PMID- 37206602 OWN - NLM STAT- PubMed-not-MEDLINE LR - 20230522 IS - 2691-171X (Electronic) IS - 2691-171X (Linking) VI - 1 IP - 2 DP - 2020 Nov TI - Chronic Recurrent Multifocal Osteomyelitis in Pediatric Crohn Disease, A Paradoxical Effect to Antitumor Necrosis Factor Alpha. PG - e007 LID - 10.1097/PG9.0000000000000007 [doi] AB - Tumor necrosis factor-alpha (TNF-alpha) inhibitors have resulted in significant progress in the treatment of chronic inflammatory diseases. However, these therapies can lead to paradoxical immune-mediated inflammatory diseases with unknown physiopathology. For the first time, we report 3 cases of paradoxical chronic recurrent multifocal osteomyelitis after infliximab or adalimumab therapy during the course of Crohn disease. The patients complained of bone pain without joint involvement. At the time of diagnosis of paradoxical reaction, all patients were in remission due to anti-TNFalpha efficiency. Trough levels of anti-TNFalpha were in the expected range, and there were no anti-anti-TNFalpha antibodies. The duration of treatment was between 2 and 26 months. Other causes of CRMO were excluded. All patients recovered after discontinuation of infliximab (n = 2) or adalimumab (n = 1). The increasing use of these therapies leads to new descriptions of paradoxical effects, which clinicians should be aware of. CI - Copyright (c) 2020 The Author(s). Published by Wolters Kluwer on behalf of European Society for Pediatric Gastroenterology, Hepatology, and Nutrition and North American Society for Pediatric Gastroenterology, Hepatology, and Nutrition. FAU - Cordesse, Anne AU - Cordesse A AD - From the Service de Nutrition et Gastroenterologie Pediatriques, APHP, Hopital Trousseau, France. FAU - Ecochard-Dugelay, Emmanuelle AU - Ecochard-Dugelay E AD - Service de Gastroenterologie et Nutrition Pediatriques, APHP, Hopital Robert Debre, France. FAU - Melki, Isabelle AU - Melki I AD - Service de pediatrie generale, maladies infectieuses et medecine interne pediatrique, APHP, Hopital Robert Debre, France. FAU - Caseris, Marion AU - Caseris M AD - Service de pediatrie generale, maladies infectieuses et medecine interne pediatrique, APHP, Hopital Robert Debre, France. FAU - Belarbi, Nadia AU - Belarbi N AD - Service d'imagerie pediatrique et foetale, APHP, Hopital Robert Debre, France. FAU - Hugot, Jean-Pierre AU - Hugot JP AD - Service de Gastroenterologie et Nutrition Pediatriques, APHP, Hopital Robert Debre, France. AD - Universite Paris Diderot, Paris, France. AD - INSERM UMR 1123 (ECEVE), Paris, France. FAU - Viala, Jerome AU - Viala J AD - Service de Gastroenterologie et Nutrition Pediatriques, APHP, Hopital Robert Debre, France. AD - Universite Paris Diderot, Paris, France. AD - INSERM UMR 1123 (ECEVE), Paris, France. FAU - Martinez-Vinson, Christine AU - Martinez-Vinson C AD - Service de Gastroenterologie et Nutrition Pediatriques, APHP, Hopital Robert Debre, France. LA - eng PT - Case Reports DEP - 20200820 PL - United States TA - JPGN Rep JT - JPGN reports JID - 101773885 PMC - PMC10191537 OTO - NOTNLM OT - Crohn disease OT - anti-TNF-alpha OT - chronic recurrent multifocal osteomyelitis OT - paradoxical effect COIS- Disclosure: C.M.V. received honoraria, speaker's fees, and/or congress fees from AbbVie, MSD, Nestle, Biogen, Adacyte. J.P.H. received congress fees from MSD, Biogen. The remaining authors report no conflicts of interest. EDAT- 2020/08/20 00:00 MHDA- 2020/08/20 00:01 PMCR- 2020/08/20 CRDT- 2023/05/19 15:43 PHST- 2020/06/24 00:00 [received] PHST- 2020/07/30 00:00 [accepted] PHST- 2020/08/20 00:01 [medline] PHST- 2020/08/20 00:00 [pubmed] PHST- 2023/05/19 15:43 [entrez] PHST- 2020/08/20 00:00 [pmc-release] AID - 10.1097/PG9.0000000000000007 [doi] PST - epublish SO - JPGN Rep. 2020 Aug 20;1(2):e007. doi: 10.1097/PG9.0000000000000007. eCollection 2020 Nov.