ArticleThe Biochemical journal2016
Loss of cargo binding in the human myosin VI deafness mutant (R1166X) leads to increased actin filament binding.
Article in The Biochemical journal, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 14 papers.
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Who cites it
14 citing papers in PubMed, 21 citations in OpenAlex.
- Methionine metabolite spermidine inhibits tumor pyroptosis by enhancing MYO6-mediated endocytosis.Nature communications · 2025Article
- The lemur tail kinase family in neuronal function and disfunction in neurodegenerative diseases.Cellular and molecular life sciences : CMLS · 2024Review
- The twoFrontiers in physiology · 2024Article
- Binding partners regulate unfolding of myosin VI to activate the molecular motor.The Biochemical journal · 2022Article
- Functional Role of Class III Myosins in Hair Cells.Frontiers in cell and developmental biology · 2021Review
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- Structure of Myosin VI/Tom1 complex reveals a cargo recognition mode of Myosin VI for tethering.Nature communications · 2019Article
- Multiple myosin motors interact with sodium/potassium-ATPase alpha 1 subunits.Molecular brain · 2018Article
- Biological function of Lemur tyrosine kinase 2 (LMTK2): implications in neurodegeneration.Molecular brain · 2018Review
- The MYO6 interactome reveals adaptor complexes coordinating early endosome and cytoskeletal dynamics.EMBO reports · 2018Article
- Myosin VI-Dependent Actin Cages Encapsulate Parkin-Positive Damaged Mitochondria.Developmental cell · 2018Article
- NDP52 activates nuclear myosin VI to enhance RNA polymerase II transcription.Nature communications · 2017Article
- MYO6 Regulates Spatial Organization of Signaling Endosomes Driving AKT Activation and Actin Dynamics.Cell reports · 2017Article
Corrections and comments
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Authors and funding
5 authors at 2 institutions in 2 countries.
Funding
Abstract
Mutations in myosin VI have been associated with autosomal-recessive (DFNB37) and autosomal-dominant (DFNA22) deafness in humans. Here, we characterise an myosin VI nonsense mutation (R1166X) that was identified in a family with hereditary hearing loss in Pakistan. This mutation leads to the deletion of the C-terminal 120 amino acids of the myosin VI cargo-binding domain, which includes the WWY-binding motif for the adaptor proteins LMTK2, Tom1 as well as Dab2. Interestingly, compromising myosin VI vesicle-binding ability by expressing myosin VI with the R1166X mutation or with single point mutations in the adaptor-binding sites leads to increased F-actin binding of this myosin in vitro and in vivo As our results highlight the importance of cargo attachment for regulating actin binding to the motor domain, we perform a detailed characterisation of adaptor protein binding and identify single amino acids within myosin VI required for binding to cargo adaptors. We not only show that the adaptor proteins can directly interact with the cargo-binding tail of myosin VI, but our in vitro studies also suggest that multiple adaptor proteins can bind simultaneously to non-overlapping sites in the myosin VI tail. In conclusion, our characterisation of the human myosin VI deafness mutant (R1166X) suggests that defects in cargo binding may leave myosin VI in a primed/activated state with an increased actin-binding ability.
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Registered trials
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.