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Upper Airway Obstruction and Tracheal Perforation Caused by Thyroid Involvement of Disseminated Mucormycosis

彌漫性毛黴菌病侵犯甲狀腺引起之上呼吸道阻塞與氣管破裂

摘要


甲狀腺的黴菌感染相的罕見因為它有結構良好的被膜包覆及擁有豐富的血管及高濃度的碘。我們在此提出一個彌漫性毛黴菌感染併甲狀腺侵犯造成嚴重上呼吸道阻塞及氣管穿孔的個案。這位57歲慢性髓性白血病(chronic myeloid myeloma)男性病患因週邊血液幹細胞移植(peripheral stem cell transplantation)及移植供應者淋巴球輸注(donor lymphocyte infusion)治療後的移植物抗宿主病(graft-versus-host disease)而服用免疫抑制劑。甲狀腺毛黴菌感染初始表現為發燒、疼痛的頸部腫塊、及暫時性甲狀腺亢進與影像學顯示左甲狀腺囊腫。因甲狀腺膿瘍的擴展,臨床進展成左側聲帶麻痺、氣管穿孔及肺部感染。彌漫性毛黴菌感染的診斷是基於頸部清創組織及皮膚切片的病理學表現為非分隔及有直角分枝的菌絲。雖然以抗黴菌藥及積極的手術清創和重建,病人仍因肺部感染進展及血循惡化而死亡。這個個案強調在治療甲狀腺毛黴菌感染時積極的監測和處理呼吸道的併發症和手術清創及抗黴菌藥的必要性。進一步地,疾病的不佳預後及困難診斷也突顯在高風險病患臨床懷疑及早期侵入性組織診斷的重要性。

並列摘要


Thyroid fungal infection is extremely rare because the thyroid gland possesses a well-developed capsule, rich vasculature and high iodine content. We report a case of disseminated mucormycosis involving the thyroid gland, which caused catastrophic upper airway obstruction and tracheal perforation. This 57-year-old man with chronic myeloid leukemia had taken immunosuppressive agents for the treatment of graft-versus-host disease, which occurred after allogenic peripheral blood stem cell transplantation and donor lymphocyte infusion. The initial presentations of thyroid mucormycosis included fever, a painful neck mass and transient hyperthyroidism, with the imaging study showing a cystic lesion occupying the left thyroid gland. With extension of the thyroid abscess, the clinical course became complicated with upper airway obstruction, palsy of the left vocal cord, tracheal perforation and pulmonary infection. The diagnosis of disseminated mucormycosis involving the thyroid gland was made on the basis of histopathology of neck debrided tissue and biopsy of concomitant skin lesions, which disclosed non-septated and right-angle branching hyphae conforming to the morphology of mucormycosis. Despite treatment with antifungal agents, as well as intensive surgical debridement and reconstruction, he eventually succumbed to progressive pulmonary infection and deterioration of his hemodynamic status. Our case emphasizes the requirement of intensive monitoring and management of airway compromise, in addition to surgical debridement and systemic antifungal therapy, for the treatment of thyroid mucormycosis. The dismal prognosis and difficulty in diagnosis of this disease highlight the importance of a high index of suspicion regarding the presence of risk factors, and early invasive tissue sampling for histological and microbiological analyses.

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