ArticleHuman molecular genetics2019
Selective serotonin reuptake inhibitors ameliorate MEGF10 myopathy.
Article in Human molecular genetics, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 9 papers.
What it found
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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
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Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
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Who cites it
9 citing papers in PubMed, 16 citations in OpenAlex.
- The role of theERJ open research · 2025Article
- Cellular and molecular alterations to muscles and neuromuscular synapses in a mouse model of MEGF10-related myopathy.Skeletal muscle · 2024Article
- Article
- The Notch signaling pathway in skeletal muscle health and disease.Muscle & nerve · 2022Review
- Congenital myopathy associated with a novel mutation inActa myologica : myopathies and cardiomyopathies : official journal of the Mediterranean Society of Myology · 2022Article
- Phenotypic Variability ofGenes · 2021Article
- Megf10 deficiency impairs skeletal muscle stem cell migration and muscle regeneration.FEBS open bio · 2021Article
- Effect of serotonin modulation on dystrophin-deficient zebrafish.Biology open · 2020Article
- Skeletal muscle in health and disease.Disease models & mechanisms · 2020Review
Corrections and comments
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Authors and funding
14 authors at 9 institutions in 1 country.
Funding
Abstract
MEGF10 myopathy is a rare inherited muscle disease that is named after the causative gene, MEGF10. The classic phenotype, early onset myopathy, areflexia, respiratory distress and dysphagia, is severe and immediately life-threatening. There are no disease-modifying therapies. We performed a small molecule screen and follow-up studies to seek a novel therapy. A primary in vitro drug screen assessed cellular proliferation patterns in Megf10-deficient myoblasts. Secondary evaluations were performed on primary screen hits using myoblasts derived from Megf10-/- mice, induced pluripotent stem cell-derived myoblasts from MEGF10 myopathy patients, mutant Drosophila that are deficient in the homologue of MEGF10 (Drpr) and megf10 mutant zebrafish. The screen yielded two promising candidates that are both selective serotonin reuptake inhibitors (SSRIs), sertraline and escitalopram. In depth follow-up analyses demonstrated that sertraline was highly effective in alleviating abnormalities across multiple models of the disease including mouse myoblast, human myoblast, Drosophila and zebrafish models. Sertraline also restored deficiencies of Notch1 in disease models. We conclude that SSRIs show promise as potential therapeutic compounds for MEGF10 myopathy, especially sertraline. The mechanism of action may involve the Notch pathway.
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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.