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Biliary-Atresia-associated Mannosidase-1-alpha-2 gene regulates biliary and ciliary morphogenesis and laterality (zebrafish)

ABSTRACT: The effect of MAN1A2 on biliary morphogenesis, left-right patterning, and ciliogenesis was evaluated with knockdown in zebrafish and subsequent RNAseq experiment/analysis.

ORGANISM(S): Danio rerio

PROVIDER: GSE138251 | GEO | 2020/11/18

REPOSITORIES: GEO

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Biliary-Atresia-associated Mannosidase-1-alpha-2 gene regulates biliary and ciliary morphogenesis and laterality

Project description:RNA sequencing was performed to evaluate dysregulated genes in EGF, TGF, developmental hedgehog and ciliogenesis pathways in human Billiary Atresia (BA)

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Biliary-Atresia-associated Mannosidase-1-alpha-2 gene regulates biliary and ciliary morphogenesis and laterality (mouse)

Project description:The effect of MAN1A2 on lung and liver development and ciliogenesis was evaluated with RNAseq analyses in knockout mice

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Biliary-Atresia-associated Mannosidase-1-alpha-2 gene regulates biliary and ciliary morphogenesis and laterality

Project description:Biliary-Atresia-associated Mannosidase-1-alpha-2 gene regulates biliary and ciliary morphogenesis and laterality

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Biliary-Atresia-associated Mannosidase-1-alpha-2 gene regulates biliary and ciliary morphogenesis and laterality (mouse)

Project description:Biliary-Atresia-associated Mannosidase-1-alpha-2 gene regulates biliary and ciliary morphogenesis and laterality (mouse)

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Biliary-Atresia-associated Mannosidase-1-alpha-2 gene regulates biliary and ciliary morphogenesis and laterality (zebrafish)

Project description:Biliary-Atresia-associated Mannosidase-1-alpha-2 gene regulates biliary and ciliary morphogenesis and laterality (zebrafish)

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Biliary-Atresia-Associated Mannosidase-1-Alpha-2 Gene Regulates Biliary and Ciliary Morphogenesis and Laterality.

Project description: Not available

| S-EPMC7662016 | biostudies-literature

Biliary organoids uncover delayed epithelial development and barrier function in biliary atresia.

Project description:Background: RNASeq was performed on organoids derived from livers of normal healthy donors and patients with biliary atresia to characterize transcriptomic signatures. Methods: Organoids generated from livers of normal healthy donors and patients with biliary atresia were cultured either in expansion (undifferentiated: 3 NCOs and 11 BACOs) or differentiation medium (differentiated: 3 BACOs). Liver tissues obtained from deceased-donor subjects served as normal controls (N=3). Total RNA was isolated from organoids and liver biopsy tissue specimens. Results: Organoids from patients with biliary atresia showed abnormal cell polarity, loss of tight junctions, increased permeability and decreased expression of genes related to epidermal growth factor (EGF)- and fibroblast growth factor 2 (FGF2)-signaling. When treated with EGF+FGF2, biliary atresia organoids expressed differentiation and functional markers with restored cell polarity. Conclusion: Organoids from biliary atresia are viable and have evidence of halted epithelial development. The induction of developmental markers, improved cell‐cell junction, and decreased epithelial permeability by EGF and FGF2 identifies potential strategies to promote epithelial maturation and function.

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Project description:We report label-free quantification of xenobiotic metabolizing enzymes (XME), transporters, redox enzymes, proteases and nucleases in 25 human liver microsomal samples, taken from patients with biliary atresia. Nearly 3500 proteins were identified and quantified. These data can be used in physiologically based pharmacokinetic models to predict appropriate drug doses drugs used in biliary atresia patients.

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Gene expression profile of human livers from patients with biliary atresia and controls (choledochocyst).

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Biliary atresia and gut environment

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