High Long-term Local Control with Sacrectomy for Primary
High-grade Bone Sarcoma in Children
Alexandre Arkader MD, Christine H. Yang BA, MS,
Vernon T. Tolo MD
Received: 25 April 2011/Accepted: 11 November 2011/Published online: 3 December 2011
? The Association of Bone and Joint Surgeons1 2011
primary sarcomas of the sacrum. However, it is unclear
whether and to what extent sacrectomies achieve local
We therefore determined the curative
potential, walking ability, continence control, and quality
of life for children after sacrectomy for primary bone
We retrospectively reviewed eight children with
high-grade primary bone sarcomas treated with sacrec-
tomy. The average age of the patients was 14 years (range,
4–18 years). There were six Ewing sarcomas and two
Sacrectomy may offer curative potential for
osteosarcomas. Five patients underwent partial and three
underwent total sacrectomies with all resection margins
free of tumor. We recorded local and distant recurrences,
functional (Musculoskeletal Tumor Society [MSTS] score)
and oncologic outcome (survival), walking ability, bladder
and bowel control, independence, and reintegration in
society. The average followup was 80 months (range, 54–
Six patients were alive without evidence of dis-
ease at last followup. Three patients had metastases
develop and two died of disease 3 and 25 months postop-
erative. MSTS scores among survivors averaged 17.5
points (range, 5–25 points). Seven of the eight patients
were able to ambulate, two independently. All initially
were incontinent, but at last followup, two were continent
and two were incontinent only under stress. Complications
occurred in seven of eight, five of whom required addi-
Despite the high incidence of neurologic
deficits and complications, sacrectomy achieved local
control in patients with high-grade bone sarcomas. Our
observations suggest long-term survival and adequate
quality of life are possible in the pediatric population.
Level of Evidence
Level IV, therapeutic study. See
Guidelines for Authors for a complete description of levels
Ewing sarcomas and osteosarcomas are the most common
primary malignant bone tumors in children, most occurring
during the first two decades, with a slight male predilection
[15, 18]. Sacral tumors are rare, and account for approximately
6% of all spinal tumors in the pediatric population [19, 24].
Each author certifies that he or she, or a member of their immediate
family, has no commercial associations (eg, consultancies, stock
ownership, equity interest, patent/licensing arrangements, etc) that
might pose a conflict of interest in connection with the submitted
All ICMJE Conflict of Interest Forms for authors and Clinical
Orthopaedics and Related Research editors and board members are
on file with the publication and can be viewed on request.
The authors certify that their institution has approved the publication
of this study that all the investigations were conducted in conformity
with ethical principles of research, and informed consent was
This work was performed at Children’s Hospital Los Angeles,
Los Angeles, CA, USA.
A. Arkader (&), V. T. Tolo
Bone and Soft-Tissue Tumors Program, Children’s Orthopaedic
Center, Children’s Hospital Los Angeles; Keck School
of Medicine, University of Southern California, 4650 W Sunset
Blvd., Mailstop #69, Los Angeles, CA 90027, USA
C. H. Yang
College of Medicine, University of Illinois
at Urbana-Champaign, Champaign, IL, USA
Clin Orthop Relat Res (2012) 470:1491–1497
and Related Research®
A Publication of The Association of Bone and Joint Surgeons®
Although Ewing sarcomas and osteosarcomas are the most
common primary malignancies of the sacrum in children,
the lumbosacral region is the most commonly involved
[9, 10, 13, 17]. Other malignant tumors of the sacrum
include chordoma, Paget’s sarcoma, and multiple myeloma,
all rarely seen in children .
Current treatment protocols include preoperative and
postoperative chemotherapy and local surgical control .
Owing to the nonspecific nature of the symptoms (eg, night
or rest pain, neurologic symptoms, and/or the presence of a
mass), and lack of anatomic barriers or tumor growth into
the pelvic region, delayed diagnosis occurs often and the
tumor may reach a large size before diagnosis [21, 31].
When feasible, radical or partial sacrectomy may be
curative; however, this may result in nerve root sacrifice,
potentially causing major neurologic deficits and functional
compromise, and creating a large dead space susceptible to
infection and difficult to reconstruct.
Owing to the rarity of sacral tumors, the observations on
long-term function and survival after sacrectomy for high-
grade sarcomas are limited to case reports [22, 28], surgical
techniques [8, 22, 31], or studies reporting cohorts that
include but are not limited to children or high-grade sar-
comas [2, 8, 22, 24–26, 32]. The overall survival after
sacrectomy depends on the tumor type, presence of
metastasis, and quality of resection (margins). It varies from
approximately 20% to 80% at an average of 5 years [8, 24–
26, 31, 32]. Among the reported cases of sacrectomy per-
formed in children for primary high-grade bone sarcomas,
the surgery has the potential to be curative and has a high
complication rate, with overall survival of approximately
80% for patients with localized disease [5, 13, 16, 17, 20].
Based on an exclusively pediatric cohort of patients who
underwent sacrectomies for primary high-grade sarcomas
of the sacrum, we sought to answer the following questions:
(1) Can sacrectomy be curative for primary bone sarcomas
of the sacrum in children? (2) Is walking ability preserved in
children who have had a sacrectomy? (3) Are bowel and
bladder function preserved in children who have had a
sacrectomy? (4) Do children who have had a sacrectomy
maintain an adequate quality of life based on integration in
society and independence (work and studies)?
Patients and Methods
We retrospectively reviewed the charts of all eight patients
who underwent a sacrectomy for a high-grade primary
sacral bone sarcoma between 1987 and 2000. We excluded
patients (number unknown) who underwent sacrectomy for
pelvic tumors extending into the sacrum and patients with
soft tissue tumors, metastatic tumors to the sacrum, and
patients who did not initially undergo treatment at our
institution. The average age of the patients at presentation
was 14 years (range, 4–18 years); six patients were male
and two were female. All patients presented with pain,
usually in the lower back and leg, of an average of
4.1 months duration (range, 1–12 months). Histologically,
there were six Ewing sarcomas and two osteosarcomas
(Table 1). The average followup was 80 months (range, 3–
204 months). No patients were lost to followup. No
patients were recalled specifically for this study; all data
were obtained from records and imaging.
From a prospectively collected electronic musculoskeletal
tumor database we collected the following information:
demographics, tumor type, and location. From the medical
records we extracted specific information such as tumor
staging, resection level, margins, reconstruction procedures,
Table 1. Patient characteristics, staging, and treatment history
Gender Diagnosis Metastases
118MaleEwing sarcomaNoneIIBR = S1; L = S2None None
2 16Male OsteosarcomaLungIIIR = L3, S3; L = all Posterior spinal
fusion; bone graft
3 13.5 Female OsteosarcomaNone IIBR = S2; L = none Sacral/iliac wing allograftNone
4 13.5MaleEwing sarcomaLungIIIR = L4; L = S3 Cotrel-Dubousset spinal
5 13 MaleEwing sarcomaNone IIBR = S3; L = L4Cotrel-Dubousset spinal
fusion; bone graft
64 Female Ewing sarcomaLungsIIIR = S2; L = S1NonePelvis
7 16.5 MaleEwing sarcoma LungsIIINone Sacral/pelvic allograftLungs
8 16MaleEwing sarcomaNoneIIBR = L5; L = L5 Sacral allograftSkull
1492Arkader et al. Clinical Orthopaedics and Related Research1
functional outcome, neurologic function, (including motor
and sensory levels, ability to walk, and bladder and bowel
continence), disease-free survival, event-free survival, and
Before surgery, all children underwent staging with MR
imaging, CT scan of the chest, and Tc-99 m ethylene
diphosphonate bone scan. According to the staging system
of Enneking et al. , four patients had Stage IIB disease
and four had Stage III disease. The four patients with Stage
III disease presented with pulmonary metastases, all of
which responded to inductive chemotherapy. All patients
received neoadjuvant chemotherapy using a standard pro-
tocol. Two patients, both with Ewing sarcoma, received
radiation therapy. One patient (Patient 6) underwent radi-
ation therapy for pelvic metastases that occurred after
sacrectomy, and the other (Patient 7) underwent preoper-
ative radiation as surgery initially was refused.
Five patients underwent partial sacrectomies and three
had total sacrectomies; patients with tumors involving S1 or
S2 underwent reconstruction surgery after resection. The
with assistance by a pediatric general surgeon. Briefly, the
technique included an initial anterior approach for exposure
of the ventral surface of the sacrum by soft tissue mobiliza-
tion,which includedligationoftheanteriorsacralartery and
internal iliac vessels when necessary. At that time, all ante-
rior retroperitoneal structures were assessed for tumor
preoperative imaging and intraoperative findings, it was
performed accordingly at the level of the foramen or just
was performed with osteotomes and the space between the
with laparotomy pads. After anterior wound closure, the
patient was positioned prone on trunk rolls. The osteotomies
of the posterior sacrum, vertebrae, and iliac wing were
on preoperative imaging studies. Once the tumor was
removed and wide margins achieved, reconstruction was
performed when needed (Table 1). The average time for
surgery was 19 hours (range, 11–32 hours), and the average
blood loss was 7000 cc (range, 3000–14,300 cc). Primary
closure was possible in all but one patient (Patient 7) who
neededa flap. Wide surgical margins (based on inking of the
all patients (Fig. 1).
Postoperatively, all patients received appropriate pain
control and physical therapy. The physical therapy differed
if reconstruction was needed and if there was iliac
involvement. In general, mobilization started as soon as the
patient was comfortable and if there was no contraindica-
tion, the patients received gait training using an assistive
device. All patients received postoperative chemotherapy
and two (Patients 7 and 8) also received postoperative
We typically evaluated patients on the following sche-
dule: 2 weeks postoperatively, followed by 6 weeks,
3 months, then every 3 months for 1 year, 4 months
another year, and every 6 months subsequently. We eval-
uated function using to the MSTS criteria described by
Enneking et al. : the score varied from 0 to 30 (best
result) and incorporated the clinician’s evaluation for pain,
function, emotional acceptance, need for support, and
walking ability (0 to 5 points in each category). We sub-
jectively documented quality of life based on patients’
psychological and occupational status, integration in soci-
ety, and independence. Complications were recorded and
classified according to Dindo et al. . Minor complica-
tions were managed clinically and major complications
needed surgical intervention.
At last followup six of the eight patients were alive with no
evidence of disease, and two patients had died of distant
disease progression. Three of the patients had late meta-
static disease; two of these three had lung metastases at
presentation that initially responded to chemotherapy, but
one (Patient 7) refused postoperative chemotherapy and
eventually died of disease 25 months postoperative. The
other patient (Patient 6) had pelvic metastases develop
3 months after surgery, which was treated successfully
with chemotherapy and radiotherapy. Another patient
(Patient 8) had skull metastases develop during the post-
operative chemotherapy regimen and died of disease
3 months after surgery.
The average MSTS score was 17.5 (range, 5–25) for the
six surviving patients, whereas the two patients who died
had scores of 0 and 2 (Table 2). All but two patients
experienced some sensory loss postoperatively. Two
patients were able to ambulate independently, five required
the use of a cane or crutch, and one was wheelchair-bound
after attempted reconstruction failed and the patient had
Postoperative, all patients initially were incontinent,
resulting in urinary tract infections secondary to neuro-
genic bladder in five patients. At last followup, two patients
were completely continent, two experienced some leaking
or incontinence only under stress, and four remained
completely incontinent, managed with self-catheterization.
According to their self-assessment at the long-term clinic
assessment, all patients finished school and performed at a
nearly normal level, including working full time.
Complications occurred in seven of the patients
(Table 2). There were four minor complications (Grades I
Volume 470, Number 5, May 2012Sacrectomy for Children with Bone Sarcoma 1493
and II) that resolved with observation and conservative
treatment, and five major complications (Grades III, IV,
and V) .
Primary high-grade bone sarcomas of the sacrum are rare,
particularly in children. Although osteosarcoma and Ewing
sarcoma are the most common primary bone malignancies
in children, less than 10% of the cases involve the spine
and approximately half of those are in the sacrum [13, 14,
23]. The current treatment for osteosarcoma and Ewing
sarcoma include neoadjuvant and adjuvant chemotherapy,
with local control performed by surgical resection .
However, it is unclear whether and to what extent sacrec-
tomy with wide excision provides local disease control. In
this study we sought to answer the following questions
regarding the treatment of primary bone sarcoma of the
sacrum in children: (1) Can sacrectomy be curative?
(2) Can children retain their walking ability after sacrec-
tomy?; (3) Can they retain bowel and bladder function?
(4) Can they be an active part of the community and be
able to finish their studies or work?
The limitations of this study relate mostly to the rare
nature of the problem. First, we lacked a control group with
patients who did not undergo surgery (specifically Ewing
sarcoma is radiosensitive and therefore some centers may
recommend radiation as local control). Second, this study
was retrospective and therefore we had no preestablished
protocol for management of these patients; rather, this
treatment was on a case-to-case basis. Third, the cohort
was small; however, that reflects the rare nature of this
The disease-free survival rate after sacrectomy for high-
grade sarcomas is unclear. A sampling of studies in the
literature for disease-free survival gives an average of 51%
(range, 25%–67%) at an average of 58.9 months followup
Fig. 1A–D A16-year-old male presented with a 3-month history of
increasing pain in the lower back and right hip. (A) A CT scan of the
lumbosacral spine and pelvis showed an aggressive, ill-defined, lytic
lesion of the hemisacrum, sacrum ala, and ilium. After the biopsy,
osteogenic sarcoma was confirmed and the patient underwent
inductive chemotherapy, followed by surgical resection. (B) AP and
(C) lateral radiographs of the lumbosacral spine show the resection,
followed by instrumentation. (D) With time, compensatory scoliosis
developed owing to pelvic obliquity that did not need formal
treatment. (Images reproduced with permission from Children’s
Orthopaedic Center, Children’s Hospital Los Angeles, Los Angeles,
1494 Arkader et al. Clinical Orthopaedics and Related Research1
(range, 1–174 months) after sacrectomy for various
tumors, mainly chordomas [4, 6, 16]. Our patients’ disease-
free survival rate of 75% suggests that, specifically for
high-grade sarcomas in children, sacrectomy has the
potential to be curative (Table 3). However, chemotherapy
is critical in the treatment of Ewing sarcoma and osteo-
sarcoma [4, 23, 30], therefore sacrectomy without effective
chemotherapy most likely would be insufficient to achieve
disease-free status. The poorer oncologic outcome of the
two patients who died does not reflect a lack of efficacy of
sacrectomy, but rather tumor biology and external factors.
One patient waited more than a year after initial onset of
symptoms before seeking treatment at our institution
compared with an average duration of 3 months for the
other patients. By presentation, the patient already was
paraplegic and incontinent and elected to undergo surgery
as a last resort after chemotherapy and radiation therapy
failed. The second patient did not respond to neoadjuvant
chemotherapy as evidenced by the near zero-percent tumor
necrosis after resection with free margins.
Table 2. Functional and oncologic outcomes
(maximum 30 points)
Patient status Followup (months) Minor complicationsMajor complications
1 23/30 NED54 Gluteal muscle tearNone
2 18/30NED 100 NoneHardware failure and deep
wound infections leading
to sepsis; needed partial
3 5/30 NED96 None Chronic sacral decubitus,
4 17/30NED 204 Superficial wound
5 17/30NED55 Perianal skin breakdown,
6 25/30NED 116None None
7 0/30DOD25 NoneAcute arterial bleeding from
hypogastric remnant, deep
wound infection leading
8 2/30 DOD3 Superficial wound
Infected allograft, spinal
NED = no evidence disease; DOD = dead of disease
Table 3. Comparison of demographics and outcome*
Study Number of patients
younger than 18 years
age – years (range)
Number of primary
(at 5 years)
Ilaslan et al.#
Ilaslan et al.#
67 (N/A)19 (4–54)67 (EWS)60%
41 (N/A)27 (8–80) 41 (OGS)N/A
Kelley et al. 32 (20) 42 (7–76) Eight (OGS and EWS)N/A
Sar & Eralp  22 (5) 26 (17–63)Two (EWS and
One of two
Ozaki et al. 15 (N/A) 17 (5–55)15 (OGS)30%
Zileli et al. 11 (N/A)42 (14–71)Five (OGS and
Two of five
Wuisman et al.  Nine (N/A) 44 (17–70) Three (OGS and
Six of nine
Current studyEight (8) 14 (4–18) Eight (OGS and EWS)75%
* After sacrectomy for primary sarcomas of the sacrum in patients younger than 18 years at time of disease onset; studies included have at least
one or more pediatric patients with a primary sacral sarcoma; OGS = osteosarcoma; EWS = Ewing sarcoma;#= mostly descriptive/epide-
miologic study; N/A = not available.
Volume 470, Number 5, May 2012 Sacrectomy for Children with Bone Sarcoma 1495
Sacrectomy is a demanding and risky procedure. There
is a high incidence of postoperative complications,
including potentially permanent neurologic deficits in
sensation, motor capabilities, and bladder and bowel con-
tinence [1, 22, 25, 26, 29]. Neurologic compromise or
deficit is a well-known result after sacrectomy. In this
series, no patients experienced the saddle-like anesthesia
described in other series after complete sacral nerve root
sacrifice [5, 18]. Others have reported that approximately
90% of patients retain the ability to ambulate, with or
without external support, after sacrectomy [8, 12]. Our
results are similar to those findings.
Bladder and bowel incontinence often occur after
sacrectomy [1, 5, 16, 18]. Most patients with unilateral
preservation of the S2 nerve root are able to retain conti-
nence , but these cut-off points are not absolute and
there are exceptions . Accordingly, although all of our
patients had at least transitory bladder incontinency, at
followup, two patients who had retained the S2 nerve root
unilaterally had continence and one patient who retained
the nerve roots from S3 down unilaterally had bladder
incontinence but was able to retain bowel continence with
Despite the high risk of complications and enduring
neurologic deficits, sacrectomy is often the best definitive
treatment (eg, local disease control) for high-grade sacral
sarcomas [23, 31]. We achieved local control in all of our
patients. Furthermore, although all patients had some
degree of neurologic deficit, all six long-term survivors
continued their education and worked full time. Our
observations show that despite their neurologic deficits and
the associated psychological impact, these patients are not
only free of disease, but also active participants in society.
The rate of postoperative complications after sacrec-
tomy is high, ranging from 32% to 67%. Of these, the most
common is wound infection [8, 22, 24, 25, 27]. Our rate of
postoperative complications (88%) is higher than those
reported in some other studies [2, 8, 22, 25, 26, 31, 32],
although this may be attributable to our relatively small
sample size. Infection also was the most common com-
plication seen in this cohort. Of the three patients with no
or only minor complications, two of the three (67%) had
not undergone any reconstruction surgery; the other five
patients with major complications underwent complex
reconstruction surgery. Tumors located below the S2 nerve
root were resected without reconstruction . Only
patients with a resection level above the S2 nerve root,
either bilaterally or unilaterally, needed reconstruction
surgery [8, 25, 32]. Therefore, the need for reconstruction,
based on the exact level of the tumor, may be considered a
risk factor for development of complications.
Our study includes one of the largest, exclusively
pediatric, series of sacrectomies for primary sarcomas of
the sacrum. Based on our observations, we believe that
sacrectomy along with systemic treatment has the ability of
being curative. Children who undergo this procedure have a
high chance of retaining walking ability, bowel and bladder
control, and likely will reintegrate in society.
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