The effective perospirone augmentation with clonazepam for treatment-resistant burning mouth syndrome: A case report.

Burning mouth syndrome (BMS) is characterized by burning sensations in the oral region without corresponding abnormalities and is often accompanied by uncomfortable sensations. Herein, we present cases of BMS in which the remaining uncomfortable sensations improved with perospirone augmentation with clonazepam. Case 1: A 61-year-old man complained of a burning pain in his tongue, a sensation of dryness and discomfort as if his tongue was sticking to a palatal plate. With the diagnosis of BMS, psychopharmacotherapy was initiated with amitriptyline. At the dose of amitriptyline 50 mg, the pain lessened but uncomfortable sensations persisted. Further attempts to alleviate symptoms by combining aripiprazole with amitriptyline, aripiprazole with mirtazapine, or aripiprazole with clonazepam were limited; however, nearly all symptoms were relieved by a combination of perospirone 8.0 mg with clonazepam 1.5 mg. Case 2: A 51-year-old woman complained of a burning sensation along with oral dryness and crumb-like feeling on her tongue. She was diagnosed with BMS and began treatment with amitriptyline. Her burning sensation improved at the dose of 25 mg, but uncomfortable sensations persisted. Augmentation of amitriptyline with aripiprazole, aripiprazole either with valproate, mirtazapine, or clonazepam failed to produce a significant improvement. However, a regimen of perospirone 6.0 mg and clonazepam 1.5 mg relieved the crumb-like sensation and pain and culminated in a stabilized condition. The reported cases suggested that multiple approaches targeting the dopaminergic circuit in basal ganglia involving the serotoninergic and GABA systems, through the administration of perospirone with clonazepam is an effective adjunctive treatment for the remaining uncomfortable sensations in patients with BMS.

Dental conditions in patients with medically unexplained oral symptoms.

Background/purpose: Dentists sometimes struggle with treating patients with unexplained symptoms, known as oral psychosomatic disorders, that do not improve with conventional treatment. Oral psychosomatic disorders do not fit the definition of psychosomatic diseases in internal medicine. To ensure appropriate dental treatment, it is important for general dentists to distinguish between oral psychosomatic disorders and psychosomatic diseases. However, relevant evaluation methods have not yet been developed. The DMFT index is widely used as an indicator of the caries status. The purpose of this study was to compare the DMFT index scores of patients with oral psychosomatic. Materials and methods: The DMFT scores of 2202 patients with oral psychosomatic disorders, 145 psychiatric inpatients, and 3940 general dental patients were statistically compared. The DMFT of patients with oral psychosomatic disorders was further compared based on the presence or absence of psychiatric history and disease. Results: The median DMFT scores of oral psychosomatic disorder patients, psychiatric inpatients, and general dental patients were 16, 22, and 10, respectively, showing a significant difference. No significant differences were found in the DMFT scores based on the presence or absence of psychiatric history in oral psychosomatic disorder patients. Conclusion: The intraoral environment of patients with oral psychosomatic disorders was worse than that of general dental patients but better than that of psychiatric inpatients. General dentists could suspect psychiatric and oral psychosomatic disorders based on the state of patients' oral environment.

Case series of drug-induced open bite: Extrapyramidal symptoms related to psychotropic medications.

Introduction: Drug-induced open bite is one of the extrapyramidal symptoms with abnormal tonus of muscles and is rarely recognized in dentistry. This is a retrospective case study to investigate clinical characteristics including detailed complaints in patients with drug-induced open bite. Methods: Of the outpatients who first visited the psychosomatic dental clinic at the Tokyo Medical and Dental University Hospital between September 2013 and September 2022, the patients diagnosed with drug-induced open bite were involved in this study. The clinical characteristics including sex, age, detailed complaints, duration of illness, abnormal findings, psychotropic medications, and other medications that were taken at the first examination, psychiatric comorbidities, the duration of psychiatric diseases, and other medical histories were collected retrospectively by reviewing their medical chart. Results: Drug-induced open bite was found in 11 patients [women: 7, men: 4, median of age: 49 (36.5, 53) years old]. Difficulty in eating especially chewing was the major complaint (9/11, 81.6%) with the duration of illness as 48.0 (16.5, 66) months. Various degrees of open bite were observed. While some showed no occlusal contact on frontal teeth, some showed occlusal contact only on the second molars; moreover, the jaw showed a horizontal slide in a few patients. Three cases could be followed up for prognosis; while in one case the drug-induced open bite improved with 6 months of follow-up, two cases did not improve, and one showed extrusion of molars. All of them had psychiatric comorbidities with the most common diagnosis being schizophrenia (n = 5) and depression (n = 5) followed by insomnia (n = 1) and autism spectrum disorder (n = 1) including duplicated diagnosis. Nine patients (81.6%) had been undergoing treatment with antipsychotics of which three patients were also taking antidepressants. Discussion: Although a drug-induced open bite is a rare symptom, prudent medical interviews about symptoms, psychiatric comorbidities, and psychotropic medication history besides oral assessment are necessary to provide a precise diagnosis and appropriate management in collaboration between dentists and psychiatrists.

Clinical Features and Variations of Pain Expressions in 834 Burning Mouth Syndrome Patients With or Without Psychiatric Comorbidities.

Introduction Burning mouth syndrome (BMS) is characterized as chronic burning pain or unpleasant discomfort in the oral region without any corresponding clinical abnormalities. The aim of this study is to investigate the difference in clinical features and the variations of pain expressions between BMS patients with and without psychiatric comorbidities. Methodology The patients with BMS who first visited between April 2016 and March 2020 were involved and the clinical data including the presence of psychiatric comorbidities and scores of self-rating depression scale (SDS), pain catastrophizing scale (PCS), and pain quality from short-form McGill pain questionnaire (SF-MPQ) were collected retrospectively. Results In 834 patients with BMS (700 females, 63.9 ± 13.1 years old), 371 patients (44.5%) had psychiatric comorbidities. There was no significant between-group difference in demographic data. However, significantly higher scores were observed in SDS (p < 0.001) and PCS (p < 0.001) in the patients with psychiatric comorbidities. Moreover, the patients with psychiatric comorbidities showed significantly stronger pain intensity (p < 0.001) besides higher scores of each descriptor in SF-MPQ. In addition, they had chosen more descriptors in SF-MPQ (p < 0.001); furthermore, the number of selected pain descriptors showed a stronger correlation with PCS than with SDS regardless of the presence of psychiatric comorbidities. Conclusion BMS patients may complain of various pain expressions regardless of the psychiatric comorbidities; however, more severe complaints relating to high pain catastrophizing are more likely in patients with psychiatric comorbidities. These results suggested that underlying anxiety exacerbated the variety of pain expressions in BMS patients with psychiatric comorbidities.

A case with burning mouth syndrome followed by dementia with Lewy bodies: a case report.

Burning mouth syndrome (BMS) is characterized by persistent oral burning sensations without corresponding organic findings. Dementia with Lewy bodies (DLB) is a common type of dementia and generally presents visual hallucination and parkinsonism as motor dysfunction besides cognitive decline. In this case report, we present a case in which DLB emerged during the treatment for BMS, with a relatively positive outcome for BMS. A 74 years-old female complained of burning pain in her mouth and a subsequent decrease in food intake. Following a diagnosis of BMS, pharmacotherapy was initiated. BMS was much improved with mirtazapine 15 mg and aripiprazole 1.0 mg, leading to the restoration of her food intake by day 180. However, BMS flared up again triggered by deteriorating physical condition of herself and that of her husband. With aripiprazole 1.5 mg and amitriptyline 25 mg, her BMS gradually improved by day 482. However, by day 510, an increase in anxiety was noted, accompanied by the occasionally misidentification of her husband on day 566. Her cognitive impairment and disorientation were also reported by her husband on the day 572, she was then immediately referred to a neurologist specialized dementia and diagnosed with DLB on the day 583. Her treatment was adjusted to include the prescription of rivastigmine which was titrated up to 9.0 mg. Considering the potential impact of amitriptyline on cognitive function, it was reduced and switched to mirtazapine; however, her oral sensations slightly got worse. Following the consultation with her neurologist, amitriptyline 10 mg was reintroduced and aripiprazole was discontinued on day 755. Remarkably, BMS gradually improved without deteriorating DLB. This case indicated the reaffirmed necessity of careful interviews for changes in daily life not only with the patients but also with their families through the medical assessments. It highlights the vigilance regarding potential cognitive decline underlying or induced as an adverse event especially when treating elderly patients with BMS. While the interaction between BMS and DLB remains unclear, this case underscores the importance of prudent diagnosis and constructing collaboration with specialists in managing BMS with the early phase of DLB.

Case report: Treatment of persistent atypical odontalgia with attention deficit hyperactivity disorder and autism spectrum disorder with risperidone and atomoxetine.

Chronic pain has recently been associated with developmental disorders [autism spectrum disorder (ASD) and attention deficit hyperactivity disorder (ADHD)]. Regarding chronic pain in adulthood, fibromyalgia, migraine, and chronic low back pain have been associated with ADHD. The ICD-11 disease classification categorizes these pain diseases as chronic primary pain, suggesting high comorbidity with developmental disorders in chronic primary pain. Atypical odontalgia (AO) is a persistent tooth pain that occurs in the absence of any of the usual dental causes, most of which are triggered by dental treatment. Conditions characterized by tooth pain with no apparent cause are also classified as chronic primary pain. Approximately half the patients with AO are diagnosed with psychiatric disorders; the most common are depression (15.4%) and anxiety disorders (10.1%). However, there are no reports on neurodevelopmental disorders comorbid with AO. In the present study, we report a case of a 46-year-old man with numerous complaints (e.g., occlusal instability, difficulty eating, difficulty speaking), who took work leave due to worsening of his symptoms after periodontal scaling ("gingival recession" and "aggressive periodontal treatment") and frequently expressed dissatisfaction and anger at the hospital, making the dental treatment difficult. After a referral to a psychiatrist specializing in chronic pain, AO and previously undiagnosed comorbidity of ASD and ADHD were confirmed. Atypical antipsychotic risperidone for ASD irritability and an ADHD medication, atomoxetine dramatically reduced anger, pain, anxiety, depression, and pain catastrophizing thoughts, leading to reduced obsession with his symptoms and less frequent complaints. After risperidone (1 mg/day) + atomoxetine (120 mg/day) were ultimately prescribed after adjustment, he was able to return to work 226 days after initiation of psychiatric treatment. Recent studies show that comorbidity of developmental disorders in patients with chronic pain is likely to be undetected. Clinicians should include screening for ASD and ADHD not only in cases of fibromyalgia, migraine, and chronic low back pain, but also in orofacial pain such as AO and other treatments for chronic primary pain. For patients diagnosed with ASD or ADHD, an effective drug therapy for ASD and ADHD should be considered.

Case Report: Auditory Hallucination Induced by Amitriptyline for the Treatment of Atypical Odontalgia.

Auditory hallucination is usually associated with psychiatric diseases and organic brain illness. It was rarely found as adverse events of antidepressants. Amitriptyline is considered as one of the first line medications for the psychopharmacotherapy of chronic pain including atypical odontalgia (AO) which shows chronic tooth pain without corresponding abnormalities. Anticholinergic adverse events induced by amitriptyline are usually bearable and not critical since the prescription dose is very low for the patients with AO. This is a first case report about the AO patients who showed auditory hallucination by the low dose of amitriptyline. A 43-years-old female, housewife, complained chronic toothache following dental procedures and was diagnosed as AO. Amitriptyline was initially prescribed 25 mg and gradually increased up to 60 mg with the improvement of AO symptoms in 7 months. Although the temporary recurrence was observed following to the retreatment of prosthodontic dental procedures, it improved in a few weeks. Therefore, the dose of amitriptyline was decreased, and the continuation dose was set 30 mg. In 24 months, the AO symptoms were very much improved; however, she reported that she had been heard the voices at midnight for a year. The voices were neighborhoods' and talking about the noise troubles she had claimed before. She had not realized that the voices were auditory hallucination since they were heard only at midnight infrequent and not bothering her daily life. At the time she reported auditory hallucination, she worried whether organic brain diseases are hiding because the frequency of voices was increased and sometimes occurred in daytime. The adverse event of amitriptyline was suspected since she had never had psychotic symptoms before. Amitriptyline was decreased and continued with the dose of 25 mg. Magnetic resonance imaging and psychiatric consultation revealed no abnormality of brain and in psychiatric aspects. After final prosthodontic treatment, the amitriptyline was discontinued in 30 months. Two months after the discontinuation, auditory hallucination was almost disappeared with no recurrence of AO. The present case report suggests that amitriptyline has possibility to induce auditory hallucination even in conventional dose throughout the treatment of chronic pain including AO.

The Effectiveness and Adverse Events of Amitriptyline and Aripiprazole in Very Elderly Patients With BMS.

Burning mouth syndrome (BMS) is defined by chronic oral burning sensations without any corresponding abnormalities. Besides amitriptyline, aripiprazole has been reported as a possible medication to manage BMS. However, especially for elderly patients, the adverse events of these medications would be a problem. The aim of the present study was to investigate the differences in the effectiveness and adverse events of amitriptyline and aripiprazole in very elderly patients with BMS. This is a retrospective comparative study of 80 years old and older patients with BMS who were initially treated with amitriptyline or aripiprazole and who were new outpatients of our department from April 2017 to March 2020. All clinical data, including sex, age, comorbid physical diseases, comorbid psychiatric disorders, the prescribed doses (initial, maximum, and effective dose), prognosis, and adverse events, were collected from their medical charts. Each medication was selected considering their medical history. Amitriptyline was prescribed in 13 patients (11 women, 82.3 ± 2.1 years old) and aripiprazole was prescribed in 27 patients (26 women, 84.2 ± 3.8 years old). There were no significant between-group differences in sex, age, duration of illness, pain intensity, salivation, and psychiatric comorbidity at the first examination. Amitriptyline clinically improved more patients (7 patients, 53.8%) with the effective dose of 10 (7.5, 15.0) mg than aripiprazole (11 patients, 40.7%) of which the effective dose was 1.0 (0.5, 1.5) mg, although there were no significant between-group differences. The adverse events of amitriptyline were found in 9 patients (69.2%) and most patients had constipation (46.2%). For aripiprazole, 7 patients (25.9%) showed adverse events, most of them reported sleep disorder (11.1%). Amitriptyline had significantly longer duration taking medication (p = 0.021) and lower discontinuation (p = 0.043) despite of higher occurrence rate of adverse events (p = 0.015) compared to aripiprazole. These results suggest that both psychopharmacotherapies with a low dose of amitriptyline and aripiprazole are effective for the very elderly patients with BMS. Furthermore, aripiprazole may have some advantages in the adverse events compared to amitriptyline; however, the low dose amitriptyline monotherapy may have more benefit in the effectiveness and tolerability over prudent collaboration with primary physicians.

Clinical Characteristics of Predominantly Unilateral Oral Cenesthopathy With and Without Neurovascular Contact.

Oral cenesthopathy (OC) is characterized by unusual oral discomfort without corresponding evidence, and it has often been categorized as "delusional disorder, somatic type". Regarding possible causative factors of OC, involvement of neurovascular contact (NVC) of the trigeminal nerve, which transmits not only pain but also thermal, tactile, and pressure sensations, has never been observed yet. This study aimed to investigate the relationship between clinical characteristics of unilateral OC and the presence of trigeminal nerve NVC. This is a retrospective comparative study that involved 48 patients having predominantly unilateral OC who visited the Psychosomatic Dentistry Clinic of Tokyo Medical and Dental University between April 2016 and February 2019. Magnetic resonance imaging was performed to assess NVC presence. The Oral Dysesthesia Rating Scale (Oral DRS) was used to assess the various oral sensations and functional impairments besides psychometric questionnaires. Clinical characteristics were retrospectively obtained from the patients' medical charts. NVC was present in 45.8% (22/48) of the patients. There was no significant difference in sex, age, psychiatric history, oral psychosomatic comorbidity, and psychometric questionnaire scores between patients with and without NVC. However, compared to the patients with NVC, the patients without NVC had significantly higher scores for overall subjective severity of OC symptoms (p = 0.008). Moreover, patients having predominantly unilateral OC without NVC showed significantly higher scores in symptom severity and functional impairment of the following parameters: movement (p = 0.030), work (p = 0.004), and social activities (p = 0.010). In addition, compared with the patients with NVC, the patients without NVC showed significantly higher averages of the total symptom severity scale (SSS) and functional impairment scale (FIS) scores in the Oral DRS (p = 0.015 and p = 0.031, respectively). Furthermore, compared with the patients with NVC, the patients without NVC had significantly higher numbers of corresponding symptoms in both the SSS and FIS (p = 0.041 and p = 0.007, respectively). While NVC may be involved in the indescribable subtle OC symptoms, more complex mechanisms may also exist in OC patients without NVC, which yield varying and more unbearable oral symptoms.

Personality Traits in Burning Mouth Syndrome Patients With and Without a History of Depression.

Objectives: So far, the strong link between neuroticism, chronic pain, and depression has been well-documented in literatures. Some suggested that they might share etiological factors, thus resulting in overlapping constructs. However, such effect has never been tested in burning mouth syndrome (BMS) patients, a complex phenomenon influenced by both neuropathic and psychopathological factors. We aim to clarify how personality affects individual's pain and pain-related experiences. Methods: Two hundred forty-eight patients with BMS provided demographic information and psychiatric history; completed Ten-Item Personality Inventory, a Visual Analog Scale of pain, and McGill Pain Questionnaire; and provided adequate parameters of depressive state, catastrophizing thinking, and central sensitization. Results: BMS patients with depression history suffered more severe clinical symptoms and scored higher in neuroticism and less in openness and extraversion than did those without psychiatric diagnoses. After age, sex, and duration of pain were controlled, neuroticism in BMS patients with depression correlates with affective dimension of pain. Instead, if psychiatric history is absent, neuroticism correlates with sensory dimension and pain intensity. In both groups, higher neuroticism, unlike other personality facets, contributed to a more severe clinical condition. Conclusion: Of the five traits, neuroticism appears to be the most crucial dimension associated with the pain symptoms and patient's conditions. This study implies that management of pain must extend beyond solely providing pain-relieving medication and must require a holistic and multidisciplinary approach.

Case Report: Iatrogenic Dental Progress of Phantom Bite Syndrome: Rare Cases With the Comorbidity of Psychosis.

Introduction: Phantom bite syndrome (PBS) is considered as the preoccupation with dental occlusion and the continual inability to adapt to changed occlusion. These patients constantly demand occlusal corrections and undergo extensive and excessive dental treatments. We present three cases with PBS-suspected iatrogenic concerns and the attribution to underlying psychosis. Case Presentation: A 70-year-old female demanded orthodontic retreatment and complained of tightness and cramped sensation of teeth in the oral cavity, uncomfortable occlusion, and pain in her neck and legs that she was convinced was induced by orthodontic treatment. However, even earlier than the orthodontic treatment, she had kept doctor shopping for over 35 years, not merely dentists but also psychiatrists, neurologists, and so on; she was diagnosed with bipolar disorder. A 48-year-old female complained of malaligned improper occlusion and demanded occlusal adjustment. These symptoms occurred in the absence of a dental trigger and were worsened by orthodontic treatment. She underwent psychiatric treatment for 15 years with a diagnosis of bipolar disorder. A 38-year-old female, who had a history of schizophrenia for over 20 years, complained of occlusal discomfort and revisited with a complaint of abnormal occlusion due to excessive dental procedures. In the last two cases, requests for dental procedures had reduced owing to the collaboration between the psychiatrists and dentists. All the cases first visited our clinic following a succession of dental visits. They were strongly convinced that occlusal correction was the only solution to their symptoms, including the symptoms of discomfort in other body parts. Their misleading perceptions were uncorrectable, and repeated dental treatments exacerbated their complaints. Moreover, the dentists overlooked the psychotic histories of the patients, while the comorbid psychosis resulted in a strict demand for dental treatment by the patients. Conclusions: The presented PBS cases with psychosis suggest that repeated dental treatments and comorbid psychosis exacerbate PBS. Moreover, their persistent demands reflecting comorbid psychosis led dentists to perform numerous procedures. Early detection of underlying psychosis and the prompt collaboration between psychiatrists and dentists are integral to help prevent complications in PBS cases with psychosis.