Literature DB >> 19332529

Two cases of deletion 2q37 associated with segregation of an unbalanced translocation 2;21: choanal atresia leading to misdiagnosis of CHARGE syndrome.

Eduardo Fernández-Rebollo1, Olga Pérez, Cristina Martinez-Bouzas, Maria Carmen Cotarelo-Pérez, Intza Garin, Jose Luis Ruibal, Gustavo Pérez-Nanclares, Luis Castaño, Guiomar Pérez de Nanclares.   

Abstract

CONTEXT: The phenotypic variability of patients with syndromes presenting with dysmorphism makes clinical diagnosis difficult, leading to an exhaustive genetic study to determine the underlying mechanism so that a proper diagnosis could be established.
OBJECTIVE: To genetically characterize siblings, the older sister diagnosed with Albright hereditary osteodystrophy and the younger one with CHARGE syndrome.
DESIGN: Clinical case report.
METHODS: Clinical, biochemical, and radiological studies were performed on the family. In addition, molecular genetic studies including sequencing of GNAS, typing of microsatellites on 2q and 21q, and multiplex ligation-dependent probe amplification of subtelomeric regions were performed, as well as confirmatory fluorescent in situ hybridization analysis.
RESULTS: The genetic analysis revealed that both sisters presented a 2q37 deletion due to the maternal unbalanced segregation of a 2;21 translocation.
CONCLUSIONS: This is the first report of a 2q37 deletion where differential diagnosis of CHARGE syndrome is needed due to the appearance of choanal atresia.

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Year:  2009        PMID: 19332529     DOI: 10.1530/EJE-08-0865

Source DB:  PubMed          Journal:  Eur J Endocrinol        ISSN: 0804-4643            Impact factor:   6.664


  5 in total

1.  The 2q37-deletion syndrome: an update of the clinical spectrum including overweight, brachydactyly and behavioural features in 14 new patients.

Authors:  Camille Leroy; Emilie Landais; Sylvain Briault; Albert David; Olivier Tassy; Nicolas Gruchy; Bruno Delobel; Marie-José Grégoire; Bruno Leheup; Laurence Taine; Didier Lacombe; Marie-Ange Delrue; Annick Toutain; Agathe Paubel; Francine Mugneret; Christel Thauvin-Robinet; Stéphanie Arpin; Cedric Le Caignec; Philippe Jonveaux; Mylène Beri; Nathalie Leporrier; Jacques Motte; Caroline Fiquet; Olivier Brichet; Monique Mozelle-Nivoix; Pascal Sabouraud; Nathalie Golovkine; Nathalie Bednarek; Dominique Gaillard; Martine Doco-Fenzy
Journal:  Eur J Hum Genet       Date:  2012-10-17       Impact factor: 4.246

2.  Structural Chromosome Abnormalities Associated with Obesity: Report of Four New subjects and Review of Literature.

Authors:  Majed J Dasouki; Erin L Youngs; Karine Hovanes
Journal:  Curr Genomics       Date:  2011-05       Impact factor: 2.236

Review 3.  Brachydactyly E: isolated or as a feature of a syndrome.

Authors:  Arrate Pereda; Intza Garin; Maria Garcia-Barcina; Blanca Gener; Elena Beristain; Ane Miren Ibañez; Guiomar Perez de Nanclares
Journal:  Orphanet J Rare Dis       Date:  2013-09-12       Impact factor: 4.123

4.  X-linked Hypophosphatemic Rickets, del(2)(q37.1;q37.3) Deletion Syndrome and Mosaic Turner Syndrome, mos 45,X/46,X, del(2)(q37.1;q37.3) in a 3-year-old Female.

Authors:  Alaina P Vidmar; Brian Miyazaki; Pedro A Sanchez-Lara; Pisit Pitukcheewanont
Journal:  J Bone Metab       Date:  2017-11-30

5.  What to consider when pseudohypoparathyroidism is ruled out: iPPSD and differential diagnosis.

Authors:  Arrate Pereda; Intza Garin; Guiomar Perez de Nanclares
Journal:  BMC Med Genet       Date:  2018-03-02       Impact factor: 2.103

  5 in total

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