A national, multicenter, retrospective study of Castleman disease in China implementing CDCN criteria
Castleman disease (CD) is a group of rare and heterogenous lymphoproliferative disorders including unicentric CD (UCD), human herpesvirus-8(HHV-8)-associated multicentric CD (HHV8-MCD), and HHV-8-negative/idiopathic multicentric CD (iMCD). Knowledge of CD mainly comes from case series or retrospecti...
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| Veröffentlicht in: | The Lancet regional health. Western Pacific 2023-05, Vol.34, p.100720-100720, Article 100720 |
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| creator | Zhang, Lu Dong, Yu-jun Peng, Hong-ling Li, Hao Zhang, Ming-zhi Wang, Hui-han Liu, Qin-hua Su, Li-ping Zhong, Li-ye Wu, Wen-jun Huang, Liang Yan, Xiao-jing Fan, Lei Tang, Wen-jiao Li, Zhen-ling Bi, Lin-tao Li, Yan Gao, Guang-xun Gao, Li Liu, Ting-bo Wei, Yong-qiang Liu, Yao Yu, Li Zhou, Hui Sun, Chun-yan Qian, Wen-bin Zou, De-hui Zhang, Hui-lai Ding, Kai-yang Wang, Xiao-bo Bai, Ou Huang, Wen-rong Chen, Bing Yang, Lin Song, Jia Gao, Da Chen, Tong Luo, Jun Wang, Shu-ye Ma, Liang-ming Fajgenbaum, David C. Li, Jian |
| description | Castleman disease (CD) is a group of rare and heterogenous lymphoproliferative disorders including unicentric CD (UCD), human herpesvirus-8(HHV-8)-associated multicentric CD (HHV8-MCD), and HHV-8-negative/idiopathic multicentric CD (iMCD). Knowledge of CD mainly comes from case series or retrospective studies, but the inclusion criteria of these studies vary because the Castleman Disease Collaborative Network (CDCN) diagnostic criteria for iMCD and UCD were not available until 2017 and 2020, respectively. Further, these criteria and guidelines have not been systematically evaluated.
In this national, multicenter, retrospective study implementing CDCN criteria, we enrolled 1634 CD patients (UCD, n = 903; MCD, n = 731) from 2000 to 2021 at 40 Chinese institutions to depict clinical features, treatment options, and prognostic factors of CD.
Among UCD, there were 162 (17.9%) patients with an MCD-like inflammatory state. Among MCD, there were 12 HHV8-MCD patients and 719 HHV-8-negative MCD patients, which included 139 asymptomatic MCD (aMCD) and 580 iMCD meeting clinical criteria. Of 580 iMCD patients, 41 (7.1%) met iMCD-TAFRO criteria, the others were iMCD-NOS. iMCD-NOS were further divided into iMCD-IPL (n = 97) and iMCD-NOS without IPL (n = 442). Among iMCD patients with first-line treatment data, a trend from pulse combination chemotherapy toward continuous treatment was observed. Survival analysis revealed significant differences between subtypes and severe iMCD (HR = 3.747; 95% CI: 2.112–6.649, p |
| doi_str_mv | 10.1016/j.lanwpc.2023.100720 |
| format | Article |
| fullrecord | <record><control><sourceid>proquest_pubme</sourceid><recordid>TN_cdi_pubmedcentral_primary_oai_pubmedcentral_nih_gov_10240357</recordid><sourceformat>XML</sourceformat><sourcesystem>PC</sourcesystem><els_id>S266660652300038X</els_id><sourcerecordid>2823499054</sourcerecordid><originalsourceid>FETCH-LOGICAL-c464t-70bb6d53cf8aadb8d1d31c4bbe85b943f06a254dfd57e56224fbe5881c9ff3f73</originalsourceid><addsrcrecordid>eNp9kUtv1TAQhSMEolXpP0DISxa9t34n2YCq0AdSBRtYW449bn2VOMF2btV_j69SqrLpypbnzJnx-arqI8Fbgok8320HHR5ms6WYsvKEa4rfVMdUSrmRWIq3L-5H1WlKO4wxFYSRFr-vjlhNG9bWzXEFFyjo7KeghzM0LkP2BkKGeIYi5DilGUz2e0ApL_YRTQ51OuUBRh2Q9Ql0AuQD6u590MiPc6mUdh_uUPet-4FM9MXL6w_VO6eHBKdP50n1--ryV3ezuf15_b27uN0YLnne1LjvpRXMuEZr2zeWWEYM73toRN9y5rDUVHDrrKhBSEq560E0DTGtc8zV7KT6uvrOSz-CPXwl6kHN0Y86PqpJe_V_Jfh7dTftFcGUYyYODp-fHOL0Z4GU1eiTgaHEDdOSFG0o422LBS9SvkpNySlFcM9zCFYHSmqnVkrqQEmtlErbp5c7Pjf9Y1IEX1YBlKT2HqJKxkMwYH0sNJSd_OsT_gJ-LKeK</addsrcrecordid><sourcetype>Open Access Repository</sourcetype><iscdi>true</iscdi><recordtype>article</recordtype><pqid>2823499054</pqid></control><display><type>article</type><title>A national, multicenter, retrospective study of Castleman disease in China implementing CDCN criteria</title><source>DOAJ Directory of Open Access Journals</source><source>EZB-FREE-00999 freely available EZB journals</source><source>PubMed Central</source><source>Alma/SFX Local Collection</source><creator>Zhang, Lu ; Dong, Yu-jun ; Peng, Hong-ling ; Li, Hao ; Zhang, Ming-zhi ; Wang, Hui-han ; Liu, Qin-hua ; Su, Li-ping ; Zhong, Li-ye ; Wu, Wen-jun ; Huang, Liang ; Yan, Xiao-jing ; Fan, Lei ; Tang, Wen-jiao ; Li, Zhen-ling ; Bi, Lin-tao ; Li, Yan ; Gao, Guang-xun ; Gao, Li ; Liu, Ting-bo ; Wei, Yong-qiang ; Liu, Yao ; Yu, Li ; Zhou, Hui ; Sun, Chun-yan ; Qian, Wen-bin ; Zou, De-hui ; Zhang, Hui-lai ; Ding, Kai-yang ; Wang, Xiao-bo ; Bai, Ou ; Huang, Wen-rong ; Chen, Bing ; Yang, Lin ; Song, Jia ; Gao, Da ; Chen, Tong ; Luo, Jun ; Wang, Shu-ye ; Ma, Liang-ming ; Fajgenbaum, David C. ; Li, Jian</creator><creatorcontrib>Zhang, Lu ; Dong, Yu-jun ; Peng, Hong-ling ; Li, Hao ; Zhang, Ming-zhi ; Wang, Hui-han ; Liu, Qin-hua ; Su, Li-ping ; Zhong, Li-ye ; Wu, Wen-jun ; Huang, Liang ; Yan, Xiao-jing ; Fan, Lei ; Tang, Wen-jiao ; Li, Zhen-ling ; Bi, Lin-tao ; Li, Yan ; Gao, Guang-xun ; Gao, Li ; Liu, Ting-bo ; Wei, Yong-qiang ; Liu, Yao ; Yu, Li ; Zhou, Hui ; Sun, Chun-yan ; Qian, Wen-bin ; Zou, De-hui ; Zhang, Hui-lai ; Ding, Kai-yang ; Wang, Xiao-bo ; Bai, Ou ; Huang, Wen-rong ; Chen, Bing ; Yang, Lin ; Song, Jia ; Gao, Da ; Chen, Tong ; Luo, Jun ; Wang, Shu-ye ; Ma, Liang-ming ; Fajgenbaum, David C. ; Li, Jian ; China Castleman Disease Network (CCDN)</creatorcontrib><description>Castleman disease (CD) is a group of rare and heterogenous lymphoproliferative disorders including unicentric CD (UCD), human herpesvirus-8(HHV-8)-associated multicentric CD (HHV8-MCD), and HHV-8-negative/idiopathic multicentric CD (iMCD). Knowledge of CD mainly comes from case series or retrospective studies, but the inclusion criteria of these studies vary because the Castleman Disease Collaborative Network (CDCN) diagnostic criteria for iMCD and UCD were not available until 2017 and 2020, respectively. Further, these criteria and guidelines have not been systematically evaluated.
In this national, multicenter, retrospective study implementing CDCN criteria, we enrolled 1634 CD patients (UCD, n = 903; MCD, n = 731) from 2000 to 2021 at 40 Chinese institutions to depict clinical features, treatment options, and prognostic factors of CD.
Among UCD, there were 162 (17.9%) patients with an MCD-like inflammatory state. Among MCD, there were 12 HHV8-MCD patients and 719 HHV-8-negative MCD patients, which included 139 asymptomatic MCD (aMCD) and 580 iMCD meeting clinical criteria. Of 580 iMCD patients, 41 (7.1%) met iMCD-TAFRO criteria, the others were iMCD-NOS. iMCD-NOS were further divided into iMCD-IPL (n = 97) and iMCD-NOS without IPL (n = 442). Among iMCD patients with first-line treatment data, a trend from pulse combination chemotherapy toward continuous treatment was observed. Survival analysis revealed significant differences between subtypes and severe iMCD (HR = 3.747; 95% CI: 2.112–6.649, p < 0.001) had worse outcome.
This study depicts a broad picture of CD, treatment options and survival information in China and validates the association between the CDCN's definition of severe iMCD and worse outcomes, requiring more intensive treatment.
Beijing Municipal Commission of Science and Technology, CAMS Innovation Fund and National High Level Hospital Clinical Research Funding.</description><identifier>ISSN: 2666-6065</identifier><identifier>EISSN: 2666-6065</identifier><identifier>DOI: 10.1016/j.lanwpc.2023.100720</identifier><identifier>PMID: 37283978</identifier><language>eng</language><publisher>England: Elsevier Ltd</publisher><subject>Castleman disease ; Multicenter study ; Survival analysis ; Treatment options</subject><ispartof>The Lancet regional health. Western Pacific, 2023-05, Vol.34, p.100720-100720, Article 100720</ispartof><rights>2023</rights><rights>2023 Published by Elsevier Ltd.</rights><rights>2023 Published by Elsevier Ltd. 2023</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c464t-70bb6d53cf8aadb8d1d31c4bbe85b943f06a254dfd57e56224fbe5881c9ff3f73</citedby><cites>FETCH-LOGICAL-c464t-70bb6d53cf8aadb8d1d31c4bbe85b943f06a254dfd57e56224fbe5881c9ff3f73</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/PMC10240357/pdf/$$EPDF$$P50$$Gpubmedcentral$$Hfree_for_read</linktopdf><linktohtml>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC10240357/$$EHTML$$P50$$Gpubmedcentral$$Hfree_for_read</linktohtml><link.rule.ids>230,314,727,780,784,864,885,27924,27925,53791,53793</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/37283978$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Zhang, Lu</creatorcontrib><creatorcontrib>Dong, Yu-jun</creatorcontrib><creatorcontrib>Peng, Hong-ling</creatorcontrib><creatorcontrib>Li, Hao</creatorcontrib><creatorcontrib>Zhang, Ming-zhi</creatorcontrib><creatorcontrib>Wang, Hui-han</creatorcontrib><creatorcontrib>Liu, Qin-hua</creatorcontrib><creatorcontrib>Su, Li-ping</creatorcontrib><creatorcontrib>Zhong, Li-ye</creatorcontrib><creatorcontrib>Wu, Wen-jun</creatorcontrib><creatorcontrib>Huang, Liang</creatorcontrib><creatorcontrib>Yan, Xiao-jing</creatorcontrib><creatorcontrib>Fan, Lei</creatorcontrib><creatorcontrib>Tang, Wen-jiao</creatorcontrib><creatorcontrib>Li, Zhen-ling</creatorcontrib><creatorcontrib>Bi, Lin-tao</creatorcontrib><creatorcontrib>Li, Yan</creatorcontrib><creatorcontrib>Gao, Guang-xun</creatorcontrib><creatorcontrib>Gao, Li</creatorcontrib><creatorcontrib>Liu, Ting-bo</creatorcontrib><creatorcontrib>Wei, Yong-qiang</creatorcontrib><creatorcontrib>Liu, Yao</creatorcontrib><creatorcontrib>Yu, Li</creatorcontrib><creatorcontrib>Zhou, Hui</creatorcontrib><creatorcontrib>Sun, Chun-yan</creatorcontrib><creatorcontrib>Qian, Wen-bin</creatorcontrib><creatorcontrib>Zou, De-hui</creatorcontrib><creatorcontrib>Zhang, Hui-lai</creatorcontrib><creatorcontrib>Ding, Kai-yang</creatorcontrib><creatorcontrib>Wang, Xiao-bo</creatorcontrib><creatorcontrib>Bai, Ou</creatorcontrib><creatorcontrib>Huang, Wen-rong</creatorcontrib><creatorcontrib>Chen, Bing</creatorcontrib><creatorcontrib>Yang, Lin</creatorcontrib><creatorcontrib>Song, Jia</creatorcontrib><creatorcontrib>Gao, Da</creatorcontrib><creatorcontrib>Chen, Tong</creatorcontrib><creatorcontrib>Luo, Jun</creatorcontrib><creatorcontrib>Wang, Shu-ye</creatorcontrib><creatorcontrib>Ma, Liang-ming</creatorcontrib><creatorcontrib>Fajgenbaum, David C.</creatorcontrib><creatorcontrib>Li, Jian</creatorcontrib><creatorcontrib>China Castleman Disease Network (CCDN)</creatorcontrib><title>A national, multicenter, retrospective study of Castleman disease in China implementing CDCN criteria</title><title>The Lancet regional health. Western Pacific</title><addtitle>Lancet Reg Health West Pac</addtitle><description>Castleman disease (CD) is a group of rare and heterogenous lymphoproliferative disorders including unicentric CD (UCD), human herpesvirus-8(HHV-8)-associated multicentric CD (HHV8-MCD), and HHV-8-negative/idiopathic multicentric CD (iMCD). Knowledge of CD mainly comes from case series or retrospective studies, but the inclusion criteria of these studies vary because the Castleman Disease Collaborative Network (CDCN) diagnostic criteria for iMCD and UCD were not available until 2017 and 2020, respectively. Further, these criteria and guidelines have not been systematically evaluated.
In this national, multicenter, retrospective study implementing CDCN criteria, we enrolled 1634 CD patients (UCD, n = 903; MCD, n = 731) from 2000 to 2021 at 40 Chinese institutions to depict clinical features, treatment options, and prognostic factors of CD.
Among UCD, there were 162 (17.9%) patients with an MCD-like inflammatory state. Among MCD, there were 12 HHV8-MCD patients and 719 HHV-8-negative MCD patients, which included 139 asymptomatic MCD (aMCD) and 580 iMCD meeting clinical criteria. Of 580 iMCD patients, 41 (7.1%) met iMCD-TAFRO criteria, the others were iMCD-NOS. iMCD-NOS were further divided into iMCD-IPL (n = 97) and iMCD-NOS without IPL (n = 442). Among iMCD patients with first-line treatment data, a trend from pulse combination chemotherapy toward continuous treatment was observed. Survival analysis revealed significant differences between subtypes and severe iMCD (HR = 3.747; 95% CI: 2.112–6.649, p < 0.001) had worse outcome.
This study depicts a broad picture of CD, treatment options and survival information in China and validates the association between the CDCN's definition of severe iMCD and worse outcomes, requiring more intensive treatment.
Beijing Municipal Commission of Science and Technology, CAMS Innovation Fund and National High Level Hospital Clinical Research Funding.</description><subject>Castleman disease</subject><subject>Multicenter study</subject><subject>Survival analysis</subject><subject>Treatment options</subject><issn>2666-6065</issn><issn>2666-6065</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2023</creationdate><recordtype>article</recordtype><recordid>eNp9kUtv1TAQhSMEolXpP0DISxa9t34n2YCq0AdSBRtYW449bn2VOMF2btV_j69SqrLpypbnzJnx-arqI8Fbgok8320HHR5ms6WYsvKEa4rfVMdUSrmRWIq3L-5H1WlKO4wxFYSRFr-vjlhNG9bWzXEFFyjo7KeghzM0LkP2BkKGeIYi5DilGUz2e0ApL_YRTQ51OuUBRh2Q9Ql0AuQD6u590MiPc6mUdh_uUPet-4FM9MXL6w_VO6eHBKdP50n1--ryV3ezuf15_b27uN0YLnne1LjvpRXMuEZr2zeWWEYM73toRN9y5rDUVHDrrKhBSEq560E0DTGtc8zV7KT6uvrOSz-CPXwl6kHN0Y86PqpJe_V_Jfh7dTftFcGUYyYODp-fHOL0Z4GU1eiTgaHEDdOSFG0o422LBS9SvkpNySlFcM9zCFYHSmqnVkrqQEmtlErbp5c7Pjf9Y1IEX1YBlKT2HqJKxkMwYH0sNJSd_OsT_gJ-LKeK</recordid><startdate>20230501</startdate><enddate>20230501</enddate><creator>Zhang, Lu</creator><creator>Dong, Yu-jun</creator><creator>Peng, Hong-ling</creator><creator>Li, Hao</creator><creator>Zhang, Ming-zhi</creator><creator>Wang, Hui-han</creator><creator>Liu, Qin-hua</creator><creator>Su, Li-ping</creator><creator>Zhong, Li-ye</creator><creator>Wu, Wen-jun</creator><creator>Huang, Liang</creator><creator>Yan, Xiao-jing</creator><creator>Fan, Lei</creator><creator>Tang, Wen-jiao</creator><creator>Li, Zhen-ling</creator><creator>Bi, Lin-tao</creator><creator>Li, Yan</creator><creator>Gao, Guang-xun</creator><creator>Gao, Li</creator><creator>Liu, Ting-bo</creator><creator>Wei, Yong-qiang</creator><creator>Liu, Yao</creator><creator>Yu, Li</creator><creator>Zhou, Hui</creator><creator>Sun, Chun-yan</creator><creator>Qian, Wen-bin</creator><creator>Zou, De-hui</creator><creator>Zhang, Hui-lai</creator><creator>Ding, Kai-yang</creator><creator>Wang, Xiao-bo</creator><creator>Bai, Ou</creator><creator>Huang, Wen-rong</creator><creator>Chen, Bing</creator><creator>Yang, Lin</creator><creator>Song, Jia</creator><creator>Gao, Da</creator><creator>Chen, Tong</creator><creator>Luo, Jun</creator><creator>Wang, Shu-ye</creator><creator>Ma, Liang-ming</creator><creator>Fajgenbaum, David C.</creator><creator>Li, Jian</creator><general>Elsevier Ltd</general><general>Elsevier</general><scope>6I.</scope><scope>AAFTH</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7X8</scope><scope>5PM</scope></search><sort><creationdate>20230501</creationdate><title>A national, multicenter, retrospective study of Castleman disease in China implementing CDCN criteria</title><author>Zhang, Lu ; Dong, Yu-jun ; Peng, Hong-ling ; Li, Hao ; Zhang, Ming-zhi ; Wang, Hui-han ; Liu, Qin-hua ; Su, Li-ping ; Zhong, Li-ye ; Wu, Wen-jun ; Huang, Liang ; Yan, Xiao-jing ; Fan, Lei ; Tang, Wen-jiao ; Li, Zhen-ling ; Bi, Lin-tao ; Li, Yan ; Gao, Guang-xun ; Gao, Li ; Liu, Ting-bo ; Wei, Yong-qiang ; Liu, Yao ; Yu, Li ; Zhou, Hui ; Sun, Chun-yan ; Qian, Wen-bin ; Zou, De-hui ; Zhang, Hui-lai ; Ding, Kai-yang ; Wang, Xiao-bo ; Bai, Ou ; Huang, Wen-rong ; Chen, Bing ; Yang, Lin ; Song, Jia ; Gao, Da ; Chen, Tong ; Luo, Jun ; Wang, Shu-ye ; Ma, Liang-ming ; Fajgenbaum, David C. ; Li, Jian</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c464t-70bb6d53cf8aadb8d1d31c4bbe85b943f06a254dfd57e56224fbe5881c9ff3f73</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2023</creationdate><topic>Castleman disease</topic><topic>Multicenter study</topic><topic>Survival analysis</topic><topic>Treatment options</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Zhang, Lu</creatorcontrib><creatorcontrib>Dong, Yu-jun</creatorcontrib><creatorcontrib>Peng, Hong-ling</creatorcontrib><creatorcontrib>Li, Hao</creatorcontrib><creatorcontrib>Zhang, Ming-zhi</creatorcontrib><creatorcontrib>Wang, Hui-han</creatorcontrib><creatorcontrib>Liu, Qin-hua</creatorcontrib><creatorcontrib>Su, Li-ping</creatorcontrib><creatorcontrib>Zhong, Li-ye</creatorcontrib><creatorcontrib>Wu, Wen-jun</creatorcontrib><creatorcontrib>Huang, Liang</creatorcontrib><creatorcontrib>Yan, Xiao-jing</creatorcontrib><creatorcontrib>Fan, Lei</creatorcontrib><creatorcontrib>Tang, Wen-jiao</creatorcontrib><creatorcontrib>Li, Zhen-ling</creatorcontrib><creatorcontrib>Bi, Lin-tao</creatorcontrib><creatorcontrib>Li, Yan</creatorcontrib><creatorcontrib>Gao, Guang-xun</creatorcontrib><creatorcontrib>Gao, Li</creatorcontrib><creatorcontrib>Liu, Ting-bo</creatorcontrib><creatorcontrib>Wei, Yong-qiang</creatorcontrib><creatorcontrib>Liu, Yao</creatorcontrib><creatorcontrib>Yu, Li</creatorcontrib><creatorcontrib>Zhou, Hui</creatorcontrib><creatorcontrib>Sun, Chun-yan</creatorcontrib><creatorcontrib>Qian, Wen-bin</creatorcontrib><creatorcontrib>Zou, De-hui</creatorcontrib><creatorcontrib>Zhang, Hui-lai</creatorcontrib><creatorcontrib>Ding, Kai-yang</creatorcontrib><creatorcontrib>Wang, Xiao-bo</creatorcontrib><creatorcontrib>Bai, Ou</creatorcontrib><creatorcontrib>Huang, Wen-rong</creatorcontrib><creatorcontrib>Chen, Bing</creatorcontrib><creatorcontrib>Yang, Lin</creatorcontrib><creatorcontrib>Song, Jia</creatorcontrib><creatorcontrib>Gao, Da</creatorcontrib><creatorcontrib>Chen, Tong</creatorcontrib><creatorcontrib>Luo, Jun</creatorcontrib><creatorcontrib>Wang, Shu-ye</creatorcontrib><creatorcontrib>Ma, Liang-ming</creatorcontrib><creatorcontrib>Fajgenbaum, David C.</creatorcontrib><creatorcontrib>Li, Jian</creatorcontrib><creatorcontrib>China Castleman Disease Network (CCDN)</creatorcontrib><collection>ScienceDirect Open Access Titles</collection><collection>Elsevier:ScienceDirect:Open Access</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>MEDLINE - Academic</collection><collection>PubMed Central (Full Participant titles)</collection><jtitle>The Lancet regional health. Western Pacific</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Zhang, Lu</au><au>Dong, Yu-jun</au><au>Peng, Hong-ling</au><au>Li, Hao</au><au>Zhang, Ming-zhi</au><au>Wang, Hui-han</au><au>Liu, Qin-hua</au><au>Su, Li-ping</au><au>Zhong, Li-ye</au><au>Wu, Wen-jun</au><au>Huang, Liang</au><au>Yan, Xiao-jing</au><au>Fan, Lei</au><au>Tang, Wen-jiao</au><au>Li, Zhen-ling</au><au>Bi, Lin-tao</au><au>Li, Yan</au><au>Gao, Guang-xun</au><au>Gao, Li</au><au>Liu, Ting-bo</au><au>Wei, Yong-qiang</au><au>Liu, Yao</au><au>Yu, Li</au><au>Zhou, Hui</au><au>Sun, Chun-yan</au><au>Qian, Wen-bin</au><au>Zou, De-hui</au><au>Zhang, Hui-lai</au><au>Ding, Kai-yang</au><au>Wang, Xiao-bo</au><au>Bai, Ou</au><au>Huang, Wen-rong</au><au>Chen, Bing</au><au>Yang, Lin</au><au>Song, Jia</au><au>Gao, Da</au><au>Chen, Tong</au><au>Luo, Jun</au><au>Wang, Shu-ye</au><au>Ma, Liang-ming</au><au>Fajgenbaum, David C.</au><au>Li, Jian</au><aucorp>China Castleman Disease Network (CCDN)</aucorp><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>A national, multicenter, retrospective study of Castleman disease in China implementing CDCN criteria</atitle><jtitle>The Lancet regional health. Western Pacific</jtitle><addtitle>Lancet Reg Health West Pac</addtitle><date>2023-05-01</date><risdate>2023</risdate><volume>34</volume><spage>100720</spage><epage>100720</epage><pages>100720-100720</pages><artnum>100720</artnum><issn>2666-6065</issn><eissn>2666-6065</eissn><abstract>Castleman disease (CD) is a group of rare and heterogenous lymphoproliferative disorders including unicentric CD (UCD), human herpesvirus-8(HHV-8)-associated multicentric CD (HHV8-MCD), and HHV-8-negative/idiopathic multicentric CD (iMCD). Knowledge of CD mainly comes from case series or retrospective studies, but the inclusion criteria of these studies vary because the Castleman Disease Collaborative Network (CDCN) diagnostic criteria for iMCD and UCD were not available until 2017 and 2020, respectively. Further, these criteria and guidelines have not been systematically evaluated.
In this national, multicenter, retrospective study implementing CDCN criteria, we enrolled 1634 CD patients (UCD, n = 903; MCD, n = 731) from 2000 to 2021 at 40 Chinese institutions to depict clinical features, treatment options, and prognostic factors of CD.
Among UCD, there were 162 (17.9%) patients with an MCD-like inflammatory state. Among MCD, there were 12 HHV8-MCD patients and 719 HHV-8-negative MCD patients, which included 139 asymptomatic MCD (aMCD) and 580 iMCD meeting clinical criteria. Of 580 iMCD patients, 41 (7.1%) met iMCD-TAFRO criteria, the others were iMCD-NOS. iMCD-NOS were further divided into iMCD-IPL (n = 97) and iMCD-NOS without IPL (n = 442). Among iMCD patients with first-line treatment data, a trend from pulse combination chemotherapy toward continuous treatment was observed. Survival analysis revealed significant differences between subtypes and severe iMCD (HR = 3.747; 95% CI: 2.112–6.649, p < 0.001) had worse outcome.
This study depicts a broad picture of CD, treatment options and survival information in China and validates the association between the CDCN's definition of severe iMCD and worse outcomes, requiring more intensive treatment.
Beijing Municipal Commission of Science and Technology, CAMS Innovation Fund and National High Level Hospital Clinical Research Funding.</abstract><cop>England</cop><pub>Elsevier Ltd</pub><pmid>37283978</pmid><doi>10.1016/j.lanwpc.2023.100720</doi><tpages>1</tpages><oa>free_for_read</oa></addata></record> |
| fulltext | fulltext |
| identifier | ISSN: 2666-6065 |
| ispartof | The Lancet regional health. Western Pacific, 2023-05, Vol.34, p.100720-100720, Article 100720 |
| issn | 2666-6065 2666-6065 |
| language | eng |
| recordid | cdi_pubmedcentral_primary_oai_pubmedcentral_nih_gov_10240357 |
| source | DOAJ Directory of Open Access Journals; EZB-FREE-00999 freely available EZB journals; PubMed Central; Alma/SFX Local Collection |
| subjects | Castleman disease Multicenter study Survival analysis Treatment options |
| title | A national, multicenter, retrospective study of Castleman disease in China implementing CDCN criteria |
| url | https://sfx.bib-bvb.de/sfx_tum?ctx_ver=Z39.88-2004&ctx_enc=info:ofi/enc:UTF-8&ctx_tim=2024-12-27T12%3A35%3A52IST&url_ver=Z39.88-2004&url_ctx_fmt=infofi/fmt:kev:mtx:ctx&rfr_id=info:sid/primo.exlibrisgroup.com:primo3-Article-proquest_pubme&rft_val_fmt=info:ofi/fmt:kev:mtx:journal&rft.genre=article&rft.atitle=A%20national,%20multicenter,%20retrospective%20study%20of%20Castleman%20disease%20in%20China%20implementing%20CDCN%20criteria&rft.jtitle=The%20Lancet%20regional%20health.%20Western%20Pacific&rft.au=Zhang,%20Lu&rft.aucorp=China%20Castleman%20Disease%20Network%20(CCDN)&rft.date=2023-05-01&rft.volume=34&rft.spage=100720&rft.epage=100720&rft.pages=100720-100720&rft.artnum=100720&rft.issn=2666-6065&rft.eissn=2666-6065&rft_id=info:doi/10.1016/j.lanwpc.2023.100720&rft_dat=%3Cproquest_pubme%3E2823499054%3C/proquest_pubme%3E%3Curl%3E%3C/url%3E&disable_directlink=true&sfx.directlink=off&sfx.report_link=0&rft_id=info:oai/&rft_pqid=2823499054&rft_id=info:pmid/37283978&rft_els_id=S266660652300038X&rfr_iscdi=true |