Key-words:
Bone cement - laminectomy - vertebral hemangioma - vertebroplasty
Introduction
Vertebral hemangiomas (VH) are considered the most common benign spinal tumor that
occurs in about 10%–12% of the population.[[1]],[[2]] They are described as benign vascular tumors formed from the vascular spaces that
are lined with endothelium and arise within the marrow spaces.[[3]] Some authors, due to the absence of aggressive histopathological criteria, have
considered them as vascular malformations or hamartomas.[[4]],[[5]],[[6]] They are mostly silent and discovered accidentally except for about 1% that are
considered symptomatic, and when they extend beyond the vertebral body, they are called
aggressive or extensive VH.[[7]],[[8]] Early presentation is usually localized pain while further symptoms develop due
to neural compression secondary to vertebral body expansion, cortical erosion, fracture,
epidural hematoma, or rarely due to cord ischemia.[[9]],[[10]],[[11]],[[12]] The presenting symptoms depend on the tumor location and the extent of spinal cord
or nerve root compression.[[13]] Even in aggressive hemangiomas, the lesion keeps its benign nature with no malignant
transformation.[[1]]
Intervention is considered only in symptomatic lesions. Different lines are described
in the literature, and the selection is still controversial.[[3]],[[14]],[[15]] These treatment modalities include radiotherapy, endovascular embolization, alcohol
injection, vertebroplasty, decompression, fixation, or even en bloc verteberectomy.
Either single or multiple lines may be combined to achieve best therapeutic outcome.[[7]],[[16]],[[17]]
Vertebroplasty using methyl methacrylate (MMA) has been first described in the management
of VHs in the late 1980s. It was proved to produce hemostatic embolization in addition
to maintain and improve the load-bearing capacity of the vertebral body.[[16]],[[18]] In cases with neurological symptoms secondary to the extension of the lesion in
the spinal canal, surgical decompression is indicated.[[7]]
The aim of the present study is to evaluate the combined use of spinal decompression
and vetebroplasty in the management of aggressive VH. All clinical, radiographic,
and operative aspects are described.
Patients and Methods
In this study, the researchers include all patients who have been presented with neurological
symptoms secondary to an aggressive solitary VH. All have been operated upon using
the spinal decompression in addition to transpedicular vertebropalsty. The researchers
have excluded patients with significant vertebral body destruction that require additional
hardware stabilization.
This study includes nine consecutive patients; 6 females and 3 males. Age of the patients
ranges between 22 and 62 years, with a mean age of 45 years. All patients have been
admitted and operated at the Department of Neurosurgery, Tanta University Hospitals
from January 2012 to January 2019. Patients' data have been retrospectively collected
from the hospital records and operative room reports. The researchers have excluded
patients with missed data from the study. Patients' data, diagnosis, and treatment
outcomes are confidentially kept private and patients are presented by specific codes.
Written consent has been obtained from all patients before the surgery.
The researchers collect and review the following data: Patients' age, sex, clinical
presentation, affected spinal level, radiological features, operative details, and
clinical outcomes.
All patients are evaluated and subjected to clinical history, general and neurological
examination, and routine laboratory investigations. Preoperative clinical examination
is carefully done and muscle strength of the lower limbs for each patient is carefully
graded according to the motor power grading system from 0 to 5. Furthermore, each
patient is assigned to a clinical grade according to the Nurick functional grading
system [[Table 1]].[[19]]
Table 1: Nurick grades
Radiological investigation
All patients are preoperatively radiologically evaluated using the X-ray films in
both anteroposterior and lateral views, computed tomography scans (CT), and magnetic
resonance imaging (MRI).
Surgical technique
All cases are operated in the prone position under general anesthesia. After confirming
the affected level using the C-Arm, a midline skin incision is performed. This is
followed by fascial and muscle dissection from the spinous process. A conventional
laminectomy opposite to the site of spinal compression is performed. In a single case
with a lesion limited to one side of the vertebral body, the researchers perform only
a unilateral foraminal decompression. After ensuring adequate decompression of the
neural structures, the researchers introduce the transpedicular vertebroplasty trocar
needle under fluoroscopic guidance. A biopsy is obtained for all cases to exclude
other possible pathologies.
Under continuous fluoroscopic guidance, the researchers inject the semi-liquid MMA
bone cement through the needle into the vertebral body with direct visualization of
the thecal sac to avoid intraoperative leakage and subsequent neurological damage.
The aim is to fill the space of the lesion as much as possible to obliterate and shrink
it [[Figure 1]]. Then, the researchers use the bipolar coagulation to perform further shrinkage
of the epidural component followed by careful excision of coagulated parts. In two
cases in which the VH invade the posterior arch of the affected vertebra, the researchers
start with decompression cranial and caudal to the affected level, followed by vertebroplasty,
and lastly by laminectomy of the affected level. This order is done to minimize the
blood loss.
Figure 1: (a and b) Two subsequent intraoperative lateral C-Arm images showing the transpedicular
needle reaching the middle of D8 vertebral body with gradual filling of vertebral
hemangioma with the radiopaque methyl methacrylate to avoid leakage
After ensuring adequate decompression of the neural structures, the wound is then
closed in layers. After recovery from the anesthesia, all patients are re-evaluated
clinically. The researchers start early mobilization of the patients when possible,
otherwise physiotherapy program for paraparetic patients has been started immediately.
Postoperative radiological evaluation using either X-ray or CT films is done to evaluate
the extent of VH filling with the MMA bone cement. The patients are discharged to
home after 24–48 h and followed in the outpatient clinic.
Follow-up of the patients clinically and with radiographs are done at 1 month and
12 months intervals.
Results
The present study includes 3 males and 6 females with a male:female ratio of 1:2.
The age at the time of presentation ranged from 22 to 62 years with a mean of 45 years.
All of them are presented with a history of progressive back pain associated lastly
with neurological complaint. The mean duration of neurological complaint is 2.78 months
(range 1–6 months). The neurological complaints are as follows: Unilateral sciatica
in 1 patient, bilateral sciatica in 2 patients, and myelopathy in 6 patients (5 of
them have subjective lower limb weakness). Three patients presented with sciatica
are intact regarding the motor power in both lower limbs. Six myelopathic patients
have a mean motor power grade of 3.67 (range 2–5) in both lower limbs and a mean Nurick
grade of 4.33 (range 3–5).
Imaging
On examination of the radiological finding, the researchers have found that the affected
spinal regions are lumbar in 3 patients and dorsal in 6 patients. Three patients have
other hemangiomas, but the presenting lesion is only one in each case. The affected
vertebrae are as follow: 2 cases in D6, 1 case in D7, 2 cases in D8, 1 case in D10,
1 case in L3, and 2 cases in L4. All lesions occupy significant part of the vertebral
body and extend to somewhat in the spinal canal [[Figure 2]] and [[Figure 3]]. One case with L4 affection has an extension limited to the medial border of the
left pedicle resulting in unilateral sciatica [[Figure 5]]. Of the six cases with dorsal spine affection, paraspinal extension is found in
4 of them [[Figure 3]] and [[Figure 4]].
Figure 2: Case 2: Male, 55 years, bilateral sciatica, (a) Sagittal magnetic resonance imaging
T2WI showed hyperintense vertebral hemangioma with salt and pepper appearance affecting
L4 body with epidural extension ventral to the thecal sac, postoperative (b) anteroposterior
and (c) lateral radiographs showing adequate filling with methyl methacrylate and
maintained vertebral body height
Figure 3: Case 3: Female, 22 years, myelopathic. magnetic resonance imaging (a) Sagittal T2WI,
(b) Axial T1WI, (c) Axial T2WI showing vertebral hemangioma in the body of D6 eroding
through both pedicles with dorsal compression of the spinal cord and with mild paraspinal
extension, (d) Axial computed tomography showing vertical trabeculation of the vertebral
body, postoperative (e) sagittal and (f) Axial computed tomography showing site of
laminectomy and filling of the body with radiopaque methyl methacrylate
Figure 4: Graph shows the change in nurick grade for cases with myelopathy before and after
surgery
Figure 5: Case 1, female, 39 years, left sciatica, (a) sagittal magnetic resonance imaging
T1WI showed hypointense vertebral hemangioma in 14, (b) axial magnetic resonance imaging
T2WI show hyperintense vertebral hemangioma in the body and eroding the medial border
of the left pedicle (arrow), (c) axial ct show destructive nature of the vertebral
hemangioma, postoperative (d) lateral and (e) anteroposterior radiographs showing
adequate filling of the vertebral hemangioma with radiopaque methyl methacrylate
The vertebral body height is maintained in all cases. In MRI studies, all lesions
appear hypointense in T1WI and hyperintense in T2WI with the salt and pepper characteristic
appearance [[Figure 3]]. In CT studies, most of the lesions show the typical vertical trabeculation of
the VH [[Figure 3]].
Operative
All cases are treated using combined spinal decompression and vertebropalsty of the
affected level. No significant intra or postoperative events are observed in this
study. The mean intraoperative blood loss is 511.11 ml (range 300–750 ml). No mortalities
are documented. The biopsies that have been obtained from all lesions reveal benign
nature of the hemangiomas.
Outcome and follow-up
On recovery from the anesthesia, no worsening of the preoperative neurological status
is observed. For the three cases presented with sciatica, the mean VAS score has dropped
from 8.33 (range 7–10) preoperatively to 2.67 (range 1–4) postoperatively. 1 month
later, all of them are free from the radicular pain.
For the six cases of myelopathy, they regain their motor power in both lower limbs
over a period of 4 weeks with a mean motor power grade of 5 and a mean Nurick grade
of 1.17 (range 0–2) [[Figure 4]]. The radiological studies that have been done postoperatively reveal near total
occlusion of the VH with the maintenance of the vertebral body height [[Figure 2]], [[Figure 3]], [[Figure 4]], [[Figure 5]], [[Figure 6]]. No clinical or radiological signs of spinal instability or recurrence are observed
over the period of follow-up.
Figure 6: Case 4: Female, 38 years, Myelopathic. magnetic resonance imaging (a) sagittal and
(b) Axial T2WI hyperintense vertebral hemangioma in the body of D8 with extension
ventral to the cord and paraspinal extension, postoperative (c) Sagittal and (d) Axial
computed tomography showing site of laminectomy and filling of the vertebral hemangioma
with radiopaque methyl methacrylate
Discussion
Virchow is the first one who describes the VH in 1867. Although they are considered
benign vascular lesions, they may have an aggressive behavior like malignant tumors.
They are found incidentally in about 10%–12% of the population. This percentage may
reach 30% in female. VH may be solitary or multiple. Dorsal spine is commonly affected
followed by lumbar and lastly by cervical and sacral areas. In about 1% of cases with
VH, they develop symptoms as they continue growing within the bony structures of the
vertebra then they have extraosseous extension either within the spinal canal or in
the paraspinal region.[[20]],[[21]],[[22]],[[23]],[[24]]
VH develops neurologic symptoms either by bony compression resulting from hypertrophy
or expansion of the affected bony parts of the vertebra, soft-tissue compression secondary
to VH extension into the epidural and foraminal space, pathological fracture of the
affected vertebral body; and/or epidural hematoma.[[2]],[[11]],[[25]]
There are different classification systems for VH. They commonly focus on the extension
of the lesion and on the patient clinical presentations. They could be simply classified
into four types or stages: Type I: Asymptomatic lesion with mild bony destruction;
Type II: Localized pain with bony destruction; Type III: Asymptomatic lesion with
epidural and/or soft-tissue extension; and Type IV: Neurological symptoms with epidural
and/or soft-tissue extension. Both types III and IV are considered aggressive types.[[6]],[[26]]
In the present study, the researchers have selected patients that have been presented
with neurological symptoms secondary to aggressive VH. In all of the cases, the normal
vertebral body height is maintained. According to the previously mentioned classification,
all are considered Type IV. Sixty-seven percent of cases were females. The affected
spinal regions are dorsal spine in 67% of cases and lumbar spine in 33% of cases.
The dorsal cases are presented with myelopathy, and the lumbar cases with sciatica.
The mean duration of neurological complaint is 2.78 months (range 1–6 months) which
means a slowly progressive course.
All of the cases are treated with the same technique which is combined surgical decompression
combined with vertebroplasty in the same session. Surgical decompression is admitted
as all of the cases have neurological symptoms secondary to extraosseous extension
of the VH. The add value of vertebroplasty is to occlude the vascular spaces of the
VH, maintain the height of the vertebral body to prevent further collapse and possible
pathological fracture, and to minimize the risk of recurrence. This combination is
associated with mean blood loss of 511.11 ml which is considered appropriate in such
procedures. No deterioration of the preoperative neurological condition or surgery-related
mortality is reported.
The combination of both modalities has achieved the desired outcome in the form of
decompression of the neural elements with relatively mild blood loss. Both resulted
in clinical cure of the patients' symptoms, preservation and stabilization of the
affected vertebral body. In addition to that, no cases of recurrence are recorded
during the follow-up period.
Several options are described in the literature for the management of aggressive VH.
These treatment modalities include radiotherapy, endovascular embolization, vertebroplasty,
transpedicular injection of absolute alcohol, surgical decompression, or any combination
of those modalities. Due to the rarity of cases, the selection between these treatment
lines is still controversial. The selection depends on many factors such as clinical
presentation, radiological features, expected outcome, possible complication, learning
curve, technical requirements, and treatment costs.[[27]],[[28]],[[29]]
Surgical decompression can be done either through posterior laminectomy or through
a more extensive approach in the form of corpectomy and/or spondylectomy. They are
indicated in the presence of rapidly progressive or severe neurological symptoms.
In spite of the beneficial effect of surgical intervention in decompressing the neural
structures and alleviating the risk of recurrence, they are associated with the significant
amount of blood loss and higher risk of mortality.[[3]],[[14]],[[16]],[[30]] They should be combined with other modalities that reduce the risk of excessive
intraoperative bleeding such as endovascular embolization of the feeding arteries,
or transpedicular injection of bone cement, alcohol, or glue to occlude the vascular
spaces within the VH.[[27]],[[28]],[[31]] Jiang et al. report intraoperative lower amount of blood loss by about 50% when
combined both vertebroplasty and laminectomy.[[6]]
Vertebroplasty is first invented by Deramond and coworkers in 1984. They use it in
a case with C1 body aggressive VH.[[32]] Vertebroplasty using MAA bone cement has proven to provide hemostasis and stabilization
when done either preoperatively of intraoperatively. In patients with aggressive VH,
vertebroplasty carries a risk of cement leakage through the eroded posterior vertebral
wall and potential neurological complications. However, it can be safely done under
direct visualization after laminectomy and with the aid of intraoperative fluoroscopy.[[7]],[[33]],[[34]]
Radiotherapy can also be used either alone or in combination with other modalities.
It can be used either before or after surgical decompression. It is used alone in
mild cases without evident neurological symptoms. It can minimize the risk of recurrence.
It carries the risk of radiation-induced myelitis, radionecrosis, and secondary malignant
transformation. It also does not provide structural support to the affected vertebral
body which may result in pathological fracture. It can be reserved for postoperative
use to minimize the risk of recurrence.[[2]],[[35]]
Ethanol injection in the VH is first used by Heiss et al. in 1994 to achieve the destruction
of the endothelium of the hemangioma and intralesional thrombosis.[[20]],[[36]] It carries the risk of transient neurological deterioration, spinal cord injury,
osteonecrosis, pathological fracture, hemodynamic instability, and asystole.[[4]],[[36]],[[37]]
Conclusions
The researchers conclude that the combined use of both surgical decompression and
intraoperative transpedicular vertebroplasty in cases with aggressive VH is considered
safe and effective modality. Both result in symptom relief, improving quality of life,
with no risk of recurrence or mortality. Preoperative selection of the ideal patient
for this modality is crucial to achieve the desired outcome.
