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两名巴德-比德尔综合征患者的胱抑素C水平异常。

Abnormal cystatin C levels in two patients with bardet-biedl syndrome.

作者信息

Hirano Makito, Ohishi Mitsuru, Yamashita Toshihide, Ikuno Yasushi, Iwahashi Hiromi, Mano Toshiyuki, Ishihara Ryu, Tanaka Ichiro, Yanagihara Keiko, Isono Chiharu, Sakamoto Hikaru, Nakamura Yusaku, Kusunoki Susumu

机构信息

Department of Neurology, Sakai Hospital Kinki University Faculty of Medicine.

出版信息

Clin Med Insights Case Rep. 2011;4:17-20. doi: 10.4137/CCRep.S6622. Epub 2011 Mar 10.

Abstract

Bardet-Biedl syndrome (BBS) is an autosomal recessive disorder characterized by central obesity, mental impairment, rod-cone dystrophy, polydactyly, hypogonadism in males, and renal abnormalities. The causative genes have been identified as BBS1-14. In the Western countries, the prevalence of this disease ranges from 1/13,500 to 1/160,000, while only a few Japanese patients have been reported in the English-language literature. The incidence of renal dysfunction or anomalies in previous reports varies considerably ranging from ∼20% to universal occurrence. We here report that two Japanese patients who had BBS with normal BUN and creatinine levels had elevated levels of cystatin C, a sensitive marker of glomerular filtration rate. A urine albumin level increased only in the elder patient. Thus, cystatin C may be useful for detecting renal abnormalities in patients with an apparent normal renal function. Because this disease is diagnosed by accumulation of symptoms, such a sensitive marker might help early diagnosis of BBS.

摘要

巴德-比埃尔综合征(BBS)是一种常染色体隐性疾病,其特征为中心性肥胖、智力障碍、视杆-视锥营养不良、多指(趾)畸形、男性性腺功能减退以及肾脏异常。已确定致病基因有BBS1 - 14。在西方国家,该病患病率为1/13500至1/160000,而英文文献中报道的日本患者仅有少数几例。既往报道中肾功能障碍或异常的发生率差异很大,从约20%到普遍发生不等。我们在此报告,两名患有BBS且血尿素氮和肌酐水平正常的日本患者,其胱抑素C水平升高,胱抑素C是肾小球滤过率的敏感标志物。仅年长患者的尿白蛋白水平升高。因此,胱抑素C可能有助于检测肾功能明显正常的患者的肾脏异常。由于该疾病通过症状累积来诊断,这样一种敏感标志物可能有助于BBS的早期诊断。

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Bardet-Biedl syndrome with syndrome X: a patient report.伴有X综合征的巴德-比埃尔综合征:一例患者报告
J Pediatr Endocrinol Metab. 2004 Jun;17(6):913-5. doi: 10.1515/jpem.2004.17.6.914.

本文引用的文献

1
Renal failure--measuring the glomerular filtration rate.肾衰竭——肾小球滤过率的测定。
Dtsch Arztebl Int. 2009 Dec;106(51-52):849-54. doi: 10.3238/arztebl.2009.0849. Epub 2009 Dec 18.
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The spectrum of renal disease in Laurence-Moon-Biedl syndrome.
N Engl J Med. 1988 Sep 8;319(10):615-8. doi: 10.1056/NEJM198809083191005.
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Laurence-Moon-Biedl syndrome accompanied by congenital hepatic fibrosis.劳-穆-比三氏综合征伴先天性肝纤维化
J Gastroenterol Hepatol. 1990 Mar-Apr;5(2):206-10. doi: 10.1111/j.1440-1746.1990.tb01826.x.

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