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Background: Giant cell tumor of bone (GCTB) is a locally aggressive neoplasm with a high propensity for recurrence following intralesional curettage. The introduction of denosumab, a RANKL inhibitor, has shown potential in facilitating joint-sparing surgery. However, concerns exist regarding its impact on local recurrence rates. This study aimed to evaluate the efficacy and safety of combined preoperative denosumab with adjuvant microwave ablation (MWA) for the treatment of high-risk GCTB., Methods: We conducted a retrospective review of 19 patients with high-risk GCTB who underwent preoperative denosumab treatment followed by curettage and adjuvant MWA. The primary outcome measure was the local recurrence rate, with secondary outcomes including functional status assessed by the Musculoskeletal Tumor Society (MSTS) score and safety profile of the treatment., Results: In this retrospective analysis, we evaluated the outcomes of 19 patients with high-risk GCTB treated with preoperative denosumab and adjuvant MWA. The median follow-up duration was 33.1 months, 3 patients (15.8%) experienced local recurrence at a median of 21.6 months postoperatively and the local recurrence-free survival was 81.2% at two years. Notably, no patient developed lung metastasis, and all recurrences were successfully managed with repeat curettage and MWA, with a mean MSTS score of 27.3. No patient required joint replacement due to tumor recurrence, resulting in a 100% joint preservation rate., Conclusion: The combination of preoperative denosumab and adjuvant MWA is a feasible and effective strategy for the management of high-risk GCTB, providing effective local control with preserved joint function. This approach may offer a surgical alternative for young patients where joint preservation is paramount., (© 2024. The Author(s).)

Background: The Musculoskeletal Tumor Society Score (MSTS) is widely used to evaluate functioning following surgery for bone and soft-tissue sarcoma. However, concerns have been raised about its content validity due to the lack of patient involvement during item development. Additionally, literature reports inconsistent results regarding data quality and structural validity. This study aimed to evaluate content, structural and construct validity of the Danish version of the MSTS for lower extremity (MSTS-LE)., Methods: The study included patients from three complete cohorts (n = 87) with bone sarcoma or giant cell tumour of bone who underwent bone resection and reconstruction surgery in hip and knee. Content validity was evaluated by linking MSTS items to frameworks of functioning, core outcome sets and semi-structured interviews. Data quality, internal consistency and factor analysis were used to assess the underlying structure of the MSTS. Construct validity was based on predefined hypotheses of correlation between the MSTS and concurrent measurements., Results: Content validity analysis revealed concerns regarding the MSTS. The MSTS did not sufficiently cover patient-important functions, the item Emotional acceptance could not be linked to the framework of functioning, the items Pain and Emotional acceptance pertained to domains beyond functioning and items' response options did not match items. A two-factor solution emerged, with the items Pain and Emotional acceptance loading highly on a second factor distinct from functioning. Internal consistency and construct validity showed values below accepted levels., Conclusion: The Danish MSTS-LE demonstrated inadequate content validity, internal consistency, and construct validity. In addition, our analyses did not support unidimensionality of the MSTS. Consequently, the MSTS-LE is not a simple reflection of the construct of functioning and the interpretation of a sum score is problematic. Clinicians and researcher should exercise caution when relying solely on MSTS scores for assessing lower extremity function. Alternative outcome measurements of functioning should be considered for the evaluation of postoperative function in this patient group., (© 2024. The Author(s).)

We present a case detailing the diagnosis and management of a periprosthetic giant cell tumour in a female patient in her 70s, who had undergone total knee arthroplasty (TKA) for primary osteoarthritis in her right knee 7 years prior. The patient reported 4 months of painful weight-bearing. Various imaging modalities, including plain radiographs, CT scans and MRI, revealed a sizeable lytic lesion beneath the TKA prosthesis, along with loosening of the tibial component.Blood tests and analyses of synovial fluid ruled out periprosthetic joint infection, and a biopsy confirmed the diagnosis of a giant cell tumour of the bone. Treatment entailed en bloc resection of the tumour and revision of the TKA using a hinged, oncological-type megaprosthesis. Surgical procedures involved careful resection of the proximal tibia, preservation of vasculature and the creation of a medial gastrocnemius muscle flap. Following surgery, the patient underwent supervised rehabilitation with a functional brace., Competing Interests: Competing interests: None declared., (© BMJ Publishing Group Limited 2024. No commercial re-use. See rights and permissions. Published by BMJ.)

SummaryGiant cell tumours of bone are benign and locally aggressive tumours that usually occur in young adults and at the epiphysial locations after physeal closure. Occurrence outside of epiphysial locations and appearance in geriatric patients is rare. We report a case of a woman in her late 60s with a giant cell tumour of the mid-shaft of the right tibia. Extended curettage and biological reconstruction were performed with autologous double-barrel fibular struts and tri-cortical iliac crest bone grafting. At the 28-month follow-up examination, we noted full bony union at both ends with successful consolidation of the fibular struts, and importantly, no evidence of recurrence or other complications was observed., Competing Interests: Competing interests: None declared., (© BMJ Publishing Group Limited 2024. No commercial re-use. See rights and permissions. Published by BMJ.)

A man in his 30s came to our clinic with a year-long history of progressive pain and swelling in his knee. Diagnostic imaging revealed a displaced patellar fracture with an osteolytic, septated lesion and thinned expanded cortex in both fracture fragments. A core needle biopsy confirmed the diagnosis of giant cell tumour. Treatment involved wide excision of the tumour and the use of polypropylene mesh and a peroneal longus tendon autograft to reconstruct the extensor mechanism of the knee joint. One year postoperatively, the patient experienced no pain, demonstrated full range of motion and showed no signs of functional impairment or local tumour recurrence. This case highlights that reconstruction of the extensor mechanism of the knee after tumour excision with synthetic mesh is an affordable, user-friendly and widely accessible method. It can address large defects effectively while minimising the risks of disease transmission and graft lengthening, resulting in satisfactory outcomes., Competing Interests: Competing interests: None declared., (© BMJ Publishing Group Limited 2024. No commercial re-use. See rights and permissions. Published by BMJ.)

Giant cell tumour (GCT) accounts for 5% of all primary bone tumours. GCT in the distal third of ulna is quite rare. We present a case of recurrent GCT in distal third of ulna with malignant features involving tenosynovium. The case was treated by wide resection of tumour and on follow up, patient recovered well with no evidence of further recurrence. Considering the features, according to the literature reviewed, is the first case of its type., Competing Interests: Competing interests: None declared., (© BMJ Publishing Group Limited 2024. No commercial re-use. See rights and permissions. Published by BMJ.)

Giant cell tumour of bone is a benign, locally aggressive osteolytic tumour that typically affects skeletally mature young individuals. It predominantly emerges within the metaphysis, extending towards the epiphysis of long bones, while occurrences in flat bones are exceptionally rare. We present a case of a woman in her late 20s who presented with a large right ischial mass. A biopsy confirmed the mass as a giant cell tumour. The tumour extended to the acetabulum, and due to the potential risk of significant bleeding and contamination during en bloc excision, a prudent approach involved initiating denosumab therapy, a monoclonal antibody targeting receptor activator of nuclear factor-κB ligand therapy, before proceeding with radical surgery. Denosumab therapy successfully rendered a previously inoperable tumour favourable for surgical intervention. We went on to perform a type 2 and 3 internal hemipelvectomy, followed by a reconstruction with a hip endoprosthesis replacement., Competing Interests: Competing interests: None declared., (© BMJ Publishing Group Limited 2023. No commercial re-use. See rights and permissions. Published by BMJ.)

The typical presentation of giant cell tumor of bone is a solitary lesion involving the meta-epiphyseal region of the long bones. The presence of more than one distinct giant cell tumor in the same patient is rare. This study reports on 7 patients with multicentric giant cell tumor of bone. Clinical and radiologic features were reviewed to evaluate the behavior of multicentric giant cell tumor of bone. Immunohistochemistry and genetic analysis for the H3F3A gene were performed to confirm the diagnosis. The knee was most frequently involved, and most of the lesions were in an ipsilateral extremity. All of the patients received surgical management with curettage or resection. The overall median follow-up was 194 months (interquartile range, 41-336 months). Five of 7 patients had local recurrence (71%), but considering the number of surgically treated lesions, the risk of local recurrence was 33% (5 local recurrences among 15 treated lesions). No lung metastases occurred. Multicentric giant cell tumor of bone tends to exhibit the same aggressive clinical behavior as solitary giant cell tumor of bone. Patients should be monitored for the occurrence of other lesions, especially in the ipsilateral extremity. [ Orthopedics . 2023;46(6):e376-e380.].

This systematic review evaluates the effects of heat treatments in de novo, residual and recurrent giant cell tumors of bone (GCTB). Studies were eligible for inclusion if one of the following treatments was administered: radiofrequency ablation (RFA), microwave ablation, argon cauterization, electrocauterization and hot liquid treatment. The primary outcome was recurrence. Secondary outcomes were complications, pain, function, and quality of life. Recurrence rates for microwave ablation as an adjuvant to intralesional curettage were 0%, 4% and 10% (3 retrospective single-group studies); for argon cauterization 4%, 8% and 26% (3 cohort studies); electrocauterization 0% to 33% (8 cohort studies); and hot liquid 9.5% and 24% (2 cohort studies). Follow-up was generally ≥24 months. Data on pain, function and quality of life were scarce. Complications included infection and secondary osteoarthritis. Current evidence does not demonstrate or exclude an effect of heat treatments on recurrence in GCTB. Further research should objectify if (subgroups of) patients benefit from these treatments.

Background: This study reports our experience in the treatment of aggressive pelvic GCT through wide resection assisted with patient-specific bone-cutting guides (PSBCGs) and subsequent reconstruction with 3D-printed personalized implants (3DPIs), aiming to present the operative technique of this method and evaluate its clinical efficacy., Methods: We retrospectively analyzed seven patients who underwent wide resection of pelvic GCT followed by reconstruction with 3DPIs from August 2019 to February 2021. There were two males and five females, with a mean age of 43 years. PSBCGs and 3DPIs were prepared using 3D-printing technology. The operational outcomes, local recurrence, radiological results, and any associated complications of this technique were assessed. And the functional outcomes were assessed according to the Musculoskeletal Tumor Society (MSTS) 93 functional score., Results: The mean follow-up time was 35.3 months (range 28-45 months). There was no intraoperative complication. Negative surgical margins were achieved in all patients. Postoperative pelvic radiographs showed that 3DPIs matched the shape and size of the bone defect. The anterior-posterior, inlet, and outlet pelvic radiograph demonstrated precise reconstruction consistent with the surgical planning. In addition, tomosynthesis-Shimadzu metal artifact reduction technology (T-SMART) showed good osseointegration at an average of three months after surgery (range 2-4 months). There was no local recurrence or tumor metastasis. The average MSTS score was 24.4 (range 23-27) at the last follow-up. Delayed wound healing was observed in one patient, and the wounds healed after debridement. Prosthesis-related complications were not detected during the follow-up, such as aseptic loosening or structure failure., Conclusions: The treatment of aggressive pelvic GCTs through wide resection assisted with PSBCGs and subsequent reconstruction with 3DPIs is a feasible method, which provides good clinical results and reasonable functional outcomes., (© 2023. BioMed Central Ltd., part of Springer Nature.)

Objective: Using a fibula autograft (FA) to reconstruct defects after en bloc resection of giant cell tumor of bone (GCTB) in the distal radius is classic but has high complication rates. We describe a novel reconstruction method employing the cooperative application of LARS® and a 3D-printed prosthesis (L-P) and investigate whether it improves postoperative outcomes., Methods: From April 2015 to August 2022, 14 patients who underwent the cooperative L-P reconstruction method after en bloc resection of distal radial GCTBs and 31 patients who received FA reconstruction were enrolled as two retrospective cohorts in this comparative study. The properties of the implants and critical surgical techniques were elaborated in the L-P group. Preoperative function, intraoperative data, and postoperative clinical, functional, and radiographic outcomes of all patients were recorded and compared between the two groups. The grip strength and range of wrist motion, including extension, flexion, radial deviation, and ulnar deviation, were measured. The Mayo modified wrist and Musculoskeletal Tumor Society scores were chosen to assess wrist function and surgical functional outcomes, respectively. Kaplan-Meier curves were generated to analyze the significant differences in complication rates and implant survival between the two groups., Results: In both groups, all 45 patients underwent the operation without complication with similar average osteotomy lengths and bleeding volumes, while a shorter operative duration was achieved in the L-P group (201.43 ± 22.87 min vs. 230.16 ± 51.44 min, P = 0.015). At a mean follow-up of 40.42 ± 18.43 months (range, 14-72 months), both reconstruction methods effectively ameliorated postoperative function. Patients who received L-P showed higher postoperative modified Mayo wrist scores (81.43 ± 5.49 vs. 71.13 ± 16.10, P = 0.003), Musculoskeletal Tumor Society scores (27.64 ± 1.34 vs. 25.06 ± 2.95, P = 0.004), and grip strength on the normal side (68.71% ± 8.00% vs. 57.81% ± 12.31%, P = 0.005) than the FA group. Better wrist extension (63.21° ± 8.99° vs. 45.32° ± 14.53°, P < 0.001) and flexion (45.36° ± 7.90° vs. 30.48° ± 12.07°, P < 0.001) were also observed in the L-P group. The complication rate was significantly higher in the FA group (29/31, 93.55%) than in the L-P group (1/14 7.14%, P < 0.001). The L-P group showed higher implant survival than the FA group, but the difference was not statistically significant., Conclusion: The cooperative application of LARS® and 3D-printed prostheses is an effective modality for reconstructing musculoskeletal defects after en bloc resection of distal radial GCTBs, which can improve functional outcomes, diminish complication rates, and promote wrist joint stability and motion., (© 2023 The Authors. Orthopaedic Surgery published by Tianjin Hospital and John Wiley & Sons Australia, Ltd.)

Background and Objectives: Denosumab is recommended for advanced giant cell tumor of bone (GCTB) that is unresectable or resectable with unacceptable morbidity. But the effect of preoperative denosumab treatment on the local control GCTB remains controversial., Methods: We conducted a study of 49 patients with GCTB in the limbs treated with denosumab before surgery and 125 patients without in our hospital from 2010 to 2017. Propensity-score matching (PSM) at a 1:1 ratio between the denosumab and control groups was performed to minimize possible selection bias, and compared the recurrence rate, limb function, and surgical degradation between the two groups., Results: The 3-year recurrence rates in the denosumab group and the control group were 20.4% and 22.9% after PSM, respectively (p = 0.702). In the denosumab group, 75.5% (n = 37/49) of patients experienced surgical downgrading. Limb joint preservation rates were 92.1% (35) for 38 patients treated with denosumab and 60.2% (71) for 118 control subjects. (p ≺ 0.001). Postoperative MSTS were higher in patients in the denosumab group than in the control group (24.1 vs. 22.6, p = 0.034)., Conclusions: Preoperative denosumab treatment did not result in an increased risk of local recurrence of GCTB. Patients with advanced GCTB may benefit from preoperative denosumab treatment for surgical downgrading and the preservation of the joint., (© 2023 The Authors. Cancer Medicine published by John Wiley & Sons Ltd.)

Background: Giant cell tumors (GTC) of bone are benign, locally aggressive tumors generally occurring in young people with a female predominance during reproductive age. Considering their worsening during pregnancy it has been suggested that pregnancy can accelerate GCT progression or favor recurrence but correlation between tumor growth and pregnancy has not yet been clarified. Aim of this study was to clarify clinical characteristics, timing and type of treatment through a literature review on GTCs occurring during pregnancy., Patients and Methods: An electronic search was performed in December 2020 in PubMed, Scopus, Embase, Medline, Cochrane Register using the keywords "giant cell tumor" AND "pregnancy" looking for papers reporting cases of giant cell tumors of the bone onset or recurred during pregnancy. The electronic search identified 212 papers; sixteen studies were selected, for a total of 32 cases., Results: The diagnosis was made during pregnancy in 24 cases and after the partum in 8 cases. 27 cases were new diagnoses while 5 cases were recurrences. Pulmonary metastases were reported in 3 patients. The treatment was performed during the pregnancy in 7 out of 32 cases; in the remaining 27 cases treatment was performed after delivery. The hormone receptor status was reported in 14 patients. Data regarding follow-up was reported for 26 out of 32 patients; three patients had local recurrences that were treated with wide resection and amputation in 2 and 1 case, respectively; at the last follow-up all patients were apparently without any evidence of disease except for three patients who had stable lung metastases., Discussion: In case of GCT during pregnancy, a multidisciplinary approach is necessary to offer the patients the best treatment in terms of mother and child's health. A correct diagnosis is necessary and not confusing tumor symptoms with ones of pregnancy is mandatory in order not to delay the diagnosis and let the tumor progress. Actually, even though pregnancy would seem to promote GCT growth and aggressiveness, the relationship is not clear. More studies are necessary to clarify this interesting aspect., Level of Evidence: IV, systematic review., (Copyright © 2022. Published by Elsevier Masson SAS.)

BACKGROUND Giant cell tumors of the bone (GCTB) are rare, locally aggressive benign neoplasms that primarily occur in the metaphyses of long bones. In less than 2% of cases, GCTBs arise in the spine, predominantly below the sacrum. We report the clinical manifestations, diagnostic approach, and successful surgical treatment of a patient with a GCTB of the thoracic spine. CASE REPORT A 21-year-old female patient presented to the Emergency Department with back pain and upper motor neuron syndrome. A thorough clinical and imaging examination revealed a tumor and pathological fracture of the T7 vertebra. Histopathological analysis confirmed the diagnosis of a GCTB. The tumor was successfully excised surgically via a posterior thoracic approach, including bilateral decompressive laminectomy, lateral costotransversectomy, and posterior corpectomy, followed by transpedicular instrumentation of the T5-T6 and T8-T9 vertebrae, and anterior arthrodesis with an autologous graft. The patient also received adjuvant radiotherapy. One year later, the patient had no signs of recurrence or physical limitations. CONCLUSIONS GCTBs located in the thoracic spine are uncommon and pose a significant challenge for healthcare professionals due to their non-specific clinical manifestations and the need for a multidisciplinary approach to their management. This case highlights the diagnostic and therapeutic implications of a GCTB of the thoracic spine and describes a successful surgical strategy resulting in complete recovery. The presented case adds to the limited published literature on GCTBs in this unusual location and further elaborates on their presentation and management.

Background: Giant cell tumor of bone (GCTB) is a benign locally aggressive tumor frequently treated with intralesional curettage and cementation. The aim of this study was to investigate the long-term incidence of arthritic changes following curettage and cementation of GCTB around the knee., Materials and Methods: This study was a retrospective review of patients with GCTB around the knee treated with curettage and cementation with a minimum follow-up of 10 years. The functional results were assessed using the Musculoskeletal Tumor Society (MSTS) score. The arthritic changes were classified using the Kellgren-Lawrence (KL) classification system of osteoarthritis., Results: This study included 119 patients, 54 males and 65 females, with a mean age of 29.4 ± 9.2 years. There were 35 (29.4%) patients with pathological fractures. There were 84 (70.6%) patients with de novo lesions and 35 (29.4%) with recurrent lesions. The mean follow-up period was 13.2 ± 3.16 years. The mean MSTS score was 28.5 ± 1.9. Overall, 25 (21%) patients developed variable degrees of arthritis of KL grade 1 (n = 7), KL grade 2 (n = 11), KL grade 3 (n = 4), and KL grade 4 (n = 3). Ten patients showed progression of arthritis during the follow-up period. Age at presentation, gender, presence of pathological fracture, whether the tumor was de novo or recurrent, and tumor location were not associated with arthritis incidence., Conclusions: Curettage and cementation can be used safely to treat GCTB around the knee. Arthritis of the knee is a possible complication, but mild grades are expected in most cases. There was no association between arthritis incidence and age, gender, pathological fractures, tumor location, or recurrent tumors., Level of Evidence: Level IV., (© 2023. The Author(s).)

Rationale: Giant cell tumor of bone is a locally aggressive and rarely metastasizing neoplasm that typically affects the ends of long bones or the axial skeleton of young to middle-aged adults. As many as 69% to 100% of giant cell tumors harbor H3F3A gene mutations, while H3F3B gene mutations have rarely been reported., Patient Concerns: A 53-year-old male patient who underwent right distal femoral tumor resection., Diagnoses: Preoperative CT plain scan indicated giant cell tumor of bone with pathological fracture. Laboratory findings were as follows: serum calcium was 2.23 mmol/L (reference range: 2.1-2.55 mmol/L) and serum phosphorus was 1.35 mmol/L (reference range: 0.81-1.45 mmol/L)., Interventions: The histological morphology showed the typical features of a conventional GCT. The immunoprecipitation analysis results were as follows: H3.3G34W(-), H3.3G34R(-), H3.3G34V(-), and H3K36M(-). Sanger sequencing showed that the H3F3A and H3F3B gene mutations were wild type. The high-throughput gene sequencing results revealed the H3F3B gene mutations H3.3p.Gly35Trp and H3.3p.Val36Leu., Outcomes: The patient was stable with no recurrence in 12 months follow-up., Lessons: Giant cell tumor of bone with H3F3B gene mutations is extremely rare. In the pathological diagnosis of bone tumors, we need to analyze clinical presentation, imaging features, histology, immunophenotype, and cytogenetic/molecular alterations, in order to get a correct diagnosis., Competing Interests: The authors have no funding and conflicts of interest to disclose., (Copyright © 2023 the Author(s). Published by Wolters Kluwer Health, Inc.)

Objectives: This meta-analysis aimed at determining the association between preoperative denosumab and the risk of local recurrence in patients with giant cell tumors of the bone., Methods and Materials: Web of Science, EMBASE, Cochrane Library, and PubMed were comprehensively searched on April 20 th , 2022. Data from the included articles were analyzed using meta-analysis. The bias of all included studies was evaluated according to ROBINS-I. Also, subgroup and sensitivity analyses were performed., Results: Eight studies with 1270 cases (195 in the denosumab group and 1075 in the control group) were eventually included. Patients receiving denosumab before curettage had a higher risk of local recurrence than those who underwent curettage alone (odds ratio: 2.29, 95% confidence intervals: 1.44-3.64, P = 0.0005). The denosumab group showed a significantly higher risk of local recurrence in most subgroup analyses, except for those with preoperative denosumab duration ≤six months/doses (P = 0.66) and sample size ranging from 100 to 180 (P = 0.69)., Conclusion: Denosumab before curettage may increase the risk of local recurrence in patients with giant cell tumor of the bone. Preoperative denosumab should be used with caution after weighing an increased risk of local recurrence against the clinical benefits and a duration time of less than six months before surgery is recommended., Competing Interests: None

Objective: The aims of this work are to present a classification of "complex fracture" and "simple fracture", to compare their features, treatments and prognosis in patients with giant cell tumour with pathologic fractures around the knee, and to determine the best surgical method for patients who have giant cell tumour around the knee with different degrees of fracture., Methods: Data from 130 patients with pathologic fractures from giant cell tumour around the knee who underwent surgical treatment from March 2000 to November 2015 at 6 institutes around China were collected and analysed. A multicentric study design was used to explore the epidemiological features and to compare differences in the surgical procedures and prognosis of the two fracture groups. The mean age at diagnosis was 37.1 years old (range, 13-77 years). The median follow-up was 126.5 months, ranging from 68 to 370 months., Results: The general clinical and imaging features of the groups of patients with simple and complex fractures, namely, sex, age, the lesion site, living or working environment, eccentric growth patterns, Campanacci grading system, and duration of symptoms before treatment, showed varying degrees of differences, but with no statistical significance (p > 0.05). The incidence rate of surrounding soft tissue mass was 35.2% (32/91) in the group with simple fractures, whereas it was 87.2% (34/39) in the group with complex fractures, which showed a significant difference (p < 0.05). Wide resection and reconstruction with joint replacement were performed more often in patients with complex fractures (61.5%, 24/39). Intralesional procedures were performed more often in patients with simple fractures (56.0%, 51/91). The difference showed significant differences (p < 0.05). The local recurrence rate was 17.6% (16/91) in the group with simple fractures, whereas it was 10.3% (4/39) in the complex fracture group, showing a significant difference (p < 0.05). A total of 2.3% of patients (n = 3,3/130) developed a skip lesion. The complication rates were 4.6% (4/87) and 14.7% (5/34), respectively, in the two groups with simple or complex fractures, showing a significant difference (p < 0.05). The mean MSTS and TESS scores with simple fractures were 26.6 (range, 13-30) and 84.1 (range, 29-100), respectively, whereas the mean scores in the group with complex fractures were 25.5 (range, 18-30) and 78.3 (range, 30-100), respectively, also showing a significant difference (p < 0.05)., Conclusion: Our classification of "simple fracture" and "complex fracture" could guide decisions regarding the best surgical method for lesions in patients who have giant cell tumour around the knee with different degrees of fracture., (© 2022. The Author(s).)

Giant cell tumor (GCT) is a benign, locally aggressive neoplasm with little incidence at the carpal bone level. We present a case of pyramidal bone GCT that required open biopsy for diagnosis. As a definitive treatment, en bloc resection of the pyramidal bone and luno-capitate arthrodesis were performed to avoid frequent relapses of these neoplasms and ensure proper functionality of the anatomical segment.

Objectives: The aim of JCOG1610 (randomized controlled phase III trial) was to confirm the superiority of preoperative denosumab to curettage with adjuvant local therapy for patients with giant cell tumor of bone without possible post-operative large bone defect., Methods: The primary endpoint was relapse-free survival and the total sample size was set at 106 patients. Patient accrual began in October 2017. However, the accrual was terminated in December 2020 due to a recommendation from the Data and Safety Monitoring Committee because of poor patient accrual. Now, we report the descriptive results obtained in this study., Results: A total of 18 patients had been registered from 13 Japanese institutions at the time of termination on December 2020. Eleven patients were assigned to Arm A (curettage and adjuvant local therapy) and 7 to Arm B (preoperative denosumab, curettage and adjuvant local therapy). Median follow-up period was 1.6 (range: 0.5-2.8) years. Protocol treatment was completed in all but one patient in Arm A who had a pathological fracture before surgery. All patients in Arm B were treated with five courses of preoperative denosumab. Relapse-free survival proportions in Arm A and B were 90.0% (95% confidence interval: 47.3-98.5) and 100% (100-100) at 1 year, and 60.0% (19.0-85.5) and 62.5% (14.2-89.3) at 2 years, respectively [hazard ratio (95% confidence interval): 1.51 (0.24-9.41)]., Conclusion: In terms of relapse-free survival, the superiority of preoperative denosumab was not observed in patients with giant cell tumor of bone without possible post-operative large bone defect., (© The Author(s) 2022. Published by Oxford University Press. All rights reserved. For permissions, please e-mail: journals.permissions@oup.com.)

En bloc resection is typically performed to treat giant cell tumors of bone (GCTB), particularly when curettage can be challenging owing to extensive bone cortex destruction with soft tissue extension. Few reports have addressed the clinical outcomes after reoperation for local recurrence in patients with GCTB who underwent en bloc resection. In this multicenter retrospective study, we investigated local recurrence, distant metastasis, malignant transformation, mortality, and limb function in patients treated for local recurrence following en bloc resection for GCTB. Among 205 patients who underwent en bloc resection for GCTB of the extremities between 1980 and 2021, we included 29 with local recurrence. En bloc resection was performed for large tumors with soft tissue extension, pathological fractures with joint invasion, complex fractures, and dispensable bones, such as the proximal fibula and distal ulna. Local re-recurrence, distant metastasis, malignant transformation, and mortality rates were 41.4% (12/29), 34.5% (10/29), 6.9% (2/29), and 6.9% (2/29), respectively. The median Musculoskeletal Tumor Society score was 26 (interquartile range, 23-28). The median follow-up period after surgery for local recurrence was 70.1 months (interquartile range, 40.5-123.8 months). Local recurrence following en bloc resection for GCTB could indicate an aggressive GCTB, necessitating careful follow-up., Competing Interests: The authors declare no conflict of interest.

There is no consensus on a treatment strategy for spinal giant cell tumor of bone (GCTB) because of the difficulty in their treatment. Treatment options often include the use of the controversial denosumab, an antibody therapy aimed at tumor shrinkage, different curettage techniques, resection, or a combination of these therapies. The current study aimed to identify treatment methods associated with favorable outcomes in patients with spinal GCTB. We retrospectively reviewed 5 patients with spinal GCTB, including patients with tumors of the sacrum, treated at our hospital between September 2011 and November 2020. Two men and 3 women were included in the study. The median follow-up period was 74 months (range: 14-108 months). We surveyed the tumor site, treatment method, denosumab use, and outcomes. The median age was 17 years (range: 17-42 years). There were 2 cases of sacral GCTB and 1 case each of lumbar, cervical, and thoracic vertebral GCTB. The comorbidities observed included hepatitis, malignant lymphoma, atopic dermatitis, and asthma. The treatment method included zoledronic acid after embolization and denosumab, denosumab only, curettage and posterior fusion, and curettage resection after embolization and anterior and posterior fusion. Denosumab was used in all cases. Three patients were continuously disease-free, 1 patient with no evidence of disease, and 1 patient alive with disease. Aggressive treatment, especially surgical treatment, may lead to good results in spinal GCTB., Competing Interests: The authors have no funding and conflicts of interest to disclose., (Copyright © 2022 the Author(s). Published by Wolters Kluwer Health, Inc.)

Background: Liquid nitrogen (LN) has been used as an adjuvant cryotherapy for bone tumors including giant-cell tumor of the bone (GCTB) to remove residual tumor cells after curettage. This study evaluated variables related to the efficacy of LN-based cryoablation in the context of adjuvant treatment of GCTB using porcine femur bone model., Methods: A porcine femur bone model was adopted to simulate intralesional cryotherapy. A LN-holding cavity (point 1, nadir) in the medial epicondyle, 4 holes (points 2-5) in the shaft situated 5, 10, 15, and 20 mm away from the proximal edge of the cavity, and 2 more holes (points 6 and 7) in the condyle cartilage (10 and 20 mm away from the distal edge of the cavity) were made. The cooling rate was compared between the 5 points. The cellular morphological changes and DNA damage in the GCTB tissue attributable to LN-based cryotherapy were determined by H&E stain and TUNEL assay. Cartilage tissue at points 6 and 7 was examined for the extent of tissue injury after cryotherapy., Results: The temperature kinetics at points 1, 2 reached the reference target and were found to be significantly better than the reference (both p < 0.05). The target temperature kinetics were not achieved at points 4 and 5, which showed a significantly lower cooling rate than the reference (both p < 0.001) without reaching the -60°C target. Compared with untreated samples, significantly higher proportion of shrunken or apoptotic cells were found at points 1-3; very small proportion were observed at points 4, 5. Significantly increased chondrocyte degeneration was observed at point 6, and was absent at point 7., Conclusion: The cryotherapy effective range was within 5 mm from nadir. Complications were restricted to within this distance. The cooling rate was unchanged after three repeated cycles of cryotherapy., Competing Interests: Conflicts of interest: Dr. Wei-Ming Chen, an editorial board member at Journal of the Chinese Medical Association , had no role in the peer review process of or decision to publish this article. The other authors declare that they have no conflicts of interest related to the subject matter or materials discussed in this article., (Copyright © 2022, the Chinese Medical Association.)

Background: Surgical complications are frequent with giant cell tumor of bone; recurrence is the best known and most widely studies; other causes of failure have been less well investigated. We therefore performed a retrospective study to identify and assess the main reasons for surgical revision., Hypothesis: Recurrence is the main cause of surgical revision in giant cell tumor of bone, but other complications, such as mechanical issues or infection, are underestimated., Patients and Methods: A single-center retrospective study included 192 patients (included from 2000 to 2016) undergoing first giant cell tumor of bone surgery in a bone tumor reference center. Surgery consisted in curettage for 152 patients (79%) and resection for 40 (21%). The 3 main reconstruction techniques were filling (136 patients; 71%), prosthesis (18 patients; 9%), and fusion (14 patients: 7%). Filling used cement in 9 cases (7%) and bone graft in 127 (93%). Cumulative incidence functions were calculated., Results: There were 171 revision procedures in 92 patients: 43 for mechanical reasons, 30 for infection, 86 for tumor recurrence, 12 for other causes. Cumulative incidence of revision at 10years was 36% (95% CI: 27-44) for recurrence, 26% (95% CI: 17-36) for mechanical causes, and 13% (95% CI: 9-19) for infection, for overall cumulative incidence of revision of 61% (95% CI: 50-69)., Discussion: Risk of all-cause surgical revision in giant cell tumor of bone was 61% at 10years, with recurrence accounting for only half of cases., Level of Evidence: IV., (Copyright © 2021. Published by Elsevier Masson SAS.)

Background: Following curettage of giant cell tumor of bone (GCTB), it is common to fill the cavity with polymethylmethacrylate (PMMA) bone cement, bone allograft, or artificial bone to maintain bone strength; however, there is a 2-14% risk of postoperative fractures. We conducted this retrospective study to clarify the risk factors for fractures after curettage for GCTB of the extremities., Methods: This study included 284 patients with GCTBs of the extremities who underwent curettage at our institutions between 1980 and 2018 after excluding patients whose cavities were not filled with anything or who had additional plate fixation. The tumor cavity was filled with PMMA bone cement alone (n = 124), PMMA bone cement and bone allograft (n = 81), bone allograft alone (n = 63), or hydroxyapatite graft alone (n = 16)., Results: Fractures after curettage occurred in 10 (3.5%) patients, and the median time from the curettage to fracture was 3.5 months (interquartile range [IQR], 1.8-8.3 months). The median postoperative follow-up period was 86.5 months (IQR, 50.3-118.8 months). On univariate analysis, patients who had GCTB of the proximal or distal femur (1-year fracture-free survival, 92.5%; 95% confidence interval [CI]: 85.8-96.2) presented a higher risk for postoperative fracture than those who had GCTB at another site (100%; p = 0.0005). Patients with a pathological fracture at presentation (1-year fracture-free survival, 88.2%; 95% CI: 63.2-97.0) presented a higher risk for postoperative fracture than those without a pathological fracture at presentation (97.8%; 95% CI: 95.1-99.0; p = 0.048). Patients who received bone grafting (1-year fracture-free survival, 99.4%; 95% CI: 95.7-99.9) had a lower risk of postoperative fracture than those who did not receive bone grafting (94.4%; 95% CI: 88.7-97.3; p = 0.003)., Conclusions: For GCTBs of the femur, especially those with pathological fracture at presentation, bone grafting after curettage is recommended to reduce the risk of postoperative fracture. Additional plate fixation should be considered when curettage and cement filling without bone grafting are performed in patients with GCTB of the femur. This should be specially performed for those patients with a pathological fracture at presentation., (© 2022. The Author(s).)

Giant-cell tumor of bone is a rare, locally aggressive and rarely metastasizing primary bone tumor. The mainstay of treatment remains controversial and is decided by the balance between adequate surgical margin and sufficient adjacent joint function. Although curettage with a high-speed burr and local adjuvants can maintain normal joint function, many reports have revealed a high local recurrence rate. Conversely, en bloc resection and reconstruction with prostheses for highly aggressive lesions have reportedly lower local recurrence rates and poorer functional outcomes. Denosumab-a full human monoclonal antibody that inhibits receptor activator of nuclear factor-kappa β ligand-was approved by the Food and Drug Authority in 2013 for use in surgically unresectable or when resection is likely to result in severe morbidity for skeletally mature adolescents and adults with giant-cell tumor of bone. However, subsequent studies have suggested that the local recurrence rate would be increased by preoperative use of denosumab. In systematic reviews of the local recurrence rate after preoperative use of denosumab, conclusions vary due to the small sample sizes of the studies reviewed. Therefore, controversy regarding the treatment of giant-cell tumor of bone is ongoing. Here, this review elucidates the management of giant-cell tumor of bone, especially with the local adjuvant and neoadjuvant use of denosumab, and presents the current, evidence-based treatment for giant-cell tumor of bone., (© The Author(s) 2022. Published by Oxford University Press. All rights reserved. For permissions, please e-mail: journals.permission@oup.com.)

Background: Due to the wide variations in location, size, local invasiveness, and treatment options, the complications associated with surgery for giant cell tumor of bone have been sporadically reported. For quality assessment, fundamental data based on large-scale surveys of complications under a universal evaluation system is needed. The Dindo-Clavien classification is an evaluation system for complications based on severity and required intervention type and is suitable for the evaluation of surgery in a heterogeneous cohort., Methods: A multi-institutional retrospective survey of 141 patients who underwent surgery for giant cell tumor of bone in the extremity was performed. The incidence and risk factors of complications, type of intervention for complication control, and impact of complications on functional and oncological outcomes were analyzed using the Dindo-Clavien classification., Results: Forty-six cases (32.6%) had one or more complications. Of them, 18 (12.8%), 11 (7.8%), and 17 (12.1%) cases were classified as Dindo-Clavien classification grade I, II, and III complications, respectively. There were no cases with grade IV or V complications. Progression in Campanacci grading (p = 0.04), resection (over curettage, p < 0.0001), reconstruction with prosthesis (p = 0.0007), and prolonged operative duration (p = 0.0002) were significant risk factors for complications. Complications had a significant impact on function (p < 0.0001). Differences in the impact of complication types and tumor location on function were confirmed. Complications had no impact on local recurrence and metastasis development., Conclusion: The Dindo-Clavien classification could provide fundamental information, under a uniform definition and classification system, on postoperative complications in patients with giant cell tumor of bone in terms of incidence, type of intervention for complication control, risk factors, and impact on functional outcome. The data are useful not only for preoperative evaluation for the risk of complications under specific conditions but also for quality assessment of surgery for giant cell tumor of bone., Competing Interests: Declaration of competing interest The authors declare that they have no conflicts of interest., (Copyright © 2021 The Japanese Orthopaedic Association. Published by Elsevier B.V. All rights reserved.)

Background: The most common reconstruction method for bone defects caused by giant cell tumor of bone (GCTB) is cement packing combined with subchondral bone grafting and extra fixation. However, this method has several limitations involving bone cement and bone graft, which may lead to poor prognosis and joint function. A titanium-based 3D-printed strut-type prosthesis, featured with excellent biocompatibility and osseointegration ability, was developed for this bone defect in our institution. The goal of this study is to comparatively analyze the biomechanical performance of reconstruction methods aimed at the identification of better operative strategy., Methods: Four different 3D finite element models were created. Model #1: Normal femur; Model #2: Femur with tumorous cavity bone defects in the distal femur; Model #3: Cavity bone defects reconstructed by cement packing combined with subchondral bone grafting and extra fixation; Model #4: Cavity bone defects reconstructed by 3D-printed strut-type prosthesis combined with subchondral bone grafting. The femoral muscle multiple forces were applied to analyze the mechanical difference among these models by finite element analysis., Results: Optimal stress and displacement distribution were observed in the normal femur. Both reconstruction methods could provide good initial stability and mechanical support. Stress distributed unevenly on the femur repaired by cement packing combined with subchondral bone grafting and extra fixation, and obvious stress concentration was found around the articular surface of this femur. However, the femur repaired by 3D-printed strut-type prosthetic reconstruction showed better performance both in displacement and stress distribution, particularly in terms of the protection of articular surface and subchondral bone., Conclusions: 3D-printed strut-type prosthesis is outstanding in precise shape matching and better osseointegration. Compared to cement packing and extra fixation, it can provide the almost same support and fixation stiffness, but better biomechanical performance and protection of subchondral bone and articular cartilage. Therefore, 3D-printed strut-type prosthetic reconstruction combined with subchondral bone grafting may be evaluated as an alternative for the treatment of GCTBs in distal femur., (© 2022. The Author(s).)

Aims: Intralesional curettage is a commonly used treatment for primary bone tumors. However, local recurrence of tumors after curettage remains a major challenge., Questions: (1) Is blood pressure related to local recurrence after intralesional curettage for benign or intermediate bone tumors? (2) What's the impact of tourniquet usage on the risk of recurrence from high blood pressure?, Methods: This retrospective study evaluated patients receiving intralesional curettage for primary bone tumors from January 2011 to January 2015. A total of 411 patients with a minimum five-year follow-up were included for analysis. Demographic and disease-related variables were first assessed in univariable analyses for local recurrence risk. When a yielded p-value was < 0.2, variables were included in multivariable analyses to identify independent risk factors for local recurrence. Patients were then stratified by tourniquet usage (use/non-use), and risk from high blood pressure was evaluated in both subgroups., Results: At an average follow-up of 6.8 ± 1.0 years, 63 of 411 patients (15.3%) experienced local recurrence. In multivariable analyses, local recurrence was associated with age (OR, 0.96; 95% CI, 0.94-0.99; p = 0.005); tumor type; lesion size (> 5 cm: OR, 3.58; 95% CI, 1.38-9.33; p = 0.009); anatomical site (proximal femur: OR, 2.49; 95% CI, 1.21-5.15; p = 0.014; proximal humerus: OR, 3.34; 95% CI, 1.61-6.92; p = 0.001); and preoperative mean arterial pressure (> 110 mmHg: OR, 2.61; 95% CI, 1.20-5.67; P = 0.015). In subgroup analyses, after adjusting for age, tumor type, lesion size, and anatomical site, tourniquet use modified the preoperative mean arterial pressure - recurrence relationship: when tourniquet was not used, preoperative mean arterial pressure predicted local recurrence (95-110 mmHg, 4.13, 1.42-12.03, p = 0.009; > 110 mmHg, 28.06, 5.27-149.30, p < 0.001); when tourniquet was used, preoperative mean arterial pressure was not related to local recurrence (all p values > 0.05)., Conclusions: A high preoperative blood pressure was related to local recurrence after intralesional curettage for primary bone tumors in our study. Tourniquet usage and controlling blood pressure might be beneficial for reducing local recurrence in patients scheduled to receive intralesional curettage for primary bone tumor treatment., Level of Evidence: Level IV, hypothesis-generating study., (© 2022. The Author(s).)

Background: Giant cell tumor of bone (GCTB) is an intermediate tumor commonly arising from the epiphysis of the distal femur and proximal tibia. Standard GCTB treatment is joint-preserving surgery performed using thorough curettage and the filling of the cavity with allo-, auto-, polymethyl methacrylate (PMMA), or synthetic bone graft. Calcium phosphate cement (CPC) is an artificial bone substitute, which has the benefit of being able to adjust defects, consequently inducing immediate mechanical strength, and promoting biological healing. Secondary osteoarthritis may occur following GCTB treatment and may need additional surgery if severe. However, details regarding surgery for secondary osteoarthritis have not been fully elucidated. There are no reports on the use of total knee arthroplasty (TKA) for the treatment of secondary osteoarthritis following CPC packing. The insertion of an alignment rod is a standard procedure in TKA; however, it was difficult to perform in this case due to CPC. Therefore, we used a computed tomography (CT)-free navigation system to assist the distal femur cut. This study presents a knee joint secondary osteoarthritis case following CPC packing for GCTB curettage that was treated with standard TKA., Case Presentation: A 67-year-old Japanese woman, who was previously diagnosed with left distal femur GCTB and was treated by curettage and CPC packing 7 years ago, complained of severe knee pain. Left knee joint plain radiography revealed Kellgren and Lawrence (K-L) grade 4 osteoarthritis without evidence of tumor recurrence. Therefore, she was scheduled for TKA. There are no reports on the cutting of a femoral condyle surface with massive CPC with accurate alignment. Because it is difficult to insert the alignment rod intramedullary and cut the femoral condyle with CPC, we planned CT-free navigation-guided surgery for accurate bone cutting using an oscillating tip saw system to prevent CPC cracks. We performed standard TKA without complications, as planned. Postoperative X-ray showed normal alignment. Knee Society Knee Score (KSKS) and Knee Society Function Score (KSFS) ameliorated from 27 and 29 to 64 and 68, respectively The patient can walk without a cane postoperatively., Conclusion: There was no report about the surface TKA guided by CT-free navigation after primary GCT surgery with CPC. We believe that this case report will help in planning salvage surgery for secondary osteoarthritis after CPC packing., (© 2022. The Author(s).)

Purpose: Giant cell tumour (GCT) of the bone is a benign but locally aggressive tumour, commonly occurs at the metaphyseal-epiphyseal junction of the distal femur, proximal tibia, and distal radius. For Campanacci grade II and III lesions of the distal radius and in cases of recurrence, we usually carry out wide resection and reconstruction. There are numerous publications on the treatment of GCT of the distal radius. Still, reports on the functional outcome using non-vascularized fibular graft arthroplasty without fusion remain limited., Method: We reviewed patients who underwent wide resection and non-vascularized fibular graft arthroplasty from 2007 to May 2014. The assessment was done with Musculoskeletal Tumour Society Score (MSTS), Toronto Extremities Scoring System (TESS) and Disability of the Arm, Shoulder and Hand (DASH) scores. We also reviewed the radiographic results., Results: Fifteen patients were recruited, of whom 10 cases used ipsilateral fibular graft and five used contralateral non-vascularized fibular graft. The average duration of follow up was 6 years (3.25-9.92 years). The average grip strength was 48.1% compared to the non-operated hand. The average MSTS score was 78.4 %, TESS score was 84%, and DASH score was 25.2. The average time to radiological union was 12.5 weeks. 64% (29-78%) of the range of movement is preserved compared to the normal side. The complication rate was 20%., Conclusion: Fibula autograft arthroplasty is a feasible method of reconstruction after distal radius resection with good functional outcomes.

PURPOSE OF THE STUDY The preferred treatment of giant cell tumor of bone is curettage with the use of local adjuvant. If the tumor spreads beyond the bone into soft tissues, en bloc excision should be performed. Intralesional curettage allows joint preservation, but it is associated with a high recurrence rate. The purpose of the study was to identify the risk factors for local recurrence and to compare the functional outcomes after both types of surgical procedures. MATERIAL AND METHODS The group included 16 patients (5 women, 11 men) with giant cell tumor of bone in distal forearm treated at the First Department of Orthopedic Surgery, St. Anne s University Hospital Brno in 2005-2019. The mean age of patients was 38 years (22-53). The follow-up period was 6.75 years (2-15) on average. The most common location of the tumor was distal radius (14). In 6 patients denosumab treatment was indicated. Based on the obtained data, we compared the effects of gender, Campanacci grade, type of surgery and administration of denosumab on the risk of local recurrence. The functional outcomes were evaluated retrospectively based on the Musculoskeletal Tumor Society scoring system for upper limb salvage surgeries. RESULTS Resection and reconstruction using an osteocartilaginous allograft was performed in 9 patients. Seven patients were treated with tumor curettage with bone cement used to fill the cavity. The group of patients who underwent curettage showed a significantly higher mean MSTS score 89% compared to the group of patients with resection with the mean MSTS score 66% (P < 0.05). Local tumor recurrence was reported in 3 patients (18.75%). No statistically significant difference was found in gender, tumor grade, radicality of surgery or administration of targeted therapy with respect to the incidence of local recurrence. Altogether 6 complications (37.5%) were observed in the group. DISCUSSION The treatment of a giant cell tumor of bone aims to completely remove the tumor and to preserve the best possible function of the limb. The complications in distal forearm involve particularly an increase incidence of local recurrence and painful or limited range of motion of the wrist. Whereas curettage with the use of local adjuvant is burdened with a higher recurrence rate, resection with allograft reconstruction of bone defect is usually associated with poorer functional outcomes. CONCLUSIONS Tumor curettage using local adjuvant is preferred in a well-circumscribed tumor and offers an excellent functional outcome. En bloc tumor resection and reconstruction using an osteocartilaginous allograft is a suitable treatment option for a locally advanced tumor with a low risk of local recurrence. Key words: giant cell tumor of bone, distal radius, distal ulna, curettage, osteocartilaginous allograft.

Background: Femoral bone deficiency is a challenging problem in revision proximal femoral replacement. The purpose of this study is to evaluate the clinical and radiological outcomes of revision proximal femoral replacement as a salvage treatment for severe bone loss after oncologic proximal replacement surgery in patient with benign giant cell tumor of bone., Methods: 16 patients (6 men and 10 women) were included in this retrospective study, with a mean age of 46.6 year at the time of revision surgery. All patients underwent revision proximal femoral replacement with the use of modular prosthesis and cortical strut allografts. The modified Harris Hip Score, Short Form 36, and musculoskeletal Tumor Society Score were used for patient evaluation. Regular follow-up was performed to evaluate the recurrence and metastases rate, limb function, and long-term complications of patients., Results: The average follow-up was 46.3 months (range, 26-75 months), during which there was no local recurrence and metastases of patient. At the latest follow-up, the mean modified Harris Hip Score was 70.6 points, which was significantly improved compared with that of preoperative ( p < 0.05). The final follow-up results of Short Form 36, Musculoskeletal Tumor Society Score, and limb-length discrepancy were also significantly improved compared to that of preoperative ( p < 0.05). At the latest follow-up, the implanted femoral stems were all stable and all cortical strut allografts were also incorporated to their own bone., Conclusion: Using modular prosthesis and cortical strut allografts in revision, proximal femur replacement is an acceptable procedure for relatively young patient with severe proximal femoral bone loss after oncologic surgery with benign giant cell tumor of bone. More attentions should be paid to reduce the risk of complications in these complex reconstructions.

The hand is an extremely rare site for giant cell tumor (GCT). There are only a few reported cases of GCT including the hand, and even fewer reporting involvement of phalanges. GCTs in small bones are typically more aggressive and have higher local recurrence and rate of metastasis in younger patients compared to long bone involvement, so the treatment is more clinically challenging in the hand. In this study, we present the management of giant cell tumors of the proximal phalanxin two patients treated with two different method; ray resection and arthrodesis using an iliac crest graft. Key words: giant cell tumor, phalanx, hand, recurrence.

Background: There is no standard treatment for giant cell tumors of the sacrum. We compared the outcomes and complications in patients with sacral giant cell tumors who underwent intralesional nerve-sparing surgery with or without (neo-) adjuvant therapies versus those who underwent non-surgical treatment (denosumab therapy and/or embolization)., Methods: We retrospectively investigated 15 cases of sacral giant cell tumors treated at two institutions between 2005 and 2020. Nine patients underwent intralesional nerve-sparing surgery with or without (neo-) adjuvant therapies, and six patients received non-surgical treatment. The mean follow-up period was 85 months for the surgical group (range, 25-154 months) and 59 months (range, 17-94 months) for the non-surgical group., Results: The local recurrence rate was 44% in the surgical group, and the tumor progression rate was 0% in the non-surgical group. There were two surgery-related complications (infection and bladder laceration) and three denosumab-related complications (apical granuloma of the tooth, stress fracture of the sacroiliac joint, and osteonecrosis of the jaw). In the surgical group, the mean modified Biagini score (bowel, bladder, and motor function) was 0.9; in the non-surgical group, it was 0.5. None of the 11 female patients became pregnant or delivered a baby after developing a sacral giant cell tumor., Conclusions: The cure rate of intralesional nerve-sparing surgery is over 50%. Non-surgical treatment has a similar risk of complications to intralesional nerve-sparing surgery and has better functional outcomes than intralesional nerve-sparing surgery, but patients must remain on therapy over time. Based on our results, the decision on the choice of treatment for sacral giant cell tumors could be discussed between the surgeon and the patient based on the tumor size and location., (© 2021. The Author(s).)

Objectives: To explore the predictive value of preoperative imaging in patients with spinal giant cell tumor of bone (GCTB) for postoperative recurrence and risk stratification., Methods: Clinical data for 62 cases of spinal GCTB diagnosed and treated at our hospital from 2008 to 2018 were identified. All patients were followed up for more than 2 years according to the clinical guidelines after surgery. Medical history data including baseline demographic and clinical characteristics, computed tomography (CT) and magnetic resonance imaging (MRI) findings of recurrent and non-recurrent patients were compared. Two musculoskeletal radiologists read the images and were blinded to the clinical data. The imaging features associated with postoperative recurrence were analyzed by multivariate logistic regression, and receiver operating characteristic (ROC) curve analysis was performed to determine the optimal cutoff value of the largest lesion diameter predicting recurrence after surgery., Results: According to whether the disease recurred within the follow-up period, patients were divided into the recurrence group and the non-recurrence group. Of 62 patients (29 males and 33 females), 17 had recurrence and 45 did not. The recurrence rate was 27.4%. The mean follow-up time was 73.66 (± 32.92) months. The three major treatments were total en bloc spondylectomy (n = 26), intralesional spondylectomy (n = 20), and curettage(n = 16). A total of 16 CT and MRI features were analyzed. A univariate analysis showed no significant difference in age, sex, treatment, multi-vertebral body involvement, location, boundary, expansile mass, residual bone crest, paravertebral soft tissue mass, CT value, and MRI signal on T1-weighted imaging (WI), T2-WI, and T2-WI fat suppression (FS) sequences (P > 0.05). The largest lesion diameter [(4.68 ± 1.79) vs (5.92 ± 2.17) cm, t = 2.287, P = 0.026] and the vertebral compression fracture (51% vs 82%, χ 2  = 5.005, P = 0.025) were significantly different between the non-recurrence and recurrence groups. Logistic regression analysis showed that both largest lesion diameter (odds ratio [OR], 1.584; 95% confidence interval [CI], 1.108-2.264; P = 0.012) and compression fracture (OR, 8.073; 95%CI, 1.481-11.003; P = 0.016) were independent predictors of postoperative recurrence. When we set the cutoff value for the largest lesion diameter at 4.2 cm, the sensitivity and specificity for distinguishing the recurrence and non-recurrence of GCTB were 94.1% and 42.2%, respectively, and the area under the curve (AUC) was 0.671. The combined model achieved a sensitivity, specificity and accuracy of 47.1%, 97.8% and 83.9%, respectively., Conclusions: In spinal GCTB, maximum lesion diameter and the vertebral compression fracture are associated with tumor recurrence after surgery, which may provide helpful information for planning personalized treatment., (© 2021 The Authors. Orthopaedic Surgery published by Chinese Orthopaedic Association and John Wiley & Sons Australia, Ltd.)

In recent years, denosumab has been used to treat giant cell tumour of bone (GCTB) not only in cases where surgery is complicated but also preoperatively to decrease the preoperative grade or to facilitate surgery for Campanacci grade II and III cases. However, there are no clear protocols regarding the preoperative use of denosumab before en bloc resection. There are a few reports of recurrent cases after en bloc resection; however, the association with the use of denosumab is unknown. We present the clinical, radiological and histopathological findings of a case of Campanacci grade III GCTB at the distal end of the ulna, which resulted in soft tissue recurrence after en bloc resection with the preoperative use of denosumab., Competing Interests: Competing interests: None declared., (© BMJ Publishing Group Limited 2021. Re-use permitted under CC BY-NC. No commercial re-use. See rights and permissions. Published by BMJ.)

Approximately 2% to 9% of giant cell tumor of bone (GCTB) metastasizes systemically, mainly to the lungs. The biological behaviors and clinical courses of lung metastases are difficult to predict, and their treatment recommendations vary, including metastasectomy and non-metastasectomy with chemotherapy (denosumab, interferon-alfa, bisphosphonates), with radiation therapy, or with observation alone. However, it is unclear whether metastasectomy for GCTB lung metastases decreases the mortality rate of these patients. Therefore, the authors performed this systematic review to compare metastasectomy and non-metastasectomy for GCTB patients with operable lung metastasis. Of the 919 relevant studies, 16 studies (138 patients) were included for analysis; 61.6% of patients had metastasectomy and 38.4% had non-metastasectomy. Analysis showed that mortality rates were similar for the patients who had metastasectomy compared with those who did not; the proportion of patients who died of disease was 7.1% in the metastasectomy group and 17.0% in the non-metastasectomy group, with an overall pooled odds ratio of 0.64 ( P =.36). Therefore, physicians should reconsider the potential risks and benefits of metastasectomy for patients with GCTB and lung metastasis, because metastasectomy does not reduce the mortality rate in these patients. [ Orthopedics . 2021;44(6):e707-e712.].

We performed a systematic analysis of existing studies to determine whether preoperative denosumab reduces the risk of local recurrence for patients with giant cell tumor of bone treated with en bloc resection and to address the optimal duration of preoperative denosumab with respect to the risk of local recurrence after en bloc resection. Denosumab did not decrease the risk of local recurrence after en bloc resection; the proportion of patients with local recurrence was 3.6% (2 of 56) in the en bloc resection with preoperative denosumab group vs 14.2% (40 of 280) in the en bloc resection alone group, with an overall pooled odds ratio of 0.76 ( P =.67). Meta-regression models revealed no association between the duration of preoperative denosumab and the odds of local recurrence after en bloc resection ( P =.83). Administration of denosumab for 3 months before en bloc resection is appropriate for sufficient bone hardening to reduce tumor cell spillage and does not result in denosumab-related complications. [ Orthopedics . 2021;44(6):326-332.].

Background: Patients with a bone sarcoma who undergo limb-sparing surgery and reconstruction with a tumor prosthesis in the lower extremity have been shown to have reduced self-reported physical function and quality of life (QoL). To provide patients facing these operations with better expectations of future physical function and to better evaluate and improve upon postoperative interventions, data from objectively measured physical function have been suggested., Questions/purposes: We sought to explore different aspects of physical function, using the International Classification of Functioning, Disability, and Health (ICF) as a framework, by asking: (1) What are the differences between patients 2 to 12 years after a bone resection and reconstruction surgery of the hip and knee following resection of a bone sarcoma or giant cell tumor of bone and age-matched controls without walking limitations in ICF body functions (ROM, muscle strength, pain), ICF activity and participation (walking, getting up from a chair, daily tasks), and QoL? (2) Within the patient group, do ICF body functions and ICF activity and participation outcome scores correlate with QoL?, Methods: Between 2006 and 2016, we treated 72 patients for bone sarcoma or giant cell tumor of bone resulting in bone resection and reconstruction with a tumor prosthesis of the hip or knee. At the timepoint for inclusion, 47 patients were alive. Of those, 6% (3 of 47) had undergone amputation in the lower limb and were excluded. A further 32% (14 of 44) were excluded because of being younger than 18 years of age, pregnant, having long transportation, palliative care, or declining participation, leaving 68% (30 of 44) for analysis. Thus, 30 patients and 30 controls with a mean age of 51 ± 18 years and 52 ± 17 years, respectively, were included in this cross-sectional study. Included patients had been treated with either a proximal femoral (40% [12 of 30]), distal femoral (47% [14 of 30]), or proximal tibia (13% [4 of 30]) reconstruction. The patients were assessed 2 to 12 years (mean 7 ± 3 years) after the resection-reconstruction. The controls were matched on gender and age (± 4 years) and included if they considered their walking capacity to be normal and had no pain in the lower extremity. Included outcome measures were: passive ROM of hip flexion, extension, and abduction and knee flexion and extension; isometric muscle strength of knee flexion, knee extension and hip abduction using a hand-held dynamometer; pain intensity (numeric rating scale; NRS) and distribution (pain drawing); the 6-minute walk test (6MWT); the 30-second chair-stand test (CST); the Toronto Extremity Salvage Score (TESS), and the European Organisation for Research and Treatment of Cancer Quality of Life Questionnaire (EORTC QLQ-C30). The TESS and the EORTC QLQ-C30 were normalized to 0 to 100 points. Higher scoring represents better status for TESS and EORTC global health and physical functioning scales. Minimum clinically important difference for muscle strength is 20% to 25%, NRS 2 points, 6MWT 14 to 31 meters, CST 2 repetitions, TESS 12 to 15 points, and EORTC QLQ-C30 5 to 20 points., Results: Compared with controls, the patients had less knee extension and hip abduction strength in both the surgical and nonsurgical limbs and regardless of reconstruction site. Mean knee extension strength in patients versus controls were: surgical limb 0.9 ± 0.5 Nm/kg versus 2.1 ± 0.6 Nm/kg (mean difference -1.3 Nm/kg [95% CI -1.5 to -1.0]; p < 0.001) and nonsurgical limb 1.7 ± 0.6 Nm/kg versus 2.2 ± 0.6 Nm/kg (mean difference -0.5 Nm/kg [95% CI -0.8 to -0.2]; p = 0.003). Mean hip abduction strength in patients versus controls were: surgical limb 1.1 ± 0.4 Nm/kg versus 1.9 ± 0.5 Nm/kg (mean difference -0.7 Nm/kg [95% CI -1.0 to -0.5]; p < 0.001) and nonsurgical limb 1.5 ± 0.4 Nm/kg versus 1.9 ± 0.5 Nm/kg (-0.4 Nm/kg [95% CI -0.6 to -0.2]; p = 0.001). Mean hip flexion ROM in patients with proximal femoral reconstructions was 113° ± 18° compared with controls 130° ± 11° (mean difference -17°; p = 0.006). Mean knee flexion ROM in patients with distal femoral reconstructions was 113° ± 29° compared with patients in the control group 146° ± 9° (mean difference -34°; p = 0.002). Eighty-seven percent (26 of 30) of the patients reported pain, predominantly in the knee, anterior thigh, and gluteal area. The patients showed poorer walking and chair-stand capacity and had lower TESS scores than patients in the control group. Mean 6MWT was 499 ± 100 meters versus 607 ± 68 meters (mean difference -108 meters; p < 0.001), mean CST was 12 ± 5 repetitions versus 18 ± 5 repetitions (mean difference -7 repetitions; p < 0.001), and median (interquartile range) TESS score was 78 (21) points versus 100 (10) points (p < 0.001) in patients and controls, respectively. Higher pain scores correlated to lower physical functioning of the EORTC QLQ-C30 (Rho -0.40 to -0.54; all p values < 0.05). Less muscle strength in knee extension, knee flexion, and hip abduction correlated to lower physical functioning of the EORTC QLQ-C30 (Rho 0.40 to 0.51; all p values < 0.05)., Conclusion: This patient group demonstrated clinically important muscle weaknesses not only in resected muscles but also in the contralateral limb. Many patients reported pain, and they showed reductions in walking and chair-stand capacity comparable to elderly people. The results are relevant for information before surgery, and assessments of objective physical function are advisable in postoperative monitoring. Prospective studies evaluating the course of physical function and which include assessments of objectively measured physical function are warranted. Studies following this patient group with repetitive measures over about 5 years could provide information about the course of physical function, enable comparisons with population norms, and lead to better-designed, targeted, and timely postoperative interventions., Level of Evidence: Level III, therapeutic study., Competing Interests: All ICMJE Conflict of Interest Forms for authors and Clinical Orthopaedics and Related Research® editors and board members are on file with the publication and can be viewed on request., (Copyright © 2021 by the Association of Bone and Joint Surgeons.)

Objective: To evaluate the prognostic significance of neutrophil-to-lymphocyte ratio (NLR) in giant cell tumor of bone (GCT)., Methods: The patients with GCT were identified in the hospital records and pre-treatment complete blood count results were acquired retrospectively. Whether preoperative NLR lymphocyte-to-monocyte ratio (LMR) and platelet-to-lymphocyte ratio (PLR) values had prognostic significance in predicting recurrence was evaluated by Receiver operating curve (ROC) analysis. Furthermore, the prognostic value of NLR was evaluated by Multivariable Cox Regression analysis., Results: There were 96 patients with GCT. It was found that only NLR values had prognostic significance for predicting recurrence (AUC:0.647; 95% CI:0.533-0.762; P=0.021). The statistically significant cut-off value of NLR for predicting recurrence was ≥2.25. NLR was ≥2.25 in 51% (n = 49) of patients. Multivariable analysis showed that NLR ≥2.25 (HR=2.9, 95% CI:1.3-6.6; p=0.009) and lung metastasis (HR=7.9, 95% CI:2.2-28.2; p=0.001) were independent factors of recurrence. In patients with lung metastasis and patients with NLR ≥2.25, recurrence was observed in a sooner period (Log rank test; p=0.001; p=0.009, respectively)., Conclusion: Our findings showed that NLR is a new and promising inflammation-based prognostic factor in GCT patients., (© 2021 Yapar A et al.)

Background: Ten years ago, we reported the results of a procedure in which we translocated the ipsilateral ulna as a vascularized autograft to reconstruct defects of the distal radius after tumor resection, with excellent functional results. At that time, wrist arthrodesis was achieved by aligning the translocated ulna with the scapholunate area of the carpus and usually the third metacarpal. This resulted in wrist narrowing. We then wondered if aligning the translocated ulna with the scaphoid and the second metacarpal would result in ulnar deviation and thereby improve grip strength. We believed lateralization would reduce the wrist narrowing that occurs with fusion to the third metacarpal and would make the cosmesis more acceptable. We also modified the incision to dororadial to make the scar less visible and thus improve the cosmesis., Questions/purposes: (1) Is there an objective improvement in grip strength and functional scores (Musculoskeletal Tumor Society [MSTS] and Mayo wrist) when the translocated ulna is lateralized and the wrist is fused with the translocated ulna and aligned with the second metacarpal versus when the translocated ulna is aligned with the third metacarpal? (2) Did lateralization caused by the wrist fusion aligned with the second metacarpal minimize wrist narrowing as measured by the circumference compared with the fusion aligned with the third metacarpal?, Methods: From 2010 and 2018, we treated 40 patients with distal radius tumors at our institution, 30 of whom had a distal radius enbloc resection. Twenty-eight patients had an ipsilateral ulna translocation and wrist arthrodesis in which the radius and translocated ulna were aligned with either the second (n = 15) or the third (n = 13) metacarpals. Two patients in the second metacarpal group and three patients in the third metacarpal group were lost to follow-up before 24 months after surgery and were excluded. A retrospective analysis of 23 patients (20 with giant cell tumors and three with malignant bone tumors) included a review of radiographs and institutional tumor database for surgical and follow-up records to study oncologic (local disease recurrence), reconstruction (union of osteotomy junctions, implant breakage or graft fracture, and wrist circumference), and functional outcomes (MSTS and Mayo wrist scores and objective grip strength assessment compared with the contralateral side). The results were compared for each study group (second metacarpal versus third metacarpal). There was no difference in the incidence of local recurrence or the time to union between the two groups. There were no implant breakages or graft fractures noted in either group., Results: Patients in the second metacarpal group lost less grip strength compared with the unoperated side in the third metacarpal group (median 12% [range -30% to 35%] versus median 28% [15% to 42%], difference of medians 16%; p = 0.006). There were no between-group differences in terms of MSTS (median 30 [24 to 30] versus median 26.5 [22 to 30], difference of medians 3.5; p = 0.21) or Mayo wrist scores (median 83 [65 to 100] versus median 72 [50 to 90], difference of medians 11; p = 0.10). The second metacarpal group also had less wrist narrowing as seen from the median difference in circumference between the operated and unoperated wrists (median narrowing 10 mm [3 to 35 mm] in the second metacarpal group versus median 30 mm [15 to 35 mm] in the third metacarpal group, difference of medians 20 mm; p = 0.04)., Conclusion: Wrist arthrodesis after ulna translocation with alignment of the translocated ulna and the second metacarpal provides a functional position with ulnar deviation that offers some improvement in grip strength but no improvement in the MSTS or Mayo scores. Radialization/lateralization of the translocated ulna achieved from the alignment with the second metacarpal decreases the reduction in the wrist circumference and therefore reduces wrist narrowing., Level of Evidence: Level III, therapeutic study., Competing Interests: Each author certifies that neither he nor she, nor any members of his or her immediate family, has funding or commercial associations (consultancies, stock ownership, equity interest, patent/licensing arrangements, etc.) that might pose a conflict of interest in connection with the submitted article. All ICMJE Conflict of Interest Forms for authors and Clinical Orthopaedics and Related Research® editors and board members are on file with the publication and can be viewed on request., (Copyright © 2020 Wolters Kluwer Health, Inc. All rights reserved.)

BACKGROUND Giant cell tumor of bone (GCTB) is a locally aggressive, intermediate tumor that rarely metastasizes. GCTB typically affects the ends of long bones and rarely involves the ribs. Curettage is typically the treatment of choice for GCTB in long bones. However, the optimal treatment of GCTB in ribs remains unclear. We report the case of a patient with asymptomatic GCTB of the first rib that was successfully treated with combined preoperative denosumab therapy and surgery via a transmanubrial approach without resection of the clavicle. CASE REPORT A healthy 27-year-old woman presented with a bone tumor involving the left first rib that was incidentally discovered on routine chest X-ray. Histological examination of core-needle biopsy specimens of the lesion led to a pathological diagnosis of GCTB. After preoperative denosumab treatment for 6 months, en bloc resection via a transmanubrial approach was performed. There were no serious postoperative complications. The patient remained free of symptoms and had no recurrence 4.5 years after surgery. CONCLUSIONS Compared with other ribs, masses located in the first rib can be challenging to treat surgically because of the clavicle and neighboring neurovascular structures. This report is the first to describe GCTB located on the anterior aspect of the first rib that was successfully treated with combined preoperative denosumab therapy and surgery via a transmanubrial approach, with no recurrence or functional impairment of the shoulder girdle.