ArticleMolecular biology of the cell2007
Dystroglycan and protein O-mannosyltransferases 1 and 2 are required to maintain integrity of Drosophila larval muscles.
Article in Molecular biology of the cell, 2007. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 40 papers.
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Who cites it
40 citing papers in PubMed, 62 citations in OpenAlex.
- An Unbiased Drug Screen in a Drosophila Model ofInternational journal of molecular sciences · 2026Article
- Review
- Neurexin facilitates glycosylation of Dystroglycan to sustain muscle architecture and function in Drosophila.Communications biology · 2024Article
- Protein O-mannosylation: one sugar, several pathways, many functions.Glycobiology · 2023Review
- Adult expression of Semaphorins and Plexins is essential for motor neuron survival.Scientific reports · 2023Article
- Alanine-scanning mutagenesis of protein mannosyl-transferase fromMicrobiology (Reading, England) · 2021Article
- High degree of conservation of the enzymes synthesizing the laminin-binding glycoepitope of α-dystroglycan.Open biology · 2021Article
- Structural, Evolutionary, and Functional Analysis of the Protein O-Mannosyltransferase Family in Pathogenic Fungi.Journal of fungi (Basel, Switzerland) · 2021Review
- Mucin-TypeFrontiers in neuroanatomy · 2021Article
- Synthesis and biological roles of O-glycans in insects.Glycoconjugate journal · 2020Review
- Profiling of the muscle-specific dystroglycan interactome reveals the role of Hippo signaling in muscular dystrophy and age-dependent muscle atrophy.BMC medicine · 2020Article
- O-Linked glycosylation in Drosophila melanogaster.Current opinion in structural biology · 2019Review
- The Neuronal Overexpression ofFrontiers in genetics · 2019Article
- Protein O-Mannosyltransferases Affect Sensory Axon Wiring and Dynamic Chirality of Body Posture in theThe Journal of neuroscience : the official journal of the Society for Neuroscience · 2018Article
- Influence of ovarian muscle contraction and oocyte growth on egg chamber elongation in Drosophila.Development (Cambridge, England) · 2016Article
- Transgenic Drosophila for Investigating DUX4 and FRG1, Two Genes Associated with Facioscapulohumeral Muscular Dystrophy (FSHD).PloS one · 2016Article
- Genetic Engineering of Dystroglycan in Animal Models of Muscular Dystrophy.BioMed research international · 2015Review
- Article
- Protein O-mannosylation is crucial for E-cadherin-mediated cell adhesion.Proceedings of the National Academy of Sciences of the United States of America · 2013Article
- N-glycosylation requirements in neuromuscular synaptogenesis.Development (Cambridge, England) · 2013Article
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
In vertebrates, mutations in Protein O-mannosyltransferase1 (POMT1) or POMT2 are associated with muscular dystrophy due to a requirement for O-linked mannose glycans on the Dystroglycan (Dg) protein. In this study we examine larval body wall muscles of Drosophila mutant for Dg, or RNA interference knockdown for Dg and find defects in muscle attachment, altered muscle contraction, and a change in muscle membrane resistance. To determine if POMTs are required for Dg function in Drosophila, we examine larvae mutant for genes encoding POMT1 or POMT2. Larvae mutant for either POMT, or doubly mutant for both, show muscle attachment and muscle contraction phenotypes identical to those associated with reduced Dg function, consistent with a requirement for O-linked mannose on Drosophila Dg. Together these data establish a central role for Dg in maintaining integrity in Drosophila larval muscles and demonstrate the importance of glycosylation to Dg function in Drosophila. This study opens the possibility of using Drosophila to investigate muscular dystrophy.
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