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莱施-奈恩综合征中肾脏氧嘌呤沉积:超声评估

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

作者信息

Stevens S K, Parker B R

机构信息

Department of Diagnostic Radiology and Nuclear Medicine, Children's Hospital, Stanford, California.

出版信息

Pediatr Radiol. 1989;19(6-7):479-80. doi: 10.1007/BF02387665.

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.

摘要

莱施-奈恩综合征是一种罕见的X连锁隐性嘌呤代谢紊乱疾病,与次黄嘌呤-鸟嘌呤磷酸核糖转移酶(HPRT)几乎完全缺乏有关。该疾病的特征为高尿酸血症、自残行为、舞蹈手足徐动症、痉挛和智力发育迟缓。嘌呤代谢异常在出生时就存在,可能在生命早期导致尿酸结晶尿和结石形成。莱施-奈恩综合征的影像学表现包括腹部X线平片上隐约可见的不透射线结石,或者如果存在肾脏疾病,静脉肾盂造影显示肾脏小且功能差。透X线结石通常由尿酸组成;然而,也有几例接受别嘌呤醇治疗的莱施-奈恩患者出现含黄嘌呤和次黄嘌呤结石的病例报道。氧嘌呤是次黄嘌呤、黄嘌呤和尿酸这些化合物的统称,它们都可能是透X线的。我们报告1例莱施-奈恩综合征病例,超声检查很容易显示出推测的肾实质氧嘌呤沉积,但在标准X线平片或静脉肾盂造影上未检测到。

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