Distribution and associations of vision-related quality of life and functional vision of children with visual impairment.

BACKGROUND: Patient-reported outcome measures (PROMs) are increasingly used in paediatric ophthalmology. However, little is known about the distribution of PROM scores among children and young people with visual impairment. AIM: To investigate the distributions and predictors of scores on the VQoL_CYP (measuring vision-related quality of life) and FVQ_CYP (measuring functional vision). METHODS: Children and young people aged 8-18 years, with visual impairment/blindness (logarithm of the minimum angle of resolution (LogMAR) worse than 0.48 in the better eye, and/or eligible visual field restriction) completed the VQoL_CYP and FVQ_CYP at home or Great Ormond Street Hospital, London, UK. Associations between VQoL_CYP and FVQ_CYP scores and sociodemographic and clinical factors were analysed using multiple linear regression models. RESULTS: Among 93 participants, VQoL_CYP scores ranged from 36.6 to 78.2 (mean=57.9, SD=8.1). FVQ_CYP scores ranged from 23.5 to 70.3 (mean=48.3, SD=10.1). Only 0.4% of the variation in VQoL_CYP scores was explained, with no associations with the variables of interest. By contrast, 21.6% of the variation in FVQ_CYP scores was explained, with a gradient of worse acuity (p<0.001) and female gender (p=0.04) associated with worse self-rated functional vision. Age, ethnicity, time of onset and stability/progression of visual impairment were not associated. DISCUSSION: Self-rated vision-related quality of life and functional vision are not readily predicted from sociodemographic or clinical characteristics that ophthalmologists measure/record. Routine use of PROMs in clinical practice can offer important insights. Use in research can provide valuable measures of effectiveness of interventions. The reference values provided will aid interpretation in both settings.

This is me: A qualitative investigation of young people's experience of growing up with visual impairment.

BACKGROUND: Childhood visual impairment (VI) has a profound impact on many aspects of childhood and adolescence. This is well-documented in cross-sectional and/or quantitative studies utilizing self-report instruments which compare children with and without VI. Young people's views on the experience of growing up with VI as a developmental, change-driven process remain largely unexplored. METHODS: As part of our broader research programme on quality of life of visually impaired children and young people in the United Kingdom, in-depth, semi-structured interviews were conducted between March and June 2015, with a stratified sample of 17 young people with VI, aged 16-19 years. An age-sensitive, empirically-based topic guide encouraged retrospective reflections on participants' experiences of growing up with VI, including age-normative and vision-specific challenges. RESULTS: Descriptions of growing up with VI largely centered on an overarching higher-order theme labelled becoming me. Four themes representing everyday activities, attitudes, preferences and perceptions in relation to i) social relationships, ii) independence and responsibilities, iii) the future, and iv) rising to challenges emerged and were used by participants in their description of three stages in which they developed a sense of self: i) laying the foundations, ii) testing the waters, and iii) this is me. Differences in manifestation of VI influenced how young people made sense of their experiences and their sense of self. CONCLUSIONS: Findings are discussed in relation to normative and vision-specific changes in psychosocial development during adolescence, including the development of identity. They highlight the need for ongoing monitoring of subjective well-being in a clinical population with a unique early life course trajectory.

Differences in Self-Rated Versus Parent Proxy-Rated Vision-Related Quality of Life and Functional Vision of Visually Impaired Children.

PURPOSE: To investigate disagreement between children's self-reported vision-related quality of life (VQoL) and functional vision (FV) and their parents' proxy-reports. DESIGN: Cross-sectional study. METHODS: A total of 152 children aged 7-18 years with visual impairment (VI) (defined by the World Health Organization), and their parents, were recruited from 22 National Health Service (NHS) ophthalmology departments in the United Kingdom. Age-appropriate versions of 2 vision-specific instruments, capturing VQoL and FV, were administered to children alongside modified versions for completion by parents on behalf of their child (ie, parent proxy-report). Disagreement between self-report and parent proxy-report was examined using the Bland-Altman (BA) method and a threshold of disagreement based on 0.5 standard deviation. Disagreement was analyzed according to participants' age, sex, and clinical characteristics, using logistic regression analyses. RESULTS: Children rated themselves as having better outcomes than their parents did, although parents both under- and overestimated their child's VQoL (mean score difference = 7.7). With each year of increasing age, there was a 1.18 (1.04-1.35) higher odds of children self-rating their VQoL better than their parents (P = .013). Although parents consistently underestimated their child's FV (mean score difference = -4.7), no characteristics were significantly associated with differences in disagreement. CONCLUSIONS: Disagreement between child self-report on the impact of VI and their parents' proxy-reports varies by age. This implies that self-report from children must remain the gold standard. Where self-reporting is not possible, parent proxy-reports may provide useful insights, but they must be interpreted with caution.

Feasibility of using patient-reported outcome measures with visually impaired children/young people attending paediatric ophthalmology clinics.

OBJECTIVE: To explore feasibility of using child/young person patient-reported outcome measures (PROMs) routinely in practice, using vision-specific instruments and paediatric ophthalmology as the exemplar. METHODS: Participants comprised patients aged 8-17 years, with visual impairment or low vision (visual acuity of the logarithm of the minimum angle of resolution (logMAR) worse than 0.3 in the better eye), attending the Department of Ophthalmology at Great Ormond Street Hospital, London, UK. All participants completed age-appropriate PROMs before attending their outpatient appointment. Half were randomly assigned to completion at home, with the choice of paper-and-pencil or electronic format. The other half were invited to complete PROMs during their hospital appointment, and randomly assigned to completion format. All participants completed a face-to-face survey exploring their attitudes and preferences. Analysis comprised survival analysis, and direct comparisons of proportions, with complementary qualitative data analysis. RESULTS: 93 patients participated. 48 (98%) completing PROMs at home chose the paper-and-pencil format. Completion at home took longer than at hospital (median=20, vs 14 min, p<0.001). Visual acuity was associated with completion time (p=0.007) and missing data (p=0.03). Overall, 52 (60%) reported a preference for completion at home but there was no clear preference for format (37 (43%) preferred either format). CONCLUSION: PROM completion at home ahead of hospital appointments may be preferable for collecting complete, high-quality datasets. Despite equipoise on preference for format, the majority of those completing at home chose the traditional paper-and-pencil format, despite impaired sight. These findings should inform implementation of child/young person PROMs into routine practice.

Attitudes, experiences, and preferences of ophthalmic professionals regarding routine use of patient-reported outcome measures in clinical practice.

BACKGROUND/OBJECTIVES: Routine use of patient-reported outcome measures (PROMs) to assess quality of health care systems is mandated in many countries and has been implemented successfully in many specialities. Ophthalmology currently lags behind. To support and inform future implementation, we investigated paediatric ophthalmic clinicians' experience of, and future training needs for, using child-appropriate vision PROMs and their views about the barriers and enablers to future routine implementation in clinical practice. METHODS: We conducted a pilot study, using an online survey to elicit the experience, attitudes, training needs and perceptions of barriers and enablers to routine PROMs use of ophthalmic health professionals in the Paediatric Ophthalmology Department at Great Ormond Street Hospital, London. A focus-group was undertaken to discuss survey results and preferences regarding presentation of PROM data. Analysis comprised descriptive statistics, presented alongside complementary qualitative data. RESULTS: Eighteen clinicians in the department completed the survey. Twenty-seven took part in the focus group. Clinicians had limited experience of using PROMs but high confidence in the potential positive impact on communication with patients, monitoring chronic conditions and clinical decision-making. Clinicians identified operational issues (collection and analysis of data) and impact (interpretation and application of data) as the two key areas for consideration. Training and information requirements before implementation were clearly articulated, alongside the benefits of using digital/electronic data capture ahead of consultations to allow efficiency and automated analysis, and presentation in an appropriate visual format alongside clinical data to ensure meaningful use. CONCLUSION: The findings of this pilot study of ophthalmic clinicians working in a specialist paediatric ophthalmology department, suggest that ophthalmic clinicians recognise the potential benefits of routine PROMs use in clinical practice. Together with existing literature outside ophthalmology relating to overcoming barriers and exploiting enablers to routine implementation, findings may be applicable in planning routine PROM implementation in paediatric ophthalmology.

A Patient-reported Outcome Measure of Functional Vision for Children and Young People Aged 8 to 18 Years With Visual Impairment.

PURPOSE: To develop age-appropriate extensions of a patient-reported outcome measure for capturing the functional impact of visual impairment on daily activities of children and young people aged 8 up to 18 years. DESIGN: Questionnaire development and validation study. METHODS: Pediatric Ophthalmology departments at Great Ormond Street Hospital and Moorfields Eye Hospital, and, in the final study phase, 20 further UK hospitals. Children and young people (aged 6-19 years) with visual impairment (acuity of the logarithm of the minimum angle of resolution (LogMAR) worse than 0.50 in the better eye) due to any cause but without significant non-ophthalmic impairments. We used our prototype FVQ_CYP for 10-15 year olds as the foundation. Twenty-nine semi-structured interviews confirmed relevance of existing, and identified new, age-specific items. Twenty-eight cognitive interviews captured information regarding comprehensibility and format. The FVQ_Child (8-12 years) and FVQ_Young Person (13-18 years) were evaluated with a national sample of 113 children and 96 young people using Rasch analysis. RESULTS: Issues emerging from interviews with children and young people were largely congruent with those elicited originally with 10-15 year olds. The 28-item FVQ_Child and 38-item FVQ_Young Person versions have goodness-of-fit statistics within the interval 0.5, 1.5 and person separation values of 5.87 and 6.09 respectively. Twenty-four overlapping "core" items enabled their calibration on the same measurement scale. Correlations with acuity (r = 0.47) demonstrated construct validity. CONCLUSIONS: The FVQ_C and FVQ_Young Person are robust age-appropriate versions of the FVQ_CYP which can be used cross-sectionally or sequentially/longitudinally across the age range of 8 up to 18 years in clinical practice and research.

An Age- and Stage-Appropriate Patient-Reported Outcome Measure of Vision-Related Quality of Life of Children and Young People with Visual Impairment.

PURPOSE: Developmentally sensitive measures of vision-related quality of life (VQoL) are needed to capture age-specific concerns about the impact of living with visual impairment (VI) in children and young people. Our objective was to use our validated VQoL instrument for children and young people 10 to 15 years of age (the VQoL_CYP) as the foundation for development of age-specific extensions. DESIGN: Questionnaire development. PARTICIPANTS: A representative sample of children and young people 6 to 19 years of age with VI, defined as visual acuity worse than 0.50 logarithm of the minimum angle of resolution in the better eye. They were recruited from pediatric ophthalmology clinics at Great Ormond Street Hospital and Moorfields Eye Hospital and, in the final phase of the study, from 20 additional United Kingdom hospitals. METHODS: Standard instrument development processes were followed across 4 phases. Twenty-nine semistructured interviews with children and young people permitted draft age-appropriate extensions. Twenty-eight cognitive interviews informed items and response options. Age-appropriate extensions were prepiloted with 49 participants to ensure feasibility and administered via a postal survey to a national sample of 160 participants for psychometric evaluation using Rasch analysis. Construct validity was evaluated through correlations with the Pediatric Quality of Life Inventory. MAIN OUTCOME MEASURES: Psychometric indices of validity and reliability of the instrument versions. RESULTS: Interviews confirmed that the existing VQoL_CYP content and format were relevant across a wider age range. Age-appropriate extensions were drafted for children (8-12 years) and young people (13-17 years). Psychometric item reduction produced 20-item child and 22-item young person versions, each with acceptable fit values, no notable differential item functioning, good measurement precision, ordered response categories and acceptable targeting, and no notable differential item functioning on items common to both. Construct validity was demonstrated through correlations with health-related quality of life (r = 0.698). CONCLUSIONS: Using an efficient child- and young person-centered approach, we developed 2 robust, age-appropriate versions of an instrument capturing VQoL that can be used cross-sectionally or sequentially across the age range of 8 to 17 years in research and clinical practice. This approach may be applicable in other rare childhood ophthalmic disorders.