Characterization of a mutant samhd1 zebrafish model implicates dysregulation of cholesterol biosynthesis in Aicardi-Goutières syndrome

Characterization of a mutant samhd1 zebrafish model implicates dysregulation of cholesterol biosynthesis in Aicardi-Goutières syndrome

Aicardi-Goutières syndrome (AGS1-9) is a genetically determined encephalopathy that falls under the type I interferonopathy disease class, characterized by excessive type I interferon (IFN-I) activity, coupled with upregulation of IFN-stimulated genes (ISGs), which can be explained by the vital role...

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Bibliographic Details
Main Authors: Sarah E. Withers, Charlie F. Rowlands, Victor S. Tapia, Frances Hedley, Ioana-Emilia Mosneag, Siobhan Crilly, Gillian I. Rice, Andrew P. Badrock, Andrew Hayes, Stuart M. Allan, Tracy A. Briggs, Paul R. Kasher
Format: Article
Language:English
Published: Frontiers Media S.A. 2023-03-01
Series:Frontiers in Immunology
Subjects:
Aicardi Goutières syndrome
cholesterol
zebrafish disease models
type I interferonopathy
SAMHD1
Online Access:https://www.frontiersin.org/articles/10.3389/fimmu.2023.1100967/full

Internet

https://www.frontiersin.org/articles/10.3389/fimmu.2023.1100967/full

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