Literature DB >> 19783189

Contiguous gene deletion syndrome in a female with ornithine transcarbamylase deficiency.

S Balasubramaniam1, C Rudduck, B Bennetts, G Peters, B Wilcken, C Ellaway.   

Abstract

OTC deficiency, a partially dominant X-linked trait, is the most frequent inborn error of the urea cycle. We describe a female patient with a contiguous gene deletion syndrome encompassing the OTC, DMD, RPGR, CYBB and XK genes, amongst others, only manifesting features of OTC deficiency. Molecular characterization was ascertained by MLPA and confirmed by CGH microarray, which revealed an 8.7 Mb deletion of the X-chromosome. Complete de novo deletion of the OTC gene led to a severe clinical phenotype in the proband. The application of high resolution molecular genetic techniques such as MLPA and array CGH, in mutation negative OTC cases allows the identification of chromosomal rearrangements, such as large deletions and provides information for accurate genetic counseling and prenatal diagnosis.

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Year:  2010        PMID: 19783189     DOI: 10.1016/j.ymgme.2009.08.007

Source DB:  PubMed          Journal:  Mol Genet Metab        ISSN: 1096-7192            Impact factor:   4.797


  9 in total

1.  Large contiguous gene deletions in Sjögren-Larsson syndrome.

Authors:  Holly Engelstad; Gael Carney; Dana S'aulis; Janae Rise; Warren G Sanger; M Katharine Rudd; Gabriele Richard; Christopher W Carr; Omar A Abdul-Rahman; William B Rizzo
Journal:  Mol Genet Metab       Date:  2011-05-30       Impact factor: 4.797

2.  Characterization of the human ornithine transcarbamylase 3' untranslated regulatory region.

Authors:  Monica Lopes-Marques; Isabel Pereira-Castro; António Amorim; Luisa Azevedo
Journal:  DNA Cell Biol       Date:  2011-11-04       Impact factor: 3.311

3.  Disease-causing mutations in the promoter and enhancer of the ornithine transcarbamylase gene.

Authors:  Yoon J Jang; Abigail L LaBella; Timothy P Feeney; Nancy Braverman; Mendel Tuchman; Hiroki Morizono; Nicholas Ah Mew; Ljubica Caldovic
Journal:  Hum Mutat       Date:  2018-01-16       Impact factor: 4.878

4.  'Double trouble': diagnostic challenges in Duchenne muscular dystrophy in patients with an additional hereditary skeletal dysplasia.

Authors:  Sandra Donkervoort; Alice Schindler; Carolina Tesi-Rocha; Allison Schreiber; Meganne E Leach; Jahannaz Dastgir; Ying Hu; Ami Mankodi; Kathryn R Wagner; Neil R Friedman; Carsten G Bönnemann
Journal:  Neuromuscul Disord       Date:  2013-08-11       Impact factor: 4.296

Review 5.  Genotype-Phenotype Correlations in Ornithine Transcarbamylase Deficiency: A Mutation Update.

Authors:  Ljubica Caldovic; Iman Abdikarim; Sahas Narain; Mendel Tuchman; Hiroki Morizono
Journal:  J Genet Genomics       Date:  2015-05-19       Impact factor: 4.275

6.  Heterogeneous lengths of copy number mutations in human coagulopathy revealed by genome-wide high-density SNP array.

Authors:  Hee-Jin Kim; Duk-Kyung Kim; Ki-Young Yoo; Chur-Woo You; Jong-Ha Yoo; Ki-O Lee; In-Ae Park; Hae-Sun Choung; Hee-Jung Kim; Min-Jung Song; Sun-Hee Kim
Journal:  Haematologica       Date:  2011-10-11       Impact factor: 9.941

7.  A deep intronic variant is a common cause of OTC deficiency in individuals with previously negative genetic testing.

Authors:  Runjun D Kumar; Lindsay C Burrage; Jan Bartos; Saima Ali; Eric Schmitt; Sandesh C S Nagamani; Cynthia LeMons
Journal:  Mol Genet Metab Rep       Date:  2021-01-08

8.  Long-term outcomes in Ornithine Transcarbamylase deficiency: a series of 90 patients.

Authors:  Anais Brassier; Stephanie Gobin; Jean Baptiste Arnoux; Vassili Valayannopoulos; Florence Habarou; Manoelle Kossorotoff; Aude Servais; Valerie Barbier; Sandrine Dubois; Guy Touati; Robert Barouki; Fabrice Lesage; Laurent Dupic; Jean Paul Bonnefont; Chris Ottolenghi; Pascale De Lonlay
Journal:  Orphanet J Rare Dis       Date:  2015-05-10       Impact factor: 4.123

9.  Gene Mutation Analysis and Prenatal Diagnosis of the Ornithine Transcarbamylase (OTC) Gene in Two Families with Ornithine Transcarbamylase Deficiency.

Authors:  Sitao Li; Yao Cai; Congcong Shi; Mengxian Liu; Bingqing Liu; Lin Lin; Xin Xiao; Hu Hao
Journal:  Med Sci Monit       Date:  2018-10-18
  9 in total

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