Abstract:
:Lesch-Nyhan syndrome (LNS), caused by the complete deficiency of hypoxanthine phosphoribosyltransferase (HPRT), is characterized by a neurological deficit, the etiology of which is still unclear. Evidence has accumulated indicating that it reflects dopamine deficiency associated with defective arborization of dopaminergic dendrites. We monitored the differentiation in vitro of dopaminergic neurons, cultured from HPRT-deficient knockout mice. The HPRT-deficient dopaminergic neurons exhibited a decelerated rate of outgrowth of dendrites in comparison to that of control neurons resulting, after 8 days in culture, in 32% smaller average total length of dendrites per neuron (P<0.025). The results suggest that the abnormal dendrite outgrowth in LNS reflects a defective developmental process.
journal_name
Neurosci Lettjournal_title
Neuroscience lettersauthors
Boer P,Brosh S,Wasserman L,Hammel I,Zoref-Shani E,Sperling Odoi
10.1016/s0304-3940(01)01716-5keywords:
subject
Has Abstractpub_date
2001-04-27 00:00:00pages
45-8issue
1eissn
0304-3940issn
1872-7972pii
S0304394001017165journal_volume
303pub_type
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