2021
DOI: 10.21203/rs.3.rs-1167543/v1
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Variation in the Accuracy of Endpoint Selection in Clinical Studies for Rare Diseases in Respect of Increasing Knowledge on Disease-Severity Measurement

Abstract: Background: Previous research assessed the accuracy of disease-severity measurement in clinical studies as a mathematical relationship between the set of endpoints selected and the disease-severity scale (DSS), a surrogate for the theoretical Neutral list of indicators representing the disease phenotype. New DSSs are continually developed, so clinical studies’ operationalisation of the Neutral list and resulting relative neutrality may vary over time. We assessed variation in the neutrality of clinical studies… Show more

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