Page 157 - Let them speak Towards a patient reported outcome measure set for

Establishing the core PRO set
meeting. Given the challenging nature of this task, the inclusion of affected
individuals with a wide range in intellectual functioning was difficult.
Although caregivers and experts also represent individuals with severe to
profound ID, the perspectives of these individuals are underrepresented,
potentially biasing the PROs towards those more relevant to individuals
with higher intellectual functioning. Additionally, the included individuals
and caregivers represent only a small fraction of the over 1500 identified
GIDs. It is also important to note that the insight into why participants
changed their responses in Delphi round two could have provided valuable
information. Several PROs, initially rated as not important by at least one
group, were considered important after receiving feedback from round
one. For example, only 24% of the caregivers rated depressive symptoms
as important in round one, increasing to 64% in round two. The specific
reasons for this shift remain unclear, as participants were not asked to
explain why they changed their response. Another limitation is that two
individuals with GID and three caregivers who attended the consensus
meeting had not participated in the Delphi study. Although participation in
the Delphi study was not a requirement, they did not go through the full
process and therefore missed feedback from round one, which may have
shaped their perspective differently. An additional limitation is that, in Delphi
round two, the PROs were ranked and the percentages of each participant
group considering a PRO important were displayed, which might have
created a bias by steering participants towards the majority opinion rather
than adhering to their own. Finally, because Dutch individuals with GID and
caregivers were more critical in rating the PROs, they had greater influence
on the final core PRO set. The set may therefore reflect their perspectives
more strongly than those of the international experts. When implementing
the set outside the Netherlands, further validation to assess its relevance
and completeness within the target population may be warranted.
Future research
In the next step, we will focus on selecting the most suitable PROMs
to measure the core PRO set. These PROMs should be short, easy to
complete, generic, and available as both self- and proxy-report. They
should be applicable to children and adults, including individuals with
learning disabilities and mild to profound ID, to encompass the whole
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