Successful Treatment of Intractable Case of Granulomatous Cheilitis with a Modified Treatment Protocol "Case Report"
Abstract
Granulomatous cheilitis (GC) is a rare, idiopathic, inflammatory disorder which usually affects young adults. It is characterized by persistent, cosmetically disturbing and persistent idiopathic lip swelling. Multiple treatment modalities have been suggested. In spite of the treatment used, recurrence of the disease is very common. We reported an intractable and recurrent case of GC characterized by a large swelling of upper lip. The diagnosis is confirmed with a combination of history, clinical examination, radiographical and histopathological evaluation. This patient was treated with modified treatment protocol with a combination of intralesional steroids, prednisolone, systemic metronidazole, and doxycycline and maintenance drug therapy with approximately no signs of relapse at twelve months’ follow-up. In conclusion, the mentioned therapeutic protocol proved a success in the treatment this an intractable case of GC with a long follow up period with no relapse. More clinical trials and case control trials are needed for enhancing our understanding of this disease for evaluating the efficacy of the various treatments and establishing a universally accepted protocol for management of GC.
Ethical Statement
This research was approved by the ethics committee at Thamar University (Faculty of Dentistry) with a reference number (3/2024)
Supporting Institution
Thamar University
Project Number
3/2024
References
- 1. Sciubba JJ, Said-Al-Naief N. Orofacial granulomatosis: presentation, pathology and management of 13 cases. J Oral Pathol Med 2003;32(10):576-585.
- 2. Wiesenfeld D, Ferguson MM, Mitchell DN, MacDonald DG, Scully C, Cochran K, et al. Oro-facial granulomatosis--a clinical and pathological analysis. Q J Med 1985;54(213):101-113.
- 3. El-Hakim M, Chauvin P. Orofacial granulomatosis presenting as persistent lip swelling: review of 6 new cases. J Oral Maxillofac Surg 2004;62(9):1114-1117.
- 4. Gupta A, Singh H. Granulomatous cheilitis: successful treatment of two recalcitrant cases with combination drug therapy. Case Rep Dermatol Med 2014;2014:509262.
- 5. Critchlow WA, Chang D. Cheilitis granulomatosa: a review. Head Neck Pathol 2014;8(2):209-213.
- 6. Martínez-Menchón T, Mahiques L, Pérez-Ferriols A, Febrer I, Vilata JJ, Fortea JM, et al. Síndrome de Melkersson-Rosenthal. Actas Dermosifiliogr. 2003;94(3):180-183.
- 7. Hornstein OP. Melkersson-Rosenthal syndrome--a challenge for dermatologists to participate in the field of oral medicine. J Dermatol 1997;24(5):281-296.
- 8. Greene RM, Rogers RS 3rd. Melkersson-Rosenthal syndrome: a review of 36 patients. J Am Acad Dermatol 1989;21(6):1263-1270.
- 9. Worsaae N, Christensen KC, Schiødt M, Reibel J. Melkersson-Rosenthal syndrome and cheilitis granulomatosa. A clinicopathological study of thirty-three patients with special reference to their oral lesions. Oral Surg Oral Med Oral Pathol 1982;54(4):404-413.
- 10. Chintagunta SR, Sana SN, Bukka KS, Padma S. Cheilitis granulomatosa: Case series. J NTR Univ Health Sci 2017;6:174–177.
- 11. Jamil R.T., Agrawal M., Gharbi A., Sonthalia S., Cheilitis granulomatosa. 2017.
- 12. Zimmer WM, Rogers RS 3rd, Reeve CM, Sheridan PJ. Orofacial manifestations of Melkersson-Rosenthal syndrome. A study of 42 patients and review of 220 cases from the literature. Oral Surg Oral Med Oral Pathol 1992;74(5):610-619.
- 13. Alawi F. Granulomatous diseases of the oral tissues: differential diagnosis and update. Dent Clin North Am 2005;49(1):203-221, x.
- 14. Patton DW, Ferguson MM, Forsyth A, James J. Oro-facial granulomatosis: a possible allergic basis. Br J Oral Maxillofac Surg 1985;23(4):235-242.
- 15. Carr RD. Is the Melkersson-Rosentha syndrome hereditary? Arch Dermatol 1966;93(4): 426-427.
- 16. McKenna KE, Walsh MY, Burrows D. The Melkersson-Rosenthal syndrome and food additive hypersensitivity. Br J Dermatol 1994;131(6):921-922
- 17. Taibjee SM, Prais L, Foulds IS. Orofacial granulomatosis worsened by chocolate: results of patch testing to ingredients of Cadbury's chocolate. Br J Dermatol 2004;150(3):595.
- 18. Fitzpatrick L, Healy CM, McCartan BE, Flint SR, McCreary CE, Rogers S. Patch testing for food-associated allergies in orofacial granulomatosis. J Oral Pathol Med 2011;40(1):10-13.
- 19. Lazarov A, Kidron D, Tulchinsky Z, Minkow B. Contact orofacial granulomatosis caused by delayed hypersensitivity to gold and mercury. J Am Acad Dermatol 2003;49(6):1117-1120.
- 20. van der Waal RI, Schulten EA, van der Meij EH, van de Scheur MR, Starink TM, van der Waal I. Cheilitis granulomatosa: overview of 13 patients with long-term follow-up--results of management. Int J Dermatol 2002;41(4):225-229.
- 21. Martínez Martínez ML, Azaña-Defez JM, Pérez-García LJ, López-Villaescusa MT, Rodríguez Vázquez M, Faura Berruga C. Granulomatous cheilitis: a report of 6 cases and a review of the literature. Actas Dermosifiliogr 2012;103(8):718-724.
- 22. Bruett Carter T, Trump Bryan R, Adams David R, Halpern Leslie R. Orofacial granulomatosis: a case treated with azithromycin pulse therapy, review of the literature and an algorithm for diagnosis. IDCases 2020;21:e00924
- 23. Kato T, Tagami H. Successful treatment of cheilitis granulomatosa with tranilast. J Dermatol 1986;13(5):402-403.
- 24. Bacci C, Valente ML. Successful treatment of cheilitis granulomatosa with intralesional injection of triamcinolone. J Eur Acad Dermatol Venereol 2010;24(3):363-364.
- 25. Barry O, Barry J, Langan S, Murphy M, Fitzgibbon J, Lyons JF. Treatment of granulomatous cheilitis with infliximab. Arch Dermatol 2005;141(9):1080-1082.
- 26. Kruse-Lösler B, Presser D, Metze D, Joos U. Surgical treatment of persistent macrocheilia in patients with Melkersson-Rosenthal syndrome and cheilitis granulomatosa. Arch Dermatol 2005;141(9):1085-1091.
- 27. Tambe S, Patil P, Modi A, Jerajani H. Metronidazole as a monotherapy in the management of granulomatous cheilitis. Indian J Dermatol Venereol Leprol 2018;84(4):491-495.
- 28. Vibhute NA, Vibhute AH, Daule NR. Cheilitis granulomatosa: a case report with review of literature. Indian J Dermatol 2013;58(3):242.
- 29. Coskun B, Saral Y, Cicek D, Akpolat N. Treatment and follow-up of persistent granulomatous cheilitis with intralesional steroid and metronidazole. J Dermatolog Treat 2004;15(5):333-335.
- 30. Stein SL, Mancini AJ. Melkersson-Rosenthal syndrome in childhood: successful management with combination steroid and minocycline therapy. J Am Acad Dermatol 1999;41(5 Pt 1):746-748.
- 31. Dar NR, Raza N, Nadeem A, Manzoor A. Granulomatous cheilitis: sustained response to combination of intralesional steroids, metronidazole and minocycline. J Coll Physicians Surg Pak 2007;17(9):566-567.
- 32. Holmes NE, Charles PGP. Safety and efficacy review of doxycycline. Clin Med Ther 2009;1:S2035.
Details
| Primary Language | English |
|---|---|
| Subjects | Oral Medicine and Pathology |
| Journal Section | Case Report |
| Authors | |
| Project Number | 3/2024 |
| Submission Date | March 13, 2025 |
| Acceptance Date | November 19, 2025 |
| Publication Date | December 29, 2025 |
| Published in Issue | Year 2025 Volume: 28 Issue: 4 |
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