BACKGROUND: As a normal variation of the cerebral venous angioarchitecture, developmental venous anomalies (DVAs) represent a rare cause of intraparenchymal hemorrhage. Different from mixed vascular lesions, such as capillary malformations, arteriovenous malformations, arteriovenous fistulas, and thrombosis, here we describe the first case of a ruptured hemodynamic aneurysm associated with a DVA and introduce the first use of hybrid operation for the treatment of a hemorrhagic DVA. METHODS: A 14-year-old girl suffered from sudden onset of headache, aphasia, and left hemiplegia. On the way of her transfer to our center, she suddenly fell into a coma Computed tomography scan demonstrated an enlarged intraparenchymal hemorrhage in the left temporal lobe. Angiography revealed a large frontal DVA with an associated hemodynamic aneurysm. Superselective angiog-raphy of left middle cerebral artery confirmed that the aneurysm was located on the turning site of arteriovenous transition. RESULTS: Considering the large hematoma and the possible occlusion of surrounding collecting veins, we attempted transarterial embolization but were unsuccessful. Hematoma evacuation and aneurysm isolation were performed in a hybrid operation room. Intraoperative angiography was used to confirm the location of the aneurysm and to recheck the result. The patient woke up 1 day later and the symptoms were relieved entirely 1 year later. CONCLUSIONS: Associated aneurysm may be a cause of intraparenchymal hemorrhage in DVAs and routine imaging monitoring is needed. Hybrid operation is a possible treatment for such complicated mixed lesions in DVA, which proved to be safe and effective in this patient.