Carra M

Courtet A

Lemale J

English

Archivio istituzionale della ricerca - Università di Ferrara

CLINICAL CHALLENGES AND IMAGES IN GIGeneralized Gingival Hyperplasia and Rectorrhagiain a 13-Year-Old BoyAlexandre Courtet,1 Julie Lemale,2 and Maria Clotilde Carra11Department of Periodontology, Odontology Unit, Rothschild Hospital (AP-HP) of Paris, and U.F.R. of Odontology, Universityof Paris, France; and 2Department of Nutrition and Gastroenterology Pediatric Unit, Trousseau Hospital (AP-HP), SorbonneUniversity, Paris, FranceQuestion: A 13-year-old boy of Arab origin was referred to the Department of Periodontology with a chief complaint ofgeneralized bleeding gums for 3 years, which had an impact on his daily quality of life. The young patient did not report anyparticular symptoms other than a chronic fatigue that hampered his sport activities. The patient’s mother mentioned 2episodes of perianal abscess that were surgically drained at the age of 6 months and 1 year and few episodes of rectorrhagiaduring childhood. She also noticed a year ago, swollen lips and mouth breathing.The patient’s body mass index is 21.5 kg/m2. The extraoral examination showed an asymmetric macrocheilia and mouthbreathing (Figure A). Periodontal examination highlighted a generalized gingival hyperplasia mainly in the vestibular areaextending beyond the mucogingival junction and covering a part of the clinical tooth crown (Figures B, C). The gingivapresented a granulomatous aspect associated with a dentomaxillary discrepancy. The plaque index and bleeding on probingindex (estimating gingival inflammation) were 42% and 48%, respectively. Previous sessions of scaling were performedonce a year by a general dentist, without any observable impact on the gingival aspect.Blood samples and gingival biopsies were taken. Normocytic nonregenerative anemia was diagnosed associated with aniron deficiency. Gingival biopsies revealed the presence of a parakeratinized epithelium with hyperplastic areas. The in-flammatory infiltrate was rich in plasma cells, noncaseating granulomas, and epithelioid cells (Figure D). The patient wasthen referred to the Pediatric Pulmonology and Gastroenterology Departments for further workup. Esophagogas-troduodenoscopy and colonoscopy were performed with biopsy at the level of the esophagus, stomach, duodenum andcolon, showing several inflammatory areas associated with numerous ulcerations.What is the most likely diagnosis?Look on page 2032 for the answer and see the Gastroenterology web site (www.gastrojournal.org) for moreinformation on submitting your favorite image to Clinical Challenges and Images in GI.Gastroenterology 2020;159:2030–2033CLINICAL CHALLENGES AND IMAGES IN GICorrespondenceAddress correspondence to: Maria Clotilde Carra, Department of Periodontology, Service of Odontology, Rothschild Hospital, AP-HP, Paris and University of ParisDiderot, U.F.R. of Odontology, 5, rue Garancière 75006 Paris, France. e-mail: mclotildecarra@gmail.com.Conflicts of interestThe authors disclose no conflicts.© 2020 by the AGA Institute0016-5085/$36.00https://doi.org/10.1053/j.gastro.2020.04.0702031CLINICAL CHALLENGES AND IMAGES IN GIAnswer to: Image 2 (Page 2030): Crohn’s DiseaseMelkersson-Rosenthal syndrome was excluded because of the lack of clinical signs. Absence of exophytic pustules on theoral mucosa ruled out pyostomatitis vegetans. Pulmonary radiologic examination (computed tomography scan) excludedsarcoidosis and tuberculosis. Biological tests for granulomatosis with polyangiitis proved positive for cytoplasmic anti-neutrophil cytoplasmic antibodies and negative for antisaccharomyces cerevisiae antibodies. The esophagogas-troduodenoscopy and colonoscopy reports were conclusive, leading to the diagnosis of Crohn’s disease (CD) localized at theesophagus, stomach, and colon and associated with gingival hyperplasia. Aphthoid ulcers were mainly observed in thetransverse and sigmoid colons (Figure E).The histopathological analyses of the gastro-intestinal biopsies showed the presence of noncaseating granulomas andepithelioid cells, with plasma cells and neutrophils in the inflammatory infiltrate (Figure F). After discussion in a multi-disciplinary meeting, immunosuppressive (azathioprine) and corticosteroid agents were prescribed associated with sup-plementations in vitamins B12 and B9 and iron to correct deficiencies. Simultaneously, periodontal treatment wasperformed by a specialized periodontist including, oral hygiene instruction with scaling and root planing.At the 3-month follow-up, a partial regression of CD is observed together with a significant reduction of the gingivalhyperplasia and inflammation (Figure G). The patient was shifted to a biological therapy with infliximab. Gingivectomy willbe eventually carried out to further decrease the gingival enlargement and promote optimal long-term plaque control, ifcomplete remission is not observed. A close monitoring of both the periodontal and intestinal manifestations is needed.Extraintestinal manifestations of CD (eg, arthritis, osteoporosis, and oral diseases) are common, especially in pediatricpatients, and may be present several years before CD diagnosis.1 It is estimated that up to 50% of CD patients present withoral manifestations.2Patients with CD are at increased risk of having impaired oral health and particularly periodontitis (odds ratio, 3.64;95% confidence interval, 2.33-5.67) compared with patients without CD.3 This susceptibility can be explained by thesimilarities in the pathogenicity between periodontitis and CD, particularly related to the inflammatory pathways anddysbiotic mechanisms in the microbial environment. This case report highlights the key role of dentists in the screening anddiagnosis of systemic diseases that might present with oral manifestations, and the necessity of a close collaboration be-tween oral health specialists and physicians.Keywords: Crohn’s disease; Periodontal disease; Extra-oral manifestations.2032CLINICAL CHALLENGES AND IMAGES IN GIReferences1. Vavricka SR, Schoepfer A, Scharl M, et al. Extraintestinal manifestations of inflammatory bowel disease. Inflamm BowelDis 2015;21:1982–1992.2. Harbord M, Annese V, Vavricka SR, et al. The first European evidence-based consensus on extra-intestinal manifestationsin inflammatory bowel disease. J Crohns Colitis 2016;10:239–254.3. She YY, Kong XB, Ge YP, et al. Periodontitis and inflammatory bowel disease: a meta-analysis. BMC Oral Health 2020;20:67.For submission instructions, please see the Gastroenterology website (www.gastrojournal.org).2033

Generalized gingival hyperplasia and rectorrhagia in a 13-year-old boy

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Generalized gingival hyperplasia and rectorrhagia in a 13-year-old boy

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