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Solitary spinal epidural cavernous haemangioma presenting as acute epidural haematoma
Solitary spinal epidural cavernous haemangioma presenting as acute epidural haematoma T a k a f u m i I n o u e MD Hisao K o g a MD M a s a m i t s u A b e MD K a z u o Tabuchi MD Department of Neurosurgery, Saga Medical School, Saga,Japan.
This report describes a case o f solitary spinal epidural cavernous h a e m a n g i o m a presenting as an acute epidural haematoma. The patient is a 50 year old diabetic female with right brachalgia and hemiparesis. Magnetic resonance imaging (MRI) demonstrated the presence o f a cervical spinal epidural haematoma. The patient underwent laminectomy and total removal o f the h a e m a t o m a and the accompanying vascular tissue. The diagnosis o f cavernous h a e m a n g i o m a was proven by histological examination. Because the source o f bleeding in m o s t cases o f spontaneous spinal epidural h a e m a t o m a is usually obscure, the authors emphasise the possibility that a small cavernous h a e m a n g i o m a can be one o f the bleeding sources in s o m e o f these cases. Journal of Clinical Neuroscience 1995, 2(3):265-268
© Pearson Professional 1995
Keywords: Spinal epidural haematoma, Cavernous haemangioma, Magnetic resonance imaging
Introduction Spinal epidural cavernous h a e m a n g i o m a s are c o m m o n l y seen as e x t e n s i o n s f r o m a v e r t e b r a l h a e m a n g i o m a . However, the report of a solitary spinal epidural cavernous h a e m a n g i o m a is r a t h e r rare a n d 34 cases have b e e n r e p o r t e d to date. ~-24 Vascular anomaly, such as AVM or h a e m a n g i o m a , is s o m e t i m e s described as the source o f spinal epidural haematomas, although in most patients with spontaneous spinal epidural h a e m a t o m a the precise source of bleeding is usually obscure. T h e r e have b e e n only three r e p o r t e d cases in which a r u p t u r e d spinal e p i d u r a l c a v e r n o u s h a e m a n g i o m a was recognised as the source of a spinal epidural h a e m a t o m a . 16, 2a, 24 We r e p o r t a r a r e case of solitary spinal e p i d u r a l cavernous h a e m a n g i o m a in a patient diagnosed as having an acute epidural h a e m a t o m a , a n d address salient points regarding the clinical presentations and MRI findings.
Case R e p o r t A 50 year old w o m a n with a 12 year history of diabetes mellitus had an attack of tingling pain radiating from her n e c k to the right s h o u l d e r on July 5, 1990. T h e pain disappeared several hours after its onset but r e c u r r e d 6
days later, f o l l o w e d by r i g h t h e m i p a r e s i s . An MRI p e r f o r m e d on July 14 showed a lesion located from C2 to C6 that was compressing the spinal cord forward within the spinal canal. T h e lesion displayed mixed intensity on T1- a n d T2-weighted, and proton-density images (Fig. 1). A hypointense b a n d was seen between the spinal cord and the lesion, w h i c h i n d i c a t e d t h e lesion was l o c a t e d extradurally. Although e n h a n c e m e n t of the dura adjacent to the lesion was evident after injection of gadoliniumDTPA (Gd-DTPA), there was no obvious e n h a n c e m e n t of the lesion p r o p e r (Fig. 2). The patient was transferred to our clinic with the diagnosis of spinal epidural haematoma. T h e n e u r o l o g i c a l e x a m i n a t i o n at admission revealed nuchal stiffness, right hemiparesis, hypaesthesia for pain and t e m p e r a t u r e below the level of C4, slightly decreased sensation of vibration in the distal part of the left lower extremity, and slight hypotonus of the anal sphincter. T h e neurological diagnosis was radiculomyelopathy at the level of C4 and diabetic n e u r o p a t h y of the left lower limb. I n v e s t i g a t i o n of p o t e n t i a l vascular lesions by spinal angiography was p r o p o s e d but was not done because of the patient's serious diabetic nephropathy. After evaluation of diabetic nephropathy and correction of high blood glucose, total laminectomy was
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p e r f o r m e d f r o m C3 to C6 on July 16. T h e e p i d u r a l h a e m a t o m a was associated with a small piece of vascular tissue that showed a t e n d e n c y to b l e e d easily d u r i n g manipulation. The tissue a d h e r e d tightly to the d u r a and was n o t attached to the adjacent vertebrae. N e i t h e r a feeding artery n o r a draining vein could be identified. T h e h a e m a t o m a a n d the vascular tissue were totally removed. T h e histological examination of the resected specimen revealed closely clustered and dilated vessels, consisting of a thin e n d o t h e l i u m and a fibrous, partially hyalinised wall. No elastic plate was seen in the vessel wall by routine elastic staining. These features were compatible with those of a cavernous h a e m a n g i o m a (Fig. 3). Although the right radicular pain disappeared after the operation, the right hemiparesis and the hypaesthesia worsened transiently, a n d a significant urinary retention occurred. An MRI o b t a i n e d after surgery disclosed no residual h a e m a t o m a (Fig. 4). T h e p a t i e n t r e c o v e r e d gradually from the neurological deficits m e n t i o n e d above.
Fig. 3 Photomicrograph of the vascular tissue showing clustered and dilated vessels of hyalinised fibrous wall with thin endothelium. (HE Stain, x 40.)
Fig. 1 Magnetic resonance (MR) images showing the lesion (arrow) of mixed intensity from C2 to C6. Left: T1- weighted sagittal image, centre: proton-density image, right: T2-weighted image.
Fig. 4 Postoperative Tl-weighted sagittal MR image. Laminectomy from C3 to C6 is shown. Residual hematoma is not seen in the spinal region.
T h r e e years later, the p a t i e n t is a l m o s t c o m p l e t e l y i n d e p e n d e n t with respect to the usual daily activity, except for a slight right hemiparesis.
Discussion Fig. 2 Tl-weighted axial MR image. A band of hypointensity (arrow head) is seen between the lesion (arrow) and the spinal cord (extradural sign). Enhancement of the dura adjacent to the lesion is shown after Gd-DTPA injection. Upper: Gd-DTPA (-), lower: Gd-DTPA (+)
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Solitary spinal epidural cavernous haemangiomas separate f r o m surrounding vertebrae are relatively rare; there have b e e n only 34 cases in the literature. 1-24A review of these cases revealed a m a r k e d p r e p o n d e r a n c e of male patients, an almost equal distribution f r o m the third to the sixth d e c a d e s o f life a n d t h e o b s e r v a t i o n t h a t t h e s e h a e m a n g i o m a s are seen most frequently in the thoracic region. The clinical presentation of cavernous haemangiomas can be rather quite variable. We u n d e r t o o k a systematic analysis of the previously described 34 cases by using the classification of spinal AVMs. 25 They were subdivided into 3 types; 8 of the h a e m a n g i o m a s were of
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Spinal epidural cavernous haemangioma Case reports
the apoplectic type, 7, 16, 18, 22-244 were o f the i n t e r m i t t e n t t y p e , 15, 20, 24 and the r e m a i n i n g 22 were of the chronic progressive type 1-14,]7,19, 2], 24(Table 1). As is evident f r o m the clinical and histological data presented, o u r present case belongs to the apoplectic type o f spinal epidural cavernous h a e m a n g i o m a . Approximately 250 cases of spinal epidural h a e m a t o m a have b e e n r e p o r t e d so far, a n d a variety o f possible aetiologies have been suggested for their occurrence; 26-29; however, m o r e than half of the epidural h a e m a t o m a s are t h o u g h t to be spontaneous. 26A vascular anomaly, such as AVM or h a e m a n g i o m a , is sometimes described as being a source of secondary spinal epidural h a e m a t o m a s , a n d it is assumed that undiscovered vascular malformations may be the m a j o r cause o f s p o n t a n e o u s spinal e p i d u r a l h a e m a t o m a s . 8,27-30 A r u p t u r e d s m a l l c a v e r n o u s h a e m a n g i o m a can be easily overlooked in a h a e m a t o m a 30, therefore a comprehensive histological examination of spontaneous spinal epidural h a e m a t o m a s is essential for the identification of a small cavernous haemangioma. 3], 32 C o m p u t e d t o m o g r a p h y (CT) m a y b e u s e f u l in detecting a large epidural h a e m a t o m a . However, a relatively small h a e m a t o m a may be misdiagnosed by this Table I
No.
modality as an e x t r u d e d disc fragment, a synovial cyst, or as an epidural t u m o u r o n selected axial Sections. s3 On the o t h e r hand, the h a e m a t o m a can be easily localised to the epidural space by MRI because of its well defined, tapered convex margins, and the presence of cerebrospinal fluid intervening between the h a e m a t o m a and the spinal cord. 33 C o m p r o m i s e of the thecal sac and spinal cord is also readily determined, especially if multiple imaging planes are used. 34, a5 To date, MRI findings of solitary spinal c a v e r n o u s h a e m a n g i o m a s have b e e n r e p o r t e d only in 5 3, 6, 7, 9 of the r e p o r t e d 34 cases. In comparison with spinal cord, a solitary spinal cavernous h a e m a n g i o m a shows iso- or hypointensity on Tl-weighted a n d h y p e r i n t e n s i t y on T 2 - w e i g h t e d i m a g e s b u t its radiographic features are nonspecific 3, 6, 36 and may be c o m p a t i b l e with the MRI f e a t u r e s o f n e u r o f i b r o m a , s c h w a n n o m a , or m e n i n g i o m a 36 H a i m e s a n d Krol p r o p o s e d that the administration of contrast m e d i u m might be helpful for the preoperative differential diagnosis of spinal cavernous h a e m a n g i o m a . 6 In our present case the cavernous h a e m a n g i o m a itself was too small to be d e t e c t e d p r e o p e r a t i v e l y . In cases w h o show a c u t e neurological deterioration, MR or angiographic study may
Summary of the reported cases of solitary spinal epidural cavernous haemangioma
Author (Year)
Age/Sex
Site
Clin Pr
Aetiol
No.
Author (Year)
Age/Sex
Site
Clin Pr
Aetiol
1
Bucy (1932) 2
58/M
Th2-4
Chr
GON
18
Yoshinaga et al. (1986)23
35 / M
Th2
Apo
AH
2
Guthkelth (1948) s
37 / F
Th2-6
Chr
GON
19
Kurose et al. (1989 )9
38 / M
Th7
Chr
GON
3
Araki et al. (1966) 1
27/M
Th5-10
Chr
GON
20
Lee et al. (1990) l°
25 / M
Th5-7
Chr
GON
4
Makk et al. (1969) 11
52 / F
Th5-6
Chr
GON
21
Lee et al. (1990) 10
60/M
Th8-10
Chr
GON
5
Fukui et al. (1978) 4
55 / M
Th4-6
Chr
GON
22
Lee et al. (1990) l°
27 / F
L5
Chr
GON
6
Fukui et al. (1978) 4
57 / M
Th5-6
Chr
GON
23
Enomoto et al. (1991) 3
42 / M
Th5-7
Chr
GON
7
Richardson et al. (1979) 2o 36 / M
C6-7
Int
GON
24
Haimes et al. (1991)6
46 / M
Th2-4
Chr
GON
8
Koyama et al. (1981) 8
52 / F C5-Thl
Chr
GON
25
Morello et al. (1991) 12
60 / M
Th7-9
Chr
GON
9
Padovani et al. (1981)18
39 / M C3-Th2
Apo
GON
26
Morello et al. (1991) 12
53 / M
Th4-6
Chr
GON
10
Nakagawaetal.(1982) TM 4 7 / M
Th3-9
Chr
GON
27
Hillman et al. (1991)24
14/F
Th2
Apo
GON
11-
Nakamura et al. (1982) 15 3 2 / M
C4-6
Int
GON
28
Hillman et a1.(1991)24
64/M
Th4-5
Chr
GON
12
Nakamuraetal.(1982) is 2 6 / M
C7-Th2
Int
GON
29
Hillman et al. (1991) 24
13 / M
Thl-4
Apo
AH
13
Padovani et al. (1982) 19
75 / M
Th3-6
Chr
GON
30
Hillman et al. (1991 )24
64/F
Th4-5
Apo
GON
14
Ojeda et al. (1986) 17
61 / M
Th3-4
Chr
GON
31
Hillman et al. (1991) 24
44 / M
L4-5
Int
CH
15
Morioka et al. (I986) 13
50 / M
Th2-3
Chr
GON
32
Isla et al. (1993) 7
70 / M
L3-4
Chr
GON
16
Ogawa et a l. (1986) 16
68 / M
Th2-5
Apo
AH
33
Isla et al. (1993) 7
16 / F C6-Th2
Apo
GON
I7
Shikata et al. (1986) 21
41/M
C7-Th4
Chr
GON
34
Sigh et al. (1993) 22
40 / M
Th7-9
Apo
GON
CH
35
Present case
50 / F
C5-6
Apo
AH
July 1 9 9 5
267
Clin Pr, Clinical Presentation; Chr, chronic progressive; Int, intermittent; Apo, apoplectic Atiology; GON, growth of nidus; AH, acute haemorrhage; CH, chronic haemorrhage
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Spinal epidural cavernous haemangioma
result in u n n e c e s s a r y time delay in the definitive surgical treatment. O n the basis o f o u r observation a n d perusal o f the literature, we suggest that a t h o r o u g h a n d m e t i c u l o u s i n t r a o p e r a t i v e investigation s h o u l d be p e r f o r m e d o n patients with a spinal epidural h a e m a t o m a in o r d e r to rule o u t a small cavernous h a e m a n g i o m a as the cause o f the h a e m a t o m a , especially in a case without preoperative MRI. Received 1 September1994 Accepted for publication 20 December 1994
Correspondence and offprint requests: Takafumi Inoue, MD Department of Neurosurgery, Saga Medical School, 5-1-1 Nabeshima, Saga 849, Japan Tel: 81-952-31-6511 Fax: 81-952-33-2517
References 1. Araki T, Yonekura M, Imakyure T. A case of extradural hemangioma. Seikei Geka To Saigai Geka 1966; 16: 37-39. 2. Bucy PC. Blood vessel tumors of the spinal canal. Surg Clin N A m e r 1932; 12: 1323-1337. 3. Enomoto H, Goto H. Spinal epidural cavernous angioma. MRI finding. Neuroradiology 1991; 33: 462. 4. Fukui M, Numaguchi Y, Sawada K, et al. Cavernous hemangioma of the central nervous system. Neurol Med Chit (Tokyo) 1978; 18: 863-871. 5. Guthkelth AN. Hemangioma involving the spinal epidural space. J Neurol Neurosurg Psychiat 1948; 11: 199-210. 6. Haimes AB, Krol G. Dumbbell-shaped spinal cavernous angioma, a case report. AJNR 1991; 12: 1021-1022. 7. Isla A, Alverez F, Morales C, Garcia BM. Spinal epidural hemangioma. J Neurosurg Sci 1993; 37: 39-42. 8. Koyama T, HanakitaJ, HandaJ. Solitary spinal epidural angiomas. Neurol Surg 1981; 9: 91-96. 9. Kurose K, Kishi H, Sadatoh T. Spinal epidural cavernous hemangioma. Neurol Med Chir (Tokyo) 1989; 29: 538542. 10. LeeJP, Wang AD, Wai YY,,Ho YS. Spinal extradural cavernous hemangioma. Surg Neurol 1990; 34: 345-351. 11. Makk L, Smiley GL. Hemangioma of spinal cord. J Kentucky Med Association 1969; 67: 825-826. 12. Morello A, Tumbiolo A, Pinto G, Lo DB. Cavernous angioma of the spinal dura. J Neurosurg Sci 1991; 35: 3135. 13. Morioka T, Hiroyuki N, Matsushima T, Hasuo K. Dumbbell-shaped spinal epidural cavernous angioma. Surg Neurol 1986; 25: 142-144. 14. Nakagawa N, Bussaka H, Takahashi M, et al. A case of spinal cavernous hemangioma. Rinsho Hoshasen 1982; 27: 943-946. 15. Nakamura T, Ubatani T, Honma T, et al. Three cases of epidural angioma with relapsing tetraplegia. Rinsho Seikei Geka 1982; 17: 181-187.
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16. Ogawa T, Kikuchi T, Ikeuchi S, Sanada S, Nakajima T, Anno I. CT findings of acute spinal epidural hematoma due to raptured cavernous angioma. Neurol Surg 1986; 14: 687-691. 17. Ojeda VJ, Arandia EG. Primary extradural neoplasms causing spinal cord compression. Aust N z J Surg 1983; 1: 77-83. 18. Padovani R, Poppi M, Pozzati E, Tognetti F, Querzola C. Spinal epidural hemangioma. Spine 1981; 6: 336-340. 19. Padovani R, Tognetti F, Proietti D, Pozzari E, Servadei E Extrathecal cavernous hemangioma. Surg Neurol 1982; 18: 463-465. 20. Richardson RR, Cerullo LJ. Spinal epidural cavernous hemangioma. Surg Neurol 1979; 12: 266-268. 21. ShikataJ, Mikawa Y, Akagi M, et al. A case report of spinal epidural cavernous angioma. Rinsho Seikei Geka 1986; 21: 939-942. 22. Singh RV, Suys S, Campbell DA, BroomeJC. Spinal extradural cavernous angioma. BrJ Neurosurg 1993; 7: 79-81. 23. Yoshinaga K, Inoue S, Minami S, et al. Two cases of spinal epidural angioma. Kanto Seikei Saigai Kaishi 1986; 17: 378-384. 24. HillmanJ, Bynke O. Solitary extradural cavernous hemangioma in the spinal canal. Report of five cases. Surg Neurol 1991; 36: 19-24. 25. Yasargil MG. Diagnosis and treatment of spinal cord arteriovenous malformations. Prog Neurol Surg 1971; 4: 355-428. 26. Bruyn GW, Bosma NJ. Spinal extradural haematoma. In: Vinken PJ and Bruyn GW (ed) : Injuries of the Spine and Spinal Cord, Part II. Handbook of Clinical Neurology. Amsterdam: North-Holland, 1976:1-30 27. Pan G, Kulkarni M, MacDougall DJ, Minor ME. Traumatic epidural hematoma of the cervical spine: diagnosis with magnetic resonance imaging. J Neurosurg 1988; 68: 798-801. 28. Wisoff HS. Spontaneous intraspinal hemorrhage. In: Wilkins RH and Rengachary SS (ed) : Neurosurgery. New York: Mcgraw-Hill, 1985:1500-1504 29. Wittebol MC, van Veelen CWM. Spontaneous spinal epidural haematoma. Etiological consideration. Clin Neurol Neurosurg 1984; 86: 265-270. 30. Cube HM. Spinal extradnral hemorrhage.J Neurosurg 1962; 19: 171-172. 31. Jellinger K. Vascular malformation of the central nervous system: a morphological overview. Neurosurg Rev 1986; 9: 177-216. 32. Lougheed WM, Hoffman HJ. Spontaneous extradural hematoma. Neurology 1960; 10: 1059-1063. 33. Rothfus WE, Chedid MK, Deeb ZL, Abla AA, MaroonJC, Sherman RL. MR imaging in the diagnosis of spinal epidural hematomas. J Comut Assist Tomogr 1987; 11: 855-854. 34. Beatty RM, Winston KR. Spontaneous cervical epidural hematoma.J Neurosurg 1984; 61: 143-148. 35. Boyd HR, Pear BL. Chronic spontaneous spinal epidural hematoma. Report of two cases.J Neurosurg 1972; 36: 239-242. 36. Burk DLJr, BrunbergJA, Kanel E, Latchaw RE, Wolf GL. Spinal and paraspinal neUrofibromatosis: surface coil MR imaging at 1.5 T. Radiology 1987; 162: 797-801.
July 1995
This report describes a case o f solitary spinal epidural cavernous h a e m a n g i o m a presenting as an acute epidural haematoma. The patient is a 50 year old diabetic female with right brachalgia and hemiparesis. Magnetic resonance imaging (MRI) demonstrated the presence o f a cervical spinal epidural haematoma. The patient underwent laminectomy and total removal o f the h a e m a t o m a and the accompanying vascular tissue. The diagnosis o f cavernous h a e m a n g i o m a was proven by histological examination. Because the source o f bleeding in m o s t cases o f spontaneous spinal epidural h a e m a t o m a is usually obscure, the authors emphasise the possibility that a small cavernous h a e m a n g i o m a can be one o f the bleeding sources in s o m e o f these cases. Journal of Clinical Neuroscience 1995, 2(3):265-268
© Pearson Professional 1995
Keywords: Spinal epidural haematoma, Cavernous haemangioma, Magnetic resonance imaging
Introduction Spinal epidural cavernous h a e m a n g i o m a s are c o m m o n l y seen as e x t e n s i o n s f r o m a v e r t e b r a l h a e m a n g i o m a . However, the report of a solitary spinal epidural cavernous h a e m a n g i o m a is r a t h e r rare a n d 34 cases have b e e n r e p o r t e d to date. ~-24 Vascular anomaly, such as AVM or h a e m a n g i o m a , is s o m e t i m e s described as the source o f spinal epidural haematomas, although in most patients with spontaneous spinal epidural h a e m a t o m a the precise source of bleeding is usually obscure. T h e r e have b e e n only three r e p o r t e d cases in which a r u p t u r e d spinal e p i d u r a l c a v e r n o u s h a e m a n g i o m a was recognised as the source of a spinal epidural h a e m a t o m a . 16, 2a, 24 We r e p o r t a r a r e case of solitary spinal e p i d u r a l cavernous h a e m a n g i o m a in a patient diagnosed as having an acute epidural h a e m a t o m a , a n d address salient points regarding the clinical presentations and MRI findings.
Case R e p o r t A 50 year old w o m a n with a 12 year history of diabetes mellitus had an attack of tingling pain radiating from her n e c k to the right s h o u l d e r on July 5, 1990. T h e pain disappeared several hours after its onset but r e c u r r e d 6
days later, f o l l o w e d by r i g h t h e m i p a r e s i s . An MRI p e r f o r m e d on July 14 showed a lesion located from C2 to C6 that was compressing the spinal cord forward within the spinal canal. T h e lesion displayed mixed intensity on T1- a n d T2-weighted, and proton-density images (Fig. 1). A hypointense b a n d was seen between the spinal cord and the lesion, w h i c h i n d i c a t e d t h e lesion was l o c a t e d extradurally. Although e n h a n c e m e n t of the dura adjacent to the lesion was evident after injection of gadoliniumDTPA (Gd-DTPA), there was no obvious e n h a n c e m e n t of the lesion p r o p e r (Fig. 2). The patient was transferred to our clinic with the diagnosis of spinal epidural haematoma. T h e n e u r o l o g i c a l e x a m i n a t i o n at admission revealed nuchal stiffness, right hemiparesis, hypaesthesia for pain and t e m p e r a t u r e below the level of C4, slightly decreased sensation of vibration in the distal part of the left lower extremity, and slight hypotonus of the anal sphincter. T h e neurological diagnosis was radiculomyelopathy at the level of C4 and diabetic n e u r o p a t h y of the left lower limb. I n v e s t i g a t i o n of p o t e n t i a l vascular lesions by spinal angiography was p r o p o s e d but was not done because of the patient's serious diabetic nephropathy. After evaluation of diabetic nephropathy and correction of high blood glucose, total laminectomy was
J. Clin. Neuroscience
Volume 2
Number 3
July 1995
265
Case reports
Spinal epidural cavernous haemangioma
p e r f o r m e d f r o m C3 to C6 on July 16. T h e e p i d u r a l h a e m a t o m a was associated with a small piece of vascular tissue that showed a t e n d e n c y to b l e e d easily d u r i n g manipulation. The tissue a d h e r e d tightly to the d u r a and was n o t attached to the adjacent vertebrae. N e i t h e r a feeding artery n o r a draining vein could be identified. T h e h a e m a t o m a a n d the vascular tissue were totally removed. T h e histological examination of the resected specimen revealed closely clustered and dilated vessels, consisting of a thin e n d o t h e l i u m and a fibrous, partially hyalinised wall. No elastic plate was seen in the vessel wall by routine elastic staining. These features were compatible with those of a cavernous h a e m a n g i o m a (Fig. 3). Although the right radicular pain disappeared after the operation, the right hemiparesis and the hypaesthesia worsened transiently, a n d a significant urinary retention occurred. An MRI o b t a i n e d after surgery disclosed no residual h a e m a t o m a (Fig. 4). T h e p a t i e n t r e c o v e r e d gradually from the neurological deficits m e n t i o n e d above.
Fig. 3 Photomicrograph of the vascular tissue showing clustered and dilated vessels of hyalinised fibrous wall with thin endothelium. (HE Stain, x 40.)
Fig. 1 Magnetic resonance (MR) images showing the lesion (arrow) of mixed intensity from C2 to C6. Left: T1- weighted sagittal image, centre: proton-density image, right: T2-weighted image.
Fig. 4 Postoperative Tl-weighted sagittal MR image. Laminectomy from C3 to C6 is shown. Residual hematoma is not seen in the spinal region.
T h r e e years later, the p a t i e n t is a l m o s t c o m p l e t e l y i n d e p e n d e n t with respect to the usual daily activity, except for a slight right hemiparesis.
Discussion Fig. 2 Tl-weighted axial MR image. A band of hypointensity (arrow head) is seen between the lesion (arrow) and the spinal cord (extradural sign). Enhancement of the dura adjacent to the lesion is shown after Gd-DTPA injection. Upper: Gd-DTPA (-), lower: Gd-DTPA (+)
266
J. Clin. Neuroscience
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Number 3
Solitary spinal epidural cavernous haemangiomas separate f r o m surrounding vertebrae are relatively rare; there have b e e n only 34 cases in the literature. 1-24A review of these cases revealed a m a r k e d p r e p o n d e r a n c e of male patients, an almost equal distribution f r o m the third to the sixth d e c a d e s o f life a n d t h e o b s e r v a t i o n t h a t t h e s e h a e m a n g i o m a s are seen most frequently in the thoracic region. The clinical presentation of cavernous haemangiomas can be rather quite variable. We u n d e r t o o k a systematic analysis of the previously described 34 cases by using the classification of spinal AVMs. 25 They were subdivided into 3 types; 8 of the h a e m a n g i o m a s were of
July 1995
Spinal epidural cavernous haemangioma Case reports
the apoplectic type, 7, 16, 18, 22-244 were o f the i n t e r m i t t e n t t y p e , 15, 20, 24 and the r e m a i n i n g 22 were of the chronic progressive type 1-14,]7,19, 2], 24(Table 1). As is evident f r o m the clinical and histological data presented, o u r present case belongs to the apoplectic type o f spinal epidural cavernous h a e m a n g i o m a . Approximately 250 cases of spinal epidural h a e m a t o m a have b e e n r e p o r t e d so far, a n d a variety o f possible aetiologies have been suggested for their occurrence; 26-29; however, m o r e than half of the epidural h a e m a t o m a s are t h o u g h t to be spontaneous. 26A vascular anomaly, such as AVM or h a e m a n g i o m a , is sometimes described as being a source of secondary spinal epidural h a e m a t o m a s , a n d it is assumed that undiscovered vascular malformations may be the m a j o r cause o f s p o n t a n e o u s spinal e p i d u r a l h a e m a t o m a s . 8,27-30 A r u p t u r e d s m a l l c a v e r n o u s h a e m a n g i o m a can be easily overlooked in a h a e m a t o m a 30, therefore a comprehensive histological examination of spontaneous spinal epidural h a e m a t o m a s is essential for the identification of a small cavernous haemangioma. 3], 32 C o m p u t e d t o m o g r a p h y (CT) m a y b e u s e f u l in detecting a large epidural h a e m a t o m a . However, a relatively small h a e m a t o m a may be misdiagnosed by this Table I
No.
modality as an e x t r u d e d disc fragment, a synovial cyst, or as an epidural t u m o u r o n selected axial Sections. s3 On the o t h e r hand, the h a e m a t o m a can be easily localised to the epidural space by MRI because of its well defined, tapered convex margins, and the presence of cerebrospinal fluid intervening between the h a e m a t o m a and the spinal cord. 33 C o m p r o m i s e of the thecal sac and spinal cord is also readily determined, especially if multiple imaging planes are used. 34, a5 To date, MRI findings of solitary spinal c a v e r n o u s h a e m a n g i o m a s have b e e n r e p o r t e d only in 5 3, 6, 7, 9 of the r e p o r t e d 34 cases. In comparison with spinal cord, a solitary spinal cavernous h a e m a n g i o m a shows iso- or hypointensity on Tl-weighted a n d h y p e r i n t e n s i t y on T 2 - w e i g h t e d i m a g e s b u t its radiographic features are nonspecific 3, 6, 36 and may be c o m p a t i b l e with the MRI f e a t u r e s o f n e u r o f i b r o m a , s c h w a n n o m a , or m e n i n g i o m a 36 H a i m e s a n d Krol p r o p o s e d that the administration of contrast m e d i u m might be helpful for the preoperative differential diagnosis of spinal cavernous h a e m a n g i o m a . 6 In our present case the cavernous h a e m a n g i o m a itself was too small to be d e t e c t e d p r e o p e r a t i v e l y . In cases w h o show a c u t e neurological deterioration, MR or angiographic study may
Summary of the reported cases of solitary spinal epidural cavernous haemangioma
Author (Year)
Age/Sex
Site
Clin Pr
Aetiol
No.
Author (Year)
Age/Sex
Site
Clin Pr
Aetiol
1
Bucy (1932) 2
58/M
Th2-4
Chr
GON
18
Yoshinaga et al. (1986)23
35 / M
Th2
Apo
AH
2
Guthkelth (1948) s
37 / F
Th2-6
Chr
GON
19
Kurose et al. (1989 )9
38 / M
Th7
Chr
GON
3
Araki et al. (1966) 1
27/M
Th5-10
Chr
GON
20
Lee et al. (1990) l°
25 / M
Th5-7
Chr
GON
4
Makk et al. (1969) 11
52 / F
Th5-6
Chr
GON
21
Lee et al. (1990) 10
60/M
Th8-10
Chr
GON
5
Fukui et al. (1978) 4
55 / M
Th4-6
Chr
GON
22
Lee et al. (1990) l°
27 / F
L5
Chr
GON
6
Fukui et al. (1978) 4
57 / M
Th5-6
Chr
GON
23
Enomoto et al. (1991) 3
42 / M
Th5-7
Chr
GON
7
Richardson et al. (1979) 2o 36 / M
C6-7
Int
GON
24
Haimes et al. (1991)6
46 / M
Th2-4
Chr
GON
8
Koyama et al. (1981) 8
52 / F C5-Thl
Chr
GON
25
Morello et al. (1991) 12
60 / M
Th7-9
Chr
GON
9
Padovani et al. (1981)18
39 / M C3-Th2
Apo
GON
26
Morello et al. (1991) 12
53 / M
Th4-6
Chr
GON
10
Nakagawaetal.(1982) TM 4 7 / M
Th3-9
Chr
GON
27
Hillman et al. (1991)24
14/F
Th2
Apo
GON
11-
Nakamura et al. (1982) 15 3 2 / M
C4-6
Int
GON
28
Hillman et a1.(1991)24
64/M
Th4-5
Chr
GON
12
Nakamuraetal.(1982) is 2 6 / M
C7-Th2
Int
GON
29
Hillman et al. (1991) 24
13 / M
Thl-4
Apo
AH
13
Padovani et al. (1982) 19
75 / M
Th3-6
Chr
GON
30
Hillman et al. (1991 )24
64/F
Th4-5
Apo
GON
14
Ojeda et al. (1986) 17
61 / M
Th3-4
Chr
GON
31
Hillman et al. (1991) 24
44 / M
L4-5
Int
CH
15
Morioka et al. (I986) 13
50 / M
Th2-3
Chr
GON
32
Isla et al. (1993) 7
70 / M
L3-4
Chr
GON
16
Ogawa et a l. (1986) 16
68 / M
Th2-5
Apo
AH
33
Isla et al. (1993) 7
16 / F C6-Th2
Apo
GON
I7
Shikata et al. (1986) 21
41/M
C7-Th4
Chr
GON
34
Sigh et al. (1993) 22
40 / M
Th7-9
Apo
GON
CH
35
Present case
50 / F
C5-6
Apo
AH
July 1 9 9 5
267
Clin Pr, Clinical Presentation; Chr, chronic progressive; Int, intermittent; Apo, apoplectic Atiology; GON, growth of nidus; AH, acute haemorrhage; CH, chronic haemorrhage
J. Clin. Neuroscience
Volume 2
Number 3
Case reports
Spinal epidural cavernous haemangioma
result in u n n e c e s s a r y time delay in the definitive surgical treatment. O n the basis o f o u r observation a n d perusal o f the literature, we suggest that a t h o r o u g h a n d m e t i c u l o u s i n t r a o p e r a t i v e investigation s h o u l d be p e r f o r m e d o n patients with a spinal epidural h a e m a t o m a in o r d e r to rule o u t a small cavernous h a e m a n g i o m a as the cause o f the h a e m a t o m a , especially in a case without preoperative MRI. Received 1 September1994 Accepted for publication 20 December 1994
Correspondence and offprint requests: Takafumi Inoue, MD Department of Neurosurgery, Saga Medical School, 5-1-1 Nabeshima, Saga 849, Japan Tel: 81-952-31-6511 Fax: 81-952-33-2517
References 1. Araki T, Yonekura M, Imakyure T. A case of extradural hemangioma. Seikei Geka To Saigai Geka 1966; 16: 37-39. 2. Bucy PC. Blood vessel tumors of the spinal canal. Surg Clin N A m e r 1932; 12: 1323-1337. 3. Enomoto H, Goto H. Spinal epidural cavernous angioma. MRI finding. Neuroradiology 1991; 33: 462. 4. Fukui M, Numaguchi Y, Sawada K, et al. Cavernous hemangioma of the central nervous system. Neurol Med Chit (Tokyo) 1978; 18: 863-871. 5. Guthkelth AN. Hemangioma involving the spinal epidural space. J Neurol Neurosurg Psychiat 1948; 11: 199-210. 6. Haimes AB, Krol G. Dumbbell-shaped spinal cavernous angioma, a case report. AJNR 1991; 12: 1021-1022. 7. Isla A, Alverez F, Morales C, Garcia BM. Spinal epidural hemangioma. J Neurosurg Sci 1993; 37: 39-42. 8. Koyama T, HanakitaJ, HandaJ. Solitary spinal epidural angiomas. Neurol Surg 1981; 9: 91-96. 9. Kurose K, Kishi H, Sadatoh T. Spinal epidural cavernous hemangioma. Neurol Med Chir (Tokyo) 1989; 29: 538542. 10. LeeJP, Wang AD, Wai YY,,Ho YS. Spinal extradural cavernous hemangioma. Surg Neurol 1990; 34: 345-351. 11. Makk L, Smiley GL. Hemangioma of spinal cord. J Kentucky Med Association 1969; 67: 825-826. 12. Morello A, Tumbiolo A, Pinto G, Lo DB. Cavernous angioma of the spinal dura. J Neurosurg Sci 1991; 35: 3135. 13. Morioka T, Hiroyuki N, Matsushima T, Hasuo K. Dumbbell-shaped spinal epidural cavernous angioma. Surg Neurol 1986; 25: 142-144. 14. Nakagawa N, Bussaka H, Takahashi M, et al. A case of spinal cavernous hemangioma. Rinsho Hoshasen 1982; 27: 943-946. 15. Nakamura T, Ubatani T, Honma T, et al. Three cases of epidural angioma with relapsing tetraplegia. Rinsho Seikei Geka 1982; 17: 181-187.
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16. Ogawa T, Kikuchi T, Ikeuchi S, Sanada S, Nakajima T, Anno I. CT findings of acute spinal epidural hematoma due to raptured cavernous angioma. Neurol Surg 1986; 14: 687-691. 17. Ojeda VJ, Arandia EG. Primary extradural neoplasms causing spinal cord compression. Aust N z J Surg 1983; 1: 77-83. 18. Padovani R, Poppi M, Pozzati E, Tognetti F, Querzola C. Spinal epidural hemangioma. Spine 1981; 6: 336-340. 19. Padovani R, Tognetti F, Proietti D, Pozzari E, Servadei E Extrathecal cavernous hemangioma. Surg Neurol 1982; 18: 463-465. 20. Richardson RR, Cerullo LJ. Spinal epidural cavernous hemangioma. Surg Neurol 1979; 12: 266-268. 21. ShikataJ, Mikawa Y, Akagi M, et al. A case report of spinal epidural cavernous angioma. Rinsho Seikei Geka 1986; 21: 939-942. 22. Singh RV, Suys S, Campbell DA, BroomeJC. Spinal extradural cavernous angioma. BrJ Neurosurg 1993; 7: 79-81. 23. Yoshinaga K, Inoue S, Minami S, et al. Two cases of spinal epidural angioma. Kanto Seikei Saigai Kaishi 1986; 17: 378-384. 24. HillmanJ, Bynke O. Solitary extradural cavernous hemangioma in the spinal canal. Report of five cases. Surg Neurol 1991; 36: 19-24. 25. Yasargil MG. Diagnosis and treatment of spinal cord arteriovenous malformations. Prog Neurol Surg 1971; 4: 355-428. 26. Bruyn GW, Bosma NJ. Spinal extradural haematoma. In: Vinken PJ and Bruyn GW (ed) : Injuries of the Spine and Spinal Cord, Part II. Handbook of Clinical Neurology. Amsterdam: North-Holland, 1976:1-30 27. Pan G, Kulkarni M, MacDougall DJ, Minor ME. Traumatic epidural hematoma of the cervical spine: diagnosis with magnetic resonance imaging. J Neurosurg 1988; 68: 798-801. 28. Wisoff HS. Spontaneous intraspinal hemorrhage. In: Wilkins RH and Rengachary SS (ed) : Neurosurgery. New York: Mcgraw-Hill, 1985:1500-1504 29. Wittebol MC, van Veelen CWM. Spontaneous spinal epidural haematoma. Etiological consideration. Clin Neurol Neurosurg 1984; 86: 265-270. 30. Cube HM. Spinal extradnral hemorrhage.J Neurosurg 1962; 19: 171-172. 31. Jellinger K. Vascular malformation of the central nervous system: a morphological overview. Neurosurg Rev 1986; 9: 177-216. 32. Lougheed WM, Hoffman HJ. Spontaneous extradural hematoma. Neurology 1960; 10: 1059-1063. 33. Rothfus WE, Chedid MK, Deeb ZL, Abla AA, MaroonJC, Sherman RL. MR imaging in the diagnosis of spinal epidural hematomas. J Comut Assist Tomogr 1987; 11: 855-854. 34. Beatty RM, Winston KR. Spontaneous cervical epidural hematoma.J Neurosurg 1984; 61: 143-148. 35. Boyd HR, Pear BL. Chronic spontaneous spinal epidural hematoma. Report of two cases.J Neurosurg 1972; 36: 239-242. 36. Burk DLJr, BrunbergJA, Kanel E, Latchaw RE, Wolf GL. Spinal and paraspinal neUrofibromatosis: surface coil MR imaging at 1.5 T. Radiology 1987; 162: 797-801.
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