부비동염에서 기원한 진균성 동맥류의 치료과정 중 발생한 코일의 이동

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1

서울의료원 신경외과학교실,

²

서울의료원 의학연구소,

³

가톨릭대학교 성빈센트병원 신경외과학교실,

⁴

성균관대학교 의과대학 삼성의료원 이비인후과학교실,

5

연세대학교 의과대학 신경외과학교실

장혁

¹

, 김도희

²

, 김정희

^1,2

, 이호진

³

, 홍상덕

⁴

, 홍창기

⁵

1

Department of Neurosurgery, Seoul Medical Center, Seoul, Korea

2

Department of Research Institute, , Seoul Medical Center, Seoul, Korea

3

Department of Neurosurgery, The Catholic University of Korea, St. Vincent’s Hospital, Suwon, Korea

4

Department of Otorhinolaryngology-Head and Neck Surgery, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Korea

5

Department of Neurosurgery, Yonsei University College of Medicine, Seoul, Korea

Hyeok Chang

¹

, Do Hee Kim

²

, Jung Hee Kim

^1,2

, Ho Jin Lee

³

, Sang Duk Hong

⁴

, Chang Ki Hong

⁵

부비동염에서 기원한 진균성 동맥류의 치료과정 중 발생한 코일의 이동

J Korean Skull Base Society 15권 1호 : 58~62, 2020

종설1 원저1 원저2

증례1 원저3

증례2 증례3 증례4 증례5 증례6

Invasive sphenoid fungal sinusitis sometimes invades the adjacent structures such as the optic nerve or cavernous sinus. Here, we present the treatment for a primarily embolized mycotic aneurysm that ruptured during endoscopic sinus surgery. This report describes the case of a 72-year-old man with a mycotic aneurysm. Although the patient’ s mycotic aneurysm was controlled by coil embolization, the subsequent endoscopic surgery resulted in massive bleeding and dislocation of the embolic coil. The ruptured vessel was stably controlled by internal carotid artery occlusion. This case emphasizes the need for careful consideration when performing endoscopic surgery for sphenoid sinusitis in patients especially with a mycotic aneurysm.

Embolic coil shift during treatment of a mycotic aneurysm stemming from a pre-existing sphenoid sinusitis

논문 접수일 : 2020년 1월 20일 논문 완료일 : 2020년 4월 23일

주소 : Department of Neurosurgery, Seoul Medical Center, 156 Sinnae-ro, Jungnang-gu, Seoul 02053, Korea Tel : +82-2-2276-7881

Fax : +82-2-2276-8537 E-mail : [email protected]

Jung Hee Kim

교신저자

Mycotic aneurysm, Sphenoid sinusitis, Transnasal endoscopic surgery, Coiled coil elimination

Key Words

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▒ INTRODUCTION

Intracranial fungal aneurysms are rare, composing only 2%-5% of intracranial aneurysms, with less than 15 cases having being reported in the 28 years since its first report in 1968.[1] There have been previous reports on the risk of infection by invasive fungal agents and bacteria in patients with sphenoid sinusitis.[2,3] Although bacterial infections are deterred by the protective arterial wall, maxillary sinusitis can still be caused by the inflammation in cerebral artery diseases.[4] Destruction of the sphenoid sinus bone tissue and the vascular barrier due to sinusitis is closely related to infectious aneurysms, frequently requiring surgical treatment due to the limitations imposed by local anatomical features. Here, we present the treatment for a primarily embolized mycotic aneurysm that ruptured during endoscopic sinus surgery (ESS).

1. Ethic statement

All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional research committee (Seoul Medical Center Ethics Committee #2019-06-005) and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.

2. Informed consent

Additional informed consent was obtained from all individual participants for whom identifying information is included in this article.

▒ CASE REPORT

A 72-year-old man came into the hospital with a right- sided headache and nasal obstruction. Upon endoscopic sinonasal examination and computed tomography (CT) Fig. 1

A B C

D

Determination of sphenoid sinusitis.

(A) Axial computed tomography (CT) showed right maxillary cyst and right sided septal deviation. (B) CT scan showed

bony erosion due to sphenoid sinusitis. (C) Magnetic resonance imaging gadolinium enhancement revealed paranasal

sinusitis involving bilateral sphenoid and ethmoid sinuses. The white arrows indicate focal bony destruction abutting to

left internal carotid artery. (D) The mycotic aneurysm was revealed with 4.8 × 6.0 mm multilobulated aneurysm.

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scan analysis, there was a right maxillary cyst and severe septal deviation (Fig. 1A).

The patient underwent right endoscopic maxillary antrostomy and septoturbinoplasty to alleviate symptoms.

Four days after surgery, the patient experienced a worsening headache, and we performed CT and magnetic resonance imaging (MRI). There was a soft tissue density in the sphenoid sinus and a bony defect around the right paraclival internal carotid artery (ICA) on the CT scan.

The MRI showed infiltrative lesions in the sphenoid sinus that crossed over to the right paraclival ICA (Fig. 1B, 1C). We performed digital subtraction angiography (DSA)

on the patient and confirmed an ICA pseudoaneurysm.

We assumed that the invasive fungal sphenoid sinusitis resulted in bony destruction and the erosion of the wall adjacent to the paraclival ICA (Fig. 1D).

A positive result on an aspergillus galactomannan test with an enzyme immunoassay on the patient’s cerebrospinal fluid enabled us to diagnose the fungal meningitis caused by the invasive fungal sinusitis. The patient received 6 mg/kg of voriconazole for 1 day, after which the dose was reduced to 4 mg/kg.

A subsequent DSA or angiography showed that the aneurysm had enlarged to 10.2 × 7.7 mm at the ICA (Fig.

Fig. 2

A B C

Expansion and treatment of mycotic aneurysm. The mycotic aneurysm expanded to (A) 10.2 × 7.7 mm (sixteenth day of admission) and (B) 13.8 × 9.6 mm (twenty-second day of admission). (C) The aneurysm was controlled by coil embolization.

Fig. 3

A B C

The suction and elimination of the embolic coil were observed by the following endoscopic sinus surgery (ESS).

(A) The secondary coil embolization was failed due to disrupted arterial vessel wall. The white arrow indicates the eliminated embolic coil by ESS. (B) A compression test revealed

contralateral blood circulation. (C) The total occlusion of the proximal internal carotid artery showed stable management of re-ruptured mycotic aneurysm.

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2A). On the twenty-second day of admission, and after administering antibiotics, coil embolization was performed to manage the aneurysm, which at that point was 13.8 × 9.6 mm in size, located in the paraclival segment of the ICA (Fig. 2B). ICA stenosis was also discovered during the coil embolization. Additional coiling, as well as stenting, were withheld due to concerns about the fragile distal aneurysm wall and sac expansion (Fig. 2C). Subsequent magnetic resonance angiography and MRI analyses revealed that the aneurysm was under control, with no noticeable change in size.

Fourteen days after coil embolization, we performed sphenoidotomy in order to eliminate the source of the fungal infection. After the procedure, we found a 3 mm bulging bluish mass like lesion. We attempted a mucosal biopsy to confirm the invasive fungal sinusitis, but this resulted in massive bleeding and exposure of the embolic coil. The patient underwent immediate aneurysm embolization. However, the secondary embolic coil failed to settle in the re-ruptured vessel due to the disruption of the arterial wall (Fig. 3A). A compression test suggested that ICA occlusion was a possible treatment option for the patient (Fig. 3B). The re-ruptured aneurysm was stably controlled by the total occlusion of the proximal ICA, with the anterior cerebral artery remaining intact (Fig. 3C).

A follow-up brain MRI showed a reduction in abnormal arterial enhancement and the alleviation of osteomyelitis.

The patient received conservative treatment and antifungal medication in the form of Voriconazole PO (per-oral) 200 mg BID (bis in die) for five months, after which the patient was transferred to a nursing hospital.

▒ DISCUSSION

We have reported on a patient with a mycotic aneurysm that had undergone coil embolization, whose coil had become dislodged due to an ESS performed after the initial coil embolization. The primary mycotic aneurysm had been stably controlled by the coil embolization,

though the subsequent re-rupture of the aneurysm due to a secondary ESS had to be managed by total proximal arterial occlusion. We discuss the relative risk factors regarding how supplementary treatment, such as an ESS, may affect the cerebrovascular system as well as the brain.

First, the incidence of mycotic aneurysms subsequent to sinusitis can be explained by the anatomical location of the sphenoid sinuses. The sphenoid sinus has a critical location in relation to the ICA, central nervous system, as well as other vascular systems that may facilitate the dissemination of infection.[5] The invasive fungal agent can spread to the ICA through the lateral sphenoid wall.

[6] Therefore, fungal sphenoid sinusitis is closely related to infectious intracranial aneurysms, and thus presents an appreciable risk when contemplating a surgical approach.

Second, the destruction of the bone and tissue barrier by invasive aspergillus sinusitis increases the risk of mycotic aneurysms.[7] Aspergillus is a ubiquitous fungus species that has angioinvasive properties and is able to digest elastic tissues through the production of elastase.

This degradation of elastin results in the destruction of the vascular wall.[8] This is especially important as elastin is a major component in many effective barriers of the human anatomy. Therefore, consideration must be given before deciding on surgical treatment for sphenoid sinusitis, due to the possibility of inflammation recurring near the sites of previous endovascular surgery, including coil embolization.

Third, the standard treatment for fungal aneurysms

accompanied by intracranial infection is antimicrobial

therapy along with surgical treatment of the infected

vessels.[9] Thus, endovascular therapy was initially

utilized in high-risk aneurysm patients, and was proposed

as a preferable treatment approach in improving mortality

rates through advances in technology, including detachable

balloons, stents, and coil embolization.[10] However,

this accessibility has also resulted in the widespread use

of surgical treatments in infectious aneurysms, and any

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additional treatment of the sphenoid sinus poses the risk of further cerebrovascular damage.

As otolaryngologists, it is all the more vital to be aware about the possibility of pseudoaneurysms caused by invasive fungal sinusitis, as well as the risk of aneurysm re-rupture, even after coil embolization. Thus, the anatomy of the sphenoid sinus itself as well as the destruction of bony and vascular barriers due to fungal infections require the utmost discretion when it comes to treatment considerations for infectious aneurysms.

CONFLICT OF INTEREST

No potential conflict of interest relevant to this article was reported.

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