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Successful treatment of ulcerative colitis with anakinra: a case report

Journal Volume 86 - 2023
Issue Fasc.4 - Case reports
Author(s) M. Truyens 1 2 3, L. Hoste 4 5, J. Geldof 1 3, A. Hoorens 6, F. Haerynck 4 5, D. Huis in ’t Veld 7, T. Lobatón 1 3
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PAGES 573-576
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DOI10.51821/86.4.11246
Affiliations:
(1) IBD Research Unit, Department of Internal Medicine and Pediatrics, Ghent University, Ghent, Belgium
(2) VIB Center for Inflammation Research, Ghent, Belgium
(3) Department of Gastroenterology, University Hospital Ghent, Ghent, Belgium
(4) Department of Pediatric Pulmonology, Infectious Diseases and Immunology, Ghent University Hospital, Ghent, Belgium
(5) Primary Immunodeficiency Research Lab, Center for Primary Immunodeficiency Ghent, Jeffrey Modell Diagnosis and Research Center, Ghent University, Ghent, Belgium
(6) Department of Pathology, Ghent University Hospital, Ghent, Belgium
(7) Department of Internal Medicine and Infectious Diseases, University Hospital, Ghent, Belgium

Currently the effect of IL-1 blockade on ulcerative colitis (UC) is still ambiguous. This case report describes a patient with UC who developed severe complications after an episode of azathioprineinduced pancytopenia including cytomegalovirus pneumonitis, hemophagocytic lymphohistiocytosis, and probable pulmonary aspergillosis. Imaging after the hospitalization revealed a severe disseminated chronic candidiasis and persisting inflammation was seen. Genetic testing revealed heterozygous variants in NOD2 and NLRP12, and cytokine testing showed an increase in IL-1Ra, IL-18, CXCL9, and CXCL10. Consequently an IL-1 mediated autoinflammatory syndrome was suspected. Simultaneously, the patient developed a corticosteroid dependent UC flare-up. Treatment with anakinra was initiated for the IL-1 mediated disease which quickly induced remission of both the inflammatory syndrome and the UC.

Keywords: Inflammatory bowel disease, IL-1, chronic disseminated candidiasis, azathioprine.

The authors declare that they have no conflict of interest.
© Acta Gastro-Enterologica Belgica.
PMID 38240554