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Clinical evaluation and molecular screening of a large consecutive series of albino patients.

Authors :
Mauri L
Manfredini E
Del Longo A
Veniani E
Scarcello M
Terrana R
Radaelli AE
Calò D
Mingoia G
Rossetti A
Marsico G
Mazza M
Gesu GP
Cristina Patrosso M
Penco S
Piozzi E
Primignani P
Source :
Journal of human genetics [J Hum Genet] 2017 Feb; Vol. 62 (2), pp. 277-290. Date of Electronic Publication: 2016 Oct 13.
Publication Year :
2017

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.

Details

Language :
English
ISSN :
1435-232X
Volume :
62
Issue :
2
Database :
MEDLINE
Journal :
Journal of human genetics
Publication Type :
Academic Journal
Accession number :
27734839
Full Text :
https://doi.org/10.1038/jhg.2016.123