{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,28]],"date-time":"2025-10-28T18:04:41Z","timestamp":1761674681805},"reference-count":27,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2001,2,28]],"date-time":"2001-02-28T00:00:00Z","timestamp":983318400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["J of Neuroscience Research"],"published-print":{"date-parts":[[2001,3,15]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Extracellular purines have essential roles in neuronal development; hence, disruptions in their metabolism as reported in Lesch Nyhan syndrome (LNS) could result in developmental abnormalities. The deficiency of hypoxanthine\u2010guanine phosphoribosyl transferase (HGPRT) in LNS leads to increased hypoxanthine and uric acid production. We have reported that HGPRT\u2010deficient B103\u20104C neuroblastoma, a neuronal model of LNS, proliferated less and differentiated more than their HGPRT\u2010positive B103 counterparts. Here, we sought to determine whether differences in proliferation and differentiation would occur when these cells were cultured in the presence of hypoxanthine or in a hypoxanthine\u2010\/serum\u2010free chemically defined media (NBMN2). In media with 1% serum, hypoxanthine (50 \u03bcM) significantly increased the proliferation of both cell lines with a greater effect on B103\u20104C cells. In 1% serum media, hypoxanthine increased differentiation of B103 but decreased B103\u20104C differentiation. In NBMN2, B103 proliferated far more than B103\u20104C, but both cell types differentiated to the same extent. These results are interpreted to suggest that elevated levels of central nervous system (CNS) hypoxanthine as reported in LNS may affect neuronal development, and to implicate hypoxanthine and abnormal neuronal development as causative factors in the etiology of LNS. J. Neurosci. Res. 63:500\u2013508, 2001. \u00a9 2001 Wiley\u2010Liss, Inc.<\/jats:p>","DOI":"10.1002\/jnr.1044","type":"journal-article","created":{"date-parts":[[2002,8,25]],"date-time":"2002-08-25T18:04:33Z","timestamp":1030298673000},"page":"500-508","source":"Crossref","is-referenced-by-count":18,"title":["Hypoxanthine impairs morphogenesis and enhances proliferation of a neuroblastoma model of Lesch Nyhan syndrome"],"prefix":"10.1002","volume":"63","author":[{"given":"Margaret H. Y.","family":"Ma","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"N. Charles","family":"Stacey","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Gerald P.","family":"Connolly","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2001,2,28]]},"reference":[{"key":"e_1_2_6_2_1","doi-asserted-by":"crossref","first-page":"4317","DOI":"10.1016\/S0021-9258(18)42836-0","article-title":"Regulation of purine nucleotide synthesis in human B lymphoblasts with both hypoxanthine\u2010guanine phosphoribosyltransferase deficiency and phosphoribosylpyrophosphate synthetase superactivity","volume":"267","author":"Becker MA","year":"1992","journal-title":"J Biol Chem"},{"key":"e_1_2_6_3_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.76.1.514"},{"key":"e_1_2_6_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0197-0186(96)00123-4"},{"key":"e_1_2_6_5_1","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1097-4547(19970801)49:3<342::AID-JNR9>3.0.CO;2-C"},{"key":"e_1_2_6_6_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM199606133342403"},{"key":"e_1_2_6_7_1","doi-asserted-by":"publisher","DOI":"10.1007\/BF01800365"},{"key":"e_1_2_6_8_1","first-page":"396","article-title":"Adenosine and hypoxanthine inhibits neurite outgrowth from human brain cells","volume":"4","author":"Korenevsky AV","year":"1999","journal-title":"Cell Mol Biol Lett"},{"key":"e_1_2_6_9_1","doi-asserted-by":"publisher","DOI":"10.1016\/0002-9343(64)90104-4"},{"key":"e_1_2_6_10_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM198111053051902"},{"key":"e_1_2_6_11_1","first-page":"412","article-title":"Morphogenesis of B103 neuroblastoma and B103\u20104C its mutant derivative: modelling Lesch Nyhan Syndrome","volume":"4","author":"Ma MHY","year":"1999","journal-title":"Cell Mol Biol Lett"},{"key":"e_1_2_6_12_1","unstructured":"MaMHY StaceyNC DuleyJA ConnollyGP.1999b.Abnormal growth of HGPRT\u2010deficient B103\u20104c neuroblastoma: a model for neuronal development in Lesch Nyhan syndrome. 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