological, physical or psychological means) or whether the condition will develop into a chronic, persistent pathology. Summarizing the literature, Tourne and Fricton [l] state that multiple methodological flaws in BMS research make the reliable interpretation of the importance of proposed causal factors and the efficacy of specific treatment modalities difficult. This case report illustrates 1) the significance of psychopathological factors in BMS; 2) the importance of a thorough psychiatric investigation (including psychodynamic-biographical history taking) to detect them; and 3) the possible usefulness of combined psychotherapy and psychopharmacotherapy in these patients.
Case Report The patient, Mrs. A, was a 42-year-old married woman with one child, who was referred by the pain clinic of our university hospital for psychiatric assessment. She complained of unbearable pain in the frontal two-thirds of the tongue and the upper palate, that existed for about 10 months, and had commenced abruptly. Mrs. A contacted her family doctor who suspected a local infectious pathology and started a therapy with an antimycotic agent, without results. Afterwards, she consulted a dentist and two different E.N.T. specialists, who could not find any local lesion or systemic causative explanation, since screening for diabetes, hematological or biochemical abnormalities, and infectious disease proved negative. Vitamins Bl and 812 failed to help. Mrs. A had no clinical signs of climacterium, and laboratory testing for estrogen deficiency was negative. After 6 months, the family doctor suggested the possibility of a psychogenic mechanism, in view of the negative somatic investigations, and sent the patient to a psychotherapist, who started a classical psychoan~ysis. At the same time, the family doctor started dosulepine (a tricyclic antidepressant) 75 mg daily, but it had to be stopped a few weeks later because oral dryness made the pain worse. Some weeks later, Mrs. A also stopped her
analysis because the therapist “did not pay any attention to her pain.“ When she eventually attended the pain clinic, she was totally hopeless about her situation. At the first interview, we saw a well-groomed woman who expressed, in a slightly histrionic way, her feelings of hopelessness and despair caused by the unbearable pain. Although she denied being manifestly depressed (“It’s just the pain that makes me feel bad”), several clinical signs of depression were obvious, such as tiredness, apathy, social isolation, diminished concentration, insomnia, and diminished appetite which had led to a weight loss of 6 kg. Her pain symptoms were less prominent in the morning and reached their culminating point at night, and her mood showed an inverse pattern and was somewhat better in the evening. Mrs. A had no manifest suicidal thoughts, but there were vague passive death wishes (“I cannot continue to live in this way”). The personal background of the patient showed a minor depressive episode 17 years ago, and she also had a familial history of depression, with a suicidal attempt by the mother and a father with a post~aumatic stress syndrome. Impressed by the severity of the patient’s depressive state, we suggested an inpatient treatment at the psychiatric ward of our hospital. We promptly started sertraline 50 mg, which was increased to 100 mg after 2 weeks. This was combined with a further psychodynamic-biographical exploration of her life history. A major issue in these sessions was her distressing childhood as a consequence of an early divorce of her parents, whereafter she felt rejected by her father with whom she was never able to develop a warm affective relationship. She also complained about her nonsatisfactory marriage, with lack of attention from her husband, and feelings of boredom and emptiness for which she blamed the absence of a meaningful occupation. A few years ago, Mrs. A had started an extramarital love affair that focused on a passionate sexual relationship. Talking about her conflicting feelings in this relationship, she mentioned that the first pain symptoms occurred 2 days after she had oral sex with her lover, followed by a statement of his which she experienced as deeply humi~ating (“You are as good as a prostitute”). It was further noteworthy that, at the onset of the symptoms, she had been worrying several days about possibly contracting AIDS. After 3 weeks of treatment, the depressive symptoms gradually disappeared and the oral pain
lklrning
simultaneously diminished to an endurable level (“Some sandy feeling in the mouth”). She was discharged from the hospital and followed up with individual psychodynamic psychotherapy, once a week, in our consultation-liaison unit. In these sessions, Mrs. A was able to talk about the emotional problems in her life, especially her disappointing relationships with men, who appeared always to reject her, as her father did. She could express her ambivalent feelings toward her lover, and see some links between her physical pain and her After a few months, she painful emotions. planned to undertake marital counseling with her husband, and looked for a job which, she hoped, would make her life more meaningful. Meanwhile, the sertraline treatment was continued. One month after discharge, the pain had completely subsided, and this situation has been maintained until now, about 1 year later, with continued sertraline therapy 50 mg daily.
Discussion This case report first suggests that psychopathological factors can play an important role in BMS. Although several authors have observed that symptoms of depression, anxiety, irritability, tension, and hypochondriacal preoccupation frequently accompany the syndrome [3-lo], much controversy exists as to whether psychopathology is to be seen as primary, or secondary to the chronic pain state [4,7]. Advocates of the latter opinion point to the fact that psychological disturbances often diminish or disappear when the pain is successfully treated [13]. Evidently, it would seem adventurous to suggest a primary psychogenic etiology for a syndrome in which the role of organic etiological or pathophysiological factors is still unclear. It may be more appropriate to postulate a multifactorial etiology, as in many other chronic pain states and somatoform disorders, in which physical changes may interact with psychological factors, such as personality, mood, anxiety/tension, and so forth [7]. Moreover, Mrs. A’s case suggests-contrary to current opinion in stomatological and odontological literature [2,7]-that what triggers the symptoms may be psychogenic as well as physiogenic. Consequently, a DSM-IV diagnosis of “pain disorder associated with psychological factors” 1141may be established, at least in this particular case. The case of this patient and its successful therapeutic course nonetheless leaves some important
Mouth
theoretical questions unanswered. One of these concerns the complex and controversial rdationship between chronic pain and depression, which has “many faces” [15] and can be considered from an interpersonal, psychodynamic, and neurobiological perspective [16,17]. More concretely, the “masked depression” concept means that a patient can hide his mood disorder by presenting predominant physical symptoms. The dynamics of some patients with organically unexplained pain suggest that pain may be interpreted as a manifestation of unresolved guilt and self-punishing tendenciescomparable to the dynamics of some depressive patients, and in line with Engel’s [X3] pain-prone patient concept. Finally, from a neurobiological viewpoint, the relationship between pain and depression may be based on common neurotransmitter systems (mainly serotonergic, although there may be some noradrenergic involvement as well) [17,19]. The development and/or maintenance of Mrs. A’s painful condition may be described from all three theoretical perspectives, the relative importance of which is, however, far from clear. Similar complexities arise with regard to antidepressant therapy in chronic pain, because these medications may have a direct analgesic effect as well as several indirect effects, by ameliorating a reactive depression and/or anxiety, or by influencing an underlying “masked depression.” Although there is little doubt that Mrs. A suffered from major depression, which obviously responded to antidepressant treatment, more direct analgesic effects on her pain, or other indirect (e.g., tension relieving) effects cannot be excluded. A final unresolved question is the contribution of concurrent psychodynamically oriented psychotherapy to the amelioration of this patient. In any case, the results of the psychotherapy exceeded merely symptomatic relief and opened for Mrs. A the possibility for more emotional well-being and greater life satisfaction. From a diagnostic viewpoint, the case of Mrs. A demonstrates that, besides a clinical-psychiatric examination, an extensive psychodynamicbiographical anamnesis may be useful in BMS patients to reveal relevant information about possible associated life stress. This may not only shed light on the patient’s particular “symptom choice’*’ but also provide significant clues for psychotherapy. From a research perspective, this case may draw attention to the fact that contemporary scientific endeavor to unravel organically unexplained syndromes may underestimate the value of “‘idio-
B. Van Houdenhove
and P. Joostens
graphic” research strategies, such as the individual case study. As a matter of fact, case reports that include an in-depth study of psychological aspects are rare in BMS literature (for a notable exception, see [9]). On the other hand, too much seems to be expected from studies using psychological testing, e.g., with anxiety/depression scales [5,20] or life event questionnaires [S]. Although case report
data should be interpreted
with caution since they
may be subject to sample bias and carry the risk of
overinference, they have important heuristic value and may as such be considered as a necessary complement of psychometric group investigations. (For a further methodological discussion of this issue, particularly with regard to the possible etiological role of life events, see 121,221). Finally, the successful outcome of this patient’s
treatment suggests that it might be useful to more systematically investigate the use of novel antidepressants, notably serotonergic re-uptake inhibitors (SSRIs), in this indication. Although until now most studies have shown that a potential analgesic effect seems more likely with classical tricyclics [19,23], there is recent evidence that SSIUs may be effective as well [24]. In any case, SSRIs are better
tolerated by BMS patients because of the absence of an~cho~nergic side effects and particularly dry mouth.
References 1. Tourne LP, F&ton JR: Burning mouth syndrome. Critical review and proposed clinical management. Oral Surg Oral Med Oral Path01 74:158-X7, 1992 2. Mott AE, Grushka M, Sessle BJ: Diagnosis and management of taste disorders and burning mouth syndrome. Dent Clin N Am 37:55-71, 1993 3. Lamb AB, Lamey PJ, Reeve PE: Burning mouth syndrome: psychological aspects, Br Dent J 165:256-260, 1988 4. Van der Ploeg HM, van der Wal N, Eijkman MAJ, van der Waal I: Psycholo~cal aspects of patients with burning mouth syndrome. Oral Surg Oral Med Oral Path01 63664668, 1987 5. Rojo L, Silvestre FJ, Bagan JV, De Vicente T: Psychiatric morbidity in burning mouth syndrome. Psychiatric interview versus depression and anxiety scales. Oral Surg Oral Med Oral Path01 75308-311, 1993 6. Rojo L, Silvestre FJ, Bagan JV, De Vicente T: Prevalence of psychopathology in burning mouth syndrome: a comparative study among patients with and without psychiatric disorders and controls. Oral Surg Oral Med Oral Path01 78312316, 1994
388
7. Eii I, Baht R, Littner MM, Kleinhauz M: Detection of psychopathological trends in glossodynia patients. Psychosom Med 56389-394, 1994 8. Eli I, Kleinhauz M, Baht R, Littner M: Antecedents of burning mouth syndrome (glossodynia). Recent life events vs psychopathologic aspects. J Dent Res 73:567-572, 1994 9. Hammaren M, Hugoson A: Clinical psychiatric assessment of patients with burning mouth syndrome resisting oral treatment. Swed Dent J 1377-88, 1989 10. Gorsky DMD, Silverman S Jr, Chinn H: Clinical characteristics and management outcome in the burning mouth syndrome. Oral Surg Oral Med Oral Path01 72:192-195, 1991 11. Feinmann C, Harris M, Cawley R: Psychogenic facial pain: presentation and treatment. Br Med J 288: 436438, 1984 12. Sharav Y, Singer E, Schmidt E, Dionne RA, Dubner R: The analgesic effect of amitriptyline on chronic facial pain. Pain 31:199-209, 1987 13. Grushka M, Sessle BJ, Miller R: Pain and personality profiles in burning mouth syndrome. Pain 28:155167, 1987 14. American Psychiatric Association: Diagnostic and Statistical Manual of Mental Disorders, 4th ed. Washington DC, 1994 15. Roy R: Many faces of depression in patients with chronic pain. Int J Psychiatry Med 12:10%119, 1982 16. Van Houdenhove B: Interpersonal and psychodynamic links between pain and depression. Eur J Psychiatry 5:177-185, 1991 17. Magni G: On the relationship between pain and depression when there is no organic lesion. Pain 31:121, 1987 18. Engel G: Psychogenic pain and the pain-prone patient. Am J Med 26899-918, 1959 19. Onghena P, Van Houde~ove B: Antidepressantinduced analgesia in chronic non-malignant pain: a meta-analysis of 39 placebo-controlled studies. Pain 49:205-219, 1992 20. Lamey PJ, Lamb AB: The usefulness of the HAD scale in assessing anxiety and depression in patients with burning mouth syndrome. Oral Surg Oral Med Oral Path01 67:39O-392, 1989 21. Van Houdenhove B, Vasquez G, Neerinckx E: Tender points or tender patients? The value of the psychiatric in-depth interview for assessing and understanding psychopatholo~cal aspects of fibromyalgia. Clin Rheum 13:470-474, 1994 22. Miller TW: Stressful life events. Integr Psychiatry 8:4453, 1992 23. Eschalier A, Me&e C, Dubray C, Ardid D: Why are antidepressants effective as pain relief? CNS Drugs 2:261-267, 1994 24. Sindrup SH, Cram LF, Brosen K, Eshoj 0, Mogensen EF: The selective serotonin reuptake inhibitor paroxetine is effective in the treatment of diabetic neuropathy symptoms. Pain 42:135-144,199O
Van Houdenhove,
M.D. and Peter Joostens, M.D.
Abstract: A case is presented that illustrates the possible role of significant life events and depression in the etiology of the ~r~iffg mouth s~drome. Furt~r~re, successful tr~tme~t with a com~~ationof a selective serotoninye-uptake b~ocki#g antidepressantf’sertraline)and psychodynamicallyoriented psychotherapyis described.
The burning mouth syndrome (BMS) is a clinical entity characterized by a painful, burning sensation in the oral area, with no visual mucosal abno~alities on clinical examination. Mostly the tongue region is involved, hence, the older term “glossodynia.” However, in half the patients, other locations are involved, e.g., the frontal twothirds of the upper palate or the lower lip. During the last decade, BMS has received a great deal of attention in the stomatological and odontological literature, and Tourne and Fricton [l] and Mott et al. [2] have critically reviewed the present state of knowledge on the syndrome. The prevalence of BMS is still uncertain, as there are no epidemiological studies on the syndrome in general population groups in different parts of the world. However, BMS does not appear to be uncommon (e.g., according to some authors, it may be seen in 5% of a general dental population), and there is a consensus among clinicians and researchers that the syndrome is most prevalent in postmenopausal females [ 1,2]. Many etiological theories have been propounded, e.g., Candida glossitis, vitamin B defiDepartment of Psychiatry, Katholielce Universiteit Leuven, Leuven, Belgium Address reprint requests to: Prof. B. Van Houdenhove, Afdelmg Liaison-psychiatric, U.Z. Pellenberg, B-3212 Lubbeek, Belgium.
General Hospital Psychiatry 17, 385-388, 1995 0 1995 Elsevier Science Inc. 655 Avenue of the Americas, New York, NY 10010
ciencies, diabetes mellitus, estrogen deficiency at menopause, local factors such as inadequate dental bungs, denture wear, pasa~c~onal oral habits, sahvary disturbances, galvanism, and so forth. In spite of intense research efforts, no concmsive organic explanation for the cause of BMS has been found, and the condition is courtly considered as “idiopathic” by most authors [1,2]. Psychopathological factors have been frequently cited as associated with BMS. Lamb et al. [3] showed an occurrence of emotional instability, anxiety, and depression in 60% of a cohort of 47 patients with BMS. Van der Ploeg et al. [4] obtained similar results, with clearly higher ratings for depression, anxiety, and neuroticism. Recently, Rojo et al. [5,6] also found controlled evidence for more affective and neurotic d~~~a~~s (depression/anxiety symptoms, obsessive symptoms, somatization tendencies, and hostility) in a subgroup of BMS patients “with psychiatric disease.” Comparable findings were recently reported by Eli et al. [7J who found more premorbid psychiatric/psychological treatment in BMS patients as well; however, no differences between patients and controls were found on a questionnaire measuring associated stressful life events [S], This was in contrast to an earlier report of Hammaren and Hugoson [9] which showed the high prevalence of distressing life events in a selected group of BMS patients, using a semis~c~red psychiatric interview. From a therapeutic perspective, several forms of local oral treatment (e.g., denture adaptation, habit control) and systemic treatment (e.g., hormone or vitamin replacements) have been tried out, with disparate results (for a review see 11,211. Some authors found that nonresponders were psy385 ISSN 0143~8343J95/$9.50 ssxx 0163-8343(95)mo61-u
B. Van Houdenhove
and P. Joostens
chologically more disturbed than those responsive to treatment [3]. Other studies demonstrated a beneficial effect of tricyclic antidepressants and benzodiazepines on BMS, and this alleviation was shown to be present in both depressed and nondepressed subjects [U&12]. According to Eli et al. [7], the individual response to pain determines whether cure is possible (by either pharmacological, physical or psychological means) or whether the condition will develop into a chronic, persistent pathology. Summarizing the literature, Tourne and Fricton [l] state that multiple methodological flaws in BMS research make the reliable interpretation of the importance of proposed causal factors and the efficacy of specific treatment modalities difficult. This case report illustrates 1) the significance of psychopathological factors in BMS; 2) the importance of a thorough psychiatric investigation (including psychodynamic-biographical history taking) to detect them; and 3) the possible usefulness of combined psychotherapy and psychopharmacotherapy in these patients.
Case Report The patient, Mrs. A, was a 42-year-old married woman with one child, who was referred by the pain clinic of our university hospital for psychiatric assessment. She complained of unbearable pain in the frontal two-thirds of the tongue and the upper palate, that existed for about 10 months, and had commenced abruptly. Mrs. A contacted her family doctor who suspected a local infectious pathology and started a therapy with an antimycotic agent, without results. Afterwards, she consulted a dentist and two different E.N.T. specialists, who could not find any local lesion or systemic causative explanation, since screening for diabetes, hematological or biochemical abnormalities, and infectious disease proved negative. Vitamins Bl and 812 failed to help. Mrs. A had no clinical signs of climacterium, and laboratory testing for estrogen deficiency was negative. After 6 months, the family doctor suggested the possibility of a psychogenic mechanism, in view of the negative somatic investigations, and sent the patient to a psychotherapist, who started a classical psychoan~ysis. At the same time, the family doctor started dosulepine (a tricyclic antidepressant) 75 mg daily, but it had to be stopped a few weeks later because oral dryness made the pain worse. Some weeks later, Mrs. A also stopped her
analysis because the therapist “did not pay any attention to her pain.“ When she eventually attended the pain clinic, she was totally hopeless about her situation. At the first interview, we saw a well-groomed woman who expressed, in a slightly histrionic way, her feelings of hopelessness and despair caused by the unbearable pain. Although she denied being manifestly depressed (“It’s just the pain that makes me feel bad”), several clinical signs of depression were obvious, such as tiredness, apathy, social isolation, diminished concentration, insomnia, and diminished appetite which had led to a weight loss of 6 kg. Her pain symptoms were less prominent in the morning and reached their culminating point at night, and her mood showed an inverse pattern and was somewhat better in the evening. Mrs. A had no manifest suicidal thoughts, but there were vague passive death wishes (“I cannot continue to live in this way”). The personal background of the patient showed a minor depressive episode 17 years ago, and she also had a familial history of depression, with a suicidal attempt by the mother and a father with a post~aumatic stress syndrome. Impressed by the severity of the patient’s depressive state, we suggested an inpatient treatment at the psychiatric ward of our hospital. We promptly started sertraline 50 mg, which was increased to 100 mg after 2 weeks. This was combined with a further psychodynamic-biographical exploration of her life history. A major issue in these sessions was her distressing childhood as a consequence of an early divorce of her parents, whereafter she felt rejected by her father with whom she was never able to develop a warm affective relationship. She also complained about her nonsatisfactory marriage, with lack of attention from her husband, and feelings of boredom and emptiness for which she blamed the absence of a meaningful occupation. A few years ago, Mrs. A had started an extramarital love affair that focused on a passionate sexual relationship. Talking about her conflicting feelings in this relationship, she mentioned that the first pain symptoms occurred 2 days after she had oral sex with her lover, followed by a statement of his which she experienced as deeply humi~ating (“You are as good as a prostitute”). It was further noteworthy that, at the onset of the symptoms, she had been worrying several days about possibly contracting AIDS. After 3 weeks of treatment, the depressive symptoms gradually disappeared and the oral pain
lklrning
simultaneously diminished to an endurable level (“Some sandy feeling in the mouth”). She was discharged from the hospital and followed up with individual psychodynamic psychotherapy, once a week, in our consultation-liaison unit. In these sessions, Mrs. A was able to talk about the emotional problems in her life, especially her disappointing relationships with men, who appeared always to reject her, as her father did. She could express her ambivalent feelings toward her lover, and see some links between her physical pain and her After a few months, she painful emotions. planned to undertake marital counseling with her husband, and looked for a job which, she hoped, would make her life more meaningful. Meanwhile, the sertraline treatment was continued. One month after discharge, the pain had completely subsided, and this situation has been maintained until now, about 1 year later, with continued sertraline therapy 50 mg daily.
Discussion This case report first suggests that psychopathological factors can play an important role in BMS. Although several authors have observed that symptoms of depression, anxiety, irritability, tension, and hypochondriacal preoccupation frequently accompany the syndrome [3-lo], much controversy exists as to whether psychopathology is to be seen as primary, or secondary to the chronic pain state [4,7]. Advocates of the latter opinion point to the fact that psychological disturbances often diminish or disappear when the pain is successfully treated [13]. Evidently, it would seem adventurous to suggest a primary psychogenic etiology for a syndrome in which the role of organic etiological or pathophysiological factors is still unclear. It may be more appropriate to postulate a multifactorial etiology, as in many other chronic pain states and somatoform disorders, in which physical changes may interact with psychological factors, such as personality, mood, anxiety/tension, and so forth [7]. Moreover, Mrs. A’s case suggests-contrary to current opinion in stomatological and odontological literature [2,7]-that what triggers the symptoms may be psychogenic as well as physiogenic. Consequently, a DSM-IV diagnosis of “pain disorder associated with psychological factors” 1141may be established, at least in this particular case. The case of this patient and its successful therapeutic course nonetheless leaves some important
Mouth
theoretical questions unanswered. One of these concerns the complex and controversial rdationship between chronic pain and depression, which has “many faces” [15] and can be considered from an interpersonal, psychodynamic, and neurobiological perspective [16,17]. More concretely, the “masked depression” concept means that a patient can hide his mood disorder by presenting predominant physical symptoms. The dynamics of some patients with organically unexplained pain suggest that pain may be interpreted as a manifestation of unresolved guilt and self-punishing tendenciescomparable to the dynamics of some depressive patients, and in line with Engel’s [X3] pain-prone patient concept. Finally, from a neurobiological viewpoint, the relationship between pain and depression may be based on common neurotransmitter systems (mainly serotonergic, although there may be some noradrenergic involvement as well) [17,19]. The development and/or maintenance of Mrs. A’s painful condition may be described from all three theoretical perspectives, the relative importance of which is, however, far from clear. Similar complexities arise with regard to antidepressant therapy in chronic pain, because these medications may have a direct analgesic effect as well as several indirect effects, by ameliorating a reactive depression and/or anxiety, or by influencing an underlying “masked depression.” Although there is little doubt that Mrs. A suffered from major depression, which obviously responded to antidepressant treatment, more direct analgesic effects on her pain, or other indirect (e.g., tension relieving) effects cannot be excluded. A final unresolved question is the contribution of concurrent psychodynamically oriented psychotherapy to the amelioration of this patient. In any case, the results of the psychotherapy exceeded merely symptomatic relief and opened for Mrs. A the possibility for more emotional well-being and greater life satisfaction. From a diagnostic viewpoint, the case of Mrs. A demonstrates that, besides a clinical-psychiatric examination, an extensive psychodynamicbiographical anamnesis may be useful in BMS patients to reveal relevant information about possible associated life stress. This may not only shed light on the patient’s particular “symptom choice’*’ but also provide significant clues for psychotherapy. From a research perspective, this case may draw attention to the fact that contemporary scientific endeavor to unravel organically unexplained syndromes may underestimate the value of “‘idio-
B. Van Houdenhove
and P. Joostens
graphic” research strategies, such as the individual case study. As a matter of fact, case reports that include an in-depth study of psychological aspects are rare in BMS literature (for a notable exception, see [9]). On the other hand, too much seems to be expected from studies using psychological testing, e.g., with anxiety/depression scales [5,20] or life event questionnaires [S]. Although case report
data should be interpreted
with caution since they
may be subject to sample bias and carry the risk of
overinference, they have important heuristic value and may as such be considered as a necessary complement of psychometric group investigations. (For a further methodological discussion of this issue, particularly with regard to the possible etiological role of life events, see 121,221). Finally, the successful outcome of this patient’s
treatment suggests that it might be useful to more systematically investigate the use of novel antidepressants, notably serotonergic re-uptake inhibitors (SSRIs), in this indication. Although until now most studies have shown that a potential analgesic effect seems more likely with classical tricyclics [19,23], there is recent evidence that SSIUs may be effective as well [24]. In any case, SSRIs are better
tolerated by BMS patients because of the absence of an~cho~nergic side effects and particularly dry mouth.
References 1. Tourne LP, F&ton JR: Burning mouth syndrome. Critical review and proposed clinical management. Oral Surg Oral Med Oral Path01 74:158-X7, 1992 2. Mott AE, Grushka M, Sessle BJ: Diagnosis and management of taste disorders and burning mouth syndrome. Dent Clin N Am 37:55-71, 1993 3. Lamb AB, Lamey PJ, Reeve PE: Burning mouth syndrome: psychological aspects, Br Dent J 165:256-260, 1988 4. Van der Ploeg HM, van der Wal N, Eijkman MAJ, van der Waal I: Psycholo~cal aspects of patients with burning mouth syndrome. Oral Surg Oral Med Oral Path01 63664668, 1987 5. Rojo L, Silvestre FJ, Bagan JV, De Vicente T: Psychiatric morbidity in burning mouth syndrome. Psychiatric interview versus depression and anxiety scales. Oral Surg Oral Med Oral Path01 75308-311, 1993 6. Rojo L, Silvestre FJ, Bagan JV, De Vicente T: Prevalence of psychopathology in burning mouth syndrome: a comparative study among patients with and without psychiatric disorders and controls. Oral Surg Oral Med Oral Path01 78312316, 1994
388
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