ArticleThe Journal of cell biology2006
M line-deficient titin causes cardiac lethality through impaired maturation of the sarcomere.
Article in The Journal of cell biology, 2006. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 51 papers.
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Who cites it
51 citing papers in PubMed.
- Titin's P-zone domains A164-167 are essential for thick filament structural arrangement.The Journal of general physiology · 2026Article
- Mending the Achilles heels of titin in cardiac and musculoskeletal disease.Biophysical reviews · 2026Review
- Location-Dependent Differences in Cardiac and Skeletal Muscle Dysfunction Associated With Truncating Titin (Circulation research · 2026Article
- Congenital Titinopathy: Comprehensive Characterization of the Most Severe End of the Disease Spectrum.Annals of neurology · 2025Article
- PROST: quantitative identification of spatially variable genes and domain detection in spatial transcriptomics.Nature communications · 2024Article
- Muscle-fiber specific genetic manipulation ofFrontiers in physiology · 2024Article
- Integration of spatial and single-cell transcriptomic data elucidates mouse organogenesis.Nature biotechnology · 2022Article
- Titin kinase ubiquitination aligns autophagy receptors with mechanical signals in the sarcomere.EMBO reports · 2021Article
- Titin M-line insertion sequence 7 is required for proper cardiac function in mice.Journal of cell science · 2021Article
- Genotype-phenotype correlations in recessive titinopathies.Genetics in medicine : official journal of the American College of Medical Genetics · 2020Article
- Maintenance of sarcomeric integrity in adult muscle cells crucially depends on Z-disc anchored titin.Nature communications · 2020Article
- Atypical ALPK2 kinase is not essential for cardiac development and function.American journal of physiology. Heart and circulatory physiology · 2020Article
- The M-band: The underestimated part of the sarcomere.Biochimica et biophysica acta. Molecular cell research · 2020Review
- Perturbation of the titin/MURF1 signaling complex is associated with hypertrophic cardiomyopathy in a fish model and in human patients.Disease models & mechanisms · 2019Article
- Cronos Titin Is Expressed in Human Cardiomyocytes and Necessary for Normal Sarcomere Function.Circulation · 2019Article
- Deleting Full Length Titin Versus the Titin M-Band Region Leads to Differential Mechanosignaling and Cardiac Phenotypes.Circulation · 2019Article
- Z-band and M-band titin splicing and regulation by RNA binding motif 20 in striated muscles.Journal of cellular biochemistry · 2018Article
- Congenital Titinopathy: Comprehensive characterization and pathogenic insights.Annals of neurology · 2018Article
- Genetic epidemiology of titin-truncating variants in the etiology of dilated cardiomyopathy.Biophysical reviews · 2017Review
- Exon- and contraction-dependent functions of titin in sarcomere assembly.Development (Cambridge, England) · 2016Article
Corrections and comments
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Authors and funding
5 authors.
Funding
Abstract
Titin, the largest protein known to date, has been linked to sarcomere assembly and function through its elastic adaptor and signaling domains. Titin's M-line region contains a unique kinase domain that has been proposed to regulate sarcomere assembly via its substrate titin cap (T-cap). In this study, we use a titin M line-deficient mouse to show that the initial assembly of the sarcomere does not depend on titin's M-line region or the phosphorylation of T-cap by the titin kinase. Rather, titin's M-line region is required to form a continuous titin filament and to provide mechanical stability of the embryonic sarcomere. Even without titin integrating into the M band, sarcomeres show proper spacing and alignment of Z discs and M bands but fail to grow laterally and ultimately disassemble. The comparison of disassembly in the developing and mature knockout sarcomere suggests diverse functions for titin's M line in embryonic development and the adult heart that not only involve the differential expression of titin isoforms but also of titin-binding proteins.
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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.