Enhancing sudden unexpected death in epilepsy (SUDEP) research through development of common data elements

Abstract

Objective: Sudden Unexpected Death in Epilepsy (SUDEP) is a fatal complication for individuals living with epilepsy and is associated with significant personal and public burden. While certain neurotransmitters and neuronal pathways have been associated with SUDEP, the exact biological mechanisms are unknown. Preclinical research has been instrumental in providing clues to the underlying pathology but is limited by a lack of standardized methodologies for describing and collecting data. A key outcome of the Basic Science working group of the 2020 SUDEP Coalition Summit was the recognition that the development of standardized tools would greatly enhance SUDEP research. Such a research infrastructure would increase experimental rigor, repeatability, reproducibility, and transparency and finally, increase the chances that preclinical SUDEP research can be translated into human SUDEP.

Methods: CURE Epilepsy assembled a Steering Committee and working groups consisting of experts in preclinical and clinical SUDEP research to develop Common Data Elements (CDEs) and Case Report Forms (CRFs) to enable standardization and translation of preclinical SUDEP data. Standardized methodology from the development of other epilepsy-related CDEs was used.

Results: The Core and Death-Related Information CRF constitutes the priority CRF for SUDEP preclinical studies. This CRF gives investigators CDEs to note details of animal models used, experiment-related information, and details about triggered and spontaneous seizures. The seizure-related death information consists of CDEs related to observations at the time of death, characteristics of fatal seizures, the posture of the animal at the time of death, diet, medications, and any adverse health conditions.

Significance: Systematic use of CDEs and CRFs in SUDEP preclinical research can help increase the rigor and transparency of research. Core CDEs along with supplemental CDEs described in the accompanying manuscript can aid investigators and groups working together toward a common goal of preventing SUDEP.

Plain language summary: Sudden Unexpected Death in Epilepsy (SUDEP) is a fatal complication of epilepsy. Preclinical research holds promise in understanding and preventing SUDEP, but its impacts are limited due to a lack of data standardization and translation among research groups. Common data elements (CDEs) are essential pieces of information for a certain field of study. CURE Epilepsy brought together a team of researchers to develop CDEs that could serve as a blueprint for all SUDEP preclinical researchers. This paper describes the SUDEP Core and Death-Related CDEs to be used with data elements presented in an accompanying paper.

Keywords: Core; SUDEP; case report form; common data elements; standardization; translation.

FIGURE 1

Timeline of all SUDEP CDE‐related events in the SUDEP Data Standardization Tools Project.

FIGURE 2

The SUDEP Data Standardization Tools Project with the priority and supplemental CRFs.