| Literature DB >> 22512483 |
Cristina Has1, Giuseppina Spartà, Dimitra Kiritsi, Lisa Weibel, Alexander Moeller, Virginia Vega-Warner, Aoife Waters, Yinghong He, Yair Anikster, Philipp Esser, Beate K Straub, Ingrid Hausser, Detlef Bockenhauer, Benjamin Dekel, Friedhelm Hildebrandt, Leena Bruckner-Tuderman, Guido F Laube.
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.Entities:
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Year: 2012 PMID: 22512483 PMCID: PMC3341404 DOI: 10.1056/NEJMoa1110813
Source DB: PubMed Journal: N Engl J Med ISSN: 0028-4793 Impact factor: 91.245