PWSA (USA) Research Grant Approved July 2000…Published Results
Am J Ment Retard. 2004 Jul;109(4):301-9.
“Effects of topiramate in adults with Prader-Willi syndrome”
(Shapira NA, Lessig MC, Lewis MH, Goodman WK, Driscoll DJ)
College of Medicine, University of Florida, Gainesville 32610, USA.
http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?cmd=Retrieve&db=pubmed&dopt=Abstract&list_uids=15176917&query_hl=1&itool=pubmed_docsum 

Prader-Willi syndrome is a multisystem neurogenetic obesity disorder with behavioral manifestations, including hyperphagia, compulsive behavior, self-injury, and mild to moderate mental retardation. In an 8-week open-label study, we evaluated adjunctive therapy with the anticonvulsant topiramate in 8 adults with Prader-Willi syndrome. Appetite was measured by a 1-hour access to food four times throughout the study and quantified with a visual analogue scale. Topiramate did not significantly change calories consumed, Body Mass Index, or increase self-reported appetite. In addition, there were no significant changes in compulsions. Surprisingly, topiramate treatment resulted in a clinically significant improvement in the self-injury (i.e., skin-picking) that is characteristic of this syndrome. Potential benefits of topiramate for self-injury should be evaluated further in controlled trials.

PWSA (USA) Research Grant Approved July 2000…Published Results
Int J Neuropsychopharmacol. 2002 Jun;5(2):141-5
“ Topiramate attenuates self-injurious behaviour in Prader-Willi Syndrome”
(Shapira NA, Lessig MC, Murphy TK, Driscoll DJ, Goodman WK).
Department of Psychiatry, Evelyn F and William L McKnight Brain Institute of the
University of Florida, Gainsville 32610-0256, USA.
http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?cmd=Retrieve&db=pubmed&dopt=Abstract&list_uids=12135538&query_hl=1&itool=pubmed_docsum 

Self-injurious behaviour (SIB), most notably skin picking, has been described by various terms in the literature ranging from neurotic/psychogenic excoriations to compulsive/pathological skin picking. Prader-Willi Syndrome (PWS) is a neurogenetic multisystem disorder characterized by infantile hypotonia, mental retardation, short stature, hypogonadism, dysmorphic features, and hyperphagia with a high risk of obesity. Psychiatric manifestations include SIBs in the form of skin picking, nail biting and rectal gouging. Topiramate is a novel anti-epileptic medication without significant liability of weight gain. There are no published reports of topiramate being utilized in PWS or SIB. We report attenuation of SIB with resultant lesion healing in three PWS adults treated with topiramate in an 8-wk open-label trial. Although our findings should be treated with caution, they suggest that double-blind or cross-over studies with topiramate are warranted to establish the possible role of topiramate in attenuating SIB in PWS and other disorders that involve SIB.

edited: 08/19/2008