Journal of Bone Oncology最新文献

{"title":"Efferocytosis-associated transcriptomic patterns characterize prognosis and immune landscape in osteosarcoma","authors":"Xueliang Song ,&nbsp;Xiaofan Tou ,&nbsp;Li Li ,&nbsp;Fengxian Wang ,&nbsp;Chengqun Qian ,&nbsp;Ru Wang ,&nbsp;Jiahong Shen","doi":"10.1016/j.jbo.2026.100743","DOIUrl":"10.1016/j.jbo.2026.100743","url":null,"abstract":"<div><h3>Background</h3><div>Osteosarcoma (OS) is the most common primary malignant bone tumor in adolescents, characterized by high heterogeneity and poor prognosis. Efferocytosis, the clearance of apoptotic cells, has been implicated in tumor progression and immune evasion, but its role in OS remains unclear.</div></div><div><h3>Methods</h3><div>We integrated TARGET-OS as a training cohort with three GEO datasets for validation. Efferocytosis pathways were quantified by ssGSEA, and WGCNA was applied to identify associated gene modules. Candidate genes were screened using univariate Cox regression, and a prognostic signature was developed with machine learning models and validated across cohorts. Functional enrichment, immune infiltration, and immunotherapy prediction analyses were performed. scRNA-seq from six OS patients and spatial transcriptomic profiling were further used to characterize the cellular distribution and communication of efferocytosis-related genes. The functions of MAGEA11 in OS were explored both in vivo and in vitro.</div></div><div><h3>Results</h3><div>The Brown module showed the strongest association with efferocytosis pathways. The StepCox + Ridge model achieved robust prognostic performance and stratified patients into risk groups with significantly different survival. Enrichment analysis revealed upregulated genes related to endothelial and nitric oxide pathways, while downregulated genes were linked to immune signaling and extracellular matrix remodeling. High-risk patients exhibited elevated M2 macrophages, altered checkpoint expression, and greater predicted sensitivity to immunotherapy. At the single-cell level, efferocytosis activity was enriched in OS cells, with MAGEA11 showing the highest expression. High-risk tumors displayed stronger intercellular signaling, particularly from OS cells and CAFs to macrophages and endothelial cells. Spatial transcriptomics confirmed enrichment of efferocytosis at tumor and interface regions, correlating positively with stromal and myeloid cells and negatively with T cells. Mechanistically, MAGEA11 promoted OS tumor growth, and drove a pro-efferocytic microenvironment by enhancing Gas6 secretion, which polarized macrophages toward an M2 phenotype and upregulated their efferocytosis receptors (MERTK/AXL).</div></div><div><h3>Conclusions</h3><div>We established an efferocytosis-related prognostic signature and elucidated its underlying mechanism wherein MAGEA11 promoted immunosuppression via a Gas6-MERTK/AXL-dependent efferocytosis circuit. This integrated study positions efferocytosis as a key driver of the OS microenvironment and a promising target for clinical intervention.</div></div>","PeriodicalId":48806,"journal":{"name":"Journal of Bone Oncology","volume":"57 ","pages":"Article 100743"},"PeriodicalIF":3.5,"publicationDate":"2026-01-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"146025341","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Clinicopathological features and survival disparities between axial and appendicular skeletal Angiosarcoma: A SEER-based analysis using competing risk models","authors":"Zhenliang Hao ,&nbsp;Xuewei Zhang ,&nbsp;Ning Yao ,&nbsp;Kai Li ,&nbsp;Luxin Lou ,&nbsp;Xuan Zheng ,&nbsp;Zhiming Guo ,&nbsp;Jiabin Chen ,&nbsp;Xuzhu Chen","doi":"10.1016/j.jbo.2026.100740","DOIUrl":"10.1016/j.jbo.2026.100740","url":null,"abstract":"<div><h3>Background</h3><div> <!-->Primary angiosarcoma of bone (ASB) is a rare and aggressive primary bone tumor. The prognostic significance of anatomical location (axial vs. appendicular skeleton) remains poorly defined. This study aimed to compare clinicopathological features and survival outcomes between these two groups.</div></div><div><h3>Methods</h3><div> <!-->Patients diagnosed with primary ASB (1975–2019) were identified from the Surveillance, Epidemiology, and End Results (SEER) database. Tumors were categorized as axial (spine, pelvis, ribs, skull) or appendicular (extremities). Overall survival was analyzed using Kaplan-Meier and Cox models. Cancer-specific mortality (CSM) was evaluated with Fine-Gray competing risk regression to calculate subdistribution hazard ratios (SHR).</div></div><div><h3>Results</h3><div> <!-->Among 458 patients, 162 (35.4%) had axial and 296 (64.6%) had appendicular tumors. Axial patients were older (median 63 vs. 46 years, p &lt; 0.001), had higher distant metastasis at diagnosis (43.2% vs. 29.1%, p = 0.002), and underwent surgery less frequently (37.0% vs. 81.1%, p &lt; 0.001). The 5-year overall survival was significantly worse for axial tumors (16.8% vs. 38.2%, p &lt; 0.001). The 5-year cumulative incidence of CSM was 69.5% for axial versus 50.8% for appendicular tumors (p &lt; 0.001). Multivariate analysis confirmed axial location as an independent predictor of higher CSM (SHR 1.62, 95% CI: 1.25–2.10, p &lt; 0.001) ,<!--> <!-->independent of the year of diagnosis.</div></div><div><h3>Conclusions</h3><div>Axial tumors represent a distinct high-risk subgroup characterized by limited surgical resectability and inferior survival. Anatomical location should be considered a critical stratification factor in clinical management and future trials.</div><div>Abbreviations: <strong>ASB,</strong> Primary angiosarcoma of the bone; <strong>SEER,</strong> Surveillance, Epidemiology, and End Results; <strong>OS,</strong> Overall Survival; <strong>CSS,</strong> Cancer-Specific Survival; <strong>CSD,</strong> Cancer-Specific Death; <strong>CIF,</strong> Cumulative Incidence Function; <strong>SHR,</strong> Subdistribution Hazard Ratio; <strong>CI,</strong> Confidence Interval; <strong>HR,</strong> Hazard Ratio.</div></div>","PeriodicalId":48806,"journal":{"name":"Journal of Bone Oncology","volume":"57 ","pages":"Article 100740"},"PeriodicalIF":3.5,"publicationDate":"2026-01-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"146079934","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Prospective external validation of a three-predictor frailty model for 90-day survival and complications following spinal metastasis surgery","authors":"Pedro Reggiani Anzuategui ,&nbsp;Glauco José Pauka Mello ,&nbsp;Ana Valéria Brunetti Rigolino ,&nbsp;Lucas Emanuel Sauer Larocca ,&nbsp;Cássio Zini ,&nbsp;Carmen Australia Paredes Marcondes Ribas","doi":"10.1016/j.jbo.2026.100739","DOIUrl":"10.1016/j.jbo.2026.100739","url":null,"abstract":"&lt;div&gt;&lt;h3&gt;Background context&lt;/h3&gt;&lt;div&gt;Surgical decision-making in patients with spinal metastases remains complex due to the need to balance potential surgical benefits with limited survival and common frailty. Predictive models can assist in this process, but their clinical utility is often limited by complexity and lack of validation.&lt;/div&gt;&lt;/div&gt;&lt;div&gt;&lt;h3&gt;Purpose&lt;/h3&gt;&lt;div&gt;To externally validate a simple three-predictor frailty model for 90-day survival and complications, and to compare its performance with other commonly used tools.&lt;/div&gt;&lt;/div&gt;&lt;div&gt;&lt;h3&gt;Study design/setting&lt;/h3&gt;&lt;div&gt;Prospective external validation study conducted at a single tertiary cancer center.&lt;/div&gt;&lt;/div&gt;&lt;div&gt;&lt;h3&gt;Patient sample&lt;/h3&gt;&lt;div&gt;A consecutive cohort of 126 patients who underwent open posterior surgery with instrumentation for spinal metastases from solid tumors between 2018 and 2024.&lt;/div&gt;&lt;/div&gt;&lt;div&gt;&lt;h3&gt;Outcome measures&lt;/h3&gt;&lt;div&gt;Primary outcomes were 90-day survival and the occurrence of postoperative complications. Secondary outcomes included 30-day, 180-day and overall survival. Model performance was evaluated through discrimination (AUC), risk stratification, accuracy for surgical indication and calibration.&lt;/div&gt;&lt;/div&gt;&lt;div&gt;&lt;h3&gt;Methods&lt;/h3&gt;&lt;div&gt;The Anzuategui model (three predictors: tumor growth rate, comorbidities, and lymphocyte count) was applied preoperatively, along with four other three-predictor models (Tomita, Modified Bauer, Van der Linden, and Sioutos). Discrimination was assessed using ROC curves. Risk stratification was evaluated using predefined low-, moderate-, and high-risk categories, analyzed through Kaplan–Meier curves and complication rates. Model accuracy for surgical indication was calculated using a 90-day survival threshold as the reference. Calibration for both 90-day survival and postoperative complications was performed by comparing category-specific predicted probabilities derived from the development cohort with observed event rates in the validation cohort.&lt;/div&gt;&lt;/div&gt;&lt;div&gt;&lt;h3&gt;Results&lt;/h3&gt;&lt;div&gt;The Anzuategui model demonstrated predictive performance for the primary outcomes comparable to the other models under evaluation. It achieved an AUC of 0.78 (95% CI: 0.70–0.85) for 90-day survival and 0.68 (95% CI: 0.59–0.76) for postoperative complications. Risk stratification showed clear separation between survival curves across the three predefined categories. Accuracy for predicting appropriate surgical indication was 70% (95% CI: 61–78), with a sensitivity of 64% and specificity of 85%. Tomita and Modified Bauer models showed comparable accuracy (75% and 74%, respectively) but lower specificity. Calibration indicated overestimation of 90-day mortality (intercept –1.75; slope 2.05) and modest miscalibration for postoperative complications (intercept –0.40; slope 0.67).&lt;/div&gt;&lt;/div&gt;&lt;div&gt;&lt;h3&gt;Conclusions&lt;/h3&gt;&lt;div&gt;The Anzuategui model demonstrated acceptable external performance, with greater validity for predicting 90-day survival than for post","PeriodicalId":48806,"journal":{"name":"Journal of Bone Oncology","volume":"57 ","pages":"Article 100739"},"PeriodicalIF":3.5,"publicationDate":"2026-01-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145982069","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Real-life use of bone-targeting agents for bone metastases in France between 2009 and 2018: Results of the OPTIMOS study","authors":"Cyrille B. Confavreux ,&nbsp;Béatrice Bouvard ,&nbsp;Nicolas Girard ,&nbsp;Pauline Bosco-Levy ,&nbsp;Clarisse Marchal ,&nbsp;Maeva Nolin ,&nbsp;Eric Lehmann ,&nbsp;Gaelle Desameric ,&nbsp;Manon Belhassen","doi":"10.1016/j.jbo.2025.100738","DOIUrl":"10.1016/j.jbo.2025.100738","url":null,"abstract":"<div><h3>Aim</h3><div>To determine the use of bone-targeting agents (BTAs) in clinical practice in France and the occurrence of skeletal-related events (SREs) in cancer patients with bone metastases.</div></div><div><h3>Methods</h3><div>This study analysed data, recorded prospectively in a French National Health Insurance database, for patients who had a first diagnosis of bone metastases between 2009 and 2018.</div></div><div><h3>Results</h3><div>A total of 6,663 patients were analysed (mean age 69.7 ± 13.2 years, 53.2 % male) corresponding to 2,363 bone metastases only patients and 4,300 patients with SREs at inclusion. The most frequent primary cancers were breast (15.8 %), prostate (13.4 %), lung (12.6 %) and digestive cancer (10.6 %). Six-hundred and twenty-one patients (9.3 %) were treated with BTAs (52.7 % with denosumab). Median [IQR] time between inclusion and BTA initiation was similar with denosumab (3.3 months [1.2–7.9]) and bisphosphonates (3.3 months [1.2–8.7]). Patients with a SRE at inclusion and early BTA initiation (≤3 months) had a significative lower incidence of a second SRE at 12 months than those with late initiation (13.6 % [95 %CI: 8.1–20.4] vs. 21.6 % [14.8–29.2] respectively; p &lt; 0.001).</div></div><div><h3>Conclusion</h3><div>BTAs are underused in bone metastases patients in France. There is an urgent need to optimise bone metastases management in accordance with ESMO 2020 guidelines.</div></div>","PeriodicalId":48806,"journal":{"name":"Journal of Bone Oncology","volume":"56 ","pages":"Article 100738"},"PeriodicalIF":3.5,"publicationDate":"2026-01-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145939112","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Pilot study of separation surgery with intraoperative radiotherapy (IORT) for spine metastasis","authors":"Baiyi Liu ,&nbsp;Dongsheng Wang ,&nbsp;Jian Zhang ,&nbsp;Bo Huang ,&nbsp;Mingying Geng ,&nbsp;Peng Liu ,&nbsp;Yaoyao Liu","doi":"10.1016/j.jbo.2025.100737","DOIUrl":"10.1016/j.jbo.2025.100737","url":null,"abstract":"<div><h3>Objective</h3><div>This study aimed to introduce a novel modified separation surgery combined with intraoperative radiotherapy (MSS-IORT) treatment strategy for spinal metastasis and evaluate its efficacy and safety.</div></div><div><h3>Methods</h3><div>A prospective study was conducted from January 2023 to June 2024. Patients with spinal metastasis exhibiting epidural spinal cord compression (ESCC) ≥ 2 grades and spinal instability neoplastic score (SINS) ≥ 7 were enrolled and underwent MSS-IORT. During the procedure, a dose of 8–10 Gy of IORT was administered to the tumor-invaded vertebrae segments during modified separation surgery. Pain intensity was assessed using the visual analog scale (VAS) preoperatively and at 1 week, 3 months, 6 months, and 12 months postoperatively. Neurological function was evaluated <em>via</em> the Frankel grade system, and functional status was measured using the Karnofsky performance scale (KPS) preoperatively and at 3, 6, and 12 months after surgery. Local control was evaluated based on X-ray, CT, or MRI examination. Survival time and perioperative complications were also documented.</div></div><div><h3>Results</h3><div>A total of 38 patients (median age: 60 years) with 46 involved vertebrae were treated with MSS-IORT. The mean operation time was 277.5 min, and the mean blood loss was 750 ml. After a mean follow-up of 174.5 days, the VAS score decreased significantly postoperatively and continued to decline over time. The KPS score increased significantly at 6 and 12 months, and the Frankel grade significantly improved at 12 months. Local control failure occurred in 3 patients, and 13 experienced adverse events without IORT.</div></div><div><h3>Conclusion</h3><div>The MSS-IORT strategy demonstrates both safety and efficacy, representing a promising treatment option for spine metastases, particularly in patients with ESCC grades ≥ 2 and SINS ≥ 7.</div></div>","PeriodicalId":48806,"journal":{"name":"Journal of Bone Oncology","volume":"56 ","pages":"Article 100737"},"PeriodicalIF":3.5,"publicationDate":"2025-12-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145939114","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Doxorubicin induces bone loss and modifies multiple cell populations in vivo – Implications for modelling of bone metastasis","authors":"Veli Kaan Aydin ,&nbsp;Lubaid Saleh ,&nbsp;Penelope Dawn Ottewell ,&nbsp;Ingunn Holen","doi":"10.1016/j.jbo.2025.100736","DOIUrl":"10.1016/j.jbo.2025.100736","url":null,"abstract":"<div><div>Doxorubicin (DOX), commonly used to treat breast cancer, is associated with cardiotoxicity and has negative effects on other organ systems, including the skeleton. DOX-induced bone damage has been demonstrated in murine models; however, results are conflicting due to the use of different doses, schedules, and rat/mouse strains. As DOX is used to limit tumour progression in models of skeletal metastasis, it is paramount to determine how the agent affects the bone microenvironment in the relevant mouse strains, to enable correct interpretation of DOX effects in tumour studies. We have therefore investigated the effects of DOX on bone structure and a range of bone and bone marrow cell populations, comparing immunocompetent and immunocompromised mice.</div><div>Groups of 7-week-old female BALB/c and BALB/c Nude mice were treated with either saline (control), 4 or 6 mg/kg DOX weekly for four weeks. Effects on bone volume and structure was determined using <em>ex vivo</em> µCT, a panel of bone marrow cell populations were quantified by flow cytometry and osteoblast/osteoclast numbers were assessed using bone histomorphometry.</div><div>DOX caused trabecular bone loss, with immunocompetent BALB/c mice being more sensitive to DOX than the immunocompromised BALB/c nude counterparts. The 6 mg/kg dose of DOX altered the ratio of bone marrow immune and haematopoietic cell populations in both groups, increasing the numbers of hematopoietic cells and progenitors, decreasing B cells and increasing the number of neutrophils. Bone marrow macrophage and monocyte numbers were increased following DOX treatment in BALB/c nude mice only. Our data demonstrate that DOX impacts a number of cell types in the bone microenvironment, highlighting the importance of considering treatment-induced bone effects when using DOX in models of bone metastasis.</div></div>","PeriodicalId":48806,"journal":{"name":"Journal of Bone Oncology","volume":"56 ","pages":"Article 100736"},"PeriodicalIF":3.5,"publicationDate":"2025-12-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145798385","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Mri-based habitat and peritumoral radiomics for predicting the proliferative activity of stromal cells in giant cell tumor of bone","authors":"Jie Xia ,&nbsp;Kunming Jiang ,&nbsp;Jinyi Zhou ,&nbsp;Lei Cao ,&nbsp;Fan Xiao ,&nbsp;Jingxuan Jiang","doi":"10.1016/j.jbo.2025.100733","DOIUrl":"10.1016/j.jbo.2025.100733","url":null,"abstract":"<div><h3>Purpose</h3><div>This study aims to explore the feasibility of MRI-based habitat and peritumoral radiomics for predicting the proliferative activity of stromal cells in giant cell tumor of bone (GCTB)<strong>.</strong></div></div><div><h3>Material and methods</h3><div>A retrospective study was performed on 133 patients<!--> <!-->(102 in training cohort and 31 in validation cohort) diagnosed with GCTB<!--> <!-->from four centers. The tumor was meticulously segmented into three distinct habitat subregions using K-means clustering, incorporating a 1-pixel peritumoral expansion to capture the microenvironments surrounding the tumor. After feature extraction and selection, habitat, intratumoral and peritumoral models integrating three different machine learning classifiers were constructed respectively to identify GCTB patients with high and low proliferation. The performance of the models was assessed by receiver operating characteristic (ROC) curves, calibration plots, and decision curve analysis (DCA). SHAP analysis was utilized to enhance model interpretability.</div></div><div><h3>Results</h3><div>Among the eligible patients, 43 (32.3 %) diagnosed with high proliferative activity of stromal cells in GCTB by pathological diagnosis. Among all models tested in the validation cohort, the Logistic Regression (LR) algorithm for habitat model exhibited superior performance in the validation cohort (AUC: 0.956, 95 % CI: 0.887–1.000). The calibration curves and DCA exhibited fit for the habitat model while providing great clinical net benefit.</div></div><div><h3>Conclusion</h3><div>MRI-based habitat radiomics had the potential to predict the proliferative activity of stromal cells in GCTB. This model may help determine optimal treatment strategies and improve patient outcomes.</div></div>","PeriodicalId":48806,"journal":{"name":"Journal of Bone Oncology","volume":"56 ","pages":"Article 100733"},"PeriodicalIF":3.5,"publicationDate":"2025-12-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145749334","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Pathological fractures as an adverse prognostic factor in chondrosarcoma: Results of a systematic review, meta-analysis and institutional case series","authors":"Julian P. Maier ,&nbsp;Ida Peiss ,&nbsp;Felix Klingler ,&nbsp;Nikos Karvouniaris ,&nbsp;Kilian Reising ,&nbsp;Hagen Schmal ,&nbsp;Georg W. Herget","doi":"10.1016/j.jbo.2025.100735","DOIUrl":"10.1016/j.jbo.2025.100735","url":null,"abstract":"<div><h3>Background</h3><div>Pathological fractures are recognized as a potential, adverse prognostic factor in primary bone sarcomas. While some studies suggest higher recurrence rates and reduced survival, robust data specific to chondrosarcoma (CS) remain limited. This study evaluates the prognostic relevance of pathological fractures (PF) in CS patients.</div></div><div><h3>Methods</h3><div>A systematic review and <em>meta</em>-analysis were conducted according to PRISMA guidelines. Eligible studies included patients with CS of the bone, with comparative groups based on fracture status and oncological outcomes. Study design, quality, and endpoints were assessed, and pooled <em>meta</em>-analysis was conducted. Additionally, clinical data from an institutional cohort was analyzed retrospectively.</div></div><div><h3>Results</h3><div>Nine studies, with a total of 1,185 patients and 245 PF cases, met the inclusion criteria. Meta-analysis revealed that PF status was significantly associated with inferior survival at 1–5 years (pooled OR 0.40; 95 % CI 0.26–0.62; p &lt; 0.0001). The increased risk of death was particularly evident in dedifferentiated CS (pooled HR 1.96; 95 % CI 1.46–2.63; p &lt; 0.00001). Institutional data supported these findings, that PF was associated with worse overall survival (HR 3.90; 95 % CI 0.69–21.98; p = 0.12) and progression-free survival (HR 3.17; 95 % CI 0.58–17.36; p = 0.18), although significance was limited by sample size.</div></div><div><h3>Conclusions</h3><div>Pathological fractures entail a significantly adverse prognosis in chondrosarcoma, particularly in high-grade and dedifferentiated subtypes and within the first 5 years of follow-up. Our institutional data corroborates <em>meta</em>-analysis findings. These results underscore the importance of personalized surgical management and intensive outcome monitoring for CS patients presenting with PF.</div></div>","PeriodicalId":48806,"journal":{"name":"Journal of Bone Oncology","volume":"56 ","pages":"Article 100735"},"PeriodicalIF":3.5,"publicationDate":"2025-12-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145698199","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Malignant and metastatic giant cell tumors of bone; clinical course of primary or secondary malignant and pulmonary metastatic variants","authors":"Floortje G.M. Verspoor ,&nbsp;Gitte G.J. Krebbekx ,&nbsp;Mylene J.C. Duivenvoorden ,&nbsp;Vaiyapuri Sumathi ,&nbsp;Scott Evans","doi":"10.1016/j.jbo.2025.100728","DOIUrl":"10.1016/j.jbo.2025.100728","url":null,"abstract":"<div><h3>Aims</h3><div>Giant cell tumor of bone (GCTB) is a rare, locally aggressive, intermediate-grade neoplasm. While typically benign, GCTB can exhibit atypical behavior, including indolent pulmonary metastases and malignant transformation. This study characterizes malignant and metastatic GCTB, distinguishing primary malignant GCTB (PM-GCTB), secondary malignant GCTB (SM-GCTB), and secondary osteosarcoma, and evaluates their outcomes.</div></div><div><h3>Methods</h3><div>A retrospectively review of 520 patients with suspected GCTB treated between 1985–2021 was performed, with malignant cases followed through 2025. Diagnosis was confirmed histologically and, where available, by H3F3A/H3F3B mutation testing. PM-GCTB was defined as a de novo high-grade sarcoma. SM-GCTB and secondary osteosarcoma were defined as malignant transformations of previously benign, histologically proven GCTB. Benign pulmonary metastases were analyzed separately. Outcomes were descriptively assessed.</div></div><div><h3>Results</h3><div>Twelve patients (2.4 %) had malignant GCTB: five PM-GCTB (1.0 %), five SM-GCTB (1.0 %), and two secondary osteosarcoma (0.4 %). Three patients (0.6 %) developed histologically benign pulmonary metastases with indolent behavior. Three SM-GCTB patients transformed within one year despite benign baseline pathology, indicating true rapid malignant progression rather than diagnostic error. Median latency to malignant transformation was 7.5 years for SM-GCTB and up to 35 years for secondary osteosarcoma. SM-GCTB showed the highest disease-specific mortality (80 %), compared to PM-GCTB (20 %) and secondary osteosarcoma (0 %). None malignant transformations occurred following denosumab or radiotherapy.</div></div><div><h3>Conclusion</h3><div>Three aggressive GCTB variants were identified: benign pulmonary metastatic GCTB, PM-GCTB, and secondary malignant transformation. SM-GCTB and osteosarcoma arise from benign GCTB but differ in morphology and prognosis. Because malignant transformation is exceptionally rare and symptomatic, a patient-centered, symptom-driven follow-up strategy is preferred over routine lifelong radiologic surveillance.</div></div>","PeriodicalId":48806,"journal":{"name":"Journal of Bone Oncology","volume":"55 ","pages":"Article 100728"},"PeriodicalIF":3.5,"publicationDate":"2025-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145614365","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"The risk of pathological fractures after intralesional curettage in Atypical Cartilaginous Tumours: A retrospective cohort study","authors":"C.H.J. Scholte ,&nbsp;K. van Langevelde ,&nbsp;D.M.J. Dorleijn ,&nbsp;M. Fiocco ,&nbsp;D. Broekhuis ,&nbsp;P.D.S. Dijkstra ,&nbsp;M.A.J. van de Sande ,&nbsp;R.J.P. van der Wal","doi":"10.1016/j.jbo.2025.100731","DOIUrl":"10.1016/j.jbo.2025.100731","url":null,"abstract":"<div><h3>Purpose</h3><div>Atypical cartilaginous tumors (ACT) are locally aggressive, cartilage-producing neoplasms, traditionally classified as chondrosarcoma grade 1. They exist within the spectrum of cartilaginous tumors, between enchondroma and high-grade chondrosarcoma. The diagnosis of ACT remains challenging, with limited consensus on optimal treatment. Despite ongoing debate, intralesional curettage is commonly performed, though the risk of pathological fractures post-treatment remains a significant complication. This study aims to investigate the impact of cortical window design on fracture risk following curettage for ACT in the distal femur.</div></div><div><h3>Methods</h3><div>We compared two types of cortical windows: smooth oval (low stress riser) and windows with sharp or predrilled rounded corners (high stress riser). A retrospective cohort of 153 patients treated between 2000 and 2020 was included. Fracture occurrence was recorded, and logistic regression models were used to assess the relationship between surgical technique and fracture risk. We corrected for void filling and tumour size.</div></div><div><h3>Results</h3><div>Our findings revealed no significant difference in fracture risk between the two types of cortical windows (OR = 1.62, 95 % CI:0.66–3.96). However, the use of cement as a void filler significantly increased fracture risk, with odds ratio (OR) approximately five times higher than those using bone chips (OR = 5.00, 95 % CI:1.81–13.88). Tumor size (cm) was also associated with fracture risk (OR 1.31, 95 % CI:1.04–1.63).</div></div><div><h3>Conclusion</h3><div>The study highlights the importance of choosing appropriate void filling material, with bone chips offering a lower fracture risk compared to cement. These results suggest that optimizing surgical techniques, particularly the choice of filling material, may reduce post-curettage fracture risk in ACT treatment.</div></div>","PeriodicalId":48806,"journal":{"name":"Journal of Bone Oncology","volume":"55 ","pages":"Article 100731"},"PeriodicalIF":3.5,"publicationDate":"2025-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145614432","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}