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Increased urinary leukotriene E(4) during febrile attacks in the hyperimmunoglobulinaemia D and periodic fever syndrome.

AbstractBACKGROUND:
The hyperimmunoglobulinaemia D and periodic fever syndrome is a hereditary periodic fever, caused by deficiency of the enzyme mevalonate kinase. It is unclear how this defect leads to recurrent fever episodes.
AIM:
To assess the involvement of cysteinyl leukotrienes in the pathogenesis of fever attacks as reflected by urinary leukotriene E(4) (LTE(4)) excretion.
METHODS:
Urinary LTE(4) was measured in seven patients while febrile and afebrile.
RESULTS:
LTE(4) was raised during fever in all subjects (46-199 nmol/mol creatinine, mean 92; normal <40). Urinary LTE(4) was normal between attacks, as well as in normal children with fever as a result of miscellaneous causes.
CONCLUSION:
Our results suggest that cysteinyl leukotrienes play a role in the pathophysiology of this disorder. As no effective treatment is yet available, leukotriene receptor antagonists might offer a new therapeutic approach for patients with the hyperimmunoglobulinaemia D and periodic fever syndrome.
AuthorsJ Frenkel, M A Willemsen, C M Weemaes, L Dorland, E Mayatepek
JournalArchives of disease in childhood (Arch Dis Child) Vol. 85 Issue 2 Pg. 158-9 (Aug 2001) ISSN: 1468-2044 [Electronic] England
PMID11466192 (Publication Type: Journal Article, Research Support, Non-U.S. Gov't)
Chemical References
  • Immunoglobulin D
  • Leukotriene E4
  • Mevalonic Acid
Topics
  • Adolescent
  • Child
  • Chromatography, Gas
  • Chromatography, High Pressure Liquid
  • Familial Mediterranean Fever (urine)
  • Humans
  • Hypergammaglobulinemia (urine)
  • Immunoglobulin D
  • Leukotriene E4 (urine)
  • Mass Spectrometry
  • Mevalonic Acid (urine)
  • Radioisotope Dilution Technique

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