Frontiers in Neurology (Dec 2020)
New Algorithms Improving PML Risk Stratification in MS Patients Treated With Natalizumab
- Inmaculada Toboso,
- Amalia Tejeda-Velarde,
- Roberto Alvarez-Lafuente,
- Rafael Arroyo,
- Harald Hegen,
- Florian Deisenhammer,
- Susana Sainz de la Maza,
- José C. Alvarez-Cermeño,
- Guillermo Izquierdo,
- Dolores Paramo,
- Pedro Oliva,
- Bonaventura Casanova,
- Eduardo Agüera-Morales,
- Diego Franciotta,
- Matteo Gastaldi,
- Oscar Fernández,
- Patricia Urbaneja,
- José M. Garcia-Dominguez,
- Fernando Romero,
- Alicia Laroni,
- Antonio Uccelli,
- Angel Perez-Sempere,
- Albert Saiz,
- Yolanda Blanco,
- Daniela Galimberti,
- Elio Scarpini,
- Carmen Espejo,
- Xavier Montalban,
- Ludwig Rasche,
- Friedemann Paul,
- Friedemann Paul,
- Inés González,
- Elena Álvarez,
- Cristina Ramo,
- Ana B. Caminero,
- Yolanda Aladro,
- Carmen Calles,
- Pablo Eguía,
- Antonio Belenguer-Benavides,
- Lluis Ramió-Torrentà,
- Ester Quintana,
- José E. Martínez-Rodríguez,
- Agustín Oterino,
- Carlos López de Silanes,
- Luis I. Casanova,
- Lamberto Landete,
- Jette Frederiksen,
- Gabriel Bsteh,
- Patricia Mulero,
- Manuel Comabella,
- Miguel A. Hernández,
- Mercedes Espiño,
- José M. Prieto,
- Domingo Pérez,
- María Otano,
- Francisco Padilla,
- Juan A. García-Merino,
- Laura Navarro,
- Alfonso Muriel,
- Lucienne Costa Frossard,
- Luisa M. Villar
Affiliations
- Inmaculada Toboso
- Immunology Department, Hospital Universitario Ramon y Cajal, Madrid, Spain
- Amalia Tejeda-Velarde
- Immunology Department, Hospital Universitario Ramon y Cajal, Madrid, Spain
- Roberto Alvarez-Lafuente
- Instituto de Investigación Sanitaria San Carlos (IDISSC), Hospital Clinico San Carlos, Madrid, Spain
- Rafael Arroyo
- Department of Neurology, Hospital Universitario Quiron Salud, Madrid, Spain
- Harald Hegen
- Department of Neurology, Medical University of Innsbruck, Innsbruck, Austria
- Florian Deisenhammer
- Department of Neurology, Medical University of Innsbruck, Innsbruck, Austria
- Susana Sainz de la Maza
- Neurology Department, Hospital Universitario Ramon y Cajal, Madrid, Spain
- José C. Alvarez-Cermeño
- Neurology Department, Hospital Universitario Ramon y Cajal, Madrid, Spain
- Guillermo Izquierdo
- Neurology Department, Hospital Universitario Virgen Macarena, Sevilla, Spain
- Dolores Paramo
- Neurology Department, Hospital Universitario Virgen Macarena, Sevilla, Spain
- Pedro Oliva
- Neurology Department, Hospital Universitario Central de Asturias, Oviedo, Spain
- Bonaventura Casanova
- Neurology Department, Hospital Universitario la Fe, Valencia, Spain
- Eduardo Agüera-Morales
- Neurology Department, Hospital Universitario Reina Sofia, Cordoba, Spain
- Diego Franciotta
- 0Istituti di Recovero e Cura a Carattere Scientifico (IRCCS) Mondino Foundation, Pavia, Italy
- Matteo Gastaldi
- 0Istituti di Recovero e Cura a Carattere Scientifico (IRCCS) Mondino Foundation, Pavia, Italy
- Oscar Fernández
- 1Neurology Department, Hospital Regional Universitario, Malaga, Spain
- Patricia Urbaneja
- 1Neurology Department, Hospital Regional Universitario, Malaga, Spain
- José M. Garcia-Dominguez
- 2Neurology Department, Hospital General Universitario Gregorio Marañón, Madrid, Spain
- Fernando Romero
- 2Neurology Department, Hospital General Universitario Gregorio Marañón, Madrid, Spain
- Alicia Laroni
- 3University of Genoa, Ospedale Policlinico San Martino, Genoa, Italy
- Antonio Uccelli
- 3University of Genoa, Ospedale Policlinico San Martino, Genoa, Italy
- Angel Perez-Sempere
- 4Neurology Department, Hospital General Universitario de Alicante, Alicante, Spain
- Albert Saiz
- 5Neurology Service, Hospital Clinic and Institut d'Investigacions Biomèdiques August Pi i Sunyer (IDIBAPS), Universitat de Barcelona, Barcelona, Spain
- Yolanda Blanco
- 5Neurology Service, Hospital Clinic and Institut d'Investigacions Biomèdiques August Pi i Sunyer (IDIBAPS), Universitat de Barcelona, Barcelona, Spain
- Daniela Galimberti
- 6Centro Dino Ferrari, Fondazione Ca' Granda, Istituti di Recovero e Cura a Carattere Scientifico (IRCCS) Ospedale Policlinico, University of Milan, Milan, Italy
- Elio Scarpini
- 6Centro Dino Ferrari, Fondazione Ca' Granda, Istituti di Recovero e Cura a Carattere Scientifico (IRCCS) Ospedale Policlinico, University of Milan, Milan, Italy
- Carmen Espejo
- 7Servei de Neurologia-Neuroimmunologia, Centre d'Esclerosi Múltiple de Catalunya, Vall d'Hebron Institut de Recerca, Hospital Universitari Vall d'Hebron, Universitat Autònoma de Barcelona, Barcelona, Spain
- Xavier Montalban
- 7Servei de Neurologia-Neuroimmunologia, Centre d'Esclerosi Múltiple de Catalunya, Vall d'Hebron Institut de Recerca, Hospital Universitari Vall d'Hebron, Universitat Autònoma de Barcelona, Barcelona, Spain
- Ludwig Rasche
- 8Department of Neurology, NeuroCure Clinical Research Center, Charité—Universitätsmedizin Berlin, Corporate Member of Freie Universität Berlin, Humboldt-Universität zu Berlin, Berlin Institute of Health, Berlin, Germany
- Friedemann Paul
- 8Department of Neurology, NeuroCure Clinical Research Center, Charité—Universitätsmedizin Berlin, Corporate Member of Freie Universität Berlin, Humboldt-Universität zu Berlin, Berlin Institute of Health, Berlin, Germany
- Friedemann Paul
- 9Experimental and Clinical Research Center, Charité—Universitätsmedizin Berlin, Max Delbrück Center for Molecular Medicine, Berlin, Germany
- Inés González
- 0Neurology Department, Hospital Alvaro Cunqueiro, Vigo, Spain
- Elena Álvarez
- 0Neurology Department, Hospital Alvaro Cunqueiro, Vigo, Spain
- Cristina Ramo
- 1Neurology Department, Hospital Germans Trias i Pujol, Badalona, Spain
- Ana B. Caminero
- 2Neurology Department, Hospital Nuestra Señora de Sonsoles, Avila, Spain
- Yolanda Aladro
- 3Neurology Department, Hospital Universitario Getafe, Getafe, Spain
- Carmen Calles
- 4Neurology Department, Hospital Universitario Son Espases, Palma de Mallorca, Spain
- Pablo Eguía
- 5Neurology Department, Hospital Doctor Jose Molina Orosa, Arrecife, Spain
- Antonio Belenguer-Benavides
- 6Neurology Department, Hospital General Universitario de Castellón, Castellón, Spain
- Lluis Ramió-Torrentà
- 7Neurology Department, Hospital Universitario Doctor Josep Trueta, Girona, Spain
- Ester Quintana
- 7Neurology Department, Hospital Universitario Doctor Josep Trueta, Girona, Spain
- José E. Martínez-Rodríguez
- 8Neurology Department, Hospital del Mar, Barcelona, Spain
- Agustín Oterino
- 9Neurology Department, Hospital Universitario Marqués de Valdecilla, Santander, Spain
- Carlos López de Silanes
- 0Neurology Department, Hospital Universitario de Torrejón, Torrejón de Ardoz, Spain
- Luis I. Casanova
- 0Neurology Department, Hospital Universitario de Torrejón, Torrejón de Ardoz, Spain
- Lamberto Landete
- 1Neurology Department, Hospital Universitario Dr. Peset, Valencia, Spain
- Jette Frederiksen
- 2Glostrup Hospital, University of Copenhagen, Copenhagen, Denmark
- Gabriel Bsteh
- Department of Neurology, Medical University of Innsbruck, Innsbruck, Austria
- Patricia Mulero
- 7Servei de Neurologia-Neuroimmunologia, Centre d'Esclerosi Múltiple de Catalunya, Vall d'Hebron Institut de Recerca, Hospital Universitari Vall d'Hebron, Universitat Autònoma de Barcelona, Barcelona, Spain
- Manuel Comabella
- 7Servei de Neurologia-Neuroimmunologia, Centre d'Esclerosi Múltiple de Catalunya, Vall d'Hebron Institut de Recerca, Hospital Universitari Vall d'Hebron, Universitat Autònoma de Barcelona, Barcelona, Spain
- Miguel A. Hernández
- 3Neurology Department, Hospital Universitario Nuestra Señora de Candelaria, Tenerife, Spain
- Mercedes Espiño
- Immunology Department, Hospital Universitario Ramon y Cajal, Madrid, Spain
- José M. Prieto
- 4Neurology Department, Hospital Clínico de Santiago, Santiago de Compostela, Spain
- Domingo Pérez
- 5Neurology Department, Hospital del Bierzo, Ponferrada, Spain
- María Otano
- 6Neurology Department, Complejo Hospitalario de Navarra, Pamplona, Spain
- Francisco Padilla
- 7Neurology Department, Hospital Clinico de Malaga, Malaga, Spain
- Juan A. García-Merino
- 8Neurology Department, Hospital Puerta de Hierro, Majadahonda, Madrid, Spain
- Laura Navarro
- 9Neurology Department, Hospital General de Elche, Elche, Spain
- Alfonso Muriel
- 0Biostatistics Unit, Hospital Univesitario Ramon y Cajal, Instituto Ramon y Cajal para la Investigación Sanitaria (IRYCIS), Madrid, Spain
- Lucienne Costa Frossard
- Neurology Department, Hospital Universitario Ramon y Cajal, Madrid, Spain
- Luisa M. Villar
- Immunology Department, Hospital Universitario Ramon y Cajal, Madrid, Spain
- DOI
- https://doi.org/10.3389/fneur.2020.579438
- Journal volume & issue
-
Vol. 11
Abstract
Overview: We assessed the role of age and disease activity as new factors contributing to establish the risk of progressive multifocal leucoencephalopathy in multiple sclerosis patients treated with natalizumab in 36 University Hospitals in Europe. We performed the study in 1,307 multiple sclerosis patients (70.8% anti-John Cunninghan virus positive antibodies) treated with natalizumab for a median time of 3.28 years. Epidemiological, clinical, and laboratory variables were collected. Lipid-specific IgM oligoclonal band status was available in 277 patients. Factors associated with progressive multifocal leucoencephalopathy onset were explored by uni- and multivariate logistic regression.Results: Thirty-five patients developed progressive multifocal leucoencephalopathy. The multivariate analysis identified anti-John Cunninghan virus antibody indices and relapse rate as the best predictors for the onset of this serious opportunistic infection in the whole cohort. They allowed to stratify progressive multifocal leucoencephalopathy risk before natalizumab initiation in individual patients [area under the curve (AUC) = 0.85]. The risk ranged from <1/3,300 in patients with anti-John Cunninghan virus antibody indices <0.9 and relapse rate >0.5, to 1/50 in the opposite case. In patients with lipid-specific IgM oligoclonal bands assessment, age at natalizumab onset, anti-John Cunninghan virus antibody indices, and lipid-specific IgM oligoclonal band status predicted progressive multifocal leucoencephalopathy risk (AUC = 0.92). The absence of lipid-specific IgM oligoclonal bands was the best individual predictor (OR = 40.94). The individual risk ranged from <1/10,000 in patients younger than 45 years at natalizumab initiation, who showed anti John Cunningham virus antibody indices <0.9 and lipid-specific IgM oligoclonal bands to 1/33 in the opposite case.Conclusions: In a perspective of personalized medicine, disease activity, anti-lipid specific IgM oligoclonal bands, anti Jonh Cunninghan virus antibody levels, and age can help tailor natalizumab therapy in multiple sclerosis patients, as predictors of progressive multifocal leucoencephalopathy.
Keywords
- multiple sclerosis
- demyelinating diseases
- biomarkers
- natalizumab
- progressive multifocal leucoencephalopathy
- disease modifying treatments