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�:	Ruptured Aneurysm At the Fenestration of the Middle Cerebral Artery Detected by Magnetic Resonance Angiography in a Patient With Systemic Lupus Erythematosus and Renal Failure

Sadaharu TABUCHI,1 Hiroki YOSHIOKA1

1Department of Neurosurgery, Tottori Prefectural Central Hospital, Tottori, Japan

Corresponding author: Sadaharu Tabuchi M.D., Ph.D.
Department of Neurosurgery, Tottori Prefectural Central Hospital, 730 Ezu, Tottori 680-0901, Japan
Tel: +81-857-26-2271; Fax: +81-857-29-3227
E-mail: Sadaharu Tabuchi -  HYPERLINK "mailto:tabuchis@pref.tottori.jp" tabuchis@pref.tottori.jp 

Key Words: arterial fenestration, cerebral aneurysm, subarachnoid hemorrhage, magnetic resonance angiography, intraoperative indocyanine green video angiography, systemic lupus erythematosus
Running title: Ruptured aneurysm at fenestration of MCA detected by MRA
Abstract

Background
Cerebral aneurysm arising at the fenestration of the middle cerebral artery (MCA) is extremely rare, with one report describing subarachnoid hemorrhage (SAH) due to this type of lesion. There have been no reports of this type of lesion occurring in a patient with systemic lupus erythematosus (SLE). 
Case description
A 47-year-old female with SLE and chronic renal failure had sudden onset of SAH. We avoided using contrast medium due to the chronic renal failure. Magnetic resonance angiography (MRA) showed the ruptured aneurysm arising at the site of fenestration of the MCA. Successful clipping, perioperative management, introducing renal dialysis after acute phase and placement of a ventriculoperitoneal (VP) shunt were performed, and the patient was discharged home with no complications. 
Conclusion
This is the first report of ruptured aneurysm associated with MCA fenestration in an SLE patient as detected by MRA. The presence and anatomical relationship of fenestration accompanied by aneurysm can be noninvasively and accurately evaluated using three-dimensional time-of-flight MRA (3D-TOF-MRA) with the volume rendering (VR) method.

Introduction

The reported incidence of fenestration of the middle cerebral artery (MCA) ranges from 0.26 to 0.28% [1]. Although MCA fenestration is extremely rare, an aneurysm can arise at the proximal end of the fenestration. The magnetic resonance (MR) angiographic features of aneurysm associated with MCA fenestration have rarely been reported. Subarachnoid hemorrhage (SAH) occurs in about 1% of patients with systemic lupus erythematosus (SLE) [2].  There are no previous reports of ruptured aneurysm at the MCA fenestration in a patient with SLE. The present report describes a case that was diagnosed preoperatively by MR angiography (MRA). This modality was used because the presence of underlying renal failure was a contraindication to other imaging modalities. As the prognosis of SLE patients is usually poor, both diagnosis and therapy were difficult and important in this case.

Case Report

A 47-year-old female with SLE had sudden onset of severe headache and was referred to our hospital. Computed tomography (CT) demonstrated SAH in the right Sylvian fissure (Fig. 1a). Considering that the patient has suffered from SLE for 10 years and had developed chronic renal failure without renal dialysis. MRA was used to identify the ruptured aneurysm. MRA was performed using a 1.5-T MR system (Signa HDXt, GE, USA). Contrast material was not used. The volume rendering (VR) method with three-dimensional time-of-flight MRA (3D-TOF-MRA) was performed (Fig. 1c,d) in addition to the usual maximum intensity projection (MIP) image (Fig. 1b). MRA revealed a fenestration at the M1 portion of the right MCA and a small saccular aneurysm at the proximal end of the fenestration (Fig. 1c,d). This was consistent with a ruptured aneurysm. A left frontotemporal craniotomy was performed to clip the neck of the aneurysm on the same day. The fenestration and ruptured aneurysm were confirmed under surgical microscopy, and the aneurysm was successfully occluded (Fig. 2a,b). Intraoperative indocyanine green (ICG) videoangiography confirmed this result (Fig. 2c). The present patient was carefully managed avoiding cerebral vasospasm and general complications during the perioperative period and introduced the renal dialysis after acute phase and had a favorable outcome.0Postoperatively, the patient developed normal pressure hydrocephalus, requiring placement of a VP shunt. The patient was ultimately discharged home in good condition. 

Discussion

Fenestration is a rare cerebrovascular variation. Fenestration of the MCA, which is present in 0.26 to 0.28% of patients, is much less frequent than fenestration of the vertebrobasilar system [1].  Most of the fenestrations reported previously were located at the proximal portion of M1 [3,4].  Rarely, aneurysms can be seen at the proximal end of the fenestration [5].  There has been only one reported case of ruptured aneurysm at this fenestration [6].  
SLE is an autoimmune systemic inflammatory disease. SAH due to ruptured aneurysm has rarely been reported in patients with SLE, where it is associated with a poor prognosis. This is the first report of SAH from a ruptured aneurysm associated with MCA fenestration in a patient with SLE. As this patient had developed chronic renal failure due to SLE, typical imaging modalities were contraindicated, and MRA was utilized for diagnosis and preoperative planning. Digital subtraction angiography (DSA) and 3D-CTA are currently considered the modality of choice for imaging and pre-surgical assessment of intracranial aneurysms. However, both methods require contrast medium, and DSA is a relatively invasive method and is associated with a risk of various complications. Some patients cannot undergo these imaging modalities due to renal failure (as in the present case) or an allergy to the contrast medium. MRA is considered a safe imaging technique because it is non-invasive, non-radioactive, technically simple and does not require the administration of a contrast medium. Furthermore, this technique allows three-dimensional imaging of intracranial structural details. Recently, the utility of the VR method with the 3D-TOF-MRA has been described [7,8].  Although the MIP technique has been generally used, VR imaging yields better results in terms of spatial recognition. Indeed, the presence and anatomical relationship between fenestration and aneurysm was characterized using this technique. This imaging technique also facilitated surgical planning in this case. This is the first report of ruptured aneurysm associated with MCA fenestration in a patient with SLE in which MRA was used to correctly diagnose the condition. 

Conflicts of Interest Disclosure

The authors did not receive any fundings for this work. 
References

Lazar ML, Bland JE, North RR, Bringewald PR (1980) Middle cerebral artery fenestration. Neurosurgery 6(3): 297-300
Baizabal Calballo JF, Cantu Brito C, Estanol B, Garcia Ramos GS (2007) Subarachnoid hemorrhage as a complication of systemic lupus erythematosus. Cerebrovasc Dis 24: 301-304
Deruty R, Pelissou-Guyotat I, Mottolese C, Bognar L, Laharotte JC, et al. (1992) Fenestration of the middle cerebral artery and aneurysm at the site of the fenestration. Neurol Res 14(5): 421-424
Uchino A, Kato A, Takase Y, Kudo S (2000) Middle cerebral artery variations detected by magnetic resonance angiography. Eur Radiol 10: 560-563
Nakamura H, Takada A, Hide T, Ushio Y (1994) Fenestration of the middle cerebral artery associated with an aneurysm. Neurol Med Chir (Tokyo) 34: 555-557
Nussbaum ES, Defillo A, Janjua TM, Nussbaum LA (2009) Fenestration of the middle cerebral artery with an associated ruptured aneurysm. J Clin Neurosci 16: 845-857
Goto M, Kunimatsu A, Shojima M, Abe O, Aoki S, et al. (2013) A pitfall of the volume rendering method with 3D Time-of-Flight MRA: A case of a branching vessel at the aneurysm neck. Magn Reson Med Sci 12(1): 53-56
Sun ZK, Li M, Li MH, Li YD, Sun WP, et al. (2012) Fenestrations accompanied by intracranial aneurysms assessed with magnetic resonance angiography. Neurol India 60 (1): 45-49


Figure legends

Figure 1: (a) Plain CT shows SAH in the right Sylvian fissure. (b) MIP image of MRA suggests the presence of a ruptured aneurysm at the right proximal M1 portion (arrow). (c) VR image of MRA shows the aneurysm arising at the fenestration of M1 (arrow). (d) Different angle of the VR image shows the anatomical relationship between the fenestration and the aneurysm (arrow). 

Figure 2: (a) Intraoperative view of the ruptured aneurysm arising at the proximal portion of the MCA fenestration. (b) Intraoperative view after clipping of the aneurysm. (c) Intraoperative ICG video angiography shows the MCA fenestration and complete disappearance of the aneurysm.









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