Page 193 - Let them speak Towards a patient reported outcome measure set for

Evaluating the core PROM set
Discussion
In this study, a GenID core PROM set was selected, and its reliability,
efficiency, and construct validity evaluated in a Dutch sample of caregivers
of children with GenID (proxy-report), adults with GenID (self-report), and
caregivers of adults with GenID (proxy-report). Most PROMIS CATs and SFs
were considered reliable, requiring completion of 4 to 13 items. The reliability
of the single-item PROMs varied. No floor or ceiling effects were observed
for PROMIS CATs and SFs, indicating that these questionnaires capture the
full spectrum of functioning in individuals with GenID. Most PROMIS CATs
and SFs were more efficient than most of the legacy subscales, reflected in
fewer items required and an absence of floor and ceiling effects, and often
showed moderate to high correlations with them, providing comparable
information. PROMIS Social Roles and Activities was able to discriminate
between adults with and without ID, whereas PROMIS Anger and Sleep
Disturbance showed less discrimination.
Although most PROMs included in the core PROM set were reliable, not
all met the reliability threshold. PROMIS SF Anger showed lower reliability
and test-retest reliability in adults and caregivers of adults. PROMIS
Fatigue for caregivers of children and adults, PROMIS Sleep Disturbance
for caregivers of adults, and PROMIS Cognitive Functioning – Abilities
for adults showed lower test-retest reliability. Some single-item PROMs
showed low test-retest reliability, such as NRS Well-being and NRS Health
for caregivers of children. A possible explanation is that complaints, such
as anger and fatigue, fluctuate more from day to day, resulting in lower test-
retest reliability. In contrast, overall cognitive functioning would be expected
to remain stable over a two-week period. However, PROMIS Cognitive
Functioning – Abilities captures subtle changes in cognition (e.g., ‘My
mind has been as sharp as usual’), which individuals with GenID may also
experience more often daily. Nevertheless, use of PROMIS SF Anger for
adults and caregivers of adults, PROMIS Fatigue for caregivers of children
and adults, PROMIS Sleep Disturbance for caregivers of adults, PROMIS
Cognitive Functioning – Abilities for adults, and some single-item PROMs
should be cautious, and further psychometric evaluation is warranted. For
example, testing these PROMs in larger GenID populations or by assessing
additional psychometric properties.
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