Ann Dermatol.  2010 May;22(2):203-205. 10.5021/ad.2010.22.2.203.

Orofacial Granulomatosis Associated with Crohn's Disease

Affiliations
  • 1Department of Dermatology, Ajou University School of Medicine, Suwon, Korea. eslee@ajou.ac.kr

Abstract

Orofacial granulomatosis (OFG) is a term used to describe swelling of the orofacial area, mainly in the lips, secondary to an underlying granulomatous inflammatory process. OFG has been reported in association with systemic conditions such as sarcoidosis and Crohn's disease (CD). OFG may precede gastrointestinal disease, such as CD, by several years and may be the only obvious focus of the disease. Herein, we report a patient with OFG and non-symptomatic ulcerations of the ileocecal valve. The patient received intralesional triamcinolone injections every 2 weeks. After 6 weeks, all oral lesions showed marked improvement. The favourable treatment response of this patient suggests that intralesional triamcinolone can be used as a treatment option for patients with CD that have oral lesions. In addition, patients presenting with OFG should be carefully evaluated for gastrointestinal signs and symptoms.

Keyword

Crohn's disease; Orofacial graulomatosis; Treatment

MeSH Terms

Crohn Disease
Gastrointestinal Diseases
Granulomatosis, Orofacial
Humans
Ileocecal Valve
Lip
Sarcoidosis
Triamcinolone
Ulcer
Triamcinolone

Figure

  • Fig. 1 (A) Marked swelling of upper and lower lips. (B, C) Cobble-stone like appearance of the buccal mucosa and sublingual space (asterisk=the site of biopsy). (D) Six weeks after intralesional triamcinolone injections every 2 weeks, the oral lesions showed marked improvement.

  • Fig. 2 (A, B) Biopsy samples from the buccal mucosa showed non-caseating granuloma with scattered perivascular chronic inflammatory cells (A: H&E, ×100, B: H&E, ×400). (C) The colonoscopic findings showed minor ulceration around the ileocecal valve (black arrow). (D) Mucosal biopsies from the terminal ileum revealed aphthous ulcerations and infiltration by inflammatory cells (H&E, ×400).


Reference

1. Triantafillidis JK, Valvi FZ, Merikas E, Peros G, Galitis ON, Gikas A. Granulomatous cheilitis associated with exacerbations of Crohn's disease: a case report. J Med Case Reports. 2008. 2:60.
Article
2. Wiesen A, David O, Katz S. Cheilitis granomatosa: Crohn's disease of the lip? J Clin Gastroenterol. 2007. 41:865–866.
3. Grave B, McCullough M, Wiesenfeld D. Orofacial granulomatosis--a 20-year review. Oral Dis. 2009. 15:46–51.
4. Al Johani K, Moles DR, Hodgson T, Porter SR, Fedele S. Onset and progression of clinical manifestations of orofacial granulomatosis. Oral Dis. 2009. 15:214–219.
Article
5. Sanderson J, Nunes C, Escudier M, Barnard K, Shirlaw P, Odell E, et al. Oro-facial granulomatosis: Crohn's disease or a new inflammatory bowel disease? Inflamm Bowel Dis. 2005. 11:840–846.
Article
6. Bogenrieder T, Rogler G, Vogt T, Landthaler M, Stolz W. Orofacial granulomatosis as the initial presentation of Crohns disease in an adolescent. Dermatology. 2003. 206:273–278.
Article
7. William T, Marsch WC, Schmidt F, Kreft B. Early oral presentation of Crohn's disease. J Dtsch Dermatol Ges. 2007. 5:678–679.
Article
8. Scully C, Cochran KM, Russell RI, Ferguson MM, Ghouri MA, Lee FD, et al. Crohn's disease of the mouth: an indicator of intestinal involvement. Gut. 1982. 23:198–201.
Article
9. Cardoso H, Nunes AC, Carneiro F, Tavarela Veloso F. Successful infliximab therapy for oral Crohn's disease. Inflamm Bowel Dis. 2006. 12:337–338.
Article
10. Mignogna MD, Fortuna G, Leuci S, Massimo A. Oral Crohn's disease: a favorable clinical response with delayed-release triamcinolone acetonide intralesional injections. Am J Gastroenterol. 2008. 103:2954–2955.
Article
Full Text Links
  • AD
Actions
Cited
CITED
export Copy
Close
Share
  • Twitter
  • Facebook
Similar articles
Copyright © 2024 by Korean Association of Medical Journal Editors. All rights reserved.     E-mail: koreamed@kamje.or.kr