| Literature DB >> 27225848 |
Daisuke Miyamichi1, Miki Asahina2, Junya Nakajima3,4, Miho Sato1, Katsuhiro Hosono1, Takahito Nomura1, Takashi Negishi5, Noriko Miyake4, Yoshihiro Hotta1, Tsutomu Ogata2, Naomichi Matsumoto4.
Abstract
Hermansky-Pudlak syndrome (HPS) is an autosomal recessive disorder characterized by oculocutaneous albinism, platelet dysfunction and ceroid deposition. We report suspected ocular albinism in two Japanese sisters, caused by mutations in the HPS6 (Hermansky-Pudlak syndrome 6) gene. Trio-based whole-exome sequencing (WES) identified novel compound heterozygous mutations in HPS6 (c.1898delC: mother origin and c.2038C>T: father origin) in the two sisters. To date, 10 associated mutations have been detected in HPS6. Although we detected no general manifestations, including platelet dysfunction, in the sisters, even in long-term follow-up, we established a diagnosis of HPS type 6 based on the HPS6 mutations and absence of dense bodies in the platelets, indicating that WES can identify cases of HPS type 6. To the best of our knowledge, this is the first report of HPS6 mutations in Japanese patients.Entities:
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Year: 2016 PMID: 27225848 DOI: 10.1038/jhg.2016.56
Source DB: PubMed Journal: J Hum Genet ISSN: 1434-5161 Impact factor: 3.172