Evidence map›Paper›PMID 33334627›Full record

ArticleJournal of voice : official journal of the Voice Foundation2023

Progressive Decline in Voice and Voice-Related Quality of Life in X-Linked Dystonia Parkinsonism.

Sungjin A Song, Criscely L Go, Patrick B Acuna, Jan Kristopher Palentinos De Guzman, Nutan Sharma, Phillip C Song

Open access · greenAbstract read
In one paragraph

Article in Journal of voice : official journal of the Voice Foundation, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
0.4field-weighted citation impact, top 38% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

2 citing papers in PubMed, 5 citations in OpenAlex.

  1. Article
  2. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

6 authors at 4 institutions in 2 countries.

Sungjin A SongDepartment of Otolaryngology, Massachusetts Eye and Ear, Boston, Massachusetts; Department of Otolaryngology, Harvard Medical School, Boston, Massachusetts. Electronic address: Sungjin_Song@meei.harvard.edu.
Criscely L GoDepartment of Neurology, Jose Reyes Memorial Medical Center, Manila, Philippines; The Collaborative Center for X-linked Dystonia Parkinsonism, Department of Neurology, Massachusetts General Hospital, Boston, Massachusetts.
Patrick B AcunaThe Collaborative Center for X-linked Dystonia Parkinsonism, Department of Neurology, Massachusetts General Hospital, Boston, Massachusetts.
Jan Kristopher Palentinos De GuzmanDepartment of Neurology, Jose Reyes Memorial Medical Center, Manila, Philippines; The Collaborative Center for X-linked Dystonia Parkinsonism, Department of Neurology, Massachusetts General Hospital, Boston, Massachusetts.
Nutan SharmaThe Collaborative Center for X-linked Dystonia Parkinsonism, Department of Neurology, Massachusetts General Hospital, Boston, Massachusetts.
Phillip C SongDepartment of Otolaryngology, Massachusetts Eye and Ear, Boston, Massachusetts; Department of Otolaryngology, Harvard Medical School, Boston, Massachusetts.
José R. Reyes Memorial Medical Center · PHMassachusetts General Hospital · USHarvard University · USMassachusetts Eye and Ear Infirmary · US

Funding

TorsinA as a key link in receptor-mediated signalingP01NS087997 · NINDS · MASSACHUSETTS GENERAL HOSPITAL · PI BREAKEFIELD, XANDRA OWENS, OZELIUS, LAURIE J. · 2015 to 2019
$6.4M
Natural history study of x-linked Dystonia ParkinsonismR21NS118541 · NINDS · MASSACHUSETTS GENERAL HOSPITAL · PI SHARMA, NUTAN · 2020 to 2021
$420k
NINDS NIH HHS P01 NS087997NINDS NIH HHS R21 NS118541
6 · The paper itself

Abstract

objectiveTo characterize the evolution of swallowing and voice in patients with X-linked dystonia parkinsonism (XDP). STUDY

designRetrospective case series.

methodsRetrospective review of 59 patients with XDP from January 2016 to January 2018. All patients underwent complete examinations and quality of life (QOL) surveys (Swallowing Quality of Life questionnaire [SWAL-QOL], Voice-Related Quality of Life [V-RQOL], and Voice Handicap Index [VHI]), and functional endoscopic examination of swallowing. We excluded patients with incomplete records or patients lost to follow-up. Univariate analysis was used to compare 2016 to 2018 Penetration-Aspiration Scale (PAS), SWAL-QOL, V-RQOL, and VHI scores.

resultsTen patients met the inclusion criteria. Nine patients had oromandibular dystonia. Voice-related measures significantly worsened with an increase in mean VHI from 81 to 109.9 (P = 0.026) and decrease in mean V-RQOL from 58 to 28 (P = 0.013). Vocal strain also significantly worsened 0.4 to 1.4 (P = 0.001). Mean PAS scores increased from 4.2 to 5.1 (P = 0.068) and mean SWAL-QOL decreased from 50.4 to 43.5 (P = 0.157). In the SWAL-QOL, the mean Eating Duration score worsened from 0.9 to 0.4 (P = 0.052) and Mental Health score declined from 10.1 to 6.1 (P = 0.077).

conclusionsBoth vocal strain and voice-related QOL measures considerably worsened over the 2-year interval in our limited group of XDP patients with no significant change in PAS scores or swallowing QOL. The findings demonstrated that the pace of disease affecting voice symptoms was different from swallowing symptoms in our study group and that changes in communication ability may be a more sensitive marker for disease progression than swallowing dysfunction.

Indexed as

DystoniaParkinsonian DisordersVoiceDystonic DisordersGenetic Diseases, X-LinkedHumansQuality of LifeRetrospective StudiesSurveys and QuestionnairesDysphagiaDysphoniaQuality of lifeXDPX-linked dystonia parkinsonism

Identifiers

PMID33334627
PMCPMC10222671
OpenAlexW3110842149

What OpenQuestion holds

Textmetadata
LicenceTDM
Read underepoch 390

Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.