Literature DB >> 17680274

Rare variant of Lesch-Nyhan syndrome without self-mutilation or nephrolithiasis.

Tanja Kersnik Levart1.   

Abstract

Lesch-Nyhan syndrome is a very rare X-linked recessive disorder characterized by mental retardation, spasticity resembling cerebral palsy, choreo-athetosis, self-mutilation and hyperuricemia. Self-mutilative behavior is a hallmark of the disease. Hyperuricemia leads to hyperuricuria and uric acid nephrolithiasis. The underlying defect is a deficiency of hypoxanthine-guanine-phosphoribosyl transferase. We report on a 7-year-old boy with Lesch-Nyhan syndrome, lacking self-mutilative behavior, who was erroneously diagnosed as having athetotic cerebral palsy. He also had no renal stones; hyperechoic renal medullary pyramids were the only renal abnormality detected and were sonographically indistinguishable from medullary nephrocalcinosis.

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Year:  2007        PMID: 17680274     DOI: 10.1007/s00467-007-0566-3

Source DB:  PubMed          Journal:  Pediatr Nephrol        ISSN: 0931-041X            Impact factor:   3.714


  7 in total

1.  A FAMILIAL DISORDER OF URIC ACID METABOLISM AND CENTRAL NERVOUS SYSTEM FUNCTION.

Authors:  M LESCH; W L NYHAN
Journal:  Am J Med       Date:  1964-04       Impact factor: 4.965

2.  Atlantoaxial subluxation with recurrent consciousness disturbance in a boy with Lesch-Nyhan syndrome.

Authors:  Jia-Woei Hou
Journal:  Acta Paediatr       Date:  2006-11       Impact factor: 2.299

3.  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

4.  Molecular basis of hypoxanthine-guanine phosphoribosyltransferase deficiency in Italian Lesch-Nyhan patients: identification of nine novel mutations.

Authors:  M Bertelli; D Randi; V Micheli; S Gallo; G Andrighetto; P Parmigiani; G Jacomelli; M Carella; C Lievore; M Pandolfo
Journal:  J Inherit Metab Dis       Date:  2004       Impact factor: 4.982

5.  Differential diagnosis of cerebral palsy: Lesch-Nyhan syndrome without self-mutilation.

Authors:  G Mitchell; R R McInnes
Journal:  Can Med Assoc J       Date:  1984-05-15       Impact factor: 8.262

6.  Hypoxanthine-guanine phosphoribosyltransferase variants: correlation of clinical phenotype with enzyme activity.

Authors:  T Page; B Bakay; E Nissinen; W L Nyhan
Journal:  J Inherit Metab Dis       Date:  1981       Impact factor: 4.982

7.  Renal sonography in long standing Lesch-Nyhan syndrome.

Authors:  I J Kenney
Journal:  Clin Radiol       Date:  1991-01       Impact factor: 2.350

  7 in total
  1 in total

1.  Chronic peritoneal dialysis in a 4-year-old boy with Lesch-Nyhan disease.

Authors:  Su Jin Kim; Young Bae Sohn; Sung Won Park; Dong-Kyu Jin; Kyung Hoon Paik
Journal:  Pediatr Nephrol       Date:  2008-12-19       Impact factor: 3.714

  1 in total

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