Idiopathic systemic capillary leak syndrome: Novel therapy for acute attacks
Patients typically present in the fifth and sixth decades of life with intermittent acute attacks of peripheral edema, hypotension, hypoalbuminemia, hemoconcentration, weight gain, and a monoclonal gammopathy.1,2 Rhabdomyolysis, compartment syndrome, and acute renal failure are common clinical findi...
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| Veröffentlicht in: | Journal of allergy and clinical immunology 2009-11, Vol.124 (5), p.1111-1113 |
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| container_title | Journal of allergy and clinical immunology |
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| creator | Dowden, Amy M., MD Rullo, Ornella J., MD Aziz, Najib, MD Fasano, Mary Beth, MD, MSPH Chatila, Talal, MD Ballas, Zuhair K., MD |
| description | Patients typically present in the fifth and sixth decades of life with intermittent acute attacks of peripheral edema, hypotension, hypoalbuminemia, hemoconcentration, weight gain, and a monoclonal gammopathy.1,2 Rhabdomyolysis, compartment syndrome, and acute renal failure are common clinical findings; however, pulmonary edema is rare.3 During an acute attack, there is an unexplained capillary hyperpermeability with a shift of plasma from the intravascular to the interstitial space.4 Acute attacks are followed by a resolution phase, with fluid shifts leading to intravascular volume overload and associated severe pulmonary edema. The usefulness of theophylline and terbutaline in this syndrome, both acutely and for prophylaxis, likely relates to their ability to increase cAMP and thus decrease endothelial cell permeability. [...]we propose a 2-step approach for treatment of acute attacks of ISCLS. |
| doi_str_mv | 10.1016/j.jaci.2009.06.043 |
| format | Article |
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The usefulness of theophylline and terbutaline in this syndrome, both acutely and for prophylaxis, likely relates to their ability to increase cAMP and thus decrease endothelial cell permeability. [...]we propose a 2-step approach for treatment of acute attacks of ISCLS.</description><identifier>ISSN: 0091-6749</identifier><identifier>EISSN: 1097-6825</identifier><identifier>DOI: 10.1016/j.jaci.2009.06.043</identifier><identifier>PMID: 19703708</identifier><language>eng</language><publisher>United States: Mosby, Inc</publisher><subject>Acute Disease ; Allergy and Immunology ; Aminophylline - therapeutic use ; Anti-Inflammatory Agents - therapeutic use ; Antibodies, Monoclonal - therapeutic use ; Capillary Leak Syndrome - drug therapy ; Capillary Leak Syndrome - immunology ; Capillary Permeability - drug effects ; Child ; Cytokines - blood ; Female ; Humans ; Immunologic Factors - therapeutic use ; Infliximab ; Male ; Middle Aged ; Mortality ; Pathogenesis ; Permeability ; Phosphodiesterase Inhibitors - therapeutic use ; Proteins ; Terbutaline - therapeutic use ; Theophylline - therapeutic use ; Tumor Necrosis Factor-alpha - antagonists & inhibitors ; Tumor Necrosis Factor-alpha - immunology</subject><ispartof>Journal of allergy and clinical immunology, 2009-11, Vol.124 (5), p.1111-1113</ispartof><rights>American Academy of Allergy, Asthma & Immunology</rights><rights>2009 American Academy of Allergy, Asthma & Immunology</rights><rights>Copyright Elsevier Limited Nov 2009</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c513t-9f4436b7311f9089f9603833c69a617a5083677cbe2c256ed2378b9011abe6413</citedby><cites>FETCH-LOGICAL-c513t-9f4436b7311f9089f9603833c69a617a5083677cbe2c256ed2378b9011abe6413</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktohtml>$$Uhttps://www.sciencedirect.com/science/article/pii/S009167490901001X$$EHTML$$P50$$Gelsevier$$H</linktohtml><link.rule.ids>314,776,780,3537,27901,27902,65534</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/19703708$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Dowden, Amy M., MD</creatorcontrib><creatorcontrib>Rullo, Ornella J., MD</creatorcontrib><creatorcontrib>Aziz, Najib, MD</creatorcontrib><creatorcontrib>Fasano, Mary Beth, MD, MSPH</creatorcontrib><creatorcontrib>Chatila, Talal, MD</creatorcontrib><creatorcontrib>Ballas, Zuhair K., MD</creatorcontrib><title>Idiopathic systemic capillary leak syndrome: Novel therapy for acute attacks</title><title>Journal of allergy and clinical immunology</title><addtitle>J Allergy Clin Immunol</addtitle><description>Patients typically present in the fifth and sixth decades of life with intermittent acute attacks of peripheral edema, hypotension, hypoalbuminemia, hemoconcentration, weight gain, and a monoclonal gammopathy.1,2 Rhabdomyolysis, compartment syndrome, and acute renal failure are common clinical findings; however, pulmonary edema is rare.3 During an acute attack, there is an unexplained capillary hyperpermeability with a shift of plasma from the intravascular to the interstitial space.4 Acute attacks are followed by a resolution phase, with fluid shifts leading to intravascular volume overload and associated severe pulmonary edema. The usefulness of theophylline and terbutaline in this syndrome, both acutely and for prophylaxis, likely relates to their ability to increase cAMP and thus decrease endothelial cell permeability. [...]we propose a 2-step approach for treatment of acute attacks of ISCLS.</description><subject>Acute Disease</subject><subject>Allergy and Immunology</subject><subject>Aminophylline - therapeutic use</subject><subject>Anti-Inflammatory Agents - therapeutic use</subject><subject>Antibodies, Monoclonal - therapeutic use</subject><subject>Capillary Leak Syndrome - drug therapy</subject><subject>Capillary Leak Syndrome - immunology</subject><subject>Capillary Permeability - drug effects</subject><subject>Child</subject><subject>Cytokines - blood</subject><subject>Female</subject><subject>Humans</subject><subject>Immunologic Factors - therapeutic use</subject><subject>Infliximab</subject><subject>Male</subject><subject>Middle Aged</subject><subject>Mortality</subject><subject>Pathogenesis</subject><subject>Permeability</subject><subject>Phosphodiesterase Inhibitors - therapeutic use</subject><subject>Proteins</subject><subject>Terbutaline - therapeutic use</subject><subject>Theophylline - therapeutic use</subject><subject>Tumor Necrosis Factor-alpha - antagonists & inhibitors</subject><subject>Tumor Necrosis Factor-alpha - immunology</subject><issn>0091-6749</issn><issn>1097-6825</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2009</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNp9kluL1TAUhYMoznH0D_ggBUGfWneSNhcRQQYvAwd9UMG3kKa7THp6m6QdOP_elHNgYB7mKbdvL7LX2oS8plBQoOJDV3TW-YIB6AJEASV_QnYUtMyFYtVTsksPNBey1BfkRYwdpDNX-jm5oFoCl6B2ZH_d-Gm2y413WTzGBYe0cXb2fW_DMevRHtL92IRpwI_Zz-kO-2y5wWDnY9ZOIbNuXTCzy2LdIb4kz1rbR3x1Xi_J329f_1z9yPe_vl9ffdnnrqJ8yXVbllzUklPaalC61QK44twJbQWVtgLFhZSuRuZYJbBhXKpaA6W2RlFSfknen3TnMN2uGBcz-OgwfXnEaY1GScEqYBVP5LtHSUapYiVAAt8-ALtpDWPqwtAKSkUFr1Si2IlyYYoxYGvm4IdklKFgtkxMZ7ZMzJaJAWFSJqnozVl6rQds7kvOISTg0wnA5Nmdx2Ci8zg6bHxAt5hm8o_rf35Q7no_emf7Ax4x3vdhIjNgfm9TsQ0FJEcB6D_-H3Qkr7s</recordid><startdate>20091101</startdate><enddate>20091101</enddate><creator>Dowden, Amy M., MD</creator><creator>Rullo, Ornella J., MD</creator><creator>Aziz, Najib, MD</creator><creator>Fasano, Mary Beth, MD, MSPH</creator><creator>Chatila, Talal, MD</creator><creator>Ballas, Zuhair K., MD</creator><general>Mosby, Inc</general><general>Elsevier Limited</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>7SS</scope><scope>7T5</scope><scope>H94</scope><scope>K9.</scope><scope>NAPCQ</scope></search><sort><creationdate>20091101</creationdate><title>Idiopathic systemic capillary leak syndrome: Novel therapy for acute attacks</title><author>Dowden, Amy M., MD ; 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however, pulmonary edema is rare.3 During an acute attack, there is an unexplained capillary hyperpermeability with a shift of plasma from the intravascular to the interstitial space.4 Acute attacks are followed by a resolution phase, with fluid shifts leading to intravascular volume overload and associated severe pulmonary edema. 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| subjects | Acute Disease Allergy and Immunology Aminophylline - therapeutic use Anti-Inflammatory Agents - therapeutic use Antibodies, Monoclonal - therapeutic use Capillary Leak Syndrome - drug therapy Capillary Leak Syndrome - immunology Capillary Permeability - drug effects Child Cytokines - blood Female Humans Immunologic Factors - therapeutic use Infliximab Male Middle Aged Mortality Pathogenesis Permeability Phosphodiesterase Inhibitors - therapeutic use Proteins Terbutaline - therapeutic use Theophylline - therapeutic use Tumor Necrosis Factor-alpha - antagonists & inhibitors Tumor Necrosis Factor-alpha - immunology |
| title | Idiopathic systemic capillary leak syndrome: Novel therapy for acute attacks |
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