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Structural basis for defective membrane targeting of mutant enzyme in human VLCAD deficiency

Authors :
Michelle S. Prew
Christina M. Camara
Thomas Botzanowski
Jamie A. Moroco
Noah B. Bloch
Hannah R. Levy
Hyuk-Soo Seo
Sirano Dhe-Paganon
Gregory H. Bird
Henry D. Herce
Micah A. Gygi
Silvia Escudero
Thomas E. Wales
John R. Engen
Loren D. Walensky
Source :
Nature Communications, Vol 13, Iss 1, Pp 1-12 (2022)
Publication Year :
2022
Publisher :
Nature Portfolio, 2022.

Abstract

Prew et al. uncovered a structural basis for human VLCAD deficiency that arises from point mutations within the enzyme’s membrane-binding region, which was shown to fold as a putative α-helical hairpin. Helix-breaking mutations selectively disrupt membrane interaction and thus homeostatic function.

Subjects

Subjects :
Science

Details

Language :
English
ISSN :
20411723
Volume :
13
Issue :
1
Database :
Directory of Open Access Journals
Journal :
Nature Communications
Publication Type :
Academic Journal
Accession number :
edsdoj.862c131a10741b096477d28809d47d0
Document Type :
article
Full Text :
https://doi.org/10.1038/s41467-022-31466-2