Literature DB >> 5074751

Partial ornithine carbamyl transferase deficiency: an inborn error of the urea cycle presenting as orotic aciduria in a male infant.

P MacLeod, S Mackenzie, C R Scriver.   

Abstract

Recurrent vomiting without apparent cause should alert the physician to the possibility of a disorder of ammonia metabolism. Crystalluria in a three-month-old male infant with a history of intermittent vomiting since birth and incipient coma led to the discovery of orotic aciduria. A diagnosis of ornithine carbamyl transferase (OCT) deficiency was derived from study of the liver after the infant had died; residual activity was about 5% of normal. Ammonia intoxication was the presumed cause of death. Overproduction of orotic acid and other pyrimidines reflects the deficiency of OCT. The possibility of genetic heterogeneity for the hereditary trait under observation must be considered because it may influence prognosis and counselling.

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Year:  1972        PMID: 5074751      PMCID: PMC1940805     

Source DB:  PubMed          Journal:  Can Med Assoc J        ISSN: 0008-4409            Impact factor:   8.262


  10 in total

1.  Hyperammonaemia. A new instance of an inborn enzymatic defect of the biosynthesis of urea.

Authors:  A RUSSELL; B LEVIN; V G OBERHOLZER; L SINCLAIR
Journal:  Lancet       Date:  1962-10-06       Impact factor: 79.321

2.  Lethal neonatal hyperammonaemia due to complete ornithine-transcarbamylase deficiency.

Authors:  A G Campbell; L E Rosenberg; P J Snodgrass; C T Nuzum
Journal:  Lancet       Date:  1971-07-24       Impact factor: 79.321

3.  Biochemical investigations of hyperammonaemia.

Authors:  B Levin; V G Oberholzer; L Sinclair
Journal:  Lancet       Date:  1969-07-26       Impact factor: 79.321

4.  Hyperammonemia due to a mutant enzyme of ornithine transcarbamylase.

Authors:  I Matsuda; S Arashima; H Nambu; Y Takekoshi; M Anakura
Journal:  Pediatrics       Date:  1971-10       Impact factor: 7.124

5.  Ornithine transcarbamylase, an isoelectric point (pI) isozyme in human liver and its deficiency.

Authors:  S Arashima; I Matsuda
Journal:  Biochem Biophys Res Commun       Date:  1971-10-01       Impact factor: 3.575

6.  Periodic attacks of lethargy in a baby with ammonia intoxication due to a congenital defect in ureogenesis.

Authors:  L M Corbeel; J P Colombo; M Van Sande; A Weber
Journal:  Arch Dis Child       Date:  1969-12       Impact factor: 3.791

7.  Hereditary orotic aciduria. II. A urinary screening test.

Authors:  L E Rogers; F S Porter
Journal:  Pediatrics       Date:  1968-09       Impact factor: 7.124

8.  Hyperammonaemia: a deficiency of liver ornithine transcarbamylase. Occurrence in mother and child.

Authors:  B Levin; J M Abraham; V G Oberholzer; E A Burgess
Journal:  Arch Dis Child       Date:  1969-04       Impact factor: 3.791

9.  Hyperammonaemia. A variant type of deficiency of liver ornithine transcarbamylase.

Authors:  B Levin; R H Dobbs; E A Burgess; T Palmer
Journal:  Arch Dis Child       Date:  1969-04       Impact factor: 3.791

10.  Enzymes in noncultured amniotic fluid cells.

Authors:  H L Nadler; A B Gerbie
Journal:  Am J Obstet Gynecol       Date:  1969-03-01       Impact factor: 8.661

  10 in total
  6 in total

1.  Hyperammonemia through deficiency of ornithine carbamyl transferase.

Authors:  J P Farriaux; J L Dhondt; L Cathelineau; J Ratel; G Fontaine
Journal:  Z Kinderheilkd       Date:  1974

2.  Hyperammonaemia in 20 families. Biochemical and genetical survey, including investigations in 3 new families.

Authors:  T Palmer; V G Oberholzer; E A Burgess; L J Butler; B Levin
Journal:  Arch Dis Child       Date:  1974-06       Impact factor: 3.791

3.  Carrier detection in ornithine transcarbamylase deficiency.

Authors:  E A Haan; D M Danks; A Grimes; N J Hoogenraad
Journal:  J Inherit Metab Dis       Date:  1982       Impact factor: 4.982

4.  Ornithine transcarbamylase deficiency in a male: strict correlation between metabolic control and plasma arginine concentration.

Authors:  U Wendel; J Wieland; H J Bremer; C Bachmann
Journal:  Eur J Pediatr       Date:  1989-01       Impact factor: 3.183

5.  Ornithine carbamoyl transferase deficiency: a neuropathological study.

Authors:  B N Harding; J V Leonard; M Erdohazi
Journal:  Eur J Pediatr       Date:  1984-02       Impact factor: 3.183

6.  Pyrimidine and purine metabolites in ornithine carbamoyl transferase deficiency.

Authors:  D R Webster; H A Simmonds; D M Barry; D M Becroft
Journal:  J Inherit Metab Dis       Date:  1981       Impact factor: 4.982

  6 in total

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