Spontaneous spinal epidural hematoma in children: a case report and literature review

Introduction

Spontaneous spinal epidural hematoma (SSEH) in children is considered a very rare neurosurgical emergency case that can cause spinal cord compression and neurological dysfunction. The incidence is estimated to be 0.1 patients per 100,000 patients per year in the adult population, while the incidence in children is still unknown (1,2). The scarcity of incidence in the children population is indicated by few SSEH reports on children (3). This article focuses on SSEH in children aged 18 years old and below because of its rarity in the literature and its specific characteristics different from adults.

SSEH is defined as hematoma within the spinal epidural space without the underlying causes of trauma or iatrogenic. Most of the underlying causes of SSEH in children are idiopathic but sometimes, it can also be due to identifiable causes such as bleeding disorders, vascular malformations, and anticoagulant therapy (4-6). The spinal compression due to SSEH may potentially cause the spinal cord or nerve root injury causing permanent neurological dysfunction which demands prompt and proper management (2,7).

We present a case of a 14-year-old boy having cervical SSEH with a very rare symptom mimicking stroke and a literature review of 81 articles with a total of 95 reported cases of SSEH in children between January 2000 to December 2023. The search strategy is summarized in Table 1. The details of these reviewed cases are available in tabular form in the Supplementary file (Appendix 1). We provide a summary of the comparison data on sex, age, clinical presentation, etiology, location of the hematoma, treatment modalities, and outcomes (Table 2). We present this article in accordance with the CARE reporting checklist (available at https://jss.amegroups.com/article/view/10.21037/jss-24-49/rc).

Case presentation

A 14-year-old previously healthy boy came to our hospital with the complaint of acute onset of posterior neck pain during singing karaoke 2 hours prior to arriving at the emergency room. There was no history of trauma on the head or spine, anticoagulant therapy, bleeding disorders, or other systemic complaints. His pain was present from the neck radiating to the right shoulder and did not improve. Before coming to the hospital, he developed a progressive weakness on the right side of his body and was not able to stand without support.

On admission, he was alert and hemodynamically stable. There was pain in the posterior neck evaluated by the visual analog scale with 7 points of 10. Neurological examination revealed right hemiparesis with manual muscle test (MMT) of 3/5 in the right extremities whereas normal on the left extremities without sensory deficits and bladder-bowel dysfunction. Laboratory blood examinations, including a thorough evaluation of the coagulation profile, were within normal range. Urgent brain and spine magnetic resonance imaging (MRI) were performed. The brain MRI result showed neither infarction, hematoma, or dissection lesions. The spine MRI revealed a right posterolateral epidural lesion in C4–C7 levels which appeared isointense on T1-weighted images (T1WI) and hyperintense on T2-weighted images (T2WI) consistent with a hyperacute spinal epidural hematoma (SEH) that caused anterior spinal cord compression to the left (Figure 1).

Figure 1 Cervical spine MRI showing SEH (white arrows) from C4 to C7 levels. (A) T1-weighted sagittal view; the SEH appeared isointense. (B) T2-weighted sagittal view; the SEH appeared hyperintense. (C) T2-weighted axial view at the level of C5/6 foramen; the SEH located at the right posterolateral compressing anteriorly the spinal cord to the left. MRI, magnetic resonance imaging; SEH, spinal epidural hematoma.

The patient underwent an emergent open-door laminoplasty (C5–C6) with partial laminectomy (C4 and C7) and evacuation of the hematoma (Figure 2). Intraoperative findings seemed to be no tumors or vascular abnormalities. After the hematoma was completely evacuated, the dorsal spinal cord appeared pulsatile indicating adequate decompression. The follow-up cervical CT scan 7 days after surgery revealed no residual hematoma (Figure 3). Spinal angiography was also performed to exclude spinal vascular malformation and showed no abnormality. His symptoms were gradually improved postoperatively. On day 1 after surgery, MMT on the right extremities improved to 4/5. The patient had a complete recovery on day 5 after surgery with no neurological deficit.

Figure 2 Open-door laminoplasty C5–C6. (A) SEH (white arrow) is exposed on the right side of the lamina. (B) Cervical laminoplasty using the laminoplasty plates. SEH, spinal epidural hematoma.

Figure 3 Post-operative CT scan. (A) Sagittal view; showed no residual hematoma in C4–C7. (B) Axial view of C6; laminoplasty using laminoplasty plates and well-decompressed spinal canal. CT, computed tomography.

All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Helsinki Declaration (as revised in 2013). Informed consent was taken from the patient’s guardians for publication of this case report and accompanying images. A copy of the written consent is available for review by the editorial office of this journal.

Discussion

In this article, SSEH refers to all SEH that is not caused by trauma which is in accordance with most of the authors. Some authors only refer to SSEH if the cause is idiopathic or unidentifiable (8,9). Others also differentiate between spontaneous and idiopathic SEHs (10). SSEH in children is very rare but clinically very important due to its compressive effect on the spinal nerve and potential permanent neurological deficits. Kreppel et al. have reported that SEHs were the most frequent type of spinal hematoma cases (74.2%) (10).

Children with SSEH have specific characteristics that are different from adults. Younger children may have difficulty expressing their symptoms clearly compared to adults, potentially leading to delays in diagnosis (11). SSEH in children may be associated with certain underlying conditions, such as bleeding disorders or vascular malformations, which are less common in adults (6). Imaging studies such as MRI are typically used for diagnosis, but sedation or anesthesia may be required in younger children, adding complexity to the diagnostic process. Treatment of SSEH in children may require specialized care tailored to their age, size, and underlying medical conditions.

Etiology

The etiology of SSEH in children is mostly idiopathic, but there are some reports of underlying causes including hemophilia, vascular malformation, and coagulopathy. There are two well-known pathogenesis of SSEH including venous origin and arterial origin. The epidural venous plexus has a thin wall and valveless making it vulnerable to any increase of intravenous pressure. Due to its continuity with the abdominal and thoracic venous system, an increase in intraabdominal or intrathoracic pressure can also increase the intravenous pressure in the epidural venous plexus which may cause venous plexus rupture and bleeding at the posterior epidural space in most of the cases (8,12-15). Beatty (16) theorized that the free epidural artery is the source of acute SSEH in the cervical. They insisted that the arterial plexus on the epidural space is anatomically vulnerable to certain mechanical movements that may overstretch the free arteries exceeding the tolerance limitation and eventually causing arterial rupture (12,16). The possible mechanism in our case is a sudden increase of intra-abdominal or intrathoracic pressure during singing karaoke which also increases the intravenous pressure of the spinal epidural veins eventually bleeding occurs. The patient then developed a progressive hemiparesis.

Clinical presentation

Symptoms and neurological deficits of SSEH cases vary depending on several factors such as age, underlying etiology, and location of the hematoma which is the most important factor. In younger patients, the symptoms are often non-specific which makes it difficult to make the diagnosis and to identify the underlying etiology. These non-specific signs and symptoms may cause delays in the diagnosis and treatment (11). Some infant patients with SSEH had complaints of general clinical presentations such as irritability (3,17-24) and torticollis (11,18,23-25). The neurological deficit due to SSEH generally occurs within a few minutes or hours by the expanded hematoma but some can appear after a few days (26). Older children can explain their complaints which makes it easier to assess compared to infant patients.

The most common clinical presentations are weakness in the lower extremities, weakness in all four extremities, neck pain, and back pain. Some also present with uncommon clinical features such as Horner syndrome (27,28), a clinical presentation like that of a spinal birth injury (29), and hemiparesis (19,22,26) mimicking stroke like in our present case. The clinical manifestations can also mimic other medical conditions such as meningitis (3,26,30,31), Guillain Barre syndrome (18,32,33), infections (20,27), and neoplasms.

The symptom of neck pain radiating to the right shoulder in our patient is an early clinical sign of spinal or nerve root compression. MRI should be performed promptly to avoid further myelopathy, particularly among children or young adults. SSEH should always be considered in similar situations (26). Early identification of the symptoms and work-up diagnosis can prevent delays in the management of SSEH.

Diagnosis

Although SSEH is a rare diagnosis, a high index of suspicion for SSEH should always be considered in the appropriate clinical presentation. The sudden onset of neck pain or back pain and progressive neurological deficit should make SSEH one of the differential diagnoses and is an indication for an emergent spinal MRI examination (34). Prompt identification of the symptoms and rapid diagnosis evaluation are important factors in reducing the delay to surgical decompression (35). In our patient, the course of symptoms was unusual. Because of the rapid workup diagnosis and early detection of cervical SSEH, we can perform surgical decompression without any delay which also contributes to the patient’s good outcome.

MRI is considered better compared to other radiographical in revealing the location of the hematoma and the extent of spinal cord compression, as well as the consistency and stage of the hematoma (6,25,36). The hematoma in our patient appears isointense on T1WI and hyperintense on T2WI indicating a hyperacute SEH with an onset time of less than 24 hours. MRI may also show abnormally enlarged blood vessels suspected of spinal vascular malformation although MRI may not always rule out vascular malformations. Spinal angiography is an essential diagnostic tool if there is suspicion of vascular malformation or in negative cases where the MRI findings cannot reveal small vascular lesions (8,9,25,37).

Treatment

Surgical decompression is the main treatment for SSEH. The available time to restore the neurological deficit is limited, therefore early surgical decompression will give an optimal chance for good functional recovery (5,38). Laminectomy with hematoma evacuation is the most common and effective decompressive procedure in SSEH treatment, however, there are some potential problems in pediatric patients because there is a risk of progressive kyphotic deformity (6). Consequently, hemilaminectomy (6,9,13,39-49), laminotomy (29,50-53), and laminoplasty (27,33,54-59) have been used as alternative options. In our case, we performed C5–C6 open-door laminoplasty with partial laminectomy at C4 and C7. The advantage of laminoplasty is that it can preserve the major posterior structure, while still allowing for spinal canal decompression.

Conservative or non-surgical treatment of SSEH can be an option in a mild neurological deficit or bleeding disorder. However, the option for conservative treatment of SSEH may also be considered in cases that coexist with serious coagulopathy or high risk for surgical treatment and is not always based on the mild clinical course alone (6,38,60). During the conservative treatment, there is still a possibility of neurological deterioration and enlargement of the hematoma. Therefore, close neurological monitoring and repeated MR imaging are necessary (15,61).

In SSEH due to bleeding disorder (hemophilia A or B), treatment with replacement clotting factor VIII (11,19-24,28,41,49,56,62-70) or factor IX (17,71-74) has resulted in the resolution of the hematoma without surgical intervention (61). Surgical resection and endovascular embolization have been reported as the treatment of SSEH with spinal arteriovenous malformation (4,9,33,36,43,57,75,76) and spinal dural arteriovenous fistula (30,77).

Outcome

The main contributing factors to the neurological outcome following SSEH treatment are the location of the hematoma (level of vertebral segments involved), the preoperative neurological condition, and the time interval from the onset to surgical decompression. Surgery performed in less than 36 hours in patients with a complete preoperative sensorimotor deficit and less than 48 hours in patients with an incomplete preoperative sensorimotor deficit are both associated with favorable outcomes (35). In our case, the patient had undergone surgery less than 12 hours from the onset. The post-surgical outcome showed a very good result. Early decompression is important and has shown a significant improvement statistically and clinically (78). Adequate surgical decompression may reduce intradural pressure and increase the blood perfusion to the spinal cord, this will eventually reduce ischemia and prevent secondary injury from occurring (79).

Several reports suggest that when the symptoms are mild and improve spontaneously after a few hours before surgery, a favorable outcome can be achieved without surgery (18,80). In hemophilia patients with SSEH, conservative treatment with clotting factor replacement has shown good outcomes (17,19-24,28,62,65-73), instead of performing high-risk surgical treatment with abnormal coagulation status unless neurological deterioration progresses rapidly (17).

Conclusions

SSEH in children is a very rare neurosurgical emergency that can cause spinal cord compression and neurological dysfunction. Prompt and proper examination is essential to establish the diagnosis and early surgical decompression. Adequate surgical decompression may reduce intradural pressure and increase the blood perfusion to the spinal cord, thus, this will eventually reduce ischemia and prevent secondary spinal injury. As a result, complete recovery can be expected.

Acknowledgments

Funding: None.

Footnote

Reporting Checklist: The authors have completed the CARE reporting checklist. Available at https://jss.amegroups.com/article/view/10.21037/jss-24-49/rc

Peer Review File: Available at https://jss.amegroups.com/article/view/10.21037/jss-24-49/prf

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://jss.amegroups.com/article/view/10.21037/jss-24-49/coif). The authors have no conflicts of interest to declare.

Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Helsinki Declaration (as revised in 2013). Informed consent was taken from the patient’s guardians for publication of this case report and accompanying images. A copy of the written consent is available for review by the editorial office of this journal.

Open Access Statement: This is an Open Access article distributed in accordance with the Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International License (CC BY-NC-ND 4.0), which permits the non-commercial replication and distribution of the article with the strict proviso that no changes or edits are made and the original work is properly cited (including links to both the formal publication through the relevant DOI and the license). See: https://creativecommons.org/licenses/by-nc-nd/4.0/.

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