El Rhaoussi Fatima Zahra, Boukhal Zineb, Haddad Fouad, Tahiri Mohamed, Hliwa Wafaa, Bellabah Ahmed, Wafaa Badre
Faculty of Medicine and Pharmacy, Hassan II University, Casablanca, MAR.
Department of Gastroenterology and Hepatology, Ibn Rochd University Hospital Center, Casablanca, MAR.
Cureus. 2024 Feb 10;16(2):e53953. doi: 10.7759/cureus.53953. eCollection 2024 Feb.
Erythromelalgia is a rare syndrome with a generally unknown etiology. Whether primary or secondary, this condition is characterized by paroxysmal episodes of erythema, pain, and heat in the extremities. We report two cases of erythromelalgia occurring after the initiation of treatment with infliximab. The first case involves a 38-year-old patient who had been followed since August 2022 for ileocolonic Crohn's disease classified as A2L3B3 according to the Montreal classification, which was resistant to treatment and required infliximab therapy. Two months after the first infusion of infliximab, the patient developed symptoms of erythromelalgia. After ruling out other potential causes through an etiological assessment and conducting a pharmacological investigation, infliximab was considered the most likely cause. Infliximab was discontinued, and symptomatic treatment was initiated, including vascular laser sessions. The patient showed significant clinical improvement. In the second case, a 16-year-old patient with ileocolonic Crohn's disease classified as A1L3B3 according to the Montreal classification was treated with ileocecal resection and received an infusion of infliximab. Sixteen days after the second infusion, she developed clinical symptoms of erythromelalgia. The etiological assessment was inconclusive. Due to a strong suspicion of erythromelalgia secondary to tumor necrosis factor (TNF) alpha inhibitor therapy, infliximab was replaced with ustekinumab. The patient also received symptomatic treatment, and her clinical condition improved, marked by the disappearance of pain.
红斑性肢痛症是一种病因通常不明的罕见综合征。无论原发性还是继发性，这种病症的特征是四肢出现红斑、疼痛和发热的阵发性发作。我们报告两例英夫利昔单抗治疗开始后发生的红斑性肢痛症病例。第一例涉及一名38岁患者，自2022年8月起因回结肠克罗恩病接受随访，根据蒙特利尔分类法分类为A2L3B3，对治疗耐药，需要英夫利昔单抗治疗。首次输注英夫利昔单抗两个月后，患者出现红斑性肢痛症症状。通过病因评估排除其他潜在原因并进行药物调查后，英夫利昔单抗被认为是最可能的病因。停用英夫利昔单抗并开始对症治疗，包括血管激光治疗。患者临床症状显著改善。第二例中，一名16岁患有回结肠克罗恩病的患者，根据蒙特利尔分类法分类为A1L3B3，接受了回盲部切除术并输注了英夫利昔单抗。第二次输注16天后，她出现了红斑性肢痛症的临床症状。病因评估尚无定论。由于强烈怀疑是肿瘤坏死因子（TNF）α抑制剂治疗继发的红斑性肢痛症，将英夫利昔单抗换成了优特克单抗。患者也接受了对症治疗，其临床状况得到改善，疼痛消失。
