David G. Hunter

• MD, PhD
• Ophthalmologist-in-Chief at Boston Children's Hospital

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 reported the identification of variants in many candidate genes. Here, we descri...

Introduction 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...

We employed high-resolution fMRI to distinguish the impacts of anisometropia and strabismus amblyopia on the evoked ocular dominance (OD) response. Sixteen amblyopic participants (eight females) plus eight individuals with normal vision (one female), participated in this study for whom we measured the difference between the response to stimulation...

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...

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...

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 ophthalmolo...

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 posterio...

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 out...

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 patter...

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 pedigrees with genetically unsolved oCCDDs, integrating analyses of pedigrees, human and animal model phenotypes, and de novo var...

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...

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 estimate...

This cross-sectional study uses a nationally representative survey of the US pediatric population to identify gaps in the vision screening pathway.

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 resecti...

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 Participan...

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...

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 surgeo...

A 5-month-old boy was referred by the genetics service for evaluation of possible strabismus. He had been born with gastroschisis and spent his first 8 weeks of life in the hospital for treatment, which included surgical correction of the abdominal wall defect. His parents had been aware of the eye misalignment, tearing, and possible trouble with p...

An elevated threshold for neuroplasticity limits visual gains with treatment of residual amblyopia in older children and adults. Acetylcholinesterase inhibitors (AChEI) can enable visual neuroplasticity and promote recovery from amblyopia in adult mice. Motivated by these promising findings, we sought to determine whether donepezil, a commercially...

This cohort study examines patterns and out-of-pocket costs of instrument-based screening among children 12 to 36 months.

Objective: The head and intraocular trauma tool (HITT) is a portable, binocular retinal polarization scanner (RPS) that detects ocular fixation with high precision to assess visuomotor function. We conducted a pilot evaluation of a prototype binocular RPS device to evaluate alterations in fixation stability, binocularity (convergence), and saccadi...

Importance: Understanding the factors associated with nasolacrimal duct probing failure in young children may help inform practice patterns. Objective: To identify factors associated with repeated nasolacrimal duct probing in young children. Design, setting, and participants: This retrospective cohort study analyzed data from the Intelligent R...

Purpose: To describe the association of smoking status with surgical intervention for TED at the population-level. Design: Retrospective cohort study. Methods: SETTING: : US clinical data registry STUDY POPULATION: : All adults (age ≥18 years) with Graves disease in the Intelligent Research in Sight (IRIS®) Registry (1/1/2013-12/31/2020). Obs...

Addressing ocular misalignment secondary to partial and complete oculomotor nerve palsy presents a special challenge to the strabismus surgeon, particularly when treating patients with binocular diplopia. We review the reported surgical options and, through illustrative cases, provide our own perspective on managing this disorder.

Purpose: Published studies of amblyopia include only patients with visual acuity (VA) worse than 20/40 in one or both eyes. This study evaluates patients diagnosed and treated as amblyopic despite not meeting traditional visual acuity criteria for amblyopia. Design: Retrospective clinical cohort study. Methods: SETTING: INSTITUTIONAL PRACTICE....

Purpose: To report the indications, operative strategies, and surgical outcomes of patients who undergo vertical and horizontal rectus muscle surgery for incomitant strabismus despite being orthophoric in primary gaze. Design: Retrospective, interventional case series. Methods: • Setting: Academic practice at Boston Children's Hospital. • Pati...

Purpose: To demonstrate the use of a novel measure of neighborhood quality, the Child Opportunity Index (COI), for investigating health disparities in pediatric ophthalmology. Methods: This study included children 2-12 years of age from a registry of patients diagnosed with amblyopia at an urban pediatric hospital between 2010 and 2014. Children...

Strabismus, the condition of misaligned eyes, can result in severe, long-lasting functional and psychosocial sequelae. This review examines existing literature that has described and quantified the psychosocial consequences of strabismus. In particular, the role of strabismus in creating social, psychological, and vocational disparities, and how th...

Purpose To quantify changes in ductions following nasal transposition of the split lateral rectus muscle (NTSLR) for treating third nerve palsy. Design Retrospective cohort study. Setting International multicentre registry. Study population A single eye from each patient with third nerve palsy treated with NTSLR with ocular motility measurements...

This cohort study used data from the IRIS Registry to assess the risk factors associated with pterygium reoperation.

Subspecialty exposure during residency can influence the future pursuit of fellowship training. In this study, we compared the trends in strabismus surgical experience reported by graduating ophthalmology residents in the United States with other categories of ophthalmic surgery. Over the 10-year period (2010-2019), there was a decline in the total...

Prisms are a noninvasive tool used to manage diplopia in patients, especially when their strabismus is small and comitant. Prisms can be used for horizontal, vertical, or oblique (but not torsional) strabismus. Ground-in prisms provide the best image quality and cosmetic appearance. Fresnel prisms are best for strabismus that is thought to be tempo...

There is a nationwide shortage of pediatric ophthalmologists, with trends in fellowship applicants foreshadowing a continued reduction in the number of active pediatric ophthalmologists in the years ahead. In this study, we investigated whether similar fellowship applicant shortages exist in other pediatric surgical subspecialties. We compared the...

Purpose To compare the reoperation rates following strabismus surgery with and without adjustable suture technique. Design Retrospective cohort study: Participants: Patients ≥ 18 years of age in the IRIS Registry who underwent strabismus surgery between January 1st, 2013 and December 31st, 2018. Methods Data were collected from the electronic hea...

A proper interaction between muscle-derived collagen XXV and its motor neuron-derived receptors protein tyrosine phosphatases σ and δ (PTP σ/δ) is indispensable for intramuscular motor innervation. Despite this, thus far, pathogenic recessive variants in the COL25A1 gene had only been detected in a few patients with isolated ocular congenital crani...

The absence of clinical tools to evaluate individual variation in the pace of aging represents a major impediment to understanding aging and maximizing health throughout life. The lens is an ideal tissue for quantitative assessment of molecular aging in vivo. Long-lived proteins in lens fiber cells are expressed during fetal life, do not undergo tu...

Microtubules are formed from heterodimers of alpha- and beta-tubulin, each of which has multiple isoforms encoded by separate genes. Pathogenic missense variants in multiple different tubulin isoforms cause brain malformations. Missense mutations in TUBB3, which encodes the neuron-specific beta-tubulin isotype, can cause congenital fibrosis of the...

Purpose Describe outcomes after treatment of Moebius syndrome (MBS) esotropia by adjustable bilateral medial rectus recession (BMR) with and without augmented superior rectus transposition (SRT) Design Retrospective case series Methods Patients meeting 2014 diagnostic criteria for MBS and treated at Boston Children's Hospital between 2003 and 201...

We summarize venture capital investment into ophthalmology companies from 2011 to 2021 and quantify early-stage investments in biotechnology, pharmaceuticals, surgical devices, and diagnostic equipment. We describe the companies and investors most active in ophthalmologic innovation.

Purpose Digital therapeutics are a new class of interventions that are software-driven and intended to treat various conditions. We developed and evaluated a dichoptic digital therapeutic for amblyopia, a neurodevelopmental disorder for which current treatments may be limited by poor adherence and residual vision deficits. Design Randomized contro...

Purpose Many clinicians treat unilateral amblyopia with glasses alone and initiate patching when needed; others start glasses and patching simultaneously. In this study, we reviewed the outcomes of the two approaches at our institution. Design Retrospective non-randomized clinical trial Methods Setting: Institutional practice. Patient Population:...

Purpose To evaluate the best-corrected visual acuity and stereoacuity gains in children >7 years of age and adults with unilateral amblyopia treated with a prototype virtual reality–based binocular amblyopia therapy. Methods In this randomized, double masked, cross-in clinical trial, patients at Boston Children’s Hospital with unilateral anisometr...

Purpose To report the results of a clinical study designed to evaluate the accuracy of the blinq pediatric vision scanner, which detects amblyopia and strabismus directly by means of retinal polarization scanning, unlike other vision screening devices, which infer possible disease based on detection of refractive risk factors. Methods Subjects 1-2...

Purpose To evaluate outcomes of botulinum toxin (BTX) injection of the inferior oblique (IO) muscles. Design Retrospective case series. Methods Setting Single center, ophthalmology department at Boston Children's Hospital Study population All patients treated with IO muscle injection of BTX (onabotulinumtoxinA) between 2010 and 2020. Observatio...

It was my privilege to serve as Editor-in-Chief for J AAPOS from 2006 to 2012. During my tenure we made many changes, not all of which were welcome or effective but most of which seemed to improve the experience of authors and our readership. It is worth taking a moment to look back upon those years to reflect on the process and effects of change a...

Purpose To identify demographic and disease-related characteristics predictive of LTFU status in amblyopia treatment and create a risk model for predicting LTFU status. Design Retrospective cohort study Methods Setting: Single center, ophthalmology department at Boston Children's Hospital (BCH). Patients: 2037 patients treated for amblyopia at BC...

Background: The effectiveness of amblyopia therapy can be limited by poor adherence. Dichoptic therapies are a new approach, but recent trials have demonstrated difficulty maintaining high adherence over extended periods of at-home treatment. We evaluated the efficacy and adherence of Luminopia One-a dichoptic treatment that applies therapeutic mo...