Stratification of amyotrophic lateral sclerosis patients: a crowdsourcing approach

  • Robert Kueffner ,
  • Neta Zach ,
  • Maya Bronfeld ,
  • Raquel Norel ,
  • Nazem Atassi ,
  • Venkat Balagurusamy ,
  • Barbara DiCamillo ,
  • Adriano Chio ,
  • Merit Cudkowicz ,
  • Donna Dillenberger ,
  • Orla Hardiman ,
  • Bruce Hoff ,
  • Joshua Knight ,
  • Melanie L. Leitner ,
  • Guang Li ,
  • Lara Mangravite ,
  • Thea Norman ,
  • Liuxia Wang ,
  • The ALS Stratification Consortium (including Lester Mackey) ,
  • Jinfeng Xiao ,
  • Wen-Chieh Fang ,
  • Jian Peng ,
  • Chen Yang ,
  • Huan-Jui Chang ,
  • Gustavo Stolovitzky ,

Scientific Reports |

Amyotrophic lateral sclerosis (ALS) is a fatal neurodegenerative disease where substantial heterogeneity in clinical presentation urgently requires a better stratification of patients for the development of drug trials and clinical care. In this study we explored stratification through a crowdsourcing approach, the DREAM Prize4Life ALS Stratification Challenge. Using data from >10,000 patients from ALS clinical trials and 1479 patients from community-based patient registers, more than 30 teams developed new approaches for machine learning and clustering, outperforming the best current predictions of disease outcome. We propose a new method to integrate and analyze patient clusters across methods, showing a clear pattern of consistent and clinically relevant sub-groups of patients that also enabled the reliable classification of new patients. Our analyses reveal novel insights in ALS and describe for the first time the potential of a crowdsourcing to uncover hidden patient sub-populations, and to accelerate disease understanding and therapeutic development.