Literature DB >> 20551993

5q11.2 deletion in a patient with tracheal agenesis.

Elisabeth M de Jong1, Hannie Douben, Bert H Eussen, Janine F Felix, Marja W Wessels, Pino J Poddighe, Peter G J Nikkels, Ronald R de Krijger, Dick Tibboel, Annelies de Klein.   

Abstract

Tracheal agenesis (TA) is a rare congenital anomaly of the respiratory tract. Many patients have associated anomalies, suggesting a syndromal phenotype. In a cohort of 12 patients, we aimed to detect copy number variations. In addition to routine cytogenetic analysis, we applied oligonucleotide array comparative genomic hybridization. Our patient cohort showed various copy number variations, of which many were parentally inherited variants. One patient had, in addition to an inherited 16p12.1 deletion, a 3.6 Mb deletion on chromosomal locus 5q11.2. This patient had a syndromic phenotype, including vertebral, anal, cardiovascular and tracheo-oesophageal associated anomalies, and other foregut-related anomalies, such as cartilage rings in the oesophagus and an aberrant right bronchus. No common deletions or duplications are found in our cohort, suggesting that TA is a genetically heterogeneous disorder.

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Year:  2010        PMID: 20551993      PMCID: PMC2987473          DOI: 10.1038/ejhg.2010.84

Source DB:  PubMed          Journal:  Eur J Hum Genet        ISSN: 1018-4813            Impact factor:   4.246


  27 in total

Review 1.  Congenital absence of the trachea.

Authors:  M B van Veenendaal; K D Liem; H A Marres
Journal:  Eur J Pediatr       Date:  2000 Jan-Feb       Impact factor: 3.183

2.  Tracheal agenesis with anomalies found in both VACTERL and TACRD associations.

Authors:  Julie L Wei; David Rodeberg; Dana M Thompson
Journal:  Int J Pediatr Otorhinolaryngol       Date:  2003-09       Impact factor: 1.675

3.  Detection of large-scale variation in the human genome.

Authors:  A John Iafrate; Lars Feuk; Miguel N Rivera; Marc L Listewnik; Patricia K Donahoe; Ying Qi; Stephen W Scherer; Charles Lee
Journal:  Nat Genet       Date:  2004-08-01       Impact factor: 38.330

4.  Diminished callus size and cartilage synthesis in alpha 1 beta 1 integrin-deficient mice during bone fracture healing.

Authors:  Erika Ekholm; Kurt D Hankenson; Hannele Uusitalo; Ari Hiltunen; Humphrey Gardner; Jyrki Heino; Risto Penttinen
Journal:  Am J Pathol       Date:  2002-05       Impact factor: 4.307

Review 5.  Tracheal agenesis in infants with VATER association.

Authors:  J M Milstein; M Lau; R G Bickers
Journal:  Am J Dis Child       Date:  1985-01

6.  Sonic hedgehog is essential to foregut development.

Authors:  Y Litingtung; L Lei; H Westphal; C Chiang
Journal:  Nat Genet       Date:  1998-09       Impact factor: 38.330

7.  Essential function of Gli2 and Gli3 in the formation of lung, trachea and oesophagus.

Authors:  J Motoyama; J Liu; R Mo; Q Ding; M Post; C C Hui
Journal:  Nat Genet       Date:  1998-09       Impact factor: 38.330

8.  Tissue-specific expression of retinoic acid receptor isoform transcripts in the mouse embryo.

Authors:  R Mollard; S Viville; S J Ward; D Décimo; P Chambon; P Dollé
Journal:  Mech Dev       Date:  2000-06       Impact factor: 1.882

9.  Tracheal agenesis and associated malformations: a comparison with tracheoesophageal fistula and the VACTERL association.

Authors:  J A Evans; J Reggin; C Greenberg
Journal:  Am J Med Genet       Date:  1985-05

10.  Multiple defects and perinatal death in mice deficient in follistatin.

Authors:  M M Matzuk; N Lu; H Vogel; K Sellheyer; D R Roop; A Bradley
Journal:  Nature       Date:  1995-03-23       Impact factor: 49.962

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  14 in total

Review 1.  Familial occurrence of the VATER/VACTERL association.

Authors:  Alina Hilger; Charlotte Schramm; Markus Draaken; Sadaf S Mughal; Gabriel Dworschak; Enrika Bartels; Per Hoffmann; Markus M Nöthen; Heiko Reutter; Michael Ludwig
Journal:  Pediatr Surg Int       Date:  2012-03-16       Impact factor: 1.827

Review 2.  Underlying genetic factors of the VATER/VACTERL association with special emphasis on the "Renal" phenotype.

Authors:  Heiko Reutter; Alina C Hilger; Friedhelm Hildebrandt; Michael Ludwig
Journal:  Pediatr Nephrol       Date:  2016-02-08       Impact factor: 3.714

3.  VACTERL Association Etiology: The Impact of de novo and Rare Copy Number Variations.

Authors:  E Brosens; H Eussen; Y van Bever; R M van der Helm; H Ijsselstijn; H P Zaveri; R Wijnen; D A Scott; D Tibboel; A de Klein
Journal:  Mol Syndromol       Date:  2013-02

4.  De novo microduplications at 1q41, 2q37.3, and 8q24.3 in patients with VATER/VACTERL association.

Authors:  Alina Hilger; Charlotte Schramm; Tracie Pennimpede; Lars Wittler; Gabriel C Dworschak; Enrika Bartels; Hartmut Engels; Alexander M Zink; Franziska Degenhardt; Annette M Müller; Eberhard Schmiedeke; Sabine Grasshoff-Derr; Stefanie Märzheuser; Stuart Hosie; Stefan Holland-Cunz; Charlotte H W Wijers; Carlo L M Marcelis; Iris A L M van Rooij; Friedhelm Hildebrandt; Bernhard G Herrmann; Markus M Nöthen; Michael Ludwig; Heiko Reutter; Markus Draaken
Journal:  Eur J Hum Genet       Date:  2013-04-03       Impact factor: 4.246

5.  VATER/VACTERL Association: Evidence for the Role of Genetic Factors.

Authors:  H Reutter; M Ludwig
Journal:  Mol Syndromol       Date:  2013-02

6.  Isl1 Regulation of Nkx2.1 in the Early Foregut Epithelium Is Required for Trachea-Esophageal Separation and Lung Lobation.

Authors:  Eugene Kim; Ming Jiang; Huachao Huang; Yongchun Zhang; Natalie Tjota; Xia Gao; Jacques Robert; Nikesha Gilmore; Lin Gan; Jianwen Que
Journal:  Dev Cell       Date:  2019-12-05       Impact factor: 12.270

Review 7.  Clinical geneticists' views of VACTERL/VATER association.

Authors:  Benjamin D Solomon; Kelly A Bear; Virginia Kimonis; Annelies de Klein; Daryl A Scott; Charles Shaw-Smith; Dick Tibboel; Heiko Reutter; Philip F Giampietro
Journal:  Am J Med Genet A       Date:  2012-11-19       Impact factor: 2.802

Review 8.  VACTERL/VATER Association.

Authors:  Benjamin D Solomon
Journal:  Orphanet J Rare Dis       Date:  2011-08-16       Impact factor: 4.123

9.  VACTERL association and mitochondrial dysfunction.

Authors:  Benjamin D Solomon; Ankita Patel; Sau Wai Cheung; Daniel E Pineda-Alvarez
Journal:  Birth Defects Res A Clin Mol Teratol       Date:  2011-02-09

10.  Cav3.2 T-type calcium channel is required for the NFAT-dependent Sox9 expression in tracheal cartilage.

Authors:  Shin-Shiou Lin; Bing-Hsiean Tzeng; Kuan-Rong Lee; Richard J H Smith; Kevin P Campbell; Chien-Chang Chen
Journal:  Proc Natl Acad Sci U S A       Date:  2014-04-28       Impact factor: 11.205

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