title: Optical coherence tomography to monitor vigabatrin toxicity in children.
abstract: OBJECTIVE
  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 < 0.01), superior (P < 0.01), and inferior (P < 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.
authors.affiliation: Children's National Health System, Washington, District of Columbia. Electronic address: coriglie@childrensnational.org.
authors.forename: Catherine
authors.initials: C
authors.lastname: Origlieri
authors.affiliation: Helen DeVos Children's Hospital, Grand Rapids, Michigan.
authors.forename: Brooke
authors.initials: B
authors.lastname: Geddie
authors.affiliation: Children's National Health System, Washington, District of Columbia.
authors.forename: Bethany
authors.initials: B
authors.lastname: Karwoski
authors.affiliation: Children's National Health System, Washington, District of Columbia.
authors.forename: Madison M
authors.initials: MM
authors.lastname: Berl
authors.affiliation: Children's National Health System, Washington, District of Columbia.
authors.forename: Nancy
authors.initials: N
authors.lastname: Elling
authors.affiliation: Children's National Health System, Washington, District of Columbia.
authors.forename: William
authors.initials: W
authors.lastname: McClintock
authors.affiliation: University of Maryland, Baltimore, Maryland.
authors.forename: Janet
authors.initials: J
authors.lastname: Alexander
authors.affiliation: Children's National Health System, Washington, District of Columbia.
authors.forename: Marlet
authors.initials: M
authors.lastname: Bazemore
authors.affiliation: Children's National Health System, Washington, District of Columbia.
authors.forename: Heather
authors.initials: H
authors.lastname: de Beaufort
authors.affiliation: Sidra Medical and Research Center, Doha, Quatar.
authors.forename: Kelly
authors.initials: K
authors.lastname: Hutcheson
authors.affiliation: Children's National Health System, Washington, District of Columbia.
authors.forename: Marijean
authors.initials: M
authors.lastname: Miller
authors.affiliation: Children's National Health System, Washington, District of Columbia.
authors.forename: Jonathan
authors.initials: J
authors.lastname: Taylormoore
authors.affiliation: Children's National Health System, Washington, District of Columbia.
authors.forename: Mohamad S
authors.initials: MS
authors.lastname: Jaafar
authors.affiliation: Children's National Health System, Washington, District of Columbia.
authors.forename: William
authors.initials: W
authors.lastname: Madigan
mesh_terms: D000293:Adolescent; D000927:Anticonvulsants / Q000633:toxicity*; D002648:Child; D002675:Child, Preschool; D004596:Electroretinography / Q000187:drug effects; D005260:Female; D006801:Humans; D007223:Infant; D008297:Male; D009412:Nerve Fibers / Q000473:pathology*; D011446:Prospective Studies; D012160:Retina / Q000187:drug effects / Q000473:pathology*; D012164:Retinal Diseases / Q000139:chemically induced / Q000175:diagnosis*; D012165:Retinal Ganglion Cells / Q000473:pathology*; D041623:Tomography, Optical Coherence; D014402:Tuberous Sclerosis / Q000188:drug therapy; D020888:Vigabatrin / Q000633:toxicity*; D014794:Visual Fields / Q000187:drug effects
chemical_list: D000927:Anticonvulsants; D020888:Vigabatrin