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Case Reports
. 2023 May 15;24(1):382.
doi: 10.1186/s12891-023-06465-z.

Tongue necrosis secondary to giant cell arteritis, successfully treated with tocilizumab: a case report

Young Min Cho  1 , Lara El Khoury  2 , Jonathan Paramo  2 , Diane Michelle Horowitz  2 , Jian Yi Li  3 , Nina Kello  2
Affiliations
Case Reports

Tongue necrosis secondary to giant cell arteritis, successfully treated with tocilizumab: a case report

Young Min Cho et al. BMC Musculoskelet Disord. .

Abstract

Background: Giant Cell Arteritis (GCA) is a large vessel vasculitis that most commonly presents with headache, scalp tenderness, jaw claudication, and vision changes. Various other, less common, manifestations have been reported in the literature such as scalp and tongue necrosis. Though most patients respond to corticosteroids, some cases of GCA are refractory to the high doses of corticosteroids.

Case presentation: We present a 73-year-old female with GCA refractory to corticosteroids presenting with tongue necrosis. This patient significantly improved with a dose of tocilizumab, an IL-6 inhibitor.

Conclusion: To the best of our knowledge, this is the first case report of a patient with refractory GCA presenting with tongue necrosis that had rapid improvement with tocilizumab. Prompt diagnosis and treatment can prevent severe outcomes such as tongue amputation in GCA patients with tongue necrosis, and tocilizumab may be effective for corticosteroid-refractory cases.

Keywords: Case report; Giant cell arteritis; Refractory; Tocilizumab; Tongue necrosis.

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Conflict of interest statement

The authors declare no competing interests.

Figures

Fig. 1
Fig. 1
A, B yellow arrows indicating the lesion on the dorsal and ventral aspect of the tongue, respectively. C, D Day 1 following tocilizumab infusion. E, F Day 2 following tocilizumab infusion, noticeable improvement of lesions. G, H Two months later, the lesions in the tongue were completely healed
Fig. 2
Fig. 2
Temporal artery biopsy results. A Hematoxylin and Eosin stain showing inflammatory cells (predominant lymphocytes and occasional eosinophils) in intima, media and adventitia and rare multinucleated giant cells in intima indicated by yellow arrow, a characteristic feature of temporal arteritis. B Elastic stain showing the fragmentation, distortion, and lack of continuity of the internal elastic lamina. C Trichrome stain showing damage of the internal elastic lamina and media, and occlusion of lumen
Fig. 3
Fig. 3
Timeline of case presentation description
Fig. 4
Fig. 4
Scheme of management plan
See this image and copyright information in PMC

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