A case report on the efficacy of vigabatrin analogue (1S, 3S)-3-amino-4-difluoromethylenyl-1-cyclopentanoic acid (CPP-115) in a patient with infantile spasms

Kyra Doumlele; Erin Conway; Julie Hedlund; Patricia Tolete; Orrin Devinsky

doi:10.1016/j.ebcr.2016.08.002

A case report on the efficacy of vigabatrin analogue (1S, 3S)-3-amino-4-difluoromethylenyl-1-cyclopentanoic acid (CPP-115) in a patient with infantile spasms

Epilepsy Behav Case Rep. 2016 Aug 21:6:67-9. doi: 10.1016/j.ebcr.2016.08.002. eCollection 2016.

Authors

Kyra Doumlele¹, Erin Conway¹, Julie Hedlund¹, Patricia Tolete¹, Orrin Devinsky¹

Affiliation

¹ New York University School of Medicine, Comprehensive Epilepsy Center, 223 East 34th Street, New York, NY 10016, USA.

Abstract

West Syndrome is characterized by infantile spasms, a hypsarrhythmic electroencephalogram (EEG) pattern, and a poor neurodevelopmental prognosis. First-line treatments include adrenocorticotrophic hormone (ACTH) and vigabatrin, but adverse effects often limit their use. CPP-115 is a high-affinity vigabatrin analogue developed to increase therapeutic potency and to limit retinal toxicity. Here, we present a child treated with CPP-115 through an investigational new drug protocol who experienced a marked reduction of seizures with no evidence of retinal dysfunction. Given the potential consequences of ongoing infantile spasms and the limitations of available treatments, further assessment of CPP-115 is warranted.

Keywords: CPP-115; Epilepsy; Epileptic spasms; Infantile spasms; West Syndrome.

Publication types

Case Reports