Introduction: Dolichoectasia describes the elongation, dilatation and tortuosity of an arterial vessel. In adults, clinical presentation of basilar dolichoectasia (BD) is most often with ischaemic stroke. True BD has been very rarely reported in childhood, but the implication is of a congenital susceptibility.
Case report: We describe two cases of infants with BD complicated by brainstem ischaemia.
Discussion: This first description of childhood BD associated with ischaemic infarction adds to the understanding of this rarely reported arteriopathy.