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Integrin α3 mutations with kidney, lung, and skin disease.
- 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.
- Subjects :
- Epidermolysis Bullosa immunology
Epidermolysis Bullosa pathology
Fatal Outcome
Female
Homozygote
Humans
Infant, Newborn
Kidney pathology
Lung diagnostic imaging
Lung pathology
Lung Diseases diagnosis
Male
Mutation
Nephrotic Syndrome congenital
Nephrotic Syndrome pathology
Radiography
Skin immunology
Skin pathology
Epidermolysis Bullosa genetics
Integrin alpha3 genetics
Lung Diseases genetics
Nephrotic Syndrome genetics
Subjects
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