Page 175 - Let them speak Towards a patient reported outcome measure set for

Evaluating the core PROM set
Selecting a core PROM set
We first established a core PRO set for GenID by identifying potentially
relevant PROs through scoping reviews and focus groups and interviews
with individuals with GenID, caregivers, and clinical experts (26, 27), as
detailed earlier in our study protocol (29). These PROs were discussed
in an expert group meeting and subsequently prioritized with individuals
with GenID, caregivers, and experts (28), resulting in a core PRO set. The
research team (NvS, MvM, LH, AvE), including PROM experts (MvM, LH),
subsequently selected PROMs to measure the corresponding PROs of
interest. We aimed to include PROMs with the following characteristics:
generic (i.e., suitable for the whole GenID population), brief, easy to
complete, available in Dutch, applicable to both children and adults, offered
as self- and proxy-report, and either free or low-cost. Where possible, we
aligned them with the Dutch generic PROM set and the international core
PROMIS instruments (17, 24, 25). If a PROMIS instrument was not available
for a specific PRO, we consulted experts in the field of that health domain
and/or PROMs to help identify the most appropriate PROM to measure
that specific PRO. Not all PROs were paired with a corresponding PROM
due to the lack of an instrument that included all characteristics described
above (e.g., applicable to both children and adults). In the absence of proxy-
report versions of the PROMs for adults, self-report versions of the PROMs
for adults were used as proxy-reports. Caregivers of adults were clearly
instructed to complete the PROMs on behalf of the individual with GenID,
not themselves.
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Evaluating the psychometric properties of the selected core PROM set
Study participants and procedure
Caregivers were eligible if they provided care for an individual aged 5 years
or older with ID or a genetic neurodevelopmental disorder associated with
ID, ranging from normal intellectual functioning to profound ID. Adults
were eligible if they were aged 18 years or older, had ID or a genetic
neurodevelopmental disorder associated with ID, ranging from normal
intellectual functioning to moderate ID, and were able to understand and
complete PROMs.
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