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Novel HPS6 mutations identified by whole-exome sequencing in two Japanese sisters with suspected ocular albinism.
- Source :
-
Journal of human genetics [J Hum Genet] 2016 Sep; Vol. 61 (9), pp. 839-42. Date of Electronic Publication: 2016 May 26. - Publication Year :
- 2016
-
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.
- Subjects :
- Alleles
Child, Preschool
Exome
Female
Fluorescein Angiography
Genes, Recessive
Genotype
High-Throughput Nucleotide Sequencing
Humans
Japan
Pedigree
Phenotype
Tomography, Optical Coherence
Albinism, Ocular diagnosis
Albinism, Ocular genetics
Intracellular Signaling Peptides and Proteins genetics
Mutation
Siblings
Subjects
Details
- Language :
- English
- ISSN :
- 1435-232X
- Volume :
- 61
- Issue :
- 9
- Database :
- MEDLINE
- Journal :
- Journal of human genetics
- Publication Type :
- Academic Journal
- Accession number :
- 27225848
- Full Text :
- https://doi.org/10.1038/jhg.2016.56