Tokunaga Mikiko, Saito Kazuyoshi, Nakatsuka Keisuke, Nakayamada Shingo, Nakano Kazuhisa, Tsujimura Shizuyo, Ota Toshiyuki, Tanaka Yoshiya
The First Department of Internal Medicine, University of Occupational and Environmental Hearth, Japan, School of Medicine.
Nihon Rinsho Meneki Gakkai Kaishi. 2004 Oct;27(5):338-44. doi: 10.2177/jsci.27.338.
A 18-year-old female had low grade fever, butterfly rush, proteinuria, leukocytopenia and hypocomplimentemia in 1988, and she was diagnosed as systemic lupus erythematosus (SLE) with lupus nephritis (WHOIIb). Treatments with prednisolone and mizoribine resulted in the remission for three years. In May 2001, she presented neurosis and polakisuria despite of the increase of prednisolone to 20 mg/day. Finally, she admitted in our hospital because of manic and repressive state and disorientaion. A brain MRI revealed high intensity lesions in bilateral basal ganglia in T2 weighted images, and cerebrospinal fluid showed elevated protein and IFN-alpha (421 IU/ml). In addition, she manifested neurogenic bladder, muscle weakness and hyperactive deep tendon reflex of bilateral lower limbs due to both supranuclear disorder and hypesthesia under the Th 10 level. Spinal MRI revealed marked atrophy and high intensity signals at the middle to lower thoracic spinal cord in T2 weighted images, indicating complication of lupus myelitis as well as cerebral involvement. Although the symptoms of CNS lupus did not respond to prednisolone, twelve monthly cyclophosphamide pulse therapy (IV-CY) has resolved urinary disturbance, muscle weakness and sensory loss, along with the improvement of both cerebral and spinal MRI images. Lupus myelitis and neurogenic bladder are the rare, but very refractory manifestation among CNS involvement of SLE. We here propose IV-CY as an invaluable choice for the treatment of not only active lupus myelitis but also neurogenic bladder resisted for steroid.
一名18岁女性于1988年出现低热、蝶形红斑、蛋白尿、白细胞减少和补体降低,被诊断为系统性红斑狼疮(SLE)伴狼疮性肾炎(WHO IIb型)。泼尼松龙和咪唑立宾治疗后缓解了三年。2001年5月,尽管泼尼松龙增至20mg/天,她仍出现神经症和多尿。最后,她因躁狂和抑郁状态以及定向障碍入住我院。脑部MRI在T2加权像上显示双侧基底节高信号病灶,脑脊液显示蛋白升高和干扰素α(421IU/ml)升高。此外,由于核上性障碍和T10水平以下感觉减退,她出现神经源性膀胱、肌肉无力和双侧下肢深腱反射亢进。脊髓MRI在T2加权像上显示胸段脊髓中下部明显萎缩和高信号,提示狼疮性脊髓炎合并脑部受累。尽管中枢神经系统狼疮症状对泼尼松龙无反应,但十二个月的环磷酰胺脉冲治疗(静脉注射环磷酰胺)已解决了排尿障碍、肌肉无力和感觉丧失,同时脑部和脊髓MRI图像也有所改善。狼疮性脊髓炎和神经源性膀胱是系统性红斑狼疮中枢神经系统受累中罕见但非常难治的表现。我们在此提出静脉注射环磷酰胺不仅是治疗活动性狼疮性脊髓炎的宝贵选择,也是治疗对类固醇耐药的神经源性膀胱的宝贵选择。