Literature DB >> 22572733

Detection of a large heterozygous deletion and a splicing defect in the CFTR transcripts from nasal swab of a Japanese case of cystic fibrosis.

Miyuki Nakakuki1, Kotoyo Fujiki, Akiko Yamamoto, Shigeru B H Ko, Lanjuan Yi, Mariko Ishiguro, Makoto Yamaguchi, Shiho Kondo, Shinsuke Maruyama, Kosuke Yanagimoto, Satoru Naruse, Hiroshi Ishiguro.   

Abstract

Cystic fibrosis (CF) is an autosomal recessive disease caused by mutations in CFTR (CF transmembrane conductance regulator). Although CF is the most common hereditary disease in Caucasians, it is rare in Asian populations. Common disease-causing mutations of CFTR in Caucasians are rarely identified in Japanese patients with CF. In the present study, CFTR transcripts from nasal swab were analyzed in a Japanese boy, in addition to conventional PCR and direct sequence of all exons, their boundaries and promoter region of the CFTR gene. The boy was diagnosed with CF by chronic respiratory infection and the elevated sweat chloride level. None of the disease-causing mutations of CFTR was detected by the conventional analysis. Cloning and sequence of the CFTR transcripts revealed a heterozygous deletion spanning exons 16, 17a and 17b. The deletion was confirmed by multiplex ligation-dependent probe amplification and the direct sequence of the junction fragment obtained from the genomic DNA by primer walking, which revealed the mutation c.2908+1085_3367+260del7201. We also identified a splicing defect: deletion/skipping of exon 1 in the CFTR transcript from the other allele. The analysis of CFTR transcripts from nasal swab is recommended in the genetic analysis of CF in Japanese.

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Year:  2012        PMID: 22572733     DOI: 10.1038/jhg.2012.46

Source DB:  PubMed          Journal:  J Hum Genet        ISSN: 1434-5161            Impact factor:   3.172


  6 in total

1.  Characterization of Δ(G970-T1122)-CFTR, the most frequent CFTR mutant identified in Japanese cystic fibrosis patients.

Authors:  Kanako Wakabayashi-Nakao; Yingchun Yu; Miyuki Nakakuki; Tzyh-Chang Hwang; Hiroshi Ishiguro; Yoshiro Sohma
Journal:  J Physiol Sci       Date:  2018-06-27       Impact factor: 2.781

2.  Functional characteristics of L1156F-CFTR associated with alcoholic chronic pancreatitis in Japanese.

Authors:  Shiho Kondo; Kotoyo Fujiki; Shigeru B H Ko; Akiko Yamamoto; Miyuki Nakakuki; Yasutomo Ito; Nikolay Shcheynikov; Motoji Kitagawa; Satoru Naruse; Hiroshi Ishiguro
Journal:  Am J Physiol Gastrointest Liver Physiol       Date:  2015-06-18       Impact factor: 4.052

3.  Screening for Regulatory Variants in 460 kb Encompassing the CFTR Locus in Cystic Fibrosis Patients.

Authors:  Jenny L Kerschner; Sujana Ghosh; Alekh Paranjapye; Wilmel R Cosme; Marie-Pierre Audrézet; Miyuki Nakakuki; Hiroshi Ishiguro; Claude Férec; Johanna Rommens; Ann Harris
Journal:  J Mol Diagn       Date:  2018-10-05       Impact factor: 5.568

4.  Genetic analysis and intracytoplasmic sperm injection outcomes of Chinese patients with congenital bilateral absence of vas deferens.

Authors:  Hongbo Cheng; Shenmin Yang; Qingxia Meng; Bo Zheng; Yidong Gu; Luyun Wang; Tao Song; Chunlu Xu; Gaigai Wang; Mutian Han; Liyan Shen; Jie Ding; Hong Li; Jun Ouyang
Journal:  J Assist Reprod Genet       Date:  2022-02-04       Impact factor: 3.412

5.  Diagnosis of cystic fibrosis in an adult Japanese male.

Authors:  Yoshitaka Tomoda; Sayuri Arai; Kentaro Kawaguchi; Shinji Nabeshima; Takeshi Orihashi; Yasuyuki Kihara; Ryoji Kouzuma; Kazutoyo Tanaka
Journal:  J Gen Fam Med       Date:  2018-01-15

6.  Case Report: Japanese Siblings of Cystic Fibrosis With a Novel Large Heterozygous Deletion in the CFTR Gene.

Authors:  Mayumi Kawase; Masato Ogawa; Takayuki Hoshina; Masumi Kojiro; Miyuki Nakakuki; Satoru Naruse; Hiroshi Ishiguro; Koichi Kusuhara
Journal:  Front Pediatr       Date:  2022-01-03       Impact factor: 3.418

  6 in total

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