A blueprint for patient and public involvement in the development of a reporting guideline for systematic reviews of outcome measurement instruments: PRISMA-COSMIN for OMIs 2024.

BACKGROUND: In recent years, projects to develop reporting guidelines have attempted to integrate the perspectives of patients and public members. Best practices for patient and public involvement (PPI) in such projects have not yet been established. We recently developed an extension of PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses), to be used for systematic reviews of outcome measurement instruments (OMIs): PRISMA-COSMIN (COnsensus-based Standards for the selection of health Measurement INstruments) for OMIs 2024. Patients and public members formed a small but impactful stakeholder group. We critically evaluated the PPI component in this project and developed recommendations for conducting PPI when developing reporting guidelines. MAIN TEXT: A patient partner was an integral research team member at the project development and grant application stage. Once the project started, five patient and public contributors (PPCs) were recruited to participate in the Delphi study; three PPCs contributed to subsequent steps. We collected quantitative feedback through surveys; qualitative feedback was garnered through a focus group discussion after the Delphi study and through debrief meetings after subsequent project activities. Feedback was thematically combined with reflections from the research team, and was predominantly positive. The following themes emerged: importance of PPI partnership, number of PPCs involved, onboarding, design of Delphi surveys, flexibility in the process, complexity of PPI in methodological research, and power imbalances. Impacts of PPI on the content and presentation of the reporting guideline were evident, and reciprocal learning between PPCs and the research team occurred throughout the project. Lessons learned were translated into 17 recommendations for future projects. CONCLUSION: Integrating PPI in the development of PRISMA-COSMIN for OMIs 2024 was feasible and considered valuable by PPCs and the research team. Our approach can be applied by others wishing to integrate PPI in developing reporting guidelines.

Sleep Patterns in Children With Blindness: A Comparison With Normally Sighted Peers.

Purpose: Studies showing problematic sleep patterns in blind and visually impaired children are often based on (parent) self-report. The purpose was to compare sleep patterns of blind children to normally sighted peers using objective measures. Methods: In this cross-sectional study, 100 blind (best-corrected visual acuity <3/60) and 100 age- and gender-matched normally sighted children aged 7 to 17 years wore a digital activity monitoring device for 1 week. Sleep quantity (i.e., total sleep time and total time in bed) and sleep quality (number of awakenings, latency, efficiency, wake after sleep onset [WASO], and sleep fragmentation index) were measured. Adjusted linear regression analyses were used to model group differences in sleep parameters. Results: Data of 163 children were included. Blind children spent significantly less total time in bed in minutes (ß, -31; 95% confidence interval, -56 to -6) and had a lower total sleep time (-41; -66 to -17), smaller number of awakenings (-2.8; -4.5 to -1.0), a lower WASO (-10; -16 to -5), and a more efficient sleep pattern (1.5; 0.1 to 2.8) compared to normally sighted children. Conclusions: Although sleep quantity and recommended hours of sleep per night were lower among blind children than normally sighted children, their sleep quality was better. This contradicts findings of self-report studies and warrants further studies to measure sleep objectively. Further, the discrepancy between previous findings and our findings regarding sleep quality may be explained by the house rules of the boarding schools attended by blind children, which may facilitate improved sleep hygiene.

Youth and family involvement in the development of a plain language trial results communication tool: CommuniKIDS.

BACKGROUND: Pediatric trials are possible through voluntary participation of children, youth (age ≤ 18 years), and their families. Despite important arguments for trialists to provide trial progress or results, and evidence that participants desire it, this information remains rarely shared with youth and their families. Little guidance exists on how trialists can best communicate trial results back to participants and their families. Guided by Liabo et al.'s framework, we describe how we developed a pediatric-specific, "plain language summary" clinical trial results template called CommuniKIDS with an adult patient partner, family partner (parent), youth advisors, and parent advisors, taking into account their unique knowledge needs and preferences. MAIN TEXT: Patient and Public Involvement (PPI) was integrated in the development of the CommuniKIDS template. In collaboration with Clinical Trials Ontario, we used a generic trial results template as a starting point. The core project leadership team included a patient partner and a family partner from project inception to completion. Five youth (ages 13-18 years) and eight parent advisors were consulted at each point of the development process through three virtual workshops conducted separately; youth workshops were led by a youth facilitator. During these workshops, advisors agreed on the importance and value of sharing trial results, and expressed their preferences on content, format, and timing of sharing trial results. PPI-led improvements included the addition of three new sections to the CommuniKIDS template: "at a glance," "side effects," and "next steps." We reflect on our PPI strategy in the context of five "values" and six "practicalities" identified as good PPI principles, and summarize lessons learned when collaborating with youth and families from this project. CONCLUSION: Involvement of a patient partner, a family partner, youth advisors, and parent advisors in the development of CommuniKIDS was critical to create a clinical trial results template that is useful and relevant to its end-users. To our knowledge, CommuniKIDS is the first to meaningfully engage youth and parents as advisors and partners in developing a plain language summary results template for pediatric trial participants and their families. Our experience of co-developing CommuniKIDS demonstrates that meaningful PPI can be achieved in trial results communication and knowledge translation practices. This report provides resources for those seeking to involve youth and families in their initiatives and in meaningfully sharing trial results.

The voluntary participation of youth aged 18 and under in clinical trials makes it possible for researchers and healthcare providers to study medications and other treatments. However, most youth and their families who take part in clinical trials do not get any information on the trial's progress or results, leaving many to wonder if anything useful came from their participation. There is an ethical obligation to give this information back to youth and their families, who might take risks by participating in trials. The aim of the CommuniKIDS project was to develop a "plain language summary" results template to share trial results back to youth and their families. Working with a patient partner, a family partner, five youth advisors (ages 13­18), and eight parent advisors, we set out to understand what youth and parents would like to see in a plain language summary of clinical trial results. The needs and preferences discussed with the advisors were included to create a child/youth health-specific template. The CommuniKIDS project is the first to involve youth and parents as advisors in developing a plain language summary results template for child/youth health trials. Here, we describe how we involved youth and parents in the development of CommuniKIDS, how the template was customized to be youth and family-friendly and reflect on lessons learned.

Measurement Properties of Patient Reported Outcome Scales: A Systematic Review.

CONTEXT: Recently a standard set for overall pediatric health outcomes in routine care was developed, which includes patient (or proxy) reported outcome measures (PROMs) for global health, cognitive functioning, and self-efficacy. OBJECTIVES: To determine whether the following PROMs have sufficient measurement properties to be used in pediatric routine care: PROMIS Pediatric and Parent Proxy Scale - Global Health 7+2, PROMIS Parent Proxy Short Form - Cognitive Function 7a, and NIH Toolbox Self-Efficacy CAT Ages 13 to 17. DATA SOURCES: Embase, Psych INFO, and Web of Science were searched from year of inception of each PROM to May 25, 2020; Medline to October 24, 2022. STUDY SELECTION: English, full-text peer-reviewed articles that evaluated measurement properties of included PROMs were eligible. DATA EXTRACTION: The COSMIN guideline for systematic reviews was used to appraise eligible studies and synthesize the overall evidence. RESULTS: Screening >4000 titles yielded 4 to 6 eligible empirical studies for each PROM. The PROMIS instruments had sufficient content validity with low-quality evidence and at least low-quality evidence for sufficient structural validity and internal consistency. The NIH Toolbox lacked essential evidence for content validity. LIMITATIONS: Assessments of measurement properties were based on information reported in the included studies; underreporting might have led to less favorable ratings. CONCLUSIONS: The PROMIS instruments assessed in this review measure their intended construct for their targeted age group; clinicians can use these PROMs in pediatric routine care. Additional studies evaluating measurement properties, including content validity, are needed for the NIH Toolbox before it should be recommended for use in clinical practice.

The evaluation of an online nurse-assisted eye-screening tool in older adults receiving home healthcare.

PURPOSE: To investigate the agreement between an online nurse-assisted eye-screening tool and reference tests in older adults receiving home healthcare and to collect user experiences. METHODS: Older adults (65+) receiving home healthcare were included. Home healthcare nurses assisted in administering the eye-screening tool at participants' homes. Approximately 2 weeks later, a researcher administered reference tests at participants' homes. Experiences from participants and home healthcare nurses were collected. Agreement in outcomes (distance and near visual acuity, with the latter being measured using two different optotypes, and macular problems) between the eye-screening tool and reference clinical testing was compared. A difference of less than ±0.15 logMAR was considered acceptable. RESULTS: A total of 40 participants were included. Here, we describe the results for the right eye; results for the left eye were similar. The mean difference between the eye-screening tool and reference tests for distance visual acuity was 0.02 logMAR. The mean difference between the eye-screening tool and reference tests using two different optotypes for near visual acuity was 0.06 and 0.03 logMAR, respectively. The majority of the individual data points were within the ±0.15 logMAR threshold (75%, 51% and 58%, respectively). The agreement between tests for macular problems was 75%. Participants and home healthcare nurses were generally satisfied with the eye-screening tool, although remarks for further improvements were made. CONCLUSIONS: The eye-screening tool is promising for nurse-assisted eye screening in older adults receiving home healthcare, with the mostly satisfactory agreement. After implementing the eye-screening tool in practice, cost-effectiveness needs to be investigated.

Psychometric properties and reference values of the Patient-Reported Outcomes Measurement Information System (PROMIS) sleep item banks in the Dutch general population.

Psychometric properties of the v1.0 Patient-Reported Outcomes Measurement Information System (PROMIS®) sleep disturbance (27 items) and sleep-related impairment (SRI; 16 items) item banks, short forms derived from the item bank, and simulated computerised adaptive test (CAT), were assessed in a representative sample of 1,006 adults from the Dutch general population. For sleep disturbance all items fitted the item response theory model. Four items showed differential item functioning (i.e., lack of measurement invariance) for age and two for language but the impact on scores (expressed as T-scores) was small. Reliable scores (r > 0.90) were found for 92.2%-96.3% of respondents with the full bank, short forms with six and eight items, and CAT, but for only 25.6% with the four-item short form. For SRI two items did not fit the item response theory model. Four items showed differential item functioning for language but the impact on T-scores was small. Reliable scores were found for 82.1% with the full bank, for 47.8%-69.5% with short forms and CAT. T-scores of 49.7 and 49.3 represent the average score of the Dutch general population for sleep disturbance and SRI, respectively. In conclusion, sufficient structural validity, reliability, and cross-cultural validity was found for the full banks but short forms of four items are not reliable enough for clinical practice. For SRI we recommend the full item bank if this is the primary outcome.

Towards standardization of measuring anxiety and depression: Differential item functioning for language and Dutch reference values of PROMIS item banks.

INTRODUCTION: The outcomes anxiety and depression are measured frequently by healthcare providers to assess the impact of a disease, but with numerous instruments. PROMIS item banks provide an opportunity for standardized measurement. Cross-cultural validity of measures and the availability of reference values are prerequisites for standardized measurement. METHODS: PROMIS Anxiety and Depression item banks were completed by 1002 representative Dutch persons. To evaluate cross-cultural validity, data from US participants in PROMIS wave 1 were used and differential item functioning (DIF) was investigated, using an iterative hybrid of logistic regression and item response theory. McFadden's pseudo R2-change of 2% was the critical threshold. The impact of any DIF on full item banks and short forms was investigated. To obtain Dutch reference values, T-scores for anxiety and depression were calculated for the complete Dutch sample, and age-group and gender subpopulations. Thresholds corresponding to normal limits, mild, moderate and severe symptoms were computed. RESULTS: In both item banks, two items had DIF but with minimal impact on population level T-scores for full item banks and short forms. The Dutch general population had a T-score of 49.9 for anxiety and 49.6 for depression, similar to the T-scores of 50.0 of the US general population. T-scores for age-group and gender subpopulations were also similar to T-scores of the US general population. Thresholds for mild, moderate and severe anxiety and depression were set to 55, 60 and 70, identical to US thresholds. CONCLUSIONS: The limited number of items with DIF and its minimal impact, enables the use of standard (US) item parameters and comparisons of scores between Dutch and US populations. The Dutch reference values provide an important tool for healthcare professionals and researchers to evaluate and interpret symptoms of anxiety and depression, stimulating the uptake of PROMIS measures, and contributing to standardized outcome measurement.

Content Validity of Patient-Reported Outcome Measures Developed for Assessing Health-Related Quality of Life in People with Type 2 Diabetes Mellitus: a Systematic Review.

PURPOSE OF REVIEW: We aimed to systematically evaluate the content validity of patient-reported outcome measures (PROMs) specifically developed to measure (aspects of) health-related quality of life (HRQOL) in people with type 2 diabetes. A systematic review was performed in PubMed and Embase of PROMs measuring perceived symptoms, physical function, mental function, social function/participation, and general health perceptions, and that were validated to at least some extent. Content validity (relevance, comprehensiveness, and comprehensibility) was evaluated using COSMIN methodology. RECENT FINDINGS: We identified 54 (different versions of) PROMs, containing 150 subscales. We found evidence for sufficient content validity for only 41/150 (27%) (subscales of) PROMs. The quality of evidence was generally very low. We found 66 out of 150 (44%) (subscales of) PROMs with evidence for either insufficient relevance, insufficient comprehensiveness, or insufficient comprehensibility. For measuring diabetes-specific symptoms, physical function, mental function, social function/participation, and general health perceptions, we identified one to 11 (subscales of) PROMs with sufficient content validity, although quality of the evidence was generally low. For measuring depressive symptoms, no PROM with sufficient content validity was identified. For each aspect of HRQL, we found at least one PROM with sufficient content validity, except for depressive symptoms. The quality of the evidence was mostly very low.

Systematic review on the measurement properties of diabetes-specific patient-reported outcome measures (PROMs) for measuring physical functioning in people with type 2 diabetes.

We aimed to systematically assess the measurement properties of diabetes-specific patient-reported outcome measures (PROMs) for measuring physical functioning, one of the core outcomes, in adults with type 2 diabetes.We performed a systematic literature search for PROMs or subscales measuring physical function that were validated to at least some extent in EMBASE and MEDLINE. Measurement properties were evaluated according to the COSMIN guideline for systematic reviews of PROMs.In total 21 articles were included, describing 12 versions of 7 unique diabetes-specific PROMs or subscales measuring physical functioning. In general, there were few high-quality studies on measurement properties of PROMs measuring physical functioning in adults with type 2 diabetes. The Dependence/Daily Life subscale of the Diabetic Foot Ulcer Scale-Short Form (DFS-SF) and the Impact of Weight on Activities of Daily Living Questionnaire (IWADL) were most extensively evaluated. Both had sufficient ratings for aspects of content validity, although with mostly very low-quality evidence. Sufficient ratings for structural validity, internal consistency, and reliability were also found for both instruments, but responsiveness was rated inconsistent for both instruments. The other PROMs or subscales often had insufficient aspects of content validity, or their unidimensionality could not be confirmed.This systematic review showed that the Dependence/Daily Life subscale of the DFS-SF and the IWADL could be used to measure physical functioning in people with type 2 diabetes in research or clinical practice, while keeping the limitations of these instruments in mind. The measurement properties that have not been evaluated extensively for these PROMs should be evaluated in future studies.The study protocol was registered in the PROSPERO database, number CRD42021234890.

Study protocol for developing, piloting and disseminating the PRISMA-COSMIN guideline: a new reporting guideline for systematic reviews of outcome measurement instruments.

BACKGROUND: Systematic reviews of outcome measurement instruments are important tools in the evidence-based selection of these instruments. COSMIN (COnsensus-based Standards for the selection of health Measurement INstruments) has developed a comprehensive and widespread guideline to conduct systematic reviews of outcome measurement instruments, but key information is often missing in published reviews. This hinders the appraisal of the quality of outcome measurement instruments, impacts the decisions of knowledge users regarding their appropriateness, and compromises reproducibility and interpretability of the reviews' findings. To facilitate sufficient, transparent, and consistent reporting of systematic reviews of outcome measurement instruments, an extension of the PRISMA (Preferred Reporting of Items for Systematic reviews and Meta-Analyses) 2020 guideline will be developed: the PRISMA-COSMIN guideline. METHODS: The PRISMA-COSMIN guideline will be developed in accordance with recommendations for reporting guideline development from the EQUATOR (Enhancing the QUAlity and Transparency Of health Research) Network. First, a candidate reporting item list will be created through an environmental literature scan and expert consultations. Second, an international Delphi study will be conducted with systematic review authors, biostatisticians, epidemiologists, psychometricians/clinimetricians, reporting guideline developers, journal editors as well as patients, caregivers, and members of the public. Delphi panelists will rate candidate items for inclusion on a 5-point scale, suggest additional candidate items, and give feedback on item wording and comprehensibility. Third, the draft PRISMA-COSMIN guideline and user manual will be iteratively piloted by applying it to systematic reviews in several disease areas to assess its relevance, comprehensiveness, and comprehensibility, along with usability and user satisfaction. Fourth, a consensus meeting will be held to finalize the PRISMA-COSMIN guideline through roundtable discussions and voting. Last, a user manual will be developed and the final PRISMA-COSMIN guideline will be disseminated through publications, conferences, newsletters, and relevant websites. Additionally, relevant journals and organizations will be invited to endorse and implement PRISMA-COSMIN. Throughout the project, evaluations will take place to identify barriers and facilitators of involving patient/public partners and employing a virtual process. DISCUSSION: The PRISMA-COSMIN guideline will ensure that the reports of systematic reviews of outcome measurement instruments are complete and informative, enhancing their reproducibility, ease of use, and uptake.

Measurement properties of the Dutch PROMIS-29 v2.1 profile in people with and without chronic conditions.

PURPOSE: To investigate the structural validity, internal consistency, measurement invariance, and construct validity of the Dutch PROMIS-29 v2.1 profile, including seven physical (e.g., pain, physical function), mental (e.g., depression, anxiety), and social (e.g., role functioning) domains of health, in a Dutch general population sample including subsamples with and without chronic diseases. METHODS: The PROMIS-29 was completed by 63,602 participants from the Lifelines cohort study. Structural validity of the PROMIS-29, including unidimensionality of each domain and the physical and mental health summary scores, was evaluated using factor analyses (criteria: CFI ≥ 0.95, TLI ≥ 0.95, RMSEA ≤ 0.06, SRMR ≤ 0.08). Internal consistency, measurement invariance (no differential item functioning (DIF) for age, gender, administration mode, educational level, ethnicity, chronic diseases), and construct validity (hypotheses on known-groups validity and correlations between domains) were assessed per domain. RESULTS: The factor structure of the seven domains was supported (CFI = 0.994, TLI = 0.993, RMSEA = 0.046, SRMR = 0.031) as was unidimensionality of each domain, both in the entire sample and the subsamples. Model fit of the physical and mental health summary scores reached the criteria, and scoring coefficients were obtained. Cronbach's alpha for the seven PROMIS-29 domains ranged from 0.75 to 0.96 in the complete sample. No DIF was detected. Of the predefined hypotheses, 78% could be confirmed. CONCLUSION: Sufficient structural validity, internal consistency and measurement invariance were found, both in the entire sample and in subsamples with and without chronic diseases. Requirements for sufficient evidence for construct validity were (almost) met for most subscales. Future studies should investigate test-retest reliability, measurement error, and responsiveness of the PROMIS-29.

Perspectives on Implementing the Patient Health Questionnaire-4 in Low-Vision Service Organizations to Screen for Depression and Anxiety.

Purpose: To describe the process of implementing a screening questionnaire for depression and anxiety, the Patient Health Questionnaire-4 (PHQ-4), in low-vision service (LVS) organizations. Methods: This study consisted of three parts: (1) a usability study combined with semistructured interviews, in which clients (n = 10) of LVS organizations expressed their preference for using the PHQ-4; (2) a feasibility study, in which the PHQ-4 was implemented on a small scale and its use was evaluated, involving health care providers (n = 6) and clients (n = 9); and (3) semistructured interviews to identify barriers and facilitators for implementing the PHQ-4 according to health care providers (n = 6) and managers (n = 4) of LVS organizations. Results were integrated into themes and linked to constructs of the Consolidated Framework for Implementation Research (CFIR). Results: Six themes were derived from the substudies: (1) quality of the intervention, (2) applicability for clients of LVS organizations, (3) attitude and needs of clients, (4) attitude of health care providers, (5) support within LVS organizations, and (6) embedment in current practice. Results could be linked to 12 CFIR constructs. The constructs "relative advantage," "patient needs and resources," and "available resources" emerged most prominently in our themes as either barrier or facilitator. Conclusions: The PHQ-4 seems an appropriate screening instrument for use in LVS organizations because of its quality and adaptable use. It might provide opportunities to timely detect depression and anxiety, but challenges in implementing the PHQ-4 should be considered. Translational Relevance: Barriers and facilitators for implementing the PHQ-4 may also apply to implementing other questionnaires in LVS organizations.

Quality of Life and Participation of Children With Visual Impairment: Comparison With Population Reference Scores.

Purpose: The purpose of this study was to investigate quality of life and participation in children aged 3 to 17 years with visual impairment (VI) compared to reference groups and between subgroups with increasing severity levels of VI. Methods: Parents of children aged 3 to 17 years (n = 500) and children aged 13 to 17 years (n = 75) completed the Child and Adolescent Scale of Participation (CASP). Children aged 7 to 17 years (n = 263) and their parents (n = 255) completed the KIDSCREEN-27 questionnaire to assess quality of life. Scores were compared to age and/or gender-appropriate population-based samples. For the CASP, a comparison was also made with children with chronic conditions or disabilities. The association between severity of VI and quality of life or participation was analyzed with linear regression models. Results: Children reported significantly worse on Physical Wellbeing and Social Support & Peers, but better on the School Environment KIDSCREEN-27 subscales compared to reference groups. Parents additionally reported worse on Autonomy & Parent Relation. Children's participation was significantly worse compared to a population-based sample, but significantly better compared to children with chronic conditions and disabilities. Having moderate or severe VI/blindness was significantly associated with worse participation, as reported by parents relative to those with no VI. Conclusions: Quality of life of children with VI is affected especially regarding Physical Wellbeing and Social Support & Peers compared to a reference population, and their participation is considerably worse. Participation was more affected in children with more severe VI. These results contribute to the understanding of the impact of VI. Interventions targeting physical health, social skills, and participation are warranted.

Dutch reference values for the Patient-Reported Outcomes Measurement Information System Scale v1.2 - Global Health (PROMIS-GH).

BACKGROUND: To add context to the impact of medical conditions, it is important to interpret and compare health outcomes across studies and populations. We aimed to determine Dutch reference values for the Patient-Reported Outcomes Measurement Information System Scale v1.2 - Global Health (PROMIS-GH). METHODS: The PROMIS-GH, also referred to as PROMIS-10, was completed by 4370 Dutch persons, representative for the 2016 Dutch population. T-scores for the mental health (GMH) and physical health (GPH) subscales, and their shorter two-item subscales, were calculated for the entire population, age groups and gender. T-scores for GMH and GPH were compared to the US reference population, representative for the 2000 US general population. Interpretability thresholds for poor, fair, good, very good and excellent GPH and GMH were calculated based on T-scores of participants, which were categorized into five groups based on their response to item Global01. For each group the mean GPH and GMH T-score was calculated and the midpoint between two adjacent means was identified, resulting in thresholds. Thresholds based on the Dutch data were compared to US thresholds. RESULTS: The Dutch population had a GMH T-score of 44.7 and a GPH T-score of 45.2, both substantially worse than the US reference population T-score of 50. Lower T-scores were also found for age-range and gender subpopulations. Dutch GMH and GPH interpretability thresholds were mostly not substantially different compared to the US thresholds, although the Dutch threshold between fair and poor mental health was considerably higher (29 vs. 38). CONCLUSIONS: This study reports reference values for the PROMIS-GH scale for the Dutch general population, including age-range and gender subpopulations. These reference values provide an important tool for healthcare professionals and researchers to better evaluate and interpret patient-reported mental health and physical health. Scores are notably worse than the US reference values. The exact reason for this remains subject for further research, although possibilities for the differences are discussed, including the presence of differential item functioning and the representativeness and recentness of the data.

Effect of Corneal Transplantation on Patient-Reported Outcomes and Potential Predictors: A Systematic Review.

PURPOSE: To investigate the effect and potential predictors of corneal transplantation on patient-reported outcomes such as quality of life, visual functioning, and mental health by systematically reviewing the literature. METHODS: Studies with 1 preoperative and at least 1 postoperative measurement were searched for in relevant literature databases. Methodological quality was assessed using the Quality Assessment Tool for Quantitative Studies, and effect sizes were calculated. RESULTS: Of 1445 unique publications, 14 studies, including 15 study designs, were described in 16 publications. Four randomized controlled trials, 1 controlled clinical trial, 1 cohort study, and 1 before-after study (BA) were of good quality; 6 BAs were of moderate quality; and 2 BAs were of weak quality. Patients generally improved 12 months after transplantation on health-related quality of life (effect size between 0.08 and -3.06), vision-related quality of life (-0.67 and -6.65), visual functioning (-0.55 and -0.63), and subjective visual symptoms (-0.31 and -0.86). Patient satisfaction was high (-0.95). Patients improved on depression (-0.31) but remained stable on anxiety (-0.05) 4 months after transplantation. Predictors of positive outcomes were lower preoperative visual acuity and visual functioning, better postoperative visual factors, younger age, and male sex. CONCLUSIONS: Corneal transplantation showed overall beneficial effects on patient-reported outcomes. Knowledge of these effects and predictors might result in better treatment, more patient-centered care, and more realistic expectations on the part of patients and ophthalmologists. Future studies should focus on not only health- and vision-related quality of life but also mental health and labor participation using longitudinal study designs.

Network Analysis of the Participation and Activity Inventory for Children and Youth (PAI-CY) 7-12 Years with Visual Impairment.

Purpose: Children with visual impairment often experience more difficulties regarding participation compared to sighted peers. The Participation and Activity Inventory for Children and Youth (PAI-CY) has recently been developed to assess their participation needs. A novel application in the field of questionnaires is the use of network analysis to explore interrelations between items in order to capture their complex interactions as a reflection of the overall construct of measurement. This study aimed to apply network modeling for the PAI-CY 7-12 from the perspectives of children and their parents. Methods: Children and their parents (n = 195) completed the 55-item PAI-CY via face-to-face interviews and a web-based survey, respectively. Internal consistency, test-retest reliability, and concordance between children and parents were investigated. Two networks were created, along with visualizations of shared and differential connections between children and parents. Results: Eight items were deleted. Network structures were dissimilar; for children, connections evolved around social contacts and school items, whereas for parents, mobility, leisure time, acceptance, self-reliance, and communication items prevailed. In the children's network, playing imaginary games, inviting a friend to play at home, and estimating the distance from others were most connected to other items. Conclusions: This study uniquely identifies connections between items of the PAI-CY 7-12, highlighting the different perspectives parents and children have on what defines participation, possibly implying that they perceive the relevance of various rehabilitation programs differently. Translational Relevance: Rehabilitation programs aimed at improving the most connected items might positively affect other items in the network, possibly improving children's participation.

First stage psychometric testing of a new instrument for adolescents with visual impairment: the Participation and Activity Inventory for Children and Youth (PAI-CY) 13-17 years.

BACKGROUND: To assess participation of children with visual impairment, the Participation and Activity Inventory for Children and Youth (PAI-CY) was recently developed. This study assessed some initial psychometric properties of the PAI-CY 13-17 years version, and investigated its feasibility. METHODS: Adolescents with visual impairment and their parents (n = 72 dyads) completed the self-report and proxy-report version of the 58-item PAI-CY, an evaluation form and several questionnaires measuring related constructs. Item deletion was informed by item responses, inter-item correlations, test-retest reliability, adolescent-parent agreement and participants' feedback. Known-group validity and concurrent validity with related questionnaires were investigated for the final item-set. RESULTS: Twelve items had > 20% missing values, whereas 39 items showed floor effects. Eight item pairs showed high inter-item correlations. Test-retest reliability was acceptable for most items (kappa ≥0.4). Evaluation forms showed that over 90% of respondents was neutral to very positive regarding several feasibility aspects such as administration time and comprehensiveness. Adolescent-parent agreement was mostly low. These results informed the deletion of three items. Known-group validity seemed adequate since PAI-CY scores were significantly worse for participants with comorbidity compared to those without. A trend towards worse scores for participants with more severe visual impairment was also observed. Correlations between the PAI-CY and related questionnaires confirmed concurrent validity. CONCLUSIONS: Initial psychometric properties of the PAI-CY 13-17 were acceptable, although more work is needed to assess other psychometric properties, such as the underlying construct. Following implementation in low vision care to assess participation needs, enabling larger samples, acceptability of the PAI-CY 13-17 to end-users should be carefully monitored, especially if alterations are made based on the current study.

Psychometric evaluation of the Participation and Activity Inventory for Children and Youth (PAI-CY) 0-2 years with visual impairment.

PURPOSE: To identify and monitor the developmental and participation needs of visually impaired (VI) children, the Participation and Activity Inventory for Children and Youth (PAI-CY) has recently been developed involving end-users as stakeholders. The aim was to investigate psychometric properties of the PAI-CY for children between 0 and 2 years. METHODS: Responses from 115 parents were included in item analyses, after which a combination of classical test theory and item response theory (IRT) was used. Internal consistency, known-group validity, and test-retest reliability at item and scale level were investigated. RESULTS: After deleting four items, the PAI-CY met IRT assumptions, i.e., unidimensionality, local independence, and monotonicity, and satisfactory model fit was obtained. Participants with more severe VI and comorbidity scored significantly worse than those with less severe VI and without comorbidity, supporting known-group validity. Satisfactory internal consistency and test-retest reliability were obtained (Cronbach's alpha 0.95, kappa 0.60-0.91, ICC 0.920). CONCLUSIONS: The PAI-CY 0-2 years has acceptable psychometric properties and can be used to systematically assess and monitor developmental and participation needs of very young children with VI from parents' perspectives in low vision practice and research. Confirmation of psychometric properties is necessary, possibly facilitating further item reduction, increased precision, and improved user-friendliness.

Cross-cultural validation of the Functional Vision Questionnaire for Children and Young People (FVQ_CYP) with visual impairment in the Dutch population: challenges and opportunities.

BACKGROUND: To assess cross-cultural validity between Dutch and English versions of the FVQ_CYP, a patient-reported outcome measure developed in the United Kingdom (UK) for children and adolescents with (severe) visual impairment or blindness (VI for brevity) to measure functional vision. METHODS: The 36-item FVQ_CYP was translated and adapted into Dutch using standard guidelines. The questionnaire was administered to Dutch children and adolescents aged 7-17 years (N = 253) with impaired vision (no restrictions regarding acuity). Data were compared to existing UK data of children and adolescents aged 10-15 years (N = 91) with VI (acuity LogMar worse than 0.48). As with the original UK FVQ_CYP validation, a rating scale model (RSM) was applied to the Dutch data. RESULTS: Minor adaptations were needed in translation-rounds. Significant differences in item responses were found between the Dutch and UK data. Item response theory assumptions were met, but fit to the RSM was unsatisfactory. Therefore, psychometric properties of the Dutch FVQ_CYP were analysed irrespective of the original model and criteria used. A graded response model led to the removal of 12 items due to missing data, low information, overlapping content and limited relevance to Dutch children. Fit indices for the remaining 24 items were adequate. CONCLUSIONS: Differences in population characteristics, distribution of responses, non-invariance at the model level and small sample sizes challenged the cross-cultural validation process. However, the Dutch adapted FVQ_CYP showed high measurement precision and broad coverage of items measuring children's functional vision. The underlying reasons for differences between countries in instrument performance are discussed with implications for future studies.

The association between visual impairment and fatigue: a systematic review and meta-analysis of observational studies.

PURPOSE: The aim was to compare fatigue levels between patients with visual impairment and controls with normal sight and to examine the association between fatigue and vision loss severity. METHODS: A systematic literature search was performed using databases of PubMed, Embase, PsycINFO and Cochrane to identify observational studies with outcomes related to fatigue (e.g. vitality subscale of the Short-Form 36, Fatigue Assessment Scale). A meta-analysis was performed using standardised mean differences (SMDs) and odds ratios (OR) to quantitatively summarise the association between visual impairment and fatigue. Sources of heterogeneity were explored by subgroup and sensitivity analyses. Study quality was assessed with the Newcastle-Ottawa scale. RESULTS: After reviewing 4477 studies, 22 studies with a total of 40 004 participants were included, of which 18 contributed to meta-analysis. Among these, eight were assessed as moderate quality studies and 10 as high quality studies. Pooled analysis involving 2500 patients and 8395 controls showed higher fatigue severity levels (S.M.D. = -0.36, 95% CI -0.50 to -0.22, 14 studies) among visually impaired patients compared to normally sighted controls. This effect size was small and persisted in sensitivity analyses that involved study quality, fatigue assessment tools and visual acuity data. Furthermore, pooled analysis of four studies including 2615 patients and 5438 controls showed a significant association between visual impairment and fatigue (OR = 2.61, 95% CI 1.69 to 4.04). Secondary meta-analysis of four studies showed no significant difference in fatigue severity (S.M.D. = 0.01, 95% CI -0.37 to 0.39) between patients with moderate visual impairment and patients with severe visual impairment or blindness. CONCLUSIONS: Current moderate to high quality evidence suggest that patients with visual impairment experience more severe fatigue symptoms than persons with normal sight. However, a limited number of available studies indicates that fatigue is not associated with severity of vision loss. Future studies are required to determine which factors and underlying mechanisms may explain the association between visual impairment and fatigue. Discussing fatigue at an early stage and developing intervention options for vision-related fatigue should be considered within the field of low vision rehabilitation.