The requirement for a disease-specific patient-reported outcome measure of dysphagia in oculopharyngeal muscular dystrophy.
content validity
dysphagia symptoms
oculopharyngeal muscular dystrophy
oropharyngeal dysphagia
patient-reported outcome
Journal
Muscle & nerve
ISSN: 1097-4598
Titre abrégé: Muscle Nerve
Pays: United States
ID NLM: 7803146
Informations de publication
Date de publication:
04 2019
04 2019
Historique:
received:
16
10
2017
revised:
11
12
2018
accepted:
15
12
2018
pubmed:
24
12
2018
medline:
6
7
2019
entrez:
22
12
2018
Statut:
ppublish
Résumé
There is no patient-reported outcome (PRO) questionnaire specifically designed to assess oropharyngeal dysphagia in oculopharyngeal muscular dystrophy (OPMD). To select a suitable questionnaire, content validity of the existing questionnaires must be assessed. This study sought (1) to identify dysphagia-related symptoms in OPMD and (2) to assess content validity of currently available PRO for the assessment of dysphagia severity in OPMD. A two-step literature review was conducted of dysphagia-related symptom identification and oropharyngeal dysphagia-related PRO. Symptoms were validated with an expert panel by using a Delphi survey. Content validity of PRO questionnaires was documented through content analysis. Ten PRO questionnaires were identified. None of the questionnaires cover the entire symptom spectrum in OPMD and thus lack content validity. The development and validation of a new PRO questionnaire to assess dysphagia in OPMD is required to establish the importance of symptomatic relief from new treatments. Muscle Nerve 59:445-450, 2019.
Types de publication
Journal Article
Research Support, Non-U.S. Gov't
Validation Study
Langues
eng
Sous-ensembles de citation
IM
Pagination
445-450Subventions
Organisme : Bioblast Pharma, LTD
Pays : International
Organisme : Junior 2 FRQS Researcher award
ID : CC#256277
Pays : International
Organisme : Junior 2 FRQS Researcher award
ID : CG#231011
Pays : International
Organisme : Groupe de recherche interdisciplinaire sur les maladies Neuromusculaires-Bioblast
Pays : International
Informations de copyright
© 2018 Wiley Periodicals, Inc.