ArticleJournal of pediatric gastroenterology and nutrition2016
Loss of a Candidate Biliary Atresia Susceptibility Gene, add3a, Causes Biliary Developmental Defects in Zebrafish.
Article in Journal of pediatric gastroenterology and nutrition, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 34 papers, 2 of them syntheses that pooled it.
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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
34 citing papers in PubMed, 2 syntheses or guidelines pooled it, 51 citations in OpenAlex.
- Systematic review of the mechanism and assessment of liver fibrosis in biliary atresia.Pediatric surgery international · 2024Pooled it
- The correlation between rs2501577 gene polymorphism and biliary atresia: a systematic review and meta-analysis.Pediatric surgery international · 2023Pooled it
- Multiple susceptibility enhancer variants increasingFrontiers in genetics · 2025Article
- Genetic background and biliary atresia.World journal of pediatric surgery · 2025Review
- Immunology of Biliary Atresia.Seminars in pediatric surgery · 2024Review
- A narrative review of genes associated with liver fibrosis in biliary atresia.Translational pediatrics · 2024Review
- Biliary atresia.Nature reviews. Disease primers · 2024Review
- Progress in Biomarkers Related to Biliary Atresia.Journal of clinical and translational hepatology · 2024Review
- Biliary atresia is associated with polygenic susceptibility in ciliogenesis and planar polarity effector genes.Journal of hepatology · 2023Article
- Loss of zebrafish pkd1l1 causes biliary defects that have implications for biliary atresia splenic malformation.Disease models & mechanisms · 2023Article
- Contribution ofInternational journal of molecular sciences · 2023Article
- Genetic Contributions to Biliary Atresia: A Developmental Cholangiopathy.Seminars in liver disease · 2023Review
- Association analysis and functional follow-up identified common variants ofFrontiers in genetics · 2023Article
- A novel model based on immune-related genes for differentiating biliary atresia from other cholestatic diseases.Pediatric surgery international · 2022Article
- Impact of EFEMP1 on the survival outcome of biliary atresia in Thai infants.Scientific reports · 2022Article
- Biliary Atresia Animal Models: Is the Needle in a Haystack?International journal of molecular sciences · 2022Review
- Role of long non-coding RNA-adducin 3 antisense RNA1 in liver fibrosis of biliary atresia.Bioengineered · 2022Article
- Genetic Factors and Their Role in the Pathogenesis of Biliary Atresia.Frontiers in pediatrics · 2022Review
- Biliary organoids uncover delayed epithelial development and barrier function in biliary atresia.Hepatology (Baltimore, Md.) · 2022Observational
- Biliary Atresia - emerging diagnostic and therapy opportunities.EBioMedicine · 2021Review
Corrections and comments
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Authors and funding
6 authors at 2 institutions in 1 country.
Funding
Abstract
objectivesBiliary atresia (BA) is a progressive fibroinflammatory cholangiopathy affecting the bile ducts of neonates. Although BA is the leading indication for pediatric liver transplantation, the etiology remains elusive. Adducin 3 (ADD3) and X-prolyl aminopeptidase 1 (XPNPEP1) are 2 genes previously identified in genome-wide association studies as potential BA susceptibility genes. Using zebrafish, we investigated the importance of ADD3 and XPNPEP1 in functional studies.
methodsTo determine whether loss of either gene leads to biliary defects, we performed morpholino antisense oligonucleotide (MO) knockdown studies targeting add3a and xpnpep1 in zebrafish. Individuals were assessed for decreases in biliary function and the presence of biliary defects. Quantitative polymerase chain reaction was performed on pooled 5 days postfertilization larvae to assess variations in transcriptional expression of genes of interest.
resultsAlthough both xpnpep1 and add3a are expressed in the developing zebrafish liver, only knockdown of add3a produced intrahepatic defects and decreased biliary function. Similar results were observed in homozygous add3a mutants. MO-mediated knockdown of add3a also showed higher mRNA expression of hedgehog (Hh) targets. Inhibition of Hh signaling rescued biliary defects caused by add3a knockdown. Combined knockdown of add3a and glypican-1 (gpc1), another mediator of Hh activity that is also a BA susceptibility gene, resulted in more severe biliary defects than knockdown of either alone.
conclusionsOur results support previous studies identifying ADD3 as a putative genetic risk factor for BA susceptibility. Our results also provide evidence that add3a may be affecting the Hh pathway, an important factor in BA pathogenesis.
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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.