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dc.creatorKomiya, Y
dc.creatorBai, Z
dc.creatorCai, N
dc.creatorLou, L
dc.creatorAl-Saadi, N
dc.creatorMezzacappa, C
dc.creatorHabas, R
dc.creatorRunnels, LW
dc.date.accessioned2021-01-22T14:47:39Z
dc.date.available2021-01-22T14:47:39Z
dc.date.issued2017-12-01
dc.identifier.issn2045-2322
dc.identifier.issn2045-2322
dc.identifier.doihttp://dx.doi.org/10.34944/dspace/4835
dc.identifier.other29142255 (pubmed)
dc.identifier.urihttp://hdl.handle.net/20.500.12613/4853
dc.description.abstract© 2017 The Author(s). In humans, germline mutations in Trpm6 cause autosomal dominant hypomagnesemia with secondary hypocalcemia disorder. Loss of Trpm6 in mice also perturbs cellular magnesium homeostasis but additionally results in early embryonic lethality and neural tube closure defects. To define the mechanisms by which TRPM6 influences neural tube closure, we functionally characterized the role of TRPM6 during early embryogenesis in Xenopus laevis. The expression of Xenopus TRPM6 (XTRPM6) is elevated at the onset of gastrulation and is concentrated in the lateral mesoderm and ectoderm at the neurula stage. Loss of XTRPM6 produced gastrulation and neural tube closure defects. Unlike XTRPM6′s close homologue XTRPM7, whose loss interferes with mediolateral intercalation, depletion of XTRPM6 but not XTRPM7 disrupted radial intercalation cell movements. A zinc-influx assay demonstrated that TRPM6 has the potential to constitute functional channels in the absence of TRPM7. The results of our study indicate that XTRPM6 regulates radial intercalation with little or no contribution from XTRPM7 in the region lateral to the neural plate, whereas XTRPM7 is mainly involved in regulating mediolateral intercalation in the medial region of the neural plate. We conclude that both TRPM6 and TRPM7 channels function cooperatively but have distinct and essential roles during neural tube closure.
dc.format.extent15623-
dc.language.isoen
dc.relation.haspartScientific Reports
dc.relation.isreferencedbySpringer Science and Business Media LLC
dc.rightsCC BY
dc.rights.urihttp://creativecommons.org/licenses/by/4.0/
dc.subjectAnimals
dc.subjectCalcium
dc.subjectCell Movement
dc.subjectEmbryonic Development
dc.subjectGene Expression Regulation, Developmental
dc.subjectGerm-Line Mutation
dc.subjectHumans
dc.subjectHypercalciuria
dc.subjectHypocalcemia
dc.subjectMagnesium
dc.subjectNephrocalcinosis
dc.subjectNeural Plate
dc.subjectNeural Tube
dc.subjectRenal Tubular Transport, Inborn Errors
dc.subjectTRPM Cation Channels
dc.subjectXenopus Proteins
dc.subjectXenopus laevis
dc.titleA Nonredundant Role for the TRPM6 Channel in Neural Tube Closure
dc.typeArticle
dc.type.genreJournal Article
dc.relation.doi10.1038/s41598-017-15855-y
dc.ada.noteFor Americans with Disabilities Act (ADA) accommodation, including help with reading this content, please contact scholarshare@temple.edu
dc.date.updated2021-01-22T14:47:34Z
refterms.dateFOA2021-01-22T14:47:39Z


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