Literature DB >> 19668106

A deep intronic mutation in the SLC12A3 gene leads to Gitelman syndrome.

Kandai Nozu1, Kazumoto Iijima, Yoshimi Nozu, Ei Ikegami, Takehide Imai, Xue Jun Fu, Hiroshi Kaito, Koichi Nakanishi, Norishige Yoshikawa, Masafumi Matsuo.   

Abstract

Many mutations have been detected in the SLC12A3 gene of Gitelman syndrome (GS, OMIM 263800) patients. In previous studies, only one mutant allele was detected in approximately 20 to 41% of patients with GS; however, the exact reason for the nonidentification has not been established. In this study, we used RT-PCR using mRNA to investigate for the first time transcript abnormalities caused by deep intronic mutation. Direct sequencing analysis of leukocyte DNA identified one base insertion in exon 6 (c.818_819insG), but no mutation was detected in another allele. We analyzed RNA extracted from leukocytes and urine sediments and detected unknown sequence containing 238bp between exons 13 and 14. The genomic DNA analysis of intron 13 revealed a single-base substitution (c.1670-191C>T) that creates a new donor splice site within the intron resulting in the inclusion of a novel cryptic exon in mRNA. This is the first report of creation of a splice site by a deep intronic single-nucleotide change in GS and the first report to detect the onset mechanism in a patient with GS and missing mutation in one allele. This molecular onset mechanism may partly explain the poor success rate of mutation detection in both alleles of patients with GS.

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Year:  2009        PMID: 19668106     DOI: 10.1203/PDR.0b013e3181b9b4d3

Source DB:  PubMed          Journal:  Pediatr Res        ISSN: 0031-3998            Impact factor:   3.756


  16 in total

1.  Clinical utility gene card for: Gitelman syndrome.

Authors:  Nine Vam Knoers; Olivier Devuyst; Erik-Jan Kamsteeg
Journal:  Eur J Hum Genet       Date:  2011-02-23       Impact factor: 4.246

2.  Natural history of genetically proven autosomal recessive Alport syndrome.

Authors:  Masafumi Oka; Kandai Nozu; Hiroshi Kaito; Xue Jun Fu; Koichi Nakanishi; Yuya Hashimura; Naoya Morisada; Kunimasa Yan; Masafumi Matsuo; Norishige Yoshikawa; Igor Vorechovsky; Kazumoto Iijima
Journal:  Pediatr Nephrol       Date:  2014-03-15       Impact factor: 3.714

3.  Recurrent deep intronic mutations in the SLC12A3 gene responsible for Gitelman's syndrome.

Authors:  Yi-Fen Lo; Kandai Nozu; Kazumoto Iijima; Takahiro Morishita; Che-Chung Huang; Sung-Sen Yang; Huey-Kang Sytwu; Yu-Wei Fang; Min-Hua Tseng; Shih-Hua Lin
Journal:  Clin J Am Soc Nephrol       Date:  2010-11-04       Impact factor: 8.237

4.  X-linked Alport syndrome caused by splicing mutations in COL4A5.

Authors:  Kandai Nozu; Igor Vorechovsky; Hiroshi Kaito; Xue Jun Fu; Koichi Nakanishi; Yuya Hashimura; Fusako Hashimoto; Koichi Kamei; Shuichi Ito; Yoshitsugu Kaku; Toshiyuki Imasawa; Katsumi Ushijima; Junya Shimizu; Yoshio Makita; Takao Konomoto; Norishige Yoshikawa; Kazumoto Iijima
Journal:  Clin J Am Soc Nephrol       Date:  2014-09-02       Impact factor: 8.237

Review 5.  Deep intronic mutations and human disease.

Authors:  Rita Vaz-Drago; Noélia Custódio; Maria Carmo-Fonseca
Journal:  Hum Genet       Date:  2017-05-12       Impact factor: 4.132

Review 6.  Inherited and acquired disorders of magnesium homeostasis.

Authors:  Matthias Tilmann Florian Wolf
Journal:  Curr Opin Pediatr       Date:  2017-04       Impact factor: 2.856

7.  Cryptic exon activation in SLC12A3 in Gitelman syndrome.

Authors:  Kandai Nozu; Yoshimi Nozu; Keita Nakanishi; Takao Konomoto; Tomoko Horinouchi; Akemi Shono; Naoya Morisada; Shogo Minamikawa; Tomohiko Yamamura; Junya Fujimura; Koichi Nakanishi; Takeshi Ninchoji; Hiroshi Kaito; Ichiro Morioka; Mariko Taniguchi-Ikeda; Igor Vorechovsky; Kazumoto Iijima
Journal:  J Hum Genet       Date:  2016-10-27       Impact factor: 3.172

8.  Molecular assay for an intronic variant in NUP93 that causes steroid resistant nephrotic syndrome.

Authors:  Rini Rossanti; Akemi Shono; Kenichiro Miura; Motoshi Hattori; Tomohiko Yamamura; Keita Nakanishi; Shogo Minamikawa; Junya Fujimura; Tomoko Horinouchi; China Nagano; Nana Sakakibara; Hiroshi Kaito; Hiroaki Nagase; Naoya Morisada; Katsuhiko Asanuma; Masafumi Matsuo; Kandai Nozu; Kazumoto Iijima
Journal:  J Hum Genet       Date:  2019-04-23       Impact factor: 3.172

9.  Evaluation of Suspected Autosomal Alport Syndrome Synonymous Variants.

Authors:  Rini Rossanti; Tomoko Horinouchi; Tomohiko Yamamura; China Nagano; Nana Sakakibara; Shinya Ishiko; Yuya Aoto; Atsushi Kondo; Sadayuki Nagai; Eri Okada; Shingo Ishimori; Hiroaki Nagase; Satoshi Matsui; Keiichi Tamagaki; Yoshifumi Ubara; Masahiko Nagahama; Yuko Shima; Koichi Nakanishi; Takeshi Ninchoji; Masafumi Matsuo; Kazumoto Iijima; Kandai Nozu
Journal:  Kidney360       Date:  2021-10-13

10.  Cerebral vasospasm after sub-arachnoid hemorrhage as a clinical predictor and phenotype for genetic association study.

Authors:  Hyungsuk Kim; Elizabeth Crago; Mirim Kim; Paula Sherwood; Yvette Conley; Samuel Poloyac; Mary Kerr
Journal:  Int J Stroke       Date:  2012-05-09       Impact factor: 5.266

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