Hayashi Yusuke, Katayama Yuko, Sakuragi Minoru, Hayashi Ayano, Kakita Hiroko, Uyama Michihiro, Marumo Satoshi, Fukui Motonari
Department of Respirology, Kitano Hospital, The Tazuke Kofukai Medical Research Institute, 2-4-20 Ohgimachi, Kita-ku, Osaka, 530-8480, Japan.
Department of Nephrology and Dialysis, Kitano Hospital, The Tazuke Kofukai Medical Research Institute, Osaka, Japan.
J Med Case Rep. 2021 Feb 1;15(1):30. doi: 10.1186/s13256-020-02614-3.
The association between a preceding malignancy and the onset of anti-neutrophil cytoplasmic antibody (ANCA)-associated vasculitis (AAV) has been reported in several studies. While the co-existence of ANCA and anti-glomerular basement membrane (GBM) antibodies in an individual patient is not a common occurrence, this double-positive disease currently has no optimal treatment method. Herein, we report a case of a double-positive disease involving the sequential development of acute kidney injury (AKI) and diffuse alveolar hemorrhage (DAH) in a patient with small cell lung cancer (SCLC).
A 75-year-old Japanese woman was diagnosed with small cell lung cancer (cT3N2M1b cStage IV) and received chemotherapy. After one cycle of chemotherapy, she experienced fever and malaise. Her serum creatinine level rapidly increased, and she tested positive for myeloperoxidase (MPO)-ANCA and anti-GBM antibody. She was diagnosed with AKI due to microscopic polyangiitis (MPA) based on renal biopsy. Corticosteroid therapy was initiated, which improved her renal dysfunction. Eight days after she was discharged from the hospital, she complained of dyspnea and bloody sputum, and her condition rapidly progressed to respiratory failure. Upon chest imaging, ground-glass opacities were seen in her bilateral lower lungs. Laboratory examinations after admission revealed a lower MPO-ANCA titer and an elevated anti-GBM antibody titer compared to her previous admission. We diagnosed her with DAH due to an anti-GBM disease. After corticosteroid pulse therapy, plasma exchange was performed five times; her oxygen saturation and chest radiologic findings improved gradually. Following five cycles of plasma exchange, her oxygen saturation recovered to 95% in room air.
To our knowledge, this is the first reported case of vasculitis caused by MPA and anti-GBM disease leading to the development of AKI and DAH during treatment of SCLC. SCLC, MPA, and anti-GBM disease may occur sequentially. A double-positive disease might have a worse prognosis; therefore, intensive therapy is more likely to achieve a better outcome.
多项研究报道了既往恶性肿瘤与抗中性粒细胞胞浆抗体（ANCA）相关血管炎（AAV）发病之间的关联。虽然个体患者中ANCA与抗肾小球基底膜（GBM）抗体同时存在并不常见，但这种双阳性疾病目前尚无最佳治疗方法。在此，我们报告一例小细胞肺癌（SCLC）患者出现双阳性疾病，先后发生急性肾损伤（AKI）和弥漫性肺泡出血（DAH）的病例。
一名75岁日本女性被诊断为小细胞肺癌（cT3N2M1b，c期IV）并接受化疗。化疗一个周期后，她出现发热和不适。血清肌酐水平迅速升高，髓过氧化物酶（MPO）-ANCA和抗GBM抗体检测呈阳性。基于肾活检，她被诊断为显微镜下多血管炎（MPA）导致的AKI。开始使用糖皮质激素治疗，肾功能障碍得到改善。出院8天后，她出现呼吸困难和咯血，病情迅速进展为呼吸衰竭。胸部影像学检查显示双侧下肺出现磨玻璃影。入院后的实验室检查显示，与之前入院相比，MPO-ANCA滴度降低，抗GBM抗体滴度升高。我们诊断她为抗GBM病导致的DAH。糖皮质激素冲击治疗后，进行了5次血浆置换；她的血氧饱和度和胸部影像学表现逐渐改善。经过5个周期的血浆置换，她在室内空气中的血氧饱和度恢复到95%。
据我们所知，这是首例报道的MPA和抗GBM病引起的血管炎，导致SCLC治疗期间发生AKI和DAH的病例。SCLC、MPA和抗GBM病可能依次发生。双阳性疾病的预后可能更差；因此，强化治疗更有可能取得更好的效果。
