Literature DB >> 2671903

Renal oxypurine deposition in Lesch-Nyhan syndrome: sonographic evaluation.

S K Stevens1, B R Parker.   

Abstract

Lesch-Nyhan syndrome is a rare X-linked recessive disorder of purine metabolism associated with a virtually complete deficiency of the enzyme hypoxanthine-guanine phosphoribosyl-transferase (HPRT). The disease is characterized by hyperuricemia, self-multilation, choreoathetosis, spasticity, and mental retardation. The abnormalities of purine metabolism are present at birth and may lead to uric acid crystalluria and stone formation early in life. Radiographic findings described in Lesch-Nyhan syndrome include faintly radio-opaque stones on abdominal radiographs or, if renal disease is present, small kidneys with poor function on intravenous urogram. Radiolucent stones are usually composed of uric acid; however, several cases of xanthine and hypoxanthine-containing calculi in Lesch-Nyhan patients receiving allopurinl therapy have also been described. Oxypurine is the collective name for the compounds hypoxanthine, xanthine, and uric acid, and all may be radiolucent. We report a case of Lesch-Nyhan syndrome with presumed renal parenchymal oxypurine deposition demonstrated readily by ultrasonography but not detected on standard radiographs or intravenous urograms.

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Year:  1989        PMID: 2671903     DOI: 10.1007/bf02387665

Source DB:  PubMed          Journal:  Pediatr Radiol        ISSN: 0301-0449


  4 in total

Review 1.  Xanthine stone formation.

Authors:  J E Seegmiller
Journal:  Am J Med       Date:  1968-11       Impact factor: 4.965

2.  Urinary xanthine stones--a rare complications of allopurinol therapy.

Authors:  M L Greene; W Y Fujimoto; J E Seegmiller
Journal:  N Engl J Med       Date:  1969-02-20       Impact factor: 91.245

3.  An autopsy case of the Lesch-Nyhan syndrome: normal HGPRT activity in liver and xanthine calculi in various tissues.

Authors:  T Mizuno; H Endoh; Y Konishi; Y Miyachi; I Akoaka
Journal:  Neuropadiatrie       Date:  1976-08

4.  Xanthine calculi in the Lesch-Nyhan syndrome.

Authors:  W A Brock; J Golden; G W Kaplan
Journal:  J Urol       Date:  1983-07       Impact factor: 7.450

  4 in total
  1 in total

1.  Serial renal sonographic evaluation of patients with Lesch-Nyhan syndrome.

Authors:  D L Rosenfeld; M P Preston; K Salvaggi-Fadden
Journal:  Pediatr Radiol       Date:  1994
  1 in total

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