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Clinical relevance of distinguishing autoimmune nodopathies from CIDP: longitudinal assessment in a large cohort.
Broers, Merel C; Wieske, Luuk; Erdag, Ece; Gürlek, Cemre; Bunschoten, Carina; van Doorn, Pieter A; Eftimov, Filip; Kuitwaard, Krista; de Vries, Juna M; de Wit, Marie-Claire Y; Nagtzaam, Mariska Mp; Franken, Suzanne C; Zhu, Louisa; Paunovic, Manuela; de Wit, Maurice; Schreurs, Marco Wj; Lleixà, Cinta; Martín-Aguilar, Lorena; Pascual-Goñi, Elba; Querol, Luis; Jacobs, Bart C; Huizinga, Ruth; Titulaer, Maarten J.
Afiliación
  • Broers MC; Department of Neurology, Erasmus MC, University Medical Center, Rotterdam, The Netherlands.
  • Wieske L; Department of Neurology, Amsterdam Neuroscience, Amsterdam UMC, location AMC, Amsterdam, The Netherlands.
  • Erdag E; Department of Neurology, Erasmus MC, University Medical Center, Rotterdam, The Netherlands.
  • Gürlek C; Department of Neuroscience, Aziz Sancar Institute of Experimental Medicine, Istanbul University, Istanbul, Turkey.
  • Bunschoten C; Department of Neurology, Erasmus MC, University Medical Center, Rotterdam, The Netherlands.
  • van Doorn PA; Department of Immunology, Erasmus MC, University Medical Center, Rotterdam, The Netherlands.
  • Eftimov F; Department of Neurology, Erasmus MC, University Medical Center, Rotterdam, The Netherlands.
  • Kuitwaard K; Department of Neurology, Erasmus MC, University Medical Center, Rotterdam, The Netherlands.
  • de Vries JM; Department of Neurology, Amsterdam Neuroscience, Amsterdam UMC, location AMC, Amsterdam, The Netherlands.
  • de Wit MY; Department of Neurology, Erasmus MC, University Medical Center, Rotterdam, The Netherlands.
  • Nagtzaam MM; Department of Neurology, Albert Schweitzer Hospital, Dordrecht, The Netherlands.
  • Franken SC; Department of Neurology, Erasmus MC, University Medical Center, Rotterdam, The Netherlands.
  • Zhu L; Department of Pediatric Neurology, Erasmus MC, Erasmus MC Sophia Children's Hospital, Rotterdam, The Netherlands.
  • Paunovic M; Department of Neurology, Erasmus MC, University Medical Center, Rotterdam, The Netherlands.
  • de Wit M; Department of Neurology, Erasmus MC, University Medical Center, Rotterdam, The Netherlands.
  • Schreurs MW; Department of Neurology, Erasmus MC, University Medical Center, Rotterdam, The Netherlands.
  • Lleixà C; Department of Immunology, Erasmus MC, University Medical Center, Rotterdam, The Netherlands.
  • Martín-Aguilar L; Department of Neurology, Erasmus MC, University Medical Center, Rotterdam, The Netherlands.
  • Pascual-Goñi E; Department of Neurology, Erasmus MC, University Medical Center, Rotterdam, The Netherlands.
  • Querol L; Department of Immunology, Erasmus MC, University Medical Center, Rotterdam, The Netherlands.
  • Jacobs BC; Neuromuscular Diseases Unit, Department of Neurology, Hospital de la Santa Creu i Sant Pau, Universitat Autònoma de Barcelona, Barcelona, Spain.
  • Huizinga R; Neuromuscular Diseases Unit, Department of Neurology, Hospital de la Santa Creu i Sant Pau, Universitat Autònoma de Barcelona, Barcelona, Spain.
  • Titulaer MJ; Neuromuscular Diseases Unit, Department of Neurology, Hospital de la Santa Creu i Sant Pau, Universitat Autònoma de Barcelona, Barcelona, Spain.
J Neurol Neurosurg Psychiatry ; 95(1): 52-60, 2023 Dec 14.
Article en En | MEDLINE | ID: mdl-37879898
BACKGROUND: The aim of this study was to determine treatment response and whether it is associated with antibody titre change in patients with autoimmune nodopathy (AN) previously diagnosed as chronic inflammatory demyelinating polyradiculoneuropathy (CIDP), and to compare clinical features and treatment response between AN and CIDP. METHODS: Serum IgG antibodies to neurofascin-155 (NF155), contactin-1 (CNTN1) and contactin-associated protein 1 (CASPR1) were detected with cell-based assays in patients diagnosed with CIDP. Clinical improvement was determined using the modified Rankin scale, need for alternative and/or additional treatments and assessment of the treating neurologist. RESULTS: We studied 401 patients diagnosed with CIDP and identified 21 patients with AN (10 anti-NF155, 6 anti-CNTN1, 4 anti-CASPR1 and 1 anti-NF155/anti-CASPR1 double positive). In patients with AN ataxia (68% vs 28%, p=0.001), cranial nerve involvement (34% vs 11%, p=0.012) and autonomic symptoms (47% vs 22%, p=0.025) were more frequently reported; patients with AN improved less often after intravenous immunoglobulin treatment (39% vs 80%, p=0.002) and required additional/alternative treatments more frequently (84% vs 34%, p<0.001), compared with patients with CIDP. Antibody titres decreased or became negative in patients improving on treatment. Treatment withdrawal was associated with a titre increase and clinical deterioration in four patients. CONCLUSIONS: Distinguishing CIDP from AN is important, as patients with AN need a different treatment approach. Improvement and relapses were associated with changes in antibody titres, supporting the pathogenicity of these antibodies.
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Polirradiculoneuropatía Crónica Inflamatoria Desmielinizante Límite: Humans Idioma: En Revista: J Neurol Neurosurg Psychiatry Año: 2023 Tipo del documento: Article País de afiliación: Países Bajos Pais de publicación: Reino Unido
Texto completo
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Polirradiculoneuropatía Crónica Inflamatoria Desmielinizante Límite: Humans Idioma: En Revista: J Neurol Neurosurg Psychiatry Año: 2023 Tipo del documento: Article País de afiliación: Países Bajos Pais de publicación: Reino Unido