Literature DB >> 3106719

A new family affected by the syndrome of hyperornithinaemia, hyperammonaemia and homocitrullinuria.

M Rodes, A Ribes, M Pineda, L Alvarez, I Fabregas, E Fernandez Alvarez, F X Coude, G Grimber.   

Abstract

We have reported three cases of hyperornithinaemia associated with hyperammonaemia and homocitrullinuria (HHH). They deal with two brothers and a sister from a family where the parents and four other children are healthy on clinical and biochemical examination. The biochemical findings in our patients indicate the existence of a defect in the transport of ornithine into the mitochondria. Cultured skin fibroblasts from two of these patients incorporated six times less [14C]ornithine into protein as compared to control cells. The most characteristic sign of the clinical picture is the progressive spastic paraparesis found in one of the cases. Ornithine supplementation and restricted protein intake may be useful in the treatment of this syndrome since after three years of treatment the clinical response was favourable and the patients showed no adverse clinical effects.

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Year:  1987        PMID: 3106719     DOI: 10.1007/bf01799492

Source DB:  PubMed          Journal:  J Inherit Metab Dis        ISSN: 0141-8955            Impact factor:   4.982


  15 in total

1.  Ornithinemia, hyperammonemia, and homocitrullinuria. A disease associated with mental retardation and possibly caused by defective mitochondrial transport.

Authors:  V Fell; R J Pollitt; G A Sampson; T Wright
Journal:  Am J Dis Child       Date:  1974-05

2.  Comparison of amino acid concentrations between plasma and erythrocytes. Studies in normal human subjects and those with metabolic disorders.

Authors:  H L Levy; E Barkin
Journal:  J Lab Clin Med       Date:  1971-10

3.  Reduced ornithine catabolism in cultured fibroblasts and phytohaemagglutinin-stimulated lymphocytes from a patient with hyperornithinaemia, hyperammonaemia and homocitrullinuria.

Authors:  R G Gray; S E Hill; R J Pollitt
Journal:  Clin Chim Acta       Date:  1982-02-05       Impact factor: 3.786

4.  Defective ornithine metabolism in the syndrome of hyperornithinaemia, hyperammonaemia and homocitrullinuria.

Authors:  V E Shih; R Mandell; A Herzfeld
Journal:  J Inherit Metab Dis       Date:  1981       Impact factor: 4.982

5.  3-Amino-2-piperidone in the urine of patients with hyperornithinemia.

Authors:  V G Oberholzer; A Briddon
Journal:  Clin Chim Acta       Date:  1978-08-01       Impact factor: 3.786

6.  The mechanism of hyperammonaemia and hyperornithinaemia in the syndrome of hyperornithinaemia, hyperammonaemia with homocitrullinuria.

Authors:  K Oyanagi; A Tsuchiyama; Y Itakura; H Sogawa; K Wagatsuma; T Nakao; S Sakamoto; A Yachi
Journal:  J Inherit Metab Dis       Date:  1983       Impact factor: 4.982

7.  Studies on the pathway from ornithine to proline in cultured skin fibroblasts with reference to the defect in hyperornithinaemia with hyperammonaemia and homocitrullinuria.

Authors:  R G Gray; S E Hill; R J Pollitt
Journal:  J Inherit Metab Dis       Date:  1983       Impact factor: 4.982

8.  Decreased transport of ornithine across the inner mitochondrial membrane as a cause of hyperornithinaemia.

Authors:  F A Hommes; C K Ho; R A Roesel; M E Coryell; B A Gordon
Journal:  J Inherit Metab Dis       Date:  1982       Impact factor: 4.982

9.  Defective ornithine metabolism in cultured skin fibroblasts from patients with the syndrome of hyperornithinemia, hyperammonemia and homocitrullinuria.

Authors:  V E Shih; R Mandell; A Herzfeld
Journal:  Clin Chim Acta       Date:  1982-02-05       Impact factor: 3.786

10.  Hyperornithinemia, hyperammonemia, and homocitrullinuria. A new disorder of amino acid metabolism associated with myoclonic seizures and mental retardation.

Authors:  V E Shih; M L Efron; H W Moser
Journal:  Am J Dis Child       Date:  1969-01
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  2 in total

1.  Comparison of ornithine metabolism in hyperornithinaemia-hyperammonaemia-homocitrullinuria syndrome, lysinuric protein intolerance and gyrate atrophy fibroblasts.

Authors:  J Botschner; D W Smith; O Simell; C R Scriver
Journal:  J Inherit Metab Dis       Date:  1989       Impact factor: 4.982

Review 2.  The hyperornithinemia-hyperammonemia-homocitrullinuria syndrome.

Authors:  Diego Martinelli; Daria Diodato; Emanuela Ponzi; Magnus Monné; Sara Boenzi; Enrico Bertini; Giuseppe Fiermonte; Carlo Dionisi-Vici
Journal:  Orphanet J Rare Dis       Date:  2015-03-11       Impact factor: 4.123

  2 in total

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