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

Vision-related quality of life and mental health outcomes of children and young people with visual impairment and their carers [ISPOR Abstracts 07 May 2023 - 10 May 2023]

Objectives To investigate the associations between vision-related quality of life (VQoL) and mental health outcomes of children and young people with visual impairment (CYP-VI) and their parents/carers, as a first step towards development of family-centred intervention. Methods Cross-sectional study included 68 CYP-VI aged 8-18 years (visual acuity of logMAR 0.50 or worse, comprising moderate and severe visual impairment and blindness in ICD-11) and their carers. Families were recruited through two paediatric ophthalmology departments and relevant vision loss charities in the United Kingdom. Children’s outcomes included the overall scores on Vision-Related Quality of Life Questionnaire for Children and Young People (VQoL_CYP) as reported by the CYP-VI themselves, and Strengths and Difficulties Questionnaire (SDQ) as reported by their carers. Carers’ outcomes were overall scores on Satisfaction With Life Scale (SWLS), Patient Health Questionnaire (PHQ-9), General Anxiety Disorder Assessment (GAD-7), and Parental Stress Scale (PSS). Associations between children’s and carers’ outcomes were analysed using Spearman’s correlation coefficient. Results Lower VQoL was associated with more overall behavioural and emotional difficulties on SDQ (r=–0.485, p<0.001), lower carer’s satisfaction with life (r=0.395, p=0.002), worse carer’s depression (r=–0.390, p=0.002) and anxiety symptoms (r=–0.315, p=0.015). VQoL was not significantly associated with parental stress levels (r=–0.196, p=0.140). More overall child’s behavioural and emotional difficulties significantly correlated with higher parental stress (r=0.363, p=0.004),worse carer’s depression (r=0.436, p<0.001) and anxiety symptoms (r=0.422, p<0.001), but not with their satisfaction with life (r=–0.159, p=0.224). Visual acuity and gender were not related with any of the measured outcomes. Older age was associated with lower VQoL (r=–0.320, p=0.010). Conclusions Our findings show moderate correlations between mental health of CYP-VI and their carers, and indicate both should be considered when assessing VQoL outcomes of CYP-VI. Interventions targeting mental health of these families may promote better VQoL of CYP-VI

Attitudes, experiences, and preferences of ophthalmicprofessionals 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

Determinants of vision-related quality of life of children and young people with visual impairment

Purpose : To investigate the broader individual, family, and environmental factors associated with vision-related quality of life (VQoL) of children and young people with visual impairment (CYP-VI), in order to identify modifiable factors that could be targets for intervention. Methods : Cross-sectional study included 152 CYP-VI aged 7-18 years (visual acuity of logMAR 0.5 or worse, comprising moderate and severe visual impairment and blindness in ICD11) attending 22 Paediatric Ophthalmology Departments in the United Kingdom who participated in the final phase of our programme to develop two vision-specific patient-reported outcome measures (PROMs), Vision-Related Quality of Life (VQoL_CYP) and Functional Vision (FV_CYP) Questionnaires for Children and Young People. CYP-VI self-completed the two PROMs. Associations with family, sociodemographic characteristics (reported by their carers), and clinical factors (derived from their clinical records) were analysed using Spearman’s correlation coefficient, Kruskal-Wallis, Wilcoxon rank-sum tests and quantile regression models. Results : The median VQoL score was 55.58 (IQR=12.06, min=17.08, max=100.00), where higher scores indicate better VQoL. Better VQoL was associated with better FV overall (r=–0.516, p<0.001), parent-reported absence of additional chronic conditions (p=0.010, dCohen=0.457), attending mainstream (versus other) school (p=0.013, dCohen=0.439), higher socio-economic status (r=0.173, p=0.038) and higher parental education level (r=0.201, p=0.018). Other child (gender, ethnicity), clinical (severity of visual impairment, onset of vision loss, rate of vision deterioration), and family (parent’s age, ethnicity, employment status; number of siblings, birth order; siblings and parent’s vision and medical status; housing tenure, cars owned) were not associated with VQoL scores. Final quantile regression model included functional vision (FV) scores and presence of additional health condition. Conclusions : Both clinical characteristics as well as non-health-related factors should be considered when assessing vision-specific outcomes of CYP-VI. Our findings and further research on other potential determinants of VQoL are useful to develop recommendations and interventions to promote better VQoL and to support healthcare professionals to provide more personalised approach towards their patients’ care

Age-and stage-appropriate measurement of vision-related quality of life (VQoL) 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

Perceptions and experiences of health care service use during the pandemic for children with uveitis: UNICORNS-C19 study

Purpose : COVID-19 has disrupted provision of and access to healthcare. Children newly diagnosed with uveitis are particularly vulnerable to these disruptions. We aimed to describe the impact of the pandemic on the experiences and perceptions of care use for families of children newly diagnosed with uveitis. Methods : UNICORNS-C19 is a cross-sectional study embedded within the Uveitis in Childhood National Prospective Cohort Study (UNICORNS), which is recruiting UK children with non-infectious uveitis in order to understand the sociodemographic, clinical and biological determinants of disease and treatment outcomes and quality of life. We distributed (postally and electronically) a modified Health Foundation / Ipsos Mori survey, which comprises 13 questions (closed and open) around health and social care. Quantitative data were analysed using descriptive statistics, free text responses were analysed using qualitative thematic analysis. A framework was developed to index and chart data into themes through an iterative process. Results : Response rate to date is 42 of the 95 UNICORNS families approached (44%). Of those who participated, 61% expressed concern over the impact of the pandemic on their health, with a third (31%) expressing difficulties in accessing essential medication, and 56% finding it harder to get basic food stuffs. Despite this, the majority expressed a positive experience with NHS services, with 72% being comfortable using their specialist hospital during the pandemic. Key themes identified in analysis included positive experiences of safety procedures and the adoption of digital health tools across different levels of care (primary to quaternary); negative experiences of poor co-ordination of care, or of delivery of synchronous telemedicine care of rare disease from primary / secondary health teams, and negative perceptions around the use of immunosuppression during the pandemic. Conclusions : The UNICORNS study is well placed to provide useful data on patient experience for those starting a rare, chronic childhood disease care pathway during a global pandemic. Our findings suggest that primary and secondary care teams require additional support in delivering / co-ordinating care for those with rare disease. UNICORN C-19 findings will inform recommendations for future service planning. This abstract was presented at the 2022 ARVO Annual Meeting, held in Denver, CO, May 1-4, 2022, and virtually

Supporting positive patient experiences for rare disease care during disruptive times: findings from a multicentre study

Introduction: We describe the perceptions and experiences of health care services during the COVID-19 pandemic of those newly diagnosed with a rare, chronic inflammatory eye disorder. Methods: We undertook a cross-sectional study nested within a longitudinal multi-centre inception cohort study. Participants were families and young people (aged under 18 years) newly diagnosed with childhood uveitis. Using a validated tool, the Health Foundation COVID-19 Survey, we captured qualitative and quantitative data. Quantitative data were analysed using descriptive summary statistics. Qualitative, free text responses were analysed using content and thematic analysis. Results: Responses received from 60 families between 1st September 2020 and 30th March 2022 were analysed. Despite two in five reporting challenges in accessing medication, the majority felt comfortable accessing healthcare services (8%, 95% CI 3 - 18%, of participants expressed discomfort, versus 28%, 95% CI 26 – 28% of general population). Thematic analysis identified five themes: the value of protected spaces to safely access care; the positive role of digital health tools the experience of immature telemedicine; disintegration of care; and dealing with uncertainty. Discussion: Our findings suggest that families of children with a rare chronic condition had greater confidence in accessing healthcare during the pandemic than the general population. Nevertheless, to ensure more robust health services for such populations in future times of disruption, developments in telemedicine should be directly informed by the experiences of those with rare disease. The development of new healthcare processes which ensure the whole healthcare team has adequate information to support families should be prioritised

Supporting positive patient experiences for rare disease care during disruptive times: findings from a national study

Aim: We describe the perceptions and experiences of healthcare services during the pandemic of those newly diagnosed with a rare, chronic eye disorder. Methods: A cross-sectional mixed-methods study nested within a multi-center inception cohort study. Participants were UK families and adolescents newly affected by childhood uveitis. Using a validated tool (Health Foundation COVID-19 Survey), we captured quantitative (analyzed using descriptive statistics) and qualitative (analyzed using content and thematic analysis) data. Results: Responses received from 60 families (September 2020–March 2022), of whom 92% felt comfortable accessing healthcare services, despite 40% reporting challenges in accessing medication. Thematic analysis identified five themes: the value of protected spaces; the positive role of digital health tools, negative experience of immature telemedicine, disintegration of care; and dealing with uncertainty. Conclusion: Our findings will support ongoing developments in care with an aim to making services more robust to future periods of disruption

2. Pathways to detection of non-infectious childhood uveitis in the UK: findings from the UNICORN cohort study

Introduction Prompt detection of childhood uveitis is key to minimising negative impact. From an internationally unique inception cohort, we report pathways to disease detection. UNICORNS is a national childhood non-infectious uveitis study with longitudinal collection of a standardised clinical dataset and patient reported outcomes. Descriptive analysis of baseline characteristics are reported. Amongst 150 recruited children (51% female, 31% non-white ethnicity) age at detection ranged from 2–18yrs (median 10). In 69%, uveitis was diagnosed following onset of symptoms: time from first symptoms to uveitis detection ranged from 0-739days (median 7days), with longer time to detection for those presenting initially to their general practitioner. Non symptomatic children were detected through JIA/other disease surveillance (16%), routine optometry review (5%) or child visual health screening (1%). Commonest underlying diagnoses at uveitis detection were JIA (17%), TINU (9%, higher than pre-pandemic reported UK disease frequency) and sarcoid (1%). 60% had no known systemic disease at uveitis detection. At disease detection, in at least one eye: 34% had structural complications (associated with greater time to detection – 17 days versus 4 days for uncomplicated presentation). The larger relative proportions of children with non-JIA uveitis reported here increase the importance of improving awareness of childhood uveitis amongst the wider clinical communities. There is scope for improvement of pathways to detection. Forthcoming analysis on the full cohort (251 recruited to date across 33 hospitals and 4 nations) will provide nationally representative data on management and the determinants of visual and broader developmental/well-being outcomes