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Expanding clinical spectrum of Caspr2 antibody-associated disease: warning on brainstem involvement and respiratory failure.
Di Giacomo, Roberta; Rossi Sebastiano, Davide; Cazzato, Daniele; Andreetta, Francesca; Pozzi, Paolo; Cenciarelli, Silvia; Deleo, Francesco; Pastori, Chiara; Didato, Giuseppe; de Curtis, Marco; Villani, Flavio.
Afiliación
  • Di Giacomo R; Epilepsy Unit, Fondazione IRCCS Istituto Neurologico Carlo Besta, Milan, Italy. Electronic address: roberta.digiacomo@istituto-besta.it.
  • Rossi Sebastiano D; Neurophysiology Unit, Fondazione IRCCS Istituto Neurologico Carlo Besta, Italy.
  • Cazzato D; Neurophysiology Unit, Fondazione IRCCS Istituto Neurologico Carlo Besta, Italy.
  • Andreetta F; Neurolgy Unit IV, Fondazione IRCCS Istituto Neurologico Carlo Besta, Milan, Italy.
  • Pozzi P; Respiratory Medicine, Sant'Antonio Abate Hospital, ASST Lariana, Como, Italy.
  • Cenciarelli S; Neurology Unit-Stroke Unit, Gubbio/Gualdo Tadino and Città di Castello Hospitals, USL Umbria 1, Perugia, Italy.
  • Deleo F; Epilepsy Unit, Fondazione IRCCS Istituto Neurologico Carlo Besta, Milan, Italy.
  • Pastori C; Epilepsy Unit, Fondazione IRCCS Istituto Neurologico Carlo Besta, Milan, Italy.
  • Didato G; Epilepsy Unit, Fondazione IRCCS Istituto Neurologico Carlo Besta, Milan, Italy.
  • de Curtis M; Epilepsy Unit, Fondazione IRCCS Istituto Neurologico Carlo Besta, Milan, Italy.
  • Villani F; Epilepsy Unit, Fondazione IRCCS Istituto Neurologico Carlo Besta, Milan, Italy; Division of Clinical Neurophysiology and Epilepsy Center, IRCCS Ospedale Policlinico San Martino, Genova, Italy.
J Neurol Sci ; 413: 116865, 2020 06 15.
Article en En | MEDLINE | ID: mdl-32371280
We report the case of a 68-year-old man who presented with ataxia, insomnia, rapidly developing cognitive decline, seizures and small vessel vasculitis. Both serum and cerebro-spinal fluid samples showed positive titre of anti-CASPR2 antibodies. Limbic encephalitis was diagnosed and immunomodulatory therapy was started with benefit. After one-year follow-up, the patient relapsed with a difficult-to-treat respiratory failure, brainstem involvement, neuropathic pain and severe dysautonomia with esophageal dysfunction. We discuss here the occurrence of life-threating complication such as respiratory dysfunction in CASPR2 limbic encephalitis. Furthermore, we showed different phenotype and treatment response during disease onset compared to relapse. This case expands the clinical spectrum of anti-CASPR2 associated disease, underlying the need for respiratory and sleep evaluation.
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Insuficiencia Respiratoria / Encefalitis Límbica Tipo de estudio: Etiology_studies / Risk_factors_studies Límite: Aged / Humans / Male Idioma: En Revista: J Neurol Sci Año: 2020 Tipo del documento: Article Pais de publicación: Países Bajos
Texto completo
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Insuficiencia Respiratoria / Encefalitis Límbica Tipo de estudio: Etiology_studies / Risk_factors_studies Límite: Aged / Humans / Male Idioma: En Revista: J Neurol Sci Año: 2020 Tipo del documento: Article Pais de publicación: Países Bajos