Literature DB >> 31424080

Whole-exome sequencing identifies a GREB1L variant in a three-generation family with Müllerian and renal agenesis: a novel candidate gene in Mayer-Rokitansky-Küster-Hauser (MRKH) syndrome. A case report.

Morten K Herlin1,2, Vang Q Le3, Allan T Højland1,4, Anja Ernst3, Henrik Okkels3, Astrid C Petersen5, Michael B Petersen1,4, Inge S Pedersen3,4.   

Abstract

The aetiology of Mayer-Rokitansky-Küster-Hauser (MRKH) syndrome, characterized by uterovaginal agenesis in 46,XX women, remains poorly understood. Since familial occurrences are rare, genetic findings reported so far only apply to a minority of mainly sporadic cases and most studies have not included other family members enabling segregation analysis. Herein, we report on the investigation of a unique three-generation family of two female cousins with MRKH syndrome and unilateral renal agenesis (RA) and two deceased male relatives with RA. We performed whole-exome sequencing (WES) in eight family members leading to the identification of a novel pathogenic (CADD = 33) c.705G>T missense variant in GREB1L, a gene recently identified as a novel cause of RA. Previous reports include several cases of female fetuses with bilateral RA and uterus agenesis, which support GREB1L as an important gene in both kidney and female genital tract development. The pedigree is compatible with autosomal dominant inheritance with incomplete penetrance following a parent-origin-specific manner, which could be due to imprinting. To our knowledge, this is the first investigation of a larger MRKH syndrome pedigree using WES, and we suggest GREB1L as a novel and promising candidate gene in the aetiology of MRKH syndrome.
© The Author(s) 2019. Published by Oxford University Press on behalf of the European Society of Human Reproduction and Embryology. All rights reserved. For permissions, please e-mail: journals.permissions@oup.com.

Entities:  

Keywords:  Mayer–Rokitansky–Küster–Hauser syndrome/Müllerian aplasia/renal agenesis/CAKUT/GREB1L/genetics/whole exome sequencing/penetrance/genomic imprinting

Mesh:

Substances:

Year:  2019        PMID: 31424080     DOI: 10.1093/humrep/dez126

Source DB:  PubMed          Journal:  Hum Reprod        ISSN: 0268-1161            Impact factor:   6.918


  10 in total

1.  A genotype-first analysis in a cohort of Mullerian anomaly.

Authors:  Weijie Tian; Na Chen; Yang Ye; Congcong Ma; Chenglu Qin; Yuchen Niu; L Xiaoxin; Lina Zhao; Hengqiang Zhao; Ze Liang; Shuang Song; Yuan Wang; Zefu Chen; Jiachen Lin; Zihui Yan; Jiali Duan; Sen Zhao; Terry Jianguo Zhang; Guixing Qiu; Zhihong Wu; Nan Wu; Lan Zhu
Journal:  J Hum Genet       Date:  2022-01-13       Impact factor: 3.172

2.  Precocious puberty or growth hormone deficiency as initial presentation in Mayer-Rokitansky-kuster-Hauser syndrome: a clinical report of 5 cases.

Authors:  Zhuanzhuan Ai; Xiaoyun Zhu; Hong Chen; Ruimin Chen
Journal:  BMC Pediatr       Date:  2022-07-14       Impact factor: 2.567

3.  Autosomal Dominantly Inherited GREB1L Variants in Individuals with Profound Sensorineural Hearing Impairment.

Authors:  Isabelle Schrauwen; Khurram Liaqat; Isabelle Schatteman; Thashi Bharadwaj; Abdul Nasir; Anushree Acharya; Wasim Ahmad; Guy Van Camp; Suzanne M Leal
Journal:  Genes (Basel)       Date:  2020-06-23       Impact factor: 4.096

4.  Protein-protein interaction network analysis applied to DNA copy number profiling suggests new perspectives on the aetiology of Mayer-Rokitansky-Küster-Hauser syndrome.

Authors:  Paola Pontecorvi; Laura Bernardini; Anna Capalbo; Simona Ceccarelli; Francesca Megiorni; Enrica Vescarelli; Irene Bottillo; Nicoletta Preziosi; Maria Fabbretti; Giorgia Perniola; Pierluigi Benedetti Panici; Antonio Pizzuti; Paola Grammatico; Cinzia Marchese
Journal:  Sci Rep       Date:  2021-01-11       Impact factor: 4.379

Review 5.  The Embryological Landscape of Mayer-Rokitansky-Kuster-Hauser Syndrome: Genetics and Environmental Factors.

Authors:  Isaac Kyei-Barffour; Miranda Margetts; Alla Vash-Margita; Emanuele Pelosi
Journal:  Yale J Biol Med       Date:  2021-12-29

6.  Variants in genes related to development of the urinary system are associated with Mayer-Rokitansky-Küster-Hauser syndrome.

Authors:  Chunfang Chu; Lin Li; Shenghui Li; Qi Zhou; Ping Zheng; Yu-Di Zhang; Ai-Hong Duan; Dan Lu; Yu-Mei Wu
Journal:  Hum Genomics       Date:  2022-03-31       Impact factor: 4.639

Review 7.  Identification of Genetic Causes in Mayer-Rokitansky-Küster-Hauser (MRKH) Syndrome: A Systematic Review of the Literature.

Authors:  Varvara Ermioni Triantafyllidi; Despoina Mavrogianni; Andreas Kalampalikis; Michael Litos; Stella Roidi; Lina Michala
Journal:  Children (Basel)       Date:  2022-06-27

8.  Genome Sequencing and Transcriptome Profiling in Twins Discordant for Mayer-Rokitansky-Küster-Hauser Syndrome.

Authors:  Rebecca Buchert; Elisabeth Schenk; Thomas Hentrich; Nico Weber; Katharina Rall; Marc Sturm; Oliver Kohlbacher; André Koch; Olaf Riess; Sara Y Brucker; Julia M Schulze-Hentrich
Journal:  J Clin Med       Date:  2022-09-23       Impact factor: 4.964

9.  Altered Expression of Candidate Genes in Mayer-Rokitansky-Küster-Hauser Syndrome May Influence Vaginal Keratinocytes Biology: A Focus on Protein Kinase X.

Authors:  Paola Pontecorvi; Francesca Megiorni; Simona Camero; Simona Ceccarelli; Laura Bernardini; Anna Capalbo; Eleni Anastasiadou; Giulia Gerini; Elena Messina; Giorgia Perniola; Pierluigi Benedetti Panici; Paola Grammatico; Antonio Pizzuti; Cinzia Marchese
Journal:  Biology (Basel)       Date:  2021-05-21

Review 10.  Mayer-Rokitansky-Küster-Hauser (MRKH) syndrome: a comprehensive update.

Authors:  Morten Krogh Herlin; Michael Bjørn Petersen; Mats Brännström
Journal:  Orphanet J Rare Dis       Date:  2020-08-20       Impact factor: 4.123

  10 in total

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