Sonomoto Koshiro, Miyamura Tomaya, Watanabe Hideyuki, Takahama Soichiro, Nakamura Masataka, Ando Hitoshi, Minami Rumi, Yamamoto Masahiro, Saito Tomoko, Imayama Shuhei, Hosokawa Chisato, Suematsu Eiichi
Department of Internal Medicine, National Hospital Organization, Kyushu Medical Center.
Nihon Rinsho Meneki Gakkai Kaishi. 2008 Apr;31(2):119-23. doi: 10.2177/jsci.31.119.
A 47-year-old man was admitted to our hospital in January, 2006 because of a huge cutaneous ulcer in his lower limb. He was diagnosed with polyarteritis nodosa due to the cutaneous ulcer, mononeuritis multiplex, muscular pain, elevated serum CRP level and from histological findings of a skin biopsy. He was initially treated with 60 mg/day of prednisolone, followed by 1000 mg/day of intravenous cyclophosphamide (IVCY) therapy. In June, skin grafting to the cutaneous ulcer was carried out, although the graft did not survive. He revealed therapy-resistance to high dose corticosteroid and IVCY therapy, and so was treated with intravenous high dose immunoglobulin therapy. Serum CRP level then decreased and in October skin grafting was once again undertaken, this time the graft successfully survived. In December, serum CRP level increased again and cutaneous ulcer relapsed, thus he was treated with leukocyte apheresis therapy, although it was ineffective. In February 2007, he subsequently received rituximab (375 mg/m(2)/week x 3). Then, serum CRP level decreased rapidly, and cutaneous ulcer also improved. Recently the efficacy of rituximab against rheumatoid arthritis, systemic lupus erythematosus, polymyositis/dermatomyositis and ANCA-associated vasculitis has been recognized. This case suggests that rituximab is also effective against corticosteroid-resistant polyarteritis nodosa.
一名47岁男性因下肢巨大皮肤溃疡于2006年1月入住我院。根据皮肤溃疡、多发性单神经炎、肌肉疼痛、血清CRP水平升高以及皮肤活检的组织学结果,他被诊断为结节性多动脉炎。他最初接受泼尼松龙60mg/天治疗,随后接受环磷酰胺1000mg/天静脉注射(IVCY)治疗。6月,对皮肤溃疡进行了植皮手术,但移植皮片未存活。他对高剂量皮质类固醇和IVCY治疗显示出治疗抵抗,因此接受了静脉注射高剂量免疫球蛋白治疗。血清CRP水平随后下降,10月再次进行植皮手术,这次移植皮片成功存活。12月,血清CRP水平再次升高,皮肤溃疡复发,因此他接受了白细胞去除术治疗,但无效。2007年2月,他随后接受了利妥昔单抗治疗(375mg/m²/周×3)。然后,血清CRP水平迅速下降,皮肤溃疡也有所改善。最近,利妥昔单抗对类风湿关节炎、系统性红斑狼疮、多发性肌炎/皮肌炎和ANCA相关血管炎的疗效已得到认可。该病例表明,利妥昔单抗对皮质类固醇抵抗的结节性多动脉炎也有效。