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 0.07×109/L to 3.3×109/L. Magnetic resonance imaging showed thickened skin of both forearms, while T2-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 0.17×109/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 (n=19), joint dysfunction (n=11), myalgia/muscle weakness (n=9), and peripheral eosinophilia (n=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.
患者男,58岁。诊断血管免疫母细胞T细胞性淋巴瘤,使用程序性细胞死亡蛋白-1抑制剂卡瑞利珠单抗治疗6个月后出现颜面、四肢及躯干皮疹伴皮肤变硬,双腕、肘和膝关节僵硬并功能受限,外周血嗜酸性粒细胞明显增多(0.07×109/L升至3.3×109/L),双前臂磁共振成像示皮下组织增厚,T2压脂序列(T2WI)示肌群间肌筋膜信号增高,增强后明显强化。右前臂皮肤活检病理示,筋膜层增厚及纤维化,淋巴细胞、浆细胞和嗜酸性粒细胞浸润。最终诊断免疫检查点抑制剂(ICI)相关嗜酸性筋膜炎(EF),予甲泼尼龙 40 mg/d+甲氨蝶呤10 mg/周+巴瑞替尼4 mg/d治疗1个月后,患者皮肤肿胀硬化较前明显减轻,关节功能改善,外周血嗜酸性粒细胞降至正常(0.17×109/L)。ICI相关EF属少见的免疫治疗相关不良反应,检索国外文献报道的20例EF患者,结合本例共21例患者,使用ICI至出现EF的中位时间12(8,15)个月,临床表现以皮肤受累(19例)、关节功能受限(11例)、肌痛/肌无力(9例)为主。16例患者外周血嗜酸性粒细胞增多,17例患者经3(1,8)个月的治疗后,临床症状改善,嗜酸性粒细胞恢复正常。提示EF是ICI的一种可致功能障碍的不良反应,接受肿瘤免疫治疗的患者应关注EF的相关症状,尽早治疗对预防长期并发症至关重要。.