An Anomalous Unilateral Single Pulmonary Vein Associated with a Bone Morphogenetic Protein Receptor II Gene Mutation
Anomalous unilateral single pulmonary vein (AUSPV), a rare congenital anomaly, is associated with an aberrant course but normal drainage, and resembles arteriovenous malformation (AVM). We treated a 26-year-old man with AUSPV in the right lung and an anomalous segmental pulmonary vein in the left lu...
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| Veröffentlicht in: | Internal Medicine 2014, Vol.53(5), pp.461-466 |
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| creator | Koyama, Kazuya Sano, Go Hata, Yoshinobu Shiraga, Nobuyuki Ota, Hiroki Sugino, Keishi Isobe, Kazutoshi Sakamoto, Susumu Takai, Yujiro Koezuka, Satoshi Makino, Takashi Otsuka, Hajime Sato, Fumitomo Sasamoto, Shuichi Iyoda, Akira Kurosaki, Atsuko Homma, Sakae |
| description | Anomalous unilateral single pulmonary vein (AUSPV), a rare congenital anomaly, is associated with an aberrant course but normal drainage, and resembles arteriovenous malformation (AVM). We treated a 26-year-old man with AUSPV in the right lung and an anomalous segmental pulmonary vein in the left lung. CT revealed a tortuous vascular shadow with an enhancement pattern identical to that of the pulmonary vein, suggesting AUSPV. This was confirmed by pulmonary angiography. Although pulmonary AVMs were not detected on angiography, microvascular AVMs could not be excluded because delayed bubbles appeared on contrast echocardiography. A genetic examination revealed a missense mutation of BMPR2. |
| doi_str_mv | 10.2169/internalmedicine.53.9245 |
| format | Article |
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We treated a 26-year-old man with AUSPV in the right lung and an anomalous segmental pulmonary vein in the left lung. CT revealed a tortuous vascular shadow with an enhancement pattern identical to that of the pulmonary vein, suggesting AUSPV. This was confirmed by pulmonary angiography. Although pulmonary AVMs were not detected on angiography, microvascular AVMs could not be excluded because delayed bubbles appeared on contrast echocardiography. A genetic examination revealed a missense mutation of BMPR2.</description><identifier>ISSN: 0918-2918</identifier><identifier>EISSN: 1349-7235</identifier><identifier>DOI: 10.2169/internalmedicine.53.9245</identifier><identifier>PMID: 24583436</identifier><language>eng</language><publisher>Japan: The Japanese Society of Internal Medicine</publisher><subject>Adult ; Angiography ; anomalous unilateral single pulmonary vein ; Arteriovenous Malformations - diagnosis ; Arteriovenous Malformations - genetics ; Arteriovenous Malformations - metabolism ; BMPR2 ; Bone Morphogenetic Protein Receptors, Type II - genetics ; Diagnosis, Differential ; DNA - genetics ; DNA Mutational Analysis ; Echocardiography ; Humans ; Lung - diagnostic imaging ; Male ; Mutation ; pulmonary angiography ; pulmonary arteriovenous malformation ; pulmonary varix ; Pulmonary Veins - abnormalities ; Tomography, X-Ray Computed</subject><ispartof>Internal Medicine, 2014, Vol.53(5), pp.461-466</ispartof><rights>2014 by The Japanese Society of Internal Medicine</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c509t-9429f0e6ac9a2f40ecb539670c3117fd27bbee34310dc4cf25b914b8de1945fa3</citedby><cites>FETCH-LOGICAL-c509t-9429f0e6ac9a2f40ecb539670c3117fd27bbee34310dc4cf25b914b8de1945fa3</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>314,776,780,1876,27903,27904</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/24583436$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Koyama, Kazuya</creatorcontrib><creatorcontrib>Sano, Go</creatorcontrib><creatorcontrib>Hata, Yoshinobu</creatorcontrib><creatorcontrib>Shiraga, Nobuyuki</creatorcontrib><creatorcontrib>Ota, Hiroki</creatorcontrib><creatorcontrib>Sugino, Keishi</creatorcontrib><creatorcontrib>Isobe, Kazutoshi</creatorcontrib><creatorcontrib>Sakamoto, Susumu</creatorcontrib><creatorcontrib>Takai, Yujiro</creatorcontrib><creatorcontrib>Koezuka, Satoshi</creatorcontrib><creatorcontrib>Makino, Takashi</creatorcontrib><creatorcontrib>Otsuka, Hajime</creatorcontrib><creatorcontrib>Sato, Fumitomo</creatorcontrib><creatorcontrib>Sasamoto, Shuichi</creatorcontrib><creatorcontrib>Iyoda, Akira</creatorcontrib><creatorcontrib>Kurosaki, Atsuko</creatorcontrib><creatorcontrib>Homma, Sakae</creatorcontrib><title>An Anomalous Unilateral Single Pulmonary Vein Associated with a Bone Morphogenetic Protein Receptor II Gene Mutation</title><title>Internal Medicine</title><addtitle>Intern. Med.</addtitle><description>Anomalous unilateral single pulmonary vein (AUSPV), a rare congenital anomaly, is associated with an aberrant course but normal drainage, and resembles arteriovenous malformation (AVM). We treated a 26-year-old man with AUSPV in the right lung and an anomalous segmental pulmonary vein in the left lung. CT revealed a tortuous vascular shadow with an enhancement pattern identical to that of the pulmonary vein, suggesting AUSPV. This was confirmed by pulmonary angiography. Although pulmonary AVMs were not detected on angiography, microvascular AVMs could not be excluded because delayed bubbles appeared on contrast echocardiography. A genetic examination revealed a missense mutation of BMPR2.</description><subject>Adult</subject><subject>Angiography</subject><subject>anomalous unilateral single pulmonary vein</subject><subject>Arteriovenous Malformations - diagnosis</subject><subject>Arteriovenous Malformations - genetics</subject><subject>Arteriovenous Malformations - metabolism</subject><subject>BMPR2</subject><subject>Bone Morphogenetic Protein Receptors, Type II - genetics</subject><subject>Diagnosis, Differential</subject><subject>DNA - genetics</subject><subject>DNA Mutational Analysis</subject><subject>Echocardiography</subject><subject>Humans</subject><subject>Lung - diagnostic imaging</subject><subject>Male</subject><subject>Mutation</subject><subject>pulmonary angiography</subject><subject>pulmonary arteriovenous malformation</subject><subject>pulmonary varix</subject><subject>Pulmonary Veins - abnormalities</subject><subject>Tomography, X-Ray Computed</subject><issn>0918-2918</issn><issn>1349-7235</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2014</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNqF0Utv1DAQAGALUdFt4S8gH7lk62cSH5eKlpUKVH1wjRxnsuvKsRfbEeLf4-2WPSAkLuODv5nRzCCEKVkyWqsL6zNEr90EgzXWw1LypWJCvkILyoWqGsbla7QgirYVK-EUnaX0RAhvG8XeoNNCWy54vUB55fHKh0m7MCf86K3TpbR2-N76jQN8O7speB1_4e9gC00pGFvIgH_avMUafwwe8JcQd9uwAQ_ZGnwbQ97jOzCwyyHi9Rpfw57NWWcb_Ft0MmqX4N3Le44erz49XH6ubr5dry9XN5WRROVKCaZGArU2SrNREDC95KpuiOGUNuPAmr4HKHNQMhhhRiZ7RUXfDkCVkKPm5-jDoe4uhh8zpNxNNhlwTnso43a0rokgRDb0_1QSQWsmnml7oCaGlCKM3S7aqayoo6Tbn6f7-zyd5N3-PCX1_UuXuS-fx8Q_9yjg6wE8paw3cAQ6lsU6-Gdl-RwOHY7QbHXswPPf6nKu7g</recordid><startdate>20140101</startdate><enddate>20140101</enddate><creator>Koyama, Kazuya</creator><creator>Sano, Go</creator><creator>Hata, Yoshinobu</creator><creator>Shiraga, Nobuyuki</creator><creator>Ota, Hiroki</creator><creator>Sugino, Keishi</creator><creator>Isobe, Kazutoshi</creator><creator>Sakamoto, Susumu</creator><creator>Takai, Yujiro</creator><creator>Koezuka, Satoshi</creator><creator>Makino, Takashi</creator><creator>Otsuka, Hajime</creator><creator>Sato, Fumitomo</creator><creator>Sasamoto, Shuichi</creator><creator>Iyoda, Akira</creator><creator>Kurosaki, Atsuko</creator><creator>Homma, Sakae</creator><general>The Japanese Society of Internal Medicine</general><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7X8</scope><scope>8FD</scope><scope>FR3</scope><scope>P64</scope><scope>RC3</scope></search><sort><creationdate>20140101</creationdate><title>An Anomalous Unilateral Single Pulmonary Vein Associated with a Bone Morphogenetic Protein Receptor II Gene Mutation</title><author>Koyama, Kazuya ; Sano, Go ; Hata, Yoshinobu ; Shiraga, Nobuyuki ; Ota, Hiroki ; Sugino, Keishi ; Isobe, Kazutoshi ; Sakamoto, Susumu ; Takai, Yujiro ; Koezuka, Satoshi ; Makino, Takashi ; Otsuka, Hajime ; Sato, Fumitomo ; Sasamoto, Shuichi ; Iyoda, Akira ; Kurosaki, Atsuko ; Homma, Sakae</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c509t-9429f0e6ac9a2f40ecb539670c3117fd27bbee34310dc4cf25b914b8de1945fa3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2014</creationdate><topic>Adult</topic><topic>Angiography</topic><topic>anomalous unilateral single pulmonary vein</topic><topic>Arteriovenous Malformations - diagnosis</topic><topic>Arteriovenous Malformations - genetics</topic><topic>Arteriovenous Malformations - metabolism</topic><topic>BMPR2</topic><topic>Bone Morphogenetic Protein Receptors, Type II - genetics</topic><topic>Diagnosis, Differential</topic><topic>DNA - genetics</topic><topic>DNA Mutational Analysis</topic><topic>Echocardiography</topic><topic>Humans</topic><topic>Lung - diagnostic imaging</topic><topic>Male</topic><topic>Mutation</topic><topic>pulmonary angiography</topic><topic>pulmonary arteriovenous malformation</topic><topic>pulmonary varix</topic><topic>Pulmonary Veins - abnormalities</topic><topic>Tomography, X-Ray Computed</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Koyama, Kazuya</creatorcontrib><creatorcontrib>Sano, Go</creatorcontrib><creatorcontrib>Hata, Yoshinobu</creatorcontrib><creatorcontrib>Shiraga, Nobuyuki</creatorcontrib><creatorcontrib>Ota, Hiroki</creatorcontrib><creatorcontrib>Sugino, Keishi</creatorcontrib><creatorcontrib>Isobe, Kazutoshi</creatorcontrib><creatorcontrib>Sakamoto, Susumu</creatorcontrib><creatorcontrib>Takai, Yujiro</creatorcontrib><creatorcontrib>Koezuka, Satoshi</creatorcontrib><creatorcontrib>Makino, Takashi</creatorcontrib><creatorcontrib>Otsuka, Hajime</creatorcontrib><creatorcontrib>Sato, Fumitomo</creatorcontrib><creatorcontrib>Sasamoto, Shuichi</creatorcontrib><creatorcontrib>Iyoda, Akira</creatorcontrib><creatorcontrib>Kurosaki, Atsuko</creatorcontrib><creatorcontrib>Homma, Sakae</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>MEDLINE - Academic</collection><collection>Technology Research Database</collection><collection>Engineering Research Database</collection><collection>Biotechnology and BioEngineering Abstracts</collection><collection>Genetics Abstracts</collection><jtitle>Internal Medicine</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Koyama, Kazuya</au><au>Sano, Go</au><au>Hata, Yoshinobu</au><au>Shiraga, Nobuyuki</au><au>Ota, Hiroki</au><au>Sugino, Keishi</au><au>Isobe, Kazutoshi</au><au>Sakamoto, Susumu</au><au>Takai, Yujiro</au><au>Koezuka, Satoshi</au><au>Makino, Takashi</au><au>Otsuka, Hajime</au><au>Sato, Fumitomo</au><au>Sasamoto, Shuichi</au><au>Iyoda, Akira</au><au>Kurosaki, Atsuko</au><au>Homma, Sakae</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>An Anomalous Unilateral Single Pulmonary Vein Associated with a Bone Morphogenetic Protein Receptor II Gene Mutation</atitle><jtitle>Internal Medicine</jtitle><addtitle>Intern. Med.</addtitle><date>2014-01-01</date><risdate>2014</risdate><volume>53</volume><issue>5</issue><spage>461</spage><epage>466</epage><pages>461-466</pages><issn>0918-2918</issn><eissn>1349-7235</eissn><abstract>Anomalous unilateral single pulmonary vein (AUSPV), a rare congenital anomaly, is associated with an aberrant course but normal drainage, and resembles arteriovenous malformation (AVM). We treated a 26-year-old man with AUSPV in the right lung and an anomalous segmental pulmonary vein in the left lung. CT revealed a tortuous vascular shadow with an enhancement pattern identical to that of the pulmonary vein, suggesting AUSPV. This was confirmed by pulmonary angiography. Although pulmonary AVMs were not detected on angiography, microvascular AVMs could not be excluded because delayed bubbles appeared on contrast echocardiography. A genetic examination revealed a missense mutation of BMPR2.</abstract><cop>Japan</cop><pub>The Japanese Society of Internal Medicine</pub><pmid>24583436</pmid><doi>10.2169/internalmedicine.53.9245</doi><tpages>6</tpages><oa>free_for_read</oa></addata></record> |
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| subjects | Adult Angiography anomalous unilateral single pulmonary vein Arteriovenous Malformations - diagnosis Arteriovenous Malformations - genetics Arteriovenous Malformations - metabolism BMPR2 Bone Morphogenetic Protein Receptors, Type II - genetics Diagnosis, Differential DNA - genetics DNA Mutational Analysis Echocardiography Humans Lung - diagnostic imaging Male Mutation pulmonary angiography pulmonary arteriovenous malformation pulmonary varix Pulmonary Veins - abnormalities Tomography, X-Ray Computed |
| title | An Anomalous Unilateral Single Pulmonary Vein Associated with a Bone Morphogenetic Protein Receptor II Gene Mutation |
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