Page 237 - Let them speak Towards a patient reported outcome measure set for

Summary
set. Chapter 6 described this process, which involved classifying and
conceptualizing (i.e., describing the content of each PRO in detail) the
PROs within a conceptual framework in collaboration with an expert group.
These PROs were then prioritized through a two-round Delphi survey and
consensus meetings with individuals with GID, caregivers, and experts.
This process resulted in the final core PRO set for children and adults with
GID. This final set included 19 PROs across all health domains, including
mobility/functioning of the lower extremities, depressive symptoms,
cognitive functioning, fatigue, and pain, underscoring the need for holistic
care and research for individuals with GID. This set fully overlapped with the
Dutch generic core PRO(M) set which is currently implemented in medical
care in the Netherlands, although the core PRO set established in this study
was more comprehensive.
Part 3: Selecting and psychometrically testing the core PROM set for GID
To adequately measure the core PRO set, PROMs were selected to form
the core PROM set. Chapter 7 outlines this selection process and the
psychometric evaluation of the selected PROMs. The final set consisted
mainly of PROMIS instruments administered through Computerized
Adaptive Testing (CAT), which tailors items to previous responses and
therefore provides shorter, more patient-specific, and reliable measurement
than conventional PROMs. The psychometric properties (i.e., reliability,
efficiency, and construct validity) of the core PROM set and conventional
legacy questionnaires, including the Pediatric Quality of Life Inventory
(PedsQL), Short Form-36 Health Survey (SF-36), Child Behavior Checklist
(CBCL), Adult Self-Report (ASR), and Adult Behavior Checklist (ABCL),
were assessed in caregivers of children with GID, adults with GID, and
caregivers of adults with GID. Overall, the core PROM set demonstrated
sufficient reliability and showed greater efficiency than most of the legacy
questionnaires. Moderate to strong correlations with legacy questionnaires
indicated that the PROM set also measured comparable constructs.
However, not all PROMs met the reliability thresholds, for example, the
PROMIS Short Form Anger for adults and for caregivers of adults or the
PROMIS CAT Fatigue for caregivers of children and adults. Their use should
therefore be approached with caution and further psychometric evaluation
is needed.
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