Razonable R R, Pulido J S, Deziel P J, Dev S, Salomão D R, Walker R C
Division of Infectious Diseases, Mayo Clinic College of Medicine, Rochester, Minnesota 55905, USA.
Transpl Infect Dis. 2008 Dec;10(6):413-8. doi: 10.1111/j.1399-3062.2008.00322.x. Epub 2008 Jun 18.
Whipple's disease is a very rare chronic multisystemic bacterial disease characterized by diarrhea, malabsorption, fever, and polyarthritis. Ocular manifestations occur very rarely. Previous reports have suggested that the use of immunosuppressive drugs appears to accelerate or exacerbate the clinical course of Whipple's disease; however, the illness has yet to be reported in the setting of transplantation. Herein, we describe what we believe is the first reported case of Whipple's disease after transplantation. The patient is a 51-year-old woman who developed progressive visual floaters and blurring of vision 30 years after living-related kidney transplantation for an autosomal-dominant polycystic kidney disease. Her allograft was functioning well on maintenance immunosuppressive therapy with azathioprine and prednisone when she developed visual abnormalities. Transient weight loss, gastrointestinal symptoms, and migratory polyarthralgia predated the onset of ocular disease by several years. The diagnosis of Whipple's bilateral vitreitis and chorioretinitis was confirmed by polymerase chain reaction analysis demonstrating Tropheryma whipplei nucleic acid in vitreous fluid and peripheral blood sample as well as by demonstration of the bacilli by cytopathology. Intraocular vancomycin, intravenous ceftriaxone, and prolonged course of oral trimethoprim-sulfamethoxazole therapy led to clinical improvement and recovery of visual acuity.
惠普尔病是一种非常罕见的慢性多系统细菌性疾病,其特征为腹泻、吸收不良、发热和多关节炎。眼部表现极为罕见。既往报告提示,使用免疫抑制药物似乎会加速或加重惠普尔病的临床病程;然而,尚未有在移植背景下发生该病的报道。在此,我们描述了我们认为是首例移植后惠普尔病的报告病例。该患者为一名51岁女性,在因常染色体显性多囊肾病接受亲属活体肾移植30年后,出现渐进性视觉飞蚊症和视力模糊。当她出现视觉异常时,其移植肾在使用硫唑嘌呤和泼尼松进行维持性免疫抑制治疗下功能良好。短暂体重减轻、胃肠道症状和游走性多关节痛在眼部疾病发作前数年就已出现。通过聚合酶链反应分析在玻璃体液和外周血样本中证实存在惠普尔嗜组织菌核酸,并通过细胞病理学证实存在杆菌,从而确诊为惠普尔双侧玻璃体炎和脉络膜视网膜炎。眼内注射万古霉素、静脉注射头孢曲松以及长时间口服甲氧苄啶 - 磺胺甲恶唑治疗导致临床症状改善和视力恢复。