Behavioral contingencies and self-mutilation in Lesch-Nyhan disease


Behavioral contingencies and self-mutilation in Lesch-Nyhan disease


Anderson L; Dancis J; Alpert M


Journal of Consulting and Clinical Psychology




Adolescent; Behavior Therapy/mt [Methods]; Child; Preschool; Humans; Lesch-Nyhan Syndrome/px [Psychology]; Lesch-Nyhan Syndrome/th [Therapy]; Male Punishment; Self Mutilation/th [Therapy]; behavioral problems; Lesch-Nyhan syndrome; psychological intervention; aversive stimulation electric shock; positive reinforcement; time-out; self-injury; self-mutilation


Lesch-Nyhan syndrome is a rare, sex-linked, recessive disease that is accompanied by severe self-mutilation, especially finger biting. Evidence is presented suggesting that parental response patterns may contribute to the genesis of the self-injurious behavior (SIB). The therapeutic effectiveness of punishment, positive reinforcement of either SIB or non-SIB, and time-out learning paradigms were evaluated in 5 Ss aged 3-13 yrs. Electric skin shock failed to suppress the behavior. Positive reinforcement of non-self-injury and time-out from social reinforcement were consistently and rapidly effective, indicating a complex interaction of genetic and environmental factors in the production of SIB. Elimination or major reductions in incidence of SIB was maintained during follow-up periods of 2 yrs. (19 ref)


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Anderson L; Dancis J; Alpert M, “Behavioral contingencies and self-mutilation in Lesch-Nyhan disease,” Pediatric Palliative Care Library, accessed September 26, 2023,