Literature DB >> 24026507

A large deletion of the AVPR2 gene causing severe nephrogenic diabetes insipidus in a Turkish family.

Emel Saglar1, Ferhat Deniz, Beril Erdem, Tugce Karaduman, Arif Yönem, Eylem Cagiltay, Hatice Mergen.   

Abstract

X-linked nephrogenic diabetes insipidus (NDI) is a rare hereditary disease caused by mutations in arginine vasopressin type 2 receptor (AVPR2) and characterized by the production of large amounts of urine and an inability to concentrate urine in response to the antidiuretic hormone vasopressin. We have identified a novel 388 bp deletion starting in intron 1 and ending in exon 2 in the AVPR2 gene in a patient with NDI and in his family. We have revealed that this mutation is a de novo mutation for the mother of the proband patient. Prospective clinical data were collected for all family members. The water deprivation test confirmed the diagnosis of diabetes insipidus. The patient has severe symptoms like deep polyuria nocturia, polydipsia, and fatigue. He was given arginine vasopressin treatment while he was a child. However, he could not get well due to his nephrogenic type of illness. Both of his nephews have the same complains in addition to failure to grow. We have sequenced all exons and intron-exon boundaries of the AVPR2 gene of all family members. The analyses of bioinformatics and comparative genomics of the deletion were done via considering the DNA level damage. AVPR2 gene mutation results in the absence of the three transmembrane domains, two extracellular domains, and one cytoplasmic domain. Three-dimensional protein structure prediction was shown. We concluded that X-linked NDI and severity of illness in this family is caused by a novel 388 bp deletion in the AVPR2 gene that is predicted to truncate the receptor protein, and also this deletion may lead to dysfunctioning in protein activity and inefficient or inadequate binding abilities.

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Year:  2013        PMID: 24026507     DOI: 10.1007/s12020-013-0043-7

Source DB:  PubMed          Journal:  Endocrine        ISSN: 1355-008X            Impact factor:   3.633


  18 in total

1.  Mutations in the AVPR2, AVP-NPII, and AQP2 genes in Turkish patients with diabetes insipidus.

Authors:  Duygu Duzenli; Emel Saglar; Ferhat Deniz; Omer Azal; Beril Erdem; Hatice Mergen
Journal:  Endocrine       Date:  2012-05-29       Impact factor: 3.633

2.  Protein structure prediction on the Web: a case study using the Phyre server.

Authors:  Lawrence A Kelley; Michael J E Sternberg
Journal:  Nat Protoc       Date:  2009       Impact factor: 13.491

Review 3.  Two new large deletions of the AVPR2 gene causing nephrogenic diabetes insipidus and a review of previously published deletions.

Authors:  Laura Anesi; Paola de Gemmis; Daniela Galla; Uros Hladnik
Journal:  Nephrol Dial Transplant       Date:  2012-08-09       Impact factor: 5.992

Review 4.  The evolutionary origin of the vasopressin/V2-type receptor/aquaporin axis and the urine-concentrating mechanism.

Authors:  Kristian Vinter Juul
Journal:  Endocrine       Date:  2012-02-29       Impact factor: 3.633

5.  Identification of mutations in the arginine vasopressin receptor 2 gene causing nephrogenic diabetes insipidus in Chinese patients.

Authors:  Chia-Hsiang Chen; Wen-Yu Chen; Hui-Lin Liu; Tze-Tze Liu; Ann-Ping Tsou; Ching-Yuang Lin; Ting Chao; Yu Qi; Kwang-Jen Hsiao
Journal:  J Hum Genet       Date:  2002       Impact factor: 3.172

Review 6.  Molecular biology of hereditary diabetes insipidus.

Authors:  T Mary Fujiwara; Daniel G Bichet
Journal:  J Am Soc Nephrol       Date:  2005-08-10       Impact factor: 10.121

Review 7.  Diabetes insipidus: diagnosis and treatment of a complex disease.

Authors:  Amgad N Makaryus; Samy I McFarlane
Journal:  Cleve Clin J Med       Date:  2006-01       Impact factor: 2.321

8.  Analysis of a novel AVPR2 mutation in a family with nephrogenic diabetes insipidus.

Authors:  Sung-Dae Moon; Ju-Hee Kim; Joo-Yun Shim; Dong-Jun Lim; Bong-Yun Cha; Je-Ho Han
Journal:  Int J Clin Exp Med       Date:  2010-11-30

9.  Novel mutation in the AVPR2 gene in a Danish male with nephrogenic diabetes insipidus caused by ER retention and subsequent lysosomal degradation of the mutant receptor.

Authors:  Lene N Nejsum; Tomas M Christensen; Joris H Robben; Graeme Milligan; Peter M T Deen; Daniel G Bichet; Klaus Levin
Journal:  NDT Plus       Date:  2011-03-02

10.  Immunological profile in a family with nephrogenic diabetes insipidus with a novel 11 kb deletion in AVPR2 and ARHGAP4 genes.

Authors:  Masaya Fujimoto; Kohsuke Imai; Kenji Hirata; Reiichi Kashiwagi; Yoichi Morinishi; Katsuhiko Kitazawa; Sei Sasaki; Tadao Arinami; Shigeaki Nonoyama; Emiko Noguchi
Journal:  BMC Med Genet       Date:  2008-05-20       Impact factor: 2.103

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  2 in total

1.  A novel AVPR2 missense mutation in an Asian family with inherited nephrogenic diabetes insipidus: A case report.

Authors:  Min Zhang; Qin Yu; Chen Chen; Jian Han; Bin Cheng; Dean Tian
Journal:  Medicine (Baltimore)       Date:  2019-04       Impact factor: 1.817

2.  A Novel Mutation in the AVPR2 Gene Causing Congenital Nephrogenic Diabetes Insipidus

Authors:  Aslı Çelebi Tayfur; Tuğçe Karaduman; Merve Özcan Türkmen; Dilara Şahin; Aysun Çaltık Yılmaz; Bahar Büyükkaragöz; Ayşe Derya Buluş; Hatice Mergen
Journal:  J Clin Res Pediatr Endocrinol       Date:  2018-07-11
  2 in total

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