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SLC45A2基因的部分缺失导致杜宾犬出现眼皮肤白化病。

A partial gene deletion of SLC45A2 causes oculocutaneous albinism in Doberman pinscher dogs.

作者信息

Winkler Paige A, Gornik Kara R, Ramsey David T, Dubielzig Richard R, Venta Patrick J, Petersen-Jones Simon M, Bartoe Joshua T

机构信息

Department of Small Animal Clinical Sciences, College of Veterinary Medicine, Michigan State University, East Lansing, Michigan, United States of America; Genetics Program, Michigan State University, East Lansing, Michigan, United States of America.

Cummings School of Veterinary Medicine, Tufts University, North Grafton, Massachusetts, United States of America.

出版信息

PLoS One. 2014 Mar 19;9(3):e92127. doi: 10.1371/journal.pone.0092127. eCollection 2014.

Abstract

The first white Doberman pinscher (WDP) dog was registered by the American Kennel Club in 1976. The novelty of the white coat color resulted in extensive line breeding of this dog and her offspring. The WDP phenotype closely resembles human oculocutaneous albinism (OCA) and clinicians noticed a seemingly high prevalence of pigmented masses on these dogs. This study had three specific aims: (1) produce a detailed description of the ocular phenotype of WDPs, (2) objectively determine if an increased prevalence of ocular and cutaneous melanocytic tumors was present in WDPs, and (3) determine if a genetic mutation in any of the genes known to cause human OCA is causal for the WDP phenotype. WDPs have a consistent ocular phenotype of photophobia, hypopigmented adnexal structures, blue irides with a tan periphery and hypopigmented retinal pigment epithelium and choroid. WDPs have a higher prevalence of cutaneous melanocytic neoplasms compared with control standard color Doberman pinschers (SDPs); cutaneous tumors were noted in 12/20 WDP (<5 years of age: 4/12; >5 years of age: 8/8) and 1/20 SDPs (p<0.00001). Using exclusion analysis, four OCA causative genes were investigated for their association with WDP phenotype; TYR, OCA2, TYRP1 and SLC45A2. SLC45A2 was found to be linked to the phenotype and gene sequencing revealed a 4,081 base pair deletion resulting in loss of the terminus of exon seven of SLC45A2 (chr4∶77,062,968-77,067,051). This mutation is highly likely to be the cause of the WDP phenotype and is supported by a lack of detectable SLC45A2 transcript levels by reverse transcriptase PCR. The WDP provides a valuable model for studying OCA4 visual disturbances and melanocytic neoplasms in a large animal model.

摘要

1976年,第一只白色杜宾犬(WDP)被美国养犬俱乐部登记注册。白色被毛颜色的新奇性导致了对这只犬及其后代的广泛品系繁育。WDP的表型与人类眼皮肤白化病(OCA)极为相似,临床医生注意到这些犬身上色素性肿物的发生率似乎很高。本研究有三个具体目标:(1)详细描述WDP的眼部表型;(2)客观确定WDP中眼和皮肤黑素细胞肿瘤的发生率是否增加;(3)确定已知导致人类OCA的任何基因中的基因突变是否是WDP表型的病因。WDP具有一致的眼部表型,即畏光症、附属器结构色素减退、虹膜呈蓝色且周边为棕褐色、视网膜色素上皮和脉络膜色素减退。与对照标准颜色杜宾犬(SDP)相比,WDP皮肤黑素细胞肿瘤的发生率更高;在12/20只WDP中发现了皮肤肿瘤(<5岁:4/12;>5岁:8/8),而在20只SDP中仅发现1只(p<0.00001)。采用排除分析方法,研究了四个OCA致病基因与WDP表型的关联;酪氨酸酶(TYR)、OCA2、酪氨酸酶相关蛋白1(TYRP1)和溶质载体家族45成员2(SLC45A2)。发现SLC45A2与该表型相关,基因测序显示有一个4081个碱基对的缺失,导致SLC45A2外显子7末端缺失(染色体4∶77,062,968 - 77,067,051)。这种突变极有可能是WDP表型的病因,逆转录聚合酶链反应未检测到SLC45A2转录水平也支持了这一点。WDP为在大型动物模型中研究OCA4视觉障碍和黑素细胞肿瘤提供了一个有价值的模型。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6982/3960214/155a0e1d9757/pone.0092127.g001.jpg

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