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Table of Contents    
CASE REPORT
Year : 2021  |  Volume : 69  |  Issue : 4  |  Page : 1048-1050

Unilateral Basal Ganglia Hyperintensity Secondary to Venous Congestion in a Case of Indirect Carotico-cavernous Fistula


1 Department of Neuroradiology and Interventional Radiology, Yashoda Hospitals, Alexander Road, Secunderabad, Telangana, India
2 Department of Neurology, Yashoda Hospitals, Alexander Road, Secunderabad, Telangana, India

Date of Submission04-May-2020
Date of Decision11-Jul-2020
Date of Acceptance13-Jul-2020
Date of Web Publication2-Sep-2021

Correspondence Address:
Suresh Giragani
Department of Neuroradiology and Interventional Radiology, Yashoda Hospitals, Alexander Road, Secunderabad, Telangana 500003
India
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Source of Support: None, Conflict of Interest: None


PMID: 34507443

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 » Abstract 


Intraparenchymal venous congestive changes secondary to carotico-cavernous fistula are not common. Isolated basal ganglia venous congestive changes in carotico-cavernous fistula have been rarely described. We report MRI findings of the unilateral basal ganglia hyperintensity, angiographic features including cortical venous reflux into a variant basal vein of Rosenthal, in a postpartum woman presenting with the left eye proptosis and the right upper limb weakness. We also describe the reversal of imaging findings and resolution of patients' symptoms after definitive treatment of the carotico-cavernous fistula by endovascular embolization


Keywords: Basal ganglia hyperintensity, basal vein of rosenthal, carotid cavernous fistula
Key Message: Indirect carotico-cavernous fistula can manifest as basal ganglia venous congestive changes due to venous reflux from arterialized cavernous sinus into an anomalous basal vein of Rosenthal.


How to cite this article:
Giragani S, Kasireddy AR, Agrawal V, Muthyala S. Unilateral Basal Ganglia Hyperintensity Secondary to Venous Congestion in a Case of Indirect Carotico-cavernous Fistula. Neurol India 2021;69:1048-50

How to cite this URL:
Giragani S, Kasireddy AR, Agrawal V, Muthyala S. Unilateral Basal Ganglia Hyperintensity Secondary to Venous Congestion in a Case of Indirect Carotico-cavernous Fistula. Neurol India [serial online] 2021 [cited 2021 Oct 18];69:1048-50. Available from: https://www.neurologyindia.com/text.asp?2021/69/4/1048/325358




An indirect carotid cavernous fistula (CCF) is a type of dural arterio venous fistula that results from an abnormal communication between the meningeal branches of internal carotid artery, external carotid arteries, and cavernous sinus in the skull base.[1] Clinical manifestations of CCF depend upon preferential venous drainage pathway effected secondary to the venous hypertension.[2] Usually, patients with CCF present with proptosis, chemosis, cranial nerve palsies etc. Very rarely they can present with venous congestive changes in brainstem and basal ganglia secondary to venous reflux in the lateral mesencephalic vein or an anomalous basal vein of Rosenthal (BVR).[3],[4] In this report, we wish to describe the isolated venous congestive changes in basal ganglia secondary to indirect carotico-cavernous fistula.


 » Case Presentation Top


A 28-year-old woman who had recent normal vaginal delivery presented with complaints of proptosis of the left eye, double vision, and weakness in the right upper limb for 3 days. On examination, there was left eye proptosis, chemosis with oculomotor nerve palsy; and she was not able to lift her right hand against gravity. A contrast-enhanced magnetic resonance imaging of brain and orbits was performed, which revealed T2 hyperintense signal involving the left lentiform nucleus, internal capsule, and caudate nucleus. The diffusion-weighted images show bright signal in the left caudate nucleus. The left cavernous sinus was showing flow related enhancement with post contrast diffuse nodular enhancement. The left superior ophthalmic vein was dilated [Figure 1]. There was associated diffuse enhancement of the extraocular muscles of the left eye with myofascial and retro orbital fat stranding. Further, a digital subtraction angiography was performed, which revealed presence of a Barrow's Type D indirect CCF involving left cavernous sinus with arterial feeders from deep temporal artery, artery of foramen rotundum, and pterygoid branches of left internal maxillary artery and meningo hypophyseal branches of left cavernous ICA. The venous drainage was preferentially superiorly and anteriorly. Posteriorly, the inferior and superior petrosal sinuses were not patent. Superiorly venous reflux was noted into the first segment of BVR and further into the left-sided striate veins [Figure 2]. The second and third parts of BVR were seen in their entirety however small in caliber, which are seen to drain into the vein of Galen (VOG). After consent, endovascular embolisation of the fistula was planned under general anesthesia. Because of occluded inferior petrosal sinus, initially left angular vein approach was attempted. However, due to tortuous junction of superior ophthalmic vein and angular vein, the cavernous sinus could not be accessed. Hence, a trans arterial approach was performed. The fistula was accessed from deep temporal artery branch of the left internal maxillary artery with Marathon micro catheter (ev3, Irvine, CA, USA) and 0.008″ mirage guide wire (ev3, Irvine, CA, USA) and embolized with liquid embolizing agent Onyx-18 (ev3, Irvine, CA, USA). Complete angiographic obliteration of the fistula was achieved [Figure 3]. At 1-month clinical follow-up, the patient had normal power in her right upper limb and a complete resolution of proptosis. On follow-up MRI at three months, there was resolution of basal ganglia hyperintensity.
Figure 1: (a) Axial T2, (b) Coronal T2, (c) axial FLAIR, (d) axial DWI images showing proptosis of the left eye and hyperintensity in left basal ganglia and DWI bright lesion in left caudate and putamen. (e) Time-of-flight MRA axial image showing flow-related enhancement of the left cavernous sinus. (f) Contrast-enhanced T1 axial image showing nodular enhancement of the left cavernous sinus

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Figure 2: (a-c) Digital subtraction angiography selective left ECA injection lateral view in arterial phase, delayed venous phase, and right anterior oblique; (d-f) Cone beam CT sagittal, axial, and oblique coronal image showing venous reflux into inferior striate vein (arrow) through prominent uncal vein (arrow head). Note the opacification of lateral mesencephalic vein (dotted arrow). Note no posterior drainage into the posterior sinuses including superior and inferior petrosal sinuses

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Figure 3: (a and b) Fluoroscopic and digital subtraction angiography images showing selective ECA cannulation with micro catheter (arrows). (c) Floroscopic spot image showing onyx cast in situ (thick arrow). (d) Left ECA injection showing complete occlusion of the fistula. (e and f) Follow-up MRI of brain at 3 months showing resolution of proptosis and basal ganglia hyper intensity

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 » Discussion Top


Indirect CCFs are usually low flow arteriovenous shunts between the meningeal branches of either the ICA or ECA and cavernous sinus.[1] The clinical manifestations and the aggressiveness of CCF are dependent predominantly on the routes of venous drainage. Clinically, patients with CCF usually present with proptosis and chemosis secondary to engorgement of the superior ophthalmic vein draining the arterialized cavernous sinus.[2] Intraparenchymal hemorrhage and venous infarction have been described in CCF associated with cortical venous reflux.[5] The venous congestion changes had been predominantly described in the brainstem and basal ganglia in the setting of venous reflux into anatomical variant of BVR. Although more extensive combined basal ganglia and brainstem congestive changes have been described in CCF,[4] selective isolated venous congestion edema in basal ganglia secondary to the CCF has not been described in the literature.

The BVR are two in number, formed by the union of inferior striate vein, anterior cerebral vein, and deep middle cerebral vein. It originates at the medial edge of temporal lobe and passes in ambient cistern to join the Great (VOG). Huang and Wolf classified BVR into three anatomical segments.[6] The first segment extends from the union of the inferior striate vein, the anterior cerebral vein, and the deep middle cerebral vein to the anterior end of cerebral peduncle. The insular veins drain into deep middle cerebral vein. The second segment extends from the cerebral peduncle to the union of the BVR with the lateral mesencephalic vein, and the third segment extends from the end of second segment to the great VOG. If any of the three segments may be hypoplastic and drainage will be redirected into cavernous sinus, superior petrosal sinus, or transverse sinus accordingly.[6] Suzuki et al. studied anatomical variations of BVR in about 250 patients (500 sides).[7] The anastomosis between the first segment and second segment were not visualized in 36.9% of cases. The first segments with such hypoplastic or aplastic anastomosis flowed into the cavernous sinus or spheno parietal sinus, whereas the second and third segments drained into VOG.[7] Also, more than one-fourth of cases with the typical type, i.e. with anastomosis between all three segments which are entering into VOG, also entered anterior sinuses or veins such as the cavernous sinus. Our index case demonstrated BVR with all three segments anastomosing with each other with drainage into VOG. However, the first segment of BVR is seen to anastomose also with the left cavernous sinus, which was dilated due to reflux from the cavernous sinus secondary to fistula. Since first segment of the BVR drains the inferior striate veins, anterior cerebral veins, and uncal veins, engorgement of this segment by the CCF explains the signal abnormalities in basal ganglia in the MRI.

The direction of venous egress may not be static; it can evolve over time with the development of venous occlusions and venous rerouting as a result of progressive venous restrictive angiopathic changes and gradual thrombosis.[8] In the index case, the prothrombotic state induced by postpartum status may have caused an acute occlusion of petrosal sinuses.[9] Probably this restricted outflow must have led to selective venous reflux anteriorly into SOV and superiorly into the inferior striate vein tributary of the BVR via the prominent uncal vein resulting in venous congestion in the basal ganglia. Intact although smaller second and third parts BVR may have led to sparing of posterior structures including brainstem from venous congestion, the reversibility of basal ganglia changes after the embolization of CCF further confirm the etiopathogenesis.


 » Conclusion Top


Venous congestion manifesting as isolated basal ganglia hyperintensity is a rare imaging finding in CCF. The association of cavernous sinus arterialization, a prominent uncal vein with reflux into striate tributaries of BVR, a thrombosed or hypoplastic inferior, and superior petrosal sinuses may explain this rare manifestation of indirect CCF. Knowledge and awareness of this unusual imaging finding in CCF may help in early diagnosis and treatment of this condition.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
 » References Top

1.
Barrow DL, Spector RH, Braun IF, Landman JA, Tindall SC, Tindall GT. Classification and treatment of spontaneous carotid-cavernous sinus fistulas. J Neurosurg 1985;62:248-56.  Back to cited text no. 1
    
2.
Kiyosue H, Mori H, Sagara Y, Hori Y, Okahara M, Nagatomi H, et al. Basal cerebral venous drainage from cavernous sinus dural arteriovenous fistulas. Neuroradiology 2009;51:175-81.  Back to cited text no. 2
    
3.
Yu J, Guo Y, Zhao S, Xu K. Brainstem edema caused by traumatic carotid-cavernous fistula: A case report and review of the literature. Exp Ther Med 2015;10:445-50.  Back to cited text no. 3
    
4.
Ract I, Drier A, Leclercq D, Sourour N, Gabrieli J, Yger M, et al. Extensive basal ganglia edema caused by a traumatic carotid-cavernous fistula: A rare presentation related to a basal vein of Rosenthal anatomical variation. J Neurosurg 2014;121:63-6.  Back to cited text no. 4
    
5.
Miyamoto N, Naito I, Takatama S, Shimizu T, Iwai T, Shimaguchi H. Clinical and angiographic characteristics of cavernous sinus dural arteriovenous fistulas manifesting as venous infarction and/or intracranial hemorrhage. Neuroradiology 2009;51:53-60.  Back to cited text no. 5
    
6.
Huang YP, Wolf BS. The basal cerebral vein and its tributaries. In: Newton TH, Poos DG, editors Radiology of the Skull and Brain (vol. 2, book 3). St. Louis, Mo: Mosby; 1974. p. 2111-54.  Back to cited text no. 6
    
7.
Suzuki Y, Ikeda H, Shimadu M, Ikeda Y, Matsumotoet K. Variations of the basal vein: Identification using three-dimensional CT angiography. AJNR Am J Neuroradiol 2001;22:670-6.  Back to cited text no. 7
    
8.
Suh DC, Lee JH, Kim SJ, Chung SJ, Choi CG, Kim HJ, et al. new concept in cavernous sinus dural arteriovenous fistula. Stroke 2005;36:1134-9.  Back to cited text no. 8
    
9.
Silvis SM, Lindgren E, Hiltunen S, Devasagayam S, Scheres LJ, Jood K, et al. Postpartum period is a risk factor for cerebral venous thrombosis. Stroke 2019;50:501-3.  Back to cited text no. 9
    


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  [Figure 1], [Figure 2], [Figure 3]



 

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