A Case of Isolated Third Nerve Palsy with Pupillary Involvement Diagnosed with Cavernous Dural Arteriovenous Fistula

CASE REPORT online © ML Comm J Neurocrit Care 2013;6:126-128 ISSN 2005-0348

Yeo Jung Kim, MD, Suk Yoon Lee, MD, Jin-ho Jung, MD, Jung Hwa Seo, MD, Eung-Gyu Kim, MD, PhD, Ki-Hwan Ji, MD, Jong Seok Bae, MD, and Sang-Jin Kim, MD Department of Neurology, Inje University College of Medicine, Busan Paik Hospital, Busan, Korea

Background: Third nerve palsy can result from lesions located anywhere along its path from the oculomotor nucleus to the nerve termi- nation within the extraocular muscles of the orbit. Common etiologies of isolated third nerve palsy with pupil involvement are intracranial aneurysm, uncal herniation, neoplasia, and traumatic and inflammatory conditions.Case Report: We present a case of a 71-year-old female with complete left third nerve palsy with pupillary involvement. She was diagnosed with cavernous dural arteriovenous fistula (dAVF) using source images of time-of-flight (TOF) magnetic resonance (MR). Cerebral angiography revealed a cavernous dAVF via a branch of the distal intracranial artery. Conclusion: Isolated third nerve palsy may be caused by cavernous dAVF, and TOF MR angiography may be a useful non-invasive pre-diagnostic tool for detecting the shunts. J Neurocrit Care 2013;6:126-128

KEY WORDS: Dural arteriovenous fistula · Third nerve palsy · Magnetic resonance angiography.

Introduction The ophthalmological evaluation revealed complete left ptosis and left third nerve palsy with mydriasis without pupil- Isolated third nerve palsy is associated with variable etiol- lary light reflex. The left eye was deviated downward and out- ogies. However, cavernous dural arteriovenous fistula( CdAVF) ward. The left pupil was dilated 7 mm and did not react to di- involvement in isolated third nerve palsy is rare and not com- rect light stimulus. The right pupil was 3 mm and reacted to monly considered as the cause of the condition. Source im- direct light stimulus. ages of time-of-flight (TOF) magnetic resonance (MR) angi- Physical examination did not reveal conjunctival injection, ography may be more useful for the detection of arteriovenous chemosis, proptosis, or eyelid swelling. The patient denied he- shunts than conventional MR imaging.1-3 We present a case of adache, ocular pain, tinnitus, and a trauma history. She had hy- left isolated third nerve palsy associated with CdAVF. pertension and a history of renal tuberculosis. She had complete left third nerve palsy with pupillary in- Case volvement. Other cranial nerve functions were normal. The brain MR image was normal, and the MR angiography show- A 71-year-old female was referred to the emergency depart- ed no evidence of intracranial aneurysm or vascular malfor- ment as a result of left ptosis and ocular nerve palsy with di- mation. However, source images of TOF MR angiography lated pupil. Three days earlier, she felt a strange sensation of revealed a prominent flow signal in the left cavernous and pe- something moving upward on her left neck. The next morn- trosal sinuses (Fig. 1). Given the putative diagnosis of dAVF ing she had complete left ptosis. in the left cavernous sinus, we performed a cerebral angiog- Her initial vitals were: blood pressure 160/100 mm Hg, raphy, which revealed a left CdAVF in a branch of the menin- pulse rate 78 bpm, and temperature 36.5ºC. gohypophyseal trunk in a distal intracranial artery. Drainage Received: November 22, 2013 / Revised: December 8, 2013 occurred primarily via the inferior petrosal sinus and was not Accepted: December 19, 2013 observed in the cortical or ophthalmic veins (Fig. 2). Our pa- Address for correspondence: Jung Hwa Seo, MD tient was treated conservatively because the amount of flow Department of Neurology, Inje University College of Medicine, Busan Paik Hospital, 75 Bokji-ro, Busanjin-gu, Busan 614-110, Korea through the dAVF shunt was small. One week following treat- Tel: +82-51-890-8768, Fax: +82-51-895-6367 ment, the left pupil was dilated; however, it reacted to direct E-mail: [email protected] light stimulus. One month later, ptosis and eyeball movement

FIGURE 1. In magnetic resonanace angiography (MR) (A), there was no evidence of aneurysm and vascular malformation. However, time of flight MR (B) revealed prominent flow signal in left cavernous and petrous B sinus (B) (arrows).

FIGURE 2. Left intracranial angiography. Cerebral angiography revealed left cavernous dural arteriovenous fistula through dural branch of me- ningohypophyseal trunk. Shunt flow was drained through the intercav- ernous sinus, right inferior petrosal sinus and clival venous plexus (arrows). Engorged ophthalmic vein was not visualized. were partially improved; however, movement of the extraoc- CdAVF is a pathological condition associated with abnor- ular muscle by the left third nerve remained limited. mal connectivity between the meningeal branch of the inter- nal or external carotid artery and the cavernous sinus. CdAVF Discussion is not uncommon in isolated cranial nerve palsy, including third nerve palsy. Several cases studies have shown that the The most common cause of isolated third nerve palsy with third nerve is frequently compromised and isolated third nerve pupillary involvement, as described here, is an aneurysm in palsy is often associated with CdAVF.4,5 A previous case se- the posterior communicating artery. The differential diagnosis ries found that CdAVF was the etiology of isolated third nerve includes intracranial hemorrhage, subarachnoid hemorrhage, palsy in eight of 11 patients.4 bacterial meningitis, cavernous sinus thrombosis, and mid- Congestive ocular symptoms have been shown to be caused brain infraction. CdAVF generally accompanies congestive by retrograde venous flow and/or pressure from cavernous si- ocular symptoms and signs, such as conjunctival injection, nus to ophthalmic vein.6 However, when fistula shunt flow is chemosis, proptosis, eyelid swelling, and ocular pain. directed toward the superior or inferior petrosal sinus rather than the ophthalmic vein, as in our case, isolated ocular motor

127 J Neurocrit Care ▌2013 ;6:126-128 nerve palsy may occur without congestive ocular symptoms.7,8 imaging of dural AV fistulas at the cavernous sinus. J Comput Assist Moreover, superoposterior drainage may compress the third Tomogr 1990;14:397-401. 9 2. Chen JC, Tsuruda JS, Halbach VV. Suspected dural arteriovenous fis- nerve against the roof of cavernous sinus. tula: results with screening MR angiography in seven patients. Radiol- The primary treatment strategies for CdAVF are endovas- ogy 1992;183:265-71. cular management, radiotherapy, and conservative care. Con- 3. Hirai T, Korogi Y, Hamatake S, Ikushima I, Sugahara T, Sigematsu Y, et al. Three-dimensional FISP imaging in the evaluation of carotid cav- servative treatment is appropriate for patients, such as ours ernous fistula: comparison with contrast-enhanced CT and spin-echo 4,6 with benign symptoms and a low-flow shunt. MR. AJNR Am J Neuroradiol 1998;19:253-9. Nevertheless, the diagnosis of CdAVF requires cerebral an- 4. Wu HC, Ro LS, Chen CJ, Chen ST, Lee TH, Chen YC, et al. Isolated ocular motor nerve palsy in dural carotid-cavernous sinus fistula. Eur giography, which is a relatively invasive procedure, and less J Neurol 2006;13:1221-5. intrusive pre-diagnostic decision tools are needed. 5. Kurata A, Suzuki S, Iwamoto K, Nakahara K, Sasaki M, Kijima C, et Contrast-enhanced computed tomography (CT) and spin- al. Dural arteriovenous fistulas in the cavernous sinus: clinical research echo MR imaging can be used to assess CdAVF.1,10 However, and treatment. ISRN Neurol 2011;2011:453834. 6. Choi BS, Park JW, Kim JL, Kim SY, Park YS, Kwon HJ, et al. Treat- in some cases CT, MR, and MR angiography may not be suf- ment Strategy Based on Multimodal Management Outcome of Cav- ficient to visualize a dAVF, and source image of TOF MR an- ernous Sinus Dural Arteriovenous Fistula (CSDAVF). Neurointerven- giography is a superior tool for detecting arteriovenous sh- tion 2011;6:6-12. 2,11 7. Eggenberger E, Lee AG, Forget TR Jr, Rosenwasser R. A bruital head- unts. ache and double vision. Surv Ophthalmol 2000;45:147-53. In our case, source image of TOF MR angiography played 8. Acierno MD, Trobe JD, Cornblath WT, Gebarski SS. Painful oculo- an important role in our pre-angiographic diagnostic deci- motor palsy caused by posterior-draining dural carotid cavernous fistulas. Arch Ophthalmol 1995;113:1045-9. sions. 9. Miyachi S, Negoro M, Handa T, Sugita K. Dural carotid cavernous si- In conclusion, brain MR and MR angiography are useful nus fistula presenting as isolated oculomotor nerve palsy. Surg Neurol methods for the evaluation of isolated third nerve palsy. How- 1993;39:105-9. ever, CdAVF is a rare etiology of isolated third nerve palsy, 10. Uchino A, Hasuo K, Matsumoto S, Masuda K. MRI of dural carotid- cavernous fistulas. Comparisons with postcontrast CT. Clin Imaging and source image of TOF MR angiography may be a useful 1992;16:263-8. non-invasive pre-diagnostic tool for detecting the shunts. 11. Noguchi K, Melhem ER, Kanazawa T, Kubo M, Kuwayama N, Seto H. Intracranial dural arteriovenous fistulas: evaluation with combined REFERENCES 3D time-of-flight MR angiography and MR digital subtraction angiography. AJR Am J Roentgenol 2004;182:183-90. 1. Komiyama M, Fu Y, Yagura H, Yasui T, Hakuba A, Nishimura S. MR

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