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PATIENT-REPORTED OUTCOMES IN RARE LYSOSOMAL...
Journal article

PATIENT-REPORTED OUTCOMES IN RARE LYSOSOMAL STORAGE DISEASES: KEY INFORMANT INTERVIEWS AND A SYSTEMATIC REVIEW PROTOCOL

Abstract

OBJECTIVES: To investigate the use, challenges and opportunities associated with using patient-reported outcomes (PROs) in studies with patients with rare lysosomal storage diseases (LSDs), we conducted interviews with researchers and health technology assessment (HTA) experts, and developed the methods for a systematic review of the literature. The purpose of the review is to identify the psychometrically sound generic and disease-specific …

Authors

Miller PA; Mulla SM; Adams-Webber T; Sivji Y; Guyatt GH; Johnston BC

Journal

International Journal of Technology Assessment in Health Care, Vol. 32, No. 6, pp. 393–399

Publisher

Cambridge University Press (CUP)

Publication Date

2016

DOI

10.1017/s0266462316000568

ISSN

0266-4623