Original articleCharacteristic retinal atrophy with secondary “inverse” optic atrophy identifies vigabatrin toxicity in children☆
Section snippets
Participants and methods
To monitor possible vigabatrin visual toxicity, the current practice at The Hospital for Sick Children (HSC) is to assess each child with seizure activity before and during drug treatment with neuro-ophthalmologic examination and with an ERG (under sedation in most cases). Electroretinograms are recorded according to the standards of The International Society for Clinical Electrophysiology of Vision (ISCEV).19 Because ERGs in young children who are still developing, ERGs are compared with
Results
During this protocol, 3 cases of a specific, identifiable, vigabatrin-associated peripheral retinal atrophy with secondary optic nerve atrophy in children have been identified and selected for this communication. The authors report the characteristic pattern of atrophy that is found in association with the peripheral field loss caused by this drug in young children. In 1 case, ERGs were recorded sequentially, and results show dramatic parallel deterioration. This clinically characteristic
Discussion
Three children, aged 12 years, 15 years, and 2 years are described, in whom a severe and atypical form of retinal and optic atrophy has been recognized in association with extended use of the anticonvulsant vigabatrin, peripheral field loss (presumed or clinically detectable), and ERG abnormalities. Because clinical assessment of peripheral visual loss, especially in its milder forms, is so difficult and unreliable in young children, clinicians have turned to investigational support of
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Manuscript no. 230580.
Technical support for electroretinogram and visual evoked potential measures was partially funded by The Hospital for Sick Children Research Institute, Toronto, Canada (seed grant), and the University of Toronto Vision Science Research Program, Toronto, Canada.