Yáñez V Jorge, Cisternas M Marcela, Saldías H Velia, Saldías P Fernando
Departamento de Enfermedades Respiratorias, Pontificia Universidad Católica de Chile, Santiago, Chile.
Rev Med Chil. 2009 Jan;137(1):88-93. Epub 2009 Apr 23.
Chronic organizing pneumonia (COP) has often been reported as a pulmonary manifestation of collagen vascular diseases, mainly rheumatoid arthritis, but the association of COP and dermatomyositis (DM) has rarely been documented. We report a 55 year-old woman with well-documented DM and a COP. She was refractory to steroids and two other immunosuppressive agents therapy (cyclophosphamide and azathioprine). Therefore, rituximab (2 x 1 g infusions) was used for treatment. During the following weeks her strength gradually increased while creatine kinase (CK), C reactive protein and erythrocyte sedimentation rate normalized. After 6 months, she had a relapse with increased muscle enzymes, fever and moderate muscle weakness. After a second course of rituximab (2 x 1 g infusions), the patient demonstrated a remarkable clinical response as indicated by an increase in muscle strength and moderate decline in creatine kinase levels. Lung abnormalities resolved significantly on high resolution chest CT scan. Thus, B-cell depletion therapy with rituximab used alone or in combination with other immunosuppressants may be a viable option in patients with polymyositis-dermatomyositis and pneumonia refractory to current therapies.
慢性机化性肺炎(COP)常被报道为胶原血管疾病的肺部表现,主要是类风湿关节炎,但COP与皮肌炎(DM)的关联鲜有文献记载。我们报告一例55岁患有确诊DM和COP的女性患者。她对类固醇及其他两种免疫抑制剂(环磷酰胺和硫唑嘌呤)治疗无效。因此,使用利妥昔单抗(2次1g静脉输注)进行治疗。在接下来的几周里,她的肌力逐渐增强,同时肌酸激酶(CK)、C反应蛋白和红细胞沉降率恢复正常。6个月后,她复发,肌肉酶升高、发热且有中度肌无力。在第二个疗程的利妥昔单抗(2次1g静脉输注)治疗后,患者出现显著临床反应,表现为肌力增加和肌酸激酶水平适度下降。高分辨率胸部CT扫描显示肺部异常明显改善。因此,对于对当前治疗无效的多发性肌炎-皮肌炎和肺炎患者,单独使用利妥昔单抗或与其他免疫抑制剂联合进行B细胞清除疗法可能是一种可行的选择。