Tetikkurt Cuneyt, Yuruyen Mehmet, Tetikkurt Seza, Bayar Nihal, Ozdemir Imran
Pulmonary Diseases Department, Cerrahpasa Medical Faculty, Istanbul University, Tanzimat sokak Serkan Apt, No 8/16 Caddebostan, Istanbul, 34728, Turkey.
Multidiscip Respir Med. 2012 Jul 17;7(1):14. doi: 10.1186/2049-6958-7-14.
A 27 year old female with Graves' disease presented with fever, exertional dyspnea and polyarthralgia. Erythema nodosum had occured three months earlier. The patient declared irregular use of propylthiouracil (PTU) for the last 8 months. Neutropenia and microscopic hematuria developed in the second week of admission. Chest X-ray showed inhomogenous pulmonary opacities, left pleural effusion and cardiomegaly. Computed tomography (CT) revealed multiple subpleural nodules, left pleural effusion, pericardial effusion, enlarged mediastinal and axillary lymph nodes. Bronchoalveolar lavage (BAL) cytology demonstrated hemosiderin laden macrophages. Histopathologic examination of the transbronchial biopsy specimen revealed a nonspecific inflammation. Serum was positive for ANA, P-ANCA, MPO-ANCA, PR3-ANCA and negative for anti-ds-DNA, C-ANCA, C3, C4 and anti-histone antibody. All symptoms resolved in two months after PTU withdrawal and starting steroid treatment. The same clinical manifestations recurred when the patient used PTU erronously one month after discharge.This is a case of PTU induced-autoimmune disease in whom the accurate distinction between drug-induced-lupus (DIL) and vasculitis was not possible due to the significant overlap of clinical and laboratory findings causing a significant diagnostic challenge for the chest physician.
一名27岁患有格雷夫斯病的女性患者出现发热、劳力性呼吸困难和多关节痛。结节性红斑在三个月前出现。患者宣称在过去8个月中不规则使用丙硫氧嘧啶(PTU)。入院第二周出现中性粒细胞减少和镜下血尿。胸部X线显示肺部不均匀阴影、左侧胸腔积液和心脏扩大。计算机断层扫描(CT)显示多个胸膜下结节、左侧胸腔积液、心包积液、纵隔和腋窝淋巴结肿大。支气管肺泡灌洗(BAL)细胞学检查显示含铁血黄素巨噬细胞。经支气管活检标本的组织病理学检查显示为非特异性炎症。血清抗核抗体(ANA)、抗髓过氧化物酶-抗中性粒细胞胞浆抗体(MPO-ANCA)、抗蛋白酶3-抗中性粒细胞胞浆抗体(PR3-ANCA)呈阳性,抗双链DNA抗体、抗中性粒细胞胞浆抗体(C-ANCA)、补体C3、C4和抗组蛋白抗体呈阴性。停用PTU并开始使用类固醇治疗两个月后,所有症状均消失。出院一个月后患者误服PTU,相同的临床表现再次出现。这是一例PTU诱发的自身免疫性疾病,由于临床和实验室检查结果存在显著重叠,无法准确区分药物性狼疮(DIL)和血管炎,给胸科医生带来了重大诊断挑战。