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无自残行为或肾结石的莱施-奈恩综合征罕见变异型。

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

作者信息

Kersnik Levart Tanja

机构信息

Department of Pediatric Nephrology, University Medical Center, Stare pravde 4, 1000 Ljubljana, Slovenia.

出版信息

Pediatr Nephrol. 2007 Nov;22(11):1975-8. doi: 10.1007/s00467-007-0566-3. Epub 2007 Aug 7.

DOI:10.1007/s00467-007-0566-3
PMID:17680274
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.

摘要

莱施-奈恩综合征是一种非常罕见的X连锁隐性疾病,其特征为智力发育迟缓、类似脑瘫的痉挛状态、舞蹈手足徐动症、自残行为和高尿酸血症。自残行为是该疾病的一个标志。高尿酸血症会导致高尿酸尿症和尿酸肾结石。潜在缺陷是次黄嘌呤-鸟嘌呤-磷酸核糖转移酶缺乏。我们报告了一名7岁患有莱施-奈恩综合征的男孩,他没有自残行为,曾被误诊为手足徐动型脑瘫。他也没有肾结石;肾髓质锥体高回声是唯一检测到的肾脏异常,在超声检查中与髓质肾钙质沉着症无法区分。

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1
Rare variant of Lesch-Nyhan syndrome without self-mutilation or nephrolithiasis.无自残行为或肾结石的莱施-奈恩综合征罕见变异型。
Pediatr Nephrol. 2007 Nov;22(11):1975-8. doi: 10.1007/s00467-007-0566-3. Epub 2007 Aug 7.
2
Differential diagnosis of cerebral palsy: Lesch-Nyhan syndrome without self-mutilation.脑瘫的鉴别诊断:无自残行为的莱施-奈恩综合征。
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3
Self-mutilation behaviour in Lesch-Nyhan syndrome.莱施-奈恩综合征中的自残行为。
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Nephrocalcinosis and Renal Failure in Lesch-Nyhan Syndrome: Report of Two Familial Cases and Review of the Literature.莱施-奈恩综合征中的肾钙质沉着症和肾衰竭:两例家族性病例报告及文献综述
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本文引用的文献

1
Atlantoaxial subluxation with recurrent consciousness disturbance in a boy with Lesch-Nyhan syndrome.患有莱施-奈恩综合征的男孩出现寰枢椎半脱位并伴有反复意识障碍。
Acta Paediatr. 2006 Nov;95(11):1500-4. doi: 10.1080/08035250600617131.
2
Molecular basis of hypoxanthine-guanine phosphoribosyltransferase deficiency in Italian Lesch-Nyhan patients: identification of nine novel mutations.意大利莱施-奈恩病患者次黄嘌呤-鸟嘌呤磷酸核糖转移酶缺乏症的分子基础:九个新突变的鉴定
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A FAMILIAL DISORDER OF URIC ACID METABOLISM AND CENTRAL NERVOUS SYSTEM FUNCTION.
Pediatr Nephrol. 2009 May;24(5):1089-90. doi: 10.1007/s00467-008-1081-x. Epub 2008 Dec 19.
一种尿酸代谢与中枢神经系统功能的家族性疾病。
Am J Med. 1964 Apr;36:561-70. doi: 10.1016/0002-9343(64)90104-4.
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Serial renal sonographic evaluation of patients with Lesch-Nyhan syndrome.对莱施-奈恩综合征患者进行肾脏超声系列评估。
Pediatr Radiol. 1994;24(7):509-12. doi: 10.1007/BF02015015.
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Hypoxanthine-guanine phosphoribosyltransferase variants: correlation of clinical phenotype with enzyme activity.次黄嘌呤 - 鸟嘌呤磷酸核糖转移酶变体:临床表型与酶活性的相关性
J Inherit Metab Dis. 1981;4(4):203-6. doi: 10.1007/BF02263652.
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Differential diagnosis of cerebral palsy: Lesch-Nyhan syndrome without self-mutilation.脑瘫的鉴别诊断:无自残行为的莱施-奈恩综合征。
Can Med Assoc J. 1984 May 15;130(10):1323-4.
7
Renal sonography in long standing Lesch-Nyhan syndrome.长期莱施-奈恩综合征的肾脏超声检查
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