Neurosurg Focus / Volume 32 / May 2012

Neurosurg Focus 32 (5):E2, 2012

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The association of cerebral AVMs and aneurysms is well established in the literature.2,4,8 Recent natural history data have clearly suggested the significance

of this association, as AVMs with associated aneurysms are more likely to have hemorrhagic presentation9,11 as well as hemorrhage on follow-up.2,4 In contrast, the fre-quency and significance of aneurysms associated with ce-rebral DAVFs is infrequently addressed, and is limited to case reports and small patient series.3,5–7,10 In this report, we review our own patient cohort of cerebral DAVFs and evaluate the association of aneurysms with cerebral DAVFs.

MethodsFifty-six patients with 70 DAVFs were treated by

the neurosurgical or interventional neuroradiology team at Brigham and Women’s Hospital from 2004 to 2011. The cerebral DAVF diagnosis was angiographically con-

firmed in all patients. In addition to background epide-miological data, we reviewed each angiogram and noted the presence of remote and/or feeding artery aneurysms in association with the DAVF.

ResultsBackground demographic and treatment information

for the entire patient cohort as well as those DAVFs associ-ated with aneurysms is provided in Table 1. Of 56 patients with cerebral DAVFs, 12 had aneurysms (21%; Tables 1 and 2). Patients with DAVFs and associated aneurysms were slightly younger (mean age 49.8 vs 58 years, re-spectively), with a slightly stronger male predilection (2.0 male-to-female ratio vs 1.2, respectively). Patients with aneurysms had DAVFs in a variety of locations without a clear predisposition for 1 particular location. Approxi-mately two-thirds of the DAVFs in each cohort had cortical venous drainage (Borden Type II or III).1 Notably, patients with DAVFs and aneurysms were more likely to have their DAVF treated surgically (75% vs 34%, respectively), and 58% of patients with DAVFs and aneurysms presented with hemorrhage compared with only 20% of patients without aneurysms (Tables 1 and 3).

Cerebral dural arteriovenous fistulas and aneurysms

Bradley a. Gross, M.d., alexander e. ropper, M.d., and rose du, M.d., ph.d.Department of Neurological Surgery, Brigham and Women’s Hospital, and Harvard Medical School, Boston, Massachusetts

Object. The association of aneurysms and cerebral arteriovenous malformations is well established in the litera-ture. Aside from a small number of case reports and small patient series, this association has not been well explored with cerebral dural arteriovenous fistulas (DAVFs). This study was designed to elucidate this relationship in the authors’ own patient cohort with DAVFs.

Methods. Cerebral angiograms of 56 patients with 70 DAVFs were reviewed for the presence of cerebral aneu-rysms. Background patient demographics, mode of presentation, and DAVF and aneurysm angiographic character-istics were noted.

Results. Twelve patients (21%) had aneurysms in addition to their DAVF. Three patients had multiple aneurysms. Of a total of 15 aneurysms, 5 (33%) occurred on DAVF feeding arteries and 10 (67%) were in remote locations. These patients more commonly presented with hemorrhage (58% vs 20% for those without aneurysms). Aneurysms were associated with DAVFs in any location (feeding artery or remote), but flow-related feeding artery aneurysms were more likely to be associated with Borden Type III DAVFs.

Conclusions. Twenty-one percent of patients with cerebral DAVFs also had aneurysms in this patient cohort. It is thus prudent to perform 6-vessel digital subtraction angiography on patients with DAVFs to rule out potential feed-ing artery and remote aneurysms. This association may be explained by flow-related phenomena, the initial inciting event leading to DAVF formation, as well as a potential genetic component or predisposition to develop these lesions.(http://thejns.org/doi/abs/10.3171/2011.12.FOCUS11336)

Key Words      •      dural arteriovenous fistula      •      aneurysm      •      epidemiology

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Abbreviations used in this paper: AICA = anterior inferior cer-ebellar artery; AVM = arteriovenous malformation; DAVF = dural arteriovenous fistula; MMA = middle meningeal artery; PMA = posterior meningeal artery; SRS = stereotactic radiosurgery.

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Of a total of 15 aneurysms, 5 (33%) were located on DAVF feeding arteries (Table 2). Thus, of 70 DAVFS overall, 5 (7%) had feeding artery aneurysms. Four of these aneurysms developed spontaneously and were all associated with Borden Type III fistulas.1 One aneurysm was an obvious pseudoaneurysm that developed adjacent to a skull base fracture, likely simultaneously with its as-sociated Type I jugular DAVF (Fig. 1). This aneurysm developed on the PMA, whereas the other 4 aneurysms were located on the AICA (2 cases), MMA (1 case), and a distal middle cerebral artery branch (1 case). Two of 5 pa-tients with feeding artery aneurysms presented with hem-orrhage (Table 3). In 1 case, subarachnoid hemorrhage was attributed to an AICA feeding artery aneurysm, con-firmed intraoperatively (Fig. 2). In another case, a large intraparenchymal hemorrhage with intraventricular ex-tension was attributed to a cortical DAVF as its feeding artery aneurysm was located extradurally on the MMA. In these 2 cases, the DAVFs were treated surgically in an expedient fashion. Two of 3 cases without hemorrhagic presentation were also treated surgically. In 1 case SRS was used, with obliteration of both the DAVF and feeding artery aneurysm observed on the patient’s 2-year follow-up angiogram.

Eight patients were found to have a total of 10 remote aneurysms. Thus, 8 (14%) of 56 patients with DAVFs also had remote aneurysms. Five of these patients presented

TABLE 1: Characteristics of 56 patients with 70 DAVFs and a subgroup of 12 patients with DAVFs and aneurysms*

CharacteristicEntire Cohort

w/ Aneurysm

w/o Aneurysm

no. of patients 56 12 44no. of DAVFs 70 12 58mean age (yrs) ± SD 56.5 ± 15.5 49.8 ± 16.9 58.0 ± 14.7M:F (ratio) 32:24 (1.3) 8:4 (2.0) 24:20 (1.2)DAVF location TS 16 1 15 SSS 12 2 10 tentorial 10 3 7 petrosal 9 1 8 torcular 5 2 3 anterior fossa 5 1 4 cavernous 4 1 3 other 9 1 (jugular) 8Borden Type I 24 4 20 II 12 0 12 III 34 8 26hemorrhagic presentation (%) 16 (29) 7 (58) 9 (20)DAVF treatment modality op w/wo embolization 29 9 20 observation 18 1 17 embolization alone 13 0 13 SRS 10 2 8

* SSS = superior sagittal sinus; TS = transverse-sigmoid.

TABLE 2: Characteristics of 12 patients with DAVFs and aneurysms*

Age (yrs), Sex

DAVF Location

Borden Type

DAVF Rupture

DAVF Treatment

Aneurysm Location Feeder

Aneurysm Rupture

Aneurysm Treatment

15, M jugular I no op PMA yes† no op46, F tentorial III no SRS AICA yes no SRS61, M SSS III no op distal MCA, PCoA yes, no no op22, M tentorial III no op AICA yes yes op52, M SSS III yes op MMA yes no op39, M cavernous I no observation ACoA no yes coil embolization53, F anterior fossa I no SRS basilar no yes coil embolization62, M torcular III no op ACoA no no observation71, F petrosal I no op PCoA, SHA no no op, observation59, M torcular III no op bilat PCoA no yes op56, F tentorial III yes op cavernous no no observation62, M TS III yes op SCA no no observation

* ACoA = anterior communicating artery; cavernous = cavernous internal carotid artery; MCA = middle cerebral artery; PCoA = posterior communicating artery; SCA = superior cerebellar artery; SHA = superior hypophyseal artery.†  Denotes pseudoaneurysm; the remainder of feeding artery aneurysms were flow related.

TABLE 3: Hemorrhage from DAVF with associated aneurysms*

DAVFNo. of

PtsHemor- rhage

Hemorrhage From DAVF

Hemorrhage From Aneurysm

w/ any aneurysm 12 7 3 4w/ feeding aneurysm 5 2 1 1w/ remote aneurysm 8 5 2 3

* Pts = patients.

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Neurosurg Focus / Volume 32 / May 2012

Cerebral dural arteriovenous fistulas and aneurysms

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with hemorrhage. In 3 cases there was diffuse subarach-noid hemorrhage from a ruptured aneurysm with inci-dental discovery of the DAVF (cavernous, anterior fossa, and torcular; Fig. 3). In 2 cases, intraparenchymal hem-orrhage was attributed to a tentorial and transverse-sig-moid DAVF with incidental discovery of a cavernous and 2-mm superior cerebellar artery aneurysm, respectively. All ruptured aneurysms and DAVFs were treated swiftly to minimize the risk of rerupture. All unruptured DAVFs were treated surgically except 2—a cavernous DAVF was carefully observed and an anterior fossa DAVF without cortical venous drainage was treated using radiosurgery. Due to their small size, most unruptured remote aneu-rysms were simply carefully observed.

DiscussionThe association of aneurysms and cerebral AVMs is

well established in the literature.2,4,8,9 Numerous natural history studies have demonstrated this relationship for approximately 20% of AVMs, with half of associated aneurysms occurring on feeding arteries, one-fourth ni-dal, and one-fourth remote.2,4,8 In our cohort of 56 pa-tients with 70 DAVFs, we found that 21% of patients had aneurysms as well, approximately one-third on feeding arteries and two-thirds in remote locations. We advocate performing 6-vessel digital subtraction angiography on patients with DAVFs to rule out potential feeding artery and remote aneurysms.

Although DAVFs are considered to be acquired le-sions, the finding that 21% of patients have concomitant aneurysms, two-thirds of which are remote (14% of pa-tients overall), suggests a potential genetic component

Fig. 1. Images obtained in a 15-year-old male without significant medical history who presented after a motor vehicle accident. Initial noncontrast axial CT revealed a traumatic subarachnoid hemorrhage (A) as well as a left occipital bone fracture (B, arrow). Digital subtraction angiography was performed 1 week after the incident to rule out vasospasm. Although no vasospasm was noted, a DAVF fed by the posterior meningeal artery with an associated feeding artery pseudoaneurysm (C, arrow) was observed on the left vertebral artery injection.

Fig. 2. Anteroposterior vertebral artery angiogram obtained in a 22-year-old man who presented with a severe headache and mild con-fusion as a result of a subarachnoid hemorrhage (Hunt and Hess Grade III). The angiogram demonstrates a tentorial DAVF with an AICA feed-ing artery aneurysm (arrow).

Fig. 3. Digital subtraction angiogram obtained in a 53-year-old woman without  a  significant medical  history who  presented with  ob-tundation due to subarachnoid hemorrhage (Hunt and Hess Grade V) from a ruptured basilar apex aneurysm. The aneurysm underwent coil embolization (arrow) and the patient made a remarkable recovery. An anterior fossa DAVF (asterisk) with an ethmoidal feeder (arrowhead) was also noted.

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or predisposition to developing these lesions as well. In addition to flow-related feeding artery aneurysms, pseu-doaneurysms due to trauma may also develop on feeding arteries (Fig. 1). The acquired pathogenesis of these pseu-doaneurysms likely occurs concomitant with the develop-ment of the DAVF itself.

One prior study10 also found that 13% of patients with DAVFs have remote aneurysms. The authors noted that all 6 patients possessing DAVFs and aneurysms were male, 3 of whom possessed anterior cranial fossa DAVFs while the other 3 had convexity DAVFs. In reviewing the literature, the study reinforced a potential predilection for anterior cranial fossa DAVFs to be associated with re-mote aneurysms. Although we also demonstrate a male predilection among patients with DAVFs and aneurysms, our cases included DAVFs in a wide variety of locations without a clear predisposition for anterior fossa lesions, although we only had 5 cases of anterior fossa DAVFs in our entire cohort.

We demonstrated that feeding artery aneurysms to DAVFs may be more commonly noted among those with direct cortical venous drainage. This relationship under-scores the difficulty of assessing the natural history of such lesions, as most DAVFs with direct cortical venous drainage alone warrant treatment, precluding longitudinal observation of these lesions. Nevertheless, our retrospec-tive analysis demonstrating more common hemorrhagic presentation among patients with DAVFs and aneurysms does reinforce an intuitively more malignant natural his-tory.

ConclusionsIn this study we demonstrated that approximately

21% of patients with DAVFs also have aneurysms, one-third of which occur on feeding arteries and the remain-der in a remote location. Feeding artery aneurysms may develop due to flow-related phenomena or due to the initial inciting event leading to DAVF formation such as trauma. Patients with DAVFs and aneurysms were more likely to present with hemorrhage, were slightly younger, and had a stronger male predilection than those without aneurysms.

Disclosure

The authors report no conflict of interest concerning the mate-rials or methods used in this study or the findings specified in this paper.

Author contributions to the study and manuscript prepara-tion include the following. Conception and design: all authors. Acquisition of data: Gross, Ropper. Analysis and interpretation of data: all authors. Drafting the article: Gross, Ropper. Critically revis-

ing the article: all authors. Reviewed submitted version of manu-script: Du. Approved the final version of the manuscript on behalf of all authors: Du. Study supervision: Du.

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Manuscript submitted November 16, 2011.Accepted December 20, 2011.Please include this information when citing this paper: DOI:

10.3171/2011.12.FOCUS11336. Address correspondence to: Rose Du, M.D., Ph.D., Department

of Neurological Surgery, Brigham and Women’s Hospital and Harvard Medical School, 75 Francis Street, Boston, Massachusetts 02115. email: rdu@partners.org.

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