ArticlePLoS genetics2017
Filamin actin-binding and titin-binding fulfill distinct functions in Z-disc cohesion.
Article in PLoS genetics, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 33 papers.
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Who cites it
33 citing papers in PubMed, 50 citations in OpenAlex.
- Article
- Drak is a potential binding partner of Drosophila Filamin.Biology open · 2026Article
- Cardiomyopathy-associated and basic residue mutations in myopalladin alter actin binding, bundling, and structural stability.Protein science : a publication of the Protein Society · 2026Article
- Genetic and Clinical Characterization of FLNC Variants in Chinese Patients with Cardiomyopathy.Journal of cardiovascular development and disease · 2025Article
- Filamin protects myofibrils from contractile damage through changes in its mechanosensory region.PLoS genetics · 2024Article
- Heat Shock Proteins: Important Helpers for the Development, Maintenance and Regeneration of Skeletal Muscles.Muscles (Basel, Switzerland) · 2023Review
- Article
- Article
- Filamin C is Essential for mammalian myocardial integrity.PLoS genetics · 2023Article
- Cytoskeletal and Cytoskeleton-Associated Proteins: Key Regulators of Cancer Stem Cell Properties.Pharmaceuticals (Basel, Switzerland) · 2022Review
- Myosin Transducer Inter-Strand Communication Is Critical for Normal ATPase Activity and Myofibril Structure.Biology · 2022Article
- Genetic Insights into Primary Restrictive Cardiomyopathy.Journal of clinical medicine · 2022Review
- Article
- Article
- Filamin C Cardiomyopathy Variants Cause Protein and Lysosome Accumulation.Circulation research · 2021Article
- Cardiomyopathy, Proximal Myopathy, Camptocormia, and Novel Filamin C (FLNC) Variant: A Case Report.The American journal of case reports · 2021Article
- Development of the indirect flight muscles of Aedes aegypti, a main arbovirus vector.BMC developmental biology · 2021Article
- The Relationship Between Microbiomes and Selective Regimes in the Sponge GenusFrontiers in microbiology · 2021Article
- Panorama of the distal myopathies.Acta myologica : myopathies and cardiomyopathies : official journal of the Mediterranean Society of Myology · 2020Review
- Cytoskeletal Remodeling in Cancer.Biology · 2020Review
Corrections and comments
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Authors and funding
3 authors at 1 institution in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Many proteins contribute to the contractile properties of muscles, most notably myosin thick filaments, which are anchored at the M-line, and actin thin filaments, which are anchored at the Z-discs that border each sarcomere. In humans, mutations in the actin-binding protein Filamin-C result in myopathies, but the underlying molecular function is not well understood. Here we show using Drosophila indirect flight muscle that the filamin ortholog Cheerio in conjunction with the giant elastic protein titin plays a crucial role in keeping thin filaments stably anchored at the Z-disc. We identify the filamin domains required for interaction with the titin ortholog Sallimus, and we demonstrate a genetic interaction of filamin with titin and actin. Filamin mutants disrupting the actin- or the titin-binding domain display distinct phenotypes, with Z-discs breaking up in parallel or perpendicularly to the myofibril, respectively. Thus, Z-discs require filamin to withstand the strong contractile forces acting on them.
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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.