Page 156 - Let them speak Towards a patient reported outcome measure set for

Chapter 6
Finally, behaviour emerged as a key outcome for GID in our qualitative
study (van Silfhout, van Muilekom, van Karnebeek, Haverman, and van
Eeghen 2025), but was not included in this Delphi study. Behaviour is a
multidimensional concept and a manifestation of underlying health issues,
such as anger, leading to externalizing behaviour, or depression, resulting
in internalizing behaviour. By including these underlying health issues in
the final core PRO set, behaviour is (partially) captured. However, further
research, for example as part of a validation study, is warranted to see if we
missed certain components of behaviour.
The core PRO set in care and research
While selecting the core PRO set, we were careful to obtain a balance
between comprehensiveness and maintaining future usability (Williamson
et al. 2017), as especially caregivers are often already overburdened and
resistant of large questionnaires. The core PRO set is relatively extensive
and diverse compared with other COS. However, it likely reflects the complex
and heterogeneous nature of individuals with GID. It is therefore important
that this set will be measured using as few items as possible to minimize
burden on respondents. In care, the PROs should be measured, assessed,
and discussed during consultations with the healthcare professional, with
the goal of adequately monitoring the functioning of affected individuals,
screening for relevant problems, and establishing personalized treatments.
In research, the set should be measured to ensure relevant outcomes for
affected individuals.
Strengths and limitations
A strength of this study is the inclusion of individuals with GID and
caregivers in the Delphi study and in one of the consensus meetings, which
is not commonly done during full COS development studies (Gargon et al.
2017). Furthermore, the participation of experts from various countries
contributed to a broad perspective, enhancing the generalizability of the
core PRO set across different countries.
A limitation of this study is that only individuals with GID who were able to
complete an online survey and/or reflect on their own responses and those
of the participant groups were included in the Delphi study and consensus
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