Sikora Przemysław, Pijanowska Monika, Majewski Marek, Bieniaś Beata, Borzecka Halina, Zajczkowska Małgorzata
Department of Pediatric Nephrology, Medical University of Lublin, ul. Chodzki 2, 20-093, Lublin, Poland,
Pediatr Nephrol. 2006 Jul;21(7):1045-7. doi: 10.1007/s00467-006-0149-8. Epub 2006 May 24.
Lesch-Nyhan syndrome is a very rare X-linked recessive disorder caused by mutation in the gene encoding enzyme hypoxanthine-guanine phosphoribosyltransferase (HPRT). A complete deficiency of HPRT leads to severe purine overproduction and to uric acid renal lithiasis as a consequence. This may be effectively prevented by administration of allopurinol; however, its overdosage may result in xanthinuria and xanthine urolithiasis. We report on a 9-year-old boy with Lesch-Nyhan syndrome who developed acute renal failure due to bilateral staghorn xanthine urolithiasis resulting from long-term treatment with excessive doses of allopurinol. To the best of our knowledge, the presented case is the first one in the literature.
莱施-奈恩综合征是一种非常罕见的X连锁隐性疾病,由编码次黄嘌呤-鸟嘌呤磷酸核糖转移酶(HPRT)的基因突变引起。HPRT完全缺乏会导致严重的嘌呤过度产生,进而导致尿酸肾结石。服用别嘌醇可有效预防这种情况;然而,过量服用可能会导致黄嘌呤尿症和黄嘌呤尿路结石。我们报告了一名9岁患有莱施-奈恩综合征的男孩,他因长期过量服用别嘌醇导致双侧鹿角状黄嘌呤尿路结石而发展为急性肾衰竭。据我们所知,该病例是文献中首例。