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A European Survey on Digestive Perianastomotic Ulcerations, a Rare Crohn-like Disorder Occurring in Children and Young Adults

Authors :
Christos Tzivinikos
Ibrahim Shamasneh
Marina Aloi
Patrizia Alvisi
Erasmo Miele
Rémi Duclaux-Loras
Jérôme Viala
Stéphanie Willot
Rosa Lima
Claire Dupont-Lucas
Mario Mašić
Julie Lemale
Daniela Prlenda-Touilleux
J. Languepin
Sanja Kolaček
Chrystèle Madre
Alexandre Fabre
Kaija-Leena Kolho
Charlotte Bergoin
Sibylle Koletzko
Jean-Pierre Hugot
Raphaël Enaud
Christine Martinez-Vinson
Maria Nachury
Alexis Mosca
Annecarin Brueckner
Emmanuelle Dugelay
Hôpital Robert Debré
University of Zagreb
Centre Hospitalier Universitaire de Saint-Etienne [CHU Saint-Etienne] (CHU ST-E)
Dr von Hauner Children's Hospital [Munich, Germany]
Ludwig-Maximilians-Universität München (LMU)
Service de pédiatrie multidisciplinaire [Hôpital de la Timone Enfants - APHM]
Hôpital de la Timone [CHU - APHM] (TIMONE)
Marseille medical genetics - Centre de génétique médicale de Marseille (MMG)
Aix Marseille Université (AMU)-Institut National de la Santé et de la Recherche Médicale (INSERM)
Centre de recherche sur l'Inflammation (CRI (UMR_S_1149 / ERL_8252 / U1149))
Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS)-Université Paris Cité (UPCité)
Centro Hospitalar do Porto
CHU Bordeaux [Bordeaux]
Hôpital Trousseau
Centre Hospitalier Régional Universitaire de Tours (CHRU Tours)
Helsingin yliopisto = Helsingfors universitet = University of Helsinki
Geriatrics Unit [Pierre-Bénite]
Université de Lyon-Centre Hospitalier Lyon Sud [CHU - HCL] (CHLS)
Hospices Civils de Lyon (HCL)-Hospices Civils de Lyon (HCL)
Ospedale Maggiore Carlo Alberto Pizzardi di Bologna
Nutrition, Inflammation et axe Microbiote-Intestin-Cerveau (ADEN)
Université de Rouen Normandie (UNIROUEN)
Normandie Université (NU)-Normandie Université (NU)-Institute for Research and Innovation in Biomedicine (IRIB)
Normandie Université (NU)-Normandie Université (NU)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS)-Université de Rouen Normandie (UNIROUEN)
Normandie Université (NU)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS)-Institut National de la Santé et de la Recherche Médicale (INSERM)
University of Naples Federico II = Università degli studi di Napoli Federico II
Hospices Civils de Lyon (HCL)
Institute for Translational Research in Inflammation - U 1286 (INFINITE (Ex-Liric))
Institut National de la Santé et de la Recherche Médicale (INSERM)-Université de Lille-Centre Hospitalier Régional Universitaire [Lille] (CHRU Lille)
CHU Limoges
Service de Neuropédiatrie et Handicaps, Hôpital Gatien de Clocheville, CHU Tours
Université de Caen Normandie (UNICAEN)
Normandie Université (NU)
Institute for Research and Innovation in Biomedicine (IRIB)
Normandie Université (NU)-Normandie Université (NU)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS)
Service de Pédiatrie Médicale [Caen]
Normandie Université (NU)-Normandie Université (NU)-CHU Caen
Normandie Université (NU)-Tumorothèque de Caen Basse-Normandie (TCBN)-Tumorothèque de Caen Basse-Normandie (TCBN)
Al Jalila Children's Specialty Hospital
Medical Genetics Unit, Shaare Zedek Medical Center
Hevrew University Medical School
Clinicum
Children's Hospital
HUS Children and Adolescents
Madre, Chrystele
Mašić, Mario
Prlenda-Touilleux, Daniela
Brueckner, Annecarin
Koletzko, Sibylle
Fabre, Alexandre
Viala, Jérome
Lima, Rosa
Enaud, Raphael
Lemale, Julie
Kolho, Kaija-Leena
Bergoin, Charlotte
Martinez-Vinson, Christine
Dugelay, Emmanuelle
Alvisi, Patrizia
Aloi, Marina
Miele, Erasmo
Duclaux-Loras, Remi
Nachury, Maria
Languepin, Jane
Willot, Stephanie
Dupont-Lucas, Claire
Mosca, Alexi
Tzivinikos, Christo
Shamasneh, Ibrahim
Kolaček, Sanja
Hugot, Jean-Pierre
Source :
Journal of Pediatric Gastroenterology and Nutrition, Journal of Pediatric Gastroenterology and Nutrition, 2021, 73 (3), pp.333-337. ⟨10.1097/MPG.0000000000003200⟩
Publication Year :
2021
Publisher :
HAL CCSD, 2021.

Abstract

International audience; Objectives: Digestive perianastomotic ulcerations (DPAU) resembling Crohn disease lesions are long-term complications of intestinal resections, occurring in children and young adults. They are known to be uncommon, severe and difficult to treat.Methods: In the absence of recommendations, we performed a large European survey among the members of the ESPGHAN working group on inflammatory bowel disease (IBD) in order to collect the experience of expert pediatric gastroenterologists on DPAU.Results: Fifty-one patients (29 boys and 22 girls) were identified from 19 centers in 8 countries. Most patients were followed after necrotizing enterocolitis (n = 20) or Hirschsprung disease (n = 11). The anastomosis was performed at a median age (interquartile range) of 6 [1–23] months, and first symptoms occurred 39 [22–106] months after surgery. Anemia was the most prevalent symptom followed by diarrhea, abdominal pain, bloating, and failure to thrive. Hypoalbuminemia, elevated CRP, and fecal calprotectin were common. Deep ulcerations were found in 59% of patients usually proximally to the anastomosis (68%). During a median follow-up of 40 [19–67] months, treatments reported to be the most effective included exclusive enteral nutrition (31/35, 88%), redo anastomosis (18/22, 82%), and alternate antibiotic treatment (37/64, 58%).Conclusions: Unfortunately, persistence of symptoms, failure to thrive, and abnormal laboratory tests at last follow-up in most of patients show the burden of DPAU lacking optimal therapy and incomplete understanding of the pathophysiology.

Details

Language :
English
ISSN :
02772116
Database :
OpenAIRE
Journal :
Journal of Pediatric Gastroenterology and Nutrition, Journal of Pediatric Gastroenterology and Nutrition, 2021, 73 (3), pp.333-337. ⟨10.1097/MPG.0000000000003200⟩
Accession number :
edsair.doi.dedup.....95c4736ccad355f64afe331d90870a03