| Literature DB >> 25898926 |
Jamal Ghoumid1,2, Florence Petit1,2, Muriel Holder-Espinasse1,2,3, Anne-Sophie Jourdain4, José Guerra1, Anne Dieux-Coeslier1, Martin Figeac5, Nicole Porchet2,3, Sylvie Manouvrier-Hanu1,2, Fabienne Escande3.
Abstract
Nail-Patella Syndrome (NPS) is a rare autosomal dominant condition comprising nail and skeletal anomalies. Skeletal features include dysplastic patellae and iliac horns, as well as scapula and elbow dysplasia. Nephropathy and glaucoma or intra-ocular hypertension can sometimes be present. NPS is due to variants affecting function in LMX1B, which encodes a LIM-homeodomain protein critical for limb, kidney and eye development. We describe the phenotype and the molecular data of 55 index patients and their 39 relatives presenting with typical NPS. We identified 38 different LMX1B anomalies, 19 of which were not reported before. In our series, 9% of families are not carriers of a LMX1B genomic alteration after extensive study of the coding and non-coding regions of the gene. One of the families showed no linkage to the LMX1B locus, raising the hypothesis of a genetic heterogeneity.Entities:
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Year: 2015 PMID: 25898926 PMCID: PMC4795216 DOI: 10.1038/ejhg.2015.77
Source DB: PubMed Journal: Eur J Hum Genet ISSN: 1018-4813 Impact factor: 4.246