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

Chrystele Madre (1) , Mario Mašić (2) , Daniela Prlenda-Touilleux (3) , Annecarin Brueckner (4) , Sibylle Koletzko (4) , Alexandre Fabre (5, 6) , Jérome Viala (7) , Rosa Lima (8) , Raphael Enaud (9) , Julie Lemale (10) , Kaija-Leena Kolho (11) , Charlotte Bergoin (12) , Christine Martinez-Vinson (1) , Emmanuelle Dugelay (1) , Patrizia Alvisi (13) , Marina Aloi (14) , Erasmo Miele (15) , Remi Duclaux-Loras (16) , Maria Nachury (17) , Jane Languepin (18) , Stephanie Willot (19) , Claire Dupont-Lucas (20, 14, 21, 22) , Alexis Mosca (1) , Christos Tzivinikos (23) , Ibrahim Shamasneh (24) , Sanja Kolaček (2) , Jean-Pierre Hugot (1)

1 Hôpital Robert Debré
2 Zagreb University Medical School
3 CHU Saint-Etienne
4 Dr von Hauner Children's Hospital [Munich, Germany]
5 Service de pédiatrie multidisciplinaire [Hôpital de la Timone Enfants - APHM]
6 MMG - Marseille medical genetics - Centre de génétique médicale de Marseille
7 CRI (UMR_S_1149 / ERL_8252 / U1149) - Centre de recherche sur l'Inflammation
8 Centro Hospitalar do Porto
9 CHU Bordeaux [Bordeaux]
10 Hôpital Trousseau
11 Helsingin yliopisto = Helsingfors universitet = University of Helsinki
12 Geriatrics Unit [Pierre-Bénite]
13 Ospedale Maggiore Carlo Alberto Pizzardi di Bologna
14 ADEN - Nutrition, Inflammation et axe Microbiote-Intestin-Cerveau
15 University of Naples Federico II = Università degli studi di Napoli Federico II
16 HCL - Hospices Civils de Lyon
17 INFINITE (Ex-Liric) - Institute for Translational Research in Inflammation - U 1286
18 CHU Limoges
19 Service de Neuropédiatrie et Handicaps, Hôpital Gatien de Clocheville, CHU Tours
20 UNICAEN - Université de Caen Normandie
21 IRIB - Institute for Research and Innovation in Biomedicine
22 Service de Pédiatrie Médicale [Caen]
23 Al Jalila Children's Specialty Hospital
24 Medical Genetics Unit, Shaare Zedek Medical Center

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.

Domains: Life Sciences [q-bio]; Human health and pathology

Chrystele Madre, Mario Mašić, Daniela Prlenda-Touilleux, Annecarin Brueckner, Sibylle Koletzko, et al.. A European Survey on Digestive Perianastomotic Ulcerations, a Rare Crohn-like Disorder Occurring in Children and Young Adults. Journal of Pediatric Gastroenterology and Nutrition, 2021, 73 (3), pp.333-337. ⟨10.1097/MPG.0000000000003200⟩. ⟨hal-03662735⟩