PRIMARY STUDY

Case reports of identical twins with developmental and epileptic encephalopathy with STXBP1 gene mutations for whom different CBD supplementations were markedly effective

Key Findings:  This case report of a set of twins with developmental and epileptic encephalopathy with STXBP1 gene mutation who both responded exceptionally well to two different add-on regimens of cannabidiol (CBD).

Type of Study:  Clinical Trial

Study Sample Size:  2

Study Result:  Positive


Cannabinoids Studied:  Cannabichromene (CBC), Cannabidiol (CBD), Cannabigerol (CBG), Cannabinol (CBN), Cannabichromenic Acid (CBC-a), Cannabigerolic Acid (CBG-a), Pharma CBD, Tetrahydrocannabivarin (THCV), Cannabitriol (CBT)

Phytocannabinoid Source:  Not Applicable

Chemotype:  Chemotype III

Terpenes Studied:  ß-Caryophyllene, Bisabolol

Receptors Studied:  GPCR 55

Ligands Studied:  GABA

DOSING DETAILS   

Study Dosing Objective:  Effective Dose

Established Protocol:  Effective dose

Route of Administration:  Oral (Ingestion), Sublingual/Oromucosal

Cannabinoid Ratio:  (CBD : Pharma CBD)   1 : 1    

Dosage Form:  CBD Supplement (1): 993.50mg/g 99.35% CBD Supplement (2): 704.30mg/g 70.43%

Starting Dose:  Twin 1: CBD supplement at 4 mg/kg/day Twin 2: CBD supplement at 2 mg/kg/day

Titration:  Twin 2: titrated up weekly by 2 mg/kg/day

Maximum Dose:  Twin 1: 400 mg/day Twin 2: 600 mg/day

Clinical Relevance:  Monozygotic twins achieved seizure suppression with CBD supplements containing different ingredients.

Additional Notes:  Parallel Dose Comparison, Dose Titration, Dose Escalation


Link to study

Authors:  Masataka Y, Miki N, Akino K, Yamamoto H, Takumi I

Journal:  Case reports of identical twins with developmental and epileptic encephalopathy with STXBP1 gene mutations for whom different CBD supplementations were markedly effective.

Year of Pub:  2024

Research Location(s):  Japan

Citation:  Masataka Y, et al. Case reports of identical twins with developmental and epileptic encephalopathy with STXBP1 gene mutations for whom different CBD supplementations were markedly effective. Epilepsy Behav Rep. 2024; 28:100720. doi: 10.1016/j.ebr.2024.100720