Literature DB >> 24368072

Dysferlin is essential for endocytosis in the sea star oocyte.

Nathalie Oulhen1, Thomas M Onorato1, Isabela Ramos1, Gary M Wessel2.   

Abstract

Dysferlin is a calcium-binding transmembrane protein involved in membrane fusion and membrane repair. In humans, mutations in the dysferlin gene are associated with muscular dystrophy. In this study, we isolated plasma membrane-enriched fractions from full-grown immature oocytes of the sea star, and identified dysferlin by mass spectrometry analysis. The full-length dysferlin sequence is highly conserved between human and the sea star. We learned that in the sea star Patiria miniata, dysferlin RNA and protein are expressed from oogenesis to gastrulation. Interestingly, the protein is highly enriched in the plasma membrane of oocytes. Injection of a morpholino against dysferlin leads to a decrease of endocytosis in oocytes, and to a developmental arrest during gastrulation. These results suggest that dysferlin is critical for normal endocytosis during oogenesis and for embryogenesis in the sea star and that this animal may be a useful model for studying the relationship of dysferlin structure as it relates to its function.
Copyright © 2014 Elsevier Inc. All rights reserved.

Entities:  

Keywords:  Dysferlin; Endocytosis; Gastrulation; Oocytes; Plasma membrane; Sea star

Mesh:

Substances:

Year:  2013        PMID: 24368072      PMCID: PMC3982204          DOI: 10.1016/j.ydbio.2013.12.018

Source DB:  PubMed          Journal:  Dev Biol        ISSN: 0012-1606            Impact factor:   3.582


  52 in total

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Review 3.  Use of sea stars to study basic reproductive processes.

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7.  Normal myoblast fusion requires myoferlin.

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Journal:  Development       Date:  2005-11-09       Impact factor: 6.868

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Journal:  Mech Dev       Date:  2003-10       Impact factor: 1.882

Review 9.  Otoferlin: a multi-C2 domain protein essential for hearing.

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  10 in total

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Authors:  Isabela Ramos; Adrian Reich; Gary M Wessel
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3.  Coupling of excitation to Ca2+ release is modulated by dysferlin.

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Review 5.  Origin and development of the germ line in sea stars.

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7.  AAV.Dysferlin Overlap Vectors Restore Function in Dysferlinopathy Animal Models.

Authors:  Patricia C Sondergaard; Danielle A Griffin; Eric R Pozsgai; Ryan W Johnson; William E Grose; Kristin N Heller; Kim M Shontz; Chrystal L Montgomery; Joseph Liu; Kelly Reed Clark; Zarife Sahenk; Jerry R Mendell; Louise R Rodino-Klapac
Journal:  Ann Clin Transl Neurol       Date:  2015-01-20       Impact factor: 4.511

8.  Exon 32 Skipping of Dysferlin Rescues Membrane Repair in Patients' Cells.

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Journal:  Microarrays (Basel)       Date:  2014-10-27
  10 in total

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