Immune-mediated cutaneous leukocytoclastic vasculitis followed by a severe acute infusion reaction after infliximab rechallenge in Crohn’s disease: a case report and review of the literature

Infliximab is widely used for Crohn’s disease (CD). Despite its efficacy, it may be associated with immune-mediated adverse events, including cutaneous vasculitis and acute infusion reactions. The temporal association between these events is clinically important and should be interpreted with caution. A 53-year-old woman of North African descent with a 5-year history of active stricturing ileocolonic CD treated with azathioprine was referred for therapeutic escalation. Infliximab was started at 5 mg/kg in combination with azathioprine 2.3 mg/kg/day. One week after the second infusion, she developed non-blanching purpuric lesions on the face and lower limbs. Skin biopsy confirmed leukocytoclastic vasculitis. According to the French pharmacovigilance method, the intrinsic causality of infliximab was considered likely (I4) for cutaneous vasculitis. Two months later, infliximab was reintroduced with hydrocortisone hemisuccinate premedication. During infusion, the patient developed chest tightness, oxygen saturation decreased to 89%, blood pressure increased to 180/120 mmHg, and erythema of the extremities appeared, followed by petechial lesions. Infliximab was stopped immediately. Symptoms improved with oxygen therapy and antihistamines. The reaction was assessed as very likely related to infliximab (I5). Infliximab was permanently discontinued, and the patient was switched to adalimumab, with good tolerance and no recurrence during 18 months of follow-up. This case suggests a possible relationship between early cutaneous leukocytoclastic vasculitis and a later severe acute infusion reaction after infliximab rechallenge. The absence of trough infliximab level and anti-drug antibody testing remains the main limitation. In patients with biopsy-proven vasculitis during infliximab therapy, rechallenge should be considered only after careful risk assessment and when safer alternatives are not feasible.