A case of vestibular schwannoma with oral burning sensation: surgical complication or burning mouth syndrome?

BACKGROUND: Despite improvements in surgical techniques, the removal of vestibular schwannoma is related to some complications. Recovery from surgical complications of vestibular schwannoma is often difficult and complications sometimes lead to permanent deficits. However, treatable trigeminal symptoms may be missed in atypical cases. CASE PRESENTATION: A 46-year-old woman complained about burning sensation on her tongue and maxilla for four years before her first visit to our clinic. She visited the neurosurgery department in a university hospital because her facial pain and burning sensation of her tongue were suddenly aggravated. She was diagnosed with vestibular schwannoma and tumour resection was performed. However, her oral pain persisted after surgery. Two months before the initial visit to our clinic, the oral pain became more severe than ever before. When the patient visited a psychiatrist due to a panic attack, the psychiatrist diagnosed her as having somatic symptom disorder and depression and referred her to our clinic. Based on the characteristics of the pain, she was diagnosed as burning mouth syndrome and treated for the same. Within 1.5 months, the pain and burning sensation of the tongue and maxilla almost completely remitted with low dose amitriptyline. CONCLUSIONS: Our case suggests that there are exceptional cases in which burning mouth syndrome and vestibular schwannoma occur simultaneously. Burning pain after vestibular schwannoma surgery cannot always be considered a complication of surgery.

A case of vestibular schwannoma mimicking burning mouth syndrome.

BACKGROUND: An oral burning sensation with unidentified cause in patients with preexisting psychosocial conditions is usually diagnosed as burning mouth syndrome. However, unexpected organic lesions may be detected in rare cases. CASE PRESENTATION: A 35-year-old woman had chief complaints of a burning sensation and numbness of the right side of the lip and tongue, as well as a dry sensation of the mouth with a taste disturbance of the right side of the tongue. The symptoms were continuous and did not show any daily fluctuations. The symptoms started without any recognizable triggering factor six months before her first visit to our clinic,. No abnormality was detected in her mouth. MRI images revealed an approximately 30 × 30 mm well-defined mass localized in the right cerebropontine angle compressing the trigeminal nerve, which was diagnosed as schwannoma of the right auditory nerve. CONCLUSIONS: It is important for clinicians to consider the possibility of brain tumors in their differential diagnosis of BMS. Although it is not always easy to eliminate all diseases that may cause an oral burning sensation in patients with BMS-like symptoms, more attention and careful examination based on the patient's psychosomatic background features and other possible causes are needed to rule out organic diseases.

Comparison of Cerebral Blood Flow Patterns in Patients with Phantom Bite Syndrome with Their Corresponding Clinical Features.

BACKGROUND: Phantom bite syndrome (PBS) is characterized by an uncomfortable sensation during occlusion without any evident abnormality. A recent case-control study with single-photon emission computed tomography (SPECT) using 99mTc-ethyl cysteinate dimer could not find the specific features of regional cerebral blood flow (rCBF), which might be due to the heterogeneity of PBS. We analyzed the brain images of PBS corresponding to the clinical features by studying PBS subgroups. METHODS: This study contributes to elucidating the pathophysiology of PBS by evaluating regional brain perfusion on SPECT and its clinical features. We performed SPECT using 99mTc-ethyl cysteinate dimer in 44 patients with PBS. The SPECT images were analyzed qualitatively and quantitatively. RESULTS: Asymmetrical rCBF patterns were detected, corresponding to symptom laterality. Patients with PBS with right-side symptoms showed right-side-predominant rCBF asymmetry in the parietal region and left-side-predominant rCBF asymmetry in the thalamus, and vice versa. Moreover, the analysis of the association between rCBF and patient behaviors revealed that patients who blamed their dentists for their symptoms tended to have a symmetrical rCBF pattern. CONCLUSION: Patients with PBS showed blood flow imbalance in the thalamus and parietal region corresponding to symptom laterality. There are two types of symmetrical and asymmetrical rCBF patterns in the pathophysiology of PBS despite similar clinical manifestations.

Pharmacotherapeutic outcomes in atypical odontalgia: determinants of pain relief.

OBJECTIVES: There has been considerable research which has focused on clarifying the origin of pain in patients with atypical odontalgia (AO), also known as "idiopathic toothache", and on identifying effective treatment, but there has been limited success so far. In this study, we assessed the outcomes of treatment and attempted to identify factors that could account for pain remission in patients with AO. PATIENTS AND METHODS: Data for 165 patients diagnosed with AO from June 2015 to August 2017 were retrospectively reviewed. The patients' sex, age, duration of pain, and psychiatric history were collected, along with information on pain intensity, depressive status, and catastrophizing scores. Responses at 4 and 16 weeks from the start of treatment were observed. The associations between potentially associated factors and outcome were investigated using Bayesian model averaging. RESULTS: A 30% reduction in pain was reported by 38 patients (46.3%) at 4 weeks and by 54 patients (65.9%) at 16 weeks. The pain intensity decreased as the depression and catastrophizing score improved; all of the changes were statistically significant (P<0.001). Four elements, that is, patient sex, depression score at baseline, pain score at 4 weeks, and change in the catastrophizing score, explained 52.5% of the variation in final outcome between individual patients. CONCLUSION: Our findings confirm the efficacy of tricyclic antidepressants (TCAs) as a treatment for AO and indicate that other medications, especially aripiprazole used in combination with a TCA, may be useful. A considerable number of patients, especially women, those with lower levels of depression at baseline, and those who responded to 4 weeks of treatment, achieved pain relief.

Change of Cerebral Blood Flow After a Successful Pharmacological Treatment of Phantom Bite Syndrome: A Case Report.

BACKGROUND: "Phantom bite syndrome," a persistent complaint of an uncomfortable bite sensation with no obvious occlusal abnormal finding, recently was suggested to be related with central nervous system dysfunction. Here, we report a case of phantom bite syndrome in which the occlusal discomfort was improved with mirtazapine and aripiprazole combination parallel with regional cerebral blood flow change. CASE REPORT: A 60-year-old-female patient came to our clinic with the chief complaint of a "loosely bite" after dental treatment and various uncomfortable sensations of body sites. One year after the medication therapy, the prosthodontic retreatment was carried out successfully, and a good outcome was obtained for over 24 months so far. In addition, a subsequent change of regional cerebral blood flow was observed in single-photon emission computed tomography: the right and left asymmetry of cerebral blood flow in the frontal lobe has disappeared along with the improvement to the symptoms improvement. CONCLUSIONS: This case suggests that some central nervous system dysfunction involving dopaminergic system might be related to the pathophysiology of phantom bite syndrome.

Current management strategies for the pain of elderly patients with burning mouth syndrome: a critical review.

Burning Mouth Syndrome (BMS), a chronic intraoral burning sensation or dysesthesia without clinically evident causes, is one of the most common medically unexplained oral symptoms/syndromes. Even though the clinical features of BMS have been astonishingly common and consistent throughout the world for hundreds of years, BMS remains an enigma and has evolved to more intractable condition. In fact, there is a large and growing number of elderly BMS patients for whom the disease is accompanied by systemic diseases, in addition to aging physical change, which makes the diagnosis and treatment of BMS more difficult. Because the biggest barrier preventing us from finding the core pathophysiology and best therapy for BMS seems to be its heterogeneity, this syndrome remains challenging for clinicians. In this review, we discuss currently hopeful management strategies, including central neuromodulators (Tricyclic Antidepressants - TCAs, Serotonin, and Norepinephrine Reuptake Inhibitors - SNRIs, Selective Serotonin Reuptake Inhibitors - SSRIs, Clonazepam) and solutions for applying non-pharmacology approaches. Moreover, we also emphasize the important role of patient education and anxiety management to improve the patients' quality of life. A combination of optimized medication with a short-term supportive psychotherapeutic approach might be a useful solution.

Psychiatric comorbidities in patients with Atypical Odontalgia.

OBJECTIVE: Atypical Odontalgia (AO) is a condition characterized by tooth pain with no apparent cause. Although psychiatric comorbidity seems to be very common, it has rarely been studied. To clarify the influence of psychiatric comorbidity on the clinical features in patients with AO, we retrospectively evaluated their examination records. METHODS: Clinical features and psychiatric diagnoses of 383 patients with AO were investigated by reviewing patients' medical records and referral letters. Psychiatric diagnoses were categorized according to the Diagnostic and Statistical Manual of Mental Disorders, Fifth Edition (DSM-5). We also analyzed visual analogue scale (VAS), self-rating depression scale (SDS), and the short-form McGill pain questionnaire (SF-MPQ) scores. RESULTS: Of the 383 patients with AO, 177 (46.2%) had comorbid psychiatric disorders. The most common were depressive disorders (15.4%) and anxiety disorders (10.1%). Serious psychotic disorders such as bipolar disorder (3.0%) and schizophrenia (1.8%) were rare. Dental trigger of AO was reported in 217 (56.7%) patients. There were no significant correlations between psychiatric comorbidities and most of the demographic features. Higher VAS and SDS scores, higher frequency of sleep disturbance, and higher ratings of "Fearful" and "Punishing-cruel" descriptors of the SF-MPQ were found in patients with psychiatric comorbidity. CONCLUSIONS: About half of AO patients had comorbid psychiatric disorders. Dental procedures are not necessarily causative factors of AO. In AO patients with comorbid psychiatric disorders, pain might have a larger emotional component than a sensory one. VAS, SDS, and SF-MPQ scores might aid in the noticing of underlying comorbid psychiatric disorders in AO patients.

Evaluating Burning Mouth Syndrome as a Comorbidity of Atypical Odontalgia: The Impact on Pain Experiences.

OBJECTIVE: This study aimed (1) to investigate the differences in clinical characteristics of patients between 2 groups, those who have atypical odontalgia (AO) only and those who have AO with burning mouth syndrome (BMS), and (2) to assess the influence of psychiatric comorbidity factors on patients' experiences. METHOD: Medical records and psychiatric referral forms of patients visiting the Psychosomatic Dentistry Clinic of Tokyo Medical and Dental University between 2013 and 2016 were reviewed. The final sample included 2 groups of 355 patients: those who have AO only (n = 272) and those who have AO with BMS (AO-BMS; n = 83). Clinicodemographic variables (gender, age, comorbid psychiatric disorders, and history of headache or sleep disturbances) and pain variables (duration of illness, pain intensity, and severity of accompanying depression) were collected. Initial pain assessment was done using the Short-Form McGill Pain Questionnaire, and depressive state was determined using the Zung Self-Rating Depression Scale. RESULTS: The average age, female ratio, and sleep disturbance prevalence in the AO-only group were significantly lower than those in AO-BMS group. AO-BMS patients rated overall pain score and present pain intensity significantly higher than did the AO-only patients (P = 0.033 and P = 0.034, respectively), emphasizing sharp (P = 0.049), hot-burning (P = 0.000), and splitting (P = 0.003) characteristics of pain. Patients having comorbid psychiatric disorders had a higher proportion of sleep disturbance in both groups and a higher proportion of depressive state in the AO-only group. CONCLUSIONS: AO-BMS patients have different epidemiological characteristics, sleep quality, and pain experiences compared to AO-only patients. The presence of psychiatric comorbidities in both groups may exacerbate sleep quality. We suggest that BMS as a comorbid oral disorder in AO patients contributes to a more intensively painful experience.