Optical coherence tomography to monitor vigabatrin toxicity in children

Copyright © 2016 by the American Association for Pediatric Ophthalmology and Strabismus. Purpose The antiepileptic drug vigabatrin is known to cause permanent loss of vision. Both visual field testing and electroretinogram are used to detect retinal damage. Adult data on optical coherence tomography (OCT) shows that retinal nerve fiber layer (RNFL) thinning may be an early indicator of vigabatrin-induced retinal toxicity. The purpose of this study was to investigate whether OCT can detect early vigabatrin-induced retinal toxicity in children. Methods Pediatric patients (≤18 years of age) requiring vigabatrin for seizure control who were followed at our institution were invited to participate. Patients were examined according to manufacturer guidelines, with most examinations taking place under general anesthesia. RNFL thickness was measured by OCT (Stratus Model 3000, Zeiss) and compared to total cumulative dose of vigabatrin. In most cases, indirect ophthalmoscopy, fundus photography, and electroretinography were also performed. Results OCT and complete dosing data was available for 19 patients. Patients with tuberous sclerosis (TS, n = 12) received higher cumulative doses (mean, 1463 g) than non-TS patients (mean, 351 g, P = 0.044). RNFL thinning was detected in the nasal (P \u3c 0.01), superior (P \u3c 0.01), and inferior (P \u3c 0.05) quadrants in patients with TS, particularly once cumulative dose exceeded 1500 g. Conclusions In our study population of patients with TS, higher cumulative doses of vigabatrin were associated with RNFL thinning in the nasal, superior, and inferior quadrants. These findings were pronounced once cumulative dose exceeded 1500 g. This pattern of RNFL thinning is similar to what has been shown in adult patients taking vigabatrin

Optical coherence tomography to monitor vigabatrin toxicity in children

Copyright © 2016 by the American Association for Pediatric Ophthalmology and Strabismus. Purpose The antiepileptic drug vigabatrin is known to cause permanent loss of vision. Both visual field testing and electroretinogram are used to detect retinal damage. Adult data on optical coherence tomography (OCT) shows that retinal nerve fiber layer (RNFL) thinning may be an early indicator of vigabatrin-induced retinal toxicity. The purpose of this study was to investigate whether OCT can detect early vigabatrin-induced retinal toxicity in children. Methods Pediatric patients (≤18 years of age) requiring vigabatrin for seizure control who were followed at our institution were invited to participate. Patients were examined according to manufacturer guidelines, with most examinations taking place under general anesthesia. RNFL thickness was measured by OCT (Stratus Model 3000, Zeiss) and compared to total cumulative dose of vigabatrin. In most cases, indirect ophthalmoscopy, fundus photography, and electroretinography were also performed. Results OCT and complete dosing data was available for 19 patients. Patients with tuberous sclerosis (TS, n = 12) received higher cumulative doses (mean, 1463 g) than non-TS patients (mean, 351 g, P = 0.044). RNFL thinning was detected in the nasal (P \u3c 0.01), superior (P \u3c 0.01), and inferior (P \u3c 0.05) quadrants in patients with TS, particularly once cumulative dose exceeded 1500 g. Conclusions In our study population of patients with TS, higher cumulative doses of vigabatrin were associated with RNFL thinning in the nasal, superior, and inferior quadrants. These findings were pronounced once cumulative dose exceeded 1500 g. This pattern of RNFL thinning is similar to what has been shown in adult patients taking vigabatrin

Schwannoma in the lower lip mucosa: Unexpected diagnosis

Schwannoma is a benign tumor arising from nerve-sheath Schwann cells. When it affects the mouth, the lips are rarely involved. A MEDLINE search in the English literature from 1969 to 2009 revealed only 17 documented cases of schwannomas of the lip. This report describes the unusual case of a 52-year-old man who developed a schwannoma of the lower lip. Histopathologic examination and immunohistochemical staining studies were performed. Although rare, schwannoma should be considered in the differential diagnosis of any nodule or mass in the oral mucosa. (Quintessence Int 2010; 41: 769-771