Gain of function p.E138A alteration in Card14 leads to psoriasiform skin inflammation and implicates genetic modifiers in disease severity.


Journal

Experimental and molecular pathology
ISSN: 1096-0945
Titre abrégé: Exp Mol Pathol
Pays: Netherlands
ID NLM: 0370711

Informations de publication

Date de publication:
10 2019
Historique:
received: 07 03 2019
revised: 18 06 2019
accepted: 16 07 2019
pubmed: 20 7 2019
medline: 28 2 2020
entrez: 20 7 2019
Statut: ppublish

Résumé

Psoriasis (PS) is a common inflammatory and incurable skin disease affecting 2-3% of the human population. Although genome-wide association studies implicate more than 60 loci, the full complement of genetic factors leading to disease is not known. Rare, highly penetrant, gain-of-function, dominantly acting mutations within the human caspase recruitment domain family, member 14 (CARD14) gene lead to the development of PS and psoriatic arthritis (PSA) (a familial p.G117S and de-novo p.E138A alteration). These residues are conserved in mouse and orthologous Knock-In (KI) mutations within Card14 were created. The Card14

Identifiants

pubmed: 31323190
pii: S0014-4800(19)30174-1
doi: 10.1016/j.yexmp.2019.104286
pmc: PMC7318844
mid: NIHMS1596590
pii:
doi:

Substances chimiques

CARD Signaling Adaptor Proteins 0
Membrane Proteins 0
Card14 protein, mouse EC 2.7.4.8
Guanylate Kinases EC 2.7.4.8
CARD14 protein, human EC 4.6.1.-
Guanylate Cyclase EC 4.6.1.2

Types de publication

Journal Article Research Support, N.I.H., Extramural Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

104286

Subventions

Organisme : NCI NIH HHS
ID : P30 CA034196
Pays : United States
Organisme : NIAMS NIH HHS
ID : R01 AR049288
Pays : United States
Organisme : NIAMS NIH HHS
ID : R01 AR050266
Pays : United States
Organisme : NIAMS NIH HHS
ID : R21 AR063781
Pays : United States

Informations de copyright

Copyright © 2019. Published by Elsevier Inc.

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Auteurs

John P Sundberg (JP)

The Jackson Laboratory, Bar Harbor, ME, USA. Electronic address: john.sundberg@jax.org.

C Herbert Pratt (CH)

The Jackson Laboratory, Bar Harbor, ME, USA.

Kathleen A Silva (KA)

The Jackson Laboratory, Bar Harbor, ME, USA.

Victoria E Kennedy (VE)

The Jackson Laboratory, Bar Harbor, ME, USA.

Wenning Qin (W)

eGenesis, Cambridge, MA, USA.

Timothy M Stearns (TM)

The Jackson Laboratory, Bar Harbor, ME, USA.

Jacqueline Frost (J)

Department of Oncological Sciences, Icahn School of Medicine at Mount Sinai, New York, NY, USA.

Beth A Sundberg (BA)

The Jackson Laboratory, Bar Harbor, ME, USA.

Anne M Bowcock (AM)

Department of Oncological Sciences, Icahn School of Medicine at Mount Sinai, New York, NY, USA; Departments of Dermatology, Genetics & Genome Sciences, Icahn School of Medicine at Mount Sinai, New York, NY, USA. Electronic address: anne.bowcock@mssm.edu.

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Classifications MeSH