Literature DB >> 23430845

Phenotype-Genotype Discrepancy Due to a 5.5-kb Deletion in the GALT Gene.

Ariadna González-del Angel1, José Velázquez-Aragón, Miguel A Alcántara-Ortigoza, Marcela Vela-Amieva, Nancy Hernández-Martínez.   

Abstract

Classical galactosemia is an autosomal recessive inborn error of metabolism caused by a deficiency of the galactose-1-phosphate uridyltransferase (GALT). More than 200 mutations have been described in the GALT gene. A 5.5-kb GALT deletion, first described in patients of Ashkenazi Jewish ancestry, may lead either to an erroneous genotype assignment of classical galactosemia or to discrepancies with parental genotypes and the expected biochemical phenotype. The presence of the 5.5-kb deletion was examined in 27 Mexican nonrelated families with at least one child with reduced GALT activity in erythrocytes and it was detected in the 5.5% (n=3) of the 54 alleles tested. The first molecular studies in three of our families showed that the genotypes of the parents were inconsistent with those of their children, which were considered initially as homozygous p.N314D-Duarte 2, but after analyzing for the presence of the 5.5-kb deletion, were reassigned as compound heterozygotes [5.5-kb deletion]+[p.N314D-Duarte 2]. Identification of the 5.5-kb deletion in Mexican patients suggests that this mutation might not be exclusive to a given ethnic group and should be tested in other populations, especially when there is a discrepancy between the genotypes of patients and parents or by incongruence between biochemical phenotype and GALT genotype. Establishing a genotype-phenotype correlation for the 5.5-kb GALT deletion and determining the appropriate management will require additional studies in patients with a G/G genotype bearing the 5.5-kb GALT deletion.

Entities:  

Year:  2011        PMID: 23430845      PMCID: PMC3509830          DOI: 10.1007/8904_2011_30

Source DB:  PubMed          Journal:  JIMD Rep        ISSN: 2192-8304


  9 in total

1.  Monitoring of biochemical status in children with Duarte galactosemia: utility of galactose, galactitol, galactonate, and galactose 1-phosphate.

Authors:  Can Ficicioglu; Christie Hussa; Paul R Gallagher; Nina Thomas; Claire Yager
Journal:  Clin Chem       Date:  2010-05-20       Impact factor: 8.327

2.  Galactosemia: deletion in the 5' upstream region of the GALT gene reduces promoter efficiency.

Authors:  M Trbusek; H Francová; L Kozák
Journal:  Hum Genet       Date:  2001-07       Impact factor: 4.132

Review 3.  Classical galactosaemia revisited.

Authors:  Annet M Bosch
Journal:  J Inherit Metab Dis       Date:  2006-07-11       Impact factor: 4.982

4.  Low allelic heterogeneity in a sample of Mexican patients with classical galactosaemia.

Authors:  J Velázquez-Aragón; M A Alcántara-Ortigoza; M Vela-Amieva; S Monroy; V Martínez-Cruz; C Todd-Quiñones; A González-del Angel
Journal:  J Inherit Metab Dis       Date:  2008-10-29       Impact factor: 4.982

5.  Mutation database for the galactose-1-phosphate uridyltransferase (GALT) gene.

Authors:  Fernanda R O Calderon; Amit R Phansalkar; David K Crockett; Martin Miller; Rong Mao
Journal:  Hum Mutat       Date:  2007-10       Impact factor: 4.878

Review 6.  The molecular biology of galactosemia.

Authors:  L J Elsas; K Lai
Journal:  Genet Med       Date:  1998 Nov-Dec       Impact factor: 8.822

7.  Origins, distribution and expression of the Duarte-2 (D2) allele of galactose-1-phosphate uridylyltransferase.

Authors:  Amanda E Carney; Rebecca D Sanders; Kerry R Garza; Lee Anne McGaha; Lora J H Bean; Bradford W Coffee; James W Thomas; David J Cutler; Natalie L Kurtkaya; Judith L Fridovich-Keil
Journal:  Hum Mol Genet       Date:  2009-02-18       Impact factor: 6.150

8.  Characterization of an unusual deletion of the galactose-1-phosphate uridyl transferase (GALT) gene.

Authors:  Bradford Coffee; Lawrence N Hjelm; Angela DeLorenzo; Ebony M Courtney; Chunli Yu; Kasinathan Muralidharan
Journal:  Genet Med       Date:  2006-10       Impact factor: 8.822

9.  Prevention of a molecular misdiagnosis in galactosemia.

Authors:  Deborah Barbouth; Tatiana Slepak; Helene Klapper; Kent Lai; Louis J Elsas
Journal:  Genet Med       Date:  2006-03       Impact factor: 8.822

  9 in total

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