Publications by authors named "Huibert J Simonsz"

38 Publications

Scope and costs of autorefraction and photoscreening for childhood amblyopia-a systematic narrative review in relation to the EUSCREEN project data.

Eye (Lond) 2021 Mar 30;35(3):739-752. Epub 2020 Nov 30.

Erasmus Medical Centre, Rotterdam, The Netherlands.

Background: Amblyopia screening can target reduced visual acuity (VA), its refractive risk factors, or both. VA testing is imprecise under 4 years of age, so automated risk-factor photoscreening appears an attractive option. This review considers photoscreening used in community services, focusing on costs, cost-effectiveness and scope of use, compared with EUSCREEN project Country Reports describing how photo- and automated screening is used internationally.

Methods: A systematic narrative review was carried out of all English language photoscreening literature to September 10th 2018, using publicly available search terms. Where costs were considered, a CASP economic evaluation checklist was used to assess data quality.

Results: Of 370 abstracts reviewed, 55 reported large-scale community photoscreening projects. Five addressed cost-effectiveness specifically, without original data. Photoscreening was a stand-alone, single, test event in 71% of projects. In contrast, 25 of 45 EUSCREEN Country Reports showed that if adopted, photoscreening often supplements other tests in established programmes and is rarely used as a stand-alone test. Reported costs varied widely and evidence of cost-effectiveness was sparse in the literature, or in international practice. Only eight (13%) papers compared the diagnostic accuracy or cost-effectiveness of photoscreening and VA testing, and when they did, cost-effectiveness of photoscreening compared unfavourably.

Discussion: Evidence that photoscreening reduces amblyopia or strabismus prevalence or improves overall outcomes is weak, as is evidence of cost-effectiveness, compared to later VA screening. Currently, the most cost-effective option seems to be a later, expert VA screening with the opportunity for a re-test before referral.
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http://dx.doi.org/10.1038/s41433-020-01261-8DOI Listing
March 2021

Increase in treatment of retinopathy of prematurity in the Netherlands from 2010 to 2017.

Acta Ophthalmol 2021 Feb 23;99(1):97-103. Epub 2020 Jul 23.

Leiden University Medical Center, Leiden, Netherlands.

Purpose: Compare patients treated for Retinopathy of Prematurity (ROP) in two consecutive periods.

Methods: Retrospective inventory of anonymized neonatal and ophthalmological data of all patients treated for ROP from 2010 to 2017 in the Netherlands, subdivided in period (P)1: 1-1-2010 to 31-3-2013 and P2: 1-4-2013 to 31-12-2016. Treatment characteristics, adherence to early treatment for ROP (ETROP) criteria, outcome of treatment and changes in neonatal parameters and policy of care were compared.

Results: Overall 196 infants were included, 57 infants (113 eyes) in P1 and 139 (275 eyes) in P2, indicating a 2.1-fold increase in ROP treatment. No differences were found in mean gestational age (GA) (25.9 ± 1.7 versus 26.0 ± 1.7 weeks, p = 0.711), mean birth weight (791 ± 311 versus 764 ± 204 grams, p = 0.967) and other neonatal risk factors for ROP. In P2, the number of premature infants born <25 weeks increased by factor 1.23 and higher oxygen saturation levels were aimed at in most centres. At treatment decision, 59.6% (P1) versus 83.5% (P2) (p = 0.263) infants were classified as Type 1 ROP (ETROP classification). Infants were treated with laser photocoagulation (98 versus 96%) and intravitreal bevacizumab (2 versus 4%). Retreatment was necessary in 10 versus 21 (p = 0.160). Retinal detachment developed in 6 versus 13 infants (p = 0.791) of which 2 versus 6 bilateral (p = 0.599).

Conclusion: In period 2, the number of infants treated according to the ETROP criteria (Type 1) increased, the number of ROP treatments, retinal detachments and retreatments doubled and the absolute number of retinal detachments increased. Neonatal data did not provide a decisive explanation, although changes in neonatal policy were reported.
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http://dx.doi.org/10.1111/aos.14501DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7891652PMC
February 2021

Long-term follow-up of an amblyopia treatment study: change in visual acuity 15 years after occlusion therapy.

Acta Ophthalmol 2021 Feb 13;99(1):e36-e42. Epub 2020 Jul 13.

Department of Ophthalmology, Erasmus University Medical Center, Rotterdam, The Netherlands.

Purpose: To determine change in visual acuity (VA) in the population of a previous amblyopia treatment study (Loudon 2006) and assess risk factors for VA decrease.

Methods: Subjects treated between 2001 and 2003 were contacted between December 2015 and July 2017. Orthoptic examination was conducted under controlled circumstances and included subjective refraction, best corrected VA, reading acuity, binocular vision, retinal fixation, cover-uncover and alternating cover test. As a measure for degree of amblyopia, InterOcular VA Difference (IOD) at the end of occlusion therapy was compared with IOD at the follow-up examination using Wilcoxon's signed-rank test. Regression analysis was conducted to determine the influence of clinical and socio-economic factors on changes in IOD.

Results: Out of 303 subjects from the original study, 208 were contacted successfully, 59 refused and 15 were excluded because of non-amblyopic cause of visual impairment. Mean IOD at end of therapy (mean age 6.4 years) was 0.11 ± 0.16 logMAR, and IOD at follow-up examination (mean age 18.3 years) was 0.09 ± 0.21 logMAR; this difference was not significant (p = 0.054). Degree of anisometropia (p = 0.008; univariable analysis), increasing anisometropia (p = 0.009; multivariable), eccentric fixation (p < 0.001; univariable and multivariable); large IOD (p < 0.001; univariable and multivariable) and non-compliance during therapy (p = 0.028; univariable) were associated with IOD increase.

Conclusion: Long-term results of occlusion therapy were good. High or increasing anisometropia, eccentric fixation and non-compliance during occlusion therapy were associated with long-term VA decrease. Subjects with poor initial VA had a larger increase despite little patching, but often showed long-term VA decrease.
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http://dx.doi.org/10.1111/aos.14499DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7891344PMC
February 2021

Endophthalmitis after strabismus surgery: incidence and outcome in relation to age, operated eye muscle, surgical technique, scleral perforation and immune state.

Acta Ophthalmol 2021 Feb 13;99(1):37-51. Epub 2020 Jun 13.

University Eye Clinic, Freiburg, Germany.

Purpose: Identify risk factors for endophthalmitis after strabismus surgery (EASS) and relate these to incidence and outcome.

Methods: Ophthalmologists, who had operated, diagnosed or treated EASS, completed a case record form with 71 questions in six domains: Preoperative, Surgery, Perforation, Postoperative, Outcome and Experts' opinion. To estimate the age-specific incidence per number of strabismus operations in the Netherlands during 1994-2013, the age distribution of Dutch cases was compared with the age-specific rates of strabismus surgery in the Dutch Registry of Strabismus Operations and with population data. Exploratory data analysis was performed. The immune state was evaluated in six patients. Five enucleated eyes were studied histopathologically.

Results: None of the 26 patients (27 eyes with EASS) were between 9 and 65 years old, except for one patient with retinal haemorrhage followed by endophthalmitis. In the Netherlands during 1994-2013, the rate of EASS was approximately one per 11 000 strabismus operations, but one per 4300 for children aged 0-3 and one per 1000 for patients 65 and older. Endophthalmitis was diagnosed on postoperative day 1-4 in children aged 0-3. In all 15 children aged 0-5, the 16 affected eyes were phthisical, eviscerated or enucleated. The involved eye muscle had been recessed in 25 of 27 cases. It was a medial rectus in 15 of 16 children aged 0-6. It was a lateral (6), inferior (2) or medial (1) rectus in elderly. Scleral perforation went unnoticed in all children (no record in three) and in two of seven elderly (no record in two). Histopathology showed transscleral scarring compatible with scleral perforation in four patients but, in a two-year-old girl who had EASS together with a transient medial rectus palsy, the sclera underneath the former suture tract was not perforated but did contain the long posterior ciliary artery.

Conclusions: Endophthalmitis after strabismus surgery (EASS) affects children and elderly, with a grave outcome in young children. It occurs after recession of the medial rectus muscle in children, and it may occur without scleral perforation. Age and perforation are key determinants that interact with other factors that determine the occurrence and fulminance of EASS.
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http://dx.doi.org/10.1111/aos.14446DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7891599PMC
February 2021

Implementation of a neonatal hearing screening programme in three provinces in Albania.

Int J Pediatr Otorhinolaryngol 2020 Jul 8;134:110039. Epub 2020 Apr 8.

Erasmus University Medical Center Rotterdam, Department of Ophthalmology, Rotterdam, the Netherlands.

Objectives: The EUSCREEN study compares the cost-effectiveness of paediatric hearing screening programmes and aims to develop a cost-effectiveness model for this purpose. Alongside and informed by the development of the model, neonatal hearing screening (NHS) is implemented in Albania. We report on the first year.

Methods: An implementation plan was made addressing objectives, target population, screening protocol, screener training, screening devices, care pathways and follow up. NHS started January 1st, 2018 in four maternity hospitals: two in Tirana, one in Pogradec and one in Kukës, representing both urban and rural areas. OAE-OAE-aABR was used to screen well infants in maternity hospitals, whereas aABR-aABR was used in neonatal intensive care units and in mountainous Kukës for all infants. Screeners' uptake and attitudes towards screening and quality of screening were assessed by distributing questionnaires and visiting the maternity hospitals. The result of screening, diagnostics, follow up and entry into early intervention were registered in a database and monitored.

Results: Screeners were keen to improve their skills in screening and considered NHS valuable for Albanian health care. The number of "fail" outcomes after the first screen was high initially but decreased to less than 10% after eight months. In 2018, 11,507 infants were born in the four participating maternity hospitals, 10,925 (94.9%) of whom were screened in the first step. For 486 infants the result of screening was not registered. For the first screen, ten parents declined, eight infants died and one infant was discharged before screening could be performed. In 1115 (10.2%) infants the test either could not be performed or the threshold was not reached; 361 (32,4%) of these did not attend the second screen. For the third screen 31 (34.4%) out of 90 did not attend. Reasons given were: parents declined (124), lived too far from screening location (95), their infant died (11), had other health issues (7), or was screened in private clinic (17), no reason given (138).

Conclusions: Implementation of NHS in Albania is feasible despite continuing challenges. Acceptance was high for the first screen. However, 32.4% of 1115 infants did not attend the second screen, after a "fail" outcome for the first test.
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http://dx.doi.org/10.1016/j.ijporl.2020.110039DOI Listing
July 2020

Prevalence of permanent neonatal hearing impairment: systematic review and Bayesian meta-analysis.

Int J Audiol 2020 06 3;59(6):475-485. Epub 2020 Feb 3.

Department of Otorhinolaryngology and Head and Neck Surgery and Department of Ophthalmology, Erasmus University Medical Center, Rotterdam, The Netherlands.

To investigate the variance in reported prevalence rates of permanent neonatal hearing impairment (HI) worldwide. A systematic review and meta-analysis was performed on reported prevalence rates of sensorineural and permanent conductive or mixed HI worse than 40 dB in neonates, detected as a result of a screening programme or audiometric study. For meta-analysis, 35 articles were selected, 25 from high-income countries and 10 from middle-income countries according to the world bank classification system. The prevalence rate of permanent uni- and bilateral HI worse than 40 dB in neonates varied from 1 to 6 per 1000, the overall prevalence was 2.21 per 1000 [1.71, 2.8]. In NICU populations the prevalence rate was higher with a larger fraction of bilateral cases. Although not significant, prevalence rates were slightly higher in Asia compared to Europe and the number of infants lost to follow-up appeared higher in countries with lower gross national income. Substantial variations exist in prevalence rates of neonatal permanent HI across countries and regions. There is a strong need for more data from low-income countries to identify demographic factors that account for this variability in reported prevalence rates. Reporting these data in a uniform way is advocated.
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http://dx.doi.org/10.1080/14992027.2020.1716087DOI Listing
June 2020

Nystagmus in patients with congenital stationary night blindness (CSNB) originates from synchronously firing retinal ganglion cells.

PLoS Biol 2019 09 12;17(9):e3000174. Epub 2019 Sep 12.

Netherlands Institute for Neuroscience, Amsterdam, the Netherlands.

Congenital nystagmus, involuntary oscillating small eye movements, is commonly thought to originate from aberrant interactions between brainstem nuclei and foveal cortical pathways. Here, we investigated whether nystagmus associated with congenital stationary night blindness (CSNB) results from primary deficits in the retina. We found that CSNB patients as well as an animal model (nob mice), both of which lacked functional nyctalopin protein (NYX, nyx) in ON bipolar cells (BCs) at their synapse with photoreceptors, showed oscillating eye movements at a frequency of 4-7 Hz. nob ON direction-selective ganglion cells (DSGCs), which detect global motion and project to the accessory optic system (AOS), oscillated with the same frequency as their eyes. In the dark, individual ganglion cells (GCs) oscillated asynchronously, but their oscillations became synchronized by light stimulation. Likewise, both patient and nob mice oscillating eye movements were only present in the light when contrast was present. Retinal pharmacological and genetic manipulations that blocked nob GC oscillations also eliminated their oscillating eye movements, and retinal pharmacological manipulations that reduced the oscillation frequency of nob GCs also reduced the oscillation frequency of their eye movements. We conclude that, in nob mice, synchronized oscillations of retinal GCs, most likely the ON-DCGCs, cause nystagmus with properties similar to those associated with CSNB in humans. These results show that the nob mouse is the first animal model for a form of congenital nystagmus, paving the way for development of therapeutic strategies.
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http://dx.doi.org/10.1371/journal.pbio.3000174DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6741852PMC
September 2019

Plusoptix photoscreener use for paediatric vision screening in Flanders and Iran.

Acta Ophthalmol 2020 Feb 14;98(1):80-88. Epub 2019 Jun 14.

Ophthalmology, Erasmus Medical Center, Rotterdam, the Netherlands.

Purpose: Photoscreening assesses risk factors for amblyopia, as an alternative to measurement of visual acuity (VA) to detect amblyopia, on the premise that its early correction could prevent development of amblyopia. We studied implementations of Plusoptix photoscreening in existing population-based screening in Flanders and Iran.

Methods: In Flanders, VA is measured at age 3, 4 and 6, photoscreening was added to existing screening at age 1 and 2.5 years in 2013. In Iran, VA is measured at ages 3-6 years, photoscreening was added at ages 3-6 years between 2011 and 2016. Plusoptix use was analysed in the literature for detection of risk factors for amblyopia and amblyopia itself, for ages 0-3 and for 4-6. A questionnaire, containing seven domains: existing vision screening, addition of photoscreening, implementation in screening program, training, attendance, diagnosis and treatment, and costs was distributed. In Iran, screening procedures were observed on site.

Results: Implementation of Plusoptix photoscreening was mainly analysed from questionnaires and interviews, its effectiveness from literature data. In Flanders, of 56 759 children photoscreened at age one (81% of children born in 2013), 9.2% had been referred, 13% of these were treated, mostly with glasses, resulting in an increase of 4-year-old children wearing glasses from 4.7% to 6.4%. In Iran, 90% of children aged 3-6 years participated in vision screening in 2016, but only those who failed the vision test were subjected to photoscreening.

Conclusions: In Flanders, the use of Plusoptix photoscreening at ages 1 and 2.5 resulted in an increase of children wearing glasses, but it remains unknown how many cases of amblyopia have been prevented. Studies are needed to determine the relation between size and sort of refractive error and strabismus, and the increased chance to develop amblyopia.
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http://dx.doi.org/10.1111/aos.14144DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7003743PMC
February 2020

High rate of failed visual-acuity measurements with the Amsterdam Picture Chart in screening at the age of 36 months.

Acta Ophthalmol 2019 Feb 3;97(1):24-28. Epub 2018 Oct 3.

Ophthalmology, Erasmus Medical Center, Rotterdam, the Netherlands.

Purpose: In the Netherlands, youth health care physicians and nurses screen all children for general health disorders at Child Health Care Centers. As part of this, the eyes are screened seven times, with the first visual acuity (VA) measurement at 36 months with the Amsterdam Picture Chart (APK). The suitability of the APK has been questioned.

Methods: Children born between July 2011 and June 2012 born in the provinces Drenthe, Gelderland and Flevoland and invited for screening at 36 months were eligible. Parents were sent the APK picture optotypes to practise with their children in advance. Data were collected from electronic screening records. The Dutch vision screening guideline prescribes that children with VA <5/6, or one line interocular difference (not logMAR, however) should be retested or referred.

Results: Of 10 809 eligible children, 1546 did not attend and 602 attended but had no VA measurement at age 36 months, 247 of these were under orthoptic treatment. Of the 8448 children examined, VA was sufficient in 5663 (67.0%) and insufficient in 1312 (15.5%). In 1400 (16.6%), the measurement of VA itself failed. In 73 (0.9%), data were missing. Of the 216 children with 2 failed VA measurements, 150 (69%) were not referred, and measurement of VA was deferred to the next general screening examination at 45 months.

Conclusion: Although most parents had practised the APK picture optotypes at home with their children, the rate of failed APK measurements plus the measurements with insufficient VA was 32.1% at 36 months. Similar rates have previously been reported for Lea Symbols and HOTV, permitting the conclusion that measurement of VA at the age of 36 months cannot be recommended as a screening test in the general population.
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http://dx.doi.org/10.1111/aos.13898DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6667895PMC
February 2019

Differences in quality-of-life dimensions of Adult Strabismus Quality of Life and Amblyopia & Strabismus Questionnaires.

Graefes Arch Clin Exp Ophthalmol 2017 Sep 29;255(9):1851-1858. Epub 2017 May 29.

Department of Social Sciences, University of Leiden, Leiden, The Netherlands.

Purpose: The Adult Strabismus Quality of Life Questionnaire (AS-20) and the Amblyopia & Strabismus Questionnaire (A&SQ) both measure health-related quality of life in strabismus patients. We evaluated to what extent these instruments cover similar domains by identifying the underlying quality-of-life factors of the combined questionnaires.

Methods: Participants were adults from a historic cohort with available orthoptic childhood data documenting strabismus and/or amblyopia. They had previously completed the A&SQ and were now asked to complete the AS-20. Factor analysis was performed on the correlation-matrix of the combined AS-20 and A&SQ data to identify common underlying factors. The identified factors were correlated with the clinical variables of angle of strabismus, degree of binocular vision, and visual acuity of the worse eye.

Results: One hundred ten patients completed both questionnaires (mean age, 44 years; range, 38-51 years). Six factors were found that together explained 78% of the total variance. The factor structure was dominated by the first four factors. One factor contained psychosocial and social-contact items, and another factor depth-perception items from both questionnaires. A third factor contained seven items-only from the AS-20-on eye strain, stress, and difficulties with reading and with concentrating. A fourth factor contained seven items-only from the A&SQ-on fear of losing the better eye and visual disorientation, specific for amblyopia. Current visual acuity of the worse eye correlated with depth-perception items and vision-related items, whereas current binocular vision correlated with psychosocial and social-contact items, in 93 patients.

Conclusions: Factor analysis suggests that the AS-20 and A&SQ measure a similar psychosocial quality-of-life domain. However, functional problems like avoidance of reading, difficulty in concentrating, eye stress, reading problems, inability to enjoy hobbies, and need for frequent breaks when reading are represented only in the AS-20. During the development of the A&SQ, asthenopia items were considered insufficiently specific for strabismus and were excluded a priori. The patients who generated the items for the AS-20 had, in majority, adulthood-onset strabismus and diplopia and were, hence, more likely to develop such complaints than our adult patients with childhood-onset strabismus and/or amblyopia.
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http://dx.doi.org/10.1007/s00417-017-3694-xDOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5554281PMC
September 2017

Visualization of Sliding and Deformation of Orbital Fat During Eye Rotation.

Transl Vis Sci Technol 2016 Jul 29;5(4). Epub 2016 Jul 29.

Department of Ophthalmology Erasmus Medical Center, Rotterdam, the Netherlands.

Purpose: Little is known about the way orbital fat slides and/or deforms during eye movements. We compared two deformation algorithms from a sequence of MRI volumes to visualize this complex behavior.

Methods: Time-dependent deformation data were derived from motion-MRI volumes using Lucas and Kanade Optical Flow (LK3D) and nonrigid registration (B-splines) deformation algorithms. We compared how these two algorithms performed regarding sliding and deformation in three critical areas: the sclera-fat interface, how the optic nerve moves through the fat, and how the fat is squeezed out under the tendon of a relaxing rectus muscle. The efficacy was validated using identified tissue markers such as the lens and blood vessels in the fat.

Results: Fat immediately behind the eye followed eye rotation by approximately one-half. This was best visualized using the B-splines technique as it showed less ripping of tissue and less distortion. Orbital fat flowed around the optic nerve during eye rotation. In this case, LK3D provided better visualization as it allowed orbital fat tissue to split. The resolution was insufficient to visualize fat being squeezed out between tendon and sclera.

Conclusion: B-splines performs better in tracking structures such as the lens, while LK3D allows fat tissue to split as should happen as the optic nerve slides through the fat. Orbital fat follows eye rotation by one-half and flows around the optic nerve during eye rotation.

Translational Relevance: Visualizing orbital fat deformation and sliding offers the opportunity to accurately locate a region of cicatrization and permit an individualized surgical plan.
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http://dx.doi.org/10.1167/tvst.5.4.9DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4981490PMC
July 2016

Patient-reported utilities in bilateral visual impairment from amblyopia and age-related macular degeneration.

BMC Ophthalmol 2016 May 17;16(1):56. Epub 2016 May 17.

Department of Ophthalmology, Erasmus MC, University Medical Center Rotterdam, PO Box 2040, NL-3000 CA, Rotterdam, The Netherlands.

Background: Utility of visual impairment caused by amblyopia is important for the cost-effectiveness of screening for amblyopia (lazy eye, prevalence 3-3.5 %). We previously measured decrease of utility in 35-year-old persons with unilateral persistent amblyopia. The current observational case-control study aimed to measure loss of utility in patients with amblyopia with recent decrease of vision in their better eye. As these patients are rare, the sample was supplemented by patients with bilateral age-related macular degeneration with similar decrease of vision.

Methods: From our out-patient department, two groups of patients with recent deterioration to bilateral visual acuity less than Snellen 0.5 (bilateral visual impairment, BVI) were recruited, with either persistent amblyopia and age-related macular degeneration (AMB + AMD), or with bilateral age-related macular degeneration (BAMD). To measure utility, the time trade-off method and the standard gamble method were applied through interviews. Correlations were sought between utility values and visual acuity, age and Visual Function Questionnaire-25 scores.

Results: Seventeen AMB + AMD patients (mean age 72.9 years), and 63 BAMD patients (mean age 79.6 years) were included in the study. Among AMB + AMD, 80 % were willing to trade lifetime in exchange for cure. The overall mean time trade-off utility was 0.925. Among BAMD, 75 % were willing to trade, utility was 0.917. Among AMB + AMD, 38 % accepted risk of death in exchange for cure, overall mean standard gamble utility was 0.999. Among BAMD, 49 % accepted risk of death, utility was 0.998. Utility was not related to visual acuity but it was to age (p = 0.02).

Conclusion: Elderly patients with BVI, caused by persistent amblyopia and age-related macular degeneration (AMD) or by bilateral AMD, had an approximately 8 % loss of TTO utility. Notably, the 8 % loss in elderly with BVI differs little from the 3.7 % loss we found previously in 35-year-old persons with unilateral amblyopia with good vision in the other eye. The moderate impact of BVI in senescence could be explained by adaptation, comorbidity, avoidance of risk and a changed percept of cure.
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http://dx.doi.org/10.1186/s12886-016-0234-0DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4869400PMC
May 2016

Concerning manuscript "Compliance and patching and atropine amblyopia treatments" by Jingyun Wang.

Vision Res 2015 Oct 1;115(Pt A):151-2. Epub 2015 Sep 1.

Department of Ophthalmology, Erasmus MC, University Medical Center Rotterdam, Rotterdam, The Netherlands.

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http://dx.doi.org/10.1016/j.visres.2015.08.002DOI Listing
October 2015

Inventory of current EU paediatric vision and hearing screening programmes.

J Med Screen 2015 Jun 5;22(2):55-64. Epub 2015 Mar 5.

Department of Ophthalmology, Erasmus University Medical Center Rotterdam, the Netherlands.

Objective: To examine the diversity in paediatric vision and hearing screening programmes in Europe.

Methods: Themes for comparison of screening programmes derived from literature were used to compile three questionnaires on vision, hearing, and public health screening. Tests used, professions involved, age, and frequency of testing seem to influence sensitivity, specificity, and costs most. Questionnaires were sent to ophthalmologists, orthoptists, otolaryngologists, and audiologists involved in paediatric screening in all EU full-member, candidate, and associate states. Answers were cross-checked.

Results: Thirty-nine countries participated; 35 have a vision screening programme, 33 a nation-wide neonatal hearing screening programme. Visual acuity (VA) is measured in 35 countries, in 71% of these more than once. First measurement of VA varies from three to seven years of age, but is usually before age five. At age three and four, picture charts, including Lea Hyvarinen, are used most; in children over four, Tumbling-E and Snellen. As first hearing screening test, otoacoustic emission is used most in healthy neonates, and auditory brainstem response in premature newborns. The majority of hearing testing programmes are staged; children are referred after 1-4 abnormal tests. Vision screening is performed mostly by paediatricians, ophthalmologists, or nurses. Funding is mostly by health insurance or state. Coverage was reported as >95% in half of countries, but reporting was often not first-hand.

Conclusion: Largest differences were found in VA charts used (12), professions involved in vision screening (10), number of hearing screening tests before referral (1-4), and funding sources (8).
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http://dx.doi.org/10.1177/0969141315572403DOI Listing
June 2015

Blinded by shiga toxin-producing O104 Escherichia coli and hemolytic uremic syndrome.

J Pediatr 2014 Aug 17;165(2):410-410.e1. Epub 2014 May 17.

Department of Hematology, Erasmus MC University Medical Center, Rotterdam, The Netherlands.

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http://dx.doi.org/10.1016/j.jpeds.2014.04.008DOI Listing
August 2014

Nationwide inventory of risk factors for retinopathy of prematurity in the Netherlands.

J Pediatr 2014 Mar 19;164(3):494-498.e1. Epub 2013 Dec 19.

Department of Ophthalmology, Leiden University Medical Center, Leiden, The Netherlands.

Objectives: To study the incidence and risk factors for retinopathy of prematurity (ROP) in the Netherlands.

Study Design: Prospective, approximating population-based study that included infants with gestational age (GA) <32 weeks and/or birth weight (BW) <1500 g born in 2009. Pediatricians and ophthalmologists of all hospitals involved in care for premature infants reported data that were matched with the national perinatal database for risk factor analysis.

Results: Of 1380 infants, median GA 29.8 weeks (IQR 28.1-31.1) and median BW 1260 g (IQR 1020-1500), ROP developed in 21.9%. Logistic regression identified GA and BW as risk factors for ROP (P < .001). After adjustment for GA and BW, additional risk factors were inhaled nitric oxide (iNO; OR 2.6, 95% CI 1.1-6.2, P = .03), stay at a neonatal intensive care unit >28 days (OR 1.6, 95% CI 1.1-2.6, P = .03), and artificial ventilation >7 days (OR 1.6, 95% CI 1.1-2.5, P = .02). Prenatal glucocorticoids (OR 0.6, 95% CI 0.4-0.8, P < .001) and female sex (OR 0.7, 95% CI 0.5-0.99, P = .04) showed a lesser incidence of ROP. iNO remained significant after correction for all significant factors (OR 2.6, 95% CI 1.1-6.2, P = .03).

Conclusion: In addition to established risk factors (GA, BW, stay at a neonatal intensive care unit >28 days, and artificial ventilation >7 days), treatment with iNO as risk factor for ROP is a novel finding.
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http://dx.doi.org/10.1016/j.jpeds.2013.11.015DOI Listing
March 2014

Effectiveness of screening for amblyopia and other eye disorders in a prospective birth cohort study.

J Med Screen 2013 Jun 19;20(2):66-72. Epub 2013 Jul 19.

Dept. of Public Health, Erasmus MC Dr. Molewaterplein 50 3015 GE Rotterdam, the Netherlands.

Objective: To establish whether the current vision screening practice in the Netherlands is effective in preventing permanent visual loss and to estimate the sensitivity of the programme.

Settings: In the Netherlands, all children are invited for preverbal (1, 3, 6-9 and 14-24 months) and preschool (36, 45, and 60-72 months) vision screening. Screening attendance is high, but the effectiveness in reducing amblyopia is unknown.

Methods: In a 7-year cohort study, 4624 children born in the city of Rotterdam between 16 September 1996 and 15 May 1997 were followed through all routine vision screening examinations. At age seven, visual acuity (VA) of children still living in Rotterdam was assessed by study orthoptists. In case of VA > 0.1 logMAR in one or both eyes, two or more logMAR lines of interocular difference or eye disorders like strabismus, children underwent a more intensive eye examination.

Results: Attendance at the 9-month screening was 89%, decreasing to about 75% at later examinations. Of preverbal tests, 2.5% were positive, and of preschool tests, 10%. In total, 19% of children had a positive vision screening test at least once. Amblyopia prevalence was 3.4%. Sensitivity of the vision screening programme was 73% and specificity 83%. At age seven, 0.7-1.2% (confirmed vs final exam) of the children had a VA > 0.3 logMAR in the worse eye compared with 2-3.9% (in literature) reported prevalence in non-screening situations. Children who were less frequently screened had a higher chance of poor vision (>0.3 logMAR) at age seven.

Conclusion: The Dutch child vision screening programme may reduce the risk of persistent amblyopia (VA > 0.3 logMAR) at age seven by more than half.
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http://dx.doi.org/10.1177/0969141313497355DOI Listing
June 2013

Accurate gaze direction measurements with free head movement for strabismus angle estimation.

IEEE Trans Biomed Eng 2013 Nov 8;60(11):3028-35. Epub 2013 Feb 8.

We present the Delft Assessment Instrument for Strabismus in Young children (DAISY) a device designed to measure angles of strabismus in young children fast and accurately. DAISY allows for unrestrained head movements by the mean of a triple camera vision system that simultaneously estimates the head rotation and the eye pose. The device combines two different methods to record bilateral eye position: corneal reflections (Purkinje images) and pupillary images. Detailed results are provided on three orthotropic subjects (age 25-27). Three different conditions were tested: 1) gaze ahead, 2) gaze ahead with different head rotations, and 3) fixed head with different eye positions. Systematic errors occurred between subjects that need further study. The system reached sufficient accuracy to be applied for the measurement of angles of strabismus, almost independent from the head pose.
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http://dx.doi.org/10.1109/TBME.2013.2246161DOI Listing
November 2013

[Blunt trauma to the eye in children: more than a black eye].

Ned Tijdschr Geneeskd 2012 ;156(40):A5262

Afd. Mondziekten, Kaak- en Aangezichtschirurgie, Erasmus MC, Rotterdam, the Netherlands.

Background: The consequences of orbital floor fractures are different in children than in adults. Children have more elastic bones and increased chance of a trapdoor fracture, a fracture in which muscle tissue may be trapped.

Case Description: A 12-year-old girl presented to the general practitioner after she had been hit in her left eye by a swing. The patient had a painful eye and double vision. She was sent home with the advice to wake her every hour during the night. The next morning she went back to the general practitioner because she had been vomiting through the night and this time she was sent to the hospital. She was diagnosed with an orbital floor fracture with an entrapment of the inferior rectus muscle and underwent immediate surgery. Unfortunately, the release of the muscle had been too late and resulted in an uncorrectable diplopia.

Conclusion: Warning signs of entrapment of the inferior rectus muscle are a painful eye with a mobility disorder in combination with nausea and vomiting. Immediate surgical intervention offers the best outcome in terms of recovery of the eye motility.
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December 2012

An educational cartoon accelerates amblyopia therapy and improves compliance, especially among children of immigrants.

Ophthalmology 2012 Nov 21;119(11):2393-401. Epub 2012 Aug 21.

Department of Ophthalmology, Erasmus MC University Medical Centre Rotterdam, Rotterdam, The Netherlands.

Purpose: We showed previously that an educational cartoon that explains without words why amblyopic children should wear their eye patch improves compliance, especially in children of immigrant parents who speak Dutch poorly. We now implemented this cartoon in clinics in low socioeconomic status (SES) areas with a large proportion of immigrants and clinics elsewhere in the Netherlands.

Design: Clinical, prospective, nonrandomized, preimplementation, and postimplementation study.

Participants: Amblyopic children aged 3 to 6 years who started occlusion therapy.

Methods: Preimplementation, children received standard orthoptic care. Postimplementation, children starting occlusion therapy received the cartoon in addition. At implementation, treating orthoptists followed a course on compliance. In low SES areas, compliance was measured electronically during 1 week.

Main Outcome Measures: The clinical effects of the cartoon-electronically measured compliance, outpatient attendance rate, and speed of reduction in interocular-acuity difference (SRIAD)-averaged over 15 months of observation.

Results: In low SES areas, 114 children were included preimplementation versus 65 children postimplementation; elsewhere in the Netherlands, 335 versus 249 children were included. In low SES areas, mean electronically measured compliance was 52.0% preimplementation versus 62.3% postimplementation (P=0.146); 41.8% versus 21.6% (P=0.043) of children occluded less than 30% of prescribed occlusion time. Attendance rates in low SES areas were 60.3% preimplementation versus 76.0% postimplementation (P=0.141), and 82.7% versus 84.5%, respectively, elsewhere in the Netherlands. In low SES areas, the SRIAD was 0.215 log/year preimplementation versus 0.316 log/year postimplementation (P=0.025), whereas elsewhere in the Netherlands, these were 0.244 versus 0.292 log/year, respectively (P=0.005; the SRIAD's improvement was significantly better in low SES areas than elsewhere, P=0.0203). This advantage remained after adjustment for confounding factors. Overall, 25.1% versus 30.1% (P=0.038) had completed occlusion therapy after 15 months.

Conclusions: After implementation of the cartoon, electronically measured compliance improved, attendance improved, acuity increased more rapidly, and treatment was shorter. This may be due, in part, to additional measures such as the course on compliance. However, that these advantages were especially pronounced in children in low SES areas with a large proportion of immigrants who spoke Dutch poorly supports its use in such areas.
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http://dx.doi.org/10.1016/j.ophtha.2012.05.035DOI Listing
November 2012

Comfort of wear and material properties of eye patches for amblyopia treatment and the influence on compliance.

Strabismus 2012 Mar;20(1):3-10

Department of Ophthalmology, Erasmus MC University, Rotterdam, the Netherlands.

Background: Occlusion therapy for amblyopia has been the mainstay of treatment for centuries, however, acceptance of the patch is often lacking. This study evaluated comfort of wear of the eye patch and assessed the mechanical properties in order to achieve a more individualized prescription.

Methods: For 8 consecutive days, parents used each of the four main brands of patches for 2 consecutive days in a randomized fashion. After 2 days a 21-item questionnaire was completed to evaluate comfort of wear for each patch. Compliance was measured electronically using the Occlusion Dose Monitor (ODM). In addition, breathing capacity at 23°C and 33°C, resistance to water penetration, opacity, and strength of adhesion to the skin were measured.

Results: Twenty-four children participated. Overall, satisfaction was moderate: large differences in discomfort when removing the patch, skin reaction, and cosmetic appearance were found. In the material measurements large differences were found in opacity and strength of adhesion to the skin. In all brands breathing capability was minimal. Answers given by the parents matched the physical properties of the eye patch. There was no difference in electronically measured compliance between patches.

Conclusions: We found large differences in comfort of wear and mechanical properties. Therefore, when prescribing a certain brand of patch, the wide variety needs to be taken into account. Further study into these properties seems warranted; especially breathing capability requires improvement since children often wear the patch for a longer period of time. This could contribute to increasing satisfaction and consequently may improve compliance.
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http://dx.doi.org/10.3109/09273972.2012.655837DOI Listing
March 2012

Determinants and outcome of unsuccessful referral after positive screening in a large birth-cohort study of population-based vision screening.

J AAPOS 2011 Jun;15(3):256-62

Department of Ophthalmology, Erasmus MC, University Medical Centre Rotterdam, Rotterdam, The Netherlands.

Purpose: The efficacy of population-based vision screening is hampered by unsuccessful referral after a positive screening test. We studied the nature and causes of unsuccessful referral in a 7-year birth cohort study of vision screening in Rotterdam, the Netherlands.

Methods: All parents of children who had been unsuccessfully referred were asked whether they recalled the referral. Reasons for noncompliance, if any, were identified using semi-structured interviews. Screening records were checked for written evidence of the referral. The parents' fluency in Dutch and their socioeconomic status were also assessed.

Results: Of the 561 screen-positive children, 129 (23%) had not been referred successfully. For the current study, 97 parents were successfully contacted. Of these, 14 parents had been willingly noncompliant and 83 said they were unaware of the referral, with 47% having poor to moderate fluency in Dutch. In 53 cases, the screening charts contained no written evidence of any referral. Amblyopia was identified in 3 of the unsuccessful referrals.

Conclusions: In this population-based screening program, 1 of 4 positively screened children was not successfully referred. Apart from parental noncompliance, the unsuccessful referrals can be explained by miscommunication, deficient documentation, and physician noncompliance with screening guidelines. An effective monitoring feedback system may improve the efficacy of child vision screening.
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http://dx.doi.org/10.1016/j.jaapos.2011.01.159DOI Listing
June 2011

Sociocultural and psychological determinants in migrants for noncompliance with occlusion therapy for amblyopia.

Graefes Arch Clin Exp Ophthalmol 2011 Dec 12;249(12):1893-9. Epub 2011 Mar 12.

Department of Ophthalmology, Room EE-1607, Erasmus MC, University Medical Center Rotterdam, PO Box 2040, 3000 CA, Rotterdam, The Netherlands.

Background: Compliance with occlusion therapy for amblyopia in children is low when their parents have a low level of education, speak Dutch poorly, or originate from another country. We determined how sociocultural and psychological determinants affect compliance.

Methods: Included were amblyopic children between the ages of 3 and 6, living in low socio-economic status (SES) areas. Compliance with occlusion therapy was measured electronically. Their parents completed an oral questionnaire, based on the "Social Position & Use of Social Services by Migrants and Natives" questionnaire that included demographics and questions on issues like education, employment, religion and social contacts. Parental fluency in Dutch was rated on a five-point scale. Regression analysis was used to describe the relationship between the level of compliance and sociocultural and psychological determinants.

Results: Data from 45 children and their parents were analyzed. Mean electronically measured compliance was 56 ± 44 percent. Children whose parents had close contact with their neighbors or who were highly dependent on their family demonstrated low levels of compliance. Children of parents who were members of a club and who had positive conceptualizations of Dutch society showed high levels of compliance. Poor compliance was also associated with low income, depression, and when patching interfered with the child's outdoor activity. Religion was not associated with compliance.

Conclusions: Poor compliance with occlusion therapy seems correlated with indicators of social cohesion. High social cohesion at micro level, i.e., family, neighbors and friends, and low social cohesion on macro level, i.e., Dutch society, are associated with noncompliance. However, such parents tend to speak Dutch poorly, so it is difficult to determine its actual cause.
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http://dx.doi.org/10.1007/s00417-011-1637-5DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC3229699PMC
December 2011

Vertical position of the orbits in nonsyndromic plagiocephaly in childhood and its relation to vertical strabismus.

J Craniofac Surg 2011 Jan;22(1):135-8

Department of Ophthalmology, Erasmus MC - Sophia Children's Hospital, Erasmus University Medical Center, Rotterdam, the Netherlands.

Purpose: To determine the existence of a correlation between the vertical angle of strabismus and the vertical angle between the orbital axes in nonsyndromic plagiocephaly in childhood.

Methods: Patients were included when diagnosed with plagiocephaly. Orthoptic measurements showed a vertical strabismus and three-dimensional computed tomographic (CT) imaging of the skull was available. Patients were excluded if plagiocephaly was part of a syndrome or if any surgical intervention had taken place before our measurements. Three-dimensional CT imaging was used to calculate the vertical angle between the orbital axes in 3 reference planes (VAO) perpendicular to a line of reference through the lower borders of the maxilla (VAOmax), both auditory canals (VAOaud), and the lower points of the external occipital protuberances (VAOocc).

Results: Fourteen patients were included (mean age, 14 mo). Three-dimensional CT measurements showed a mean (SD) VAOmax of 1.70 (2.31) degrees, VAOaud of -1.54 (1.46) degrees, and VAOocc of -2.06 (4.29) degrees (a negative value indicates that the eye on the affected side was situated lower in the head). The mean vertical angle of strabismus was -2.39 (4.69) degrees in gaze toward the affected side, 3.66 (3.77) degrees in gaze ahead, and 8.14 (5.63) degrees in gaze toward the nonaffected side. The Pearson test showed no significant correlations.

Conclusions: The clinical observation that vertical strabismus in adult plagiocephaly is correlated with the vertical angle of the orbital axes could not be confirmed in young children.
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http://dx.doi.org/10.1097/SCS.0b013e3181f6f814DOI Listing
January 2011

Amblyopia and strabismus questionnaire: Rasch analysis.

Invest Ophthalmol Vis Sci 2010 Dec;51(12):6898; author reply 6899-900

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http://dx.doi.org/10.1167/iovs.10-5761DOI Listing
December 2010

Rotterdam AMblyopia screening effectiveness study: detection and causes of amblyopia in a large birth cohort.

Invest Ophthalmol Vis Sci 2010 Jul 20;51(7):3476-84. Epub 2010 Jan 20.

Departments of Public Health, Erasmus Medical Center (MC), University Medical Center Rotterdam, Rotterdam, The Netherlands.

PURPOSE. The Dutch population-based child health monitoring program includes regular preverbal (age range, 1-24 months) and preschool (age range, 36-72 months) vision screening. This study is on the contribution of an organized vision screening program to the detection of amblyopia. METHODS. A 7-year birth cohort study of 4624 children was started in 1996/1997 in Rotterdam. Vision screening data were obtained from the child screening centers. Treating orthoptists working at the regional ophthalmology departments provided information about diagnosis and treatment. The diagnosis was reviewed by two experts. The parents provided additional information on their child's eye history through written questionnaires and telephone interviews. At age 7 years, the children underwent a final examination by the study orthoptists. RESULTS. Of the 3897 children still living in Rotterdam by 2004, 2964 (76.1%) underwent the final examination. Amblyopia was diagnosed in 100 (3.4%) of these (95% CI, 2.7-4.0). At age 7, 23% had visual acuity >0.3 logMAR. Amblyopia was caused by refractive error (n = 42), strabismus (n = 19), combined-mechanism (n = 30), deprivation (n = 7), or unknown (n = 2). Eighty-three amblyopia cases had been detected before age 7. Amblyopia detection followed positive results in vision screening in 56 children, either preverbal (n = 15) or preschool (n = 41). Twenty-six other amblyopes were self-referred (n = 12, before a first positive screening test), especially strabismic or combined-mechanism amblyopia; data were uncertain for one other positively screened amblyopic child. Amblyopia remained undetected until age 7 due to unsuccessful referral (n = 4, three with visual acuity >0.3 logMAR at age 7) or false-negative screening (n = 13). CONCLUSIONS. Most cases of amblyopia were detected by vision screening with visual acuity measurement. Preverbal screening contributed little to the detection of refractive amblyopia.
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http://dx.doi.org/10.1167/iovs.08-3352DOI Listing
July 2010

Construct validation of the Amblyopia and Strabismus Questionnaire (A&SQ) by factor analysis.

Graefes Arch Clin Exp Ophthalmol 2009 Sep 3;247(9):1263-8. Epub 2009 Jun 3.

Department of Ophthalmology, Erasmus MC, University Medical Center Rotterdam, NL-3000 CA Rotterdam, The Netherlands.

Background: The Amblyopia and Strabismus Questionnaire (A&SQ) was previously developed to assess quality of life (QoL) in amblyopia and/or strabismus patients. Here, factor analysis with Varimax rotation was employed to confirm that the questions of the A&SQ correlated to dimensions of quality of life (QoL) in such patients.

Methods: Responses on the A&SQ from three groups were analyzed: healthy adults (controls) (n = 53), amblyopia and/or strabismus patients (n = 72), and a historic cohort of amblyopes born between 1962-1972 and occluded between 1968-1974 (n = 173). The correlations among the responses to the 26 A&SQ items were factor-analysed by Principal Component Analysis (PCA). As the development of the A&SQ was intuitive-deductive, it was expected that the pattern of correlation could be explained by the five a priori hypothesized dimensions: fear of losing the better eye, distance estimation, visual disorientation, diplopia, and social contact and cosmetic problems. Distribution of questions along the factors derived by PCA was examined by orthogonal Varimax rotation.

Results: Data from 296 respondents were analyzed. PCA provided that six factors (cutoff point eigenvalue >1.0) accumulatively explained 70.5% of the variance. All A&SQ dimensions but one matched with four factors found by Varimax rotation (factor loadings >0.50), while two factors pertained to the fifth dimension. The six factors explained 33.7% (social contact and cosmetic problems); 10.3% (near distance estimation); 8.7% (diplopia); 7.2% (visual disorientation); 6.3% (fear of losing the better eye); and 4.3% (far distance estimation), together 70.48% of the item variance.

Conclusion: The highly explained variance in the A&SQ scores by the factors found by the PCA confirmed the a priori hypothesized dimensions of this QoL instrument.
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http://dx.doi.org/10.1007/s00417-009-1112-8DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC2720588PMC
September 2009

The Amblyopia and Strabismus Questionnaire: English translation, validation, and subscales.

Am J Ophthalmol 2007 Feb 20;143(2):305-310. Epub 2006 Oct 20.

Retina Foundation of Southwest, Dallas, Texas 75231, USA.

Purpose: To establish the English-language version of the Amblyopia and Strabismus Questionnaire (ASQE).

Design: Prospective cohort study.

Methods: A structured translation process was followed to generate the ASQE, a 26-item instrument (originally in Dutch) containing five subscales for fear of losing the better eye, distance estimation, visual disorientation, double vision, and social contact and appearance. The ASQE was administered to 150 adults in a private practice setting. All had strabismus with or without amblyopia and visual acuity of 20/50 or better in at least one eye. Subjects also completed a brief disability questionnaire, and they were further characterized by levels of unilateral vision loss, diplopia, and asthenopia.

Results: ASQE scores were highly correlated with the disability questionnaire outcomes (r = -.76, P < .0001). Internal consistency reliability (Cronbach alpha) of the subscales ranged from 0.80 to 0.92. Strong correlations were found between clinical characteristics and the ASQE (total score and subscale scores).

Conclusions: The ASQE showed good psychometric properties that are in line with those of the original instrument. This, combined with strong correlations between ASQE scores and clinical characterization of the participants, establishes the ASQE as a useful tool for use in populations with strabismus and/or amblyopia.
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http://dx.doi.org/10.1016/j.ajo.2006.09.046DOI Listing
February 2007