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.

A Pilot Evaluation of an Educational Video to Support Consent to a Pediatric Malignancy Biobank.

Introduction: The collection of biological specimens is necessary to support basic and translational research. However, the complexity of biobanking introduces numerous ethical issues, particularly regarding informed consent. Objective: To evaluate the acceptability and perceived benefits of an educational video facilitating the consent process for the Children's Cancer Centre Biobank. Methods: We invited individuals who had previously consented to be (or their child to be) part of the Biobank, and health professionals who were involved in obtaining consent. Participants watched the video and completed a purpose-designed online survey. Results: A total of 16 health professionals (invited = 30) and 15 patients/caregivers (invited = 127) participated. Most patients/caregivers felt informed about the Biobank at consent, however, noted how overwhelmed they were at the time and that they did not engage with the written information. Overall, both patients/caregivers and health professionals rated the video favorably regarding the information provided and format. Participants valued that it was simple and clear, with several health professionals noting the need for linguistic translations to better support the families they work with. Most patients/caregivers agreed that the video provided enough information to begin considering participation. This aligned with the health professionals' feedback that the video was most effective when used as a conversation starter to help formalize the written consent. Conclusion: Our findings suggest that our video is an acceptable and beneficial tool to assist in the Biobank consenting process, from both the perspective of decision-makers and health professionals obtaining consent. It appears particularly valuable as a precursor to an interactive, formal consent discussion. Further work is required to determine whether our video has a significant impact on outcomes such as decision-making satisfaction and knowledge, and to determine the value to adolescents.

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.

Systems-Level Change to Alleviate Barriers to Cancer Clinical Trial Access for Adolescents and Young Adults in Australia.

Purpose: International data demonstrate association between clinical trial participation and reduced cancer mortality. Adolescents and young adults (AYA) have low clinical trial enrollment rates. We established a program to understand local barriers and develop targeted solutions that lead to greater AYA clinical trial participation. Methods: A steering committee (SC) with expertise in adult and pediatric oncology, research ethics, and consumer representation was formed. The SC mapped barriers related to AYA trial access and established working groups (WGs) around three themes. Results: The Regulatory Awareness WG identified a lack of understanding of processes that support protocol approval for clinical trials across the AYA age range. A guideline to raise awareness was developed. The Access WG identified challenges for young adults (18-25 years) to access a pediatric hospital to enroll in a pediatric trial. A procedure was developed to streamline applications for access. The first six applications using this procedure have been successful. The Availability WG identified lack of pediatric-adult oncology reciprocal relationships as a barrier to awareness of open trials, and future collaboration. An AYA Craft Group Framework was established to grow relationships within tumor streams across institutions; two craft groups are now operating locally. An additional achievement was a successful request to the Therapeutic Goods Administration for Australian adoption of the Food and Drug Administration Guidance on Considerations for the Inclusion of Adolescent Patients in Adult Oncology Clinical Trials. Conclusion: This multipronged approach to improving AYA clinical trial access has relevance for other health environments. Our knowledge products are available as an online toolkit.

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.

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.

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.

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.

High-priority and low-priority drug-drug interactions in different international electronic health record systems: A comparative study.

OBJECTIVES: To investigate whether alert warnings for high-priority and low-priority drug-drug interactions (DDIs) were present in five international electronic health record (EHR) systems, to compare and contrast the severity level assigned to them, and to establish the proportion of alerts that were overridden. METHODS: We conducted a comparative, retrospective, multinational study using a convenience sample of 5 EHRs from the U.S., U.K., Republic of Korea and Belgium. RESULTS: Of the 15 previously defined, high-priority, class-based DDIs, alert warnings were found to exist for 11 in both the Korean and UK systems, 9 in the Belgian system, and all 15 in the two US systems. The specific combinations that were included in these class-based DDIs varied considerably in number, type and level of severity amongst systems. Alerts were only active for 8.4% (52/619) and 52.4% (111/212) of the specific drug-drug combinations contained in the Belgian and UK systems, respectively. Hard stops (not possible to override) existed in the US and UK systems only. The override rates for high-priority alerts requiring provider action ranged from 56.7% to 83.3%. Of the 33 previously defined low-priority DDIs, active alerts existed only in the US systems, for three class-based DDIs. The majority were non-interruptive. CONCLUSIONS: Alert warnings existed for most of the high-priority DDIs in the different EHRs but overriding them was easy in most of the systems. In addition to validating the high- and low-priority DDIs, this study reported a lack of standardization in DDI levels across different international knowledge bases.

Analysis of variations in the display of drug names in computerized prescriber-order-entry systems.

PURPOSE: The variations in how drug names are displayed in computerized prescriber-order-entry (CPOE) systems were analyzed to determine their contribution to potential medication errors. METHODS: A diverse set of 10 inpatient and outpatient CPOE system vendors and self-developed CPOE systems in 6 U.S. healthcare institutions was evaluated. A team of pharmacists, physicians, patient-safety experts, and informatics experts created a CPOE assessment tool to standardize the assessment of CPOE features across the systems studied. Hypothetical scenarios were conducted with test patients to study the medication ordering workflow and ways in which medications were displayed in each system. Brand versus generic drug name ordering was studied at 1 large outpatient system to understand why prescribers ordered both brand and generic forms of the same drug. RESULTS: Widespread variations in the display of drug names were observed both within and across the 6 study sites and 10 systems, including the inconsistent display of brand and generic names. Some displayed drugs differently even on the same screen. Combination products were often displayed inconsistently, and some systems required prescribers to know the first drug listed in the combination in order for the correct product to appear in a search. It also appeared that prescribers may have prescribed both brand and generic forms of the same medication, creating the potential for drug duplication errors. CONCLUSION: A review of 10 CPOE systems revealed that medication names were displayed inconsistently, which can result in confusion or errors in reviewing, selecting, and ordering medications.

Efficiency and usability of a near field communication-enabled tablet for medication administration.

BACKGROUND: Barcode-based technology coupled with the electronic medication administration record (e-MAR) reduces medication errors and potential adverse drug events (ADEs). However, many current barcode-enabled medication administration (BCMA) systems are difficult to maneuver and often require multiple barcode scans. We developed a prototype, next generation near field communication-enabled medication administration (NFCMA) system using a tablet. OBJECTIVE: We compared the efficiency and usability of the prototype NFCMA system with the traditional BCMA system. METHODS: We used a mixed-methods design using a randomized observational cross-over study, a survey, and one-on-one interviews to compare the prototype NFCMA system with a traditional BCMA system. The study took place at an academic medical simulation center. Twenty nurses with BCMA experience participated in two simulated patient medication administration scenarios: one using the BCMA system, and the other using the prototype NFCMA system. We collected overall scenario completion time and number of medication scanning attempts per scenario, and compared those using paired t tests. We also collected participant feedback on the prototype NFCMA system using the modified International Business Machines (IBM) Post-Study System Usability Questionnaire (PSSUQ) and a semistructured interview. We performed descriptive statistics on participant characteristics and responses to the IBM PSSUQ. Interview data was analyzed using content analysis with a qualitative description approach to review and categorize feedback from participants. RESULTS: Mean total time to complete the scenarios using the NFCMA and the BCMA systems was 202 seconds and 182 seconds, respectively (P=.09). Mean scan attempts with the NFCMA was 7.6 attempts compared with 6.5 attempts with the BCMA system (P=.12). In the usability survey, 95% (19/20) of participants agreed that the prototype NFCMA system was easy to use and easy to learn, with a pleasant interface. Participants expressed interest in using the NFCMA tablet in the hospital; suggestions focused on implementation issues, such as storage of the mobile devices and infection control methods. CONCLUSIONS: The NFCMA system had similar efficiency to the BCMA system in a simulated scenario. The prototype NFCMA system was well received by nurses and offers promise to improve nurse medication administration efficiency.

Prenatal growth and early postnatal influences on adult motor cortical excitability.

OBJECTIVE: Suboptimal prenatal growth may adversely influence motor neurophysiologic development and predispose the individual to greater risk of neurodegenerative disorders in later life. We investigated the influences of prenatal growth and the postnatal environment on motor cortical function in young adults. METHODS: Transcranial magnetic stimulation was used to construct corticospinal stimulus-response curves for 35 young adults (mean age: 28 +/- 0.5 years; 19 males) born >or=37 weeks' gestation. Birth weight centile was calculated relative to maternal size, parity, ethnicity, gender, and gestation. Handgrip strength and dexterity were measured separately. Regression analyses assessed the influence of prenatal (birth weight centile and gestation) and postnatal (socioeconomic indices and maternal education) factors on corticospinal parameters, strength, and dexterity scores. RESULTS: Lower birth weight was associated with increased interhemispheric asymmetry in motor threshold and increased cortical stimulus-response curve slope. A shorter gestation predicted a larger area under this curve in the right hand. High motor threshold was predicted by greater environmental adversity in early postnatal life, but not by prenatal factors. Higher birth weight centile and lower motor threshold were associated with greater educational achievement. CONCLUSIONS: Poor in utero growth and mild prematurity are associated with altered corticospinal excitability in adulthood. An early postnatal environment with less early postnatal socioeconomic disadvantage and having a mother with a completed high school education partly ameliorates this. While altered cortical development has some functional consequences already evident in early adulthood, it may have a later, additional adverse impact on aging-related changes in motor function.

Facilitation of cortically evoked potentials with motor imagery during post-exercise depression of corticospinal excitability.

This study examined whether muscle fatigue alters the facilitatory effect of motor imagery on corticospinal excitability. We aimed to determine if post-exercise depression of potentials evoked magnetically from the motor cortex is associated with alterations in internally generated movement plans. In experiment 1, motor-evoked potentials (MEPs) were recorded from two right hand and two right forearm muscles, at rest and during motor imagery of a maximal handgrip contraction, in eight neurologically normal subjects, before and after a 2-min maximal voluntary handgrip contraction. Resting MEP amplitude was facilitated by motor imagery in three of the four muscles, but consistently only in two. Motor imagery also reduced the trial-to-trial variability of resting MEPs. Following the exercise, resting MEP amplitude was depressed reliably in only one muscle engaged in the task, although two other muscles exhibited some depression. Motor imagery MEPs were smaller after exercise, but the degree of facilitation compared to the rest MEP was unchanged. In experiment 2, TMS intensity was increased after exercise-induced MEP depression so that the MEP amplitude matched the pre-exercise baseline. The amplitude of the MEP facilitated with motor imagery was not altered by MEP depression, nor was it increased when the TMS intensity was increased. These results suggest, at least with a simple motor task, that while post-exercise depression reduces corticospinal excitability, it does not appear to significantly affect the strength of the input to the motor cortex from those areas of the brain responsible for the storage and generation of internal representations of movement.