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Diagnosis and treatment of unicameral and aneurysmal bone cysts in children.

Bone cysts are commonly encountered clinical problems in the pediatric age group. The most common types of cysts are the unicameral and aneurysmal bone cysts. These benign lesions vary in their aggressiveness, clinical behavior and treatment. Both of these lesions are poorly understood in terms of their etiology, but effective treatment exists. These lesions represent a source of great consternation to both clinicians and families, for they weaken the bone and may be confused with malignant lesions. This review will focus on the two most commonly encountered cystic lesions in the pediatric population.

Bone Cysts, Aneurysmal↗

Treatment of chondroblastoma of the calcaneus with a secondary aneurysmal bone cyst using endoscopic curettage without bone grafting.

Chondroblastoma is a relatively rare benign bone tumor. Approximately 7% of chondroblastomas occur in the calcaneus, and 17% of chondroblastoma associated with cystic lesions. We report a case of a chondroblastoma in the calcaneus with a secondary aneurysmal bone cyst treated successfully by endoscopic curettage without bone grafting. New bone formation is facilitated by minimal damage to the bone and soft tissue. The cosmetic results of this procedure are good. Two years later, the patient is asymptomatic with no radiographic evidence of recurrence. Endoscopic curettage without bone grafting is a promising new treatment for chondroblastoma.

Adult↗

Unicameral bone cysts treated by injection of bone marrow or methylprednisolone.

In 79 consecutive patients with unicameral bone cysts we compared the results of aspiration and injection of bone marrow with those of aspiration and injection of steroid. All were treated by the same protocol. The only difference was the substance injected into the cysts. The mean radiological follow-up to detect activity in the cyst was 44 months (12 to 108). Of the 79 patients, 14 received a total of 27 injections of bone marrow and 65 a total of 99 injections of steroid. Repeated injections were required in 57% of patients after bone marrow had been used and in 49% after steroid. No complications were noted in either group. In this series no advantage could be shown for the use of autogenous injection of bone marrow compared with injection of steroid in the management of unicameral bone cysts.

Bone Cysts↗

Demineralized bone implants for nonunion fractures, bone cysts, and fibrous lesions.

Demineralized bone implants were used to treat eight patients with fracture nonunion, five patients with bone cysts, and eight patients with fibrous lesions. Five of the eight patients with nonunions had had previous unsuccessful attempts at reconstruction. After fixation and implantation with either human or bovine demineralized bone, all eight fractures healed. For those patients with nonunion fractures, mean followup time was 8 years, 5 months, and longest followup was 15 years, 3 months. The cystic lesions in five patients included three typical aneurysmal bone cysts and two recurrent unicameral bone cysts. The aneurysmal bone cysts had excellent healing and bone remodeling. The two unicameral bone cysts were repacked more densely after 1 year and healed. For these patients with bone cysts, the mean followup time was 12 years, 5 months, and longest followup was 15 years, 1 month. The various fibrous lesions in seven of the eight patients were healed within 6 months, with only one requiring repacking. For these patients with fibrous bone lesions, the mean followup time was 9 years, 8 months, and longest followup was 14 years. A biopsy of the lesions in five patients was performed and in two cases showed osteoblasts and new bone around small particles of the implants. These results with long term followup show that allogeneic or xenogeneic demineralized bone implants offer a reasonable alternative for the treatment of typical nonunion fractures, bone cysts, and fibrous lesions of bone.

Adolescent↗

Treatment of aneurysmal bone cysts by introduction of demineralized bone and autogenous bone marrow.

BACKGROUND: On the assumption that an aneurysmal bone cyst has an intrinsic potential to heal by ossification, a new, minimally invasive protocol was developed. Demineralized bone powder mixed with bone-marrow aspirate was introduced into the cyst to halt the expansion phase and to allow the cyst to ossify. We hypothesized that, in order to induce bone-healing, cells from the cyst are needed to respond to the inductive material but that curettage or extensive surgery is not necessary. The goals of the present study were to assess cyst-healing and to determine the prevalence of recurrence associated with this new procedure. METHODS: Thirteen biopsy-proven primary aneurysmal bone cysts were entered through a small incision, and a paste of demineralized bone and autologous bone marrow was introduced with an applicator. The study group included three male and ten female patients with a mean age of 16.6 years. The cyst was located in a long bone in six patients, the pelvis in five patients, and the scapular glenoid and the calcaneus in one patient each. Five patients had not received treatment previously, whereas one had had a preoperative embolization and seven had recurrent lesions that had been treated previously. RESULTS: After a mean duration of follow-up of 3.9 years, healing was achieved in eleven patients. CONCLUSIONS: This minimally invasive method is able to promote the self-healing of a primary aneurysmal bone cyst. As no curettage is required, the proposed treatment avoids extensive surgery and blood loss and is convenient for the treatment of poorly accessible lesions such as those occurring in the pelvis. LEVEL OF EVIDENCE: Therapeutic Level IV.

Adolescent↗

Diagnosing aneurysmal and unicameral bone cysts with magnetic resonance imaging.

The differential between aneurysmal bone cysts and unicameral bone cysts usually is clear clinically and radiographically. Occasionally there are cases in which the diagnosis is not clear. Because natural history and treatment are different, the ability to distinguish between these two entities before surgery is important. The authors reviewed, in a blinded fashion, the preoperative magnetic resonance images to investigate criteria that could be used to differentiate between the two lesions. All patients had operative or pathologic confirmation of an aneurysmal bone cyst or unicameral bone cyst. The authors analyzed the preoperative magnetic resonance images of 14 patients with diagnostically difficult bone cysts (eight children with unicameral bone cysts and six children with aneurysmal bone cysts) and correlated these findings with diagnosis after biopsy or cyst aspiration and contrast injection. The presence of a double density fluid level within the lesion strongly indicated that the lesion was an aneurysmal bone cyst, rather than a unicameral bone cyst. Other criteria that suggested the lesion was an aneurysmal bone cyst were the presence of septations within the lesion and signal characteristics of low intensity on T1 images and high intensity on T2 images. The authors identified a way of helping to differentiate between aneurysmal bone cysts and unicameral bone cysts on magnetic resonance images. Double density fluid level, septation, and low signal on T1 images and high signal on T2 images strongly suggest the bone cyst in question is an aneurysmal bone cyst, rather than a unicameral bone cyst. This may be helpful before surgery for the child who has a cystic lesion for which radiographic features do not allow a clear differentiation of unicameral bone cyst from aneurysmal bone cyst.

Adolescent↗

Classic adamantinoma with osteofibrous dysplasia-like foci and secondary aneurysmal bone cyst.

Adamantinoma, a rare bone lesion of the tibia and fibula, has two distinct variants, classic adamantinoma and osteofibrous dysplasia-like adamantinoma. Composite lesions have not been described. Aneurysmal bone cyst is a benign cystic lesion which may also occur in the tibia and fibula. We report an unusual case of classic adamantinoma with osteofibrous dysplasia-like areas and foci of secondary aneurysmal bone cyst with prominent giant cells. A lesion was diagnosed in a 17-year-old girl with a 14-year history of a slowly enlarging left tibial mass and increasing deformity. Pathologically, the predominant pattern was classic adamantinoma, with minor foci of osteofibrous dysplasia-like adamantinoma and areas of secondary aneurysmal bone cyst with abundant multinucleated giant cells. We report the clinical, radiologic, and pathologic features of this case, and summarize lesions associated with secondary aneurysmal bone cyst. To our knowledge, the association of adamantinoma with secondary aneurysmal bone cyst has not been previously reported.

Adolescent↗

Zoledronate induces apoptosis in cells from fibro-cellular membrane of unicameral bone cyst (UBC).

Unicameral bone cyst (UBC) is a benign cystic lesion in children which is prone to fracture. Various treatments are available, but recurrence after different types of percutaneous injection therapy can cause bone destruction and pathologic fracture. The potential therapeutic effects of anti-resorptive agents, such as bisphosphonates, have not been investigated for UBC. The objective of this study was to characterize the cells from the fibro-cellular membrane of unicameral bone cyst (UBC cells) and to determine whether zoledronate, a nitrogen-containing bisphosphonate, could induce apoptosis in UBC cells. Flow cytometry and immunoblotting were performed in order to determine whether zoledronate induced apoptosis. Cells derived from normal human trabecular bones were used as controls against UBC cells to compare the effect of zoledronate in inducing apoptosis. Immunohisto/cytochemistry (IHC/ICC) and mini-array analyses were performed on tissues and cultured cells. Isolated peripheral blood mononuclear cells were incubated with conditioned media from the UBC cells to determine whether they are capable of inducing osteoclastogenesis. UBC membrane is composed of cells staining positively with CD68, SDF-1, STRO-1 and RANKL, but in vitro cells showed no staining with antibodies to CD68 and STRO-1, suggesting that there was a clonal selection of stromal cells during cell culture. UBC cells also express RUNX2 (runt-related transcription factor-2, core binding factor-1), a key transcription factor for osteoblastic differentiation. In addition, media collected from UBC cells induced a generation of multi-nucleated osteoclast-like cells of peripheral blood mononuclear cells. Zoledronate induced apoptosis of UBC cells in a dose-dependent manner. Apoptosis was evidenced by induction of the active cleaved form of caspase-3. The baseline apoptotic fractions were similar in UBC cells and trabecular bone cells. However, in the overall apoptotic fractions in this study, trabecular bone cells showed 17.2% of apoptosis, significantly lower than 24.2% of UBC cells (p-value=0.007). With the various zoledronate concentrations, mean apoptotic fractions of trabecular bone cells was 19.2%, significantly lower than 27.8% of UBC cells (p-value=0.040). With GGOH co-treatment in various zoledronate concentrations, 15.1% apoptosis was shown in trabecular bone cells, which was not significantly lower than 20.6% of UBC cells (p-value=0.076). This data suggests that zoledronate causes apoptosis in both UBC and trabecular bone cells by inhibition of the mevalonate pathway. In addition to the known anti-osteoclastogenic effect of bisphosphonates, the GGOH inhibitory effects of zoledronate were more prominent in UBC cells than trabecular bone cells, indicating their potential therapeutic role in UBC.

Apoptosis↗

Contribution to the vascular origin of the unicameral bone cyst.

Vascular occlusion between bone cyst and intramedullary venous system seems to be the primum movens in the genesis of a unicameral bone cyst. High speed injection of saline and alternative aspiration injection of the cyst fluid and reaming of the medullary cavity clearly open vascular channels connecting the bone cyst to the intramedullary venous system. This was monitored by pre- and postmanipulative radiographs. This technique was employed in 12 patients with a unicameral bone cyst. All but one went on to healing with increasing diameter of the cortical bone and filling of the cystic cavity.

Adolescent↗

Aneurysmal cyst, bone type, primary in an artery.

An aneurysmal bone cyst-like reaction involving the left common carotid artery bifurcation in an otherwise healthy 7-year-old boy is described. The patient presented with a 6-week history of an enlarging pulsatile neck mass. The mass was excised with connection of arteries on either side with a saphenous vein graft. There were no associated intracranial arterial aneurysms or associated skeletal lesions. A history of trauma could not be elicited. To the best of our knowledge this is the first report of an aneurysmal cyst, bone type, involving a large blood vessel.

Aneurysm↗

Unicameral and aneurysmal bone cysts.

Unicameral and aneurysmal bone cysts are considered tumorlike conditions of unclear origin. The diagnosis of unicameral bone cysts is almost always based on the radiographic appearance, whereas aneurysmal bone cyst imaging may sometimes mimic a sarcomatous lesion. Several pathogenetic hypotheses [correction of hypothesis] reported in literature have been described. Classifications have been proposed to detect the activity of the cysts and to predict the prognostic behavior. The results observed with different options of treatment have been discussed.

Bone Cysts↗

Bone cysts containing silicone particles in bones adjacent to a carpal silastic implant.

Silastic implants for a wide variety of medical purposes are in current and frequent use worldwide. Only recently there have been reports of the migration of silicone to the surrounding tissues via lymphatics. In the present material of nine cases with carpal implants followed for more than two years, bone cysts developed in the surrounding bones on five occasions. The only cysts so far investigated thoroughly contained foreign body reaction, and silicone could be detected by electron probe microanalysis. A long-term follow-up is suggested whenever these implants are used.

Adult↗

[Aneurysmal bone cyst of the frontal bone (author's transl)].

A six year-old boy. He complained of a swelling of the left forehead since October of 1971, the region of his upper eyelid has then been gradually swollen. He was admitted to our institute on the 24th of February, 1973, without the past history of head trauma. We found that the swollen region had a diameter of about 4 centimeters covered from the left upper eyelid to the forehead with a slight tenderness on pressure. We had no neurological findings. According to the results of skull X rays, the superior margin of left orbit and zygomatic process of frontal bone were swollen and like honey combs. An irregular, long and narrow osteolytic legion was found, which was about 4 centimeters long and 5 centimeters wide. According to the results of the left selective external carotid angiography, after injection of 60% Urografin, for more than 2.5- 10 seconds, at the left frontal bone an abnormal shadow (patchy contrast filling) was noted, which was about 4 centimeters long and 5 centimeters wide. After the direct injection of Urografid into the lesion, the cyst of one centimeter long and 3 centimeters wide was observed at the zygomatic process of the frontal bone. Operation was performed to excise the outer plate of the swollen bone and to curette the lesion after the ligature of the left external carotid artery. Histological examination showed many blood lakes and some multinuclear giant cells in the specimens and we diagnosed it was an aneurysmall bone cyst. This case is the first one of aneurysmall bone cyst confirmed by the selective external carotid angiography and the direct puncture of lesion.

Bone Cysts↗

Radiographic features of an aneurysmal bone cyst of the orbit.

Aneurysmal bone cyst is an unusual benign solitary lesion of bone occurring rarely in the orbit. In this report, we present computed tomography and magnetic resonance images of an orbital aneurysmal bone cyst involving the frontal bone in a 16-month-old boy. The aneurysmal bone cyst exhibited bone destruction, new bone formation of the orbital roof and fluid-fluid levels in the lesion following hemorrhage. These radiographic features may be useful for diagnosing orbital aneurysmal bone cysts.

Bone Cysts, Aneurysmal↗