Invasive pulmonary mucormycosis: rare presentation with pulmonary eosinophilia

Abstract Background Fungi can cause a variety of infectious diseases, including invasive mycosis and non-invasive mycosis, as well as allergic diseases. The different forms of mycosis usually have been described as mutually exclusive, independent entities, with few descriptions of overlapping cases....

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Main Authors: Taizou Hirano, Mitsuhiro Yamada, Kei Sato, Koji Murakami, Tokiwa Tamai, Yoshiya Mitsuhashi, Tsutomu Tamada, Hisatoshi Sugiura, Naomi Sato, Ryoko Saito, Junya Tominaga, Akira Watanabe, Masakazu Ichinose
Format: Article
Language:English
Published: BMC 2017-04-01
Series:BMC Pulmonary Medicine
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Online Access:http://link.springer.com/article/10.1186/s12890-017-0419-1
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author Taizou Hirano
Mitsuhiro Yamada
Kei Sato
Koji Murakami
Tokiwa Tamai
Yoshiya Mitsuhashi
Tsutomu Tamada
Hisatoshi Sugiura
Naomi Sato
Ryoko Saito
Junya Tominaga
Akira Watanabe
Masakazu Ichinose
author_facet Taizou Hirano
Mitsuhiro Yamada
Kei Sato
Koji Murakami
Tokiwa Tamai
Yoshiya Mitsuhashi
Tsutomu Tamada
Hisatoshi Sugiura
Naomi Sato
Ryoko Saito
Junya Tominaga
Akira Watanabe
Masakazu Ichinose
author_sort Taizou Hirano
collection DOAJ
description Abstract Background Fungi can cause a variety of infectious diseases, including invasive mycosis and non-invasive mycosis, as well as allergic diseases. The different forms of mycosis usually have been described as mutually exclusive, independent entities, with few descriptions of overlapping cases. Here, we describe the first reported case of a patient with the complication of pulmonary eosinophilia in the course of invasive mucormycosis. Case presentation A 74-year-old Japanese man with asthma-COPD overlap underwent emergency surgery for a ruptured abdominal aortic aneurysm. The surgery was successful, but fever and worsening dyspnea appeared and continued from postoperative day (POD) 10. A complete blood count showed leukocytosis with neutrophilia and eosinophilia, and the chest X-ray showed consolidation of the left upper lung at POD 15. We suspected nosocomial pneumonia together with an exacerbation of the asthma-COPD overlap, and both antibiotics and bronchodilator therapy were initiated. However, the symptoms, eosinophilia and imaging findings deteriorated. We then performed a bronchoscopy, and bronchoalveolar lavage (BAL) fluid analysis revealed an increased percentage of eosinophils (82% of whole cells) as well as filamentous fungi. We first suspected that this was a case of allergic bronchopulmonary mycosis (ABPM) caused by Aspergillus infection and began corticosteroid therapy with an intravenous administration of voriconazole at POD 27. However, the fungal culture examination of the BAL fluid revealed mucormycetes, which were later identified as Cunninghamella bertholletiae by PCR and DNA sequencing. We then switched the antifungal agent to liposomal amphotericin B for the treatment of the pulmonary mucormycosis at POD 29. Despite replacing voriconazole with liposomal amphotericin B, the patient developed septic shock and died at POD 39. The autopsy revealed that filamentous fungi had invaded the lung, heart, thyroid glands, kidneys, and spleen, suggesting that disseminated mucormycosis had occurred. Conclusions We describe the first reported case of pulmonary mucormycosis with pulmonary eosinophilia caused by Cunninghamella bertholletiae, which resulted in disseminated mucormycosis. Although it is a rather rare case, two important conclusions can be drawn: i) mycosis can simultaneously cause both invasive infection and a host allergic reaction, and ii) Cunninghamella bertholletiae rarely infects immunocompetent patients.
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spelling doaj.art-33f6db705997497a9959b78e9698ecbd2022-12-21T18:14:51ZengBMCBMC Pulmonary Medicine1471-24662017-04-011711610.1186/s12890-017-0419-1Invasive pulmonary mucormycosis: rare presentation with pulmonary eosinophiliaTaizou Hirano0Mitsuhiro Yamada1Kei Sato2Koji Murakami3Tokiwa Tamai4Yoshiya Mitsuhashi5Tsutomu Tamada6Hisatoshi Sugiura7Naomi Sato8Ryoko Saito9Junya Tominaga10Akira Watanabe11Masakazu Ichinose12Department of Respiratory Medicine, Tohoku University Graduate School of MedicineDepartment of Respiratory Medicine, Tohoku University Graduate School of MedicineDepartment of Respiratory Medicine, Tohoku University Graduate School of MedicineDepartment of Respiratory Medicine, Tohoku University Graduate School of MedicineDepartment of Respiratory Medicine, Tohoku University Graduate School of MedicineDepartment of Respiratory Medicine, Tohoku University Graduate School of MedicineDepartment of Respiratory Medicine, Tohoku University Graduate School of MedicineDepartment of Respiratory Medicine, Tohoku University Graduate School of MedicineDepartment of Anatomic Pathology, Tohoku University Graduate School of MedicineDepartment of Anatomic Pathology, Tohoku University Graduate School of MedicineDepartment of Diagnostic Radiology, Tohoku University Graduate School of MedicineResearch Division for Development of Anti-Infective Agents, Institute of Development, Aging and Cancer, Tohoku UniversityDepartment of Respiratory Medicine, Tohoku University Graduate School of MedicineAbstract Background Fungi can cause a variety of infectious diseases, including invasive mycosis and non-invasive mycosis, as well as allergic diseases. The different forms of mycosis usually have been described as mutually exclusive, independent entities, with few descriptions of overlapping cases. Here, we describe the first reported case of a patient with the complication of pulmonary eosinophilia in the course of invasive mucormycosis. Case presentation A 74-year-old Japanese man with asthma-COPD overlap underwent emergency surgery for a ruptured abdominal aortic aneurysm. The surgery was successful, but fever and worsening dyspnea appeared and continued from postoperative day (POD) 10. A complete blood count showed leukocytosis with neutrophilia and eosinophilia, and the chest X-ray showed consolidation of the left upper lung at POD 15. We suspected nosocomial pneumonia together with an exacerbation of the asthma-COPD overlap, and both antibiotics and bronchodilator therapy were initiated. However, the symptoms, eosinophilia and imaging findings deteriorated. We then performed a bronchoscopy, and bronchoalveolar lavage (BAL) fluid analysis revealed an increased percentage of eosinophils (82% of whole cells) as well as filamentous fungi. We first suspected that this was a case of allergic bronchopulmonary mycosis (ABPM) caused by Aspergillus infection and began corticosteroid therapy with an intravenous administration of voriconazole at POD 27. However, the fungal culture examination of the BAL fluid revealed mucormycetes, which were later identified as Cunninghamella bertholletiae by PCR and DNA sequencing. We then switched the antifungal agent to liposomal amphotericin B for the treatment of the pulmonary mucormycosis at POD 29. Despite replacing voriconazole with liposomal amphotericin B, the patient developed septic shock and died at POD 39. The autopsy revealed that filamentous fungi had invaded the lung, heart, thyroid glands, kidneys, and spleen, suggesting that disseminated mucormycosis had occurred. Conclusions We describe the first reported case of pulmonary mucormycosis with pulmonary eosinophilia caused by Cunninghamella bertholletiae, which resulted in disseminated mucormycosis. Although it is a rather rare case, two important conclusions can be drawn: i) mycosis can simultaneously cause both invasive infection and a host allergic reaction, and ii) Cunninghamella bertholletiae rarely infects immunocompetent patients.http://link.springer.com/article/10.1186/s12890-017-0419-1MucormycosisPulmonary eosinophiliaCunninghamella bertholletiae
spellingShingle Taizou Hirano
Mitsuhiro Yamada
Kei Sato
Koji Murakami
Tokiwa Tamai
Yoshiya Mitsuhashi
Tsutomu Tamada
Hisatoshi Sugiura
Naomi Sato
Ryoko Saito
Junya Tominaga
Akira Watanabe
Masakazu Ichinose
Invasive pulmonary mucormycosis: rare presentation with pulmonary eosinophilia
BMC Pulmonary Medicine
Mucormycosis
Pulmonary eosinophilia
Cunninghamella bertholletiae
title Invasive pulmonary mucormycosis: rare presentation with pulmonary eosinophilia
title_full Invasive pulmonary mucormycosis: rare presentation with pulmonary eosinophilia
title_fullStr Invasive pulmonary mucormycosis: rare presentation with pulmonary eosinophilia
title_full_unstemmed Invasive pulmonary mucormycosis: rare presentation with pulmonary eosinophilia
title_short Invasive pulmonary mucormycosis: rare presentation with pulmonary eosinophilia
title_sort invasive pulmonary mucormycosis rare presentation with pulmonary eosinophilia
topic Mucormycosis
Pulmonary eosinophilia
Cunninghamella bertholletiae
url http://link.springer.com/article/10.1186/s12890-017-0419-1
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