Damla Tuncer Budanur1 / Merve Şirin2 / Elif Sepet1 / Meral Ünür2 / Mine Güllüoğlu3 / Mustafa Serdar Cantez4 / Özlem Durmaz Uğurcan4
1Istanbul University, Faculty of Dentistry, Department of Pediatric Dentistry, Istanbul, Turkey
2Istanbul University, Faculty of Dentistry, Department of Oral & Maxillofacial Surgery, Istanbul, Turkey
3 Istanbul Faculty of Medicine, Department of Pathology, Division of Gastroenterology, Hepatology and Nutrition Istanbul, Turkey
4Istanbul Faculty of Medicine, Department of Pediatrics, Division of Gastroenterology, Hepatology and Nutrition Istanbul, Turkey
Background: Crohn’s disease (CD) and ulcerative colitis (UC) are the two major relapsing conditions of inflammatory bowel diseases. Case Report: A case of Crohn’s disease with orofacial manifestations in a 10 year old girl is described. She had suffered from fever, dysphagia, arthralgia, painful recurrent ulcers of the oral mucosa and swelling of the lower lip lasting over 6 weeks. Clinical examination and the punch biopsy from the buccal mucosa revealed major recurrent aphthous ulcerations. A partial regression and significant relief of lesions were achieved two weeks after the treatment, but the patient suffered from abdominal pain, irregular bowel movements, arthritis, multiple hyperplastic and swollen mucosal folds, after 3 months. The patient was referred to a pediatric gastroenterologist. Esophagogastroduodenoscopy showed pyloric ulcer formation. Abdominal ultrasound showed increased thickening of the ileal wall with multiple enlarged lympadenopathies in the periileal region. Colonoscopy images showed deep ulcers with surrounding erythema. The histopathological examination of biopsies from the terminal ileum and the colon showed basal plasmacytosis, minimal crypt distortions and aphthous ulcerations. The diagnosis of Orofacial Crohn’s disease was made. Exclusive enteral nutrition for 8 weeks, followed by azathiopurine treatment was started with an excellent clinical response on abdominal and oral symptoms. Conclusion: Diagnosis of the disease by dentists and other clinicians through the evaluation of oral clinical findings is very rare. Mucocutaneous and granulomatous lesions of the oral cavity should alert the clinician to pursue an underlying systemic cause. Early communication with a gastroenterologist can help early diagnosis of Crohn’s disease for better patient management and prognosis.
- Baumgart DC, Sandborn WJ. Inflammatory bowel disease: clinical aspects and established and evolving therapies. Lancet, 2007;369:1641-1657.[Web of Science]
- Cho JH, Weaver CT. The genetics of inflammatory bowel disease. Gastroenterol, 2007;133:1327-1339.[Web of Science]
- Baumgart DC, Carding SR. Inflammatory bowel disease: cause and immunobiology. Lancet, 2007;369:1627-1640.[Web of Science]
- Abraham C, Cho JH. Inflammatory bowel disease. N Engl J Med, 2009;361:2066-2078.
- Crohn’s Disease. National Digestive Diseases Information Clearinghouse (NDDIC) website. Available at: https://www.niddk.nih.gov/ Accessed: 12 June 2014.
- Baumgart DC, Sandborn WJ. Crohn’s disease. Lancet, 2012;380:1590-1605.
- Boraz RA. Oral manifestations of Crohn disease: Update of the literature and report of a case. J Dent Child, 1988;55:72-74.
- Itzkowitz SH. Inflammatory bowel disease and cancer. Gastroenterol Clin North Am, 1997;26:129-139.[Web of Science][Crossref]
- Lourenço SV, Hussein TP, Bologna SB, Sipahi AM, Nico MM. Oral manifestations of inflammatory bowel disease: a review based on the observation of six cases. J Eur Acad Dermatol Venereol, 2010;24:204-207.[Crossref][Web of Science]
- Zbar AP, Ben-Horin S, Beer-Gabel M, Eliakim R. Oral Crohn’s disease: is it a separable disease from orofacial granulomatosis? A review. J Crohns Colitis, 2012;6:135-142.[Crossref][Web of Science]
- Alawi F. Granulomatous diseases of the oral tissues: differential diagnosis and update Dent Clin North Am, 2005;49:203-221.
- Cosnes J. Tobacco and IBD: Relevance in the understanding of disease mechanisms and clinical practice. Best Pract Res Clin Gastroenterol, 2004;18:481-496.[Crossref]
- Daley DT, Armstrong JE. Oral manifestations of gastrointestinal diseases. Can J Gastroenterol, 2007;21:241-244.[Crossref][Web of Science]
- Franke A, McGovern DP, Barrett JC, Wang K, Radford- Smith GL, Ahmad T et al. Genome-wide meta-analysis increases to 71 the number of confirmed Crohn’s disease susceptibility loci. Nat Genet, 2010;42:1118-1125.[Web of Science][Crossref]
- Lu Z-K, Chen Z-R, Zhu J-Y, Xu Y, Hua X. Analysis of the association of single nucleotide polymorphisms of interleukin-23 receptor (IL-23R) and inflammatory bowel disease in a Chinese Han cohort. Oncotarget, 2016;7:67851-67856.[Web of Science]
- Liu JZ, Anderson CA. Genetic studies of Crohn’s disease: Past, present and future. Best Pract Res Clin Gastroenterol, 2014;28:373-386.[Crossref][Web of Science]
- Stavropoulos F, Katz J, Guelmann M, Bimstein E. Oral Ulcerations as a Sign of Crohn’s Disease in a Pediatric Patient: A Case Report. Pediatr Dent, 2004;26:355-358.
- Van Rheenen PF, Van de Vijver E, Fidler V. Faecal calprotectin for screening of patients with suspected inflammatory bowel disease: diagnostic meta-analysis. BMJ, 2010;341:c3369.
- O’Sullivan S, Gilmer JF, Medina C. Matrix Metalloproteinases in Inflammatory Bowel Disease: An Update. Mediat Inflamm, 2015;2015:964131.[Web of Science]
- Gurram B, Joeckel R, Stephens M. Nutrition in pediatric inflammatory bowel disease. Nutr Clin Practice, 2010;25:335-339.[Web of Science][Crossref]
- Wilkins T, Jarvis K, Patel J. Diagnosis and Management of Crohn’s Disease. Am Fam Physician, 2011;84:1365-1375.
- Ruemmele F, Veres G, Kolho KL, Levine A, Escher JC. Consensus Guidelines of ECCO/ ESPGHAN on the medical management of pediatric Crohn’s disease. J Crohns Colitis, 2014;8:1179-1207.[Crossref][Web of Science]
- Boirivant M, Cossu A. Inflammatory bowel disease. Oral Dis, 2012;18:1-15.[Web of Science][Crossref]
- Harty S, Fleming P, Rowland M, Crushell E, McDermott M, Drumm B et al. A prospective study of the oral manifestations of Crohn’s disease. Clin Gastroenterol Hepatol, 2005;3:886-891.[Crossref]
- Hussey S, Rowland M, Fleming P. Outcomes of oral Crohn’s disease in children. Can J Gastroenterol, 2007;21:5211.
Citation Information: Balkan Journal of Dental Medicine, ISSN (Online) 2335-0245, DOI: https://doi.org/10.1515/bjdm-2017-0021. Export Citation