The first Hermansky-Pudlak syndrome type 9 patient with two novel variants in Chinese population.
Teng LiuYefeng YuanDayong BaiXingfeng YaoTianjiao ZhangQiaorong HuangZhan QiLin YangXiumin YangWei LiAihua WeiPublished in: The Journal of dermatology (2021)
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.
Keyphrases
- case report
- end stage renal disease
- copy number
- newly diagnosed
- genome wide
- ejection fraction
- chronic kidney disease
- peritoneal dialysis
- prognostic factors
- physical activity
- autism spectrum disorder
- multiple sclerosis
- high throughput
- single cell
- white matter
- blood brain barrier
- brain injury
- intellectual disability
- duchenne muscular dystrophy
- functional connectivity
- muscular dystrophy