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Coexistence of diffuse panbronchiolitis and sarcoidosis revealed during splenectomy: a case report
BMC Pulmonary Medicine ( IF 2.6 ) Pub Date : 2020-03-30 , DOI: 10.1186/s12890-020-1117-y
Tomohiro Akaba , Kiyoshi Takeyama , Mitsuko Kondo , Fumi Kobayashi , Asako Okabayashi , Tatsuo Sawada , Etsuko Tagaya

Sarcoidosis is a systemic granulomatous disease caused by CD4+ cell-dominant inflammation. Meanwhile, diffuse panbronchiolitis is a chronic inflammatory respiratory disease predominantly caused by CD8+ lymphocytes and neutrophils. Herein, we report a rare case of sarcoidosis in which the clinical presentation had become evident as diffuse panbronchiolitis after splenectomy for sarcoidosis. A 23-year-old Japanese woman was referred to our hospital due to splenomegaly of unknown etiology. Upon admission, chest computed tomography scan revealed centrilobular and randomly distributed small nodules in both lungs. Bronchoalveolar lavage revealed a high proportion of lymphocytes and a decreased CD4/CD8 ratio. However, the biopsy specimens obtained from both the liver and lungs revealed noncaseating epithelioid granulomas, which confirmed the diagnosis of sarcoidosis. The patient underwent splenectomy due to progressive cytopenia and high risk of splenic rupture. After the surgery, the condition of the patient was consistently good for 3 months. Then, she gradually developed productive cough and dyspnea. Both sinus and chest computed tomography scan revealed chronic paranasal sinusitis and deterioration of centrilobular nodules in both lung fields, respectively. The second bronchoalveolar lavage revealed a high proportion of neutrophils, and the bronchoalveolar lavage fluid tested positive for Hemophilus influenzae. The titer of cold agglutinin was elevated, thereby confirming the diagnosis of diffuse panbronchiolitis. On the basis of the clinical and radiological findings, the condition of the patient improved with low-dose macrolide therapy for 3 months. The coexistence of sarcoidosis and diffuse panbronchiolitis has not been previously reported, and the hidden profiles of diffuse panbronchiolitis may have been revealed by splenectomy.

中文翻译:

脾切除术中发现弥漫性全细支气管炎和结节病并存:一例

结节病是由CD4 +细胞为主的炎症引起的系统性肉芽肿性疾病。同时,弥漫性全细支气管炎是一种慢性炎性呼吸系统疾病,主要由CD8 +淋巴细胞和中性粒细胞引起。在此,我们报告了结节病的罕见病例,其中结节切除术在脾切除后的临床表现已表现为弥漫性全细支气管炎。由于病因不明的脾肿大,一名23岁的日本妇女被转诊到我们医院。入院时,胸部计算机断层扫描显示两个肺中有小叶小结节和随机分布。支气管肺泡灌洗显示淋巴细胞比例高,CD4 / CD8比降低。但是,从肝脏和肺部获取的活检标本均显示非干酪样上皮样肉芽肿,这证实了结节病的诊断。由于进行性血细胞减少和脾破裂的高风险,患者接受了脾切除术。手术后,患者的病情一直良好,持续了3个月。然后,她逐渐发展为生产性咳嗽和呼吸困难。鼻窦和胸部计算机断层扫描均分别显示了慢性鼻旁鼻窦炎和两个肺野中小叶小结节的恶化。第二次支气管肺泡灌洗显示中性粒细胞比例很高,并且支气管肺泡灌洗液检测为流感嗜血杆菌阳性。冷凝集素的效价升高,从而证实了弥漫性全细支气管炎的诊断。根据临床和放射学结果,低剂量大环内酯类药物治疗3个月后患者的病情得到改善。
更新日期:2020-04-22
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