Abstract
The role of arginine as an essential amino was evaluated in four children with one of the deficiencies of carbamyl phosphate synthetase, ornithine transcarbamylase, argininosuccinate synthetase, and argininosuccinase. Within 15-68 h after arginine deprivation nitrogen accumulated as ammonium or glutamine or both, but glutamine was quantitatively the largest nitrogen accumulation product. Concomitantly plasma and urinary urea levels decreased. Resumption of arginine intake (or citrulline in the case of ornithine transcarbamylase deficiency) promptly led to correction of the hyperammonemia, hyperglutaminemia and hypoargininemia. Ornithine was an unsatisfactory substitute for arginine. Arginine deprivation did not interfere with carbamyl phosphate synthesis as manifested by orotic aciduria. It is concluded that arginine is an indispensable amino acid for children with inborn errors of ureagenesis and its absence results in the rapid onset of symptomatic hyperammonemia.
MeSH Terms
Amino Acid Metabolism, Inborn Errors/metabolism
Ammonia/blood
Arginine/physiology,therapeutic use
Argininosuccinate Synthase/deficiency
Argininosuccinic Aciduria
Blood Urea Nitrogen
Carbamoyl-Phosphate Synthase (Ammonia)/deficiency
Child, Preschool
Female
Glutamine/blood
Humans
Infant
Infant, Newborn
Male
Ornithine/blood
Ornithine Carbamoyltransferase Deficiency Disease
Urea/biosynthesis
Chemicals
Glutamine
Ammonia
Urea
Arginine
Ornithine
Carbamoyl-Phosphate Synthase (Ammonia)
Argininosuccinate Synthase
Authors & Affiliations
1 authors, click to expand affiliations / ORCID
Brusilow S W
References (10)
10 references, click to expand
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