Cerebral hyperperfusion syndrome (CHS) is a failure of autoregulation after a revascularization procedure. It has rarely been reported in patients with no pre-existing cerebral hypoperfusion.
We present a rare case of a patient who underwent stent graft implantation to treat postirradiated carotid blowout syndrome. The patient developed hypertension, focal neurological deficit, and seizures after the procedure; neuroimages revealed ipsilateral cerebral edema, swelling, and increased cerebral perfusion. CHS was diagnosed based on clinical and radiological findings. The patient recovered gradually after receiving supportive care.
Owing to the risk of CHS, monitoring for cerebral perfusion and prevention of hypertension is suggested for patients who undergo stent graft placement for postirradiated carotid blowout syndrome.
Cerebral hyperperfusion syndrome (CHS) is an uncommon complication experienced by patients who have undergone revascularization for neurovascular disorders. CHS is most often reported in patients who have undergone carotid endarterectomy or carotid artery stenting, although it has also been reported in patients who have undergone other intracranial procedures [
A 66-year-old male patient presented with bloody discharge from a left neck wound for 3 days. The patient had a history of nasopharyngeal cancer diagnosed 16 years prior and had received concurrent chemoradiotherapy and left neck dissection for the initial and recurrent disease. The patient’s neck was heavily treated. He received 70 Gy in the left upper neck, 50 Gy in the right upper neck, and 116 Gy in the bilateral lower neck. Neoplastic disease was reported to be stable for the preceding 11 years. However, extensive radiation necrosis of the neck soft tissue occurred 6 years prior to the patient’s presentation at our clinic. Twelve months before presentation, the patient underwent total laryngectomy, esophagectomy, C6 vertebral corpectomy, and pectoralis major myocutaneous flap reconstruction owing to pharyngocutaneous fistula and deep neck infection. Poor healing and infection of the patient’s chronic neck wound developed several times thereafter. The patient was admitted to our hospital for antibiotic therapy.
Sudden-onset massive left neck wound bleeding occurred 3 days after admission. An otolaryngologist on duty immediately applied epinephrine gauze packing to the wound. On examination, high blood pressure (212/130 mmHg) was noted. Emergency computed tomography angiography (CTA) revealed a pseudoaneurysm in the left distal common carotid artery (
CBS is a life-threatening condition requiring emergency management [
Conceptually, the diagnosis of CHS is based on elevation of cerebral blood flow in the affected territory. CHS is commonly diagnosed based on clinical and radiological features because there is no consensus on the diagnostic criteria for CHS [
Although impaired cerebral autoregulation is the mechanism most commonly presumed to underlie the pathophysiology of CHS, other theories have been proposed. Injury from free radicals, baroreflex breakdown, and abnormal trigeminovascular reflux may also play a role in the development of CHS [
CHS can occur after stent graft placement in the postirradiated neck without luminal stenosis. Practitioners must be familiar with the presentation and diagnosis of CHS and should carefully monitor the cerebral perfusion and systemic blood pressure of patients with this condition.
This study was approved by the Research Ethics Committee of the National Taiwan University Hospital (No. RIND9561703046). The requirement for written informed consent was waived.
No potential conflict of interest relevant to this article.
Conceptualization: YHL. Data curation: CWL, TWL. Formal analysis: YCH. Methodology: CWL, YCH, TWL. Visualization: YHL. Writing–original draft: YCH. Writing–review & editing: YCH, YHL.
Left carotid blowout syndrome diagnosed using preoperative computed tomography angiography. (A) Axial computed tomography (CT) revealing irregular focal protrusion of the left common carotid artery (arrow). (B) Sagittal reformatted CT depicting the location of a pseudoaneurysm (arrow) in relation to the surrounding free flap. No obvious stenosis noted in the carotid artery.
Endovascular treatment for carotid blowout syndrome. (A) Lateral view of the left common carotid revealing a pseudoaneurysm of the left distal common carotid artery (black arrow) corresponding to the computed tomography angiography finding. (B, C) After placement of a stent graft (white arrows) and coils (white arrowhead), the pseudoaneurysm was obliterated. The diameter of the carotid artery did not change considerably after the procedure.
Postoperative neuroimaging findings. (A) Non-contrast computed tomography (CT) revealing substantial sulcal effacement at the left frontoparietal lobe, suggestive of cerebral swelling. No frank hypodensity or hemorrhage is observed. (B, C) CT perfusion revealing elevated cerebral blood flow (B) and cerebral blood volume (C) of the left posterior frontal and parietal lobe, and CT angiography revealing dilatation of the left convexity leptomeningeal arteries compared to those in the contralateral right hemisphere. (D, E) Magnetic resonance imaging revealed subcortical hyperintensity on the fluid-attenuated inversion recovery image but not on the diffusion-weighted image, suggestive of vasogenic edema.