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Lenticulostriate artery (LSA) aneurysms are uncommon. Here, we report one case of ruptured LSA aneurysm which is related to Moyamoya disease (MMD). Surgical treatment of this aneurysm is challenging because of its deep location and complex neural structures around the LSA. We report one case treated with endovascular Onyx embolization, successfully and review LSA aneurysm associated with MMD.

A Ruptured lenticulostriate artery aneurysm in moyamoya disease treated with Onyx embolization