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Oral diadochokinetic markers of X-linked dystonia-parkinsonism.

Title: Oral diadochokinetic markers of X-linked dystonia-parkinsonism.
Authors: Kao TH; MGH Institute of Health Professions, 36 1st Ave, Charlestown Navy Yard, Boston, MA, 02129, United States. Electronic address: tkao@mghihp.edu.; Rowe HP; Department of Speech, Language, and Hearing Sciences, Boston University, 677 Beacon St, Boston, MA, 02215, United States. Electronic address: hprowe@bu.edu.; Green JR; MGH Institute of Health Professions, 36 1st Ave, Charlestown Navy Yard, Boston, MA, 02129, United States. Electronic address: jgreen2@mghihp.edu.; Stipancic KL; Department of Communicative Disorders and Sciences, University at Buffalo, Buffalo, NY, 14214, United States. Electronic address: klstip@buffalo.edu.; Sharma N; Department of Neurology, Massachusetts General Hospital and Harvard Medical School, Boston, MA, 02114, United States; The Collaborative Center for X-linked Dystonia-Parkinsonism, Massachusetts General Hospital, Charlestown, MA, 02129, United States. Electronic address: nsharma@partners.org.; de Guzman JK; Department of Neurology, Jose Reyes Memorial Medical Center, Manila, Metro Manila, 1012, Philippines; Sunshine Care Foundation, The Health Centrum, Roxas City, Capiz, 5800, Philippines. Electronic address: jekneuromd511@gmail.com.; Supnet-Wells ML; Department of Neurology, Massachusetts General Hospital and Harvard Medical School, Boston, MA, 02114, United States; The Collaborative Center for X-linked Dystonia-Parkinsonism, Massachusetts General Hospital, Charlestown, MA, 02129, United States. Electronic address: msupnet@mgh.harvard.edu.; Acuna P; Department of Neurology, Massachusetts General Hospital and Harvard Medical School, Boston, MA, 02114, United States; The Collaborative Center for X-linked Dystonia-Parkinsonism, Massachusetts General Hospital, Charlestown, MA, 02129, United States. Electronic address: pacuna1@mgh.harvard.edu.; Perry BJ; MGH Institute of Health Professions, 36 1st Ave, Charlestown Navy Yard, Boston, MA, 02129, United States. Electronic address: bjperry@mghihp.edu.
Source: Parkinsonism & related disorders [Parkinsonism Relat Disord] 2024 Mar; Vol. 120, pp. 105991. Date of Electronic Publication: 2024 Jan 04.
Publication Type: Journal Article
Language: English
Journal Info: Publisher: Elsevier Science Country of Publication: England NLM ID: 9513583 Publication Model: Print-Electronic Cited Medium: Internet ISSN: 1873-5126 (Electronic) Linking ISSN: 13538020 NLM ISO Abbreviation: Parkinsonism Relat Disord Subsets: MEDLINE
Imprint Name(s): Original Publication: Kidlington, Oxford, U.K. ; Tarrytown, NY : Elsevier Science, c1995-
MeSH Terms: Dystonia*/genetics ; Dystonic Disorders*/genetics ; Genetic Diseases, X-Linked*/complications ; Genetic Diseases, X-Linked*/genetics ; Parkinsonian Disorders*/genetics; Humans ; Dysarthria
Abstract: Introduction: X-linked dystonia-parkinsonism (XDP) is a neurodegenerative disorder that may result in severe speech impairment. The literature suggests that there are differences in the speech of individuals with XDP and healthy controls. This study aims to examine the motor speech characteristics of the mixed dystonia-parkinsonism phase of XDP.; Method: We extracted acoustic features representing coordination, consistency, speed, precision, and rate from 26 individuals with XDP and 26 controls using Praat, MATLAB, and R software. Group demographics were compared using descriptive statistics. A one-way analysis of variance (ANOVA) with Tukey's post hoc test was used to test for acoustic differences between the two groups.; Results: The XDP group had significantly lower consistency, speed, precision, and rate than controls (p 
Competing Interests: Declaration of competing interest This work was supported by the Massachusetts General Hospital Collaborative Center for X-linked Dystonia-Parkinsonism, Boston, MA; the National Institutes of Health (NIH)National Institute on Deafness and Other Communication Disorders [grant number K24DC0016312]; and the NIHNational Institute of Neurological Disorders and Stroke [grant number K23NS123369]. The funding sources did not participate in the study design; data collection, analysis, and interpretation; report writing; or decision to submit the article for publication. The authors declare that there are no conflicts of interest relevant to this work.
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Grant Information: K23 NS123369 United States NS NINDS NIH HHS; K24 DC016312 United States DC NIDCD NIH HHS
Contributed Indexing: Keywords: Acoustic analysis; Mixed dystonia-parkinsonism phenotype; Motor speech; X-linked dystonia-parkinsonism (XDP)
SCR Disease Name: Dystonia 3, Torsion, X-Linked
Entry Date(s): Date Created: 20240107 Date Completed: 20240226 Latest Revision: 20250302
Update Code: 20260130
PubMed Central ID: PMC10922526
DOI: 10.1016/j.parkreldis.2024.105991
PMID: 38184995
Database: MEDLINE

Journal Article