Literature DB >> 18287946

A model of behavioral treatments for self-mutilation behavior in Lesch-Nyhan syndrome.

Eric A Zilli1, Michael E Hasselmo.   

Abstract

Lesch-Nyhan syndrome is a rare genetic disorder, caused by a mutation in the gene coding for the enzyme hypoxanthine-guanine phosphoribosyltransferase, which is characterized by hyperuricemia and its associated symptoms along with motor disorders and compulsive self-mutilation. We show that the temporal difference learning algorithm that has been often used to interpret dopaminergic activity in the basal ganglia offers an explanation for the self-mutilation behaviors. We propose that a dysfunctional dopamine signal inadvertently reinforces early, accidental injurious behavior that is initially caused by clumsiness owing to the motor disorders. Simulations of this model reproduce findings on the results of behavioral treatments for dealing with self-mutilation behaviors.

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Year:  2008        PMID: 18287946      PMCID: PMC2408668          DOI: 10.1097/WNR.0b013e3282f5f76f

Source DB:  PubMed          Journal:  Neuroreport        ISSN: 0959-4965            Impact factor:   1.837


  25 in total

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Review 8.  Delineation of the motor disorder of Lesch-Nyhan disease.

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4.  Congenital anomalies-associated Riga-Fede disease as an early manifestation of Lesch-Nyhan syndrome: rare entities in the same pediatric patient-a case report.

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