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Peer-reviewedCC BYCNS neuroscience & therapeutics

Long-Term Drop-Seizure Response Patterns to Cannabidiol According to Genetic Status in Lennox-Gastaut Syndrome.

  • Ji-Hoon Na1
  • Hee Yeon Cho1
  • Hayoon Jeon1
  • Hyunjoo Lee1
  • Young-Mock Lee1
  1. 1Department of Pediatrics, Division of Pediatric Neurology, Gangnam Severance Hospital, Yonsei University College of Medicine, Seoul, Republic of Korea.
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Abstract

Background

Lennox-Gastaut syndrome (LGS) is a genetically heterogeneous developmental and epileptic encephalopathy in which variability in long-term cannabidiol (CBD) response remains incompletely understood. We evaluated whether long-term drop-seizure response trajectories to CBD differ according to genetic status.Methods

This single-center retrospective longitudinal study analyzed systematically collected clinical follow-up data from 61 patients with LGS treated with pharmaceutical-grade CBD; 46 patients with ≥ 24 months of follow-up were included. Patients were classified as pathogenic/likely pathogenic variant-positive (PV+) or variant-negative (PV-). Drop-seizure reduction trajectories were assessed at 3-month intervals over 24 months.Results

Drop-seizure reduction improved rapidly and was sustained over 24 months in both PV+ and PV- groups, with no significant differences between groups at the prespecified comparison time points. Longitudinal response trajectories showed substantial overlap, and sustained response was observed in 76.1% of patients.Conclusion

Long-term drop-seizure response to CBD in LGS was sustained but not stratified by genetic status, supporting a syndrome-level treatment approach irrespective of genetic findings.

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Cite

BibTeXRIS
BibTeX
@article{na2026longterm,
  title = {{Long-Term Drop-Seizure Response Patterns to Cannabidiol According to Genetic Status in Lennox-Gastaut Syndrome.}},
  author = {Ji-Hoon Na and Hee Yeon Cho and Hayoon Jeon and Hyunjoo Lee and Young-Mock Lee},
  journal = {CNS neuroscience \& therapeutics},
  year = {2026},
  month = nov,
  doi = {10.1002/cns.71221},
  url = {https://europepmc.org/article/MED/42968542},
  note = {CC BY},
}
RIS
TY  - JOUR
TI  - Long-Term Drop-Seizure Response Patterns to Cannabidiol According to Genetic Status in Lennox-Gastaut Syndrome.
AU  - Ji-Hoon Na
AU  - Hee Yeon Cho
AU  - Hayoon Jeon
AU  - Hyunjoo Lee
AU  - Young-Mock Lee
PY  - 2026
DA  - 2026/11/01
JO  - CNS neuroscience & therapeutics
DO  - 10.1002/cns.71221
UR  - https://europepmc.org/article/MED/42968542
AB  - Background  Lennox-Gastaut syndrome (LGS) is a genetically heterogeneous developmental and epileptic encephalopathy in which variability in long-term cannabidiol (CBD) response remains incompletely understood. We evaluated whether long-term drop-seizure response trajectories to CBD differ according to genetic status.Methods  This single-center retrospective longitudinal study analyzed systematically collected clinical follow-up data from 61 patients with LGS treated with pharmaceutical-grade CBD; 46 patients with ≥ 24 months of follow-up were included. Patients were classified as pathogenic/likely pathogenic variant-positive (PV+) or variant-negative (PV-). Drop-seizure reduction trajectories were assessed at 3-month intervals over 24 months.Results  Drop-seizure reduction improved rapidly and was sustained over 24 months in both PV+ and PV- groups, with no significant differences between groups at the prespecified comparison time points. Longitudinal response trajectories showed substantial overlap, and sustained response was observed in 76.1% of patients.Conclusion  Long-term drop-seizure response to CBD in LGS was sustained but not stratified by genetic status, supporting a syndrome-level treatment approach irrespective of genetic findings.
SN  - 1755-5930
SN  - 1755-5949
ER  -