Unilateral Carotid-Cavernous Fistula Associated With Primitive Persistent Trigeminal Artery Leading to Bilateral Ophthalmic Presentations

In our previous accepted article (1), we described a case with contralateral ocular manifestations of a left primitive; persistent trigeminal artery (PPTA)-induced carotidcavernous fistula (CCF).

Letters to the Editor Nirali Vora, MD Department of Neurology and Neurological Sciences, Stanford University, Stanford, California The authors report no conflicts of interest. REFERENCES 1. Tugizova M, Siegel DT, Huang S, Su E, Subramanian PS, Beres S, Vora N. Case series: atypical optic neuritis after COVID-19 Unilateral Carotid-Cavernous Fistula Associated With Primitive Persistent Trigeminal Artery Leading to Bilateral Ophthalmic Presentations I n our previous accepted article (1), we described a case with contralateral ocular manifestations of a left primitive persistent trigeminal artery (PPTA)-induced carotidcavernous fistula (CCF). Interestingly, 3 months after the previous intervention, she experienced the same problems of her left eye, so she revisited our hospital. On examination, we found conjunctival injection with corkscrew vessel pattern, no ocular vaccination. J Neuroophthalmol. 2022;1–3. (doi: 10.1097/ WNO.0000000000001519). 2. Stubgen J. A literature review of optic neuritis following vaccination against virus infections. Autoimmun Rev. 2013;12:990–997. 3. Helmchen C, Buttler GM, Markewitz R, Hummel K, Wiendl H, Boppel T. Acute bilateral optic/chiasm neuritis with longitudinal extensive transverse myelitis in longstanding stable multiple sclerosis following vector-based vaccination against the SARSCoV-2. J Neurol. 2022;269:49–54. 4. Centers for Disease Control and Prevention. Dengue in the US states and territories. 2020. Available at: https:// www.cdc.gov/dengue/areaswithrisk/in-the-us.html. Accessed January 22, 2022. hypertension, and no motility restriction in the left side (Fig. 1A). Her visual acuities were normal. Digital subtraction angiography demonstrated a new fistula from the basal arterial side of PPTA in the left side (Fig. 2A–C). We performed another coil embolization to repair this (Fig. 2D). Three days later, her left proptosis and conjunctival injection improved (Fig. 1B). Thus, this case turned out to be a unilateral PPTA causing bilateral ophthalmic findings with late onset of the ipsilateral abnormality. Chen et al described a case with bilateral PPTA, but only one fistula was identified (2). In addition, Demartini reported a patient suffering from traumatic unilateral CCF with contralateral ophthalmic manifestations (3). However, this clinical scenario seems to be extremely unique to the best of our knowledge. FIG. 1. Left proptosis and conjunctival injection before (A) and after (B) intervention. e38 Letters to the Editor: J Neuro-Ophthalmol 2023; 43: e37-39 Copyright © North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited. Letters to the Editor FIG. 2. A. Left vertebral angiography showed the fistula from basilar artery into the left cavernous sinus (short arrow) through primitive persistent trigeminal artery (PPTA) (long arrow). B. 3D reconstruction showed the basilar artery (BA), left cavernous sinus (LCS), left orbital vein (LOV). C. The PPTA from the left internal carotid artery (arrow) had already been occluded by coil at previous intervention. D. The PPTA (arrows) was occluded by coil after intervention. Tung T. Hoang, MD Ophthalmology Department, Hanoi Medical University, Hanoi, Vietnam Ophthalmology Unit, Hanoi Medical University Hospital, Hanoi, Vietnam Save Sight Institute, The University of Sydney School of Medicine, Australia Cuong N. Nguyen, MD Radiology Department, Hanoi Medical University Hospital, Hanoi, Vietnam Hieu L. Nguyen, MD, PhD Cardiology Center, Hanoi Medical University Hospital, Hanoi, Vietnam Letters to the Editor: J Neuro-Ophthalmol 2023; 43: e37-39 Prem S. Subramanian, MD, PhD Ophthalmology Department, University of Colorado School of Medicine, Aurora, Colorado The authors report no conflicts of interest. T. T. Hoang and C. N. Nguyen share the co-first authorship. REFERENCES 1. Hoang TT, Nguyen AQ, Nguyen HL, Subramanian PS, Pham VT. Contralateral ocular manifestations of a carotid cavernous fistula associated with primitive persistent trigeminal artery. J Neuroophthalmol Forthcoming 2021. Publish Ahead of Print. Available at: https://journals.lww.com/jneuro-ophthalmology/Citation/9000/ Contralateral_Ocular_Manifestations_of_a_Carotid.98803.aspx. 2. Chen D, Chen CJ, Chen JJ, Tseng YC, Hsu HL, Ku JW. Bilateral persistent trigeminal arteries with unilateral trigeminal artery to cavernous sinus fistula. A case report. Interv Neuroradiol. 2013;19:339–343. 3. Demartini Z Jr, Liebert F, Gatto LA, Jung TS, Rocha C Jr, Santos AM, Unilateral KGL Direct carotid cavernous fistula causing bilateral ocular manifestation. Case Rep Ophthalmol. 2015;6:482–487. e39 Copyright © North American Neuro-Ophthalmology Society. Unauthorized reproduction of this article is prohibited.