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• W2268832644 abstract "Intraorbital arteriovenous fistulas (AVFs) are a rare subtype of dural AVF (dAVF). Dural AVFs are abnormal direct connections (fistulas) between a meningeal artery and a dural venous sinus. They make up 10%–15% of all AV shunting cerebral vascular malformations.1Newton TH Cronqvist S. Involvement of dural arteries in intracranial arteriovenous malformations.Radiology. 1969; 93: 1071-1078Crossref PubMed Scopus (407) Google Scholar The incidence of dAVF at various locations as reported in literature is as follows: transverse sinus 50%, cavernous sinus 16%, tentorium cerebelli 12%, and superior sagittal sinus 8%.2Lasjaunias P Chiu M ter Brugge K et al.Neurological manifestations of intracranial dural arteriovenous malformations.J Neurosurg. 1986; 64: 724-730Crossref PubMed Scopus (512) Google Scholar AV shunts in the orbit are quite rare. Only 5 cases of intraorbital AVFs were reported in the literature based on a search of the Medline database. The first 2 documented cases of intraorbital AVFs were described by Serbinenko in 1978.3Serbinenko FA Padalko PI. [Orbital arteriovenous anastomoses].Zh Vopr Neirokhir Im N N Burdenko. 1978; : 16-21Google Scholar In 1999, Ohtsuka reported a patient with spontaneous AVFs of the orbit.4Ohtsuka K Hashimoto M. Clinical findings in a patient with spontaneous arteriovenous fistulas of the orbit.Am J Ophthalmol. 1999; 127: 736-737Abstract Full Text Full Text PDF PubMed Scopus (21) Google Scholar Caragine described 2 cases of intraorbital AVFs treated by transvenous endovascular occlusion.5Caragine Jr., LP Halbach VV Dowd CF Higashida RT. Intraorbital arteriovenous fistulae of the ophthalmic veins treated by transvenous endovascular occlusion: technical case report.Neurosurgery. 2006; 58 (ONS-E170; discussion ONS-E)Google ScholarClinical ophthalmic symptoms and signs of dAVFs are similar to the ocular features of carotid-cavernous sinus fistulas (CCF), such as proptosis, diplopia, and engorged episcleral vessels.6de Keizer R. Carotid-cavernous and orbital arteriovenous fistulas: ocular features, diagnostic and hemodynamic considerations in relation to visual impairment and morbidity.Orbit. 2003; 22: 121-142Crossref PubMed Scopus (138) Google Scholar Symptoms are caused by increased pressure in the cavernous sinus, which is transferred to the orbit through the superior and inferior ophthalmic veins.Superior ophthalmic vein thrombosis is usually found in cases of orbital cellulitis, idiopathic orbital inflammation, and vascular malformation.7Lai PF Cusimano MD. The spectrum of cavernous sinus and orbital venous thrombosis: a case and a review.Skull Base Surg. 1996; 6: 53-59Crossref PubMed Scopus (25) Google Scholar Stiebel-Kalish observed angiographic evidence of ophthalmic vein thrombosis in 11 of 85 patients with dural CCF.8Stiebel-Kalish H Setton A Nimii Y et al.Cavernous sinus dural arteriovenous malformations: patterns of venous drainage are related to clinical signs and symptoms.Ophthalmology. 2002; 109: 1685-1691Abstract Full Text Full Text PDF PubMed Scopus (76) Google Scholar Ophthalmic vein thrombosis secondary to AVF is associated with endothelial damage caused by high blood flow and the venous stasis from the fistula.Orbital AVFs are low-flow fistulas, like indirect CCFs, which sometimes close spontaneously.6de Keizer R. Carotid-cavernous and orbital arteriovenous fistulas: ocular features, diagnostic and hemodynamic considerations in relation to visual impairment and morbidity.Orbit. 2003; 22: 121-142Crossref PubMed Scopus (138) Google Scholar However, if progressive vision loss and severe proptosis occur, surgery is necessary. Transvenous embolization is generally used as the first treatment. To the best of our knowledge, this is the first report of a rare case of intraorbital AVF of the ophthalmic vein treated successfully using thrombosed superior ophthalmic vein approach.An 81-year-old woman came to the emergency department with progressive eyelid swelling and conjunctival chemosis over the right eye for 3 weeks (Fig. 1). The right eye also had proptosis, ptosis, and limited movement in all directions. There was no history of head trauma. The patient had hypertension and a history of partial mastectomy for right breast cancer. The patient had raised intraocular pressure in the right eye (23 mm Hg). Visual acuity was 6/30 in the right eye and 6/15 in the left eye. Fundal examination revealed diffuse retinal hemorrhage in the right eye. Orbital computed tomography reported engorgement and thrombosis of the right superior ophthalmic vein (SOV; Fig 2). Cerebral angiography demonstrated early opacification of reverse flow of right inferior ophthalmic vein with feeding arteries from small branch of right internal carotid artery (ICA) and external carotid artery (ECA) but no cavernous sinus enhancement (Fig. 3). Nonvisualization of right superior ophthalmic vein was compatible with thrombosis, which was also detected by computed tomography. A schematic illustration of this dual-ophthalmic vein fistula is shown in Figure 4.Figure 2Computed tomography showed right eye proptosis and engorged right superior ophthalmic vein.View Large Image Figure ViewerDownload (PPT)Figure 3A, Right external common carotid angiogram showing the dual arteriovenous fistula. Note opacification of the right inferior ophthalmic vein (arrow). B, Right internal common carotid angiogram showing the dual arteriovenous fistula. Note opacification of the right inferior ophthalmic vein (double arrow). C, Right carotid angiogram revealed the fistula is obliterated. ECA, external carotid artery; ICA, internal carotid artery.View Large Image Figure ViewerDownload (PPT)Figure 4Schematic illustration of dual arteriovenous fistula with thrombotic superior ophthalmic vein. SOV, superior ophthalmic vein; IOV, inferior ophthalmic v; ICA, internal carotid artery; ECA, external carotid artery.View Large Image Figure ViewerDownload (PPT)Transvenous embolization via the petrosal sinus was attempted but could not be advanced further. Therefore, direct exposure of the right SOV via anterior orbitotomy was performed in the operating room under general anesthesia (Fig. 5). The vessel was punctured with an angiocatheter and the guidewire was slowly advanced and passed through the thrombosed SOV. When the vein was punctured, there was no bleeding; once the thrombosed segment was passed, the catheter filled with blood in a pulsatile fashion. A microcatheter was then inserted through the angiocatheter. The inferior ophthalmic vein was packed with cyanoacrylate glue delivered through the microcatheter under fluoroscopic guidance. Cerebral angiography after embolization showed occlusion of the fistula (Fig. 3). One month after embolization, vision in the left eye recovered to 6/20 and the right extraocular muscles could all move freely. It showed complete remission in right eye.Figure 5Direct exposure engorged superior ophthalmic vein by anterior orbitotomy.View Large Image Figure ViewerDownload (PPT)Orbital AVFs are low-flow fistulas and rarely life threatening. Closure of fistula should be considered in patients with neurologic symptoms or progressive vision loss. The ocular symptoms most often are mild in the case of indirect CCFs. The severity in the present case was related to the direct connection from ECA and ICA to the ophthalmic vein.Serbinenko described 2 cases with orbital AV anastomoses between the branches of the internal maxillary artery and inferior ophthalmic vein. Surgical management consisted of ligation of the internal maxillary artery with muscular emboli.3Serbinenko FA Padalko PI. [Orbital arteriovenous anastomoses].Zh Vopr Neirokhir Im N N Burdenko. 1978; : 16-21Google Scholar Caragine reported 2 cases of intraorbital AVF. One had superior ophthalmic vein supplied by the ophthalmic artery and the anterior division of the middle meningeal artery, whereas the other AVF was between the internal maxillary artery and inferior orbital varix.5Caragine Jr., LP Halbach VV Dowd CF Higashida RT. Intraorbital arteriovenous fistulae of the ophthalmic veins treated by transvenous endovascular occlusion: technical case report.Neurosurgery. 2006; 58 (ONS-E170; discussion ONS-E)Google Scholar Ohtsuka described a case in which the ophthalmic and facial artery supplied the superior ophthalmic vein.4Ohtsuka K Hashimoto M. Clinical findings in a patient with spontaneous arteriovenous fistulas of the orbit.Am J Ophthalmol. 1999; 127: 736-737Abstract Full Text Full Text PDF PubMed Scopus (21) Google Scholar No thrombosed ophthalmic vein was noted in the 5 cases just described. In the present case, the fistula was between the right ophthalmic vein and small branch of the right ICA and ECA. Two branches of the carotid artery flowing into the ophthalmic artery induced a higher blood flow and resulted in severe thrombosis of SOV. The inferior ophthalmic vein was partially thrombosed.Venous thrombi are caused mainly by venous stasis and hypercoagulability. Vessel bifurcation sites proximal to the fistula are prone to development of thrombosis.9Sivanesan S How T.V Bakran A Sites of stenosis in AV fistulae for haemodialysis access.Nephrol Dial Transplant. 1999; 14: 118-120Crossref PubMed Scopus (101) Google Scholar Acute angulation, relative narrowness, and bifurcation in the SOV wound explain the development of thrombosis in the present case. Thrombosis of the SOV is generally associated with exacerbation of orbital symptoms, as evidenced by the severe conjuncitval chemosis and diplopia in all gaze direction in this case.8Stiebel-Kalish H Setton A Nimii Y et al.Cavernous sinus dural arteriovenous malformations: patterns of venous drainage are related to clinical signs and symptoms.Ophthalmology. 2002; 109: 1685-1691Abstract Full Text Full Text PDF PubMed Scopus (76) Google ScholarCatheterization of the thrombosed SOV may be difficult because of the fibrotic and fragile structure of the vein, resulting in failure to approach to the aim site. However, SOV approach provides a direct and short access to the cavernous sinus and enables complete closure of the fistula. In Yilmaz’s series, dural CCFs can be successfully treated through the SOV, even when the vein is thrombosed.10Guven Yilmaz S Yazici B Cetinkaya A Yagci A. Embolization of dural carotid-cavernous fistulas via the thrombosed superior ophthalmic vein.Ophthal Plast Reconstr Surg. 2013; 29: 272-276Crossref Scopus (6) Google Scholar In the present case, we exposed the SOV in a direct anterior orbitotomy as the engorged thrombosed SOV is easy to identify and the procedure, with a small incision wound, is effortless. During the procedure, the angiocatheter passed through the thombosed SOV successfully and embolized the ophthalmic vein smoothly. Additionally, Badilla embolized the CCF through a lateral orbitotomy by avoiding the thrombosed segment after an unsuccessful anterior cannulation approach.11Badilla J Haw C Rootman J. Superior ophthalmic vein cannulation through a lateral orbitotomy for embolization of a cavernous dural fistula.Arch Ophthalmol. 2007; 125: 1700-1702Crossref Scopus (16) Google Scholar We concluded that the dissection can be performed more posteriorly or via lateral orbitotomy when difficult to advance the catheter through a thrombosed SOV. Transvenous embolization of CCF by direct surgical approach has been described in some cases12Baldauf J Spuler A Hoch HH et al.Embolization of indirect carotid-cavernous sinus fistulas using the superior ophthalmic vein approach.Acta Neurol Scand. 2004; 110: 200-204Crossref PubMed Scopus (26) Google Scholar and case series.13Gioulekas J Mitchell P Tress B McNab AA. Embolization of carotid cavernous fistulas via the superior ophthalmic vein.Aust N Z J Ophthalmol. 1997; 25: 47-53Crossref PubMed Scopus (25) Google Scholar, 14Goldberg RA Goldey SH Duckwiler G Vinuela F. Management of cavernous sinus-dural fistulas. Indications and techniques for primary embolization via the superior ophthalmic vein.Arch Ophthalmol. 1996; 114: 707-714Crossref PubMed Scopus (118) Google Scholar, 15Miller NR Monsein LH Debrun GM et al.Treatment of carotid-cavernous sinus fistulas using a superior ophthalmic vein approach.J Neurosurg. 1995; 83: 838-842Crossref PubMed Scopus (124) Google Scholar To our knowledge, treatment of an intraorbital AVF via the superior ophthalmic vein in the present case has not been reported in the literature.ConclusionWe found that transvenous embolization through an enlarged and thrombotic SOV is a safe, direct, and efficient method that allows endovascular glue occlusion of dural AVF of the ophthalmic vein with excellent angiographic and clinical results. Intraorbital arteriovenous fistulas (AVFs) are a rare subtype of dural AVF (dAVF). Dural AVFs are abnormal direct connections (fistulas) between a meningeal artery and a dural venous sinus. They make up 10%–15% of all AV shunting cerebral vascular malformations.1Newton TH Cronqvist S. Involvement of dural arteries in intracranial arteriovenous malformations.Radiology. 1969; 93: 1071-1078Crossref PubMed Scopus (407) Google Scholar The incidence of dAVF at various locations as reported in literature is as follows: transverse sinus 50%, cavernous sinus 16%, tentorium cerebelli 12%, and superior sagittal sinus 8%.2Lasjaunias P Chiu M ter Brugge K et al.Neurological manifestations of intracranial dural arteriovenous malformations.J Neurosurg. 1986; 64: 724-730Crossref PubMed Scopus (512) Google Scholar AV shunts in the orbit are quite rare. Only 5 cases of intraorbital AVFs were reported in the literature based on a search of the Medline database. The first 2 documented cases of intraorbital AVFs were described by Serbinenko in 1978.3Serbinenko FA Padalko PI. [Orbital arteriovenous anastomoses].Zh Vopr Neirokhir Im N N Burdenko. 1978; : 16-21Google Scholar In 1999, Ohtsuka reported a patient with spontaneous AVFs of the orbit.4Ohtsuka K Hashimoto M. Clinical findings in a patient with spontaneous arteriovenous fistulas of the orbit.Am J Ophthalmol. 1999; 127: 736-737Abstract Full Text Full Text PDF PubMed Scopus (21) Google Scholar Caragine described 2 cases of intraorbital AVFs treated by transvenous endovascular occlusion.5Caragine Jr., LP Halbach VV Dowd CF Higashida RT. Intraorbital arteriovenous fistulae of the ophthalmic veins treated by transvenous endovascular occlusion: technical case report.Neurosurgery. 2006; 58 (ONS-E170; discussion ONS-E)Google Scholar Clinical ophthalmic symptoms and signs of dAVFs are similar to the ocular features of carotid-cavernous sinus fistulas (CCF), such as proptosis, diplopia, and engorged episcleral vessels.6de Keizer R. Carotid-cavernous and orbital arteriovenous fistulas: ocular features, diagnostic and hemodynamic considerations in relation to visual impairment and morbidity.Orbit. 2003; 22: 121-142Crossref PubMed Scopus (138) Google Scholar Symptoms are caused by increased pressure in the cavernous sinus, which is transferred to the orbit through the superior and inferior ophthalmic veins. Superior ophthalmic vein thrombosis is usually found in cases of orbital cellulitis, idiopathic orbital inflammation, and vascular malformation.7Lai PF Cusimano MD. The spectrum of cavernous sinus and orbital venous thrombosis: a case and a review.Skull Base Surg. 1996; 6: 53-59Crossref PubMed Scopus (25) Google Scholar Stiebel-Kalish observed angiographic evidence of ophthalmic vein thrombosis in 11 of 85 patients with dural CCF.8Stiebel-Kalish H Setton A Nimii Y et al.Cavernous sinus dural arteriovenous malformations: patterns of venous drainage are related to clinical signs and symptoms.Ophthalmology. 2002; 109: 1685-1691Abstract Full Text Full Text PDF PubMed Scopus (76) Google Scholar Ophthalmic vein thrombosis secondary to AVF is associated with endothelial damage caused by high blood flow and the venous stasis from the fistula. Orbital AVFs are low-flow fistulas, like indirect CCFs, which sometimes close spontaneously.6de Keizer R. Carotid-cavernous and orbital arteriovenous fistulas: ocular features, diagnostic and hemodynamic considerations in relation to visual impairment and morbidity.Orbit. 2003; 22: 121-142Crossref PubMed Scopus (138) Google Scholar However, if progressive vision loss and severe proptosis occur, surgery is necessary. Transvenous embolization is generally used as the first treatment. To the best of our knowledge, this is the first report of a rare case of intraorbital AVF of the ophthalmic vein treated successfully using thrombosed superior ophthalmic vein approach. An 81-year-old woman came to the emergency department with progressive eyelid swelling and conjunctival chemosis over the right eye for 3 weeks (Fig. 1). The right eye also had proptosis, ptosis, and limited movement in all directions. There was no history of head trauma. The patient had hypertension and a history of partial mastectomy for right breast cancer. The patient had raised intraocular pressure in the right eye (23 mm Hg). Visual acuity was 6/30 in the right eye and 6/15 in the left eye. Fundal examination revealed diffuse retinal hemorrhage in the right eye. Orbital computed tomography reported engorgement and thrombosis of the right superior ophthalmic vein (SOV; Fig 2). Cerebral angiography demonstrated early opacification of reverse flow of right inferior ophthalmic vein with feeding arteries from small branch of right internal carotid artery (ICA) and external carotid artery (ECA) but no cavernous sinus enhancement (Fig. 3). Nonvisualization of right superior ophthalmic vein was compatible with thrombosis, which was also detected by computed tomography. A schematic illustration of this dual-ophthalmic vein fistula is shown in Figure 4. Transvenous embolization via the petrosal sinus was attempted but could not be advanced further. Therefore, direct exposure of the right SOV via anterior orbitotomy was performed in the operating room under general anesthesia (Fig. 5). The vessel was punctured with an angiocatheter and the guidewire was slowly advanced and passed through the thrombosed SOV. When the vein was punctured, there was no bleeding; once the thrombosed segment was passed, the catheter filled with blood in a pulsatile fashion. A microcatheter was then inserted through the angiocatheter. The inferior ophthalmic vein was packed with cyanoacrylate glue delivered through the microcatheter under fluoroscopic guidance. Cerebral angiography after embolization showed occlusion of the fistula (Fig. 3). One month after embolization, vision in the left eye recovered to 6/20 and the right extraocular muscles could all move freely. It showed complete remission in right eye. Orbital AVFs are low-flow fistulas and rarely life threatening. Closure of fistula should be considered in patients with neurologic symptoms or progressive vision loss. The ocular symptoms most often are mild in the case of indirect CCFs. The severity in the present case was related to the direct connection from ECA and ICA to the ophthalmic vein. Serbinenko described 2 cases with orbital AV anastomoses between the branches of the internal maxillary artery and inferior ophthalmic vein. Surgical management consisted of ligation of the internal maxillary artery with muscular emboli.3Serbinenko FA Padalko PI. [Orbital arteriovenous anastomoses].Zh Vopr Neirokhir Im N N Burdenko. 1978; : 16-21Google Scholar Caragine reported 2 cases of intraorbital AVF. One had superior ophthalmic vein supplied by the ophthalmic artery and the anterior division of the middle meningeal artery, whereas the other AVF was between the internal maxillary artery and inferior orbital varix.5Caragine Jr., LP Halbach VV Dowd CF Higashida RT. Intraorbital arteriovenous fistulae of the ophthalmic veins treated by transvenous endovascular occlusion: technical case report.Neurosurgery. 2006; 58 (ONS-E170; discussion ONS-E)Google Scholar Ohtsuka described a case in which the ophthalmic and facial artery supplied the superior ophthalmic vein.4Ohtsuka K Hashimoto M. Clinical findings in a patient with spontaneous arteriovenous fistulas of the orbit.Am J Ophthalmol. 1999; 127: 736-737Abstract Full Text Full Text PDF PubMed Scopus (21) Google Scholar No thrombosed ophthalmic vein was noted in the 5 cases just described. In the present case, the fistula was between the right ophthalmic vein and small branch of the right ICA and ECA. Two branches of the carotid artery flowing into the ophthalmic artery induced a higher blood flow and resulted in severe thrombosis of SOV. The inferior ophthalmic vein was partially thrombosed. Venous thrombi are caused mainly by venous stasis and hypercoagulability. Vessel bifurcation sites proximal to the fistula are prone to development of thrombosis.9Sivanesan S How T.V Bakran A Sites of stenosis in AV fistulae for haemodialysis access.Nephrol Dial Transplant. 1999; 14: 118-120Crossref PubMed Scopus (101) Google Scholar Acute angulation, relative narrowness, and bifurcation in the SOV wound explain the development of thrombosis in the present case. Thrombosis of the SOV is generally associated with exacerbation of orbital symptoms, as evidenced by the severe conjuncitval chemosis and diplopia in all gaze direction in this case.8Stiebel-Kalish H Setton A Nimii Y et al.Cavernous sinus dural arteriovenous malformations: patterns of venous drainage are related to clinical signs and symptoms.Ophthalmology. 2002; 109: 1685-1691Abstract Full Text Full Text PDF PubMed Scopus (76) Google Scholar Catheterization of the thrombosed SOV may be difficult because of the fibrotic and fragile structure of the vein, resulting in failure to approach to the aim site. However, SOV approach provides a direct and short access to the cavernous sinus and enables complete closure of the fistula. In Yilmaz’s series, dural CCFs can be successfully treated through the SOV, even when the vein is thrombosed.10Guven Yilmaz S Yazici B Cetinkaya A Yagci A. Embolization of dural carotid-cavernous fistulas via the thrombosed superior ophthalmic vein.Ophthal Plast Reconstr Surg. 2013; 29: 272-276Crossref Scopus (6) Google Scholar In the present case, we exposed the SOV in a direct anterior orbitotomy as the engorged thrombosed SOV is easy to identify and the procedure, with a small incision wound, is effortless. During the procedure, the angiocatheter passed through the thombosed SOV successfully and embolized the ophthalmic vein smoothly. Additionally, Badilla embolized the CCF through a lateral orbitotomy by avoiding the thrombosed segment after an unsuccessful anterior cannulation approach.11Badilla J Haw C Rootman J. Superior ophthalmic vein cannulation through a lateral orbitotomy for embolization of a cavernous dural fistula.Arch Ophthalmol. 2007; 125: 1700-1702Crossref Scopus (16) Google Scholar We concluded that the dissection can be performed more posteriorly or via lateral orbitotomy when difficult to advance the catheter through a thrombosed SOV. Transvenous embolization of CCF by direct surgical approach has been described in some cases12Baldauf J Spuler A Hoch HH et al.Embolization of indirect carotid-cavernous sinus fistulas using the superior ophthalmic vein approach.Acta Neurol Scand. 2004; 110: 200-204Crossref PubMed Scopus (26) Google Scholar and case series.13Gioulekas J Mitchell P Tress B McNab AA. Embolization of carotid cavernous fistulas via the superior ophthalmic vein.Aust N Z J Ophthalmol. 1997; 25: 47-53Crossref PubMed Scopus (25) Google Scholar, 14Goldberg RA Goldey SH Duckwiler G Vinuela F. Management of cavernous sinus-dural fistulas. Indications and techniques for primary embolization via the superior ophthalmic vein.Arch Ophthalmol. 1996; 114: 707-714Crossref PubMed Scopus (118) Google Scholar, 15Miller NR Monsein LH Debrun GM et al.Treatment of carotid-cavernous sinus fistulas using a superior ophthalmic vein approach.J Neurosurg. 1995; 83: 838-842Crossref PubMed Scopus (124) Google Scholar To our knowledge, treatment of an intraorbital AVF via the superior ophthalmic vein in the present case has not been reported in the literature. ConclusionWe found that transvenous embolization through an enlarged and thrombotic SOV is a safe, direct, and efficient method that allows endovascular glue occlusion of dural AVF of the ophthalmic vein with excellent angiographic and clinical results. We found that transvenous embolization through an enlarged and thrombotic SOV is a safe, direct, and efficient method that allows endovascular glue occlusion of dural AVF of the ophthalmic vein with excellent angiographic and clinical results." @default.
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• W2268832644 title "Intraorbital arteriovenous fistula of the ophthalmic vein—embolization using the thombosed superior ophthalmic vein approach" @default.
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