Page 134 - Let them speak Towards a patient reported outcome measure set for

Chapter 6
Abstract
Background Improving care and research for individuals with genetic
intellectual disabilities (GID) requires the identification and measurement
of relevant patient reported outcomes (PROs). PROs represent the patient
perspective on their health status. Currently, a myriad of potentially irrelevant
PROs is being measured for individuals with GID. Therefore, the aim of this
study is to identify the most relevant PROs through a Delphi survey and
consensus meetings, and develop a generic core PRO set applicable to the
whole GID population to be used in care and research.
Methods PROs, previously identified through a comprehensive literature
review and a qualitative study, were integrated and conceptualised into a pilot
generic core PRO set with an expert group. This pilot set was presented in a
two-round Delphi survey with individuals with GID, caregivers, and experts.
Consensus was set at 60% or more of all participant groups rating a PRO
as important for inclusion in the final core PRO set, or as not important for
exclusion. The Delphi surveys were followed by two consensus meetings
with individuals with GID, caregivers, and experts to reach consensus on
the undecided PROs.
Results Twelve individuals with GID, 21 caregivers, and 28 experts (total n =
61) participated in the first Delphi round. Twenty-nine PROs were presented
to the participants. In the first round, consensus was reached on one
important PRO ‘fatigue’. In the second round, consensus was reached on
12 important PROs: fatigue, sleep, physical functioning/activities, quality of
life, social functioning/participation, perceived health, cognitive functioning,
depressive symptoms, mobility/functioning of the lower extremities,
receptive communication, expressive communication, and sensory over-
responsivity. During the consensus meetings, consensus was reached
on seven additional important PROs: pain, anxiety/stress, anger, vision,
hearing, gastrointestinal symptoms, and mental functioning. This resulted
in a final generic core PRO set including the 19 PROs.
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