| Literature DB >> 27734839 |
Lucia Mauri1, Emanuela Manfredini1, Alessandra Del Longo2, Emanuela Veniani1, Manuela Scarcello1, Roberta Terrana2, Adriano Egidio Radaelli3, Donata Calò3, Giuseppe Mingoia4, Antonella Rossetti4, Giovanni Marsico2, Marco Mazza2, Giovanni Pietro Gesu1, Maria Cristina Patrosso1, Silvana Penco1, Elena Piozzi2, Paola Primignani2.
Abstract
Oculocutaneous albinism (OCA) is characterized by hypopigmentation of the skin, hair and eye, and by ophthalmologic abnormalities caused by a deficiency in melanin biosynthesis. In this study we recruited 321 albino patients and screened them for the genes known to cause oculocutaneous albinism (OCA1-4 and OCA6) and ocular albinism (OA1). Our purpose was to detect mutations and genetic frequencies of the main causative genes, offering to albino patients an exhaustive diagnostic assessment within a multidisciplinary approach including ophthalmological, dermatological, audiological and genetic evaluations. We report 70 novel mutations and the frequencies of the major causative OCA genes that are as follows: TYR (44%), OCA2 (17%), TYRP1 (1%), SLC45A2 (7%) and SLC24A5 (<0.5%). An additional 5% of patients had GPR143 mutations. In 19% of cases, a second reliable mutation was not detected, whereas 7% of our patients remain still molecularly undiagnosed. This comprehensive study of a consecutive series of OCA/OA1 patients allowed us to perform a clinical evaluation of the different OCA forms.Entities:
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Year: 2016 PMID: 27734839 DOI: 10.1038/jhg.2016.123
Source DB: PubMed Journal: J Hum Genet ISSN: 1434-5161 Impact factor: 3.172