Successful endovascular treatment of intractable epistaxis due to ...

CASE REPORT

Eben L. Rosenthal, MD, Section Editor

SUCCESSFUL ENDOVASCULAR TREATMENT OF INTRACTABLE EPISTAXIS DUE TO RUPTURED INTERNAL CAROTID ARTERY PSEUDOANEURYSM SECONDARY TO INVASIVE FUNGAL SINUSITIS Shaner-Yeun Jao, MD,1 Hsu-Huei Weng, MD, MPH,1,2,3 Ho-Fai Wong, MD,4 Wen-Hung Wang, MD,5 Yuan-Hsiung Tsai, MD1 1

Department of Diagnostic Radiology, Chang Gung Memorial Hospital, College of Medicine and School of Medical Technology, Chang Gung University, Chiayi, Taiwan. E-mail: [email protected] 2 Department of Respiratory Care, Chang Gung Institute of Technology, Chiayi, Taiwan and Chang Gung Memorial Hospital, College of Medicine and School of Medical Technology, Chang Gung University, Chiayi, Taiwan 3 Institute of Occupational Safety and Health, College of Health Sciences, Kaohsiung Medical University, Kaoshiung, Taiwan and Chang Gung Memorial Hospital, College of Medicine and School of Medical Technology, Chang Gung University, Chiayi, Taiwan 4 Department of Diagnostic Radiology, Chang Gung Memorial Hospital, College of Medicine and School of Medical Technology, Chang Gung University, Linko, Taiwan 5 Department of Otolaryngology–Head & Neck Surgery, Chang Gung Memorial Hospital, College of Medicine and School of Medical Technology, Chang Gung University Chiayi, Taiwan Accepted 16 September 2009 Published online 1 December 2009 in Wiley Online Library (wileyonlinelibrary.com). DOI: 10.1002/hed.21305

Abstract: Background. Mycotic pseudoaneurysm from the cavernous segment of the internal carotid artery (ICA) secondary to an invasive aspergillus sinusitis is rare. Surgical intervention with ICA ligation is generally accepted for most mycotic aneurysms or pseudoaneurysms. When presented with massive epistaxis due to a fungal aspergillus ICA invasion, mortality rates are high. Methods. We present the case of a 76-year-old man who developed intractable epistaxis due to a mycotic pseudoaneurysm arising from the cavernous segment of the right ICA. Results. The patient was successfully treated by endovascular embolization at the orifice of the pseudoaneurysm followed by the total ICA trapping technique using electrolytically Guglielmi detachable coils (GDCs) and injection of N-butyl-2cyanoacrylate (n-BCA). The patient survived for 7 months but eventually died of urosepsis and cardiorespiratory failure. Conclusion. Endovascular embolization is a feasible and lifesaving approach for emergent management of massive intractable epistaxis secondary to a complicated invasive fungal sinusC 2009 Wiley Periodicals, Inc. Head Neck 33: 437–440, 2011 itis. V Keywords: embolization; epistaxis; fungal sinusitis; internal carotid artery; pseudoaneurysm

Epistaxis is a common clinical problem that is usually easily controlled. Patients who fail initial conservCorrespondence to: Y.-H. Tsai This work was partially supported by grant CMRPG660332 from the Chang Gung Medical Research Fund, Chang Gung Memorial Hospital, Chiayi, Taiwan. C 2009 Wiley Periodicals, Inc. V

Epistaxis from Carotid Artery Pseudoaneurysm

ative therapy require endoscopic cauterization, surgical ligation, or transarterial embolization.1 Embolization successfully controls epistaxis in 70% to 96% of cases.2 Pseudoaneurysm of the internal carotid artery (ICA) is an uncommon etiology of epistaxis, arising from conditions including transsphenoidal surgery for tumor or sinus disease, penetrating or blunt trauma, primary and salvage radical neck surgery, mycotic infections, radiation therapy, vessel dissection, head and neck malignancy, and other diseases.3 Mortality for these patients is high without early diagnosis and management. Carotid ligation was first used for those pseudoaneurysms, but, in cases such as infectious mycotic aneurysm and radiation-induced pseudoaneurysm, the tissue plane and anatomy are often destroyed so that surgical intervention is not feasible. Newer endovascular embolization techniques such as endovascular trapping, stentassisted coiling, and stent-graft implantation are commonly used in cases of traumatic or atherosclerotic aneurysms, but a noninvasive, rapid approach can also be used to manage fungal mycotic ICA pseudoaneurysm. Here we describe a rare case of a complicated invasive aspergillus sinusitis presenting with massive, intractable epistaxis secondary to an ICA pseudoaneurysm in the orbital apex, which was successfully treated with endovascular embolization. CASE REPORT

A 76-year-old man with a history of hypertension and well-controlled diabetes was admitted to our institution

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FIGURE 1. Axial CT scan of paranasal sinus showing widening of the right superior ophthalmic fissure (short arrow) and erosion of the lateral wall and roof of the right sphenoid sinus (long arrow). An infiltrative mass occupies the entire right sphenoid sinus, extending into the right orbital apex. Note mild protrusion of the right eye.

because of a progressive, throbbing right temporal headache radiating to the right cheek for 3 months. Physical examination revealed a mild fever, tenderness over the right temporal side, and decreased sensation in the right cheek. There was also mild protrusion of his right eye with right eyeball movement slightly restricted in all directions. A CT scan of the paranasal sinuses disclosed an infiltrative mass in the right posterior ethmoid and sphenoid sinuses with destruction of the roof and lateral wall of the right sphenoidal sinus. The superior orbital fissure and ptergopalatine fossa were widened with extension of the mass into the right orbital apex (Figure 1). The patient underwent functional endoscopic sinus surgery (FESS) with subsequent biopsy of the right paranasal sinuses. A histopathologic report revealed necrotic debris, mixed inflammatory cell infiltration, and fungal spores with branching hyphae at acute angles consistent with aspergillosis. Besides several sinus debridements and combined anti-fungal (amphotericin-B) and anti-bacterial (augmentin) medication, the 8 weeks of hospital stay were uneventful and the patient was eventually discharged with oral antifungal medication. However, after a week, he was readmitted because of a highgrade fever and worsening temporal headache. Intravenous fungal and bacterial antibiotics were resumed, amphotericin-B was changed to voriconazole, and the patient was also given itraconazole. One week later, the patient presented with sudden, severe bulging of his right eye and intermittent epistaxis. An MRI of the orbit and brain showed invasive sinusitis with intracranial involvement. The T1-weighted postgadolinium images showed localized pooling of contrast medium around the right orbital apex (Figure 2). Massive intractable epistaxis eventually followed, despite nasal packing. An emergent angiogram revealed pseudo-

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FIGURE 2. T1-weighted post-gadolinium MR image shows localized pooling of contrast medium attached to a vascular structure, supposedly the ophthalmic artery in the right orbital apex (arrow). An iso-low signal intensity soft tissue mass appears to invade the right orbital apex and right posterior ethmoid sinus, causing a mass effect on the dura in the right middle cranial fossa.

aneurysm formation about 2 cm long arising from the anterior segment of the cavernous right ICA (Figure 3). The right ophthalmic artery was not well visualized and there was also narrowing of the infraclinoid portion of the distal ICA. Ligation of ICA was contemplated, but in the presence of severe infection, endovascular embolization was selected. A balloon test occlusion was performed to assess the risk of stroke following total ICA trapping and was tolerated for 30 minutes without any abnormal neurologic signs. Electrolytically, Guglielmi detachable coils (GDCs; Target Therapeutics, Fremont, CA) placed at the orifice of the pseudoaneurysm with

FIGURE 3. Conventional angiography shows a long fusiform pseudoaneurysm directly arising from the supposed ophthalmic segment of the right internal carotid artery (short arrow). Note the irregular narrowing of the cavernous segment of the right internal carotid artery (long arrow).


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FIGURE 4. MR angiography of brain shows right middle cerebral artery (arrow) with compensatory blood supply, probably from the anterior communicating artery. The right ICA is not opacified.

subsequent embolization of the parent ICA. Further total occlusion of the ICA was achieved with injection of 25% n-BCA (Cordis, Miami Lakes, FL). Epistaxis ceased immediately after total sacrifice of the ICA. An MRI and MR angiography of the brain after 10 days confirmed complete occlusion of the right ICA with good collateral filling of the right middle cerebral artery (Figure 4). Tiny infarcts observed at the ipsilateral borderzone territories were asymptomatic. After 3 weeks, the patient was discharged home with oral voriconazole and other medications. The patient had no further episodes of epistaxis. Aside from intermittent temporal headaches, the patient showed no signs of fever or progressive neurologic deficits during regular follow-up in our outpatient department. However, after 7 months, he was readmitted to our hospital for urosepsis and eventually died of cardiorespiratory failure. DISCUSSION

Fungal sinusitis is classified as either allergic, noninvasive, chronic invasive, or acute fulminant infection.4 The vast majority are noninvasive with a good prognosis. The invasive type is characterized by extension of hyphae through the sinus mucosa and walls, followed by invasive spread into contiguous structures.5 The aspergillus species have an angioinvasive nature due to its ability to produce the enzyme elastase. The elastin in the arterial walls offers resistance to bacterial invasion, but cannot protect against fungi such as aspergillus species.6 The growth of fungal hyphae further contributes to weakening of the vessel wall. Repeated pulsations exploit this weakness and lead to aneurysm formation, which, left alone, can lead to rupture. In most reports, massive epistaxis due to extracranial ICA rupture with pseudoaneurysm formation are secondary to skull base or facial trauma, sinus sur-


geries, and nasal intubation.7 In our case, the massive epistaxis resulted from the aggressive aspergillus organism causing bony erosion and arterial wall rupture. This occurred despite combined anti-fungal and anti-bacterial treatment. The sinus debridement (FESS), although it was performed several times, did not cause any complications. While the pseudoaneurysm noted during the carotid angiogram was initially thought to arise from the ophthalmic artery, this idea was dismissed when this artery was not visualized. Most likely this was caused by turbulent blood flow directed toward the usual location of the ophthalmic artery origin. Surgical management with ligation of the affected artery is the technique of choice for extracranial carotid noninfectious aneurysms.8 However, with site infection, limited access to the aneurysm and the need for immediate treatment of massive epistaxis, as in our case, an alternative, minimally invasive technique is warranted. Recent reports advocate endovascular embolization for such aneurysms.9 The efficacy of this minimally invasive procedure is appealing, but few cases with long-term follow-up after a complicated invasive aspergillus sinusitis have been described in the English literature. Hot et al8 reported a case using GDCs for endovascular treatment of an extracranial carotid artery pseudoaneurysm complicated by aspergillus sinusitis. He reported a high mortality rate of 80% (8 of 10) when fungal sinusitis is associated with an intracranial or extracranial ICA aneurysm. Only 2 patients survived, including a case undergoing endovascular embolization, while the rest of the patients received no interventional procedure. Hurst et al5 also reported an internal carotid artery pseudoaneurysm caused by invasive aspergillus sinusitis. Although the aneurysm was obturated by endovascular coils, it extended intradurally, causing a fatal subarachnoid hemorrhage. The optimal approach for endovascular embolization of a pseudoaneurysm is to occlude the lesion while preserving the parent artery. This technique is commonly applied in cases of traumatic or atherosclerotic aneurysms. However, for mycotic aneurysm, especially for lesions with extensive inflammation around the site or with long segmental vascular involvement, use of ICA trapping to occlude the involved segment is preferred. Moreover, the inflammation may invade another vascular segment and lead to further pseudoaneurysm formation and even bleeding. Stent-graft implantation has recently been used for cerebrovascular diseases such as dissecting pseudoaneurysm, carotid-cavernous fistula, and carotid blow-out syndrome.10 This approach may be an alternative for sealing the mycotic pseudoaneurysm, especially for patients who fail the balloon test occlusion. However, further investigation is necessary to determine its efficacy and long-term results. In summary, emergent angiography and therapeutic endovascular embolization can be life saving in


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cases where conservative management of epistaxis has failed and surgery is not feasible. For patients with an extensive fungal infection adjacent to or surrounding the ICA, total ICA occlusion may be preferable to other ICA-preserving endovascular techniques. Endovascular embolization offers an effective and minimally invasive therapeutic alternative for fungal internal carotid pseudoaneurysms.

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5. Hurst RW, Judkins A, Bolger W, Chu A, Loevner LA. Mycotic aneurysm and cerebral infarction resulting from fungal sinusitis: imaging and pathologic correlation. AJNR Am J Neuroradiol 2001;22:858–863. 6. Kothary MH, Chase T Jr, Macmillan JD. Correlation of elastase production by some strains of Aspergillus fumigatus with ability to cause pulmonary invasive aspergillosis in mice. Infect Immun 1984;43:320–325. 7. Chen D, Concus AP, Halbach VV, Cheung SW. Epistaxis originating from traumatic pseudoaneurysm of the internal carotid artery: diagnosis and endovascular therapy. Laryngoscope 1998;108:326–331. 8. Hot A, Mazighi M, Lecuit M, et al. Fungal internal carotid artery aneurysms: successful embolization of an Aspergillus-associated case and review. Clin Infect Dis 2007;45:e156–161. 9. Reisner A, Marshall GS, Bryant K, Postel GC, Eberly SM. Endovascular occlusion of a carotid pseudoaneurysm complicating deep neck space infection in a child. Case report. J Neurosurg 1999;91:510–514. 10. Hoppe H, Barnwell SL, Nesbit GM, Petersen BD. Stentgrafts in the treatment of emergent or urgent carotid artery disease: review of 25 cases. J Vasc Interv Radiol 2008;19:31–41.


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