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Propylthiouracil-Associated Antineutrophil Cytoplasmic Antibodies Resulted Microscopic Polyangiitis and Pulmonary-Renal Syndrome: A Case Report and Literature Review

Propylthiouracil相關之抗嗜中性白血球細胞質抗體併發多發性微小血管炎及肺腎症候狀:一病例報告及文獻回顧

摘要


Propylthiouracil (PTU)是屬於thionamide類治療甲狀腺功能亢進的藥物。PTU會引起許多藥物副作用,包括白血球低下、皮疹、發燒、關節炎、血管炎及類似紅斑性狼瘡症候群。另外,有使用PTU的病患發生抗嗜中性白血球細胞質抗體相關血管炎的報告。肺腎症候群是其中一個不常見但會危及生命的PTU相關抗嗜中性白血球細胞質抗體陽性的血管炎表現。本文報告一位四十五歲女性Graves' disease患者,在接受PTU治療期間以血痰、正血球性貧血和顯微性血尿來表現的肺腎症候群。血清中的胞核旁抗嗜中性白血球細胞質抗體呈陽性,尿液也有變形紅血球的發現。在PTU停藥及免疫抑制劑的治療下,發燒、血尿和肺出血的情況都有改善,但是抗嗜中性白血球細胞質抗體仍持續陽性數個月。抗嗜中性白血球細胞質抗體相關之肺腎症候群成為新報告的PTU併發症。回溯文獻,在使用PTU的患者,使用的期間越是延長,體內被偵測到抗嗜中性白血球細胞質抗體陽性的比率也越高。相反的,其它thionamide類的藥物,如carbimazole和methimazole,少有抗嗜中性白血球細胞質抗體相關的血管炎病例被報告。因此,在需要長期使用抗甲狀腺藥物治療甲狀腺亢進時,可以優先考慮carbimazole或methimazole。

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並列摘要


Propylthiouracil (PTU) is a thionamide used for hyperthyroidism. Numbers of PTU induced adverse reactions such as leukopenia, rash, fever, arthritis, vasculitis, and lupus-like syndrome are reported. In addition, antineutrophil cytoplasmic antibody (ANCA)-associated vasculitis during PTU therapy was reported. Pulmonary-renal syndrome is one infrequent but life-threatening presentation of PTU-associated ANCA-positive vasculitis. We report a 45-year-old woman with pulmonary-renal syndrome presented as hemoptysis, normocytic anemia, and microscopic hematuria during the treatment of Graves' disease with PTU. Her serum perinuclear-ANCA was positive and dysmorphic red blood cells were found in urinalysis. After discontinuing PTU and starting immunosuppressive agent treatment, the fever, hematuria, and pulmonary hemorrhage subsided but the p-ANCA was still present several months later. ANCA-associated pulmonary-renal syndrome may be a complication of PTU therapy. The proportion of ANCA-positive case increases with the prolongation of PTU therapy. In contrast, there are rare reports of ANCA-associated vasculitis in the other thionamide i.e. carbimazole and methimazole. Therefore, carbimazole or methimazole may be the drug of choice when long-term treatment for hyperthyroidism is required.

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