| Literature DB >> 33543539 |
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.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