Intrathecal injection of methotrexate and dexamethasone for vasculitis granuloma of the fourth ventricle: a case report and literature review
Granulomatosis with polyangiitis (GPA) is a pauci-immune small vessel vasculitis characterised by neutrophil-mediated vasculitis and granuloma. The presence of intracranial parenchymal space-occupying lesions is rarely seen in GPA patients. In this manuscript, we report a case of GPA with granuloma...
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| Veröffentlicht in: | Clinical rheumatology 2024-03, Vol.43 (3), p.1217-1226 |
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| creator | Li, Chunyan Wang, Yu Lin, Yupei Gong, Qianla Wu, Bangcui Zheng, Wendan Tian, Yingying Chen, Yong Tian, Mei |
| description | Granulomatosis with polyangiitis (GPA) is a pauci-immune small vessel vasculitis characterised by neutrophil-mediated vasculitis and granuloma. The presence of intracranial parenchymal space-occupying lesions is rarely seen in GPA patients. In this manuscript, we report a case of GPA with granuloma of the fourth ventricle accompanied by obstructive hydrocephalus. Treatment with glucocorticoids (GCs) and multiple immunosuppressants cyclophosphamide (CYC), mycophenolate mofetil (MMF), and rituximab (RTX) showed poor efficacy in this case. After removal of the granuloma by craniotomy, GPA relapsed within 3 months. Under the premise of GC and MMF treatment combined with intrathecal injection of dexamethasone (DXM) and methotrexate (MTX), the intracranial granuloma gradually shrank, and the patient’s general condition was alleviated, showing that this is an effective treatment method.
Key Points
• To date, there are few reports of granulomatous vasculitis combined with granuloma of the fourth ventricle, and our case is the second.
• In this case, multiple immunosuppressants and rituximab were ineffective treatments, and the intracranial granuloma was effectively controlled by intrathecal injection of dexamethasone (DXM) and methotrexate (MTX).
• Based on this report, it can be suggested that intrathecal injection is effective in treating patients with GPA and central nervous system involvement, but large-scale sample studies are needed. |
| doi_str_mv | 10.1007/s10067-023-06777-4 |
| format | Article |
| fullrecord | <record><control><sourceid>proquest_cross</sourceid><recordid>TN_cdi_proquest_miscellaneous_2885541239</recordid><sourceformat>XML</sourceformat><sourcesystem>PC</sourcesystem><sourcerecordid>3153612532</sourcerecordid><originalsourceid>FETCH-LOGICAL-c359t-ac6a6f368e861f8959bdbad9e73e3acec5bd868d384e73dbe243e3d0988d2b743</originalsourceid><addsrcrecordid>eNqFUc1u1DAQthAVXQovwAFZ4tJLqP8S29xQRUulSlzgbDn2pJtVYi-2s8BD8M51ugUkDnDxWDPfj2Y-hF5R8pYSIi9yfTvZEMabWqVsxBO0oYKLRmuhn6INkZI0nGp1ip7nvCOEMKXpM3TKpaZCcblBP29CSbZswdkJj2EHrowx4DjgGco2lgTfbQFsg8e-ftemzTEAHmLCB5vdMo1lzPgu2bBMcbYrtcrV-ZLKFh-g6o9ugnfYYmcz4AT7mMqDYqVCNV_S2j2M8O0FOhnslOHlYz1DX64-fL782Nx-ur65fH_bON7q0ljX2W7gnQLV0UHpVve-t16D5MCtA9f2XnXKcyVqy_fARB14opXyrJeCn6Hzo-4-xa8L5GLmMTuYJhsgLtlw2vKOspaz_0KZUm0rKOO6Qt_8Bd3VI4S6iGGaKVFPLlZvdkS5FHNOMJh9GmebfhhKzJqrOeZqaq7mIVezkl4_Si_9DP435VeQFcCPgFxH4Q7SH-9_yN4DbVWw3w</addsrcrecordid><sourcetype>Aggregation Database</sourcetype><iscdi>true</iscdi><recordtype>article</recordtype><pqid>2928414844</pqid></control><display><type>article</type><title>Intrathecal injection of methotrexate and dexamethasone for vasculitis granuloma of the fourth ventricle: a case report and literature review</title><source>MEDLINE</source><source>Springer Nature - Complete Springer Journals</source><creator>Li, Chunyan ; Wang, Yu ; Lin, Yupei ; Gong, Qianla ; Wu, Bangcui ; Zheng, Wendan ; Tian, Yingying ; Chen, Yong ; Tian, Mei</creator><creatorcontrib>Li, Chunyan ; Wang, Yu ; Lin, Yupei ; Gong, Qianla ; Wu, Bangcui ; Zheng, Wendan ; Tian, Yingying ; Chen, Yong ; Tian, Mei</creatorcontrib><description>Granulomatosis with polyangiitis (GPA) is a pauci-immune small vessel vasculitis characterised by neutrophil-mediated vasculitis and granuloma. The presence of intracranial parenchymal space-occupying lesions is rarely seen in GPA patients. In this manuscript, we report a case of GPA with granuloma of the fourth ventricle accompanied by obstructive hydrocephalus. Treatment with glucocorticoids (GCs) and multiple immunosuppressants cyclophosphamide (CYC), mycophenolate mofetil (MMF), and rituximab (RTX) showed poor efficacy in this case. After removal of the granuloma by craniotomy, GPA relapsed within 3 months. Under the premise of GC and MMF treatment combined with intrathecal injection of dexamethasone (DXM) and methotrexate (MTX), the intracranial granuloma gradually shrank, and the patient’s general condition was alleviated, showing that this is an effective treatment method.
Key Points
• To date, there are few reports of granulomatous vasculitis combined with granuloma of the fourth ventricle, and our case is the second.
• In this case, multiple immunosuppressants and rituximab were ineffective treatments, and the intracranial granuloma was effectively controlled by intrathecal injection of dexamethasone (DXM) and methotrexate (MTX).
• Based on this report, it can be suggested that intrathecal injection is effective in treating patients with GPA and central nervous system involvement, but large-scale sample studies are needed.</description><identifier>ISSN: 0770-3198</identifier><identifier>EISSN: 1434-9949</identifier><identifier>DOI: 10.1007/s10067-023-06777-4</identifier><identifier>PMID: 37914837</identifier><language>eng</language><publisher>Cham: Springer International Publishing</publisher><subject>Anti-Neutrophil Cytoplasmic Antibody-Associated Vasculitis - complications ; Case Based Review ; Case reports ; case studies ; Central nervous system ; Cyclophosphamide ; Dexamethasone ; Dexamethasone - therapeutic use ; Fourth Ventricle ; glucocorticoids ; Granuloma ; Granuloma - complications ; Granuloma - drug therapy ; Granulomas ; Granulomatosis ; Granulomatosis with Polyangiitis - complications ; Humans ; Hydrocephalus ; Immunosuppressive agents ; Immunosuppressive Agents - therapeutic use ; Injection ; Injections, Spinal ; intrathecal injection ; Leukocytes (neutrophilic) ; Literature reviews ; Medicine ; Medicine & Public Health ; Methotrexate ; Methotrexate - therapeutic use ; Mycophenolate mofetil ; Mycophenolic acid ; Mycophenolic Acid - therapeutic use ; patients ; Rheumatology ; Rituximab ; Rituximab - therapeutic use ; Vasculitis ; Ventricle ; Ventricles (cerebral)</subject><ispartof>Clinical rheumatology, 2024-03, Vol.43 (3), p.1217-1226</ispartof><rights>The Author(s), under exclusive licence to International League of Associations for Rheumatology (ILAR) 2023. Springer Nature or its licensor (e.g. a society or other partner) holds exclusive rights to this article under a publishing agreement with the author(s) or other rightsholder(s); author self-archiving of the accepted manuscript version of this article is solely governed by the terms of such publishing agreement and applicable law.</rights><rights>2023. The Author(s), under exclusive licence to International League of Associations for Rheumatology (ILAR).</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><cites>FETCH-LOGICAL-c359t-ac6a6f368e861f8959bdbad9e73e3acec5bd868d384e73dbe243e3d0988d2b743</cites><orcidid>0009-0006-1531-643X ; 0000-0001-5025-041X ; 0009-0007-1471-4150 ; 0000-0001-5538-5081 ; 0000-0002-1633-2988 ; 0000-0003-3746-6958 ; 0009-0003-7287-898X ; 0000-0003-0510-782X ; 0009-0002-8062-9652</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://link.springer.com/content/pdf/10.1007/s10067-023-06777-4$$EPDF$$P50$$Gspringer$$H</linktopdf><linktohtml>$$Uhttps://link.springer.com/10.1007/s10067-023-06777-4$$EHTML$$P50$$Gspringer$$H</linktohtml><link.rule.ids>314,776,780,27903,27904,41467,42536,51298</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/37914837$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Li, Chunyan</creatorcontrib><creatorcontrib>Wang, Yu</creatorcontrib><creatorcontrib>Lin, Yupei</creatorcontrib><creatorcontrib>Gong, Qianla</creatorcontrib><creatorcontrib>Wu, Bangcui</creatorcontrib><creatorcontrib>Zheng, Wendan</creatorcontrib><creatorcontrib>Tian, Yingying</creatorcontrib><creatorcontrib>Chen, Yong</creatorcontrib><creatorcontrib>Tian, Mei</creatorcontrib><title>Intrathecal injection of methotrexate and dexamethasone for vasculitis granuloma of the fourth ventricle: a case report and literature review</title><title>Clinical rheumatology</title><addtitle>Clin Rheumatol</addtitle><addtitle>Clin Rheumatol</addtitle><description>Granulomatosis with polyangiitis (GPA) is a pauci-immune small vessel vasculitis characterised by neutrophil-mediated vasculitis and granuloma. The presence of intracranial parenchymal space-occupying lesions is rarely seen in GPA patients. In this manuscript, we report a case of GPA with granuloma of the fourth ventricle accompanied by obstructive hydrocephalus. Treatment with glucocorticoids (GCs) and multiple immunosuppressants cyclophosphamide (CYC), mycophenolate mofetil (MMF), and rituximab (RTX) showed poor efficacy in this case. After removal of the granuloma by craniotomy, GPA relapsed within 3 months. Under the premise of GC and MMF treatment combined with intrathecal injection of dexamethasone (DXM) and methotrexate (MTX), the intracranial granuloma gradually shrank, and the patient’s general condition was alleviated, showing that this is an effective treatment method.
Key Points
• To date, there are few reports of granulomatous vasculitis combined with granuloma of the fourth ventricle, and our case is the second.
• In this case, multiple immunosuppressants and rituximab were ineffective treatments, and the intracranial granuloma was effectively controlled by intrathecal injection of dexamethasone (DXM) and methotrexate (MTX).
• Based on this report, it can be suggested that intrathecal injection is effective in treating patients with GPA and central nervous system involvement, but large-scale sample studies are needed.</description><subject>Anti-Neutrophil Cytoplasmic Antibody-Associated Vasculitis - complications</subject><subject>Case Based Review</subject><subject>Case reports</subject><subject>case studies</subject><subject>Central nervous system</subject><subject>Cyclophosphamide</subject><subject>Dexamethasone</subject><subject>Dexamethasone - therapeutic use</subject><subject>Fourth Ventricle</subject><subject>glucocorticoids</subject><subject>Granuloma</subject><subject>Granuloma - complications</subject><subject>Granuloma - drug therapy</subject><subject>Granulomas</subject><subject>Granulomatosis</subject><subject>Granulomatosis with Polyangiitis - complications</subject><subject>Humans</subject><subject>Hydrocephalus</subject><subject>Immunosuppressive agents</subject><subject>Immunosuppressive Agents - therapeutic use</subject><subject>Injection</subject><subject>Injections, Spinal</subject><subject>intrathecal injection</subject><subject>Leukocytes (neutrophilic)</subject><subject>Literature reviews</subject><subject>Medicine</subject><subject>Medicine & Public Health</subject><subject>Methotrexate</subject><subject>Methotrexate - therapeutic use</subject><subject>Mycophenolate mofetil</subject><subject>Mycophenolic acid</subject><subject>Mycophenolic Acid - therapeutic use</subject><subject>patients</subject><subject>Rheumatology</subject><subject>Rituximab</subject><subject>Rituximab - therapeutic use</subject><subject>Vasculitis</subject><subject>Ventricle</subject><subject>Ventricles (cerebral)</subject><issn>0770-3198</issn><issn>1434-9949</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2024</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNqFUc1u1DAQthAVXQovwAFZ4tJLqP8S29xQRUulSlzgbDn2pJtVYi-2s8BD8M51ugUkDnDxWDPfj2Y-hF5R8pYSIi9yfTvZEMabWqVsxBO0oYKLRmuhn6INkZI0nGp1ip7nvCOEMKXpM3TKpaZCcblBP29CSbZswdkJj2EHrowx4DjgGco2lgTfbQFsg8e-ftemzTEAHmLCB5vdMo1lzPgu2bBMcbYrtcrV-ZLKFh-g6o9ugnfYYmcz4AT7mMqDYqVCNV_S2j2M8O0FOhnslOHlYz1DX64-fL782Nx-ur65fH_bON7q0ljX2W7gnQLV0UHpVve-t16D5MCtA9f2XnXKcyVqy_fARB14opXyrJeCn6Hzo-4-xa8L5GLmMTuYJhsgLtlw2vKOspaz_0KZUm0rKOO6Qt_8Bd3VI4S6iGGaKVFPLlZvdkS5FHNOMJh9GmebfhhKzJqrOeZqaq7mIVezkl4_Si_9DP435VeQFcCPgFxH4Q7SH-9_yN4DbVWw3w</recordid><startdate>20240301</startdate><enddate>20240301</enddate><creator>Li, Chunyan</creator><creator>Wang, Yu</creator><creator>Lin, Yupei</creator><creator>Gong, Qianla</creator><creator>Wu, Bangcui</creator><creator>Zheng, Wendan</creator><creator>Tian, Yingying</creator><creator>Chen, Yong</creator><creator>Tian, Mei</creator><general>Springer International Publishing</general><general>Springer Nature B.V</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>7T5</scope><scope>H94</scope><scope>K9.</scope><scope>7X8</scope><scope>7S9</scope><scope>L.6</scope><orcidid>https://orcid.org/0009-0006-1531-643X</orcidid><orcidid>https://orcid.org/0000-0001-5025-041X</orcidid><orcidid>https://orcid.org/0009-0007-1471-4150</orcidid><orcidid>https://orcid.org/0000-0001-5538-5081</orcidid><orcidid>https://orcid.org/0000-0002-1633-2988</orcidid><orcidid>https://orcid.org/0000-0003-3746-6958</orcidid><orcidid>https://orcid.org/0009-0003-7287-898X</orcidid><orcidid>https://orcid.org/0000-0003-0510-782X</orcidid><orcidid>https://orcid.org/0009-0002-8062-9652</orcidid></search><sort><creationdate>20240301</creationdate><title>Intrathecal injection of methotrexate and dexamethasone for vasculitis granuloma of the fourth ventricle: a case report and literature review</title><author>Li, Chunyan ; Wang, Yu ; Lin, Yupei ; Gong, Qianla ; Wu, Bangcui ; Zheng, Wendan ; Tian, Yingying ; Chen, Yong ; Tian, Mei</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c359t-ac6a6f368e861f8959bdbad9e73e3acec5bd868d384e73dbe243e3d0988d2b743</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2024</creationdate><topic>Anti-Neutrophil Cytoplasmic Antibody-Associated Vasculitis - complications</topic><topic>Case Based Review</topic><topic>Case reports</topic><topic>case studies</topic><topic>Central nervous system</topic><topic>Cyclophosphamide</topic><topic>Dexamethasone</topic><topic>Dexamethasone - therapeutic use</topic><topic>Fourth Ventricle</topic><topic>glucocorticoids</topic><topic>Granuloma</topic><topic>Granuloma - complications</topic><topic>Granuloma - drug therapy</topic><topic>Granulomas</topic><topic>Granulomatosis</topic><topic>Granulomatosis with Polyangiitis - complications</topic><topic>Humans</topic><topic>Hydrocephalus</topic><topic>Immunosuppressive agents</topic><topic>Immunosuppressive Agents - therapeutic use</topic><topic>Injection</topic><topic>Injections, Spinal</topic><topic>intrathecal injection</topic><topic>Leukocytes (neutrophilic)</topic><topic>Literature reviews</topic><topic>Medicine</topic><topic>Medicine & Public Health</topic><topic>Methotrexate</topic><topic>Methotrexate - therapeutic use</topic><topic>Mycophenolate mofetil</topic><topic>Mycophenolic acid</topic><topic>Mycophenolic Acid - therapeutic use</topic><topic>patients</topic><topic>Rheumatology</topic><topic>Rituximab</topic><topic>Rituximab - therapeutic use</topic><topic>Vasculitis</topic><topic>Ventricle</topic><topic>Ventricles (cerebral)</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Li, Chunyan</creatorcontrib><creatorcontrib>Wang, Yu</creatorcontrib><creatorcontrib>Lin, Yupei</creatorcontrib><creatorcontrib>Gong, Qianla</creatorcontrib><creatorcontrib>Wu, Bangcui</creatorcontrib><creatorcontrib>Zheng, Wendan</creatorcontrib><creatorcontrib>Tian, Yingying</creatorcontrib><creatorcontrib>Chen, Yong</creatorcontrib><creatorcontrib>Tian, Mei</creatorcontrib><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>ProQuest Health & Medical Complete (Alumni)</collection><collection>MEDLINE - Academic</collection><collection>AGRICOLA</collection><collection>AGRICOLA - Academic</collection><jtitle>Clinical rheumatology</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Li, Chunyan</au><au>Wang, Yu</au><au>Lin, Yupei</au><au>Gong, Qianla</au><au>Wu, Bangcui</au><au>Zheng, Wendan</au><au>Tian, Yingying</au><au>Chen, Yong</au><au>Tian, Mei</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Intrathecal injection of methotrexate and dexamethasone for vasculitis granuloma of the fourth ventricle: a case report and literature review</atitle><jtitle>Clinical rheumatology</jtitle><stitle>Clin Rheumatol</stitle><addtitle>Clin Rheumatol</addtitle><date>2024-03-01</date><risdate>2024</risdate><volume>43</volume><issue>3</issue><spage>1217</spage><epage>1226</epage><pages>1217-1226</pages><issn>0770-3198</issn><eissn>1434-9949</eissn><abstract>Granulomatosis with polyangiitis (GPA) is a pauci-immune small vessel vasculitis characterised by neutrophil-mediated vasculitis and granuloma. The presence of intracranial parenchymal space-occupying lesions is rarely seen in GPA patients. In this manuscript, we report a case of GPA with granuloma of the fourth ventricle accompanied by obstructive hydrocephalus. Treatment with glucocorticoids (GCs) and multiple immunosuppressants cyclophosphamide (CYC), mycophenolate mofetil (MMF), and rituximab (RTX) showed poor efficacy in this case. After removal of the granuloma by craniotomy, GPA relapsed within 3 months. Under the premise of GC and MMF treatment combined with intrathecal injection of dexamethasone (DXM) and methotrexate (MTX), the intracranial granuloma gradually shrank, and the patient’s general condition was alleviated, showing that this is an effective treatment method.
Key Points
• To date, there are few reports of granulomatous vasculitis combined with granuloma of the fourth ventricle, and our case is the second.
• In this case, multiple immunosuppressants and rituximab were ineffective treatments, and the intracranial granuloma was effectively controlled by intrathecal injection of dexamethasone (DXM) and methotrexate (MTX).
• Based on this report, it can be suggested that intrathecal injection is effective in treating patients with GPA and central nervous system involvement, but large-scale sample studies are needed.</abstract><cop>Cham</cop><pub>Springer International Publishing</pub><pmid>37914837</pmid><doi>10.1007/s10067-023-06777-4</doi><tpages>10</tpages><orcidid>https://orcid.org/0009-0006-1531-643X</orcidid><orcidid>https://orcid.org/0000-0001-5025-041X</orcidid><orcidid>https://orcid.org/0009-0007-1471-4150</orcidid><orcidid>https://orcid.org/0000-0001-5538-5081</orcidid><orcidid>https://orcid.org/0000-0002-1633-2988</orcidid><orcidid>https://orcid.org/0000-0003-3746-6958</orcidid><orcidid>https://orcid.org/0009-0003-7287-898X</orcidid><orcidid>https://orcid.org/0000-0003-0510-782X</orcidid><orcidid>https://orcid.org/0009-0002-8062-9652</orcidid></addata></record> |
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| subjects | Anti-Neutrophil Cytoplasmic Antibody-Associated Vasculitis - complications Case Based Review Case reports case studies Central nervous system Cyclophosphamide Dexamethasone Dexamethasone - therapeutic use Fourth Ventricle glucocorticoids Granuloma Granuloma - complications Granuloma - drug therapy Granulomas Granulomatosis Granulomatosis with Polyangiitis - complications Humans Hydrocephalus Immunosuppressive agents Immunosuppressive Agents - therapeutic use Injection Injections, Spinal intrathecal injection Leukocytes (neutrophilic) Literature reviews Medicine Medicine & Public Health Methotrexate Methotrexate - therapeutic use Mycophenolate mofetil Mycophenolic acid Mycophenolic Acid - therapeutic use patients Rheumatology Rituximab Rituximab - therapeutic use Vasculitis Ventricle Ventricles (cerebral) |
| title | Intrathecal injection of methotrexate and dexamethasone for vasculitis granuloma of the fourth ventricle: a case report and literature review |
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