Browsing by Author "Dropol, Anastasia"

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    Health-related quality of life in children with inflammatory brain disease
    (2018-11-20) Liu, Elina; Twilt, Marinka; Tyrrell, Pascal N; Dropol, Anastasia; Sheikh, Shehla; Gorman, Mark; Kim, Susan; Cabral, David A; Forsyth, Rob; Van Mater, Heather; Li, Suzanne; Huber, Adam M; Stringer, Elizabeth; Muscal, Eyal; Wahezi, Dawn; Toth, Mary; Dolezalova, Pavla; Kobrova, Katerina; Ristic, Goran; Benseler, Susanne M
    Abstract Objective To quantify the impact of inflammatory brain diseases in the pediatric population on health-related quality of life, including the subdomains of physical, emotional, school and social functioning. Methods This was a multicenter, observational cohort study of children (< 18 years of age) diagnosed with inflammatory brain disease (IBrainD). Patients were included if they had completed at least one Health Related Quality of Life Questionnaire (HRQoL). HRQoL was measured using the Pediatric Quality of Life Inventory Version 4.0 (PedsQL) Generic Core Scales, which provided a total score out of 100. Analyses of trends were performed using linear regression models adjusted for repeated measures over time. Results In this study, 145 patients were included of which 80 (55%) were females. Cognitive dysfunction was the most common presenting symptoms (63%), and small vessel childhood primary angiitis of the CNS was the most common diagnosis (33%). The mean child’s self-reported PedsQL total score at diagnosis was 68.4, and the mean parent’s proxy-reported PedsQL score was 63.4 at diagnosis. Child’s self-reported PedsQL scores reflected poor HRQoL in 52.9% of patients at diagnosis. Seizures or cognitive dysfunction at presentation was associated with statistically significant deficits in HRQoL. Conclusion Pediatric IBrainD is associated with significantly diminished health-related quality of life. Future research should elucidate why these deficits occur and interventions should focus on improving HRQoL in the most affected subdomains, in particular for children presenting with seizures and cognitive dysfunction.
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    ItemOpen Access
    Proceedings of the 23rd Paediatric Rheumatology European Society Congress: part two
    (2017-05-30) Lomakina, Olga; Alekseeva, Ekaterina; Valieva, Sania; Bzarova, Tatiana; Nikishina, Irina; Zholobova, Elena; Rodionovskaya, Svetlana; Kaleda, Maria; Nakagishi, Yasuo; Shimizu, Masaki; Mizuta, Mao; Yachie, Akihiro; Sugita, Yuko; Okamoto, Nami; Shabana, Kousuke; Murata, Takuji; Tamai, Hiroshi; Smith, Eve M; Yin, Peng; Jorgensen, Andrea L; Beresford, Michael W; Smith, Eve M; Eleuteri, Antonio; Goilav, Beatrice; Lewandowski, Laura; Phuti, Angel; Wahezi, Dawn; Rubinstein, Tamar; Jones, Caroline; Newland, Paul; Marks, Stephen; Corkhill, Rachel; Ekdawy, Diana; Pilkington, Clarissa; Tullus, Kjell; Putterman, Chaim; Scott, Chris; Fisher, Antony C; Beresford, Michael W; Smith, Eve M; Lewandowski, Laura; Phuti, Angel; Jorgensen, Andrea; Scott, Chris; Beresford, Michael W; Batu, Ezgi D; Kosukcu, Can; Taskiran, Ekim; Akman, Sema; Ozturk, Kubra; Sozeri, Betul; Unsal, Erbil; Ekinci, Zelal; Bilginer, Yelda; Alikasifoglu, Mehmet; Ozen, Seza; Lythgoe, Hanna; Beresford, Michael W; Brunner, Hermine I; Gulati, Gaurav; Jones, Jordan T; Altaye, Mekibib; Eaton, Jamie; Difrancesco, Mark; Yeo, Joo G; Leong, Jingyao; Bathi, Loshinidevi D T; Arkachaisri, Thaschawee; Albani, Salvatore; Abdelrahman, Nagla; Beresford, Michael W; Leone, Valentina; Groot, Noortje; Shaikhani, D.; Bultink, I. E M; Bijl, M.; Dolhain, R. J E M; Teng, Y. K O; Zirkzee, E.; de Leeuw, K.; Fritsch-Stork, R.; Kamphuis, S. S M; Wright, Rachael D; Smith, Eve M; Beresford, Michael W; Abdawani, Reem; Al Shaqshi, Laila; Al Zakwani, Ibrahim; Gormezano, Natali W; Kern, David; Pereira, Oriany L; Esteves, Gladys C C; Sallum, Adriana M; Aikawa, Nadia E; Pereira, Rosa M; Silva, Clovis A; Bonfa, Eloisa; Beckmann, Jessica; Bartholomä, Nora; Venhoff, Nils; Henneke, Philipp; Salzer, Ulrich; Janda, Ales; Boteanu, Alina L; Corral, Sandra G; Giraldo, Alberto S; Gámir, Mariluz G; Mendoza, Antonio Z; Adrovic, Amra; Dedeoglu, Reyhan; Sahin, Sezgin; Barut, Kenan; Koka, Aida; Oztunc, Funda; Kasapcopur, Ozgur; Rodriguez-Lozano, Ana L; Rivas-Larrauri, Francisco; de la Puente, Silvestre G; Alves, Andressa G F; Giacomin, Maria F D A; Farhat, Juliana; Braga, Alfésio L F; Sallum, Adriana M E; Campos, Lúcia M D A; Pereira, Luiz A A; Lichtenfels, Ana J D F C; Silva, Clóvis A; Farhat, Sylvia C L; Acar, Banu; Ozcakar, Z. B; Çakar, Nilgün; Uncu, Nermin; Gür, Gökçe; Özdel, Semanur; Yalçınkaya, Fatoş; Scott, Christiaan; Brice, Nicky; Nourse, Peter; Lewandowski, Laura; Arango, Christine; Mosquera, Angela C; Malagon, Clara; Sakamoto, Ana P; Silva, Clovis A; Silva, Marco F C D; Lopes, Ananadreia S; Russo, Gleice C S; Sallum, Adriana E M; Kozu, Katia; Bonfá, Eloisa; Saad-Magalhães, Claudia; Pereira, Rosa M R; Len, Claudio A; Terreri, Maria T; Suri, Deepti; Didel, Siyaram; Rawat, Amit; Singh, Surjit; Maritsi, Despoina; Onoufriou, MArgarita; Vougiouka, Olga; Tsolia, Maria; Bosak, Edi P; Vidović, Mandica; Lamot, Mirta; Lamot, Lovro; Harjaček, Miroslav; Van Nieuwenhove, Erika; Liston, Adrian; Wouters, Carine; Tahghighi, Fatemeh; Ziaee, Vahid; Raeeskarami, Seid-Reza; Aguiar, Francisca; Pereira, Sandra; Rodrigues, Mariana; Moura, Cláudia; Rocha, Gustavo; Guimarães, Hercília; Brito, Iva; Aguiar, Francisca; Fonseca, Rita; Rodrigues, Mariana; Brito, Iva; Horneff, Gerd; Klein, Ariane; Minden, Kirsten; Huppertz, Hans-Iko; Weller-Heinemann, Frank; Kuemmerle-Deschner, Jasmin; Haas, J-Peter; Hospach, Anton; Menendez-Castro, Ricardo; Huegle, Boris; Haas, Johannes-Peter; Swart, Joost; Giancane, Gabriella; Bovis, Francesca; Castagnola, Elio; Groll, Andreas; Horneff, Gerd; Huppertz, Hans-Iko; Lovell, Daniel J; Wolfs, Tom; Hofer, Michael; Alekseeva, Ekaterina; Panaviene, Violeta; Nielsen, Susan; Anton, Jordi; Uettwiller, Florence; Stanevicha, Valda; Trachana, Maria; Marafon, Denise P; Ailioaie, Constantin; Tsitsami, Elena; Kamphuis, Sylvia; Herlin, Troels; Doležalová, Pavla; Susic, Gordana; Flatø, Berit; Sztajnbok, Flavio; Pistorio, Angela; Martini, Alberto; Wulffraat, Nico; Ruperto, Nicolino; Gattorno, Marco; Brucato, Antonio; Finetti, Martina; Lazaros, George; Maestroni, Silvia; Carraro, Mara; Cumetti, Davide; Carobbio, Alessandra; Lorini, Monia; Rimini, Alessandro; Marcolongo, Renzo; Valenti, Anna; Erre, Gian L; Belli, Riccardo; Gaita, Fiorenzo; Sormani, Maria P; Ruperto, Nicolino; Imazio, Massimo; Martini, Alberto; Abinun, Mario; Smith, Nicola; Rapley, Tim; McErlane, Flora; Kearsley-Fleet, Lianne; Hyrich, Kimme L; Foster, Helen; Ruperto, Nicolino; Lovell, Daniel J; Tzaribachev, Nikolay; Zeft, Andrew; Cimaz, Rolando; Stanevicha, Valda; Horneff, Gerd; Bohnsack, John; Griffin, Thomas; Carrasco, Ruy; Trachana, Maria; Dare, Jason; Foeldvari, Ivan; Vehe, Richard; Bovis, Francesca; Simon, Teresa; Martini, Alberto; Brunner, Hermine; Verazza, S.; Davì, S.; Consolaro, A.; Insalaco, A.; Gerloni, V.; Cimaz, R.; Zulian, F.; Pastore, S.; Corona, F.; Conti, G.; Barone, P.; Cattalini, M.; Cortis, E.; Breda, L.; Olivieri, A. N; Civino, A.; Podda, R.; Rigante, D.; La Torre, F.; D’Angelo, G.; Jorini, M.; Gallizzi, R.; Maggio, M. C; Consolini, R.; De Fanti, A.; Alpigiani, M. G; Martini, A.; Ravelli, A.; Sozeri, Betul; Kısaarslan, Aysenur P; Gunduz, Zubeyde; Dusunsel, Ruhan; Dursun, Ismail; Poyrazoglu, Hakan; Kuchinskaya, Ekaterina; Abduragimova, Farida; Kostik, Mikhail; Sundberg, Erik; Omarsdottir, Soley; Klevenvall, Lena; Erlandsson-Harris, Helena; Basbozkurt, Gokalp; Erdemli, Ozge; Simsek, Dogan; Yazici, Fatih; Karsioglu, Yildirim; Tezcaner, Aysen; Keskin, Dilek; Ozkan, Huseyin; Acikel, Cengizhan; Ozen, Seza; Demirkaya, Erkan; Orbán, Ilonka; Sevcic, Krisztina; Brodszky, Valentin; Kiss, Emese; Tekko, Ismaiel A; Rooney, Madeleine; McElnay, James; Taggart, Cliff; McCarthy, Helen; Donnelly, Ryan F; Abinun, Mario; Slatter, Mary; Nademi, Zohreh; Friswell, Mark; Foster, Helen; Jandial, Sharmila; McErlane, Flora; Flood, Terence; Hambleton, Sophie; Gennery, Andrew; Cant, Andrew; Finetti, Martina; Bovis, Francesca; Swart, Joost; Doležalová, Pavla; Tsitsami, Elena; Trachana, Maria; Demirkaya, Erkan; Duong, Phoi-Ngoc; Koné-Paut, Isabelle; Vougiouka, Olga; Marafon, Denise P; Cimaz, Rolando; Filocamo, Giovanni; Gamir, María L; Stanevicha, Valda; Sanner, Helga; Carenini, Laura; Wulffraat, Nico; Martini, Alberto; Ruperto, Nicolino; Topdemir, Mesut; Basbozkurt, Gokalp; Karslioglu, Yildirim; Ozkan, Huseyin; Acikel, Cengizhan; Demirkaya, Erkan; Gok, Faysal; Zholobova, Elena; Tsurikova, Nadezhda; Ligostaeva, Elena; Ramchurn, Navdha R; Friswell, Mark; Kostareva, O.; Nikishina, I.; Arsenyeva, S.; Rodionovskaya, S.; Kaleda, M.; Alexeev, D.; Dursun, Ismail D; Sozeri, Betul; Kısaarslan, Aysenur P; Dusunsel, Ruhan; Poyrazoglu, Hakan; Poyrazoglu, Hakan; Murias, Sara; Barral, Estefania; Alcobendas, Rosa; Enriquez, Eugenia; Remesal, Agustin; de Inocencio, Jaime; Castro, Tania M; Lotufo, Simone A; Freye, Tatjana; Carlomagno, Raffaella; Zumbrunn, Thomas; Bonhoeffer, Jan; Schneider, Elvira C; Kaiser, Daniela; Hofer, Michaël; Hentgen, Véronique; Woerner, Andreas; Schwarz, Tobias; Klotsche, Jens; Niewerth, Martina; Horneff, Gerd; Haas, Johannes-Peter; Hospach, Anton; Huppertz, Hans-Iko; Ganser, Gerd; Minden, Kirsten; Jeyaratnam, Jerold; ter Haar, Nienke; Kasapcopur, Ozgur; Rigante, Donato; Dedeoglu, Fatma; Baris, Ezgi; Vastert, Sebastiaan; Wulffraat, Nico; Frenkel, Joost; Hausmann, Jonathan S; Lomax, Kathleen G; Shapiro, Ari; Durrant, Karen L; Brogan, P. A; Hofer, M.; Kuemmerle-Deschner, J. B; Lauwerys, B.; Speziale, A.; Leon, K.; Wei, X.; Laxer, R. M; Signa, Sara; Rusmini, Marta; Campione, Elena; Chiesa, Sabrina; Grossi, Alice; Omenetti, Alessia; Caorsi, Roberta; Viglizzo, Gianmaria; Martini, Alberto; Ceccherini, Isabella; Gattorno, Marco; Federici, Silvia; Frenkel, Joost; Ozen, Seza; Lachmann, Helen; Finetti, Martina; Martini, Alberto; Ruperto, Nicola; Gattorno, Marco; Federici, Silvia; Vanoni, Federica; Ozen, Seza; Hofer, Michaël; Frenkel, Joost; Lachmann, Helen; Martini, Alberto; Ruperto, Nicola; Gattorno, Marco; Gomes, Sonia M; Omoyinmi, Ebun; Arostegui, Juan I; Gonzalez-Roca, Eva; Eleftheriou, Despina; Klein, Nigel; Brogan, Paul; Volpi, Stefano; Santori, Elettra; Picco, Paolo; Pastorino, Claudia; Caorsi, Roberta; Rice, Gillian; Tesser, Alessandra; Martini, Alberto; Crow, Yanick; Candotti, Fabio; Gattorno, Marco; Barut, Kenan; Sahin, Sezgin; Adrovic, Amra; Sinoplu, Ada B; Yucel, Gozde; Pamuk, Gizem; Kasapcopur, Ozgur; Damian, Laura O; Lazea, Cecilia; Sparchez, Mihaela; Vele, Paulina; Muntean, Laura; Albu, Adriana; Rednic, Simona; Lazar, Calin; Mendonça, Leonardo O; Pontillo, Alessandra; Kalil, Jorge; Castro, Fabio M; Barros, Myrthes T; Pardeo, Manuela; Messia, Virginia; De Benedetti, Fabrizio; Insalaco, Antonella; Malighetti, Giorgia; Gorio, Chiara; Ricci, Francesca; Parissenti, Ilaria; Montesano, Paola; Bonafini, Barbara; Medeghini, Veronica; Cattalini, Marco; Giordano, Lucio; Zani, Giulia; Ferraro, Rosalba; Vairo, Donatella; Giliani, Silvia; Cattalini, Marco; Maggio, Maria C; Luppino, Girolamo; Corsello, Giovanni; Fernandez, Maria I G; Montesinos, Berta L; Vidal, Adriana R; Gorospe, Juan I A; Penades, Inmaculada C; Rafiq, Nadia K; Wynne, Karen; Hussain, Khalid; Brogan, Paul A; Ang, Elizabeth; Ng, Nicholas; Kacar, Ayla; Gucenmez, Ozge A; Makay, Balahan; Unsal, Sevket E; Sahin, Yasin; Barut, Kenan; Kutlu, Tufan; Cullu-Cokugras, Fugen; Sahin, Sezgin; Adrovic, Amra; Ayyildiz-Civan, Hasret; Kasapcopur, Ozgur; Erkan, Tulay; Abdawani, Reem; Al Zuhbi, Sana; Abdalla, Eiman; Russo, Ricardo A; Katsicas, María M; Caorsi, Roberta; Minoia, Francesca; Viglizzo, Gianmaria; Grossi, Alice; Chiesa, Sabrina; Picco, Paolo; Ravelli, Angelo; Gattorno, Marco; Bhattad, Sagar; Rawat, Amit; Gupta, Anju; Suri, Deepti; Pandiarajan, Vignesh; Nada, Ritambhra; Tiewsoh, Kaara; Hawkins, Philip; Rowczenio, Dorota; Singh, Surjit; Fingerhutova, Sarka; Franova, Jana; Prochazkova, Leona; Hlavackova, Eva; Dolezalova, Pavla; Evrengül, Havva; Yüksel, Selçuk; Doğan, Mustafa; Gürses, Dolunay; Evrengül, Harun; De Pauli, Silvia; Pastore, Serena; Bianco, Anna M; Severini, Giovanni M; Taddio, Andrea; Tommasini, Alberto; Salugina, Svetlana O; Fedorov, Evgeny; Kamenets, Elena; Zaharova, Ekaterina; Kaleda, Maria; Salugina, Svetlana O; Fedorov, Evgeny; Kamenets, Elena; Zaharova, Ekaterina; Kaleda, Maria; Sleptsova, Tatiana; Alexeeva, Ekaterina; Savostyanov, Kirill; Pushkov, Alexander; Bzarova, Tatyana; Valieva, Saniya; Denisova, Rina; Isayeva, Kseniya; Chistyakova, Evgeniya; Lomakina, Olga; Soloshenko, Margarita; Kaschenko, Elena; Kaneko, Utako; Imai, Chihaya; Saitoh, Akihiko; Teixeira, Vitor A; Ramos, Filipa O; Costa, Manuela; Aviel, Yonatan B; Fahoum, Shafe; Brik, Riva; Özçakar, Zeynep B; Çakar, Nilgün; Uncu, Nermin; Celikel, Banu A; Yalcinkaya, Fatos; Schiappapietra, Benedetta; Davi’, Sergio; Mongini, Federica; Giannone, Luisa; Bava, Cecilia; Alpigiani, Maria G; Martini, Alberto; Ravelli, Angelo; Consolaro, Alessandro; Lazarevic, Dragana S; Vojinovic, Jelena; Susic, Gordana; Basic, Jelena; Giancane, Gabriella; Muratore, Valentina; Marzetti, Valentina; Quilis, Neus; Benavente, Belen S; Alongi, Alessandra; Civino, Adele; Quartulli, Lorenzo; Consolaro, Alessandro; Martini, Alberto; Ravelli, Angelo; Januskeviciute, Giedre; van Dijkhuizen, Pieter; Muratore, Valentina; Giancane, Gabriella; Schiappapietra, Benedetta; Martini, Alberto; Ravelli, Angelo; Consolaro, Alessandro; Groot, N.; van Dijk, W.; Bultink, I. E M; Bijl, M.; Dolhain, R. J E M; Teng, Y. K O; Zirkzee, E.; de Leeuw, K.; Fritsch-Stork, R.; Kamphuis, S. S M; Groot, Noortje; Kardolus, A.; Bultink, I. E M; Bijl, M.; Dolhain, R. J E M; Teng, Y. K O; Zirkzee, E.; de Leeuw, K.; Fritsch-Stork, R.; Kamphuis, S. S M; Suárez, Raul G; Nordal, Ellen B; Rypdal, Veronika G; Berntson, Lillemor; Ekelund, Maria; Aalto, Kristiina; Peltoniemi, Suvi; Zak, Marek; Nielsen, Susan; Glerup, Mia; Herlin, Troels; Arnstad, Ellen D; Fasth, Anders; Rygg, Marite; Duarte, Ana C; Sousa, Sandra; Teixeira, Lídia; Cordeiro, Ana; Santos, Mª J; Mourão, Ana F; Santos, Maria J; Eusébio, Mónica; Lopes, Ana; Oliveira-Ramos, Filipa; Salgado, Manuel; Estanqueiro, Paula; Melo-Gomes, José; Martins, Fernando; Costa, José; Furtado, Carolina; Figueira, Ricardo; Brito, Iva; Branco, Jaime C; Fonseca, João E; Canhão, Helena; Mourão, Ana F; Santos, Maria J; Eusébio, Mónica; Lopes, Ana; Oliveira-Ramos, Filipa; Salgado, Manuel; Estanqueiro, Paula; Melo-Gomes, José; Martins, Fernando; Costa, José; Furtado, Carolina; Figueira, Ricardo; Brito, Iva; Branco, Jaime C; Fonseca, João E; Canhão, Helena; Coda, Andrea; Cassidy, Samuel; West, Kerry; Hendry, Gordon; Grech, Debra; Jones, Julie; Hawke, Fiona; Grewal, Davinder S; Coda, Andrea; Jones, Julie; Grech, Debra; Grewal, Davinder S; Foley, Charlene; Killeen, Orla; MacDermott, Emma; Veale, Douglas; Fearon, Ursula; Konukbay, Dilek; Demirkaya, Erkan; Tarakci, Ela; Arman, Nilay; Barut, Kenan; Şahin, Sezgin; Adrovic, Amra; Kasapcopur, Ozgur; Munro, Jane; Consolaro, Alessandro; Morgan, Esi; Riebschleger, Meredith; Horonjeff, Jennifer; Strand, Vibeke; Bingham, Clifton; Collante, Ma. T M; Ganeva, Margarita; Stefanov, Stefan; Telcharova, Albena; Mihaylova, Dimitrina; Saraeva, Radoslava; Tzveova, Reni; Kaneva, Radka; Tsakova, Adelina; Temelkova, Katya; Picarelli, Maria M C; Danzmann, Luiz C; Barbé-Tuana, Florencia; Grun, Lucas K; Jones, Marcus H; Frković, Marijan; Ištuk, Karla; Birkić, Ika; Sršen, Saša; Jelušić, Marija; Smith, Nicola; Jandial, Sharmila; Easton, Alan; Quarmby, Rachael; Khubchandani, Raju; Chan, Mercedes; Rapley, Tim; Foster, Helen; Srp, Radoslav; Kobrova, Katerina; Franova, Jana; Fingerhutova, Sarka; Nemcova, Dana; Hoza, Jozef; Uher, Michal; Saifridova, Melania; Linkova, Lenka; Dolezalova, Pavla; Charuvanij, Sirirat; Leelayuwattanakul, Isree; Pacharapakornpong, Thita; Vallipakorn, Sakda A; Lerkvaleekul, Butsabong; Vilaiyuk, Soamarat; Muratore, Valentina; Giancane, Gabriella; Lanni, Stefano; Alongi, Alessandra; Alpigiani, Maria G; Martini, Alberto; Ravelli, Angelo; Consolaro, Alessandro; Alongi, Alessandra; Bovis, Francesca; Minoia, Francesca; Davì, Sergio; Martini, Alberto; Ruperto, Nicolino; Cron, Randy Q; Ravelli, Angelo; Passarelli, Chiara; Pardeo, Manuela; Pisaneschi, Elisa; Novelli, Antonio; De Benedetti, Fabrizio; Bracaglia, Claudia; Bracaglia, Claudia; Marafon, Denise P; Caiello, Ivan; de Graaf, Kathy; Guilhot, Florence; Ferlin, Walter; Davi’, Sergio; Schulert, Grant; Ravelli, Angelo; Grom, Alexi A; Nelson, Robert; de Min, Cristina; De Benedetti, Fabrizio; Holzinger, Dirk; Kessel, Christoph; Fall, Ndate; Grom, Alexei; de Jager, Wilco; Vastert, Sebastiaan; Strippoli, Raffaele; Bracaglia, Claudia; Sundberg, Erik; Horne, Anna; Ehl, Stephan; Ammann, Sandra; Lehmberg, Kai; De Benedetti, Fabrizio; Beutel, Karin; Foell, Dirk; Minoia, Francesca; Horne, AnnaCarin; Bovis, Francesca; Davì, Sergio; Pagani, Laura; Espada, Graciela; Gao, Yi-jin; Insalaco, Antonella; Lehmberg, Kai; Sanner, Helga; Shenoi, Susan; Weitzman, Sheila; Ruperto, Nicolino; Martini, Alberto; Cron, Randy Q; Ravelli, Angelo; Prencipe, Giusi; Caiello, Ivan; Pascarella, Antonia; Bracaglia, Claudia; Ferlin, Walter G; Chatel, Laurence; Strippoli, Raffaele; de Min, Cristina; De Benedetti, Fabrizio; Jacqmin, Philippe; De Graaf, Kathy; Ballabio, Maria; Nelson, Robert; Johnson, Zoë; Ferlin, Walter; Lapeyre, Geneviève; de Benedetti, Fabrizio; Cristina, de M; Wakiguchi, Hiroyuki; Hasegawa, Shunji; Hirano, Reiji; Okazaki, Fumiko; Nakamura, Tamaki; Kaneyasu, Hidenobu; Ohga, Shouichi; Yamazaki, Kazuko; Nozawa, Tomo; Kanetaka, Taichi; Ito, Shuichi; Yokota, Shumpei; McLellan, Kirsty; MacGregor, Ishbel; Martin, Neil; Davidson, Joyce; Kuemmerle-Deschner, Jasmin; Hansmann, Sandra; Wulffraat, Nico; Eikelberg, Andreas; Haug, Iris; Schuller, Sabrina; Benseler, Susanne M; Nazarova, Liliia S; Danilko, Kseniia V; Malievsky, Viktor A; Viktorova, Tatiana V; Mauro, Angela; Omoyinmi, Ebun; Barnicoat, Angela; Brogan, Paul; Foley, Charlene; Killeen, Orla; MacDermott, Emma; Veale, Douglas; Foley, Charlene; Killeen, Orla; MacDermott, Emma; Veale, Douglas; Gomes, Sonia M; Omoyinmi, Ebun; Hurst, Jane; Canham, Nathalie; Eleftheriou, Despina; Klein, Nigel; Lacassagne, Sandrine; Brogan, Paul; Wiener, Anastasia; Hügle, Boris; Denecke, Bernd; Costa-Filho, Ivan; Haas, Johannes P; Tenbrock, Klaus; Popp, David; Boltjes, Arjan; Rühle, Frank; Herresthal, Stefanie; de Jager, Wilco; van Wijk, Femke; Schultze, Joachim; Stoll, Monika; Klotz, Luisa; Vogl, Thomas; Roth, Johannes; Quesada-Masachs, Estefania; de la Sierra, Daniel Á; Prat, Marina G; Sánchez, Ana M M; Borrell, Ricardo P; Barril, Sara M; Gallo, Mónica M; Caballero, Consuelo M; Chyzheuskaya, Iryna; Byelyaeva, Lyudmyla M; Filonovich, Rostislav M; Khrustaleva, Helena K; Zajtseva, Larisa I; Yuraga, Tamara M; Chyzheuskaya, Iryna; Byelyaeva, Lyudmyla M; Filonovich, Rostislav M; Khrustaleva, Helena K; Zajtseva, Larisa I; Yuraga, Tamara M; Giner, Thomas; Hackl, Lukas; Albrecht, Julia; Würzner, Reinhard; Brunner, Juergen; Pastore, Serena; Minute, Marta; Parentin, Fulvio; Tesser, Alessandra; Nocerino, Agostino; Taddio, Andrea; Tommasini, Alberto; Nørgaard, Mette; Herlin, Troels; Alberdi-Saugstrup, Mikel; Zak, Marek S; Nielsen, Susan M; Herlin, Troels; Nordal, Ellen; Berntson, Lillemor; Fasth, Anders; Rygg, Marite; Müller, Klaus G; Avramovič, Mojca Z; Dolžan, Vita; Toplak, Nataša; Avčin, Tadej; Ruperto, N.; Lovell, D. J; Wallace, C.; Toth, M.; Foeldvari, I.; Bohnsack, J.; Milojevic, D.; Rabinovich, C.; Kingsbury, D.; Marzan, K.; Quartier, P.; Minden, K.; Chalom, E.; Horneff, G.; Kuester, R. M; Dare, J.; Heinrich, M.; Kupper, H.; Kalabic, J.; Martini, A.; Brunner, H. 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    Towards a histological diagnosis of childhood small vessel CNS vasculitis
    (2024-12-28) Nouri, Maryam N.; Dropol, Anastasia; Tyrrell, Pascal N.; Sheikh, Sheila; Twilt, Marinka; Michaud, Jean; Ellezam, Benjamin; Sarnat, Harvey B.; Dunham, Christopher; Schutz, Peter W.; Keith, Julia; Munoz, David G.; Vinters, Harry V.; Hawkins, Cynthia; Benseler, Susanne M.
    Abstract Background Primary small vessel CNS vasculitis (sv-cPACNS) is a challenging inflammatory brain disease in children. Brain biopsy is mandatory to confirm the diagnosis. This study aims to develop and validate a histological scoring tool for diagnosing small vessel CNS vasculitis. Methods A standardized brain biopsy scoring instrument was developed and applied to consecutive full-thickness brain biopsies of pediatric cases and controls at a single center. Stains included immunohistochemistry and Hematoxylin & Eosin. Nine North American neuropathologists, blinded to patients’ presentation, diagnosis, and therapy, scored de-identified biopsies independently. Results A total of 31 brain biopsy specimens from children with sv-cPACNS, 11 with epilepsy, and 11 with non-vasculitic inflammatory brain disease controls were included. Angiocentric inflammation in the cortex or white matter increases the likelihood of sv-cPACNS, with odds ratios (ORs) of 3.231 (95CI: 0.914–11.420, p = 0.067) and 3.923 (95CI: 1.13–13.6, p = 0.031). Moderate to severe inflammation in these regions is associated with a higher probability of sv-cPACNS, with ORs of 5.56 (95CI: 1.02–29.47, p = 0.046) in the cortex and 6.76 (95CI: 1.26–36.11, p = 0.025) in white matter. CD3, CD4, CD8, and CD20 cells predominated the inflammatory infiltrate. Reactive endothelium was strongly associated with sv-cPACNS, with an OR of 8.93 (p = 0.001). Features reported in adult sv-PACNS, including granulomas, necrosis, or fibrin deposits, were absent in all biopsies. The presence of leptomeningeal inflammation in isolation was non-diagnostic. Conclusion Distinct histological features were identified in sv-cPACNS biopsies, including moderate to severe angiocentric inflammatory infiltrates in the cortex or white matter, consisting of CD3, CD4, CD8, and CD20 cells, alongside reactive endothelium with specificity of 95%. In the first study of its kind proposing histological criteria for evaluating brain biopsies, we aim to precisely characterize the type and severity of the inflammatory response in patients with sv-cPACNS; this can enable consolidation of this population to assess outcomes and treatment methodologies comprehensively.