Literature DB >> 29702541

Novel Variations of FANCA Gene Provokes Fanconi Anemia: Molecular Diagnosis in a Special Chinese Family.

Niu Li1,2, Aiyun Song1, Lixia Ding1, Hua Zhu3, Guoqiang Li2, Yan Miao1, Jian Wang2, Benshang Li1, Jing Chen1.   

Abstract

Fanconi anemia (FA) is a rare autosomal recessive or X-linked disorder with highly variable clinical manifestations and an incidence of ∼1 to 5 in 1 million births. To date, 15 bona fide FA genes have been reported to be responsible for the known FA complementation groups and the FANCA gene accounts for almost 60%. In the present study, we report a special Chinese family, which has 2 children with classic FA characteristics. Via 2-step analysis of the whole-exome sequencing data and verification using multiplex ligation-dependent probe amplification test, one child was found to have a novel compound heterozygous mutation of a splicing variant (c.1471-1G>A) and a large intragenic deletion (exons 23-30 del) of the FANCA gene. The other child had the same splicing variant and another novel large deletion (exons 1-18 del) in the FANCA gene. Clone sequencing showed the c.1471-1G>A variant generate an altered transcript with 1 cryptic splice site in intron 15, resulting in a premature termination codon (p.Val490HisfsX6). This study not only shows the complexity of FA molecular diagnosis via comprehensively studying the FA pathogenic genes and the mutational spectrum, but also has significant reference value for the future molecular diagnosis of FA.

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Year:  2018        PMID: 29702541     DOI: 10.1097/MPH.0000000000001197

Source DB:  PubMed          Journal:  J Pediatr Hematol Oncol        ISSN: 1077-4114            Impact factor:   1.289


  1 in total

1.  Novel FANCA mutation in the first fully-diagnosed patient with Fanconi anemia in Polish population - case report.

Authors:  Anna Repczynska; Agata Pastorczak; Katarzyna Babol-Pokora; Jolanta Skalska-Sadowska; Malgorzata Drozniewska; Wojciech Mlynarski; Olga Haus
Journal:  Mol Cytogenet       Date:  2020-08-10       Impact factor: 2.009

  1 in total

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