Kikukawa Yoshitaka, Hata Hiroyuki, Ueda Mitsuharu, Yamashita Taro, Nasu Singo, Ide Kazuhiko, Ueno Shikiko, Ando Yukio, Mitsuya Hiroaki, Okuno Yutaka
Departments of Hematology, Rheumatology, and Infectious Disease, Kumamoto University Graduate School of Medicine, Japan.
Intern Med. 2016;55(18):2707-12. doi: 10.2169/internalmedicine.55.5815. Epub 2016 Sep 15.
A 70-year-old woman with Charcot-Marie-Tooth disease (CMT) suffered from nephrotic syndrome and a renal biopsy revealed non-AA amyloid depositions that contained immunoglobulin light chain λ. Her serum λ free LC was elevated to 80.8 mg/L and she was diagnosed with primary amyloid light-chain (AL) amyloidosis. She was subsequently treated with lenalidomide, cyclophosphamide, and dexamethasone (RCD). After 14 cycles of RCD, she achieved complete remission. Her serum albumin levels gradually normalized to 3.1 g/dL. No exacerbation of neurologic symptoms related to CMT was observed. Thus, RCD may be a well-tolerated and effective regimen for treating AL amyloidosis in patients with CMT disease.
一名患有夏科-马里-图斯病(CMT)的70岁女性患有肾病综合征,肾活检显示非AA型淀粉样蛋白沉积,其中含有免疫球蛋白轻链λ。她的血清λ游离轻链升高至80.8mg/L,被诊断为原发性淀粉样轻链(AL)淀粉样变性。随后她接受了来那度胺、环磷酰胺和地塞米松(RCD)治疗。经过14个周期的RCD治疗,她实现了完全缓解。她的血清白蛋白水平逐渐恢复正常至3.1g/dL。未观察到与CMT相关的神经症状加重。因此,RCD可能是治疗CMT病患者AL淀粉样变性的耐受性良好且有效的方案。