Journal of the Neurological Sciences, 1990, 98:121-138
121
Elsevier JNS 03363
The spontaneous spinal epidural hematoma A study of the etiology Rob J.M. Groen and Hans Ponssen Department of Neurosurgery, Free University Hospital, Amsterdam (The Netherlands)
(Received 19 January, 1990) (Revised, received 1 March, 1990) (Accepted 5 March, 1990)
SUMMARY From the literature 199 cases of spontaneous spinal epidural hematoma (SSEH) are analyzed. With these data and the vascular anatomical characteristics of the spinal epidural space, the theories on the etiology of the SSEH are discussed. There seems to be no relationship between the SSEH and arterial hypertension. The age highly affects the level-distribution of the SSEH. The authors have the opinion that the posterior internal vertebral venous plexus plays an important part in the etiology of the SSEH. In the literature the role of a vascular anomaly in the etiology of the SSEH is overestimated. The cluster-type dural artery can easily be mistaken for a vascular anomaly.
Key words: Spontaneous spinal epidural hematoma; Level-distribution; Arterial hypertension; Posterior internal vertebral venous plexus; Cluster-type artery; Vascular anomaly
INTRODUCTION The spontaneous spinal epidural hematoma (SSEH) is a rare acute condition, initially described in literature by Jackson (1869) and Bain (1897). The classical clinical Correspondence to: R. J. M. Groen, Department of Neurology,UniversityHospital, P.O. Box 30.001, 9700 RB Groningen,The Netherlands.
0022-510X/90/$03.50 © 1990 Elsevier Science Publishers B.V. (Biomedical Division)
122 picture regards an acute onset of severe irradiating back-pain, followed by the signs and symptoms of a rapidly evolving nerve root and/or spinal cord compression, the latter depending on the site of the bleeding. The prognosis is determined m prompt management by means of a decompressive laminectomy and evacuation of the hematoma (Bruyn et al. 1976). In the last two decades many authors reported their cases, contributing to the series of SSEH in the international medical literature. As possible etiological factors, minor trauma (Cooper 1967; Devadiga et al. 1973; Hehman et al. 1968; Svien et al. 1950; Verbrugghen 1946), whooping cough (Jackson 1869), sneezing (Pear 1972; Scott et al. 1976), voiding (Bain 1897), vomiting (Kaplan et al. 1949), lifting (Correa et al. 1978: Ghanem et al. 1978: Lepoire et al. 1961: Rao et al. 1966) (all assumed to produce an acute increase of the pressure in the spinal epidural veins), pregnancy (Bidzinski 1966: Suzuki et al. 1977: Yonekawa et al. 1975), hypertension (Ainslie 1958; Lougheed 1960), atherosclerosis (Ainslie 1958), anticoagulants (Alderman 1956; Busse et al. 1972: Cloward et al. 1955; Devadiga etal. I973: yon Fischbach et al. 1972; Goulon et al. 1967; Harik et al. 1971 ; Weigert 1961: Winer et al. 1959) or bleeding diathesis (Amyes et al. 1955: Caltiauw 1988: Keety e~ al. 1972: London et al. 1974: Stanley et al. 1983) are mentioned. In other cases none of these factors could be found (Chavany et al. 1949: Jackson 1869; Shenkin et al. 19451. A vascular anomaly is thought to be an important source of SSEH (Banerjee 1974; Combelles et al. 1983: Cooper 1967: Cube 1962: Dauch 1986: Dawson 1963: Foo et al. 1980; Kaplan et al. 1949: Koyama et al. 1982; Maxwell et al. 1967: Mtiller et al. 1982; Nichols et al. 1956; Packe et al. 1978; Scott et al. 1976; Solero et al. 1980: Tsai et al. 1975). Nevertheless. in most postoperatively investigated hematomas no histological evidence for a vascular malformation could be found. Other authors (Ainslie 1958; Amyes et al. 1955; Gold 1963; Jackson 1869; Lepoire et al. 1961: Liebeskind et al. 1975; Pear 1972) have the opinion that the internal spinal venous plexus is the major bleeding source. In contrast, Beatty et al. (1984) and B are~o et al. (19871 believe in an arterial genesis of the S S EH. However that may be, a rather large number of acute spinal epidural hematomas is found to be spontaneous m origin (Foo et al. 198 t : Gold 1963; Lougheed et al. 1960: Markham et al. 1967; Schultz et al. 1953). The different ideas from the literature about the etiology of the SS EH are exclusively theoretical. As far as to our knowledge no supportive evidence has been given by any author and no study is published about any relationship between the anatomical properties of the epidural space and the localization of the SSEH. Since the clinical and diagnostic features as well as the postoperative course of the S SEH have been thoroughly discussed in the literature, these items will be left out of consideration in this article.
MATERIAL AND METHODS
Reviewing the world literature, we made a selection of 197 sufficiently documented cases of SSEH. Hematomas following a trauma with spinal dislocations or fractures, or after procedures like epidural anaesthesia, diagnostic lumbar punction or operation, were excluded. Spinal epidural hemorrhage together with spinal subdural
123 TABLE 1 S U M M A R Y O F 199 CASES OF S P O N T A N E O U S SPINAL E P I D U R A L H E M A T O M A S Author
Year
Ainslie
1958
Alderman Amyes Amyot Aring Bain Banerjee
1956 1955 1969 1952 1897 1974
Barefio Beatty
1987 1984
Bidzinski Bruyn
1966 1976
Busse
1972
Calliauw
1988
Carroll Chavany Combelles
1981 1949 1983
Cooper Correa Cube Dauch Dawson
1967 1978 1962 1986 1963
Devadiga
1973
Fischerbach Foo Gauthier Ghanem Giagheddu Gold
1972 1980 1963 1978 1964 1963
Sex F M F M M M M F M M M F M F M M M F F F M M M M M F F F F F M M F M F F M M M F M M F M M F M M F M M F F
Age (yr) 73 70 63 57 57 1, 83 62 18 34 22 46 65 37 26 50 69 60 55 57 57 49 13 20 39 63 72 70 73 67 28 39 67 57 63 72 14 78 29 11 19 15 41 65 57 33 34 8 60 62 72 62 54 67
History Ht Ht Ht Ht, Ac
Ht, Ac
Ht SLE Pregnant Ac CML Moschc. Ac Ac Ac
Hemoph. Ht, Ac
Ac Ac Ac Ac Ac Colon Ca Bypass
Ac Ac Hemang.
Ht Ht Ht Ht
Level D8-DI0 C7 C2-C4 LI-L3 DI l-L4 C5-C7 L3-L5 C2-C3 C7-D1 D7-D9 C3-C6 C2-C7 C3-C7 D2-D5 L2-L5 C6-D5 D 1 l-L3 L3-S 1 D2-D4 D10-D1 D9-L1 C7-D9 C7-D3 D4-D8 D12-L3 C3-C6 LI-IA C6-D2 C3-C4 C4-C7 C3-C5 L2-IA DII-LI C4-C6 DI0-DI2 C3-C5 C2-D2 C6-D1 D3 D2-D6 L2-L5 L5 L5 C7-D3 C2-C7 D8-DI0 DI-D3 C5-C6 D9-L2 C5-C7 C7-D2 C5-C6 D7-D12
Histopathology Unknown Hematoma Unknown Unknown Unknown Unknown Unknown Hematoma Hematoma Hematoma Hematoma Hematoma Hematoma Hematoma Unknown Unknown Unknown Unknown Unknown Hematoma Unknown Hematoma Hematoma Unknown Hematoma Unknown Unknown Hematoma Unknown Unknown Unknown Unknown Hematoma Unknown Hematoma Unknown Unknown Hemangioma Hematoma Venous ang. Venous ang. Hematoma Hematoma Unknown Vasc. malf. Hematoma Hematoma Hematoma Unknown Unknown Unknown Unknown Hematoma
124 T A B L E 1 (continued) Author
Year
Sex
Age (yr)
History
Level
Histopathology
Goulon Griponissiotis Groen (not published) Grollmus
1967 1963 1989
1987
Harris Hassin Hehman Hirai Home
1969 1935 1968 1970 1977
Jackson F.
1963
79 60 29 33 15 29 44 34 66 32 1, 75 44 27 78 1, 17
Ht Ac
Gruszkiewicz
F M M M M M M M M F M M F F F
C6-D 1 C2-C6 D3-D5 D1-D2 C6-DI C6-D1 D1-D3 D5-D7 L5- S 1 D10 C5-D9 C3-C5 C7-D1 D10-Dll D1-D5
Hematoma Unknown Hematoma Hematoma Hemat0ma Hematoma Hematoma Hematoma Hematoma Hematoma Hematoma Unknown Unknown Unknown Hematoma
Jackson R. Jacobson
1869 1966
Jost Kaplan
1970 1949
Khatib Klossek von
1966 1984
Koyama Kuchiwaki Lazorthes
1982 1973 1968
Lecuire Lepoire
1966 1961
Lesoin Levitan
1965 1983
L6vy
1964
Liebeskind Lin Locke London Lougheed
1975 1961 1976 1974 1960
F F F M M F M M M F M M F F F F M M M M M F F M F M F M M M F M M F M
14 61 60 48 63 39 43 86 36 40 18 67 62 23 72 34 32 19 15 33 63 58 90 67 58 23 53 36 50 33 74 67 57 54 55
C1-C7 D2-D4 D12-L2 C5-C6 L1 D5-D6 D9-L1 D12-L4 C5-C7 L2-L5 D1 D1 I-L1 D4-D5 C4-C5 D5-D6 D6-D8 C5-C7 C6-D2 C6-D 1 C7-D2 L4-SI L4 L3 L2-L4 C5-D 1 C7-D2 LI-L3 D5-D6 C2-S 1 D3-D6 D10-LI D 1 l-L5 D3-D5 D5-D10 DI1-S1
Hematoma Hematoma Unknown Unknown Unknown Hematoma Hematoma Unknown Unknown Unknown Venous ang. Unknown Unknown Hematoma Unknown Hematoma Unknown Unknown Unknown Unknown Unknown Unknown Unknown Hematoma Hematoma Hematoma Hematoma Hematoma Hematoma Vasc; malf. Hematoma Unknown Hematoma Unknown Unknown
1975
Ht, Ac Arthr.
Ht Whooping cough Ht Ht, Ac Ac Ac
Ac Ht
Ac
Ac
Ht, Ac Ac Ht Ac Alcohol. Ht Ht
Ht, Ac
125 TABLE 1 (continued) Author
Year
Sex
Age (yr)
History
Level
Histopathology
Lowrey
1959
1967
Maxwell Mayer
1957 1963
McQuarrie
1978
MOiler
1982
Nichols Oldenkott Packer
1956 1966 1978
Panitz Parman
1975 1980
Pear
1972
71 23 52 79 45 56 4 17 49 65 33 55 32 71 65 59 53 15 50 13 17 26 34 15 68 66 75 76 47 73 13 71 63 46
Arthr.
Markham
F M M F M F M F M F F F F F M F F M M F M F M M M M F M M M M M M M
C5-D5 D 11 D12-L2 D8-D 11 C7-D 1 D10-D12 D2-D3 C7-D1 C3-D 1 D7-L3 C4-D3 D 11 C7-D2 C5-C7 D5-D10 D9-D10 D9-D 11 C6-D1 D7-D9 C7-D7 D9 D2 D8-D10 D5-D6 Dll-D12 L5-S 1 L3-L4 C4-C6 D2 L1-S1 D5-D8 D8-DI0 L5 D9-L 1
Unknown Unknown Unknown Hematoma Hematoma Hematoma Hematoma Vasc. malf. Hematoma Unknown Unknown Unknown Unknown Hemangioma Vasc. malf. Hematoma Vasc. malf. Hematoma Unknown Vasc. malf. Vasc. malf. Hematoma Unknown Hematoma Unknown Unknown Unknown Hematoma Unknown Unknown Hematoma Hematoma Hematoma Hematoma
F M M M F F F M M M M F F F M F F
74 21 63 55 2,5 76 63 17 54 11 20 60 78 56 28 6 53
C5-C7 D2-D6 L4-$2 L3-L4 C5-D5 C4-D2 D12-L2 L4-L5 C4-C6 C3-C7 C6-C7 D5-D6 C6-C7 D9-D10 D 12-L 1 D1-D3 C4-C7
Hematoma Hematoma Unknown Unknown Hematoma Unknown Hematoma Hematoma Hematoma Hematoma Hematoma Unknown Unknown Unknown Unknown Hematoma Hematoma
Pendl
1971
Peserico Phillips Piontud Pomm6 Posnikoff Quequet
1959 1981 1979 1959 1968 1987
Rao Rathe RebeUo
1966 1969 1966
Reddy
1972
Robertson Russman
1979 1971
DM
Ht Ac Ht DM
Ac
Postpartum
Ht
Ac Ht, Ac Ac Ht Ac, M. BOrger
Ac
Thromb. Ht
Ht
126 TABLE 1 (continued) Author
Year
Sex
Age (yr)
History
Level
Histopathology
Scharfetter
1972
Schicke Schiffer Schultz
1970 1984 1953
1976
Shenkin
1945
Solero Spill ter Spurny
1980 1989 1964
Stanley Strain Suzuki N. Suzuki S. Svien Telerman Tsai
1983 1964 1977 1968 1950 1972 1975
Valle6 Vapalahti
1982 1975
Ventureyra VerBrugghen Verhagen Weigert Whaley Wirier Wittebol
1979 1946 1986 1961 1962 1959 1984
65 60 63 60 56 24 79 56 34 63 52 1, 75 42 38 15 67 70 t3 57 29 23 67 48 60 20 t7 1, 83 63 63 66 ~ 8 75 70 43 52 70 6 75 52 70 20 62 67 45
Ht Ht Ac Ac Arthr.
Scott
F M F M M M M F M F M M M M F F M M M F F M M M [, M F M F M F M M M M M F F M M F F M M M
D6-LI D9-L1 D l-L3 D12-LI L1-L3 D2-D4 L2-L4 D10-D12 D9-L1 C3-D2 C5-D3 D1-D7 D2-D5 C5-D1 D8-DI0 D9-L2 D 1l-L4 D5-D8 C2-D2 DI-D3 D6-D9 L3-L4 DI2-L4 C4-C7 D3-D4 L3-L4 C5-C7 D12-L1 D9-D 12 D12-L4 D11-LI D1-D3 C5-C7 C4-D2 D10-D12 D10-D12 D6-D11 C6-D6 C4-D1 C4-C7 D1-D3 C4-C6 C3-C6 DI I-L3 C4-D1
Unknown Unknown Hematoma Unknown Unknown Unknown Hematoma Hematoma Hematoma Unknown Unknown Unknown Unknown Vasc. malf. Hematoma Unknown Unknown Hematoma Unknown Unknown Unknown Hematoma Unknown Unknown Hematoma Unknown Hematoma Unknown Unknown Unknown Unknown Hematoma Unknown Unknown Hematoma Unknown Unknown Hematoma Unknown Hematoma Unknown Hematoma Hematoma Unknown Hernatoma
Yonekawa Zilkha
1975 1983
Zuccarello
1980
Ht
Ht, Ac Ac Hemoph. Ac Pregnant
Ac Ht
Ac Ac Ac Arthr. Ac Ac Ac Ht. Ac
CARA Ht. DM Pregnant Ht. Ac Ac
a n d / o r a r a c h n o i d a l e f f u s i o n s o r c o m b i n a t i o n s w i t h t u m o r s in t h e s p i n a l c a n a l a l s o w e r e left o u t o f t h e d i s c u s s i o n . I n c l u d e d are p a t i e n t s w i t h h y p e r t e n s i o n , c a r d i o v a s c u l a r disease, anticoagulant therapy or bleeding diathesis, m i n o r t r a u m a or physical exertion w i t h o u t s p i n a l f r a c t u r e o r d i s l o c a t i o n , a n d all o t h e r c a s e s o f S S E H .
127 50 n u m b
40
e r
o
30
f P a
t I e n t
2o
lO
0-10
10-20 20-30 30-40
40-50 50-60 60-70 70-80 80-80 90-100
age (in yeare)
Fig. 1. Age distribution (decades) in 198 SSEH patients.
With 2 cases that were operated in our own department, we collected 199 patients (Table 1). This concerned 82 females and 117 males, aged between 14 months and 90 years (mean age 46.5 years). The male/female ratio is 1.4: 1. The age distribution of 198 patients is shown in Fig. 1. One patient was left out because of unknown age (Vapalahti and Kuurne 1975). The largest number of patients (53 ~o) is aged between 50 and 80 years. Few patients older than 80 years are reported, possibly because of the small contribution of this age-group in the population.
RESULTS
103 patients had no medical history. Hypertension was present in 26 patients and 11 patients both were hypertensive and adjusted on oral anticoagulants. A group of 40 patients had increased bleeding tendency due to the use of oral anticoagulants (36 patients), alcohol abuse (1 patient), hemophilia (2 patients) or M. Moschcowitz (1 patient). The history of the entire group of 199 patients is described in Table 2. The relationship between the age-distribution and the existence of hypertension and/or the use of anticoagulants is described in Table 3 (n = 73). Hypertension was present in 26 patients, 36 patients used oral anticoagulants and 11 patients were hypertensive as well as adjusted on oral anticoagulants. In 194 cases (97 ~o) the hematoma was confirmed by operation and in 5 cases after autopsy. The predominant site of the SSEH was posterolateral (99~'o) in the spinal epidural space. Only 2 (1 ~o) were located anterior. Histopathological investigation after operation or autopsy was performed in 99 cases (49.7~o). Only in 13 cases (13~o) this showed a more or less defined "vascular
128 TABLE 2 H I S T O R Y O F 199 C A S E S W I T H S P O N T A N E O U S
SPINAL EPIDURAL HEMORRHAGE
History
Score
Healthy . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . Coagulopathies
t03
* iatrogenic ( A c ) . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . * disease . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . Hypertension ( H t ) . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . Both Ht and Ac . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .
37 4 26 11
Systemic disease (SLE/DM/RA/etc.) .................................................... ....................................................................... Malignancy .................................................................... Cardiovasc. disease . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . Toxicosis . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . Whooping cough . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . Resp. disease . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . Fam. hemangiomata .............................................................
: ......
Pregnancy
Total . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .
7 3 2 2 1 1 1 l
199
TABLE 3 AGE-DISTRIBUTION RELATED TO THE COEXISTENCE OF HYPERTENSION AND/OR THE U S E O F A N T I C O A G U L A N T S I N 73 O U T O F 199 P A T I E N T S W I T H S P O N T A N E O U S : S P I N A L EPIDURAL HEMORRHAGE
A g e (yr)
Hypertension
Anticoagulants
Both
30-40 40-50 50-60 60-70 70-80
1 4 13 8
3 7 '~ 12 5
2 2 5 2
3 10 15 30 15
total
26
36
11
73
Total
anomaly". These findings are separately listed and specified in detail in Table 4. According to the histological accounts, in six patients a (hem)angioma was diagnosed and in 7 patients an unspecified "arterial andlor venous malformation" was found. The SSEHs were ranged according to the spinal segments involved. Fig. 2 pictures the distribution of the levels. The total number of segments is 763, resulting in an average of 3.8 segments per patient. The levels C5 down to and including D2 comprise one third (33.2~o) of the total number of segments. A second peak appeared at the level D 12. The relationship between level, age (in decades) and sex was analyzed. There appeared to be an equally dispersion between both sexes, according to age and level.
129
TABLE 4 SUMMARY OF 13 PATIENTS WITH ABNORMAL FINDINGS AFTER HISTOPATHOLOGICAL EXAMINATION Author
Sex
Age
Level
Operation
Histopathology
Cube (1962) Dawson (1963)
M
29
C6-D1
two angiomatous lesions
F
19
D2-D6
M
15
L2-L5
Foo ( 1980) Koyama (1982) Lougheed (1960)
M
33
C2-C7
distended venous channels and a vase. malformation infected hematoma with tangled blood vessels no source found
M
18
DI
abnormal vessel
M
33
D3-D6
profuse bleeding
Mayer (1963) Miiller (1982)
F
17
C7-DI
0clotattachedtofatandvessels
dilated vasc. channels; hemangioma vasc. nodule, vase. channels; venous angioma vase. nodule, vase. channels; angioma arteries and veins; vascular malformation channels with many veins and capillaries; venous angioma numerous veins with little muscle and thin fibrous walls; vase. malform. dilated vessels; angioma
F
71
C5-C7
organized clot
M F F
65 53 13
D5-DI0 D9-DI 1 C7-D7
M
17
D9
venous convolution unknown abnormal vessels coiled over lamina D4-D6 abnormal vessels
M
38
C5-D1
Packer (1978)
Solero (1980)
bleeding epidural vessels around blood clot
arteries and veins; A.-V.-angioma unknown; vase. malformation pathol, veins; vase. malform. clotted blood, 3-4 arteries; vase. rnalform. multiple thick channels; A.-V.malforrn. thick-walled venous vessels; venous malform.
8
6
r ¢
e
?
4
8
2
0 C12 3 4 5 6 7D12
3 4 5 6 7 8 9101112L12 oplnsI eegmente
Fig. 2. Level distribution (%) in 199 SSEH patients.
3 4 5S12
130 20
15 P
e r c e n t
10
$
C12
3 4 5 6 7 D1 2 3 4 5 6 7 8 g 10 1112L1 2 3 4 5 81 2
epinal eeQmente m
0-40 yeare
~
40-80 years
Fig. 3. Level distribution (%) related to age in 196 SSEH patients.
On the contrary the level-distribution showed to depend strongly on the age. The localization of a SSEH at the lower dorsal and lumbosacral segments under the age of 40 years (n = 74) appeared to be an exception. This phenomenon is exemplified in Fig. 3, as well as the level-distribution of the patients aging between 40 and 80 years (n = 122). One patient with unknown age and 2 patients older than 80 years were left out. The interval chosen (40 years), results from the remarkable change in the level distribution that can be observed after the 4th decade. This suggests the existence of two separate groups among the series of SSEH.
DISCUSSION
In the past decades several studies are published on the spinal epidural hematoma (Bruyn et al. 1976: Foo et al. 1981 : Gauthier 1963; von Klossek et al. 1984: Lrvy et al. 1963; Markham et al. 1967; Rebello et al. 1966; Tsai et al. 1975). Our review of 199 patients, concerning strictly SSEH, pointed out some differences in comparison to the findings of other studies. Some authors included patients with(non-spontaneous) spinal epidural hemorrhage after epidural anaesthesia (Foo et al. 1981; Helperin et al. 1971), lumbar puncture (Dutac et al. 1975; Foo et al. 1981) or spinal tumours (Foo et al. 1981 : Griponissiotis et al. 1963), In other articles we met an incomplete documentation, which made critical study difficult (yon Klossek et al. 1984). With regard to the agedistribution and sex-ratiO, t h e discussion witl be brief. The data suggest that the SSEH occurs more frequently in males (1.4: 1 in our series) and at every age. Some authors (Bruyn et al. 1976; Dauch 1986: Jellinger 1975) made a differentiation between children (0-18 years) and adults. However no arguments were given to support this distinction. The number of S S E H in our series is highest between
131 the age of 50 and 80 years (53 ~o). Many authors came to this conclusion (Bruyn et al. 1976; Combelles et al. 1983; Dauch 1986; Foo et al. 1981; Jellinger 1975; von Klossek et al. 1984; L6vy et al. 1963; Piotrowski et al. 1979; Tsai et al. 1975), but up to this moment its significance is unclear. In quite a number of reports arterial hypertension and anticoagulant therapy are mentioned as important pathogenetic factors (Ainslie 1958, Alderman 1956; Cloward et al. 1955; Hassin et al. 1935; Jellinger 1975; Lougheed et al. 1960; Mayer 1963; Oldenkott et al. 1966; Pear 1972; Weigert 1961; Winer et al. 1959). Ainslie (1958) feels that the increase of bleedings after the age of 50 years is based on the coexistence of hypertension and the use of anticoagulants. Some others (Bruyn et al. 1976; Schicke et al. 1970) doubt this strongly. According to the histories of the patients, indeed a relationship is suggested between the occurrence of a SSEH and the coexistence of hypertension and/or the use of anticoagulants. Of 114 patients between 30 and 70 years, 58 (50.8~o)had cardio-vascular problems, from which 27 patients (23.7~o) suffered hypertension (Table 3). However, comparison of these findings with the data of the Hypertension Detection and Follow-up Program Cooperative Group (1977) makes obvious that there is no difference between the percentage of hypertension in our group and the percentage of hypertensive persons (aging 30-69 years) in a population of 158 906 individuals (25.3~o) that underwent home-screening of their blood pressure (Ingelfinger et al. 1987). Therefore we feel that conclusions about a causal relationship between hypertension and S S EH can not be drawn. The role of anticoagulants likewise seems uncertain, but we have no data to support this idea. The etiology of the SSEH is discussed by many authors. Some of them support the theory of the epidural hemorrhage being caused by a rupture of a spinal epidural vein in the venous plexus encircling the spinal dura. They postulate the rupture of a weakened vessel in a pre-existing abnormal epidural venous plexus as the basic pathologic process (Bannerjee et al. 1974; Dauch 1986; Gold 1963; Kaplan et al. 1949) and regard the internal spinal venous plexus as a "locus minoris resistentiae" (Ghanem et al. 1978; Schicke et al. 1970). Liebeskind (1975) stated that the cervico-thoracic and the thoraco-lumbar junctions are the sites of least resistance in the normal curved spine, suggesting mechanical factors to be of etiological and localizational importance. Indeed, in our series the epidural hematomas are most common at these segments. Beatty et al. (1984) assumed an arterial hemorrhage, in a review of 43 cases of spontaneous cervical epidural hematoma. Robertson et al. (1979) mentioned a rupture of a so-called cryptic vascular anomaly as a possible etiologic factor, analogous to what is said to be the most common cause of non-traumatic intracranial hemorrhage in children (Menkes 1974). A rupture of such a small cryptic epidural angiomatous structure could be responsible for some cases of SSEH (Robertson et al. 1979). In many publications the authors propose the existence of a true vascular anomaly as the cause of SSEH (Banerjee et al. 1974; Combelles et al. 1983; Cube 1962; Dauch 1986; Dawson 1963; Foo et al. 1980; Kaplan et al. 1949; Koyama et al. 1982; Lougheed et al. 1960; Maxwell et al. 1957; Mtlller et al. 1982; Nichols et al. 1956; Packer et al. 1978 ; Scott et al. 1976; Solero et al. 1980; Tsai et al. 1975). Such an anomaly might well explain the occasional occurrence of back and radicular pain without neurologic deficit which has preceded the final attack
132 by months or years in some cases of SSEH (Jellinger 1975). The inability of some authors to reveal a hemangioma by pathological examination, not necessarily means that such lesions can not be pointed out as the source of SSEH (Cube 1962). An unrecognized vascular anomaly might be demonstrated more frequently by the use of serial histological sections (Lougheed et al. 1960). The failure to visualize these malformations in SSEH could be related to the small size of these anomalies or to thrombosis after the initial episode of bleeding (Devadiga et al. 1973). Summarizing the different causes mentioned in literature, the possible ruptures of epidural veins, arteries, cryptic angiomas, vascular malformations or hemangiomas and spinal angiomas are advocated, but none of the authors managed to give (strong) supportive arguments for their theories, nor statistically neither on anatomical basis. The internal epidural venous plexus is subdividedinto a posterior and an anterior part, the first being the largest (Clemens 1961). The anterior internal plexus is covered by the posterior longitudinal vertebral ligament and to some extend it is sheltered from the epidural space (Luyendijk 1962). Mayer (1963) noticed that never an explanation is offered as to why the spinal epidural hematomas are usually situated in the posterior epidural space. We feel this phenomenon to be elucidated by the absence of a shielding structure like the posterior longitudinal vertebral ligament, as well as by the fact that the posterior plexus is much bigger than the anterior internal spinal venous plexus (Clemens 1961; Luyendijk 1962). The venous theory is supported by the fact that the posterior internal vertebral venous plexus shows an increased convolution of veins in the cervico-dorsal area (Clemens 1961). With regard to the level distribution in the age group between 0 and 40 years, as exemplified in Fig. 4. the largest segmental involvement is at this level (C6-D3). A simular picture is seen in the second age group (40-80 years), however in this series a second manifest peak occurs at the dorso-lumbar change-over (D10-L3). Clemens (1961) noticed a (much smaller) increase of veins in the lumbar posterior internal venous plexus, however this does explain nor the clear age differences neither the frequency peak at D 12. Therefore the local volume of the posterior internal venous plexus can not answer all the questions about the age and segmental distribution of the S S EH. As illustrated, aging plays an important part in the level distribution, concerning the involvement of the dorso-lumbar region. However, it seems very unlikely that only degenerative changes of the spinal column and/or the spinal epidural vascular structures are the explanation for this phenomenon. In our review 13 hematomas are reported to be the result of a ruptured epidural angioma resp. vascular malformation (see Table 4). According to the histological accounts in these cases, we have some marginal notes. Dawson (1963) (case 2), Foo et al. (1980) and MOiler et al. (1982) (case 1) detected structures with "intimate union of arteries and veins" in their histological preparations. Their reports of these small "vascular anomalies" made us think of the definition of the cluster type artery, as reported by Lazorthes et al. (1970). As far as we know, these structures are never mentioned in discussions about the etiology of the SSEH. Because of the unacquaintedness with these vascular clews, the cluster arteries can easily be mistaken for a "'vascular malformation" (Lazorthes et al. 1970) and erroneously considered responsible for the
133 SSEH. In some reports (Dawson (1963) (case 1), Koyama et al. (1982), Lougheed et al. (1960), Mayer (1963), Mtlller et al. (1982) (cases 2 and 3), Solero et al. (1980)) the authors concluded to a vascular anomaly after meeting with venous clusters in an epidural hematoma (Table 4). In our opinion it should be taken into consideration that in these cases the hematomas enclosed congested or distended vessels of the posterior internal vertebral venous plexus. In the literature the vertebral hemangioma, in particular the purely epidural hemangioma, is regarded as a very rare condition. Most cases of spinal epidural hemangiomas are vertebral angiomas extending into or invading the vertebral epidural space (Guthkelch 1948; Vogelsang 1970). Purely epidural hemangiomas almost always seem to occur in the thoracic spinal canal, predominantly in females (Umbach et al. 1976). The histological structure varies from cavernous hemangioma to less mature hemangio-endothelioma and hemangio-blastoma (Guthkelch 1948). The clinical signs of hemangiomas of the spinal epidural space are those of progressive paraplegia of varying rapidity of onset, but usually this developes within a few months. Some cases with temporary remission are known. Only one case of sudden and complete paraplegia was noticed and was shown at autopsy to have resulted from an epidural hemorrhage from the vessels of an epidural vascular tumour (Guthkelch 1948). Scharfetter (1972) found it remarkable that never an acute epidural hemorrhage was mentioned in publications about spinal epidural vascular malformations. With regard to the clinical history of spinal (epidural) (hem)angiomas and the doubtful results of histological examination in SSEH in this review, we question the predominant role of a vascular malformation in the pathogenesis of the SSEH. This opinion seems to be affirmed by the recent report of a SSEH in a patient with a vertebro-epidural hemangioma (postoperatively diagnosed by angiography) several segments above the actual spinal epidural hematoma (ter Spill et al. 1989).
CONCLUSIONS
(1) The anatomical dimensions of the posterior internal vertebral venous plexus and the level distribution in 199 cases of SSEH, indicate that the majority of SSEH result from a rupture of this particular vascular network. (2) The cluster type artery can easily be mistaken for a vascular anomaly, because of its remarkable morphology and the unacquaintedness of the clinician with this structure. (3) The vertebral and/or epidural hemangioma plays an insignificant role in the etiology of the SSEH. (4) The rupture of a spinal epidural artery (cluster- or spiral-type) might be responsible for some cases of SSEH. (5) Despite many advocating reports, arterial hypertension does not play a role in the pathogenesis of the SSEH. (6) The SSEH at the lower dorsal level mainly occurs after the fourth decade. Before that age, an epidural hematoma extending to or originating from the lower dorsal
134 a n d l u m b a r s e g m e n t s is a n e x c e p t i o n . A t t h i s m o m e n t t h i s o b s e r v a t i o n c a n n o t b e e x p l a i n e d in a n a t o m i c a l , f u n c t i o n a l o r p a t h o p h y s i o l o g i c a l t e r m s . T h i s wilt b e a n i t e m f o r f u r t h e r study,
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121
Elsevier JNS 03363
The spontaneous spinal epidural hematoma A study of the etiology Rob J.M. Groen and Hans Ponssen Department of Neurosurgery, Free University Hospital, Amsterdam (The Netherlands)
(Received 19 January, 1990) (Revised, received 1 March, 1990) (Accepted 5 March, 1990)
SUMMARY From the literature 199 cases of spontaneous spinal epidural hematoma (SSEH) are analyzed. With these data and the vascular anatomical characteristics of the spinal epidural space, the theories on the etiology of the SSEH are discussed. There seems to be no relationship between the SSEH and arterial hypertension. The age highly affects the level-distribution of the SSEH. The authors have the opinion that the posterior internal vertebral venous plexus plays an important part in the etiology of the SSEH. In the literature the role of a vascular anomaly in the etiology of the SSEH is overestimated. The cluster-type dural artery can easily be mistaken for a vascular anomaly.
Key words: Spontaneous spinal epidural hematoma; Level-distribution; Arterial hypertension; Posterior internal vertebral venous plexus; Cluster-type artery; Vascular anomaly
INTRODUCTION The spontaneous spinal epidural hematoma (SSEH) is a rare acute condition, initially described in literature by Jackson (1869) and Bain (1897). The classical clinical Correspondence to: R. J. M. Groen, Department of Neurology,UniversityHospital, P.O. Box 30.001, 9700 RB Groningen,The Netherlands.
0022-510X/90/$03.50 © 1990 Elsevier Science Publishers B.V. (Biomedical Division)
122 picture regards an acute onset of severe irradiating back-pain, followed by the signs and symptoms of a rapidly evolving nerve root and/or spinal cord compression, the latter depending on the site of the bleeding. The prognosis is determined m prompt management by means of a decompressive laminectomy and evacuation of the hematoma (Bruyn et al. 1976). In the last two decades many authors reported their cases, contributing to the series of SSEH in the international medical literature. As possible etiological factors, minor trauma (Cooper 1967; Devadiga et al. 1973; Hehman et al. 1968; Svien et al. 1950; Verbrugghen 1946), whooping cough (Jackson 1869), sneezing (Pear 1972; Scott et al. 1976), voiding (Bain 1897), vomiting (Kaplan et al. 1949), lifting (Correa et al. 1978: Ghanem et al. 1978: Lepoire et al. 1961: Rao et al. 1966) (all assumed to produce an acute increase of the pressure in the spinal epidural veins), pregnancy (Bidzinski 1966: Suzuki et al. 1977: Yonekawa et al. 1975), hypertension (Ainslie 1958; Lougheed 1960), atherosclerosis (Ainslie 1958), anticoagulants (Alderman 1956; Busse et al. 1972: Cloward et al. 1955; Devadiga etal. I973: yon Fischbach et al. 1972; Goulon et al. 1967; Harik et al. 1971 ; Weigert 1961: Winer et al. 1959) or bleeding diathesis (Amyes et al. 1955: Caltiauw 1988: Keety e~ al. 1972: London et al. 1974: Stanley et al. 1983) are mentioned. In other cases none of these factors could be found (Chavany et al. 1949: Jackson 1869; Shenkin et al. 19451. A vascular anomaly is thought to be an important source of SSEH (Banerjee 1974; Combelles et al. 1983: Cooper 1967: Cube 1962: Dauch 1986: Dawson 1963: Foo et al. 1980; Kaplan et al. 1949: Koyama et al. 1982; Maxwell et al. 1967: Mtiller et al. 1982; Nichols et al. 1956; Packe et al. 1978; Scott et al. 1976; Solero et al. 1980: Tsai et al. 1975). Nevertheless. in most postoperatively investigated hematomas no histological evidence for a vascular malformation could be found. Other authors (Ainslie 1958; Amyes et al. 1955; Gold 1963; Jackson 1869; Lepoire et al. 1961: Liebeskind et al. 1975; Pear 1972) have the opinion that the internal spinal venous plexus is the major bleeding source. In contrast, Beatty et al. (1984) and B are~o et al. (19871 believe in an arterial genesis of the S S EH. However that may be, a rather large number of acute spinal epidural hematomas is found to be spontaneous m origin (Foo et al. 198 t : Gold 1963; Lougheed et al. 1960: Markham et al. 1967; Schultz et al. 1953). The different ideas from the literature about the etiology of the SS EH are exclusively theoretical. As far as to our knowledge no supportive evidence has been given by any author and no study is published about any relationship between the anatomical properties of the epidural space and the localization of the SSEH. Since the clinical and diagnostic features as well as the postoperative course of the S SEH have been thoroughly discussed in the literature, these items will be left out of consideration in this article.
MATERIAL AND METHODS
Reviewing the world literature, we made a selection of 197 sufficiently documented cases of SSEH. Hematomas following a trauma with spinal dislocations or fractures, or after procedures like epidural anaesthesia, diagnostic lumbar punction or operation, were excluded. Spinal epidural hemorrhage together with spinal subdural
123 TABLE 1 S U M M A R Y O F 199 CASES OF S P O N T A N E O U S SPINAL E P I D U R A L H E M A T O M A S Author
Year
Ainslie
1958
Alderman Amyes Amyot Aring Bain Banerjee
1956 1955 1969 1952 1897 1974
Barefio Beatty
1987 1984
Bidzinski Bruyn
1966 1976
Busse
1972
Calliauw
1988
Carroll Chavany Combelles
1981 1949 1983
Cooper Correa Cube Dauch Dawson
1967 1978 1962 1986 1963
Devadiga
1973
Fischerbach Foo Gauthier Ghanem Giagheddu Gold
1972 1980 1963 1978 1964 1963
Sex F M F M M M M F M M M F M F M M M F F F M M M M M F F F F F M M F M F F M M M F M M F M M F M M F M M F F
Age (yr) 73 70 63 57 57 1, 83 62 18 34 22 46 65 37 26 50 69 60 55 57 57 49 13 20 39 63 72 70 73 67 28 39 67 57 63 72 14 78 29 11 19 15 41 65 57 33 34 8 60 62 72 62 54 67
History Ht Ht Ht Ht, Ac
Ht, Ac
Ht SLE Pregnant Ac CML Moschc. Ac Ac Ac
Hemoph. Ht, Ac
Ac Ac Ac Ac Ac Colon Ca Bypass
Ac Ac Hemang.
Ht Ht Ht Ht
Level D8-DI0 C7 C2-C4 LI-L3 DI l-L4 C5-C7 L3-L5 C2-C3 C7-D1 D7-D9 C3-C6 C2-C7 C3-C7 D2-D5 L2-L5 C6-D5 D 1 l-L3 L3-S 1 D2-D4 D10-D1 D9-L1 C7-D9 C7-D3 D4-D8 D12-L3 C3-C6 LI-IA C6-D2 C3-C4 C4-C7 C3-C5 L2-IA DII-LI C4-C6 DI0-DI2 C3-C5 C2-D2 C6-D1 D3 D2-D6 L2-L5 L5 L5 C7-D3 C2-C7 D8-DI0 DI-D3 C5-C6 D9-L2 C5-C7 C7-D2 C5-C6 D7-D12
Histopathology Unknown Hematoma Unknown Unknown Unknown Unknown Unknown Hematoma Hematoma Hematoma Hematoma Hematoma Hematoma Hematoma Unknown Unknown Unknown Unknown Unknown Hematoma Unknown Hematoma Hematoma Unknown Hematoma Unknown Unknown Hematoma Unknown Unknown Unknown Unknown Hematoma Unknown Hematoma Unknown Unknown Hemangioma Hematoma Venous ang. Venous ang. Hematoma Hematoma Unknown Vasc. malf. Hematoma Hematoma Hematoma Unknown Unknown Unknown Unknown Hematoma
124 T A B L E 1 (continued) Author
Year
Sex
Age (yr)
History
Level
Histopathology
Goulon Griponissiotis Groen (not published) Grollmus
1967 1963 1989
1987
Harris Hassin Hehman Hirai Home
1969 1935 1968 1970 1977
Jackson F.
1963
79 60 29 33 15 29 44 34 66 32 1, 75 44 27 78 1, 17
Ht Ac
Gruszkiewicz
F M M M M M M M M F M M F F F
C6-D 1 C2-C6 D3-D5 D1-D2 C6-DI C6-D1 D1-D3 D5-D7 L5- S 1 D10 C5-D9 C3-C5 C7-D1 D10-Dll D1-D5
Hematoma Unknown Hematoma Hematoma Hemat0ma Hematoma Hematoma Hematoma Hematoma Hematoma Hematoma Unknown Unknown Unknown Hematoma
Jackson R. Jacobson
1869 1966
Jost Kaplan
1970 1949
Khatib Klossek von
1966 1984
Koyama Kuchiwaki Lazorthes
1982 1973 1968
Lecuire Lepoire
1966 1961
Lesoin Levitan
1965 1983
L6vy
1964
Liebeskind Lin Locke London Lougheed
1975 1961 1976 1974 1960
F F F M M F M M M F M M F F F F M M M M M F F M F M F M M M F M M F M
14 61 60 48 63 39 43 86 36 40 18 67 62 23 72 34 32 19 15 33 63 58 90 67 58 23 53 36 50 33 74 67 57 54 55
C1-C7 D2-D4 D12-L2 C5-C6 L1 D5-D6 D9-L1 D12-L4 C5-C7 L2-L5 D1 D1 I-L1 D4-D5 C4-C5 D5-D6 D6-D8 C5-C7 C6-D2 C6-D 1 C7-D2 L4-SI L4 L3 L2-L4 C5-D 1 C7-D2 LI-L3 D5-D6 C2-S 1 D3-D6 D10-LI D 1 l-L5 D3-D5 D5-D10 DI1-S1
Hematoma Hematoma Unknown Unknown Unknown Hematoma Hematoma Unknown Unknown Unknown Venous ang. Unknown Unknown Hematoma Unknown Hematoma Unknown Unknown Unknown Unknown Unknown Unknown Unknown Hematoma Hematoma Hematoma Hematoma Hematoma Hematoma Vasc; malf. Hematoma Unknown Hematoma Unknown Unknown
1975
Ht, Ac Arthr.
Ht Whooping cough Ht Ht, Ac Ac Ac
Ac Ht
Ac
Ac
Ht, Ac Ac Ht Ac Alcohol. Ht Ht
Ht, Ac
125 TABLE 1 (continued) Author
Year
Sex
Age (yr)
History
Level
Histopathology
Lowrey
1959
1967
Maxwell Mayer
1957 1963
McQuarrie
1978
MOiler
1982
Nichols Oldenkott Packer
1956 1966 1978
Panitz Parman
1975 1980
Pear
1972
71 23 52 79 45 56 4 17 49 65 33 55 32 71 65 59 53 15 50 13 17 26 34 15 68 66 75 76 47 73 13 71 63 46
Arthr.
Markham
F M M F M F M F M F F F F F M F F M M F M F M M M M F M M M M M M M
C5-D5 D 11 D12-L2 D8-D 11 C7-D 1 D10-D12 D2-D3 C7-D1 C3-D 1 D7-L3 C4-D3 D 11 C7-D2 C5-C7 D5-D10 D9-D10 D9-D 11 C6-D1 D7-D9 C7-D7 D9 D2 D8-D10 D5-D6 Dll-D12 L5-S 1 L3-L4 C4-C6 D2 L1-S1 D5-D8 D8-DI0 L5 D9-L 1
Unknown Unknown Unknown Hematoma Hematoma Hematoma Hematoma Vasc. malf. Hematoma Unknown Unknown Unknown Unknown Hemangioma Vasc. malf. Hematoma Vasc. malf. Hematoma Unknown Vasc. malf. Vasc. malf. Hematoma Unknown Hematoma Unknown Unknown Unknown Hematoma Unknown Unknown Hematoma Hematoma Hematoma Hematoma
F M M M F F F M M M M F F F M F F
74 21 63 55 2,5 76 63 17 54 11 20 60 78 56 28 6 53
C5-C7 D2-D6 L4-$2 L3-L4 C5-D5 C4-D2 D12-L2 L4-L5 C4-C6 C3-C7 C6-C7 D5-D6 C6-C7 D9-D10 D 12-L 1 D1-D3 C4-C7
Hematoma Hematoma Unknown Unknown Hematoma Unknown Hematoma Hematoma Hematoma Hematoma Hematoma Unknown Unknown Unknown Unknown Hematoma Hematoma
Pendl
1971
Peserico Phillips Piontud Pomm6 Posnikoff Quequet
1959 1981 1979 1959 1968 1987
Rao Rathe RebeUo
1966 1969 1966
Reddy
1972
Robertson Russman
1979 1971
DM
Ht Ac Ht DM
Ac
Postpartum
Ht
Ac Ht, Ac Ac Ht Ac, M. BOrger
Ac
Thromb. Ht
Ht
126 TABLE 1 (continued) Author
Year
Sex
Age (yr)
History
Level
Histopathology
Scharfetter
1972
Schicke Schiffer Schultz
1970 1984 1953
1976
Shenkin
1945
Solero Spill ter Spurny
1980 1989 1964
Stanley Strain Suzuki N. Suzuki S. Svien Telerman Tsai
1983 1964 1977 1968 1950 1972 1975
Valle6 Vapalahti
1982 1975
Ventureyra VerBrugghen Verhagen Weigert Whaley Wirier Wittebol
1979 1946 1986 1961 1962 1959 1984
65 60 63 60 56 24 79 56 34 63 52 1, 75 42 38 15 67 70 t3 57 29 23 67 48 60 20 t7 1, 83 63 63 66 ~ 8 75 70 43 52 70 6 75 52 70 20 62 67 45
Ht Ht Ac Ac Arthr.
Scott
F M F M M M M F M F M M M M F F M M M F F M M M [, M F M F M F M M M M M F F M M F F M M M
D6-LI D9-L1 D l-L3 D12-LI L1-L3 D2-D4 L2-L4 D10-D12 D9-L1 C3-D2 C5-D3 D1-D7 D2-D5 C5-D1 D8-DI0 D9-L2 D 1l-L4 D5-D8 C2-D2 DI-D3 D6-D9 L3-L4 DI2-L4 C4-C7 D3-D4 L3-L4 C5-C7 D12-L1 D9-D 12 D12-L4 D11-LI D1-D3 C5-C7 C4-D2 D10-D12 D10-D12 D6-D11 C6-D6 C4-D1 C4-C7 D1-D3 C4-C6 C3-C6 DI I-L3 C4-D1
Unknown Unknown Hematoma Unknown Unknown Unknown Hematoma Hematoma Hematoma Unknown Unknown Unknown Unknown Vasc. malf. Hematoma Unknown Unknown Hematoma Unknown Unknown Unknown Hematoma Unknown Unknown Hematoma Unknown Hematoma Unknown Unknown Unknown Unknown Hematoma Unknown Unknown Hematoma Unknown Unknown Hematoma Unknown Hematoma Unknown Hematoma Hematoma Unknown Hernatoma
Yonekawa Zilkha
1975 1983
Zuccarello
1980
Ht
Ht, Ac Ac Hemoph. Ac Pregnant
Ac Ht
Ac Ac Ac Arthr. Ac Ac Ac Ht. Ac
CARA Ht. DM Pregnant Ht. Ac Ac
a n d / o r a r a c h n o i d a l e f f u s i o n s o r c o m b i n a t i o n s w i t h t u m o r s in t h e s p i n a l c a n a l a l s o w e r e left o u t o f t h e d i s c u s s i o n . I n c l u d e d are p a t i e n t s w i t h h y p e r t e n s i o n , c a r d i o v a s c u l a r disease, anticoagulant therapy or bleeding diathesis, m i n o r t r a u m a or physical exertion w i t h o u t s p i n a l f r a c t u r e o r d i s l o c a t i o n , a n d all o t h e r c a s e s o f S S E H .
127 50 n u m b
40
e r
o
30
f P a
t I e n t
2o
lO
0-10
10-20 20-30 30-40
40-50 50-60 60-70 70-80 80-80 90-100
age (in yeare)
Fig. 1. Age distribution (decades) in 198 SSEH patients.
With 2 cases that were operated in our own department, we collected 199 patients (Table 1). This concerned 82 females and 117 males, aged between 14 months and 90 years (mean age 46.5 years). The male/female ratio is 1.4: 1. The age distribution of 198 patients is shown in Fig. 1. One patient was left out because of unknown age (Vapalahti and Kuurne 1975). The largest number of patients (53 ~o) is aged between 50 and 80 years. Few patients older than 80 years are reported, possibly because of the small contribution of this age-group in the population.
RESULTS
103 patients had no medical history. Hypertension was present in 26 patients and 11 patients both were hypertensive and adjusted on oral anticoagulants. A group of 40 patients had increased bleeding tendency due to the use of oral anticoagulants (36 patients), alcohol abuse (1 patient), hemophilia (2 patients) or M. Moschcowitz (1 patient). The history of the entire group of 199 patients is described in Table 2. The relationship between the age-distribution and the existence of hypertension and/or the use of anticoagulants is described in Table 3 (n = 73). Hypertension was present in 26 patients, 36 patients used oral anticoagulants and 11 patients were hypertensive as well as adjusted on oral anticoagulants. In 194 cases (97 ~o) the hematoma was confirmed by operation and in 5 cases after autopsy. The predominant site of the SSEH was posterolateral (99~'o) in the spinal epidural space. Only 2 (1 ~o) were located anterior. Histopathological investigation after operation or autopsy was performed in 99 cases (49.7~o). Only in 13 cases (13~o) this showed a more or less defined "vascular
128 TABLE 2 H I S T O R Y O F 199 C A S E S W I T H S P O N T A N E O U S
SPINAL EPIDURAL HEMORRHAGE
History
Score
Healthy . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . Coagulopathies
t03
* iatrogenic ( A c ) . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . * disease . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . Hypertension ( H t ) . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . Both Ht and Ac . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .
37 4 26 11
Systemic disease (SLE/DM/RA/etc.) .................................................... ....................................................................... Malignancy .................................................................... Cardiovasc. disease . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . Toxicosis . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . Whooping cough . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . Resp. disease . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . Fam. hemangiomata .............................................................
: ......
Pregnancy
Total . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .
7 3 2 2 1 1 1 l
199
TABLE 3 AGE-DISTRIBUTION RELATED TO THE COEXISTENCE OF HYPERTENSION AND/OR THE U S E O F A N T I C O A G U L A N T S I N 73 O U T O F 199 P A T I E N T S W I T H S P O N T A N E O U S : S P I N A L EPIDURAL HEMORRHAGE
A g e (yr)
Hypertension
Anticoagulants
Both
30-40 40-50 50-60 60-70 70-80
1 4 13 8
3 7 '~ 12 5
2 2 5 2
3 10 15 30 15
total
26
36
11
73
Total
anomaly". These findings are separately listed and specified in detail in Table 4. According to the histological accounts, in six patients a (hem)angioma was diagnosed and in 7 patients an unspecified "arterial andlor venous malformation" was found. The SSEHs were ranged according to the spinal segments involved. Fig. 2 pictures the distribution of the levels. The total number of segments is 763, resulting in an average of 3.8 segments per patient. The levels C5 down to and including D2 comprise one third (33.2~o) of the total number of segments. A second peak appeared at the level D 12. The relationship between level, age (in decades) and sex was analyzed. There appeared to be an equally dispersion between both sexes, according to age and level.
129
TABLE 4 SUMMARY OF 13 PATIENTS WITH ABNORMAL FINDINGS AFTER HISTOPATHOLOGICAL EXAMINATION Author
Sex
Age
Level
Operation
Histopathology
Cube (1962) Dawson (1963)
M
29
C6-D1
two angiomatous lesions
F
19
D2-D6
M
15
L2-L5
Foo ( 1980) Koyama (1982) Lougheed (1960)
M
33
C2-C7
distended venous channels and a vase. malformation infected hematoma with tangled blood vessels no source found
M
18
DI
abnormal vessel
M
33
D3-D6
profuse bleeding
Mayer (1963) Miiller (1982)
F
17
C7-DI
0clotattachedtofatandvessels
dilated vasc. channels; hemangioma vasc. nodule, vase. channels; venous angioma vase. nodule, vase. channels; angioma arteries and veins; vascular malformation channels with many veins and capillaries; venous angioma numerous veins with little muscle and thin fibrous walls; vase. malform. dilated vessels; angioma
F
71
C5-C7
organized clot
M F F
65 53 13
D5-DI0 D9-DI 1 C7-D7
M
17
D9
venous convolution unknown abnormal vessels coiled over lamina D4-D6 abnormal vessels
M
38
C5-D1
Packer (1978)
Solero (1980)
bleeding epidural vessels around blood clot
arteries and veins; A.-V.-angioma unknown; vase. malformation pathol, veins; vase. malform. clotted blood, 3-4 arteries; vase. rnalform. multiple thick channels; A.-V.malforrn. thick-walled venous vessels; venous malform.
8
6
r ¢
e
?
4
8
2
0 C12 3 4 5 6 7D12
3 4 5 6 7 8 9101112L12 oplnsI eegmente
Fig. 2. Level distribution (%) in 199 SSEH patients.
3 4 5S12
130 20
15 P
e r c e n t
10
$
C12
3 4 5 6 7 D1 2 3 4 5 6 7 8 g 10 1112L1 2 3 4 5 81 2
epinal eeQmente m
0-40 yeare
~
40-80 years
Fig. 3. Level distribution (%) related to age in 196 SSEH patients.
On the contrary the level-distribution showed to depend strongly on the age. The localization of a SSEH at the lower dorsal and lumbosacral segments under the age of 40 years (n = 74) appeared to be an exception. This phenomenon is exemplified in Fig. 3, as well as the level-distribution of the patients aging between 40 and 80 years (n = 122). One patient with unknown age and 2 patients older than 80 years were left out. The interval chosen (40 years), results from the remarkable change in the level distribution that can be observed after the 4th decade. This suggests the existence of two separate groups among the series of SSEH.
DISCUSSION
In the past decades several studies are published on the spinal epidural hematoma (Bruyn et al. 1976: Foo et al. 1981 : Gauthier 1963; von Klossek et al. 1984: Lrvy et al. 1963; Markham et al. 1967; Rebello et al. 1966; Tsai et al. 1975). Our review of 199 patients, concerning strictly SSEH, pointed out some differences in comparison to the findings of other studies. Some authors included patients with(non-spontaneous) spinal epidural hemorrhage after epidural anaesthesia (Foo et al. 1981; Helperin et al. 1971), lumbar puncture (Dutac et al. 1975; Foo et al. 1981) or spinal tumours (Foo et al. 1981 : Griponissiotis et al. 1963), In other articles we met an incomplete documentation, which made critical study difficult (yon Klossek et al. 1984). With regard to the agedistribution and sex-ratiO, t h e discussion witl be brief. The data suggest that the SSEH occurs more frequently in males (1.4: 1 in our series) and at every age. Some authors (Bruyn et al. 1976; Dauch 1986: Jellinger 1975) made a differentiation between children (0-18 years) and adults. However no arguments were given to support this distinction. The number of S S E H in our series is highest between
131 the age of 50 and 80 years (53 ~o). Many authors came to this conclusion (Bruyn et al. 1976; Combelles et al. 1983; Dauch 1986; Foo et al. 1981; Jellinger 1975; von Klossek et al. 1984; L6vy et al. 1963; Piotrowski et al. 1979; Tsai et al. 1975), but up to this moment its significance is unclear. In quite a number of reports arterial hypertension and anticoagulant therapy are mentioned as important pathogenetic factors (Ainslie 1958, Alderman 1956; Cloward et al. 1955; Hassin et al. 1935; Jellinger 1975; Lougheed et al. 1960; Mayer 1963; Oldenkott et al. 1966; Pear 1972; Weigert 1961; Winer et al. 1959). Ainslie (1958) feels that the increase of bleedings after the age of 50 years is based on the coexistence of hypertension and the use of anticoagulants. Some others (Bruyn et al. 1976; Schicke et al. 1970) doubt this strongly. According to the histories of the patients, indeed a relationship is suggested between the occurrence of a SSEH and the coexistence of hypertension and/or the use of anticoagulants. Of 114 patients between 30 and 70 years, 58 (50.8~o)had cardio-vascular problems, from which 27 patients (23.7~o) suffered hypertension (Table 3). However, comparison of these findings with the data of the Hypertension Detection and Follow-up Program Cooperative Group (1977) makes obvious that there is no difference between the percentage of hypertension in our group and the percentage of hypertensive persons (aging 30-69 years) in a population of 158 906 individuals (25.3~o) that underwent home-screening of their blood pressure (Ingelfinger et al. 1987). Therefore we feel that conclusions about a causal relationship between hypertension and S S EH can not be drawn. The role of anticoagulants likewise seems uncertain, but we have no data to support this idea. The etiology of the SSEH is discussed by many authors. Some of them support the theory of the epidural hemorrhage being caused by a rupture of a spinal epidural vein in the venous plexus encircling the spinal dura. They postulate the rupture of a weakened vessel in a pre-existing abnormal epidural venous plexus as the basic pathologic process (Bannerjee et al. 1974; Dauch 1986; Gold 1963; Kaplan et al. 1949) and regard the internal spinal venous plexus as a "locus minoris resistentiae" (Ghanem et al. 1978; Schicke et al. 1970). Liebeskind (1975) stated that the cervico-thoracic and the thoraco-lumbar junctions are the sites of least resistance in the normal curved spine, suggesting mechanical factors to be of etiological and localizational importance. Indeed, in our series the epidural hematomas are most common at these segments. Beatty et al. (1984) assumed an arterial hemorrhage, in a review of 43 cases of spontaneous cervical epidural hematoma. Robertson et al. (1979) mentioned a rupture of a so-called cryptic vascular anomaly as a possible etiologic factor, analogous to what is said to be the most common cause of non-traumatic intracranial hemorrhage in children (Menkes 1974). A rupture of such a small cryptic epidural angiomatous structure could be responsible for some cases of SSEH (Robertson et al. 1979). In many publications the authors propose the existence of a true vascular anomaly as the cause of SSEH (Banerjee et al. 1974; Combelles et al. 1983; Cube 1962; Dauch 1986; Dawson 1963; Foo et al. 1980; Kaplan et al. 1949; Koyama et al. 1982; Lougheed et al. 1960; Maxwell et al. 1957; Mtlller et al. 1982; Nichols et al. 1956; Packer et al. 1978 ; Scott et al. 1976; Solero et al. 1980; Tsai et al. 1975). Such an anomaly might well explain the occasional occurrence of back and radicular pain without neurologic deficit which has preceded the final attack
132 by months or years in some cases of SSEH (Jellinger 1975). The inability of some authors to reveal a hemangioma by pathological examination, not necessarily means that such lesions can not be pointed out as the source of SSEH (Cube 1962). An unrecognized vascular anomaly might be demonstrated more frequently by the use of serial histological sections (Lougheed et al. 1960). The failure to visualize these malformations in SSEH could be related to the small size of these anomalies or to thrombosis after the initial episode of bleeding (Devadiga et al. 1973). Summarizing the different causes mentioned in literature, the possible ruptures of epidural veins, arteries, cryptic angiomas, vascular malformations or hemangiomas and spinal angiomas are advocated, but none of the authors managed to give (strong) supportive arguments for their theories, nor statistically neither on anatomical basis. The internal epidural venous plexus is subdividedinto a posterior and an anterior part, the first being the largest (Clemens 1961). The anterior internal plexus is covered by the posterior longitudinal vertebral ligament and to some extend it is sheltered from the epidural space (Luyendijk 1962). Mayer (1963) noticed that never an explanation is offered as to why the spinal epidural hematomas are usually situated in the posterior epidural space. We feel this phenomenon to be elucidated by the absence of a shielding structure like the posterior longitudinal vertebral ligament, as well as by the fact that the posterior plexus is much bigger than the anterior internal spinal venous plexus (Clemens 1961; Luyendijk 1962). The venous theory is supported by the fact that the posterior internal vertebral venous plexus shows an increased convolution of veins in the cervico-dorsal area (Clemens 1961). With regard to the level distribution in the age group between 0 and 40 years, as exemplified in Fig. 4. the largest segmental involvement is at this level (C6-D3). A simular picture is seen in the second age group (40-80 years), however in this series a second manifest peak occurs at the dorso-lumbar change-over (D10-L3). Clemens (1961) noticed a (much smaller) increase of veins in the lumbar posterior internal venous plexus, however this does explain nor the clear age differences neither the frequency peak at D 12. Therefore the local volume of the posterior internal venous plexus can not answer all the questions about the age and segmental distribution of the S S EH. As illustrated, aging plays an important part in the level distribution, concerning the involvement of the dorso-lumbar region. However, it seems very unlikely that only degenerative changes of the spinal column and/or the spinal epidural vascular structures are the explanation for this phenomenon. In our review 13 hematomas are reported to be the result of a ruptured epidural angioma resp. vascular malformation (see Table 4). According to the histological accounts in these cases, we have some marginal notes. Dawson (1963) (case 2), Foo et al. (1980) and MOiler et al. (1982) (case 1) detected structures with "intimate union of arteries and veins" in their histological preparations. Their reports of these small "vascular anomalies" made us think of the definition of the cluster type artery, as reported by Lazorthes et al. (1970). As far as we know, these structures are never mentioned in discussions about the etiology of the SSEH. Because of the unacquaintedness with these vascular clews, the cluster arteries can easily be mistaken for a "'vascular malformation" (Lazorthes et al. 1970) and erroneously considered responsible for the
133 SSEH. In some reports (Dawson (1963) (case 1), Koyama et al. (1982), Lougheed et al. (1960), Mayer (1963), Mtlller et al. (1982) (cases 2 and 3), Solero et al. (1980)) the authors concluded to a vascular anomaly after meeting with venous clusters in an epidural hematoma (Table 4). In our opinion it should be taken into consideration that in these cases the hematomas enclosed congested or distended vessels of the posterior internal vertebral venous plexus. In the literature the vertebral hemangioma, in particular the purely epidural hemangioma, is regarded as a very rare condition. Most cases of spinal epidural hemangiomas are vertebral angiomas extending into or invading the vertebral epidural space (Guthkelch 1948; Vogelsang 1970). Purely epidural hemangiomas almost always seem to occur in the thoracic spinal canal, predominantly in females (Umbach et al. 1976). The histological structure varies from cavernous hemangioma to less mature hemangio-endothelioma and hemangio-blastoma (Guthkelch 1948). The clinical signs of hemangiomas of the spinal epidural space are those of progressive paraplegia of varying rapidity of onset, but usually this developes within a few months. Some cases with temporary remission are known. Only one case of sudden and complete paraplegia was noticed and was shown at autopsy to have resulted from an epidural hemorrhage from the vessels of an epidural vascular tumour (Guthkelch 1948). Scharfetter (1972) found it remarkable that never an acute epidural hemorrhage was mentioned in publications about spinal epidural vascular malformations. With regard to the clinical history of spinal (epidural) (hem)angiomas and the doubtful results of histological examination in SSEH in this review, we question the predominant role of a vascular malformation in the pathogenesis of the SSEH. This opinion seems to be affirmed by the recent report of a SSEH in a patient with a vertebro-epidural hemangioma (postoperatively diagnosed by angiography) several segments above the actual spinal epidural hematoma (ter Spill et al. 1989).
CONCLUSIONS
(1) The anatomical dimensions of the posterior internal vertebral venous plexus and the level distribution in 199 cases of SSEH, indicate that the majority of SSEH result from a rupture of this particular vascular network. (2) The cluster type artery can easily be mistaken for a vascular anomaly, because of its remarkable morphology and the unacquaintedness of the clinician with this structure. (3) The vertebral and/or epidural hemangioma plays an insignificant role in the etiology of the SSEH. (4) The rupture of a spinal epidural artery (cluster- or spiral-type) might be responsible for some cases of SSEH. (5) Despite many advocating reports, arterial hypertension does not play a role in the pathogenesis of the SSEH. (6) The SSEH at the lower dorsal level mainly occurs after the fourth decade. Before that age, an epidural hematoma extending to or originating from the lower dorsal
134 a n d l u m b a r s e g m e n t s is a n e x c e p t i o n . A t t h i s m o m e n t t h i s o b s e r v a t i o n c a n n o t b e e x p l a i n e d in a n a t o m i c a l , f u n c t i o n a l o r p a t h o p h y s i o l o g i c a l t e r m s . T h i s wilt b e a n i t e m f o r f u r t h e r study,
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