Literature DB >> 1250313

Urea-cycle enzyme deficiencies and an increased nitrogen load producing hyperammonemia in Reye's syndrome.

P J Snodgrass, G R DeLong.   

Abstract

Assay of urea-cycle enzymes in liver tissue showed ornithine transcarbamylase activities of 18 to 72 per cent of the normal mean in eight patients with Reye's syndrome, below the range of normal in seven of eight, and, in six cases, as low as those in females with X-linked deficiency of this enzyme. Carbamyl phosphate synthetase activities were less than 32 per cent of controls in two patients. Argininosuccinate synthetase and lyase activities were normal in seven patients. Arginase was normal in two biopsy specimens, but below normal in four of five autopsy specimens. The Km's for ornithine and carbamyl phosphate, pH optimum, and heat lability of ornithine transcarbamylase were normal. Two patients excreted 0.64 and 0.58 g per kilogram per day of urinary nitrogen at the peak of hyperammonemia, in spite of peritoneal dialysis. The hyperammonemia of Reye's syndrome apparently results from excess waste nitrogen that overwhelms the ability of reduced ornithine transcarbamylase (and occasionally carbamyl phosphate synthetase) to detoxify the ammonia load.

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Year:  1976        PMID: 1250313     DOI: 10.1056/NEJM197604152941601

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


  8 in total

1.  Neonatal hyperammonaemia with complete absence of liver carbamyl phosphate synthetase activity.

Authors:  S Mantagos; S Tsagaraki; E A Burgess; V Oberholzer; T Palmer; J Sacks; S Baibas; T Valaes
Journal:  Arch Dis Child       Date:  1978-03       Impact factor: 3.791

2.  Liver pathology in transient neonatal hyperammonemia.

Authors:  A Zimmermann; C Bachmann; I Högger; J Gehler; A Mielfried; E Mönch; H Peters
Journal:  Virchows Arch A Pathol Anat Histopathol       Date:  1983

3.  Recurrent, familial Reye-like syndrome with a new complex amino and organic aciduria.

Authors:  O N Elpeleg; E Christensen; H Hurvitz; D Branski
Journal:  Eur J Pediatr       Date:  1990-07       Impact factor: 3.183

4.  Murine adenovirus infection of SCID mice induces hepatic lesions that resemble human Reye syndrome.

Authors:  L Pirofski; M S Horwitz; M D Scharff; S M Factor
Journal:  Proc Natl Acad Sci U S A       Date:  1991-05-15       Impact factor: 11.205

5.  Interactions of aspirin and other potential etiologic factors in an animal model of Reye syndrome.

Authors:  D R Deshmukh; H F Maassab; M Mason
Journal:  Proc Natl Acad Sci U S A       Date:  1982-12       Impact factor: 11.205

Review 6.  Reye's syndrome: a clinical review.

Authors:  J F Crocker; P C Bagnell
Journal:  Can Med Assoc J       Date:  1981-02-15       Impact factor: 8.262

7.  Humanized liver mouse model with transplanted human hepatocytes from patients with ornithine transcarbamylase deficiency.

Authors:  Go Sugahara; Chihiro Yamasaki; Ami Yanagi; Suzue Furukawa; Yuko Ogawa; Akinari Fukuda; Shin Enosawa; Akihiro Umezawa; Yuji Ishida; Chise Tateno
Journal:  J Inherit Metab Dis       Date:  2020-12-30       Impact factor: 4.982

Review 8.  Acute hepatic failure in children.

Authors:  C A Riely
Journal:  Yale J Biol Med       Date:  1984 Mar-Apr
  8 in total

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