Serotonergic approaches to the modification of behavior in the Lesch-Nyhan syndrome

Serotonergic approaches to the modification of behavior in the Lesch-Nyhan syndrome

Nyhan W L; Johnson H G; Kaufman I A; Jones K L

Applied Research in Mental Retardation

1980

behavioral problems; Lesch-Nyhan syndrome; pharmacologic intervention; 5-hydroxy-tryptophan; self-injurious behavior

In a series of patients with the Lesch-Nyhan syndrome a pharmacologic attempt was made to alter the balance of biogenic amines in the central nervous system by increasing the level of serotin. Patients were treated with 5-hydroxytryptophan in combination with a peripheral decarboxylase inhibitor carbidopa and with imipramine. Most patients had a striking alteration in self-mutilative behavior. However, within 1–3 months each became tolerant and this pharmacologic effect could not be produced again even a year later. These observations suggest that the characteristic behavior of the patient with the Lesch-Nyhan syndrome is related to neurotransmitter balance, and that there may be ways to modify it.

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