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VIGABATRIN TOXICITY IN INFANCY IS ASSOCIATED WITH RETINAL DEFECT IN ADOLESCENCE: A Prospective Observational Study
Tom Wright1, Ananthavalli Kumarappah, Aphrodite Stavropoulos
1*Department of Ophthalmology and Vision Science, The Hospital for Sick Children, Toronto, Ontario, Canada; and †University of Toronto, Toronto, Ontario, Canada.
Insights
Vigabatrin (VGB) treatment in childhood can cause retinal toxicity, leading to long-term visual field loss and optic nerve damage in adolescence. Early electroretinogram monitoring is crucial for identifying these vision defects.
Area of Science:
- Ophthalmology
- Neuroscience
- Pediatric Neurology
Background:
- Vigabatrin (VGB) is an anti-epileptic drug with known potential for retinal toxicity.
- Early identification of VGB-attributed retinal defects is essential for monitoring long-term visual function.
Purpose of the Study:
- To investigate the association between early childhood electroretinogram-defined vigabatrin (VGB) retinal toxicity and adolescent visual system defects.
- To assess long-term visual field and retinal nerve fiber layer (RNFL) thickness after VGB discontinuation.
Main Methods:
- Prospective cross-sectional study of 24 children with prior VGB exposure.
- Group I (10 children) had electroretinogram-diagnosed VGB retinal defects; Group II (14 children) did not.
- Monocular visual fields (Goldmann perimetry) and optic nerve head RNFL thickness (OCT) were measured.
Main Results:
- Visual field loss was observed in Group I participants using Goldmann kinetic perimetry.
- Optical coherence tomography (OCT) revealed significant RNFL attenuation in Group I eyes compared to Group II.
- OCT data showed distinct differences in RNFL thickness between the two groups.
Conclusions:
- Early electroretinogram findings of VGB-attributed retinal toxicity are associated with persistent visual field defects in adolescence.
- Retinal nerve fiber layer attenuation is a measurable consequence of VGB toxicity identified in infancy.
- These findings highlight the importance of long-term ophthalmological monitoring for children treated with VGB.
Purpose:
The purpose was to determine whether vigabatrin (VGB) (Sabril)-attributed retinal toxicity defined by electroretinogram in early childhood is associated with visual system defect in adolescents after discontinuation of VGB.
Methods:
This prospective cross-sectional study included 24 children previously treated with VGB and monitored in early childhood by electroretinogram for VGB-attributed retinal defects. Ten had been diagnosed with VGB-attributed retinal defect (Group I) and 14 had no VGB-attributed retinal defect (Group II). Outcome measures were extent of monocular visual fields using Goldmann kinetic perimetry and RNFL thickness at the optic nerve head, using optical coherence tomography.
Results:
Of those able to complete testing (6 eyes Group I and 16 eyes Group II), Goldmann results revealed results of visual field loss in Group I and not in Group II. The optical coherence tomography results demonstrated attenuation of the RNFL in all 6 eyes of Group I participants and in only 1 eye of 10 Group II participants. Optical coherence tomography data were nonoverlapping between Group 1 and Group II eyes.
Conclusion:
The VGB-attributed retinal toxicity identified by means of electroretinogram in infancy was associated with visual field loss and RNFL attenuation of the retinal nerve when tested in adolescence.
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