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ACUTE SPINAL CORD COMPRESSION FROM AN

EXTRAOSSEOUS VERTEBRAL HEMANGIOMA WITH


HEMORRHAGIC COMPONENTS: A CASE REPORT
Tarkan Ergun, MD, a Hatice Lakadamyali, MD,a Huseyin Lakadamyali, MD,b and Amir Mukaddem, MD c

ABSTRACT

Objective: This case report presents a patient with acute compression myelopathy caused by acute hemorrhage of a
thoracic vertebral hemangioma extending into the epidural space.
Clinical Features: A 22-year-old male patient experiencing back pain for 5 months presented to our medical
facility complaining of sudden onset numbness and muscle weakness in the lower extremities.
Intervention and Outcome: Magnetic resonance imaging of the thoracic spine revealed a T5-level mass involving
predominantly the posterior vertebral elements, extending into the epidural area, and showing significant gadolinium
enhancement. Hemorrhagic signal changes were noted within the epidural component of the mass. In addition, the
epidural mass component was noted to significantly compress the spinal cord. The patient was referred for emergency
surgery with the preliminary diagnosis of epidural vertebral hemangioma with hemorrhagic component; a decompression
laminectomy was performed without preoperative angiography. The patient's complaints improved completely after
surgery, and radiotherapy was instituted for the residual tumor tissue.
Conclusion: The presence of acute or subacute myelopathic symptoms is usually suggestive for malignancy or
metastasis. However, in young patients, vertebral hemangioma, causing acute hemorrhage, should be considered in the
differential diagnosis. Decompression surgery should be done in such cases before neurological symptoms
become irreversible. (J Manipulative Physiol Ther 2007;30:602-606)
Key Indexing Terms: Hemorrhage; Spinal Cord Compression; Hemangioma

ertebral hemangioma (VH) is a developmental canal due to expansion of the vertebral corpus caused by the

V hamartoma consisting of vascular tissue. These


lesions have no malignant potential because they
do not grow by mitotic division.1 According to information
hemangioma.3 Acute spinal cord compression cases sec-
ondary to a hemorrhage into the epidural space as with our
patient are extremely rare4; thus, no standard therapy exists
obtained from autopsy studies, the incidence of VH ranges for such cases. However, immediate surgical intervention is
from 10% to 12%.2 These tumors usually produce no necessary in cases of acute compressive myelopathy before
symptoms and are mostly diagnosed incidentally. In rare the symptoms become irreversible.
cases, a VH compresses the spinal cord and nerve roots, We present a patient with thoracic VH that extended into
which causes back pain, radiculopathy, and myelopathy. the epidural space and caused acute spinal cord compression
The mechanisms underlying the compressive symptoms as a result of bleeding from the lesion.
in VH cases are mostly related to narrowing of the spinal

CASE REPORT
a
Specialist, Department of Radiology, Alanya Teaching and A 22-year-old man, complaining of sudden numbness and
Medical Research Center, Baskent University, Alanya, Turkey. minimal muscle weakness in both legs and back pain of
b
Specialist, Department of Pulmonology, Alanya Teaching and 5 months' duration presented to our institution. Physical
Medical Research Center, Baskent University, Alanya, Turkey.
c examination revealed tenderness in the thoracic region on
Specialist, Department of Neurosurgery, Alanya Teaching and
Medical Research Center, Baskent University, Alanya, Turkey. deep palpation and hyperactive deep tendon reflexes in the
Submit requests for reprints to: Tarkan Ergun, MD, Specialist, lower extremities. Motor examination revealed a 4/5 muscle
Baskent Universitesi, Alanya Hastanesi, Radyoloji Bolumu, 07400 weakness score in the lower extremities. The preliminary
Alanya, Antalya, Turkey. (eQmail: tarkanergun@yahoo.com). diagnosis was herniation of the nucleus pulposus at the
Paper submitted December 16, 2006; in revised form May 2, thoracic region. Magnetic resonance imaging (MRI) of the
2007; accepted June 9, 2007.
0161-4754/$32.00 thoracic vertebral column revealed a T5-level soft-tissue
Copyright © 2007 by National University of Health Sciences. mass that involved both the anterior and posterior vertebral
doi:10.1016/j.jmpt.2007.06.007 elements, extended into the epidural space, caused expansion

602
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Journal of Manipulative and Physiological Therapeutics Ergun et al 603
Volume 30, Number 8 Hemorrhagic Hemangioma

Fig 2. A post intravenous gadolinium injection T1-weighted image


of the same level reveals a significant osseous enhancement of the
vertebral corpus and posterior elements affected by the heman-
gioma and significant enhancement of the extraosseous component
of the hemangioma extending into the posterior epidural space and
right paravertebral region (arrow).

of the laminae and pedicles on both sides, and significantly


compressed the spinal cord. Magnetic resonance imaging
studies also revealed a T2-T7 level epidural soft-tissue mass
with multiple hyperintense areas of hemorrhage in the T1-
weighted sequences; these contained a small amount of fat
in the fat suppression sequences, which were enhanced by
the administration of intravenous contrast media (Figs 1A-B
and 2). The MRI features of the lesion suggested a VH, but
the expansive nature of the tumor and its extension into the
epidural space suggested a possible malignancy. The
clinical characteristics of the lesion did not indicate an
infectious process.
Taking into consideration the young age of the patient and
the radiological features of the lesion, we suspected a
hemorrhagic VH extending to the epidural area, and the
patient was referred for immediate surgery.
After determination of the surgical level using C-arm
fluoroscopy, the paraspinal muscles were dissected at the
T5-level with a posterior approach. Posterior decompres-
sion was accomplished by removing the vertebral laminae.
The posterior epidural tumoral tissue and hematoma were
resected as much as possible. The acute blood loss due
to intratumoral hemorrhage comprised 2 L, and a 2-unit
Fig 1. A, Sagittal T1-weighted image shows a soft-tissue mass
extending into the epidural space, causing significant compression intraoperative blood transfusion was done. The region
and anterior displacement of the spinal cord (arrows). B, A was covered with thrombin-soaked gel foam to prevent
significant hyperintense hemorrhagic component is visible within further hemorrhage.
the epidural space; the mass cannot be suppressed in the T2- The results of histopathologic examination of the
weighted fat-suppression sequences.
partially resected mass showed that the lesion was a VH.

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604 Ergun et al Journal of Manipulative and Physiological Therapeutics
Hemorrhagic Hemangioma October 2007

Postoperative radiotherapy was instituted to prevent recur- contrast media.8 The MRI studies provide additional
rence of the residual mass. The total radiotherapy dose was information about the aggressiveness of the hemangioma,
given in 20 fractions of 40 Gy (5 fractions per week), and no depict the location and extraosseous extension of the tumor,
complications were encountered. The patient's symptoms and reveal the extent of the neural compression caused by the
improved completely in the third day after surgery, and he lesion. Aggressive tumors with a potential to compress the
was symptom-free at the follow-up visit 6 months after spinal cord appear as highly vascular soft-tissue masses in
the operation. the MRI sequences; these are identified by their isointense
presence in the T1-weighted images, hyperintense signal
in the T2-weighted series, and significant contrast enhance-
DISCUSSION ment after an intravenous contrast injection. The fat
A VH is usually a solitary lesion located in the component, the presence of which is a good prognostic
vertebral corpus, although it may occasionally involve the sign, is hyperintense in both the T1- and T2-weighted
posterior vertebral elements. Vertebral hemangiomas series and is suppressed in the fat-suppression series.9 The
develop most frequently in the thoracic vertebrae.5 Usually hemorrhagic component appears hyperintense in both
asymptomatic for a lifetime, VHs may cause symptoms in sequences, as with our case, but cannot be suppressed in
1% of cases, the most common symptom being back pain. the fat-suppression sequences.
It may rarely cause neurological symptoms and compres- The clinical and radiological findings in our patient were
sive myelopathy and thus should be considered in the concordant. The MRI high-vascular soft-tissue component
differential diagnosis of metabolic, inflammatory, and ratio of the mass was high, which indicates aggressive tumor
neoplastic myelopathies. behavior. The fat-tissue component ratio of the mass was
The physiopathologic mechanism of a VH that com- low, and the hemorrhagic component ratio of the mass
presses the spinal cord involves the angiomatous hypertro- was significant.
phy of the vertebral arch and the widening of the vertebral The differential diagnosis of spinal tumors is principally
corpus, which leads to the deformation and narrowing of the based on the localization of the tumor (Fig 3). They are
vertebral canal, extension of the tumor into the epidural classified in this concern as intramedullary, intradural/
space, collapse of the vertebral corpus, and (rarely) extramedullary, and extradural (osseous) spinal tumors.10
expansion of the tumor vessels and subsequent epidural Intramedullary spinal cord tumors comprise nearly 25% of
hemorrhage. In our patient, the tumor caused the expansion all spinal tumors, mostly being ependymomas in the adults
of the vertebral laminae and pedicles, and the resultant and astrocytomas in children. Intramedullary tumors gen-
significant expansion of the epidural area compressed the erally cause expansion of the spinal cord. Most encountered
spinal cord and nerve roots. intradural/extramedullary tumors are schwannomas and
Hemangiomas that extend extradurally may cause spinal neurofibromas. These lesions usually expand the spinal
pain, radiculopathy, progressive paraparesis, or acute para- neural foramina and erode the vertebral bodies. Radiologi-
paresis as a result of their epidural mass effect. The cally, they are visualized as smooth-contoured lesions that
progression of VHs is often insidious.2 As in our patient, may show cystic changes.6 The second most frequent
the sudden hemorrhage or thrombosis of the hemangioma intradural/extramedullary tumor is meningioma, being
can cause acute clinical symptoms resulting from the rapid second to nerve sheath tumors. Its most frequent location
enlargement of the lesion.6,7 is the thoracic region. It has a broad dural base, a typical
Ninety percent of the VHs that produce symptoms isointense appearance in the T1-weighted sequence, a
develop in the thoracic vertebral column, as was the case hypointense appearance in the T2-weighted sequences
in our patient, and 75% of those lesions occur between levels (with respect to the spinal cord), and shows significant
T3 and T9. The second most frequent site of development is homogeneous gadolinium enhancement after an intravenous
the lumbar vertebral column. Hemangiomas are usually injection. Its dural tail can be traced. In contrast to nerve
located in the vertebral bodies and may extend into the sheath tumors, meningiomas extend rarely into the extra-
posterior elements. They rarely affect all the posterior dural region.8
elements, extending into the epidural space, as in our case. Extradural tumors are the most frequently seen spinal
Vertebral hemangiomas appear as parallel vertical lines or tumors. The osseous lesions can either be primary or
as a honeycomb in the vertebral corpus in plain roentgen- metastatic. Vertebral hemangioma is the most commonly
ograms. Its characteristic computed tomography appearance encountered lesion. The second most common tumor is
is a “polka dot” appearance within the bony structure due to osteoblastoma, which accounts for 10% of the spinal
thickened trabeculae, secondary to cystic vascular structures. osseous tumors. It is an expansile osteolytic lesion with
The MRI images of a VH reveal a lesion that is isointense occasional epidural component. It has low/medium signal
with the spinal cord in T1-weighted images, hyperintense in intensity in the T1-weighted and high signal intensity in the
T2-weighted images, and characterized by significant T2-weighted series and may show gadolinium enhance-
contrast enhancement after the intravenous injection of ment. Aneurysmal bone cyst is an expansile and aggressive

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Journal of Manipulative and Physiological Therapeutics Ergun et al 605
Volume 30, Number 8 Hemorrhagic Hemangioma

Asymptomatic VHs do not need follow-up imaging or


therapy. Symptomatic ones, on the other hand, can be treated
either with surgery, transarterial embolization, radiotherapy,
vertebroplasty, or direct ethanol injection. Vertebral heman-
giomas causing pain only and having no extraosseous
extension can be treated with transarterial embolization,
vertebroplasty, or radiotherapy, whereas those with progres-
sive neurological deficits should be treated with surgery.13
The percutaneous vertebroplasty technique involves
injection of acrylic cement, usually polymethylmethacrylate
(PMMA), into the vertebral body. The mechanism of action
of this procedure is not entirely clear, but it is believed that
stabilization of microfractures, prevention of further com-
pression, and PMMA-chemical ablation of pain-sensitive
neural roots in the vertebral body provide cure to the pain.
However, this procedure does not obliterate the hemangioma
and does not prevent its further progression; thus, it is
preserved for lesions causing pain only and is not suitable for
lesions showing extraosseous extension. 14 In addition,
PMMA-related spinal cord injuries have been reported
secondary to PMMA escaping into the spinal canal.
Vertebral hemangioma is a radiosensitive lesion, which
is why radiotherapy is one of the therapeutic modalities and
is especially preferred in the lesions causing pain only.2 It is
Fig 3. Drawings of the normal thoracic spinal column: axial view also reported to be effective in VH lesions that cause
(A) and sagittal view (B).
neurological deficit. However, because its effect appears late
in the course of therapy, its use alone in cases of VH with
bone lesion having multilocular blood spaces. It is mostly neurological symptoms is debated. The average radiation
encountered in the lumbar region and shows a tendency to dose is 30 to 40 Gy. This suggested that dose of radiation
affect the neighboring vertebra, passing through the bears no risk for spinal cord injury, but radiation-related
intervertebral disk. Its characteristic computed tomography sarcoma development in the late period has been reported.15
appearance is a multiloculated expansile cystic mass lesion The preferred therapeutic modality in cases with progres-
containing fluid-fluid levels.11 The giant-cell bone tumor is sive neurological deficit should be preoperative transarterial
mostly located in the sacrum. The vertebral body lesion embolization followed by surgical decompression.13 The
extends to the posterior elements, into the disk space, and to surgical approach (anterior or posterior) or the surgical
the neighboring vertebral body. Its characteristic radiological procedure of choice depends on the localization of the
appearance is an expansile lytic lesion with no significant lesion and the rate of progression of the neurological
sclerotic border. It may have solid and cystic components. derangement. In all cases with fast and progressive
The solid component has medium signal intensity in the neurological deficit, laminectomy and immediate decom-
T1-weighted series and is hypointense in the T2-weighted pression are mandatory.2,16
series, in opposite to chordoma, plasmocytoma, and meta- In cases of neurological deficit secondary to neural canal
static lesions. stenosis caused by osseous tumor growth only (without
Chordomas are most frequently located in the sacrococcy- extraosseous tumor extension), simple decompression lami-
geal region and have 60% amorphous calcifications content. nectomy would be sufficient. However, when extraosseous
Chondrosarcoma destroys the bone structure, and its chon- tumor components are present, postoperative radiotherapy
droid matrix can be traced. Advanced cases may have soft- should be instituted after reducing the aggressive (vascular)
tissue components. The lesion shows ring- or arch-shaped tumor components because it prevents growth and recur-
septal contrast enhancements with intravenous gadolinium. rence of the residual tumor. In cases of extraosseous tumors
Osteosarcoma has an osteoblastic tendency and shows originating from the vertebral corpus and cord compression
hypointense MRI signal intensity in both the T1- and T2- due to extraosseous tumor extension, corpectomy or
weighted sequences.10 The typical MRI signal of metastatic vertebrectomy should be preferred. The most basic reason
bone tumors, such as myeloma, breast cancer, lung cancer, for perioperative morbidity in VH surgery is intraoperative
prostate cancer, and leukemia/lymphoma, is hypointense in the blood loss or postoperative epidural hematoma. These
T1-weighted and hyperintense in the T2-weighted sequences, complications are decreased using preoperative transarterial
the lesions being of different size and separate.12 embolization.16 The period between initial neurological

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606 Ergun et al Journal of Manipulative and Physiological Therapeutics
Hemorrhagic Hemangioma October 2007

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