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Germline hypomorphic CARD11 mutations in severe atopic disease
- Source :
- Nature Genetics. 49:1192-1201
- Publication Year :
- 2017
- Publisher :
- Springer Science and Business Media LLC, 2017.
-
Abstract
- Few monogenic causes for severe manifestations of common allergic diseases have been identified. Through next-generation sequencing on a cohort of patients with severe atopic dermatitis with and without comorbid infections, we found eight individuals, from four families, with novel heterozygous mutations in CARD11, which encodes a scaffolding protein involved in lymphocyte receptor signaling. Disease improved over time in most patients. Transfection of mutant CARD11 expression constructs into T cell lines demonstrated both loss-of-function and dominant-interfering activity upon antigen receptor-induced activation of nuclear factor-κB and mammalian target of rapamycin complex 1 (mTORC1). Patient T cells had similar defects, as well as low production of the cytokine interferon-γ (IFN-γ). The mTORC1 and IFN-γ production defects were partially rescued by supplementation with glutamine, which requires CARD11 for import into T cells. Our findings indicate that a single hypomorphic mutation in CARD11 can cause potentially correctable cellular defects that lead to atopic dermatitis.
- Subjects :
- Amino Acid Transport System ASC
Male
0301 basic medicine
Glutamine
T-Lymphocytes
Lymphocyte
T cell
DNA Mutational Analysis
mTORC1
Mechanistic Target of Rapamycin Complex 1
Biology
Lymphocyte Activation
Jurkat cells
Dermatitis, Atopic
Cohort Studies
Minor Histocompatibility Antigens
Jurkat Cells
03 medical and health sciences
Germline mutation
Antigen
Interferon
Genetics
medicine
Humans
Germ-Line Mutation
Genes, Dominant
TOR Serine-Threonine Kinases
NF-kappa B
Atopic dermatitis
medicine.disease
Pedigree
3. Good health
CARD Signaling Adaptor Proteins
030104 developmental biology
medicine.anatomical_structure
Guanylate Cyclase
Multiprotein Complexes
Immunology
Female
medicine.drug
Subjects
Details
- ISSN :
- 15461718 and 10614036
- Volume :
- 49
- Database :
- OpenAIRE
- Journal :
- Nature Genetics
- Accession number :
- edsair.doi.dedup.....0771c386e540f21e44863d09e0748ebd
- Full Text :
- https://doi.org/10.1038/ng.3898