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隐匿性肺出血:丙硫氧嘧啶诱导血管炎的罕见表现。

Occult pulmonary hemorrhage as a rare presentation of propylthiouracil-induced vasculitis.

机构信息

Division of Allergy, Asthma, and Rheumatology, Department of Pediatrics, Chang Gung Memorial Hospital, 5 Fu-Hsin St, Kweishan, Taoyuan, Taiwan.

出版信息

Pediatrics. 2011 Jan;127(1):e245-9. doi: 10.1542/peds.2010-0138. Epub 2010 Dec 6.

Abstract

Propylthiouracil, a drug commonly used to treat hyperthyroidism, is known to cause antineutrophil cytoplasmic antibody (ANCA)-associated vasculitis as a rare complication. The wide clinical spectrum of propylthiouracil-induced vasculitis ranges from mild forms with rash and/or arthralgia to severe forms with renal or pulmonary involvement, which can be critical and life-threatening if left unrecognized and untreated. Given its rarity and exceedingly variable clinical presentations, diagnosis may be challenging, and delayed diagnosis is not uncommon without a high index of suspicion, as illustrated by this report of a 17-year-old girl with Graves' disease who developed occult pulmonary hemorrhage as an overlooked rare presentation of ANCA-associated vasculitis after administration of propylthiouracil. Associated clinical features included fever, fatigue, palpable purpura, polyarthritis, and nephritis. Positive findings on chest radiography prompted the bronchoalveolar lavage procedure, which led to the identification of pulmonary hemorrhage. Skin biopsy showed leukocytoclastic vasculitis. Serologic test results were positive for perinuclear ANCA, cytoplasmic ANCA, myeloperoxidase-ANCA, proteinase 3-ANCA, and cryoglobulins but negative for antinuclear antibody, anti-double-stranded DNA, rheumatoid factor, and anti-hepatitis C virus antibody. The symptoms resolved after discontinuation of propylthiouracil and a few months of corticosteroids and azathioprine. This report highlights the necessity for physicians to keep alert for the protean manifestations of propylthiouracil-induced vasculitis.

摘要

丙硫氧嘧啶是一种常用于治疗甲状腺功能亢进症的药物,已知可引起抗中性粒细胞胞质抗体(ANCA)相关性血管炎作为罕见的并发症。丙硫氧嘧啶诱导的血管炎的广泛临床谱从伴有皮疹和/或关节炎的轻度形式到伴有肾脏或肺部受累的严重形式不等,如果不被识别和治疗,可能会变得严重并危及生命。鉴于其罕见性和极其多变的临床表现,诊断可能具有挑战性,并且如果没有高度怀疑,延迟诊断并不罕见,正如本报告中一名 17 岁 Graves 病女孩的情况所示,在给予丙硫氧嘧啶后,隐匿性肺出血作为被忽视的 ANCA 相关性血管炎的罕见表现出现。相关的临床特征包括发热、疲劳、可触及的紫癜、多发性关节炎和肾炎。胸部 X 线摄影的阳性发现促使进行支气管肺泡灌洗程序,从而确定了肺出血。皮肤活检显示白细胞碎裂性血管炎。血清学检测结果为核周型 ANCA、细胞质型 ANCA、髓过氧化物酶-ANCA、蛋白酶 3-ANCA 和冷球蛋白阳性,但抗核抗体、抗双链 DNA、类风湿因子和抗丙型肝炎病毒抗体阴性。在停用丙硫氧嘧啶和数月的皮质类固醇和硫唑嘌呤后,症状得到缓解。本报告强调了医生必须警惕丙硫氧嘧啶诱导的血管炎的多变表现。

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