Literature DB >> 4017276

Deficiency of fumarylacetoacetase without hereditary tyrosinemia.

E A Kvittingen, A L Børresen, O Stokke, C B van der Hagen, S O Lie.   

Abstract

A variant of the enzyme fumarylacetoacetase (FAH) (E.C.3.7.1.2) in healthy individuals, determined by the enzyme activity, is reported. Analysis of family members of probands with low FAH activity suggests that the enzyme variant causing low activity could be the product of a pseudodeficiency gene. Assumed homozygotes for this gene have only slightly higher enzyme activity than patients with the metabolic disorder hereditary tyrosinemia I (hepatorenal type). No clinical abnormalities have been found in association with the postulated gene.

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Year:  1985        PMID: 4017276     DOI: 10.1111/j.1399-0004.1985.tb02039.x

Source DB:  PubMed          Journal:  Clin Genet        ISSN: 0009-9163            Impact factor:   4.438


  6 in total

1.  The human fumarylacetoacetase gene: characterisation of restriction fragment length polymorphisms and identification of haplotypes in tyrosinemia type 1 and pseudodeficiency.

Authors:  H Rootwelt; E A Kvittingen; K Høie; E Agsteribbe; M Hartog; H van Faassen; R Berger
Journal:  Hum Genet       Date:  1992-05       Impact factor: 4.132

2.  Identification of a frequent pseudodeficiency mutation in the fumarylacetoacetase gene, with implications for diagnosis of tyrosinemia type I.

Authors:  H Rootwelt; E Brodtkorb; E A Kvittingen
Journal:  Am J Hum Genet       Date:  1994-12       Impact factor: 11.025

3.  Fumarylacetoacetase measurement as a mass-screening procedure for hereditary tyrosinemia type I.

Authors:  C Laberge; A Grenier; J P Valet; J Morissette
Journal:  Am J Hum Genet       Date:  1990-08       Impact factor: 11.025

4.  Oral loading of homogentisic acid in controls and in obligate heterozygotes for hereditary tyrosinemia type I.

Authors:  C Laberge; A Lescault; A Grenier; J Morrisette; R Gagné; P Gadbois; J Halket
Journal:  Am J Hum Genet       Date:  1990-08       Impact factor: 11.025

5.  Tyrosinaemia type 1 and glutathione synthetase deficiency: two disorders with reduced hepatic thiol group concentrations and a liver 4-fumarylacetoacetate hydrolase deficiency.

Authors:  A J Lloyd; R G Gray; A Green
Journal:  J Inherit Metab Dis       Date:  1995       Impact factor: 4.982

6.  A missense mutation (Q279R) in the fumarylacetoacetate hydrolase gene, responsible for hereditary tyrosinemia, acts as a splicing mutation.

Authors:  N Dreumont; J A Poudrier; A Bergeron; H L Levy; F Baklouti; R M Tanguay
Journal:  BMC Genet       Date:  2001-06-29       Impact factor: 2.797

  6 in total

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