Literature DB >> 9211195

Inherited defects of purine and pyrimidine metabolism: laboratory methods for diagnosis.

M Duran1, L Dorland, E E Meuleman, P Allers, R Berger.   

Abstract

The diagnosis of the majority of the known inherited defects of purine and pyrimidine metabolism can be achieved by analysing urinary excretion profiles. A quantitative measurement of the urinary uric acid/creatinine ratio should be the first approach for purine defects. The general screening system involves separation of the bases and nucleosides by reversed-phase high-performance liquid chromatography and multiwavelength UV detection. The catabolic defects of pyrimidine degradation can be diagnosed by gas chromatography-mass spectrometry as used for organic acids. For the detection of adenylosuccinase deficiency, several simple but effective thin-layer chromatographic methods are available. Techniques such as liquid chromatography-mass spectrometry, direct nega-tiveion fast-atom bombardment mass spectrometry, and proton nuclear magnetic resonance spectroscopy give promising results, but are not yet being used on a large scale. Patients should keep to a simple diet and preferably be free of medication in order to allow a reliable interpretation of the analytical data.

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Year:  1997        PMID: 9211195     DOI: 10.1023/a:1005360907238

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


  20 in total

1.  Dihydropyrimidinuria.

Authors:  M Duran; P Rovers; P K de Bree; C H Schreuder; H Beukenhorst; L Dorland; R Berger
Journal:  Lancet       Date:  1990-09-29       Impact factor: 79.321

2.  Dihydropyrimidinase deficiency presenting in infancy with severe developmental delay.

Authors:  M J Henderson; K Ward; H A Simmonds; J A Duley; P M Davies
Journal:  J Inherit Metab Dis       Date:  1993       Impact factor: 4.982

3.  Simple method of measurement of orotic acid and orotidine in urine.

Authors:  S W Brusilow; E Hauser
Journal:  J Chromatogr       Date:  1989-09-01

4.  Urine test for adenylosuccinase deficiency in autistic children.

Authors:  J Maddocks; T Reed
Journal:  Lancet       Date:  1989-01-21       Impact factor: 79.321

5.  Rapid, high-resolution, two-dimensional amino acid chromatography on micro scale chromatograms.

Authors:  S K Wadman; H F de Jonge; P K de Bree
Journal:  Clin Chim Acta       Date:  1969-07       Impact factor: 3.786

Review 6.  Inborn errors of the purine nucleotide cycle: adenylosuccinase deficiency.

Authors:  G Van den Berghe; M F Vincent; J Jaeken
Journal:  J Inherit Metab Dis       Date:  1997-06       Impact factor: 4.982

7.  Urinary excretion of thymine and uracil in a two-year-old child with a malignant tumor of the brain.

Authors:  G Berglund; J Greter; S Lindstedt; G Steen; J Waldenström; U Wass
Journal:  Clin Chem       Date:  1979-07       Impact factor: 8.327

8.  Urinary excretion of methylated purines in man and in the rat after the administration of theophylline.

Authors:  A H van Gennip; J Grift; E J van Bree-Blom; D Ketting; S K Wadman
Journal:  J Chromatogr       Date:  1979-08-21

9.  Dihydropyrimidine dehydrogenase deficiency leading to thymine-uraciluria. An inborn error of pyrimidine metabolism.

Authors:  R Berger; S A Stoker-de Vries; S K Wadman; M Duran; F A Beemer; P K de Bree; J J Weits-Binnerts; T J Penders; J K van der Woude
Journal:  Clin Chim Acta       Date:  1984-08-31       Impact factor: 3.786

10.  Diagnosis of inherited adenylosuccinase deficiency by thin-layer chromatography of urinary imidazoles and by automated cation exchange column chromatography of purines.

Authors:  P K de Bree; S K Wadman; M Duran; H Fabery de Jonge
Journal:  Clin Chim Acta       Date:  1986-05-15       Impact factor: 3.786

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  8 in total

1.  The application of HPLC/ESI tandem mass spectrometry on urine-soaked filter-paper strips for the screening of disorders of purine and pyrimidine metabolism.

Authors:  T Ito; A B van Kuilenburg; A H Bootsma; A J Haasnoot; A van Cruchten; Y Wada; A H van Gennip
Journal:  J Inherit Metab Dis       Date:  2000-06       Impact factor: 4.982

2.  Dihydropyrimidinase deficiency and congenital microvillous atrophy: coincidence or genetic relation?

Authors:  B Assmann; G F Hoffmann; L Wagner; C Bräutigam; H W Seyberth; M Duran; A B Van Kuilenburg; R Wevers; A H Van Gennip
Journal:  J Inherit Metab Dis       Date:  1997-09       Impact factor: 4.982

3.  Partial hypoxanthine-guanine phosphoribosyltransferase deficiency presenting as acute renal failure.

Authors:  Sarah Cherian; Charles H Crompton
Journal:  Pediatr Nephrol       Date:  2005-10-21       Impact factor: 3.714

Review 4.  Inborn errors of purine and pyrimidine metabolism.

Authors:  A Jurecka
Journal:  J Inherit Metab Dis       Date:  2009-03-15       Impact factor: 4.982

5.  Inborn errors of pyrimidine metabolism: clinical update and therapy.

Authors:  Shanti Balasubramaniam; John A Duley; John Christodoulou
Journal:  J Inherit Metab Dis       Date:  2014-07-17       Impact factor: 4.982

6.  Inborn errors of purine metabolism: clinical update and therapies.

Authors:  Shanti Balasubramaniam; John A Duley; John Christodoulou
Journal:  J Inherit Metab Dis       Date:  2014-06-28       Impact factor: 4.982

Review 7.  Uric acid changes in urine and plasma: an effective tool in screening for purine inborn errors of metabolism and other pathological conditions.

Authors:  R E Simoni; L N L Ferreira Gomes; F B Scalco; C P H Oliveira; F R Aquino Neto; M L Costa de Oliveira
Journal:  J Inherit Metab Dis       Date:  2007-05-19       Impact factor: 4.982

8.  New anthropometry-based age- and sex-specific reference values for urinary 24-hour creatinine excretion based on the adult Swiss population.

Authors:  Valentina Forni Ogna; Adam Ogna; Philippe Vuistiner; Menno Pruijm; Belen Ponte; Daniel Ackermann; Luca Gabutti; Nima Vakilzadeh; Markus Mohaupt; Pierre-Yves Martin; Idris Guessous; Antoinette Péchère-Bertschi; Fred Paccaud; Murielle Bochud; Michel Burnier
Journal:  BMC Med       Date:  2015-02-27       Impact factor: 8.775

  8 in total

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