A role of glycosphingolipids in tube morphogenesis and neuronal polarity in C. elegans

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	A role of glycosphingolipids in tube morphogenesis and neuronal polarity in C. elegans
	Cui, M.; Ying, R.; Jiang, X.; Zhang, H.
	2018-08-01
Source Publication	EMBO Workshop-Chemical Biology 2018
Abstract	Hereditary sensory and autonomic neuropathy type 1 (HSAN1) is a rare autosomal dominant inherited peripheral neuropathy. Mutations in subunits of serine palmitoyltransferase have been linked to the majority of HSAN1 cases. Despite extensive investigation, the molecular pathogenesis of HSAN1 remains controversial. Herein, we established a C. elegans model of HSAN1 by generating a sptl-1(c363g) mutation, encoding SPTL-1(C121W) and equivalent to human SPTLC1C133W, at the C. elegans genomic locus through CRISPR. sptl-1(c363g) homozygous mutants are larval lethal with epithelial polarity defect. sptl-1(c363g)/+ heterozygotes as well as SPTL-1(C121W)-overexpressing animals exhibited neuronal morphology and axon-dendrite polarity defects with concomitant locomotion and sensory deficits, implicating physiological resemblance of the C. elegans model with human patients. sptl-1(c363g)-derived defects were rescued by overexpressing a ceramide glucosyltransferase CGT-3, demonstrating that SPTLC1C133W contribute to HSAN1 pathogenesis through disrupting glucosylceramide biosynthesis. Genetic interactions between SPTL-1(C121W) and the mutations in clathrin adaptor AP3 and kinesin suggested that glycosphingolipids regulate neuronal polarity through participating in polarized vesicle trafficking.
Keyword	Glycosphingolipids tube morphogenesis neuronal polarity C. elegans
Language	英語English
The Source to Article	PB_Publication
PUB ID	46759
Document Type	Conference paper
Collection	DEPARTMENT OF BIOMEDICAL SCIENCES Faculty of Health Sciences
Recommended Citation GB/T 7714	Cui, M.,Ying, R.,Jiang, X.,et al. A role of glycosphingolipids in tube morphogenesis and neuronal polarity in C. elegans[C], 2018.
APA	Cui, M.., Ying, R.., Jiang, X.., & Zhang, H. (2018). A role of glycosphingolipids in tube morphogenesis and neuronal polarity in C. elegans. EMBO Workshop-Chemical Biology 2018.

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