Noelia Cabaleiro-Raña, Carmen Álvarez-Reguera, Evelin Cecilia Cervantes Pérez, Lucía Romar de Las Heras, Diego Santos-Álvarez, Carlos Álvarez Álvarez, Susana Romero-Yuste
{"title":"巨细胞动脉炎的舌坏死:一个基于病例的回顾。","authors":"Noelia Cabaleiro-Raña, Carmen Álvarez-Reguera, Evelin Cecilia Cervantes Pérez, Lucía Romar de Las Heras, Diego Santos-Álvarez, Carlos Álvarez Álvarez, Susana Romero-Yuste","doi":"10.1007/s00296-025-05969-2","DOIUrl":null,"url":null,"abstract":"<p><p>Lingual necrosis is a rare but serious complication of giant cell arteritis (GCA). Diagnosis can be difficult due to its atypical presentation, particularly when it occurs without the usual GCA symptoms. We present the case of a 59-year-old female with a history of migraines and smoking, who developed severe tongue pain and neck discomfort. Glucocorticoid therapy was promptly initiated due to clinical suspicion of GCA. However, when the steroid dosage was reduced, the patient's symptoms worsened despite initial improvement. Tocilizumab was subsequently introduced, and by discharge, there was a marked reduction in tongue swelling and evidence of progressive healing. A review of 55 reported cases of lingual necrosis, including ours, revealed an average age of 77.8 years and a female predominance. Hypertension was the most common cardiovascular risk factor, and 32.7% of cases presented with tongue necrosis as the initial manifestation. Most patients had their diagnosis confirmed by temporal artery biopsy, however in other cases, imaging verified the diagnosis. Even though glucocorticoids were still the primary treatment, 13 patients needed other immunosuppressive medications. Tocilizumab has demonstrated promising results in reducing glucocorticoid exposure and improving remission rates. This case highlights the importance of considering GCA in the differential diagnosis of lingual necrosis, even in younger patients or those with atypical presentations. To avoid irreparable consequences, early detection and timely treatment beginning are essential. Tocilizumab may be used as an effective therapeutic option for cases that don't respond to glucocorticoids.</p>","PeriodicalId":21322,"journal":{"name":"Rheumatology International","volume":"45 10","pages":"226"},"PeriodicalIF":2.9000,"publicationDate":"2025-09-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Lingual necrosis in giant-cell arteritis: a case-based review.\",\"authors\":\"Noelia Cabaleiro-Raña, Carmen Álvarez-Reguera, Evelin Cecilia Cervantes Pérez, Lucía Romar de Las Heras, Diego Santos-Álvarez, Carlos Álvarez Álvarez, Susana Romero-Yuste\",\"doi\":\"10.1007/s00296-025-05969-2\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Lingual necrosis is a rare but serious complication of giant cell arteritis (GCA). Diagnosis can be difficult due to its atypical presentation, particularly when it occurs without the usual GCA symptoms. We present the case of a 59-year-old female with a history of migraines and smoking, who developed severe tongue pain and neck discomfort. Glucocorticoid therapy was promptly initiated due to clinical suspicion of GCA. However, when the steroid dosage was reduced, the patient's symptoms worsened despite initial improvement. Tocilizumab was subsequently introduced, and by discharge, there was a marked reduction in tongue swelling and evidence of progressive healing. A review of 55 reported cases of lingual necrosis, including ours, revealed an average age of 77.8 years and a female predominance. Hypertension was the most common cardiovascular risk factor, and 32.7% of cases presented with tongue necrosis as the initial manifestation. Most patients had their diagnosis confirmed by temporal artery biopsy, however in other cases, imaging verified the diagnosis. Even though glucocorticoids were still the primary treatment, 13 patients needed other immunosuppressive medications. Tocilizumab has demonstrated promising results in reducing glucocorticoid exposure and improving remission rates. This case highlights the importance of considering GCA in the differential diagnosis of lingual necrosis, even in younger patients or those with atypical presentations. To avoid irreparable consequences, early detection and timely treatment beginning are essential. 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Lingual necrosis in giant-cell arteritis: a case-based review.
Lingual necrosis is a rare but serious complication of giant cell arteritis (GCA). Diagnosis can be difficult due to its atypical presentation, particularly when it occurs without the usual GCA symptoms. We present the case of a 59-year-old female with a history of migraines and smoking, who developed severe tongue pain and neck discomfort. Glucocorticoid therapy was promptly initiated due to clinical suspicion of GCA. However, when the steroid dosage was reduced, the patient's symptoms worsened despite initial improvement. Tocilizumab was subsequently introduced, and by discharge, there was a marked reduction in tongue swelling and evidence of progressive healing. A review of 55 reported cases of lingual necrosis, including ours, revealed an average age of 77.8 years and a female predominance. Hypertension was the most common cardiovascular risk factor, and 32.7% of cases presented with tongue necrosis as the initial manifestation. Most patients had their diagnosis confirmed by temporal artery biopsy, however in other cases, imaging verified the diagnosis. Even though glucocorticoids were still the primary treatment, 13 patients needed other immunosuppressive medications. Tocilizumab has demonstrated promising results in reducing glucocorticoid exposure and improving remission rates. This case highlights the importance of considering GCA in the differential diagnosis of lingual necrosis, even in younger patients or those with atypical presentations. To avoid irreparable consequences, early detection and timely treatment beginning are essential. Tocilizumab may be used as an effective therapeutic option for cases that don't respond to glucocorticoids.
期刊介绍:
RHEUMATOLOGY INTERNATIONAL is an independent journal reflecting world-wide progress in the research, diagnosis and treatment of the various rheumatic diseases. It is designed to serve researchers and clinicians in the field of rheumatology.
RHEUMATOLOGY INTERNATIONAL will cover all modern trends in clinical research as well as in the management of rheumatic diseases. Special emphasis will be given to public health issues related to rheumatic diseases, applying rheumatology research to clinical practice, epidemiology of rheumatic diseases, diagnostic tests for rheumatic diseases, patient reported outcomes (PROs) in rheumatology and evidence on education of rheumatology. Contributions to these topics will appear in the form of original publications, short communications, editorials, and reviews. "Letters to the editor" will be welcome as an enhancement to discussion. Basic science research, including in vitro or animal studies, is discouraged to submit, as we will only review studies on humans with an epidemological or clinical perspective. Case reports without a proper review of the literatura (Case-based Reviews) will not be published. Every effort will be made to ensure speed of publication while maintaining a high standard of contents and production.
Manuscripts submitted for publication must contain a statement to the effect that all human studies have been reviewed by the appropriate ethics committee and have therefore been performed in accordance with the ethical standards laid down in an appropriate version of the 1964 Declaration of Helsinki. It should also be stated clearly in the text that all persons gave their informed consent prior to their inclusion in the study. Details that might disclose the identity of the subjects under study should be omitted.