Literature DB >> 1248389

[Lesch-Nyhan syndrome without self-mutilation: biochemical and morphological studies on blood cells (author's transl)].

W Schneider, E Morgenstern, I Schindera.   

Abstract

Absent enzyme activity of hypoxanthine-guanine-phosphoribosyl transferase in erythrocytes was demonstrated in the blood from a 21-year-old man with spastic tetraplegia, choreoathetosis, oligophrenia, and hyperruricaemia which developed after birth. But there was no tendency towards self-mutilation, considered to be characteristic of the full-blown picture of the disease. This case thus runs counter to the view that the enzyme defect is the cause of the inevitable, stereotyped change in behaviour. Unusual morphological changes of all blood-cell systems confirm the dependence of nucleic acid synthesis on the "salvage pathway" of the purine nucleotides, which is abnormal in this inherited disease.

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Year:  1976        PMID: 1248389     DOI: 10.1055/s-0028-1104054

Source DB:  PubMed          Journal:  Dtsch Med Wochenschr        ISSN: 0012-0472            Impact factor:   0.628


  3 in total

1.  Hyperuricaemia and choreoathetosis in a child without mental retardation or self-mutilation - a new HPRT variant.

Authors:  R P Gottlieb; M M Koppel; W L Nyhan; B Bakay; E Nissinen; M Borden; T Page
Journal:  J Inherit Metab Dis       Date:  1982       Impact factor: 4.982

2.  [Properties of hypoxanthineguanine-phosphoribosyltransferase (HGPRTase) in a gout patient with partial deficiency of this enzyme (author's transl)].

Authors:  W Gröbner; N Zöllner
Journal:  Klin Wochenschr       Date:  1979-01-15

3.  Disassembly of microtubules in the Lesch-Nyhan Syndrome? (Lesch-Nyhan syndrome and microtubules).

Authors:  W Schneider; E Morgenstern; H J Reimers
Journal:  Klin Wochenschr       Date:  1979-02-15
  3 in total

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