دورية أكاديمية
أعرض في EDS

Hepatocystin is Essential for TRPM7 Function During Early Embryogenesis.

التفاصيل البيبلوغرافية
العنوان: Hepatocystin is Essential for TRPM7 Function During Early Embryogenesis.
المؤلفون: Overton JD; Rutgers-Robert Wood Johnson Medical School, Dept. of Pharmacology, Piscataway, 08854, USA., Komiya Y; Rutgers-Robert Wood Johnson Medical School, Dept. of Pharmacology, Piscataway, 08854, USA., Mezzacappa C; Temple University, Dept. of Biology, Philadelphia, 19122, USA., Nama K; Temple University, Dept. of Biology, Philadelphia, 19122, USA., Cai N; Rutgers-Robert Wood Johnson Medical School, Dept. of Pharmacology, Piscataway, 08854, USA., Lou L; Rutgers-Robert Wood Johnson Medical School, Dept. of Pharmacology, Piscataway, 08854, USA., Fedeles SV; Yale University School of Medicine, Dept. of Internal Medicine, New Haven, 06510, USA., Habas R; Temple University, Dept. of Biology, Philadelphia, 19122, USA., Runnels LW; Rutgers-Robert Wood Johnson Medical School, Dept. of Pharmacology, Piscataway, 08854, USA.
المصدر: Scientific reports [Sci Rep] 2015 Dec 16; Vol. 5, pp. 18395. Date of Electronic Publication: 2015 Dec 16.
نوع المنشور: Journal Article; Research Support, N.I.H., Extramural
اللغة: English
بيانات الدورية: Publisher: Nature Publishing Group Country of Publication: England NLM ID: 101563288 Publication Model: Electronic Cited Medium: Internet ISSN: 2045-2322 (Electronic) Linking ISSN: 20452322 NLM ISO Abbreviation: Sci Rep Subsets: MEDLINE
أسماء مطبوعة: Original Publication: London : Nature Publishing Group, copyright 2011-
مواضيع طبية MeSH: Embryo, Nonmammalian/*embryology , Embryonic Development/*physiology , Gastrula/*embryology , Glucosidases/*metabolism , TRPM Cation Channels/*metabolism , Xenopus Proteins/*metabolism, Animals ; Cell Line ; Glucosidases/genetics ; Humans ; Mice ; TRPM Cation Channels/genetics ; Xenopus Proteins/genetics ; Xenopus laevis
مستخلص: Mutations in protein kinase C substrate 80K-H (PRKCSH), which encodes for an 80 KDa protein named hepatocystin (80K-H, PRKCSH), gives rise to polycystic liver disease (PCLD). Hepatocystin functions as the noncatalytic beta subunit of Glucosidase II, an endoplasmic reticulum (ER)-resident enzyme involved in processing and quality control of newly synthesized glycoproteins. Patients harboring heterozygous germline mutations in PRKCSH are thought to develop renal cysts as a result of somatic loss of the second allele, which subsequently interferes with expression of the TRP channel polycystin-2 (PKD2). Deletion of both alleles of PRKCSH in mice results in embryonic lethality before embryonic day E11.5. Here, we investigated the function of hepatocystin during Xenopus laevis embryogenesis and identified hepatocystin as a binding partner of the TRPM7 ion channel, whose function is required for vertebrate gastrulation. We find that TRPM7 functions synergistically with hepatocystin. Although other N-glycosylated proteins are critical to early development, overexpression of TRPM7 in Xenopus laevis embryos was sufficient to fully rescue the gastrulation defect caused by loss of hepatocystin. We observed that depletion of hepatocystin in Xenopus laevis embryos decreased TRPM7 expression, indicating that the early embryonic lethality caused by loss of hepatocystin is mainly due to impairment of TRPM7 protein expression.
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معلومات مُعتمدة: GM078172 United States GM NIGMS NIH HHS; S10 OD010572 United States OD NIH HHS; GM080753 United States GM NIGMS NIH HHS; R01 GM078172 United States GM NIGMS NIH HHS; R01 GM080753 United States GM NIGMS NIH HHS
المشرفين على المادة: 0 (TRPM Cation Channels)
0 (TRPM7 protein, Xenopus)
0 (Xenopus Proteins)
EC 3.2.1.- (Glucosidases)
تواريخ الأحداث: Date Created: 20151217 Date Completed: 20161028 Latest Revision: 20190417
رمز التحديث: 20231215
مُعرف محوري في PubMed: PMC4680877
DOI: 10.1038/srep18395
PMID: 26671672
قاعدة البيانات: MEDLINE
الوصف
تدمد:2045-2322
DOI:10.1038/srep18395