Lingual necrosis as a sentinel ischemic manifestation of giant cell arteritis: a case report

Autores

DOI:

https://doi.org/10.5327/2525-5711.530

Palavras-chave:

Case report, Giant cell arteritis, Ischemia, Oral mucosa, Vasculitis

Resumo

Giant cell arteritis (GCA) is the most common systemic vasculitis affecting individuals aged ≥50 years and is characterized by granulomatous inflammation of large- and medium-sized arteries, which may lead to ischemic complications. Lingual necrosis is a rare but severe manifestation and is often associated with diagnostic challenges. We report the case of a 52-year-old woman who presented with rapidly progressive, painful ulceration of the tongue. Within 72 hours, the lesion evolved into a large necrotic ulcer, resulting in significant functional impairment. Clinical examination revealed scalp and temporal tenderness and an ischemic-appearing ulcer. Additional findings included new-onset temporal headache, tongue claudication, and morning stiffness of the shoulders and neck. Laboratory findings were unremarkable. Systemic corticosteroid therapy led to rapid clinical and symptomatic improvement. Complete healing was achieved within two months, with full resolution of signs/symptoms after four months. To the best of our knowledge, this is the first documented case of GCA-associated lingual necrosis reported in Venezuela.

 

Biografia do Autor

Jovienmily Villegas-Graterol, Department of Oral and Maxillofacial Surgery and Traumatology, Hospital Universitario de Caracas, Caracas, Venezuela.

Department of Oral and Maxillofacial Surgery and Traumatology, Hospital Universitario de Caracas, Caracas, Venezuela.

Luis Dariel Reyes-Quezada, Department of Oral and Maxillofacial Surgery and Traumatology, Hospital Universitario de Caracas, Caracas, Venezuela.

Department of Oral and Maxillofacial Surgery and Traumatology, Hospital Universitario de Caracas, Caracas, Venezuela.

Fabiana Valentina Vásquez-Rondón, Department of Oral and Maxillofacial Surgery and Traumatology, Hospital Universitario de Caracas, Caracas, Venezuela.

Department of Oral and Maxillofacial Surgery and Traumatology, Hospital Universitario de Caracas, Caracas, Venezuela.

María Gabriela Contreras, Department of Oral and Maxillofacial Surgery and Traumatology, Hospital Universitario de Caracas, Caracas, Venezuela.

Department of Oral and Maxillofacial Surgery and Traumatology, Hospital Universitario de Caracas, Caracas, Venezuela.

José Alcides Almeida de Arruda, School of Dentistry, Universidade Federal de Alfenas, Alfenas, Brazil.

School of Dentistry, Universidade Federal de Alfenas, Alfenas, Brazil.

Mariana Villarroel-Dorrego, School of Dentistry, Universidad Central de Venezuela, Caracas, Venezuela.

School of Dentistry, Universidad Central de Venezuela, Caracas, Venezuela.

School of Dentistry, Universidad Santa María, Caracas, Venezuela.

Referências

1. Dua AB, Husainat NM, Kalot MA, Byram K, Springer JM, James KE, Chang Lin Y, Turgunbaev M, Villa-Forte A, Abril A, Langford C, Maz M, Chung SA, Mustafa RA. Giant cell arteritis: a systematic review and meta-analysis of test accuracy and benefits and harms of common treatments. ACR Open Rheumatol. 2021;3(7):429-441. doi: 10.1002/acr2.11226.

2. Ponte C, Grayson PC, Robson JC, Suppiah R, Gribbons KB, Judge A, Craven A, Khalid S, Hutchings A, Watts RA, Merkel PA, Luqmani RA; DCVAS Study Group. 2022 American College of Rheumatology/EULAR classification criteria for giant cell arteritis. Ann Rheum Dis. 2022;81(12):1647-1653. doi: 10.1136/ard-2022-223480.

3. Salvarani C, Cantini F, Hunder GG. Polymyalgia rheumatica and giant-cell arteritis. Lancet. 2008;372(9634):234-45. doi: 10.1016/S0140-6736(08)61077-6.

4. Cabaleiro-Raña N, Álvarez-Reguera C, Cervantes Pérez EC, Romar de Las Heras L, Santos-Álvarez D, Álvarez Álvarez C, Romero-Yuste S. Lingual necrosis in giant-cell arteritis: a case-based review. Rheumatol Int. 2025;45(10):226. doi: 10.1007/s00296-025-05969-2.

5. Zadik Y, Findler M, Maly A, Rushinek H, Czerninski R. A 78-year-old woman with bilateral tongue necrosis. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2011;111(1):15-9. doi: 10.1016/j.tripleo.2010.09.001.

6. Howard S, Cremin MD. Acute parenchymatous glossitis with gangrene of the tongue. Lancet 1959;274:410-11.

7. Gagnier JJ, Kienle G, Altman DG, Moher D, Sox H, Riley D; CARE Group. The CARE guidelines: consensus-based clinical case reporting guideline development. J Med Case Rep. 2013;7:223. doi: 10.1186/1752-1947-7-223.

8. Dos Reis LA, Faustino ISP, Vargas PA, Dos Santos-Silva AR, Lopes MA. Lingual necrosis leading to the diagnosis of giant cell arteritis. Spec Care Dentist. 2021;41(3):408-410. doi: 10.1111/scd.12571.

9. Hunder GG, Bloch DA, Michel BA, Stevens MB, Arend WP, Calabrese LH, Edworthy SM, Fauci AS, Leavitt RY, Lie JT, et al. The American College of Rheumatology 1990 criteria for the classification of giant cell arteritis. Arthritis Rheum. 1990;33(8):1122-8. doi: 10.1002/art.1780330810.

10. Chehem Daoud Chehem F, de Mornac D, Feuillet F, Liozon E, Samson M, Bonnotte B, de Boysson H, Guffroy A, Balquet MH, Ledoult E, Lavigne C, Trefond L, Smets P, Bodard Q, Fenot M, Richez C, Duffau P, Guillaud C, Espitia O, Agard C; French Study Group for Large Vessel Vasculitis (GEFA). Giant cell arteritis associated with scalp, tongue or lip necrosis: a French multicenter case control study. Semin Arthritis Rheum. 2024;64:152348. doi: 10.1016/j.semarthrit.2023.152348.

11. Soowamber ML, Bond M, Touma Z, Langford C, Sanchez-Alvarez C, Abril A, Aydin SZ, Buttgereit F, Camellino D, Cid MC, Grayson PC, Hellmich B, Lichliter W, Kermani TA, Khalidi NA, Mackie SL, Matteson EL, Maz M, Merkel PA, Monach PA, Neill L, Ponte C, Salvarani C, Schmidt WA, Villiger PM, Warrington KJ, Whitlock M, Ramiro S, Dejaco C. A glossary of signs and symptoms of giant cell arteritis. Ann Rheum Dis. 2025;84(11):1877-1887. doi: 10.1016/j.ard.2025.06.2126.

12. Breuer GS, Nesher R, Nesher G. Negative temporal artery biopsies: eventual diagnoses and features of patients with biopsy-negative giant cell arteritis compared to patients without arteritis. Clin Exp Rheumatol. 2008;26(6):1103-6.

13. Kermani TA, Schmidt J, Crowson CS, Ytterberg SR, Hunder GG, Matteson EL, Warrington KJ. Utility of erythrocyte sedimentation rate and C-reactive protein for the diagnosis of giant cell arteritis. Semin Arthritis Rheum. 2012;41(6):866-71. doi: 10.1016/j.semarthrit.2011.10.005.

14. Grzybowski A, Justynska A. Giant cell arteritis with normal ESR and/or CRP is rare, but not unique! Eye (Lond). 2013;27(12):1418-9. doi: 10.1038/eye.2013.208.

15. Martínez-Taboada VM, Blanco R, Armona J, Uriarte E, Figueroa M, Gonzalez-Gay MA, Rodriguez-Valverde V. Giant cell arteritis with an erythrocyte sedimentation rate lower than 50. Clin Rheumatol. 2000;19(1):73-5. doi: 10.1007/s100670050017.

16. Hall JK. Giant-cell arteritis. Curr Opin Ophthalmol. 2008;19(6):454-60. doi: 10.1097/ICU.0b013e328310da01.

17. Narváez J, Vidal-Montal P, Maymó-Paituvi P, Palacios-Olid J, Aguilar-Coll M, Roig-Kim M, de Daniel-Bisbe L, Nolla JM. Further expanding the clinical spectrum of giant cell arteritis: Non-classical organ involvement and uncommon complications. Semin Arthritis Rheum. 2025;74:152817. doi: 10.1016/j.semarthrit.2025.152817.

18. Mukhtyar CB, Nikiphorou E, Bartoletti A, Dey M, Ducker G, Misra DP, Monti S, Studenic P, Berti A, Blockmans D, Brouwer E, Buttgereit F, Cannings K, Cid MC, Conway R, Christ L, Dejaco C, Keller KK, Kingham L, Merkel PA, Mohammad AJ, Mollan SP, Nielsen BD, Ponte C, Saadoun D, Schmidt WA, Warrington KJ, Hellmich B. 2025 EULAR recommendations for the management of polymyalgia rheumatica and primary large vessel vasculitis. Ann Rheum Dis. 2026; S0003-4967(26)00357-2. doi: 10.1016/j.ard.2026.06.009.

19. Stone JH, Tuckwell K, Dimonaco S, Klearman M, Aringer M, Blockmans D, Brouwer E, Cid MC, Dasgupta B, Rech J, Salvarani C, Schett G, Schulze-Koops H, Spiera R, Unizony SH, Collinson N. Trial of tocilizumab in giant-cell arteritis. N Engl J Med. 2017;377(4):317-328. doi: 10.1056/NEJMoa1613849.

20. Antonio AA, Santos RN, Abariga SA. Tocilizumab for giant cell arteritis. Cochrane Database Syst Rev. 2022;5(5):CD013484. doi: 10.1002/14651858.CD013484.pub3.

Downloads

Publicado

2026-11-05

Como Citar

1.
Villegas-Graterol J, Reyes-Quezada LD, Vásquez-Rondón FV, Contreras MG, de Andrade BAB, de Arruda JAA, et al. Lingual necrosis as a sentinel ischemic manifestation of giant cell arteritis: a case report. J Oral Diagn [Internet]. 5º de novembro de 2026 [citado 14º de novembro de 2026];. Disponível em: https://www.joraldiagnosis.com/revista/article/view/530

Edição

Seção

Relato de Caso