Refractory Necrotizing Vasculitis of the Oral Mucosa-A Case Report

壞死性血管炎是一個血管發炎及壞死的疾病，單獨口腔黏膜反復壞死性血管炎是罕見的病例。本文報告一位41歲口腔黏膜反覆壞死性血管炎之男性病患，病患第一個口腔病灶是在2001年6月於右上後方區域有口鼻竇瘻管。病患於2003年2月接受廣泛切除及前側大腿游離皮瓣重建右上後方病灶後，於2003年5月，反復於右下牙齦出現潰瘍性病灶，診斷確定爲口腔黏膜壞死性血管炎。病患因下顎骨暴露於口腔中，於2003年7月接受廣泛切除及前臂游離皮瓣重建右下方病灶。術後追蹤期間，於2003年11月，右上後方區域再次出現潰瘍性病灶。病患接受脈衡療法及服用維持性免疫抑制藥物後，右上後方區域潰瘍性病灶痊癒，且未再出現其他口腔病灶。此病患口腔黏膜反復壞死性血管炎，經過先後兩次的游離皮瓣及脈衡療法，才成功的治癒。口腔黏膜反覆壞死性血管炎是罕見的疾病，臨床上也不易鑑別診斷，本病例報告提出病患之臨床病症、治療過程和預後，以提供日後相似病症鑑別診斷之參考及相關研究之基礎。

關鍵字

壞死性血管炎；口腔黏膜；游離皮瓣

並列摘要

Necrotizing vasculitis is an inflammatory disease together with necrosis of blood vessels. Refractory necrotizing vasculitis solely in the oral mucosa is rare. We report a case of a 41 year-old male with refractory necrotizing vasculitis lesion of the oral mucosa. An initial intraoral ulcerative lesion with oroantral fistula of the right upper posterior area was noted in June, 2001. In Feb, 2003 the patient underwent excision of the lesion together with partial maxillectomy and was reconstructed with an anterior lateral thigh free flap. However, he suffered from another refractory, ulcerative lesion over the right lower gingiva with mandible bone exposure in May, 2003. The diagnosis of necrotizing vasculitis of the oral mucosa was confirmed by pathological exam. The patient received a second operation of excision, marginal mandibulectomy, and forearm free flap reconstruction in July, 2003. During post-op follow-up period, recurrent, ulcerative lesion of the right upper area was noted again in Nov, 2003. The patient received steroid pulse therapy and maintenances immunosuppressive drugs. The refractory lesion of right upper area was healed and no more oral lesions were noted. Necrotizing vasculitis of the oral mucosa was treated successfully with two surgical excisions with free flap reconstructions, and steroid pulse therapy. The refractory necrotizing vasculitis of the oral mucosa is difficult for diagnosis. We reported the disease profile, treatment course, and prognosis of the patient for the differential diagnosis of similar patient and the basis of further studies.

並列關鍵字

Necrotizing vasculitis ； Oral mucosa ； Free flap

國際替代計量

Refractory Necrotizing Vasculitis of the Oral Mucosa-A Case Report

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