Late Intracerebral Hemorrhage After Successful Endovascular Closure of a Carotid-Cavernous Fistula: A Case Report and Updated Review

Abstract

Background and Clinical Significance: Intracerebral hemorrhage (ICH) is a very rare complication following endovascular closure of direct carotid-cavernous fistulas (CCFs). When reported, ICH typically appears within the first 48 h after CCF closure. We performed an extensive literature review, starting from the case of a 48-year-old patient presenting with an intracerebral hemorrhage after CCF closure. Case Presentation: A 48-year-old woman with arterial hypertension developed an intracerebral hemorrhage in the right frontal lobe 12 days after successful closure of a traumatic CCF. The patient exhibited acute neurological deterioration in a previously hypoperfused territory. A narrative review identifies the classical molecular theory of hemodynamic dysregulation, known as Normal Perfusion Pressure Breakthrough (NPPB), as the principal pathophysiological mechanism. Other mechanisms such as oxidative stress, microglial activation, blood-brain barrier disruption, metalloproteinase expression, and possible genetic alterations such as ICA1L variants are also implicated. Conclusions: This case underscores the importance of considering molecular mechanisms in the pathophysiology of delayed post-endovascular treatment of ICH, as well as the need for hemodynamic monitoring and follow-up in patients with vascular comorbidities.

Keywords: carotid-cavernous sinus fistula; endovascular procedures; intracerebral hemorrhage.

Figure 1

Radiological evolution of intracerebral hemorrhage following successful closure of a carotid-cavernous fistula (CCF). (A) Carotid arteriography showing a right-sided CCF with bilateral venous drainage. Note the absence of contrast opacification in the right middle cerebral artery (MCA) and right anterior cerebral artery (ACA), indicating impaired arterial flow prior to treatment. (B) CT angiography (CTA) revealing hyperdense and tortuous venous structures in the right hemisphere, consistent with abnormal venous drainage secondary to the ipsilateral CCF. (C) Immediate post-procedural arteriography acquired after endovascular occlusion of the CCF; arrows highlight the reappearance of the right ACA and MCA, suggesting reestablishment of normal arterial circulation. (D) CTA performed twelve days after CCF closure demonstrates a right frontal lobe intracerebral hemorrhage, reflecting delayed vascular reperfusion injury. (E) Follow-up arteriography one month later shows stable vessel patency and resolution of venous congestion. The asterisk identifies the residual mass effect produced by the hematoma. (R: right, L: left).

Figure 2

Selective angiography from: At the top, an angiographic image depicts the carotid-cavernous fistula prior to treatment. The lower image shows the angiography after treatment, demonstrating the post-intervention vascular status.