Literature DB >> 19248183

2q31.2q32.3 deletion syndrome: report of an adult patient.

Paolo Prontera1, Laura Bernardini, Gabriela Stangoni, Anna Capalbo, Daniela Rogaia, Carmela Ardisia, Antonio Novelli, Bruno Dallapiccola, Emilio Donti.   

Abstract

A 36-year-old patient with a disorder characterized by severe mental retardation, behavioral problems, dysmorphic face, "muscular build," and hand/foot anomalies, is reported. Following a diagnosis of de novo pericentric inversion of chromosome 8 based on standard cytogenetic analysis, a subsequent 75 kb array-CGH investigation disclosed a deletion spanning for about 13.7 Mb in the 2q31.2q32.3 region. Whole painting of chromosome 8 established the intrachromosomal nature of the rearrangement and FISH analysis with locus-specific probes confirmed the deletion on the long arm of chromosome 2. The deleted region, clinical outcome, and medical history in this patient are mainly superimposable to those reported in a published 8-year-old boy, suggesting that this genomic segment is prone to rearrangements and its hemizygosity gives rise to a clinically recognizable syndrome. The role of some genes mapping in the deleted region and related with distinct disorders is discussed. Interestingly, deletion of MSTN gene, a negative regulator of muscle growth, was associated in our patient with a "muscular build," a feature which could be regarded as a handle for clinical recognition of this syndrome.

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Year:  2009        PMID: 19248183     DOI: 10.1002/ajmg.a.32688

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  9 in total

1.  Aicardi syndrome associated with autosomal genomic imbalance: coincidence or evidence for autosomal inheritance with sex-limited expression?

Authors:  P Prontera; A Bartocci; V Ottaviani; I Isidori; D Rogaia; C Ardisia; G Guercini; A Mencarelli; E Donti
Journal:  Mol Syndromol       Date:  2013-04-11

2.  Duplication at chromosome 2q31.1-q31.2 in a family presenting syndactyly and nystagmus.

Authors:  Jamal Ghoumid; Joris Andrieux; Bernard Sablonnière; Sylvie Odent; Nathalie Philippe; Xavier Zanlonghi; Pascale Saugier-Veber; Thomas Bardyn; Sylvie Manouvrier-Hanu; Muriel Holder-Espinasse
Journal:  Eur J Hum Genet       Date:  2011-06-08       Impact factor: 4.246

Review 3.  Role of titin in cardiomyopathy: from DNA variants to patient stratification.

Authors:  James S Ware; Stuart A Cook
Journal:  Nat Rev Cardiol       Date:  2017-12-14       Impact factor: 32.419

Review 4.  Molecular Therapies for Muscular Dystrophies.

Authors:  Ava Y Lin; Leo H Wang
Journal:  Curr Treat Options Neurol       Date:  2018-06-21       Impact factor: 3.598

5.  The use of array-CGH in a cohort of Greek children with developmental delay.

Authors:  Emmanouil Manolakos; Annalisa Vetro; Konstantinos Kefalas; Stamatia-Maria Rapti; Eirini Louizou; Antonios Garas; George Kitsos; Lefteris Vasileiadis; Panagiota Tsoplou; Makarios Eleftheriades; Panagiotis Peitsidis; Sandro Orru; Thomas Liehr; Michael B Petersen; Loretta Thomaidis
Journal:  Mol Cytogenet       Date:  2010-11-09       Impact factor: 2.009

6.  A phase 1/2a follistatin gene therapy trial for becker muscular dystrophy.

Authors:  Jerry R Mendell; Zarife Sahenk; Vinod Malik; Ana M Gomez; Kevin M Flanigan; Linda P Lowes; Lindsay N Alfano; Katherine Berry; Eric Meadows; Sarah Lewis; Lyndsey Braun; Kim Shontz; Maria Rouhana; Kelly Reed Clark; Xiomara Q Rosales; Samiah Al-Zaidy; Alessandra Govoni; Louise R Rodino-Klapac; Mark J Hogan; Brian K Kaspar
Journal:  Mol Ther       Date:  2014-10-17       Impact factor: 11.454

7.  Integrated allelic, transcriptional, and phenomic dissection of the cardiac effects of titin truncations in health and disease.

Authors:  Angharad M Roberts; James S Ware; Daniel S Herman; Sebastian Schafer; John Baksi; Alexander G Bick; Rachel J Buchan; Roddy Walsh; Shibu John; Samuel Wilkinson; Francesco Mazzarotto; Leanne E Felkin; Sungsam Gong; Jacqueline A L MacArthur; Fiona Cunningham; Jason Flannick; Stacey B Gabriel; David M Altshuler; Peter S Macdonald; Matthias Heinig; Anne M Keogh; Christopher S Hayward; Nicholas R Banner; Dudley J Pennell; Declan P O'Regan; Tan Ru San; Antonio de Marvao; Timothy J W Dawes; Ankur Gulati; Emma J Birks; Magdi H Yacoub; Michael Radke; Michael Gotthardt; James G Wilson; Christopher J O'Donnell; Sanjay K Prasad; Paul J R Barton; Diane Fatkin; Norbert Hubner; Jonathan G Seidman; Christine E Seidman; Stuart A Cook
Journal:  Sci Transl Med       Date:  2015-01-14       Impact factor: 17.956

8.  Critical region in 2q31.2q32.3 deletion syndrome: Report of two phenotypically distinct patients, one with an additional deletion in Alagille syndrome region.

Authors:  Susana Isabel Ferreira; Eunice Matoso; Margarida Venâncio; Jorge Saraiva; Joana B Melo; Isabel Marques Carreira
Journal:  Mol Cytogenet       Date:  2012-05-02       Impact factor: 2.009

9.  Case report: early aortic valve degeneration associated with interstitial deletion of chromosome 2-46,XX,del (2)(q31.3; q32.2).

Authors:  Raymond Pfister; Clara Knowles; Matthias Kirsch
Journal:  J Cardiothorac Surg       Date:  2020-04-21       Impact factor: 1.637

  9 in total

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