Long-term disability trajectories in primary progressive MS patients: A latent class growth analysis

Alessio Signori, Guillermo Izquierdo, Alessandra Lugaresi, Raymond Hupperts, Francois Grand’Maison, Patrizia Sola, Dana Horakova, Eva Havrdova, Alexandre Prat, Marc Girard, Pierre Duquette, Cavit Boz, Pierre Grammond, Murat Terzi, Bhim Singhal, Raed Alroughani, Thor Petersen, Cristina Ramo, Celia Oreja-Guevara, Daniele SpitaleriVahid Shaygannejad, Helmut Butzkueven, Tomas Kalincik, Vilija Jokubaitis, Mark Slee, Ricardo Fernandez Bolaños, Jose Luis Sanchez-Menoyo, Eugenio Pucci, Franco Granella, Jeannette Lechner-Scott, Gerardo Iuliano, Stella Hughes, Roberto Bergamaschi, Bruce Taylor, Freek Verheul, Maria Edite Rio, Maria Pia Amato, Seyed Aidin Sajedi, Nastaran Majdinasab, Vincent Van Pesch, Maria Pia Sormani, Maria Trojano

Research output: Contribution to journalArticle

Abstract

Background: Several natural history studies on primary progressive multiple sclerosis (PPMS) patients detected a consistent heterogeneity in the rate of disability accumulation. Objectives: To identify subgroups of PPMS patients with similar longitudinal trajectories of Expanded Disability Status Scale (EDSS) over time. Methods: All PPMS patients collected within the MSBase registry, who had their first EDSS assessment within 5 years from onset, were included in the analysis. Longitudinal EDSS scores were modeled by a latent class mixed model (LCMM), using a nonlinear function of time from onset. LCMM is an advanced statistical approach that models heterogeneity between patients by classifying them into unobserved groups showing similar characteristics. Results: A total of 853 PPMS (51.7% females) from 24 countries with a mean age at onset of 42.4 years (standard deviation (SD): 10.8 years), a median baseline EDSS of 4 (interquartile range (IQR): 2.5–5.5), and 2.4 years of disease duration (SD: 1.5 years) were included. LCMM detected three different subgroups of patients with a mild (n = 143; 16.8%), moderate (n = 378; 44.3%), or severe (n = 332; 38.9%) disability trajectory. The probability of reaching EDSS 6 at 10 years was 0%, 46.4%, and 81.9% respectively. Conclusion: Applying an LCMM modeling approach to long-term EDSS data, it is possible to identify groups of PPMS patients with different prognosis.

Original languageEnglish
Pages (from-to)642-652
Number of pages11
JournalMultiple Sclerosis Journal
Volume24
Issue number5
DOIs
Publication statusPublished - Apr 1 2018

Keywords

  • clinical trials
  • disability
  • heterogeneity
  • long-term
  • Primary progressive multiple sclerosis
  • trajectories

ASJC Scopus subject areas

  • Neurology
  • Clinical Neurology

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    Signori, A., Izquierdo, G., Lugaresi, A., Hupperts, R., Grand’Maison, F., Sola, P., Horakova, D., Havrdova, E., Prat, A., Girard, M., Duquette, P., Boz, C., Grammond, P., Terzi, M., Singhal, B., Alroughani, R., Petersen, T., Ramo, C., Oreja-Guevara, C., ... Trojano, M. (2018). Long-term disability trajectories in primary progressive MS patients: A latent class growth analysis. Multiple Sclerosis Journal, 24(5), 642-652. https://doi.org/10.1177/1352458517703800