Disease-related PSS1 mutant impedes the formation and function of osteoclasts.

Glycerophospholipids Lenz-Majewski syndrome Lipidomics Phospholipids Phospholipids/Biosynthesis Phospholipids/Metabolism osteoclasts phosphatidylserine

Journal

Journal of lipid research
ISSN: 1539-7262
Titre abrégé: J Lipid Res
Pays: United States
ID NLM: 0376606

Informations de publication

Date de publication:
Nov 2023
Historique:
received: 06 02 2023
revised: 01 09 2023
accepted: 04 09 2023
medline: 20 11 2023
pubmed: 16 9 2023
entrez: 15 9 2023
Statut: ppublish

Résumé

Phosphatidylserine (PS) is an acidic phospholipid that is involved in various cellular events. Heterologous dominant mutations have been identified in the gene encoding PS synthase 1 (PSS1) in patients with a congenital disease called Lenz-Majewski syndrome (LMS). Patients with LMS show various symptoms, including craniofacial/distal-limb bone dysplasia and progressive hyperostosis. The LMS-causing gain-of-function mutants of PSS1 (PSS1

Identifiants

pubmed: 37714410
pii: S0022-2275(23)00116-5
doi: 10.1016/j.jlr.2023.100443
pmc: PMC10641532
pii:
doi:

Substances chimiques

Phospholipids 0
phospholipid serine base exchange enzyme EC 2.7.8.-

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

100443

Informations de copyright

Copyright © 2023 The Authors. Published by Elsevier Inc. All rights reserved.

Déclaration de conflit d'intérêts

Conflict of interest The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.

Auteurs

Sari Sugahara (S)

Department of Health Chemistry, Graduate School of Pharmaceutical Sciences, The University of Tokyo, Tokyo, Japan.

Yuki Ishino (Y)

Department of Health Chemistry, Graduate School of Pharmaceutical Sciences, The University of Tokyo, Tokyo, Japan.

Koki Sawada (K)

Department of Health Chemistry, Graduate School of Pharmaceutical Sciences, The University of Tokyo, Tokyo, Japan.

Tsumugi Iwata (T)

Department of Health Chemistry, Graduate School of Pharmaceutical Sciences, The University of Tokyo, Tokyo, Japan.

Yuta Shimanaka (Y)

Department of Health Chemistry, Graduate School of Pharmaceutical Sciences, The University of Tokyo, Tokyo, Japan.

Junken Aoki (J)

Department of Health Chemistry, Graduate School of Pharmaceutical Sciences, The University of Tokyo, Tokyo, Japan.

Hiroyuki Arai (H)

Department of Health Chemistry, Graduate School of Pharmaceutical Sciences, The University of Tokyo, Tokyo, Japan.

Nozomu Kono (N)

Department of Health Chemistry, Graduate School of Pharmaceutical Sciences, The University of Tokyo, Tokyo, Japan. Electronic address: nozomu@mol.f.u-tokyo.ac.jp.

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Classifications MeSH