Literature DB >> 31953843

Next-generation sequencing through multi-gene panel testing for diagnosis of hereditary ichthyosis in Chinese.

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.
© 2020 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.

Entities:  

Keywords:  Chinese; congenital ichthyosis; mutation; next-generation sequence

Mesh:

Substances:

Year:  2020        PMID: 31953843     DOI: 10.1111/cge.13704

Source DB:  PubMed          Journal:  Clin Genet        ISSN: 0009-9163            Impact factor:   4.438


  4 in total

1.  Compound Heterozygous Mutations in TGM1 Causing a Severe Form of Lamellar Ichthyosis: A Case Report.

Authors:  Jing Zeng; Baihui Shan; Lu Guo; Sha Lv; Fuqiu Li
Journal:  Pharmgenomics Pers Med       Date:  2022-06-07

Review 2.  Current Strategies for the Gene Therapy of Autosomal Recessive Congenital Ichthyosis and Other Types of Inherited Ichthyosis.

Authors:  Daria S Chulpanova; Alisa A Shaimardanova; Aleksei S Ponomarev; Somaia Elsheikh; Albert A Rizvanov; Valeriya V Solovyeva
Journal:  Int J Mol Sci       Date:  2022-02-24       Impact factor: 5.923

3.  Case Report: Chanarin-Dorfman Syndrome: A Novel Homozygous Mutation in ABHD5 Gene in a Chinese Case and Genotype-Phenotype Correlation Analysis.

Authors:  Bo Liang; He Huang; Jiaxiang Zhang; Gang Chen; Xiangsheng Kong; Mengting Zhu; Peiguang Wang; Lili Tang
Journal:  Front Genet       Date:  2022-03-28       Impact factor: 4.599

4.  Genomic basis for skin phenotype and cold adaptation in the extinct Steller's sea cow.

Authors:  Diana Le Duc; Akhil Velluva; Molly Cassatt-Johnstone; Remi-Andre Olsen; Sina Baleka; Chen-Ching Lin; Johannes R Lemke; John R Southon; Alexander Burdin; Ming-Shan Wang; Sonja Grunewald; Wilfried Rosendahl; Ulrich Joger; Sereina Rutschmann; Thomas B Hildebrandt; Guido Fritsch; James A Estes; Janet Kelso; Love Dalén; Michael Hofreiter; Beth Shapiro; Torsten Schöneberg
Journal:  Sci Adv       Date:  2022-02-04       Impact factor: 14.136

  4 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.