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BMC Musculoskeletal Disorders Oct 2019The treatment of aneurysmal bone cysts (ABCs) has evolved and less invasive methods have been tried. Denosumab is a monoclonal antibody which inhibits osteoclasts. It...
BACKGROUND
The treatment of aneurysmal bone cysts (ABCs) has evolved and less invasive methods have been tried. Denosumab is a monoclonal antibody which inhibits osteoclasts. It has been shown to be effective in giant cell tumour of bone (GCT) of bone and hence promises some effect also in ABC. We report on 6 patients treated with Denosumab and compare our results to the cases already published.
METHODS
Data of 6 patients with ABCs and patients whose treatment included Denosumab were retrospectively analyzed. Denosumab was used at a dose of 120 mg on days 1, 8, 15 and 29, and every 4 weeks thereafter. In some of these patients the dose was reduced at the end of the treatment. Clinical and radiological responses were evaluated.
RESULTS
In 4 female and 2 male patients with a mean age of 17 years (range: 6-30 years) the lesions were located in the sacrum (2), in distal radius, distal femur, talus and pelvis. One of the sacral lesions healed after 12 months and has stayed stable for 3 years since. The second patient received 2 years of therapy with recalcification, but recurred 1 year later and is under renewed therapy. The pelvic lesion improved but recurred. This patient has a 13-years history of intermittent therapy including surgery, two pregnancies and remains in a stable situation. The lesion of the talus did not improve with Denosumab after surgery and was complicated by destruction of the ankle joint with osteoarthritis. Recurrent lesions of the distal femur and the distal radius, previously treated by curettage and bone grafting healed under Denosumab and have remained stable for 2 and 3 years, respectively. One case of severe hypercalcemia was observed in a 7-year old child 6 months after discontinuation of Denosumab.
CONCLUSION
Denosumab provides a treatment option for ABCs in anatomically critical locations. Adjuvant application might reduce the rate of local recurrence. In young patients, severe rebound hypercalcemia months after discontinuation of Denosumab may occur.
Topics: Adolescent; Adult; Bone Cysts, Aneurysmal; Child; Denosumab; Dose-Response Relationship, Drug; Drug Administration Schedule; Female; Humans; Hypercalcemia; Male; Off-Label Use; Recurrence; Retrospective Studies; Secondary Prevention; Tomography, X-Ray Computed; Treatment Outcome; Young Adult
PubMed: 31630689
DOI: 10.1186/s12891-019-2855-y -
Anales de Pediatria (Barcelona, Spain :... Aug 2015
Topics: Bone Cysts, Aneurysmal; Child; Humans; Male
PubMed: 25617976
DOI: 10.1016/j.anpedi.2014.12.001 -
An aneurysmal bone cyst at T1 treated with bone grafts containing calcitonin and methylprednisolone.Journal of Orthopaedic Surgery (Hong... 2019Aneurysmal bone cysts (ABCs) rarely trigger pathological fractures. Various surgical and nonsurgical treatments have been reported for this condition. Herein, we present...
Aneurysmal bone cysts (ABCs) rarely trigger pathological fractures. Various surgical and nonsurgical treatments have been reported for this condition. Herein, we present the examination findings and treatment for a 15-year-old girl who initially presented with adolescent idiopathic scoliosis and mild back pain, but subsequently experienced severe back pain. Magnetic resonance imaging revealed an ABC at T1, with an associated pathological fracture. We successfully treated the patient using posterior fixation with instrumentation, curettage, and bone grafts combined with calcitonin and methylprednisolone (mPSL). At 3 years post-surgery, there was no ABC recurrence and only mild back pain persisted. To our knowledge, this is the first report of open surgery (curettage and fixation) with local intralesional administration of calcitonin and mPSL for an ABC-induced pathological spinal fracture. We believe that this treatment is an effective option for ABCs associated with a pathological spinal fracture.
Topics: Adolescent; Bone Cysts, Aneurysmal; Bone Density Conservation Agents; Bone Transplantation; Calcitonin; Curettage; Female; Fractures, Spontaneous; Glucocorticoids; Humans; Magnetic Resonance Imaging; Methylprednisolone; Neoplasm Recurrence, Local; Thoracic Vertebrae
PubMed: 30943849
DOI: 10.1177/2309499019839626 -
Journal of Orthopaedic Surgery and... Apr 2021The primary aim of our study was to evaluate the comparative efficacy and safety profile of curettage and mixed bone grafting without instrument or with elastic... (Comparative Study)
Comparative Study
Comparative efficacy and safety profile for the treatment of humeral bone cysts in children: curettage and mixed bone grafting either with or without elastic intramedullary nailing.
PURPOSE
The primary aim of our study was to evaluate the comparative efficacy and safety profile of curettage and mixed bone grafting without instrument or with elastic intramedullary nailing in the treatment of humeral bone cyst in children.
METHODS
Our retrospective study included a total of 48 children harboring humeral bone cyst in our hospital from August 2012 to February 2019. The patients enrolled were divided into elastic nailing group with the application of elastic intramedullary nailing (n = 25) and control group without using instrument (n = 23) during the management of curettage and mixed bone grafting. The following medical outcomes of the two groups were monitored and recorded: the amount of intraoperative blood loss, operation time and postoperative full weight-bearing time, in addition to postoperative clinical effects after 1 year, the function and pain level of shoulder joint before and 1, 3, 6, 9, 12, and 16 months after operation. Follow-up radiographic outcomes were reviewed to observe bone healing, local recurrence and internal fixation loosening, and other postoperative complications.
RESULTS
The clinical curative effect of the elastic nailing group was higher than that of the control group 16 months after operation (96.00% > 73.91%, P < 0.05). The intraoperative blood loss and postoperative full weight-bearing time in the elastic nailing group were less than those in the control group (P < 0.05), but the operation time was statistically insignificant between the two groups (P > 0.05). Before operation, the shoulder joint function of the two groups was comparable (P > 0.05), while the function showed remarkably better outcome in the elastic nailing group than control group 1 to 16 months after operation (P < 0.05). Before operation, the pain level of the two groups was comparable (P > 0.05), while 1 to 16 months after operation, the pain level of the elastic nailing group was significantly lower than that of the control group (P < 0.05). Patients in both groups were followed up for 16 months. Mixed bone grafting fusion was indicated by imaging CT and X-ray during the follow-up period, with an average fusion time of 11.3 ± 1.2 months (range, 8-16 months). Three months after operation, there was 1 case of incomplete pathological fracture in the control group, while no related complications occurred in the elastic nailing group. Moreover, no tumor recurrence was observed in the two groups. The two groups were comparable in terms of the incidence of complications (P > 0.05).
CONCLUSION
Children with humeral cyst treated with curettage and mixed bone grafting with the additional use of elastic intramedullary nailing exerted superior results to those without using instrument as there are beneficial outcomes and safety profile and no complications.
Topics: Adolescent; Bone Cysts; Bone Nails; Bone Transplantation; Child; Curettage; Elasticity; Female; Follow-Up Studies; Fracture Fixation, Intramedullary; Humans; Humerus; Male; Safety; Tomography, X-Ray Computed; Treatment Outcome
PubMed: 33823909
DOI: 10.1186/s13018-020-02130-6 -
Frontiers in Endocrinology 2021Aneurysmal bone cysts (ABCs) are rare benign pseudotumoral bone lesions with potential aggressive behavior due to the extensive destruction of surrounding bone....
Aneurysmal bone cysts (ABCs) are rare benign pseudotumoral bone lesions with potential aggressive behavior due to the extensive destruction of surrounding bone. Traditionally, these tumors were treated with open surgery, but there is more and more a shift to less invasive procedures. In particular, treatment for spinal ABCs is generally unsatisfactory due to the risk of morbidity, neurological impairment and recurrence, and there is a need for innovative therapies. Denosumab has been reported as a useful treatment in giant cell tumors of bone (GCTB), so its efficacy has been tested also in other fibro-osseus lesions affecting children and adolescents, such as spinal aneurysmal bone cysts. The pediatric literature is limited to case reports and small series, all of which highlight the efficacy of this treatment on lesions growth and associated bone pain. Some of these reports have already reported well known side effects associated with denosumab, such as hypocalcemia at the beginning of the treatment, and rebound hypercalcemia at the discontinuation. The latter seems to be more frequent in children and adolescents than in adults, probably due to the higher baseline bone turnover in children. In addition, the use of denosumab in young patients could affect both bone modeling and remodeling, even if the consequences on the growing skeleton have not been reported in detail. Here we describe the case of a spinal ABC diagnosed in an 8-year old young boy which was not accessible to surgery but responded favorably to denosumab. Our aim is to describe the rapid changes in mineral and bone homeostasis in this patient, that required advice from the experts of the European Reference Network (ERN) for rare bone and endocrine diseases.
Topics: Adolescent; Bone Cysts, Aneurysmal; Bone Density; Bone Density Conservation Agents; Bone Remodeling; Bone and Bones; Child; Child Development; Denosumab; Follow-Up Studies; Genu Valgum; Humans; Male; Minerals; Spinal Diseases
PubMed: 34335473
DOI: 10.3389/fendo.2021.698963 -
BMC Musculoskeletal Disorders Nov 2021The irregular anatomical shape and complex structures of irregular bones make it more difficult to repair and reconstruct bone defects in irregular bones than in the...
Individualized 3D printing-assisted repair and reconstruction of neoplastic bone defects at irregular bone sites: exploration and practice in the treatment of scapular aneurysmal bone cysts.
BACKGROUND
The irregular anatomical shape and complex structures of irregular bones make it more difficult to repair and reconstruct bone defects in irregular bones than in the long bones of the extremities. Three-dimensional (3D) printing technology can help to overcome the technical limitations of irregular bone repair by generating simulations that enable structural integration of the lesion area and bone structure of the donor site in all directions and at multiple angles. Thus, personalized and accurate treatment plans for restoring anatomical structure, muscle attachment points, and maximal function can be made. The present study aimed to investigate the ability of 3D printing technology to assist in the repair and reconstruction of scapular aneurysmal ABC defects.
METHODS
The study included seven patients with ABCs of the scapula. Based on computed tomography (CT) data for the patient, the scapula (including the defect) and pelvis were reconstructed using Mimics Medical software. The reconstructed scapula model was printed using a 3D printer. Before the operation, the model was used to design the surgical approach and simulate the operation process, to determine the length and radius of the plate and the number and direction of screws, and to determine the bone mass of the ilium and develop reasonable strategies for segmentation and distribution. The operation time, amount of bleeding, length and radius of the plate, and direction and number of screws were recorded.
RESULTS
The average duration of follow-up was 25.6 months, and none of the seven patients experienced recurrence during the follow-up period. The surgical approach, the length and radius of internal fixation, and the number and direction of screws were consistent with the designed operation plan. Patients gradually recovered the anatomical structure of the scapula and function of the shoulder joint.
CONCLUSIONS
In the treatment of bone defects caused by irregular bone tumors, 3D printing technology combined with surgery has the advantages of less trauma, short operation time, less bleeding and reducing the difficulty of operation, which can reduce the waste of bone graft, and more complete reconstruction of the anatomical structure of the defective bone.
Topics: Bone Cysts, Aneurysmal; Bone Plates; Fracture Fixation, Internal; Humans; Printing, Three-Dimensional; Scapula
PubMed: 34823490
DOI: 10.1186/s12891-021-04859-5 -
Management of juvenile and aneurysmal bone cysts: a systematic literature review with meta-analysis.European Journal of Trauma and... Feb 2023Numerous approaches to the management of juvenile and aneurysmal bone cysts (ABC) are described in the specialist literature together with discussion of the associated... (Meta-Analysis)
Meta-Analysis
PURPOSE
Numerous approaches to the management of juvenile and aneurysmal bone cysts (ABC) are described in the specialist literature together with discussion of the associated healing and recurrence rates. Since there is currently no evidence-based treatment standard for these conditions, the aim of this systematic literature review with meta-analysis was to examine the different management approaches, evaluate the corresponding clinical outcomes and, as appropriate, to formulate a valid treatment recommendation.
METHODS
A systematic search on OVID Medline based on a pre-existing search strategy returned 1333 publications. Having defined inclusion and exclusion criteria and analysis of the relevant full texts, 167 publications were included in the descriptive analysis and 163 in the meta-analysis. For this purpose, different subgroups were created, based on the type of cyst and the therapeutic procedure. Those subgroups were then analysed in relation to their healing rates, the number of recurrences and complication rates.
RESULTS
For aneurysmal bone cysts, both surgical removal and Doxycycline injection lead to excellent outcomes (98% healing) and low recurrence rates (6% and 11% resp.). Curettage (91% healing), including its combination with autologous cancellous bone graft (96% healing), showed very good healing rates but higher recurrence rates (22% and 15%, resp.), which were however improved by preoperative selective arterial embolization. A critical view must be taken of radiotherapy (90% healing) and the injection of alcohol (92% healing) because of their high complication rates (0.43/cyst and 0.42/cyst, resp.). In the management of juvenile bone cysts, surgical interventions like curettage and cancellous bone graft (87% healing) are far superior to non-surgical approaches (51% healing), furthermore, the application of autologous cancellous bone graft reduced the recurrence rate (3% recurrence) compared to curettage alone (20% recurrence). In subgroup analysis, treatment by ESIN was found to produce excellent outcomes (100% healing), though the patient collectives were small.
CONCLUSION
Surgical procedures to treat aneurysmal bone cysts appear to be the method of choice whereby Doxycycline injection may be an alternative. A surgical approach should be preferred in the treatment of juvenile bone cysts.
Topics: Humans; Bone Cysts, Aneurysmal; Doxycycline; Bone Cysts; Curettage; Bone Transplantation; Treatment Outcome
PubMed: 35989377
DOI: 10.1007/s00068-022-02077-9 -
Medicine Jun 2020Unicameral bone cyst (UBC) is a benign fluid-filled lesion, mainly located in the metaphyses of long bones in children and adolescents. Elastic stable intramedullary... (Comparative Study)
Comparative Study Observational Study
Unicameral bone cyst (UBC) is a benign fluid-filled lesion, mainly located in the metaphyses of long bones in children and adolescents. Elastic stable intramedullary nail (ESIN) is adopted in our institute for UBCs since 2010, and bone grafting was performed simultaneously. This study aims to evaluate the efficacy of ESIN decompression combined with different bone graft materials.All patients with the diagnoses of UBCs of the humerus, treated with ESINs and bone grafting between January 2010 and June 2018, were analyzed retrospectively. The bone grafting included injectable calcium sulfate, a mixture of the autologous iliac bone and allogeneic bone. All patients were categorized into 2 groups: ICS (injectable calcium sulfate) group and MIX (a mixture of the autologous iliac bone and allogeneic bone, ratio: 1:3) group. All the information was collected from the Hospital Database.In all, 17 patients (8.8 ± 2.3-year-old, male 8, female 9) in the ICS group and 19 patients (8.9 ± 1.9-year-old, male 10, female 9) in the MIX group were included in this study. Patients in both groups were followed up for more than 2 years (average, 3.4 ± 1.3 years). No recurrence was observed in either group. There was no significant difference between the two groups concerning the patient's demographic parameters, including sex, age, and affected side. All patients in both groups displayed excellent and good shoulder function, and there was no significant difference between the 2 groups (P = .29). As for the Capanna classification, there was no significant difference between these 2 groups (P = .78).Intramedullary nailing has the advantage of a minimally invasive procedure, immediate stability, and continuous decompression. ICS showed similar results as a mixture of the autologous iliac bone and allogeneic bone.
Topics: Bone Cysts; Bone Nails; Bone Transplantation; Calcium Sulfate; Child; Decompression, Surgical; Female; Fracture Fixation, Intramedullary; Fractures, Spontaneous; Humans; Humerus; Ilium; Male; Retrospective Studies; Transplantation, Homologous; Treatment Outcome
PubMed: 32502025
DOI: 10.1097/MD.0000000000020563 -
Journal of Medical Case Reports Nov 2022Aneurysmal bone cysts are benign bone tumors that not uncommonly involves the spine. However, this involvement can cause scoliosis, albeit rarely. This report focuses on...
BACKGROUND
Aneurysmal bone cysts are benign bone tumors that not uncommonly involves the spine. However, this involvement can cause scoliosis, albeit rarely. This report focuses on the importance of proper management for complete tumor resection to prevent recurrence and spinal deformity.
CASE PRESENTATION
A 12-year-old Middle Eastern boy, with a history of T11 aneurysmal bone cyst resection and bone grafting carried out at another hospital, presented with spine deformity of 4 months' duration. The deformity was not associated with pain or neurological deficit. A whole-spine magnetic resonance imaging with contrast confirmed the recurrence of the aneurysmal bone cyst. Posterior spinal instrumentation with corpectomy of T11 was then performed, and confirmed with histopathology the recurrence of aneurysmal bone cyst. Two years post-corpectomy, deformity correction was done from T5-L4.
CONCLUSION
Management of aneurysmal bone cysts requires meticulous planning and full excision to prevent recurrence, especially in the growing spine. If neglected, it can cause major spinal deformities and cord compression, which places a medical burden on the patient and family. To avoid such complications, treating aneurysmal bone cysts along with scoliosis correction can prevent deformity progression.
Topics: Male; Humans; Child; Scoliosis; Bone Cysts, Aneurysmal; Spine; Neurosurgical Procedures; Bone Transplantation
PubMed: 36443749
DOI: 10.1186/s13256-022-03685-0 -
The Cochrane Database of Systematic... Feb 2017Simple bone cysts, also known as a unicameral bone cysts or solitary bone cysts, are the most common type of benign bone lesion in growing children. Cysts may lead to... (Review)
Review
BACKGROUND
Simple bone cysts, also known as a unicameral bone cysts or solitary bone cysts, are the most common type of benign bone lesion in growing children. Cysts may lead to repeated pathological fracture (fracture that occurs in an area of bone weakened by a disease process). Occasionally, these fractures may result in symptomatic malunion. The main goals of treatment are to decrease the risk of pathological fracture, enhance cyst healing and resolve pain. Despite the numerous treatment methods that have been used for simple bone cysts in long bones of children, there is no consensus on the best procedure. This is an update of a Cochrane review first published in 2014.
OBJECTIVES
To assess the effects (benefits and harms) of interventions for treating simple bone cysts in the long bones of children, including adolescents.We intended the following main comparisons: invasive (e.g. injections, curettage, surgical fixation) versus non-invasive interventions (e.g. observation, plaster cast, restricted activity); different categories of invasive interventions (i.e. injections, curettage, drilling holes and decompression, surgical fixation and continued decompression); different variations of each category of invasive intervention (e.g. different injection substances: autologous bone marrow versus steroid).
SEARCH METHODS
We searched the Cochrane Bone, Joint and Muscle Trauma Group Specialised Register, the Cochrane Central Register of Controlled Trials, MEDLINE, Embase, the China National Knowledge Infrastructure Platform, trial registers, conference proceedings and reference lists. Date of last search: April 2016.
SELECTION CRITERIA
Randomised and quasi-randomised controlled trials evaluating methods for treating simple bone cysts in the long bones of children.
DATA COLLECTION AND ANALYSIS
Two review authors independently screened search results and performed study selection. We resolved differences in opinion between review authors by discussion and by consulting a third review author. Two review authors independently assessed risk of bias and data extraction. We summarised data using risk ratios (RRs) or mean differences (MDs), as appropriate, and 95% confidence intervals (CIs). We used the Grading of Recommendations Assessment, Development, and Evaluation (GRADE) system to assess the overall quality of the evidence.
MAIN RESULTS
In this update in 2017, we did not identify any new randomised controlled trials (RCT) for inclusion. We identified one ongoing trial that we are likely to include in a future update. Accordingly, our results are unchanged. The only included trial is a multicentre RCT conducted at 24 locations in North America and India that compared bone marrow injection with steroid (methylprednisolone acetate) injection for treating simple bone cysts. Up to three injections were planned for participants in each group. The trial involved 90 children (mean age 9.5 years) and presented results for 77 children at two-year follow-up. Although the trial had secure allocation concealment, it was at high risk of performance bias and from major imbalances in baseline characteristics. Reflecting these study limitations, we downgraded the quality of evidence by two levels to 'low' for most outcomes, meaning that we are unsure about the estimates of effect. For outcomes where there was serious imprecision, we downgraded the quality of evidence by a further level to 'very low'.The trial provided very low quality evidence that fewer children in the bone marrow injection group had radiographically assessed healing of bone cysts at two years than in the steroid injection group (9/39 versus 16/38; RR 0.55 favouring steroid injection, 95% CI 0.28 to 1.09). However, the result was uncertain and may be compatible with no difference or small benefit favouring bone marrow injection. Based on an illustrative success rate of 421 children with healed bone cysts per 1000 children treated with steroid injections, this equates to 189 fewer (95% CI 303 fewer to 38 more) children with healed bone cysts per 1000 children treated with bone marrow injections. There was low quality evidence of a lack of difference between the two interventions at two years in functional outcome, based on the Activity Scale for Kids function score (0 to 100; higher scores equate to better outcome: MD -0.90; 95% CI -4.26 to 2.46) or in pain assessed using the Oucher pain score. There was very low quality evidence of a lack of differences between the two interventions for adverse events: subsequent pathological fracture (9/39 versus 11/38; RR 0.80, 95% CI 0.37 to 1.70) or superficial infection (two cases in the bone marrow group). Recurrence of bone cyst, unacceptable malunion, return to normal activities, and participant satisfaction were not reported.
AUTHORS' CONCLUSIONS
The available evidence is insufficient to determine the relative effects of bone marrow versus steroid injections, although the bone marrow injections are more invasive. Noteably, the rate of radiographically assessed healing of the bone cyst at two years was well under 50% for both interventions. Overall, there is a lack of evidence to determine the best method for treating simple bone cysts in the long bones of children. Further RCTs of sufficient size and quality are needed to guide clinical practice.
Topics: Adolescent; Bone Cysts; Bone Marrow Transplantation; Child; Glucocorticoids; Humans; Methylprednisolone; Methylprednisolone Acetate; Randomized Controlled Trials as Topic
PubMed: 28158933
DOI: 10.1002/14651858.CD010847.pub3