{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,4]],"date-time":"2026-10-04T20:09:23Z","timestamp":1791144563928,"version":"4.1.0"},"reference-count":39,"publisher":"Wiley","issue":"8","license":[{"start":{"date-parts":[[2010,6,29]],"date-time":"2010-06-29T00:00:00Z","timestamp":1277769600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Birth Defects Research"],"published-print":{"date-parts":[[2010,8]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p><jats:bold>BACKGROUND:<\/jats:bold> Folate one\u2010carbon metabolism has been implicated as a determinant of susceptibility to neural tube defects (NTDs), owing to the preventive effect of maternal folic acid supplementation and the higher risk associated with markers of diminished folate status. <jats:bold>METHODS:<\/jats:bold> Folate one\u2010carbon metabolism was compared in <jats:italic>curly tail<\/jats:italic> (<jats:italic>ct\/ct<\/jats:italic>) and genetically matched congenic (+<jats:italic><jats:sup>ct<\/jats:sup>\/+<jats:sup>ct<\/jats:sup><\/jats:italic>) mouse strains using the deoxyuridine suppression test in embryonic fibroblast cells and by quantifying s\u2010adenosylmethionine (SAM) and s\u2010adenosylhomocysteine (SAH) in embryos using liquid chromatography tandem mass spectrometry. A possible genetic interaction between <jats:italic>curly tail<\/jats:italic> and a null allele of 5,10\u2010methylenetetrahydrofolate reductase (MTHFR) was investigated by generation of compound mutant embryos. <jats:bold>RESULTS:<\/jats:bold> There was no deficit in thymidylate biosynthesis in <jats:italic>ct\/ct<\/jats:italic> cells, but incorporation of exogenous thymidine was lower than in +<jats:sup><jats:italic>ct<\/jats:italic><\/jats:sup>\/+<jats:sup><jats:italic>ct<\/jats:italic><\/jats:sup> cells. In +<jats:sup><jats:italic>ct<\/jats:italic><\/jats:sup>\/+<jats:sup><jats:italic>ct<\/jats:italic><\/jats:sup> embryos the SAM\/SAH ratio was diminished by dietary folate deficiency and normalized by folic acid or <jats:italic>myo\u2010<\/jats:italic>inositol treatment, in association with prevention of NTDs. In contrast, folate deficiency caused a significant increase in the SAM\/SAH ratio in <jats:italic>ct\/ct<\/jats:italic> embryos. Loss of MTHFR function in <jats:italic>curly tail<\/jats:italic> embryos significantly reduced the SAM\/SAH ratio but did not cause cranial NTDs or alter the frequency of caudal NTDs. <jats:bold>CONCLUSIONS:<\/jats:bold>\n<jats:italic>Curly tail<\/jats:italic> fibroblasts and embryos, in which <jats:italic>Grhl3<\/jats:italic> expression is reduced, display alterations in one\u2010carbon metabolism, particularly in the response to folate deficiency, compared to genetically matched congenic controls in which <jats:italic>Grhl3<\/jats:italic> is unaffected. However, unlike folate deficiency, diminished methylation potential appears to be insufficient to cause cranial NTDs in the <jats:italic>curly tail<\/jats:italic> strain, nor does it increase the frequency of caudal NTDs. Birth Defects Research (Part A), 2010. \u00a9 2010 Wiley\u2010Liss, Inc.<\/jats:p>","DOI":"10.1002\/bdra.20690","type":"journal-article","created":{"date-parts":[[2010,6,29]],"date-time":"2010-06-29T14:51:16Z","timestamp":1277823076000},"page":"612-618","source":"Crossref","is-referenced-by-count":24,"title":["Neural tube defects induced by folate deficiency in mutant <i>curly tail<\/i> (<i>Grhl3<\/i>) embryos are associated with alteration in folate one\u2010carbon metabolism but are unlikely to result from diminished methylation"],"prefix":"10.1002","volume":"88","author":[{"given":"Sandra C. 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