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Purpose: Plant-derived highly purified cannabidiol (CBD) reduced the frequency of seizures associated with Lennox-Gastaut syndrome (LGS) and Dravet syndrome (DS) and improved the overall condition of patients in placebo-controlled phase 3 clinical trials. Anecdotal reports also suggest a positive effect on nonseizure outcomes. In this study, we aimed to identify, through a caregiver survey which nonseizure outcomes were most likely to change in these patients., Methods: The BEhavior, COgnition, and More with Epidiolex® (BECOME) was a 20-minute, cross-sectional, online survey that was developed with extensive input from caregivers, healthcare professionals, and epilepsy researchers, and was based on questions from validated measures and previously published caregiver reports. US-based caregivers (from Jazz Pharmaceuticals patient/caregiver database) of people with LGS or DS who were treated with CBD (Epidiolex®, 100 mg/mL oral solution) for ≥3 months were asked to compare the past month to the period before CBD initiation and rate their impression of changes using symmetrical Likert scales., Results: A total of 498 caregivers (97% parents) of patients with LGS (80%) or DS (20%) completed the survey. Mean (range) age of patients was 16 (1-73) years, and 52% were male. Patients were taking a median CBD dose of 14 mg/kg/d and median 4 concomitant antiseizure medications. A large proportion of respondents reported improvements in ≥1 survey question for all nonseizure-related domains: alertness, cognition, and executive function (85%); emotional functioning (82%); language and communication (79% in nonverbal patients and 74% in verbal); activities of daily living (51%); sleep (51%); and physical functioning (46%). Respondents reported improvements in seizure-related domains, including overall seizure frequency (85%), overall seizure severity (76%), seizure-free days per week for ≥1 seizure type (67%), and seizure freedom during the past month (16%). The majority of respondents who reported reduction in seizure frequency also reported improvements in nonseizure outcomes domains (51-80%). However, improvements in nonseizure outcomes (18-56%) were also reported in patients who either had no change or worsening of seizure frequency., Conclusions: This survey characterized and quantified caregiver impression of changes in the seizure and nonseizure outcomes in patients taking add-on CBD treatment. Overall, 93% of caregivers reported planning to continue CBD treatment, primarily because of reduced seizure burden but also because of improvements in nonseizure-related outcomes. Despite the limitations that are associated with a retrospective survey-based study design, these results support further evaluation of the effect of CBD treatment on nonseizure outcomes among patients with LGS or DS., Competing Interests: Declaration of Competing Interest ATB received speakers’ fees from BioMarin and advisory panel fees from Biogen, Biohaven, and Encoded. TD-S is the Executive Director of the LGS Foundation and a consultant for the Chan Zuckerberg Initiative and Neurelis. MAM is the Executive Director of the Dravet Syndrome Foundation. SRD has nothing to disclose. NMDL was an employee of Jazz Pharmaceuticals at the time this study was conducted and may hold stock and/or stock options in Jazz Pharmaceuticals, plc. MSP received safety monitoring committee fees from Stoke Therapeutics; consulting fees from BioMarin, Encoded Therapeutics, Greenwich Biosciences (now part of Jazz Pharmaceuticals Inc), Neurelis, Stoke Therapeutics, Taysha, and Zogenix; speakers bureau fees/advisory board fees from Zogenix and Bright Minds; research funds paid to Cook Children’s from Encoded Therapeutics, Greenwich Biosciences (now part of Jazz Pharmaceuticals Inc), Marinus, Ovid, Stoke Therapeutics, and Zogenix; speakers fees from nobelPharma; and advisory board fees from Eisai., (Copyright © 2024 The Authors. Published by Elsevier B.V. All rights reserved.)