Vasculitis associated with large granular lymphocyte (LGL) leukemia: Presentation and treatment outcomes of 11 cases
Abstract Objective The association between vasculitis and large granular lymphocyte (LGL) leukemia has rarely been reported or investigated. Thus, we assessed the clinical and biological phenotypes of LGL leukemia associated with vasculitis. Results We studied a series of 11 patients displaying LGL...
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| creator | Audemard, Alexandra, MD Lamy, Thierry, MD, PhD Bareau, Benoît, MD Sicre, Flore, MD Suarez, Felipe, MD Truquet, Florence, PharmD Salaun, Véronique, PharmD Macro, Magaret, MD Verneuil, Laurence, MD, PhD Lobbedez, Thierry, MD Castrale, Cindy, MD Boutemy, Jonathan, MD Cheze, Stéphane, MD Geffray, Loïk, MD Schleinitz, Nicolas, MD, PhD Rey, Jérôme, MD Lazaro, Estibaliz, MD, PhD Guillevin, Loïc, MD, PhD Bienvenu, Boris, MD, PhD |
| description | Abstract Objective The association between vasculitis and large granular lymphocyte (LGL) leukemia has rarely been reported or investigated. Thus, we assessed the clinical and biological phenotypes of LGL leukemia associated with vasculitis. Results We studied a series of 11 patients displaying LGL leukemia associated with vasculitis (LAV). The mean age at diagnosis of LGL leukemia was 60.3 years; there were nine women and two men. The mean follow-up period was 45 months. The main LGL lineage was T-LGL (10 patients), and only one NK-LGL was identified. Clinical and biological features of T-LGL leukemia were compared with those from the 2009 French T-LGL registry. We did not find any relevant differences except that patients with LAV were predominantly female ( p < 0.05). The most frequently observed vasculitis was cryoglobulinemia ( n = 5). Three patients presented with cutaneous leukocytoclastic angiitis, two patients had ANCA-negative microscopic polyangiitis, and one patient had giant cell arteritis. The main clinical features involved the skin, e.g., purpura (91%), arthralgia (37%), peripheral neuritis (27%), and renal glomerulonephritis (18%). The most frequent histologic finding was leucocytoclastic vasculitis (54%). The rate of complete remission was high; i.e., 80%. A minority of patients had a vasculitis relapse (27%). Three patients (27%) died; one death was related to LGL leukemia (acute infection) and the two other deaths were related to vasculitis (both with heart failure). Conclusion We conclude that vasculitis is overrepresented in the population of LGL patients, LAV predominantly affects women, vasculitis preferentially affects the small vessels, and LAV has high rate of complete response. |
| doi_str_mv | 10.1016/j.semarthrit.2013.07.002 |
| format | Article |
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Thus, we assessed the clinical and biological phenotypes of LGL leukemia associated with vasculitis. Results We studied a series of 11 patients displaying LGL leukemia associated with vasculitis (LAV). The mean age at diagnosis of LGL leukemia was 60.3 years; there were nine women and two men. The mean follow-up period was 45 months. The main LGL lineage was T-LGL (10 patients), and only one NK-LGL was identified. Clinical and biological features of T-LGL leukemia were compared with those from the 2009 French T-LGL registry. We did not find any relevant differences except that patients with LAV were predominantly female ( p < 0.05). The most frequently observed vasculitis was cryoglobulinemia ( n = 5). Three patients presented with cutaneous leukocytoclastic angiitis, two patients had ANCA-negative microscopic polyangiitis, and one patient had giant cell arteritis. The main clinical features involved the skin, e.g., purpura (91%), arthralgia (37%), peripheral neuritis (27%), and renal glomerulonephritis (18%). The most frequent histologic finding was leucocytoclastic vasculitis (54%). The rate of complete remission was high; i.e., 80%. A minority of patients had a vasculitis relapse (27%). Three patients (27%) died; one death was related to LGL leukemia (acute infection) and the two other deaths were related to vasculitis (both with heart failure). Conclusion We conclude that vasculitis is overrepresented in the population of LGL patients, LAV predominantly affects women, vasculitis preferentially affects the small vessels, and LAV has high rate of complete response.</description><identifier>ISSN: 0049-0172</identifier><identifier>EISSN: 1532-866X</identifier><identifier>DOI: 10.1016/j.semarthrit.2013.07.002</identifier><identifier>PMID: 24326032</identifier><language>eng</language><publisher>United States: Elsevier Inc</publisher><subject>Adult ; Aged ; Female ; Glucocorticoids - therapeutic use ; Humans ; Immunosuppressive Agents - therapeutic use ; Large granular lymphocyte leukemia ; Leukemia, Large Granular Lymphocytic - complications ; Leukemia, Large Granular Lymphocytic - drug therapy ; LGL leukemia ; Male ; Middle Aged ; Rheumatology ; Treatment Outcome ; Vasculitis ; Vasculitis - complications ; Vasculitis - drug therapy</subject><ispartof>Seminars in arthritis and rheumatism, 2013-12, Vol.43 (3), p.362-366</ispartof><rights>Elsevier Inc.</rights><rights>2013 Elsevier Inc.</rights><rights>Copyright © 2013 Elsevier Inc. All rights reserved.</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c429t-4dc46330b7e41aaa714ca70938a82cab008ce48a0aaaf1716a06498a9125d5873</citedby><cites>FETCH-LOGICAL-c429t-4dc46330b7e41aaa714ca70938a82cab008ce48a0aaaf1716a06498a9125d5873</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktohtml>$$Uhttps://www.sciencedirect.com/science/article/pii/S0049017213001509$$EHTML$$P50$$Gelsevier$$H</linktohtml><link.rule.ids>314,776,780,3537,27901,27902,65306</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/24326032$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Audemard, Alexandra, MD</creatorcontrib><creatorcontrib>Lamy, Thierry, MD, PhD</creatorcontrib><creatorcontrib>Bareau, Benoît, MD</creatorcontrib><creatorcontrib>Sicre, Flore, MD</creatorcontrib><creatorcontrib>Suarez, Felipe, MD</creatorcontrib><creatorcontrib>Truquet, Florence, PharmD</creatorcontrib><creatorcontrib>Salaun, Véronique, PharmD</creatorcontrib><creatorcontrib>Macro, Magaret, MD</creatorcontrib><creatorcontrib>Verneuil, Laurence, MD, PhD</creatorcontrib><creatorcontrib>Lobbedez, Thierry, MD</creatorcontrib><creatorcontrib>Castrale, Cindy, MD</creatorcontrib><creatorcontrib>Boutemy, Jonathan, MD</creatorcontrib><creatorcontrib>Cheze, Stéphane, MD</creatorcontrib><creatorcontrib>Geffray, Loïk, MD</creatorcontrib><creatorcontrib>Schleinitz, Nicolas, MD, PhD</creatorcontrib><creatorcontrib>Rey, Jérôme, MD</creatorcontrib><creatorcontrib>Lazaro, Estibaliz, MD, PhD</creatorcontrib><creatorcontrib>Guillevin, Loïc, MD, PhD</creatorcontrib><creatorcontrib>Bienvenu, Boris, MD, PhD</creatorcontrib><title>Vasculitis associated with large granular lymphocyte (LGL) leukemia: Presentation and treatment outcomes of 11 cases</title><title>Seminars in arthritis and rheumatism</title><addtitle>Semin Arthritis Rheum</addtitle><description>Abstract Objective The association between vasculitis and large granular lymphocyte (LGL) leukemia has rarely been reported or investigated. Thus, we assessed the clinical and biological phenotypes of LGL leukemia associated with vasculitis. Results We studied a series of 11 patients displaying LGL leukemia associated with vasculitis (LAV). The mean age at diagnosis of LGL leukemia was 60.3 years; there were nine women and two men. The mean follow-up period was 45 months. The main LGL lineage was T-LGL (10 patients), and only one NK-LGL was identified. Clinical and biological features of T-LGL leukemia were compared with those from the 2009 French T-LGL registry. We did not find any relevant differences except that patients with LAV were predominantly female ( p < 0.05). The most frequently observed vasculitis was cryoglobulinemia ( n = 5). Three patients presented with cutaneous leukocytoclastic angiitis, two patients had ANCA-negative microscopic polyangiitis, and one patient had giant cell arteritis. The main clinical features involved the skin, e.g., purpura (91%), arthralgia (37%), peripheral neuritis (27%), and renal glomerulonephritis (18%). The most frequent histologic finding was leucocytoclastic vasculitis (54%). The rate of complete remission was high; i.e., 80%. A minority of patients had a vasculitis relapse (27%). Three patients (27%) died; one death was related to LGL leukemia (acute infection) and the two other deaths were related to vasculitis (both with heart failure). Conclusion We conclude that vasculitis is overrepresented in the population of LGL patients, LAV predominantly affects women, vasculitis preferentially affects the small vessels, and LAV has high rate of complete response.</description><subject>Adult</subject><subject>Aged</subject><subject>Female</subject><subject>Glucocorticoids - therapeutic use</subject><subject>Humans</subject><subject>Immunosuppressive Agents - therapeutic use</subject><subject>Large granular lymphocyte leukemia</subject><subject>Leukemia, Large Granular Lymphocytic - complications</subject><subject>Leukemia, Large Granular Lymphocytic - drug therapy</subject><subject>LGL leukemia</subject><subject>Male</subject><subject>Middle Aged</subject><subject>Rheumatology</subject><subject>Treatment Outcome</subject><subject>Vasculitis</subject><subject>Vasculitis - complications</subject><subject>Vasculitis - drug therapy</subject><issn>0049-0172</issn><issn>1532-866X</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2013</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNqNkU2PEzEMhiMEYrsLfwHluBxmcD6ameGABCt2QaoEEh_iFrkZd5vuzKQkGVD_Pam6gMSJky37tS0_L2NcQC1AmBe7OtGIMW-jz7UEoWpoagD5gC3EUsmqNebbQ7YA0F0FopFn7DylHYAQBprH7ExqJQ0ouWD5KyY3Dz77xDGl4Dxm6vlPn7d8wHhL_DbiNJeUD4dxvw3ukIlfrm5Wz_lA8x2NHl_yj5ESTRmzDxPHqec5EuaxlHiYswsjJR42XAjuMFF6wh5tcEj09D5esC_Xbz9fvatWH27eX71eVU7LLle6d9ooBeuGtEDERmiHDXSqxVY6XAO0jnSLUHob0QiDYHTXYifksl-2jbpgl6e9-xi-z5SyHX1yNAw4UZiTFdo0RrVGySJtT1IXQ0qRNnYffUF8sALskbnd2b_M7ZG5hcYW5mX02f2VeT1S_2fwN-QieHMSUPn1h6dok_M0Oep9JJdtH_z_XHn1zxI3-Mk7HO7oQGkX5jgVllbYJC3YT0fvj9YLVWxfFmi_ABofrek</recordid><startdate>20131201</startdate><enddate>20131201</enddate><creator>Audemard, Alexandra, MD</creator><creator>Lamy, Thierry, MD, PhD</creator><creator>Bareau, Benoît, MD</creator><creator>Sicre, Flore, MD</creator><creator>Suarez, Felipe, MD</creator><creator>Truquet, Florence, PharmD</creator><creator>Salaun, Véronique, PharmD</creator><creator>Macro, Magaret, MD</creator><creator>Verneuil, Laurence, MD, PhD</creator><creator>Lobbedez, Thierry, MD</creator><creator>Castrale, Cindy, MD</creator><creator>Boutemy, Jonathan, MD</creator><creator>Cheze, Stéphane, MD</creator><creator>Geffray, Loïk, MD</creator><creator>Schleinitz, Nicolas, MD, PhD</creator><creator>Rey, Jérôme, MD</creator><creator>Lazaro, Estibaliz, MD, PhD</creator><creator>Guillevin, Loïc, MD, PhD</creator><creator>Bienvenu, Boris, MD, PhD</creator><general>Elsevier Inc</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></search><sort><creationdate>20131201</creationdate><title>Vasculitis associated with large granular lymphocyte (LGL) leukemia: Presentation and treatment outcomes of 11 cases</title><author>Audemard, Alexandra, MD ; Lamy, Thierry, MD, PhD ; Bareau, Benoît, MD ; Sicre, Flore, MD ; Suarez, Felipe, MD ; Truquet, Florence, PharmD ; Salaun, Véronique, PharmD ; Macro, Magaret, MD ; Verneuil, Laurence, MD, PhD ; Lobbedez, Thierry, MD ; Castrale, Cindy, MD ; Boutemy, Jonathan, MD ; Cheze, Stéphane, MD ; Geffray, Loïk, MD ; Schleinitz, Nicolas, MD, PhD ; Rey, Jérôme, MD ; Lazaro, Estibaliz, MD, PhD ; Guillevin, Loïc, MD, PhD ; Bienvenu, Boris, MD, PhD</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c429t-4dc46330b7e41aaa714ca70938a82cab008ce48a0aaaf1716a06498a9125d5873</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2013</creationdate><topic>Adult</topic><topic>Aged</topic><topic>Female</topic><topic>Glucocorticoids - therapeutic use</topic><topic>Humans</topic><topic>Immunosuppressive Agents - therapeutic use</topic><topic>Large granular lymphocyte leukemia</topic><topic>Leukemia, Large Granular Lymphocytic - complications</topic><topic>Leukemia, Large Granular Lymphocytic - drug therapy</topic><topic>LGL leukemia</topic><topic>Male</topic><topic>Middle Aged</topic><topic>Rheumatology</topic><topic>Treatment Outcome</topic><topic>Vasculitis</topic><topic>Vasculitis - complications</topic><topic>Vasculitis - drug therapy</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Audemard, Alexandra, MD</creatorcontrib><creatorcontrib>Lamy, Thierry, MD, PhD</creatorcontrib><creatorcontrib>Bareau, Benoît, MD</creatorcontrib><creatorcontrib>Sicre, Flore, MD</creatorcontrib><creatorcontrib>Suarez, Felipe, MD</creatorcontrib><creatorcontrib>Truquet, Florence, PharmD</creatorcontrib><creatorcontrib>Salaun, Véronique, PharmD</creatorcontrib><creatorcontrib>Macro, Magaret, MD</creatorcontrib><creatorcontrib>Verneuil, Laurence, MD, PhD</creatorcontrib><creatorcontrib>Lobbedez, Thierry, MD</creatorcontrib><creatorcontrib>Castrale, Cindy, MD</creatorcontrib><creatorcontrib>Boutemy, Jonathan, MD</creatorcontrib><creatorcontrib>Cheze, Stéphane, MD</creatorcontrib><creatorcontrib>Geffray, Loïk, MD</creatorcontrib><creatorcontrib>Schleinitz, Nicolas, MD, PhD</creatorcontrib><creatorcontrib>Rey, Jérôme, MD</creatorcontrib><creatorcontrib>Lazaro, Estibaliz, MD, PhD</creatorcontrib><creatorcontrib>Guillevin, Loïc, MD, PhD</creatorcontrib><creatorcontrib>Bienvenu, Boris, MD, PhD</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><jtitle>Seminars in arthritis and rheumatism</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Audemard, Alexandra, MD</au><au>Lamy, Thierry, MD, PhD</au><au>Bareau, Benoît, MD</au><au>Sicre, Flore, MD</au><au>Suarez, Felipe, MD</au><au>Truquet, Florence, PharmD</au><au>Salaun, Véronique, PharmD</au><au>Macro, Magaret, MD</au><au>Verneuil, Laurence, MD, PhD</au><au>Lobbedez, Thierry, MD</au><au>Castrale, Cindy, MD</au><au>Boutemy, Jonathan, MD</au><au>Cheze, Stéphane, MD</au><au>Geffray, Loïk, MD</au><au>Schleinitz, Nicolas, MD, PhD</au><au>Rey, Jérôme, MD</au><au>Lazaro, Estibaliz, MD, PhD</au><au>Guillevin, Loïc, MD, PhD</au><au>Bienvenu, Boris, MD, PhD</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Vasculitis associated with large granular lymphocyte (LGL) leukemia: Presentation and treatment outcomes of 11 cases</atitle><jtitle>Seminars in arthritis and rheumatism</jtitle><addtitle>Semin Arthritis Rheum</addtitle><date>2013-12-01</date><risdate>2013</risdate><volume>43</volume><issue>3</issue><spage>362</spage><epage>366</epage><pages>362-366</pages><issn>0049-0172</issn><eissn>1532-866X</eissn><abstract>Abstract Objective The association between vasculitis and large granular lymphocyte (LGL) leukemia has rarely been reported or investigated. Thus, we assessed the clinical and biological phenotypes of LGL leukemia associated with vasculitis. Results We studied a series of 11 patients displaying LGL leukemia associated with vasculitis (LAV). The mean age at diagnosis of LGL leukemia was 60.3 years; there were nine women and two men. The mean follow-up period was 45 months. The main LGL lineage was T-LGL (10 patients), and only one NK-LGL was identified. Clinical and biological features of T-LGL leukemia were compared with those from the 2009 French T-LGL registry. We did not find any relevant differences except that patients with LAV were predominantly female ( p < 0.05). The most frequently observed vasculitis was cryoglobulinemia ( n = 5). Three patients presented with cutaneous leukocytoclastic angiitis, two patients had ANCA-negative microscopic polyangiitis, and one patient had giant cell arteritis. The main clinical features involved the skin, e.g., purpura (91%), arthralgia (37%), peripheral neuritis (27%), and renal glomerulonephritis (18%). The most frequent histologic finding was leucocytoclastic vasculitis (54%). The rate of complete remission was high; i.e., 80%. A minority of patients had a vasculitis relapse (27%). Three patients (27%) died; one death was related to LGL leukemia (acute infection) and the two other deaths were related to vasculitis (both with heart failure). Conclusion We conclude that vasculitis is overrepresented in the population of LGL patients, LAV predominantly affects women, vasculitis preferentially affects the small vessels, and LAV has high rate of complete response.</abstract><cop>United States</cop><pub>Elsevier Inc</pub><pmid>24326032</pmid><doi>10.1016/j.semarthrit.2013.07.002</doi><tpages>5</tpages></addata></record> |
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| subjects | Adult Aged Female Glucocorticoids - therapeutic use Humans Immunosuppressive Agents - therapeutic use Large granular lymphocyte leukemia Leukemia, Large Granular Lymphocytic - complications Leukemia, Large Granular Lymphocytic - drug therapy LGL leukemia Male Middle Aged Rheumatology Treatment Outcome Vasculitis Vasculitis - complications Vasculitis - drug therapy |
| title | Vasculitis associated with large granular lymphocyte (LGL) leukemia: Presentation and treatment outcomes of 11 cases |
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