Matteo Bramuzzo1, Paolo Lionetti2, Erasmo Miele3, Claudio Romano4, Serena Arrigo5,6, Sabrina Cardile7, Giovanni Di Nardo8, Maria Teresa Illiceto9, Maria Pastore10, Enrico Felici11, Maurizio Fuoti12, Claudia Banzato13, Michele Citrano14, Mauro Congia15, Lorenzo Norsa16, Elena Pozzi17, Giovanna Zuin18, Anna Agrusti19, Martina Bianconi20, Claudia Grieco3, Fabiola Giudici21,22, Marina Aloi23, Patrizia Alvisi24. 1. Gastroenterology, Digestive Endoscopy and Nutrition Unit, Institute for Maternal and Child Health, IRCCS "Burlo Garofolo," Trieste, Italy. 2. Department NEUROFARBA, University of Florence, Meyer Children's Hospital, Florence, Italy. 3. Department of Translational Medical Sciences, Section of Pediatrics, University of Naples "Federico II," Naples, Italy. 4. Unit of Pediatric Gastroenterology and Cystic Fibrosis, Department of Human Pathology in Adulthood and Childhood "G. Barresi," University of Messina, Messina, Italy. 5. Pediatric Gastroenterology and Endoscopy Unit, Institute "Giannina Gaslini," Genoa, Italy. 6. Department of Pediatrics, "F. Del Ponte" Hospital, University of Insubria, Varese, Italy. 7. Department of Hepatology, Gastroenterology and Nutrition, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy. 8. NESMOS Department, Faculty of Medicine and Psychology, Sapienza University of Rome, Sant'Andrea University Hospital, Rome, Italy. 9. Pediatric Gastroenterology and Endoscopic Unit, Department of Pediatrics, "Santo Spirito" Hospital, Pescara, Italy. 10. Pediatric Department, "Casa Sollievo della Sofferenza" Hospital, IRCCS, San Giovanni Rotondo, Italy. 11. Pediatric and Pediatric Emergency Unit, "Umberto Bosio" Center for Digestive Diseases, The Children Hospital, AO SS Antonio e Biagio e Cesare Arrigo, Alessandria, Italy. 12. Pediatric Gastroenterology and Endoscopy Unit Children's Hospital, ASST Spedali Civili, Brescia, Italy. 13. Department of Surgical Sciences, Dentistry, Gynecology and Pediatrics, Pediatric Division, University of Verona, Verona, Italy. 14. Department of Pediatrics, "Ospedali Riuniti Villa Santa Sofia-Cervello," Palermo, Italy. 15. Pediatric Clinic and Rare Diseases, Microcitemic Pediatric Hospital Antonio Cao, Azienda Ospedaliera Brotzu, Cagliari, Italy. 16. Pediatric Hepatology Gastroenterology and Transplantation, Papa Giovanni XXIII Hospital, Bergamo, Italy. 17. Department of Pediatrics, Vittore Buzzi Children's Hospital, University of Milan, Milan, Italy. 18. Pediatric Department, University of Milano Bicocca, FMBBM, San Gerardo Hospital, Monza, Italy. 19. Department of Medicine, Surgery, and Health Sciences, University of Trieste, Trieste, Italy. 20. Department of Health Sciences, University of Florence, Meyer children's Hospital, Florence, Italy. 21. Biostatistics Unit, Department of Medicine, Surgery and Health Sciences, Cattinara Hospital, University of Trieste, Trieste, Italy. 22. Unit of Biostatistics, Epidemiology and Public Health, Department of Cardiac, Thoracic, Vascular Sciences and Public Health, University of Padua, Padua, Italy. 23. Women's and Children's Health Department, Pediatric Gastroenterology and Hepatology Unit, Sapienza University of Rome, Rome, Italy. 24. Pediatric Gastroenterology Unit, Maggiore Hospital, Bologna, Italy.
Abstract
BACKGROUND: Adult patients with both inflammatory bowel disease (IBD) and celiac disease (CeD) have peculiar phenotypic features. This study aimed at describing the characteristics and natural history of children with both IBD and CeD. METHODS: This was a case-control study based on a national registry. Cases included children diagnosed with both IBD and CeD. Two matched IBD controls without CeD, and 2 matched CeD controls were selected for each case. Inflammatory bowel disease phenotype and natural history, comprising growth and pubertal development, were compared between groups. RESULTS: Forty-nine (1.75%) patients with IBD and CeD were identified out of 2800 patients with IBD. Compared with patients with IBD alone, patients with IBD and CeD presented more frequently with autoimmune diseases (odds ratio, 2.81; 95% CI, 0.97-8.37; P = 0.04). Ileocolonic localization (46.1% vs 73.1%), treatment with azathioprine (46.2% vs 71.2%), and anti-TNF biologics (46.2% vs 69.2%) were less common in patients with Crohn's disease and CeD than in patients with Crohn's disease alone. Patients with ulcerative colitis and CeD had an increased risk of colectomy despite similar medical treatments compared with patients with ulcerative colitis alone (13.0% vs 0%). Pubertal delay was more common in patients with IBD and CeD compared with patients with IBD alone (14.9% vs 3.2%; odds artio, 5.24; 95% CI, 1.13-33.0; P = 0.02) and CeD alone (14.9% vs 1.1%; P = 0.002). CONCLUSIONS: Children with IBD and CeD may have peculiar features with a higher risk for autoimmune diseases, colectomy, and pubertal delay compared with IBD alone.
BACKGROUND: Adult patients with both inflammatory bowel disease (IBD) and celiac disease (CeD) have peculiar phenotypic features. This study aimed at describing the characteristics and natural history of children with both IBD and CeD. METHODS: This was a case-control study based on a national registry. Cases included children diagnosed with both IBD and CeD. Two matched IBD controls without CeD, and 2 matched CeD controls were selected for each case. Inflammatory bowel disease phenotype and natural history, comprising growth and pubertal development, were compared between groups. RESULTS: Forty-nine (1.75%) patients with IBD and CeD were identified out of 2800 patients with IBD. Compared with patients with IBD alone, patients with IBD and CeD presented more frequently with autoimmune diseases (odds ratio, 2.81; 95% CI, 0.97-8.37; P = 0.04). Ileocolonic localization (46.1% vs 73.1%), treatment with azathioprine (46.2% vs 71.2%), and anti-TNF biologics (46.2% vs 69.2%) were less common in patients with Crohn's disease and CeD than in patients with Crohn's disease alone. Patients with ulcerative colitis and CeD had an increased risk of colectomy despite similar medical treatments compared with patients with ulcerative colitis alone (13.0% vs 0%). Pubertal delay was more common in patients with IBD and CeD compared with patients with IBD alone (14.9% vs 3.2%; odds artio, 5.24; 95% CI, 1.13-33.0; P = 0.02) and CeD alone (14.9% vs 1.1%; P = 0.002). CONCLUSIONS: Children with IBD and CeD may have peculiar features with a higher risk for autoimmune diseases, colectomy, and pubertal delay compared with IBD alone.