Bilateral internal caroid artery agenesis is a very rare congenital anomaly, which may be accompanied by various types of associated vascular abnormalities, included intracranial aneurysms or dolichoectatic change of posterior circulation. In this article, we present unique, and to the best of our knowledge, the first case of bilateral internal carotid agenesis associated with basilar artery fenestration, which resembles intra-arterial floating thrombus.
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