{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,27]],"date-time":"2026-09-27T06:22:21Z","timestamp":1790490141713,"version":"4.1.0"},"reference-count":75,"publisher":"The Company of Biologists","issue":"2","license":[{"start":{"date-parts":[[2021,1,15]],"date-time":"2021-01-15T00:00:00Z","timestamp":1610668800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/www.biologists.com\/user-licence-1-1\/"}],"funder":[{"DOI":"10.13039\/501100001659","name":"Deutsche Forschungsgemeinschaft","doi-asserted-by":"crossref","award":["GRK2318"],"award-info":[{"award-number":["GRK2318"]}],"id":[{"id":"10.13039\/501100001659","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/501100001659","name":"Deutsche Forschungsgemeinschaft","doi-asserted-by":"crossref","award":["CRC958"],"award-info":[{"award-number":["CRC958"]}],"id":[{"id":"10.13039\/501100001659","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/501100004895","name":"European Social Fund","doi-asserted-by":"crossref","id":[{"id":"10.13039\/501100004895","id-type":"DOI","asserted-by":"crossref"}]},{"name":"Eunice Kennedy Shriver National Institute of Child Health and Human Development","award":["R01HD099191"],"award-info":[{"award-number":["R01HD099191"]}]},{"DOI":"10.13039\/100000057","name":"National Institute of General Medical Sciences","doi-asserted-by":"crossref","award":["R01GM104853"],"award-info":[{"award-number":["R01GM104853"]}],"id":[{"id":"10.13039\/100000057","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2021,1,15]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>Pathogenic mutations in the endocytic receptor LRP2 in humans are associated with severe neural tube closure defects (NTDs) such as anencephaly and spina bifida. Here, we have combined analysis of neural tube closure in mouse and in the African Clawed Frog Xenopus laevis to elucidate the etiology of Lrp2-related NTDs. Lrp2 loss of function impaired neuroepithelial morphogenesis, culminating in NTDs that impeded anterior neural plate folding and neural tube closure in both model organisms. Loss of Lrp2 severely affected apical constriction as well as proper localization of the core planar cell polarity (PCP) protein Vangl2, demonstrating a highly conserved role of the receptor in these processes, which are essential for neural tube formation. In addition, we identified a novel functional interaction of Lrp2 with the intracellular adaptor proteins Shroom3 and Gipc1 in the developing forebrain. Our data suggest that, during neurulation, motifs within the intracellular domain of Lrp2 function as a hub that orchestrates endocytic membrane removal for efficient apical constriction, as well as PCP component trafficking in a temporospatial manner.<\/jats:p>","DOI":"10.1242\/dev.195008","type":"journal-article","created":{"date-parts":[[2021,1,26]],"date-time":"2021-01-26T06:00:13Z","timestamp":1611640813000},"source":"Crossref","is-referenced-by-count":59,"title":["Neural tube closure requires the endocytic receptor Lrp2 and its functional interaction with intracellular scaffolds"],"prefix":"10.1242","volume":"148","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-1476-7531","authenticated-orcid":false,"given":"Izabela","family":"Kowalczyk","sequence":"first","affiliation":[{"name":"Disorders of the Nervous System, Max Delbr\u00fcck Center for Molecular Medicine in the Helmholtz Association, Robert R\u00f6ssle Strasse 10, 13125 Berlin, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chanjae","family":"Lee","sequence":"additional","affiliation":[{"name":"Department of Molecular Biosciences, The University of Texas at Austin, Austin, TX 78712, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elisabeth","family":"Schuster","sequence":"additional","affiliation":[{"name":"University of Hohenheim, Institute of Biology, Department of Zoology, Garbenstrasse 30, 70599 Stuttgart, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Josefine","family":"Hoeren","sequence":"additional","affiliation":[{"name":"University of Hohenheim, Institute of Biology, Department of Zoology, Garbenstrasse 30, 70599 Stuttgart, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Valentina","family":"Trivigno","sequence":"additional","affiliation":[{"name":"University of Hohenheim, Institute of Biology, Department of Zoology, Garbenstrasse 30, 70599 Stuttgart, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Levin","family":"Riedel","sequence":"additional","affiliation":[{"name":"Disorders of the Nervous System, Max Delbr\u00fcck Center for Molecular Medicine in the Helmholtz Association, Robert R\u00f6ssle Strasse 10, 13125 Berlin, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jessica","family":"G\u00f6rne","sequence":"additional","affiliation":[{"name":"Disorders of the Nervous System, Max Delbr\u00fcck Center for Molecular Medicine in the Helmholtz Association, Robert R\u00f6ssle Strasse 10, 13125 Berlin, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"John B.","family":"Wallingford","sequence":"additional","affiliation":[{"name":"Department of Molecular Biosciences, The University of Texas at Austin, Austin, TX 78712, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1663-8378","authenticated-orcid":false,"given":"Annette","family":"Hammes","sequence":"additional","affiliation":[{"name":"Disorders of the Nervous System, Max Delbr\u00fcck Center for Molecular Medicine in the Helmholtz Association, Robert R\u00f6ssle Strasse 10, 13125 Berlin, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3397-8972","authenticated-orcid":false,"given":"Kerstin","family":"Feistel","sequence":"additional","affiliation":[{"name":"University of Hohenheim, Institute of Biology, Department of Zoology, Garbenstrasse 30, 70599 Stuttgart, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2021,1,26]]},"reference":[{"key":"2021042701103867500_DEV195008C1","doi-asserted-by":"publisher","first-page":"2728","DOI":"10.1091\/mbc.e02-11-0767","article-title":"Myo6 facilitates the translocation of endocytic vesicles from cell peripheries","volume":"14","author":"Aschenbrenner","year":"2003","journal-title":"Mol. 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