Tentorial dural arteriovenous fistulas are rare and complex lesions in deep locations with unusual vascular anatomy and critical surrounding neuroanatomy. A rare case presenting a complex fistula with a giant venous draining ampulae, causing headaches and visual troubles is presented. We describe the case of a 52-year-old woman admitted in our department for headaches and visual troubles. Magnetic resonance imaging and cerebral angiography showed a tentorial dural arteriovenous fistula draining in a giant tentorial venous ampulae and leptomeningeal veins. The patient was embolised via an arterial route with a good clinical and radiological result. However, 4 days later she presented a sudden change of her clinical status with coma, left hemiparesis and a right midriasis. The cerebral computed tomography scan showed a huge occipital haemorrhagic mass and a severe cerebral oedema. An emergent surgical procedure was decided realising evacuation of the occipital haematoma and a complete resection of the giant venous ampoule. The neck of the ampulae was sutured and clipped at its dural entrance. Postoperatively a new embolisation was realised because of persistent of a small dural fistulae with occipital leptomeningeal drainage. The patient recovered rapidly with only a residual hemianopsy. Treatment of dural AV malformation represent a serious challenge. Our report describes an unusual case of a tentorial dural complex fistula treated by an endovascular procedure with secondary clinical aggravation that needed emergent surgical therapy. Even in a case for good immediate radiological result after endovascular procedure, dural arteriovenous fistulas with giant venous ampulae and important venous engorgement, need closed follow-up, because of the possibility of aggravation secondary to venous thrombosis and haemorrhage. Treatment and patophysiology of the aggravation mechanism are discussed.