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J Hum Genet ; 62(2): 277-290, 2017 Feb.
Artigo em Inglês | MEDLINE | ID: mdl-27734839

RESUMO

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.


Assuntos
Albinismo Oculocutâneo/diagnóstico , Albinismo Oculocutâneo/genética , Antígenos de Neoplasias/genética , Antiporters/genética , Proteínas do Olho/genética , Glicoproteínas de Membrana/genética , Proteínas de Membrana/genética , Proteínas de Membrana Transportadoras/genética , Oxirredutases/genética , Adulto , Idoso , Testes Genéticos , Humanos , Masculino , Melaninas/biossíntese , Pessoa de Meia-Idade
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