Thrombotic microangiopathy in adult Still's disease
Adult Still's disease (ASD) is a rare systemic disorder characterized by fever, arthralgia, cutaneous rash, and lymphadenopathy, with high polymorphonuclear leucocytosis and low glycosylated ferritinaemia. Kidney involvement has been reported rarely. We present a patient with ASD who developed...
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Veröffentlicht in: | Scandinavian journal of rheumatology 2005-10, Vol.34 (5), p.399-403 |
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container_title | Scandinavian journal of rheumatology |
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creator | Quéméneur, T. Noel, L-H. Kyndt, X. Droz, D. Fleury, D. Binaut, R. Lemaitre, V. Gobert, P. Vanhille, P. |
description | Adult Still's disease (ASD) is a rare systemic disorder characterized by fever, arthralgia, cutaneous rash, and lymphadenopathy, with high polymorphonuclear leucocytosis and low glycosylated ferritinaemia. Kidney involvement has been reported rarely. We present a patient with ASD who developed haemolytic uraemic syndrome (HUS). The 42-year-old patient was admitted for unexplained fever related to ASD according to Yamaguchi's classification criteria. As Still's disease was resistant to prednisone, high-dose intravenous immunoglobulins (IV Ig) were administered. During the follow-up the patient developed acute renal failure and non-immune haemolytic anaemia with high levels of antiphospholipid antibodies (IgG anticardiolipin antibodies and anti- 2 glycoprotein 1 antibodies). Renal biopsy disclosed thrombotic microangiopathy (TMA) with arteriolar and glomerular involvement. Treatment with steroids and intravenous IV Ig was reinitiated but renal function worsened towards end-stage renal failure. In this case, we suggest that antiphospholipid antibodies could have promoted arteriolar and glomerular TMA. HUS may be the cause of acute renal failure in Still's disease. |
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Kidney involvement has been reported rarely. We present a patient with ASD who developed haemolytic uraemic syndrome (HUS). The 42-year-old patient was admitted for unexplained fever related to ASD according to Yamaguchi's classification criteria. As Still's disease was resistant to prednisone, high-dose intravenous immunoglobulins (IV Ig) were administered. During the follow-up the patient developed acute renal failure and non-immune haemolytic anaemia with high levels of antiphospholipid antibodies (IgG anticardiolipin antibodies and anti- 2 glycoprotein 1 antibodies). Renal biopsy disclosed thrombotic microangiopathy (TMA) with arteriolar and glomerular involvement. Treatment with steroids and intravenous IV Ig was reinitiated but renal function worsened towards end-stage renal failure. In this case, we suggest that antiphospholipid antibodies could have promoted arteriolar and glomerular TMA. HUS may be the cause of acute renal failure in Still's disease.</description><identifier>ISSN: 0300-9742</identifier><identifier>EISSN: 1502-7732</identifier><identifier>DOI: 10.1080/03009740510026689</identifier><identifier>PMID: 16234190</identifier><identifier>CODEN: SJRHAT</identifier><language>eng</language><publisher>Colchester: Informa UK Ltd</publisher><subject>Acute Kidney Injury - etiology ; Acute Kidney Injury - immunology ; Adult ; Antibodies, Antiphospholipid - immunology ; Biological and medical sciences ; Diseases of the osteoarticular system ; Hemolytic-Uremic Syndrome - etiology ; Hemolytic-Uremic Syndrome - immunology ; Humans ; Immunoglobulins, Intravenous ; Inflammatory joint diseases ; Male ; Medical sciences ; Microcirculation ; Renal Circulation ; Steroids - therapeutic use ; Still's Disease, Adult-Onset - complications ; Still's Disease, Adult-Onset - drug therapy ; Still's Disease, Adult-Onset - immunology ; Thrombosis - etiology ; Thrombosis - immunology</subject><ispartof>Scandinavian journal of rheumatology, 2005-10, Vol.34 (5), p.399-403</ispartof><rights>Informa UK Ltd All rights reserved: reproduction in whole or part not permitted</rights><rights>2005 INIST-CNRS</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c412t-e44d21ea59e62d895b1f663d020780f191301816c41de187f9d5c5a457f65c3a3</citedby><cites>FETCH-LOGICAL-c412t-e44d21ea59e62d895b1f663d020780f191301816c41de187f9d5c5a457f65c3a3</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://www.tandfonline.com/doi/pdf/10.1080/03009740510026689$$EPDF$$P50$$Ginformahealthcare$$H</linktopdf><linktohtml>$$Uhttps://www.tandfonline.com/doi/full/10.1080/03009740510026689$$EHTML$$P50$$Ginformahealthcare$$H</linktohtml><link.rule.ids>314,777,781,27905,27906,61200,61235,61381,61416</link.rule.ids><backlink>$$Uhttp://pascal-francis.inist.fr/vibad/index.php?action=getRecordDetail&idt=17214017$$DView record in Pascal Francis$$Hfree_for_read</backlink><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/16234190$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Quéméneur, T.</creatorcontrib><creatorcontrib>Noel, L-H.</creatorcontrib><creatorcontrib>Kyndt, X.</creatorcontrib><creatorcontrib>Droz, D.</creatorcontrib><creatorcontrib>Fleury, D.</creatorcontrib><creatorcontrib>Binaut, R.</creatorcontrib><creatorcontrib>Lemaitre, V.</creatorcontrib><creatorcontrib>Gobert, P.</creatorcontrib><creatorcontrib>Vanhille, P.</creatorcontrib><title>Thrombotic microangiopathy in adult Still's disease</title><title>Scandinavian journal of rheumatology</title><addtitle>Scand J Rheumatol</addtitle><description>Adult Still's disease (ASD) is a rare systemic disorder characterized by fever, arthralgia, cutaneous rash, and lymphadenopathy, with high polymorphonuclear leucocytosis and low glycosylated ferritinaemia. Kidney involvement has been reported rarely. We present a patient with ASD who developed haemolytic uraemic syndrome (HUS). The 42-year-old patient was admitted for unexplained fever related to ASD according to Yamaguchi's classification criteria. As Still's disease was resistant to prednisone, high-dose intravenous immunoglobulins (IV Ig) were administered. During the follow-up the patient developed acute renal failure and non-immune haemolytic anaemia with high levels of antiphospholipid antibodies (IgG anticardiolipin antibodies and anti- 2 glycoprotein 1 antibodies). Renal biopsy disclosed thrombotic microangiopathy (TMA) with arteriolar and glomerular involvement. Treatment with steroids and intravenous IV Ig was reinitiated but renal function worsened towards end-stage renal failure. In this case, we suggest that antiphospholipid antibodies could have promoted arteriolar and glomerular TMA. HUS may be the cause of acute renal failure in Still's disease.</description><subject>Acute Kidney Injury - etiology</subject><subject>Acute Kidney Injury - immunology</subject><subject>Adult</subject><subject>Antibodies, Antiphospholipid - immunology</subject><subject>Biological and medical sciences</subject><subject>Diseases of the osteoarticular system</subject><subject>Hemolytic-Uremic Syndrome - etiology</subject><subject>Hemolytic-Uremic Syndrome - immunology</subject><subject>Humans</subject><subject>Immunoglobulins, Intravenous</subject><subject>Inflammatory joint diseases</subject><subject>Male</subject><subject>Medical sciences</subject><subject>Microcirculation</subject><subject>Renal Circulation</subject><subject>Steroids - therapeutic use</subject><subject>Still's Disease, Adult-Onset - complications</subject><subject>Still's Disease, Adult-Onset - drug therapy</subject><subject>Still's Disease, Adult-Onset - immunology</subject><subject>Thrombosis - etiology</subject><subject>Thrombosis - immunology</subject><issn>0300-9742</issn><issn>1502-7732</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2005</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNqF0DtPwzAQB3ALgWgpfAAWlAWYAneOH7GYUMVLqsRAmSPXcYirPIqdDP32pGqkDkiwnIf73en8J-QS4Q4hhXtIAJRkwBGACpGqIzJFDjSWMqHHZLrrxwOgE3IWwhoAmJLqlExQ0IShgilJlqVv61XbORPVzvhWN1-u3eiu3EauiXTeV1300bmqug1R7oLVwZ6Tk0JXwV6M74x8Pj8t56_x4v3lbf64iA1D2sWWsZyi1VxZQfNU8RUWQiQ5UJApFKgwAUxRDDq3mMpC5dxwzbgsBDeJTmbkZr9349vv3oYuq10wtqp0Y9s-ZCKVAEP5F1JgIBhXA8Q9HD4agrdFtvGu1n6bIWS7SLNfkQ4zV-PyflXb_DAxZjiA6xHoYHRVeN0YFw5OUmSAuysf9s41RetrXVpddaXR3mbrtvfNEOUfZ_wAmduOAg</recordid><startdate>20051001</startdate><enddate>20051001</enddate><creator>Quéméneur, T.</creator><creator>Noel, L-H.</creator><creator>Kyndt, X.</creator><creator>Droz, D.</creator><creator>Fleury, D.</creator><creator>Binaut, R.</creator><creator>Lemaitre, V.</creator><creator>Gobert, P.</creator><creator>Vanhille, P.</creator><general>Informa UK Ltd</general><general>Taylor & Francis</general><scope>IQODW</scope><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>7T5</scope><scope>H94</scope><scope>7X8</scope></search><sort><creationdate>20051001</creationdate><title>Thrombotic microangiopathy in adult Still's disease</title><author>Quéméneur, T. ; Noel, L-H. ; Kyndt, X. ; Droz, D. ; Fleury, D. ; Binaut, R. ; Lemaitre, V. ; Gobert, P. ; Vanhille, P.</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c412t-e44d21ea59e62d895b1f663d020780f191301816c41de187f9d5c5a457f65c3a3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2005</creationdate><topic>Acute Kidney Injury - etiology</topic><topic>Acute Kidney Injury - immunology</topic><topic>Adult</topic><topic>Antibodies, Antiphospholipid - immunology</topic><topic>Biological and medical sciences</topic><topic>Diseases of the osteoarticular system</topic><topic>Hemolytic-Uremic Syndrome - etiology</topic><topic>Hemolytic-Uremic Syndrome - immunology</topic><topic>Humans</topic><topic>Immunoglobulins, Intravenous</topic><topic>Inflammatory joint diseases</topic><topic>Male</topic><topic>Medical sciences</topic><topic>Microcirculation</topic><topic>Renal Circulation</topic><topic>Steroids - therapeutic use</topic><topic>Still's Disease, Adult-Onset - complications</topic><topic>Still's Disease, Adult-Onset - drug therapy</topic><topic>Still's Disease, Adult-Onset - immunology</topic><topic>Thrombosis - etiology</topic><topic>Thrombosis - immunology</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Quéméneur, T.</creatorcontrib><creatorcontrib>Noel, L-H.</creatorcontrib><creatorcontrib>Kyndt, X.</creatorcontrib><creatorcontrib>Droz, D.</creatorcontrib><creatorcontrib>Fleury, D.</creatorcontrib><creatorcontrib>Binaut, R.</creatorcontrib><creatorcontrib>Lemaitre, V.</creatorcontrib><creatorcontrib>Gobert, P.</creatorcontrib><creatorcontrib>Vanhille, P.</creatorcontrib><collection>Pascal-Francis</collection><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>Immunology Abstracts</collection><collection>AIDS and Cancer Research Abstracts</collection><collection>MEDLINE - Academic</collection><jtitle>Scandinavian journal of rheumatology</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Quéméneur, T.</au><au>Noel, L-H.</au><au>Kyndt, X.</au><au>Droz, D.</au><au>Fleury, D.</au><au>Binaut, R.</au><au>Lemaitre, V.</au><au>Gobert, P.</au><au>Vanhille, P.</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Thrombotic microangiopathy in adult Still's disease</atitle><jtitle>Scandinavian journal of rheumatology</jtitle><addtitle>Scand J Rheumatol</addtitle><date>2005-10-01</date><risdate>2005</risdate><volume>34</volume><issue>5</issue><spage>399</spage><epage>403</epage><pages>399-403</pages><issn>0300-9742</issn><eissn>1502-7732</eissn><coden>SJRHAT</coden><abstract>Adult Still's disease (ASD) is a rare systemic disorder characterized by fever, arthralgia, cutaneous rash, and lymphadenopathy, with high polymorphonuclear leucocytosis and low glycosylated ferritinaemia. Kidney involvement has been reported rarely. We present a patient with ASD who developed haemolytic uraemic syndrome (HUS). The 42-year-old patient was admitted for unexplained fever related to ASD according to Yamaguchi's classification criteria. As Still's disease was resistant to prednisone, high-dose intravenous immunoglobulins (IV Ig) were administered. During the follow-up the patient developed acute renal failure and non-immune haemolytic anaemia with high levels of antiphospholipid antibodies (IgG anticardiolipin antibodies and anti- 2 glycoprotein 1 antibodies). Renal biopsy disclosed thrombotic microangiopathy (TMA) with arteriolar and glomerular involvement. Treatment with steroids and intravenous IV Ig was reinitiated but renal function worsened towards end-stage renal failure. In this case, we suggest that antiphospholipid antibodies could have promoted arteriolar and glomerular TMA. HUS may be the cause of acute renal failure in Still's disease.</abstract><cop>Colchester</cop><pub>Informa UK Ltd</pub><pmid>16234190</pmid><doi>10.1080/03009740510026689</doi><tpages>5</tpages></addata></record> |
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subjects | Acute Kidney Injury - etiology Acute Kidney Injury - immunology Adult Antibodies, Antiphospholipid - immunology Biological and medical sciences Diseases of the osteoarticular system Hemolytic-Uremic Syndrome - etiology Hemolytic-Uremic Syndrome - immunology Humans Immunoglobulins, Intravenous Inflammatory joint diseases Male Medical sciences Microcirculation Renal Circulation Steroids - therapeutic use Still's Disease, Adult-Onset - complications Still's Disease, Adult-Onset - drug therapy Still's Disease, Adult-Onset - immunology Thrombosis - etiology Thrombosis - immunology |
title | Thrombotic microangiopathy in adult Still's disease |
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