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Integrin α3 mutations with kidney, lung, and skin disease.

Authors :
Has C
Spartà G
Kiritsi D
Weibel L
Moeller A
Vega-Warner V
Waters A
He Y
Anikster Y
Esser P
Straub BK
Hausser I
Bockenhauer D
Dekel B
Hildebrandt F
Bruckner-Tuderman L
Laube GF
Source :
The New England journal of medicine [N Engl J Med] 2012 Apr 19; Vol. 366 (16), pp. 1508-14.
Publication Year :
2012

Abstract

Integrin α(3) is a transmembrane integrin receptor subunit that mediates signals between the cells and their microenvironment. We identified three patients with homozygous mutations in the integrin α(3) gene that were associated with disrupted basement-membrane structures and compromised barrier functions in kidney, lung, and skin. The patients had a multiorgan disorder that included congenital nephrotic syndrome, interstitial lung disease, and epidermolysis bullosa. The renal and respiratory features predominated, and the lung involvement accounted for the lethal course of the disease. Although skin fragility was mild, it provided clues to the diagnosis.

Details

Language :
English
ISSN :
1533-4406
Volume :
366
Issue :
16
Database :
MEDLINE
Journal :
The New England journal of medicine
Publication Type :
Academic Journal
Accession number :
22512483
Full Text :
https://doi.org/10.1056/NEJMoa1110813