Literature DB >> 6188953

Evidence for liver disease preceding amino acid abnormalities in hereditary tyrosinemia.

M K Hostetter, H L Levy, H S Winter, G J Knight, J E Haddow.   

Abstract

In hereditary tyrosinemia, an autosomal recessive disorder of infants, it has been postulated that hepatic toxicity arising from defective degradation of tyrosine accounts for the severe liver disease that is a feature of this condition. We measured the concentration of alpha-fetoprotein, a marker for liver disease, and of amino acid in cord blood from three infants with hereditary tyrosinemia and found that the concentration of alpha-fetoprotein was greatly increased at birth, whereas the level of tyrosine was normal or not specifically elevated, and that hypertyrosinemia developed only postnatally. These results indicate that liver disease is prenatal in hereditary tyrosinemia and that therapy aimed at reduction of the elevated tyrosine level is unlikely to be of fundamental value.

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Year:  1983        PMID: 6188953     DOI: 10.1056/NEJM198305263082105

Source DB:  PubMed          Journal:  N Engl J Med        ISSN: 0028-4793            Impact factor:   91.245


  10 in total

1.  Hereditary tyrosinemia type 1 from a single center in Egypt: clinical study of 22 cases.

Authors:  Hanaa El-Karaksy; Mona Fahmy; Mona El-Raziky; Nehal El-Koofy; Rokaya El-Sayed; Mohamed S Rashed; Hasan El-Kiki; Ahmad El-Hennawy; Nabil Mohsen
Journal:  World J Pediatr       Date:  2011-06-01       Impact factor: 2.764

2.  Coagulation defect of congenital tyrosinaemia.

Authors:  S Ozsoylu
Journal:  Arch Dis Child       Date:  1985-02       Impact factor: 3.791

3.  DNA ploidy abnormalities in the liver of children with hereditary tyrosinemia type I. Correlation with histopathologic features.

Authors:  C Zerbini; D S Weinberg; K A Hollister; A R Perez-Atayde
Journal:  Am J Pathol       Date:  1992-05       Impact factor: 4.307

4.  Significant increase of succinylacetone within the first 12 h of life in hereditary tyrosinemia type 1.

Authors:  Jan-Ulrich Schlump; Ertan Mayatepek; Ute Spiekerkoetter
Journal:  Eur J Pediatr       Date:  2009-10-08       Impact factor: 3.183

5.  Fumarylacetoacetate hydrolase deficient pigs are a novel large animal model of metabolic liver disease.

Authors:  Raymond D Hickey; Shennen A Mao; Jaime Glorioso; Joseph B Lillegard; James E Fisher; Bruce Amiot; Piero Rinaldo; Cary O Harding; Ronald Marler; Milton J Finegold; Markus Grompe; Scott L Nyberg
Journal:  Stem Cell Res       Date:  2014-05-14       Impact factor: 2.020

6.  Severe neurological crisis in a patient with hereditary tyrosinaemia type I after interruption of NTBC treatment.

Authors:  J-U Schlump; C Perot; K Ketteler; M Schiff; E Mayatepek; U Wendel; U Spiekerkoetter
Journal:  J Inherit Metab Dis       Date:  2008-05-20       Impact factor: 4.982

7.  Visceral pathology of hereditary tyrosinemia type I.

Authors:  P Russo; S O'Regan
Journal:  Am J Hum Genet       Date:  1990-08       Impact factor: 11.025

8.  Mildly elevated succinylacetone and normal liver function in compound heterozygotes with pathogenic and pseudodeficient FAH alleles.

Authors:  Hao Yang; Francis Rossignol; Denis Cyr; Rachel Laframboise; Shu Pei Wang; Jean-François Soucy; Marie-Thérèse Berthier; Yves Giguère; Paula J Waters; Grant A Mitchell
Journal:  Mol Genet Metab Rep       Date:  2017-12-27

9.  Type 1 tyrosinemia in Finland: a nationwide study.

Authors:  Linnea Äärelä; Pauliina Hiltunen; Tea Soini; Nina Vuorela; Heini Huhtala; Pasi I Nevalainen; Markku Heikinheimo; Laura Kivelä; Kalle Kurppa
Journal:  Orphanet J Rare Dis       Date:  2020-10-12       Impact factor: 4.123

Review 10.  Recommendations for the management of tyrosinaemia type 1.

Authors:  Corinne de Laet; Carlo Dionisi-Vici; James V Leonard; Patrick McKiernan; Grant Mitchell; Lidia Monti; Hélène Ogier de Baulny; Guillem Pintos-Morell; Ute Spiekerkötter
Journal:  Orphanet J Rare Dis       Date:  2013-01-11       Impact factor: 4.123

  10 in total

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