Unilateral upper lung-field pulmonary fibrosis radiologically consistent with pleuroparenchymal fibroelastosis after thoracic surgery: Clinical and radiological courses with autopsy findings |
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| Affiliation: | 1. Department of Respiratory Medicine, Kanagawa Cardiovascular and Respiratory Center, Yokohama, Japan;2. Department of Internal Medicine, Mito Medical Center, University of Tsukuba, Mito, Japan;3. Department of Pathology, Kanagawa Cardiovascular and Respiratory Center, Yokohama, Japan;4. Department of Pathology, Graduate School of Medicine, Yokohama-City University, Yokohama, Japan;5. Department of Radiology, Kanagawa Cardiovascular and Respiratory Center, Yokohama, Japan;1. Department of Respiratory Medicine, Tokyo Medical and Dental University, 1-5-45 Yushima, Bunkyo-ku, Tokyo, 113-8519, Japan;2. Department of Respiratory Medicine, Hiratsuka Kyosai Hospital, 9-11 Oiwake, Hiratsuka, Kanagawa, 254-8502, Japan;3. School of Data Science, Yokohama City University, 1-50-1, Mutsuura Higashi, Kanazawa-ku, Yokohama, Kanagawa, 236-8501, Japan;1. Department of Clinical Oncology, Hiroshima University Hospital, Hiroshima, Japan;2. Department of Respiratory Internal Medicine, Hiroshima University Hospital, Hiroshima, Japan;3. Department of Molecular and Internal Medicine, Graduate School of Biomedical and Health Sciences, Hiroshima University, Hiroshima, Japan;4. Department of Diagnostic Radiology, Graduate School of Biomedical and Health Sciences, Hiroshima University, Hiroshima, Japan;5. Department of Emergency and Critical Care Medicine, Graduate School of Biomedical and Health Sciences, Hiroshima University, Hiroshima, Japan;6. Department of Physical Analysis and Therapeutic Sciences, Graduate School of Biomedical and Health Sciences, Hiroshima University, Hiroshima, Japan;1. Department of Respiratory Medicine, Cellular Transplantation Biology, Kanazawa University Graduate School of Medicine, 13-1, Takara-machi, Kanazawa, Ishikawa 920-8641, Japan;2. Department of Biomedical Imaging and Image-Guided Therapy, Medical University of Vienna, Waehringer Guertel 18-20, Vienna 1090 Austria;3. Department of Respiratory Medicine, Komatsu Municipal Hospital, Ho-60, Mukaimoto-ori-machi, Komatsu 923-8560, Japan;4. Department of Radiology, Kinki Central Hospital of Mutual Aid Association of Public School Teachers, 3-1 Kurumazuka, Itami, Hyogo 664-8533, Japan;5. Department of Radiology, Faculty of Medicine, Saga University, 5-1-1 Nabeshima, Saga 849-8501, Japan;6. Division of Diagnostic Pathology, Saitama Prefectural Cardiovascular and Respiratory Center, 1696 Itai, Kumagaya City, Saitama 360-0105, Japan;7. Section of Diagnostic Pathology, Kanazawa University Graduate School of Medicine, 13-1, Takara-machi, Kanazawa, Ishikawa 920-8641, Japan;8. Department of Respiratory Medicine, Kanazawa Municipal Hospital, 3-7-3, Heiwa-machi, Kanazawa, Ishikawa 921-8105, Japan;9. Department of Respiratory Medicine, National Hospital Organization Nanao Hospital, 3-1, Mattoumachi-hachibu, Nanao, Ishikawa, Japan;1. Department of Respiratory Medicine, Fukuoka University Hospital, Fukuoka, Japan;2. Department of Respiratory Medicine, Kanagawa Cardiovascular and Respiratory Center, Kanagawa, Japan;3. Department of Respiratory Medicine, Cellular Translation Biology, Kanazawa University Graduate School of Medicine, Japan;4. Department of Respiratory Medicine, JR Tokyo General Hospital, Japan;5. Department of Respiratory Medicine, National Hospital Organization Kinki-Chuo Chest Medical Center, Japan;6. Department of Respiratory Medicine, Chiba University Hospital, Japan;7. Department of Respiratory Medicine, Tosei General Hospital, Japan;8. Second Division, Department of Internal Medicine, Hamamatsu University School of Medicine, Japan;9. Department of Analytic Human Pathology, Nippon Medical School, Tokyo, Japan;10. Department of Clinical Research, National Hospital Organization Tokyo National Hospital, Japan;11. Department of Radiology, Kinki Central Hospital of Mutual Aid Association of Public School Teachers, Hyogo, Japan;12. Department of Diagnostic Radiology, Saitama International Medical Center, Saitama Medical University, Saitama, Japan;13. Department of Pathology, Japanese Red Cross Medical Center, Tokyo, Japan;14. Division of Pathology, Saitama Cardiovascular and Respiratory Center, Saitama, Japan |
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| Abstract: | BackgroundPleuroparenchymal fibroelastosis (PPFE) is a rare idiopathic interstitial pneumonia characterized by pleural and parenchymal involvements predominantly in the upper lobes. Unilateral upper-lung field pulmonary fibrosis (upper-PF) radiologically consistent with PPFE was recently reported in patients with a history of open thoracotomy and presented with impaired thoracic movements in the operated side with unknown mechanisms. This retrospective study aimed to elucidate the clinical and radiological courses and pathological findings of unilateral upper-PF.MethodsAll the consecutive patients diagnosed as having unilateral upper-PF between March 2012 and April 2018 were included. Radiological images and clinical courses before and after the diagnosis were thoroughly reviewed.ResultsFourteen patients were included. Unilateral upper-PF was diagnosed after a median of 4.8 years from the open thoracotomy or video-assisted thoracic surgery for treating lung or esophageal cancer, or bronchiectasis. Before or at diagnosis, 12 (85.7%) of 14 patients developed aberrant intrathoracic/extrathoracic air suggestive of pleural fistula, although the degree was slight. Of note, the upper-PF lesion apparently deteriorated once aberrant air emerged in all the patients. After diagnosis, the upper-PF lesion transformed into cystic lesion in 9 patients, 4 of whom eventually developed pulmonary aspergillosis. The prognosis was poor, with a median overall survival of 49.3 months. The autopsy in one patient demonstrated findings consistent with PPFE and chronic pleuritis.ConclusionsUnilateral upper-PF developed after thoracic surgeries and had many clinical, radiological, and pathological characteristics in common with idiopathic PPFE. Our results indicate that the commonly observed aberrant air may be correlated with disease development and progression. |
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| Keywords: | Pleural fistula Chronic pleuritis Unilateral Pleuroparenchymal fibroelastosis Aberrant air BMI" },{" #name" :" keyword" ," $" :{" id" :" kwrd0040" }," $$" :[{" #name" :" text" ," _" :" body mass index CT" },{" #name" :" keyword" ," $" :{" id" :" kwrd0050" }," $$" :[{" #name" :" text" ," _" :" computed tomography FDG-PET" },{" #name" :" keyword" ," $" :{" id" :" kwrd0060" }," $$" :[{" #name" :" text" ," _" :" 18F-fluorodeoxyglucose positron emission tomography FEV1" },{" #name" :" keyword" ," $" :{" id" :" kwrd0070" }," $$" :[{" #name" :" text" ," _" :" forced expiratory volume in 1 s FVC" },{" #name" :" keyword" ," $" :{" id" :" kwrd0080" }," $$" :[{" #name" :" text" ," _" :" forced vital capacity PPFE" },{" #name" :" keyword" ," $" :{" id" :" kwrd0090" }," $$" :[{" #name" :" text" ," _" :" pleuro-parenchymal fibroelastosis SUVmax" },{" #name" :" keyword" ," $" :{" id" :" kwrd0100" }," $$" :[{" #name" :" text" ," _" :" maximum standardized uptake value VATS" },{" #name" :" keyword" ," $" :{" id" :" kwrd0110" }," $$" :[{" #name" :" text" ," _" :" video-assisted thoracic surgery |
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