Monica M Schmitt1, Elise M N Ferré1, Michelly Sampaio De Melo2, Megan A Cooper3, Martha M Quezado2, Theo Heller4, Michail S Lionakis1. 1. Fungal Pathogenesis Section, Laboratory of Clinical Immunology and Microbiology (LCIM), National Institute of Allergy & Infectious Diseases (NIAID), NIH, Bethesda, MD, USA. 2. Laboratory of Pathology, Center for Cancer Research, National Cancer Institute (NCI), NIH, Bethesda, MD, USA. 3. Department of Pediatrics, Division of Rheumatology/Immunology, Washington University School of Medicine, St. Louis, MO, USA. 4. Translational Hepatology Section, Liver Diseases Branch, National Institute of Diabetes and Digestive and Kidney Diseases (NIDDK), NIH, Bethesda, MD, USA.
Abstract
Introduction: Autoimmune polyendocrinopathy-candidiasis-ectodermal dystrophy (APECED) is a prototypic monogenic autoimmune disorder caused by AIRE deficiency-mediated impaired central immune tolerance. Although multiple endocrine and non-endocrine tissues are affected in APECED, the colon is an uncommon target of autoimmune attack. Mycophenolate is a potent immunomodulatory medication that is used to treat autoimmune manifestations in patients with APECED and other autoimmune diseases. Methods: We reviewed the clinical, laboratory, genetic, histological, and treatment data of mycophenolate-induced colitis in our cohort of 104 APECED patients. Discussion: Among 10 mycophenolate-treated APECED patients, four (40%) developed reversible biopsy-proven mycophenolate-induced colitis characterized by an inflammatory bowel disease-like and/or graft-versus-host disease-like histological pattern. Mycophenolate-induced colitis appears to be a common complication in patients with APECED for which clinicians should maintain a high index of suspicion.
Introduction: Autoimmune polyendocrinopathy-candidiasis-ectodermal dystrophy (APECED) is a prototypic monogenic autoimmune disorder caused by AIRE deficiency-mediated impaired central immune tolerance. Although multiple endocrine and non-endocrine tissues are affected in APECED, the colon is an uncommon target of autoimmune attack. Mycophenolate is a potent immunomodulatory medication that is used to treat autoimmune manifestations in patients with APECED and other autoimmune diseases. Methods: We reviewed the clinical, laboratory, genetic, histological, and treatment data of mycophenolate-induced colitis in our cohort of 104 APECED patients. Discussion: Among 10 mycophenolate-treated APECED patients, four (40%) developed reversible biopsy-proven mycophenolate-induced colitis characterized by an inflammatory bowel disease-like and/or graft-versus-host disease-like histological pattern. Mycophenolate-induced colitis appears to be a common complication in patients with APECED for which clinicians should maintain a high index of suspicion.
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