Unicameral Bone Cysts

Ross M. Wilkins, MD, MS

Abstract

Unicameral, or solitary, bone cysts are unusual tumors seen in the ends of long history of previous fracture, but for
bones in skeletally immature persons. The etiology of these lesions is poorly any single cyst, these factors are
understood. Various hypotheses have included dysplastic processes, synovial unreliable.
cysts, and abnormalities in the local circulation. Most patients present with a
nondisplaced pathologic fracture, but occasionally cysts are found incidentally.
Plain radiographs typically show a symmetric lesion with cortical thinning and Pathogenesis
expansion of the cortical boundaries. Once diagnosed, unicameral bone cysts con-
tinue to be a treatment dilemma. Traditional methods, such as prednisolone ther- Various mechanisms have been
apy, usually involve multiple anesthetics and injections and are associated with proposed for the pathogenesis of
high recurrence rates. Major surgical procedures, such as wide exposure, curet- unicameral bone cysts. On the
tage, and bone grafting, may be somewhat more effective, but still carry with them basis of electron-microscopic find-
significant morbidity and recurrence rates. Newer techniques involving percuta- ings, Mirra et al3 considered them
neous grafting with allograft or bone substitutes or a combination of the two are to be intraosseous synovial cysts.
promising in light of their low complication rate and lower reoperation rate. Jaffe and Lichtenstein 4 observed
J Am Acad Orthop Surg 2000;8:217-224 dysplastic areas, which they be-
lieved developed in response to
trauma. Cohen 5,6 hypothesized
that the cyst forms as a response to
Unicameral bone cysts have been these lesions, which represent about venous occlusion in the intramed-
recognized for many years as benign 3% of biopsied bone tumors, is elu- ullary space.
lesions; however, they remain trou- sive. They usually present with a Histologic examination of these
blesome in regard to their diagnosis pathologic fracture (Fig. 1). Such lesions has been relatively unre-
and treatment. Virchow originally fractures occur through thin, weak- warding in regard to their patho-
described these lesions in 1891 as ened bone and are generally not genesis. Generally, the cyst walls
“cystic structures,” which he hypoth- grossly displaced, nor are they diffi- are lined with a fibrous membrane,
esized occurred as abnormalities in cult to treat. The cysts have a pre- with occasional giant cells. There is
the local circulation. Unicameral dilection to occur in males more fre- no evidence of endothelial lining.1
bone cysts are also known as soli- quently than in females (2.5:1). Most It has been proposed that there are
tary, or simple, bone cysts. Multi- patients (reportedly as many as 85%) synovial cells in the lining, resem-
loculated bone cysts are usually are under 20 years of age.1
included in this category. Since the Once diagnosed, unicameral
first description of these cysts, many bone cysts continue to be a dilemma
authors have presented series detail- for the clinician because the natural Dr. Wilkins is Assistant Clinical Professor of
ing the clinical characteristics and history and management remain Orthopaedics, University of Colorado School of
diagnostic features of this disease controversial. Historically, recur- Medicine, Denver.
entity and discussing treatment rence rates have ranged from 20%
modalities. to 50% after the various forms of Reprint requests: Dr. Wilkins, Institute for
Limb Preservation, Denver Orthopedic
Unicameral bone cysts are be- treatment.2 Classification of indi-
Specialists, PC, Suite 5000, 1601 East 19th
nign, fluid-filled cavities that tend to vidual lesions in a way that pre- Avenue, Denver, CO 80218
expand and weaken the local area. dicts their natural history has been
They are generally seen in the metaph- difficult. Various authors have at- Copyright 2000 by the American Academy of
yseal areas of long bones in skeletal- tempted to determine prognosis on Orthopaedic Surgeons.
ly immature persons. The etiology of the basis of patient age, site, size, or

Vol 8, No 4, July/August 2000 217

Unicameral Bone Cysts

bling the type A and B cells seen in strated the role that these factors ing the interstitial pressure by mul-
synovial tissue.3 The fluid within may have by showing that injection tiple perforations may cause cyst
the cyst has been analyzed and of cyst fluid into mouse bone involution.11
shown to contain high levels of caused bone resorption.
oxygen-free-radical scavengers and More recent research supports
prostaglandins (prostaglandin E2, the theory that a vascular occlusion Clinical Features
interleukin-1, and proteolytic en- phenomenon occurs within the
zymes).7 These substances, which cyst.10 The pressures within a cyst The symptoms of unicameral bone
cause bone resorption, may play a are elevated above venous pres- cysts are most often brought on by
role in the formation and growth of sures. It appears that if radiopaque trauma. On examination, the area is
cysts. The fluid appears to be inter- dye is injected into the cyst with slightly warm and swollen. Radio-
stitial fluid transudate or exudate. enough pressure, the dye can be graphs usually reveal a nondis-
The cyst fluid has a lower total pro- extruded into the venous system of placed or minimally displaced frac-
tein content than serum but higher the limb. Reestablishing these out- ture through an area of very thin,
levels of protein-bound hydroxy- flow channels may assist in the expanded cortical bone. Occasion-
proline, lactate, and alkaline phos- involution of the cyst. 10 Others ally, a fragment of the cyst wall has
phatase.8,9 Komiya et al7 demon- have proposed that simply lower- fractured and fallen into the fluid
cavity. This is evidenced by the
radiographic “fallen leaf sign” 12
(Fig. 2).
The differential diagnosis in-
cludes aneurysmal bone cyst and
fibrous dysplasia. When additional
studies are warranted, magnetic
resonance imaging most accurately
delineates the central fluid collec-
tion. If a pathologic fracture has
occurred, a fluid level may be visu-
alized, mimicking the appearance
of an aneurysmal bone cyst. There
is no convincing evidence, however,
that a unicameral bone cyst will
convert to an aneurysmal bone cyst
or other bone lesion.
Unicameral bone cysts usually
occur in younger patients. The
most common site is the femur, fol-
lowed by the proximal humerus.
At diagnosis, many cysts are imme-
diately adjacent to, and appear to
Admission 8 wk postop 3 yr postop
involve, the epiphyseal growth
plate, which supports the theory
A B C
that this is a growth disturbance
Figure 1 A, A 9-year-old boy complained of pain in his right arm subsequent to a fall rather than a true tumorous pro-
while running. A pathologic fracture through the cystic area in his proximal humerus was cess. Minor growth disturbances
barely discernible on this anteroposterior radiograph. The patient was immobilized, and
healing was allowed to occur. The patient subsequently underwent percutaneous injection, occasionally occur (10% of cases in
by means of a two-needle technique, of a mixture of demineralized bone matrix and autolo- one study 13). When fractures do
gous marrow aspirate from his iliac crest. Three weeks postoperatively, healing of the frac- become evident, they rarely in-
ture and some opacification of the cystic areas were demonstrated. The superior cortical
perforation was made with a 5-mm cannulated needle device; the inferior perforation was volve the growth plate itself.
made with a bone-marrow aspiration needle. B, Cortical hypertrophy and further opacifi- It is the opinion of many authors
cation of the cystic area were noted 8 weeks postoperatively. The patient was released to that the proximity of a cyst to the
normal activities. C, Radiolucency persisted at 3 years postoperatively; however, the thick-
ness of the cortical margins was maintained, and a slightly increased diameter of the diaph- growth plate and its size at diagno-
ysis was evident. The patient was asymptomatic and participated in contact sports. sis are directly related to the prog-
nosis.2 Diaphyseal cysts do occur

218 Journal of the American Academy of Orthopaedic Surgeons


Figure 2 A 9-year-old boy fractured his
proximal humerus playing baseball. The
“fallen leaf sign” (arrow) is occasionally
seen in fractures through unicameral bone
cysts. The cortical fragment becomes dis-
lodged from the margin at the time of frac-
ture and literally floats to the bottom of the
cystic structure.
and can fracture. Others have pro-
posed that patient age and patho-
logic fracture are directly related to
the future problems that may be
encountered.14,15 It is evident that
these cysts progress from active to
quiescent to an involutional stage
in the course of their natural his-
tory. The difficulty for the clinician
is to assess the current stage of the
cyst at the time of diagnosis. Treat-
ment of an active cyst may be
unsuccessful, whereas treatment of
a quiescent or involutional cyst
may be successful but unnecessary.
The cyst usually progressively
shrinks as the patient approaches
skeletal maturity and may heal
spontaneously after growth is com-
pleted.16
Vol 8, No 4, July/August 2000
Ross M. Wilkins, MD, MS
Treatment In light of this statistic, the consen-
sus is that the surgeon should allow
Indications the cyst to heal before proceeding
There are two basic scenarios in with treatment. By waiting, inter-
which a unicameral bone cyst is nal fixation can usually be avoided.
diagnosed. Occasionally, the cyst is The exception is when the fracture
discovered incidentally during is in a high-stress weight-bearing
investigation for another complaint. area, such as the femur.
Usually, however, the cyst is symp- Surgical treatment of UBCs re-
tomatic or is associated with a mains controversial. Suggestions
pathologic fracture (68% of cases in range from a hemicylindrical subto-
one study15) (Fig. 1). tal resection18 to a saline injection.10
In the first situation, it is often Interpreting reported clinical series
difficult to decide whether the cyst presents a dilemma because it is
is in the active, latent, or involutional virtually impossible to ascertain
stage. The mere size of the cyst it- whether the cysts in these studies
self is probably of less importance were active, latent, or involutional
than the structural properties of the when they were treated. Few stud-
area. The strength of a cylinder is ies have reported a clear and pre-
proportional to the square of its cise set of criteria for treatment.15
diameter. Therefore, unless there is
a tremendous amount of cortical
thinning, there may not be a compa-
rable decrease in strength as a cyst
expands the cortical margins. If a
cyst is discovered incidentally in an
asymptomatic patient, it may be
reasonable to choose close observa-
tion rather than a surgical proce-
dure. If the cyst is active and obvi-
ously enlarging during observation
(3 to 6 months), treatment may be
appropriate. If, however, a cyst
remains asymptomatic and the
patient is able to maintain normal
activities, continued observation is
warranted, because the cyst may
eventually resolve on its own. One
exception to this guideline is when a
large cyst involves the subtrochan-
teric region of the femur. Early
treatment may be needed to avoid
fracture due to the high forces to
which that area is normally subjected.
A cyst that is symptomatic has
A B
an incompetent osseous structure
and has undergone either an obvi- Figure 3 A, Anteroposterior view of a uni-
ous or an undetected pathologic cameral bone cyst in the proximal fibula in
an 8-year-old boy who had sustained a
fracture. Some authors have sug- pathologic fracture while running. B,
gested that such a cyst will then Cortical thickening and partial obliteration
undergo an involutional process of the space was seen at 3 months. The
patient returned to normal activities, and
and heal (Fig. 3). However, in observation was continued. He was asymp-
closely observed series, this occurs tomatic 3 years after the fracture.
less than 10% of the time17 (Fig. 4).
219
Unicameral Bone Cysts

A B C D E

Figure 4 A, A unicameral bone cyst in a 14-year-old boy who sustained a pathologic fracture while throwing a baseball. B, At 6 months,
the cyst appeared to be active, and healing was delayed at the fracture site. The patient had continued symptoms. C, Fluoroscopic view
shows a pituitary rongeur being inserted to obtain a percutaneous biopsy specimen by means of a cannula system. D, With the diagnosis
of unicameral bone cyst confirmed on frozen section, the area was grafted percutaneously with calcium sulfate pellets and demineralized
bone matrix. E, The area of the cyst and fracture showed cortical hypertrophy at 3 months postoperatively, and the calcium sulfate pellets
had been resorbed. The patient resumed his activities in competitive baseball as a pitcher and remained asymptomatic.

Injection Techniques
In an early paper, Scaglietti 19
described the technique of injecting
methylprednisolone into unicameral
bone cysts. He reported successful
healing of the cyst with much less
morbidity than with resection or
curettage plus bone grafting. The
procedure involves injecting meth-
ylprednisolone into the cyst under
fluoroscopic control while using
radiopaque dye to confirm entry
into the cyst. Aspiration of the cyst
is done prior to injection. The
return of clear, straw-colored fluid
is confirmatory of the diagnosis. If
grossly bloody fluid is encountered,
a formal biopsy is advised to ascer-
tain whether the lesion is an aneu-
rysmal bone cyst or another type of
lesion. The cyst is then flushed with
saline, and methylprednisolone is
injected with either a one-needle or
a two-needle technique. The two-
needle technique allows efflux of
the saline and excess fluid through
an outflow needle in another region
of the cyst.
Although in principle this proce-
dure would seem to be advanta-
geous by decreasing the morbidity
due to a major surgical procedure,
unfortunately it has not proved to
be very effective and usually in-
volves multiple injections and anes-
thetics.19 Overall, a review of the
literature revealed recurrence rates
of 15% to 88% after an average of
three injections. 20-22 It is unclear
what effect the methylprednisolone
actually has on the local anatomy.
Some authors advocate using only
normal saline, because they feel
that the mechanical disturbance of
the injection is the important factor,
rather than the agent itself.10 Other
authors also recommend that multi-
ple percutaneous perforations be
made in the cyst to normalize its
local circulation and disrupt any
venous obstruction.11
Surgical Techniques
Resection or curettage plus bone
grafting has been employed as the
definitive treatment for unicameral
bone cysts. However, in published
series, the recurrence rates have
ranged as high as 45%.2,18 The high
rates of recurrence may be due to the
fact that surgically treated cysts are
more active, more aggressive, and
likely to be recurrent. Active uni-
cameral bone cysts tend to be treated
more aggressively from the outset.
Most published series are reports
from tertiary referral centers, where
the patient populations have cysts
with a more difficult clinical course.

220 Journal of the American Academy of Orthopaedic Surgeons


The technique of resection or
curettage is relatively straightfor-
ward. Once an approach has been
made to the bone, a cortical win-
dow is made, which allows access
to the entire contents of the cavity
(Fig. 5). The clear fluid should be
removed, and the fibrous mem-
brane curetted from the cyst wall.
If the cyst is immediately adjacent to
or involves the epiphyseal growth
plate, care must be taken to avoid
injury to the plate. It is not neces-
sary to remove structural bone from
the outer cyst wall. It is also not ne-
cessary to use adjunctive materials,
such as phenol or liquid nitrogen,
to perform this procedure. Such
materials have secondary complica-
tions and may interfere with graft
and bone healing.15 Furthermore,
A B
D E
Vol 8, No 4, July/August 2000
to employ these toxic and damag-
ing agents is to imply that the local
cells are the primary etiologic fac-
tors. There is no evidence to sup-
port this hypothesis. Neer et al 2
found no difference in the rate of re-
currence when phenol was used,
and Schreuder et al15 reported a 5%
rate of complications related to the
use of liquid nitrogen.
The choice of autologous bone
graft or a substitute is dependent on
the orthopaedic surgeon’s prefer-
ence. Autologous bone marrow,
allograft, demineralized bone matrix
(DBM), and other bone substitute
materials have been used success-
fully, thus sparing the patient the
morbidity of an autograft harvesting
site.23,24 Allograft bone chips have
proved effective in the treatment of
Ross M. Wilkins, MD, MS
cysts15,25,26 (Fig. 5). Calcium sulfate
in the form of plaster of paris has
been used with a good success rate
and a low recurrence rate (11%).23
The injection of autologous bone
marrow is also effective, but this
procedure requires several injec-
tions14,27 (Figs. 1 and 6). Deminera-
lized bone matrix, an osteoinductive
material, can be injected percuta-
neously into cysts28 (Figs. 1 and 6).
In the author’s series of 11 patients
who received one percutaneous
injection of DBM, no further treat-
ment was required in any case after
2 years of follow-up, and 9 of the 11
cysts were entirely obliterated.
An alternative technique in-
volves the use of a combination of
DBM (AlloGro, AlloSource, Den-
ver) and calcium sulfate (OsteoSet,
C
Figure 5 A, Anteroposterior radiograph
of a 21-year-old man with a stress fracture
through a cystic lesion in his left femoral
neck. B, Computed tomogram through
the lesion. C, Photomicrograph of a biop-
sy specimen of a unicameral bone cyst
(original magnification ×225). Note the
bland-appearing fibrous tissue and occa-
sional giant cells. D, Anteroposterior
view of the hip after the area was curetted
and grafted with demineralized bone
matrix and cancellous chips. Cannulated
screws were placed to stabilize the area
and prevent fracture. E, Follow-up film at
6 months shows opacification in the area
of the lesion. The patient had resumed all
activities, including sports and running.
221
Unicameral Bone Cysts

wise healthy children, the initial

fracture through a unicameral bone
cyst can usually be weathered with-
out much difficulty. However,
with repeated fractures, the psycho-
logical consequences for young
patients and their families can be
significant. The children become
tentative in their daily activities,
because of either their own fear of
fracture or that of their parents.
Occasionally, family members be-
come obsessed with the “fragility”
of the child, which may drastically
alter the young patient’s lifestyle
and attitudes. Whichever treatment
modality is utilized, it should be the
A B C D procedure that will return the pa-
tient to early normal activities and
Figure 6 A, Lateral view of the leg of an 11-year-old girl in whom a fracture through a cyst sufficiently heal the area so that
occurred while she was doing cartwheels. She was treated conservatively with cast immo-
bilization. B, Cortical thickening and remodeling were demonstrated 8 months after the there is no further concern about
fracture, but the cyst persisted and appeared to be enlarging. The patient was asympto- fracture with minimal trauma.
matic, and observation was continued. She was allowed to maintain normal activities.
C, At 28 months after the original fracture, the patient was having pain once again during
cheerleading activities. Radiograph shows partial involution of the proximal cystic area but
continued growth of the distal area. Percutaneous grafting was performed with the use of a Summary
mixture of DBM and autologous marrow aspirated from her iliac crest. D, Radiograph
obtained 2 years postoperatively shows cortical thickening and involution of the cystic
areas. The patient continued her cheerleading activities without pain or problems. Unicameral bone cysts are relative-
ly rare tumors, which are usually
found in children. The etiology of
these cysts is unclear, although the-
Wright Medical Technology, Ar- 20 months), no patient required ories range from dysplastic pro-
lington, Tenn).29 The technique in- additional treatment or sustained a cesses to venous occlusion. The ac-
volves perforation of the cyst wall fracture. All patients subsequently tivity of any one individual cyst is
with a specially designed trocar sys- returned to normal activities. There difficult to predict. If the cyst is
tem (Wright Medical Technology) has been only one recurrence, in a found incidentally and the patient
(Fig. 4). A biopsy specimen is ob- young patient who has remained is asymptomatic, observation for
tained, and the cyst is then irrigated asymptomatic and has not required several months may help determine
with normal saline under fluoro- further surgery. The remaining 10 whether the cyst is active, quies-
scopic control and packed through patients healed uneventfully after cent, or involutional. If the cyst is
the trocar with a 50:50 mixture (by one procedure and had no subse- quite large and obviously active or
volume) of DBM and calcium sul- quent fractures. is associated with a pathologic frac-
fate pellets (Fig. 7). This effectively ture, treatment should be consid-
obliterates the cyst space and allows ered. Few cysts become completely
rapid bone growth stimulation. As Complications obliterated subsequent to a patho-
the calcium sulfate dissolves over 6 logic fracture, but the cyst may
to 8 weeks, the DBM stimulates While the rate of complications fol- have sufficient cortical thickening
local bone to grow into the vacated lowing injection or surgery is low, to provide a stable construct. In
space. the relative morbidity of each treat- this situation, continued observa-
In one series,29 11 patients with ment modality should be assessed. tion may be appropriate, and surgi-
unicameral bone cysts were treated The most common complication of cal treatment may be unnecessary.
by using this technique. Seven cysts treating unicameral bone cysts is However, if it is evident that the
occurred in skeletally immature recurrence of the lesion after treat- cyst is still active after appropriate
patients. After an average follow- ment and development of a subse- fracture healing, more aggressive
up period of 12 months (range, 4 to quent fracture. In active and other- treatment should be considered. It

222 Journal of the American Academy of Orthopaedic Surgeons

 Ross M. Wilkins, MD, MS

A B C D

Figure 7 A, Lateral radiograph of a 12-year-old boy who had sustained three previous fractures of his distal tibia shows a multiloculated
bone cyst of the distal tibial metaphysis. The patient had become very tentative in his daily activities and refused to participate in any
physical education classes or sports. B, Film obtained 1 week after the lesion was curetted and percutaneously grafted with a mixture of
calcium sulfate (OsteoSet) and DBM (AlloGro). Notice that the anterior portion of the cyst was not grafted. C, Postoperative film
obtained at 8 weeks shows corticalization of the boundaries of the grafted areas. A small anterior cyst remnant persists. The patient had
resumed all athletic activities and was asymptomatic. D, Additional remodeling in the intramedullary area was demonstrated at 6
months. The patient had maintained a normal active lifestyle, including downhill skiing and soccer.

appears that more recent percuta-
neous treatment techniques are as
effective as prednisolone injection
and formal curettage with bone
grafting and usually require only
one anesthetic and procedure.
Accurate determination of the
stage of activity of the cyst at pre-
sentation, coupled with appropri-
ate care, is the key to successful
treatment. Surgical treatment may
not be necessary for quiescent or
involutional cysts. However, ac-
tive, progressive cysts require
intervention. The ideal surgical
procedure is predictable and suc-
cessful in obliterating the cyst in
one attempt.

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