Primary malignant tumors of the cervical spine are
rare1 but challenging because of the spine’s unique structure
and proximity to vital structures. A thorough clinical and imaging
investigation is necessary for the oncological and surgical staging of these
lesions. New operative techniques for aggressive en bloc resection of these
tumors through combined anterior and posterior approaches and reconstruction of
the spinal column have improved the oncological outcome with low local
recurrence rates and increased patient survival.2-11
Malignant bone tumors are rare, accounting for 0.4%-1% of all
tumors.1 Only 10% of these affect the spine, with 4.2%-6.3%
affecting the mobile spine above the sacrum.1,12-14 In children and
adolescents, malignant tumors account for <30% of="" all="" spine="" tumors,="" whereas="" in="" adults,="" approximately="" 75%="" of="" spine="" tumors="" are="">30%>15 The
male to female ratio is 2:1, with an even distribution among all spine
levels.1 Multilevel involvement may be noted, and at diagnosis, the
tumor may extend to the paravertebral soft tissues.13
Myeloma and solitary plasmatocytoma are the most common primary
malignant tumors of the adult spine, accounting for approximately 30% of all
primary malignant spine tumors. In the Mayo Clinic Registry, only 3% of all
myeloma cases affected the cervical spine.1 Solitary plasmatocytomas
are diagnosed in approximately 3% of myelomas, with 50% occurring in
middle-aged men (mean age 50 years).16,17
Non-plasma cell lymphomas account for approximately 10% of
primary malignant lesions. In the Mayo Clinic Registry, only 0.8% of all
lymphoma cases affected the cervical spine.1 Non-plasma cell
lymphomas are associated with epidural, rather than bony
Chordomas of the spine are the most common solid spine tumors,
accounting for 33% of the primary malignant tumors of the spine.16
Eighty-five percent of all chordomas occur in the sacrum or the
clivus.18,19 In the Mayo Clinic Registry, only 6% of all chordoma
cases affected the cervical spine.1 These tumors are thought to
arise from notochordal remnants in the vertebral disks. They are considered
low-grade, slow-growing, locally invasive, indolent malignancies, that behave
aggressively in the region of their origin and metastasize late in their course.
Chondrosarcomas are the second most common solid tumors,
accounting for approximately 10% of primary malignant spine tumors. In the Mayo
Clinic Registry, only 1.5% of all chondrosarcomas affected the cervical
spine.1 Most chondrosarcomas are relatively indolent and tend to
recur locally before distant spreading occurs. Various low to highly malignant
histologic subtypes of chondrosarcomas have been reported.20-21
Osteosarcomas and Ewing’s sarcomas are highly malignant bone
lesions, accounting for 10% of malignant primary tumors of the spine.
Osteosarcomas occur in the spine in <3% of="" any="" other="" skeletal="">3%>11,22,23 In the Mayo Clinic Registry, only 0.5% of all
osteosarcomas and only 0.4% of all Ewing’s sarcomas affected the cervical
spine.1 Ewing’s tumors occur primarily in
children.24 Osteosarcomas may rise primarily in the spine (usually
seen in adults), or they may occur secondarily in areas previously irradiated
or in patients with Paget’s disease (usually seen after age 60 years).
Primary malignant tumors of the cervical spine usually manifest
pain as the primary symptom. However, they may be diagnosed incidentally on
plain radiographs or physical examination. Severe night pain preventing sleep
or waking the patient is a common symptom.3,13 Spasm, scoliosis, or
torticollis; neck stiffness and tenderness, and dysphagia due to compression of
the esophagus may be associated symptoms.9 General symptoms such as
weight loss and anorexia can also occur.12
Neurological deficits are common; however, the incidence of
neurological deficits at diagnosis is reported to be <>25
Aggressive tumors produce cord or root compression more rapidly than indolent
In upper cervical spine tumors, pain usually is the initial
symptom.26 Neurologic deficits in these patients are less frequent
and occur late in the disease process because of the wide spinal canal at this
level. More likely, neurologic symptoms result from spine instability rather
than direct compression from the tumor.
In lower cervical spine tumors, an insidious onset of cervical
pain, usually is the initial symptom.12,13 Radicular pain is most
common in cases with soft-tissue or bony encroachment on the foramina and may
be associated with hypesthesia and muscle weakness.
Plain radiographs remain the first imaging tool in the diagnosis
and evaluation of malignant tumors of the cervical spine.1,2
Standard imaging features include a radiolucent and destructive lesion or
severe osteopenia of one or more vertebrae, often associated with pathologic
fracture (Figure 1A). Other findings include the loss of a pedicle on an
anteroposterior (AP) view, an osteoblastic lesion, the destruction of one or
more vertebrae with preservation of the disks, or a paraspinal soft-tissue
mass, with or without calcification. Collapse of the vertebral body with
radiographic appearance of vertebra plana has been reported.27
Secondary spine deformities, such as kyphotic or scoliotic angulation, may also
Bone scintigraphy shows increased radioisotope uptake in the
majority of malignant spinal tumors (Figure 1B). The disadvantage is the low
specificity due to the high rates of false-positive findings in coexisting
degenerative spondylosis in older patients and pyogenic infections of the
spine. In most cases, hematopoietic and lymphoproliferative tumors, such as
leukemia and plasma-cell dyskrasias plasma-cell malignancies produce little, if
any, osteoblastic reaction and therefore are not evident on isotope bone
Computed tomography (CT) is more sensitive than plain films and
more accurate than magnetic resonance imaging (MRI) in imaging bone lesions
(Figure 1C). Computed tomography is most useful in oncological staging for the
CT-guided needle biopsy and the surgical staging and instrumentation planning
of patients with diagnosed malignant tumors of the spine.28 Computed
tomography and MRI provide the most detailed views of the transverse and
longitudinal extent of the tumors, the presence of a reactive tissue margin, or
the invasion of the epidural space (Figures 1D and
Magnetic resonance imaging is most useful in the evaluation and
differential diagnosis of intra- and extradural spinal lesions. Spine tumors
usually are evident on MRI before canal compromise or instability develops.
However, MRI may not be feasible in some cases (eg, in patients with pacemakers
or other metallic implants and claustrophobia or
Angiography and selective arterial embolization can be performed
for vascular tumors.18,31-33
Biopsy is the final step in the preoperative work-up for the
determination of the histological type and staging of the tumor. In vascular
lesions, embolization is indicated prior to biopsy.33
Three types of biopsy techniques are used: percutaneous needle
(fine-needle aspiration or CT-guided core biopsy), open incision, and
Fine-needle aspiration provides a small amount of tissue and is
associated with a high rate of false-negative results. In the cervical spine,
its use is limited, and generally is performed to confirm metastatic disease,
local recurrence of a known lesion, or lesions with a limited differential
Percutaneous CT-guided core biopsy although more difficult, has
been performed successfully in the cervical spine.36 It is
considered the best way to diagnose primary or secondary spine malignancies,
with a low incidence of tumor spreading in the surrounding tissues.
Open incision biopsy should be performed in cases where
sufficient tissue is required for immunohistochemical, molecular, and
microbiological analyses. Open biopsy of cervical spine lesions is best
obtained through an anterior approach rather than by laminectomy. This prevents
contamination of the epidural space by violation of the pseudocapsule of the
tumor.34 Meticulous hemostasis should be maintained. Although often
impossible in the cervical spine, biopsy with an adequate margin of healthy
tissue should be included, and all contaminated tissue and hematomas should be
excised at the definitive surgical procedure.1,2,12,39 Excisional
biopsy should be reserved for tumors of the posterior elements of the cervical
It is recommended that the surgeon performing the definitive
procedure also be involved in the biopsy and preoperative staging of the
In few cases, biopsy can be avoided especially when imaging
features are strongly consistent with a certain diagnosis such as recurrent
primary tumor, metastatic disease, and in cases in which a certain diagnosis,
such as multiple myeloma, can be provided.
The purpose of staging is to identify the location and local or
systemic extent of the lesion and to confirm whether it is a primary or
metastatic, benign or malignant tumor.12
The principles of the Enneking staging system of musculoskeletal
neoplasms are also applied to the spine.2,34,42,43 At the initial
examination, tumors are staged according to histologic type, compartmental
location, and the presence or absence of metastases.
The Enneking staging system2,34,43 divides benign bone
tumors into three stages: stage 1, also called latent or inactive; stage 2,
active; and stage 3, aggressive (Figures 2A-2C). Stage 3 tumors generally do
not have a true capsule, or the capsule is thin and discontinuous, and the
tumor extends outside of the vertebral compartment of origin. Because of the
high rate of local recurrences and probable malignant degeneration, treatment
should focus on an aggressive surgical approach, with a safe, wide margin
surgical excision of the lesion, in addition to adjuvant therapy as
According to the same system, malignant tumors are staged as low
grade (stage I), high grade (stage II), or any grade tumor with distant
metastases (stage III) (Figures 2D-2G). Low-grade, stage I malignant tumors are
further subdivided into stage IA (tumors remaining inside the vertebra) and
stage IB (tumors extending into the paravertebral compartments). No true
capsule but a thick pseudocapsule of reactive tissue is associated with these
lesions. The major difference between stage 3 primary benign and stage I
malignant tumors is the potential for satellite lesions or foci of active tumor
within the pseudocapsule, which often are neglected when inadequate resection
is performed. The treatment of choice for these tumors is wide en bloc excision
with adjuvant megavoltage irradiation.2
High-grade, stage II malignant bone tumors are also subdivided
into stage IIA (intracompartmental tumors within the vertebra) and stage IIB
(tumors extending outside the vertebra into the surrounding paravertebral
region). Stage III includes the same lesions as stages I and II, but with
distant metastases, in addition to intra- (stage IIIA) and extra-compartmental
(stage IIIB) extension. Because of the rapid tumor growth, no reactive tissue
layer is formed and continuous seeding of tumor nodules (satellite nodules) and
nodules at some distance from the main lesion (skip metastases) are observed.
Extension can also occur into the epidural space.
Surgical staging is the final step in the preoperative work-up of
the patient. The unique anatomy of the vertebra and the difficulty of spine
tumor resection led to the first surgical classification system proposed by
Weinstein, Boriani, and Biagini (WWB staging system), which has been further
modified and tested.2,34,42,44 In the WBB staging system, the
vertebra in the transaxial plane is divided into 12 radiating zones (numbered 1
to 12 clockwise) and 5 layers (named A to E from paravertebral extraosseous
region to dural involvement) (Figure 3). The extent of the tumor is identified
by the number of sectors occupied and the letters of the layers
involved.2,42 The transverse and longitudinal extent of these tumors
is confirmed with CT and MRI, and sometimes with
|Figure 2: Stage 1 benign tumors (A). The tumor is inactive and contained within its
capsule (1). Stage 2 benign tumors (B). The tumor is growing, and the capsule
(1) is thin and bordered by a pseudocapsule of reactive tissue (2). Stage 3
benign tumors (C). The aggressiveness of these tumors is evident by the wide
reaction of healthy tissue (2), and the capsule (1) is thin and discontinued.
Stage IA malignant tumors (D). The capsule, if any, is thin (1) and the
pseudocapsule (2) is wide and contains an island of tumor (3). Stage IB
malignant tumors (E). The capsule, if any, is thin (1), and the pseudocapsule
(2) is wide and contains an island of tumor (3). The tumoral mass is growing
outside the compartment of occurrence. Stage IIA malignant tumors (F). The
pseudocapsule (2) is infiltrated by tumor (3), and the island of tumor can be
found far from the main tumoral mass (skip metastasis, 4). Stage IIB malignant
tumors (G). The pseudocapsule (2) is infiltrated by tumor (3), which is growing
outside the vertebra. An island of tumor can be found far from the main tumoral
mass (skip metastasis, 4). (Reprinted with permission from Boriani S et al.
Primary bone tumors of the spine. Terminology and surgical staging.
Spine. 1997; 22:1036-1044. Copyright © 1997. Lippincott Williams and Wilkins.)|
Figure 3: Weinstein, Boriani, and Biagini (WBB) Surgical Staging System. The
transverse extension of the vertebral tumor is described with reference to 12
radiating zones (numbered 1 to 12 in a clockwise order) and to 5 concentric
layers (A to E, from the paravertebral extraosseous compartments to the dural
involvement). The longitudinal extent of the tumor is recorded according to the
levels involved. (Reprinted with permission from Boriani S et al. Primary bone
tumors of the spine. Terminology and surgical staging. Spine.
1997; 22:1036-1044. Copyright © 1997. Lippincott Williams & Wilkins.)
Figure 4: Classifications of different anatomic sites of the vertebra. 1=vertebral
body; 2=pedicle; 3=lamina, transverse, and spinous processes; 4=spinal canal
(epidural space); and 5=paravertebral area. (Reprinted with permission from
Tomita K et al. Total en bloc spondylectomy: a new surgical technique for
primary malignant vertebral tumors. Spine. 1997; 22:324-333.
Copyright © 1997. Lippincott Williams & Wilkins.)
A modification of the Enneking oncologic staging system was
proposed by Tomita et al,11 who incorporated a description of the
affected anatomic site and extent of the tumor. This system has been reported
in relation to the thoracolumbar spine; however, it is relevant to the cervical
spine as well. According to this classification system, the vertebral body is
divided into five anatomic sites: the body (1), the pedicle (2), the lamina and
spinous process (3), the epidural canal (4), and the paravertebral area (5),
with the numbers reflecting the common sequence of tumor progression (Figure
4). Intra-compartmental lesions are in sites (1), (2), and (3), and
extracompartmental lesions in sites (4), (5), and (6). A type (7) lesion is one
with multiple lesions or “skip” metastases (Figure 5). The purpose of
this surgical system is to further define the lesions that can be excised en
bloc. This can be attempted in lesions (2) to (5) and with a relative
indication in lesions (1) and (6). According to the author, wide en bloc
surgical resection is not recommended in lesions type (7).
Management of primary malignant tumors of the cervical spine is
challenging. Complete excision of the tumor is the goal of treatment. Until
recently, because of the unique anatomic structure of the cervical vertebra,
the proximity to major vessels and the spinal cord, and the difficulty
attaining wide excision of the spinal tumor, most surgical procedures included
curettage and piecemeal intralesional tumor resection. As a result, incomplete
tumor excision, tumor cell contamination of the surrounding tissues, and high
rates of local recurrences and distal metastases were
Lymphoproliferative and plasma cell neoplasms usually are
radiosensitive even in response to low doses of conventional radiation.
Systemic chemotherapy is also frequently indicated. Surgery is rarely
necessary. Indications include progressive neurologic deficits, due to spinal
instability, spinal cord pressure or nerve entrapment by tumor mass, or
pathologic fracture, or recurrent lesions after maximum dose
irradiation.13,17 The prognosis for solitary plasmacytoma is better
than multiple myeloma, with a higher mean survival rate.16
|Figure 6: CT showing a lytic lesion in the left C5 vertebral body, sclerotic borders,
and widening of the left C5-C6 foramen (A). CT showing an osteolytic lesion
with lobular pattern accompanied by soft tissue extension (white arrow) in the
vertebral body of C5 (B). T1-weighted MRI showing a slight increase in
intensity in the lateral side of C5 with soft tissue extension (C). Schematic
illustrating the technique used for cutting the left pedicle of C5 using the
threadwire saw (D). Postoperative lateral radiograph showing anterior interbody
fusion of C5 to C7 with the plate (E). (Reprinted with permission from Fujita T
et al. Chordoma in the cervical spine managed with en bloc excision.
Spine. 1999; 24:1848-1851. Copyright © 1999. Lippincott Williams & Wilkins.)
Figure 5: Schematic diagram of the surgical classification of vertebral tumors.
(Reprinted with permission from Tomita K et al. Total en bloc spondylectomy: a
new surgical technique for primary malignant vertebral tumors. Spine.
1997; 22:324-333. Copyright © 1997. Lippincott Williams & Wilkins.)
The goal of treatment for chordomas of the cervical spine is en
bloc surgical resection (Figure 6).18,49 However, in most cases,
only marginal or intralesional resection is feasible.42,49
Intralesional excision is associated with high recurrence
rates.13,49 Chordoma is not sensitive to chemotherapy and only
high-dose radiation therapy seems to slow the evolution of the
Chondrosarcomas are usually low-grade tumors and are resistant to
irradiation and chemotherapy.16 The treatment of choice is wide
surgical resection with wide surgical margins, which in some instances requires
total en bloc spondylectomy.21 These tumors tend to recur locally
with prolonged disease-free periods before distant metastases occur.
The natural course of osteosarcomas usually is rapid and a poor
prognosis is anticipated.13,22 To improve survival, wide tumor
resection is recommended with safe margins.13 Although adjuvant
radiotherapy has been reported when wide resection is not feasible,
osteosarcomas are not radiosensitive. Total en bloc spondylectomy is the
treatment of choice for osteosarcomas of the spine, in addition to neoadjuvant
and postoperative chemotherapy.9,10,13,22,50
Multi-agent chemotherapy followed by wide tumor resection is the
mainstay of treatment for Ewing’s sarcoma.13 In cases of
neurologic deterioration, spinal decompression may precede chemotherapy.
High-dose radiation therapy and chemotherapy may be used as adjuvant to
surgical treatment or alone when operative treatment is not
Several studies describing the surgical excision of primary
malignant or aggressive benign spine tumors using combined anterior and
posterior surgical approaches have been reported.4,8,11,21,22,50-52
However, the combined anteroposterior procedures may be more extensive and
traumatic than the single posterior or anterior approach and may increase the
chance of contamination by tumor cells and subsequent local tumor
Total en bloc spondylectomy has been described for complete tumor
resection and oncologic management of primary malignant vertebral tumors of the
thoracolumbar spine.11,12 The procedure is performed in two steps,
consisting of en bloc laminectomy after bilateral pediculotomy using a
stainless-steel threadwire saw (step I), and en bloc corpectomy followed by
anterior fusion with instrumentation and spacer grafting and posterior spinal
instrumentation (step II).10,47,48,51
The major risks in total en bloc spondylectomy are mechanical
damage to adjacent neural structures during the excision of the pedicles,
possibility of tumor cell contamination during pediculotomy, injury of major
vessels during dissection of the anterior aspect of the vertebral body, blood
perfusion disturbance of the spinal cord at the level of surgery, and excessive
bleeding from the internal vertebral vein and epidural venous
The use of the threadwire saw is believed to minimize tumor cell
contamination of the surrounding tissues or the surgical incision.9
No major complications such as mechanical damage, dural tears or leakage of
cerebrospinal fluid, damage to the nerve roots, neurological complications, or
signs of aseptic necrosis, such as bone absorption or collapse and sclerotic
changes, have been reported with the use of threadwire
In the cervical spine, wide surgical resections have been
attempted using techniques similar to total “en bloc” spondylectomy.
In a recent case report by Fujita et al,18 a chordoma was located on
one side of the C5 vertebral body (Figure 6). A special modification was made
to allow the threadwire saw to be hooked inside the pedicle. This modification
involved ligating the left vertebral artery followed by making troughs in the
vertebral body and endplates of C6 and C4. However, the use of the threadwire
saw resulted in an intralesional margin on the dura mater near the insertion of
the left C6 root because the tumor had invaded the neural foramen at that level.
Cohen et al52 described a technique for total
spondylectomy for osteosarcoma involving the cervical spine. The method
involved separately staged posterior and anterior approaches (Figure 7).
However, the resection described in this case was by definition intralesional.
Ligation of a vertebral artery may sometimes be inevitable to
avoid massive hemorrhage during tumor resection surgery. Although several
reports18,57-59 on this approach exist, because of its vital blood
supply and the possibility of other coexisting occlusive disease that might
affect the contralateral vertebral artery later in the future, the vertebral
artery should be preserved whenever possible. In a recent study,57
the vertebral artery was unilaterally ligated with no postoperative signs of
spinal cord, brain stem, or cerebellum ischemic dysfunction. Ligation of a
vertebral artery should be performed for safe margin tumor resection, only when
the tumor is located on the side of the minor or equal diameter of vertebral
artery, which should always be confirmed with preoperative angiography.
For the treatment of chordomas originating at the cranio-cervical
junction, the lateral transfacetal retrovascular approach has been reported
avoiding neuraxis retraction.60 This procedure is considered an
indication for anterior located vertebral body tumors with lateral extension
and vertebral artery invasion, with or without an intradural mass. It allows
for spinal cord decompression, C2 corpectomy without C1 or C2 hemilaminectomy,
and anterolateral reconstruction and posterior instrumentation. It is
considered restrictive when a spacious cephalad exposure is necessary, as in
In the cervicothoracic junction, tumor resection often is
troublesome because of the thoracic cage and adjacent neurovascular structures.
A combined cervicothoracic surgical approach of anterior cervical access,
median sternotomy, and anterior thoracotomy has been reported,61
resulting in gross tumor resection, with maintenance of the sternoclavicular
joint and sparing of the uninvolved neurovascular structures.
Wide excision or total en bloc cervical spondylectomy usually
results in spinal instability, thus spine reconstruction is necessary at the
level of the resection. Autografts (eg, iliac crest, fibula, or ribs),
allografts, and artificial spacers have all been used to achieve anterior spine
fusion in reconstructive surgery.52,53,62,63 Recently, structural
titanium mesh cages have been designed with varying available diameters and
heights, and great resistance and maintenance of spinal alignment for anterior
vertebral body structural support.53,64,65 Inside these cages,
autograft or allograft can be inserted for bone fusion.
Preoperative selective arterial embolization after spinal
angiography and adjuvant radiation therapy or chemotherapy can be performed as
indicated from the preoperative oncological and surgical staging of the
Postoperative adjuvant chemotherapy has been shown to be
effective in decreasing local recurrence and improving oncological outcome and
survival of patients with osteosarcoma or Ewing’s sarcoma of the cervical
spine. Wound lavage with a cis-platinum solution has been reported after
resection of osteosarcomas, although the merit of this practice is
|Figure 7: Total cervical spondylectomy
for osteosarcoma of the C6 vertebra. Illustrations showing the posterior
approach for resection of the posterior elements of C5 to C7, exposure of the
nerve roots and vertebral arteries bilaterally, transpendicular C6 partial
vertebrectomy, placement of a temporary spacer in the vertebrectomy defect,
segmental instrumentation and fusion using lateral mass screws (C3 to C5),
pedicle screws (T1 to T3) and cross-links. (A) Illustrations showing the
anterior approach for bilateral exposure of the vertebral arteries and nerve
roots, and en bloc resection from C5 to C7 vertebral bodies including the C5-C6
and C6-C7 intervertebral disks, and reconstruction with a titanium cage filled
with allograft and cervical plate from C4 to T1 (B). (Reprinted with permission
from Cohen ZR et al. Total cervical spondylectomy for primary osteogenic
sarcoma. Case report and description of operative technique. J Neurosurg Spine.
2002; 97:386-392. Copyright © 2002. American Association of Neurological Surgeons.)
Primary malignant tumors of the cervical spine are rare. Neck or
radicular pain is the most common initial symptom. Appropriate clinical and
radiographic evaluation of the patient, biopsy, oncologic and surgical staging,
and adequate operative techniques and adjuvant therapies can improve clinical,
oncologic, and functional outcome. Preoperative angiography and selective
arterial embolization, adjuvant radiation therapy, or chemotherapy can be
performed as indicated.
Surgery is the main treatment for most cervical spine malignant
tumors. En bloc resection with a wide surgical margin results in a better
prognosis. Total en bloc spondylectomy is a new, promising surgical technique
to allow complete tumor resection and improve clinical outcome; however it is
difficult to perform in the cervical spine. Ligation of a verterbal artery may
be inevitable to achieve safe surgical margins during excision of a cervical
spine tumor. Posterior alone or combined anterior and posterior surgical
approaches can facilitate complete tumor resection with low rates of local
tumor recurrence. Adjuvant irradiation or chemotherapy is indicated for certain tumor histologies.
From the First Department of Orthopedics, Athens University
Medical School, Athens, Greece; the Department of Orthopedics, Mayo
Clinic, Rochester Minn; the Department of Orthopedics and Traumatology,
University of Crete, Medical School Heraklion, Crete, Greece; and the Division of Medical Oncology, Mayo Clinic, Rochester Minn.
The authors have no industry relationships to declare.
Reprint requests: Panayiotis J. Papagelopoulos, MD, DSc, 4
Christovassili St, 154 51 N Psychikon, Athens, Greece.
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