Aneurysmal Bone Cyst: A Review of 150 Patients

Abstract

We have reviewed a series of 150 aneurysmal bone cysts treated over the last 20 years.
The lesions were principally located in the tibia, femur, pelvis, humerus, and spine and, in most cases, presented the imaging appearance originally described by Jaffe and Lichtenstein as a blowout with thin cortices.
Only one of the patients was believed to have an osteoblastoma of the spine with secondary development of an aneurysmal bone cyst, and none of the patients developed additional lesions. The patients were treated primarily with curettage and implantation of allograft chips or polymethylmethacrylate, but some patients were treated with insertion of autografts or allografts. The local recurrence rate was 20%, which is consistent with that reported by other centers.
Aneurysmal bone cysts are enigmatic lesions of unknown cause and presentation and are difficult to distinguish from other lesions. Overall, the treatment is satisfactory, but it is possible that newer approaches, such as improved magnetic resonance imaging studies, may help diagnose the lesions and allow the physicians to plan for more effective treatment protocols.

Full text

Aneurysmal Bone Cyst: A Review of 150 Patients
Henry J. Mankin, Francis J. Hornicek, Eduardo Ortiz-Cruz, Jorge Villafuerte, and Mark C. Gebhardt
ABSTRACT
Purpose
We have reviewed a series of 150 aneurysmal bone cysts treated over the last 20 years.
Patients and Methods
The lesions were principally located in the tibia, femur, pelvis, humerus, and spine and, in
most cases, presented the imaging appearance originally described by Jaffe and Lichtenstein
as a blowout with thin cortices.
Results
Only one of the patients was believed to have an osteoblastoma of the spine with secondary
development of an aneurysmal bone cyst, and none of the patients developed additional
lesions. The patients were treated primarily with curettage and implantation of allograft chips
or polymethylmethacrylate, but some patients were treated with insertion of autografts
or allografts. The local recurrence rate was 20%, which is consistent with that reported by
other centers.
Conclusion
Aneurysmal bone cysts are enigmatic lesions of unknown cause and presentation and are
difficult to distinguish from other lesions. Overall, the treatment is satisfactory, but it is possible
that newer approaches, such as improved magnetic resonance imaging studies, may help
diagnose the lesions and allow the physicians to plan for more effective treatment protocols.
J Clin Oncol 23:6756-6762. © 2005 by American Society of Clinical Oncology
INTRODUCTION
Despite a descriptive history of more than
60 years, the nature, character, and optimal
treatment of aneurysmal bone cysts remain
obscure. The lesion was first described by Jaffe
and Lichtenstein
1
in 1942, was subsequently
further defined by both of these authors,
2,3
and became known as Jaffe-Lichtenstein dis-
ease. Despite attempts on the part of investiga-
tors to establish a relationship of the disorder
to other entities, the term aneurysmal bone
cyst remains purely descriptive. It does not
provide any concept of pathogenesis or causa-
tion mechanisms, and efforts on the part of a
number of investigators to discover a genetic
or neoplastic cause have failed.
4-18
Examina-
tion of the tissue at the time of surgery has, in
the past, demonstrated a blood-filled cavity
within an expanded region of the bone, and
the cells that line the cyst wall show fibrous
components, macrophages, giant cells, and is-
lands of bone.
1,7-9,19-24
The term aneurysmal
seems to relate to the blowout distension, and
the word cyst reflects the fact that the tumor
often presents as a blood-filled cavity.
7,9,25
Oc-
casionally in prior studies, there have been
findings suggesting the possibility that the an-
eurysmal cyst is actually a result of hemor-
rhagic degradative events occurring in patients
with other lesions including giant cell tumor,
hemangioma, chondroblastoma, osteoblas-
toma, nonossifying fibroma, fibrous dysplasia,
chondromyxoid fibroma, eosinophilic granu-
loma, and other tumors.
1,7,9,19-24
Of greater
concern is the possibility that the lesion is not
From the Orthopedic Oncology Service,
Massachusetts General Hospital and
Children’s Hospital, Harvard Medical
School, Boston, MA.
Submitted February 15, 2005; accepted
May 6, 2005.
Authors’ disclosures of potential con-
flicts of interest are found at the end of
this article.
Address reprint requests to Henry J.
Mankin, MD, Orthopedic Surgery,
Massachusetts General Hospital,
Boston, MA 02114; e-mail: hmankin@
partners.org.
© 2005 by American Society of Clinical
Oncology
0732-183X/05/2327-6756/$20.00
DOI: 10.1200/JCO.2005.15.255
JOURNAL OF CLINICAL ONCOLOGY
ORIGINAL REPORT
VOLUME 23 䡠 NUMBER 27 䡠 SEPTEMBER 20 2005
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an aneurysmal cyst but a partially necrotic and extremely
vascular telangiectatic osteosarcoma, which has a high rate
of metastasis.
7,9
Many reports have appeared that describe patients with
aneurysmal bone cysts.
3,6,8,14,16,22,24,27,28,31-36
The lesions
are more common in patients in the first two decades of life
rather than in later years
7,8,28,31,37
and seem to be slightly
more frequent in females than males.
7,8,28-30
The major sites
of occurrence, according to a number of authors, are the
femur, tibia, humerus, spine, and pelvis, and although tu-
mors arising in the small bones of the hands and feet are
seen occasionally, there are fewer of such instances in most
series.
3,7,9,14,16,24,32-36
The tumors are usually metaphyseal or
diaphyseal and are most often eccentrically located, which is an
important distinguishing radiographic feature from unicam-
eral bone cyst in patients in their second decade.
7-9,12,20,24,38,39
The appearance of the lesions support the word blowout and
show marked thinning of the cortex over the site, with only
minimal bone formation; all of which are sometimes best seen
on a computed tomography (CT) or a magnetic resonance
imagining (MRI) scan.
38,39
Controversy exists regarding opti-
mal treatment, and regardless of techniques reported, there
remains a recurrence rate that ranges from 5% to greater than
40%.
7,24,28-34
At present, curettage and insertion of bone graft
or polymethylmethacrylate are the principal techniques
used,
7,20,24,29,31,33,34
but in the past, radiation has been used.
40
In several trials, sclerosing substances, bone substitutes, a n d
other agents seemed to be less effective than conventional
curettage.
41-43
Our purpose in presenting this material is to re-
view the data obtained over the last 20 years regarding patients
Fig 1. An x-ray of an aneurysmal cyst arising in the proximal humerus in a
child. Note the irregularity of the cortices and the expansion of the bone. The
tumor was painful not only because of its structure but also because of a
small pathologic fracture on the lateral side.
Table 1. Demographic Data for 150 Patients With Aneurysmal Bone Cyst
Characteristic No. of Patients
Age, years
Mean 18
Standard deviation 12
Range 3-62
Sex
Male 69
Female 81
Follow-up, years
Mean 6
Standard deviation 7
Range 1-20
Anatomic sites
Tibia 37
Proximal 25
Middle 5
Distal 7
Femur 26
Proximal 9
Middle 4
Distal 13
Fibula 16
Proximal 7
Middle 2
Distal 7
Pelvis 13
Humerus 10
Proximal 7
Middle 2
Distal 1
Clavicle 10
Foot 8
Ulna 5
Proximal 1
Middle 3
Distal 1
Lumbar spine 5
Cervical spine 3
Scapula 3
Sacrum 2
Ribs 2
Dorsal spine 1
Aneurysmal Bone Cyst: Recurrence and Outcome
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with aneurysmal bone cysts to define the appearance, anatomic
site, complications, and results of various forms of treatment.
PATIENTS AND METHODS
The Orthopedic Oncology Service at the Massachusetts General
and Children’s Hospitals has maintained a computerized database
containing information regarding more than 17,000 patients with
bone and soft tissue tumors treated over the last 30 years.
44
Study
of the system provided information regarding 223 patients with
aneurysmal bone cyst, but only 150 of these patients who had been
observed for as long as 20 years had sufficiently accurate descrip-
tions of treatment protocols, definition of pathology, and recent
assessment of outcomes to allow statistical analysis (Table 1). The
system used to gather information regarding the patients did not,
in any way, violate patient confidentiality and was approved by the
hospitals’ institutional review boards.
Sixty-nine of the 150 patients were male, and 81 were female.
The mean age of the patients was 18 ⫾ 12 years, with a range of 3
to 62 years. The mean follow-up time for the patients was 6 ⫾ 7
years (range, 1 to 20 years). The anatomic locations for the lesions
are listed in Table 1, and as noted, the most common sites were the
tibia, femur, fibula, pelvis, humerus, clavicle, foot, and lumbar
spine. The patients’ complaints at the time of the initial visit were
almost always concerning pain at the site and sometimes indicated
pain and numbness extending down the limb. The patients were
often disabled by the pain and almost always presented with ten-
derness over the site of the lesion. Examples of the imaging studies
are shown in Figures 1 and 2, and as noted, the lesions are metaph-
yseal or diaphyseal in location, are usually eccentric with thin
cortices, and, in most cases, present the classic blowout appear-
ance (Fig 3). Gross structure is shown in Figure 4, which displays
the thin cortices, the expansion, and the chambers often filled with
blood. The histologic patterns are shown in Figures 5 and 6. The
patterns demonstrate thin cortices; blood elements in the central
portion of the tumor; and an array of benign-appearing macro-
phages, lymphocytes, fibroblasts, bone-forming cells, and giant
cells, which are sometimes atypical in terms of structure and
nuclear distribution.
As shown in Table 2, 130 patients were treated with curettage,
and of these patients, 101 had lesions that were packed with
allograft bone, and 20 had lesions that were packed with polym-
ethylmethacrylate cement. Twenty of the patients had an excision
or resection of the lesion, and 11 of these patients received an
intercalary allograft transplantation.
Statistical studies used analysis of variance and Mantel-
Haenzel and Fisher’s exact tests using
␹
2
analysis. The systems
were provided by BMDP Statistical Software (Los Angeles, CA).
P ⬍ .05 was considered statistically significant.
RESULTS
None of the patients died of disease, and there were no
amputations. The principal problem that the patients en-
countered with their treatment of the aneurysmal bone
cysts was local recurrence, which occurred in 30 (20%) of
the 150 patients at 1.2 ⫾ 0.7 years (range, 0.3 to 3 years) after
discovery of the lesion. There was no statistical difference
for sex. Patient age did not seem to have a significant effect
on outcome, although the rate of local recurrence was
slightly increased in younger patients (Table 3). Anatomic
site seemed to make some difference in rates of local recur-
rence. The recurrence rate for the 10 patients with lesions
of the clavicle was 50%, and the recurrence rate for the 13
Fig 2. An enormous aneurysmal bone
cyst of the proximal humerus. Note the
size of the lesion and the blowout appear-
ance with thin cortices and periosteal new
bone on the shaft. Despite the suggestion
that this was a giant cell tumor, the histology
was classic for an aneurysmal bone cyst.
The lesion was curetted out, and a segment
of allograft radius was inserted (B).
Mankin et al
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patients with tumors in the distal femur was 46%, whereas
other sites showed few or no recurrences (Table 3).
Of considerable interest was the recurrence rate data
for the various operative procedures performed by our
group over the 20⫹ years of experience in the treatment of
patients with aneurysmal bone cysts. The recurrence rate
for the 121 patients who were treated with curettage and
packing with either allograft chips or polymethylmethacry-
late was 22%, which was considerably greater than the 5%
recurrence rate for patients who underwent resection and
either an autograft or an allograft implantation (Table 2).
The patients who developed a recurrence required sub-
sequent surgery, which consisted of another curettage and
packing or resection of the site and autograft or intercalary
allograft implantation. Of the 150 patients, 34 required a
second operation and 13 required a third operation for
recurrences or structural problems related to failed systems.
One of the patients was treated with sclerotherapy, and two
patients were treated with local injections of alcohol and
other agents, all of which were successful. Three of the
patients developed fractures through the weakened bony
part, which, although not related to recurrence, required
Fig 3. A typical site and structure for an
aneurysmal bone cyst of the distal tibia in a
12-year-old female patient. The eccentric
location and the marked thinning of the
cortex are characteristic.
Fig 4. Classic gross appearance of an aneurysmal bone cyst of a rib. The
blood-filled chamber is irregular in structure, and there are islands of bone
and fibrous tissue. The bone is expanded and irregular in shape with the
typical blowout appearance.
Fig 5. Histologic appearance of the margin of the tissue from the rib lesion
shown in Figure 4. The bone is irregular in structure, and a fibrous layer
separates it from the region of the blood-filled cavity.
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further surgery. The 11 allograft implantations were all
rated as good or excellent at 6 ⫾ 6 years (range, 1 to 20
years), and only one of the grafts sustained a fracture, which
healed after replacement of the fixation. Only one of the
patients in our series was found to have tumor suggestive of
an osteoblastoma of L4 at the time of biopsy. The lesion was
curetted and packed with allograft chips, and there has been
no recurrence reported to date.
Fifty-seven of the 150 patients had the DNA content of
the tissue obtained at surgery studied by flow cytometric
analysis.
45
The mean value for the diploid peak was 92 ⫾ 4.3
(range, 86 to 98), and the mean value for the G2 ⫹M was
4.7 ⫾ 3.2 (range, 0. to 14). The average mean DNA index
was 1.06 ⫾ 0.04 (range, 1 to 1.2). No aneuploid peaks were
encountered. These values can be interpreted as being char-
acteristic of benign tumors.
45
DISCUSSION
As stated in the Introduction, despite the long experience of
radiologists, pathologists, and orthopedists with aneurysmal
Table 2. Recurrence Rate for 150 Patients According to Various Parameters
Parameter
No. of
Patients
Recurrences Proximal Middle Distal
No. %
No. of
Patients
No. of
Recurrences
No. of
Patients
No. of
Recurrences
No. of
Patients
No. of
Recurrences
Sex
ⴱ
Male 69 13 21
Female 81 21 19
Patient age
ⴱ
⬍ 10 years 29 7 24
10-20 years 76 16 21
⬎ 20 years 45 7 16
Anatomic site
Tibia 37 4 11 25 3 5 0 7 1
Femur 26 9 35 9 1 4 2 13 6
Fibula 16 4 25 7 1 2 0 7 3
Pelvis 13 2 15
Humerus 10 2 20 7 2 2 0 1 0
Clavicle 10 5 50
Foot 8 2 25
Ulna 5 1 20 1 1 3 0 1 0
Lumbar spine 5 0
Cervical spine 3 1 33
Scapula 3 0
Sacrum 2 0
Ribs 2 0
Dorsal spine 1 0
NOTE. Recurrence rate: 30 recurrences (20%) at 1.2 ⫾ 0.7 years (range, 0.3 to 3 years).
ⴱ
Not significant.
Fig 6. A high-power view of the tissue shown in Figure 5 showing the
cellular components, which include fibroblasts and many monocytic cells
along with blood cells. Several giant cells are present, but the appearance of
the tissue does not suggest a giant cell tumor.
Table 3. Effect of Treatment on Recurrence Rate
Treatment Method
No. of
Patients
Recurrences
No. %
Curettage and pack with allograft chips 101 21 21
Curettage and pack with PMMA 20 5 25
Resect or excise and autograft implantation 9 1 11
Biopsy, curettage, and no implantation 9 3 33
Resect or excise and allograft implantation 11 0 0
Abbreviation: PMMA, polymethylmethacrylate.
Mankin et al
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bone cysts, there is limited knowledge regarding the cause
of the lesion, its natural history, and the results of treat-
ment.
1,4,5-7,9-17
The concept that the lesion represents a
vascular degenerative process for some benign bone lesions
is an attractive one, but the pathologic findings, with rare
exception, do not really support this proposal. Few pathologic
specimens contain tissues that are highly characteristic or di-
agnostic of giant cell tumor, chondroblastoma, hemangioma,
osteoblastoma, nonossifying fibroma, fibrous dysplasia, chon-
dromyxoid fibroma, and other tumors.
6,7,9-11,15-17,19,21,26,28,29
Furthermore, the recurrences after surgical treatment do not
show evidence of such lesions, particularly aggressive lesions
such as giant cell tumor. Only one of our 150 patients showed
a finding suggestive of an osteoblastoma at the site of the
aneurysmal bone cyst of L4. At best, it was a difficult decision
based on the similarity of the repair process to the histologic
pattern for osteoblastoma, and the lesion has not recurred at
more than 2 years since the surgery.
Of some importance is the difficulty that can occur in
diagnosing these lesions. The imaging studies, even CTs and
MRIs, sometimes do not provide clearly diagnostic criteria
for the diagnosis of aneurysmal bone cyst, and aneurysmal
bone cyst is sometimes added on to a list of diagnoses
including eosinophilic granuloma, giant cell tumor, nonos-
sifying fibroma, unicameral bone cyst, fibrous dysplasia,
chondroblastoma, chondrosarcoma, chondromyxoid fi-
broma, Ewing’s tumor, and, in older patients, metastatic
carcinoma or myeloma.
7-9,12,20,24,38,39
The lesions are often
eccentric and irregular in structure and sometimes show
calcification in the central areas. As a rule, the cortex is thin,
but there is rarely a cortical defect or a soft tissue mass. CT
and MRI are often helpful in defining the extent of the
lesion and establishing the diagnosis. A biopsy is often
helpful, and many of our patients underwent a needle bi-
opsy before definitive treatment. Needle biopsies are some-
times a problem because the material obtained may consist
of mostly blood elements. Often, an open biopsy and frozen
section are necessary to establish the diagnosis.
The recurrence rate in this series was 20%, which
should be considered high compared with other series and
other benign tumors.
7
Part of this problem could conceiv-
ably be related to the methods of treatment over the 20 years
during which these patients were treated. The recurrence
rate for patients treated in the earlier years was higher than
for patients who were treated more recently (approximately
26% v 17%, respectively). In the past, we used curettage
alone, but our principal current approach was biopsy fol-
lowed by curettage and then implantation of allograft chips
or, more recently, polymethylmethacrylate. Autograft im-
plantations or utilization of intercalary allografts were quite
successful but were, for the most part, used for patients with
lesions that were large or seemed to threaten the integrity of
the bone and were used less frequently for patients who
experienced failure of their primary procedure.
Although none of our patients died or required an
amputation, a number of them had some relatively minor
disability as a result of the tumor and its treatment. Thus, it
is our opinion that aneurysmal bone cyst is sometimes an
aggressive lesion that is difficult to treat. Lesions that occur
in the proximal femur should perhaps be treated more
aggressively, partly because of the high rate of local recur-
rence and the risk of fracture. The most appropriate tech-
niques for some of these tumors are primary resective
surgery and allograft implantation. Patients with lesions of
the proximal or mid fibula or clavicle or body of the scapula
could be treated by resection alone, and lesions of the foot
might be best treated by resection and arthrodesis using
autograft. According to our series, such treatment would at
least reduce the local recurrence rate considerably and
probably reduce the degree of even minimal disability re-
ported by some of these young patients.
In this last analysis, aneurysmal bone cyst remains an
enigma, not only regarding causation, but also regarding
clinical and imaging diagnosis and optimal treatment.
There is perhaps some hope for implantation of newer
agents, such as the bone substitutes, to aid in healing of the
lesions. Although there are now some markers that are
alleged to be specific for aneurysmal bone cyst,
1,9,12-15,18
there is still no system to establish the diagnosis or to sup-
port different methods of treatment and, thus, reduce the
problems encountered by the patient and the surgeon.
■■■
Authors’ Disclosures of Potential
Conflicts of Interest
The authors indicated no potential conflicts of interest.
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