Abstract:
:In humans, congenital deficiency of the enzyme hypoxanthine-guanine phosphoribosyltransferase (HPRT) results in a disorder known as the Lesch-Nyhan syndrome. Patients with this disorder exhibit a prominent neurobehavioral phenotype that results in part from dysfunction of catecholaminergic systems in the striatum. HPRT-deficient mice produced as animal models for this syndrome curiously exhibit no spontaneous neurobehavioral abnormalities. However, the present study demonstrates that HPRT-deficient mice are more sensitive than their HPRT-normal littermates to the ability of amphetamine to stimulate locomotor or stereotypic behaviors. This behavioral supersensitivity to amphetamine indicates the existence of an underlying subclinical abnormality of catecholaminergic systems in the brains of HPRT-deficient mice, analogous to findings in human Lesch-Nyhan patients.
journal_name
Behav Neuroscijournal_title
Behavioral neuroscienceauthors
Jinnah HA,Gage FH,Friedmann Tdoi
10.1037//0735-7044.105.6.1004subject
Has Abstractpub_date
1991-12-01 00:00:00pages
1004-12issue
6eissn
0735-7044issn
1939-0084journal_volume
105pub_type
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