Dystonia Research Articles

Oromandibular dystonia and temporomandibular disorders

BackgroundThe aim of this study was to characterize clinical features of patients with oromandibular dystonia (OMD) who had temporomandibular disorder (TMD) symptoms. MethodsA retrospective chart review of patients seeking treatment at a tertiary-level orofacial pain clinic from January 2015 through December 2020 was undertaken. The inclusionary criteria consisted of a diagnosis of OMD (International Classification of Diseases, Revision 10 code G24.4), which had been confirmed by a neurologist. ResultsEleven patients met the inclusion criteria. Focal dystonia and jaw deviation OMD were the most frequent diagnoses. A dental procedure was a triggering or aggravating factor in 36.4% of patients. All but 2 patients had a sensory trick, or tactile stimulus to a particular body part, and approximately one-half of the patients used an oral appliance as a sensory trick device. All but 1 patient had received a diagnosis of TMD, with myofascial pain of the masticatory muscles being the most prevalent diagnosis. Four patients had received a recommendation for orthodontic treatment. About one-half of the patients had undergone 1 or more invasive dental or maxillofacial surgical interventions to address their dystonia. Anxiety was the most common psychological comorbidity. ConclusionsBecause patients with OMD commonly experience TMD symptoms, they can receive a misdiagnosis of TMD while the OMD is overlooked. Practical ImplicationsOwing to concomitant TMD symptoms, patients most often seek dental consultations and undergo treatments such as orthodontic interventions and temporomandibular joint surgeries. A dentist’s competency in recognizing these patients can prevent unnecessary procedures and facilitate appropriate patient care.

DIPS (Dystonia Image-based Programming of Stimulation: a prospective, randomized, double-blind crossover trial)

IntroductionDeep brain stimulation of the internal globus pallidus is an effective treatment for dystonia. However, there is a large variability in clinical outcome with up to 25% non-responders even in highly selected primary dystonia patients. In a large cohort of patients we recently demonstrated that the variable clinical outcomes of pallidal DBS for dystonia may result to a large degree by the exact location and stimulation volume within the pallidal region. Here we test a novel approach of programing based on these insights: we first defined probabilistic maps of anti-dystonic effects by aggregating individual electrode locations and volumes of tissue activated of > 80 patients collected in a multicentre effort. We subsequently modified the algorithms to be able to test all possible stimulation settings of de novo patients in silico based on the expected clinical outcome and thus potentially predict the best possible stimulation parameters for the individual patients.MethodsWithin the framework of a BMBF-funded study, this concept of a computer-based prediction of optimal stimulation parameters for patients with dystonia will be tested in a randomized, controlled crossover study. The main parameter for clinical efficacy and primary endpoint is based on the blinded physician rating of dystonia severity reflected by Clinical Dystonia Rating Scales for both interventions (best clinical settings and model predicted settings) after 4 weeks of continuous stimulation. The primary endpoint is defined as “successful treatment with model predicted settings” (yes or no). The value is “yes” if the motor symptoms with model predicted settings are equal or better (tolerance 5% of absolute difference in percentages) to clinical settings. Secondary endpoints will include measures of quality of life, calculated energy consumption of the neurostimulation system and physician time for programming.PerspectiveWe envision, that computer-guided deep brain stimulation programming in silico might provide optimal stimulation settings for patients with dystonia without the burden of months of programming sessions. The study protocol is designed to evaluate which programming method is more effective in controlling motor symptom severity and improving quality of life in dystonia (best clinical settings and model predicted settings).Trial registration Registered with ClinicalTrials.gov on Oct 27, 2021 (NCT05097001).

Deep Brain Stimulation Treating Dystonia: A Systematic Review of Targets, Body Distributions and Etiology Classifications.

Background: Deep brain stimulation (DBS) is a typical intervention treating drug-refractory dystonia. Currently, the selection of the better target, the GPi or STN, is debatable. The outcomes of DBS treating dystonia classified by body distribution and etiology is also a popular question.Objective: To comprehensively compare the efficacy, quality of life, mood, and adverse effects (AEs) of GPi-DBS vs. STN-DBS in dystonia as well as in specific types of dystonia classified by body distribution and etiology.Methods: PubMed, Embase, the Cochrane Library, and Google Scholar were searched to identify studies of GPi-DBS and STN-DBS in populations with dystonia. The efficacy, quality of life, mood, and adverse effects were quantitatively compared. Meta-regression analyses were also performed. This analysis has been registered in PROSPERO under the number CRD42020146145.Results: Thirty five studies were included in the main analysis, in which 319 patients underwent GPI-DBS and 113 patients underwent STN-DBS. The average follow-up duration was 12.48 months (range, 3–49 months). The GPI and STN groups were equivalent in terms of efficacy, quality of life, mood, and occurrence of AEs. The focal group demonstrated significantly better disability symptom improvement (P = 0.012) than the segmental and generalized groups but showed less SF-36 enhancement than the segmental group (P < 0.001). The primary groups exhibited significantly better movement and disability symptom improvements than the secondary non-hereditary group (P < 0.005), which demonstrated only disability symptom improvement compared with the secondary hereditary group (P < 0.005). The primary hereditary and idiopathic groups had a significantly lower frequency of AEs than the secondary non-hereditary group (P < 0.005). The correlation between disability symptom improvement and movement symptom improvement was also significant (P < 0.05).Conclusion: GPi-DBS and STN-DBS were both safe and resulted in excellent improvement in efficacy and quality of life in patients with dystonia. Compared with patients with segmental dystonia, patients with focal dystonia demonstrated better improvement in dystonia symptoms but less enhancement of quality of life. Those with primary dystonia had a better response to DBS in terms of efficacy than those with secondary dystonia. Patients who exhibit a significant improvement in movement symptoms might also exhibit excellent improvement in disability symptoms.

Pharmacological perturbation reveals deficits in D2 receptor responses in Thap1 null mice.

The primary dystonia DYT6 is caused by mutations in the transcription factor Thanatos‐associated protein 1 (THAP1). To understand THAP1’s functions, we generated mice lacking THAP1 in the nervous system. THAP1 loss causes locomotor deficits associated with transcriptional changes. Since many of the genes misregulated involve dopaminergic signaling, we pharmacologically challenged the two striatal canonical dopamine pathways: the direct, regulated by the D1 receptor, and the indirect, regulated by the D2 receptor. We discovered that depleting THAP1 specifically interferes with the D2 receptor responses, pointing to a selective misregulation of the indirect pathway in DYT6 with implications for pathogenesis and treatment.

Is Generalized and Segmental Dystonia Accompanied by Impairments in the Dopaminergic System?

Background: The pathogenesis of dystonia is remarkably diverse. Some types of dystonia, such as DYT5 (DYT-GCH1) and tardive dystonia, are related to dysfunction of the dopaminergic system. Furthermore, on pathological examination, cell loss in the substantia nigra (SN) of patients with dystonia has been reported, suggesting that impaired dopamine production may be involved in DYT5 and in other types of dystonia.Objectives: To investigate functional dopaminergic impairments, we compared patients with dystonia and those with Parkinson's disease (PD) with normal controls using neuromelanin-sensitive magnetic resonance imaging (NM-MRI) and dopamine transporter single photon emission computed tomography (DAT SPECT).Methods: A total of 18, 18, and 27 patients with generalized or segmental dystonia, patients with PD, and healthy controls, respectively, were examined using NM-MRI. The mean area corresponding to NM in the SN (NM-SN) was blindly quantified. DAT SPECT was performed on 17 and eight patients with dystonia and PD, respectively. The imaging data of DAT SPECT were harmonized with the Japanese database using striatum phantom calibration. These imaging data were compared between patients with dystonia or PD and controls from the Japanese database in 256 healthy volunteers using the calibrated specific binding ratio (cSBR). The symptoms of dystonia were evaluated using the Fahn–Marsden Dystonia Rating Scale (FMDRS), and the correlation between the results of imaging data and FMDRS was examined.Results: The mean areas corresponding to NM in the SN (NM-SN) were 31 ± 4.2, 28 ± 3.8, and 43 ± 3.8 pixels in patients with dystonia, PD, and in healthy controls, respectively. The mean cSBRs were 5 ± 0.2, 2.8 ± 0.2, 9.2 (predictive) in patients with dystonia, PD, and in healthy controls, respectively. The NM-SN area (r = −0.49, p < 0.05) and the cSBR (r = −0.54, p < 0.05) were inversely correlated with the FMDRS. There was no significant difference between the dystonia and PD groups regarding NM-SN (p = 0.28). In contrast, the cSBR was lower in patients with PD than in those with dystonia (p < 0.5 × 10−6).Conclusions: Impairments of the dopaminergic system may be involved in developing generalized and segmental dystonia. SN abnormalities in patients with dystonia were supposed to be different from degeneration in PD.

Tardive dyskinesia

Tardive dyskinesia is a group of hyperkinetic and hypokinetic movement disorders, following the administration of dopamine receptor-blocking drugs. The severity of these syndromes varies from soft forms to the development of life-degrading situations. Phenomenologically tardive dyskinesia can be represented both in isolation and in various combinations. Recognition of these syndromes early in the development of tardive dyskinesia can optimize therapeutic treatment and reduce the risk of severe complications. As a means of treatment, deutetrabenazine or valbenazine are used as first-line drugs, with resistance to therapy and in severe cases, drugs of other groups are used (amantadine, baclofen, botulinum toxin type A, clonazepam, donepezil, gabapentin, ginkgo biloba, levetiracetam, melatonin, pregabalin, thiamine, verapamil, vitamin B6, vitamin E). Our own experience of 12 patients with tardive dystonia showed the effeciency of local injections of botulinum toxin.

A sensory trick for monitored anaesthesia care.

A sensory trick for monitored anaesthesia care.

Comparison of cognitive performance between patients with Parkinson\u2019s disease and dystonia using an intraoperative recognition memory test

Neuroscientific studies on the function of the basal ganglia often examine the behavioral performance of patients with movement disorders, such as Parkinson’s disease (PD) and dystonia (DT), while simultaneously examining the underlying electrophysiological activity during deep brain stimulation surgery. Nevertheless, to date, there have been no studies comparing the cognitive performance of PD and DT patients during surgery. In this study, we assessed the memory function of PD and DT patients with the Montreal Cognitive Assessment (MoCA) and the Mini-Mental State Examination (MMSE). We also tested their cognitive performance during the surgery using a continuous recognition memory test. The results of the MoCA and MMSE failed to reveal significant differences between the PD and DT patients. Additionally, no significant difference was detected by the intraoperative memory test between the PD and DT patients. The intraoperative memory test scores were highly correlated with the MMSE scores and MoCA scores. Our data suggest that DT patients perform similarly to PD patients in cognitive tests during surgery, and intraoperative memory tests can be used as a quick memory assessment tool during surgery.

Marie Vidailhet: recognising the importance of teamwork

The city of Toulouse in southern France is filled with a rich history of heroes, adventure, and challenges; where Concorde made its first test flight and the French National Centre for Space Studies obtained stunning satellite images of Earth. It is also the birthplace of Marie Vidailhet, professor of neurology and head of the Movement Disorders and Parkinson's Disease department at the Salpêtrière Hospital—the “cradle of neurology” in France (Sorbonne University, Paris). Bilateral, pallidal, deep-brain stimulation in primary generalised dystonia: a prospective 3 year follow-up studyBilateral pallidal stimulation provides sustained motor benefit after 3 years. Mild long-term improvements in quality of life and attention were also observed. Full-Text PDF Bilateral pallidal deep brain stimulation for the treatment of patients with dystonia-choreoathetosis cerebral palsy: a prospective pilot studyBilateral pallidal neurostimulation could be an effective treatment option for patients with dystonia-choreoathetosis CP. However, given the heterogeneity of motor outcomes and the small sample size, results should be interpreted with caution. The optimum placement of the leads seemed to be a crucial, but not exclusive, factor that could affect a good outcome. Full-Text PDF

Acute Dystonia with Paroxetine – Case Presentation, Possible Mechanism and Clinical Implications

Acute Dystonia with Paroxetine – Case Presentation, Possible Mechanism and Clinical Implications

Sensory tricks modulate corticocortical and corticomuscular connectivity in cervical dystonia

ObjectiveTo examine interactions between cortical areas and between cortical areas and muscles during sensory tricks in cervical dystonia (CD). MethodsThirteen CD patients and thirteen age-matched healthy controls performed forewarned reaction time tasks, sensory tricks, and two tasks replicating aspects of the tricks (moving necks/arms). Control subjects mimicked sensory tricks. Corticocortical and corticomuscular coherence values were calculated from surface electrodes placed over motor, premotor, and sensory cortical areas and dystonic muscles. ResultsDuring initial preparation (after the warning stimulus), the only between-task difference was found in the γ-band corticocortical coherence (higher during tricks than during voluntary neck movements). With movements (before/after the imperative stimulus), the γ-band coherence of CD patients significantly increased during tricks but decreased during voluntary movements, while opposite trends were observed in healthy subjects. Additionally, the α- and β-band coherence decreased in healthy subjects during movements. Between the two patient subgroups (typical vs. forcible tricks), only those with typical tricks showed significant decrease in corticomuscular coherence during tricks. ConclusionsObserved changes in the corticocortical coherence suggest that sensory tricks improve cortical function, which reduces corticomuscular connectivity and the dystonia. SignificanceWe demonstrated that sensory tricks fundamentally affect sensorimotor integration in CD, both in movement preparation and execution.

TOR1A mutation-related isolated childhood-onset generalised dystonia in South Africa.

Childhood-onset generalised dystonia is commonly caused by TOR1A mutations and is known to respond well to pallidal deep-brain stimulation (DBS) surgery. The incidence and prevalence of monogenic dystonia in individuals from Africa and specifically of African ancestry are unknown, and no local cases of TOR1A mutation dystonia are found in the literature. To describe our experience with the outcome of TOR1A mutation-positive patients with isolated generalised dystonia (IGD) of childhood onset who were treated with pallidal DBS. All patients with TOR1A mutations from Steve Biko Academic Hospital and the Pretoria Neurology Institute in Pretoria, South Africa (SA), who underwent DBS for IGD of childhood onset were identified. We conducted a retrospective analysis of their demographics, clinical presentation and time to generalisation, genetic status and family history, and response to DBS treatment of the internal segment of the globus pallidus (GPi), utilising pre- and post-surgical scores of the United Dystonia Rating Scale (UDRS). Three patients, all of black African ancestry, were identified. The median age at onset was 12 years and the median time to surgery from dystonia generalisation was 3 years. Two children presented with cervical-onset dystonia. Two patients were related, representing the only two with a positive family history. All three patients had a positive outcome after surgery, with improvement of 67 - 90% on the UDRS recorded at last follow-up. TOR1A mutations are found in SA patients of black African ancestry, with age of onset and generalisation comparable to those described in international studies. However, onset with cervical dystonia was more common than previously reported. Response to GPi DBS was excellent in all patients.

A2A receptors in mouse model of DYT1 primary dystonia

Adenosine A2A receptors are highly expressed in caudate-putamen, and external globus pallidus (GPe), but in trace in internal globus pallidus/entopeduncular nucleus. A2A upregulation in striatal-pallidal pathway is associated with levodopa dyskinesias in Parkinson's disease. Instead, A2A downregulation has been reported in striatum of Huntington Disease. We investigated distribution of A2A receptors in basal ganglia of Tor1a+/− knock-out mouse model DYT1 primary dystonia.

Specific movement and disability improvements in Burke-Fahn-Marsden Dystonia Rating Scale derived from pallidotomy in refractory patients to medical treatment

BackgroundDystonia is a movement disorder associated with significant disability and is usually refractory to medical treatment. Pallidotomy may decrease dystonic movements. The aim of this study was to quantify movement and disability improvements through Burke-Fahn-Marsden Dystonia Rating Scale (BFMDRS). MethodsWe carried out a longitudinal clinical study in patients with refractory primary and secondary dystonia, who underwent radiofrequency (RF) unilateral and bilateral lesions on the postero-ventro-lateral globus pallidus internus (GPi), evaluating the outcomes through BFMDRS and variables as age, time of evolution, etiology, body distribution, planned target coordinates, and lesion size, during a mean follow-up time of 35.67 months. ResultsNine RF pallidotomies were performed on 6 patients, 7 right-sided and 2 left-sided; three patients were treated unilaterally for one occasion, while the others underwent 2 surgeries, including one staged bilateral procedure. Mean BFMDRS scores for movement were 38.5 preoperative and 25.5 postoperative, and for disability were 20.4 preoperative and 17.3 postoperative. We noticed improvement in movement (32.54%, p = 0.001) and disability (17.23%, p = 0.002). There was one right GPi and internal capsule (IC) infarction with contralateral hemiparesis as sequelae. ConclusionsRF pallidotomy is an effective and accessible procedure to reduce BFMDRS scores in refractory dystonia if patients are correctly selected by severity, evolution, and disability as determining factors.

Diagnostic criteria for blepharospasm: A multicenter international study

BackgroundThere are no widely accepted criteria to aid the physician in diagnosing BSP. ObjectiveTo validate recently proposed diagnostic criteria for blepharospasm in a larger and geographically diverse population and to develop a screening system for blepharospasm. MethodsVideo-recordings from 211 blepharospasm patients and 166 healthy/disease controls were examined by 8 raters. Agreement for presence of orbicularis oculi spasms, sensory trick, and increased blinking was measured by k statistics. Inability to voluntarily suppress the spasms was asked by the examiner but not captured in the video. Patients/controls were also requested to fill a self-administered questionnaire addressing relevant blepharospasm clinical aspects. The diagnosis at each site was the gold standard for sensitivity/specificity. ResultsAll the study items yielded satisfactory inter/intra-observer agreement. Combination of items rather than each item alone reached satisfactory sensitivity/specificity. The combined algorithm started with recognition of spasms followed by sensory trick. In the absence of a sensory trick, including “increased blinking” or “inability to voluntarily suppress the spasms” or both items yielded 88–92% sensitivity and 79–83% specificity. No single question of the questionnaire yielded high sensitivity/specificity. Serial application of the questionnaire to our blepharospasm and control subjects and subsequent clinical examination of subjects screening positive by the validated diagnostic algorithms yielded 78–81% sensitivity and 83–91% specificity. ConclusionThese results support the use of proposed diagnostic criteria in multi-ethnic, multi-center cohorts. We also propose a case-finding procedure to screen blepharospasm in a given population with less effort than would be required by examination of all subjects.

Neurological Side Effects of Psychotropic Medications

This article focuses on potential neurological side effects secondary to psychotropic agents. Clinicians often have to manage these relatively commonly occurring neurological side effects. These potential side effects, if not managed in a timely manner, could be a significant negative factor impacting compliance and tolerability as well as efficacy. This article reviews mechanism of action, assessment, diagnosis, and management of extrapyramidal side effects including akathisia, acute dystonia, parkinsonism, and tardive dyskinesia, as well as motor tics, tremors, myoclonus, seizures, and delirium. Prescribing clinicians need to be cognizant of these potential side effects and knowledgeable in assessing and managing them. [ Psychiatr Ann . 2021;51(9):402–409.]

A-72 Neuropsychological Functioning in Primary Dystonia: A Multi-Domain Meta-Analysis

Abstract Objective Primary dystonia is conventionally seen as a motor disorder, though growing literature indicates cognitive dysfunction among persons with primary dystonia (PWD). Here, we completed a meta-analysis comparing cognition on clinical measures between PWD and normal controls. Method We searched PubMed, Embase, Cochrane Library, Scopus, and PsycINFO using a uniform search-strategy to locate original research comparing cognition between PWD and control samples. All analyses were modeled under random-effects. We used Hedge’s g as a bias-corrected estimate of effect size. Between-study heterogeneity was assessed using Cochran’s Q and I2. Results The initial search strategy yielded 866 results. Twenty studies were analyzed (PWD n = 739, control n = 865; 254 effect sizes extracted). Meta-analysis showed a significant combined effect size of primary dystonia across all studies (g = −0.55, p &amp;lt; 0.001), with low heterogeneity (Q = 23.60, p = 0.21, I2 = 19.49). Trim-and-fill procedure estimated 6 studies missing due to publication bias (adjusted g = −0.47, Q = 44.58). Within-domain effects of primary dystonia were: Motor/Non-Motor Speed = −0.76, Global Cognition/Orientation = −0.65, Language = −0.62, Executive Functioning = −0.50, Learning/Memory = −0.46, Visuospatial/Construction = −0.44, and Simple/Complex Attention = −0.36. Heterogeneity was generally low within domains. Effects were comparable between Speed tasks with (g = −0.85) and without (g = −0.80) a motor component. Meta-regressions indicated age, education, gender, and disease duration were not related to effect sizes. Conclusions PWD consistently demonstrated lower performances on neuropsychological tests compared to controls. Effect sizes were generally moderate in strength, with smallest effects in Simple/Complex Attention, and largest in Motor/Non-Motor Speed. Within the Speed domain, results suggested cognitive slowing beyond effects from motor symptoms. This quantitative summary indicates that PWD experience difficulties in multiple aspects of cognition, as detected by neuropsychological tests.

A case of delayed-onset multiple oculogyric crisis and torticollis episodes after low dose intramuscular haloperidol in a non-neuroleptic drug overdose setting

This report documents a rare case of delayed-onset multiple acute dystonias after treatment with low dose intramuscular (IM) haloperidol lactate injection in a setting of non-neuroleptic drug overdose. The drug–drug interactions between haloperidol and high levels of paracetamol and naproxen are deliberated upon. A 25-year-old Asian female was admitted after an intentional overdose of medications (paracetamol, naproxen and pregabalin). She received 5 mg of IM haloperidol injection for agitation. 21 hours later she experienced mild intermittent ocular deviation in an upward and outward direction and generalised stiffness, which were self-resolving. An hour later, she required another 2.5 mg of IM haloperidol injection for further agitation. In the 35 hours following her first IM haloperidol (13 hours after the second IM haloperidol), she developed a total of three episodes of oculogyric crisis (OGC) with torticollis. Each episode was treated promptly with IM diphenhydramine 25 mg, and there was remission of symptoms within 15 minutes of treatment. An objective causality assessment revealed a definite relationship between the episodes of acute dystonia with IM haloperidol therapy. Where oral alternatives and IM atypical antipsychotics/benzodiazepines are unavailable, rapid tranquillisation with a high-potency typical antipsychotic is a possibility. However, consideration should be made to combine haloperidol with an anticholinergic agent as prophylaxis against acute dystonia, especially in the setting of drug overdose, even if it is that of a non-neuroleptic drug (in this case, paracetamol and naproxen).

Sleep-related hypermotor seizures of insulo-opercular origin: A review of 27 cases

Introduction . Insular epilepsy is known to be a “great mimicker” of various types of focal epilepsy, including sleep-related hypermotor epilepsy (SHE) in about 15% of cases. Because SHE tends to have a frontal origin, misdiagnosis can result in a poor surgical outcome when epilepsy surgery is considered. In this study, we reviewed the existing literature to identify electro-clinical features suggestive of an insulo-opercular (IO) origin in SHE. Methodology . We found 11 case series of SHE or insular epilepsy with sufficiently detailed patient data. We identified 27 patients with IO SHE from 6 studies. When available, the semiology patterns (SP), early nonmotor manifestations, alteration of consciousness (AOC), EEG and MRI features and histopathology were collected. Results . SP analysis of the 27 identified patients showed a wide variety of indiscriminate SP, including hyperkinetic and/or dystonic features. Early nonmotor manifestations were observed in 80%, most commonly somatosensory auras (44%). Laryngeal constriction, pain, oral and widespread paresthesia were observed in IO SHE but not in frontal SHE. AOC was uncommon (20%). EEG was rarely suggestive of an IO origin (17%) and MRI was diagnostic in 48%. Focal cortical dysplasia was the most common diagnosis (78%). Following surgery, 83% were seizure-free (follow-up: 12–60 months). Conclusions . Few electro-clinical features, except early non-motor manifestations, are suggestive of an IO onset in SHE. Therefore, in MRI-negative patients, a heightened vigilance for subtle subjective or behavioral signs is warranted to avoid misdiagnosis and poor surgical outcome.

The prevalence and characteristics of misophonia in Ankara, Turkey: population-based study.

Misophonia is defined as significant distress (anger, distress or disgust) when exposed to certain sounds that would not affect most people, such as lip smacking or gum chewing. Although misophonia is common, the aetiology, prevalence and effective treatments are largely unknown. Based on our proposed diagnostic criteria, we examined the prevalence of misophonia and its relationship with clinical and demographic variables in a large representative population sample. We used a household sample (N = 541) of all residents aged >15 years, living in 300 homes randomly selected in Ankara city centre, Turkey. All participants were assessed at their homes by trained interviewers, for sociodemographic variables, misophonic sounds and related factors, using a semi-structured interview (the Misophonia Interview Schedule) developed for the current research. The current misophonia diagnosis prevalence was 12.8% (n = 69 of 541), although 427 (78.9%) participants reported at least one sound that was distressing. The mean number of misophonic sounds was 8.6 (s.d. 8.9, range 0-44); the figure was 17.6 in those with misophonia compared with 7.3 in those without misophonia. Of those with misophonia, only 5.8% contacted services for their condition. Predictors of misophonia diagnosis included younger age, family history of misophonia and previous contact with mental health services. Our study showed that misophonia is common in the general population, may cause significant disruption in daily life and is undertreated. Although more evidence is needed to classify misophonia as a psychiatric disorder, our findings support others who claim that the condition belongs to the group of mental disorders.