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Proximal occlusion for infectious carotid-cavernous fistula caused by invasive fungal sinusitis: illustrative case
Akihiro Okada1,2, Takeshi Kawauchi1, Yukinori Terada1
1Department of Neurosurgery, Osaka Red Cross Hospital, Osaka, Osaka, Japan.
Background:
Thirteen cases of fungal cavernous-carotid aneurysms (CCAs) attributable to invasive fungal sinusitis have been reported in the literature. These reports included only 1 case of carotid-cavernous fistula (CCF). In this report, the authors present a case of CCF attributable to the rupture of an infected CCA caused by invasive fungal sinusitis that required proximal occlusion for treatment.
Observations:
A 56-year-old man with poorly controlled diabetes experienced a sudden loss of vision in his left eye. Cranial imaging revealed invasive sphenoid sinusitis and CCF attributable to a ruptured infected CCA, as well as an unruptured infected supraclinoid internal carotid artery (ICA) aneurysm. After 3 weeks of medical treatment, the patient developed sudden impairment of higher function. MRI revealed occlusion of the distal ICA attributable to thrombosis of the supraclinoid ICA aneurysm. The anterior circulation was perfused from the contralateral side via the anterior communicating artery. Proximal occlusion with coils was performed, leading to ablation of the CCF.
Lessons:
This is the first reported case of spontaneous thrombosis of an infected distal cerebral aneurysm, and proximal occlusion with coils was necessary to treat CCF caused by the rupture of a fungal CCA. This case could help guide future treatment. https://thejns.org/doi/10.3171/CASE25336.
Insights
This case study details a rare fungal cavernous-carotid aneurysm (CCA) rupture causing a carotid-cavernous fistula (CCF). Treatment involved proximal occlusion, highlighting a novel approach for this fungal sinusitis complication.
Area of Science:
- Neurology
- Infectious Diseases
- Vascular Surgery
Background:
- Fungal cavernous-carotid aneurysms (CCAs) are rare, with few reported cases linked to invasive fungal sinusitis.
- Carotid-cavernous fistulas (CCFs) are an infrequent complication of fungal CCAs.
Purpose of the Study:
- To report a unique case of CCF resulting from a ruptured fungal CCA due to invasive fungal sinusitis.
- To describe the successful treatment of this rare condition using proximal occlusion.
Main Methods:
- A 56-year-old male with diabetes presented with vision loss and was diagnosed with invasive sphenoid sinusitis, a ruptured infected CCA, and an unruptured supraclinoid internal carotid artery (ICA) aneurysm.
- After medical management, the patient experienced neurological decline due to distal ICA occlusion from aneurysm thrombosis. Treatment involved proximal coil occlusion to ablate the CCF.
Main Results:
- The patient developed CCF from a ruptured fungal CCA and subsequent distal ICA occlusion from aneurysm thrombosis.
- Proximal occlusion with coils successfully ablated the CCF, preserving anterior circulation via collateral pathways.
Conclusions:
- This is the first reported instance of spontaneous thrombosis in an infected distal cerebral aneurysm.
- Proximal occlusion proved effective in managing CCF secondary to a ruptured fungal CCA, offering guidance for future cases.
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