Encephalo-Arterio-Synangiosis with Cranioplasty after Treatment of Acute Subdural Hematoma Associated with Subcortical Hemorrhage Due to Unilateral Moyamoya Disease

Abstract

Moyamoya disease is often diagnosed after intracranial hemorrhage in adult patients. Here, we report a case of unilateral moyamoya disease treated with indirect revascularization combined with cranioplasty after treatment for acute subdural hematoma and subcortical hemorrhage. A middle-aged woman with disturbed consciousness was transferred to our hospital. Computed tomography (CT) revealed an acute subdural hematoma with left temporoparietal subcortical hemorrhage. Three-dimensional CT angiography indicated a scarcely enhanced left middle cerebral artery (MCA) that was suspected to be delayed or nonfilling due to increased intracranial pressure. Subsequently, hematoma evacuation and external decompression were performed. Postoperative digital subtraction angiography (DSA) revealed stenosis of the left MCA and moyamoya vessels, indicating unilateral moyamoya disease. Forty-five days after the initial procedure, we performed encephalo-arterio-synangiosis (EAS) using the superficial temporal artery simultaneously with cranioplasty for the skull defect. The modified Rankin Scale score of the patient one year after discharge was 1, and the repeat DSA showed good patency of the EAS. Revascularization using EAS in the second step can be an option for revascularization for hemorrhagic moyamoya disease if the patient required cranioplasty for postoperative skull defect after decompressive craniotomy.

Figure 1

Preoperative radiological examinations. (a) Computed tomography (CT) showed subcortical temporoparietal hemorrhage (arrow) and subdural hematoma (arrowheads) with midline shift of the cerebrum as well as uncal herniation. (b) Three-dimensional CT angiography indicated scarcely enhanced the left middle cerebral artery (arrow heads): (left) anterior-posterior and (right) posterior-anterior view.

Figure 2

Images of the initial procedure. (a) Photograph showing a huge subdural hematoma after opening the dura mater. (b) Photograph after evacuation of the hematoma showing a swollen brain surface. (c) Intraoperative microscopic photograph showing the left internal carotid artery (arrow) and M1 segment of the hypoplastic left middle cerebral artery (MCA) (arrowheads). (d) Intraoperative microscopic showing a narrow M2 segment of the hypoplastic left MCA (arrowheads). (e) Computed tomography immediately after surgery, confirming adequate decompression and removal of the hematoma.

Figure 3

Images after initial procedure. (a) Digital subtraction angiography (DSA) of the right common carotid artery (CCA) after the initial surgery showing no remarkable abnormality. (b) DSA of the left CCA showed stenosis of the middle cerebral artery and moyamoya vessels (arrowheads). (c) Intraoperative photograph demonstrating encephalo-arterio-synangiosis between the brain surface and the parietal branch of the superficial temporal artery (STA) (arrowheads). (d) Three-dimensional computed tomography after the second surgery showing the structure of the implanted bone flap and STA.

Figure 4

Radiological findings after the second surgery. (a) Repeat magnetic resonance angiography showing the patent vessel of encephalo-arterio-synangiosis (arrow). The patent superficial temporal artery (STA) (arrow) and revascularized M4 segment of the middle cerebral artery (MCA) (arrow heads) were confirmed by digital subtraction angiography (DSA) of the external carotid artery (ECA); (b) left anterior-oblique and (c) lateral view. Three-dimensional DSA of the ECA verified good patency of the parietal branch of the STA (arrow) and the collateralized MCA (arrow heads); (d) left anterior-oblique view.