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Does Crohn's Disease with Concomitant Orofacial Granulomatosis Represent a Distinctive Disease Subtype?

Journal article
Authors Gita Gale
Gudmundur Vignir Sigurdsson
Sofia M Östman
P. Malmborg
K. Hogkil
Bengt Hasséus
Mats Jontell
Robert Saalman
Published in Inflammatory Bowel Diseases
Volume 22
Issue 5
Pages 1071-1077
ISSN 1078-0998
Publication year 2016
Published at Institute of Odontology
Institute of Clinical Sciences, Department of Pediatrics
Institute of Biomedicine, Department of Infectious Medicine
Pages 1071-1077
Language en
Keywords inflammatory bowel disease, mucosal pathology, pediatric gastroenterology, inflammatory-bowel-disease, northern stockholm county, oral, manifestations, phenotypic characteristics, onset, variants, children, cohort, susceptibility, metaanalysis, Gastroenterology & Hepatology
Subject categories Clinical Medicine


Background: Although orofacial granulomatosis (OFG) may present as a separate clinical entity, it often seems in conjunction with various systemic diseases, of which Crohn's disease (CD) is one of the most common. The aim of this study was to investigate whether CD with concomitant OFG represents a distinctive disease subtype. Methods: Twenty-one patients with CD and concomitant OFG (CD+OFG group) were included in the study. As the reference group, a cohort of 39 patients with CD but without OFG (CD-R group) was used. Demographic data and clinical characteristics were recorded at the time of diagnosis. The 2 groups were compared using multivariate analyses. Results: The percentage of patients with intestinal inflammation in the upper gastrointestinal tract was significantly higher in the CD+OFG group, as compared with the CD-R group (81% versus 33%; P < 0.001). Furthermore, ileocolonic inflammation was significantly more common in the CD+OFG patients (81% versus 46%; P = 0.013). In addition, perianal disease was more frequently observed in the CD+OFG group (48% versus 18%; P = 0.033). Significantly more patients showed evidence of granulomas in the primary endoscopy in the CD+OFG group than in the CD-R group (81% versus 38%; P = 0.003). Conclusion: The data from this study suggest that the presence of CD in conjunction with OFG represents a distinctive subphenotype of CD that is characterized by extensive inflammation, perianal disease, and pronounced granuloma formation in the intestine.

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