Idiopathic multicentric Castleman disease with positive antiphospholipid antibody: atypical and undiagnosed autoimmune disease?
Idiopathic multicentric Castleman disease (iMCD) is a systemic disorder characterized by systemic inflammation and organ dysfunction associated with an increase in pro-inflammatory cytokines. Some patients with iMCD are positive for autoantibodies, although their significance and relationship with s...
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Veröffentlicht in: | Journal of Clinical and Experimental Hematopathology 2022, Vol.62(2), pp.99-105 |
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creator | Nishimura, Yoshito Nishikori, Asami Sawada, Haruki Czech, Torrey Otsuka, Yuki Nishimura, Midori Filiz Mizuno, Hiroki Sawa, Naoki Momose, Shuji Ohsawa, Kumiko Otsuka, Fumio Sato, Yasuharu |
description | Idiopathic multicentric Castleman disease (iMCD) is a systemic disorder characterized by systemic inflammation and organ dysfunction associated with an increase in pro-inflammatory cytokines. Some patients with iMCD are positive for autoantibodies, although their significance and relationship with specific associated autoimmune diseases are unclear. This study retrospectively analyzed the clinicopathological features of iMCD patients focusing on autoantibodies. Among 63 iMCD patients in our database, 19 were positive for at least one autoantibody. Among the 19, we identified five with plasma cell type (PC)-iMCD lymph node histopathology and positive anti-phospholipid antibodies. These patients were likely to have thrombocytopenia, anasarca, fever, reticulin fibrosis or renal insufficiency, organomegaly (TAFRO) symptoms, and thrombotic events. The present study suggests that patients with undiagnosed or atypical autoimmune diseases, including anti-phospholipid syndrome (APS), were treated for iMCD. APS may present with thrombocytopenia or even multi-organ failure, which overlap with clinical presentations of iMCD. Due to differences in the treatment regimen and follow-up, recognition of the undiagnosed autoimmune disease process in those suspected of iMCD is essential. Our study highlights the importance of complete exclusion of differential diagnoses in patients with iMCD in their diagnostic workup. |
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Some patients with iMCD are positive for autoantibodies, although their significance and relationship with specific associated autoimmune diseases are unclear. This study retrospectively analyzed the clinicopathological features of iMCD patients focusing on autoantibodies. Among 63 iMCD patients in our database, 19 were positive for at least one autoantibody. Among the 19, we identified five with plasma cell type (PC)-iMCD lymph node histopathology and positive anti-phospholipid antibodies. These patients were likely to have thrombocytopenia, anasarca, fever, reticulin fibrosis or renal insufficiency, organomegaly (TAFRO) symptoms, and thrombotic events. The present study suggests that patients with undiagnosed or atypical autoimmune diseases, including anti-phospholipid syndrome (APS), were treated for iMCD. APS may present with thrombocytopenia or even multi-organ failure, which overlap with clinical presentations of iMCD. Due to differences in the treatment regimen and follow-up, recognition of the undiagnosed autoimmune disease process in those suspected of iMCD is essential. Our study highlights the importance of complete exclusion of differential diagnoses in patients with iMCD in their diagnostic workup.</description><identifier>ISSN: 1346-4280</identifier><identifier>EISSN: 1880-9952</identifier><identifier>DOI: 10.3960/jslrt.21038</identifier><identifier>PMID: 35249898</identifier><language>eng</language><publisher>Japan: The Japanese Society for Lymphoreticular Tissue Research</publisher><subject>autoimmune, anti-phospholipid syndrome ; idiopathic plasmacytic lymphadenopathy ; multicentric Castleman disease ; Original</subject><ispartof>Journal of Clinical and Experimental Hematopathology, 2022, Vol.62(2), pp.99-105</ispartof><rights>2022 by The Japanese Society for Lymphoreticular Tissue Research</rights><rights>2022 by The Japanese Society for Lymphoreticular Tissue Research 2022 The Japanese Society for Lymphoreticular Tissue Research</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c573t-b196af11f09ffcf24bc431a224d9d18d823f80e107740095335d7077697e6c223</citedby><cites>FETCH-LOGICAL-c573t-b196af11f09ffcf24bc431a224d9d18d823f80e107740095335d7077697e6c223</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC9353850/pdf/$$EPDF$$P50$$Gpubmedcentral$$Hfree_for_read</linktopdf><linktohtml>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC9353850/$$EHTML$$P50$$Gpubmedcentral$$Hfree_for_read</linktohtml><link.rule.ids>230,314,727,780,784,864,885,1883,27924,27925,53791,53793</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/35249898$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Nishimura, Yoshito</creatorcontrib><creatorcontrib>Nishikori, Asami</creatorcontrib><creatorcontrib>Sawada, Haruki</creatorcontrib><creatorcontrib>Czech, Torrey</creatorcontrib><creatorcontrib>Otsuka, Yuki</creatorcontrib><creatorcontrib>Nishimura, Midori Filiz</creatorcontrib><creatorcontrib>Mizuno, Hiroki</creatorcontrib><creatorcontrib>Sawa, Naoki</creatorcontrib><creatorcontrib>Momose, Shuji</creatorcontrib><creatorcontrib>Ohsawa, Kumiko</creatorcontrib><creatorcontrib>Otsuka, Fumio</creatorcontrib><creatorcontrib>Sato, Yasuharu</creatorcontrib><title>Idiopathic multicentric Castleman disease with positive antiphospholipid antibody: atypical and undiagnosed autoimmune disease?</title><title>Journal of Clinical and Experimental Hematopathology</title><addtitle>J Clin Exp Hematopathol</addtitle><description>Idiopathic multicentric Castleman disease (iMCD) is a systemic disorder characterized by systemic inflammation and organ dysfunction associated with an increase in pro-inflammatory cytokines. Some patients with iMCD are positive for autoantibodies, although their significance and relationship with specific associated autoimmune diseases are unclear. This study retrospectively analyzed the clinicopathological features of iMCD patients focusing on autoantibodies. Among 63 iMCD patients in our database, 19 were positive for at least one autoantibody. Among the 19, we identified five with plasma cell type (PC)-iMCD lymph node histopathology and positive anti-phospholipid antibodies. These patients were likely to have thrombocytopenia, anasarca, fever, reticulin fibrosis or renal insufficiency, organomegaly (TAFRO) symptoms, and thrombotic events. The present study suggests that patients with undiagnosed or atypical autoimmune diseases, including anti-phospholipid syndrome (APS), were treated for iMCD. APS may present with thrombocytopenia or even multi-organ failure, which overlap with clinical presentations of iMCD. Due to differences in the treatment regimen and follow-up, recognition of the undiagnosed autoimmune disease process in those suspected of iMCD is essential. Our study highlights the importance of complete exclusion of differential diagnoses in patients with iMCD in their diagnostic workup.</description><subject>autoimmune, anti-phospholipid syndrome</subject><subject>idiopathic plasmacytic lymphadenopathy</subject><subject>multicentric Castleman disease</subject><subject>Original</subject><issn>1346-4280</issn><issn>1880-9952</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2022</creationdate><recordtype>article</recordtype><recordid>eNpVkctPxCAQxonR-D55N72bKo8-wINms_GVmHjRM5kFumXTlqZQzZ7818WubvTAwMz8-CbwIXRG8CUTBb5a-WYIl5RgxnfQIeEcp0LkdDeeWVakGeX4AB15v8I4K_KC7aMDltNMcMEP0eeTtq6HUFuVtGMTrDJdGGIyBx8a00KXaOsNeJN82FAnvfM22HeTQBdsXzsfV2N7q6fCwun1dQJh3VsFTSzpZOy0hWXnvInIGJxt27Ezv6K3J2ivgsab05_9GL3d373OH9Pnl4en-ew5VXnJQrogooCKkAqLqlIVzRYqYwQozbTQhGtOWcWxIbgsM4xFzliuy5gUojSFopQdo5uNbj8uWqOnV0Ij-8G2MKylAyv_dzpby6V7l4LljOc4ClxsBNTgvB9Mtb1LsPz2QU4-yMmHSJ__Hbdlfz8-ArMNsPIBlmYLwBAtaMyPWEElncK36Lanahik6dgXTwWhIQ</recordid><startdate>20220101</startdate><enddate>20220101</enddate><creator>Nishimura, Yoshito</creator><creator>Nishikori, Asami</creator><creator>Sawada, Haruki</creator><creator>Czech, Torrey</creator><creator>Otsuka, Yuki</creator><creator>Nishimura, Midori Filiz</creator><creator>Mizuno, Hiroki</creator><creator>Sawa, Naoki</creator><creator>Momose, Shuji</creator><creator>Ohsawa, Kumiko</creator><creator>Otsuka, Fumio</creator><creator>Sato, Yasuharu</creator><general>The Japanese Society for Lymphoreticular Tissue Research</general><general>JSLRT</general><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>5PM</scope></search><sort><creationdate>20220101</creationdate><title>Idiopathic multicentric Castleman disease with positive antiphospholipid antibody: atypical and undiagnosed autoimmune disease?</title><author>Nishimura, Yoshito ; Nishikori, Asami ; Sawada, Haruki ; Czech, Torrey ; Otsuka, Yuki ; Nishimura, Midori Filiz ; Mizuno, Hiroki ; Sawa, Naoki ; Momose, Shuji ; Ohsawa, Kumiko ; Otsuka, Fumio ; Sato, Yasuharu</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c573t-b196af11f09ffcf24bc431a224d9d18d823f80e107740095335d7077697e6c223</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2022</creationdate><topic>autoimmune, anti-phospholipid syndrome</topic><topic>idiopathic plasmacytic lymphadenopathy</topic><topic>multicentric Castleman disease</topic><topic>Original</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Nishimura, Yoshito</creatorcontrib><creatorcontrib>Nishikori, Asami</creatorcontrib><creatorcontrib>Sawada, Haruki</creatorcontrib><creatorcontrib>Czech, Torrey</creatorcontrib><creatorcontrib>Otsuka, Yuki</creatorcontrib><creatorcontrib>Nishimura, Midori Filiz</creatorcontrib><creatorcontrib>Mizuno, Hiroki</creatorcontrib><creatorcontrib>Sawa, Naoki</creatorcontrib><creatorcontrib>Momose, Shuji</creatorcontrib><creatorcontrib>Ohsawa, Kumiko</creatorcontrib><creatorcontrib>Otsuka, Fumio</creatorcontrib><creatorcontrib>Sato, Yasuharu</creatorcontrib><collection>PubMed</collection><collection>CrossRef</collection><collection>PubMed Central (Full Participant titles)</collection><jtitle>Journal of Clinical and Experimental Hematopathology</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Nishimura, Yoshito</au><au>Nishikori, Asami</au><au>Sawada, Haruki</au><au>Czech, Torrey</au><au>Otsuka, Yuki</au><au>Nishimura, Midori Filiz</au><au>Mizuno, Hiroki</au><au>Sawa, Naoki</au><au>Momose, Shuji</au><au>Ohsawa, Kumiko</au><au>Otsuka, Fumio</au><au>Sato, Yasuharu</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Idiopathic multicentric Castleman disease with positive antiphospholipid antibody: atypical and undiagnosed autoimmune disease?</atitle><jtitle>Journal of Clinical and Experimental Hematopathology</jtitle><addtitle>J Clin Exp Hematopathol</addtitle><date>2022-01-01</date><risdate>2022</risdate><volume>62</volume><issue>2</issue><spage>99</spage><epage>105</epage><pages>99-105</pages><artnum>21038</artnum><issn>1346-4280</issn><eissn>1880-9952</eissn><abstract>Idiopathic multicentric Castleman disease (iMCD) is a systemic disorder characterized by systemic inflammation and organ dysfunction associated with an increase in pro-inflammatory cytokines. Some patients with iMCD are positive for autoantibodies, although their significance and relationship with specific associated autoimmune diseases are unclear. This study retrospectively analyzed the clinicopathological features of iMCD patients focusing on autoantibodies. Among 63 iMCD patients in our database, 19 were positive for at least one autoantibody. Among the 19, we identified five with plasma cell type (PC)-iMCD lymph node histopathology and positive anti-phospholipid antibodies. These patients were likely to have thrombocytopenia, anasarca, fever, reticulin fibrosis or renal insufficiency, organomegaly (TAFRO) symptoms, and thrombotic events. The present study suggests that patients with undiagnosed or atypical autoimmune diseases, including anti-phospholipid syndrome (APS), were treated for iMCD. APS may present with thrombocytopenia or even multi-organ failure, which overlap with clinical presentations of iMCD. Due to differences in the treatment regimen and follow-up, recognition of the undiagnosed autoimmune disease process in those suspected of iMCD is essential. Our study highlights the importance of complete exclusion of differential diagnoses in patients with iMCD in their diagnostic workup.</abstract><cop>Japan</cop><pub>The Japanese Society for Lymphoreticular Tissue Research</pub><pmid>35249898</pmid><doi>10.3960/jslrt.21038</doi><tpages>7</tpages><oa>free_for_read</oa></addata></record> |
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subjects | autoimmune, anti-phospholipid syndrome idiopathic plasmacytic lymphadenopathy multicentric Castleman disease Original |
title | Idiopathic multicentric Castleman disease with positive antiphospholipid antibody: atypical and undiagnosed autoimmune disease? |
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