- Email: [email protected]
Spinal Epidural Hematoma Associated with Epidural Metastasis After Minor Trauma
Journal Pre-proof Spinal epidural hematoma associated with epidural metastasis after minor trauma: a case report Ryuya Maejima, MD, Masahiro Aoyama, MD, PhD, Masahito Hara, MD, PhD, Shigeru Miyachi, MD, PhD PII:
S1878-8750(19)32551-3
DOI:
https://doi.org/10.1016/j.wneu.2019.09.110
Reference:
WNEU 13409
To appear in:
World Neurosurgery
Received Date: 24 July 2019 Revised Date:
22 September 2019
Accepted Date: 23 September 2019
Please cite this article as: Maejima R, Aoyama M, Hara M, Miyachi S, Spinal epidural hematoma associated with epidural metastasis after minor trauma: a case report World Neurosurgery (2019), doi: https://doi.org/10.1016/j.wneu.2019.09.110. This is a PDF file of an article that has undergone enhancements after acceptance, such as the addition of a cover page and metadata, and formatting for readability, but it is not yet the definitive version of record. This version will undergo additional copyediting, typesetting and review before it is published in its final form, but we are providing this version to give early visibility of the article. Please note that, during the production process, errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain. © 2019 Elsevier Inc. All rights reserved.
Maejima et al.
Spinal epidural hematoma associated with epidural metastasis after minor trauma: a case report
Spinal epidural hematoma with epidural metastasis
Ryuya Maejima, MD,1 Masahiro Aoyama, MD, PhD,1,2 Masahito Hara, MD, PhD,2 Shigeru Miyachi, MD, PhD1
1
Department of Neurosurgery, Aichi Medical University, 1-1 Yazakokarimata, Nagakute,
Aichi, 480-1195, Japan 2
Spine Center, Aichi Medical University, 1-1 Yazakokarimata, Nagakute, Aichi,
480-1195, Japan
Corresponding author: Ryuya Maejima Department of Neurosurgery, Aichi Medical University, 1-1 Yazakokarimata, Nagakute, Aichi, 480-1195, Japan Tel: +81-561-62-3311, Fax: +81-561-62-2879 Email: [email protected]
Key words: epidural metastasis, small cell carcinoma, spinal epidural hematoma, spinal surgery
1
Maejima et al.
Introduction Spinal epidural hematoma (SEH) rarely occurs, with an incidence estimated to be 0.1 patients per 100,000 patients per year.1 Spinal cord compression due to mass effect results in sudden pain, progressive movement disorder and sensory disturbance according to the lesion size and location.1,2,3 Most usually occur without the presence of any known etiological factors, such as trauma, vascular pathology, anticoagulation, or a tumor.2,4 The classical clinical features are symptoms indicating rapidly evolving nerve root and/or spinal cord compression. Epidural metastatic tumors are a hemorrhagic factor and far more likely than primary spinal neoplasms to cause spinal compression; however, the mass is unusually located in the epidural space itself and not in the vertebral column.5 Here, we present a rare case of cervical epidural hematoma related to epidural metastasis after minor trauma.
Case description History and Examination A 76-year-old man who had untreated Stage III rectal cancer and severe chronic obstructive pulmonary disease (COPD) presented with mild left hemiparesis after minor trauma (falling on his buttocks). His neck was not directly injured. The next day, his symptoms progressively worsened. He consulted a local doctor, and magnetic resonance imaging (MRI) showed a suspected epidural hematoma at the level of C3-4. Therefore, he was referred and transferred to our hospital for treatment. Upon arrival in our hospital, he suffered from complete quadriplegia and severe disturbance of sensation. He did not have posterior cervical pain. A physical examination showed normal anal tonus and no bladder and rectal disturbance. A cervical MRI performed in our hospital 1
Maejima et al.
showed lesions with a size of 1.8*0.5*1.1 cm extending from the C3 to C4 level in the left posterior epidural space; the lesion was high on T2-weighted imaging, low with a high portion inside on T1-weighted imaging and high with a low portion inside on short-T1 inversion recovery (STIR). The spinal cord was compressed, and T2-weighted imaging showed mild hyperintense signal (Fig. 1). T2 imaging also showed high intensity in the left C3-5 posterior supporting tissues. The vertebrae themselves showed normal findings. Computed tomography (CT) showed a high-density epidural mass that was mildly enhanced (Fig. 2). In addition, connective tissue associated with the interlamina and interspinous processes at C3/4 and C/5 and the sequential posterior cervical muscles adjacent to the C5 vertebra were also enhanced. The epidural mass was suspected to be a hematoma because his symptoms had rapidly evolved, and we considered the possibility of tumors. We checked the patient’s blood test, which did not show thrombocytopenia or coagulation disorder. Removal of the epidural mass with C3-5 laminectomy was performed for decompression of the spinal cord and to achieve a definite diagnosis.
Operation — A C3-5 laminectomy was performed to sufficiently expose the hematoma. A skin incision was made, and the muscles were divided at the midline. The posterior supporting tissue showed no abnormality. After the C3-5 lamina was removed in an unblock fashion, the hematoma was confirmed and removed by irrigation and suction. In the left epidural space, venous bleeding continued. At that point, a hemorrhagic lesion was found to be stuck to the dura and had the appearance of a yellow ligament (Fig. 3). It was visually indistinguishable from a yellow ligament, but its strong 2
Maejima et al.
adhesion to the dura matter and hemorrhagic region were abnormal. It was coagulated and partially resected for pathological diagnosis. Similar tissue was confirmed on the right side and collected for analysis. Expanded duraplasty was not performed because the tension of the dura matter was not severely affected after the removal of the lesion.
Pathological findings — Frozen and permanent sections of the left lesion were analyzed by hematoxylin and eosin staining and showed atypical cells with scant cytoplasm and a high N/C ratio (Fig. 4). The sample contained collagen fibers. The cell nuclei were dyed by chromatin. In addition, immunostaining revealed atypical cells with AE1/AE3 (+, dot-like), CD56 (+), chromogranin A(-), synaptophysin (+), TTF-1 (+), NKX3.1 (-), and CD20 (-) and that 90% expressed Ki-67. The pathologist made a diagnosis of metastatic carcinoma (small cell carcinoma). The specimen resected on the right side was connective tissue and was not malignant. The sample from the right lesion also showed no malignancy.
Postoperative course — Postoperatively, the patient’s neurological condition improved a little. Manual muscle testing (MMT) of the left upper limb was 2/5, MMT of the other limb was 3/5, and he recovered from paresthesia. MRI showed that the lesion had disappeared but that an intramedullary high-intensity signal at the C4 level remained (Fig. 5). However, his pulmonary status was severe. Because he suffered from pneumothorax, pneumonia, COPD and fatigue of the respiratory muscle, he could not be weaned off the ventilator. After he was diagnosed with small cell carcinoma, a whole body CT was performed for the detection of primary lesions and showed nodules at the hilum and pleura of the right 3
Maejima et al.
lung. Therefore, he was diagnosed with Stage IV lung small cell carcinoma. Enhanced MRI did not reveal any mass in the epidural space. Based on this performance status, pulmonologists assessed that active treatment was not applicable to this patient. During preparation for a hospital transfer to palliative care, his respiratory condition worsened, and he expired at postoperative 4 weeks. An autopsy was not performed on this patient because of his family’s will.
Discussion — To our knowledge, this report is the first case of SEH related to epidural metastatic tumors caused by minor trauma. Spinal epidural hematoma is a rarely occurring condition. Approximately 40% of cases with SEH have no definite etiological factors.2 Others have been shown to result from vascular malformation, anticoagulant therapy, severe trauma or in rare cases, tumors.3,4,6 SEH classically shows rapidly progressive symptoms that evolve into nerve root and/or spinal cord compression followed by acute onset of severe and radiating pain.1,2,3 MRI is the diagnostical tool of choice for the detection of spinal epidural hematomas. T1-weigted images of the pathognomonic region will appear isointense with the cord, and T2-weighted images will show hyperdensity in the periphery with a central hypodense area in the early period.1,2 In this case, we first suspected hematoma because of MRI findings and his symptoms. On the other hand, images showed not only epidural mass but also connective tissue and muscle, both of which were enhanced and suggested other causes, such as neoplasms. We propose that findings related to the extra-spinal canal tend to be overlooked. In cases of SEH with unknown causes, sometimes an analysis of this region can help to make a diagnosis, and enhanced CT or MRI is important. 4
Maejima et al.
The bleeding source for SEH is usually a vein. Because its wall is fragile and has no valves, the spinal venous system is weak when exposed to stress, such as high blood pressure, increased intrathoracic and abdominal pressure caused by sneezing, bearing down and falling or other minor trauma.10,11 In addition, hematogenous metastases to the spine are common and believed to be delivered by a venous route. Primary tumors predominantly metastasize to the breast, lung and prostate, and this tendency has been attributed to the finding that metastases from these tumors spread through the vertebral venous system, widely known as Baston’s plexus.12 Our case has limited value because there was no follow up or autopsy. It is difficult to say whether the patient’s life was further shortened by the epidural hematoma and paralysis. However, enough factors were evaluated to support an important hypothesis based on clinical features, images, and intraoperative and pathological findings. We hypothesize that in our case, the spinal venous system that had obtained hematogenous metastasis became fragile and was bleeding because of acute elevated venous pressure caused by the patient falling on his behind. In a previous similar case, the author estimated that the bleeding was due to the fragility of the epidural venous plexuses.13 — Kim RY reported that 5% of all patients with systemic cancer who are autopsied have pathologic evidence of a tumor invading the extradural space.9 Spinal cord compression caused by epidural metastatic tumors leads to progressive neurological deficits. Epidural metastasis commonly arises from the extension of metastases located in the adjacent vertebral column in the paravertebral spaces through the intervertebral foramina or, rarely, into the epidural space itself.5 In our case, the lesion was located in the epidural space. In addition, neurological images showed abnormal signals in the posterior connective tissue and muscle as well as the interlamina and interspinous processes, 5
Maejima et al.
which are considered to be associated with epidural tumor and neoplasm seeding. In the past, Lin HS reported a similar case which did not have connective tissue lesions but did have lesions in the vertebral bodies and lamina.13 Here, we suggest that the spinal metastatic lesions were quite unlikely to be present only in the epidural space; other lesions have been recognized in peripheral structures, including the vertebral bodies, laminae and connective tissue. In cases of epidural hematoma with abnormal signs on MRI and CT in peripheral structures, hemorrhage-associated metastasis should be considered. — In our case, emergency surgical treatment was chosen because of the rapidly progressive course and the need for a diagnosis. Treatment for epidural hematoma is generally conservative therapy, medication and surgery. Surgical treatment is needed in cases with severe symptoms.7,8 Moreover, direct surgery is helpful for diagnosis. In this case, no obvious massive lesion was confirmed, but the lesion did look like a yellow ligament with an indistinct border. The pathological diagnosis revealed that a similar contralateral mass was a normal ligament. No obvious masses were identified in the posterior cervical muscles or interlaminar and interspinous process connective tissue. The only operative characteristic was strong adhesion. However, the pathological sample showed tumor cells throughout. We hypothesized that this malignancy was so invasive that it had spread to a variety of tissues. Enhanced lesions of not only the spinal canal but also the surrounding tissue related to the spinal canal showed the possibility of infiltrating cancer. Attention was needed during surgery because the lesion did not have a massive form or unique color.
6
Maejima et al.
Conclusion This is the first report of an SEH related to epidural metastasis after minor trauma. In particular, metastatic lesions located in the epidural space itself are so uncommon that it is usually difficult to distinguish them from hematomas based on neurological images alone. In the case of epidural hematoma, especially in cancer-carrying or elderly patients, a hemorrhage originating in the metastasis should be kept in mind during the differential diagnosis.
Declaration of interest None. All authors who are members of the Japan Neurosurgical Society (JNS) have registered online Self-reported COI disclosure statement forms through the website for JNS members.
Funding sources None.
7
Maejima et al.
References 1) Holtas S, Heiling M, Lonntoft M. Spontaneous spinal epidural hematoma: findings at MR imaging and clinical correlation. Radiology. 1996;199:409 413 2) Kreppel D, Antoniadis G, Seeling W: Spinal hematoma: a literature survey with meta-analysis of 613 patients. Neurosurg Rev. 2003;26:1-49 3) Groen RJ, van Alphen HA: Operative treatment of spontaneous spinal epidural hematomas: a study of the factors determining postoperative outcome. Neurosurgery. 1996;39:494 509 4) Cetinalp NE, Oktay K, Ozsoy KM: Spontaneous spinal epidural hematoma mimicking a cerebrovascular disease. Neurol India. 2017;65:1434-1435 5) Byrne TN: Spinal cord compression from epidural metastases. N Engl J Med. 1992;27:614 619 6) Harris DJ, Fomasier VL, Livingston KE: Hemangiopericytoma of the spinal canal. Report of three cases. J Neurosurg. 1978;49:914-920 7) Alexiadou-Rudolf C, Ernestus RI, Nanassis K, Lanfermann H, Klug N: Acute nontraumatic spinal epidural hematomas. An important differential diagnosis in spinal emergencies. Spine. 1998;23:1810-1813 8) Liao CC, Lee ST, Hsu WC, Chen LR, Lui TN, Lee SC: Experience in the surgical management of spontaneous spinal epidural hematoma. J Neurosurg. 2004;100:38-45 9) Kim RY: Extradural spinal cord compression from metastatic tumor. Ala Med. 1990;60:10-15
8
Maejima et al.
10) Groen RJ, Ponssen H: The spontaneous spinal epidural hematoma. A study of the etiology. J Neurol Sci. 1990;98:121-138 11) Kaplan LI, Denker PG: Acute epidural epidural hemorrhage. Am J Surg. 1949;78:356-361 12) Baston OV: The function of the vertebral veins and their role in the spread of metastases. Ann Surg. 1940;112:138-149 13) Lin HS, Chen SJ: Metastatic carcinoma related long segment thoracic spinal epidural hematoma: a case report. Spine. 2009;34:266-268
9
Maejima et al.
Figure legends Figure 1. Preoperative magnetic resonance images (MRI): sagittal view showing the lesion extending from the C3 to C4 level (T2 weighted image: A, T1 weighted image: B, short-T1 inversion recovery (STIR): C). The lesion was located at the left posterior epidural space (axial view on T2-weighted image). T2 axial imaging also showed high intensity in the left C3-5 posterior supporting tissues.
Figure 2. Enhanced computed tomography (sagittal view: A, axial view: B) showing a high-density epidural mass. Connective tissue of the interlamina and interspinous processes at the C3/4 and C/5 levels and sequential posterior cervical muscles adjacent to the C5 vertebra were also enhanced.
Figure 3. Intraoperative view showing hemorrhagic lesions adhered to the dura. A black arrow indicates the dura mater, and a white arrow indicates lesions.
Figure 4. Histopathological examination a frozen and permanent section stained by hematoxylin and eosin to show atypical cells (low power field:A) . The cells showed scant cytoplasm and had a high N/C ratio (high power field: B).
Figure 5. Preoperative magnetic resonance image (T2-weighted image): the lesion had disappeared, but an intramedullary high-intensity signal at the C4 level remained.
10
Abbreviation list COPD, chronic obstructive pulmonary disease; CT, computed tomography; MRI, magnetic resonance imaging; MMT, manual muscle testing; SEH, spinal epidural hematoma
1
Disclosure-Conflict of Interest
Type of article: Case report
Title of the article: Spinal epidural hematoma associated with epidural metastasis after minor trauma: a case report
Contributor 1.
Ryuya Maejima (1)
2.
Masahiro Aoyama (1) (2)
3.
Masahito Hara (2)
(1) Department of Neurosurgery, Aichi Medical University, 1-1 Yazakokarimata, Nagakute, Aichi, 480-1195, Japan (2) Spine Center, Aichi Medical University, 1-1 Yazakokarimata, Nagakute, Aichi, 4801195, Japan
Corresponding Author: RYUYA MAEJIMA, MD.
Source(s) of support: None
Presentation at a meeting: No
Conflicting Interest: None
Acknowledgement:
No employees from any sponsoring agencies or industries contributed to this work. The corresponding author obtained written permission from all contributors prior to including their names in this work. The author contributions are as follows. Conception and design: R. Maejima. Acquisition of clinical data: all authors. Drafting the article: R. Maejima. Approved the final version of the manuscript on behalf of all authors: M. Aoyama. Supervision: M. Hara.
S1878-8750(19)32551-3
DOI:
https://doi.org/10.1016/j.wneu.2019.09.110
Reference:
WNEU 13409
To appear in:
World Neurosurgery
Received Date: 24 July 2019 Revised Date:
22 September 2019
Accepted Date: 23 September 2019
Please cite this article as: Maejima R, Aoyama M, Hara M, Miyachi S, Spinal epidural hematoma associated with epidural metastasis after minor trauma: a case report World Neurosurgery (2019), doi: https://doi.org/10.1016/j.wneu.2019.09.110. This is a PDF file of an article that has undergone enhancements after acceptance, such as the addition of a cover page and metadata, and formatting for readability, but it is not yet the definitive version of record. This version will undergo additional copyediting, typesetting and review before it is published in its final form, but we are providing this version to give early visibility of the article. Please note that, during the production process, errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain. © 2019 Elsevier Inc. All rights reserved.
Maejima et al.
Spinal epidural hematoma associated with epidural metastasis after minor trauma: a case report
Spinal epidural hematoma with epidural metastasis
Ryuya Maejima, MD,1 Masahiro Aoyama, MD, PhD,1,2 Masahito Hara, MD, PhD,2 Shigeru Miyachi, MD, PhD1
1
Department of Neurosurgery, Aichi Medical University, 1-1 Yazakokarimata, Nagakute,
Aichi, 480-1195, Japan 2
Spine Center, Aichi Medical University, 1-1 Yazakokarimata, Nagakute, Aichi,
480-1195, Japan
Corresponding author: Ryuya Maejima Department of Neurosurgery, Aichi Medical University, 1-1 Yazakokarimata, Nagakute, Aichi, 480-1195, Japan Tel: +81-561-62-3311, Fax: +81-561-62-2879 Email: [email protected]
Key words: epidural metastasis, small cell carcinoma, spinal epidural hematoma, spinal surgery
1
Maejima et al.
Introduction Spinal epidural hematoma (SEH) rarely occurs, with an incidence estimated to be 0.1 patients per 100,000 patients per year.1 Spinal cord compression due to mass effect results in sudden pain, progressive movement disorder and sensory disturbance according to the lesion size and location.1,2,3 Most usually occur without the presence of any known etiological factors, such as trauma, vascular pathology, anticoagulation, or a tumor.2,4 The classical clinical features are symptoms indicating rapidly evolving nerve root and/or spinal cord compression. Epidural metastatic tumors are a hemorrhagic factor and far more likely than primary spinal neoplasms to cause spinal compression; however, the mass is unusually located in the epidural space itself and not in the vertebral column.5 Here, we present a rare case of cervical epidural hematoma related to epidural metastasis after minor trauma.
Case description History and Examination A 76-year-old man who had untreated Stage III rectal cancer and severe chronic obstructive pulmonary disease (COPD) presented with mild left hemiparesis after minor trauma (falling on his buttocks). His neck was not directly injured. The next day, his symptoms progressively worsened. He consulted a local doctor, and magnetic resonance imaging (MRI) showed a suspected epidural hematoma at the level of C3-4. Therefore, he was referred and transferred to our hospital for treatment. Upon arrival in our hospital, he suffered from complete quadriplegia and severe disturbance of sensation. He did not have posterior cervical pain. A physical examination showed normal anal tonus and no bladder and rectal disturbance. A cervical MRI performed in our hospital 1
Maejima et al.
showed lesions with a size of 1.8*0.5*1.1 cm extending from the C3 to C4 level in the left posterior epidural space; the lesion was high on T2-weighted imaging, low with a high portion inside on T1-weighted imaging and high with a low portion inside on short-T1 inversion recovery (STIR). The spinal cord was compressed, and T2-weighted imaging showed mild hyperintense signal (Fig. 1). T2 imaging also showed high intensity in the left C3-5 posterior supporting tissues. The vertebrae themselves showed normal findings. Computed tomography (CT) showed a high-density epidural mass that was mildly enhanced (Fig. 2). In addition, connective tissue associated with the interlamina and interspinous processes at C3/4 and C/5 and the sequential posterior cervical muscles adjacent to the C5 vertebra were also enhanced. The epidural mass was suspected to be a hematoma because his symptoms had rapidly evolved, and we considered the possibility of tumors. We checked the patient’s blood test, which did not show thrombocytopenia or coagulation disorder. Removal of the epidural mass with C3-5 laminectomy was performed for decompression of the spinal cord and to achieve a definite diagnosis.
Operation — A C3-5 laminectomy was performed to sufficiently expose the hematoma. A skin incision was made, and the muscles were divided at the midline. The posterior supporting tissue showed no abnormality. After the C3-5 lamina was removed in an unblock fashion, the hematoma was confirmed and removed by irrigation and suction. In the left epidural space, venous bleeding continued. At that point, a hemorrhagic lesion was found to be stuck to the dura and had the appearance of a yellow ligament (Fig. 3). It was visually indistinguishable from a yellow ligament, but its strong 2
Maejima et al.
adhesion to the dura matter and hemorrhagic region were abnormal. It was coagulated and partially resected for pathological diagnosis. Similar tissue was confirmed on the right side and collected for analysis. Expanded duraplasty was not performed because the tension of the dura matter was not severely affected after the removal of the lesion.
Pathological findings — Frozen and permanent sections of the left lesion were analyzed by hematoxylin and eosin staining and showed atypical cells with scant cytoplasm and a high N/C ratio (Fig. 4). The sample contained collagen fibers. The cell nuclei were dyed by chromatin. In addition, immunostaining revealed atypical cells with AE1/AE3 (+, dot-like), CD56 (+), chromogranin A(-), synaptophysin (+), TTF-1 (+), NKX3.1 (-), and CD20 (-) and that 90% expressed Ki-67. The pathologist made a diagnosis of metastatic carcinoma (small cell carcinoma). The specimen resected on the right side was connective tissue and was not malignant. The sample from the right lesion also showed no malignancy.
Postoperative course — Postoperatively, the patient’s neurological condition improved a little. Manual muscle testing (MMT) of the left upper limb was 2/5, MMT of the other limb was 3/5, and he recovered from paresthesia. MRI showed that the lesion had disappeared but that an intramedullary high-intensity signal at the C4 level remained (Fig. 5). However, his pulmonary status was severe. Because he suffered from pneumothorax, pneumonia, COPD and fatigue of the respiratory muscle, he could not be weaned off the ventilator. After he was diagnosed with small cell carcinoma, a whole body CT was performed for the detection of primary lesions and showed nodules at the hilum and pleura of the right 3
Maejima et al.
lung. Therefore, he was diagnosed with Stage IV lung small cell carcinoma. Enhanced MRI did not reveal any mass in the epidural space. Based on this performance status, pulmonologists assessed that active treatment was not applicable to this patient. During preparation for a hospital transfer to palliative care, his respiratory condition worsened, and he expired at postoperative 4 weeks. An autopsy was not performed on this patient because of his family’s will.
Discussion — To our knowledge, this report is the first case of SEH related to epidural metastatic tumors caused by minor trauma. Spinal epidural hematoma is a rarely occurring condition. Approximately 40% of cases with SEH have no definite etiological factors.2 Others have been shown to result from vascular malformation, anticoagulant therapy, severe trauma or in rare cases, tumors.3,4,6 SEH classically shows rapidly progressive symptoms that evolve into nerve root and/or spinal cord compression followed by acute onset of severe and radiating pain.1,2,3 MRI is the diagnostical tool of choice for the detection of spinal epidural hematomas. T1-weigted images of the pathognomonic region will appear isointense with the cord, and T2-weighted images will show hyperdensity in the periphery with a central hypodense area in the early period.1,2 In this case, we first suspected hematoma because of MRI findings and his symptoms. On the other hand, images showed not only epidural mass but also connective tissue and muscle, both of which were enhanced and suggested other causes, such as neoplasms. We propose that findings related to the extra-spinal canal tend to be overlooked. In cases of SEH with unknown causes, sometimes an analysis of this region can help to make a diagnosis, and enhanced CT or MRI is important. 4
Maejima et al.
The bleeding source for SEH is usually a vein. Because its wall is fragile and has no valves, the spinal venous system is weak when exposed to stress, such as high blood pressure, increased intrathoracic and abdominal pressure caused by sneezing, bearing down and falling or other minor trauma.10,11 In addition, hematogenous metastases to the spine are common and believed to be delivered by a venous route. Primary tumors predominantly metastasize to the breast, lung and prostate, and this tendency has been attributed to the finding that metastases from these tumors spread through the vertebral venous system, widely known as Baston’s plexus.12 Our case has limited value because there was no follow up or autopsy. It is difficult to say whether the patient’s life was further shortened by the epidural hematoma and paralysis. However, enough factors were evaluated to support an important hypothesis based on clinical features, images, and intraoperative and pathological findings. We hypothesize that in our case, the spinal venous system that had obtained hematogenous metastasis became fragile and was bleeding because of acute elevated venous pressure caused by the patient falling on his behind. In a previous similar case, the author estimated that the bleeding was due to the fragility of the epidural venous plexuses.13 — Kim RY reported that 5% of all patients with systemic cancer who are autopsied have pathologic evidence of a tumor invading the extradural space.9 Spinal cord compression caused by epidural metastatic tumors leads to progressive neurological deficits. Epidural metastasis commonly arises from the extension of metastases located in the adjacent vertebral column in the paravertebral spaces through the intervertebral foramina or, rarely, into the epidural space itself.5 In our case, the lesion was located in the epidural space. In addition, neurological images showed abnormal signals in the posterior connective tissue and muscle as well as the interlamina and interspinous processes, 5
Maejima et al.
which are considered to be associated with epidural tumor and neoplasm seeding. In the past, Lin HS reported a similar case which did not have connective tissue lesions but did have lesions in the vertebral bodies and lamina.13 Here, we suggest that the spinal metastatic lesions were quite unlikely to be present only in the epidural space; other lesions have been recognized in peripheral structures, including the vertebral bodies, laminae and connective tissue. In cases of epidural hematoma with abnormal signs on MRI and CT in peripheral structures, hemorrhage-associated metastasis should be considered. — In our case, emergency surgical treatment was chosen because of the rapidly progressive course and the need for a diagnosis. Treatment for epidural hematoma is generally conservative therapy, medication and surgery. Surgical treatment is needed in cases with severe symptoms.7,8 Moreover, direct surgery is helpful for diagnosis. In this case, no obvious massive lesion was confirmed, but the lesion did look like a yellow ligament with an indistinct border. The pathological diagnosis revealed that a similar contralateral mass was a normal ligament. No obvious masses were identified in the posterior cervical muscles or interlaminar and interspinous process connective tissue. The only operative characteristic was strong adhesion. However, the pathological sample showed tumor cells throughout. We hypothesized that this malignancy was so invasive that it had spread to a variety of tissues. Enhanced lesions of not only the spinal canal but also the surrounding tissue related to the spinal canal showed the possibility of infiltrating cancer. Attention was needed during surgery because the lesion did not have a massive form or unique color.
6
Maejima et al.
Conclusion This is the first report of an SEH related to epidural metastasis after minor trauma. In particular, metastatic lesions located in the epidural space itself are so uncommon that it is usually difficult to distinguish them from hematomas based on neurological images alone. In the case of epidural hematoma, especially in cancer-carrying or elderly patients, a hemorrhage originating in the metastasis should be kept in mind during the differential diagnosis.
Declaration of interest None. All authors who are members of the Japan Neurosurgical Society (JNS) have registered online Self-reported COI disclosure statement forms through the website for JNS members.
Funding sources None.
7
Maejima et al.
References 1) Holtas S, Heiling M, Lonntoft M. Spontaneous spinal epidural hematoma: findings at MR imaging and clinical correlation. Radiology. 1996;199:409 413 2) Kreppel D, Antoniadis G, Seeling W: Spinal hematoma: a literature survey with meta-analysis of 613 patients. Neurosurg Rev. 2003;26:1-49 3) Groen RJ, van Alphen HA: Operative treatment of spontaneous spinal epidural hematomas: a study of the factors determining postoperative outcome. Neurosurgery. 1996;39:494 509 4) Cetinalp NE, Oktay K, Ozsoy KM: Spontaneous spinal epidural hematoma mimicking a cerebrovascular disease. Neurol India. 2017;65:1434-1435 5) Byrne TN: Spinal cord compression from epidural metastases. N Engl J Med. 1992;27:614 619 6) Harris DJ, Fomasier VL, Livingston KE: Hemangiopericytoma of the spinal canal. Report of three cases. J Neurosurg. 1978;49:914-920 7) Alexiadou-Rudolf C, Ernestus RI, Nanassis K, Lanfermann H, Klug N: Acute nontraumatic spinal epidural hematomas. An important differential diagnosis in spinal emergencies. Spine. 1998;23:1810-1813 8) Liao CC, Lee ST, Hsu WC, Chen LR, Lui TN, Lee SC: Experience in the surgical management of spontaneous spinal epidural hematoma. J Neurosurg. 2004;100:38-45 9) Kim RY: Extradural spinal cord compression from metastatic tumor. Ala Med. 1990;60:10-15
8
Maejima et al.
10) Groen RJ, Ponssen H: The spontaneous spinal epidural hematoma. A study of the etiology. J Neurol Sci. 1990;98:121-138 11) Kaplan LI, Denker PG: Acute epidural epidural hemorrhage. Am J Surg. 1949;78:356-361 12) Baston OV: The function of the vertebral veins and their role in the spread of metastases. Ann Surg. 1940;112:138-149 13) Lin HS, Chen SJ: Metastatic carcinoma related long segment thoracic spinal epidural hematoma: a case report. Spine. 2009;34:266-268
9
Maejima et al.
Figure legends Figure 1. Preoperative magnetic resonance images (MRI): sagittal view showing the lesion extending from the C3 to C4 level (T2 weighted image: A, T1 weighted image: B, short-T1 inversion recovery (STIR): C). The lesion was located at the left posterior epidural space (axial view on T2-weighted image). T2 axial imaging also showed high intensity in the left C3-5 posterior supporting tissues.
Figure 2. Enhanced computed tomography (sagittal view: A, axial view: B) showing a high-density epidural mass. Connective tissue of the interlamina and interspinous processes at the C3/4 and C/5 levels and sequential posterior cervical muscles adjacent to the C5 vertebra were also enhanced.
Figure 3. Intraoperative view showing hemorrhagic lesions adhered to the dura. A black arrow indicates the dura mater, and a white arrow indicates lesions.
Figure 4. Histopathological examination a frozen and permanent section stained by hematoxylin and eosin to show atypical cells (low power field:A) . The cells showed scant cytoplasm and had a high N/C ratio (high power field: B).
Figure 5. Preoperative magnetic resonance image (T2-weighted image): the lesion had disappeared, but an intramedullary high-intensity signal at the C4 level remained.
10
Abbreviation list COPD, chronic obstructive pulmonary disease; CT, computed tomography; MRI, magnetic resonance imaging; MMT, manual muscle testing; SEH, spinal epidural hematoma
1
Disclosure-Conflict of Interest
Type of article: Case report
Title of the article: Spinal epidural hematoma associated with epidural metastasis after minor trauma: a case report
Contributor 1.
Ryuya Maejima (1)
2.
Masahiro Aoyama (1) (2)
3.
Masahito Hara (2)
(1) Department of Neurosurgery, Aichi Medical University, 1-1 Yazakokarimata, Nagakute, Aichi, 480-1195, Japan (2) Spine Center, Aichi Medical University, 1-1 Yazakokarimata, Nagakute, Aichi, 4801195, Japan
Corresponding Author: RYUYA MAEJIMA, MD.
Source(s) of support: None
Presentation at a meeting: No
Conflicting Interest: None
Acknowledgement:
No employees from any sponsoring agencies or industries contributed to this work. The corresponding author obtained written permission from all contributors prior to including their names in this work. The author contributions are as follows. Conception and design: R. Maejima. Acquisition of clinical data: all authors. Drafting the article: R. Maejima. Approved the final version of the manuscript on behalf of all authors: M. Aoyama. Supervision: M. Hara.