181 Psychometrics of the pediatric PROMIS measures in hemophilia Dutch language or children who were unable to complete the PROMs were excluded, as determined by the treating clinician. The Medical Ethics Committees of the participating centers approved this study. All participants signed online informed consent. 2.2. Measurements 2.2.1. Patient characteristics The sociodemographic questionnaire included questions about the caregivers (eg, country of birth, educational level, marital status), the child (eg, position in family, school, sports), and clinical characteristics/variables (eg, type and severity of hemophilia, treatment, bleeding episodes, comorbidities). 2.2.2. PROMIS pediatric measures Six PROMIS pediatric measures were assessed as CAT: V2.0 Pain Interference [35], V2.0 Fatigue [36], V2.0 Anxiety [37], V2.0 Depressive Symptoms [37], V2.0 Mobility [38], and V2.0 Peer Relationships [39]. For two domains, no CAT was available; therefore, we used the fixed scales; V2.0 Anger 9a scale [40] and V1.0 Global Health scale (7+2) [41]. All PROMIS pediatric measures use a 7-day recall period. Items are scored on a 5-point Likert scale ranging from 1 (“never”) to 5 (“almost always”), except for the domains Mobility (ranging from “not able to” to “with no trouble”) and Global Health (response categories differ for each item, eg, ranging from “excellent” to “poor”). The CAT automatically stopped when the standard error of the estimate (SE) was ≤ 3.2 (90% reliability) and/or a maximum of 12 items was administered. PROMIS total scores were calculated by transforming the item scores into a T-score ranging from 0 to 100. For all PROMIS pediatric measures, higher scores represent more of the construct (eg, more pain interference or better peer relationships). The scores of the total scales were calculated with use of the PROMIS Assessment Centre Scoring Service (https://www.assessmentcenter.net/ac_scoringservice). 2.2.3. Legacy instruments The Haemo-QoL is a widely-used disease-specific instrument developed for the assessment of HRQoL of children with hemophilia [42]. The Haemo-QoL consists of different age versions. For this study, we used the Dutch versions for children 8 to 12 years (64 items) and adolescents 13 to 16 years (including children aged 17; 77 items). The Haemo-QoL measures 10 domains (Physical Health, Feeling, Attitude, Family, Friends, Coping, Other People, Sport and School, Dealing, and treatment), and two additional domains for the adolescent version (Future and Relationship). 7
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