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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
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
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 a two-needle technique. The two- authors also recommend that multi-
In an early paper, Scaglietti 19 needle technique allows efflux of ple percutaneous perforations be
described the technique of injecting the saline and excess fluid through made in the cyst to normalize its
methylprednisolone into unicameral an outflow needle in another region local circulation and disrupt any
bone cysts. He reported successful of the cyst. venous obstruction.11
healing of the cyst with much less Although in principle this proce-
morbidity than with resection or dure would seem to be advanta- Surgical Techniques
curettage plus bone grafting. The geous by decreasing the morbidity Resection or curettage plus bone
procedure involves injecting meth- due to a major surgical procedure, grafting has been employed as the
ylprednisolone into the cyst under unfortunately it has not proved to definitive treatment for unicameral
fluoroscopic control while using be very effective and usually in- bone cysts. However, in published
radiopaque dye to confirm entry volves multiple injections and anes- series, the recurrence rates have
into the cyst. Aspiration of the cyst thetics.19 Overall, a review of the ranged as high as 45%.2,18 The high
is done prior to injection. The literature revealed recurrence rates rates of recurrence may be due to the
return of clear, straw-colored fluid of 15% to 88% after an average of fact that surgically treated cysts are
is confirmatory of the diagnosis. If three injections. 20-22 It is unclear more active, more aggressive, and
grossly bloody fluid is encountered, what effect the methylprednisolone likely to be recurrent. Active uni-
a formal biopsy is advised to ascer- actually has on the local anatomy. cameral bone cysts tend to be treated
tain whether the lesion is an aneu- Some authors advocate using only more aggressively from the outset.
rysmal bone cyst or another type of normal saline, because they feel Most published series are reports
lesion. The cyst is then flushed with that the mechanical disturbance of from tertiary referral centers, where
saline, and methylprednisolone is the injection is the important factor, the patient populations have cysts
injected with either a one-needle or rather than the agent itself.10 Other with a more difficult clinical course.
The technique of resection or to employ these toxic and damag- cysts15,25,26 (Fig. 5). Calcium sulfate
curettage is relatively straightfor- ing agents is to imply that the local in the form of plaster of paris has
ward. Once an approach has been cells are the primary etiologic fac- been used with a good success rate
made to the bone, a cortical win- tors. There is no evidence to sup- and a low recurrence rate (11%).23
dow is made, which allows access port this hypothesis. Neer et al 2 The injection of autologous bone
to the entire contents of the cavity found no difference in the rate of re- marrow is also effective, but this
(Fig. 5). The clear fluid should be currence when phenol was used, procedure requires several injec-
removed, and the fibrous mem- and Schreuder et al15 reported a 5% tions14,27 (Figs. 1 and 6). Deminera-
brane curetted from the cyst wall. rate of complications related to the lized bone matrix, an osteoinductive
If the cyst is immediately adjacent to use of liquid nitrogen. material, can be injected percuta-
or involves the epiphyseal growth The choice of autologous bone neously into cysts28 (Figs. 1 and 6).
plate, care must be taken to avoid graft or a substitute is dependent on In the author’s series of 11 patients
injury to the plate. It is not neces- the orthopaedic surgeon’s prefer- who received one percutaneous
sary to remove structural bone from ence. Autologous bone marrow, injection of DBM, no further treat-
the outer cyst wall. It is also not ne- allograft, demineralized bone matrix ment was required in any case after
cessary to use adjunctive materials, (DBM), and other bone substitute 2 years of follow-up, and 9 of the 11
such as phenol or liquid nitrogen, materials have been used success- cysts were entirely obliterated.
to perform this procedure. Such fully, thus sparing the patient the An alternative technique in-
materials have secondary complica- morbidity of an autograft harvesting volves the use of a combination of
tions and may interfere with graft site.23,24 Allograft bone chips have DBM (AlloGro, AlloSource, Den-
and bone healing.15 Furthermore, proved effective in the treatment of ver) and calcium sulfate (OsteoSet,
A B C
D E
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- Accurate determination of the involutional cysts. However, ac-
neous treatment techniques are as stage of activity of the cyst at pre- tive, progressive cysts require
effective as prednisolone injection sentation, coupled with appropri- intervention. The ideal surgical
and formal curettage with bone ate care, is the key to successful procedure is predictable and suc-
grafting and usually require only treatment. Surgical treatment may cessful in obliterating the cyst in
one anesthetic and procedure. not be necessary for quiescent or one attempt.
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