Literature DB >> 18321894

Higher incidence of linked malformations in siblings of Mayer-Rokitansky-Küster-Hauser-syndrome patients.

M Wottgen1, S Brucker, S P Renner, P L Strissel, R Strick, A Kellermann, D Wallwiener, M W Beckmann, P Oppelt.   

Abstract

BACKGROUND: Mayer-Rokitansky-Küster-Hauser (MRKH) syndrome is a malformation of the female genital tract (vaginal aplasia, rudimentary uterus, normal fallopian tubes and high ovaries). The incidence is one in 4000 female newborns. The aim of the present study was to record genital and associated malformations among siblings and relatives of MRKH patients in order to draw possible conclusions regarding the etiology of the syndrome: heredity (dominant versus recessive) or spontaneous malformation.
METHODS: Using a standardized questionnaire, affected MRKH patients were asked about other cases of MRKH and/or associated malformations among siblings and relatives.
RESULTS: No other cases of MRKH syndrome had occurred among the siblings or relatives of 73 MRKH patients; however, 13 associated malformations were recorded among a total of 103 siblings. Musculoskeletal malformations were markedly increased (3.27 times higher) in comparison with the prevalence of congenital malformations among newborns in the normal population.
CONCLUSIONS: This study shows that dominant inheritance cannot play a role in the etiology of MRKH syndrome, as no further cases of MRKH syndrome occurred among any of the siblings. The study provides support for the view that the syndrome has a multifactorial pathogenesis. Siblings/relatives of MRKH patients should be examined for associated musculoskeletal/urogenital malformations.

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Year:  2008        PMID: 18321894     DOI: 10.1093/humrep/den059

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


  6 in total

Review 1.  Malformation syndromes associated with disorders of sex development.

Authors:  John M Hutson; Sonia R Grover; Michele O'Connell; Samuel D Pennell
Journal:  Nat Rev Endocrinol       Date:  2014-06-10       Impact factor: 43.330

2.  A combination of transcriptome and methylation analyses reveals embryologically-relevant candidate genes in MRKH patients.

Authors:  Katharina Rall; Gianmaria Barresi; Michael Walter; Sven Poths; Karina Haebig; Karin Schaeferhoff; Birgitt Schoenfisch; Olaf Riess; Diethelm Wallwiener; Michael Bonin; Sara Brucker
Journal:  Orphanet J Rare Dis       Date:  2011-05-28       Impact factor: 4.123

3.  Genetic Syndromes and Genes Involved in the Development of the Female Reproductive Tract: A Possible Role for Gene Therapy.

Authors:  Mt Connell; Cm Owen; Jh Segars
Journal:  J Genet Syndr Gene Ther       Date:  2013

4.  Rare genital malformations in women's health research: sociodemographic, regional, and disease-related characteristics of patients with Mayer-Rokitansky-Küster-Hauser syndrome.

Authors:  Sara Yvonne Brucker; Leonie-Sophia Pösch; Joachim Graf; Alexander N Sokolov; Norbert Schaeffeler; Andrea Kronenthaler; Hanna Hiltner; Anke Wagner; Esther Ueding; Monika A Rieger; Dorit Schöller; Diana Stefanescu; Kristin Katharina Rall; Diethelm Wallwiener; Elisabeth Simoes
Journal:  BMC Womens Health       Date:  2020-06-29       Impact factor: 2.809

5.  An Association between EMX2 Variations and Mayer-Rokitansky-Küster-Hauser Syndrome: A Case-Control Study of Chinese Women.

Authors:  Haiping Li; Shi Liao; Guangnan Luo; Haixia Li; Shuai Wang; Zhimin Li; Xiping Luo
Journal:  J Healthc Eng       Date:  2022-04-13       Impact factor: 3.822

Review 6.  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
  6 in total

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