Abstract
A coordinate relationship between the activities of two sequential enzymes in the de novo pyrimidine biosynthetic pathway has been demonstrated in human red cells. The two enzymes, orotidylate phosphoribosyltransferase and decarboxylase are responsible for the conversion of orotic acid to uridine-5'-monophosphate. Fractionation of red cells, on the basis of increase of specific gravity with cell age, has revealed that these two enzymes have a marked but equal degree of lability in the ageing red cell. It is postulated that orotidylate phosphoribosyltransferase and decarboxylase form an enzyme-enzyme complex, and that the sequential deficiency of these two enzymes in hereditary orotic aciduria may reflect a structural abnormality in this complex. In patients receiving allopurinol, the activities of both enzymes are coordinately increased, and this increase appears to be due, at least in part, to stabilization of both orotidylate phosphoribosyltransferase and decarboxylase in the ageing red cell. Allopurinol ribonucleotide is an in vitro inhibitor of orotidine-5'-monophosphate decarboxylase and requires the enzyme hypoxanthineguanine phosphoribosyltransferase for its synthesis. However, the administration of allopurinol to patients lacking this enzyme results in orotidinuria and these patients have elevated orotidylate phosphoribosyltransferase and decarboxylase activities in their erythrocytes. Evidence is presented that the chief metabolite of allopurinol, oxipurinol, with a 2,4-diketo pyrimidine ring is capable of acting as an analogue of orotic acid. It is postulated that the in vivo formation of oxipurinol ribonucleotide, catalyzed by orotidylate phosphoribosyltransferase, after allopurinol administration, leads to inhibition of orotidine-5'-monophosphate decarboxylase. This inhibition results in the urinary excretion of excessive amounts of orotidine and orotic acid, and "pseudo-substrate" stabilization of orotidylate phosphoribosyltransferase and decarboxylase.
MeSH Terms
Adenine/metabolism
Allopurinol/pharmacology,therapeutic use
Carbon Isotopes
Carboxy-Lyases/metabolism
Centrifugation, Density Gradient
Depression, Chemical
Erythrocytes/enzymology,metabolism
Humans
Hypoxanthines/metabolism
Lesch-Nyhan Syndrome/drug therapy,metabolism
Nucleosides/urine
Orotic Acid/urine
Transferases/metabolism
Uracil Nucleotides/biosynthesis
Chemicals
Carbon Isotopes
Hypoxanthines
Nucleosides
Uracil Nucleotides
Orotic Acid
Allopurinol
Transferases
Carboxy-Lyases
Adenine
Authors & Affiliations
3 authors, click to expand affiliations / ORCID
Fox R M
Wood M H
O'Sullivan W J
References (28)
28 references, click to expand
-
Bacterial preparation of orotidine-5'-phosphate and uridine-5'-phosphate.
Nature. 1958 Oct 18;182(4642):1090-1
PMID: 13590232
-
Orotic aciduria. Differing enzyme patterns.
Am J Med. 1969 Aug;47(2):332-6
PMID: 5808247
-
Drugs and uric acid.
Annu Rev Pharmacol. 1969;9:345-62
PMID: 4953095
-
Genetic regulatory mechanisms in the synthesis of proteins.
J Mol Biol. 1961 Jun;3:318-56
PMID: 13718526
-
Red cell glucose-6-phosphate and 6-phosphogluconic dehydrogenases and nucleoside phosphorylase.
Science. 1958 Jun 6;127(3310):1338-9
PMID: 13555886
-
Studies on the control of pyrimidine biosynthesis in human diploid cell strains. II. Effects of 5-azaorotic acid, barbituric acid, and pyrimidine precursors on cellular phenotype.
Proc Natl Acad Sci U S A. 1967 May;57(5):1267-74
PMID: 5231733
-
Enzyme activities in human red blood cells of different age groups.
Isr J Med Sci. 1966 May-Jun;2(3):291-6
PMID: 4224847
-
A specific enzyme defect in gout associated with overproduction of uric acid.
Proc Natl Acad Sci U S A. 1967 Jun;57(6):1735-9
PMID: 4291947
-
Dihydroorotic acid dehydrogenase activity of human diploid cell strains.
Science. 1968 May 3;160(3827):539-41
PMID: 5644059
-
Allopurinol: alteration in pyrimidine metabolism in man.
Science. 1970 Jul 24;169(3943):388-90
PMID: 5450375
-
Elevated AMP pyrophosphorylase activity in congenital IMP pyrophosphorylase deficiencey (Lesch-Nyhan disease).
J Lab Clin Med. 1969 Nov;74(5):732-41
PMID: 5350202
-
Purification of yeast inorganic pyrophosphatase.
J Biol Chem. 1951 Sep;192(1):87-94
PMID: 14917654
-
3-RIBOSYLPURINES. I. SYNTHESIS OF (3-RIBOSYLURIC ACID) 5'-PHOSPHATE AND (3-RIBOSYLXANTHINE) 5'-PHOSPHATE BY A PYRIMIDINE RIBONUCLEOTIDE PYROPHOSPHORYLASE OF BEEF ERYTHROCYTES.
J Biol Chem. 1964 Aug;239:2580-6
PMID: 14235538
-
Studies on the control of pyrimidine biosynthesis in human diploid cell strains, I. Effect of 6-azauridine on cellular phenotype.
Proc Natl Acad Sci U S A. 1967 Apr;57(4):925-32
PMID: 5231355
-
Hereditary orotic aciduria. II. A urinary screening test.
Pediatrics. 1968 Sep;42(3):423-8
PMID: 5677483
-
DETERMINATION OF DENSITY DISTRIBUTION OF RED CELL POPULATION.
J Lab Clin Med. 1964 Oct;64:668-74
PMID: 14233155
-
Coordination of the synthesis of the enzymes in the pyrimidine pathway of E. coli.
J Mol Biol. 1962 Dec;5:618-34
PMID: 13970180
-
PYRIMIDINE METABOLISM IN MAN. IV. THE ENZYMATIC DEFECT OF OROTIC ACIDURIA.
J Clin Invest. 1961 Apr;40(4):656-64
PMID: 16695858
-
Genetic control of enzyme activity in higher organisms.
Biochem Genet. 1970 Feb;4(1):105-25
PMID: 4192088
-
A FAMILIAL DISORDER OF URIC ACID METABOLISM AND CENTRAL NERVOUS SYSTEM FUNCTION.
Am J Med. 1964 Apr;36:561-70
PMID: 14142409
-
Nucleotide biosynthesis from preformed purines in mammalian cells: regulatory mechanisms and biological significance.
Prog Nucleic Acid Res Mol Biol. 1970;10:87-119
PMID: 4910307
-
PURIFICATION AND PROPERTIES OF OROTIC ACID-DECARBOXYLATING ENZYMES FROM CALF THYMUS.
J Biol Chem. 1964 Dec;239:4245-9
PMID: 14247677
-
Purification and kinetic properties of brain orotidine 5'-phosphate decarboxylase.
J Biol Chem. 1968 Jul 25;243(14):3924-9
PMID: 5661716
-
Orotidinuria induced by allopurinol.
Science. 1970 May 15;168(3933):861-2
PMID: 5444063
-
Ribonucleosides of allopurinol and oxoallopurinol. Isolation from human urine, enzymatic synthesis, and characterization.
J Biol Chem. 1967 Jun 10;242(11):2675-82
PMID: 6028796
-
XANTHINE AS A PRECURSOR OF NUCLEIC ACID PURINES IN THE MOUSE.
Biochim Biophys Acta. 1965 Mar 15;95:505-6
PMID: 14342542
-
Effects of allopurinol and oxipurinol on purine synthesis in cultured human cells.
J Clin Invest. 1970 Mar;49(3):602-9
PMID: 5415686
-
Substrate stabilization: genetically controlled reciprocal relationship of two human enzymes.
Science. 1970 Feb 6;167(3919):887-9
PMID: 5410854