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March 27, 2026Therapeutic Advances in Rare Disease0 citationsOpen Access

Could an outcome-based agreement be operationalized using real-world data from the Canadian Neuromuscular Disease Registry? Perspectives from an expert-led assessment in spinal muscular atrophy

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AMArif MithaVHVictoria HodgkinsonSWSusi Vander Wyk

Key Points

  • This research aims to identify suitable SMA health outcomes for an OBA and evaluate CNDR data collection processes.
  • Conducted expert-led focus group discussions with selected participants.
  • Evaluated eight SMA health outcomes for suitability in OBA using criteria like data readiness and interpretability.
  • Assessed CNDR data tracking processes to identify gaps and propose solutions.
  • Collected stakeholder feedback to validate findings and insights.
  • Five SMA health outcomes were identified as suitable for OBA.
  • Three process gaps in CNDR were identified with proposed solutions.
  • A future process flow for CNDR was developed to enhance RWE generation for OBA.

Abstract

Background: Outcome-based agreements (OBAs) may facilitate earlier patient access to promising therapies, particularly when evidence is limited. The authors of this paper investigated how to operationalize an OBA using real-world data (RWD) from the Canadian Neuromuscular Disease Registry (CNDR). Objective: The first objective of this research was to determine which spinal muscular atrophy (SMA) health outcomes in the CNDR are suitable for an OBA. The second objective was to evaluate the current process of data collection in the CNDR and explore how to operationalize data processes to support an OBA, including identifying gaps and proposing solutions. Design: This qualitative expert-led assessment was conducted through a series of focus group discussions with selected experts. Methods: A selected group of experts participated in 17 focus group meetings. The ability of the CNDR to generate real-world evidence (RWE) for an OBA was evaluated for eight SMA health outcomes. The criteria used were data readiness, interpretability, and timeframe within the CNDR. Next, the processes involved were evaluated, specifically to determine how data tracking within the CNDR could be operationalized for an OBA, including identifying gaps and possible solutions. Based on the findings, the group proposed a future state for an OBA process using the CNDR. The group followed up with stakeholder feedback interviews to validate the findings of this research and to gather insights. Results: Five SMA outcomes within the CNDR were identified as potentially suitable outcomes for OBA. Three process gaps were identified in the current state of the CNDR, and corresponding solutions were proposed. A proposed future-state process flow for CNDR was developed to support RWE generation for OBA. Conclusions: Expert consultations suggest that operationalizing an OBA using CNDR RWD is feasible.

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Cite This Study

Mitha et al. (2026) studied this question.

synapsesocial.com/papers/69c620be15a0a509bde194cehttps://doi.org/10.1177/26330040261433035
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