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A case of ruptured aneurysm associated with spinal arteriovenous malformation presenting with hematomyelia: case report
A Case of Ruptured Aneurysm Associated with Spinal Arteriovenous Malformation Presenting with Hematomyelia: Case Report Makoto Sakamoto, M.D.,* Takashi Watanabe, M.D., Ph.D.,* and Hisayo Okamoto, M.D., Ph.D.† *Department of Neurosurgery, Institute of Neurological Sciences, Faculty of Medicine, Tottori University and †Department of Neurosurgery, Takashima Hospital, Yonago, Japan
Sakamoto M, Watanabe T, Okamoto H. A case of ruptured aneurysm associated with spinal arteriovenous malformation presenting with hematomyelia: case report. Surg Neurol 2002;57:438 – 42.
KEY WORDS
BACKGROUND
pinal cord arteriovenous malformation (SCAVM) associated with spinal aneurysm is not especially rare, but cases presenting with hematomyelia are relatively rare compared to those with SAH. Patients presenting with hematomyelia frequently have severe neurological deficits, and their outcomes are poor. Although there have been many reports on SCAVM [1– 4,6,8 –21], many problems still remain unsolved in the management of such patients, especially regarding the choice of treatment—surgery [2,20] or endovascular obliteration [4,10,11,14,19]. Several authors have reported that the prognoses of untreated patients who suffered from SCAVM were poor [11,16,18], especially in cases accompanied by aneurysms because of a high incidence of rupture of the aneurysm [3,11,14]. Endovascular treatment for SCAVM has recently been developed, but recanalization of the lesion after embolization has occurred occasionally [4,8, 10,14,19]. We report a case of SCAVM associated with ruptured spinal aneurysms presenting with hematomyelia, which was treated successfully by direct surgical obliteration of the feeding arteries and nidus.
Spinal cord arteriovenous malformation (AVM) associated with spinal aneurysm is not particularly rare, but cases presenting with hematomyelia are relatively rare compared to those with subarachnoid hemorrhage (SAH). We report a rare case of successfully treated spinal AVM associated with ruptured aneurysm presenting with hematomyelia. CASE DESCRIPTION
A 52-year-old male was admitted to our hospital with sudden onset of tetraplegia, respiratory disturbance, and superficial sensory disturbance. Computed tomography revealed hematomyelia at the level of C3– 4. Gadoliniumenhanced magnetic resonance imaging showed small, enhanced lesions. Angiography revealed an intradural perimedullary arteriovenous malformation associated with two aneurysms on the feeding arteries. Administration of high-dose methylprednisolone gradually ameliorated his symptoms. Direct surgical obliteration was performed on the 30th day after the onset. The bilateral C3 cervical radicular arteries and the nidus were coagulated. Angiography performed after surgery showed neither the aneurysms nor the nidus. He was discharged with only mild weakness in the left upper extremity and mild left hypesthesia 3 months after surgery, and was fully independent. CONCLUSION
We report a case of hematomyelia caused by ruptured aneurysm associated with spinal arteriovenous malformation that was successfully treated with surgical obliteration. © 2002 by Elsevier Science Inc.
Spinal AVM, spinal aneurysm, hematomyelia.
S
Case Report Address reprint requests to: Dr Makoto Sakamoto, Department of Neurosurgery, Institute of Neurological Sciences, Faculty of Medicine, Tottori University, 36-1 Nishi-cho, Yonago, Tottori Pref. 683-8504, Japan. Received August 30, 2001; accepted January 3, 2002. 0090-3019/02/$–see front matter PII S0090-3019(02)00728-0
A 52-year-old male presented with sudden weakness of the extremities and was transferred to Takashima Hospital on March 23, 1999. On arrival, © 2002 by Elsevier Science Inc. 655 Avenue of the Americas, New York, NY 10010
Spinal Aneurysm Presenting Hematomyelia
1
CT scan of the cervical spine at the level of C2 on admission revealed hematomyelia.
his consciousness level was almost alert, but he also exhibited tetraplegia, labored respiration, and bilateral superficial sensory disturbance below the level of C4. Deep sensory and sphincter functions were preserved. Emergency computed tomography (CT) scan revealed an intramedullary high-density area at the level of C2– 4, suggesting hematomyelia (Figure 1). He underwent nasotracheal intubation for artificial respiration and was administered 6 g/day methylprednisolone for 48 h to reduce spinal cord edema. Magnetic resonance (MR) imaging revealed a spinal cord swelling at the level of C2– 4 on T2-weighted images and gadolinium-enhanced MR imaging showed a dumbbell-shaped enhancement; the medial lesion was intramedullary, and the other lesion was perimedullary (Figure 2). His tetraplegia, sensory disturbance and left phrenic nerve palsy were gradually ameliorated, then he underwent selective angiography on March 29. The serial left
Axial gadolinium-enhanced MR image of the cervical spine at the level of C2–3 on admission revealed a dumbbell shaped lesion. Its medial portion seemed to be intramedullary, and the other portion perimedullary.
2
Surg Neurol 439 2002;57:438 –42
vertebral angiograms first revealed an aneurysm originating from the left C3 radicular artery. After 0.3 s, another aneurysm and small conglomerated vessels were visualized, and then dilated draining veins were observed (Figure 3). The right vertebral angiogram showed the aneurysms via the left C3 radicular artery but not the nidus and/or draining vein (Figure 4). Together with the findings on CT and MRI, the lesions were assumed to be an intradural and perimedullary spinal AVM originating from both C3 radicular arteries with perimedullary aneurysms on the feeding arteries. The cause of hematomyelia was presumed to be rupture of the aneurysm buried in the spinal cord. Just after the vertebral angiogram, his consciousness level and left hemiparesis transiently deteriorated, perhaps because of a thromboembolic event. We had to stop angiography at that time. His neurological deficits almost disappeared within a week. He underwent angiography again on April 12, but the appearances of the lesions were unchanged. On April 23, 30 days after the onset, he underwent surgical obliteration of both feeding arteries and the nidus of the AVM via a C2– 4 posterior laminotomy. The nidus of the AVM was identified caudal to the left C3 nerve root. Beneath the left C3 nerve root, the left C3 radicular artery was identified and there was an aneurysm on its cranial side. The right C3 radicular artery was identified beside the right C3 nerve root. The bilateral C3 radicular arteries feeding the nidus of the AVM were obliterated in their distal portion with bipolar coagulation, and then we coagulated the nidus of the AVM. The color of the nidus, its draining vein, and the extramedullary aneurysm were darkened and the tension of the spinal aneurysm was consequently decreased. We could not observe the ruptured aneurysm or hematomyelia during the operation. There was no evidence of subarachnoid hemorrhage; the cerebrospinal fluid was clear and no hemosiderin deposition was observed around the operative field. Immediately after the operation, the patient’s neurological condition did not worsen compared to the preoperative state. Angiography performed on the day after surgery showed neither the aneurysms nor the AVM, and the posterior spinal artery was preserved. Complete obliteration of the aneurysms and the SCAVM was revealed by follow-up angiography performed 2 months later. Postoperatively, the patient experienced continuous improvement of tetraplegia, and 3 months after the surgery he was discharged with only mild weakness in the left upper extremity and left mild hypesthesia, and returned to his former activities.
440 Surg Neurol 2002;57:438 –42
Serial anteroposterior left vertebral angiograms first revealed an aneurysm originating from the left C3 radicular artery (A). After 0.3 s, another aneurysm and small conglomerated vessels were visualized (B). The dilated draining vein was then demonstrated (C).
3
Sakamoto et al
Spinal Aneurysm Presenting Hematomyelia
Anteroposterior right vertebral angiogram showed the aneurysms via the right C3 radicular artery but neither the nidus nor the draining vein.
4
Discussion The management of SCAVM associated with a spinal aneurysm has remained controversial. Several authors have reported that SCAVM associated with a spinal aneurysm was present in 2.2–7.7% of all types of SCAVM [3,14]. The natural history of patients with untreated SCAVM is poor [11,16,18], with high mortality and morbidity, and in cases with spinal aneurysms is poorer than in those with SCAVM without aneurysm because of the high incidence of hemorrhage from the aneurysms [3,11,14]. Treatments for these lesions include two options; direct microsurgical obliteration [2, 20] and an endovascular approach [4,10,11,14,19]. For both treatments, it is important to detect the accurate location of the lesion in relation to the anterior spinal artery, feeding artery and draining vein. When the anterior spinal artery is involved in the SCAVM, the risks associated with treating the lesion may be high [11]. Recently, endovascular embolization has been widely undertaken as a treatment for SCAVM,
Surg Neurol 441 2002;57:438 –42
but recanalization of SCAVM after endovascular embolization has been reported by several authors [4,8,12,14,19]. Embolization alone sometimes cannot achieve an angiographically complete cure. As direct surgical obliteration and/or excision of SCAVM provides a therapeutic means to permanently eliminate the flow through the SCAVM in most patients, it should be the treatment of choice when feasible. Because the nidus of the SCAVM in our case was extramedullary, we assumed it would be possible to approach the feeding vessels and the nidus, and chose to perform direct surgical obliteration of the SCAVM. Most spinal aneurysms are associated with SCAVM, although there have been a few reports of isolated spinal aneurysms [3]. It is said that development of the aneurysm is attributed to increased blood flow and hemodynamic stress caused by SCAVM [3,14]. In our case, the aneurysms showed spontaneous regression immediately after surgical obliteration of the feeding arteries and the nidus, and finally disappeared. We coagulated not only the feeding arteries of the AVM, but also the nidus, so that complete obliteration of the AVM and aneurysms was accomplished. If we had not coagulated the nidus of the AVM, recanalization might have occurred. The classification of SCAVMs is based on their location and angiographic features. These lesions are classified as dural arteriovenous fistula (AVF) or intradural SCAVM [2,15,18]. The intradural AVMs were further classified as intramedullary AVMs (juvenile and glomus type) and perimedullary AVFs. Intradural perimedullary AVF presents with either hemorrhage or, more commonly, myelopathy because of venous hypertension [15,16]. Patients with SCAVM associated with aneurysm often present with subarachnoid hemorrhage. Our case presented with hematomyelia, which is uncommon for this type of SCAVM. It seemed that recent hemorrhage had occurred from the perimedullary aneurysm on the left radicular artery, and extended into the medulla. Glucocorticosteroids have been extensively used in the clinical treatment of traumatic spinal cord injuries [5,7]. The rationale for their use chiefly consists of an expectation of reduction of posttraumatic spinal cord edema and inhibition of lipid peroxidation. There have been few reports on the efficacy of administration of high-dose glucocorticosteroids for patients with non-traumatic spinal cord injuries, such as hemorrhagic episodes of SCAVM. We administered high-dose methylprednisolone to this patient with spinal cord injury caused by hematomyelia, and observed dramatic
442 Surg Neurol 2002;57:438 –42
amelioration of his respiratory disturbance, tetraplegia, and sensory disturbance. It seems that administration of high dose methylpredonisolone is efficacious, not only for traumatic spinal injury but also for nontraumatic lesions. In conclusion, we report a case of hematomyelia caused by ruptured aneurysm associated with spinal arteriovenous malformation that was treated with surgical obliteration.
Sakamoto et al
14.
15. 16.
REFERENCES 1. Avman N, Ozkal E, Gokben B. Aneurysm and arteriovenous malformation of the spinal cord. Surg Neurol 1979;11(1):5– 6. 2. Barrow DL, Colohan AR, Dawson R. Intradural perimedullary arteriovenous fistulas (type IV spinal cord arteriovenous malformations). J Neurosurg 1994; 81(2):221–9. 3. Biondi A, Merland JJ, Hodes JE, Pruvo JP, Reizine D. Aneurysms of spinal arteries associated with intramedullary arteriovenous malformations. I. Angiographic and clinical aspects. AJNR Am J Neuroradiol 1992;13(3):913–22. 4. Biondi A, Merland JJ, Hodes JE, Aymard A, Reizine D. Aneurysms of spinal arteries associated with intramedullary arteriovenous malformations. II. Results of AVM endovascular treatment and hemodynamic considerations. AJNR Am J Neuroradiol 1992;13(3): 923–31. 5. Bracken MB, Shepard MJ, Holford TR, et al. Administration of methylprednisolone for 24 or 48 h or tirilazad mesylate for 48 h in the treatment of acute spinal cord injury. Results of the Third National Acute Spinal Cord Injury Randomized Controlled Trial. National Acute Spinal Cord Injury Study. JAMA 1997;277(20): 1597– 604. 6. Di Chiro G, Wener L. Angiography of the spinal cord. A review of contemporary techniques and applications. J Neurosurg 1973;39(1):1–29. 7. Hall ED. The neuroprotective pharmacology of methylprednisolone. J Neurosurg 1992;76(1):13–22. 8. Hall WA, Oldfield EH, Doppman JL. Recanalization of spinal arteriovenous malformations following embolization. J Neurosurg 1989;70(5):714 –720. 9. Hopkins CA, Wilkie FL, Voris DC. Extramedullary aneurysm of the spinal cord. Case report. J Neurosurg 1966;24(6):1021–3. 10. Kahara VJ, Seppanen SK, Kuurne T, Laasonen EM. Diagnosis and embolizing of spinal arteriovenous malformations. Ann Med 1997;29(5):377– 82. 11. Konan AV, Raymond J, Roy D. Transarterial embolization of aneurysms associated with spinal cord arteriovenous malformations. Report of four cases. J Neurosurg 1999;90(1 Suppl):148 –54. 12. Kunc Z, Bret J. Diagnosis and treatment of vascular malformations of the spinal cord. J Neurosurg 1969; 30(4):436 – 45. 13. Malek AM, Halbach VV, Phatouros CC, et al. Spinal dural arteriovenous fistula with an associated feeding
17. 18.
19.
20. 21.
artery aneurysm: case report. Neurosurgery 1999; 44(4):877– 80. Miyamoto S, Kikuchi H, Karasawa J, Ikota T, Nagata I. Spinal cord arteriovenous malformations associated with spinal aneurysms. Neurosurgery 1983;13(5):577– 80. Morgan MK. Outcome from treatment for spinal arteriovenous malformation. Neurosurg Clin N Am 1999; 10(1):113–119. Mourier KL, Gobin YP, George B, Lot G, Merland JJ. Intradural perimedullary arteriovenous fistulae: results of surgical and endovascular treatment in a series of 35 cases. Neurosurgery 1993;32(6):885–91; discussion 891. Oldfield EH, Doppman JL. Spinal arteriovenous malformations. Clin Neurosurg 1988;34:161– 83. Rosenblum B, Oldfield EH, Doppman JL, Di Chiro G. Spinal arteriovenous malformations: a comparison of dural arteriovenous fistulas and intradural AVM’s in 81 patients. J Neurosurg 1987;67(6):795– 802. Touho H, Karasawa J, Ohnishi H, Yamada K, Shibamoto K. Superselective embolization of spinal arteriovenous malformations using the Tracker catheter. Surg Neurol 1992;38(2):85–94. Watson JC, Oldfield EH. The surgical management of spinal dural vascular malformations. Neurosurg Clin N Am 1999;10(1):73– 87. Yasargil MG, DeLong WB, Guarnaschelli JJ. Complete microsurgical excision of cervical extramedullary and intramedullary vascular malformations. Surg Neurol 1975;4(2):211–24.
COMMENTARY
While most neurosurgeons will never see the combination of intradural spinal arteriovenous malformation presenting with sudden tetraplegia because of hematomyelia as well as aneurysms of the two feeding arteries, I found reading this case report fascinating. Resolving the tetralegia with 6 g per day of methyprednisolone was a breakthrough. The surgical procedure, coagulation of the nidus and the distal feeding arteries, was well planned. Leaving the proximal arteries and the aneurysms alone both spared the spinal circulation and allowed the aneurysms to close themselves. This again demonstrated that aneurysms associated with the feeding vessels of an arteriovenous malformation are dependent upon the flow to the AVM. This cannot be emphasized too often. It is gratifying that all the wise decisions culminated in this patient’s excellent recovery. Leonard I. Malis, M.D. Neurosurgeon New York, New York
Sakamoto M, Watanabe T, Okamoto H. A case of ruptured aneurysm associated with spinal arteriovenous malformation presenting with hematomyelia: case report. Surg Neurol 2002;57:438 – 42.
KEY WORDS
BACKGROUND
pinal cord arteriovenous malformation (SCAVM) associated with spinal aneurysm is not especially rare, but cases presenting with hematomyelia are relatively rare compared to those with SAH. Patients presenting with hematomyelia frequently have severe neurological deficits, and their outcomes are poor. Although there have been many reports on SCAVM [1– 4,6,8 –21], many problems still remain unsolved in the management of such patients, especially regarding the choice of treatment—surgery [2,20] or endovascular obliteration [4,10,11,14,19]. Several authors have reported that the prognoses of untreated patients who suffered from SCAVM were poor [11,16,18], especially in cases accompanied by aneurysms because of a high incidence of rupture of the aneurysm [3,11,14]. Endovascular treatment for SCAVM has recently been developed, but recanalization of the lesion after embolization has occurred occasionally [4,8, 10,14,19]. We report a case of SCAVM associated with ruptured spinal aneurysms presenting with hematomyelia, which was treated successfully by direct surgical obliteration of the feeding arteries and nidus.
Spinal cord arteriovenous malformation (AVM) associated with spinal aneurysm is not particularly rare, but cases presenting with hematomyelia are relatively rare compared to those with subarachnoid hemorrhage (SAH). We report a rare case of successfully treated spinal AVM associated with ruptured aneurysm presenting with hematomyelia. CASE DESCRIPTION
A 52-year-old male was admitted to our hospital with sudden onset of tetraplegia, respiratory disturbance, and superficial sensory disturbance. Computed tomography revealed hematomyelia at the level of C3– 4. Gadoliniumenhanced magnetic resonance imaging showed small, enhanced lesions. Angiography revealed an intradural perimedullary arteriovenous malformation associated with two aneurysms on the feeding arteries. Administration of high-dose methylprednisolone gradually ameliorated his symptoms. Direct surgical obliteration was performed on the 30th day after the onset. The bilateral C3 cervical radicular arteries and the nidus were coagulated. Angiography performed after surgery showed neither the aneurysms nor the nidus. He was discharged with only mild weakness in the left upper extremity and mild left hypesthesia 3 months after surgery, and was fully independent. CONCLUSION
We report a case of hematomyelia caused by ruptured aneurysm associated with spinal arteriovenous malformation that was successfully treated with surgical obliteration. © 2002 by Elsevier Science Inc.
Spinal AVM, spinal aneurysm, hematomyelia.
S
Case Report Address reprint requests to: Dr Makoto Sakamoto, Department of Neurosurgery, Institute of Neurological Sciences, Faculty of Medicine, Tottori University, 36-1 Nishi-cho, Yonago, Tottori Pref. 683-8504, Japan. Received August 30, 2001; accepted January 3, 2002. 0090-3019/02/$–see front matter PII S0090-3019(02)00728-0
A 52-year-old male presented with sudden weakness of the extremities and was transferred to Takashima Hospital on March 23, 1999. On arrival, © 2002 by Elsevier Science Inc. 655 Avenue of the Americas, New York, NY 10010
Spinal Aneurysm Presenting Hematomyelia
1
CT scan of the cervical spine at the level of C2 on admission revealed hematomyelia.
his consciousness level was almost alert, but he also exhibited tetraplegia, labored respiration, and bilateral superficial sensory disturbance below the level of C4. Deep sensory and sphincter functions were preserved. Emergency computed tomography (CT) scan revealed an intramedullary high-density area at the level of C2– 4, suggesting hematomyelia (Figure 1). He underwent nasotracheal intubation for artificial respiration and was administered 6 g/day methylprednisolone for 48 h to reduce spinal cord edema. Magnetic resonance (MR) imaging revealed a spinal cord swelling at the level of C2– 4 on T2-weighted images and gadolinium-enhanced MR imaging showed a dumbbell-shaped enhancement; the medial lesion was intramedullary, and the other lesion was perimedullary (Figure 2). His tetraplegia, sensory disturbance and left phrenic nerve palsy were gradually ameliorated, then he underwent selective angiography on March 29. The serial left
Axial gadolinium-enhanced MR image of the cervical spine at the level of C2–3 on admission revealed a dumbbell shaped lesion. Its medial portion seemed to be intramedullary, and the other portion perimedullary.
2
Surg Neurol 439 2002;57:438 –42
vertebral angiograms first revealed an aneurysm originating from the left C3 radicular artery. After 0.3 s, another aneurysm and small conglomerated vessels were visualized, and then dilated draining veins were observed (Figure 3). The right vertebral angiogram showed the aneurysms via the left C3 radicular artery but not the nidus and/or draining vein (Figure 4). Together with the findings on CT and MRI, the lesions were assumed to be an intradural and perimedullary spinal AVM originating from both C3 radicular arteries with perimedullary aneurysms on the feeding arteries. The cause of hematomyelia was presumed to be rupture of the aneurysm buried in the spinal cord. Just after the vertebral angiogram, his consciousness level and left hemiparesis transiently deteriorated, perhaps because of a thromboembolic event. We had to stop angiography at that time. His neurological deficits almost disappeared within a week. He underwent angiography again on April 12, but the appearances of the lesions were unchanged. On April 23, 30 days after the onset, he underwent surgical obliteration of both feeding arteries and the nidus of the AVM via a C2– 4 posterior laminotomy. The nidus of the AVM was identified caudal to the left C3 nerve root. Beneath the left C3 nerve root, the left C3 radicular artery was identified and there was an aneurysm on its cranial side. The right C3 radicular artery was identified beside the right C3 nerve root. The bilateral C3 radicular arteries feeding the nidus of the AVM were obliterated in their distal portion with bipolar coagulation, and then we coagulated the nidus of the AVM. The color of the nidus, its draining vein, and the extramedullary aneurysm were darkened and the tension of the spinal aneurysm was consequently decreased. We could not observe the ruptured aneurysm or hematomyelia during the operation. There was no evidence of subarachnoid hemorrhage; the cerebrospinal fluid was clear and no hemosiderin deposition was observed around the operative field. Immediately after the operation, the patient’s neurological condition did not worsen compared to the preoperative state. Angiography performed on the day after surgery showed neither the aneurysms nor the AVM, and the posterior spinal artery was preserved. Complete obliteration of the aneurysms and the SCAVM was revealed by follow-up angiography performed 2 months later. Postoperatively, the patient experienced continuous improvement of tetraplegia, and 3 months after the surgery he was discharged with only mild weakness in the left upper extremity and left mild hypesthesia, and returned to his former activities.
440 Surg Neurol 2002;57:438 –42
Serial anteroposterior left vertebral angiograms first revealed an aneurysm originating from the left C3 radicular artery (A). After 0.3 s, another aneurysm and small conglomerated vessels were visualized (B). The dilated draining vein was then demonstrated (C).
3
Sakamoto et al
Spinal Aneurysm Presenting Hematomyelia
Anteroposterior right vertebral angiogram showed the aneurysms via the right C3 radicular artery but neither the nidus nor the draining vein.
4
Discussion The management of SCAVM associated with a spinal aneurysm has remained controversial. Several authors have reported that SCAVM associated with a spinal aneurysm was present in 2.2–7.7% of all types of SCAVM [3,14]. The natural history of patients with untreated SCAVM is poor [11,16,18], with high mortality and morbidity, and in cases with spinal aneurysms is poorer than in those with SCAVM without aneurysm because of the high incidence of hemorrhage from the aneurysms [3,11,14]. Treatments for these lesions include two options; direct microsurgical obliteration [2, 20] and an endovascular approach [4,10,11,14,19]. For both treatments, it is important to detect the accurate location of the lesion in relation to the anterior spinal artery, feeding artery and draining vein. When the anterior spinal artery is involved in the SCAVM, the risks associated with treating the lesion may be high [11]. Recently, endovascular embolization has been widely undertaken as a treatment for SCAVM,
Surg Neurol 441 2002;57:438 –42
but recanalization of SCAVM after endovascular embolization has been reported by several authors [4,8,12,14,19]. Embolization alone sometimes cannot achieve an angiographically complete cure. As direct surgical obliteration and/or excision of SCAVM provides a therapeutic means to permanently eliminate the flow through the SCAVM in most patients, it should be the treatment of choice when feasible. Because the nidus of the SCAVM in our case was extramedullary, we assumed it would be possible to approach the feeding vessels and the nidus, and chose to perform direct surgical obliteration of the SCAVM. Most spinal aneurysms are associated with SCAVM, although there have been a few reports of isolated spinal aneurysms [3]. It is said that development of the aneurysm is attributed to increased blood flow and hemodynamic stress caused by SCAVM [3,14]. In our case, the aneurysms showed spontaneous regression immediately after surgical obliteration of the feeding arteries and the nidus, and finally disappeared. We coagulated not only the feeding arteries of the AVM, but also the nidus, so that complete obliteration of the AVM and aneurysms was accomplished. If we had not coagulated the nidus of the AVM, recanalization might have occurred. The classification of SCAVMs is based on their location and angiographic features. These lesions are classified as dural arteriovenous fistula (AVF) or intradural SCAVM [2,15,18]. The intradural AVMs were further classified as intramedullary AVMs (juvenile and glomus type) and perimedullary AVFs. Intradural perimedullary AVF presents with either hemorrhage or, more commonly, myelopathy because of venous hypertension [15,16]. Patients with SCAVM associated with aneurysm often present with subarachnoid hemorrhage. Our case presented with hematomyelia, which is uncommon for this type of SCAVM. It seemed that recent hemorrhage had occurred from the perimedullary aneurysm on the left radicular artery, and extended into the medulla. Glucocorticosteroids have been extensively used in the clinical treatment of traumatic spinal cord injuries [5,7]. The rationale for their use chiefly consists of an expectation of reduction of posttraumatic spinal cord edema and inhibition of lipid peroxidation. There have been few reports on the efficacy of administration of high-dose glucocorticosteroids for patients with non-traumatic spinal cord injuries, such as hemorrhagic episodes of SCAVM. We administered high-dose methylprednisolone to this patient with spinal cord injury caused by hematomyelia, and observed dramatic
442 Surg Neurol 2002;57:438 –42
amelioration of his respiratory disturbance, tetraplegia, and sensory disturbance. It seems that administration of high dose methylpredonisolone is efficacious, not only for traumatic spinal injury but also for nontraumatic lesions. In conclusion, we report a case of hematomyelia caused by ruptured aneurysm associated with spinal arteriovenous malformation that was treated with surgical obliteration.
Sakamoto et al
14.
15. 16.
REFERENCES 1. Avman N, Ozkal E, Gokben B. Aneurysm and arteriovenous malformation of the spinal cord. Surg Neurol 1979;11(1):5– 6. 2. Barrow DL, Colohan AR, Dawson R. Intradural perimedullary arteriovenous fistulas (type IV spinal cord arteriovenous malformations). J Neurosurg 1994; 81(2):221–9. 3. Biondi A, Merland JJ, Hodes JE, Pruvo JP, Reizine D. Aneurysms of spinal arteries associated with intramedullary arteriovenous malformations. I. Angiographic and clinical aspects. AJNR Am J Neuroradiol 1992;13(3):913–22. 4. Biondi A, Merland JJ, Hodes JE, Aymard A, Reizine D. Aneurysms of spinal arteries associated with intramedullary arteriovenous malformations. II. Results of AVM endovascular treatment and hemodynamic considerations. AJNR Am J Neuroradiol 1992;13(3): 923–31. 5. Bracken MB, Shepard MJ, Holford TR, et al. Administration of methylprednisolone for 24 or 48 h or tirilazad mesylate for 48 h in the treatment of acute spinal cord injury. Results of the Third National Acute Spinal Cord Injury Randomized Controlled Trial. National Acute Spinal Cord Injury Study. JAMA 1997;277(20): 1597– 604. 6. Di Chiro G, Wener L. Angiography of the spinal cord. A review of contemporary techniques and applications. J Neurosurg 1973;39(1):1–29. 7. Hall ED. The neuroprotective pharmacology of methylprednisolone. J Neurosurg 1992;76(1):13–22. 8. Hall WA, Oldfield EH, Doppman JL. Recanalization of spinal arteriovenous malformations following embolization. J Neurosurg 1989;70(5):714 –720. 9. Hopkins CA, Wilkie FL, Voris DC. Extramedullary aneurysm of the spinal cord. Case report. J Neurosurg 1966;24(6):1021–3. 10. Kahara VJ, Seppanen SK, Kuurne T, Laasonen EM. Diagnosis and embolizing of spinal arteriovenous malformations. Ann Med 1997;29(5):377– 82. 11. Konan AV, Raymond J, Roy D. Transarterial embolization of aneurysms associated with spinal cord arteriovenous malformations. Report of four cases. J Neurosurg 1999;90(1 Suppl):148 –54. 12. Kunc Z, Bret J. Diagnosis and treatment of vascular malformations of the spinal cord. J Neurosurg 1969; 30(4):436 – 45. 13. Malek AM, Halbach VV, Phatouros CC, et al. Spinal dural arteriovenous fistula with an associated feeding
17. 18.
19.
20. 21.
artery aneurysm: case report. Neurosurgery 1999; 44(4):877– 80. Miyamoto S, Kikuchi H, Karasawa J, Ikota T, Nagata I. Spinal cord arteriovenous malformations associated with spinal aneurysms. Neurosurgery 1983;13(5):577– 80. Morgan MK. Outcome from treatment for spinal arteriovenous malformation. Neurosurg Clin N Am 1999; 10(1):113–119. Mourier KL, Gobin YP, George B, Lot G, Merland JJ. Intradural perimedullary arteriovenous fistulae: results of surgical and endovascular treatment in a series of 35 cases. Neurosurgery 1993;32(6):885–91; discussion 891. Oldfield EH, Doppman JL. Spinal arteriovenous malformations. Clin Neurosurg 1988;34:161– 83. Rosenblum B, Oldfield EH, Doppman JL, Di Chiro G. Spinal arteriovenous malformations: a comparison of dural arteriovenous fistulas and intradural AVM’s in 81 patients. J Neurosurg 1987;67(6):795– 802. Touho H, Karasawa J, Ohnishi H, Yamada K, Shibamoto K. Superselective embolization of spinal arteriovenous malformations using the Tracker catheter. Surg Neurol 1992;38(2):85–94. Watson JC, Oldfield EH. The surgical management of spinal dural vascular malformations. Neurosurg Clin N Am 1999;10(1):73– 87. Yasargil MG, DeLong WB, Guarnaschelli JJ. Complete microsurgical excision of cervical extramedullary and intramedullary vascular malformations. Surg Neurol 1975;4(2):211–24.
COMMENTARY
While most neurosurgeons will never see the combination of intradural spinal arteriovenous malformation presenting with sudden tetraplegia because of hematomyelia as well as aneurysms of the two feeding arteries, I found reading this case report fascinating. Resolving the tetralegia with 6 g per day of methyprednisolone was a breakthrough. The surgical procedure, coagulation of the nidus and the distal feeding arteries, was well planned. Leaving the proximal arteries and the aneurysms alone both spared the spinal circulation and allowed the aneurysms to close themselves. This again demonstrated that aneurysms associated with the feeding vessels of an arteriovenous malformation are dependent upon the flow to the AVM. This cannot be emphasized too often. It is gratifying that all the wise decisions culminated in this patient’s excellent recovery. Leonard I. Malis, M.D. Neurosurgeon New York, New York