Baumann, T.

Lyrer, P.

Radü, E.

Sollberger, M.

Steck, A.

Wetzel, S.

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J Neurol (2005) 252 : 372–373DOI 10.1007/s00415-005-0657-7M. SollbergerP. LyrerT. BaumannE. W. RadüA. J. SteckS. G. WetzelIsolated bilateralabducent nerve palsy dueto a spontaneous left-sidedural carotid cavernousfistula Type Barrow CReceived: 11 May 2004Received in revised form: 7 September 2004Accepted: 10 September 2004Published online: 30 March 2005Sirs: Carotid cavernous fistulas(CCFs) are commonly associatedwith congestive orbito-ocularfeatures – in most cases ipsilateralto the fistula side [1, 2]. As a rareentity, we describe a case of iso-lated bilateral abducent nerve palsydue to a spontaneous left-side CCF,whose presence was proven bymagnetic resonance (MR) digitalsubtraction angiography (DSA).A 63-year old woman developedacute horizontal diplopia associ-ated with nausea, without preced-ing trauma or illness. The symp-toms disappeared within a fewhours, reappeared again 10 dayslater and persisted thereafter. Hermedical history included an essen-tial arterial hypertension.Physical examination showed abilateral, left-dominant abducentnerve palsy and a pulsatile bruitover the left carotid arteries. Onrequest the patient told us about anintermittent, pulsatile left-sidetinnitus, which had started about 6months prior to the occurrence ofthe diplopia. The funduscopicexamination and the visual acuitywere normal. Time-of-flight MRangiography of the cerebral vesselsand color-coded Doppler sonogra-phy were normal. MR DSA [3]showed filling of the cavernoussinus during the early arterialphase (Fig. 1A), proving the pres-ence of a CCF. A second moresubtle finding – only detected afterfurther analysis of the MR DSAimages – was the presence of smallflow voids adjacent to the cav-ernous segment of the internalcarotid artery on T2-weighted MRimages of the brain (Fig. 1B). Onintra-arterial DSA the fistula wasseen to be filled only by branchesof the left maxillary artery(Fig. 1C). Cavernous outflowdrained into the left superiorophthalmic vein, which showed afocal stenosis in its proximal seg-ment, into the left middle cerebralvein and bilaterally into petrosalveins.The embolization with micro-particles of the major feedingbranches arising from the maxil-lary and the middle meningealarteries resulted in a marked re-duction of the fistula flow. A fewhours after embolization the pul-satile tinnitus disappeared andwithin about 10 weeks the bilateralabducent nerve palsy graduallyregressed. MR DSA 6 weeks afterembolization showed signs of aslight persistent fistula, and intra-arterial DSA 18 weeks after em-bolization disclosed a completeocclusion of the fistula.Our patient presented anisolated left-dominant bilateralabducent nerve palsy due to aspontaneous left-side dural CCFType Barrow C [4]. Apart from thediplopia the only other clinicalsymptom was a left-side pulsatiletinnitus, which is a well-knownfeature of dural arteriovenousfistulas (DAVFs).Abducent nerve palsy is themost common cranial nerve palsyassociated with CCF [2], but iso-lated bilateral abducent nerve palsydue to a unilateral CCF is a rareentity [5]. Isolated abducent nerveLETTER TO THE EDITORSJON 1657Fig. 1 A: MR DSA image during the early arterialphase. Note the early flow into the cavernous sinus(arrow) from a prominent artery (arrowhead) on theleft side. From the anatomical course the feeding ves-sel is probably the maxillary artery. However, becausethere is only one image plane (frontal view), leadingto limited spatial resolution compared to conven-tional DSA, a detailed characterisation of the feedingvessels is not possible. B: T2-weighted image at thelevel of the cavernous sinus. Note the signal loss lo-cated medially to the internal carotid artery (arrow),representing flow-voids caused by fast flowing ves-sels of the fistula. C: Conventional DSA image (ob-tained in the arterial phase with the catheter posi-tioned in the left maxillary artery) depicts multiplefeeders of the CCF0372_0373_Sollberger_JON_1657  29.03.2005  14:44 Uhr  Seite 3723734. Barrow DL, Spector RH, et al. (1985)Classification and treatment of sponta-neous carotid-cavernous sinus fistulas. JNeurosurg 62(2):248–2565. Lee KY, Kim SM, et al. (1998) Isolated bi-lateral abducens nerve palsy due tocarotid cavernous dural arteriovenousfistula. Yonsei Med J 39(3):283–2866. Leonard TJ, Moseley IF, et al. (1984)Ophthalmoplegia in carotid cavernoussinus fistula. Br J Ophthalmol 68(2):128–1347. Mironov A (1994) Pathogenetical con-sideration of spontaneous dural arteri-ovenous fistulas (DAVFs). Acta Neu-rochir (Wien) 131(1–2):45–588. Cloft HJ, Jensen ME, et al. (1998) Elec-tive stenting of the extracranial carotidarteries. Acad Radiol 5(8):578–580; dis-cussion 581–582M. Sollberger () · P. Lyrer · T. Baumann ·E. W. Radü · A. J. Steck · S. G. WetzelDept. of NeurologyUniversity Hospital of BaselPetersgraben 44031 Basel, Switzerlandpalsy is thought to be less due tothe mass effect of the distendedsinuses, than to the venous hyper-tension with nerve ischemia asso-ciated with arteriovenous shunting[1, 6]. Interestingly orbito-ocularcongestive features were not ob-served in our case, despite the ante-grade drainage into the superiorophthalmic vein. The reason forthis was most likely the segmentalnarrowing of the vein in its proxi-mal course, leading also to retro-grade cavernous outflow. This nar-rowing, which has already beendescribed in cases with DAVFs [7],probably represents a postthrom-botic state.The exact anatomical depictionof the fistula was only possible withthe high spatial and temporal reso-lution of intra-arterial DSA, whichis without doubt the method of ref-erence for imaging these patholo-gies. On the other hand, intra-arterial DSA is time-consuming,expensive, and bears a small risk ofneurological complications [8]. Wetherefore advocate to incorporateinto the imaging algorithm MRDSA, a two-dimensional, contrast-enhanced angiographic techniquewith a high temporal resolutionwhich proved useful in our case notonly for the detection but as wellfor the follow-up of the treatedfistula.References1. Meyers PM, Halbach VV, et al. (2002)Dural carotid cavernous fistula: defini-tive endovascular management andlong-term follow-up. Am J Ophthalmol134(1):85–922. Klisch J, Huppertz HJ, et al. (2003)Transvenous treatment of carotid cav-ernous and dural arteriovenous fistulae:results for 31 patients and review of theliterature. Neurosurgery 53(4):836–856;discussion 856–8573. Wetzel SG, Bilecen D, et al. (2000) Cere-bral dural arteriovenous fistulas: detec-tion by dynamic MR projection angiog-raphy. AJR Am J Roentgenol 174(5):1293–12950372_0373_Sollberger_JON_1657  29.03.2005  14:44 Uhr  Seite 373

Isolated bilateralabducent nerve palsy dueto a spontaneous left-sidedural carotid cavernousfistula Type Barrow C

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Isolated bilateralabducent nerve palsy dueto a spontaneous left-sidedural carotid cavernousfistula Type Barrow C

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