Pubblicazioni

Autori:

Milano, Chiara; Saylam, Ezgi; Papi, Claudia; Marmolejo, Laura; Sankovic, Alexandra; Reinecke, Raphael; Kerstens, Jeroen; Pizzanelli, Chiara; Simabukuro, Mateus Mistieri; Benaiteau, Marie; Joubert, Bastien; Gastaldi, Matteo; Dutra, Lívia Almeida; Leypoldt, Frank; Jansen, Mareike; Kawachi, Izumi; Akiyama, Hisanao; Boy, Nikolas; Bozzetti, Silvia; Christensen, Peter Broegger; Businaro, Pietro; Dinoto, Alessandro; Friedman-Korn, Tal; Fujiwara, Urara; Fukumoto, Tatsuya; Fuse, Kenshiro; Hooshmand, Sam Ishmael; Hurst, Lauren; Iorio, Raffaele; Korn-Lubetzki, Isabelle; Rodrigues, André Filipe Lucchi; Mariotto, Sara; Michael, Johannes; Morimoto, Yuko; Neuteboom, Rinze Frederik; Piquet, Amanda L; Rossetti, Andrea O; Rumpler-Kreiner, Sabine; Santoro, Jonathan D; Schwendinger, Florian; Shen, Brenda; Syrbe, Steffen; Troger, Johannes; Wagner, Ingrid; Bien, Christian G; Martinez-Hernandez, Eugenia; Armangue, Thais; Höftberger, Romana; Iizuka, Takahiro; Titulaer, Maarten J; Honnorat, Jerome; Graus, Francesc; Dalmau, Josep O; Magaña, Setty; Spatola, Marianna

Titolo:

Clinical Characterization and Long-Term Outcome in Children and Adults With Anti-AMPA Receptor Encephalitis

Anno:

2025

Tipologia prodotto:

Articolo in Rivista

Tipologia ANVUR:

Articolo su rivista

Lingua:

Inglese

Formato:

Elettronico

Referee:

Sì

Nome rivista:

NEUROLOGY® NEUROIMMUNOLOGY & NEUROINFLAMMATION

ISSN Rivista:

2332-7812

N° Volume:

12

Numero o Fascicolo:

5

Intervallo pagine:

1-15

Parole chiave:

Anti-AMPA Receptor; Encephalitis; Child; Adult; long-term follow-up; outcome

Breve descrizione dei contenuti:

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.

Pagina Web:

https://doi.org/10.1212/nxi.0000000000200453

Id prodotto:

146876

Handle IRIS:

11562/1167968

ultima modifica:

27 settembre 2025

Citazione bibliografica:

Milano, Chiara; Saylam, Ezgi; Papi, Claudia; Marmolejo, Laura; Sankovic, Alexandra; Reinecke, Raphael; Kerstens, Jeroen; Pizzanelli, Chiara; Simabukuro, Mateus Mistieri; Benaiteau, Marie; Joubert, Bastien; Gastaldi, Matteo; Dutra, Lívia Almeida; Leypoldt, Frank; Jansen, Mareike; Kawachi, Izumi; Akiyama, Hisanao; Boy, Nikolas; Bozzetti, Silvia; Christensen, Peter Broegger; Businaro, Pietro; Dinoto, Alessandro; Friedman-Korn, Tal; Fujiwara, Urara; Fukumoto, Tatsuya; Fuse, Kenshiro; Hooshmand, Sam Ishmael; Hurst, Lauren; Iorio, Raffaele; Korn-Lubetzki, Isabelle; Rodrigues, André Filipe Lucchi; Mariotto, Sara; Michael, Johannes; Morimoto, Yuko; Neuteboom, Rinze Frederik; Piquet, Amanda L; Rossetti, Andrea O; Rumpler-Kreiner, Sabine; Santoro, Jonathan D; Schwendinger, Florian; Shen, Brenda; Syrbe, Steffen; Troger, Johannes; Wagner, Ingrid; Bien, Christian G; Martinez-Hernandez, Eugenia; Armangue, Thais; Höftberger, Romana; Iizuka, Takahiro; Titulaer, Maarten J; Honnorat, Jerome; Graus, Francesc; Dalmau, Josep O; Magaña, Setty; Spatola, Marianna, Clinical Characterization and Long-Term Outcome in Children and Adults With Anti-AMPA Receptor Encephalitis «NEUROLOGY® NEUROIMMUNOLOGY & NEUROINFLAMMATION» , vol. 12 , n. 5 , 2025 , pp. 1-15

Consulta la scheda completa presente nel repository istituzionale della Ricerca di Ateneo