Department of Neurosurgery, Iwate Medical University (AO), Division of Neurosurgery, Institute of Brain Diseases (TT, TY) and Department of Ophthalmology (MK), Tohoku University School of Medicine, Sendai, Japan Neurosurgery 31; 1102-1104, 1992 ABSTRACT: A RARE CASE is presented of a nonruptured aneurysm of the intraorbital ophthalmic artery in which successful resection of the aneurysm resulted in improvement of preoperative progressive signs caused by the mass ef-fect of the aneurysm. The surgical management of this rare entity is dis-cussed with special attention given to the collateral circulation of the ophthalmic artery from the external carotid artery. KEY WORDS: Aneurysm; Collateral circulation; Ophthalmic artery; Orbit An intraorbital ophthalmic artery aneurysm is extremely rare, compared with a carotid-ophthalmic aneurysm arising from the wall of the internal carotid artery (2). We report a case of intraorbital ophthalmic aneurysm in which the patient presented with progressive symptoms of central scotoma with exophthalmos. The aneurysm was successfully resected, and visual function was maintained. When surgery is required, preservation of visual function should be considered. Based on our operative findings, surgical treat-ment of the aneurysm of the intraorbital segment of the ophthalmic artery is discussed. CASE REPORT A 63-year-old woman who had exophthalmos and a 2-week history of worsening of right visual acuity was referred. Her visual acuity was 20/200 in the right eye and 20/20 in the left eye. She had corneal opacity and posterior synechia in the right eye caused by old keratitis that occurred at the age of 10. She also had central scotoma in the right eye that was progressive during a period of 3 months. Examination At admission, the patient showed 2 mm of exophthalmos, enough visual acuity in the right eye to count fingers, a pale optic disc, and a central scotoma of the right visual field. Visual functions in the left eye were normal. Third and 6th nerve palsies and Marcus Gunn pupil of the right eye were also noted. A computed tomographic scan and magnetic resonance imaging disclosed a deep orbital mass and
Surgery Right orbital unroofing by a subfrontal-pterional approach disclosed a saccular aneurysm, 5 mm in diameter, near the orbital apex and on the ophthalmic artery running along the inferolateral aspect of the optic nerve (the first part of the ophthalmic artery, as defined by Hayreh and Dass [4]). The ophthalmic artery crossed over the optic nerve (the second part), and the lacrimal artery stemmed from the angular portion of the ophthalmic artery, which was distal to the aneurysm. The thick wall of the aneurysm was tightly attached to the inferolateral aspect of the optic nerve and compressed it. Because the aneurysm was contiguous with the ophthalmic artery, neck clipping of the aneurysm was impossible. Therefore, it was trapped and resected. Histological findings Histological findings showed the pseudolumen of the resected aneurysm to have formed in the arterial wall. The thickened wall was mostly composed of collagenous tissue, and both lamina elastica and muscularis were absent. Neither atherosclerotic changes nor any inflammatory process was observed. Postoperative course One month postoperatively, the patient had only light perception in the temporal field of the right eye. She showed 3rd nerve palsy with ptosis. With the exception of optic atrophy, in spite of the sacrifice of the ophthalmic artery, her fundi were not remarkable. Four months after the operation, eye movement had fully recovered, visual acuity had improved, and she could again count fingers. A postoperative carotid angiogram demonstrated a filling defect of the ophthalmic artery and disappearance of the ophthalmic aneurysm. The choroidal crescent was fed by a branch of the right external carotid artery. DISCUSSION Because there have been few detailed reports on intraorbital ophthalmic aneurysms, their clinical features are not fully understood. Common neurological symptoms and signs include progressive reduction of visual acuity, visual field defect, and exophthalmos, all of which derive from the mass effect of the aneurysm (7,10-13). Rupture of the intraorbital ophthalmic aneurysm is an extremely rare occurrence. Meyerson and Lazar (9), however, reported a case that showed a sudden onset of blindness and total ophthalmoplegia with massive subconjunctival and periorbital hemorrhage. Despite the removal of the intraorbital aneurysm and hematoma, visual acuity remained no light
Redistribution of this article permitted only in accordance with the publisher’s copyright provisions.
AUTHOR(S): Ogawa, Akira, M.D.; Tominaga, Teiji, M.D.; Yoshimoto, Takashi, M.D.; Kiyosawa, Motohiro, M.D.
slight exophthalmos in the right eye (Fig. 1). A carotid angiogram revealed an aneurysm arising from the intraorbital segment of the ophthalmic artery (Fig. 2). Because the aneurysm was located on the inferolateral side of the optic nerve near the orbital apex, compression of the optic nerve and 3rd and 6th nerves by this aneurysm was considered to be responsible for both the reduction of visual acuity and the disturbance of eye movement.
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Neurosurgery 1992-98 December 1992, Volume 31, Number 6 1102 Intraorbital Ophthalmic Artery Aneurysm: Case Report Case Report
Received, November 19, 1991. Accepted, July 7, 1992. Reprint requests: Akira Ogawa, M.D., Department of Neurosurgery, Iwate Medical University, 19-1, Uchimaru, Morioka, Japan 020.
1.
2. 3. 4. 5. 6. 7. 8.
9. 10. 11. 12.
13.
Brown GC, Magargal LE, Sergott R: Acute obstruction of the retinal and choroidal circulations. Ophthalmology 93:1373-1382, 1986. Day AL: Aneurysms of the ophthalmic segment: A clinical and anatomical analysis. J Neurosurg 72:677-691, 1990. Gillilan LA: The collateral circulation of the human orbit. Arch Ophthalmol 65:684-694, 1961. Hayreh SS, Das R: The ophthalmic artery II. Intraorbital course. Br J Ophthalmol 46:165185, 1962. Hayreh SS: The ophthalmic artery III. Branches. Br J Ophthalmol 46:212-247, 1962. Hayreh SS: Arteries of the orbit in the human being. Br J Surg 50:938-953, 1963. Jain KK: Saccular aneurysm of the ophthalmic artery. Am J Ophthalmol 69:997-998, 1970. Johnson HC, Walker AE: Angiographic diagnosis of spontaneous thrombosis of the internal and common carotid arteries. J Neurosurg 8:631- 659, 1951. Meyerson L, Lazar SJ: Intraorbital aneurysm of the ophthalmic artery. Br J Ophthalmol 55:199- 204, 1971. Raitta C: Ophthalmic artery aneurysm causing optic atrophy and enlargement of the optic foramen. Br J Ophthalmol 52:707-709, 1968. Rengachary SS, Kishore PRS: Intraorbital ophthalmic aneurysms and arteriovenous fistulae. Surg Neurol 9:35-41, 1978. Rubinstein MK, Wilson G, Levin DC: Intraorbital aneurysms of the ophthalmic artery: Report of a unique case and review of the literature. Arch Ophthalmol 80:42-44, 1968. Wheeler EC, Baker HL: The ophthalmic arterial complex in angiographic diagnosis. Radiology 83:26-35, 1964.
COMMENTS The authors have reported an extremely rare form of cerebrovascular disease: an intraorbital ophthalmic artery aneurysm producing visual loss and ophthalmoparesis. Their case is even more unique because their histological description suggests a dissecting aneurysm. Their management by excision was apparently very successful in reversing the patient's symptoms, but it is tempting to consider whether simple proximal clipping of the ophthalmic artery origin might not have been less invasive and equally effective, particularly in light of the
Redistribution of this article permitted only in accordance with the publisher’s copyright provisions.
REFERENCES: (1-13)
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perception. Thus, although the intraorbital ophthalmic aneurysm rarely ruptures, once it does, the recovery of visual acuity is hopeless. Therefore, surgery should be performed not only to remove the mass effect caused by the aneurysm but also to prevent rupture. To achieve this goal, such operative procedures as resection or clipping of the aneurysm are the only radical measures possible. In the past, in fact, an attempt to ligate the carotid artery failed to achieve total thrombosis of the aneurysm (10). However, neck clipping of similar aneurysms, documented in the literature as well as in our own case, is difficult (7,9,10). Consequently, it may be necessary to resect the aneurysm. Therefore, at the time of surgery, particular attention should be paid to preserving visual acuity. The ophthalmic artery is divided into the intracranial course, the intracanalicular course that lies in the optic canal, and the intraorbital course. Furthermore, the intraorbital course can be divided into three parts. The first part extends from the point at which the artery enters the orbit to the point where it bends to become the second part. In the second part, the artery crosses over or under the optic nerve from the inferolateral to the superomedial direction. The third part extends from the point at which the second part bends to its termination (4,5). The ocular circulation is supplied by the central retinal and the ciliary arteries that usually arise at the junction of the first and second parts of the ophthalmic artery (1,5). Intraorbital ophthalmic artery aneurysms appear to occur in the first part (7,10,12,13) or the second part (9,11) of the ophthalmic artery. When the aneurysm is located in the second part of the ophthalmic artery distal to the origin of the central retinal and the ciliary arteries, resection of the aneurysm does not lead to ocular ischemia. In the presented case, in which the aneurysm was located in the first part of the ophthalmic artery, although the ophthalmic artery was trapped to remove the aneurysm, preoperative visual acuity was preserved. As evidenced by a postoperative carotid angiogram, collateral circulation from the external carotid artery successfully prevented the induction of ocular ischemia (Fig. 3). It is known that the anastomosis between the branches arising from the third part of the ophthalmic artery and the branches of the external carotid arteries is one of the major collateral channels between the internal and external carotid arteries (3,6). This collateral blood supply is adequate to prevent permanent blindness after occlusion of the internal carotid or ophthalmic artery in 90% of patients (8). Therefore, there is some possibility that ocular ischemia can be avoided by collateral circulation after the resection of the aneurysm, as described here. In conclusion, at the time of surgery, it is necessary to trap the ophthalmic artery without sacrifice of the central retinal and ciliary arteries. Even if an aneurysm is located in the first part of the ophthalmic artery, resection of an aneurysm, which should be the treatment of choice, seems possible because of collateral circulation from the external carotid artery.
aneurysm's dissection characteristics.
Arthur L. Day Gainesville, Florida
Redistribution of this article permitted only in accordance with the publisher’s copyright provisions.
Visual loss from ophthalmic artery aneurysms is a common phenomenon, occurring in approximately one-third of cases. Visual loss is usually caused by giant aneurysms (greater than 2.5 cm) that elevate the optic nerve against the falciform fold, just before the nerve's entry into the optic canal. Less commonly, visual loss can be produced by the aneurysms arising below the dural ring, compressing the optic nerve within the optic canal. This type of aneurysm usually arises from the clinoidal segment of the carotid artery and may not be visible to the surgeon when dissection is limited to the subarachnoid space. Often, this lesion is found in association with an ophthalmic artery origin within the clinoidal segment, a variation that occurs in 8 to 10% of individuals. The case described herein identifies a third (and very rare) site of aneurysm origin affiliated with the ophthalmic artery: the orbit. The intraorbital ophthalmic artery has become an extracerebral vessel in this position, and thus would be expected to have a thicker adventitial and muscular layer, thereby influencing the pathological cause, shape, and risks of hemorrhage from rupture. The authors have exhibited excellent judgment and anatomic knowledge in the management of this case.
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H. Hunt Batjer Dallas, Texas
Figure 3. A schematic diagram illustrates the location of the aneurysm on the ophthalmic artery with reference to the collateral circulation between the ophthalmic and the external carotid arteries. Arrowheads indicate the sites of the resection of the aneurysm.
Redistribution of this article permitted only in accordance with the publisher’s copyright provisions.
Figure 2. A preoperative right carotid angiogram shows an aneurysm located in the intraorbital segment of the ophthalmic artery. A, anteroposterior projection, B, lateral projection.
Downloaded from https://academic.oup.com/neurosurgery/article-abstract/31/6/1102/2752138 by University of Texas Southwestern Medical Center at Dallas user on 10 October 2018
Figure 1. A preoperative magnetic resonance imaging scan (T2-weighted image) shows an ophthalmic aneurysm as a dilated flow void area (arrowheads) at the right orbital apex.
Surgery Right orbital unroofing by a subfrontal-pterional approach disclosed a saccular aneurysm, 5 mm in diameter, near the orbital apex and on the ophthalmic artery running along the inferolateral aspect of the optic nerve (the first part of the ophthalmic artery, as defined by Hayreh and Dass [4]). The ophthalmic artery crossed over the optic nerve (the second part), and the lacrimal artery stemmed from the angular portion of the ophthalmic artery, which was distal to the aneurysm. The thick wall of the aneurysm was tightly attached to the inferolateral aspect of the optic nerve and compressed it. Because the aneurysm was contiguous with the ophthalmic artery, neck clipping of the aneurysm was impossible. Therefore, it was trapped and resected. Histological findings Histological findings showed the pseudolumen of the resected aneurysm to have formed in the arterial wall. The thickened wall was mostly composed of collagenous tissue, and both lamina elastica and muscularis were absent. Neither atherosclerotic changes nor any inflammatory process was observed. Postoperative course One month postoperatively, the patient had only light perception in the temporal field of the right eye. She showed 3rd nerve palsy with ptosis. With the exception of optic atrophy, in spite of the sacrifice of the ophthalmic artery, her fundi were not remarkable. Four months after the operation, eye movement had fully recovered, visual acuity had improved, and she could again count fingers. A postoperative carotid angiogram demonstrated a filling defect of the ophthalmic artery and disappearance of the ophthalmic aneurysm. The choroidal crescent was fed by a branch of the right external carotid artery. DISCUSSION Because there have been few detailed reports on intraorbital ophthalmic aneurysms, their clinical features are not fully understood. Common neurological symptoms and signs include progressive reduction of visual acuity, visual field defect, and exophthalmos, all of which derive from the mass effect of the aneurysm (7,10-13). Rupture of the intraorbital ophthalmic aneurysm is an extremely rare occurrence. Meyerson and Lazar (9), however, reported a case that showed a sudden onset of blindness and total ophthalmoplegia with massive subconjunctival and periorbital hemorrhage. Despite the removal of the intraorbital aneurysm and hematoma, visual acuity remained no light
Redistribution of this article permitted only in accordance with the publisher’s copyright provisions.
AUTHOR(S): Ogawa, Akira, M.D.; Tominaga, Teiji, M.D.; Yoshimoto, Takashi, M.D.; Kiyosawa, Motohiro, M.D.
slight exophthalmos in the right eye (Fig. 1). A carotid angiogram revealed an aneurysm arising from the intraorbital segment of the ophthalmic artery (Fig. 2). Because the aneurysm was located on the inferolateral side of the optic nerve near the orbital apex, compression of the optic nerve and 3rd and 6th nerves by this aneurysm was considered to be responsible for both the reduction of visual acuity and the disturbance of eye movement.
Downloaded from https://academic.oup.com/neurosurgery/article-abstract/31/6/1102/2752138 by University of Texas Southwestern Medical Center at Dallas user on 10 October 2018
Neurosurgery 1992-98 December 1992, Volume 31, Number 6 1102 Intraorbital Ophthalmic Artery Aneurysm: Case Report Case Report
Received, November 19, 1991. Accepted, July 7, 1992. Reprint requests: Akira Ogawa, M.D., Department of Neurosurgery, Iwate Medical University, 19-1, Uchimaru, Morioka, Japan 020.
1.
2. 3. 4. 5. 6. 7. 8.
9. 10. 11. 12.
13.
Brown GC, Magargal LE, Sergott R: Acute obstruction of the retinal and choroidal circulations. Ophthalmology 93:1373-1382, 1986. Day AL: Aneurysms of the ophthalmic segment: A clinical and anatomical analysis. J Neurosurg 72:677-691, 1990. Gillilan LA: The collateral circulation of the human orbit. Arch Ophthalmol 65:684-694, 1961. Hayreh SS, Das R: The ophthalmic artery II. Intraorbital course. Br J Ophthalmol 46:165185, 1962. Hayreh SS: The ophthalmic artery III. Branches. Br J Ophthalmol 46:212-247, 1962. Hayreh SS: Arteries of the orbit in the human being. Br J Surg 50:938-953, 1963. Jain KK: Saccular aneurysm of the ophthalmic artery. Am J Ophthalmol 69:997-998, 1970. Johnson HC, Walker AE: Angiographic diagnosis of spontaneous thrombosis of the internal and common carotid arteries. J Neurosurg 8:631- 659, 1951. Meyerson L, Lazar SJ: Intraorbital aneurysm of the ophthalmic artery. Br J Ophthalmol 55:199- 204, 1971. Raitta C: Ophthalmic artery aneurysm causing optic atrophy and enlargement of the optic foramen. Br J Ophthalmol 52:707-709, 1968. Rengachary SS, Kishore PRS: Intraorbital ophthalmic aneurysms and arteriovenous fistulae. Surg Neurol 9:35-41, 1978. Rubinstein MK, Wilson G, Levin DC: Intraorbital aneurysms of the ophthalmic artery: Report of a unique case and review of the literature. Arch Ophthalmol 80:42-44, 1968. Wheeler EC, Baker HL: The ophthalmic arterial complex in angiographic diagnosis. Radiology 83:26-35, 1964.
COMMENTS The authors have reported an extremely rare form of cerebrovascular disease: an intraorbital ophthalmic artery aneurysm producing visual loss and ophthalmoparesis. Their case is even more unique because their histological description suggests a dissecting aneurysm. Their management by excision was apparently very successful in reversing the patient's symptoms, but it is tempting to consider whether simple proximal clipping of the ophthalmic artery origin might not have been less invasive and equally effective, particularly in light of the
Redistribution of this article permitted only in accordance with the publisher’s copyright provisions.
REFERENCES: (1-13)
Downloaded from https://academic.oup.com/neurosurgery/article-abstract/31/6/1102/2752138 by University of Texas Southwestern Medical Center at Dallas user on 10 October 2018
perception. Thus, although the intraorbital ophthalmic aneurysm rarely ruptures, once it does, the recovery of visual acuity is hopeless. Therefore, surgery should be performed not only to remove the mass effect caused by the aneurysm but also to prevent rupture. To achieve this goal, such operative procedures as resection or clipping of the aneurysm are the only radical measures possible. In the past, in fact, an attempt to ligate the carotid artery failed to achieve total thrombosis of the aneurysm (10). However, neck clipping of similar aneurysms, documented in the literature as well as in our own case, is difficult (7,9,10). Consequently, it may be necessary to resect the aneurysm. Therefore, at the time of surgery, particular attention should be paid to preserving visual acuity. The ophthalmic artery is divided into the intracranial course, the intracanalicular course that lies in the optic canal, and the intraorbital course. Furthermore, the intraorbital course can be divided into three parts. The first part extends from the point at which the artery enters the orbit to the point where it bends to become the second part. In the second part, the artery crosses over or under the optic nerve from the inferolateral to the superomedial direction. The third part extends from the point at which the second part bends to its termination (4,5). The ocular circulation is supplied by the central retinal and the ciliary arteries that usually arise at the junction of the first and second parts of the ophthalmic artery (1,5). Intraorbital ophthalmic artery aneurysms appear to occur in the first part (7,10,12,13) or the second part (9,11) of the ophthalmic artery. When the aneurysm is located in the second part of the ophthalmic artery distal to the origin of the central retinal and the ciliary arteries, resection of the aneurysm does not lead to ocular ischemia. In the presented case, in which the aneurysm was located in the first part of the ophthalmic artery, although the ophthalmic artery was trapped to remove the aneurysm, preoperative visual acuity was preserved. As evidenced by a postoperative carotid angiogram, collateral circulation from the external carotid artery successfully prevented the induction of ocular ischemia (Fig. 3). It is known that the anastomosis between the branches arising from the third part of the ophthalmic artery and the branches of the external carotid arteries is one of the major collateral channels between the internal and external carotid arteries (3,6). This collateral blood supply is adequate to prevent permanent blindness after occlusion of the internal carotid or ophthalmic artery in 90% of patients (8). Therefore, there is some possibility that ocular ischemia can be avoided by collateral circulation after the resection of the aneurysm, as described here. In conclusion, at the time of surgery, it is necessary to trap the ophthalmic artery without sacrifice of the central retinal and ciliary arteries. Even if an aneurysm is located in the first part of the ophthalmic artery, resection of an aneurysm, which should be the treatment of choice, seems possible because of collateral circulation from the external carotid artery.
aneurysm's dissection characteristics.
Arthur L. Day Gainesville, Florida
Redistribution of this article permitted only in accordance with the publisher’s copyright provisions.
Visual loss from ophthalmic artery aneurysms is a common phenomenon, occurring in approximately one-third of cases. Visual loss is usually caused by giant aneurysms (greater than 2.5 cm) that elevate the optic nerve against the falciform fold, just before the nerve's entry into the optic canal. Less commonly, visual loss can be produced by the aneurysms arising below the dural ring, compressing the optic nerve within the optic canal. This type of aneurysm usually arises from the clinoidal segment of the carotid artery and may not be visible to the surgeon when dissection is limited to the subarachnoid space. Often, this lesion is found in association with an ophthalmic artery origin within the clinoidal segment, a variation that occurs in 8 to 10% of individuals. The case described herein identifies a third (and very rare) site of aneurysm origin affiliated with the ophthalmic artery: the orbit. The intraorbital ophthalmic artery has become an extracerebral vessel in this position, and thus would be expected to have a thicker adventitial and muscular layer, thereby influencing the pathological cause, shape, and risks of hemorrhage from rupture. The authors have exhibited excellent judgment and anatomic knowledge in the management of this case.
Downloaded from https://academic.oup.com/neurosurgery/article-abstract/31/6/1102/2752138 by University of Texas Southwestern Medical Center at Dallas user on 10 October 2018
H. Hunt Batjer Dallas, Texas
Figure 3. A schematic diagram illustrates the location of the aneurysm on the ophthalmic artery with reference to the collateral circulation between the ophthalmic and the external carotid arteries. Arrowheads indicate the sites of the resection of the aneurysm.
Redistribution of this article permitted only in accordance with the publisher’s copyright provisions.
Figure 2. A preoperative right carotid angiogram shows an aneurysm located in the intraorbital segment of the ophthalmic artery. A, anteroposterior projection, B, lateral projection.
Downloaded from https://academic.oup.com/neurosurgery/article-abstract/31/6/1102/2752138 by University of Texas Southwestern Medical Center at Dallas user on 10 October 2018
Figure 1. A preoperative magnetic resonance imaging scan (T2-weighted image) shows an ophthalmic aneurysm as a dilated flow void area (arrowheads) at the right orbital apex.
