Shrinking Lung Syndrome Successfully Treated with Rituximab and Cyclophosphamide
Shrinking lung syndrome (SLS) is an uncommon feature of systemic lupus erythematosus (SLE) characterized by dyspnea, pleuritic chest pain, diaphragmatic elevation, restrictive ventilatory defect and reduced respiratory muscle strength as measured by volitional tests. We report the case of a 28-year-...
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| Veröffentlicht in: | Respiration 2012-01, Vol.84 (2), p.144-149 |
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| creator | Langenskiöld, Elisabeth Bonetti, Alessandro Fitting, Jean William Heinzer, Raphaël Dudler, Jean Spertini, François Lazor, Romain |
| description | Shrinking lung syndrome (SLS) is an uncommon feature of systemic lupus erythematosus (SLE) characterized by dyspnea, pleuritic chest pain, diaphragmatic elevation, restrictive ventilatory defect and reduced respiratory muscle strength as measured by volitional tests. We report the case of a 28-year-old woman with overlapping features of SLE and Sjögren syndrome who developed severe SLS while receiving corticosteroids and azathioprine for severe polyarthritis. She was treated with a combination of rituximab and cyclophosphamide, which led to a dramatic improvement in her clinical condition and respiratory function tests. The increase in vital capacity was one of the highest among 35 published cases of SLS. Thus, restoring a near-normal lung function is an achievable goal in SLS, and the use of rituximab, with or without concomitant cyclophosphamide, certainly deserves further study in this setting. |
| doi_str_mv | 10.1159/000334947 |
| format | Article |
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We report the case of a 28-year-old woman with overlapping features of SLE and Sjögren syndrome who developed severe SLS while receiving corticosteroids and azathioprine for severe polyarthritis. She was treated with a combination of rituximab and cyclophosphamide, which led to a dramatic improvement in her clinical condition and respiratory function tests. The increase in vital capacity was one of the highest among 35 published cases of SLS. Thus, restoring a near-normal lung function is an achievable goal in SLS, and the use of rituximab, with or without concomitant cyclophosphamide, certainly deserves further study in this setting.</description><identifier>ISSN: 0025-7931</identifier><identifier>EISSN: 1423-0356</identifier><identifier>DOI: 10.1159/000334947</identifier><identifier>PMID: 22301521</identifier><language>eng</language><publisher>Basel, Switzerland: S. 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Thus, restoring a near-normal lung function is an achievable goal in SLS, and the use of rituximab, with or without concomitant cyclophosphamide, certainly deserves further study in this setting.</description><subject>Adult</subject><subject>Antibodies, Monoclonal, Murine-Derived - administration & dosage</subject><subject>Antirheumatic Agents - administration & dosage</subject><subject>Autoimmune diseases</subject><subject>Chest Pain - etiology</subject><subject>Chest Pain - physiopathology</subject><subject>Cyclophosphamide - administration & dosage</subject><subject>Drug Monitoring - methods</subject><subject>Drug therapy</subject><subject>Drug Therapy, Combination</subject><subject>Dyspnea - etiology</subject><subject>Dyspnea - physiopathology</subject><subject>Female</subject><subject>Humans</subject><subject>Lung diseases</subject><subject>Lung Diseases - drug therapy</subject><subject>Lung Diseases - etiology</subject><subject>Lung Diseases - physiopathology</subject><subject>Lupus Erythematosus, Systemic - complications</subject><subject>Lupus Erythematosus, Systemic - drug therapy</subject><subject>Lupus Erythematosus, Systemic - physiopathology</subject><subject>Novel Insights from Clinical Practice</subject><subject>Pleura - physiopathology</subject><subject>Pleurisy - drug therapy</subject><subject>Pleurisy - etiology</subject><subject>Pleurisy - physiopathology</subject><subject>Respiratory Function Tests - methods</subject><subject>Respiratory Muscles - physiopathology</subject><subject>Rituximab</subject><subject>Severity of Illness Index</subject><subject>Sjogren's Syndrome - complications</subject><subject>Sjogren's Syndrome - drug therapy</subject><subject>Sjogren's Syndrome - physiopathology</subject><subject>Treatment Outcome</subject><issn>0025-7931</issn><issn>1423-0356</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2012</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><sourceid>8G5</sourceid><sourceid>ABUWG</sourceid><sourceid>AFKRA</sourceid><sourceid>AZQEC</sourceid><sourceid>BENPR</sourceid><sourceid>CCPQU</sourceid><sourceid>DWQXO</sourceid><sourceid>GNUQQ</sourceid><sourceid>GUQSH</sourceid><sourceid>M2O</sourceid><recordid>eNpd0M9LwzAUB_AgipvTg3eRghc9VJO8pmmOUvwFA8XNc0nbdO3WXyYN2v_eyOYOXpJAPu_x3hehc4JvCWHiDmMMEIiAH6ApCSj4GFh4iKYYU-ZzAWSCToxZY0xYFNFjNKEU3JuSKXpblLpqN1W78ubWHYuxzXXXKG9hs0wZU9i6Hr2lVnJQufdVDaX3Xg32u2pk6sk29-Ixq7u-7ExfyqbK1Sk6KmRt1NnunqGPx4dl_OzPX59e4vu5n0EoBl9gTkjOIIBQRlkhAhqkAREKQFARSsW4YLhIJacQKSIksJxwgILwkEXuH2boetu3192nVWZImspkqq5lqzprEoKBcgh5BI5e_aPrzurWTeeUYEBoBNipm63KdGeMVkXSa7elHh1KfmNO9jE7e7nraNNG5Xv5l6sDF1uwkXql9B7s6n8APLl-VQ</recordid><startdate>20120101</startdate><enddate>20120101</enddate><creator>Langenskiöld, Elisabeth</creator><creator>Bonetti, Alessandro</creator><creator>Fitting, Jean William</creator><creator>Heinzer, Raphaël</creator><creator>Dudler, Jean</creator><creator>Spertini, François</creator><creator>Lazor, Romain</creator><general>S. 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administration & dosage</topic><topic>Antirheumatic Agents - administration & dosage</topic><topic>Autoimmune diseases</topic><topic>Chest Pain - etiology</topic><topic>Chest Pain - physiopathology</topic><topic>Cyclophosphamide - administration & dosage</topic><topic>Drug Monitoring - methods</topic><topic>Drug therapy</topic><topic>Drug Therapy, Combination</topic><topic>Dyspnea - etiology</topic><topic>Dyspnea - physiopathology</topic><topic>Female</topic><topic>Humans</topic><topic>Lung diseases</topic><topic>Lung Diseases - drug therapy</topic><topic>Lung Diseases - etiology</topic><topic>Lung Diseases - physiopathology</topic><topic>Lupus Erythematosus, Systemic - complications</topic><topic>Lupus Erythematosus, Systemic - drug therapy</topic><topic>Lupus Erythematosus, Systemic - physiopathology</topic><topic>Novel Insights from Clinical Practice</topic><topic>Pleura - physiopathology</topic><topic>Pleurisy - drug therapy</topic><topic>Pleurisy - etiology</topic><topic>Pleurisy - physiopathology</topic><topic>Respiratory Function Tests - methods</topic><topic>Respiratory Muscles - physiopathology</topic><topic>Rituximab</topic><topic>Severity of Illness Index</topic><topic>Sjogren's Syndrome - complications</topic><topic>Sjogren's Syndrome - drug therapy</topic><topic>Sjogren's Syndrome - physiopathology</topic><topic>Treatment Outcome</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Langenskiöld, Elisabeth</creatorcontrib><creatorcontrib>Bonetti, Alessandro</creatorcontrib><creatorcontrib>Fitting, Jean William</creatorcontrib><creatorcontrib>Heinzer, Raphaël</creatorcontrib><creatorcontrib>Dudler, Jean</creatorcontrib><creatorcontrib>Spertini, François</creatorcontrib><creatorcontrib>Lazor, Romain</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>ProQuest Central (Corporate)</collection><collection>Career & Technical Education Database</collection><collection>Nursing & Allied Health Database</collection><collection>Health & Medical Collection</collection><collection>ProQuest Central (purchase pre-March 2016)</collection><collection>Medical Database (Alumni Edition)</collection><collection>ProQuest Pharma Collection</collection><collection>Hospital Premium Collection</collection><collection>Hospital Premium Collection (Alumni Edition)</collection><collection>ProQuest Central (Alumni) (purchase pre-March 2016)</collection><collection>Research Library (Alumni Edition)</collection><collection>ProQuest Central (Alumni Edition)</collection><collection>ProQuest Central UK/Ireland</collection><collection>ProQuest Central Essentials</collection><collection>ProQuest Central</collection><collection>ProQuest One Community College</collection><collection>ProQuest Central Korea</collection><collection>Health Research Premium Collection</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>ProQuest Central Student</collection><collection>Research Library Prep</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Nursing & Allied Health Database (Alumni Edition)</collection><collection>Health & Medical Collection (Alumni Edition)</collection><collection>Medical Database</collection><collection>Research Library</collection><collection>Research Library (Corporate)</collection><collection>Nursing & Allied Health Premium</collection><collection>ProQuest One Academic Eastern Edition (DO NOT USE)</collection><collection>ProQuest One Academic</collection><collection>ProQuest One Academic UKI Edition</collection><collection>ProQuest Central China</collection><collection>ProQuest Central Basic</collection><collection>MEDLINE - Academic</collection><jtitle>Respiration</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Langenskiöld, Elisabeth</au><au>Bonetti, Alessandro</au><au>Fitting, Jean William</au><au>Heinzer, Raphaël</au><au>Dudler, Jean</au><au>Spertini, François</au><au>Lazor, Romain</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Shrinking Lung Syndrome Successfully Treated with Rituximab and Cyclophosphamide</atitle><jtitle>Respiration</jtitle><addtitle>Respiration</addtitle><date>2012-01-01</date><risdate>2012</risdate><volume>84</volume><issue>2</issue><spage>144</spage><epage>149</epage><pages>144-149</pages><issn>0025-7931</issn><eissn>1423-0356</eissn><abstract>Shrinking lung syndrome (SLS) is an uncommon feature of systemic lupus erythematosus (SLE) characterized by dyspnea, pleuritic chest pain, diaphragmatic elevation, restrictive ventilatory defect and reduced respiratory muscle strength as measured by volitional tests. We report the case of a 28-year-old woman with overlapping features of SLE and Sjögren syndrome who developed severe SLS while receiving corticosteroids and azathioprine for severe polyarthritis. She was treated with a combination of rituximab and cyclophosphamide, which led to a dramatic improvement in her clinical condition and respiratory function tests. The increase in vital capacity was one of the highest among 35 published cases of SLS. Thus, restoring a near-normal lung function is an achievable goal in SLS, and the use of rituximab, with or without concomitant cyclophosphamide, certainly deserves further study in this setting.</abstract><cop>Basel, Switzerland</cop><pub>S. Karger AG</pub><pmid>22301521</pmid><doi>10.1159/000334947</doi><tpages>6</tpages><oa>free_for_read</oa></addata></record> |
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| ispartof | Respiration, 2012-01, Vol.84 (2), p.144-149 |
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| subjects | Adult Antibodies, Monoclonal, Murine-Derived - administration & dosage Antirheumatic Agents - administration & dosage Autoimmune diseases Chest Pain - etiology Chest Pain - physiopathology Cyclophosphamide - administration & dosage Drug Monitoring - methods Drug therapy Drug Therapy, Combination Dyspnea - etiology Dyspnea - physiopathology Female Humans Lung diseases Lung Diseases - drug therapy Lung Diseases - etiology Lung Diseases - physiopathology Lupus Erythematosus, Systemic - complications Lupus Erythematosus, Systemic - drug therapy Lupus Erythematosus, Systemic - physiopathology Novel Insights from Clinical Practice Pleura - physiopathology Pleurisy - drug therapy Pleurisy - etiology Pleurisy - physiopathology Respiratory Function Tests - methods Respiratory Muscles - physiopathology Rituximab Severity of Illness Index Sjogren's Syndrome - complications Sjogren's Syndrome - drug therapy Sjogren's Syndrome - physiopathology Treatment Outcome |
| title | Shrinking Lung Syndrome Successfully Treated with Rituximab and Cyclophosphamide |
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