The Syndrome of Spontaneous Spinal Epidural Hematoma

Report of Three Cases

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Acute compression of the spinal cord or cauda equina caused by spontaneous epidural hematoma is relatively rare. Sadka31 reviewed 12 published cases and added two more. Schultz, et al.,32 Lougheed and Hoffman,22 and Lépoire, et al.,20 brought the total to 46 cases, to which we are now adding three cases.

Case 1. A 79-year-old white woman was admitted to the San Jose Hospital on January 28, 1961. At 4:00 a.m. that morning she had awakened with severe pain in the thoraco-lumbar region, which was followed

Acute compression of the spinal cord or cauda equina caused by spontaneous epidural hematoma is relatively rare. Sadka31 reviewed 12 published cases and added two more. Schultz, et al.,32 Lougheed and Hoffman,22 and Lépoire, et al.,20 brought the total to 46 cases, to which we are now adding three cases.

Case 1. A 79-year-old white woman was admitted to the San Jose Hospital on January 28, 1961. At 4:00 a.m. that morning she had awakened with severe pain in the thoraco-lumbar region, which was followed by bowel incontinence. A few minutes later her right leg became so weak that she could not walk. Before the sudden onset of her symptoms the patient had been active despite mild cardiac insufficiency and diabetes mellitus.

Examination. The bladder was distended to the umbilicus and catheterization was necessary. Neurological examination January 29, approximately 30 hours after onset of symptoms, revealed flaccid paralysis of both legs, with areflexia, and loss of all sensory perception below the level of T-10 bilaterally.

Spinal puncture revealed a pressure of 100 mm of water, xanthochromic fluid, and a protein content of 2,800 mg%. X-rays of the lumbar spine were normal except for osteoporosis. Myelography revealed a complete block at the level of the disc space between T-11 and T-12.

Operation. Laminectomy was performed approximately 33 hours after onset of symptoms and 12 hours after onset of paraplegia (H.N.L.). The laminae of T-8 through T-11 were resected. A thick, unencapsulated, recently-clotted mass of blood was removed from the epidural space. No abnormalities were found to account for the origin of this clot.

Postoperative course. Twenty-four hours after surgery, the patient could move both feet and the sensory level had dropped to mid-thigh bilaterally. Twelve days after surgery she could rotate the right leg medially, and on the thirteenth day she was transferred to a convalescent home. At the time of last examination, November 13, 1961, 10 months after surgery, she was walking well with the aid of a stroller, although her right leg dragged somewhat. Sensory testing was within normal limits. The right ankle jerk was decreased, but the remainder of the tendon reflexes in the legs were normal. A positive Babinski sign was present on the left. Bowel control had returned to a satisfactory degree, but a retention catheter within the bladder was still necessary.

The cause of the epidural bleeding in this case was unexplained. There was nothing to suggest that trauma was a factor, and the presence of diabetes mellitus was believed to be coincidental.

Case 2. A 45-year-old white man was admitted to the O'Connor Hospital on May 28, 1962, approximately 24 hours after falling 8 feet from a truck. He was stunned at the time, and immediately afterward complained of pain between the shoulder blades. During the next 12 hours he developed pain in both arms and weakness of the right arm, hand, and leg.

Examination. Neurological examination approximately 20 hours after injury revealed weakness of both arms and hands, which was more pronounced on the right side. Marked weakness of the right leg was noted. Pain perception was decreased below the level of T-3 on the left and C-8 on the right. Temperature sensation was decreased below T-3 on the left, while vibratory sensaation was decreased in the left leg. Tendon reflexes were increased in the right arm and leg. The Babinski sign was present on the right. Abdominal and cremasteric reflexes were absent bilaterally. Myelography was performed, revealing a partial block at the level of the disc space at C7-T1. The spinal fluid protein was 77 mg%.

The morning after hospital admission, catheterization of the bladder was necessary; 1,100 ml of urine were released. X-rays of the cervical spine were normal.

Operation. Laminectomy was performed approximately 24 hours after injury and 18 hours after the onset of motor weakness (G.E.S.). The lamina of C-7 was removed bilaterally. In the epidural space there was a large, recently-clotted mass of blood which extended anterolaterally on the right and dorsomedially on the left. It was thicker on the right. After removal of the clot with cup forceps and suction, the dura was opened. The spinal cord appeared somewhat edematous but no other abnormalities were observed.

Postoperative course. The patient's motor and sensory findings improved each day. Seven days after surgery, however, while being turned in bed, he suddenly developed dyspnea, cyanosis, and hypotension, and died a few minutes later. Autopsy revealed a massive pulmonary embolism which had originated in the right leg. The cervical cord showed no gross abnormality. Microscopic sections of the cervical cord at the level of C-7 were normal except for a small focus of gliosis in the gray substance on the right.

In this case, injury appears to have been a contributing factor to the onset of the epidural hematoma. Since there was no radiographic evidence of spinal injury and no source of the bleeding was found at surgery or autopsy, it is difficult to understand why such an unusual lesion developed despite the recent trauma. Two cases reported by Hopkins and by Shenkin15,33 were similar (see Table 1, Cases 3 and 8).

TABLE 1

Reported cases of spinal epidural hematoma

Case No.Age (yrs) SexSitePrecipitating FactorCourseSurgeryAuthor
114 FC1–C7None.6½ days of increasing paralysis.Jackson16 1869
218 FC2–C3Straining at stool.Quadriplegia in 2 hours.Bain4 1897
340 MLumbarShovelling coal.Paraplegia in ½ hour.Hopkins15 1899
435 MT6–T7Fell 10 ft.Paraplegia in 24 hrs.YesJonas17 1911
5Adult FC3-S1Fell off bicycle.Paraplegia in 5 days.Reid30 1925
632 FT10Fell down.Paraplegia in 2 days.YesHassin14 1935
742 FT2–T5Turned in bed suddenly.Paraplegia in 2 hours.YesShenkin33 1945
8 FT7–T10Infection?Paraplegia in 2 weeks.YesShenkin33 1945
975 MC5–C7Fell 4 feet on buttocks.Paraplegia 1 hour.YesVerBrugghen35 1946
1044 MCerv.Sneezing fit.Rapid paralysis.NoOldak27 1948
1139 FT5–T6Vomiting.Paraplegia in 3 hrs.YesKaplan18 1949
1243 FT9–L1None.Paraplegia in 3 days.YesKaplan18 1949
1370 FL1–L3Straining at micturition.Paraplegia in 3 hours.YesChavany7 1949
1467 ML3–L4Fell on buttocks.Partial lesion in 2 weeks.YesSvien34 1950
1573 FT8–T10Straining at micturition.Paraplegia in 1 hour.YesSadka31 1953
1670 MC7–T2None.Paraplegia in 4 days.Sadka31 1953
1724 MT2–T4Lifting.Paraplegia in 15 hrs.YesSchultz32 1953
1879 ML2–L4None.Paraplegia in few min.YesSchultz32 1953
1956 FT10–L1Bending forward.Paraplegia in 3 hours.YesSchultz32 1953
2034 MT9–L1None.Paraplegia in few hrs.YesSchultz32 1953
2157 MC1–T10Turned neck suddenly.Polycythemia vera.NoAmyes2 1955 #1
2270 MT3–T7Onset while asleep.Immediate paraplegia.YesAmyes2 1955 #2
2357 MT11–L4Walking along street.Paraplegia in 30 min.YesAmyes2 1955 #3
2415 MC6–T1None.Paraplegia in few hrs.YesNichols26 1956
254 MT2–T4Relapsing chest pain 36 hours.Sudden paraplegia.YesMaxwell & Puletti24 1957
2673 FT8–T10Getting in bed.Immediate paraplegia.YesAinslie1 1958 #1
2770 MC7While sitting in chair.Gradual quadriparesis 6 days.NoAinslie1 1958 #2
2863 FC2–C4Spontaneous onset.Quadriparesis in 2 days.YesAinslie1 1958 #3
2955 ML3–L4Carrying water jugs.Paraplegia in 24 hrs.YesPommé29 1959
30 Peserico & Svien28 1959M C5–C7Spontaneous onset.Relapsing neck pain 16 days, then sudden paralysis.YesComplete recovery.74
3171 FC5–T5Fell out of chair.Paraplegia in 4 hours.YesLowrey23 1959 #1
3223 MT11Lifting. Low back pain.Paraparesis 4 days later.YesLowrey23 1959 #2
3352 MT12–L2Pulling a chair.Sudden paralysis 1 day later.YesLowrey23 1959 #3
34 Lougheed & Hoffman22 1960 #1M T3–T6Carrying light object. Back pains 2 years.Paraplegia in 30 min.YesPartial recovery.33
3574 FT10–L1Spontaneous. Sudden backache.Gradual paralysis in 36 hours.YesLougheed & Hoffman22 1960 #2
3667 MT11–L5Asleep in bed. Sudden back pain.Paraplegia in 12 hrs.YesLougheed & Hoffman22 1960 #3
3757 MT3–T5Epigastric pain and back pain.Sudden paraplegia 56 hrs. later.YesLougheed & Hoffman22 1960 #4
3854 FT5–T10Awoke in bed with back pain.Paralysis 6 hours later.YesLougheed & Hoffman22 1960 #5
39 Lougheed & Hoffman22 1960 #6M T11–S1Sawing wood.Bachaches 5 years. Paralysis in ½ hours.YesNearly complete recovery.55
4032 MC5–T1Spontaneous.Paralysis in ½ hour.YesLepoire20 1961 #1
4119 MC6–T2Spontaneous.Paralysis in few hrs.YesLepoire20 1962 #2
4213 MC6–T1Awoke in bed with back pain.Paralysis in 2 hours.YesLepoire20 1961 #3
4333 MC7–T2Spontaneous.Intermittent paralysis.YesLepoire20 1961 #4
4453 FT11–L3Turned in bed. Back pain.Paralysis in 3 days.YesLin21 1961 (Case 2)
4529 MC6–C7None.Paraplegia in few hrs.YesCube9 1962
4626 FT2–T5Spontaneous.Paraplegia in 1 hour.YesBidzinski5 1966
4779 FT8–T10Awoke in bed with pain.Paraplegia in 28 hrs.YesMarkham #1
4845 MC6–T1Fell 8 ft.Triplegia in 24 hrs.YesMarkham #2
4956 FT9–L1Bending forward.Paraplegia in 3 hrs.YesMarkham #3

Case 3. A 56-year-old housewife was admitted to the O'Connor Hospital on July 15, 1962. Twenty-eight hours earlier she had bent over to pick up a towel and experienced severe low-back pain with radiation into both legs. A physician examined her at home 3 hours later, suspected a ruptured lumbar disc, and administered a hypodermic for pain. Shortly after his departure, she attempted to walk to the bathroom but her legs would not support her and she fell. She refused to be moved from the floor for approximately 12 hours; by then she had acute urinary retention.

The patient had had variable hypertension for several years. There was no history of previous backache.

Examination. Neurological examination on admission revealed flaccid paraplegia from the hips down, and absence of tendon reflexes in both legs. The Babinski sign was not obtained. Complete loss of sensation for touch, pain, and vibration was found below the junction of the middle and upper thirds of the thighs. X-rays of the lumbar spine were normal. The spinal fluid was clear and colorless, with normal cell count, but the protein content was 358 mg%. The manometric tests were normal. Myelography revealed a complete block at the level of L-1 (Fig. 1).

Fig. 1.
Fig. 1.

Myelogram in lateral plant. Arrow points to level of block at L-1.

Operation. On July 16, surgery was begun 33 hours after the onset of symptoms and 30 hours after the onset of motor paralysis (J.W.M.). The laminae of T-9 through T-12 were removed bilaterally. An extensive, unencapsulated mass of clotted blood was found extending from the rostral margin of L-1 to the caudal margin of T-9, occupying the dorsal aspect of the dural sac and both lateral recesses. The clot was considerably thicker along the left aspect of the dura which, in fact, was displaced somewhat to the right. The clot was thickest posterior to the body of T-10 and lateral to the dura at that level. Upon removing the clot with cup forceps, several large epidural veins were seen, and active bleeding occurred. The wound was closed without opening the dura. Microscopic examination of the surgical specimen revealed clotted erythrocytes, very few lymphocytes, scattered fibrin strands, and loose areolar tissue, but no evidence of vascular malformation or neoplasm.

Postoperative course. Six hours after surgery, the sensory level had dropped to midtibial level and the Babinski sign was present bilaterally. Two days postoperatively she was able to move both legs and had some sensory perception in both feet. Nine days postoperatively she was able to stand and walk a few steps with assistance and by the 15th day was able to walk 20 feet, although the left leg was weaker than the right. She was dismissed August 2; by then she was voiding well. At 1 year she had normal motor and sensory responses in the legs, although her gait was slightly unsteady and wide-based. Three years later she was hospitalized elsewhere for treatment of vesico-vaginal fistula which developed after radiation therapy for carcinoma of the cervix. At that time, there had been no recurrences of spinal cord symptoms.

Discussion

The 46 reported cases of spinal epidural hematoma, plus our three cases, are summarized in Table 1.

Symptoms and Signs

Of the 49 cases reviewed, 28 were male and 21 female; age varied from 1½ to 79 years. The greatest number of cases occurred at 51–60 years (Table 2).

TABLE 2

Age at time of onset

Age (yrs)No. of Cases
0–102
11–205
21–304
31–409
41–504
51–6010
61–707
71–808
49

Pain in the spinal region, motor weakness, numbness, and urinary retention were the main symptoms, and usually in that sequence. The severe pain was usually of sudden onset and initially localized at the level of the lesion; later it tended to become more extensive, but still referred to the spinal region. Radicular pain soon followed, along the distribution of the affected nerve roots. As Svien, et al.,34 have noted, the symptoms may resemble those of a ruptured intervertebral disc.

The onset of pain was usually related to seemingly insignificant effort, such as straining at stool (Case 2), micturition (Cases 13 and 15), bending forward (Cases 19 and 49), turning in bed (Cases 7 and 44), lifting (Cases 17 and 32), or shoveling coal (Case 3), and even sneezing (Case 27). The onset of symptoms during sleep also occurred in some cases (Cases 22, 36, 38, 42, and 47). Motor weakness progressed quickly, sometimes unrealized by the patient because he was soon prostrate in bed. In most cases there was profound paraparesis within 3 hours after onset of symptoms. Flaccid motor weakness and diminished tendon reflexes occurred more often than spasticity and hyperreflexia. Numbness was experienced by most patients, and sensory levels corresponding to the cord lesion were clearly definable. Urinary retention occurred in the majority of cases.

It is interesting that the diagnosis of the first reported case16 was made by Sir William Jenner, in 1869, purely on the history and physical findings.

X-ray and Spinal Fluid Studies

In the majority of cases the epidural hematoma was localized to two or three vertebral segments (Table 3). The most extensive lesion extended from C-3 to S-1 (Reid30). Radiographs of the suspected spinal level were obtained in 40 of the 49 cases (Table 4). These were reported as essentially negative in all cases except Case 20, which showed evidence of an “old” compression fracture at the level of the lesion. Myelography was performed in 33 cases; a significant lesion was demonstrated in 30 cases, while in 3 the examination was regarded as “negative.”

TABLE 3

Level of lesion

LevelNo. of Cases
Cervical8
Cervical-Thoracic9
Cervical-Sacral1
Thoracic15
Thoracic-Lumbar11
Lumbar5
49
TABLE 4

X-ray and spinal fluid studies

CaseSpinal X-rayMyelographyManometric TestCSF Protein (mg%)
1
2
3
4
5
6NegativePositive
7NegativePositivePositive
8NegativePositivePositive
9Negative
10????
11NegativePartial180
12NegativePositive
13NegativePositivePartial22
14NegativePositive
15NegativeNegativeNegative200
16NegativeNegativeNegative172
17NegativePositivePositive50
18NegativePositiveNegative194
19NegativePositive
20Old frac.Negative196
21NegativePositive
22NegativePositivePositive238
23NegativePositive
24NegativePositiveNegative28
25NegativePositiveNegative136
26NegativePositiveNegative200
27NegativePositiveNegative172
28NegativePositive178
29PositivePositive1,000
30NegativePositivePositive195
31Negative41
32NegativePositivePositive425
33NegativePositivePositive122
34NegativePositive
35Positive
36NegativePositive
37NegativePositiveNegative73
38NegativeNegative108
39Positive
40NegativePositivePositive230
41NegativePositive
42NegativePositivePositive
43NegativePositivePositive
44NegativePositive61
45NegativePositive42
46NegativePositiveElevated
47NegativePositive2,800
48NegativePositive77
49NegativePositiveNegative358

The manometric test was reported in 29 of the 49 cases. It revealed a block in 17 cases, partial block in 2 cases, and was negative in 10 cases. Cerebrospinal fluid protein, reported in 27 of the 49 cases, varied from 22 mg% to 2,800 mg%; in 23 cases, it was over 45 mg% and in 4 cases it was under 45 mg%.

Although x-rays of the spine at the suspected level are normal in nearly all cases, they are useful in excluding bony abnormalities from the differential diagnosis. Elevation of spinal fluid protein may be expected, but normal values do not exclude the possibility of spinal epidural hematoma. We believe myelography should be done in cases of suspected intraspinal bleeding to determine the presence or absence of a surgical lesion. Cube9 stated that myelography may be hazardous in such cases, but we do not share this opinion. To rely on history and neurological examination only may lead to a negative surgical exploration, because there are other conditions that may resemble epidural spinal hematoma.

Therapy

Eight cases received no surgical treatment; all died within 9 days. Laminectomy was performed in 41 cases; 10 of these patients died within 5 months. Of the 33 cases who survived at least 5 weeks following laminectomy, 16 were restored to normal or were greatly improved. A moderate permanent disability occurred in 9 cases, while in 6 others there was no appreciable improvement.

Etiology

In 44 of the 49 cases, there was no significant trauma. In the remaining five cases (Cases 4, 5, 9, 14, and 48) trauma appears to have been a contributory but not a primary cause (Table 5).

TABLE 5

Role of trauma

Degree of TraumaNo. of Cases
Significant5
Minimal19
No trauma known25
49

Cube9 stated that in 13 of the 19 cases of spinal epidural hemorrhage which he reviewed the bleeding arose from an epidural hemangioma. This statement cannot be corroborated since our review of these cases revealed but one case in which there was a verified epidural hemangioma, namely, that reported by Cube. However, this prompted us to review some related references on spinal epidural hemangiomas;6,11–14,19,25 we found no mention of epidural hematoma caused by such a lesion.

Anticoagulant therapy has been reported as an etiological factor in some cases of spontaneous spinal epidural hematoma.2,8,10,21,36 We excluded this group of cases from our study.

Summary

We have described three cases of spontaneous spinal epidural hematoma with cord compression and reviewed 46 similar cases. We believe that this condition is a distinct clinical entity which should be considered in the differential diagnosis of acute spinal cord syndromes or ruptured intervertebral discs.

References

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Presented at the Second Annual Meeting of the Federation of Western Societies of Neurological Science, San Francisco, California, March 3, 1966.

Article Information

Clinical Assistant Professor of Neurological Surgery, Stanford University, Palo Alto, California.

© AANS, except where prohibited by US copyright law.

Headings

Figures

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    Myelogram in lateral plant. Arrow points to level of block at L-1.

References

1.

AinslieJ. P. Paraplegia due to spontaneous extradural or subdural haemorrhage. Br. J. Surg. 195845:565567.AinslieBr. J. Surg.45:565–567.

2.

AldermanD. B. Extradural spinal-cord hematoma. Report of a case due to dicumarol and review of the literature. New Engl. J. Med.1956255:839842.AldermanNew Engl. J. Med.255: 839–842.

3.

AmyesE. W.VogelP. J.RaneyR. B. Spinal cord compression due to spontaneous epidural hemorrhage; report of 3 cases. Bull. Los Ang. neurol. Soc.195520:18.AmyesVogelRaneyBull. Los Ang. neurol. Soc.20:1–8.

4.

BainW. A case of haematorrachis. Br. med. J.18972:455.BainBr. med. J.2:455.

5.

BidzinskiJ. Spontaneous spinal epidural hematoma during pregnancy. Case report. J. Neurosurg.196624:1017.BidzinskiJ. Neurosurg.24:1017.

6.

BrionS.NetskyM. G.ZimmermanH. Vascular malformations of the spinal cord. A. M. A. Archs Neurol. Psychiatry195268:339361.BrionNetskyZimmermanA. M. A. Archs Neurol. Psychiatry68:339–361.

7.

ChavanyJ. A.TaptasJ. N.PeckerJ. A propos d'une variété exceptionnelle d'hématorachis; l'hématome épidural spontané.Presse méd.194957:869871.ChavanyTaptasPeckerPresse méd.57:869–871.

8.

ClowardR. B.YuhlE. T. Spontaneous intraspinal hemorrhage and paraplegia complicating dicumarol therapy. 19555:600602.ClowardYuhl5:600–602.

9.

CubeH. M. Spinal extradural hemorrhage. J. Neurosurg.196219:171172.CubeJ. Neurosurg.19:171–172.

10.

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11.

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