Literature DB >> 19865097

A comprehensive analysis reveals mutational spectra and common alleles in Chinese patients with oculocutaneous albinism.

Aihua Wei1, Yu Wang, Yan Long, Yi Wang, Xiaoli Guo, Zhiyong Zhou, Wei Zhu, Juntao Liu, Xuming Bian, Shi Lian, Wei Li.   

Abstract

Oculocutaneous albinism (OCA) is a heterogeneous recessive disorder with hypopigmentation in the skin, hair, and eyes. At least 16 genes have been identified as causative genes for human OCA. No comprehensive analysis has been conducted to study the spectral distribution of OCA in Chinese patients. We screened 127 unrelated and unselected Chinese OCA patients for mutations in the TYR, OCA2, TYRP1, SLC45A2, and HPS1 genes. We found that the spectrum of mutational genes and alleles of OCA is population specific. OCA1 is the most common (70.1% of cases) form of Chinese OCA, whereas OCA2, OCA4, and HPS1 account for 10.2%, 12.6%, and 1.6%, respectively. No apparent pathological mutation of TYRP1 has been found. Thirty-eight previously unreported mutational alleles were identified from these OCA patients and were not found in 100 nonalbinism subjects. Of the TYR mutational alleles, 81.1% were clustered on exons 1 and 2. Ten common alleles account for 74.6% of the mutational TYR alleles in Chinese OCA1 patients. The p.D160H allele accounts for 55.6% of the mutational SLC45A2 alleles in Chinese OCA4 patients. These results provide useful information for the establishment of an optimized strategy of gene diagnosis and genetic counseling of Chinese OCA patients.

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Year:  2009        PMID: 19865097     DOI: 10.1038/jid.2009.339

Source DB:  PubMed          Journal:  J Invest Dermatol        ISSN: 0022-202X            Impact factor:   8.551


  24 in total

1.  Homozygosity mapping in albinism patients using a novel panel of 13 STR markers inside the nonsyndromic OCA genes: introducing 5 novel mutations.

Authors:  Faravareh Khordadpoor-Deilamani; Mohammad Taghi Akbari; Morteza Karimipoor; Gholam Reza Javadi
Journal:  J Hum Genet       Date:  2016-01-28       Impact factor: 3.172

2.  Molecular analysis of Korean patients with oculocutaneous albinism.

Authors:  Shin Hae Park; Hyojin Chae; Yonggoo Kim; Myungshin Kim
Journal:  Jpn J Ophthalmol       Date:  2011-11-01       Impact factor: 2.447

3.  The BLOS1-interacting protein KXD1 is involved in the biogenesis of lysosome-related organelles.

Authors:  Qing Yang; Xin He; Lin Yang; Zhiyong Zhou; Andrew R Cullinane; Aihua Wei; Zhe Zhang; Zhenhua Hao; Aili Zhang; Min He; Yaqin Feng; Xiang Gao; William A Gahl; Marjan Huizing; Wei Li
Journal:  Traffic       Date:  2012-05-28       Impact factor: 6.215

Review 4.  DNA variations in oculocutaneous albinism: an updated mutation list and current outstanding issues in molecular diagnostics.

Authors:  Dimitre R Simeonov; Xinjing Wang; Chen Wang; Yuri Sergeev; Monika Dolinska; Matthew Bower; Roxanne Fischer; David Winer; Genia Dubrovsky; Joan Z Balog; Marjan Huizing; Rachel Hart; Wadih M Zein; William A Gahl; Brian P Brooks; David R Adams
Journal:  Hum Mutat       Date:  2013-04-30       Impact factor: 4.878

5.  Identification of a Homozygous Missense Mutation in the TYR Gene in a Chinese Family with OCA1.

Authors:  Yan Wang; Yi-Fan Zhou; Na Shen; Yao-Wu Zhu; Kun Tan; Xiong Wang
Journal:  Curr Med Sci       Date:  2018-10-20

6.  Mutation spectrum of the TYR and SLC45A2 genes in patients with oculocutaneous albinism.

Authors:  Jung Min Ko; Jung-Ah Yang; Seon-Yong Jeong; Hyon-Ju Kim
Journal:  Mol Med Rep       Date:  2012-01-25       Impact factor: 2.952

7.  Complex interactions of Tyrp1 in the eye.

Authors:  Hong Lu; Liyuan Li; Edmond R Watson; Robert W Williams; Eldon E Geisert; Monica M Jablonski; Lu Lu
Journal:  Mol Vis       Date:  2011-09-22       Impact factor: 2.367

8.  Evident hypopigmentation without other ocular deficits in Dutch patients with oculocutaneous albinism type 4.

Authors:  C C Kruijt; N E Schalij-Delfos; G C de Wit; R J Florijn; M M van Genderen
Journal:  Sci Rep       Date:  2021-06-02       Impact factor: 4.379

9.  SLC45A2 protein stability and regulation of melanosome pH determine melanocyte pigmentation.

Authors:  Linh Le; Iliana E Escobar; Tina Ho; Ariel J Lefkovith; Emily Latteri; Kirk D Haltaufderhyde; Megan K Dennis; Lynn Plowright; Elena V Sviderskaya; Dorothy C Bennett; Elena Oancea; Michael S Marks
Journal:  Mol Biol Cell       Date:  2020-09-23       Impact factor: 4.138

10.  Hermansky-Pudlak syndrome: Mutation update.

Authors:  Marjan Huizing; May C V Malicdan; Jennifer A Wang; Hadass Pri-Chen; Richard A Hess; Roxanne Fischer; Kevin J O'Brien; Melissa A Merideth; William A Gahl; Bernadette R Gochuico
Journal:  Hum Mutat       Date:  2020-01-23       Impact factor: 4.700

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