Abstract
Background and aims
Burning mouth syndrome (BMS) and atypical facial pain (AFP) are often persistent idiopathic pain conditions that mainly affect middle-aged and elderly women. They have both been associated with various psychiatric disorders. This study examined current and lifetime prevalence of psychiatric axis I (symptom-based) and II (personality) disorders in patients with chronic idiopathic orofacial pain, and investigated the temporal relationship of psychiatric disorders and the onset of orofacial pain.
Method
Forty patients with BMS and 23 patients with AFP were recruited from Turku university hospital clinics. Mean age of the patients was 62.3 years (range 35–84) and 90% were female. BMS and AFP diagnoses were based on thorough clinical evaluation, and all patients had undergone clinical neurophysiological investigations including blink reflex and thermal quantitative tests. Current and lifetime DSM-IV diagnoses of axis I and II disorders were made on clinical basis with the aid of SCID-I and II-interviews. The detected prevalence rates and their 95% confidence intervals based on binomial distribution were compared to three previous large population-based studies.
Results
Of the 63 patients, 26 (41.3%) had had an axis I disorder that preceded the onset of orofacial pain, and 33 (52.4%) had had a lifetime axis I disorder. Rate of current axis I disorders was 36.5%, indicating that only about 16% of lifetime disorders had remitted, and they tended to run chronic course. The most common lifetime axis I disorders were major depression (30.2%), social phobia (15.9%), specific phobia (11.1%), and panic disorder (7.9%). Twelve patients (19.0%) had at least one cluster C personality disorder already before the emergence of orofacial pain. Patients with cluster C personality disorders are characterized as fearful and neurotic. None of the patients had cluster A (characterized as odd and eccentric) or B (characterized as dramatic, emotional or erratic) personality disorders. The most common personality disorders were obsessive–compulsive personality (14.3%), dependent personality (4.8%), and avoidant personality (3.2%). The majority of the patients (54%) had also one or more chronic pain conditions other than orofacial pain. In almost all patients (94%) they were already present at the onset of orofacial pain.
Conclusions
Our results suggest that major depression, persistent social phobia, and neurotic, fearful, and obsessive–compulsive personality characteristics are common in patients with chronic idiopathic orofacial pain. Most psychiatric disorders precede the onset of orofacial pain and they tend to run a chronic course.
Implications
We propose that the high psychiatric morbidity, and comorbidity to other chronic pain conditions, in chronic idiopathic orofacial pain can be best understood in terms of shared vulnerability to both chronic pain and specific psychiatric disorders, most likely mediated by dysfunctional brain dopamine activity.
1 Introduction
Burning mouth syndrome (BMS) and atypical facial pain (AFP) are persistent idiopathic pain conditions that mainly affect middle-aged and elderly women [1,2]. BMS is characterized by burning, usually bilateral oral mucosal pain, typically reported at more than one site [3,4]. It is likely that with stringent diagnostic exclusion criteria its prevalence in general adult population is between 1% and 5% [3,4,5,6]. AFP is described as a deep or superficial diffuse pain with constant or fluctuating intensity, typically spreading unilaterally out over a large area and not following peripheral neuroanatomic distributions [1,7]. No studies have been made of the prevalence of AFP in the general population, but it is obviously rare compared to BMS [1].
The exact pathophysiologies of BMS and AFP are unknown. Both peripheral and central neuropathic mechanisms have been suggested. PET studies have found alterations in brain dopamine activity in both BMS [8,9] and AFP [10], and disturbed inhibitory function of the cingulated gyrus in AFP [11]. Also, a functional magnetic resonance imaging study implied brain hypoactivity in BMS [12]. On the other hand, some indications of peripheral neuropathy have been revealed in both BMS [7,13,14,15,16] and AFP [7].
Between 1930 and 1980, most psychiatrists believed that idiopathic facial pain is either a hysterical conversion symptom or a symptom of an underlying psychiatric disorder [17,18,19,20]. In studies using structured psychiatric interviews, about half of the patients with BMS and AFP have had at least one life-time axis I disorder, depressive disorders being most frequent, followed by anxiety disorders [21,22,23,24,25,26]. Neurotic, hostile, emotionally detached and obsessive–compulsive personality traits have been found to be common among patients with chronic idiopathic orofacial pain [19,21,23,27,28,29,30]. To our knowledge, the prevalence of personality disorders in patients with chronic idiopathic orofacial pain has been investigated using structured clinical interview in only one study. Maina et al. [31] found that 86% of their 70 patients with BMS had at least one personality disorder, dependent and obsessive–compulsive personalities being the most common ones.
The aims of this study were: (a) to examine current and life-time prevalence of psychiatric axis I and II disorders in patients with chronic idiopathic orofacial pain, and (b) to investigate the temporal relationship of psychiatric disorders and orofacial pain.
2 Materials and methods
2.1 Patients
Patients with BMS and AFP were recruited for psychiatric interview from the Departments of Neurology or Oral diseases of Turku University Hospital. BMS and AFP diagnoses were based on thorough clinical evaluation described in detail earlier [7,13,32]. All patients with AFP had been previously examined by a neurologist and a dentist, an all except for one by an otorhinolaryngologist. In addition, all patients had undergone clinical neurophysiological testing including blink reflex test and thermal quantitative sensory testing. Patients were recruited for psychiatric evaluation by a letter including complete description of the study and by a telephone call few days later. Exclusion criterion for the study was severe cognitive decline, and four patients were excluded. 13 patients refused to participate in the psychiatric evaluation and one patient could not be reached. Forty patients with BMS and 23 patients with AFP formed the study group, and the participation rate among eligible patients was 82%. Mean age of the patients was 62.3 years (SD 11.3, range 35–84) and the majority of them were female (Table 1). The study protocol was approved by the joint Ethical Committee of Turku University Hospital and University of Turku, and all participants gave their written informed consent.
Characteristics of 63 patients with chronic idiopathic orofacial pain: 40 patients with burning mouth syndrome (BMS) and 23 patients with atypical facial pain (AFP).
| Patients with BMS | Patients with AFP | P value | All patients | |
|---|---|---|---|---|
| Sex | ||||
| Male (%) | 3 (7.5) | 3 (13.0) | 0.66 | 6 (9.5) |
| Female (%) | 37 (92.5) | 20 (87.0) | 57 (90.5) | |
| Age, mean (SD, range) | 64.9 (9.7,35–84) | 57.7 (12.5,37–81) | 0.013 | 62.3 (11.3,35–84) |
| Socioeconomic class (%) | ||||
| Unskilled | 9 (22.5) | 1 (4.3) | 0.098 | 10 (15.9) |
| Blue collar[a] | 27 (67.5) | 17 (73.9) | 44 (69.8) | |
| White collar[a] | 4 (10.0) | 5 (21.7) | 9 (14.3) | |
| Managerial | 0 (0) | 0 (0) | 0 (0) | |
| Duration of orofacial pain in years, mean (SD, range) | 7.4 (4.2,1–18) | 11.9 (8.6, 2–35) | 0.026 | 9.1 (11.3,1–35) |
| Any lifetime | 22 (55.0) | 11 (47.8) | 0.58 | 33 (52.4) |
| Axis I (symptom-based) disorder (%)[b] | ||||
| Depressive disorder | 14 (35.0) | 6 (26.1) | 0.46 | 20 (31.7) |
| Bipolar disorder | 1 (2.5) | 0 (0) | 1.0 | 1 (1.7) |
| Anxiety disorder | 19 (47.5) | 7 (30.4) | 0.18 | 26 (41.2) |
| Alcohol dependence | 0 (0) | 2 (8.7) | 0.13 | 2 (3.3) |
| Any lifetime | 6 (15.9) | 6 (26.1) | 0.14 | 12 (19.0) |
| Axis II (personality) disorder (%)[b] | ||||
| Obsessive-compulsive personality | 5 (12.5) | 4 (17.4) | 0.59 | 9 (15.0) |
| Avoidant personality | 1 (2.5) | 1 (4.3) | 1.0 | 2 (3.2) |
| Dependent personality | 2 (5.0) | 1 (4.3) | 1.0 | 3 (4.8) |
2.2 Psychiatric assessment
Psychiatric interviews were conducted by physicians, who were trained to use the instruments. Current (previous month) and life-time DSM-IV [33] diagnoses of axis I disorders were made on clinical basis with the aid of the structured clinical interview for DSM-IV axis I disorders (SCID-I) [34]. Personality disorders were assessed independently of axis I disorders with the SCID-II interview [35]. Lifetime axis I and II disorders were further divided into disorders with onset before and after the onset of orofacial pain. Patients’ personal history and history of somatic complaints were also recorded. Patients’ somatic and psychiatric patient records of Turku University Hospital were available for interviewers. Duration of psychiatric interviews ranged from 2 to 4 h.
2.2 Statistical methods
Descriptive statistics, such as means, standard deviations, ranges for continuous variables, and frequencies and percentages for categorical variables, were used to assess the patients’ back-ground characteristics and diagnostic distribution. Comparisons between the BMS and AFP patients were carried out with Students t-tests, chi-square tests and Fisher’s exact tests. 95% confidence intervals (CIs) based on binomial distribution were calculated for the rates of psychiatric disorders. CIs based on the rates in 63 patients are relatively broad. Two prevalence rates diverge significantly from each other if their 95% CIs do not overlap. In an attempt to avoid comparing two broad CIs with each other for every disorder, we did not use a specific comparison group for this study. Instead, to assist the reader to compare the prevalence rates between our sample and general population, we calculated 95% CIs based on binomial distribution for three previous large population-based studies, referred in the discussion, from their original data [36,37,38,39]. Statistical analyses were conducted using SAS version 8.00-software (SAS Institute, 1999).
3 Results
3.1 Comparison between BMS and AFP patients
Patients with BMS were an average 7.2 years older and the duration of their orofacial pain was an average 4.5 years shorter compared with AFP patients (Table 1). Of the 40 BMS patients, 22 (55%) had at least one lifetime axis I disorder and 6 (16%) at least one personality disorder. The corresponding figures in the group of 23 AFP patients were 11 (48%) and 6 (26%), respectively, and the differences between BMS and AFP patients were not significant (Table 1). The prevalence rates of all axis I disorders between BMS and AFP patients were compared also separately, and no significant differences emerged. The only nearly significant difference (P = 0.052) was in the rate of lifetime panic disorder, which was somewhat more common in AFP patients (17.4% vs. 2.5%). Because there were no differences in the rates of axis I and II disorders between BMS and AFP patients, the prevalence rates of the combined group (N = 63) are presented in Table 2.
Rates of DSM-lV axis I and II psychiatric disorders, by onset, in 63 patients with chronic idiopathic orofacial pain: 40 patients with burning mouth syndrome (BMS) and 23 patients with atypical facial pain (AFP).
| Diagnosis | Onset before orofacial pain | Onset after orofacial pain | Current (previous month) | Lifetime | ||||
|---|---|---|---|---|---|---|---|---|
|
|
|
|
|
|||||
| N | % (95% CI) | N | % (95% CI) | N | % (95% CI) | N | % (95% CI) | |
| Axis I disorders | ||||||||
| Major depressive disorder | 13 | 20.6 (11.5–32.7) | 6 | 9.5 (3.6–19.6) | 8 | 12.7 (5.6–23.5) | 19 | 30.2 (19.2–43.0) |
| Chronic depression | 1 | 1.6 (0.04–8.5) | 0 | 0 | 1 | 1.6 (0.04–8.5) | 1 | 1.6 (0.04–8.5) |
| Bipolar l disorder | 1 | 1.6 (0.04–11.0) | 0 | 0 | 1 | 1.6 (0.04–8.5) | 1 | 1.6 (0.04–8.5) |
| Generalized anxiety disorder | 0 | 0 | 2 | 3.2 (0.4–11.0) | 1 | 1.6 (0.04–8.5) | 2 | 3.2 (0.4–11.0) |
| Specific phobia | 6 | 9.5 (3.6–19.6) | 1 | 1.6 (0.04–8.5) | 7 | 11.1 (4.6–21.6) | 7 | 11.1 (4.6–21.6) |
| Social phobia | 10 | 15.9 (7.9–27.3) | 0 | 0 | 8 | 12.7 (5.6–23.5) | 10 | 15.9 (7.9–27.3) |
| Panic disorder | 3 | 4.8 (1.0–13.3) | 2 | 3.2 (0.4–11.0) | 3 | 4.8 (1.0–13.3) | 5 | 7.9 (2.6–17.6) |
| Post-traumatic stress disorder | 2 | 3.2 (0.4–11.0) | 0 | 0 | 1 | 1.6 (0.04–8.5) | 2 | 3.2 (0.4–11.0) |
| Alcohol dependence | 1 | 1.6 (0.04–8.5) | 1 | 1.6 (0.04–8.5) | 1 | 1.6 (0.04–8.5) | 2 | 3.2 (0.4–11.0) |
| Any axis-I disorder | 26 | 41.3 (29.0–54.4) | 7 | 11.1 (4.6–21.6) | 23 | 36.5 (24.7–49.6) | 33 | 52.4 (39.4–65.1) |
| Axis II personality disorders | ||||||||
| Obsessive-compulsive personality | 9 | 14.3 (6.7–25.4) | 0 | 0 | 9 | 14.3 (6.7–25.4) | 9 | 14.3 (6.7–25.4) |
| Avoidant personality | 2 | 3.2 (0.4–11.0) | 0 | 0 | 2 | 3.2 (0.4–11.0) | 2 | 3.2 (0.4–11.0) |
| Dependent personality | 3 | 4.8 (1.0–13.3) | 0 | 0 | 3 | 4.8 (1.0–13.3) | 3 | 4.8 (1.0–13.3) |
| Any personality disorder | 12 | 19.0 (10.2–30.1) | 0 | 0 | 12 | 19.0 (10.2–30.1) | 12 | 19.0 (10.2–30.1) |
3.2 Axis I disorders
In DSM-IV [33], axis I refers to disorders causing clear-cut psychiatric symptoms, e.g. anxiety or depression. Of the 63 patients assessed, 33 (52.4%, 95% CI = 39.4–65.1%) had at least one lifetime axis I disorder (Table 2). The most common lifetime disorders were major depression (30.2%, 95% CI = 19.2–43.0%), social phobia (15.9%, 95% CI = 7.9–27.3%), specific phobia (11.1%, 95% CI = 4.6–21.6%) and panic disorder (7.9%, 95% CI = 2.6–17.6%). None of the patients had a psychotic disorder. The specific phobias in seven patients were: (number of patients with the phobia in parenthesis, one patient had two phobias): high places (2), closed places (2), snakes (1), flying (1), spiders (1), and deep water (1).
Comorbidity among lifetime axis I disorders was observed in 15 (45%) of the 33 patients. Eight of the patients had two and seven patients three comorbid axis I disorders. The majority of axis I disorders (79%) had their onset before the emergence of orofacial pain. Psychiatric disorders of patients tended to run a chronic course. Rate of all current axis I disorders was 36.5% (95% CI = 24.7–49.6%), indicating that only about 16% of lifetime disorders had remitted.
3.3 Axis II disorders
In DSM-IV [33], axis II refers to personality disorders. They are divided into three clusters, A, B and C. Patients in the cluster A are characterized as odd and eccentric, in the cluster B as dramatic, emotional or erratic, and in the cluster C as fearful and neurotic. Personality disorders were found in 12 patients (19%, 95% CI = 10.2–30.1%) and they all preceded the onset of orofacial pain. The personality disorders present among the patients were obsessive–compulsive personality (14.3%, 95% CI = 6.7–25.4%), dependent personality (4.8%, 95% CI = 1.0–13.3%) and avoidant personality (3.2%, 95% CI = 0.4–11.0%). Comorbidity among personality disorders was observed in two patients. Eight out of 12 patients (67%) with personality disorders had also a life-time axis I disorder, and about one-fourth of the patients with a lifetime axis I disorder (8 of 33, 24.2%) had a comorbid personality disorder. All three personality disorders present among our patients belonged to the cluster C.
3.4 Chronic painful physical condition other than orofacial pain
Lifetime chronic painful physical conditions were found in 34 patients (54.0%, 95% CI = 40.1–66.6%). The localizations of the painful conditions were (number of patients with the painful condition in parenthesis, many patients had more than one pain condition): joint/articular (18), limb (5), back (15), headache (4), gastrointestinal pain (8), and coronary pain (2). In 17 patients (27.0%) chronic pain condition was comorbid with an axis I disorder and in five patients (7.9%) with an axis II disorder. In almost all patients (32 out of 34, 94%) chronic painful physical conditions were already present at the onset of orofacial pain.
4 Discussion
The main findings of the present study were high rates of major depression and cluster C personality disorders, lack of cluster A and B personality disorders, and a tendency of the psychiatric disorders to run a chronic course. In the present study, the rate of current (previous month) major depression (12.7%, 95% CI = 5.6–23.5%) was markedly higher than both the 12-month rate of major depression in the National Comorbidity Survey Replication (NCS-R) in the U.S. (6.7%, 95% CI = 6.2–7.2%) [36], and the 12-month rate of major depression among females aged 30 years and over in the Health 2000 Study in Finland (4.9%, 95% CI = 5.5–7.2%) [38]. The rate of lifetime major depression (30.2%, 95% CI 19.2–43.0%) was significantly higher than in the NCS-R survey (16.6%, 95% CI = 15.8–17.4%) [37]. Majority of the depressive disorders (14/20, 70%) among our subjects preceded the onset of orofacial pain. This finding is in line with earlier reports of high rates of depressive disorders already before the onset of orofacial pain [18,24,40].
In our subjects, the lifetime rates of anxiety disorders were only slightly higher than in population surveys. The lifetime rate of the most common anxiety disorder in our sample, social phobia, 15.9% (95% CI = 7.9–27.3%), was little higher than in the NCS-R survey, 12.1% [36]. The one-month rate of social phobia, 12.7% (95% CI = 5.6–23.5%), was significantly higher than the 12-month rate of social phobia in Finnish female population aged 30 years and over (0.9%, 95% CI = 0.6–1.3%) in the Health 2000 Study [38]. This insinuates that in patients with BMS and AFP social phobia is both more common and more persistent than in the general population. In all patients with social phobia its onset preceded the emergence of facial pain. The lifetime rate of specific phobia, 11.1% (95% CI = 4.6–21.6%), was about the same as in the NCS-R survey (12.1% and 12.5%) [37]. The lifetime rates of panic disorder (7.9%, 95% CI = 2.6–17.6%), generalized anxiety disorder (3.2%, 95% CI = 0.4–11.0%), post-traumatic stress disorder (3.2%, 95% CI = 0.4–11.0%) and alcohol dependence (3.2%, 95% CI = 0.4–11.0%) did not differ significantly from the lifetime rates in the NCS-R survey (4.7%, 5.7%, 6.8% and 5.4%, respectively) [37].
In our sample, the one-month and lifetime rates of at least one axis I disorder were 36.5% (95% CI = 24.7–49.6%) and 52.4% (95% CI = 39.4–65.1%), respectively. The one-month rate was significantly higher than the12-month rate among Finnish females aged 30 years and over, 11.8% (95% CI = 10.7–12.9%), in the Health 2000 Study [38], but the lifetime rate was only slightly higher than in the NCS-R survey [37], 46.4% (95% CI = 45.1–47.7%). These findings imply that in patients with BMS and AFP, axis I disorders tend to run a more chronic course than in the general population. Majority of axis I disorders, 26/33, 79%, preceded the onset of orofacial pain.
In the present study group, 19.0% (95% CI = 10.2–30.1%) of the patients had at least one personality disorder. This rate is significantly higher than in the NCS-R survey (9.1%, 95% CI = 8.4–9.9%) [39]. All personality disorders in our sample belonged to the ‘fearful and neurotic’ cluster, cluster C, and all of them preceded the onset of orofacial pain. There were no patients with either cluster A or cluster B personality disorders. The rate of cluster C personality disorders in the NCS-R survey, 6.0% (95% CI = 5.4–6.6%) [39], was strikingly lower than the rate in the present sample. In our patients, the rate of obsessive–compulsive personality was particularly high, 14.3% (95% CI = 6.7–25.4%) compared with the rate in the NCS-R survey, 2.4% [39]. The lack of cluster B personality disorders can be partly explained by the high age of our patients (mean 62.3 years), because cluster B disorders tend to decline with advancing age [41]. Our results differ markedly from the previous findings by Maina et al. [31]. In their sample of 102 patients with BMS, at least one personality disorder was found in 85.7% (95% CI = 75.3–92.9%) of the patients [31]. In line with our results, also in their material the highest prevalence rate was found for cluster C personality disorders (42.8%, 95% CI = 31.1–55.2%), and obsessive–compulsive personality disorder was the most common disorder (30.0%, 95% CI = 19.6–42.1%) [31]. It is of interest, that a high rate of cluster C personality disorders, obsessive–compulsive personality disorder in particular, has also been found in patients with fibromyalgia [42]. As a whole, these results are in line with previous studies evaluating personality traits, suggesting that patients with AP and BMS often tend to be neurotic and obsessive–compulsive [19,21,23,28,29,30].
Lifetime chronic painful physical conditions other than orofacial pain were found in 34 patients (54.0%, 95% CI = 40.1–66.6%), and in almost all patients (94%) chronic pain condition preceded the onset of orofacial pain. In a large survey of a general population sample in five European countries, only 28.6% (95% CI 27.7–29.5%) of the females aged 65 or over reported having at least one chronic painful physical condition [43]. This suggests that idiopathic orofacial pain has a tendency to be comorbid with other chronic pain conditions. Türp et al. [44] have also found that in the majority of female patients with persistent facial pain, the pain distribution was widespread.
Theoretically, chronic idiopathic pain could be associated with psychiatric disorders by several mechanisms [45,46]: (a) there could be a common genetic predisposition to both chronic pain and psychiatric disorders mediated, e.g. by low brain dopamine activity [8–10,47,48]; (b) stress could predispose a patient to both psychiatric disorders and pain by causing reduction of dopamine output in brain [49]; (c) psychiatric disorders could induce alterations in brain functions, which in turn expose patients to pain disorders [50]; (d) chronic idiopathic pain could be either a psychiatric symptom or a psychiatric disorders per se [18,20]; (e) chronic pain could predispose a patient to psychiatric disorders by altering reward-aversion circuitry [51]; and (f) suffering caused by pain could expose a patient to psychiatric disorders and even initiate personality changes [28,45].
Major depression, social phobia and cluster C personality disorders are clinically quite different psychiatric conditions. Nevertheless, both major depression and social phobia have been linked with decreased, and obsessive–compulsive personality with dysfunctional brain dopamine activity [52,57,58]. Cluster C personality disorders, in general, are characterized by low novelty seeking, neuroticism, persistence, high harm avoidance and fearfulness–traits, which all have been connected to low brain dopamine activity [59,60,61,62]. On the other hand, cluster B personality disorders, which were totally absent in our material, are characterized by extroversion, high novelty seeking, impulsivity and low harm avoidance, traits which have been associated with a high brain dopamine activity [60,61,62,63]. Pain is a non-motor symptom of Parkinson disease [64], and increasing evidence imply that reduced dopamine activity is associated with pain reception in general [65,66], and also with chronic idiopathic orofacial pain [8–10,47,67] and fibromyalgia [68,69].
Vulnerability caused by reduced dopamine activity could explain the fact that BMS and AFP affect mostly elderly women. Both post-mortem [70] and PET-studies [71,72] have shown that dopamine activity in brain declines with advancing age. PET-studies have found gender differences in brain dopamine activity, particularly in older age groups [73,74], and, in the rat brain, estradiol treatment increases dopamine receptor density [75]. Taken together, results of the present study and previous findings considering the role of dopamine in pain and particular psychiatric disorders, are in accordance with the assumptions (a)–(c) above. Both axis I and II comorbidity is characteristic for psychiatric disorders [36]. Absence of cluster A and B personality disorders, and a high comorbidity with only specific axis I disorders in our patients, do not give support to mechanism (d) above. With an assumption that our patients could recall the temporal relationships of the various disorders accurately, the finding that 79% of axis I disorders and all personality disorders preceded the onset of orofacial pain makes assumptions (e) and (f) unlikely.
The strengths of our study include thorough diagnostic evaluation of all patients by a multidisciplinary team with several medical specialities, and the assessment of both axis I and II psychiatric disorders with structured instruments. This study has also some limitations. First, our patients were originally referred to the university clinic, and therefore, the findings may not be generalized to all patients with chronic idiopathic orofacial pain. Second, the lack of a comparison group can be regarded as a shortcoming. On the other hand, it is often more productive to compare the broad CIs of a relatively small sample with the narrow CIs of a large population sample than with the broad CIs of a small comparison group [76]. Third, psychiatric and personality disorders may have been more common among those thirteen patients who refused to participate. Fourth, the reliability of retrospective diagnoses and temporal relationship between psychiatric disorders and the onset of orofacial pain may have been compromised by memory disturbances. However, both in a previous study using the same interview method [76] and in the present study, there were no major discrepancies between the somatic and psychiatric symptoms reported by the patients, and their previous hospital records.
5 Conclusions
Our results suggest that major depression, persistent social phobia, and neurotic, fearful, and obsessive–compulsive personality characteristics are common in patients with chronic idiopathic orofacial pain. Most psychiatric disorders precede the onset of orofacial pain and they tend to run a chronic course. We propose that the high psychiatric morbidity in chronic idiopathic orofacial pain can be best understood in terms of shared vulnerability to both chronic pain and specific psychiatric disorders, most likely mediated by dysfunctional brain dopamine activity.
DOI of refers to article: 10.1016/j.sjpain.2011.08.003.
Acknowledgements
This study was supported by the Finnish Medical Foundation and the Sigrid Juselius Foundation.
References
[1] Woda A, Pionchon P. A unified concept of idiopathic orofacial pain: clinical features. J Orofac Pain 1999;13:172–84.Search in Google Scholar
[2] Woda A, Tubert-Jeannin S, Bouhassira D, Attal N, Fleiter B, Goulet J-P, Gremeau-Richard C, Navez ML, Picard P, Pionchon P, Albuisson E. Towards a new taxonomy of idiopathic orofacial pain. Pain 2005;116:396–406.Search in Google Scholar
[3] Bergdahl M, Bergdahl J. Burning mouth syndrome: prevalence and associated factors. J Oral Pathol Med 1999;28:350–4.Search in Google Scholar
[4] Scala A, Checchi L, Montevecchi M, Marini I. Update on burning mouth syndrome: overview and patient management. Crit Rev Oral Biol Med 2003;14:275–91.Search in Google Scholar
[5] Bergdahl J, Anneroth G. Burning mouth syndrome: literature review and model for research and management. J Oral Pathol Med 1993;22:433–8.Search in Google Scholar
[6] Lipton JA, Ship JA, Larach-Robinson D. Estimated prevalence and distribution of reported orofacial pain in the United States. J Am Den Assoc 1993;124:115–21.Search in Google Scholar
[7] Forssell H, Tenovuo O, Silvoniemi P, Jääskeläinen S. Differences and similarities between atypical facial pain and trigeminal neuropathic pain. Neurology 2007;69:1451–9.Search in Google Scholar
[8] Jääskeläinen SK, Rinne JO, Forssell H, Tenovuo O, Kaasinen V, Sonninen P, Bergman J. Role of dopaminergic system in chronic pain—a fluorodopa-PET study. Pain 2001;90:257–60.Search in Google Scholar
[9] Hagelberg N, Forssell H, Aalto S, Rinne JO, Scheinin H, Taiminen T, Aalto S, Luutonen S, Någren K, Jääskeläinen S. Striatal dopamine D1 and D2 receptors in burning mouth syndrome. Pain 2003;101:149–54.Search in Google Scholar
[10] Hagelberg N, Forssell H, Aalto S, Rinne JO, Scheinin H, Taiminen T, Någren K, Eskola O, Jääskeläinen SK. Altered dopamine D2 receptor binding in atypical facial pain. Pain 2003;106:43–8.Search in Google Scholar
[11] Derbyshire SWG, Jones AKP, Devani P, Friston KJ, Feinman C, Harris M, Pearce S, Watson JD, Frackowiak RS. Cerebral responses to pain in patients with atypical facial pain measured by positron emission tomography. J Neurol Neurosurg Psychiatry 1994;57:1166–72.Search in Google Scholar
[12] Albuquerque RJC, de Leeuw R, Carlson CR, Okeson JP, Miller CS, Andersen AH. Cerebral activation during thermal stimulation of patients who have burning mouth disorder: an fMRI study. Pain 2006;122:223–34.Search in Google Scholar
[13] Forssell H, Jääskeläinen S, Tenovuo O, Hinkka S. Sensory dysfunction in burning mouth syndrome. Pain 2002;99:41–7.Search in Google Scholar
[14] Lauria G, Majorana A, Borgna M, Lombardi R, Penza P, Padovani A, Sapelli P. Trigeminal small-fibre sensory neuropathy causes burning mouth syndrome. Pain 2005;115:332–7.Search in Google Scholar
[15] Yilmaz Z, Renton T, Yiangou Y, Zakrzewska J, Chessell IP, Bountra C, Anand P. Burning mouth syndrome as a trigeminal small fibre neuropathy: increased heat and capsaicin receptor TRPV1 in nerve fibres correlates with pain score. J Clin Neurosci 2007;14:864–71.Search in Google Scholar
[16] Greméau-Richard C, Dubray C, Aublet-Cuvelier B, Ughetto S, Woda A. Effect of lingual nerve block on burning mouth syndrome (stomatodynia): a randomized controlled trial. Pain 2010;149:27–32.Search in Google Scholar
[17] Gilpin SF. Glossodynia. JAMA 1936;106:1722–4.Search in Google Scholar
[18] Lascelles RG. Atypical facial pain and depression. Br J Psychiatry 1966;112:651–9.Search in Google Scholar
[19] Feinmann C, Harris M, Cawley R. Psychogenic facial pain: presentation and treatment. BMJ 1984;288:436–8.Search in Google Scholar
[20] Ott G, Ott C. Glossodynia—psychodynamic basis and results of psychopathometric investigations. J Psychosomatic Res 1992;36:677–86.Search in Google Scholar
[21] Remick RA, Blasberg B, Campos PE, Miles JE. Psychiatric disorders associated with atypical facial pain. Can J Psychiatry 1983;28:178–81.Search in Google Scholar
[22] 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 Pathol 1993;75:308–11.Search in Google Scholar
[23] Rojo L, Silvestre FJ, Bagan JV. Prevalence of psychopathology in burning mouth syndrome. A comparative study among patients with and without psychiatric disorders and controls. Oral Surg Oral Med Oral Pathol 1994;73:312–6.Search in Google Scholar
[24] Bogetto F, Maina G, Ferro G, Carbone M, Gandolfo S. Psychiatric comorbidity in patients with burning mouth syndrome. Psychosom Med 1998;60:378–85.Search in Google Scholar
[25] Nicholson M, Wilkinson G, Field E, Longman L, Fitzgerald B. A pilot study: stability of psychiatric diagnoses over 6 months in burning mouth syndrome. J Psychosom Res 2000;49:1–2.Search in Google Scholar
[26] Lang E, Kaltenhäuser M, Seidler S, Mattenklodt P, Neundörfer B. Persistent idiopathic facial pain exists independent of somatosensory input from the painful region: findings from quantitative sensory functions and somatotopy of the primary somatosensory cortex. Pain 2005;118:80–91.Search in Google Scholar
[27] Solomon S, Lipton RB. Atypical facial pain: a review. Semin Neurol 1988;8:332–8.Search in Google Scholar
[28] Gouda JJ, Brown JA. Atypical facial pain and other pain syndromes. Neurosurg Clin North Am 1997;8:87–100.Search in Google Scholar
[29] Bergdahl J, Anneroth G, Perris H. Personality characteristics of patients with resistant burning mouth syndrome. Acta Odontol Scand 1995;53:7–11.Search in Google Scholar
[30] Trikkas G, Nikolatou O, Samara C, Bazopoulou-Kyrkanodou E, Rabavilas AD, Christodoulou GN. Glossodynia: personality characteristics and psychopathology. Psychother Psychosom 1996;65:163–8.Search in Google Scholar
[31] Maina G, Albert U, Gandolfo S, Vitalucci A, Bogetto F. Personality disorders in patients with burning mouth syndrome. J Personal Disord 2005;19:84–93.Search in Google Scholar
[32] Jääskeläinen SK, Forssell H, Tenovuo O. Electrophysiological testing of the trigeminofacial system: aid in the diagnosis of atypical facial pain. Pain 1999;80:191–200.Search in Google Scholar
[33] American Psychiatric Association, editor. Diagnostic and statistical manual of mental disorders (DSM-IV). 4th ed. Washington, DC: APA; 1994.Search in Google Scholar
[34] First MB, Spitzer RL, Gibbon M, Williams JBW. Structured clinical interview for DSM-IV axis I disorders (SCID-I, 4/97 version). New York, NY: Biometrics Research Department, New York State Psychiatric Institute; 1997.Search in Google Scholar
[35] First MB, Spitzer RL, Gibbon M, Williams JBW, Benjamin L. Structured clinical interview for DSM-IV axis II disorders (SCID-II). New York, NY: Biometrics Research Department, New York State Psychiatric Institute; 1997.Search in Google Scholar
[36] Kessler RC, Chiu WT, Demler O, Walters EE. Prevalence, severity, and comorbidity of 12-month DSM-IV disorders in the National Comorbidity Survey Replication. Arch Gen Psychiatry 2005;62:617–27.Search in Google Scholar
[37] Kessler RC, Berglund P, Demler O, Jin R, Walters EE. Lifetime prevalence and age-of-onset distributions of DSM-IV disorders in the National Comorbidity Survey Replication. Arch Gen Psychiatry 2005;62:593–602.Search in Google Scholar
[38] Pirkola SP, Isometsä E, Suvisaari J, Aro H, Joukamaa M, Poikolainen K, Koskinen S, Aromaa A, Lönnqvist J. DSM-IV mood-, anxietyand alcohol use disorders and their comorbidity in the Finnish general population: results from the Health 2000 Study. Soc Psychiatry Psychiatr Epidemiol 2005;40:1–10.Search in Google Scholar
[39] Lenzenweger MF, Lane MC, Loranger AW, Kessler RC. DSM-IV personality disorders in the National Comorbidity Survey Replication. Biol Psychiatry 2007;62:553–64.Search in Google Scholar
[40] Lamey P-J, Lamb AB. Prospective study of aetiological factors in burning mouth syndrome. BMJ 1988;296:1243–6.Search in Google Scholar
[41] Reich J, Nduaguba M, Yates W. Age and sex distribution of DCM III personality cluster traits in a community population. Compr Psychiatry 1988;29: 298–303.Search in Google Scholar
[42] Uguz F, Cicek E, Salli A, Karahan AY, Albayrak I, Kaya N, Ugurlu H. Axis I and II psychiatric disorders in patients with fibromyalgia. Gen Hosp Psychiatry 2010;32:105–7.Search in Google Scholar
[43] Ohayon MM, Scatzberg AF. Using chronic pain to predict depressive morbidity in the general population. Arch Gen Psychiatry 2003;60:39–47.Search in Google Scholar
[44] Türp JC, Kowalski CJ, O’Leary N, Stohler CS. Pain maps from facial pain patients indicate a broad pain geography. J Dent Res 1998;77:1465–72.Search in Google Scholar
[45] Dersh J, Polatin PB, Gatchel RJ. Chronic pain and psychopathology: research findings and theoretical considerations. Psychosom Med 2002;64:773–86.Search in Google Scholar
[46] Diatchenko L, Nackley AG, Slade GD, Fillingim RB, Maixner W. Idiopathic pain disorders—pathways of vulnerability. Pain 2006;123:226–30.Search in Google Scholar
[47] Jääskeläinen SK, Forssell H, Tenovuo O. Abnormalities of the blink reflex in burning mouth syndrome. Pain 1997;73:455–60.Search in Google Scholar
[48] Zubieta JK, Heitzeg MM, Smith YR, Bueller JA, Xu K, Xu Y, Koeppe RA, Stohler CS, Goldman D. COMT val158 met genotype affects mu-opioid neurotransmitter responses to a pain stressor. Science 2003;299:1240–3.Search in Google Scholar
[49] Wood PB. Stress and dopamine: implications for the pathophysiology of chronic widespread pain. Med Hypothesis 2004;62:420–4.Search in Google Scholar
[50] Strigo IA, Simmons AN, Matthews SC, Craig AD, Paulus MP. Association of major depressive disorder with altered functional brain response during anticipation and processing of heat pain. Arch Gen Psychiatry 1998;65:1275–84.Search in Google Scholar
[51] Borsook D, Becerra L, Carlezon WA, Shaw M, Renshaw P, Elman I, Levine J. Reward-aversion circuitry in analgesia and pain: implications for psychiatric disorders. Eur J Pain 2007;11:7–20.Search in Google Scholar
[52] Tiihonen J, Kuikka J, Bergström K, Lepola U, Koponen H, Leinonen E. Dopamine reuptake site densities in patients with social phobia. Am J Psychiatry 1997;154:239–42.Search in Google Scholar
[53] Lambert G, Johansson M, Ågren H, Friberg P. Reduced brain norepinephrine and dopamine release in treatment-refractory depressive illness. Arch Gen Psychiatry 2000;57:787–93.Search in Google Scholar
[54] Schneier FR, Liebowitch MR, Abi-Dargham A, Zea-Ponce Y, Lin SH, Laruelle M. Low dopamine D2 receptor binding potential in social phobia. Am J Psychiatry 2000;157:457–9.Search in Google Scholar
[55] Klimek V, Schenck JE, Han H, Stockmeier CA, Ordway GA. Dopaminergic abnormalities in amygdaloid nuclei in major depression: a post-mortem study. Biol Psychiatry 2002;52:740–8.Search in Google Scholar
[56] Meyer JH, Ginovart N, Boovariwala A, Sagrati S, Hussey D, Garcia A, Young T, Praschak-Rieder N, Wilson AA, Houele S. Elevated monoamine oxidase A levels in the brain: an explanation for the monoamine imbalance of major depression. Arch Gen Psychiatry 2006;63:1209–16.Search in Google Scholar
[57] Olver JS, O’Keefe G, Jones GR, Burrows GD, Tochon-Danguy HJ, Ackermann U, Scott A, Norman TR. Dopamine D1 receptor binding in the striatum of patients with obsessive–compulsive disorder. J Affect Disord 2009;114:321–6.Search in Google Scholar
[58] Perani D, Garibotto V, Gprini A, Moresco RM, Henin M, Panzacchi A, Matarrese M, Carpinelli A, Ballodi L, Fazio F. In vivo PET study of 5HT2A serotonin and D2 dopamine dysfunction in drug-naïve obsessive–compulsive disorder. Neuroimage 2008;42:306–14.Search in Google Scholar
[59] Tomer R, Aharon-Petez J. Novelty seeking and harm avoidance in Parkinson’s disease: effects of asymmetric dopamine deficiency. J Neurol Neurosurg Psychiatry 2004;75:972–5.Search in Google Scholar
[60] Conrad R, Schilling G, Bausch C, Nadstawek J, Wartenberg HC, Wegener I, Geiser F, Imbierowicz K, Liedtke R. Temperament and character personality profiles and personality disorders in chronic pain patients. Pain 2007;133: 197–209.Search in Google Scholar
[61] Nyman ES, Loukola A, Varolo T, Ekelund J, Veijola J, Joukamaa M, Taanila A, Pouta A, Miettunen J, Freimer N, Järvelin M-R, Peltonen L. Impact of the dopamine receptor gene family on temperament traits in a population-based cohort. Am J Med Genet B Neuropsychiatr Genet 2009;150:854–65.Search in Google Scholar
[62] Zald DH, Cowan RL, Riccardi P, Baldwin RM, Ansari MS, Li R, Shelby ES, Smith CE, McHugo M, Kessler RM. Midbrain dopamine receptor availability is inversely associated with novelty-seeking traits in humans. J Neurosci 2008;28: 14372–8.Search in Google Scholar
[63] Hess C, Reif A, Strobel A, Boreatti-Hümmer A, Heine M, Lesch K-P, Jacop CP. A functional dopamine-beta-hydroxylase gene promoter polymorphism is associated with impulsive personality styles, but not with affective disorders. J Neural Transm 2009;116:121–30.Search in Google Scholar
[64] Defazio G, Bearardelli A, Fabbrini G, Martino D, Fincati E, Fiaschi A, Moretto G, Abbruzzese G, Marchese R, Bonuccelli U, Del Dotto P, Barone P, De Vivo E, Albanese A, Antonini A, Canesi M, Lopiano L, Zibetti M, Nappi G, Martignoni E, Lamberti P, Tinazzi M. Pain as a nonmotor symptom of Parkinson disease. Arch Neurol 2008;65:1191–4.Search in Google Scholar
[65] Magnusson JE, Fisher K. The involvement of dopamine in nociception: the role of D1 and D2 receptors in the dorsolateral striatum. Brain Res 2000;855: 260–6.Search in Google Scholar
[66] Hagelberg N, Jääskeläinen SK, Martikainen IK, Mansikka H, Forssell H, Scheinin H, Hietala J, Pertovaara A. Striatal dopamine D2 receptors in modulation of pain in humans: a review. Eur J Pharmacol 2004;500:187–92.Search in Google Scholar
[67] Burkey AR, Carstens E, Jasmin L. Dopamine reuptake inhibition in the rostral agranular insular cortex produces antinociception. J Neurosci 1999;19:4169–79.Search in Google Scholar
[68] Wood PB, Schweinhardt P, Jaeger E, Dagher A, Hakyemez H, Rabiner EA, Bushnell MC, Chizh BA. Fibromyalgia patients show an abnormal dopamine response to pain. Eur J Neurosci 2007;25:3576–82.Search in Google Scholar
[69] Wood PB, Patterson II JC, Sunderland JJ, Tainter KH, Glabus MF, Lilien DL. Reduced presynaptic dopamine activity in fibromyalgia syndrome demonstrated with positron emission tomography: a pilot study. J Pain 2007;8:51–8.Search in Google Scholar
[70] Scherman D, Desnos C, Darchen F, Pollack P, Javoy-Agid F, Agid Y. Striatal dopamine deficiency in Parkinson’s disease: role of aging. Ann Neurol 1989;26:551–7.Search in Google Scholar
[71] Rinne JO, Hietala J, Ruotsalainen U, Saki E, Laihnen A, Någren K, Lehikoinen P, Oikonen V, Syvälahti E. Decrease in human striatal dopamine D2 receptor density with age: a PET study with [11C] raclopride. J Cereb Blood Flow Metab 1993;13:310–4.Search in Google Scholar
[72] Bäckman L, Ginovart N, Dixon RA, Robins Wahlin T-B, Wahlin Å, Halldin C, Farde L. Age-related cognitive deficits mediated by changes in the striatal dopamine system. Am J Psychiatry 2000;157:635–7.Search in Google Scholar
[73] Pohjalainen T, Rinne JO, Någren K, Syvälaht E, Hietala J. Sex differences in the striatal dopamine D2 receptor binding characteristics in vivo. Am J Psychiatry 1998;155:768–73.Search in Google Scholar
[74] Kaasinen V, Någren K, Hietala J, Farde L, Rinne JO. Sex differences in extrastriatal dopamine D2 -like receptors in the human brain. Am J Psychiatry 2001;158:308–11.Search in Google Scholar
[75] Di Paolo T. Modulation of brain dopamine transmission by sex steroids. Rev Neurosci 1994;5:27–42.Search in Google Scholar
[76] Koponen S, Taiminen T, Portin R, Himanen L, Isoniemi H, Heinonen H, Hinkka S, Tenovuo O. Axis I and II psychiatric disorders after traumatic brain injury: a 30-year follow-up study. Am J Psychiatry 2002;159:1315–21.Search in Google Scholar
© 2011 Scandinavian Association for the Study of Pain
