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ANATOMICAL AND FUNCTIONAL OUTCOMES OF BEVACIZUMAB TREATMENT IN PEDIATRIC AUTOSOMAL RECESSIVE BESTROPHINOPATHY.

Publication ,  Journal Article
Jacobs, M; El-Rashedy, M; Fowler, N; Shirkey, B; Kitchens, J; Maldonado, RS
Published in: Retin Cases Brief Rep
May 1, 2024

PURPOSE: The purpose of this study was to report functional and anatomical outcomes of anti-VEGF treatment in eyes with autosomal recessive bestrophinopathy (ARB) presenting in the first decade of life. METHODS: The study included case series of four eyes from two siblings with compound heterozygous mutations in the BEST1 gene who were treated with eight monthly intravitreal bevacizumab injections. Response to treatment was analyzed using color fundus photography, fundus autofluorescence, optical coherence tomography, OCT angiography, and microperimetry. RESULTS: Patient 1 (male, age 9 years) had visual acuity of 20/20 in the right eye and 20/50 in the left eye. Patient 2 (female, age 10 years) had visual acuity of 20/25 in the right eye and 20/20 in the left eye. All eyes had multifocal subretinal deposition of lipofuscin and subretinal fluid, and three eyes had choroidal neovascularization (CNV). Lipofuscin material reabsorbed in 2 of 4 eyes, the CNV regressed in 3 of 3 eyes, a bacillary detachment resolved in 1 of 1 eye, but the subretinal fluid did not change. Functional improvement in visual acuity was noted, but MP showed scattered areas of reduced retinal sensitivity. No ocular or systemic side effects were detected. CONCLUSION: Anti-VEGF treatment of choroidal neovascularization in eyes with ARB resulted in anatomical changes that were only clinically significant in the eye with decreased visual acuity. The hyporeflective subretinal material remained unchanged suggesting a nonexudative cause. These findings provide new insights into the management of ARB, especially in pediatric subjects with CNV.

Duke Scholars

Published In

Retin Cases Brief Rep

DOI

EISSN

1937-1578

Publication Date

May 1, 2024

Volume

18

Issue

3

Start / End Page

378 / 386

Location

United States

Related Subject Headings

  • Visual Acuity
  • Treatment Outcome
  • Tomography, Optical Coherence
  • Retinal Diseases
  • Male
  • Intravitreal Injections
  • Humans
  • Fluorescein Angiography
  • Female
  • Eye Diseases, Hereditary
 

Citation

APA
Chicago
ICMJE
MLA
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Jacobs, M., El-Rashedy, M., Fowler, N., Shirkey, B., Kitchens, J., & Maldonado, R. S. (2024). ANATOMICAL AND FUNCTIONAL OUTCOMES OF BEVACIZUMAB TREATMENT IN PEDIATRIC AUTOSOMAL RECESSIVE BESTROPHINOPATHY. Retin Cases Brief Rep, 18(3), 378–386. https://doi.org/10.1097/ICB.0000000000001390
Jacobs, Mitchell, May El-Rashedy, Nicholas Fowler, Belinda Shirkey, John Kitchens, and Ramiro S. Maldonado. “ANATOMICAL AND FUNCTIONAL OUTCOMES OF BEVACIZUMAB TREATMENT IN PEDIATRIC AUTOSOMAL RECESSIVE BESTROPHINOPATHY.Retin Cases Brief Rep 18, no. 3 (May 1, 2024): 378–86. https://doi.org/10.1097/ICB.0000000000001390.
Jacobs M, El-Rashedy M, Fowler N, Shirkey B, Kitchens J, Maldonado RS. ANATOMICAL AND FUNCTIONAL OUTCOMES OF BEVACIZUMAB TREATMENT IN PEDIATRIC AUTOSOMAL RECESSIVE BESTROPHINOPATHY. Retin Cases Brief Rep. 2024 May 1;18(3):378–86.
Jacobs, Mitchell, et al. “ANATOMICAL AND FUNCTIONAL OUTCOMES OF BEVACIZUMAB TREATMENT IN PEDIATRIC AUTOSOMAL RECESSIVE BESTROPHINOPATHY.Retin Cases Brief Rep, vol. 18, no. 3, May 2024, pp. 378–86. Pubmed, doi:10.1097/ICB.0000000000001390.
Jacobs M, El-Rashedy M, Fowler N, Shirkey B, Kitchens J, Maldonado RS. ANATOMICAL AND FUNCTIONAL OUTCOMES OF BEVACIZUMAB TREATMENT IN PEDIATRIC AUTOSOMAL RECESSIVE BESTROPHINOPATHY. Retin Cases Brief Rep. 2024 May 1;18(3):378–386.

Published In

Retin Cases Brief Rep

DOI

EISSN

1937-1578

Publication Date

May 1, 2024

Volume

18

Issue

3

Start / End Page

378 / 386

Location

United States

Related Subject Headings

  • Visual Acuity
  • Treatment Outcome
  • Tomography, Optical Coherence
  • Retinal Diseases
  • Male
  • Intravitreal Injections
  • Humans
  • Fluorescein Angiography
  • Female
  • Eye Diseases, Hereditary