Literature DB >> 25691421

Partial MEF2C deletion in a Cypriot patient with severe intellectual disability and a jugular fossa malformation: review of the literature.

George A Tanteles1, Angelos Alexandrou, Paola Evangelidou, Marina Gavatha, Violetta Anastasiadou, Carolina Sismani.   

Abstract

Deletions or intragenic mutations involving the MEF2C gene on chromosome 5q14.3 have generally been associated with a relatively uniform phenotype characterized by severe developmental delay, absent speech, stereotypies, absent or limited gait abilities, lack of a typical facial gestalt and scarcity of major malformations. We report on a patient of Cypriot descent with a de novo, approximately 147 kb in size, partial MEF2C deletion removing exons 1 to 3. He had a history of severe intellectual disability with absent speech, poor eye contact, hand stereotypies and a wide-based gait. A broad-based, shallow jugular pit with an overlying vascular malformation was also present. Partial MEF2C deletions have only been reported in a very small number of patients and have on occasion been associated with relatively milder phenotypes. We present a patient of Cypriot descent with such a deletion and review previously published literature on partial MEF2C gene deletions postulating a key role of the first few exons in the pathogenesis of the disease.
© 2015 Wiley Periodicals, Inc.

Entities:  

Keywords:  Cyprus; MEF2C; array-CGH; intellectual disability; partial deletion

Mesh:

Substances:

Year:  2015        PMID: 25691421     DOI: 10.1002/ajmg.a.36945

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


  6 in total

Review 1.  Chromosomal Microarrays: Understanding Genetics of Neurodevelopmental Disorders and Congenital Anomalies.

Authors:  Jill A Rosenfeld; Ankita Patel
Journal:  J Pediatr Genet       Date:  2016-05-30

2.  Identification of a novel 15.5 kb SHOX deletion associated with marked intrafamilial phenotypic variability and analysis of its molecular origin.

Authors:  Angelos Alexandrou; Ioannis Papaevripidou; Kyriakos Tsangaras; Ioanna Alexandrou; Marios Tryfonidis; Violetta Christophidou-Anastasiadou; Eleni Zamba-Papanicolaou; George Koumbaris; Vassos Neocleous; Leonidas A Phylactou; Nicos Skordis; George A Tanteles; Carolina Sismani
Journal:  J Genet       Date:  2016-12       Impact factor: 1.166

Review 3.  Further Clinical Delineation of the MEF2C Haploinsufficiency Syndrome: Report on New Cases and Literature Review of Severe Neurodevelopmental Disorders Presenting with Seizures, Absent Speech, and Involuntary Movements.

Authors:  Irena Vrečar; Josie Innes; Elizabeth A Jones; Helen Kingston; William Reardon; Bronwyn Kerr; Jill Clayton-Smith; Sofia Douzgou
Journal:  J Pediatr Genet       Date:  2017-04-12

4.  Genotypes and Phenotypes of MEF2C Haploinsufficiency Syndrome: New Cases and Novel Point Mutations.

Authors:  Lin Wan; Xinting Liu; Linyan Hu; Huimin Chen; Yulin Sun; Zhichao Li; Zhenfang Wang; Zhi Lin; Liping Zou; Guang Yang
Journal:  Front Pediatr       Date:  2021-05-13       Impact factor: 3.418

Review 5.  Progress on the roles of MEF2C in neuropsychiatric diseases.

Authors:  Zhikun Zhang; Yongxiang Zhao
Journal:  Mol Brain       Date:  2022-01-06       Impact factor: 4.041

6.  The Cell Adhesion Molecule L1 Interacts with Methyl CpG Binding Protein 2 via Its Intracellular Domain.

Authors:  Gabriele Loers; Ralf Kleene; Maria Girbes Minguez; Melitta Schachner
Journal:  Int J Mol Sci       Date:  2022-03-24       Impact factor: 5.923

  6 in total

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