Journal of Periodontal and Implant Science

Korean Academy of Periodontology (대한치주과학회)
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Role of gingival manifestation in diagnosis of granulomatosis with polyangiitis (Wegener's granulomatosis)

  • Sung, Iel-Yong (Department of Dentistry, Ulsan University Hospital, University of Ulsan College of Medicine) ;
  • Kim, Young-Min (Department of Pathology, Ulsan University Hospital, University of Ulsan College of Medicine) ;
  • Cho, Yeong-Cheol (Department of Dentistry, Ulsan University Hospital, University of Ulsan College of Medicine) ;
  • Son, Jang-Ho (Department of Dentistry, Ulsan University Hospital, University of Ulsan College of Medicine)
  • Received : 2015.08.28
  • Accepted : 2015.11.27
  • Published : 2015.12.31
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Abstract

Purpose: This report describes a case of granulomatosis with polyangiitis (GPA) in which the gingival manifestation was crucial in both making an early diagnosis and possibly in deciding the approach to treatment. Methods: A 57-year-old sailor presented to the Department of Dentistry at Ulsan University Hospital complaining of gingival swelling since approximately 2 months. He had orofacial granulomatous lesions and the specific gingival manifestation of strawberry gingivitis. Results: The diagnosis of GPA was made on the basis of clinical symptoms and signs, and confirmed by the presence of the anti-neutrophil cytoplasmic antibody and a positive biopsy. The patient was admitted to the hospital and subsequently placed on a disease-modifying therapy regimen that included methotrexate and prednisone. Conclusions: Identification of the gingival manifestation of the disease permitted an early diagnosis and prompt therapy in a disease in which time is a crucial factor. Because of its rapid progression and potentially fatal outcome, an early diagnosis of GPA is important. Therefore, dentists should be aware of the oral signs and symptoms of such systemic diseases.

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References

  1. Jennette JC. Nomenclature and classification of vasculitis: lessons learned from granulomatosis with polyangiitis (Wegener's granulomatosis). Clin Exp Immunol 2011;164 Suppl 1:7-10. https://doi.org/10.1111/j.1365-2249.2011.04357.x
  2. Hernandez G, Serrano C, Porras L, Lopez-Pintor R, Rubio L, Yanes J. Strawberry-like gingival tumor as the first clinical sign of Wegener's granulomatosis. J Periodontol 2008;79:1297-303. https://doi.org/10.1902/jop.2008.070572
  3. Guidelli GM, Tenti S, Pascarelli NA, Galeazzi M, Fioravanti A. Granulomatosis with polyangiitis and intravenous immunoglobulins: a case series and review of the literature. Autoimmun Rev 2015;14: 659-64. https://doi.org/10.1016/j.autrev.2015.03.005
  4. Siar CH, Yeo KB, Nakano K, Nagatsuka H, Tsujigiwa H, Tomida M, et al. Strawberry gingivitis as the first presenting sign of Wegener's granulomatosis: report of a case. Eur J Med Res 2011;16:331-4. https://doi.org/10.1186/2047-783X-16-7-331
  5. Almouhawis HA, Leao JC, Fedele S, Porter SR. Wegener's granulomatosis: a review of clinical features and an update in diagnosis and treatment. J Oral Pathol Med 2013;42:507-16. https://doi.org/10.1111/jop.12030
  6. Martinez Del Pero M, McKiernan D, Jani P. Presentation and initial assessment of ENT problems in patients with granulomatosis with polyangiitis (Wegener's). J Laryngol Otol 2014;128:730-7. https://doi.org/10.1017/S0022215114001492
  7. Ponniah I, Shaheen A, Shankar KA, Kumaran MG. Wegener's granulomatosis: the current understanding. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2005;100:265-70. https://doi.org/10.1016/j.tripleo.2005.04.018
  8. Bhatt V, Hall TJ. Strawberry gingival enlargement as only manifestation of Wegeners granulomatosis. Br J Oral Maxillofac Surg 2009;47:500.
  9. Bajema IM, Hagen EC, van der Woude FJ, Bruijn JA. Wegener's granulomatosis: a meta-analysis of 349 literary case reports. J Lab Clin Med 1997;129:17-22. https://doi.org/10.1016/S0022-2143(97)90157-8
  10. Troiano G, Dioguardi M, Giannatempo G, Laino L, Testa NF, Cocchi R, et al. Orofacial granulomatosis: clinical signs of different pathologies. Med Princ Pract 2015;24:117-22. https://doi.org/10.1159/000369810
  11. Wegener F. On generalised septic vessel diseases. By Friedrich Wegener, 1937 (translation). Thorax 1987;42:918-9. https://doi.org/10.1136/thx.42.12.918
  12. Falk RJ, Gross WL, Guillevin L, Hoffman GS, Jayne DR, Jennette JC, et al. Granulomatosis with polyangiitis (Wegener's): an alternative name for Wegener's granulomatosis. Arthritis Rheum 2011; 63:863-4. https://doi.org/10.1002/art.30286
  13. Berden A, Goceroglu A, Jayne D, Luqmani R, Rasmussen N, Bruijn JA, et al. Diagnosis and management of ANCA associated vasculitis. BMJ 2012;344:e26. https://doi.org/10.1136/bmj.e26
  14. Hoffman GS, Langford CA, Specks U. Granulomatosis with polyangiitis (Wegener's). In: Hoffman GS, Weyand CM, Langford CA, Goronzy JJ, editors. Inflammatory diseases of blood vessels. 2nd ed. Chichester, West Sussex: Blackwell Publishing Ltd; 2012. p.238-51.
  15. Khan AM, Elahi F, Hashmi SR, Mahida KH, Ingrams DR. Wegener's granulomatosis: a rare, chronic and multisystem disease. Surgeon 2006;4:45-52. https://doi.org/10.1016/S1479-666X(06)80021-1
  16. Comarmond C, Cacoub P. Granulomatosis with polyangiitis (Wegener): clinical aspects and treatment. Autoimmun Rev 2014;13: 1121-5. https://doi.org/10.1016/j.autrev.2014.08.017
  17. Patten SF, Tomecki KJ. Wegener's granulomatosis: cutaneous and oral mucosal disease. J Am Acad Dermatol 1993;28:710-8. https://doi.org/10.1016/0190-9622(93)70098-E
  18. Liam CK. Hyperplastic gingivitis: an oral manifestation of Wegener's granulomatosis. Postgrad Med J 1993;69:754. https://doi.org/10.1136/pgmj.69.815.754
  19. Stewart C, Cohen D, Bhattacharyya I, Scheitler L, Riley S, Calamia K, et al. Oral manifestations of Wegener's granulomatosis: a report of three cases and a literature review. J Am Dent Assoc 2007; 138:338-48. https://doi.org/10.14219/jada.archive.2007.0166
  20. Leavitt RY, Fauci AS, Bloch DA, Michel BA, Hunder GG, Arend WP, et al. The American College of Rheumatology 1990 criteria for the classification of Wegener's granulomatosis. Arthritis Rheum 1990;33:1101-7.
  21. Savige J, Dimech W, Fritzler M, Goeken J, Hagen EC, Jennette JC, et al. Addendum to the International Consensus Statement on testing and reporting of antineutrophil cytoplasmic antibodies. Quality control guidelines, comments, and recommendations for testing in other autoimmune diseases. Am J Clin Pathol 2003;120: 312-8. https://doi.org/10.1309/WAEPADW0K4LPUHFN
  22. van der Woude FJ, Rasmussen N, Lobatto S, Wiik A, Permin H, van Es LA, et al. Autoantibodies against neutrophils and monocytes: tool for diagnosis and marker of disease activity in Wegener's granulomatosis. Lancet 1985;1:425-9.
  23. Flossmann O, Berden A, de Groot K, Hagen C, Harper L, Heijl C, et al. Long-term patient survival in ANCA-associated vasculitis. Ann Rheum Dis 2011;70:488-94. https://doi.org/10.1136/ard.2010.137778
  24. Wung PK, Stone JH. Therapeutics of Wegener's granulomatosis. Nat Clin Pract Rheumatol 2006;2:192-200.
  25. Semple D, Keogh J, Forni L, Venn R. Clinical review: Vasculitis on the intensive care unit -- part 2: treatment and prognosis. Crit Care 2005;9:193-7. https://doi.org/10.1186/cc3256
  26. Hellmich B, Kausch I, Doehn C, Jocham D, Holl-Ulrich K, Gross WL. Urinary bladder cancer in Wegener's granulomatosis: is it more than cyclophosphamide? Ann Rheum Dis 2004;63:1183-5. https://doi.org/10.1136/ard.2004.023937
  27. Allen CM, Camisa C, Salewski C, Weiland JE. Wegener's granulomatosis: report of three cases with oral lesions. J Oral Maxillofac Surg 1991;49:294-8. https://doi.org/10.1016/0278-2391(91)90224-A
  28. Alawi F. Granulomatous diseases of the oral tissues: differential diagnosis and update. Dent Clin North Am 2005;49:203-21. https://doi.org/10.1016/j.cden.2004.07.012
  29. Ghalayani P, Hajisadeghi S, Babadi F. Extragingival pyogenic granuloma associated with medication: Report of an unusual case. Dent Res J (Isfahan) 2014;11:400-4.

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