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
Department of Neurology, Sakai Hospital Kinki University Faculty of Medicine.
Clin Med Insights Case Rep. 2011;4:17-20. doi: 10.4137/CCRep.S6622. Epub 2011 Mar 10.
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.
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。