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Biallelic WRN Mutations in Newly Identified Japanese Werner Syndrome Patients
Author(s) -
Yoshiro Maezawa,
Hisaya Kato,
Minoru Takemoto,
Aki Watanabe,
Masaya Koshizaka,
Takahiro Ishikawa,
Forough Sargolzaeiaval,
Masafumi Kuzuya,
Hiroshi Wakabayashi,
Takashi Kusaka,
Koutaro Yokote,
Junko Oshima
Publication year - 2018
Publication title -
molecular syndromology
Language(s) - English
Resource type - Journals
SCImago Journal Rank - 0.609
H-Index - 36
eISSN - 1661-8777
pISSN - 1661-8769
DOI - 10.1159/000489055
Subject(s) - werner syndrome , compound heterozygosity , genetics , mutation , gene , medicine , helicase , biology , microbiology and biotechnology , rna
Werner syndrome (WS) is a rare autosomal recessive disorder characterized by systemic accelerated aging. It is caused by pathogenic variants of the WRN gene that encodes a nuclear helicase. In this report, we describe 4 newly identified WS cases among those referred to the Japanese Werner Consortium, Chiba University, Japan. All 4 cases were compound heterozygotes of the Japanese founder mutation, c.3139-1G>C, and a novel null pathogenic variant, c.1587G>A, c.2448+1G>A, or c.3233+1G>T, or an amino acid substitution variant, c.1720G>A, p.Gly574Arg. These 3 null pathogenic variants were not previously described. The p. Gly574Arg was previously reported in a European patient, and the identification of the second p. Gly574Arg case, with classical WS features, further confirmed the pathogenic nature of this variant. For the case with c.3233+1G>T, we determined the phase of 2 disease-causing mutations and demonstrated that they are on different chromosomes. This assay would be particularly important for those cases with ambiguous clinical diagnosis.

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