| Literature DB >> 31953843 |
Ruhong Cheng1,2, Jianying Liang1,2, Yue Li1,2, Jia Zhang1,2, Cheng Ni1,2, Hong Yu1,2, Xiangsheng Kong3, Ming Li1,2, Zhirong Yao1,2.
Abstract
Inherited ichthyoses are a heterogeneous group of rare disorders related to over 40 genes. To identify underlying molecular causes in inherited ichthyosis among Chinese and to correlate genotype and phenotype, 35 probands clinically diagnosed inherited ichthyosis, except ichthyosis vulgaris and X-linked ichthyosis, were included in our study. Molecular analysis was performed using next-generation sequencing (NGS) through multi-gene panel testing targeting all ichthyosis-related genes. Genetic variants causative for the ichthyosis were identified in 32 of 35 investigated patients. In all, 43 causative mutations across 12 genes were disclosed, including 16 novel variants. Thirteen keratinopathic ichthyosis, fourteen autosomal recessive congenital ichthyosis (ARCI) including one caused by mutations in SDR9C7, and five syndromic ichthyoses were confirmed. Four probands, with presumptive ARCI, turned out to be keratinopathic ichthyosis (2), neutral lipid storage disease (1), and Sjogren-Larsson syndrome (1), respectively. Next-generation technology has been demonstrated to be an effective tool in diagnosing inherited ichthyosis constituting a diverse group of cornification disorders. Our study further expands mutation spectrum and clinical phenotype associated with inherited ichthyosis in Chinese.Entities:
Keywords: Chinese; congenital ichthyosis; mutation; next-generation sequence
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Year: 2020 PMID: 31953843 DOI: 10.1111/cge.13704
Source DB: PubMed Journal: Clin Genet ISSN: 0009-9163 Impact factor: 4.438