Propranolol as a treatment option in Gorham-Stout syndrome: A case report
Summary Gorham–Stout syndrome is a very rare syndrome characterized by progressive angiomatosis and lymphangiomas involving multiple organs. We describe herein a girl with progressive Gorham–Stout syndrome since the age of 13 years. Her disease involved the mediastinum, pericardium, vertebrae, ribs,...
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| Veröffentlicht in: | Pediatric pulmonology 2014-04, Vol.49 (4), p.417-419 |
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| container_title | Pediatric pulmonology |
| container_volume | 49 |
| creator | Nir, Vered Guralnik, Ludmila Livnat, Galit Bar-Yoseph, Ronen Hakim, Fahed Ilivitzki, Anat Bentur, Lea |
| description | Summary
Gorham–Stout syndrome is a very rare syndrome characterized by progressive angiomatosis and lymphangiomas involving multiple organs. We describe herein a girl with progressive Gorham–Stout syndrome since the age of 13 years. Her disease involved the mediastinum, pericardium, vertebrae, ribs, and skull and did not respond to interferon and bisphosphonates. Propranolol administration was initiated at the age of 18 years and was associated with improvement in pulmonary function tests and involution of the mediastinal and hilar hemangiomatous lesions. The possible beneficial effect of propranolol in Gorham–Stout syndrome should be further investigated. Pediatr Pulmonol. 2014; 49:417–419. © 2013 Wiley Periodicals, Inc. |
| doi_str_mv | 10.1002/ppul.22869 |
| format | Article |
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Gorham–Stout syndrome is a very rare syndrome characterized by progressive angiomatosis and lymphangiomas involving multiple organs. We describe herein a girl with progressive Gorham–Stout syndrome since the age of 13 years. Her disease involved the mediastinum, pericardium, vertebrae, ribs, and skull and did not respond to interferon and bisphosphonates. Propranolol administration was initiated at the age of 18 years and was associated with improvement in pulmonary function tests and involution of the mediastinal and hilar hemangiomatous lesions. The possible beneficial effect of propranolol in Gorham–Stout syndrome should be further investigated. Pediatr Pulmonol. 2014; 49:417–419. © 2013 Wiley Periodicals, Inc.</description><identifier>ISSN: 8755-6863</identifier><identifier>EISSN: 1099-0496</identifier><identifier>DOI: 10.1002/ppul.22869</identifier><identifier>PMID: 24000203</identifier><language>eng</language><publisher>United States: Blackwell Publishing Ltd</publisher><subject>Adolescent ; Female ; Gorham-Stout syndrome ; Humans ; Osteolysis, Essential - drug therapy ; propranolol ; Propranolol - therapeutic use</subject><ispartof>Pediatric pulmonology, 2014-04, Vol.49 (4), p.417-419</ispartof><rights>2013 Wiley Periodicals, Inc.</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c4269-3a518229504a9147473088a20e50aff4f62250a5e0924fb4f55e8d6d1beea05f3</citedby></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://onlinelibrary.wiley.com/doi/pdf/10.1002%2Fppul.22869$$EPDF$$P50$$Gwiley$$H</linktopdf><linktohtml>$$Uhttps://onlinelibrary.wiley.com/doi/full/10.1002%2Fppul.22869$$EHTML$$P50$$Gwiley$$H</linktohtml><link.rule.ids>314,776,780,1411,27903,27904,45553,45554</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/24000203$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Nir, Vered</creatorcontrib><creatorcontrib>Guralnik, Ludmila</creatorcontrib><creatorcontrib>Livnat, Galit</creatorcontrib><creatorcontrib>Bar-Yoseph, Ronen</creatorcontrib><creatorcontrib>Hakim, Fahed</creatorcontrib><creatorcontrib>Ilivitzki, Anat</creatorcontrib><creatorcontrib>Bentur, Lea</creatorcontrib><title>Propranolol as a treatment option in Gorham-Stout syndrome: A case report</title><title>Pediatric pulmonology</title><addtitle>Pediatr Pulmonol</addtitle><description>Summary
Gorham–Stout syndrome is a very rare syndrome characterized by progressive angiomatosis and lymphangiomas involving multiple organs. We describe herein a girl with progressive Gorham–Stout syndrome since the age of 13 years. Her disease involved the mediastinum, pericardium, vertebrae, ribs, and skull and did not respond to interferon and bisphosphonates. Propranolol administration was initiated at the age of 18 years and was associated with improvement in pulmonary function tests and involution of the mediastinal and hilar hemangiomatous lesions. The possible beneficial effect of propranolol in Gorham–Stout syndrome should be further investigated. Pediatr Pulmonol. 2014; 49:417–419. © 2013 Wiley Periodicals, Inc.</description><subject>Adolescent</subject><subject>Female</subject><subject>Gorham-Stout syndrome</subject><subject>Humans</subject><subject>Osteolysis, Essential - drug therapy</subject><subject>propranolol</subject><subject>Propranolol - therapeutic use</subject><issn>8755-6863</issn><issn>1099-0496</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2014</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNpdkMFKAzEQhoMoWqsXH0ACXrysJtkkm3grxVahaEGLx5C2s7h1d7MmWbRv77bVHjzND_P9w_AhdEHJDSWE3TZNW94wpqQ-QD1KtE4I1_IQ9VQmRCKVTE_QaQgrQrqdpsfohPEuM5L20OPUu8bb2pWuxDZgi6MHGyuoI3ZNLFyNixqPnX-3VfISXRtxWNdL7yq4wwO8sAGwh8b5eIaOclsGOP-dfTQb3b8OH5LJ8_hxOJgkC86kTlIrqGJMC8KtpjzjWUqUsoyAIDbPeS4Z65IAohnP5zwXAtRSLukcwBKRp310vbvbePfZQoimKsICytLW4NpgqCCKM5FK1aFX_9CVa33dfbehMqo5Fxvq8pdq5xUsTeOLyvq1-ZPUAXQHfBUlrPd7SsxGv9noN1v9ZjqdTbap6yS7ThEifO871n8YmaWZMG9PYzOSk-xNDIcmS38AGTKEyQ</recordid><startdate>201404</startdate><enddate>201404</enddate><creator>Nir, Vered</creator><creator>Guralnik, Ludmila</creator><creator>Livnat, Galit</creator><creator>Bar-Yoseph, Ronen</creator><creator>Hakim, Fahed</creator><creator>Ilivitzki, Anat</creator><creator>Bentur, Lea</creator><general>Blackwell Publishing Ltd</general><general>Wiley Subscription Services, Inc</general><scope>BSCLL</scope><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>K9.</scope><scope>7X8</scope></search><sort><creationdate>201404</creationdate><title>Propranolol as a treatment option in Gorham-Stout syndrome: A case report</title><author>Nir, Vered ; Guralnik, Ludmila ; Livnat, Galit ; Bar-Yoseph, Ronen ; Hakim, Fahed ; Ilivitzki, Anat ; Bentur, Lea</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c4269-3a518229504a9147473088a20e50aff4f62250a5e0924fb4f55e8d6d1beea05f3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2014</creationdate><topic>Adolescent</topic><topic>Female</topic><topic>Gorham-Stout syndrome</topic><topic>Humans</topic><topic>Osteolysis, Essential - drug therapy</topic><topic>propranolol</topic><topic>Propranolol - therapeutic use</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Nir, Vered</creatorcontrib><creatorcontrib>Guralnik, Ludmila</creatorcontrib><creatorcontrib>Livnat, Galit</creatorcontrib><creatorcontrib>Bar-Yoseph, Ronen</creatorcontrib><creatorcontrib>Hakim, Fahed</creatorcontrib><creatorcontrib>Ilivitzki, Anat</creatorcontrib><creatorcontrib>Bentur, Lea</creatorcontrib><collection>Istex</collection><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>MEDLINE - Academic</collection><jtitle>Pediatric pulmonology</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Nir, Vered</au><au>Guralnik, Ludmila</au><au>Livnat, Galit</au><au>Bar-Yoseph, Ronen</au><au>Hakim, Fahed</au><au>Ilivitzki, Anat</au><au>Bentur, Lea</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Propranolol as a treatment option in Gorham-Stout syndrome: A case report</atitle><jtitle>Pediatric pulmonology</jtitle><addtitle>Pediatr Pulmonol</addtitle><date>2014-04</date><risdate>2014</risdate><volume>49</volume><issue>4</issue><spage>417</spage><epage>419</epage><pages>417-419</pages><issn>8755-6863</issn><eissn>1099-0496</eissn><abstract>Summary
Gorham–Stout syndrome is a very rare syndrome characterized by progressive angiomatosis and lymphangiomas involving multiple organs. We describe herein a girl with progressive Gorham–Stout syndrome since the age of 13 years. Her disease involved the mediastinum, pericardium, vertebrae, ribs, and skull and did not respond to interferon and bisphosphonates. Propranolol administration was initiated at the age of 18 years and was associated with improvement in pulmonary function tests and involution of the mediastinal and hilar hemangiomatous lesions. The possible beneficial effect of propranolol in Gorham–Stout syndrome should be further investigated. Pediatr Pulmonol. 2014; 49:417–419. © 2013 Wiley Periodicals, Inc.</abstract><cop>United States</cop><pub>Blackwell Publishing Ltd</pub><pmid>24000203</pmid><doi>10.1002/ppul.22869</doi><tpages>3</tpages></addata></record> |
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| ispartof | Pediatric pulmonology, 2014-04, Vol.49 (4), p.417-419 |
| issn | 8755-6863 1099-0496 |
| language | eng |
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| source | MEDLINE; Wiley Online Library Journals Frontfile Complete |
| subjects | Adolescent Female Gorham-Stout syndrome Humans Osteolysis, Essential - drug therapy propranolol Propranolol - therapeutic use |
| title | Propranolol as a treatment option in Gorham-Stout syndrome: A case report |
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