Miyata Y, Okano R, Kuratomi Y
Department of General Internal Medicine, Omiya Medical Center, Jichi Medical School. f1p4
Nihon Kyobu Shikkan Gakkai Zasshi. 1997 Oct;35(10):1093-8.
A 51-year-old woman was admitted to our hospital with exertional dyspnea, swelling and stiffness in her fingers. Raynaud's phenomenon and mammary and axillary lymphadenopathy. She had received silicone augmentation mammoplasty 30 years ago, and had since noticed bilateral mammary and axillary lymphadenopathy that was stable in size. In the 2 years before admittance she had become aware of an exacerbation of the lymphadenopathy had begun to experience and exertional dyspnea several months before admission suggesting a connective tissue disease. Physical examination revealed symmetrical weakness of the proximal limb muscles and fine crackles in the base of both lungs. Elevated myogenic enzymes, inflammatory reactions, and positive anti-SSA antibody were noted. Based upon these findings, muscle and lip biopsy results, myogenic EMG, and an apple tree appearance on sialography, a differential diagnosis of polymypositis or sjögren's syndrome was made. Axillary lymph node biopsy findings were consistent with silicone lymphadenitis. In addition, chest roentgenogram and HRCT (which revealed decreased lung volumes and interstitial opacities with no honeycombing, present predominatly in the subpleural space), pulmonary function tests (decreased VC and DLco), bronchoalveolar lavage (elevated total cell count and neutrophil and eosinophil fractions), and transbronchial lung biopsy specimens (unevently distributed alveolitis with fibrosis) indicated concurrent interstitial pneumonia. The clinical correlation between exacerbation of silicone lymphadenopathy and the development of connective tissue disease with accompanying interstitial pneumonia strongly suggested human adjuvant disease (HAD) as the pathogenesis. To our knowledge, interstitial pneumonia associated with HAD is rare.
一名51岁女性因劳力性呼吸困难、手指肿胀和僵硬入院。伴有雷诺现象以及乳腺和腋窝淋巴结病。她30年前接受过硅胶隆乳术,此后双侧乳腺和腋窝淋巴结病大小稳定。入院前2年,她意识到淋巴结病加重,入院前几个月开始出现劳力性呼吸困难,提示结缔组织病。体格检查发现近端肢体肌肉对称性无力,双肺底部有细湿啰音。肌源性酶升高、炎症反应以及抗SSA抗体阳性。基于这些发现、肌肉和唇部活检结果、肌源性肌电图以及唾液造影显示的苹果树样外观,做出了多肌炎或干燥综合征的鉴别诊断。腋窝淋巴结活检结果与硅胶淋巴结炎一致。此外,胸部X线片和高分辨率CT(显示肺容积减小和间质模糊,无蜂窝状改变,主要位于胸膜下间隙)、肺功能测试(肺活量和一氧化碳弥散量降低)、支气管肺泡灌洗(总细胞数、中性粒细胞和嗜酸性粒细胞比例升高)以及经支气管肺活检标本(不均匀分布的肺泡炎伴纤维化)表明并发间质性肺炎。硅胶淋巴结病加重与结缔组织病伴发间质性肺炎之间的临床相关性强烈提示人类佐剂病(HAD)为发病机制。据我们所知,与HAD相关的间质性肺炎很少见。