Literature DB >> 17786115

Alagille syndrome with deletion 20p12.2-p12.3 and hypoplastic left heart.

Marie Leema P Robert1, Tony Lopez, John Crolla, Shuwen Huang, Carol Owen, Lisa Burvill-Holmes, Oliver Stumper, Peter D Turnpenny.   

Abstract

We report a male patient born at 37-weeks gestation, weighing 1.885 kg (<0.4th centile). Shortly after birth, he was diagnosed with hypoplastic left heart syndrome for which he underwent a Norwood procedure. Subsequently, he developed problems with failure to thrive and developmental delay. At the age of 4 years his delay in growth and development led to further investigations, which revealed a small de-novo interstitial deletion of chromosome 20p12.2. JAGGED1 haploinsufficiency was confirmed by fluorescence in situ hybridization. Array-comparative genomic hybridization analysis confirmed and quantified an approximate 5.4 Mb interstitial deletion involving the chromosomal region 20p12.2-p12.3. This precise interstitial deletion has not been previously reported. Further clinical evaluation revealed posterior embryotoxon and butterfly vertebrae. He has normal liver function tests, facial features consistent with Alagille syndrome, and mild learning difficulties. To our knowledge this is the first report of Alagille syndrome associated with hypoplastic left heart syndrome.

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Year:  2007        PMID: 17786115     DOI: 10.1097/MCD.0b013e3282358d21

Source DB:  PubMed          Journal:  Clin Dysmorphol        ISSN: 0962-8827            Impact factor:   0.816


  3 in total

Review 1.  Alagille syndrome: pathogenesis, diagnosis and management.

Authors:  Peter D Turnpenny; Sian Ellard
Journal:  Eur J Hum Genet       Date:  2011-09-21       Impact factor: 4.246

2.  Microdeletion 20p12.3 involving BMP2 contributes to syndromic forms of cleft palate.

Authors:  Trilochan Sahoo; Aaron Theisen; Pedro A Sanchez-Lara; Michael Marble; Daniela N Schweitzer; Beth S Torchia; Allen N Lamb; Bassem A Bejjani; Lisa G Shaffer; Yves Lacassie
Journal:  Am J Med Genet A       Date:  2011-06-10       Impact factor: 2.802

3.  Cleft palate in a multigenerational family with a microdeletion of 20p12.3 involving BMP2.

Authors:  Eli S Williams; Kim A Uhas; Brian P Bunke; Kathryn B Garber; Christa L Martin
Journal:  Am J Med Genet A       Date:  2012-09-10       Impact factor: 2.802

  3 in total

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