ArticleJournal of molecular medicine (Berlin, Germany)2015
Animal model of Sar1b deficiency presents lipid absorption deficits similar to Anderson disease.
Article in Journal of molecular medicine (Berlin, Germany), 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 26 papers.
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26 citing papers in PubMed, 47 citations in OpenAlex.
- Case Report: Neonatal-onset chylomicron retention disease presenting as isolated failure to thrive with compound heterozygousFrontiers in pediatrics · 2026Article
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- TANGO1 Dances to Export of Procollagen from the Endoplasmic Reticulum.Fibrosis (Hong Kong, China) · 2023Article
- High-fat diet reveals the impact of Sar1b defects on lipid and lipoprotein profile and cholesterol metabolism.Journal of lipid research · 2023Article
- Article
- Sorting and Export of Proteins at the Endoplasmic Reticulum.Cold Spring Harbor perspectives in biology · 2023Review
- Small sequence variations between two mammalian paralogs of the small GTPase SAR1 underlie functional differences in coat protein complex II assembly.The Journal of biological chemistry · 2020Article
- Metabolic coessentiality mapping identifies C12orf49 as a regulator of SREBP processing and cholesterol metabolism.Nature metabolism · 2020Article
- Distinct roles for luminal acidification in apical protein sorting and trafficking in zebrafish.The Journal of cell biology · 2020Article
- Consequences of mutations in the genes of the ER export machinery COPII in vertebrates.Cell stress & chaperones · 2020Review
- Key roles of Arf small G proteins and biosynthetic trafficking for animal development.Small GTPases · 2019Review
- SAR1B GTPase is necessary to protect intestinal cells from disorders of lipid homeostasis, oxidative stress, and inflammation.Journal of lipid research · 2019Article
- GRIK5 Genetically Regulated Expression Associated with Eye and Vascular Phenomes: Discovery through Iteration among Biobanks, Electronic Health Records, and Zebrafish.American journal of human genetics · 2019Article
- ATF4 overexpression induces early onset of hyperlipidaemia and hepatic steatosis and enhances adipogenesis in zebrafish.Scientific reports · 2017Article
- Muscle fatigue: general understanding and treatment.Experimental & molecular medicine · 2017Review
- A genetic screen for zebrafish mutants with hepatic steatosis identifies a locus required for larval growth.Journal of anatomy · 2017Article
- Regulation of the Sar1 GTPase Cycle Is Necessary for Large Cargo Secretion from the Endoplasmic Reticulum.Frontiers in cell and developmental biology · 2017Review
- Role of Intestinal LXRα in Regulating Post-prandial Lipid Excursion and Diet-Induced Hypercholesterolemia and Hepatic Lipid Accumulation.Frontiers in physiology · 2017Article
- Recent discoveries on absorption of dietary fat: Presence, synthesis, and metabolism of cytoplasmic lipid droplets within enterocytes.Biochimica et biophysica acta · 2016Review
- Zebrafish Models for Dyslipidemia and Atherosclerosis Research.Frontiers in endocrinology · 2016Review
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6 authors at 3 institutions in 2 countries.
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Abstract
Anderson disease (ANDD) or chylomicron retention disease (CMRD) is a rare, hereditary lipid malabsorption syndrome associated with mutations in the SAR1B gene that is characterized by failure to thrive and hypocholesterolemia. Although the SAR1B structure has been resolved and its role in formation of coat protein II (COPII)-coated carriers is well established, little is known about the requirement for SAR1B during embryogenesis. To address this question, we have developed a zebrafish model of Sar1b deficiency based on antisense oligonucleotide knockdown. We show that zebrafish sar1b is highly conserved among vertebrates; broadly expressed during development; and enriched in the digestive tract organs, brain, and craniofacial skeleton. Consistent with ANDD symptoms of chylomicron retention, we found that dietary lipids in Sar1b-deficient embryos accumulate in enterocytes. Transgenic expression analysis revealed that Sar1b is required for growth of exocrine pancreas and liver. Furthermore, we found abnormal differentiation and maturation of craniofacial cartilage associated with defects in procollagen II secretion and absence of select, neuroD-positive neurons of the midbrain and hindbrain. The model presented here will help to systematically dissect developmental roles of Sar1b and to discover molecular and cellular mechanisms leading to organ-specific ANDD pathology. Key messages: Sar1b depletion phenotype in zebrafish resembles Anderson disease deficits. Sar1b deficiency results in multi-organ developmental deficits. Sar1b is required for dietary cholesterol uptake into enterocytes.
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