Page 213 - Let them speak Towards a patient reported outcome measure set for

General discussion
instruments using computer adaptive testing (CAT). In CAT, each item is
selected based on previous responses (13), providing instruments that are
shorter, more patient-centered, and more reliable than conventional PROMs
(14, 15). In addition, self-reports for affected individuals are used less
frequently, particularly for those with ID, because the administered PROM
items are considered too complex for them to understand and reflect upon
(16). This challenge was not addressed in this thesis. Further evaluation is
therefore required to determine whether the PROM set is understandable
for the entire GID population.
Third, PROMs had often not been sufficiently psychometrically tested
in the GID population (17), or had only been evaluated in a specific GID
subgroup (18). We addressed this by assessing the reliability, efficiency,
and construct validity of the core PROM set among caregivers of children
with GID, adults with GID, and caregivers of adults with GID, and showed
that the set was largely reliable and more efficient than most conventional
legacy questionnaires.
Finally, PROMs were not yet routinely used in GID care, largely because
available instruments measure potentially irrelevant PROs, are lengthy and
complex, and have not been sufficiently psychometrically tested in the GID
population. Consequently, it also remains uncertain whether these PROMs
accurately measure disease progression and treatment effects in research
with individuals with GID. We did not address the implementation of the
core PROM set in care and research in this thesis and this issue, therefore,
still warrants attention.
8
The core PRO set
While the ultimate yield of this thesis was the development of a core PROM
set intended for use in both clinical care and research, the core PRO set is
of equal importance. These PROs should inform care, research, and policy.
In clinical practice, care pathways should be established for each PRO. For
instance, while mental functioning (e.g., anxiety) is included in the set, clear
and structured care pathways for individuals with GID experiencing mental
health problems are often absent. ID and mental healthcare are often poorly
integrated, with organization varying by region. This fragmentation makes
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