Literature DB >> 25620158

Oculo-facio-cardio-dental (OFCD) syndrome: the first Italian case of BCOR and co-occurring OTC gene deletion.

C Di Stefano1, B Lombardo2, C Fabbricatore3, C Munno3, I Caliendo4, F Gallo5, L Pastore3.   

Abstract

Oculo-facio-cardio-dental (OFCD) syndrome is a rare genetic disorder affecting ocular, facial, dental and cardiac systems. The syndrome is an X-linked dominant trait and it might be lethal in males. This syndrome is usually caused by mutations in the BCL6 interacting co-repressor gene (BCOR). We described a female child with mild phenotype of oculo-facio-cardio-dental syndrome. Array-comparative genomic hybridization (a-CGH) analysis revealed a de novo heterozygous deletion in the Xp11.4 region of approximately 2.3 Mb, involving BCOR and ornithine carbamoyl-transferase (OTC) genes. The deletion observed was subsequently confirmed by real time PCR. In this study we report a first case with co-occurrence of BCOR and OTC genes completely deleted in OFCD syndrome.
Copyright © 2015 Elsevier B.V. All rights reserved.

Entities:  

Keywords:  BCOR; OTC; Oculo-facio-cardio-dental syndrome; a-CGH

Mesh:

Substances:

Year:  2015        PMID: 25620158     DOI: 10.1016/j.gene.2015.01.044

Source DB:  PubMed          Journal:  Gene        ISSN: 0378-1119            Impact factor:   3.688


  8 in total

1.  Expression of Normal or Mutated X-Linked BCOR Transcripts in OFCD iPSCs.

Authors:  I El Ayachi; X-Y Zou; X Yan; Y Lou; G T-J Huang
Journal:  J Dent Res       Date:  2019-11-27       Impact factor: 6.116

2.  Combined aCGH and Exome Sequencing Analysis Improves Autism Spectrum Disorders Diagnosis: A Case Report.

Authors:  Annaluisa Ranieri; Iolanda Veneruso; Ilaria La Monica; Maria Grazia Pascale; Lucio Pastore; Valeria D'Argenio; Barbara Lombardo
Journal:  Medicina (Kaunas)       Date:  2022-04-07       Impact factor: 2.948

3.  Ocular findings in a patient with oculofaciocardiodental (OFCD) syndrome and a novel BCOR pathogenic variant.

Authors:  Yujia Zhou; Antonina Wojcik; Victoria R Sanders; Bahram Rahmani; Sudhi P Kurup
Journal:  Int Ophthalmol       Date:  2017-10-22       Impact factor: 2.031

4.  A novel deletion mutation in the BCOR gene is associated with oculo-facio-cardio-dental syndrome: a case report.

Authors:  Qian Hu; Jingqun Mai; Qinqin Xiang; Bin Zhou; Shanling Liu; Jing Wang
Journal:  BMC Pediatr       Date:  2022-02-07       Impact factor: 2.125

5.  Identification of a De Novo Deletion by Using A-CGH Involving PLNAX2: An Interesting Candidate Gene in Psychomotor Developmental Delay.

Authors:  Noemi Falcone; Annaluisa Ranieri; Andrea Vitale; Lucio Pastore; Barbara Lombardo
Journal:  Medicina (Kaunas)       Date:  2022-04-08       Impact factor: 2.948

6.  D-aspartate oxidase gene duplication induces social recognition memory deficit in mice and intellectual disabilities in humans.

Authors:  Barbara Lombardo; Marco Pagani; Arianna De Rosa; Marcella Nunziato; Sara Migliarini; Martina Garofalo; Marta Terrile; Valeria D'Argenio; Alberto Galbusera; Tommaso Nuzzo; Annaluisa Ranieri; Andrea Vitale; Eleonora Leggiero; Anna Di Maio; Noemi Barsotti; Ugo Borello; Francesco Napolitano; Alessandra Mandarino; Marco Carotenuto; Uriel Heresco-Levy; Massimo Pasqualetti; Paolo Malatesta; Alessandro Gozzi; Francesco Errico; Francesco Salvatore; Lucio Pastore; Alessandro Usiello
Journal:  Transl Psychiatry       Date:  2022-08-01       Impact factor: 7.989

7.  OFCD syndrome and extraembryonic defects are revealed by conditional mutation of the Polycomb-group repressive complex 1.1 (PRC1.1) gene BCOR.

Authors:  Michelle Y Hamline; Connie M Corcoran; Joseph A Wamstad; Isabelle Miletich; Jifan Feng; Jamie L Lohr; Myriam Hemberger; Paul T Sharpe; Micah D Gearhart; Vivian J Bardwell
Journal:  Dev Biol       Date:  2020-07-18       Impact factor: 3.148

8.  Genetic analysis resolves differential diagnosis of a familial syndromic dilated cardiomyopathy: A new case of Alström syndrome.

Authors:  Barbara Lombardo; Valeria D'Argenio; Emanuele Monda; Andrea Vitale; Martina Caiazza; Lucia Sacchetti; Lucio Pastore; Giuseppe Limongelli; Giulia Frisso; Cristina Mazzaccara
Journal:  Mol Genet Genomic Med       Date:  2020-05-12       Impact factor: 2.183

  8 in total

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