ArticleDevelopmental biology2012
Mutations in vacuolar H+ -ATPase subunits lead to biliary developmental defects in zebrafish.
Article in Developmental biology, 2012. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 16 papers.
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Who cites it
16 citing papers in PubMed, 27 citations in OpenAlex.
- Forward genetics combined with unsupervised classifications identified zebrafish mutants affecting biliary system formation.Developmental biology · 2024Article
- Defective Lipid Droplet-Lysosome Interaction Causes Fatty Liver Disease as Evidenced by Human Mutations in TMEM199 and CCDC115.Cellular and molecular gastroenterology and hepatology · 2022Article
- Single-cell transcriptomic analysis of zebrafish cranial neural crest reveals spatiotemporal regulation of lineage decisions during development.Cell reports · 2021Article
- Loss of vacuolar-type H+-ATPase induces caspase-independent necrosis-like death of hair cells in zebrafish neuromasts.Disease models & mechanisms · 2021Article
- A Model Construction of Starvation Induces Hepatic Steatosis and Transcriptome Analysis in Zebrafish Larvae.Biology · 2021Article
- Microarray Expression Profiling and Raman Spectroscopy Reveal Anti-Fatty Liver Action of Berberine in a Diet-Induced Larval Zebrafish Model.Frontiers in pharmacology · 2019Article
- Atp6ap2 ablation in adult mice impairs viability through multiple organ deficiencies.Scientific reports · 2017Article
- Loss of a Candidate Biliary Atresia Susceptibility Gene, add3a, Causes Biliary Developmental Defects in Zebrafish.Journal of pediatric gastroenterology and nutrition · 2016Article
- Methylation Microarray Studies Highlight PDGFA Expression as a Factor in Biliary Atresia.PloS one · 2016Article
- Zebrafish: an important tool for liver disease research.Gastroenterology · 2015Review
- The V-ATPase accessory protein Atp6ap1b mediates dorsal forerunner cell proliferation and left-right asymmetry in zebrafish.Developmental biology · 2015Article
- Identification of Annexin A4 as a hepatopancreas factor involved in liver cell survival.Developmental biology · 2014Article
- Zebrafish models of human liver development and disease.Comprehensive Physiology · 2013Review
- Article
- Evidence from human and zebrafish that GPC1 is a biliary atresia susceptibility gene.Gastroenterology · 2013Article
- The zebrafish mutants for the V-ATPase subunits d, ac45, E, H and c and their variable pigment dilution phenotype.BMC research notes · 2013Article
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Authors and funding
12 authors at 2 institutions in 1 country.
Funding
Abstract
We identified three zebrafish mutants with defects in biliary development. One of these mutants, pekin (pn), also demonstrated generalized hypopigmentation and other defects, including disruption of retinal cell layers, lack of zymogen granules in the pancreas, and dilated Golgi in intestinal epithelial cells. Bile duct cells in pn demonstrated an accumulation of electron dense bodies. We determined that the causative defect in pn was a splice site mutation in the atp6ap2 gene that leads to an inframe stop codon. atp6ap2 encodes a subunit of the vacuolar H(+)-ATPase (V-H(+)-ATPase), which modulates pH in intracellular compartments. The Atp6ap2 subunit has also been shown to function as an intracellular renin receptor that stimulates fibrogenesis. Here we show that mutants and morphants involving other V-H(+)-ATPase subunits also demonstrated developmental biliary defects, but did not demonstrate the inhibition of fibrogenic genes observed in pn. The defects in pn are reminiscent of those we and others have observed in class C VPS (vacuolar protein sorting) family mutants and morphants, and we report here that knockdown of atp6ap2 and vps33b had an additive negative effect on biliary development. Our findings suggest that pathways which are important in modulating intracompartmental pH lead to defects in digestive organ development, and support previous studies demonstrating the importance of intracellular sorting pathways in biliary development.
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