Literature DB >> 26453481

The relationship between physical symptoms and health-related quality of life in oculopharyngeal muscular dystrophy.

Sarah Youssof1.   

Abstract

INTRODUCTION: Oculopharyngeal muscular dystrophy (OPMD) causes ptosis, dysphagia, and limb weakness. Health-related quality of life (HRQoL) and its relationship to physical symptoms was investigated.
METHODS: The 36-item Short Form (SF-36) was completed by 89 participants in the U.S. OPMD Registry. Multiple hierarchical regression was used to determine the relative contributions of dysphagia severity and lower extremity functional impairment to the physical (PCS) and mental (MCS) components of the SF-36.
RESULTS: HRQoL was reduced in OPMD compared with population norms. Lower extremity functional impairment explained a significant proportion of variance in PCS and MCS. Dysphagia symptom severity explained a moderate amount of variance only in MCS. Dysphagia symptom severity had the strongest associations with general health perception and social functioning domains.
CONCLUSIONS: Lower extremity functional impairment in OPMD deserves attention due to its large influence on HRQoL. Both generic and dysphagia-specific measures are necessary to assess HRQoL in OPMD.
© 2015 Wiley Periodicals, Inc.

Entities:  

Keywords:  dysphagia; mobility; oculopharyngeal muscular dystrophy; quality of life; rare disease registry

Mesh:

Year:  2016        PMID: 26453481      PMCID: PMC4826630          DOI: 10.1002/mus.24932

Source DB:  PubMed          Journal:  Muscle Nerve        ISSN: 0148-639X            Impact factor:   3.217


  27 in total

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2.  Pain location and intensity impacts function in persons with myotonic dystrophy type 1 and facioscapulohumeral dystrophy with chronic pain.

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4.  Autologous myoblast transplantation for oculopharyngeal muscular dystrophy: a phase I/IIa clinical study.

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6.  [Proximal weakness and exercise-induced pain as initial symptom of oculopharyngeal muscular dystrophy].

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8.  Hip flexion weakness is associated with impaired mobility in oculopharyngeal muscular dystrophy: a retrospective study with implications for trial design.

Authors:  Sarah Youssof; Ronald Schrader; David Bear; Leslie Morrison
Journal:  Neuromuscul Disord       Date:  2014-11-20       Impact factor: 4.296

9.  The NIH Office of Rare Diseases Research patient registry Standard: a report from the University of New Mexico's Oculopharyngeal Muscular Dystrophy Patient Registry.

Authors:  Shamsi Daneshvari; Sarah Youssof; Philip J Kroth
Journal:  AMIA Annu Symp Proc       Date:  2013-11-16

10.  A multicentre postal survey investigating the contribution of illness perceptions, coping and optimism to quality of life and mood in adults with muscle disease.

Authors:  Christopher D Graham; John Weinman; Reza Sadjadi; Trudie Chalder; Richard Petty; Mike G Hanna; Chris Turner; Matt Parton; Paul Maddison; Aleksandar Radunovic; Cheryl Longman; Yvonne Robb; Kate Bushby; David Hilton-Jones; Michael R Rose
Journal:  Clin Rehabil       Date:  2013-11-15       Impact factor: 3.477

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  5 in total

1.  Novel mouse models of oculopharyngeal muscular dystrophy (OPMD) reveal early onset mitochondrial defects and suggest loss of PABPN1 may contribute to pathology.

Authors:  Katherine E Vest; Brittany L Phillips; Ayan Banerjee; Luciano H Apponi; Eric B Dammer; Weiting Xu; Dinghai Zheng; Julia Yu; Bin Tian; Grace K Pavlath; Anita H Corbett
Journal:  Hum Mol Genet       Date:  2017-09-01       Impact factor: 6.150

2.  Mental health care for rare disease in the UK - recommendations from a quantitative survey and multi-stakeholder workshop.

Authors:  Rosa Spencer-Tansley; Nick Meade; Farhana Ali; Amy Simpson; Amy Hunter
Journal:  BMC Health Serv Res       Date:  2022-05-14       Impact factor: 2.908

3.  Dysphagia-related quality of life in oculopharyngeal muscular dystrophy: Psychometric properties of the SWAL-QOL instrument.

Authors:  Sarah Youssof; Carol Romero-Clark; Teddy Warner; Emily Plowman
Journal:  Muscle Nerve       Date:  2017-02-12       Impact factor: 3.217

Review 4.  Health-Related Quality of Life (HRQoL) in Idiopathic Inflammatory Myopathy: A Systematic Review.

Authors:  Valérie Leclair; Malin Regardt; Sophie Wojcik; Marie Hudson
Journal:  PLoS One       Date:  2016-08-09       Impact factor: 3.240

Review 5.  Recent Progress in Oculopharyngeal Muscular Dystrophy.

Authors:  Satoshi Yamashita
Journal:  J Clin Med       Date:  2021-03-29       Impact factor: 4.241

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