دورية أكاديمية
أعرض في EDS

Knockout of zebrafish desmin genes does not cause skeletal muscle degeneration but alters calcium flux.

التفاصيل البيبلوغرافية
العنوان: Knockout of zebrafish desmin genes does not cause skeletal muscle degeneration but alters calcium flux.
المؤلفون: Kayman Kürekçi G; Department of Medical Biology, Hacettepe University Faculty of Medicine, 06100, Ankara, Turkey., Kural Mangit E; Department of Medical Biology, Hacettepe University Faculty of Medicine, 06100, Ankara, Turkey.; Laboratory Animals Research and Application Centre, Hacettepe University, 06100, Ankara, Turkey., Koyunlar C; Department of Medical Biology, Hacettepe University Faculty of Medicine, 06100, Ankara, Turkey.; Department of Hematology, Erasmus MC, 3015 CN, Rotterdam, The Netherlands., Unsal S; Department of Medical Biology, Hacettepe University Faculty of Medicine, 06100, Ankara, Turkey.; Department of Cancer and Inflammation, The Institute of Molecular Medicine, University of Southern Denmark, 5000, Odense C, Denmark., Saglam B; Department of Biophysics, Hacettepe University Faculty of Medicine, 06100, Ankara, Turkey., Ergin B; Department of Biophysics, Hacettepe University Faculty of Medicine, 06100, Ankara, Turkey., Gizer M; Department of Stem Cell Sciences, Hacettepe University Graduate School of Health Sciences, 06100, Ankara, Turkey., Uyanik I; Department of Electrical and Electronics Engineering, Hacettepe University Faculty of Engineering, 06800, Ankara, Turkey., Boustanabadimaralan Düz N; Department of Medical Biology, Hacettepe University Faculty of Medicine, 06100, Ankara, Turkey., Korkusuz P; Department of Histology and Embryology, Hacettepe University Faculty of Medicine, 06100, Ankara, Turkey., Talim B; Pathology Unit, Department of Pediatrics, Hacettepe University Faculty of Medicine, 06100, Ankara, Turkey., Purali N; Department of Biophysics, Hacettepe University Faculty of Medicine, 06100, Ankara, Turkey., Hughes SM; Randall Centre for Cell and Molecular Biophysics, New Hunt's House, Guy's Campus, King's College London, London, SE1 1UL, United Kingdom., Dincer PR; Department of Medical Biology, Hacettepe University Faculty of Medicine, 06100, Ankara, Turkey. pdincer@hacettepe.edu.tr.
المصدر: Scientific reports [Sci Rep] 2021 Apr 05; Vol. 11 (1), pp. 7505. Date of Electronic Publication: 2021 Apr 05.
نوع المنشور: Journal Article; Research Support, Non-U.S. Gov't
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: Gene Knockout Techniques*, Calcium/*metabolism , Desmin/*genetics , Muscle, Skeletal/*metabolism , Muscle, Skeletal/*pathology , Zebrafish/*genetics, Animals ; Base Sequence ; Desmin/metabolism ; Embryo, Nonmammalian/metabolism ; Gene Expression Profiling ; Gene Expression Regulation, Developmental ; Larva/genetics ; Muscle Fibers, Skeletal/pathology ; Muscle, Skeletal/ultrastructure ; Mutation/genetics ; Neuromuscular Junction/pathology ; RNA, Messenger/genetics ; RNA, Messenger/metabolism ; Zebrafish/embryology
مستخلص: Desmin is a muscle-specific intermediate filament protein that has fundamental role in muscle structure and force transmission. Whereas human desmin protein is encoded by a single gene, two desmin paralogs (desma and desmb) exist in zebrafish. Desma and desmb show differential spatiotemporal expression during zebrafish embryonic and larval development, being similarly expressed in skeletal muscle until hatching, after which expression of desmb shifts to gut smooth muscle. We generated knockout (KO) mutant lines carrying loss-of-function mutations for each gene by using CRISPR/Cas9. Mutants are viable and fertile, and lack obvious skeletal muscle, heart or intestinal defects. In contrast to morphants, knockout of each gene did not cause any overt muscular phenotype, but did alter calcium flux in myofibres. These results point to a possible compensation mechanism in these mutant lines generated by targeting nonsense mutations to the first coding exon.
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معلومات مُعتمدة: G1001029 United Kingdom MRC_ Medical Research Council; MR/N021231/1 United Kingdom MRC_ Medical Research Council
المشرفين على المادة: 0 (Desmin)
0 (RNA, Messenger)
SY7Q814VUP (Calcium)
تواريخ الأحداث: Date Created: 20210406 Date Completed: 20211028 Latest Revision: 20220419
رمز التحديث: 20231215
مُعرف محوري في PubMed: PMC8021586
DOI: 10.1038/s41598-021-86974-w
PMID: 33820917
قاعدة البيانات: MEDLINE
الوصف
تدمد:2045-2322
DOI:10.1038/s41598-021-86974-w