{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,18]],"date-time":"2026-09-18T12:14:54Z","timestamp":1789733694304,"version":"4.0.1"},"reference-count":20,"publisher":"Wiley","issue":"8","license":[{"start":{"date-parts":[[2014,5,9]],"date-time":"2014-05-09T00:00:00Z","timestamp":1399593600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["R01NS083897"],"award-info":[{"award-number":["R01NS083897"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["5T32NS051156-09"],"award-info":[{"award-number":["5T32NS051156-09"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Genesis"],"published-print":{"date-parts":[[2014,8]]},"abstract":"<jats:title>Summary<\/jats:title><jats:p><jats:italic>Magoh<\/jats:italic> encodes a core component of the exon junction complex (EJC), which binds mRNA and regulates mRNA metabolism. <jats:italic>Magoh<\/jats:italic> is highly expressed in proliferative tissues during development. EJC components have been implicated in several developmental disorders including TAR syndrome, Richieri\u2013Costa\u2013Pereira syndrome, and intellectual disability. Existing germline null <jats:italic>Magoh<\/jats:italic> mice are embryonic lethal as homozygotes and perinatal lethal as heterozygotes, precluding detailed analysis of embryonic and postnatal functions. Here, we report the generation of a new genetic tool to dissect temporal and tissue\u2010specific roles for <jats:italic>Magoh<\/jats:italic> in development and adult homeostasis. This <jats:italic>Magoh<\/jats:italic> conditional allele has two <jats:italic>loxP<\/jats:italic> sites flanking the second exon. Ubiquitous Cre\u2010mediated deletion of the floxed allele in a heterozygous mouse (<jats:italic>Magoh<jats:sup>del\/+<\/jats:sup><\/jats:italic>) causes 50% reduction of both <jats:italic>Magoh<\/jats:italic> mRNA and protein. <jats:italic>Magoh<jats:sup>del\/+<\/jats:sup><\/jats:italic> mice exhibit both microcephaly and hypopigmentation, thus phenocopying germline haploinsufficient <jats:italic>Magoh<\/jats:italic> mice. Using <jats:italic>Emx1\u2010Cre<\/jats:italic>, we further show that conditional <jats:italic>Magoh<\/jats:italic> deletion in neural progenitors during embryonic development also causes microcephaly. We anticipate this novel conditional allele will be a valuable tool for assessing tissue\u2010specific roles for <jats:italic>Magoh<\/jats:italic> in mammalian development and postnatal processes. genesis 52:752\u2013758, 2014. \u00a9 2014 Wiley Periodicals, Inc.<\/jats:p>","DOI":"10.1002\/dvg.22788","type":"journal-article","created":{"date-parts":[[2014,4,27]],"date-time":"2014-04-27T07:56:38Z","timestamp":1398585398000},"page":"752-758","source":"Crossref","is-referenced-by-count":23,"title":["Generation of a <i>Magoh<\/i> conditional allele in mice"],"prefix":"10.1002","volume":"52","author":[{"given":"John J.","family":"McMahon","sequence":"first","affiliation":[{"name":"Department of Molecular Genetics and Microbiology Duke University Medical Center Durham North Carolina"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lei","family":"Shi","sequence":"additional","affiliation":[{"name":"Department of Molecular Genetics and Microbiology Duke University Medical Center Durham North Carolina"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Debra L.","family":"Silver","sequence":"additional","affiliation":[{"name":"Department of Molecular Genetics and Microbiology Duke University Medical Center Durham North Carolina"},{"name":"Department of Cell Biology Duke University Medical Center Durham North Carolina"},{"name":"Department of Neurobiology and Duke Institute for Brain Sciences Duke University Medical Center Durham North Carolina"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2014,5,9]]},"reference":[{"key":"e_1_2_4_2_1","doi-asserted-by":"publisher","DOI":"10.1038\/mp.2010.59"},{"key":"e_1_2_4_3_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng.1083"},{"key":"e_1_2_4_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cell.2010.09.014"},{"key":"e_1_2_4_5_1","doi-asserted-by":"publisher","DOI":"10.4161\/rna.8.1.13618"},{"key":"e_1_2_4_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajhg.2013.11.020"},{"key":"e_1_2_4_7_1","doi-asserted-by":"publisher","DOI":"10.1038\/nrn3586"},{"key":"e_1_2_4_8_1","doi-asserted-by":"publisher","DOI":"10.1093\/emboj\/20.22.6424"},{"key":"e_1_2_4_9_1","doi-asserted-by":"publisher","DOI":"10.1093\/embo-reports\/kve245"},{"key":"e_1_2_4_10_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/ddn110"},{"key":"e_1_2_4_11_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/ddt035"},{"key":"e_1_2_4_12_1","doi-asserted-by":"publisher","DOI":"10.1101\/gad.1163204"},{"key":"e_1_2_4_13_1","doi-asserted-by":"publisher","DOI":"10.1038\/nature02351"},{"key":"e_1_2_4_14_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cell.2010.09.036"},{"key":"e_1_2_4_15_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng1862"},{"key":"e_1_2_4_16_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ydbio.2013.01.004"},{"key":"e_1_2_4_17_1","doi-asserted-by":"publisher","DOI":"10.1038\/nn.2527"},{"key":"e_1_2_4_18_1","doi-asserted-by":"publisher","DOI":"10.4161\/rna.25827"},{"key":"e_1_2_4_19_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng2100"},{"key":"e_1_2_4_20_1","doi-asserted-by":"publisher","DOI":"10.1093\/nar\/gkh029"},{"key":"e_1_2_4_21_1","doi-asserted-by":"publisher","DOI":"10.1006\/geno.1999.6064"}],"container-title":["genesis"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fdvg.22788","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/dvg.22788","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,10,6]],"date-time":"2023-10-06T10:54:01Z","timestamp":1696589641000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/dvg.22788"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2014,5,9]]},"references-count":20,"journal-issue":{"issue":"8","published-print":{"date-parts":[[2014,8]]}},"alternative-id":["10.1002\/dvg.22788"],"URL":"https:\/\/doi.org\/10.1002\/dvg.22788","archive":["Portico"],"relation":{},"ISSN":["1526-954X","1526-968X"],"issn-type":[{"value":"1526-954X","type":"print"},{"value":"1526-968X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2014,5,9]]}}}