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Background: An aplastic or twig-like middle cerebral artery (Ap/T-MCA) is a rare congenital anomaly that can present with both ischemic and hemorrhagic stroke. The etiology of this pathology has remained unclear. Here, we report 2 cases of intracerebral hemorrhage (ICH) owing to an Ap/T-MCA in pregnant patients. Case Description: In both patients, cerebral angiography revealed a steno-occlusive lesion and an abnormal arterial network on the unilateral middle cerebral artery. One patient was treated conservatively for a putaminal hemorrhage, and a cesarean section was performed uneventfully 6 months after onset of the ICH. The other patient underwent a craniotomy for evacuation of the lobar hemorrhage. Subsequently, a cesarean section was performed uneventfully. Both patients gradually recovered without significant disabilities. Conclusions: An Ap/T-MCA is a rare congenital anomaly and is a potential cause of ICH for pregnant patients. A cesarean section is a useful option for pregnant patients with this condition. Key words: Cesarean section, Intracerebral hemorrhage, Middle cerebral artery, Moyamoya disease, Pregnancy, Twig-like MCA