Literature DB >> 27225848

Novel HPS6 mutations identified by whole-exome sequencing in two Japanese sisters with suspected ocular albinism.

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.

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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


  20 in total

1.  Mutation of a new gene causes a unique form of Hermansky-Pudlak syndrome in a genetic isolate of central Puerto Rico.

Authors:  Y Anikster; M Huizing; J White; Y O Shevchenko; D L Fitzpatrick; J W Touchman; J G Compton; S J Bale; R T Swank; W A Gahl; J R Toro
Journal:  Nat Genet       Date:  2001-08       Impact factor: 38.330

2.  Albinism and Hermansky-Pudlak syndrome in Puerto Rico.

Authors:  C J Witkop; M Nuñez Babcock; G H Rao; F Gaudier; C G Summers; F Shanahan; K R Harmon; D Townsend; H O Sedano; R A King
Journal:  Bol Asoc Med P R       Date:  1990-08

3.  A new genetic isolate with a unique phenotype of syndromic oculocutaneous albinism: clinical, molecular, and cellular characteristics.

Authors:  Nira Schreyer-Shafir; Marjan Huizing; Yair Anikster; Ziva Nusinker; Idit Bejarano-Achache; Genia Maftzir; Luba Resnik; Amanda Helip-Wooley; Wendy Westbroek; Libe Gradstein; Ada Rosenmann; Anat Blumenfeld
Journal:  Hum Mutat       Date:  2006-11       Impact factor: 4.878

4.  A germline mutation in BLOC1S3/reduced pigmentation causes a novel variant of Hermansky-Pudlak syndrome (HPS8).

Authors:  Neil V Morgan; Shanaz Pasha; Colin A Johnson; John R Ainsworth; Robin A J Eady; Ban Dawood; Carole McKeown; Richard C Trembath; Jonathan Wilde; Steve P Watson; Eamonn R Maher
Journal:  Am J Hum Genet       Date:  2005-11-28       Impact factor: 11.025

5.  Hermansky-Pudlak syndrome is caused by mutations in HPS4, the human homolog of the mouse light-ear gene.

Authors:  Tamio Suzuki; Wei Li; Qing Zhang; Amna Karim; Edward K Novak; Elena V Sviderskaya; Simon P Hill; Dorothy C Bennett; Alex V Levin; H Karel Nieuwenhuis; Chin-To Fong; Claudio Castellan; Bianca Miterski; Richard T Swank; Richard A Spritz
Journal:  Nat Genet       Date:  2002-02-11       Impact factor: 38.330

6.  Platelet morphology in Parkinson's disease: an electron microscopic study.

Authors:  S A Factor; E Ortof; M P Dentinger; R Mankes; K D Barron
Journal:  J Neurol Sci       Date:  1994-03       Impact factor: 3.181

7.  Biallelic Mutations in Nuclear Pore Complex Subunit NUP107 Cause Early-Childhood-Onset Steroid-Resistant Nephrotic Syndrome.

Authors:  Noriko Miyake; Hiroyasu Tsukaguchi; Eriko Koshimizu; Akemi Shono; Satoko Matsunaga; Masaaki Shiina; Yasuhiro Mimura; Shintaro Imamura; Tomonori Hirose; Koji Okudela; Kandai Nozu; Yuko Akioka; Motoshi Hattori; Norishige Yoshikawa; Akiko Kitamura; Hae Il Cheong; Shoji Kagami; Michiaki Yamashita; Atsushi Fujita; Satoko Miyatake; Yoshinori Tsurusaki; Mitsuko Nakashima; Hirotomo Saitsu; Kenichi Ohashi; Naoko Imamoto; Akihide Ryo; Kazuhiro Ogata; Kazumoto Iijima; Naomichi Matsumoto
Journal:  Am J Hum Genet       Date:  2015-09-24       Impact factor: 11.025

8.  Organelle biogenesis: en BLOC exchange for RAB32 and RAB38.

Authors:  Michael S Marks
Journal:  Curr Biol       Date:  2012-11-20       Impact factor: 10.834

9.  Genotype-phenotype correlation and mutation spectrum in a large cohort of patients with inherited retinal dystrophy revealed by next-generation sequencing.

Authors:  Xiu-Feng Huang; Fang Huang; Kun-Chao Wu; Juan Wu; Jie Chen; Chi-Pui Pang; Fan Lu; Jia Qu; Zi-Bing Jin
Journal:  Genet Med       Date:  2014-11-06       Impact factor: 8.822

10.  Targeted exome sequencing identified novel USH2A mutations in Usher syndrome families.

Authors:  Xiu-Feng Huang; Ping Xiang; Jie Chen; Dong-Jun Xing; Na Huang; Qingjie Min; Feng Gu; Yi Tong; Chi-Pui Pang; Jia Qu; Zi-Bing Jin
Journal:  PLoS One       Date:  2013-05-30       Impact factor: 3.240

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  7 in total

1.  Identification of novel variants in ten patients with Hermansky-Pudlak syndrome by high-throughput sequencing.

Authors:  Jose María Bastida; Sara Morais; Veronica Palma-Barqueros; Rocio Benito; Nuria Bermejo; Mutlu Karkucak; Maria Trapero-Marugan; Natalia Bohdan; Mónica Pereira; Ana Marin-Quilez; Jorge Oliveira; Yusuf Yucel; Rosario Santos; Jose Padilla; Kamila Janusz; Catarina Lau; Marta Martin-Izquierdo; Eduarda Couto; Juan Francisco Ruiz-Pividal; Vicente Vicente; Jesus Maria Hernández-Rivas; Jose Ramon González-Porras; Maria Luisa Lozano; Margarida Lima; Jose Rivera
Journal:  Ann Med       Date:  2019-04-16       Impact factor: 4.709

2.  Clinical characteristics of a Japanese patient with Bardet-Biedl syndrome caused by BBS10 mutations.

Authors:  Kentaro Kurata; Katsuhiro Hosono; Akiko Hikoya; Akihiko Kato; Hirotomo Saitsu; Shinsei Minoshima; Tsutomu Ogata; Yoshihiro Hotta
Journal:  Jpn J Ophthalmol       Date:  2018-04-17       Impact factor: 2.447

3.  Severe bleeding with subclinical oculocutaneous albinism in a patient with a novel HPS6 missense variant.

Authors:  Chen G Han; Kevin J O'Brien; Lea M Coon; Julie A Majerus; Laryssa A Huryn; Sara G Haroutunian; Nagabhishek Moka; Wendy J Introne; Ellen Macnamara; William A Gahl; May Christine V Malicdan; Dong Chen; Koyamangalath Krishnan; Bernadette R Gochuico
Journal:  Am J Med Genet A       Date:  2018-10-04       Impact factor: 2.802

4.  A novel mutation causes Hermansky-Pudlak syndrome type 4 with pulmonary fibrosis in 2 siblings from China.

Authors:  Wenjuan Wu; Keqin Lin; Yanni Yang; ZhaoXing Dong; Tao Zhang; Wen Lei; Weimin Yang; Zhaoqing Yang
Journal:  Medicine (Baltimore)       Date:  2019-08       Impact factor: 1.817

Review 5.  Clinical Application of Genome and Exome Sequencing as a Diagnostic Tool for Pediatric Patients: a Scoping Review of the Literature.

Authors:  Hadley Stevens Smith; J Michael Swint; Seema R Lalani; Jose-Miguel Yamal; Marcia C de Oliveira Otto; Stephan Castellanos; Amy Taylor; Brendan H Lee; Heidi V Russell
Journal:  Genet Med       Date:  2018-05-14       Impact factor: 8.822

6.  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

7.  Two Novel Homozygous HPS6 Mutations (Double Mutant) Identified by Whole-Exome Sequencing in a Saudi Consanguineous Family Suspected for Oculocutaneous Albinism.

Authors:  Sajjad Karim; Samah Saharti; Nofe Alganmi; Zeenat Mirza; Ahmed Alfares; Shereen Turkistany; Manal Al-Attas; Hend Noureldin; Khadega Al Sakkaf; Heba Abusamra; Mohammed Al-Qahtani; Adel Abuzenadah
Journal:  Life (Basel)       Date:  2021-12-23
  7 in total

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