Background: Eosinophilic enterocolitis (EEC) is an emerging distinct inflammatory bowel disease of unknown etiology. There are no published data on the effect of infliximab (IFX) or adalimumab (ADA) for the treatment of refractory cases.
Methods: A report of all pediatric cases with EEC treated with anti-tumor necrosis factor, identified after an open international call.
Results: We describe here the first 8 children with refractory EEC who were treated with IFX (75% boys; mean age at diagnosis 8.6 ± 4.03 [range 1.6–14 years]; mean age at IFX treatment 11.7 ± 4.4 [range 4.2–16 years]). Allergic and infectious causes of EEC were excluded in all cases. Rapid and complete clinical remission was documented in 6 (75%) children following the induction infusions: 3 (38%) with endoscopic remission, 2 (25%) with endoscopic improvement, and 1 unknown. Four of the 6 responders had secondary loss of response and were switched to ADA, 3 of whom with sustained remission using high doses. Overall, the 6 responders were followed for a median of 7 years (range 4–12; interquartile range 6.4–8.8 years) without evidence of developing Crohn disease or ulcerative colitis. The only case with macroscopic findings on endoscopy was a primary nonresponder.
Conclusions: IFX and ADA may be effective in cases of refractory idiopathic EEC; however, because this is an uncontrolled report, further prospective studies are warranted.
*Pediatric Gastroenterology Unit, Shaare Zedek Medical Center, Hebrew University of Jerusalem, Jerusalem, Israel
†Pediatric Gastroenterology, UMC Utrecht, The Netherlands
‡Pediatric Gastroenterology, Royal Hospital for Sick Children, Glasgow, UK
§Mercy Hospital, Kansas City, MO
||Division of Pediatric Gastroenterology, Hepatology, and Nutrition
¶Department of Pathology, The Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada.
Address correspondence and reprint requests to Dan Turner, MD, PhD, Pediatric Gastroenterology and Nutrition Unit, Shaare Zedek Medical Center, POB 3235, Jerusalem 91031, Israel (e-mail: firstname.lastname@example.org).
Received 21 August, 2012
Accepted 27 November, 2012
R.K.R. is supported by an NHS Research Scotland career fellowship award. A.M.M. is supported by an Early Researcher Award from the Ontario Ministry of Research and Innovation and a CDHNF/NASPGHAN George Ferry Young Investigator Development Award and funded by a Canadian Institute of Health Research Operating Grant (MOP119457).
R.K.R. received consultation fees, research grant, and honorarium from MSD, and Abbott; D.T. received consultation fees, a research grant, and an honorarium, from MSD, Janssen, and Abbott. The other authors report no conflicts of interest.