Ouyang Z M, Ma J D, Yang Z H, Mo Y Q, Zou Y W, Dai L
Department of Rheumatology, Sun Yat-sen Memorial Hospital, Sun Yat-sen University, Guangzhou 510120, China.
Departments of Radiology, Sun Yat-Sen Memorial Hospital, Sun Yat-sen University, Guangzhou 510120, China.
Zhonghua Nei Ke Za Zhi. 2023 Feb 1;62(2):182-187. doi: 10.3760/cma.j.cn112138-20220403-00245.
A 58-year-old male patient with angioimmunoblastic T-cell lymphoma developed a rash and skin tightness on the face, limbs, and trunk together with joint stiffness and dysfunction after 6 months of treatment with the programmed cell death protein-1 inhibitor camrelizumab. Laboratory tests revealed progressive eosinophilia over 6 months, with the eosinophil count increasing from 0.07×10/L to 3.3×10/L. Magnetic resonance imaging showed thickened skin of both forearms, while T-weighted imaging showed markedly increased signal intensity within the myofascia. Skin biopsy of the right forearm showed thickened and fibrosed fascia and infiltration of inflammatory cells, including lymphocytes, plasma cells, and eosinophils. The patient was diagnosed with immune checkpoint inhibitor (ICI)-induced eosinophilic fasciitis (EF). After beginning treatment with methylprednisolone (40 mg daily), methotrexate (10 mg/week), and baricitinib (4 mg daily), his symptoms of skin tightness and joint dysfunction significantly improved within 1 month, and his peripheral blood eosinophil count decreased to 0.17×10/L. ICI-induced EF is a rare immune-related adverse reaction. To date, only 20 cases have been reported in published foreign literature, and their clinical characteristics are summarized here. The time from ICI treatment to EF was 12 (8,15) months, and the main clinical manifestations included skin involvement (=19), joint dysfunction (=11), myalgia/muscle weakness (=9), and peripheral eosinophilia (=16). After treatment, the clinical symptoms of EF improved in 17 patients, and eosinophil counts returned to normal after 3 (1,8) months. EF is a dysfunctional adverse response to ICI therapy. Tumor patients undergoing immunotherapy should be monitored for symptoms of EF. Early treatment is essential for preventing complications.
一名58岁的血管免疫母细胞性T细胞淋巴瘤男性患者,在接受程序性细胞死亡蛋白1抑制剂卡瑞利珠单抗治疗6个月后,面部、四肢和躯干出现皮疹、皮肤紧绷,同时伴有关节僵硬和功能障碍。实验室检查显示,6个月来嗜酸性粒细胞持续增多,嗜酸性粒细胞计数从0.07×10⁹/L增至3.3×10⁹/L。磁共振成像显示双前臂皮肤增厚,T2加权成像显示肌筋膜内信号强度明显增加。右前臂皮肤活检显示筋膜增厚、纤维化,并有淋巴细胞、浆细胞和嗜酸性粒细胞等炎性细胞浸润。该患者被诊断为免疫检查点抑制剂(ICI)诱导的嗜酸性筋膜炎(EF)。在开始使用甲泼尼龙(每日40mg)、甲氨蝶呤(每周10mg)和巴瑞替尼(每日4mg)治疗后,其皮肤紧绷和关节功能障碍症状在1个月内明显改善,外周血嗜酸性粒细胞计数降至0.17×10⁹/L。ICI诱导的EF是一种罕见的免疫相关不良反应。迄今为止,国外已发表的文献中仅报道了20例,现将其临床特征总结如下。从ICI治疗到发生EF的时间为12(8,15)个月,主要临床表现包括皮肤受累(n=19)、关节功能障碍(n=11)、肌痛/肌肉无力(n=9)和外周嗜酸性粒细胞增多(n=16)。治疗后,17例患者的EF临床症状得到改善,嗜酸性粒细胞计数在3(1,8)个月后恢复正常。EF是ICI治疗的一种功能失调性不良反应。接受免疫治疗的肿瘤患者应监测EF症状。早期治疗对于预防并发症至关重要。