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Comparative Study of Paraneoplastic and Nonparaneoplastic Autoimmune Encephalitis With GABABR Antibodies.
Lamblin, Florian; Kerstens, Jeroen; Muñiz-Castrillo, Sergio; Vogrig, Alberto; Goncalves, David; Rogemond, Veronique; Picard, Geraldine; Villard, Marine; Pinto, Anne-Laurie; Van Coevorden-Hameete, Marleen H; De Bruijn, Marienke A; De Vries, Juna M; Schreurs, Marco; Tyvaert, Louise; Hopes, Lucie; Aupy, Jerome; Marchal, Cecile; Psimaras, Dimitri; Kremer, Laurent; Bourg, Veronique; Antoine, Jean-Christophe G; Wang, Adrien; Kahane, Philippe; Demeret, Sophie; Ahle, Guido; Sempere, Vicente Peris; Timestit, Noemie; Nourredine, Mikail; Maureille, Aurelien; Benaiteau, Marie; Joubert, Bastien; Mignot, Emmanuel; Titulaer, Maarten J; Honnorat, Jerome.
Afiliación
  • Lamblin F; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Kerstens J; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Muñiz-Castrillo S; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Vogrig A; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Goncalves D; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Rogemond V; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Picard G; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Villard M; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Pinto AL; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Van Coevorden-Hameete MH; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • De Bruijn MA; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • De Vries JM; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Schreurs M; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Tyvaert L; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Hopes L; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Aupy J; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Marchal C; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Psimaras D; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Kremer L; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Bourg V; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Antoine JG; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Wang A; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Kahane P; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Demeret S; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Ahle G; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Sempere VP; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Timestit N; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Nourredine M; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Maureille A; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Benaiteau M; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Joubert B; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Mignot E; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Titulaer MJ; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
  • Honnorat J; From the French Reference Center on Paraneoplastic Neurological Syndrome and Autoimmune Encephalitis (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Hospices Civils de Lyon; Institut MeLiS INSERM U1314/CNRS UMR 5284 (F.L., V.R., G.P., M.V., A.-L.P., M.B., B.J., J.H.), Université Claude Bernard
Neurol Neuroimmunol Neuroinflamm ; 11(3): e200229, 2024 May.
Article en En | MEDLINE | ID: mdl-38657198
ABSTRACT
BACKGROUND AND

OBJECTIVES:

While patients with paraneoplastic autoimmune encephalitis (AE) with gamma-aminobutyric-acid B receptor antibodies (GABABR-AE) have poor functional outcomes and high mortality, the prognosis of nonparaneoplastic cases has not been well studied.

METHODS:

Patients with GABABR-AE from the French and the Dutch Paraneoplastic Neurologic Syndromes Reference Centers databases were retrospectively included and their data collected; the neurologic outcomes of paraneoplastic and nonparaneoplastic cases were compared. Immunoglobulin G (IgG) isotyping and human leukocyte antigen (HLA) genotyping were performed in patients with available samples.

RESULTS:

A total of 111 patients (44/111 [40%] women) were enrolled, including 84 of 111 (76%) paraneoplastic and 18 of 111 (16%) nonparaneoplastic cases (cancer status was undetermined for 9 patients). Patients presented with seizures (88/111 [79%]), cognitive impairment (54/111 [49%]), and/or behavioral disorders (34/111 [31%]), and 54 of 111 (50%) were admitted in intensive care unit (ICU). Nonparaneoplastic patients were significantly younger (median age 54 years [range 19-88] vs 67 years [range 50-85] for paraneoplastic cases, p < 0.001) and showed a different demographic distribution. Nonparaneoplastic patients more often had CSF pleocytosis (17/17 [100%] vs 58/78 [74%], p = 0.02), were almost never associated with KTCD16-abs (1/16 [6%] vs 61/70 [87%], p < 0.001), and were more frequently treated with second-line immunotherapy (11/18 [61%] vs 18/82 [22%], p = 0.003). However, no difference of IgG subclass or HLA association was observed, although sample size was small (10 and 26 patients, respectively). After treatment, neurologic outcome was favorable (mRS ≤2) for 13 of 16 (81%) nonparaneoplastic and 37 of 84 (48%) paraneoplastic cases (p = 0.03), while 3 of 18 (17%) and 42 of 83 (51%) patients had died at last follow-up (p = 0.008), respectively. Neurologic outcome no longer differed after adjustment for confounding factors but seemed to be negatively associated with increased age and ICU admission. A better survival was associated with nonparaneoplastic cases, a younger age, and the use of immunosuppressive drugs.

DISCUSSION:

Nonparaneoplastic GABABR-AE involved younger patients without associated KCTD16-abs and carried better neurologic and vital prognoses than paraneoplastic GABABR-AE, which might be due to a more intensive treatment strategy. A better understanding of immunologic mechanisms underlying both forms is needed.
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Texto completo: 1 Colección: 01-internacional Banco de datos: MEDLINE Asunto principal: Autoanticuerpos / Receptores de GABA-B / Síndromes Paraneoplásicos del Sistema Nervioso / Encefalitis / Enfermedad de Hashimoto Límite: Adult / Aged / Aged80 / Female / Humans / Male / Middle aged Idioma: En Revista: Neurol Neuroimmunol Neuroinflamm Año: 2024 Tipo del documento: Article
Texto completo
Imprimir
XML
PubMed Links
Buscar en Google

Texto completo: 1 Colección: 01-internacional Banco de datos: MEDLINE Asunto principal: Autoanticuerpos / Receptores de GABA-B / Síndromes Paraneoplásicos del Sistema Nervioso / Encefalitis / Enfermedad de Hashimoto Límite: Adult / Aged / Aged80 / Female / Humans / Male / Middle aged Idioma: En Revista: Neurol Neuroimmunol Neuroinflamm Año: 2024 Tipo del documento: Article