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Research priorities in spasmodic dysphonia.

ABSTRACT: OBJECTIVE:To identify research priorities to increase understanding of the pathogenesis, diagnosis, and improved treatment of spasmodic dysphonia. STUDY DESIGN AND SETTING:A multidisciplinary working group was formed that included both scientists and clinicians from multiple disciplines (otolaryngology, neurology, speech pathology, genetics, and neuroscience) to review currently available information on spasmodic dysphonia and to identify research priorities. RESULTS:Operational definitions for spasmodic dysphonia at different levels of certainty were recommended for diagnosis and recommendations made for a multicenter multidisciplinary validation study. CONCLUSIONS:The highest priority is to characterize the disorder and identify risk factors that may contribute to its onset. Future research should compare and contrast spasmodic dysphonia with other forms of focal dystonia. Development of animal models is recommended to explore hypotheses related to pathogenesis. Improved understanding of the pathophysiology of spasmodic dysphonia should provide the basis for developing new treatment options and exploratory clinical trials. SIGNIFICANCE:This document should foster future research to improve the care of patients with this chronic debilitating voice and speech disorder by otolaryngology, neurology, and speech pathology.

SUBMITTER: Ludlow CL

PROVIDER: S-EPMC2643054 | biostudies-literature | 2008 Oct

REPOSITORIES: biostudies-literature

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Research priorities in spasmodic dysphonia.

Ludlow Christy L CL Adler Charles H CH Berke Gerald S GS Bielamowicz Steven A SA Blitzer Andrew A Bressman Susan B SB Hallett Mark M Jinnah H A HA Juergens Uwe U Martin Sandra B SB Perlmutter Joel S JS Sapienza Christine C Singleton Andrew A Tanner Caroline M CM Woodson Gayle E GE

Otolaryngology--head and neck surgery : official journal of American Academy of Otolaryngology-Head and Neck Surgery 20081001 4

<h4>Objective</h4>To identify research priorities to increase understanding of the pathogenesis, diagnosis, and improved treatment of spasmodic dysphonia.<h4>Study design and setting</h4>A multidisciplinary working group was formed that included both scientists and clinicians from multiple disciplines (otolaryngology, neurology, speech pathology, genetics, and neuroscience) to review currently available information on spasmodic dysphonia and to identify research priorities.<h4>Results</h4>Operat ...[more]

PMID: 18922334

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Project description:ImportanceThe relative outcomes of onabotulinum toxin A injections for treatment of adductor spasmodic dysphonia (ADSD), ADSD with lateral laryngeal tremor (ADSD+LT), and lateral LT without ADSD are unclear.ObjectiveTo compare the outcomes of onabotulinum toxin A treatment on ADSD, ADSD+LT, and lateral LT without ADSD.Design, setting, and participantsA retrospective cohort study was conducted from January 1, 1990, to September 30, 2016, at a tertiary referral voice center. Participants included 817 patients treated with onabotulinum toxin A injections for diagnosis of ADSD, ADSD+LT, and lateral LT without ADSD.ExposureInjection of onabotulinum toxin A into the thyroarytenoid/lateral cricoarytenoid muscle complex.Main outcomes and measuresData from patient diaries were used to evaluate patient-perceived effectiveness of onabotulinum toxin A injection. Primary outcomes were (1) patient-reported good voice days (voice breaks or tremor minimized to patient satisfaction) and (2) percentage of injections in which maximal voice quality was reached (significant or complete reduction in vocal tremor or spasms during a treatment cycle). Multivariate analysis of variance tests compared differences in outcomes between groups. Subanalysis was performed to compare outcomes in patients with isolated LT with those who had mixed tremor (lateral with concomitant anterior-posterior and/or vertical components).ResultsOf 817 patients treated with onabotulinum toxin A injections for laryngeal movement disorders, 548 patients (12 771 injection sessions) met inclusion criteria (ADSD: n = 328, ADSD+LT: n = 77, lateral LT without ADSD: n = 143). Of these, 408 (80.8%) were women; mean (SD) age was 57.2 (13.7) years. Among patients with tremor, those with isolated LT had better outcomes than those with mixed tremor. In adjusted analysis, good voice days in patients with ADSD, ADSD+LT, and lateral LT without ADSD were 81.1, 75.4, and 71.3 days, respectively (partial η2, 0.05; 95% CI, 0.01-0.09). The percentage of maximally beneficial injections was 88.1% for ADSD, 83.4% for ADSD+LT, and 70.4% for LT without ADSD (partial η2, 0.12; 95% CI, 0.06-0.17).Conclusions and relevanceOnabotulinum toxin A injections into the thyroarytenoid/lateral cricoarytenoid muscle complex are an effective treatment for ADSD, ADSD+LT, and LT without ADSD; however, the greatest effectiveness was observed among patients with ADSD. Defining tremor directionality may help to prognosticate the effectiveness of onabotulinum toxin A injection among patients presenting with tremor components.

Project description:ImportanceA roadblock for research on adductor spasmodic dysphonia (ADSD), abductor SD (ABSD), voice tremor (VT), and muscular tension dysphonia (MTD) is the lack of criteria for selecting patients with these disorders.ObjectiveTo determine the agreement among experts not using standard guidelines to classify patients with ABSD, ADSD, VT, and MTD, and develop expert consensus attributes for classifying patients for research.Design, setting and participantsFrom 2011 to 2016, a multicenter observational study examined agreement among blinded experts when classifying patients with ADSD, ABSD, VT or MTD (first study). Subsequently, a 4-stage Delphi method study used reiterative stages of review by an expert panel and 46 community experts to develop consensus on attributes to be used for classifying patients with the 4 disorders (second study). The study used a convenience sample of 178 patients clinically diagnosed with ADSD, ABSD, VT MTD, vocal fold paresis/paralysis, psychogenic voice disorders, or hypophonia secondary to Parkinson disease. Participants were aged 18 years or older, without laryngeal structural disease or surgery for ADSD and underwent speech and nasolaryngoscopy video recordings following a standard protocol.ExposuresSpeech and nasolaryngoscopy video recordings following a standard protocol.Main outcomes and measuresSpecialists at 4 sites classified 178 patients into 11 categories. Four international experts independently classified 75 patients using the same categories without guidelines after viewing speech and nasolaryngoscopy video recordings. Each member from the 4 sites also classified 50 patients from other sites after viewing video clips of voice/laryngeal tasks. Interrater κ less than 0.40 indicated poor classification agreement among rater pairs and across recruiting sites. Consequently, a Delphi panel of 13 experts identified and ranked speech and laryngeal movement attributes for classifying ADSD, ABSD, VT, and MTD, which were reviewed by 46 community specialists. Based on the median attribute rankings, a final attribute list was created for each disorder.ResultsWhen classifying patients without guidelines, raters differed in their classification distributions (likelihood ratio, χ2 = 107.66), had poor interrater agreement, and poor agreement with site categories. For 11 categories, the highest agreement was 34%, with no κ values greater than 0.26. In external rater pairs, the highest κ was 0.23 and the highest agreement was 38.5%. Using 6 categories, the highest percent agreement was 73.3% and the highest κ was 0.40. The Delphi method yielded 18 attributes for classifying disorders from speech and nasolaryngoscopic examinations.Conclusions and relevanceSpecialists without guidelines had poor agreement when classifying patients for research, leading to a Delphi-based development of the Spasmodic Dysphonia Attributes Inventory for classifying patients with ADSD, ABSD, VT, and MTD for research.

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Research priorities in spasmodic dysphonia.

Publications

Research priorities in spasmodic dysphonia.

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