2025 Volume 12 Pages 215-219
Spontaneous spinal epidural hematoma is a clinical entity as a hematoma occurring in the spinal epidural space with unknown etiology. It is known to sometimes show spontaneous regression, and recurrence is rare and repeating even rarer. We had an experience of repeated recurrent spontaneous spinal epidural hematoma, eventually leading to surgery.
A 25-year-old man, a habitual exerciser of strength training, had a sudden onset of cervical back pain during sleep the same night after training. Radiological examinations revealed a cervical epidural hematoma but no vascular anomalies, and the patient's symptoms and the hematoma resolved rapidly and spontaneously in a day. Within 2.5 years, he had another three more similar hemorrhagic episodes with rapid and spontaneous resolution. On the 5th episode, the patient underwent surgical treatment with removal of the epidural tissue and packing of the epidural space. The pathological diagnosis was only the normal connective tissue and veins. He has had no recurrence in the 1.5 years after surgery with the same strength training as before.
The appropriate timing and method of treatment for recurrent spontaneous spinal epidural hematoma with rapid resolution is still under debate, but surgery should be considered if there are multiple recurrences. It is important to collapse the epidural space with packing to prevent further recurrence.
Spontaneous spinal epidural hematoma (SSEH) is a rare clinical entity as a hematoma occurring in the spinal, usually cervico-thoracic, epidural space without a traumatic or postoperative history, presenting with acute neck and back pain, sometimes with radicular symptoms.1-4) Although several factors have been reported to cause SSEH, including the use of anticoagulation or antiplatelet agents, coagulopathies, and vascular anomalies such as arteriovenous malformations (AVMs) or angiomas, it still seems difficult to identify a specific etiology in 40%-60% of SSEH cases.1,3,5) Some authors have recommended urgent surgical treatment of SSEH, similar to traumatic or postoperative epidural hematoma,4-6) but others have reported spontaneous resolution of SSEH within a few days without surgery.7-11) Recurrence of SSEH after spontaneous resolution is even rarer, and the underlying mechanism and indications for invasive treatment remain unclear.12-15) We report here a unique experience of repeated recurrence and rapid resolution of SSEH, ultimately leading to surgery, in the case of a habitual exerciser of intense strength training including repetitive heavy-weight barbell lifts and squats for bodybuilding purposes.
A 25-year-old man was admitted to our hospital by ambulance with a sudden onset of cervical back pain during sleep and with motor weakness and sensory disturbance in his left upper extremity. He had no hematologic diseases and had never taken anticoagulants or antiplatelet agents. He had no history of trauma or spinal surgery before admission, but was a habitual and hard exerciser, doing intense strength training and his routine training in the evening of the day. Magnetic resonance imaging (MRI) on admission showed a relatively left-sided epidural hematoma at levels of C6-7 with right antero-lateral spinal cord compression (Fig. 1A-C). Under careful observation, the patient's symptoms continued to improve and MRI in the evening of the same day showed almost complete resolution of the hematoma and slightly thickening of the dura mater (Fig. 1D). After spontaneous resolution of the hematoma, further evaluation including heavily T2-weighted MRI, contrast-enhanced computed tomography, and angiography, was performed to identify the source of the hemorrhage. We found only one finding suspecting a dilated venous-like structure in the left epidural space at the level of C6 (Fig. 2), but we could not rule it out as a definite abnormal finding. No AVM or other obvious vascular abnormality was found. He was discharged on the 8th day after admission with no neurological symptoms. At 16 months after the first episode, he was admitted to our hospital again by ambulance in the middle of the night with severe pain in his left upper limb and neck, similar symptoms to the previous episode. MRI also showed similar findings to the ones at the first admission. The day after admission, the symptoms had completely improved and the hematoma on MRI had disappeared, and he was discharged without any surgical treatment again. Although he had significantly reduced the intensity of his habitual strength training in response to our suggestion, he had two further episodes with similar clinical and radiological courses of SSEH within 16 months of the second episode. We performed another selective cervical DSA under careful review with a slow manual injection of contrast medium, but no abnormal blood vessels could be found. On the night of the fourth discharge with complete resolution of clinical symptoms and radiological hematoma, he had the same clinical symptoms of sudden neck pain and left motor weakness. Although the symptoms were mild on arrival and no hematoma was seen on imaging, the symptoms were obvious and we considered this to be the fifth episode of very short-term bleeding and resorption.
Magnetic resonance imaging (MRI) in the first episode of epidural hematoma.
Sagittal T1-weighted and T2-weighted images (A and B) show an epidural hematoma at levels of C6-7, and an axial T2-weighted image (C) at level C6 shows compression of the spinal cord to the right anterolaterally by a hematoma. Half a day later, the sagittal T2-weighted image (D) shows resolution of the hematoma.
Investigation into the cause of the spontaneous epidural hematoma after spontaneous resolution of the hematoma. A thin-slice coronal heavily T2-weighted image (A) shows the enlarged vascular-like structure (white arrow) in the left epidural space, and the axial and reconstructed coronal contrast-enhanced CT images (B and C) also show the slightly enhanced structure (red arrow) in the left epidural space at the level of C6.
CT: computed tomography
We decided to perform direct observation surgery with epidural space packing to prevent further recurrence. A left unilateral laminectomy of C6 with the lower part of C5 and the upper part of C7 was performed. Observation of the epidural space revealed enlarged venous structures shown in the previous evaluation in addition to the fatty tissue and yellow ligament, but no angiomas or AVMs (Fig. 3A). We removed these tissues with coagulation of large veins, after confirming them as true venous structures with indocyanine green fluorescence (Fig. 3B). After clearly exposing the dural surfaces, we tightly packed the epidural spaces with cellulose cotton and fibrin glue (Fig. 3C). The pathological diagnosis of the removed tissue showed only normal fatty tissues with a normal venous structure. The patient was discharged with no neurological deficits and has not experienced any recurrence in the 1.5 years since surgery while performing the same strength training as before.
Operative views after the left unilateral laminectomy at level of C6.
The enlarged venous structure is observed in the left lateral epidural space (A), and it is confirmed by indocyanine green fluorescence angiography (B). After coagulation of the vein and cleaning of the epidural tissues, the epidural space was packed with cellulose cotton and fibrin glue (C).
Our case is so unique in the clinical course of repeated recurrent SSEH with rapid improvement of symptoms and the radiological findings with only conservative treatment, eventually leading to only five events in 3 years. Despite persistent evaluation, including MRI with different sequences and selective spinal angiography, we could not identify a definitive etiology to cause SSEH and therefore could not perform invasive surgical treatment. The intervals between events became shorter and shorter, with the last event occurring within a few days of the previous one and having resolved on MRI on arrival, although the patient had obvious symptoms. Therefore, we had to recognize that the cervical epidural condition was so unstable that it could easily bleed out. We then planned an operation to prevent further bleeding.
The location of recurrent SSEH, in this case, was similar in all events and was observed in the posterolateral part of the cervical region extending over two to three segments, which has been reported as the most common location of SSEH.1,3-5,16) Intraoperative observation of the epidural space revealed abundant enlarged venous structures than usually observed, but the pathological examination of the removed tissue did not reveal any abnormal vascular lesions such as AVMs or others, only normal venous structures. From an anatomical point of view, it is well known that the posterior epidural venous plexus contains thin-walled, valveless veins that are directly affected by fluctuations in abdominal and thoracic pressure, resulting in a collapse and causing SSEH.4,16) There has been interest in the increased venous pressure associated with coughing, sneezing, and straining as a possible trigger for SSEH.17,18) Several authors have reported the unique performances that lead to a positive venous pressure for the presentation of SSEH in normal healthy patients, such as fast bowling while playing cricket,19) lifting heavy pipes in a manufacturing plant,20) making two thousand traditional Korean deep bows at a funeral,21) or professional diving with the pressure changes in the underwater environment.22) The present patient was also a habitual and hard exerciser of intense strength training, although not a professional. He usually performed lifting heavy-weight barbells, squatting while carrying a barbell, and other practices for 1-2 hrs. The Valsalva maneuver may have been exacerbated by improper breathing during these activities, resulting in increased abdominal and thoracic pressure. The fact that the hematoma developed a little later rather than during or immediately after such exercise also suggests that SSEH is caused by the collapse of the congested veins. In addition, the abnormal flexion and rotation of the cervical spine may have contributed to the rupture of the venous plexus.
The mechanism of rapid and spontaneous resolution of SSEH is not fully understood.7-11) A previous author has suggested that a hematoma component may be easily reabsorbed into the epidural fatty areolar tissue, which contains a rich vascular network and may even drain through the intervertebral foramen.14) In our case, even after multiple bleeding events, we could not find any findings such as hemosiderin deposition or post-bleeding degeneration in the epidural space observed during surgery. Therefore, we speculate that the venous plexus of the region has become so fragile after the first and second hemorrhage, leading to further repetition of events and that the hematoma is unlikely to be coagulated and contains almost exclusively a fluid component. In such a condition, the higher pressure in the intrathecal space than in the epidural space may also contribute to the rapid disappearance of the hematoma. In addition, the thickening of the dura mater on MRI after the resolution of the hematoma may be related to a mechanism of spontaneous resolution.
After experiencing complete spontaneous resolution of SSEH, the timing of the surgical decision was very difficult even for repeated recurrent SSEH. There have been rare case reports of recurrent SSEH after spontaneous resolution.12-15) However, in such reports, there were a few cases in which major neurological symptoms remained even after surgical decompression of laminectomies and hematoma evacuation after recurrent bleeding with presenting severe symptoms.13) In the present case, the SSEH resolved rapidly after the fifth episode of hemorrhage, but we decided that it was no longer dangerous to treat conservatively and then planned to observe the relevant epidural space directly, including the questionable abnormal vascular findings sometimes seen on MRI. We also wondered about the appropriate surgical procedure and finally performed a unilateral laminectomy on the hematoma side only. Although there were no hematomas to be removed on careful observation, we decided that it was important to remove the structures in the space clearly and to completely collapse the epidural space by packing with cellulose cotton and fibrin glue. Finally, we would like to emphasize here that the real key point of the surgical procedure to prevent recurrent SSEH should not be how to remove the hematoma, but how to collapse the epidural space in which hematoma can easily accumulate.
ConclusionsWe had a rare experience of repeated recurrence of SSEH in a case of habitual strength training. The cause of recurrent SSEH in this case may have been the fragile venous structures in the epidural tissue and the increasing venous pressure associated with hard strength training. Appropriate management of recurrent SSEH with rapid resolution is difficult, but surgery should be considered if bleeding recurred several times. It is important to collapse the epidural space with packing to prevent further recurrence.
Informed consent was obtained from the present patient.
There are no conflicts of interest.
