ASAKO OKABAYASHI
   Department   School of Medicine(Institute of Oriental Medicine), School of Medicine
   Position  
Article types Case report
Language English
Peer review Non peer reviewed
Title Coexistence of diffuse panbronchiolitis and sarcoidosis revealed during splenectomy: a case report.
Journal Formal name:BMC pulmonary medicine
Abbreviation:BMC Pulm Med
ISSN code:14712466/14712466
Domestic / ForeginForegin
Volume, Issue, Page 20(1),pp.77
Author and coauthor AKABA Tomohiro†, TAKEYAMA Kiyoshi, KONDO Mitsuko, KOBAYASHI Fumi, OKABAYASHI Asako, SAWADA Tatsuo, TAGAYA Etsuko
Publication date 2020/03
Summary 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