Re-examining the characteristics of pediatric multiple sclerosis in the era of antibody-associated demyelinating syndromes
The discovery of anti-myelin oligodendrocyte glycoprotein (MOG)-IgG and anti-aquaporin 4 (AQP4)-IgG and the observation on certain patients previously diagnosed with multiple sclerosis (MS) actually have an antibody-mediated disease mandated re-evaluation of pediatric MS series. To describe the char...
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| Veröffentlicht in: | European journal of paediatric neurology 2022-11, Vol.41, p.8-18 |
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| creator | Yılmaz, Ünsal Gücüyener, Kıvılcım Yavuz, Merve Öncel, İbrahim Saltık, Sema Ünver, Olcay Yılmaz, Sanem Erol, İlknur Öztoprak, Ülkühan Elitez, Duygu Aykol Direk, Meltem Çobanoğulları Bodur, Muhittin Teber, Serap Yılmaz, Ünsal Anlar, Banu Gücüyener, Kıvılcım Ünver, Olcay Çıtak Kurt, Ayşegül Neşe Tosun, Ayşe Yılmaz, Sanem Özgör, Bilge Öztoprak, Ülkühan Aykol, Duygu Yıldız, Edibe Pembegül Yarar, Coşkun Haspolat, Şenay İncecik, Faruk Kutluk, Gültekin Dundar, Nihal Olgac Öncel, İbrahim Demir, Ercan Türkdoğan, Dilşad Yalnızoğlu, Dilek Akbaş, Salih Güleç, Ayten Yılmaz, Deniz Ayanoğlu, Müge Kanmaz, Seda Beşen, Şeyda Öztürk, Selcan Yüksel, Deniz Gürkaş, Esra Oktay, Seçil Serin, Hepsen Mine Karadağ, Meral Hakkı Akbeyaz, İsmail Yiş, Uluç Polat, Burçin Gönüllü Okan, Mehmet Sait Bektaş, Ömer Orgun, Leman Tekin Günbey, Ceren Gültutan, Pembe Aksoy, Erhan Akyüz, Gülcan Tekgül, Hasan Kürekçi, Fulya Kurul, A. Semra Hız Çarman, Kürşat Bora Duman, Özgür Kömür, Mustafa Yıldırım, Miraç Alıcı, Nurettin Gümüş, Hakan Polat, Muzaffer Konuşkan, Bahadır Güngör, Olcay Mert, Gülen Gül Edizer, Selvinaz Mıhçı, Filiz Yavuz, Merve Toker, Rabia Tütüncü Şahin, Sevim Gencpinar, Pinar Yıldırım, Elif Yüksel, Ersin Ekici, Arzu Deniz, Adnan Yayici Köken, Özlem Okuyaz, Çetin Süt, Nurşah Yeniay Atasoy, Ergin Yetkin, Mehmet Fatih Bilgin, Neslihan Tekin, Hande Gazeteci Özçelik, Aysima Türköz, Ayşe Nur Cavusoglu, Dilek Özkan, Mehbare Tekin, Emine Koç, Habibe Sarıgeçili, Esra Sarıtaş, Serdar Ayça, Senem Şenoğlu, Mine Çiğdem Kamaşak, Tülay Keskin, Filiz Karaoğlu, Pakize İpek, Rojan Acer, Hamit |
| description | The discovery of anti-myelin oligodendrocyte glycoprotein (MOG)-IgG and anti-aquaporin 4 (AQP4)-IgG and the observation on certain patients previously diagnosed with multiple sclerosis (MS) actually have an antibody-mediated disease mandated re-evaluation of pediatric MS series.
To describe the characteristics of recent pediatric MS cases by age groups and compare with the cohort established before 2015.
Data of pediatric MS patients diagnosed between 2015 and 2021 were collected from 44 pediatric neurology centers across Türkiye. Clinical and paraclinical features were compared between patients with disease onset before 12 years (earlier onset) and ≥12 years (later onset) as well as between our current (2015–2021) and previous ( |
| doi_str_mv | 10.1016/j.ejpn.2022.08.006 |
| format | Article |
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Semra Hız ; Çarman, Kürşat Bora ; Duman, Özgür ; Kömür, Mustafa ; Yıldırım, Miraç ; Alıcı, Nurettin ; Gümüş, Hakan ; Polat, Muzaffer ; Konuşkan, Bahadır ; Güngör, Olcay ; Mert, Gülen Gül ; Edizer, Selvinaz ; Mıhçı, Filiz ; Yavuz, Merve ; Toker, Rabia Tütüncü ; Şahin, Sevim ; Gencpinar, Pinar ; Yıldırım, Elif ; Yüksel, Ersin ; Ekici, Arzu ; Deniz, Adnan ; Yayici Köken, Özlem ; Okuyaz, Çetin ; Süt, Nurşah Yeniay ; Atasoy, Ergin ; Yetkin, Mehmet Fatih ; Bilgin, Neslihan ; Tekin, Hande Gazeteci ; Özçelik, Aysima ; Türköz, Ayşe Nur ; Cavusoglu, Dilek ; Özkan, Mehbare ; Tekin, Emine ; Koç, Habibe ; Sarıgeçili, Esra ; Sarıtaş, Serdar ; Ayça, Senem ; Şenoğlu, Mine Çiğdem ; Kamaşak, Tülay ; Keskin, Filiz ; Karaoğlu, Pakize ; İpek, Rojan ; Acer, Hamit</creator><creatorcontrib>Yılmaz, Ünsal ; Gücüyener, Kıvılcım ; Yavuz, Merve ; Öncel, İbrahim ; Saltık, Sema ; Ünver, Olcay ; Yılmaz, Sanem ; Erol, İlknur ; Öztoprak, Ülkühan ; Elitez, Duygu Aykol ; Direk, Meltem Çobanoğulları ; Bodur, Muhittin ; Teber, Serap ; Yılmaz, Ünsal ; Anlar, Banu ; Gücüyener, Kıvılcım ; Ünver, Olcay ; Çıtak Kurt, Ayşegül Neşe ; Tosun, Ayşe ; Yılmaz, Sanem ; Özgör, Bilge ; Öztoprak, Ülkühan ; Aykol, Duygu ; Yıldız, Edibe Pembegül ; Yarar, Coşkun ; Haspolat, Şenay ; İncecik, Faruk ; Kutluk, Gültekin ; Dundar, Nihal Olgac ; Öncel, İbrahim ; Demir, Ercan ; Türkdoğan, Dilşad ; Yalnızoğlu, Dilek ; Akbaş, Salih ; Güleç, Ayten ; Yılmaz, Deniz ; Ayanoğlu, Müge ; Kanmaz, Seda ; Beşen, Şeyda ; Öztürk, Selcan ; Yüksel, Deniz ; Gürkaş, Esra ; Oktay, Seçil ; Serin, Hepsen Mine ; Karadağ, Meral ; Hakkı Akbeyaz, İsmail ; Yiş, Uluç ; Polat, Burçin Gönüllü ; Okan, Mehmet Sait ; Bektaş, Ömer ; Orgun, Leman Tekin ; Günbey, Ceren ; Gültutan, Pembe ; Aksoy, Erhan ; Akyüz, Gülcan ; Tekgül, Hasan ; Kürekçi, Fulya ; Kurul, A. Semra Hız ; Çarman, Kürşat Bora ; Duman, Özgür ; Kömür, Mustafa ; Yıldırım, Miraç ; Alıcı, Nurettin ; Gümüş, Hakan ; Polat, Muzaffer ; Konuşkan, Bahadır ; Güngör, Olcay ; Mert, Gülen Gül ; Edizer, Selvinaz ; Mıhçı, Filiz ; Yavuz, Merve ; Toker, Rabia Tütüncü ; Şahin, Sevim ; Gencpinar, Pinar ; Yıldırım, Elif ; Yüksel, Ersin ; Ekici, Arzu ; Deniz, Adnan ; Yayici Köken, Özlem ; Okuyaz, Çetin ; Süt, Nurşah Yeniay ; Atasoy, Ergin ; Yetkin, Mehmet Fatih ; Bilgin, Neslihan ; Tekin, Hande Gazeteci ; Özçelik, Aysima ; Türköz, Ayşe Nur ; Cavusoglu, Dilek ; Özkan, Mehbare ; Tekin, Emine ; Koç, Habibe ; Sarıgeçili, Esra ; Sarıtaş, Serdar ; Ayça, Senem ; Şenoğlu, Mine Çiğdem ; Kamaşak, Tülay ; Keskin, Filiz ; Karaoğlu, Pakize ; İpek, Rojan ; Acer, Hamit ; Turkish Pediatric Multiple Sclerosis Study Group</creatorcontrib><description>The discovery of anti-myelin oligodendrocyte glycoprotein (MOG)-IgG and anti-aquaporin 4 (AQP4)-IgG and the observation on certain patients previously diagnosed with multiple sclerosis (MS) actually have an antibody-mediated disease mandated re-evaluation of pediatric MS series.
To describe the characteristics of recent pediatric MS cases by age groups and compare with the cohort established before 2015.
Data of pediatric MS patients diagnosed between 2015 and 2021 were collected from 44 pediatric neurology centers across Türkiye. Clinical and paraclinical features were compared between patients with disease onset before 12 years (earlier onset) and ≥12 years (later onset) as well as between our current (2015–2021) and previous (<2015) cohorts.
A total of 634 children (456 girls) were enrolled, 89 (14%) were of earlier onset. The earlier-onset group had lower female/male ratio, more frequent initial diagnosis of acute disseminated encephalomyelitis (ADEM), more frequent brainstem symptoms, longer interval between the first two attacks, less frequent spinal cord involvement on magnetic resonance imaging (MRI), and lower prevalence of cerebrospinal fluid (CSF)-restricted oligoclonal bands (OCBs). The earlier-onset group was less likely to respond to initial disease-modifying treatments. Compared to our previous cohort, the current series had fewer patients with onset <12 years, initial presentation with ADEM-like features, brainstem or cerebellar symptoms, seizures, and spinal lesions on MRI. The female/male ratio, the frequency of sensorial symptoms, and CSF-restricted OCBs were higher than reported in our previous cohort.
Pediatric MS starting before 12 years was less common than reported previously, likely due to exclusion of patients with antibody-mediated diseases. The results underline the importance of antibody testing and indicate pediatric MS may be a more homogeneous disorder and more similar to adult-onset MS than previously thought.
•The incidence of pediatric MS is estimated as 0.68 per 100,000 children in Turkey.•Careful exclusion of MOG-IgG disease and other MS mimics allowed accurate description of pediatric MS in this series.•No cases of primary progressive MS were recorded among the 634 pediatric MS patients.•Onset before 12 years and ADEM-like presentation were less frequent than previously reported.•Pediatric MS appears more homogeneous and more similar to adult-onset MS than previously thought.</description><identifier>ISSN: 1090-3798</identifier><identifier>ISSN: 1532-2130</identifier><identifier>EISSN: 1532-2130</identifier><identifier>DOI: 10.1016/j.ejpn.2022.08.006</identifier><identifier>PMID: 36137476</identifier><language>eng</language><publisher>England: Elsevier Ltd</publisher><subject>Anti-AQP4-IgG ; Anti-MOG-IgG ; Antibody ; Autoantibodies ; Demyelinating ; Encephalomyelitis, Acute Disseminated ; Female ; Humans ; Immunoglobulin G ; Magnetic Resonance Imaging ; Male ; Multiple Sclerosis - diagnostic imaging ; Myelin-Oligodendrocyte Glycoprotein ; Neuromyelitis Optica ; Pediatric multiple sclerosis</subject><ispartof>European journal of paediatric neurology, 2022-11, Vol.41, p.8-18</ispartof><rights>2022 European Paediatric Neurology Society</rights><rights>2022 European Paediatric Neurology Society. Published by Elsevier Ltd. All rights reserved.</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c356t-205a3a7140ef34f7d8d4211e3ec6ae94bc3e344c474bbcc88c58df57ef3b933a3</citedby><cites>FETCH-LOGICAL-c356t-205a3a7140ef34f7d8d4211e3ec6ae94bc3e344c474bbcc88c58df57ef3b933a3</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktohtml>$$Uhttps://www.sciencedirect.com/science/article/pii/S1090379822001246$$EHTML$$P50$$Gelsevier$$H</linktohtml><link.rule.ids>314,776,780,3537,27901,27902,65306</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/36137476$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Yılmaz, Ünsal</creatorcontrib><creatorcontrib>Gücüyener, Kıvılcım</creatorcontrib><creatorcontrib>Yavuz, Merve</creatorcontrib><creatorcontrib>Öncel, İbrahim</creatorcontrib><creatorcontrib>Saltık, Sema</creatorcontrib><creatorcontrib>Ünver, Olcay</creatorcontrib><creatorcontrib>Yılmaz, Sanem</creatorcontrib><creatorcontrib>Erol, İlknur</creatorcontrib><creatorcontrib>Öztoprak, Ülkühan</creatorcontrib><creatorcontrib>Elitez, Duygu Aykol</creatorcontrib><creatorcontrib>Direk, Meltem Çobanoğulları</creatorcontrib><creatorcontrib>Bodur, Muhittin</creatorcontrib><creatorcontrib>Teber, Serap</creatorcontrib><creatorcontrib>Yılmaz, Ünsal</creatorcontrib><creatorcontrib>Anlar, Banu</creatorcontrib><creatorcontrib>Gücüyener, Kıvılcım</creatorcontrib><creatorcontrib>Ünver, Olcay</creatorcontrib><creatorcontrib>Çıtak Kurt, Ayşegül Neşe</creatorcontrib><creatorcontrib>Tosun, Ayşe</creatorcontrib><creatorcontrib>Yılmaz, Sanem</creatorcontrib><creatorcontrib>Özgör, Bilge</creatorcontrib><creatorcontrib>Öztoprak, Ülkühan</creatorcontrib><creatorcontrib>Aykol, Duygu</creatorcontrib><creatorcontrib>Yıldız, Edibe Pembegül</creatorcontrib><creatorcontrib>Yarar, Coşkun</creatorcontrib><creatorcontrib>Haspolat, Şenay</creatorcontrib><creatorcontrib>İncecik, Faruk</creatorcontrib><creatorcontrib>Kutluk, Gültekin</creatorcontrib><creatorcontrib>Dundar, Nihal Olgac</creatorcontrib><creatorcontrib>Öncel, İbrahim</creatorcontrib><creatorcontrib>Demir, Ercan</creatorcontrib><creatorcontrib>Türkdoğan, Dilşad</creatorcontrib><creatorcontrib>Yalnızoğlu, Dilek</creatorcontrib><creatorcontrib>Akbaş, Salih</creatorcontrib><creatorcontrib>Güleç, Ayten</creatorcontrib><creatorcontrib>Yılmaz, Deniz</creatorcontrib><creatorcontrib>Ayanoğlu, Müge</creatorcontrib><creatorcontrib>Kanmaz, Seda</creatorcontrib><creatorcontrib>Beşen, Şeyda</creatorcontrib><creatorcontrib>Öztürk, Selcan</creatorcontrib><creatorcontrib>Yüksel, Deniz</creatorcontrib><creatorcontrib>Gürkaş, Esra</creatorcontrib><creatorcontrib>Oktay, Seçil</creatorcontrib><creatorcontrib>Serin, Hepsen Mine</creatorcontrib><creatorcontrib>Karadağ, Meral</creatorcontrib><creatorcontrib>Hakkı Akbeyaz, İsmail</creatorcontrib><creatorcontrib>Yiş, Uluç</creatorcontrib><creatorcontrib>Polat, Burçin Gönüllü</creatorcontrib><creatorcontrib>Okan, Mehmet Sait</creatorcontrib><creatorcontrib>Bektaş, Ömer</creatorcontrib><creatorcontrib>Orgun, Leman Tekin</creatorcontrib><creatorcontrib>Günbey, Ceren</creatorcontrib><creatorcontrib>Gültutan, Pembe</creatorcontrib><creatorcontrib>Aksoy, Erhan</creatorcontrib><creatorcontrib>Akyüz, Gülcan</creatorcontrib><creatorcontrib>Tekgül, Hasan</creatorcontrib><creatorcontrib>Kürekçi, Fulya</creatorcontrib><creatorcontrib>Kurul, A. Semra Hız</creatorcontrib><creatorcontrib>Çarman, Kürşat Bora</creatorcontrib><creatorcontrib>Duman, Özgür</creatorcontrib><creatorcontrib>Kömür, Mustafa</creatorcontrib><creatorcontrib>Yıldırım, Miraç</creatorcontrib><creatorcontrib>Alıcı, Nurettin</creatorcontrib><creatorcontrib>Gümüş, Hakan</creatorcontrib><creatorcontrib>Polat, Muzaffer</creatorcontrib><creatorcontrib>Konuşkan, Bahadır</creatorcontrib><creatorcontrib>Güngör, Olcay</creatorcontrib><creatorcontrib>Mert, Gülen Gül</creatorcontrib><creatorcontrib>Edizer, Selvinaz</creatorcontrib><creatorcontrib>Mıhçı, Filiz</creatorcontrib><creatorcontrib>Yavuz, Merve</creatorcontrib><creatorcontrib>Toker, Rabia Tütüncü</creatorcontrib><creatorcontrib>Şahin, Sevim</creatorcontrib><creatorcontrib>Gencpinar, Pinar</creatorcontrib><creatorcontrib>Yıldırım, Elif</creatorcontrib><creatorcontrib>Yüksel, Ersin</creatorcontrib><creatorcontrib>Ekici, Arzu</creatorcontrib><creatorcontrib>Deniz, Adnan</creatorcontrib><creatorcontrib>Yayici Köken, Özlem</creatorcontrib><creatorcontrib>Okuyaz, Çetin</creatorcontrib><creatorcontrib>Süt, Nurşah Yeniay</creatorcontrib><creatorcontrib>Atasoy, Ergin</creatorcontrib><creatorcontrib>Yetkin, Mehmet Fatih</creatorcontrib><creatorcontrib>Bilgin, Neslihan</creatorcontrib><creatorcontrib>Tekin, Hande Gazeteci</creatorcontrib><creatorcontrib>Özçelik, Aysima</creatorcontrib><creatorcontrib>Türköz, Ayşe Nur</creatorcontrib><creatorcontrib>Cavusoglu, Dilek</creatorcontrib><creatorcontrib>Özkan, Mehbare</creatorcontrib><creatorcontrib>Tekin, Emine</creatorcontrib><creatorcontrib>Koç, Habibe</creatorcontrib><creatorcontrib>Sarıgeçili, Esra</creatorcontrib><creatorcontrib>Sarıtaş, Serdar</creatorcontrib><creatorcontrib>Ayça, Senem</creatorcontrib><creatorcontrib>Şenoğlu, Mine Çiğdem</creatorcontrib><creatorcontrib>Kamaşak, Tülay</creatorcontrib><creatorcontrib>Keskin, Filiz</creatorcontrib><creatorcontrib>Karaoğlu, Pakize</creatorcontrib><creatorcontrib>İpek, Rojan</creatorcontrib><creatorcontrib>Acer, Hamit</creatorcontrib><creatorcontrib>Turkish Pediatric Multiple Sclerosis Study Group</creatorcontrib><title>Re-examining the characteristics of pediatric multiple sclerosis in the era of antibody-associated demyelinating syndromes</title><title>European journal of paediatric neurology</title><addtitle>Eur J Paediatr Neurol</addtitle><description>The discovery of anti-myelin oligodendrocyte glycoprotein (MOG)-IgG and anti-aquaporin 4 (AQP4)-IgG and the observation on certain patients previously diagnosed with multiple sclerosis (MS) actually have an antibody-mediated disease mandated re-evaluation of pediatric MS series.
To describe the characteristics of recent pediatric MS cases by age groups and compare with the cohort established before 2015.
Data of pediatric MS patients diagnosed between 2015 and 2021 were collected from 44 pediatric neurology centers across Türkiye. Clinical and paraclinical features were compared between patients with disease onset before 12 years (earlier onset) and ≥12 years (later onset) as well as between our current (2015–2021) and previous (<2015) cohorts.
A total of 634 children (456 girls) were enrolled, 89 (14%) were of earlier onset. The earlier-onset group had lower female/male ratio, more frequent initial diagnosis of acute disseminated encephalomyelitis (ADEM), more frequent brainstem symptoms, longer interval between the first two attacks, less frequent spinal cord involvement on magnetic resonance imaging (MRI), and lower prevalence of cerebrospinal fluid (CSF)-restricted oligoclonal bands (OCBs). The earlier-onset group was less likely to respond to initial disease-modifying treatments. Compared to our previous cohort, the current series had fewer patients with onset <12 years, initial presentation with ADEM-like features, brainstem or cerebellar symptoms, seizures, and spinal lesions on MRI. The female/male ratio, the frequency of sensorial symptoms, and CSF-restricted OCBs were higher than reported in our previous cohort.
Pediatric MS starting before 12 years was less common than reported previously, likely due to exclusion of patients with antibody-mediated diseases. The results underline the importance of antibody testing and indicate pediatric MS may be a more homogeneous disorder and more similar to adult-onset MS than previously thought.
•The incidence of pediatric MS is estimated as 0.68 per 100,000 children in Turkey.•Careful exclusion of MOG-IgG disease and other MS mimics allowed accurate description of pediatric MS in this series.•No cases of primary progressive MS were recorded among the 634 pediatric MS patients.•Onset before 12 years and ADEM-like presentation were less frequent than previously reported.•Pediatric MS appears more homogeneous and more similar to adult-onset MS than previously thought.</description><subject>Anti-AQP4-IgG</subject><subject>Anti-MOG-IgG</subject><subject>Antibody</subject><subject>Autoantibodies</subject><subject>Demyelinating</subject><subject>Encephalomyelitis, Acute Disseminated</subject><subject>Female</subject><subject>Humans</subject><subject>Immunoglobulin G</subject><subject>Magnetic Resonance Imaging</subject><subject>Male</subject><subject>Multiple Sclerosis - diagnostic imaging</subject><subject>Myelin-Oligodendrocyte Glycoprotein</subject><subject>Neuromyelitis Optica</subject><subject>Pediatric multiple sclerosis</subject><issn>1090-3798</issn><issn>1532-2130</issn><issn>1532-2130</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2022</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNp9kEtv1TAQRq2qqC_6B1igLLtJ8CuxI3WDKh6VKiEhWFvOeNL6KnFS2xdx-fU43MKS1czifN9oDiFvGG0YZd27XYO7NTScct5Q3VDanZAL1gpecyboadlpT2uhen1OLlPaUUp7ybszci46JpRU3QX59RVr_GlnH3x4rPITVvBko4WM0afsIVXLWK3ovM3RQzXvp-zXCasEE8Yl-VT58CeG0W6oDdkPizvUNqUFSgpd5XA-4OSDzduNdAguLjOm1-TVaKeE1y_zinz_-OHb3ef64cun-7v3DzWItss1p60VVjFJcRRyVE47yRlDgdBZ7OUAAoWUIJUcBgCtodVubFWhh14IK67IzbF3jcvzHlM2s0-A02QDLvtkuGKq01y0uqD8iEL5LUUczRr9bOPBMGo252ZnNudmc26oNsV5Cb196d8PM7p_kb-SC3B7BLB8-cNjNAk8BihWI0I2bvH_6_8NTvSWJQ</recordid><startdate>202211</startdate><enddate>202211</enddate><creator>Yılmaz, Ünsal</creator><creator>Gücüyener, Kıvılcım</creator><creator>Yavuz, Merve</creator><creator>Öncel, İbrahim</creator><creator>Saltık, Sema</creator><creator>Ünver, Olcay</creator><creator>Yılmaz, Sanem</creator><creator>Erol, İlknur</creator><creator>Öztoprak, Ülkühan</creator><creator>Elitez, Duygu Aykol</creator><creator>Direk, Meltem Çobanoğulları</creator><creator>Bodur, Muhittin</creator><creator>Teber, Serap</creator><creator>Yılmaz, Ünsal</creator><creator>Anlar, Banu</creator><creator>Gücüyener, Kıvılcım</creator><creator>Ünver, Olcay</creator><creator>Çıtak Kurt, Ayşegül Neşe</creator><creator>Tosun, Ayşe</creator><creator>Yılmaz, Sanem</creator><creator>Özgör, Bilge</creator><creator>Öztoprak, Ülkühan</creator><creator>Aykol, Duygu</creator><creator>Yıldız, Edibe Pembegül</creator><creator>Yarar, Coşkun</creator><creator>Haspolat, Şenay</creator><creator>İncecik, Faruk</creator><creator>Kutluk, Gültekin</creator><creator>Dundar, Nihal Olgac</creator><creator>Öncel, İbrahim</creator><creator>Demir, Ercan</creator><creator>Türkdoğan, Dilşad</creator><creator>Yalnızoğlu, Dilek</creator><creator>Akbaş, Salih</creator><creator>Güleç, Ayten</creator><creator>Yılmaz, Deniz</creator><creator>Ayanoğlu, Müge</creator><creator>Kanmaz, Seda</creator><creator>Beşen, Şeyda</creator><creator>Öztürk, Selcan</creator><creator>Yüksel, Deniz</creator><creator>Gürkaş, Esra</creator><creator>Oktay, Seçil</creator><creator>Serin, Hepsen Mine</creator><creator>Karadağ, Meral</creator><creator>Hakkı Akbeyaz, İsmail</creator><creator>Yiş, Uluç</creator><creator>Polat, Burçin Gönüllü</creator><creator>Okan, Mehmet Sait</creator><creator>Bektaş, Ömer</creator><creator>Orgun, Leman Tekin</creator><creator>Günbey, Ceren</creator><creator>Gültutan, Pembe</creator><creator>Aksoy, Erhan</creator><creator>Akyüz, Gülcan</creator><creator>Tekgül, Hasan</creator><creator>Kürekçi, Fulya</creator><creator>Kurul, A. Semra Hız</creator><creator>Çarman, Kürşat Bora</creator><creator>Duman, Özgür</creator><creator>Kömür, Mustafa</creator><creator>Yıldırım, Miraç</creator><creator>Alıcı, Nurettin</creator><creator>Gümüş, Hakan</creator><creator>Polat, Muzaffer</creator><creator>Konuşkan, Bahadır</creator><creator>Güngör, Olcay</creator><creator>Mert, Gülen Gül</creator><creator>Edizer, Selvinaz</creator><creator>Mıhçı, Filiz</creator><creator>Yavuz, Merve</creator><creator>Toker, Rabia Tütüncü</creator><creator>Şahin, Sevim</creator><creator>Gencpinar, Pinar</creator><creator>Yıldırım, Elif</creator><creator>Yüksel, Ersin</creator><creator>Ekici, Arzu</creator><creator>Deniz, Adnan</creator><creator>Yayici Köken, Özlem</creator><creator>Okuyaz, Çetin</creator><creator>Süt, Nurşah Yeniay</creator><creator>Atasoy, Ergin</creator><creator>Yetkin, Mehmet Fatih</creator><creator>Bilgin, Neslihan</creator><creator>Tekin, Hande Gazeteci</creator><creator>Özçelik, Aysima</creator><creator>Türköz, Ayşe Nur</creator><creator>Cavusoglu, Dilek</creator><creator>Özkan, Mehbare</creator><creator>Tekin, Emine</creator><creator>Koç, Habibe</creator><creator>Sarıgeçili, Esra</creator><creator>Sarıtaş, Serdar</creator><creator>Ayça, Senem</creator><creator>Şenoğlu, Mine Çiğdem</creator><creator>Kamaşak, Tülay</creator><creator>Keskin, Filiz</creator><creator>Karaoğlu, Pakize</creator><creator>İpek, Rojan</creator><creator>Acer, Hamit</creator><general>Elsevier Ltd</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>202211</creationdate><title>Re-examining the characteristics of pediatric multiple sclerosis in the era of antibody-associated demyelinating syndromes</title><author>Yılmaz, Ünsal ; Gücüyener, Kıvılcım ; Yavuz, Merve ; Öncel, İbrahim ; Saltık, Sema ; Ünver, Olcay ; Yılmaz, Sanem ; Erol, İlknur ; Öztoprak, Ülkühan ; Elitez, Duygu Aykol ; Direk, Meltem Çobanoğulları ; Bodur, Muhittin ; Teber, Serap ; Yılmaz, Ünsal ; Anlar, Banu ; Gücüyener, Kıvılcım ; Ünver, Olcay ; Çıtak Kurt, Ayşegül Neşe ; Tosun, Ayşe ; Yılmaz, Sanem ; Özgör, Bilge ; Öztoprak, Ülkühan ; Aykol, Duygu ; Yıldız, Edibe Pembegül ; Yarar, Coşkun ; Haspolat, Şenay ; İncecik, Faruk ; Kutluk, Gültekin ; Dundar, Nihal Olgac ; Öncel, İbrahim ; Demir, Ercan ; Türkdoğan, Dilşad ; Yalnızoğlu, Dilek ; Akbaş, Salih ; Güleç, Ayten ; Yılmaz, Deniz ; Ayanoğlu, Müge ; Kanmaz, Seda ; Beşen, Şeyda ; Öztürk, Selcan ; Yüksel, Deniz ; Gürkaş, Esra ; Oktay, Seçil ; Serin, Hepsen Mine ; Karadağ, Meral ; Hakkı Akbeyaz, İsmail ; Yiş, Uluç ; Polat, Burçin Gönüllü ; Okan, Mehmet Sait ; Bektaş, Ömer ; Orgun, Leman Tekin ; Günbey, Ceren ; Gültutan, Pembe ; Aksoy, Erhan ; Akyüz, Gülcan ; Tekgül, Hasan ; Kürekçi, Fulya ; Kurul, A. Semra Hız ; Çarman, Kürşat Bora ; Duman, Özgür ; Kömür, Mustafa ; Yıldırım, Miraç ; Alıcı, Nurettin ; Gümüş, Hakan ; Polat, Muzaffer ; Konuşkan, Bahadır ; Güngör, Olcay ; Mert, Gülen Gül ; Edizer, Selvinaz ; Mıhçı, Filiz ; Yavuz, Merve ; Toker, Rabia Tütüncü ; Şahin, Sevim ; Gencpinar, Pinar ; Yıldırım, Elif ; Yüksel, Ersin ; Ekici, Arzu ; Deniz, Adnan ; Yayici Köken, Özlem ; Okuyaz, Çetin ; Süt, Nurşah Yeniay ; Atasoy, Ergin ; Yetkin, Mehmet Fatih ; Bilgin, Neslihan ; Tekin, Hande Gazeteci ; Özçelik, Aysima ; Türköz, Ayşe Nur ; Cavusoglu, Dilek ; Özkan, Mehbare ; Tekin, Emine ; Koç, Habibe ; Sarıgeçili, Esra ; Sarıtaş, Serdar ; Ayça, Senem ; Şenoğlu, Mine Çiğdem ; Kamaşak, Tülay ; Keskin, Filiz ; Karaoğlu, Pakize ; İpek, Rojan ; Acer, Hamit</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c356t-205a3a7140ef34f7d8d4211e3ec6ae94bc3e344c474bbcc88c58df57ef3b933a3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2022</creationdate><topic>Anti-AQP4-IgG</topic><topic>Anti-MOG-IgG</topic><topic>Antibody</topic><topic>Autoantibodies</topic><topic>Demyelinating</topic><topic>Encephalomyelitis, Acute Disseminated</topic><topic>Female</topic><topic>Humans</topic><topic>Immunoglobulin G</topic><topic>Magnetic Resonance Imaging</topic><topic>Male</topic><topic>Multiple Sclerosis - diagnostic imaging</topic><topic>Myelin-Oligodendrocyte Glycoprotein</topic><topic>Neuromyelitis Optica</topic><topic>Pediatric multiple sclerosis</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Yılmaz, Ünsal</creatorcontrib><creatorcontrib>Gücüyener, Kıvılcım</creatorcontrib><creatorcontrib>Yavuz, Merve</creatorcontrib><creatorcontrib>Öncel, İbrahim</creatorcontrib><creatorcontrib>Saltık, Sema</creatorcontrib><creatorcontrib>Ünver, Olcay</creatorcontrib><creatorcontrib>Yılmaz, Sanem</creatorcontrib><creatorcontrib>Erol, İlknur</creatorcontrib><creatorcontrib>Öztoprak, Ülkühan</creatorcontrib><creatorcontrib>Elitez, Duygu Aykol</creatorcontrib><creatorcontrib>Direk, Meltem Çobanoğulları</creatorcontrib><creatorcontrib>Bodur, Muhittin</creatorcontrib><creatorcontrib>Teber, Serap</creatorcontrib><creatorcontrib>Yılmaz, Ünsal</creatorcontrib><creatorcontrib>Anlar, Banu</creatorcontrib><creatorcontrib>Gücüyener, Kıvılcım</creatorcontrib><creatorcontrib>Ünver, Olcay</creatorcontrib><creatorcontrib>Çıtak Kurt, Ayşegül Neşe</creatorcontrib><creatorcontrib>Tosun, Ayşe</creatorcontrib><creatorcontrib>Yılmaz, Sanem</creatorcontrib><creatorcontrib>Özgör, Bilge</creatorcontrib><creatorcontrib>Öztoprak, Ülkühan</creatorcontrib><creatorcontrib>Aykol, Duygu</creatorcontrib><creatorcontrib>Yıldız, Edibe Pembegül</creatorcontrib><creatorcontrib>Yarar, Coşkun</creatorcontrib><creatorcontrib>Haspolat, Şenay</creatorcontrib><creatorcontrib>İncecik, Faruk</creatorcontrib><creatorcontrib>Kutluk, Gültekin</creatorcontrib><creatorcontrib>Dundar, Nihal Olgac</creatorcontrib><creatorcontrib>Öncel, İbrahim</creatorcontrib><creatorcontrib>Demir, Ercan</creatorcontrib><creatorcontrib>Türkdoğan, Dilşad</creatorcontrib><creatorcontrib>Yalnızoğlu, Dilek</creatorcontrib><creatorcontrib>Akbaş, Salih</creatorcontrib><creatorcontrib>Güleç, Ayten</creatorcontrib><creatorcontrib>Yılmaz, Deniz</creatorcontrib><creatorcontrib>Ayanoğlu, Müge</creatorcontrib><creatorcontrib>Kanmaz, Seda</creatorcontrib><creatorcontrib>Beşen, Şeyda</creatorcontrib><creatorcontrib>Öztürk, Selcan</creatorcontrib><creatorcontrib>Yüksel, Deniz</creatorcontrib><creatorcontrib>Gürkaş, Esra</creatorcontrib><creatorcontrib>Oktay, Seçil</creatorcontrib><creatorcontrib>Serin, Hepsen Mine</creatorcontrib><creatorcontrib>Karadağ, Meral</creatorcontrib><creatorcontrib>Hakkı Akbeyaz, İsmail</creatorcontrib><creatorcontrib>Yiş, Uluç</creatorcontrib><creatorcontrib>Polat, Burçin Gönüllü</creatorcontrib><creatorcontrib>Okan, Mehmet Sait</creatorcontrib><creatorcontrib>Bektaş, Ömer</creatorcontrib><creatorcontrib>Orgun, Leman Tekin</creatorcontrib><creatorcontrib>Günbey, Ceren</creatorcontrib><creatorcontrib>Gültutan, Pembe</creatorcontrib><creatorcontrib>Aksoy, Erhan</creatorcontrib><creatorcontrib>Akyüz, Gülcan</creatorcontrib><creatorcontrib>Tekgül, Hasan</creatorcontrib><creatorcontrib>Kürekçi, Fulya</creatorcontrib><creatorcontrib>Kurul, A. Semra Hız</creatorcontrib><creatorcontrib>Çarman, Kürşat Bora</creatorcontrib><creatorcontrib>Duman, Özgür</creatorcontrib><creatorcontrib>Kömür, Mustafa</creatorcontrib><creatorcontrib>Yıldırım, Miraç</creatorcontrib><creatorcontrib>Alıcı, Nurettin</creatorcontrib><creatorcontrib>Gümüş, Hakan</creatorcontrib><creatorcontrib>Polat, Muzaffer</creatorcontrib><creatorcontrib>Konuşkan, Bahadır</creatorcontrib><creatorcontrib>Güngör, Olcay</creatorcontrib><creatorcontrib>Mert, Gülen Gül</creatorcontrib><creatorcontrib>Edizer, Selvinaz</creatorcontrib><creatorcontrib>Mıhçı, Filiz</creatorcontrib><creatorcontrib>Yavuz, Merve</creatorcontrib><creatorcontrib>Toker, Rabia Tütüncü</creatorcontrib><creatorcontrib>Şahin, Sevim</creatorcontrib><creatorcontrib>Gencpinar, Pinar</creatorcontrib><creatorcontrib>Yıldırım, Elif</creatorcontrib><creatorcontrib>Yüksel, Ersin</creatorcontrib><creatorcontrib>Ekici, Arzu</creatorcontrib><creatorcontrib>Deniz, Adnan</creatorcontrib><creatorcontrib>Yayici Köken, Özlem</creatorcontrib><creatorcontrib>Okuyaz, Çetin</creatorcontrib><creatorcontrib>Süt, Nurşah Yeniay</creatorcontrib><creatorcontrib>Atasoy, Ergin</creatorcontrib><creatorcontrib>Yetkin, Mehmet Fatih</creatorcontrib><creatorcontrib>Bilgin, Neslihan</creatorcontrib><creatorcontrib>Tekin, Hande Gazeteci</creatorcontrib><creatorcontrib>Özçelik, Aysima</creatorcontrib><creatorcontrib>Türköz, Ayşe Nur</creatorcontrib><creatorcontrib>Cavusoglu, Dilek</creatorcontrib><creatorcontrib>Özkan, Mehbare</creatorcontrib><creatorcontrib>Tekin, Emine</creatorcontrib><creatorcontrib>Koç, Habibe</creatorcontrib><creatorcontrib>Sarıgeçili, Esra</creatorcontrib><creatorcontrib>Sarıtaş, Serdar</creatorcontrib><creatorcontrib>Ayça, Senem</creatorcontrib><creatorcontrib>Şenoğlu, Mine Çiğdem</creatorcontrib><creatorcontrib>Kamaşak, Tülay</creatorcontrib><creatorcontrib>Keskin, Filiz</creatorcontrib><creatorcontrib>Karaoğlu, Pakize</creatorcontrib><creatorcontrib>İpek, Rojan</creatorcontrib><creatorcontrib>Acer, Hamit</creatorcontrib><creatorcontrib>Turkish Pediatric Multiple Sclerosis Study Group</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>European journal of paediatric neurology</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Yılmaz, Ünsal</au><au>Gücüyener, Kıvılcım</au><au>Yavuz, Merve</au><au>Öncel, İbrahim</au><au>Saltık, Sema</au><au>Ünver, Olcay</au><au>Yılmaz, Sanem</au><au>Erol, İlknur</au><au>Öztoprak, Ülkühan</au><au>Elitez, Duygu Aykol</au><au>Direk, Meltem Çobanoğulları</au><au>Bodur, Muhittin</au><au>Teber, Serap</au><au>Yılmaz, Ünsal</au><au>Anlar, Banu</au><au>Gücüyener, Kıvılcım</au><au>Ünver, Olcay</au><au>Çıtak Kurt, Ayşegül Neşe</au><au>Tosun, Ayşe</au><au>Yılmaz, Sanem</au><au>Özgör, Bilge</au><au>Öztoprak, Ülkühan</au><au>Aykol, Duygu</au><au>Yıldız, Edibe Pembegül</au><au>Yarar, Coşkun</au><au>Haspolat, Şenay</au><au>İncecik, Faruk</au><au>Kutluk, Gültekin</au><au>Dundar, Nihal Olgac</au><au>Öncel, İbrahim</au><au>Demir, Ercan</au><au>Türkdoğan, Dilşad</au><au>Yalnızoğlu, Dilek</au><au>Akbaş, Salih</au><au>Güleç, Ayten</au><au>Yılmaz, Deniz</au><au>Ayanoğlu, Müge</au><au>Kanmaz, Seda</au><au>Beşen, Şeyda</au><au>Öztürk, Selcan</au><au>Yüksel, Deniz</au><au>Gürkaş, Esra</au><au>Oktay, Seçil</au><au>Serin, Hepsen Mine</au><au>Karadağ, Meral</au><au>Hakkı Akbeyaz, İsmail</au><au>Yiş, Uluç</au><au>Polat, Burçin Gönüllü</au><au>Okan, Mehmet Sait</au><au>Bektaş, Ömer</au><au>Orgun, Leman Tekin</au><au>Günbey, Ceren</au><au>Gültutan, Pembe</au><au>Aksoy, Erhan</au><au>Akyüz, Gülcan</au><au>Tekgül, Hasan</au><au>Kürekçi, Fulya</au><au>Kurul, A. Semra Hız</au><au>Çarman, Kürşat Bora</au><au>Duman, Özgür</au><au>Kömür, Mustafa</au><au>Yıldırım, Miraç</au><au>Alıcı, Nurettin</au><au>Gümüş, Hakan</au><au>Polat, Muzaffer</au><au>Konuşkan, Bahadır</au><au>Güngör, Olcay</au><au>Mert, Gülen Gül</au><au>Edizer, Selvinaz</au><au>Mıhçı, Filiz</au><au>Yavuz, Merve</au><au>Toker, Rabia Tütüncü</au><au>Şahin, Sevim</au><au>Gencpinar, Pinar</au><au>Yıldırım, Elif</au><au>Yüksel, Ersin</au><au>Ekici, Arzu</au><au>Deniz, Adnan</au><au>Yayici Köken, Özlem</au><au>Okuyaz, Çetin</au><au>Süt, Nurşah Yeniay</au><au>Atasoy, Ergin</au><au>Yetkin, Mehmet Fatih</au><au>Bilgin, Neslihan</au><au>Tekin, Hande Gazeteci</au><au>Özçelik, Aysima</au><au>Türköz, Ayşe Nur</au><au>Cavusoglu, Dilek</au><au>Özkan, Mehbare</au><au>Tekin, Emine</au><au>Koç, Habibe</au><au>Sarıgeçili, Esra</au><au>Sarıtaş, Serdar</au><au>Ayça, Senem</au><au>Şenoğlu, Mine Çiğdem</au><au>Kamaşak, Tülay</au><au>Keskin, Filiz</au><au>Karaoğlu, Pakize</au><au>İpek, Rojan</au><au>Acer, Hamit</au><aucorp>Turkish Pediatric Multiple Sclerosis Study Group</aucorp><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Re-examining the characteristics of pediatric multiple sclerosis in the era of antibody-associated demyelinating syndromes</atitle><jtitle>European journal of paediatric neurology</jtitle><addtitle>Eur J Paediatr Neurol</addtitle><date>2022-11</date><risdate>2022</risdate><volume>41</volume><spage>8</spage><epage>18</epage><pages>8-18</pages><issn>1090-3798</issn><issn>1532-2130</issn><eissn>1532-2130</eissn><abstract>The discovery of anti-myelin oligodendrocyte glycoprotein (MOG)-IgG and anti-aquaporin 4 (AQP4)-IgG and the observation on certain patients previously diagnosed with multiple sclerosis (MS) actually have an antibody-mediated disease mandated re-evaluation of pediatric MS series.
To describe the characteristics of recent pediatric MS cases by age groups and compare with the cohort established before 2015.
Data of pediatric MS patients diagnosed between 2015 and 2021 were collected from 44 pediatric neurology centers across Türkiye. Clinical and paraclinical features were compared between patients with disease onset before 12 years (earlier onset) and ≥12 years (later onset) as well as between our current (2015–2021) and previous (<2015) cohorts.
A total of 634 children (456 girls) were enrolled, 89 (14%) were of earlier onset. The earlier-onset group had lower female/male ratio, more frequent initial diagnosis of acute disseminated encephalomyelitis (ADEM), more frequent brainstem symptoms, longer interval between the first two attacks, less frequent spinal cord involvement on magnetic resonance imaging (MRI), and lower prevalence of cerebrospinal fluid (CSF)-restricted oligoclonal bands (OCBs). The earlier-onset group was less likely to respond to initial disease-modifying treatments. Compared to our previous cohort, the current series had fewer patients with onset <12 years, initial presentation with ADEM-like features, brainstem or cerebellar symptoms, seizures, and spinal lesions on MRI. The female/male ratio, the frequency of sensorial symptoms, and CSF-restricted OCBs were higher than reported in our previous cohort.
Pediatric MS starting before 12 years was less common than reported previously, likely due to exclusion of patients with antibody-mediated diseases. The results underline the importance of antibody testing and indicate pediatric MS may be a more homogeneous disorder and more similar to adult-onset MS than previously thought.
•The incidence of pediatric MS is estimated as 0.68 per 100,000 children in Turkey.•Careful exclusion of MOG-IgG disease and other MS mimics allowed accurate description of pediatric MS in this series.•No cases of primary progressive MS were recorded among the 634 pediatric MS patients.•Onset before 12 years and ADEM-like presentation were less frequent than previously reported.•Pediatric MS appears more homogeneous and more similar to adult-onset MS than previously thought.</abstract><cop>England</cop><pub>Elsevier Ltd</pub><pmid>36137476</pmid><doi>10.1016/j.ejpn.2022.08.006</doi><tpages>11</tpages></addata></record> |
| fulltext | fulltext |
| identifier | ISSN: 1090-3798 |
| ispartof | European journal of paediatric neurology, 2022-11, Vol.41, p.8-18 |
| issn | 1090-3798 1532-2130 1532-2130 |
| language | eng |
| recordid | cdi_proquest_miscellaneous_2717682358 |
| source | MEDLINE; Elsevier ScienceDirect Journals |
| subjects | Anti-AQP4-IgG Anti-MOG-IgG Antibody Autoantibodies Demyelinating Encephalomyelitis, Acute Disseminated Female Humans Immunoglobulin G Magnetic Resonance Imaging Male Multiple Sclerosis - diagnostic imaging Myelin-Oligodendrocyte Glycoprotein Neuromyelitis Optica Pediatric multiple sclerosis |
| title | Re-examining the characteristics of pediatric multiple sclerosis in the era of antibody-associated demyelinating syndromes |
| url | https://sfx.bib-bvb.de/sfx_tum?ctx_ver=Z39.88-2004&ctx_enc=info:ofi/enc:UTF-8&ctx_tim=2025-02-08T16%3A40%3A57IST&url_ver=Z39.88-2004&url_ctx_fmt=infofi/fmt:kev:mtx:ctx&rfr_id=info:sid/primo.exlibrisgroup.com:primo3-Article-proquest_cross&rft_val_fmt=info:ofi/fmt:kev:mtx:journal&rft.genre=article&rft.atitle=Re-examining%20the%20characteristics%20of%20pediatric%20multiple%20sclerosis%20in%20the%20era%20of%20antibody-associated%20demyelinating%20syndromes&rft.jtitle=European%20journal%20of%20paediatric%20neurology&rft.au=Y%C4%B1lmaz,%20%C3%9Cnsal&rft.aucorp=Turkish%20Pediatric%20Multiple%20Sclerosis%20Study%20Group&rft.date=2022-11&rft.volume=41&rft.spage=8&rft.epage=18&rft.pages=8-18&rft.issn=1090-3798&rft.eissn=1532-2130&rft_id=info:doi/10.1016/j.ejpn.2022.08.006&rft_dat=%3Cproquest_cross%3E2717682358%3C/proquest_cross%3E%3Curl%3E%3C/url%3E&disable_directlink=true&sfx.directlink=off&sfx.report_link=0&rft_id=info:oai/&rft_pqid=2717682358&rft_id=info:pmid/36137476&rft_els_id=S1090379822001246&rfr_iscdi=true |