Figure 5.

Vitamin B6-responsive epilepsy in plpbp^−/− zebrafish larvae. Survival in mutants was moderately improved using PLP (A) but showed a better response that was clearly dose-dependent with pyridoxine (B). (C) Five-minute trace recordings of 11 dpf zebrafish larvae showing increased hyperactivity in the mutants which was alleviated with 10 mM pyridoxine treatment, as measured by (D) time spent in fast movements and (E) distance moved in fast movements. (F) Highest seizure-like behaviour category identified by blinded observers. Only untreated mutant larvae showed evidence of S2 or S3 seizure-like activity. (G) Electrographic activity in mutant larvae was normalized by treatment with 5 mM pyridoxine. (H) Example electrophysiology recordings of untreated and treated mutant larvae. Significance: **P < 0.01, *P < 0.05. PN = pyridoxine; WT = wild-type.