Literature DB >> 7906500

Inborn errors of urea synthesis.

M L Batshaw1.   

Abstract

Inborn errors of urea synthesis can present in the newborn period as a catastrophic illness or later in childhood or adulthood with an indolent course punctuated by hyperammonemic episodes. Because symptoms mimic other neuropsychiatric disorders, it is common for there to be a delay in diagnosis, often with dire consequences. Diagnosis relies on the combination of clinical suspicion and the measurement of ammonium, lactate, and amino acids in plasma and organic acids and orotic acid in urine. Treatment involves nitrogen restriction combined with the stimulation of alternate pathways of waste nitrogen excretion. More recently liver transplantation has been performed as enzyme replacement therapy. The outcome is poor in children who survive prolonged neonatal hyperammonemic coma, with most manifesting developmental disabilities. The etiology of neuronal injury in this disorder is unclear but may involve some combination of ammonia/amino acid accumulation, neurotransmitter alterations, and excitotoxic injury. Gene therapy holds the promise of improved treatment in the future.

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Year:  1994        PMID: 7906500     DOI: 10.1002/ana.410350204

Source DB:  PubMed          Journal:  Ann Neurol        ISSN: 0364-5134            Impact factor:   10.422


  14 in total

1.  Fatal hyperammonemic coma caused by ornithine transcarbamylase deficiency in a woman.

Authors:  T Perpoint; L Argaud; Q Blanc; D Robert
Journal:  Intensive Care Med       Date:  2001-11-07       Impact factor: 17.440

2.  In vivo nitrogen metabolism in ornithine transcarbamylase deficiency.

Authors:  M Yudkoff; Y Daikhin; I Nissim; A Jawad; J Wilson; M Batshaw
Journal:  J Clin Invest       Date:  1996-11-01       Impact factor: 14.808

3.  Differential inhibition by hyperammonemia of the electron transport chain enzymes in synaptosomes and non-synaptic mitochondria in ornithine transcarbamylase-deficient spf-mice: restoration by acetyl-L-carnitine.

Authors:  K Qureshi; K V Rao; I A Qureshi
Journal:  Neurochem Res       Date:  1998-06       Impact factor: 3.996

Review 4.  Cerebral edema and liver disease: Classic perspectives and contemporary hypotheses on mechanism.

Authors:  Eric M Liotta; W Taylor Kimberly
Journal:  Neurosci Lett       Date:  2020-02-05       Impact factor: 3.046

5.  ECMO as a Platform for Rapid Ammonia Removal in a Neonate with Multienzyme Urea Cycle Disorder.

Authors:  Kelechi Ikeri; Vilmaris Quinones Cardona; Abena Hagan-Brown; Megan Young; Michael Schneider; Ogechukwu Menkiti
Journal:  J Extra Corpor Technol       Date:  2020-03

6.  Human carbamoyl-phosphate synthetase: insight into N-acetylglutamate interaction and the functional effects of a common single nucleotide polymorphism.

Authors:  V Ahuja; S G Powers-Lee
Journal:  J Inherit Metab Dis       Date:  2008-08-09       Impact factor: 4.982

7.  Molecular genetic research into carbamoyl-phosphate synthase I: molecular defects and linkage markers.

Authors:  M L Summar
Journal:  J Inherit Metab Dis       Date:  1998       Impact factor: 4.982

8.  Developing adenoviral-mediated in vivo gene therapy for ornithine transcarbamylase deficiency.

Authors:  S E Raper; J M Wilson; M Yudkoff; M B Robinson; X Ye; M L Batshaw
Journal:  J Inherit Metab Dis       Date:  1998       Impact factor: 4.982

9.  In vivo measurement of ureagenesis with stable isotopes.

Authors:  M Yudkoff; Y Daikhin; X Ye; J M Wilson; M L Batshaw
Journal:  J Inherit Metab Dis       Date:  1998       Impact factor: 4.982

Review 10.  Neurological implications of urea cycle disorders.

Authors:  A L Gropman; M Summar; J V Leonard
Journal:  J Inherit Metab Dis       Date:  2007-11-23       Impact factor: 4.982

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