ArticleMechanisms of development2016
The zebrafish goosepimples/myosin Vb mutant exhibits cellular attributes of human microvillus inclusion disease.
Article in Mechanisms of development, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 15 papers.
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Who cites it
15 citing papers in PubMed, 31 citations in OpenAlex.
- Modeling the cell biology of monogenetic intestinal epithelial disorders.The Journal of cell biology · 2024Review
- Voltage-sensing phosphatase (Vsp) regulates endocytosis-dependent nutrient absorption in chordate enterocytes.Communications biology · 2022Article
- UNC45A deficiency causes microvillus inclusion disease-like phenotype by impairing myosin VB-dependent apical trafficking.The Journal of clinical investigation · 2022Article
- Case Report:Frontiers in genetics · 2022Article
- Experimental Studies on the Impact of the Projected Ocean Acidification on Fish Survival, Health, Growth, and Meat Quality; Black Sea Bream (Animals : an open access journal from MDPI · 2021Article
- Advanced Microscopy for Liver and Gut Ultrastructural Pathology in Patients with MVID and PFIC Caused by MYO5B Mutations.Journal of clinical medicine · 2021Article
- Tools of the trade: studying actin in zebrafish.Histochemistry and cell biology · 2020Review
- The Rab11 effectors Fip5 and Fip1 regulate zebrafish intestinal development.Biology open · 2020Article
- Multiple epithelia are required to develop teeth deep inside the pharynx.Proceedings of the National Academy of Sciences of the United States of America · 2020Article
- Article
- Loss of myosin Vb promotes apical bulk endocytosis in neonatal enterocytes.The Journal of cell biology · 2019Article
- Microridges are apical epithelial projections formed of F-actin networks that organize the glycan layer.Scientific reports · 2019Article
- A V0-ATPase-dependent apical trafficking pathway maintains the polarity of the intestinal absorptive membrane.Development (Cambridge, England) · 2019Article
- MYO5B, STX3, and STXBP2 mutations reveal a common disease mechanism that unifies a subset of congenital diarrheal disorders: A mutation update.Human mutation · 2018Review
- Intestinal epithelial cell polarity defects in disease: lessons from microvillus inclusion disease.Disease models & mechanisms · 2018Review
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Authors and funding
6 authors at 4 institutions in 1 country.
Funding
Abstract
Microvillus inclusion disease (MVID) is a life-threatening enteropathy characterised by malabsorption and incapacitating fluid loss due to chronic diarrhoea. Histological analysis has revealed that enterocytes in MVID patients exhibit reduction of microvilli, presence of microvillus inclusion bodies and intestinal villus atrophy, whereas genetic linkage analysis has identified mutations in myosin Vb gene as the main cause of MVID. In order to understand the cellular basis of MVID and the associated formation of inclusion bodies, an animal model that develops ex utero and is tractable genetically as well as by microscopy would be highly useful. Here we report that the intestine of the zebrafish goosepimples (gsp)/myosin Vb (myoVb) mutant shows severe reduction in intestinal folds - structures similar to mammalian villi. The loss of folds is further correlated with changes in the shape of enterocytes. In striking similarity with MVID patients, zebrafish gsp/myoVb mutant larvae exhibit microvillus atrophy, microvillus inclusions and accumulation of secretory material in enterocytes. We propose that the zebrafish gsp/myoVb mutant is a valuable model to study the pathophysiology of MVID. Furthermore, owing to the advantages of zebrafish in screening libraries of small molecules, the gsp mutant will be an ideal tool to identify compounds having therapeutic value against MVID.
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