Refractory multicentric reticulohistiocytosis treated by infliximab: two cases.
Refractory multicentric reticulohistiocytosis treated by infliximab: two cases.
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英夫利昔单抗治疗难治性多中心网状组织细胞增多症：二例。
DOI：
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发表时间：
2005
影响因子：
3.7
通讯作者：
A. Kahan
中科院分区：
文献类型：
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作者：
J. Sellam;C. Deslandre;F. Dubreuil;S. Arfi;A. Kahan
我们报告了英夫利昔单抗（一种单克隆抗tnfalpha抗体）在两例难治性皮肤和关节多中心网状组织细胞增多症（MRH）患者中的效果。一名37岁女性和一名53岁女性患有多发性关节炎，与MRH相关的面部皮疹和手上的结节性病变，对多种药物：carilysine，皮质类固醇，羟氯喹和细胞毒性药物均难治。英夫利昔单抗剂量为3mg /kg，然后增加到5mg /kg，与甲氨喋呤或硫唑嘌呤联合使用，对疾病的皮肤表现有效，但对多发性关节炎无效。改用依那西普没有改善第二例患者的多发性关节炎。一些数据表明，TNFalpha参与MRH，但根据我们的病例，抗TNFalpha治疗需要进一步评估难治性MRH患者。
We report the effect of infliximab, a monoclonal anti-TNFalpha antibody, in two patients with refractory cutaneous and articular multicentric reticulohistiocytosis (MRH). One 37-year-old woman and one 53-year-old woman with polyarthritis, facial rash and nodular lesions on the hands related to MRH were refractory to multiple agents: cariolysine, corticosteroids, hydroxychloroquine and cytotoxic agents. Infliximab at 3 mg/kg which was then increased to 5 mg/kg in combination with methotrexate or azathioprine was effective on cutaneous manifestations of the disease but not on polyarthritis. A switch to etanercept did not improve polyarthritis in the second patient. Some data suggest that TNFalpha is involved in MRH, but based on our cases anti-TNFalpha therapy needs further evaluation in patients with refractory MRH.