Literature DB >> 17081814

Phenotypic variability of a 4q34-->qter inherited deletion: MRKH syndrome in the daughter, cardiac defect and Fallopian tube cancer in the mother.

Claude Bendavid1, Laurent Pasquier, Tanguy Watrin, Karine Morcel, Josette Lucas, Isabelle Gicquel, Christèle Dubourg, Catherine Henry, Véronique David, Sylvie Odent, Jean Levêque, Isabelle Pellerin, Daniel Guerrier.   

Abstract

Terminal deletions of the long arm of chromosome 4 are associated with a recognizable phenotype consisting of dysmorphic facial features, cleft palate, upper and lower limb malformations, cardiac defects and growth and mental retardation. Here we report on two female patients, a mother and her daughter, carrying the same 4q34-->qter deletion but presenting with a different phenotype. The mother's presentation is consistent with previous findings in patients with terminal deletions of the long arm of chromosome 4. However, she presented at the age of 54years with bilateral serous carcinoma of the Fallopian tubes, a rare gynaecologic cancer that might be attributed to the haploinsufficiency of the tumor suppressor gene FAT. The daughter presented isolated congenital aplasia of the uterus and vagina, the prime feature of the MRKH syndrome. This has not been described before in association with a 46,XX,del(4)(q34qter).

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Year:  2006        PMID: 17081814     DOI: 10.1016/j.ejmg.2006.09.003

Source DB:  PubMed          Journal:  Eur J Med Genet        ISSN: 1769-7212            Impact factor:   2.708


  11 in total

1.  4q34.1-q35.2 deletion in a boy with phenotype resembling 22q11.2 deletion syndrome.

Authors:  Goran Cuturilo; Björn Menten; Aleksandar Krstic; Danijela Drakulic; Ida Jovanovic; Vojislav Parezanovic; Milena Stevanovic
Journal:  Eur J Pediatr       Date:  2011-07-22       Impact factor: 3.183

2.  Clinical utility gene card for: Mayer-Rokitansky-Küster-Hauser syndrome.

Authors:  Karine Morcel; Bruno Dallapiccola; Laurent Pasquier; Tanguy Watrin; Laura Bernardini; Daniel Guerrier
Journal:  Eur J Hum Genet       Date:  2011-09-07       Impact factor: 4.246

3.  Developmental profiles of the murine palatal methylome.

Authors:  Ratnam S Seelan; Savitri N Appana; Partha Mukhopadhyay; Dennis R Warner; Guy N Brock; M Michele Pisano; Robert M Greene
Journal:  Birth Defects Res A Clin Mol Teratol       Date:  2013-04-03

4.  Genetic analyses in a variant of Mayer-Rokitansky-Kuster-Hauser syndrome (MURCS association).

Authors:  Gerda Hofstetter; Nicole Concin; Christian Marth; Tuula Rinne; Martin Erdel; Andreas Janecke
Journal:  Wien Klin Wochenschr       Date:  2008       Impact factor: 1.704

5.  Utero-vaginal aplasia (Mayer-Rokitansky-Küster-Hauser syndrome) associated with deletions in known DiGeorge or DiGeorge-like loci.

Authors:  Karine Morcel; Tanguy Watrin; Laurent Pasquier; Lucie Rochard; Cédric Le Caignec; Christèle Dubourg; Philippe Loget; Bernard-Jean Paniel; Sylvie Odent; Véronique David; Isabelle Pellerin; Claude Bendavid; Daniel Guerrier
Journal:  Orphanet J Rare Dis       Date:  2011-03-15       Impact factor: 4.123

6.  Evaluation of SHOX copy number variations in patients with Müllerian aplasia.

Authors:  Maria Sandbacka; Mervi Halttunen; Varpu Jokimaa; Kristiina Aittomäki; Hannele Laivuori
Journal:  Orphanet J Rare Dis       Date:  2011-08-02       Impact factor: 4.123

Review 7.  Mayer-Rokitansky-Küster-Hauser (MRKH) syndrome.

Authors:  Karine Morcel; Laure Camborieux; Daniel Guerrier
Journal:  Orphanet J Rare Dis       Date:  2007-03-14       Impact factor: 4.123

8.  Recurrent microdeletion at 17q12 as a cause of Mayer-Rokitansky-Kuster-Hauser (MRKH) syndrome: two case reports.

Authors:  Laura Bernardini; Stefania Gimelli; Cristina Gervasini; Massimo Carella; Anwar Baban; Giada Frontino; Giancarlo Barbano; Maria Teresa Divizia; Luigi Fedele; Antonio Novelli; Frédérique Béna; Faustina Lalatta; Monica Miozzo; Bruno Dallapiccola
Journal:  Orphanet J Rare Dis       Date:  2009-11-04       Impact factor: 4.123

9.  Frequent loss of heterozygosity and altered expression of the candidate tumor suppressor gene 'FAT' in human astrocytic tumors.

Authors:  Kunzang Chosdol; Anjan Misra; Sachin Puri; Tapasya Srivastava; Parthaprasad Chattopadhyay; Chitra Sarkar; Ashok K Mahapatra; Subrata Sinha
Journal:  BMC Cancer       Date:  2009-01-07       Impact factor: 4.430

Review 10.  Studying Müllerian duct anomalies - from cataloguing phenotypes to discovering causation.

Authors:  Laura Santana González; Mara Artibani; Ahmed Ashour Ahmed
Journal:  Dis Model Mech       Date:  2021-06-23       Impact factor: 5.758

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