Literature DB >> 31304537

LMO2 gene deletions significantly worsen the prognosis of Wilms' tumor development in patients with WAGR syndrome.

Andrey V Marakhonov1,2, Tatyana A Vasilyeva1, Anna A Voskresenskaya3, Natella V Sukhanova4, Vitaly V Kadyshev1, Sergey I Kutsev1,5, Rena A Zinchenko1,5.   

Abstract

WAGR syndrome (OMIM #194072) is a rare genetic disorder that consists of development of Wilms' tumor (nephroblastoma), aniridia, genitourinary anomalies and intellectual disability (mental retardation). It is associated with WAGR-region deletions in the 11p13 chromosome region. Our previous study of congenital aniridia patients revealed a noticeable number of aniridia patients with WAGR-region deletions but without Wilms' tumor in their medical history. We assessed the involvement of other neighboring genes from affected chromosome regions in the patients with and without Wilms' tumor. Reliable confidence was obtained for the LMO2 gene, which is significantly more often deleted in patients with nephroblastoma. Thus, our study presents genetic evidence that the development of Wilms tumors in WAGR syndrome patients should be attributed to the deletion of WT1 and LMO2 rather than WT1 only.
© The Author(s) 2019. Published by Oxford University Press. All rights reserved. For Permissions, please email: journals.permissions@oup.com.

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Year:  2019        PMID: 31304537     DOI: 10.1093/hmg/ddz168

Source DB:  PubMed          Journal:  Hum Mol Genet        ISSN: 0964-6906            Impact factor:   6.150


  6 in total

1.  Characterization of Associated Nonclassical Phenotypes in Patients with Deletion in the WAGR Region Identified by Chromosomal Microarray: New Insights and Literature Review.

Authors:  Vanessa Sodré de Souza; Gabriela Corassa Rodrigues da Cunha; Beatriz R Versiani; Claudiner Pereira de Oliveira; Maria Teresa Alves Silva Rosa; Silviene F de Oliveira; Patricia N Moretti; Juliana F Mazzeu; Aline Pic-Taylor
Journal:  Mol Syndromol       Date:  2022-02-11

2.  Epidemiology of Rare Hereditary Diseases in the European Part of Russia: Point and Cumulative Prevalence.

Authors:  Rena A Zinchenko; Eugeny K Ginter; Andrey V Marakhonov; Nika V Petrova; Vitaly V Kadyshev; Tatyana P Vasilyeva; Oksana U Alexandrova; Alexander V Polyakov; Sergey I Kutsev
Journal:  Front Genet       Date:  2021-08-30       Impact factor: 4.772

3.  Preferentially Paternal Origin of De Novo 11p13 Chromosome Deletions Revealed in Patients with Congenital Aniridia and WAGR Syndrome.

Authors:  Tatyana A Vasilyeva; Andrey V Marakhonov; Natella V Sukhanova; Sergey I Kutsev; Rena A Zinchenko
Journal:  Genes (Basel)       Date:  2020-07-17       Impact factor: 4.096

4.  Identification of a 6-month-old baby with a combination of WAGR and Potocki-Shaffer contiguous deletion syndromes by SNP array testing.

Authors:  Yan Meng; Jun Yang; Chan Tian; Jie Qiao
Journal:  Hereditas       Date:  2020-05-23       Impact factor: 3.271

5.  A sporadic case of congenital aniridia caused by pericentric inversion inv(11)(p13q14) associated with a 977 kb deletion in the 11p13 region.

Authors:  Tatyana A Vasilyeva; Andrey V Marakhonov; Marina E Minzhenkova; Zhanna G Markova; Nika V Petrova; Natella V Sukhanova; Philipp A Koshkin; Denis V Pyankov; Ilya V Kanivets; Sergey A Korostelev; Irina A Krynskaya; Nadezhda V Shilova; Sergey I Kutsev; Vitaly V Kadyshev; Rena A Zinchenko
Journal:  BMC Med Genomics       Date:  2020-09-18       Impact factor: 3.063

6.  Unusual Presentation in WAGR Syndrome: Expanding the Phenotypic and Genotypic Spectrum of the Diseases.

Authors:  Qiwei Wang; Xulin Zhang; Tingfeng Qin; Dongni Wang; Xiaoshan Lin; Yuanyuan Zhu; Haowen Tan; Lanqin Zhao; Jing Li; Zhuoling Lin; Haotian Lin; Weirong Chen
Journal:  Genes (Basel)       Date:  2022-08-12       Impact factor: 4.141

  6 in total

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