Page 82 - Let them speak Towards a patient reported outcome measure set for

Chapter 4
Abstract
Background The impact of genetic intellectual disability (GID) on daily
life is significant. To better understand the impact of GID, it is essential to
measure relevant patient reported outcomes (PROs). The aim of this study
is to provide an overview of PROs used for individuals with GID, laying the
groundwork for a future generic core PRO set for GID.
Methods To identify PROs used for individuals with GID, results of two
literature reviews were integrated; 1) PROs extracted from a scoping
review on outcomes in clinical trials, and 2) PROs identified from a scoping
review on core outcome sets (COSs) for specific GIDs through a search in
MEDLINE (Ovid), PsycINFO, Embase, and the COMET database. Descriptive
analyses were performed.
Results In the first scoping review, 66 different PROs were identified. In the
second scoping review, 22 different PROs were identified. After integrating
PROs, 18 unique PROs remained, which were classified into a conceptual
framework. Most frequently reported PROs were quality of life, perceived
health, cognitive functioning, anxiety/stress, and depressive symptoms.
Conclusion This study provides an overview of PROs used for individuals
with GID. These results will assist in developing a generic core PRO set for
GID, to harmonize PROs used in care and research.
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