Page 220 - Let them speak Towards a patient reported outcome measure set for

Chapter 8
In addition, recruitment of individuals with GID and their caregivers was
challenging and time-consuming across studies. Many potential participants
were overburdened and therefore reluctant to engage in research. This may
also have led to selection bias, with an over-representation of affected
individuals and caregivers with greater capacity to cope and/or a higher
socio-economic status, underscoring the need for validation of the core
PRO(M) set in diverse subgroups. This aspect further highlights the
importance of designing studies that minimize participant burden. For
example, by conducting research with individuals with GID within their own
care organizations, schools, or neighborhoods, offering the option of online
participation, and providing study information that is simple and accessible.
Lastly, the psychometric properties of the core PROM set were evaluated
only in caregivers of children with GID, adults with GID, and caregivers of
adults with GID. Psychometric testing is also still required in caregivers of
young children and in children themselves. Use of the core PROM set in
these two groups should therefore be undertaken with caution. Moreover,
only adults with moderate ID to normal intellectual functioning participated
in the psychometric study. We considered individuals with severe to
profound ID unable to complete the self-report measures because of limited
cognitive abilities. Consequently, the set cannot yet be used as a self-report
tool for this group. Further research is needed to determine whether, and in
what way, individuals with severe to profound ID may be able to self-report.
International applicability
International stakeholders were involved throughout the development of
the core PRO set. European patient representatives participated in focus
groups and interviews to ensure the identification of PROs relevant across
countries. In the Delphi study and consensus meetings, international
healthcare professionals and patient representatives contributed to the
final selection of PROs. However, no patients or caregivers from outside
the Netherlands participated (although some patient representatives might
also have been caregivers) and individuals with multiple ethnic backgrounds
were under-represented. It is therefore uncertain whether the set fully
reflects the priorities of the international and multi-ethnic population with
GID. This may be particularly relevant because, in the Delphi process, Dutch
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