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ANATOMICAL AND FUNCTIONAL OUTCOMES OF BEVACIZUMAB TREATMENT IN PEDIATRIC AUTOSOMAL RECESSIVE BESTROPHINOPATHY
Mitchell Jacobs1, May El-Rashedy1, Nicholas Fowler1
1Department of Ophthalmology and Visual Sciences, University of Kentucky, Lexington, Kentucky.
Retinal Cases & Brief Reports
|February 2, 2023
Summary
Anti-VEGF treatment for autosomal recessive bestrophinopathy (ARB) showed some anatomical improvements like reduced lipofuscin and choroidal neovascularization (CNV) regression. However, functional gains were limited, with subretinal fluid persisting.
Area of Science:
- Ophthalmology
- Genetics
- Retinal Diseases
Background:
- Autosomal recessive bestrophinopathy (ARB) is a genetic retinal disorder.
- Early-onset ARB can present with subretinal fluid and choroidal neovascularization (CNV).
- Anti-VEGF therapy is a potential treatment for CNV in various retinal conditions.
Purpose of the Study:
- To evaluate the functional and anatomical outcomes of anti-VEGF treatment in pediatric ARB.
- To assess the efficacy of intravitreal bevacizumab in eyes with ARB and CNV.
Main Methods:
- Case series of four eyes from two siblings with compound heterozygous BEST1 mutations.
- Treatment involved eight monthly intravitreal bevacizumab injections.
- Evaluated using fundus photography, autofluorescence, OCT, OCT angiography, and microperimetry.
Main Results:
- Improvement in visual acuity was noted in some eyes.
- Lipofuscin reabsorbed in 50% of eyes; CNV regressed in all treated eyes.
- Subretinal fluid persisted, and microperimetry showed reduced retinal sensitivity in some areas.
Conclusions:
- Anti-VEGF therapy led to anatomical changes in pediatric ARB eyes with CNV.
- Clinical significance was limited, particularly where subretinal fluid persisted.
- Findings suggest ARB may have nonexudative components and inform management strategies for pediatric CNV.
