Together with the observation of a median overall survival (OS) of more than 12 months in this heavily pretreated cohort since the start of lenvatinib therapy (Fig. 1c), these data suggest a clinically meaningful response to the treatment with lenvatinib in FGFR2-driven iCCA, even with daily doses of 12 mg or less (Fig. 1a, b). Here, FGFR2 is linked to infantile convulsions and choreoathetosis.