Osteonecrosis of Maxilla in Wegener's Granulomatosis: Case Report

상악골 괴사를 동반한 베게너 육아종증: 증례보고

  • Kim, Il-Kyu (Devision of Oral and Maxillofacial Surgery, Department of Dentistry, College of Medicine, Inha University) ;
  • Chang, Jae-Won (Devision of Oral and Maxillofacial Surgery, Department of Dentistry, College of Medicine, Inha University) ;
  • Ju, Sang-Hyun (Devision of Oral and Maxillofacial Surgery, Department of Dentistry, College of Medicine, Inha University) ;
  • Pyeon, Young-Hoon (Devision of Oral and Maxillofacial Surgery, Department of Dentistry, College of Medicine, Inha University) ;
  • Jung, Bum-Sang (Devision of Oral and Maxillofacial Surgery, Department of Dentistry, College of Medicine, Inha University) ;
  • Han, Ji-Young (Department of Pathology, College of Medicine, Inha University)
  • 김일규 (인하대학교 의과대학 치과학교실 구강악안면외과) ;
  • 장재원 (인하대학교 의과대학 치과학교실 구강악안면외과) ;
  • 주상현 (인하대학교 의과대학 치과학교실 구강악안면외과) ;
  • 편영훈 (인하대학교 의과대학 치과학교실 구강악안면외과) ;
  • 정범상 (인하대학교 의과대학 치과학교실 구강악안면외과) ;
  • 한지영 (인하대학교 의과대학 병리학교실)
  • Received : 2012.02.13
  • Accepted : 2012.05.04
  • Published : 2012.05.30

Abstract

Wegener's granulomatosis (WG) is an autoimmune disease of unknown etiology characterized by the triad of necrotizing granulomatous lesion in the upper and lower respiratory tracts or both, disseminated vasculitis involving both small arteries and veins, and necrotizing glomerulonephritis. The most common oral lesions associated with WG are ulceration and strawberry gingivitis. A 47-years old man in medical care associated WG was consulted our Division of Oral and Maxillofacial Surgery for the chief complaint of toothaches. Pre-operative panorama showed the alveolar radiolucency and the loss of lamina dura regarding the left upper teeth. An oropharyngeal magnetic resonance imaging also revealed the increased bone marrow signal intensity on the left maxilla. Under the impression of maxillary osteonecrosis due to WG, maxillary saucerization with removal of involved teeth was performed. We obtained good results and report the first case of WG in Korea, with the review of literatures regarding oral and general systemic features.

Keywords

References

  1. Wegener F. Uber eine eigenartige rhinogene granulomatose mit besondere beteilgung des arteriensystems and der nieren. Beitrage Pathologie Anatomie 1939;102:36-51.
  2. Stewart C, Cohen D, Bhattacharyya I, 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
  3. van der Woude FJ, Rasmussen N, Lobatto S, et al. Autoantibodies against neutrophils and monocytes: tool for diagnosis and marker of disease activity in Wegener's granulomatosis. Lancet 1985;1:425-9.
  4. Gubbels SP, Barkhuizen A, Hwang PH. Head and neck manifestations of Wegener's granulomatosis. Otolaryngol Clin North Am 2003;36:685-705. https://doi.org/10.1016/S0030-6665(03)00023-9
  5. Carter LM, Brizman E. Lingual infarction in Wegener's Granulomatosis: a case report and review of the literature. Head Face Med 2008;4:19. https://doi.org/10.1186/1746-160X-4-19
  6. Lilly J, Juhlin T, Lew D, Vincent S, Lilly G. Wegener's granulomatosis presenting as oral lesions: a case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1998;85:153-7. https://doi.org/10.1016/S1079-2104(98)90418-8
  7. Napier SS, Allen JA, Irwin CR, McCluskey DR. Strawberry gums: a clinicopathological manifestation diagnostic of Wegener's granulomatosis? J Clin Pathol 1993;46:709-12. https://doi.org/10.1136/jcp.46.8.709
  8. Ruokonen H, Helve T, Arola J, Hietanen J, Lindqvist C, Hagstrom J. "Strawberry like" gingivitis being the first sign of Wegener's granulomatosis. Eur J Intern Med 2009;20:651-3. https://doi.org/10.1016/j.ejim.2009.04.007
  9. Scott J, Finch LD. Wegener's granulomatosis presenting as gingivitis. Review of the clinical and pathologic features and report of a case. Oral Surg Oral Med Oral Pathol 1972;34:920-33. https://doi.org/10.1016/0030-4220(72)90230-7
  10. Rodgers H, Quirke P, Lipkin GW, Brownjohn AM. Infarction of the tongue in Wegener's granulomatosis. Br J Clin Pract 1992;46:268-9.
  11. Bachmeyer C, Petitjean B, Testart F, Richecoeur J, Ammouri W, Blum L. Lingual necrosis as the presenting sign of Wegener's granulomatosis. Clin Exp Dermatol 2006;31:321-2. https://doi.org/10.1111/j.1365-2230.2005.02053.x
  12. Duna GF, Galperin C, Hoffman GS. Wegener's granulomatosis. Rheum Dis Clin North Am 1995;21:949-86.
  13. Aasarod K, Iverson BM, Hammerstrom J, Bostad L, Vatten L, Jorstad S. Wegener's granulomatosis: clinical course in 108 patients with renal involvement. Nephrol Dial Transplant 2000;15:611-8. https://doi.org/10.1093/ndt/15.5.611
  14. Crean SJ, Adams R, Bennett J. Sublingual gland involvement in systemic Wegener's granulomatosis: a case report. Int J Oral Maxillofac Surg 2002;31:104-6. https://doi.org/10.1054/ijom.2001.0144
  15. Shiboski CH, Regezi JA, Sanchez HC, Silverman S Jr. Oral lesions as the first clinical sign of microscopic polyangiitis: a case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002;94:707-11. https://doi.org/10.1067/moe.2002.129178
  16. Leavitt RY, Fauci AS, Bloch DA, et al. The American College of Rheumatology 1990 criteria for the classification of Wegener's granulomatosis. Arthritis Rheum 1990;33:1101-7.
  17. Ye S, Yang CD. How could we make a diagnosis of Wegener's granulomatosis? Clin Rheumatol 2007;26:784-6. https://doi.org/10.1007/s10067-005-0196-8
  18. Mahr A, Girard T, Agher R, Guillevin L. Analysis of factors predictive of survival based on 49 patients with systemic Wegener's granulomatosis and prospective follow-up. Rheumatology (Oxford) 2001;40:492-8. https://doi.org/10.1093/rheumatology/40.5.492