Clinical characterization and long-term outcome in children and adults with anti-AMPA receptor encephalitis
Background and Objectives - Anti-alpha-amino-3-hydroxy-5-methyl-4-isoxazole-propionic acid receptor (anti-AMPAR) encephalitis manifests as limbic encephalitis in adults and is often associated with cancer. Although some reports suggest that it may occur in children, the clinical features in this pop...
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| Dokumenttyp: | Article (Journal) |
| Sprache: | Englisch |
| Veröffentlicht: |
September 2025
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| In: |
Neurology: Neuroimmunology & Neuroinflammation ; official journal of the American Academy of Neurology
Year: 2025, Jahrgang: 12, Heft: 5, Pages: 1-15 |
| ISSN: | 2332-7812 |
| DOI: | 10.1212/NXI.0000000000200453 |
| Online-Zugang: | Verlag, kostenfrei, Volltext: https://doi.org/10.1212/NXI.0000000000200453 Verlag, kostenfrei, Volltext: https://www.neurology.org/doi/10.1212/NXI.0000000000200453 
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| Verfasserangaben: | Chiara Milano, Ezgi Saylam, Claudia Papi, Laura Marmolejo, Alexandra Sankovic, Raphael Reinecke, Jeroen Kerstens, Chiara Pizzanelli, Mateus Mistieri Simabukuro, Marie Benaiteau, Bastien Joubert, Matteo Gastaldi, Lívia Almeida Dutra, Frank Leypoldt, Mareike Jansen, Izumi Kawachi, Hisanao Akiyama, Nikolas Boy, Silvia Bozzetti, Peter Broegger Christensen, Pietro Businaro, Alessandro Dinoto, Tal Friedman-Korn, Urara Fujiwara, Tatsuya Fukumoto, Kenshiro Fuse, Sam Ishmael Hooshmand, Lauren Hurst, Raffaele Iorio, Isabelle Korn-Lubetzki, André Filipe Lucchi Rodrigues, Sara Mariotto, Johannes Michael, Yuko Morimoto, Rinze Frederik Neuteboom, Amanda L. Piquet, Andrea O. Rossetti, Sabine Rumpler-Kreiner, Jonathan D. Santoro, Florian Schwendinger, Brenda Shen, Steffen Syrbe, Johannes Troger, Ingrid Wagner, Christian G. Bien, Eugenia Martinez-Hernandez, Thais Armangue, Romana Höftberger, Takahiro Iizuka, Maarten J. Titulaer, Jerome Honnorat, Francesc Graus, Josep O. Dalmau, Setty Magaña, and Marianna Spatola |
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| 245 | 1 | 0 | |a Clinical characterization and long-term outcome in children and adults with anti-AMPA receptor encephalitis |c Chiara Milano, Ezgi Saylam, Claudia Papi, Laura Marmolejo, Alexandra Sankovic, Raphael Reinecke, Jeroen Kerstens, Chiara Pizzanelli, Mateus Mistieri Simabukuro, Marie Benaiteau, Bastien Joubert, Matteo Gastaldi, Lívia Almeida Dutra, Frank Leypoldt, Mareike Jansen, Izumi Kawachi, Hisanao Akiyama, Nikolas Boy, Silvia Bozzetti, Peter Broegger Christensen, Pietro Businaro, Alessandro Dinoto, Tal Friedman-Korn, Urara Fujiwara, Tatsuya Fukumoto, Kenshiro Fuse, Sam Ishmael Hooshmand, Lauren Hurst, Raffaele Iorio, Isabelle Korn-Lubetzki, André Filipe Lucchi Rodrigues, Sara Mariotto, Johannes Michael, Yuko Morimoto, Rinze Frederik Neuteboom, Amanda L. Piquet, Andrea O. Rossetti, Sabine Rumpler-Kreiner, Jonathan D. Santoro, Florian Schwendinger, Brenda Shen, Steffen Syrbe, Johannes Troger, Ingrid Wagner, Christian G. Bien, Eugenia Martinez-Hernandez, Thais Armangue, Romana Höftberger, Takahiro Iizuka, Maarten J. Titulaer, Jerome Honnorat, Francesc Graus, Josep O. Dalmau, Setty Magaña, and Marianna Spatola |
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| 500 | |a Gesehen am 28.11.2025 | ||
| 520 | |a Background and Objectives - Anti-alpha-amino-3-hydroxy-5-methyl-4-isoxazole-propionic acid receptor (anti-AMPAR) encephalitis manifests as limbic encephalitis in adults and is often associated with cancer. Although some reports suggest that it may occur in children, the clinical features in this population, as well as the prognostic factors and long-term outcomes in children and adults, are unknown. - Methods - We performed a retrospective, international collaborative study of patients with anti-AMPAR encephalitis. Clinical information was reviewed, together with data from published pediatric patients. Clinical features of children and adults were compared with nonparametric tests. Survival rates (Kaplan-Meier curves) were compared using log-rank tests. Prognostic factors of poor outcome (modified Rankin Scale score >2) were identified using logistic regression models. - Results - A total of 115 patients were included, of whom 84 (71 adults, 13 children) had only AMPAR antibodies and 31 (27 adults, 4 children) had additional concurrent neural antibodies. Among patients with AMPAR antibodies alone, tumors were identified in 37 adults (56%) and none of the children (p < 0.0001). Children were more likely than adults to have behavioral/psychiatric symptoms (5/13, 39%, vs 8/71, 11%, p = 0.026) at onset, cerebellar dysfunction (6/13, 46%, vs 7/68, 10% p = 0.005) or movement disorders (5/13, 39%, vs 8/67, 12%, p = 0.032) during the disease course, and extratemporal brain MRI lesions (4/9, 44%, vs 5/44, 11%, p = 0.035). Among 34 patients with prolonged follow-up (>24 months), long-term neurocognitive sequelae were reported in 23 (68%), all adults. Failure to respond to first-line immunotherapy at multivariable analysis predicted a poor outcome (OR 8.0, 95% CI 1.1-59.2, p = 0.043). Among the 31 patients with concurrent neural autoantibodies, 22 (79%) had a tumor; those with high-risk antibodies had lower survival rates (p = 0.008). - Discussion - Children and adults with anti-AMPAR encephalitis show distinct clinical-radiologic features. At long-term follow-up, 68% of patients, all adults, have neurologic sequelae, with failure to respond to first-line immunotherapy being associated with worse outcomes. | ||
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