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postnatal days 11 and 15, within the cortex and/or hippocampus

(Fig. 4B).

The percentage of mice showing electroclinical seizures reached

a peak at postnatal day 10, and then a second peak at postnatal

day 14 (Fig. 4F). Neither the frequency nor the duration of elec-

troclinical seizures changed appreciably with age. Seizure fre-

quency ﬂuctuated with a mean frequency of 1.6  0.6 ictal

events per hour at postnatal day 14 (Fig. 4F). The mean duration

of seizures for animals over all recorded ages was 120  12 s

(Fig. 4G).

Homozygous LGI1

/

mice die

prematurely

All homozygous LGI1

/

mice died prematurely, and Kaplan–Meier

curves revealed a mean lifetime of 16 days (n = 25; SD = 1.8).

500 ms

300 μV

LGI1-/-

onset

1.5 sec

500 μV

300 μV

500 ms

500 μV

1.5 sec

onset

end

LGI1-/-

LGI1+/-

1.5 sec

500 μV

1.5 sec

500 μV

Wild-type

Figure 3 Video–EEG recordings of LGI1

/

, LGI1

+/

and wild-type mice. (A) Epidural EEG recording in a postnatal day 10 LGI1

/

mouse showing the onset of an electroclinical seizure (upper trace corresponds to the right cortex and bottom trace to the left cortex).

Behavioural modiﬁcations were correlated with EEG changes: 1 = immobility; 2 = repeated clonies of the four limbs, agitation;

3 = myoclonic jerks of the trunk. In the lower panel, expanded EEG traces show low voltage fast activities (LVFA) following spikes,

and bursts of polyspikes (PS) with increasing duration. No EEG abnormality was seen in age-matched LGI1

+/

(B) or wild-type (C) mice.

(D) Epidural EEG recording in a postnatal day 10 LGI1

/

mouse. In the lower panel, expanded EEG traces show initial spikes discharge

with increasing amplitude, and then pseudo-periodic slow potentials with over-imposed polyspike activity. Note the asymmetry at the end

of the seizure.

2754 | Brain 2010: 133; 2749–2762 E. Chabrol et al.

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