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David G Hunter Boston Children’s Hospital, Harvard Medical School, Boston, MA, USACorrespondence: David G Hunter Email david.hunter@childrens.harvard.edu In their article, “Drive-by Photoscreening,”1 Keffalos, Martin, and Arnold make impressive, hands-on efforts to modify vision screening devices to enable their continued use during a pandemic. I would like to offer two clarifications. View the original paper by Keffalos and colleagues

David G Hunter Department of Ophthalmology, Boston Children’s Hospital, Harvard Medical School, Boston, MA, 02115, USACorrespondence: David G Hunter Department of OphthalmologyBoston Children’s Hospital, Harvard Medical School, 300 Longwood Ave, Boston, MA 02115, USATel +1 617-355-6766Email david.hunter@childrens.harvard.edu I write to clarify some of the conclusions made by Dr. Arnold in his study of the blinq.™ vision screener.1 The goal of vision screening is early detection of amblyopia and strabismus, which until now, due to limitations in technology, could only be achieved by detecting refractive risk factors. Unfortunately, risk factor screening results in under-detection of patients with strabismus and over referral of children who would not benefit from treatment.2 The blinq. device was not designed to detect refractive risk factors; instead, the device detects amblyopia directly by performing a binocular retinal polarization scan to identify reduced binocularity, microstrabismus, and fixation instability without regard to refractive status.3 In his study, Dr. Arnold evaluated the ability of blinq. to detect amblyopia risk factors, but the performance of blinq. and all vision screening technology should be evaluated in the context of how well it identifies patients who meet diagnostic criteria for amblyopia and strabismus.4 Out of concern that his study design might create confusion among readers, I contacted Dr. Arnold, who kindly provided his de-identified data to allow me to determine which patients had referral warranted disease, not just risk factors. View the original paper by Arnold

Surgical management of strabismus remains a challenge because surgical success rates, short-term and long-term, are not ideal. Adjustable suture strabismus surgery has been available for decades as a tool to potentially enhance the surgical outcomes. Intellectually, it seems logical that having a second chance to improve the outcome of a strabismus procedure should increase the overall success rate and reduce the reoperation rate. Yet, adjustable suture surgery has not gained universal acceptance, partly because Level 1 evidence of its advantages is lacking, and partly because the learning curve for accurate decision making during suture adjustment may span a decade or more. In this review we describe the indications, techniques, and published results of adjustable suture surgery. We will discuss the option of 'no adjustment' in cases with satisfactory alignment with emphasis on recent advances allowing for delayed adjustment. The use of adjustable sutures in special circumstances will also be reviewed. Consistently improved outcomes in the adjustable arm of nearly all retrospective studies support the advantage of the adjustable option, and strabismus surgeons are advised to become facile in the application of this approach. [ABSTRACT FROM AUTHOR]

We employed high-resolution fMRI to distinguish the impacts of anisometropia and strabismus amblyopia on the evoked ocular dominance (OD) response. Sixteen amblyopic participants (8 females) plus 8 individuals with normal vision (1 female), participated in this study for whom, we measured the difference between the response to stimulation of the two eyes, across areas V1-V4.In controls, the evoked OD response formed the expected striped pattern within V1. Compared to controls, the OD response in amblyopic participants formed larger fused patches that extended into downstream visual areas. Moreover, both anisometropic and strabismic participants showed elevated OD responses across V1-V4.Beyond these common effects, and despite similar densities of amblyopia between the two groups, strabismus and anisometropia had differential impacts on the OD bias, binocular response and correlation between V1 depth levels. Specifically, we found a greater increase in the size of V1 portion that responded preferentially to fellow eye stimulation in anisometropic compared to strabismic individuals. We also found a greater difference between the amplitudes of the response to binocular stimulation, in those regions that responded preferentially to the fellow vs. amblyopic eye, in anisometropic compared to strabismic participants. In contrast, strabismic participants demonstrated increased correlation between the OD responses evoked within V1 superficial and deep depths, whereas anisometropic individuals did not.These results provide the primary direct functional evidence for distinct impacts of strabismus and anisometropia on the mesoscale functional organization of the human visual system, thus extending what was inferred previously about amblyopia from animal models. Significance Statement Amblyopia is a developmental disorder caused by perturbations to normal binocular visual experience during the critical period impact. Despite its high prevalence, our current understanding of amblyopia impacts on the mesoscale functional organization of primary visual area (V1) is mostly based on invasive techniques in animals. In this study we showed the primary direct evidence for the distinct impacts of anisometropia versus strabismus (two major causes of amblyopia) on the fMRI activity evoked within the human visual cortex. Our findings also confirmed the hypothesized link between the evoked OD response and the interocular visual acuity difference in amblyopia., (Copyright © 2025 the authors.)

Introduction: Botulinum toxin is an alternative to conventional strabismus surgery for treatment of acute, acquired, comitant esotropia (AACE). Previous studies suggest that the 2 treatment approaches may be equally effective for 6 months. The purpose of our study was to determine whether botulinum toxin remains as effective as strabismus surgery for 36 months after treatment., Design: Multicenter, retrospective, nonrandomized, comparative, clinical, noninferiority study., Methods: Setting: Two tertiary care pediatric hospitals., Study Population: 76 children with AACE followed for at least 36 months after treatment., Intervention: Treatment with either botulinum toxin ("BTX group") or strabismus surgery ("surgery group")., Main Outcome Measure: Success rate at 36 months (horizontal deviation of 10 prism diopters or less and evidence of binocular vision)., Results: There were 44 patients in the BTX group and 32 patients in the surgery group with a median deviation of 35 PD in both groups (range 10-55). The duration of general anesthesia (6 versus 71 min, P < .0001) and time in the postanesthesia care unit (40 versus 95 min, P < .0001) were significantly shorter in the BTX group. At 36 months, the success rate was 72% in the BTX group and 56% in the surgery group with a similar median deviation and median stereoacuity. BTX was noninferior to surgery at 36 months (95%CI for difference in success rate (BTX minus surgery) was -5% to +38%). At 36 months, the median time from esotropia onset to any intervention was 6.5 months without treatment success and 4 months in those with treatment success (P < .05)., Conclusions/relevance: Botulinum toxin was noninferior to strabismus surgery in the treatment of AACE at 36 months while reducing the duration of general anesthesia. Longer delay from esotropia onset to treatment was an independent risk factor for worse sensorimotor outcomes irresepctive of the type of treatment., (Copyright © 2025 Elsevier Inc. All rights reserved.)

Purpose: To evaluate the cost-utility of Luminopia and CureSight as therapy for amblyopia compared to current common amblyopic treatments such as glasses, atropine drops, and patching., Design: Cost analysis based on data from published randomized control trials (RCTs)., Subjects: None; based on data from the Luminopia, CureSight and atropine RCTs., Methods: A cost-utility analysis was performed using patient preference-based time trade-off utility values from previous literature. Costs for eye examinations were calculated using reimbursement data; device costs for duration of treatment were provided by sales representatives of Luminopia and CureSight. All treatments were inclusive of the cost of eyeglasses. Visual acuity (VA) and stereoacuity outcomes were extrapolated from the randomized control trials (RCTs) for atropine, Luminopia and CureSight. Quality-adjusted life-year (QALY) was calculated by multiplying utility gain, a value correlated with visual acuity gain, by length of time of benefit., Main Outcome Measures: Cost, cost per quality-adjusted life-year, and cost per stereoacuity gain., Results: The cost to treat amblyopia with glasses alone for 12 weeks was $514. The cost of treating with patching for 12 weeks was $540 and with atropine for 16 weeks was $652, while the cost of treating with Luminopia or CureSight for 12 weeks was $1951 and $1564 or $1814, respectively. Treatment with glasses alone or patching for 12-weeks resulted in a cost/QALY gained of $427 and $101 respectively. Atropine treatment for 16 weeks resulted in a cost/QALY gained of $151. The cost/QALY for 12-week Luminopia treatment was $618 versus $368 or $427 for 12-week CureSight treatment and $314 or $354 for 16-week CureSight treatment (p<0.05). Cost per stereoacuity gain for 12-week treatment duration was $6421/log arcsec (glasses), $1801/log arcsec (patching), and $3007/log arcsec or $3488 (CureSight)., Conclusion: Treatment of amblyopia with Luminopia or CureSight is cost-effective in comparison to established willingness-to-pay thresholds and can provide a viable treatment option, especially for those who are unable to tolerate patching or atropine penalization. Cost effectiveness based on VA gain of Luminopia and CureSight were comparable., (Copyright © 2025. Published by Elsevier Inc.)

Ocular torticollis is traditionally attributed to eye misalignment, nystagmus, ptosis, or refractive error. We present 3 pediatric cases of acquired torticollis caused by a foreign body beneath the upper eyelid. The head posturing presumably developed to minimize contact of the foreign body with the corneal surface and mitigate ocular discomfort. Torticollis resolved in all cases after removal of the foreign bodies. These cases demonstrate that acquired torticollis in a child can be a clinical sign of an ocular surface foreign body., (Copyright © 2024 American Association for Pediatric Ophthalmology and Strabismus. Published by Elsevier Inc. All rights reserved.)

This cross-sectional study used data from the American Academy of Ophthalmology IRIS Registry (Intelligent Research in Sight) and machine learning algorithms to identify pediatric ophthalmologists based on physician coding patterns. A random forest model achieved an area under the receiver operating characteristic curve of 0.98, sensitivity of 0.98, and specificity of 0.88 when classifying pediatric eye specialists in the test validation cohort. Algorithm-based approaches to identify pediatric ophthalmologists using procedure codes may offer new avenues to determine the scope, scale, and trajectory of pediatric eye care delivery., (Copyright © 2024 American Association for Pediatric Ophthalmology and Strabismus. Published by Elsevier Inc. All rights reserved.)

Purpose: Bardet-Biedl Syndrome (BBS) is a rare autosomal recessive ciliopathy. Within corneal development, primary cilia serve a critical role. We sought to investigate the association of BBS with corneal astigmatism among a cohort of patients with BBS., Methods: This was a cross-sectional, retrospective study performed at a pediatric ophthalmology department of a tertiary hospital. The study enrolled 45 patients with genetically confirmed Bardet-Biedl syndrome, encompassing a total of 90 eyes observed from February 2011 to August 2021. Spherical and cylindrical refractive errors and keratometry outcome measures, including diopter (D) values at the flattest and steepest axes, were recorded. Corneal astigmatism of greater than 3D is considered extreme corneal astigmatism based on previously published data., Results: Among 45 patients (M:26; F:19), the mean age was 16.4 ± 8.2 years, and the mean best-corrected visual acuity was 20/60. The most common molecular diagnosis was BBS1 , seen in 24 of 45 (53.3%). Among all the patients, the mean spherical refractive error was -2.9 ± 3.8D. The mean cylindrical refractive error was 2.6 ± 1.5D. The mean keratometry values at the flattest axis was 43.5 ± 5.3D (39.4-75.0) and at the steepest axis was 47.2 ± 7.3D(41.5-84.0). Among all the patients with BBS, the mean corneal astigmatism was 3.7 ± 1.0D(0.5-7.1), which is considered extreme., Conclusion: A cohort of individuals with BBS demonstrated high corneal astigmatism. These results suggest an association between corneal astigmatism and primary ciliary dysfunction and may assist in clinical management and future therapeutic targets among BBS and other corneal disorders.

Importance: Racial, ethnic, and sex disparities exist in US clinical study enrollment, and the prevalence of these disparities in Pediatric Eye Disease Investigator Group (PEDIG) clinical studies has not been thoroughly assessed., Objective: To evaluate racial, ethnic, and sex representation in PEDIG clinical studies compared with the 2010 US Census pediatric population., Design, Setting, and Participants: This cross-sectional analysis examined PEDIG clinical studies based in the US from December 1, 1997 to September 12, 2022, 41 of which met inclusion criteria of a completed study, a study population younger than 18 years, and 1 or more accompanying publication. Data analysis was performed between November 2023 and February 2024., Exposure: Study participant race, ethnicity, and sex for each clinical study, as collected from peer-reviewed publications, patient-enrollment datasets, and ClinicalTrials.gov., Main Outcomes and Measures: Median enrollment percentages of female, White, Black, Hispanic, Asian, and other race participants were calculated and compared with the 2010 US Census pediatric population using a 1-sample Wilcoxon rank test. Proportionate enrollment was defined as no difference on a 1-sample Wilcoxon rank test if P ≥ .05. If P < .05, we determined if the median enrollment percentage was greater than or less than 2010 US Census proportion to determine if enrollees were underrepresented or overrepresented. To calculate the magnitude of overrepresentation or underrepresentation, enrollment-census difference (ECD) was defined as the difference between groups' median enrollment percentage and percentage representation in the 2010 US Census. Compound annual growth rate (CAGR) was used to measure temporal trends in enrollment, and logistic regression analysis was used to analyze factors that may have contributed to proportionate representation outcomes., Results: A total of 11 658 study participants in 41 clinical studies were included; mean (SD) participant age was 5.9 (2.8) years and 5918 study participants (50.8%) were female. In clinical studies meeting inclusion criteria, White participants were overrepresented (ECD, 0.19; 95% CI, 0.10-0.28; P < .001). Black participants (ECD, -0.07; 95% CI, -0.10 to -0.03; P < .001), Asian participants (ECD, -0.03; 95% CI, -0.04 to -0.02; P < .001), and Hispanic participants (ECD, -0.09; 95% CI, -0.13 to -0.05; P < .001) were underrepresented. Female participants were represented proportionately (ECD, 0.004; 95% CI, -0.036 to 0.045; P = .21). White and Asian participants demonstrated a decreasing trend in study enrollment from 1997 to 2022 (White: CAGR, -1.5%; 95% CI, -2.3% to -0.6%; Asian: CAGR, -1.7%; 95% CI, -2.0% to -1.4%), while Hispanic participants demonstrated an increasing enrollment trend (CAGR, 7.2%; 95% CI, 3.7%-10.7%)., Conclusions and Relevance: In this retrospective cross-sectional study of PEDIG clinical studies from December 1, 1997 to September 12, 2022, Black, Hispanic, and Asian participants were underrepresented, White participants were overrepresented, and female participants were represented proportionally. Trends suggested increasing enrollment of Hispanic participants and decreasing enrollment of White participants over time. This study demonstrates an opportunity to advocate for increased enrollment of underrepresented groups in pediatric ophthalmology clinical studies.

Fluorescein angiography is a fluorescent dye-based imaging procedure, most commonly indicated in the pediatric setting to evaluate peripheral retinal vascular lesions. Fluorescein dye is organic, water soluble, and largely excreted renally, with a reassuring safety profile at therapeutic doses. While toxicity with intrathecal overdose has been reported, the effect of intravenous exposure to supratherapeutic levels has not been previously documented in the literature. We report the case of a 10-month-old girl with incontinentia pigmenti who received more than four times the recommended dosage of fluorescein during scheduled fluorescein angiography and developed marked yellowing of skin, sclera, stool, and urine. She was seen by the toxicology team and underwent monitoring for 8 hours before being discharged, with no adverse consequences detected during follow-up., (Copyright © 2024 American Association for Pediatric Ophthalmology and Strabismus. Published by Elsevier Inc. All rights reserved.)

Purpose: To functionally evaluate novel human sequence-derived candidate genes and variants for unsolved ocular congenital cranial dysinnervation disorders (oCCDDs)., Methods: Through exome and genome sequencing of a genetically unsolved human oCCDD cohort, we previously identified variants in 80 strong candidate genes. Here, we further prioritized a subset of these (43 human genes, 57 zebrafish genes) using a G0 CRISPR/Cas9-based knockout assay in zebrafish and generated F2 germline mutants for seventeen. We tested the functionality of variants of uncertain significance in known and novel candidate transcription factor-encoding genes through protein binding microarrays., Results: We first demonstrated the feasibility of the G0 screen by targeting known oCCDD genes phox2a and mafba . 70-90% of gene-targeted G0 zebrafish embryos recapitulated germline homozygous null-equivalent phenotypes. Using this approach, we then identified three novel candidate oCCDD genes ( SEMA3F, OLIG2 , and FRMD4B ) with putative contributions to human and zebrafish cranial motor development. In addition, protein binding microarrays demonstrated reduced or abolished DNA binding of human variants of uncertain significance in known and novel sequence-derived transcription factors PHOX2A (p.(Trp137Cys)), MAFB (p.(Glu223Lys)), and OLIG2 (p.(Arg156Leu))., Conclusions: This study nominates three strong novel candidate oCCDD genes ( SEMA3F, OLIG2 , and FRMD4B ) and supports the functionality and putative pathogenicity of transcription factor candidate variants PHOX2A p.(Trp137Cys), MAFB p.(Glu223Lys), and OLIG2 p.(Arg156Leu). Our findings support that G0 loss-of-function screening in zebrafish can be coupled with human sequence analysis and protein binding microarrays to aid in prioritizing oCCDD candidate genes/variants.

Introduction: Congenital enophthalmos is a rare condition characterized by posterior displacement of the globe, often associated with bony orbital anomalies or whole globe development defects. The purpose of this report is to present two unrelated cases of congenital enophthalmos secondary to anomalous accessory orbital bands and to describe characteristics of orbital imaging that differentiate this condition from the other causes., Methods: The case records of two patients who presented with congenital enophthalmos and were discovered to have anomalous accessory orbital extraocular muscle bands were reviewed. The clinical features, initial diagnosis, high resolution magnetic resonance imaging (MRI) findings, and surgical outcomes were noted. A 3-dimensional reconstruction model was used to understand the approach and surgical management in one of the cases., Results: Both patients presented with unilateral severe enophthalmos, globe retraction, and restricted ocular motility in all directions since birth. High-resolution MRI of the orbits revealed a short anomalous band, isointense to the muscle, arising from a rectus muscle belly and attaching to the posteroinferior part of the globe adjacent to the optic nerve. The caliber of the extraocular muscles and ocular motor nerves was normal. In one patient, surgery was not pursued due to the extreme posterior location of the band with proximity to the optic nerve. In the other patient, the deviation did not improve, despite successfully severing the accessory band, due to extensive scarring., Conclusion: Anomalous accessory orbital extraocular muscle bands are a rare and often overlooked cause of congenital enophthalmos when associated with limited ocular motility. Imaging the orbit can aid in diagnosis and help differentiate it from other causes. Safe surgical approaches to address the problem are limited, and available approaches may not be effective. These two cases highlight that the management of accessory extraocular muscle bands causing enophthalmos can be extremely challenging and difficult to improve even with intensive surgical intervention.

Purpose: To identify genetic etiologies and genotype/phenotype associations for unsolved ocular congenital cranial dysinnervation disorders (oCCDDs)., Methods: We coupled phenotyping with exome or genome sequencing of 467 probands (550 affected and 1108 total individuals) with genetically unsolved oCCDDs, integrating analyses of pedigrees, human and animal model phenotypes, and de novo variants to identify rare candidate single nucleotide variants, insertion/deletions, and structural variants disrupting protein-coding regions. Prioritized variants were classified for pathogenicity and evaluated for genotype/phenotype correlations., Results: Analyses elucidated phenotypic subgroups, identified pathogenic/likely pathogenic variant(s) in 43/467 probands (9.2%), and prioritized variants of uncertain significance in 70/467 additional probands (15.0%). These included known and novel variants in established oCCDD genes, genes associated with syndromes that sometimes include oCCDDs (e.g., MYH10, KIF21B, TGFBR2, TUBB6), genes that fit the syndromic component of the phenotype but had no prior oCCDD association (e.g., CDK13, TGFB2), genes with no reported association with oCCDDs or the syndromic phenotypes (e.g., TUBA4A, KIF5C, CTNNA1, KLB, FGF21), and genes associated with oCCDD phenocopies that had resulted in misdiagnoses., Conclusion: This study suggests that unsolved oCCDDs are clinically and genetically heterogeneous disorders often overlapping other Mendelian conditions and nominates many candidates for future replication and functional studies., (Copyright © 2024. Published by Elsevier Inc.)

Purpose: To compare the efficacy of botulinum toxin injections to strabismus surgery in children with acute, acquired, comitant esotropia (ACE), and to investigate factors predicting success., Design: International, multi-center nonrandomized comparative study METHODS: Setting: Cloud-based survey., Study Population: Children aged 2 to 17 years who underwent a single surgical intervention for ACE., Interventions: Botulinum toxin injection ("chemodenervation" group) or strabismus surgery ("surgery" group)., Main Outcome Measures: Primary measure: success rate at 6 months in propensity-matched cohort, defined as total horizontal deviation of 10 prism diopters or less with evidence of binocular single vision. Secondary measure: Risk factors for poor outcomes in the full cohort., Results: Surgeons from 19 centers contributed. There were 74 patients in the chemodenervation group and 97 patients in the surgery group. In the propensity-matched data (n = 98), success rate was not significantly different at 6 months (70.2% vs 79.6%; P = .2) and 12 months (62.9% vs 77.8%; P = .2), but was significantly lower in the chemodenervation group at 24 months (52% vs 86.4%; P = .015). Irrespective of treatment modality, treatment delay was associated with lower success rates at 6 months, with median time from onset to intervention 4.5 months (interquartile range (IQR): 2.1, 6.7) in the success group and 7.7 months (IQR: 5.6, 10.1) in the failure group (P < .001)., Conclusions: In children with ACE, success rate after chemodenervation was similar to that of surgery for up to 12 months but lower at 24 months. Those with prompt intervention and no amblyopia had the most favorable outcomes, regardless of treatment modality., (Copyright © 2024 Elsevier Inc. All rights reserved.)

Purpose: To compare the incidence of strabismus after upper and lower blepharoplasty in the United States., Methods: Retrospective cohort study of adults (age ≥18 years) in the IRIS Registry (Intelligent Research in Sight) who underwent blepharoplasty between January 1, 2013 and December 31, 2020. The primary outcome was the Kaplan-Meier estimated cumulative incidence of strabismus diagnosis and surgery within 3 years of blepharoplasty. Multivariable Cox regression was used to determine the association of blepharoplasty type with strabismus diagnosis and surgery, adjusting for patient age, sex, and geographic region., Results: Blepharoplasty was performed in 368,623 patients (median [interquartile range] age, 69 [63-75] years, and 69% female). Compared with those undergoing upper eyelid blepharoplasty, patients treated with lower eyelid blepharoplasty were slightly younger (median age, 66 vs. 69 years; p < 0.001) and more likely to be female (71% vs. 69%; p < 0.001). There was a greater 3-year incidence of strabismus diagnosis (2.0% vs. 1.5%; p < 0.001) and a greater 3-year incidence of strabismus surgery (0.15% vs. 0.06%; p = 0.003) for individuals undergoing lower vs. upper blepharoplasty. After adjusting for age, sex, and geographic region, lower blepharoplasty was associated with a higher 3-year risk of strabismus diagnosis (HR, 1.49; 95% CI, 1.23-1.81; p < 0.001) and surgery (HR, 2.53; 95% CI, 1.27-5.03; p = 0.008)., Conclusions: This registry-based analysis found that individuals undergoing lower eyelid blepharoplasty were at higher risk of strabismus compared with those undergoing upper eyelid blepharoplasty. Using large databases to understand the incidence of complications of frequently performed procedures may improve ophthalmologists' ability to provide data-driven counseling on surgical risks prior to intervention., (Copyright © 2024 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.)

Amblyopia is a developmental disorder associated with reduced performance in visually guided tasks, including binocular navigation within natural environments. To help understand the underlying neurological disorder, we used fMRI to test the impact of amblyopia on the functional organization of scene-selective cortical areas, including the posterior intraparietal gyrus scene-selective (PIGS) area, a recently discovered region that responds selectively to ego-motion within naturalistic environments (Kennedy et al., 2024). Nineteen amblyopic adults (10 female) and thirty age-matched controls (12 female) participated in this study. Amblyopic participants spanned a wide range of amblyopia severity, based on their interocular visual acuity difference and stereoacuity. The visual function questionnaire (VFQ-39) was used to assess the participants' perception of their visual capabilities. Compared to controls, we found weaker scene-selective activity within the PIGS area in amblyopic individuals. By contrast, the level of scene-selective activity across the occipital place area (OPA), parahippocampal place area (PPA), and retrosplenial cortex (RSC)) remained comparable between amblyopic and control participants. The subjects' scores on "general vision" (VFQ-39 subscale) correlated with the level of scene-selective activity in PIGS. These results provide novel and direct evidence for amblyopia-related changes in scene-processing networks, thus enabling future studies to potentially link these changes across the spectrum of documented disabilities in amblyopia., Competing Interests: Conflict of interest statement: Authors declare no competing financial interests.

We report the case of an 80-year-old man with restrictive strabismus in lateral gaze following multiple oculoplastic procedures for idiopathic epiphora. Despite excellent initial response to nasal conjunctival recession with lysis of adhesions and a miminal recession of the medial rectus muscle, the patient suffered recurrence of diplopia associated with limitation of abduction due to aggressive, deep, subconjunctival scarring. Given the history of oral lichen planus (LP), the patient was diagnosed with ocular involvement of LP. He underwent a second conjunctival recession, this time accompanied by an intensive LP treatment regimen. Nine months after surgery, he remained diplopia free and orthophoric in primary gaze. Surgeons treating restrictive strabismus in patients with LP should consider implementing systemic and topical immunosuppressive treatment simultaneously with surgical management., (Copyright © 2024 American Association for Pediatric Ophthalmology and Strabismus. Published by Elsevier Inc. All rights reserved.)

Purpose: To identify sociodemographic factors associated with the visual outcomes of retinoblastoma survivors., Methods: Retrospective cohort study using a US-based clinical data registry. All individuals < 18 years of age with a history of retinoblastoma in the Intelligent Research in Sight (IRIS®) Registry (1/1/2013-12/31/2020). The primary outcome was visual acuity below the threshold for legal blindness (20/200 or worse) in at least one eye. Multivariable logistic regression was used to evaluate the association between visual outcomes and age, sex, laterality, race, ethnicity, type of insurance, and geographic location., Results: This analysis included 1545 children with a history of retinoblastoma. The median length of follow-up was 4.1 years (IQR, 2.2-5.9 years) and the median age at most recent clinical visit was 12 years (IQR, 8-16 years). Retinoblastoma was unilateral in 54% of cases. Poor vision in at least one eye was identified in 78% of all children and poor vision in both eyes in 17% of those with bilateral disease. Poor visual outcomes were associated with unilateral diagnosis (OR, 1.55; 95% CI,1.13-2.12; p  = .007), Black race (OR, 2.03; 95% CI, 1.19-3.47; p  = .010), Hispanic ethnicity (OR, 1.65; 95% CI, 1.16-2.37; p  = .006), and non-private insurance (OR, 1.47; 95% CI, 1.02-2.10; p  = .037)., Conclusions: Poor visual outcomes appear to be more common among Black, Hispanic, and publicly insured children with a history of retinoblastoma, raising concerns regarding healthcare inequities. Primary care physicians should ensure that young children receive red reflex testing during routine visits and consider retinoblastoma in the differential diagnosis of abnormal eye exams.

Purpose: To investigate the prevalence and risk of new-onset abducens nerve palsy and acute-onset diplopia following mRNA COVID-19 vaccination., Methods: In this retrospective, population-based study, patient data from the COVID-19 Research Network of TriNetX was searched via the TriNetX Analytics platform for patients who received specific vaccinations based on Common Procedural Technology codes. We recorded instances of newly diagnosed abducens nerve palsy and diplopia within 21 days following each vaccination event., Results: Of the 3,545,224 patients (mean age at vaccination, 46.2 ± 21.3 years) who received the mRNA COVID-19 vaccine, 12 (<0.0001%) patients had a new diagnosis of abducens nerve palsy and 453 (0.013%) had acute-onset diplopia within 21 days of first dose of COVID-19 vaccination. After propensity score matching, the relative risk for new abducens nerve palsy diagnosis after the first dose of COVID-19 vaccination was not significantly different from that after influenza (RR, 0.77), Tdap (RR, 1.0), or the second dose of the COVID-19 vaccinations (RR, 1.00). Furthermore, there was a lower risk of abducens nerve palsy diagnosis after the first dose of the COVID-19 vaccination compared with the risk after COVID-19 infection (RR, 0.15)., Conclusions: The risk of a new abducens nerve palsy diagnosis following the first dose of the COVID-19 vaccine is lower than the risk associated with COVID-19 infection itself. There is no evidence to suggest a causal relationship between COVID-19 vaccination and the development of abducens nerve palsy., (Copyright © 2024 American Association for Pediatric Ophthalmology and Strabismus. Published by Elsevier Inc. All rights reserved.)

Down syndrome (DS) is the most common chromosomal disorder in humans. DS is associated with increased prevalence of several ocular sequelae, including characteristic blue-dot cerulean cataract. DS is accompanied by age-dependent accumulation of Alzheimer's disease (AD) amyloid-β (Aβ) peptides and amyloid pathology in the brain and comorbid early-onset Aβ amyloidopathy and colocalizing cataracts in the lens. Quasi-elastic light scattering (QLS) is an established optical technique that noninvasively measures changes in protein size distributions in the human lens in vivo. In this cross-sectional study, lenticular QLS correlation time was decreased in adolescent subjects with DS compared to age-matched control subjects. Clinical QLS was consistent with alterations in relative particle hydrodynamic radius in lenses of adolescents with DS. These correlative results suggest that noninvasive QLS can be used to evaluate molecular changes in the lenses of individuals with DS., Competing Interests: Declaration of competing interest Goldstein (Cognoptix, Rebion); Hunter (Rebion, Luminopia). None of the reported entities contributed financial support for or had access to results from this study. No other disclosures were reported., (Copyright © 2024. Published by Elsevier Ltd.)

Purpose: Although strabismus surgery outcomes can be objectively measured, patient perception of results may differ. We present surgical outcomes graded by a prospective, "goal-determined metric" and compare these outcomes to results of a patient satisfaction survey., Design: Validity analysis comparing a clinical "goal-determined metric" to patient satisfaction., Methods: Goal-determined metric outcomes (2018-2021) for 2 surgeons treating esotropia or exotropia for diplopia control or reconstructive goals were collected. Inclusion required completion of postoperative examination 2-6 months after surgery and a satisfaction survey., Results: Record review identified 275 patients; 228 (median age 41 years [interquartile range 13-59]) met inclusion criteria. For the entire cohort, 87% were graded as "excellent" outcomes, and 78% of patients were overall "very satisfied." Agreement between patients' and surgeons' grading was 75% to 79% for all reconstructive surgery and for treatment of diplopia from esotropia. Agreement was lower, although not statistically different, for treatment of diplopia from exotropia (64%, 95% CI 43%-80%; P = .184). Preoperative risk factors, concurrent vertical or oblique surgery, and sex did not affect outcomes or satisfaction. Performance of activities requiring distance viewing improved more than performance of activities at near after esotropia-diplopia surgery (odds ratio 3.0, 95% CI 1.5-6.4; P = .004). For reconstructive cases achieving "much better" eye alignment, 62% and 72% (previously esotropic and exotropic) reported enhanced self-confidence., Conclusions: Outcomes graded by goal-determined metric correlated well with many aspects of patient satisfaction. Patient-perceived improvement in appearance was important regardless of goal. Greater improvement in performance of activities requiring distance rather than near viewing characterized treatment of diplopia from esotropia., (Copyright © 2023 Elsevier Inc. All rights reserved.)

Purpose: Individuals with amblyopia experience central vision deficits, including loss of visual acuity, binocular vision, and stereopsis. In this study, we examine the differences in peripheral binocular imbalance in children with anisometropic amblyopia, strabismic amblyopia, and typical binocular vision to determine if there are systematic patterns of deficits across the visual field., Methods: This prospective cohort study recruited 12 participants with anisometropic amblyopia, 10 with strabismic amblyopia, and 10 typically sighted controls (age range, 5-18 years). Binocular imbalance was tested at 0°, 4°, and 8° eccentricities (4 angular locations each) using band-pass filtered Auckland optotypes (5 cycles per optotype) dichoptically presented with differing contrast to each eye. The interocular contrast ratio was adjusted until the participant reported each optotype with equal frequency., Results: Participants with anisometropic and strabismic amblyopia had a more balanced contrast ratio, or decreased binocular imbalance, at 4° and 8° eccentricities as compared with central vision. Participants with strabismic amblyopia had significantly more binocular imbalance in the periphery as compared with individuals with anisometropic amblyopia or controls. A linear mixed effects model showed a main effect for strabismic amblyopia and eccentricity on binocular imbalance across the visual field., Conclusions: There is evidence of decreased binocularity deficits, or interocular suppression, in the periphery in anisometropic and strabismic amblyopia as compared with controls. Notably, those with strabismic amblyopia exhibited more significant peripheral binocular imbalance. These variations in binocularity across the visual field among different amblyopia subtypes may necessitate tailored approaches for dichoptic treatment.

Importance: Strabismus is a common ocular disorder of childhood. There is a clear genetic component to strabismus, but it is not known if esotropia and exotropia share genetic risk factors., Objective: To determine whether genetic duplications associated with esotropia are also associated with exotropia., Design, Setting, and Participants: This was a cross-sectional study conducted from November 2005 to December 2023. Individuals with constant or intermittent exotropia of any magnitude or a history of surgery for exotropia were recruited from pediatric ophthalmic practices. Data were analyzed from March to December 2023., Exposure: Genetic duplication., Main Outcomes and Measures: Presence of genetic duplications at 2p11.2, 4p15.2, and 10q11.22 assessed by digital droplet polymerase chain reaction. Orthoptic measurements and history of strabismus surgery were performed., Results: A total of 234 individuals (mean [SD] age, 19.5 [19.0] years; 127 female [54.3%]) were included in this study. The chromosome 2 duplication was present in 1.7% of patients with exotropia (4 of 234; P = .40), a similar proportion to the 1.4% of patients with esotropia (23 of 1614) in whom it was previously reported and higher than the 0.1% of controls (4 of 3922) previously reported (difference, 1.6%; 95% CI, 0%-3.3%; P < .001). The chromosome 4 duplication was present in 3.0% of patients with exotropia (7 of 234; P = .10), a similar proportion to the 1.7% of patients with esotropia (27 of 1614) and higher than the 0.2% of controls (6 of 3922) in whom it was previously reported (difference, 2.8%; 95% CI, 0.6%-5.0%; P < .001). The chromosome 10 duplication was present in 6.0% of patients with exotropia (14 of 234; P = .08), a similar proportion to the 4% of patients with esotropia (64 of 1614) and higher than the 0.4% of controls (18 of 3922) in whom it was previously reported (difference, 5.6%; 95% CI, 2.5%-8.6%; P < .001). Individuals with a duplication had higher mean (SD) magnitude of deviation (31 [13] vs 22 [14] prism diopters [PD]; difference, 9 PD; 95% CI, 1-16 PD; P = .03), were more likely to have constant (vs intermittent) exotropia (70% vs 29%; difference, 41%; 95% CI, 20.8%-61.2%; P < .001), and had a higher rate of exotropia surgery than those without a duplication (58% vs 34%; difference, 24%; 95% CI, 3%-44%; P = .02)., Conclusions and Relevance: In this cross-sectional study, results suggest that the genetic duplications on chromosomes 2, 4, and 10 were risk factors for exotropia as well as esotropia. These findings support the possibility that esotropia and exotropia have shared genetic risk factors. Whether esotropia or exotropia develops in the presence of these duplications may be influenced by other shared or independent genetic variants or by environmental factors.

We employed high-resolution functional MRI (fMRI) to distinguish the impacts of anisometropia and strabismus (the two most frequent causes of amblyopia) on the evoked ocular dominance (OD) response. Sixteen amblyopic participants (8 females), comprising 8 individuals with strabismus, 7 with anisometropia, 1 with deprivational amblyopia, along with 8 individuals with normal visual acuity (1 female), participated in this study for whom, we measured the difference between the response to stimulation of the two eyes, across early visual areas (V1-V4). In controls, as expected from the organization of OD columns, the evoked OD response formed a striped pattern that was mostly confined to V1. Compared to controls, the OD response in amblyopic participants formed larger fused patches that extended into downstream visual areas. Moreover, both anisometropic and strabismic participants showed stronger OD responses in V1, as well as in downstream visual areas V2-V4. Although this increase was most pronounced in V1, the correlation between the OD response level and the interocular visual acuity difference (measured behaviorally) was stronger in higher-level visual areas (V2-V4). Beyond these common effects, and despite similar densities of amblyopia between the anisometropic and strabismic participants, we found a greater increase in the size of V1 portion that responded preferentially to fellow eye stimulation in anisometropic compared to strabismic individuals. We also found a greater difference between the amplitudes of the response to binocular stimulation, in those regions that responded preferentially to the fellow vs. amblyopic eye, in anisometropic compared to strabismic subjects. In contrast, strabismic subjects demonstrated increased correlation between the OD responses evoked within V1 superficial and deep cortical depths, whereas anisometropic subjects did not. These results provide some of the first direct functional evidence for distinct impacts of strabismus and anisometropia on the mesoscale functional organization of the human visual system, thus extending what was inferred previously about amblyopia from animal models.

This cross-sectional study used data from a large nationwide registry to describe the factors associated with use of botulinum toxin injections for adults with strabismus in the United States. Botulinum toxin injections were performed on 3.1% of adults undergoing an intervention for strabismus between 2013 and 2020. Adults treated with botulinum toxin injections were more likely to be older and female. Compared to non-Hispanic White patients, non-Hispanic Black patients were three times less likely to receive treatment with botulinum toxin after adjusting for age, sex, geographic region, and type of insurance. Efforts to understand the factors contributing to disparities in the use of botulinum toxin for strabismus may lead to opportunities for more equitable access to this intervention., (Copyright © 2024 American Association for Pediatric Ophthalmology and Strabismus. Published by Elsevier Inc. All rights reserved.)

Importance: There is no consensus on the optimal surgical treatment for children with intermittent exotropia (IXT)., Objective: To compare the 5-year reoperation rates for children with IXT treated with horizontal muscle strabismus surgery using bilateral lateral rectus recession (BLR) vs unilateral lateral rectus recession with medial rectus resection (RR)., Design, Setting, and Participants: This cohort study examined data obtained from the Intelligent Research in Sight (IRIS) Registry on 7482 children (age, <18 years) with IXT who underwent horizontal eye muscle strabismus surgery between January 1, 2013, and December 31, 2017. Children undergoing initial surgeries involving 3 or more horizontal muscles, vertical muscles, or reoperations were excluded., Main Outcomes and Measures: The primary outcome was the adjusted cumulative incidence of repeat horizontal muscle surgery within 5 years after the initial surgery. Reoperation risk was analyzed using adjusted hazard ratios (AHRs) derived from multivariable Cox regression models, adjusting for individual demographic and surgical factors (age, sex, race and ethnicity, US Census region, and surgeon subspecialty). Data were analyzed between January 16 and September 20, 2023., Results: The study included 7482 children (median [IQR] age at initial surgery, 6 [4-9] years; 3945 females [53%]) with IXT treated with horizontal muscle strabismus surgery. Bilateral lateral rectus recession was performed more frequently than RR (85.3% vs 14.7%, P < .001), especially in younger children (rates of BLR vs RR by age: age 0 to ≤4 years, 88.4% vs 11.6%; age 5 to ≤11 years, 84.7% vs 15.3%; age 12 to ≤17 years, 78.1% vs 21.9%; P < 0.001). After data adjustment, the 5-year cumulative incidence of reoperation was 21.3% (95% CI, 20.1%-22.5%). The adjusted 5-year cumulative incidence of reoperation was higher for BLR than for RR (22.2% vs 17.2%; difference, 4.9%; 95% CI, 1.9%-8.0%). Unilateral lateral rectus recession with medial rectus resection was associated with a lower 5-year reoperation risk compared with BLR (AHR, 0.77; 95% CI, 0.64-0.93). Younger age at time of initial surgery was associated with a higher reoperation risk (AHR per 1-year decrease, 1.09; 95% CI, 1.07-1.11) after adjusting for all other covariates., Conclusions and Relevance: In this nationwide registry, approximately 1 in 5 children with IXT underwent reoperation within 5 years after the initial surgery. Children treated with RR were less likely to require a reoperation within 5 years compared with those treated with BLR. Further efforts to identify modifiable risk factors for reoperation are needed to reduce the surgical burden and improve outcomes for children with IXT.

Objective: To quantify changes in ductions following nasal transposition of the split lateral rectus muscle (NTSLR) for treating third nerve palsy., Design: Retrospective cohort study., Participants: A single eye from each patient with third nerve palsy treated with NTSLR with ocular motility measurements., Methods: Observation of changes in pre- and postoperative ductions. Outcome measures including patient demographic and surgical factors associated with the ability to adduct beyond the midline after NTSLR were evaluated using multivariable logistic regression., Results: A total of 116 patients met the inclusion criteria for this study. The NTSLR significantly decreased abduction (median of 0 limitation [interquartile range (IQR), 0-0] prior to surgery to -4 [IQR, -4 to -3] after NTSLR; p < 0.001), with a corresponding improvement in adduction (median, -5 [IQR, -5 to -4] prior to surgery to -4 [IQR, -4 to -3] after NTSLR; p < 0.001). There was no change in median supraduction or infraduction after NTSLR (p > 0.05). The ability to adduct beyond the midline after NTSLR was demonstrated in 42% of patients. Although not statistically significant, a trend toward a postoperative ability to adduct beyond the midline was seen in patients who had concurrent superior oblique muscle tenotomy (odds ratio [OR] = 5.08; 95% CI, 0.91-40.9) or who were designated with partial rather than complete third nerve palsy (OR = 2.29; 95% CI, 0.82-6.70)., Conclusions: NTSLR improves the horizontal midline positioning of eyes with third nerve palsy. Most eyes lose the ability to abduct, but some regain a modest ability to adduct while vertical ductions remain unchanged., (Copyright © 2022 Canadian Ophthalmological Society. Published by Elsevier Inc. All rights reserved.)

Importance: Untreated refractive error contributes to the racial, ethnic, and socioeconomic disparities in visual function of adolescent children in the US., Objective: To describe patterns in vision testing as a function of age among US adolescents and identify sociodemographic factors associated with vision testing., Design, Setting, and Participants: This cross-sectional study used data from the National Survey of Children's Health (2018-2019), a nationally representative survey of the noninstitutionalized US pediatric population. A total of 24 752 adolescent children (aged 12 to <18 years) were included. Data were analyzed from March 22 to August 11, 2023., Main Outcomes and Measures: The primary outcome was the caregiver report of vision testing within the last 12 months. Linear regression was used to describe the patterns in reported vision testing as a function of participant age. Logistic regression was used to describe the association of sociodemographic factors with the report of vision testing in each setting., Results: Among 24 752 adolescents, the median (IQR) age was 14 (13-16) years; 12 918 (weighted, 51%) were male. Vision testing in any setting within the previous year was reported by caregivers of 18 621 adolescents (weighted, 74%). Vision testing was reported to have occurred at an eye clinic in 13 323 participants (weighted, 51%), at a primary care clinic in 5230 participants (weighted, 22%), at a school in 2594 participants (weighted, 11%), and at a health center in 635 participants (weighted, 4%). The percentage of adolescents reported to have vision tested decreased with age (-1.3% per year; 95% CI, -2.5% to 0% per year) due to a decrease in testing in primary care and school settings. After adjusting for age and sex, there were lower odds of vision testing reported for adolescents who were uninsured vs insured (adjusted odds ratio [AOR], 0.81; 95% CI, 0.76-0.87), had caregivers with less than vs greater than high school education (AOR, 0.89; 95% CI, 0.84-0.95), and were from a family born outside vs inside the US (AOR, 0.90; 95% CI, 0.82-0.98)., Conclusions and Relevance: In this cross-sectional study, vision testing in adolescents decreased as a function of age due to fewer reported tests performed in primary care and school-based settings. Relative to children in socioeconomically advantaged families, those from disadvantaged families were less likely to report receiving vision testing in clinical settings. Efforts to expand the role of school-based vision testing for older adolescents from disadvantaged backgrounds may enable opportunities to address disparities in untreated refractive error.

This study used data from the annual fellowship survey over 7 academic years (2014-15 to 2020-21) to describe the trends in surgical experience for pediatric ophthalmology and strabismus fellows and to quantify the impact of the COVID-19 pandemic on trainee surgical volume. The overall number of procedures performed by fellows in the primary surgeon role declined during the first academic year impacted by the pandemic but recovered in the second year. There was an increase in the number of intraocular cases performed per year during the 7-year study interval., (Copyright © 2023 American Association for Pediatric Ophthalmology and Strabismus. Published by Elsevier Inc. All rights reserved.)

Purpose: To compare amblyopia treatment outcomes between patients with neurodevelopmental disorders and their typically developing peers., Methods: Of 2,311 patients diagnosed with amblyopia between 2010 and 2014 at Boston Children's Hospital, 460 met inclusion criteria (age 2-12 with anisometropic, strabismic, or mixed amblyopia [interocular difference (IOD) ≥2 lines]). Treatment and visual outcomes were analyzed according to neurodevelopmental status: neurodevelopmental delay (DD) versus typical development (TD)., Results: The DD group (n = 54) and TD group (n = 406) were similar in demographics, amblyogenic risk factors, baseline visual measures, prescribed therapy, and adherence (P ≥ 0.10). Between-visit follow-up time was longer for the DD group (0.65 [0.42- 0.97] years) than for the TD group (0.5 [0.36-0.82] years; P = 0.023). IOD improved similarly in each group by the last visit (DD, -0.15 logMAR [-0.31 to -0.02]; TD, -0.2 logMAR [-0.38 to -0.1]; P = 0.09). Each group reached amblyopia resolution by the last visit at similar frequencies (DD, 23/54 [43%]; TD, 211/406 [52%]; P > 0.2). DD diagnosis did not independently influence amblyopia resolution (HR, 0.77; 95% CI, 0.53-1.12; P = 0.17), but each additional month of interval time between follow-up visits reduced the likelihood of resolution by 2.7% (HR, 0.67; 95% CI, 0.51-0.87; P = 0.003)., Conclusions: Patients with DD and those with TD responded similarly to amblyopia therapy; however, follow-up intervals were longer in patients with DD and correlated with the likelihood of persistent amblyopia, suggesting that greater efforts at assuring follow-up may benefit patients with DD., (Copyright © 2023 American Association for Pediatric Ophthalmology and Strabismus. Published by Elsevier Inc. All rights reserved.)

Objectives: To describe trends in vision screening based on insurance claims for young children in the United States., Methods: This cross-sectional study used administrative claims data from the 2010-2019 IBM MarketScan Commercial Claims and Encounters Database. We included children aged 1 to <5 years at the beginning of each calendar year. The primary outcome was a vision screening claim within 12 months for chart-based or instrument-based screening. Linear regression was used to evaluate trends over time in vision screening claims and practitioner payment., Results: This study included a median of 810 048 (interquartile range, 631 523 - 1 029 481) children between 2010 and 2019 (mean [standard deviation] age, 2.5 [1.1] years; 48.7% female). The percentage of children with vision screening claims increased from 16.7% in 2010 to 44.3% in 2019 (difference, 27.5%; 95% confidence interval, 27.4% to 27.7%). Instrument-based screening claims, which were identified in <0.2% of children in 2010, increased to 23.4% of children 1 to <3 years old and 14.4% of children 3 to <5 years old by 2019. From 2013 to 2018, the average of the median practitioner payment for instrument-based screening was $23.70, decreasing $2.10 per year during this time (95% confidence interval, $0.85 to $3.34; P = .009)., Conclusions: Vision screening claims among young children nearly tripled over the last decade, and this change was driven by increased instrument-based screening for children aged <3 years. Further investigation is needed to determine whether the decreasing trends in practitioner payment for screening devices will reduce the adoption of vision screening technology in clinical practice., (Copyright © 2023 by the American Academy of Pediatrics.)

Purpose: Published studies of amblyopia include only patients with visual acuity (VA) worse than 20/40 in one or both eyes. The purpose of this study is to evaluate patients diagnosed and treated as amblyopic despite not meeting traditional VA criteria for amblyopia., Design: Retrospective clinical cohort study., Methods: Setting: Institutional practice., Patient Population: All patients diagnosed with amblyopia at Boston Children's Hospital between 2010 and 2014., Inclusion Criteria: VA better than 20/40 but not correctable to 20/20 in one or both eyes; age 2 to 12 years., Observations: Demographics, VA, baseline characteristics., Outcome Measures: Resolution, defined as VA 20/20 in both eyes; stereopsis at the last follow-up., Results: Of 2311 patients reviewed, 464 (20.1%) had subthreshold amblyopia. A majority (61.7%) had an amblyogenic factor, most commonly anisometropia (32.8%). Patients were followed for a median of 3.1 years; nearly all (97.5%) were treated. Of 318 patients who returned for follow-up, 47.8% achieved resolution, including 55.7% of treatment-naïve patients, and 62.5% (5 of 8 patients) offered observation alone. Median stereopsis improved by 0.4 log units in those who achieved resolution, with no change in those with persistent amblyopia. In the multivariate analysis, a longer length of follow-up was significantly associated with resolution of subthreshold amblyopia (odds ratio: 1.38; 95% confidence interval: 1.22-1.57, P < .001)., Conclusions: Patients with subthreshold amblyopia represent a sizeable cohort in real-world amblyopia practice. When offered treatment, half achieved 20/20 vision in both eyes with improved stereopsis as well. Further studies are needed to assess whether observation alone would result in similar outcomes., (Copyright © 2022 Elsevier Inc. All rights reserved.)