Transition from paediatric to adult ophthalmology services: what matters most to young people with visual impairment

Purpose: To identify the views and experiences and thus the transition-related needs of young people with visual impairment (VI), so as to inform future practice and policies. Patients and methods: Qualitative study of 17 young people aged 16–19 years (ie the conventional transition age threshold) with VI (best-corrected acuity logMAR worse than 0.48) and without additional impairments, drawn from a sampling frame of paediatric ophthalmology patients attending Great Ormond Street Hospital and Moorfields Eye Hospital (London, UK). In-depth, semistructured interviews were conducted to elicit their experiences, preferences, and attitudes towards transitioning within health care. Qualitative thematic analysis identified themes related to participants’ experience of transition. Results: Eight of 17 participants had transitioned out of paediatric ophthalmology services, 7 had not, and 2 were unsure. Their views and experiences varied. Only 2 of those who had transitioned preferred their prior paediatric service, and 1 still in a paediatric services did not want to transition. Age-appropriate communication and physical clinical environment were two key components of care, both associated with greater confidence to self-manage health care in the future as an adult. Emotional attachment to paediatric services/teams was associated with reluctance to transition. Conclusions: Generic guidance on transition is broadly applicable to children/young people with VI. Age-appropriate communication and appropriate physical clinical environments may be optimally delivered through adolescent ophthalmology services bridging paediatric and adult provision. Lack of research on transitions in paediatric ophthalmology has thus far restricted intervention studies; our findings serve to aid in developing an evidence base to achieve this

Measuring quality of life in muscular dystrophy

OBJECTIVES: The objectives of this study were to develop a conceptual model of quality of life (QOL) in muscular dystrophies (MDs) and review existing QOL measures for use in the MD population. METHODS: Our model for QOL among individuals with MD was developed based on a modified Delphi process, literature review, and input from patients and patient advocacy organizations. Scales that have been used to measure QOL among patients with MD were identified through a literature review and evaluated using the COSMIN (Consensus-Based Standards for the Selection of Health Measurement Instruments) checklist. RESULTS: The Comprehensive Model of QOL in MD (CMQM) captures 3 broad domains of QOL (physical, psychological, and social), includes factors influencing self-reported QOL (disease-related factors, support/resources, and expectations/aspirations), and places these concepts within the context of the life course. The literature review identified 15 QOL scales (9 adult and 6 pediatric) that have been applied to patients with MD. Very few studies reported reliability data, and none included data on responsiveness of the measures to change in disease progression, a necessary psychometric property for measures included in treatment and intervention studies. No scales captured all QOL domains identified in the CMQM model. CONCLUSIONS: Additional scale development research is needed to enhance assessment of QOL for individuals with MD. Item banking and computerized adaptive assessment would be particularly beneficial by allowing the scale to be tailored to each individual, thereby minimizing respondent burden

Known-groups validity of the Patient-Reported Outcomes Measurement Information System (PROMIS®) in adolescents and young adults with special healthcare needs

PURPOSE: To examine known-groups validity of the Patient-Reported Outcomes Measurement Information System (PROMIS(®)) Short Forms (SFs) for adolescents and young adults with special health care needs (SHCN) using data collected from the PROMIS Linking Study. METHODS: 292 adolescents aged 14–17.9 years and 300 young adults aged 18–20 years with SHCN from the PROMIS Linking Study were used for analyses. Presence of SHCN was classified into 3 categories (medication use, service use, and functional limitations). HRQoL was measured with the PROMIS Pediatric and Adult SFs. Differences in health-related quality of life (HRQoL) domain scores between SHCN sample and the norms of the PROMIS pediatric and adult calibration samples, respectively, were compared using t-tests. Associations of HRQoL scores with the presence of individual SHCN categories were tested using linear regression. RESULTS: All HRQoL domain scores for the SHCN samples were significantly worse than the respective calibration samples. Adolescents and young adults with SHCN-related service use and functional limitations reported worse HRQoL than those without service use and functional limitations (p’s<0.01). CONCLUSIONS: PROMIS Pediatric and Adult SFs possess satisfactory known-groups validity related to SHCN status