ArticleDevelopmental biology2014
Dysferlin is essential for endocytosis in the sea star oocyte.
Article in Developmental biology, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 13 papers.
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Who cites it
13 citing papers in PubMed, 15 citations in OpenAlex.
- Peptide-phosphorodiamidate morpholino oligomer therapy for dysferlinopathy induces pseudoexon skipping and restoration of functional protein.JCI insight · 2026Article
- Utilization of Targeted RNA-Seq for the Resolution of Variant Pathogenicity and Enhancement of Diagnostic Yield in Dysferlinopathy.Journal of personalized medicine · 2023Article
- Article
- Systemic Delivery of Dysferlin Overlap Vectors Provides Long-Term Gene Expression and Functional Improvement for Dysferlinopathy.Human gene therapy · 2018Article
- Coupling of excitation to CaThe Journal of physiology · 2017Article
- Regeneration in bipinnaria larvae of the bat star Patiria miniata induces rapid and broad new gene expression.Mechanisms of development · 2016Article
- Differential Nanos 2 protein stability results in selective germ cell accumulation in the sea urchin.Developmental biology · 2016Article
- Exon 32 Skipping of Dysferlin Rescues Membrane Repair in Patients' Cells.Journal of neuromuscular diseases · 2015Article
- AAV.Dysferlin Overlap Vectors Restore Function in Dysferlinopathy Animal Models.Annals of clinical and translational neurology · 2015Article
- Simple perfusion apparatus for manipulation, tracking, and study of oocytes and embryos.Fertility and sterility · 2015Article
- Two-pore channels function in calcium regulation in sea star oocytes and embryos.Development (Cambridge, England) · 2014Article
- DNA Microarray Analysis on the Genes Differentially Expressed in the Liver of the Pufferfish, Takifugu rubripes, Following an Intramuscular Administration of Tetrodotoxin.Microarrays (Basel, Switzerland) · 2014Article
- Origin and development of the germ line in sea stars.Genesis (New York, N.Y. : 2000) · 2014Review
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Authors and funding
4 authors at 1 institution in 1 country.
Funding
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.
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