Literature DB >> 33543539

The first Hermansky-Pudlak syndrome type 9 patient with two novel variants in Chinese population.

Teng Liu1,2, Yefeng Yuan3, Dayong Bai4, Xingfeng Yao5, Tianjiao Zhang1, Qiaorong Huang1, Zhan Qi3, Lin Yang6, Xiumin Yang1, Wei Li2,3, Aihua Wei1.   

Abstract

Hermansky-Pudlak syndrome 9 (HPS-9) is a recessive disorder caused by BLOC1S6 gene. There are only four variants identified from four HPS-9 patients so far. Here, we reported the first HPS-9 patient in a Chinese population. He had brownish-yellow hair, white skin, brown irises with visual acuity, photophobia and nystagmus. Two novel variants, c.148G>T (p.Glu50*) and c.351dupT (p.Ile118Tyrfs*10) in BLOC1S6 gene were identified by whole-exome sequencing (WES). Absence of platelet dense granules was found by whole-mount platelet electron microscopy and Western blotting assays showed the destabilized BLOC-1 subunits. He had recurrent bruising and was found to have abnormal brain waves by electroencephalogram, but did not develop thrombopenia, immunodeficiency or other symptoms reported in other HPS-9 patients. This is the first case report of BLOC-1 mutation in a Chinese population and our findings expand the mutational spectrum of HPS genes.
© 2021 Japanese Dermatological Association.

Entities:  

Keywords:  Hermansky-Pudlak syndrome type 9; albinism; biogenesis of lysosome-related organelles complex 1; pathological variant; whole-exome sequencing

Year:  2021        PMID: 33543539     DOI: 10.1111/1346-8138.15762

Source DB:  PubMed          Journal:  J Dermatol        ISSN: 0385-2407            Impact factor:   4.005


  2 in total

1.  A zinc transporter, transmembrane protein 163, is critical for the biogenesis of platelet dense granules.

Authors:  Yefeng Yuan; Teng Liu; Xiahe Huang; Yuanying Chen; Weilin Zhang; Ting Li; Lin Yang; Quan Chen; Yingchun Wang; Aihua Wei; Wei Li
Journal:  Blood       Date:  2021-04-01       Impact factor: 22.113

Review 2.  A Novel Likely Pathogenic Variant in the BLOC1S5 Gene Associated with Hermansky-Pudlak Syndrome Type 11 and an Overview of Human BLOC-1 Deficiencies.

Authors:  Doris Boeckelmann; Mira Wolter; Barbara Käsmann-Kellner; Udo Koehler; Lea Schieber-Nakamura; Barbara Zieger
Journal:  Cells       Date:  2021-10-01       Impact factor: 6.600

  2 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.