Two Kinds of Cystic Lung Lesions with Pulmonary Lymphangioleiomyomatosis in a Male
A 34-year-old male with frequent recurrence of right pneumothorax was admitted to our hospital. He was a current smoker and outwardly male without genital aplasia. He was diagnosed as tuberous sclerosis complex (TSC) at 2 year-old and underwent transcatheter arterial embolization for right renal hem...
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| Veröffentlicht in: | Annals of Thoracic and Cardiovascular Surgery 2017, Vol.23(1), pp.36-39 |
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| creator | Yamanaka, Sumitaka Mizobuchi, Teruaki Kurihara, Masatoshi |
| description | A 34-year-old male with frequent recurrence of right pneumothorax was admitted to our hospital. He was a current smoker and outwardly male without genital aplasia. He was diagnosed as tuberous sclerosis complex (TSC) at 2 year-old and underwent transcatheter arterial embolization for right renal hemorrhage due to renal tumor 2 years ago. Chest Computed Tomography showed that he had multiple tiny round cystic lesions with thin wall in both lungs. The recurrent pneumothorax was expected to be associated with TSC-Lymphangioleiomyomatosis (LAM). Video-assisted thoracic surgery was successfully performed. The operative and histological findings revealed that the bullae were classified into two groups; emphysematous bullae and bullae due to LAM. His postoperative course was uneventful. TSC-LAM is extremely rare, but in some cases the clinical recognition might be escaped due to subtle findings of bullae in early LAM, resulting in diagnosis as spontaneous pneumothorax. |
| doi_str_mv | 10.5761/atcs.cr.16-00095 |
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He was a current smoker and outwardly male without genital aplasia. He was diagnosed as tuberous sclerosis complex (TSC) at 2 year-old and underwent transcatheter arterial embolization for right renal hemorrhage due to renal tumor 2 years ago. Chest Computed Tomography showed that he had multiple tiny round cystic lesions with thin wall in both lungs. The recurrent pneumothorax was expected to be associated with TSC-Lymphangioleiomyomatosis (LAM). Video-assisted thoracic surgery was successfully performed. The operative and histological findings revealed that the bullae were classified into two groups; emphysematous bullae and bullae due to LAM. His postoperative course was uneventful. TSC-LAM is extremely rare, but in some cases the clinical recognition might be escaped due to subtle findings of bullae in early LAM, resulting in diagnosis as spontaneous pneumothorax.</description><identifier>ISSN: 1341-1098</identifier><identifier>EISSN: 2186-1005</identifier><identifier>DOI: 10.5761/atcs.cr.16-00095</identifier><identifier>PMID: 27349306</identifier><language>eng</language><publisher>Japan: The Editorial Committee of Annals of Thoracic and Cardiovascular Surgery</publisher><subject>Adult ; Biopsy ; Blister - diagnostic imaging ; Blister - etiology ; Blister - surgery ; Case Report ; emphysematous bullae ; Humans ; Immunohistochemistry ; lymphangioleiomyomatosis ; Lymphangioleiomyomatosis - diagnostic imaging ; Lymphangioleiomyomatosis - etiology ; Lymphangioleiomyomatosis - surgery ; Male ; pneumothorax ; Pneumothorax - etiology ; Pulmonary Emphysema - diagnostic imaging ; Pulmonary Emphysema - etiology ; Pulmonary Emphysema - surgery ; Recurrence ; Smoking - adverse effects ; Thoracic Surgery, Video-Assisted ; Tomography, X-Ray Computed ; Treatment Outcome ; Tuberous Sclerosis - complications ; Tuberous Sclerosis - diagnosis ; tuberous sclerosis complex ; video-assisted thoracic surgery</subject><ispartof>Annals of Thoracic and Cardiovascular Surgery, 2017, Vol.23(1), pp.36-39</ispartof><rights>2017 The Editorial Committee of Annals of Thoracic and Cardiovascular Surgery</rights><rights>2017 Annals of Thoracic and Cardiovascular Surgery 2017</rights><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c549t-6e48c7ef285dda2d999e0763fdc361929adfa4ecd148e47bc0e35b9cc9161df03</citedby><cites>FETCH-LOGICAL-c549t-6e48c7ef285dda2d999e0763fdc361929adfa4ecd148e47bc0e35b9cc9161df03</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC5347486/pdf/$$EPDF$$P50$$Gpubmedcentral$$H</linktopdf><linktohtml>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC5347486/$$EHTML$$P50$$Gpubmedcentral$$H</linktohtml><link.rule.ids>230,314,724,777,781,882,1877,27905,27906,53772,53774</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/27349306$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Yamanaka, Sumitaka</creatorcontrib><creatorcontrib>Mizobuchi, Teruaki</creatorcontrib><creatorcontrib>Kurihara, Masatoshi</creatorcontrib><title>Two Kinds of Cystic Lung Lesions with Pulmonary Lymphangioleiomyomatosis in a Male</title><title>Annals of Thoracic and Cardiovascular Surgery</title><addtitle>ATCS</addtitle><description>A 34-year-old male with frequent recurrence of right pneumothorax was admitted to our hospital. He was a current smoker and outwardly male without genital aplasia. He was diagnosed as tuberous sclerosis complex (TSC) at 2 year-old and underwent transcatheter arterial embolization for right renal hemorrhage due to renal tumor 2 years ago. Chest Computed Tomography showed that he had multiple tiny round cystic lesions with thin wall in both lungs. The recurrent pneumothorax was expected to be associated with TSC-Lymphangioleiomyomatosis (LAM). Video-assisted thoracic surgery was successfully performed. The operative and histological findings revealed that the bullae were classified into two groups; emphysematous bullae and bullae due to LAM. His postoperative course was uneventful. TSC-LAM is extremely rare, but in some cases the clinical recognition might be escaped due to subtle findings of bullae in early LAM, resulting in diagnosis as spontaneous pneumothorax.</description><subject>Adult</subject><subject>Biopsy</subject><subject>Blister - diagnostic imaging</subject><subject>Blister - etiology</subject><subject>Blister - surgery</subject><subject>Case Report</subject><subject>emphysematous bullae</subject><subject>Humans</subject><subject>Immunohistochemistry</subject><subject>lymphangioleiomyomatosis</subject><subject>Lymphangioleiomyomatosis - diagnostic imaging</subject><subject>Lymphangioleiomyomatosis - etiology</subject><subject>Lymphangioleiomyomatosis - surgery</subject><subject>Male</subject><subject>pneumothorax</subject><subject>Pneumothorax - etiology</subject><subject>Pulmonary Emphysema - diagnostic imaging</subject><subject>Pulmonary Emphysema - etiology</subject><subject>Pulmonary Emphysema - surgery</subject><subject>Recurrence</subject><subject>Smoking - adverse effects</subject><subject>Thoracic Surgery, Video-Assisted</subject><subject>Tomography, X-Ray Computed</subject><subject>Treatment Outcome</subject><subject>Tuberous Sclerosis - complications</subject><subject>Tuberous Sclerosis - diagnosis</subject><subject>tuberous sclerosis complex</subject><subject>video-assisted thoracic surgery</subject><issn>1341-1098</issn><issn>2186-1005</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2017</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNpVkcFv2yAUh9HUaU273neaOPbiFAzG5jJpipq1qqdVVXtGBJ4TKhsysBf5v5-zdFF64SHxvY-n90PoCyXzohT0RvcmzU2cU5ERQmTxAc1yWomMElKcoRllnE53WZ2ji5ReCWGVEOQTOs9LxiUjYoaenncBPzhvEw4NXoypdwbXg1_jGpILPuGd6zf4cWi74HUccT122432axdacKEbQ6f7kFzCzmONf-oWPqOPjW4TXL3VS_SyvH1e3GX1rx_3i-91Zgou-0wAr0wJTV4V1urcSimBlII11jBBZS61bTQHYymvgJcrQ4AVK2mMpILahrBL9O3g3Q6rDqwB30fdqm103TSoCtqp9y_ebdQ6_FEF4yWvxCS4fhPE8HuA1KvOJQNtqz2EISla5aKkpCz2KDmgJoaUIjTHbyhR-yjUPgploqJC_Ytiavl6Ot6x4f_uJ2B5AF5Tr9dwBHScMmjhYMyZovvjxHwEzEZHBZ79BXnDoes</recordid><startdate>20170101</startdate><enddate>20170101</enddate><creator>Yamanaka, Sumitaka</creator><creator>Mizobuchi, Teruaki</creator><creator>Kurihara, Masatoshi</creator><general>The Editorial Committee of Annals of Thoracic and Cardiovascular Surgery</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>5PM</scope></search><sort><creationdate>20170101</creationdate><title>Two Kinds of Cystic Lung Lesions with Pulmonary Lymphangioleiomyomatosis in a Male</title><author>Yamanaka, Sumitaka ; Mizobuchi, Teruaki ; Kurihara, Masatoshi</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c549t-6e48c7ef285dda2d999e0763fdc361929adfa4ecd148e47bc0e35b9cc9161df03</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2017</creationdate><topic>Adult</topic><topic>Biopsy</topic><topic>Blister - diagnostic imaging</topic><topic>Blister - etiology</topic><topic>Blister - surgery</topic><topic>Case Report</topic><topic>emphysematous bullae</topic><topic>Humans</topic><topic>Immunohistochemistry</topic><topic>lymphangioleiomyomatosis</topic><topic>Lymphangioleiomyomatosis - diagnostic imaging</topic><topic>Lymphangioleiomyomatosis - etiology</topic><topic>Lymphangioleiomyomatosis - surgery</topic><topic>Male</topic><topic>pneumothorax</topic><topic>Pneumothorax - etiology</topic><topic>Pulmonary Emphysema - diagnostic imaging</topic><topic>Pulmonary Emphysema - etiology</topic><topic>Pulmonary Emphysema - surgery</topic><topic>Recurrence</topic><topic>Smoking - adverse effects</topic><topic>Thoracic Surgery, Video-Assisted</topic><topic>Tomography, X-Ray Computed</topic><topic>Treatment Outcome</topic><topic>Tuberous Sclerosis - complications</topic><topic>Tuberous Sclerosis - diagnosis</topic><topic>tuberous sclerosis complex</topic><topic>video-assisted thoracic surgery</topic><toplevel>online_resources</toplevel><creatorcontrib>Yamanaka, Sumitaka</creatorcontrib><creatorcontrib>Mizobuchi, Teruaki</creatorcontrib><creatorcontrib>Kurihara, Masatoshi</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>PubMed Central (Full Participant titles)</collection><jtitle>Annals of Thoracic and Cardiovascular Surgery</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Yamanaka, Sumitaka</au><au>Mizobuchi, Teruaki</au><au>Kurihara, Masatoshi</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Two Kinds of Cystic Lung Lesions with Pulmonary Lymphangioleiomyomatosis in a Male</atitle><jtitle>Annals of Thoracic and Cardiovascular Surgery</jtitle><addtitle>ATCS</addtitle><date>2017-01-01</date><risdate>2017</risdate><volume>23</volume><issue>1</issue><spage>36</spage><epage>39</epage><pages>36-39</pages><issn>1341-1098</issn><eissn>2186-1005</eissn><abstract>A 34-year-old male with frequent recurrence of right pneumothorax was admitted to our hospital. He was a current smoker and outwardly male without genital aplasia. He was diagnosed as tuberous sclerosis complex (TSC) at 2 year-old and underwent transcatheter arterial embolization for right renal hemorrhage due to renal tumor 2 years ago. Chest Computed Tomography showed that he had multiple tiny round cystic lesions with thin wall in both lungs. The recurrent pneumothorax was expected to be associated with TSC-Lymphangioleiomyomatosis (LAM). Video-assisted thoracic surgery was successfully performed. The operative and histological findings revealed that the bullae were classified into two groups; emphysematous bullae and bullae due to LAM. His postoperative course was uneventful. TSC-LAM is extremely rare, but in some cases the clinical recognition might be escaped due to subtle findings of bullae in early LAM, resulting in diagnosis as spontaneous pneumothorax.</abstract><cop>Japan</cop><pub>The Editorial Committee of Annals of Thoracic and Cardiovascular Surgery</pub><pmid>27349306</pmid><doi>10.5761/atcs.cr.16-00095</doi><tpages>4</tpages><oa>free_for_read</oa></addata></record> |
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| source | J-STAGE Free; MEDLINE; Elektronische Zeitschriftenbibliothek - Frei zugängliche E-Journals; PubMed Central |
| subjects | Adult Biopsy Blister - diagnostic imaging Blister - etiology Blister - surgery Case Report emphysematous bullae Humans Immunohistochemistry lymphangioleiomyomatosis Lymphangioleiomyomatosis - diagnostic imaging Lymphangioleiomyomatosis - etiology Lymphangioleiomyomatosis - surgery Male pneumothorax Pneumothorax - etiology Pulmonary Emphysema - diagnostic imaging Pulmonary Emphysema - etiology Pulmonary Emphysema - surgery Recurrence Smoking - adverse effects Thoracic Surgery, Video-Assisted Tomography, X-Ray Computed Treatment Outcome Tuberous Sclerosis - complications Tuberous Sclerosis - diagnosis tuberous sclerosis complex video-assisted thoracic surgery |
| title | Two Kinds of Cystic Lung Lesions with Pulmonary Lymphangioleiomyomatosis in a Male |
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