マツモト　エツコ   Matsumoto Etsuko   松本　悦子    所属   医学部 医学科（東京女子医科大学病院）    職種   教授・基幹分野長
論文種別	症例報告
言語種別	英語
査読の有無	査読なし
表題	Coexistence of diffuse panbronchiolitis and sarcoidosis revealed during splenectomy: a case report.
掲載誌名	正式名：BMC pulmonary medicine 略　 称：BMC Pulm Med ISSNコード：14712466/14712466
掲載区分	国外
巻・号・頁	20(1),pp.77
著者・共著者	AKABA Tomohiro†, TAKEYAMA Kiyoshi, KONDO Mitsuko, KOBAYASHI Fumi, OKABAYASHI Asako, SAWADA Tatsuo, TAGAYA Etsuko
担当区分	最終著者
発行年月	2020/03
概要	BACKGROUND: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.CASE PRESENTATION: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.CONCLUSIONS: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.
DOI	10.1186/s12890-020-1117-y
PMID	32228530