From the Department of Neurosurgery, Kitasato University School of
Medicine, Kanagawa, Japan.
Case Description—Initial angiography performed in a 39-year-old
man showed thrombosis of the superior sagittal sinus (SSS) and the
right transverse sinus (TS) but no vascular malformations. Follow-up
angiography 29 months later revealed recanalization
of the SSS and the TS, retrograde cortical venous drainage which
suggested that thrombosis of the sinuses probably propagated into the
adjacent parietal cortical veins, and development of a dural-pial AVM
at or near the site of thrombi in more than one cortical vein. Complete
surgical excision of the lesion was accomplished without neurological
deterioration.
Conclusions—The present case suggests the possibility that
the pial AVM is not only a congenital condition but also may develop as
an acquired lesion.
CT scan revealed a low-density area with a mass effect in the right
frontal lobe (Figure 1
Immediately after admission, anticoagulant therapy was initiated. Both
the headache and the left hemiparesis improved gradually and since,
there were no clearly abnormal findings on neurological examination
other than a slight exaggeration of the deep tendon reflex on the left
side, the patient was discharged. Although MRI performed on September
27, 1996, revealed recanalization of the SSS and
the right TS, at the same time a large number of flow void signals
suggestive of a vascular malformation made their appearance in the
right parietal lobe. On December 19 of the same year, right selective
internal carotid angiography demonstrated a pial AVM in the right
parietal region, supplied by branches from the middle cerebral artery
and draining into the cortical veins (Figure 3A
Operative and Postoperative Findings
In addition, the pial AVM, whose feeding arteries were 3 branches of
the middle cerebral artery, was seen surrounding the same superficial
draining veins; we removed these structures in their entirety. The
structures removed were found histologically to be pial
AVM tissue. The resulting defect of the dura was repaired with the
epicranial aponeurosis beneath the skin flap with watertight
suturing.
Postoperative selective internal and external carotid angiography
revealed no vascular malformation. The postoperative course was
uneventful, and there was no deterioration in the patient's
neurological condition.
Kerber and Newton6 suggest that minute
arteriovenous shunts, which are normally present within the dura
mater, might enlarge in the presence of increased venous pressure and
lead to angiographically significant arteriovenous shunting.
Furthermore, Lawton et al2 speculate that an
increased cortical venous pressure induces angiogenic activity either
directly or indirectly by decreasing cerebral perfusion and increasing
ischemia and that dural AVM formation may be the result of
aberrant angiogenesis.
On the other hand, pial AVMs are generally considered congenital
vascular malformations. Indeed, there are very few studies (only 3
relevant reports could be found7 8 9 ) in which
the presence of a pial AVM is ruled out in initial angiography but is
later discovered to have developed. However, the present
case is the first reported in which a pial AVM is thought to have newly
formed after a sinus thrombosis.
Lyons et al10 reported that angiogenic growth
factors such as basic fibroblast growth factor appeared in the cerebral
tissue surrounding an area of cerebral ischemia. In our case,
the pial AVM occurred adjacent to the posterior part of the
ischemic region (Figure 1
Kader et al11 recently described pediatric
cases in which pial AVMs recurred after total resection of the original
lesions had been verified, and they have begun to cast doubt on the
theory of cerebral AVMs as congenital anomalies. The present case
also is significant in that it calls into question the theory that a
pial AVM is an exclusively congenital phenomenon.
Ravens12 examined anastomoses in the vascular bed
of the human brain. According to his findings, arteriovenous
anastomoses were present in both the cerebral cortex and white
matter in the normal human brain.
We suggest the possibility that the pial AVM in the present case
developed because closed arteriovenous fistulas that were originally
present somehow became patent when cerebral ischemia
occurred.
Received March 3, 1998;
revision received May 18, 1998;
accepted May 18, 1998.
2.
Lawton MT, Jacobowitz R, Spetzler RF. Redefined role
of angiogenesis in the pathogenesis of dural arteriovenous
malformations. J Neurosurg. 1997;87:267–274.[Medline]
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Awad IA, Little JR, Akrawi WP, Ahl J.
Intracranial dural arteriovenous malformations: factors predisposing to
an aggressive neurological course. J Neurosurg. 1990;72:832–850.[Medline]
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Houser OW, Campbell JK, Campbell RJ, Sundt TM.
Arteriovenous malformation affecting the transverse dural venous sinus:
an acquired lesion. Mayo Clin Proc. 1979;54:651–661.[Medline]
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Newton TH, Cronqvist S. Involvement of dural arteries
in intracranial arteriovenous malformations. Radiology. 1969;93:1071–1078.[Medline]
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Kerber CW, Newton TH. The macro and microvasculature
of the dura mater. Neuroradiology. 1973;6:175–179.[Medline]
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Miyasaka Y, Kurata A, Saegusa H, Yuzawa I, Utsuki S,
Ohwada T. Dural-pial arteriovenous malformation with unusual venous
drainage: case report. Neurol Med Chir (Tokyo). 1996;36:91–95.[Medline]
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Sasaki T, Hoya K, Kinone K, Kirino T. Postsurgical
development of dural arteriovenous malformations after transpetrosal
and transtentorial operation: case report. Neurosurgery. 1995;37:820–825.[Medline]
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Schmit BP, Burrows PE, Kuban K, Goumnerova L, Scott
RM. Acquired cerebral arteriovenous malformation in a child with
moyamoya disease: case report. J Neurosurg. 1996;84:677–680.[Medline]
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Lyons MK, Anderson RE, Meyer FB. Basic fibroblast
growth factor promotes in vivo cerebral angiogenesis in chronic
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Kader A, Goodrich JT, Sonstein WJ, Stein BM, Carmel PW,
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© 1998 American Heart Association, Inc.
Case Report
Dural-Pial Arteriovenous Malformation After Sinus Thrombosis
Abstract
Top
Abstract
Introduction
Case Report
Discussion
References
Background—We report an unusual case
of acquired dural-pial arteriovenous malformation (AVM) following sinus
thrombosis.
Key Words: angiogenesis • cerebral arteriovenous malformations • sinus thrombosis
Introduction
Top
Abstract
Introduction
Case Report
Discussion
References
Dural arteriovenous
malformations (AVMs) appear to be acquired rather than congenital
lesions, and it is well known that they often develop at the site of a
sinus thrombosis.1 2 3 4 On the other, hand, pial
AVMs are generally considered to be congenital malformations. We report
a case of acquired dural-pial AVM following superior sagittal and
transverse sinus thrombosis and discuss the causes both of the unusual
location of the dural AVM and of the development of the pial lesion in
this case.
Case Report
Top
Abstract
Introduction
Case Report
Discussion
References
The patient was a 39-year-old man who, on July 27, 1994,
experienced headache and vomiting, followed 2 days later by weakness in
the upper part of his left arm. He was hospitalized 4 days later, and
neurological examinations indicated left hemiparesis. 
), and on enhanced
CT an empty delta sign was observed in the superior sagittal sinus
(SSS). MRI showed sinus thrombosis of the SSS and the right transverse
sinus (TS). However, MRI revealed no abnormal findings suggestive of a
vascular malformation. Because no image of the anterior half of the SSS
or the right TS could be detected in angiograms of the right common
carotid artery, a diagnosis of sinus thrombosis of the SSS and the
right TS was made (Figure 2A and 2B).
Nevertheless, no dural or pial AVMs were found (Figure 2C and 2D).
View larger version (141K):
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Figure 1. Axial CT scan without contrast enhancement showing
a low-density area with mass effect in the right frontal lobe.
View larger version (135K):
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Figure 2. A and B, Right common carotid angiograms showing
15° oblique anteroposterior (A) and lateral (B) views of the venous
phase. Occlusion of the superior sagittal sinus and the right
transverse sinus is demonstrated. C and D, Right common carotid
angiograms of the lateral (C) and anteroposterior (D) views of the
arterial phase, demonstrating no vascular malformations. and 3B). It was also noted that the
SSS was patent but there was a reflux of blood into the cortical veins,
and the venous routes between the cortical veins and the SSS were
occluded (Figure 3C). Meanwhile, right selective external carotid
angiography revealed a dural AVM supplied by the superficial temporal
artery and the middle meningeal arteries and also draining into the
cortical veins (Figure 3D).
View larger version (140K):
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Figure 3. A and B, Selective right internal carotid
angiograms of the lateral (A) and anteroposterior (B) views of the
arterial phase, demonstrating a pial AVM supplied by
branches from the middle cerebral artery and draining to the cortical
vein. C, Right internal carotid angiogram of the lateral view of the
venous phase, demonstrating the patency of the superior sagittal sinus.
D, Selective right external carotid angiograms of the lateral view of
the arterial phase, demonstrating a pial AVM fed by the
superficial temporal and middle meningeal arteries and draining to the
cortical vein.
After a negative result was obtained in a provocation test
conducted on March 24, 1996, before direct surgery, the feeder of the
pial AVM was selectively occluded with use of a liquid coil (Target
Therapeutics), and a parieto-occipital craniotomy was
performed. The superficial temporal artery was found to pierce
the cranium, becoming a feeding artery. When the dura mater was opened,
several draining veins that were red veins were observed on the surface
of the cerebrum. These draining veins on the cerebral surface formed
vascular connections together with the dural AVM fed by the middle
meningeal arteries at 2 sites (Figure 4).
A section of the dura measuring 4x6 cm was resected together with a
dural AVM and the connections of the cortical vein. Many of the vessels
seen on histological examination were dilated and
appeared to be dysplastic, which confirmed the presence of an AVM in
the dura.
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Figure 4. Intraoperative view showing the opening of
the dura mater. On the surface of the cerebrum, red veins acting as
draining veins can be seen. These draining veins (arrow) on the
cerebral surface formed vascular connections (arrowhead) together with
the dural AVM fed by the middle meningeal arteries at 2 sites.
Discussion
Top
Abstract
Introduction
Case Report
Discussion
References
Newton and Cronqvist5 classify intracranial
AVMs into 3 types on the basis of their arterial supply:
pure pial, mixed pial and dural, and pure dural. Mixed pial and dural
malformations receive their blood supply not only from cerebral or
cerebellar arteries but also from meningeal vessels. Therefore, the
vascular malformation presented here was termed a dural-pial
type. It has been emphasized that development of dural AVMs correlates
positively with venous hypertension due to sinus
thrombosis.1 2 Usually, dural AVMs develop
adjacent to a thrombosed sinus.1 2 3 4 The
present case was unique in that the dural component of the AVM was
not located at the site of a sinus thrombosis. Although the SSS and the
TS were recanalized, as shown in Figure 3C
, retrograde cortical venous
drainage was found. This suggests that thrombosis of the SSS and the TS
probably propagated into the adjacent cortical veins, that the
connections between the cortical veins and the sinuses had been
obliterated, and that increased cortical venous pressure persisted.
Therefore, it was considered that cortical venous hypertension due to
thrombosis of a cortical vein may have played an important role in the
development of the dural AVM in this case. ), an area in which obstruction of the
connections between the cortical veins and the sinus was present
and a reduction of cerebral perfusion due to the increased cortical
venous pressure persisted. Thus, the possibility that the increased
expression of angiogenic factors and the resulting neovascularization
played a part in the development of an acquired pial AVM was
suggested.
Acknowledgments
The authors wish to thank C.W.P. Reynolds for his editorial
assistance.
Footnotes
Reprint requests to Dr Ryusui Tanaka, Department of Neurosurgery, Kitasato University School of Medicine, 1-15-1 Kitasato, Sagamihara, Kanagawa, 228-8555 Japan.
References
Top
Abstract
Introduction
Case Report
Discussion
References
1.
Herman JM, Spetzler RF, Bederson JB, Kurbat JM,
Zabramski JM. Genesis of dural arteriovenous malformation in a rat
model. J Neurosurg. 1995;83:539–545.[Medline]
[Order article via Infotrieve]
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