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对一大组连续白化病患者进行临床评估和分子筛查。

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

作者信息

Mauri Lucia, Manfredini Emanuela, Del Longo Alessandra, Veniani Emanuela, Scarcello Manuela, Terrana Roberta, Radaelli Adriano Egidio, Calò Donata, Mingoia Giuseppe, Rossetti Antonella, Marsico Giovanni, Mazza Marco, Gesu Giovanni Pietro, Cristina Patrosso Maria, Penco Silvana, Piozzi Elena, Primignani Paola

机构信息

Medical Genetics Unit, Department of Laboratory Medicine, ASST Grande Ospedale Metropolitano Niguarda, Milan, Italy.

Pediatric Ophthalmology Unit, ASST Grande Ospedale Metropolitano Niguarda, Milan, Italy.

出版信息

J Hum Genet. 2017 Feb;62(2):277-290. doi: 10.1038/jhg.2016.123. Epub 2016 Oct 13.

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.

摘要

眼皮肤白化病(OCA)的特征是皮肤、毛发和眼睛色素减退,以及由黑色素生物合成缺陷引起的眼科异常。在本研究中,我们招募了321名白化病患者,并对已知导致眼皮肤白化病(OCA1 - 4和OCA6)和眼白化病(OA1)的基因进行了筛查。我们的目的是检测主要致病基因的突变和基因频率,在包括眼科、皮肤科、听力学和遗传学评估的多学科方法中,为白化病患者提供详尽的诊断评估。我们报告了70个新突变以及主要致病OCA基因的频率,具体如下:TYR(44%)、OCA2(17%)、TYRP1(1%)、SLC45A2(7%)和SLC24A5(<0.5%)。另外5%的患者有GPR143突变。在19%的病例中,未检测到第二个可靠的突变,而我们7%的患者在分子水平上仍未得到诊断。对一系列连续的OCA/OA1患者进行的这项综合研究使我们能够对不同类型的OCA进行临床评估。

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