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ANGIOEDEMA ASSOCIATED WITH CROHN'S DISEASE: RESPONSE TO BIOLOGICS
V Huang, F HabalUniversity Health Network, University of Toronto, Toronto, ON
Aims: To present the first known case report of chronic idiopathic urticaria with angioedema coexistent with Crohn's disease that was successfully treated with anti-TNF-alpha agents, and to propose a hypothesis of the common pathophysiology of angioedema and Crohn's disease.
Methods: A case review of a patient with Crohn's disease and angioedema is presented. A literature review was conducted on angioedema in inflammatory bowel diseases and a hypothesis generated.
Results: A 46-year-old patient with terminal ileum Crohn's disease and ankylosing spondylitis presented with recurrent angioedema and urticaria. Investigations ruled out hereditary angioedema, and environmental or food allergen triggers. She was diagnosed with chronic idiopathic urticaria with angioedema, and was treated with a trial of IVIG immunotherapy, danazol, prednisone, and hydroxyzine. Due to ongoing bowel and arthritic complaints, she was started on Infliximab infusions and within two treatments, she had complete resolution of the angioedema and urticaria, as well as of the bowel and arthritic symptoms. Unfortunately she developed allergic reactions to the Infliximab and was switched to another anti-TNF alpha agent, adalumimab. Since then, she has had no further angioedema or urticaria, and her Crohn's disease has been quiescent.
Conclusions: Studies have shown that there is a strong autoimmune involvement in the pathophysiology of urticaria and angioedema. There are increases in pro-inflammatory cytokines, such as IL-1 beta, IL-12p70, TNF-alpha, IL-6, IL-10, and IL-17, in chronic idiopathic urticaria. Crohn's disease also has autoimmune involvement, and there is evidence for an altered cytokine milieu leading to mucosal inflammation. Recent studies have shown that T-cell production of certain cytokines play a strong role. One common thread in the pathophysiology of chronic idiopathic urticaria and Crohn's disease is the derangement in cytokine levels, in particular, IL-17, and TNF-alpha. The IL-17 cytokines are T-cell derived cytokines that stimulate various cells to secrete cytokines and chemokines, and therefore play an important role in many autoimmune diseases. It has been shown that IL-17A positive cells are increased in the inflamed mucosa of IBD patients, and IL-17F mRNA expression is elevated in the mucosa of Crohn's disease patients. Infliximab and Adalumimab are anti-TNF-alpha agents that block the inflammatory cascade. Given the similarity in cytokine derangements found in chronic idiopathic urticaria and in Crohn's disease, medications that target these cytokines, such as anti-TNF-alpha agents should be effective in both conditions. This case brings to attention the common pathophysiology between autoimmune diseases, and the need for further research looking into the changes in the cytokine milieu as potential targets for treatment.
Figure) Angioedema and urticaria of the face.