Infantile spasms in Down syndrome: good response to a short course of vigabatrin.
OBJECTIVE
To evaluate the efficacy of vigabatrin (VGB) in the treatment of infantile spasms (ISs) associated with Down syndrome (DS) and to assess the feasibility of early discontinuation to reduce the possible retinal toxicity.


METHODS
Five children with ISs with DS were treated with vigabatrin as first-line monotherapy in an open prospective study. The short-term response was evaluated, and VGB was continued in responders. The treatment was stopped after 6 months in children who were still spasm free.


RESULTS
Four children of five became spasm free with VGB, three of them responding within 1 week. This response was maintained during the 6 months of VGB treatment. After VGB discontinuation, and with a follow-up ranging from 2 to 4 years, none of the responders experienced spasm recurrence or other types of seizures.


CONCLUSIONS
This study confirms the efficacy of VGB in ISs associated with DS. Moreover, it shows that the duration of VGB treatment can be reduced to 6 months without relapse of ISs. This short treatment might reduce the risk of developing visual field constriction.
Nabbout
R
R
Department of Pediatrics, Hôpital Hôtel Dieu de France, Université St. Joseph, Beirut, Lebanon.
Melki
I
I
Gerbaka
B
B
Dulac
O
O
Akatcherian
C
C
D000293:Adolescent; D017668:Age of Onset; D000927:Anticonvulsants / Q000008:administration & dosage / Q000627:therapeutic use*; D002648:Child; D015897:Comorbidity; D004314:Down Syndrome / Q000453:epidemiology*; D004334:Drug Administration Schedule; D005240:Feasibility Studies; D005260:Female; D006801:Humans; D007223:Infant; D008297:Male; D011446:Prospective Studies; D013036:Spasms, Infantile / Q000188:drug therapy* / Q000453:epidemiology; D016896:Treatment Outcome; D020888:Vigabatrin / Q000008:administration & dosage / Q000627:therapeutic use*
D000927:Anticonvulsants; D020888:Vigabatrin