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Summary Background Screening for amblyopia in early childhood is done in many countries to ensure that affected children are detected and treated within the critical period, and achieve a level of vision in their amblyopic eye that would be useful should they lose vision in their non-amblyopic eye later in life. We aimed to investigate the risk, causes, and outcomes of visual impairment attributable to loss of vision in the non-amblyopic eye. Methods For 24 months from July, 1997, national surveillance was done to identify all individuals in the UK with unilateral amblyopia (acuity worse than 6/12) who had newly acquired vision loss in the non-amblyopic eye, resulting in acuity of worse than 6/12 or visual-field restriction precluding driving. Information about participants was obtained at presentation and 1 year later. Participants were categorised as having socially significant visual impairment, or visual impairment, severe visual impairment, or blindness, in accordance with WHO taxonomy. Findings Of 370 eligible individuals, at presentation 104 (28%) had socially significant visual impairment, 180 (49%) visual impairment, and 86 (23%) severe visual impairment or blindness. The minimum risk of permanent visual impairment by age 95 years was 32·9 (95% CI 29·1–36·9) per 100 000 total population. The projected lifetime risk of vision loss for an individual with amblyopia was at least 1·2% (95% CI 1·1–1·4). Only 36 (35%) of 102 people previously in paid employment were able to continue. Interpretation In the UK, where screening for amblyopia is under review, risk of serious vision loss affecting the non-amblyopic eye and its results are greater than that previously assumed. Thus, in addition to the benefits of improved vision in the amblyopic eye, treatment of amblyopia during childhood is a potentially valuable strategy to prevent incapacitating vision loss later in life.

Importance Despite the existing country-specific strategies tackling social inequalities in visual health in adults, little is known about trends in visual function in childhood and its association with social position. Objective To investigate the distribution of childhood visual function in the United Kingdom and associations with early-life social position between 1961 and 1986, a period of significant social change. Design, Setting, and Participants Longitudinal cohort study using harmonized data sets from the British 1946, 1958, and 1970 national birth cohorts. In total, 14 283 cohort members with complete data on visual acuity at age 15 or 16 years, measured in 1961, 1974, and 1986, respectively, for each cohort, and social position were assessed. Main Outcomes and Measures Using habitual distance visual acuity (with correction if prescribed), participants were assigned to a visual function category ranging from bilateral normal to visual impairment/severe visual impairment/blindness ( International Statistical Classification of Diseases, Tenth Revision, Clinical Modification ). Distribution of visual function over time and associations with social position (risk ratios [RRs] and 95% confidence intervals) were analyzed. Results Complete data were available for 3152 participants (aged 15 years; 53% boys [n = 1660]) in the 1946 Medical Research Council National Survey of Health and Development, 6683 participants (aged 16 years; 51% boys [n = 3420]) in the 1958 National Child Development Study, and 4448 participants (aged 16 years; 48% boys [n = 2156]) in the 1970 British Birth Cohort Study. The proportion of children with bilateral normal vision decreased by 1.3% (95% CI, −5.1% to 2.7%) in 1974 and 1.7% (95% CI, −5.9% to 2.7%) in 1986. The risk of overall impaired vision increased by 1.20 times (95% CI, 1.01-1.43) and the risk of visual impairment/severe visual impairment/blindness by 1.75 times (95% CI, 1.03-2.98) during this period. Girls were consistently at increased risk of all vision impairment categories. Higher social position at birth and in childhood was associated with reduced risk of visual impairment/severe visual impairment/blindness (RR, 0.58; 95% CI, 0.20-1.68) and unilateral impairment (RR, 0.89; 95% CI, 0.72-1.11), respectively. Conclusions and Relevance Our study provides evidence of temporal decline in childhood visual function between 1961 and 1986. Despite the limited power of the analysis owing to the small sample size of those with impaired vision, we found an emergence of a contribution of sociodemographic status to the cohort effect that may be the antecedent of the current picture of childhood blindness. Equally, early-life social position may also have contributed to the current social patterning in visual function in older adults in the United Kingdom. These findings highlight the potential value of targeting children in national ophthalmic public policies tackling inequalities.

Refractive error (RE) is a complex, multifactorial disorder characterized by a mismatch between the optical power of the eye and its axial length that causes object images to be focused off the retina. The two major subtypes of RE are myopia (nearsightedness) and hyperopia (farsightedness), which represent opposite ends of the distribution of the quantitative measure of spherical refraction. We performed a fixed effects meta-analysis of genome-wide association results of myopia and hyperopia from 9 studies of European-derived populations: AREDS, KORA, FES, OGP-Talana, MESA, RSI, RSII, RSIII and ERF. One genome-wide significant region was observed for myopia, corresponding to a previously identified myopia locus on 8q12 (p = 1.25×10-8), which has been reported by Kiefer et al. as significantly associated with myopia age at onset and Verhoeven et al. as significantly associated to mean spherical-equivalent (MSE) refractive error. We observed two genome-wide significant associations with hyperopia. These regions overlapped with loci on 15q14 (minimum p value = 9.11×10-11) and 8q12 (minimum p value 1.82×10-11) previously reported for MSE and myopia age at onset. We also used an intermarker linkage- disequilibrium-based method for calculating the effective number of tests in targeted regional replication analyses. We analyzed myopia (which represents the closest phenotype in our data to the one used by Kiefer et al.) and showed replication of 10 additional loci associated with myopia previously reported by Kiefer et al. This is the first replication of these loci using myopia as the trait under analysis. "Replication-level" association was also seen between hyperopia and 12 of Kiefer et al.'s published loci. For the loci that show evidence of association to both myopia and hyperopia, the estimated effect of the risk alleles were in opposite directions for the two traits. This suggests that these loci are important contributors to variation of refractive error across the distribution.

As visual impairment (VI) due to adverse drug reactions (ADR) is rare in adults and children, there is an incomplete evidence base to inform guidance for screening and for counseling patients on the potential risks of medications. We report on suspected drugs and the eye conditions found in a national study of incidence of diagnosis of visual impairment due to suspected ADR. Case ascertainment was via the British Ophthalmological Surveillance Unit (BOSU), between March 2010 and February 2012, with follow-up after 6 months. Case definition: any child or adult with bilateral or unilateral visual impairment due to a suspected ADR, using distance acuity worse than Snellen 6/18 (logMAR 0.48) in the better eye (bilateral) or affected eye (unilateral). Anonymized patient information on potential cases was provided by managing ophthalmologists, comprising visual status before and after suspected ADR, ophthalmic condition attributable to the ADR, preexisting eye disease and prescribed medications at the time of the ADR. Permanency and causality of the visual impairment were confirmed by the managing clinician, after 6 months, using the WHO Uppsala Monitoring Committee criteria. Over 2 years, 36 eligible cases were reported of whom 23 had permanent VI. While most cases were due to drugs known to have adverse side-effects, some were unanticipated sporadic cases. Visual impairment due to ADRs is rare. However, with for example, increasing polypharmacy in the elderly, monitoring of ocular ADRs, although challenging, is necessary.

Aim:\ud To identify patient-reported outcome measures (PROMs) specifically developed and used to assess the impact of ophthalmic disorders in children and to systematically assess their quality as a basis for recommendations about their use in clinical and research settings.\ud \ud Methods:\ud A systematic review of the literature was performed in MEDLINE, EMBASE, PsychINFO, CINAHL and AMED, supplemented by a grey literature search. Papers reporting development and validation of questionnaire instruments for assessing patient-reported outcomes of an ophthalmic disorder in patients aged 2–18 years were included. Quality was assessed by examining the purpose and psychometric properties of the instruments. Strengths and limitations were summarised with recommendations regarding use.\ud \ud Results:\ud Search identified 17 instruments. Of these, 11 were condition-specific and six were intended for a broader population of children and young people with visual impairment regardless of the ophthalmic condition. Three were developed for use in a specific trial and two are still in development.\ud \ud Conclusions:\ud Paediatric ophthalmology PROM development and application is a developing field and new instruments are needed. There is scope for improvement in this area through (a) clarity of definitions of the underlying constructs intended to be measured at the onset of development of new instruments, (b) application of child-centred approaches and (c) adherence to extant guidance and best practice in questionnaire instrument development.

BACKGROUND: The prevalence and causes of blindness in children vary widely between regions. Few epidemiological data are available on the relative importance of the major congenital anomalies of the globe (i.e., microphthalmos, anophthalmos, coloboma) as causes of blindness in children. The aim of this study was to determine the re-gional variation in the proportion of severe visual impairment and blindness due to congenital abnormalities of the globe in children in schools for the blind and in those identified through Community Based Rehabilitation programs. Other objectives were to estimate the prevalence of blindness due to major congenital abnormalities, and to investigate their etiology. METHODS: Data on the causes of blindness in children were collected between 1990 and 1998 using standard methods, definitions and reporting form in 26 countries. Children were examined in schools for the blind and in Community Based Rehabilitation programs. RESULTS: Of 7,113 children aged 3-15 years with severe visual impairment and blindness examined, 762 (10.7%) had microphthalmos, 161 (2.3%) had anophthalmos, and 96 (1.3%) had coloboma. There are large regional differences in the proportion of severe visual loss in blind school children, ranging from 1.4% in Cuba to 33.2% in Sri Lanka. Severe visual loss due to congenital abnormalities of the globe is estimated to affect between 0.4 and 16.2/100,000 children in the countries studied. An underlying cause could not be identified in 84.2%. CONCLUSIONS: Major congenital abnormalities of the globe are important causes of severe visual loss in children, particularly in Asian countries. Further research into etiology is warranted in order to plan prevention programs.

Objectives: To determine the mode of detection and timing of ophthalmic assessment of a nationally representative group of children with congenital and infantile cataract. Design: Cross sectional study. Setting: United Kingdom. Subjects: All children born in the United Kingdom and aged 15 years or under in whom congenital or infantile cataract was newly diagnosed between October 1995 and September 1996. Main outcome measures: Proportion of cases detected through routine ocular examination and proportion assessed by an ophthalmologist by 3 months and 1 year of age. Results: Data were complete for 235 (95%) of 248 children identified. Of these, 83 (35%) were detected at the routine newborn examination and 30 (12%) at the 6-8 week examination; 82 children presented symptomatically. 137 (57%) children had been assessed by an ophthalmologist by the age of 3 months but 78 (33%) were not examined until after 1 year of age. In 91 cases the child9s carers suspected an eye defect before cataract was diagnosed. Conclusions: A substantial proportion of children with congenital and infantile cataract are not diagnosed by 3 months of age, although routine ocular examination of all newborn and young infants is recommended nationally. Strategies to achieve earlier detection through screening and surveillance are required.

Since vision is the major sensory modality in humans, normal vision is important for the general development of a child.1 2 Visual impairment has significant implications for the affected child and family in terms of education, future employment, and personal and social welfare throughout life. Using the World Health Organisation (WHO) classification of levels of visual impairment3 (shown in table 1 and used throughout this paper), it is estimated that globally almost one in 1000 children are blind, which is less than a tenth of the prevalence in adults.4 5 However, the 1.5 million blind children in the world account for about 75 million person years of blindness,6 equivalent to the burden due to cataract related blindness, which accounts for 40% of adult blindness worldwide.7 As the combined categories of low vision in childhood (table 1) are three to 10 times more common than blindness,5 6 the total burden of childhood visual impairment is considerable. It has been estimated that globally, the financial costs of childhood blindness incurred by countries, in terms of care and productivity lost, are between 6 and 27 thousand million US dollars.8 Most of this is accounted for by children in high income countries where childhood blindness is less common but life expectancy and earning capacity are greater than in low income countries. Reduction of avoidable visual impairment remains an important international public health goal. View this table: Table 1 WHO classification of levels of visual impairment (adapted from International Classification of Diseases, 10th revision3) Knowledge of the magnitude and causes of childhood visual impairment is important to the planning, provision, and evaluation of educational and health services for affected children. This is also essential information for all strategies to reduce visual disability in the population. The purpose of this paper …

To explore current challenges surrounding the development and application of PROMs/PREMs in paediatric healthcare1, a workshop was held for healthcare professionals and parent representatives from University College London (UCL) Institute of Child Health (ICH) and two clinical partners, Great Ormond Street Hospital and Moorfields Eye Hospital. The workshop aimed to establish an academic-clinical-service user collaborative network to support local development and translation into clinical practice of paediatric PROMs/PREMs. The workshop report summarises invited presentations, small group discussions and key local issues. The report is permanently archived at UCL Discovery and publicly available.

To develop a novel age-appropriate measure of functional vision (FV) for self-reporting by visually impaired (VI) children and young people.Questionnaire development.A representative patient sample of VI children and young people aged 10 to 15 years, visual acuity of the logarithm of the minimum angle of resolution (logMAR) worse than 0.48, and a school-based (nonrandom) expert group sample of VI students aged 12 to 17 years.A total of 32 qualitative semistructured interviews supplemented by narrative feedback from 15 eligible VI children and young people were used to generate draft instrument items. Seventeen VI students were consulted individually on item relevance and comprehensibility, instrument instructions, format, and administration methods. The resulting draft instrument was piloted with 101 VI children and young people comprising a nationally representative sample, drawn from 21 hospitals in the United Kingdom. Initial item reduction was informed by presence of missing data and individual item response pattern. Exploratory factor analysis (FA) and parallel analysis (PA), and Rasch analysis (RA) were applied to test the instrument's psychometric properties.Psychometric indices and validity assessment of the Functional Vision Questionnaire for Children and Young People (FVQ_CYP).A total of 712 qualitative statements became a 56-item draft scale, capturing the level of difficulty in performing vision-dependent activities. After piloting, items were removed iteratively as follows: 11 for high percentage of missing data, 4 for skewness, and 1 for inadequate item infit and outfit values in RA, 3 having shown differential item functioning across age groups and 1 across gender in RA. The remaining 36 items showed item fit values within acceptable limits, good measurement precision and targeting, and ordered response categories. The reduced scale has a clear unidimensional structure, with all items having a high factor loading on the single factor in FA and PA. The summary scores correlated significantly with visual acuity.We have developed a novel, psychometrically robust self-report questionnaire for children and young people-the FVQ_CYP-that captures the functional impact of visual disability from their perspective. The 36-item, 4-point unidimensional scale has potential as a complementary adjunct to objective clinical assessments in routine pediatric ophthalmology practice and in research.

Importance Large-scale generic studies offer detailed information on potential risk factors for refractive error across the life course, but ophthalmic examination in such cases to determine the refractive error phenotype is challenging and costly. Thus, refractive status is commonly assigned using questionnaires. In a population survey, often only a few condition-specific self-reported questions can be included, so the questions used must be effective in ruling in those who have the trait of interest and ruling out those who do not. Objective To determine the accuracy of identification of refractive status using self-reported age at and/or reason for first use of glasses or contact lenses (optical correction). Design, Setting, and Participants The UK Biobank study, a cross-sectional epidemiologic study, included 117 278 participants aged 40 to 69 years in 6 regional centers in England and Wales. Data for the present study were assessed from June 2009 to July 2010. Patients underwent autorefraction measurement. Spherical equivalent in the more extreme eye was used to categorize myopia (−1.00 diopter [D] or more extreme) and hypermetropia (+1.00 D or more extreme). Main Outcomes and Measures Sensitivity and specificity of the reason for optical correction were assessed using autorefraction as the gold standard. Receiver operating characteristic curves assessed the accuracy of self-reported age at first use of optical correction and incremental improvement with addition of the reason. Results Of the 95 240 participants who reported using optical correction (55.6% female; mean [SD] age, 57.7 [7.5] years), 92 121 (96.7%) provided their age at first use and 93 156 (97.8%) provided the reason. For myopia, sensitivity of the reason for optical correction was 89.1% (95% CI, 88.7%-89.4%), specificity was 83.7% (95% CI, 83.4%-84.0%), and positive and negative predictive values were 72.7% (95% CI, 72.2%-73.1%) and 94.0% (95% CI, 93.8%-94.2%), respectively. The area under the curve was 0.829 (95% CI, 0.826-0.831) and improved to 0.928 (95% CI, 0.926-0.930) with combined information. By contrast, self-report of the reason for optical correction of hypermetropia had low sensitivity (38.1%; 95% CI, 37.6%-38.6%), and the area under the curve with combined information was 0.713 (95% CI, 0.709-0.716). Conclusions and Relevance In combination, self-report of the reason for and age at first use of optical correction are accurate in identifying myopia. These findings indicate an agreed set of questions could be implemented effectively in large-scale generic population-based studies to increase opportunities for integrated research on refractive error leading to development of novel prevention or treatment strategies.

Aims:\ud To investigate agreement between children with visual impairment (VI) and their parents on their ratings of the child's vision-related quality of life (VQoL) and functional vision (FV) using two novel self-report patient-reported outcome measures developed for this population.\ud \ud Methods:\ud 99 children aged 10–15 years (mean age=12.2, SD=1.9) with VI (best corrected acuity (logarithm of the minimum angle of resolution) 0.50 or worse in better eye) and their parents participated in a national postal survey, completing the child and proxy versions of our novel instruments assessing VQoL and FV of children with VI—the vision-related quality of life instrument for children and young people (VQoL_CYP) and the functional vision questionnaire for children and young people (FVQ_CYP), respectively. Parent-child agreement was investigated using the Bland-Altman (BA) method. Variation across key sociodemographic and clinical characteristics was examined using the Intraclass Correlation Coefficient.\ud \ud Results:\ud Average parental ratings of their child's VQoL and FV were significantly lower than the children's own ratings, but the range of disagreement was wide, with parents both overestimating and underestimating their child's VQoL (mean score difference=5.7, BA limits of agreement (LOA): lower −22.10 (CI 95% −24.61 to 19.59) and upper 33.50 (CI 95% 30.99 to 36.01)), but more consistently underestimating the child's FV (mean score difference=−11.8, BA LOA: lower −39.60 (CI 95% −42.12 to 37.08) and upper 16 (CI 95% 13.48 to 18.52)). There was variation in agreement by some child characteristics, including vision level, time of onset and course of VI progression.\ud \ud Conclusions:\ud Visually impaired children and their parents perceive the broader impact of living with VI very differently. There is value in routine capture of information independently from children and their parents for comprehensively gauging the impact of childhood VI and tailoring appropriate interventions.

OBJECTIVES--To investigate the importance of vitamin A deficiency as a cause of childhood blindness and severe visual impairment (SVI) in India. DESIGN AND SETTING--Cross sectional survey of children with visual acuity less than 6/60 in the better eye (SVI/blindness), attending 22 schools for the blind in nine states of India. MAIN OUTCOME MEASURE--Proportion of children with blindness or SVI from corneal scarring attributable to vitamin A deficiency. RESULTS--245 of 1318 (18.6%) of children had SVI/blindness attributable to vitamin A deficiency. The proportion of SVI/blindness due to vitamin A deficiency ranged from 7.5% (7/93) in Kerala to 26.7% (27/101) in Madhya Pradesh. In Tamil Nadu, 7.5% (9/120) of children in the capital city blind school had SVI/blindness due to vitamin A deficiency, compared with 30.4% (51/168) in a blind school in a rural area of the same state. CONCLUSION--Overall, vitamin A deficiency is the single most important cause of childhood blindness and SVI in India. There are marked variations by state and also between urban and rural locations.

Myopia is the most common human eye disorder and it results from complex genetic and environmental causes. The rapidly increasing prevalence of myopia poses a major public health challenge. Here, the CREAM consortium performs a joint meta-analysis to test single-nucleotide polymorphism (SNP) main effects and SNP × education interaction effects on refractive error in 40,036 adults from 25 studies of European ancestry and 10,315 adults from 9 studies of Asian ancestry. In European ancestry individuals, we identify six novel loci (FAM150B-ACP1, LINC00340, FBN1, DIS3L-MAP2K1, ARID2-SNAT1 and SLC14A2) associated with refractive error. In Asian populations, three genome-wide significant loci AREG, GABRR1 and PDE10A also exhibit strong interactions with education (P, This report by the Consortium for Refractive Error and Myopia uses gene-environment-wide interaction study (GEWIS) to identify genetic loci that affect environmental influence in myopia development, and identifies ethnic specific genetic loci that attribute to eye refractive errors.

Myopia is a complex genetic disorder and a common cause of visual impairment among working age adults. Genome-wide association studies have identified susceptibility loci on chromosomes 15q14 and 15q25 in Caucasian populations of European ancestry. Here, we present a confirmation and meta-analysis study in which we assessed whether these two loci are also associated with myopia in other populations. The study population comprised 31 cohorts from the Consortium of Refractive Error and Myopia (CREAM) representing 4 different continents with 55,177 individuals; 42,845 Caucasians and 12,332 Asians. We performed a meta-analysis of 14 single nucleotide polymorphisms (SNPs) on 15q14 and 5 SNPs on 15q25 using linear regression analysis with spherical equivalent as a quantitative outcome, adjusted for age and sex. We calculated the odds ratio (OR) of myopia versus hyperopia for carriers of the top-SNP alleles using a fixed effects meta-analysis. At locus 15q14, all SNPs were significantly replicated, with the lowest P value 3.87 × 10−12 for SNP rs634990 in Caucasians, and 9.65 × 10−4 for rs8032019 in Asians. The overall meta-analysis provided P value 9.20 × 10−23 for the top SNP rs634990. The risk of myopia versus hyperopia was OR 1.88 (95 % CI 1.64, 2.16, P

Background The purpose of this study was to investigate current patterns of management and outcomes of intermittent distance exotropia [X(T)] in the UK. Methods This was an observational cohort study which recruited 460 children aged < 12 years with previously untreated X(T). Eligible subjects were enrolled from 26 UK hospital ophthalmology clinics between May 2005 and December 2006. Over a 2-year period of follow-up, clinical data were prospectively recorded at standard intervals from enrolment. Data collected included angle, near stereoacuity, visual acuity, control of X(T) measured with the Newcastle Control Score (NCS), and treatment. The main outcome measures were change in clinical outcomes (angle, stereoacuity, visual acuity and NCS) in treated and untreated X(T), 2 years from enrolment (or, where applicable, 6 months after surgery). Change over time was tested using the chi-square test for categorical, Wilcoxon test for non-parametric and paired-samples t-test for parametric data. Results At follow-up, data were available for 371 children (81% of the original cohort). Of these: 53% (195) had no treatment; 17% (63) had treatment for reduced visual acuity only (pure refractive error and amblyopia); 13% (50) had non surgical treatment for control (spectacle lenses, occlusion, prisms, exercises) and 17% (63) had surgery. Only 0.5% (2/371) children developed constant exotropia. The surgically treated group was the only group with clinically significant improvements in angle or NCS. However, 8% (5) of those treated surgically required second procedures for overcorrection within 6 months of the initial procedure and at 6-month follow-up 21% (13) were overcorrected. Conclusions Many children in the UK with X(T) receive active monitoring only. Deterioration to constant exotropia, with or without treatment, is rare. Surgery appears effective in improving angle of X(T) and NCS, but rates of overcorrection are high.

Children with visual and ocular disorders form a heterogeneous group with differing ocular and systemic disorders, and visual, other sensory, motor and global developmental impairments which impact the level and type of support they need in order to function to their highest capabilities.

BACKGROUND/AIMS Congenital ocular anomalies contribute significantly to childhood visual morbidity, with congenital cataract being a major cause of visual impairment throughout the world. As in many other countries, a National Congenital Anomaly System (NCAS) exists in England and Wales to monitor the frequency of ocular and other anomalies in order to identify new public health hazards and inform aetiological research. The aim of this study was to assess level of ascertainment by the NCAS of children with congenital cataract. METHODS Using independent ophthalmic and paediatric national active surveillance schemes, all infants (⩽1 year) newly diagnosed with congenital and infantile cataract in England and Wales in 1 year from September 1995 were identified. These notifications were compared with those made independently to the NCAS during the same period. The proportion of cases identified by the active surveillance schemes and also notified to the NCAS was determined. RESULTS 10% (15/149) of eligible children with newly diagnosed congenital or infantile cataract were actually notified to the NCAS. A higher proportion of those diagnosed as neonates (16%, 14/85) than in later infancy (2%, 1/64) was ascertained through the NCAS. There is a need for better verification of notifications and reported information in the NCAS. CONCLUSION Currently, ascertainment of congenital cataract through the NCAS is low and the system is likely to be insensitive to small but important changes in risk factors for this disorder. This limits its use for monitoring secular and other trends in ocular anomalies. Strategies to improve its future use are discussed, including enhancing the awareness and participation of ophthalmic professionals involved in managing children with anomalies.

AIMS To determine the visual outcome and complications of lens aspiration with intraocular lens implantation in children aged 5 years and under. METHODS The hospital notes of all children aged 5 years and under, who had undergone lens aspiration with intraocular lens implantation between January 1994 and September 1998, and for whom follow up data of at least 1 year were available, were reviewed. RESULTS Of 50 children who underwent surgery, 45 were eligible based on the follow up criteria. 34 children had bilateral cataracts and, of these, 30 had surgery on both eyes. Cataract was unilateral in 11 cases; thus, 75 eyes of 45 children had surgery. Cataracts were congenital in 28 cases, juvenile in 16, and traumatic in one case. The median age at surgery was 39 months (range 11–70 months). Follow up ranged from 12–64 months (median 36 months). Of 34 children with bilateral disease, 25 (73.5%) had a final best corrected visual acuity of 6/12 or better, while seven (20.5%) achieved 6/18 or less; in one child the vision improved from UCUSUM to CSM but another, who had only one eye operated on, was unable to fix or follow with this eye preoperatively or 2 years postoperatively. Of 11 children with unilateral cataract, five (45.5%) had a final best corrected visual of 6/12 or better, and six (54.5%) 6/18 or less. A mild fibrinous uveitis occurred in 20 (28.2%) eyes in the immediate postoperative period, but resolved with topical steroids. One child had a vitreous wick postoperatively requiring surgical division. Glaucoma, endophthalmitis, or retinal detachment have not been observed so far in any patient postoperatively. CONCLUSION From this series the authors suggest that, in children aged 5 years and under, lens aspiration with intraocular lens implantation is a safe procedure, with a good visual outcome in the short term. Further studies are needed to investigate these outcomes in the long term.

OBJECTIVES: In industrialized countries, there are established programs of childhood vision screening and surveillance, but little is known about their performance. We investigated the patterns of presentation/detection and early treatment of a nationally representative cohort of children with severe visual impairment or blindness (SVI/BL) in 1 year (2000) in the United Kingdom. METHODS: All children who were younger than 16 years and had a new diagnosis of SVI/BL were identified by active surveillance through the British Ophthalmological and Pediatric Surveillance Units. Data that were collected up to 1 year after diagnosis included sociodemographic characteristics, detection of SVI/BL, nonophthalmic disorders/impairments, ophthalmic findings, and early management. RESULTS: Of 439 identified children, 65% were younger than 1 year at diagnosis, 28% were of nonwhite ethnicity, and 40% in the worst quintile of deprivation score. A total of 77% had associated nonophthalmic disorders/impairments. Although 70% had established symptoms or signs at diagnosis by a health professional, parents had suspected blindness in only 47%. A quarter of isolated SVI/BL was detected through routine vision screening; however, 46% of children's SVI/BL and associated nonophthalmic disorders/impairments were diagnosed through a clinical surveillance examination undertaken because of high risk for a specific eye disease. CONCLUSIONS: The “patient journey” of children with visual impairment is markedly influenced by the presence of additional impairments/chronic diseases. Parents' understanding of normal visual development needs to be improved. Increasingly, new evidence-based formal programs of clinical (ophthalmic) surveillance are needed in response to the changing population of children who are at risk for blinding eye disease.

To describe a child-centered approach to identifying content for a novel self-report questionnaire for assessing vision-related quality of life (QoL) of visually impaired (VI) or blind (BL) children and young people.Questionnaire development.A stratified random patient sample of children and young people who are VI/BL (visual acuity in the better eye Snellen6/18; logarithm of the minimum angle of resolution0.51) as the result of any visual disorder, but in the absence of any other significant impairment, aged 10 to 15 years (N=49); and a convenience school-based sample of children and young people who are VI/BL and aged 10 to 17 years (N=29).Individual interviews were conducted with a stratified random sample of 32 children and young people, aged 10 to 15 years, who were VI/BL. The interviews followed a topic guide based on vision-related issues identified from a focus group of affected children and young people, combined with a literature review and consultations with professionals. Collaborative qualitative thematic analysis was undertaken and used to derive draft items of the instrument, using the children's own language wherever possible. Items were reduced, rephrased, and refined through individual consultation, as well as an expert reference group of children and young people who were VI/BL, and supplemented by the research team's consensus.A draft 47-item instrument.A total of 874 potential questionnaire items were initially generated spanning the following domains: social relations, acceptance, and participation; independence and autonomy; psychological and emotional well-being; future-aspirations and fears; functioning-home, school, and leisure; and treatment of eye condition. This was eventually reduced to a 47-item instrument with each item presented as a vignette describing a QoL issue from an "illustrative" child's perspective. Thus, the responding child reports on how much he/she is presently like and how much he/she wishes to be like that child, using a 4-point Likert-type scale.We demonstrate that a child-centered approach to identifying the content for a self-report vision-related QoL questionnaire is feasible. We suggest this approach is critical to accurately capturing children and young peoples' subjective perspectives on the impact of living with impaired vision.

Purpose:\ud To investigate patterns of participation of visually impaired (VI) children and their families in health services research.\ud \ud Methods:\ud The authors compared clinical and sociodemographic characteristics of children and their families who participated with those who did not participate in two studies of quality of life (QoL) of VI children. In Study 1, the authors interviewed VI children and adolescents, aged 10 to 15 years, about their vision-related quality of life (VRQoL) as the first phase of a program to develop a VRQoL instrument for this population. One hundred seven children with visual impairment (visual acuity in the better eye LogMar worse than 0.51) were invited to participate in the interviews. Study 2 investigated health-related quality of life (HRQoL) of VI children using an existing generic instrument, administered in a postal survey. 151 VI children and adolescents, aged 2 to 16 years, with hereditary retinal disorders were invited to participate in the survey.\ud \ud Results:\ud The overall participation level was below 50%. In both studies, participants from white ethnic and more affluent socioeconomic backgrounds were overrepresented. Participation did not vary by age, sex, or clinical characteristics.\ud \ud Conclusions:\ud The authors suggest that there are barriers to participation in child- and family-centered research on childhood visual disability for children from socioeconomically deprived or ethnic minority groups. They urge assessment and reporting of participation patterns in further health services research on childhood visual disability. Failure to recognize that there are “silent voices” is likely to have important implications for equitable and appropriate service planning and provision for VI children.

To report the prevalence and distribution of eye conditions and visual impairment and associations with early social and biological factors using parental report of diagnosed eye conditions and additional chronic illnesses.Population-based, cross-sectional study.We included 14 981 children, aged 3 years, participating in the United Kingdom Millennium Cohort Study.Data on demographic, socioeconomic, and maternal and child health factors were obtained by maternal report through structured interview and verbatim reports of diagnosed eye problems and additional chronic illnesses were recorded. Multinomial regression analyses were used to calculate risk ratios of the association of eye disease (with or without associated visual impairment), with socioeconomic and early life factors.Parental report of diagnosed eye conditions and other chronic illnesses.Overall, at 3 years, 5.7% (95% confidence interval, 5.2-6.3%; n = 881) of children had ≥ 1 eye condition with 0.24% (0.15-0.3%; n = 45) reported to have associated visual impairment. In the majority, time of onset was reported to be the first year of life. Eye disorders without report of visual impairment were independently associated with lower socioeconomic status, decreasing birth weight, and prematurity. Visual impairment was more likely in those of low birthweight for gestational age and from an ethnic minority group. Maternal illnesses during pregnancy were associated with eye disease without reported visual impairment, as was white ethnicity.Good research opportunities exist within the context of population-based general health surveys to use parental report to estimate minimum prevalence, investigate associations of eye disease with a broad range of environmental factors, and as a mechanism for "flagging" individuals with eye disease in a population sample for further study. Our findings regarding the association of parentally reported childhood eye disease with early life factors such as modest degrees of prematurity, ethnicity and maternal ill-health warrant further investigation.The authors have no proprietary or commercial interest in any of the materials discussed in this article.

To investigate factors associated with early management of intermittent exotropia (X(T)) in hospital eye departments in the UK in a prospective cohort study.An inception cohort of 460 children aged12 years with previously untreated X(T) (mean age 3.6 years, 55.9% girls) was recruited from 26 UK hospital children's eye clinics and orthoptic departments. Participants received a standard ophthalmic examination at recruitment and orthoptic assessment at three-monthly intervals thereafter. The influence of severity of exotropia (control measured by Newcastle Control Score (NCS), and angle of strabismus, visual acuity and stereoacuity) and age on the type of management was investigated.Within the first 12 months following recruitment, 297 (64.6%) children received no treatment, either for impaired visual acuity or for strabismus. Ninety-six (21%) children had treatment for impaired visual acuity. Eighty-nine (19.4%) received treatment for strabismus (22 of whom also received treatment for defective visual acuity); in 54 (11.7%) treatment was non-surgical and in 35 (7.6%) eye muscle surgery was performed. Children with poor (score 7-9) control of strabismus at recruitment were more likely to have surgery than children with good (score 1-3) control (p0.001). Children who had no treatment were younger (mean age 3.38 years) than those who were treated (mean 4.07 years) (p0.001). Stereoacuity and size of the angle of strabismus did not influence the type of management received.X(T) can be a presenting sign of reduced visual acuity. Most children with well controlled X(T) receive no treatment within 12 months following presentation.

Purpose To investigate how visual function in mid-adult life is associated with health and social outcomes and, using life-course epidemiology, whether it is influenced by early life biological and social factors. Design Population-based cohort study. Participants Nine thousand three hundred thirty members of the 1958 British birth cohort at age 44 or 45 years. Methods Distance, near, and stereo vision were assessed as part of a broader biomedical examination. Logistic, multinomial, and proportional odds ordinal logistic regression were used, as appropriate, to assess the association between these vision functions and both key early life influences and health and social outcomes in mid-adult life. Main Outcome Measures Distance, near, and stereo acuities and health and social outcomes. Results In mid-adult life, vision function (across the full spectrum of both type and level of function) is associated with unemployment resulting from permanent sickness, lower socioeconomic status, and poorer general health (for example, for blindness; odds ratios were 2.5, 2.6, and 1.2, respectively). Also, impaired visual functions in mid-adult life are associated with a low birthweight, being small for gestational age, maternal smoking in pregnancy, and markers of socioeconomic deprivation in childhood (for example, for impaired distance acuity; odds ratios were 1.4, 1.3, 1.02, and 1.1, respectively). Conclusions Although relatively uncommon in working-age adults, impaired vision can have important adverse consequences, which highlights the value of investigating visual function in the broader context of health and social functioning. In addition, visual function in adult life may be influenced directly by key prenatal and childhood biological and social determinants of general health. Thus, application of life-course epidemiology to complex chronic ophthalmic diseases of adult life such as glaucoma or macular degeneration is likely to prove valuable in elucidating whether and how biological, social, and lifestyle factors contribute to the cause. Financial Disclosure(s) The author(s) have no proprietary or commercial interest in any materials discussed in this article.

To describe the prevalence of impaired vision and its relative burden, together with the prevalence of impaired vision-related quality of life (VRQOL), and investigate associations with social outcomes in a contemporary and nationally representative population of working age adults.Population-based cross-sectional study.We included 9330 members of the 1958 British birth cohort at age 44 and 45 years."Habitual" and "best achieved" distance visual acuity in each eye, binocular near vision acuity and stereoacuity (three dimensional/depth perception) were tested during a broader biomedical examination. VRQOL was assessed using the Vision-related Quality of Life Core Measure 1 (VCM1), a validated, 10-item, self-complete instrument. Logistic and proportional odds ordinal logistic regression were used to calculate odds ratios (ORs) of the association of VRQOL with visual acuities and social outcomes.Distance, near, and stereo acuities and VRQOL and social outcomes.Of the 1.3% (124) of those with visual loss that precluded driving, a further 0.75% (70) were visually impaired or severely visually impaired and 0.15% (14) blind, the latter accounting for 19% total population (all ages) burden of blindness. Impairment of VRQOL is strongly associated with impaired distance, near, and stereo vision, as well as with adverse occupational and other social outcomes. However, VRQOL impairment is also sometimes reported with unilateral or mild bilateral visual loss.Although impaired vision in working age adults is relatively uncommon, it confers important adverse consequences for the "health and wealth" of the public. This may be captured best by assessment of VRQOL in addition to objective visual function. Ophthalmic disorders occurring or impacting in middle life should be given a higher priority than currently in national and international strategies against avoidable visual disability.Proprietary or commercial disclosure may be found after the references.

PURPOSE. Myopia is a common complex trait that affects up to 60% of some populations. Its development is influenced by multiple genes and environmental factors. PAX6 and SOX2 are genes with fundamental roles in ocular growth and development, and they have been linked with myopia in a recent linkage study. The authors investigated the roles of PAX6 and SOX2 in common myopia as part of a broader association study of refractive error. METHODS. Five hundred ninety-six persons from the 1958 British Birth Cohort, a nationally representative population, were randomly selected from the outer tertiles of the refractive error (RE) distribution and were genotyped using 25 tagSNPs across PAX6 and 3 tagSNPs across SOX2 and their putative control regions. This experiment had 80% power to exclude either gene contributing more than 10% of the variance of refractive error. RESULTS. All SNPs were in Hardy-Weinberg equilibrium, and the genotyping failure rate was less than 5%. Accounting for multiple testing, no significant association (P < 0.05) was found between any of the SNPs or haplotypes and refractive error. CONCLUSIONS. PAX6 and SOX2 are obvious candidates in RE genetic studies because of their biological roles and prior linkage studies. The present findings strongly suggest refractive error is not directly affected in this population by variants in either gene or by their known promoters/enhancers. The authors suggest that neither PAX6 nor SOX2 should be prioritized in the international search for genetic modifiers of refractive error. Their findings contribute to broader understanding of the pathophysiology of refractive error and highlight the critical role of replication in genetic research on complex disorders.

PURPOSE. Pediatric glaucoma is a rare, potentially blinding condition, yet, in the United Kingdom, there is a paucity of contemporary epidemiologic and clinical data regarding this condition. The British Infantile and Childhood Glaucoma (BIG) Eye Study is the first national population-based study conducted to examine the incidence, detection patterns, current management, and intraocular pressure (IOP) control at 1 year in children with newly diagnosed glaucoma in the United Kingdom. METHODS. A prospective study was conducted wherein children in the United Kingdom and Republic of Ireland aged 16 years with newly diagnosed primary or secondary glaucoma, were identified by consultant ophthalmologists through active surveillance from December 2001 until November 2002. Eligible cases were re-evaluated 12 months after notification. RESULTS. Of the 99 eligible children with newly diagnosed glaucoma, 47 had primary and 52 secondary glaucoma. The annual incidence of diagnosis of primary congenital glaucoma (PCG) in Great Britain was 5.41 in 100,000 (1/18,500) live births and in the Republic of Ireland, 3.31 in 100,000 (1/ 30,200). The incidence of PCG in children of Pakistani origin was almost nine times that of Caucasians. IOP control of 21 mm Hg was achieved in 94% with medications (60% without medications) in cases of PCG and in 86% with medications (28% without medications) in cases of secondary glaucoma. CONCLUSIONS. The British annual incidence of PCG diagnosis is comparable to that reported for other similar populations. Ethnic minorities from South Asia are at significantly increased risk of PCG. Successful IOP control in PCG after surgery in Britain is comparable to that in the published literature. (Invest Ophthalmol Vis Sci. 2007;48:4100‐4106) DOI:10.1167/

Understanding genetic and environmental factors that together contribute to the development of myopia is an international research priority. We have investigated the feasibility and accuracy of identifying and classifying refractive error, without formal refraction, as a means of easily identifying affected individuals in a large-scale, non-ophthalmological, and population-based survey. At age 44/45 years, members of the 1958 British birth cohort underwent a biomedical, community survey. Refractive error (autorefraction) was measured and categorised by spherical equivalent (SE) measurement; myopia (SE of −1.00 or more extreme), hypermetropia (+1.00 or more extreme), or emmetropia (−0.99 to +0.99). Lenses of prescribed distance glasses, if worn, were assessed as minifying, magnifying, or making no difference to a standard viewed image and cohort members reported on ‘short’ or ‘long’ sightedness. A total of 2499 cohort members, randomly selected, had formal refraction (autorefraction) and 917 (36.7%) of these individuals had their prescribed distance glasses examined. Sensitivities for myopia and hypermetropia using examination of glasses were over 80% and positive predictive values were 95 and 65% respectively whereas self-report of ‘short-sightedness’ or ‘long-sightedness’ had poor accuracy. We suggest examination of prescribed distance glasses can be an effective method of ‘screening’ for refractive error in the field, especially where prevalence is high.

A survey of a sample of UK paediatricians was carried out to identify the practices and determine the training of those involved in routine surveillance examinations to detect ophthalmic disorders in infants. The findings indicate important variation in current practices and raise concerns about both undergraduate and postgraduate training in ophthalmic assessment of infants.

Aim: To report the usefulness of uncorrected distance visual acuity (DVA) at 16 years to “screen” for myopia status and to assess the lifetime risk of myopia, based on a national birth cohort. Methods: 1867 members of the 1958 British birth cohort for whom there were data on acuity at 16 years had autorefraction, as part of a biomedical survey, at 45 years. Reduced uncorrected DVA at age 16 years (6/12 or worse in both eyes) was compared with adult refraction (spherical equivalent). Results: Only a quarter of individuals in the population studied who had developed myopia by 45 years of age had reduced acuity at 16 years of age. Notably, half of all adults with moderate myopia (−2.99 to −5.99) and 31% (11/35) with severe myopia (⩾−6) had good uncorrected DVA in both eyes at 16 years of age. Thus, sensitivities were low, ranging from 16% for all myopia (cut-off point spherical equivalent −0.5) to 69% for severe myopia (cut-off point spherical equivalent −6). However, a high (91%) lifetime probability of primary myopia (spherical equivalent ⩾−0.5) given a reduced uncorrected DVA at 16 years was found. Conclusion: In this population, reduced uncorrected DVA in childhood is an inaccurate and inappropriate intermediate “phenotype” for capturing adult myopia status. However, our findings support assessment of DVA in secondary school children as an effective method of identifying refractive error (both myopia and hypermetropia).

PURPOSE. To report long-term postoperative visual acuity in a nationally representative group of children with congenital/ infantile cataract and to investigate the factors associated with poor vision.METHODS. All children aged less than 16 years in the United Kingdom who had newly diagnosed congenital/infantile cataract in a 12-month period during 1995-1996 (the British Congenital/ infantile Cataract Study) were traced through their managing ophthalmologists. Outcome data were collected at least 6 years after diagnosis, by using specifically designed questionnaires. Ordinal regression analysis identified factors associated with postoperative acuity.RESULTS. Of 153 children who had surgery, complete data were available in 122 (85%). Median age at follow-up was 7 and 6.91 years, respectively, for bilateral and unilateral disease. Median age at surgery was 4.57 months in bilateral and 2.99 months in unilateral cases, with 40% and 45%, respectively, of children operated on by 3 months. Median (range) postoperative acuity was 6/ 18 (6/ 5, no perception of light) in bilateral and 6/60 (6/ 5, no perception of light) in unilateral disease. Poor compliance with occlusion was the factor most strongly associated with poorer acuity in both unilateral and bilateral disease: the odds of worse vision in unilateral cataract were 7.92 times greater with < 50% versus 100% compliance (95% CI 1.68-37.26). In bilateral disease, odds of worse vision were reduced with each month of decreasing age at surgery (0.98, 95% CI 0.94-0.99), but increased by the presence of additional medical conditions (3.53, 95% CI 1.08-11.44) and the presence of postoperative ocular complications (2.94, 95% CI 1.38-6.51).CONCLUSIONS. These findings support a secular improvement in postoperative acuity in bilateral, and to a lesser extent, unilateral disease. Nevertheless early detection of congenital cataract through effective newborn screening and improving concordance with occlusion both remain priorities. Further improvements in outcomes in unilateral disease are necessary before parents can be advised universally that treatment will achieve a functionally useful "spare" eye.

Background The British Paediatric Surveillance Unit (BPSU) was established in 1986 to facilitate national surveillance of uncommon paediatric disorders. This study investigated the effectiveness of using multiple source reporting and capture–recapture analysis to maximize case ascertainment in studies undertaken through the BPSU. Methods Structured review of all surveillance studies completed through the BPSU. Quantitative and qualitative analysis of the effectiveness of multiple reporting sources and capture–recapture methods was made. Results Of 71 studies undertaken through the BPSU, 59 were included in this review and 38 used additional data sources. Established national sources were most readily adapted for use as secondary data sources, including routine health data, communicable disease and specialty-specific surveillance units, whilst the involvement of parent groups and the media was less successful. Six studies employed capture–recapture techniques to estimate the completeness of case ascertainment. Conclusions Active surveillance through the BPSU remains a timely and reliable primary source of cases, but employing additional reporting sources is effective in enhancing case ascertainment. When the assumptions for its valid use are met, capture–recapture analysis allows the estimation of completeness of ascertainment. It is essential to define the purpose of an additional source at the outset of a study and to ensure that subsequent analysis is appropriate.

Objective To determine the association of amblyopia with diverse educational, health, and social outcomes in order to inform current debate about population screening for this condition.Design, setting, and participants Comparison of 8432 people with normal vision in each eye with 429 (4.8%) people with amblyopia (childhood Unilateral reduced acuity when tested with correction and unaccounted for by eye disease) from the 1958 British birth cohort, with respect to subsequent health and social functioning.Results No functionally or clinically significant differences between people with and without amblyopia in education outcomes, behavioural difficulties or social maladjustment, participation in social activities, Unintended injuries (school, workplace, or road traffic accidents as driver), general or mental health and mortality, paid employment, or occupation based social class trajectories.Conclusions It may be difficult to distinguish, at population level, between the lives of people with amblyopia and those without, in terms of several important outcomes. A pressing need exists for further concerted research on what it means to have amblyopia and, specifically, how this varies with severity and how it changes with treatment, so that screening programmes can best serve those who have the most to gain from early identification.

Dear Editor, In their recent article, Dawes et al. report prevalence estimates for visual impairment and dual sensory problems in middle age and associations between visual impairment and demographic factors, using data from UK Biobank. UK Biobank recruited half a million individuals with a subsample of 122 000 having an enhanced ophthalmic examination which included distance visual acuity and autorefraction. As the group within the UK Biobank Eyes and Vision Consortium undertaking investigation of vision function and refractive error, we have two areas of concern in relation to this paper. Firstly, as UK Biobank is not a population sample, it is inappropriate to cite an estimate of frequency as true population prevalence. There are few population based studies with which to compare these findings but in our study of a UK population sample (birth cohort) of subjects aged 44 years, the prevalence of habitual and best corrected distance visual acuity in those with low vision were 1.23% and 0.75% respectively, notably higher than equivalent estimates in this study (Supplemental table 1). Secondly, the statistical approaches used in the paper to obtain standardised frequency estimates used 2001 census data rather than the more appropriate 2011 census data. In addition, the comparative statistics in Table 2 should have compared the UK population and the subsample of the UK Biobank with ophthalmic data, rather than the entire UK Biobank population. More importantly, these approaches cannot address the issues of bias which have led to the low prevalence estimates of visual impairment in the study population. For example, there are a significant number of subjects excluded who were invited but did not have an acuity test as they were visually-impaired (self-reported reason for no test). Equally, there are a large number of individuals who were recorded as having reduced visual acuity who were known to have been tested without their habitual optical correction. Finally, the authors have assumed that the most common cause of visual impairment was uncorrected refractive error without confirming this using the refraction data available on subjects. We agree with the authors that the size and coverage of UK Biobank allow estimates of association between health outcomes and demographic and socio-economic factors to be generalizable to the wider population, but this requires the methodological issues we have outlined to be taken into account when interpreting or applying the findings of this paper.

Background Public health initiatives to prevent childhood blindness have emphasised the importance of effective intervention for congenital cataract. Primary intraocular lens implantation, an important recent management innovation, has been rapidly adopted despite the absence of robust supporting evidence. The IoLunder2 study aimed to provide much needed insights into visual and adverse outcomes after surgery with and without primary intraocular lens implantation in children aged under 2 years. Methods Active surveillance case identification of cataract surgery and systematic data collection were undertaken through the British Congenital Cataract Interest Group (comprising 173 ophthalmologists who manage affected children in the UK and Ireland), which identified more children undergoing surgery for cataract than were reported to the National Health Service statutory databases. Multivariable regression analysis was used to estimate associations of intraocular lens use with visual outcome (assessed with standard hospital-specific but harmonised protocols) and incidence of adverse events at 1 year after surgery, with adjustment for all relevant confounding factors (eg, ocular comorbidity or age at surgery), which were agreed a priori on the basis of previous research or biological plausibility. All reported odds ratios (ORs) are fully adjusted. Findings Of 221 children, 56 of 131 with bilateral cataract and 48 of 90 with unilateral cataract had primary intraocular lens implantation. Implantation was independently associated with better visual outcome in bilateral (OR 5·4, 95% CI 1·09–26·4, p=0·04) but not unilateral disease, and with increased odds of reoperation (bilateral OR 5·5, 95% CI 2·3–13·2, p=0·002; unilateral 16·7, 4·1–68·9, p=0·009). Intraocular lens use did not reduce the odds of postoperative glaucoma, the key sight-threatening complication, after surgery in bilateral or unilateral cataract. Interpretation Use of intraocular lenses in young children, particularly in settings where follow-up is limited, should be critically reassessed. The absence of visual benefit and the lack of a previously postulated protective effect against postoperative glaucoma question the value of intraocular lens implantation in unilateral disease. The association between intraocular lens implantation and better early visual outcomes in bilateral disease needs to be balanced against the risk of reoperation and exposure to general anaesthetics during a key neurodevelopmental period. Funding Ulverscroft Vision Research Group, Great Ormond Street Hospital/UCL Institute of Child Health National Institute for Health Research Biomedical Research Centre, and Moorfields Eye Hospital NHS Foundation Trust/UCL Institute of Ophthalmology National Institute for Health Research Biomedical Research Centre.

A global problem requiring more research measuring outcomes that matter to children and their parents

Aims: To investigate whether there is an association between congenital colour vision defects (CVD) and occupational choice and employment history, in order to inform the debate about the value of universal childhood screening for these disorders. Methods: Participants were 6422 males and 6112 females from the 1958 British birth cohort, followed from birth to 33 years, whose colour vision was assessed (Ishihara test) at 11 years. Results: A total of 431 males (6.7%) had CVD. Men with CVD had pursued some careers for which normal colour vision is currently regarded as essential; for example, eight men (3.1%) with CVD were in the police, armed forces, or fire-fighting service at 33 years compared to 141 men (3.8%) with normal colour vision. They were, however, under-represented compared to those with normal colour vision, in other occupations; for example, no men with CVD were employed in electrical and electronic engineering at 33 years compared to 15 men (0.4%) with normal colour vision. Conclusions: The findings of this study suggest there is little to be gained by continuing with existing school screening programmes for CVD, whose primary purpose is to advise affected children against certain careers. Other ways of informing young people about potential occupational difficulties and pathways for referral for specialist assessment are likely to be more useful.

Objective. Key worker programs for families of children with disabilities, to promote information provision, emotional support, and liaisons among different agencies, have long been advocated but not extensively implemented. We report the impact on the experiences of parents and the practices of health care professionals of a novel, hospital-based, key worker service (Community Link Team [CLT]), implemented in the pediatric ophthalmology department of Great Ormond Street Hospital (London, United Kingdom). Design, Setting, and Participants. The CLT included 2 members, 1 of whom was present during the first outpatient assessment by the consultant ophthalmologist of any child newly diagnosed as visually impaired (corrected acuity of 6/18 or worse in the better eye) and accompanied the family during other assessments performed during that visit. A dedicated room was used by the CLT members to spend time with each family after completion of the clinical assessments. The CLT members reiterated and/or clarified clinical information already provided, specifically advised the families about visual stimulation programs and the benefits and purpose of visual impairment certification, and provided information about educational and social services. The same CLT member met the family at subsequent visits to the department and acted as the first point of contact for parents. Parents of children newly diagnosed with visual impairment and/or ophthalmic disorders at Great Ormond Street Hospital participated in a 2-stage study to assess their needs, their views about the processes of care, and their overall satisfaction. The study included a questionnaire survey with 2 standard instruments, ie, the Measure of Processes of Care, specifically developed and used to assess parents’ views of the degree to which health services for a range of childhood disorders are family-centered, and the short form of the Client Satisfaction Questionnaire, used to assess overall parental satisfaction or dissatisfaction with services in the preceding year, as in other studies of parental satisfaction with pediatric services. This was followed by in-depth individual interviews with a subsample of parents who returned completed questionnaires. The views of families with experience with the new service (CLT) were compared with those without. The experiences of health care professionals before and after implementation of the service were elicited through group interviews and were compared. We recognized that any differences would be attributable to both the direct effects of the CLT, ie, actual services provided by the team, and indirect effects, ie, broader changes in approaches or practices within the department resulting from shifting roles and responsibilities regarding specific elements of management. Therefore, both the specific tasks/activities undertaken by the CLT and broader changes in practices within the department were identified. Results. Seventy-nine families from the pre-CLT group and 68 from the post-CLT group (68% and 65% of those invited, respectively) participated in the questionnaire survey, of which 29 and 19 (71% and 79% of those invited), respectively, took part in interviews. The 2 groups were comparable with respect to sociodemographic and clinical characteristics. Parents and health care professionals agreed that the CLT provided important information and facilitated access to specific services, while providing both emotional and social support and facilitating meetings with other families with children with similar conditions. A number of key generic components of the service were identified. First, provision, within the outpatient setting, of a dedicated “quiet room” and office space for key workers was an essential physical requirement. Second, early identification of the key workers as the parents’ point of contact was essential; this was achieved in this case by the CLT members attending the first consultation, combined with their detailed debriefing of families at the end of the outpatient visit. Third, the adoption of certain tasks by the key workers, including some previously undertaken by ophthalmologists, helped to define the liaison role of the program. These tasks included discussing the process and benefits of visual impairment certification, contacting the advisory teacher for the visually impaired, and providing written reports to educational and social services; analogous tasks would exist for other disabilities. Conclusions. Research on the needs of families of visually impaired children has been limited but indicates that, as with other childhood disabilities, the greatest needs during the critical period around diagnosis are for information, especially about educational and social services, and emotional support from professionals, informal and formal social networks, and support groups. Although not widely implemented or studied, key worker programs for families of visually impaired children, particularly in the context of multidisciplinary visual impairment teams, have been advocated, on the basis of their potential to facilitate coordination of health, educational, and social services. The model of such provision evaluated in this study reflects the fact that it was established as an outpatient service in a tertiary referral center for pediatric ophthalmology in the United Kingdom, with the specific structure and specialized roles for health care professionals that this requires. Different models might be more suitable in other settings in the United Kingdom or elsewhere. However, the important general lessons learned should guide implementation of such services for families of children with other disabilities. The recently launched National Service Framework for Children provides a new context and standards for meeting the needs of disabled children and their families in the United Kingdom and may also guide initiatives elsewhere. The findings of this study support implementation of programs for information provision, support, and liaison by key workers in all specialized centers for the assessment and diagnosis of children with serious visual problems. Implementation of similar services for families with children with other disabilities is likely to be equally valuable.

Aim: To investigate the barriers to, and degree and nature of bias in, participation in health services research by parents of children with visual impairment. Methods: Parents of children newly diagnosed with ophthalmic disorders at Great Ormond Street Hospital, London, participated in a study to elicit their health service experiences and needs through a postal questionnaire survey followed by in-depth interviews. The participating and non-participating families were compared at different stages of recruitment, according to sociodemographic and clinical characteristics. Results: 20% (55) of all eligible families could not be invited to participate because of out of date contact details for either the family and/or family doctor in the hospital and/or community record systems. Completed questionnaires were received from 67% (147/221) of contacted families, although only 6% actively declined to take part. Compared to non-participating parents, those who took part were more likely to be white British, from higher socioeconomic groups, have English as their main language, and have no other visually impaired family members. There were no significant differences according to the clinical characteristics of their affected children. Conclusions: Families from socioeconomically deprived and ethnic minority groups are likely to be less visible than others in health services research on childhood visual impairment. Geographical mobility in families of young children with visual disability poses a potentially important obstacle to engaging them in research on their experiences of health services. These findings indicate the importance of addressing potential biases in the design and interpretation of future studies, to ensure equity in recommendations for policy and practice, and in implementation of services.

Purpose. Parents' input is critical to clinical management in pediatric ophthalmology. The importance of providing parents with appropriate information to enable them to participate effectively is recognized. However, little is known about the range of sources parents use to learn about their child's ophthalmic condition, which sources they find most useful, and how this relates to their understanding.Methods. Cross-sectional survey of the parents or usual care-givers of children with diverse oplithalmic disorders, diagnosed at least 1 year earlier, who attended pediatric ophthalmology clinics at Great Ormond Street Hospital, London, during 1 week in August 2001.Results. Eighty-nine percent (n = 58) of parents with eligible children participated. Most parents received information from more than one source, with ophthalmologists (79%) and family practitioners (42%) being the two most frequently reported. Family support groups and voluntary organizations (29%) and the Internet (23%) were less commonly cited than anticipated. Parents reported receiving verbal information much more frequently than written information from professionals working with their children. Although 72% (n = 42) of parents could correctly name their child's diagnosis, only 46% (n = 27) were able to describe correctly the nature and impact of the disorder(s). Ophthalmologists were ranked as the most important source overall.Conclusions. The findings emphasize the key role of ophthalmic professionals in improving parental education directly, as well their responsibilities and opportunities to do so through supporting and shaping information provision through other sources, especially colleagues in primary care and the Internet. (Invest Ophthalmol Vis Sci. 2003;44:2457-2460) DOI: 10.1167/iovs.02-1184.

Amblyopia arises from abnormal visual experiences in early childhood. Improved function of the amblyopic eye after visual loss in the non-amblyopic eye could be a model for residual neural plasticity. We aimed to establish the likelihood of, and predictive factors for, this improvement in function. We identified 254 individuals aged 11 years or older with unilateral amblyopia who were visually impaired after loss of vision in their non-amblyopic eye but had no other disorder affecting their amblyopic eye. 25 (10%) of 254 people had improved visual acuity in their amblyopic eye. These findings suggest there is some plasticity in the visual system of a few visually mature individuals with amblyopia, which warrants further study. Children should remain the focus of detection and treatment.