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Abnormal cystatin C levels in two patients with bardet-biedl syndrome.两名巴德-比德尔综合征患者的胱抑素C水平异常。
Clin Med Insights Case Rep. 2011;4:17-20. doi: 10.4137/CCRep.S6622. Epub 2011 Mar 10.
2
New criteria for improved diagnosis of Bardet-Biedl syndrome: results of a population survey.改善巴德-比德尔综合征诊断的新标准:一项人群调查结果
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The First Nationwide Survey and Genetic Analyses of Bardet-Biedl Syndrome in Japan.日本巴德-比埃尔综合征的首次全国性调查及基因分析
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Clinical characteristics of a Japanese patient with Bardet-Biedl syndrome caused by BBS10 mutations.一名由BBS10基因突变引起的巴德-比德尔综合征日本患者的临床特征。
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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
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The importance of renal impairment in the natural history of Bardet-Biedl syndrome.肾功能损害在巴德-比德尔综合征自然病程中的重要性。
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引用本文的文献

1
Clinical characteristics of a Japanese patient with Bardet-Biedl syndrome caused by BBS10 mutations.一名由BBS10基因突变引起的巴德-比德尔综合征日本患者的临床特征。
Jpn J Ophthalmol. 2018 Jul;62(4):458-466. doi: 10.1007/s10384-018-0591-8. Epub 2018 Apr 17.
2
The First Nationwide Survey and Genetic Analyses of Bardet-Biedl Syndrome in Japan.日本巴德-比埃尔综合征的首次全国性调查及基因分析
PLoS One. 2015 Sep 1;10(9):e0136317. doi: 10.1371/journal.pone.0136317. eCollection 2015.

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.
2
Mechanistic insights into Bardet-Biedl syndrome, a model ciliopathy.对巴德-比德尔综合征(一种典型的纤毛病)的机制性见解。
J Clin Invest. 2009 Mar;119(3):428-37. doi: 10.1172/JCI37041. Epub 2009 Mar 2.
3
Treatable fluctuating mental impairment in a patient with Bardet-Biedl syndrome.一名患有巴德-比德尔综合征的患者出现可治疗的波动性精神障碍。
Clin Neurol Neurosurg. 2009 Jan;111(1):102-4. doi: 10.1016/j.clineuro.2008.08.008. Epub 2008 Oct 19.
4
New criteria for improved diagnosis of Bardet-Biedl syndrome: results of a population survey.改善巴德-比德尔综合征诊断的新标准:一项人群调查结果
J Med Genet. 1999 Jun;36(6):437-46.
5
The importance of microalbuminuria as an indicator of incipient diabetic nephropathy: therapeutic implications.微量白蛋白尿作为早期糖尿病肾病指标的重要性:治疗意义
Ann Med. 1997 Oct;29(5):439-45. doi: 10.3109/07853899708999374.
6
Renal histopathology of Laurence-Moon-Biedl syndrome: tubulointerstitial nephritis without specific glomerular changes.劳伦斯-穆恩-比德尔综合征的肾脏组织病理学:肾小管间质性肾炎,无特异性肾小球改变。
Nephron. 1988;49(4):337-8. doi: 10.1159/000185089.
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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.
8
The cardinal manifestations of Bardet-Biedl syndrome, a form of Laurence-Moon-Biedl syndrome.巴德-比德尔综合征(一种劳伦斯-穆恩-比德尔综合征)的主要表现。
N Engl J Med. 1989 Oct 12;321(15):1002-9. doi: 10.1056/NEJM198910123211503.
9
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.

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.

DOI:10.4137/CCRep.S6622
PMID:21769262
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC3096431/
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。