Pulmonary capillary hemangiomatosis diagnosed by pathology of explanted lungs: a unique etiology serves as a key of clinical diagnosis
A 27-year-old female patient had presented progressing exertional dyspnea due to pulmonary hypertension. Chest CT revealed diffusely spread patchy ground-glass opacities sparing subpleural parenchymal areas suggesting the diagnosis of pulmonary veno-occlusive disease (PVOD). Despite the diagnosis of...
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| Veröffentlicht in: | General thoracic and cardiovascular surgery 2019-03, Vol.67 (3), p.332-335 |
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| container_title | General thoracic and cardiovascular surgery |
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| creator | Wada, Hironobu Nakajima, Takahiro Suzuki, Hidemi Anazawa, Rie Narita, Tomoharu Terada, Jiro Yoshida, Shigetoshi Tatsumi, Koichiro Nakatani, Yukio Yoshino, Ichiro |
| description | A 27-year-old female patient had presented progressing exertional dyspnea due to pulmonary hypertension. Chest CT revealed diffusely spread patchy ground-glass opacities sparing subpleural parenchymal areas suggesting the diagnosis of pulmonary veno-occlusive disease (PVOD). Despite the diagnosis of PVOD, she was somehow managed by a repetitive escalation of the epoprostenol dose and oxygen supply during the 12-month waiting period until successful bilateral lung transplantation was performed. Pathology demonstrated capillary proliferation in alveolar septae with scarce lesions of narrowed and/or occluded postcapillary small veins, leading to the final diagnosis of pulmonary capillary hemangiomatosis (PCH), not PVOD. We herein present a case of PCH diagnosed after lung transplantation with a focus on its etiology and a key to clinical diagnosis. |
| doi_str_mv | 10.1007/s11748-018-0950-x |
| format | Article |
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Chest CT revealed diffusely spread patchy ground-glass opacities sparing subpleural parenchymal areas suggesting the diagnosis of pulmonary veno-occlusive disease (PVOD). Despite the diagnosis of PVOD, she was somehow managed by a repetitive escalation of the epoprostenol dose and oxygen supply during the 12-month waiting period until successful bilateral lung transplantation was performed. Pathology demonstrated capillary proliferation in alveolar septae with scarce lesions of narrowed and/or occluded postcapillary small veins, leading to the final diagnosis of pulmonary capillary hemangiomatosis (PCH), not PVOD. We herein present a case of PCH diagnosed after lung transplantation with a focus on its etiology and a key to clinical diagnosis.</description><identifier>ISSN: 1863-6705</identifier><identifier>EISSN: 1863-6713</identifier><identifier>DOI: 10.1007/s11748-018-0950-x</identifier><identifier>PMID: 29804176</identifier><language>eng</language><publisher>Singapore: Springer Singapore</publisher><subject>Cardiac Surgery ; Cardiology ; Case Report ; Case reports ; Disease ; Etiology ; Hemoptysis ; Lung transplants ; Lymphatic system ; Medicine ; Medicine & Public Health ; Pathology ; Patients ; Pulmonary arteries ; Pulmonary hypertension ; Surgical Oncology ; Thoracic Surgery ; Veins & arteries ; Ventilation</subject><ispartof>General thoracic and cardiovascular surgery, 2019-03, Vol.67 (3), p.332-335</ispartof><rights>The Japanese Association for Thoracic Surgery 2018</rights><rights>The Japanese Association for Thoracic Surgery 2018.</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c396t-7696f9104f9ae893d72042bb9d9af12508ed0aff3778b74c609a5a8c2bcff2713</citedby><cites>FETCH-LOGICAL-c396t-7696f9104f9ae893d72042bb9d9af12508ed0aff3778b74c609a5a8c2bcff2713</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://link.springer.com/content/pdf/10.1007/s11748-018-0950-x$$EPDF$$P50$$Gspringer$$H</linktopdf><linktohtml>$$Uhttps://www.proquest.com/docview/2918739929?pq-origsite=primo$$EHTML$$P50$$Gproquest$$H</linktohtml><link.rule.ids>315,781,785,21393,21394,27929,27930,33535,33536,33749,33750,41493,42562,43664,43810,51324,64390,64392,64394,72474</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/29804176$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Wada, Hironobu</creatorcontrib><creatorcontrib>Nakajima, Takahiro</creatorcontrib><creatorcontrib>Suzuki, Hidemi</creatorcontrib><creatorcontrib>Anazawa, Rie</creatorcontrib><creatorcontrib>Narita, Tomoharu</creatorcontrib><creatorcontrib>Terada, Jiro</creatorcontrib><creatorcontrib>Yoshida, Shigetoshi</creatorcontrib><creatorcontrib>Tatsumi, Koichiro</creatorcontrib><creatorcontrib>Nakatani, Yukio</creatorcontrib><creatorcontrib>Yoshino, Ichiro</creatorcontrib><title>Pulmonary capillary hemangiomatosis diagnosed by pathology of explanted lungs: a unique etiology serves as a key of clinical diagnosis</title><title>General thoracic and cardiovascular surgery</title><addtitle>Gen Thorac Cardiovasc Surg</addtitle><addtitle>Gen Thorac Cardiovasc Surg</addtitle><description>A 27-year-old female patient had presented progressing exertional dyspnea due to pulmonary hypertension. Chest CT revealed diffusely spread patchy ground-glass opacities sparing subpleural parenchymal areas suggesting the diagnosis of pulmonary veno-occlusive disease (PVOD). Despite the diagnosis of PVOD, she was somehow managed by a repetitive escalation of the epoprostenol dose and oxygen supply during the 12-month waiting period until successful bilateral lung transplantation was performed. Pathology demonstrated capillary proliferation in alveolar septae with scarce lesions of narrowed and/or occluded postcapillary small veins, leading to the final diagnosis of pulmonary capillary hemangiomatosis (PCH), not PVOD. We herein present a case of PCH diagnosed after lung transplantation with a focus on its etiology and a key to clinical diagnosis.</description><subject>Cardiac Surgery</subject><subject>Cardiology</subject><subject>Case Report</subject><subject>Case reports</subject><subject>Disease</subject><subject>Etiology</subject><subject>Hemoptysis</subject><subject>Lung transplants</subject><subject>Lymphatic system</subject><subject>Medicine</subject><subject>Medicine & Public Health</subject><subject>Pathology</subject><subject>Patients</subject><subject>Pulmonary arteries</subject><subject>Pulmonary hypertension</subject><subject>Surgical Oncology</subject><subject>Thoracic Surgery</subject><subject>Veins & arteries</subject><subject>Ventilation</subject><issn>1863-6705</issn><issn>1863-6713</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2019</creationdate><recordtype>article</recordtype><sourceid>ABUWG</sourceid><sourceid>AFKRA</sourceid><sourceid>BENPR</sourceid><sourceid>CCPQU</sourceid><recordid>eNp1kctu1TAQhi1ERS_wAGyQJTZsAraT-MIOVUCRKtFFWVuOY6cujh0ySXXOC_Dc-DRtkZCQbHmk-eaf8fwIvabkPSVEfABKRSMrQstVLal2z9AJlbyuuKD186eYtMfoFOCWkJZL2r5Ax0xJ0lDBT9DvqzWOOZl5j62ZQoyH6MaNJg0hj2bJEAD3wQwpg-txt8eTWW5yzMMeZ4_dboomLSUT1zTAR2zwmsKv1WG3hI0CN985wKYc_NPdV9kYUrAmPgoHeImOvIngXj28Z-jHl8_X5xfV5fev384_XVa2VnypBFfcK0oar4yTqu4FIw3rOtUr4ylriXQ9Md7XQshONJYTZVojLeus96ws5Qy923SnOZcpYdFjAOvKt5PLK-gixwllQjQFffsPepvXOZXpNFNUiloppgpFN8rOGWB2Xk9zGMsSNSX6YJLeTNLFJH0wSe9KzZsH5bUbXf9U8ehKAdgGQEmlwc1_W_9f9Q-6lZ-a</recordid><startdate>20190301</startdate><enddate>20190301</enddate><creator>Wada, Hironobu</creator><creator>Nakajima, Takahiro</creator><creator>Suzuki, Hidemi</creator><creator>Anazawa, Rie</creator><creator>Narita, Tomoharu</creator><creator>Terada, Jiro</creator><creator>Yoshida, Shigetoshi</creator><creator>Tatsumi, Koichiro</creator><creator>Nakatani, Yukio</creator><creator>Yoshino, Ichiro</creator><general>Springer Singapore</general><general>Springer Nature B.V</general><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>3V.</scope><scope>7X7</scope><scope>7XB</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>BENPR</scope><scope>CCPQU</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>K9.</scope><scope>M0S</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>7X8</scope></search><sort><creationdate>20190301</creationdate><title>Pulmonary capillary hemangiomatosis diagnosed by pathology of explanted lungs: a unique etiology serves as a key of clinical diagnosis</title><author>Wada, Hironobu ; Nakajima, Takahiro ; Suzuki, Hidemi ; Anazawa, Rie ; Narita, Tomoharu ; Terada, Jiro ; Yoshida, Shigetoshi ; Tatsumi, Koichiro ; Nakatani, Yukio ; Yoshino, Ichiro</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c396t-7696f9104f9ae893d72042bb9d9af12508ed0aff3778b74c609a5a8c2bcff2713</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2019</creationdate><topic>Cardiac Surgery</topic><topic>Cardiology</topic><topic>Case Report</topic><topic>Case reports</topic><topic>Disease</topic><topic>Etiology</topic><topic>Hemoptysis</topic><topic>Lung transplants</topic><topic>Lymphatic system</topic><topic>Medicine</topic><topic>Medicine & Public Health</topic><topic>Pathology</topic><topic>Patients</topic><topic>Pulmonary arteries</topic><topic>Pulmonary hypertension</topic><topic>Surgical Oncology</topic><topic>Thoracic Surgery</topic><topic>Veins & arteries</topic><topic>Ventilation</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Wada, Hironobu</creatorcontrib><creatorcontrib>Nakajima, Takahiro</creatorcontrib><creatorcontrib>Suzuki, Hidemi</creatorcontrib><creatorcontrib>Anazawa, Rie</creatorcontrib><creatorcontrib>Narita, Tomoharu</creatorcontrib><creatorcontrib>Terada, Jiro</creatorcontrib><creatorcontrib>Yoshida, Shigetoshi</creatorcontrib><creatorcontrib>Tatsumi, Koichiro</creatorcontrib><creatorcontrib>Nakatani, Yukio</creatorcontrib><creatorcontrib>Yoshino, Ichiro</creatorcontrib><collection>PubMed</collection><collection>CrossRef</collection><collection>ProQuest Central (Corporate)</collection><collection>Health & Medical Collection</collection><collection>ProQuest Central (purchase pre-March 2016)</collection><collection>Hospital Premium Collection</collection><collection>Hospital Premium Collection (Alumni Edition)</collection><collection>ProQuest Central (Alumni) (purchase pre-March 2016)</collection><collection>ProQuest Central (Alumni Edition)</collection><collection>ProQuest Central UK/Ireland</collection><collection>ProQuest Central</collection><collection>ProQuest One Community College</collection><collection>Health Research Premium Collection</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Health & Medical Collection (Alumni Edition)</collection><collection>ProQuest One Academic Eastern Edition (DO NOT USE)</collection><collection>ProQuest One Academic</collection><collection>ProQuest One Academic UKI Edition</collection><collection>MEDLINE - Academic</collection><jtitle>General thoracic and cardiovascular surgery</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Wada, Hironobu</au><au>Nakajima, Takahiro</au><au>Suzuki, Hidemi</au><au>Anazawa, Rie</au><au>Narita, Tomoharu</au><au>Terada, Jiro</au><au>Yoshida, Shigetoshi</au><au>Tatsumi, Koichiro</au><au>Nakatani, Yukio</au><au>Yoshino, Ichiro</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Pulmonary capillary hemangiomatosis diagnosed by pathology of explanted lungs: a unique etiology serves as a key of clinical diagnosis</atitle><jtitle>General thoracic and cardiovascular surgery</jtitle><stitle>Gen Thorac Cardiovasc Surg</stitle><addtitle>Gen Thorac Cardiovasc Surg</addtitle><date>2019-03-01</date><risdate>2019</risdate><volume>67</volume><issue>3</issue><spage>332</spage><epage>335</epage><pages>332-335</pages><issn>1863-6705</issn><eissn>1863-6713</eissn><abstract>A 27-year-old female patient had presented progressing exertional dyspnea due to pulmonary hypertension. Chest CT revealed diffusely spread patchy ground-glass opacities sparing subpleural parenchymal areas suggesting the diagnosis of pulmonary veno-occlusive disease (PVOD). Despite the diagnosis of PVOD, she was somehow managed by a repetitive escalation of the epoprostenol dose and oxygen supply during the 12-month waiting period until successful bilateral lung transplantation was performed. Pathology demonstrated capillary proliferation in alveolar septae with scarce lesions of narrowed and/or occluded postcapillary small veins, leading to the final diagnosis of pulmonary capillary hemangiomatosis (PCH), not PVOD. We herein present a case of PCH diagnosed after lung transplantation with a focus on its etiology and a key to clinical diagnosis.</abstract><cop>Singapore</cop><pub>Springer Singapore</pub><pmid>29804176</pmid><doi>10.1007/s11748-018-0950-x</doi><tpages>4</tpages></addata></record> |
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| subjects | Cardiac Surgery Cardiology Case Report Case reports Disease Etiology Hemoptysis Lung transplants Lymphatic system Medicine Medicine & Public Health Pathology Patients Pulmonary arteries Pulmonary hypertension Surgical Oncology Thoracic Surgery Veins & arteries Ventilation |
| title | Pulmonary capillary hemangiomatosis diagnosed by pathology of explanted lungs: a unique etiology serves as a key of clinical diagnosis |
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