Rotationplasty is a surgical technique primarily used in pediatric patients with malignant bone tumors around the knee. Its application in adults is rare, and outcomes are poorly defined. This study aimed to compare long-term QoL, function, and gait between patients treated with rotationplasty in childhood versus adulthood.
Methods
This cross-sectional post-hoc predefined secondary analysis included 33 patients treated between 1980–2002 (9 adults, 24 minors). Assessments included the mental and physical component score of the Short Form-36 (SF-36, MCS/ PCS), overall satisfaction, radiographs (Kellgren-Lawrence) of the pseudo-knee, contralateral ankle, and hips, Toronto Extremity Salvage Score (TESS), Musculoskeletal Tumor Society Score (MSTS), energy cost of comfortable walking during a 6-minute walk test, and 3-Dimenional gait analysis. Group comparisons were performed across all outcomes. Additionally, a systematic literature search identified published adult cases.
Results
People who underwent surgery during adulthood reported significantly higher physical QoL compared to those operated in childhood (MD 6.1, 95 % CI 0.0–12.3; p = 0.05). No significant differences were observed for the SF-36 MCS, TESS, MSTS, energy cost, sagittal-plane biomechanics, and radiographic OA prevalence. Adults exhibited a significantly shorter stride length (p < 0.01) and a longer double support phase (p = 0.01) compared to those treated in childhood. The literature review (22 studies, 51 patients) demonstrated overall favorable outcomes, though no objective gait data were reported.
Conclusion
Adults who undergo rotationplasty can achieve long-term quality of life, functional, and gait outcomes at least comparable to those operated on in childhood. These findings suggest that rotationplasty may be a viable reconstructive option in adults.
{"title":"Rotationplasty performed in adults versus minors: a comparative study of long-term quality of life, functional and biomechanical outcomes","authors":"Gitte G.J. Krebbekx , N.F.J. Waterval , M.A. Brehm , M.J.C. Duivenvoorden , I.N. Sierevelt , J.A.M. Bramer , G.M.M.J. Kerkhoffs , F.G.M. Verspoor","doi":"10.1016/j.jbo.2025.100732","DOIUrl":"10.1016/j.jbo.2025.100732","url":null,"abstract":"<div><h3>Background</h3><div>Rotationplasty is a surgical technique primarily used in pediatric patients with malignant bone tumors around the knee. Its application in adults is rare, and outcomes are poorly defined. This study aimed to compare long-term QoL, function, and gait between patients treated with rotationplasty in childhood versus adulthood.</div></div><div><h3>Methods</h3><div>This cross-sectional post-hoc predefined secondary analysis included 33 patients treated between 1980–2002 (9 adults, 24 minors). Assessments included the mental and physical component score of the Short Form-36 (SF-36, MCS/ PCS), overall satisfaction, radiographs (Kellgren-Lawrence) of the pseudo-knee, contralateral ankle, and hips, Toronto Extremity Salvage Score (TESS), Musculoskeletal Tumor Society Score (MSTS), energy cost of comfortable walking during a 6-minute walk test, and 3-Dimenional gait analysis. Group comparisons were performed across all outcomes. Additionally, a systematic literature search identified published adult cases.</div></div><div><h3>Results</h3><div>People who underwent surgery during adulthood reported significantly higher physical QoL compared to those operated in childhood (MD 6.1, 95 % CI 0.0–12.3; <em>p</em> = 0.05). No significant differences were observed for the SF-36 MCS, TESS, MSTS, energy cost, sagittal-plane biomechanics, and radiographic OA prevalence. Adults exhibited a significantly shorter stride length (p < 0.01) and a longer double support phase (p = 0.01) compared to those treated in childhood. The literature review (22 studies, 51 patients) demonstrated overall favorable outcomes, though no objective gait data were reported.</div></div><div><h3>Conclusion</h3><div>Adults who undergo rotationplasty can achieve long-term quality of life, functional, and gait outcomes at least comparable to those operated on in childhood. These findings suggest that rotationplasty may be a viable reconstructive option in adults.</div></div>","PeriodicalId":48806,"journal":{"name":"Journal of Bone Oncology","volume":"55 ","pages":"Article 100732"},"PeriodicalIF":3.5,"publicationDate":"2025-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145684794","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}
Pub Date : 2025-12-01DOI: 10.1016/j.jbo.2025.100730
Halil İbrahim Ellez , Hüseyin Salih Semiz , Ferhat Ekinci , Atike Pınar Erdoğan , Fatih Kuş , Fatih Karataş , Ertuğrul Bayram , Kubilay Karaboyun , Havva Yeşil Çınkır , Nilgün Yıldırım , Melek Karakurt Eryılmaz , Esma Türkmen Bekmez , Özkan Alan , Melike Özçelik , Yakup Düzköprü , Teoman Şakalar , Naziye Ak , Yusuf İlhan , Tuğba Yavuzşen , Nazım Serdar Turhal
Background
Denosumab, a monoclonal antibody against receptor activator of nuclear factor kappa-B ligand (RANKL), is widely used to prevent skeletal-related events (SREs) in patients with bone metastases from solid tumours. However, its safety in individuals with advanced chronic kidney disease (CKD), particularly regarding severe hypocalcaemia and skeletal complications, is not well defined.
Methods
We conducted a retrospective, multicentre study within the Turkish Oncology Group including patients with breast, prostate, or lung cancer who received denosumab between January 2011 and December 2022. Demographic and clinical data, CKD stage, prior fractures, serum calcium levels, episodes of hypocalcaemia, concomitant medications, and adverse events were recorded. Primary endpoints were the incidences of grade ≥ 3 hypocalcaemia and other grade ≥ 3 toxicities; secondary endpoints included skeletal-related events and overall survival.
Results
We analysed 264 patients from 17 oncology centres. Overall, 18 patients (6.8 %) experienced grade ≥ 3 toxicity, including 16 cases of severe hypocalcaemia and two of renal function decline. Among 42 patients with baseline estimated glomerular filtration rate (eGFR) < 60 mL/min, 13 (31.0 %) developed grade ≥ 3 toxicity (11 hypocalcaemia, two renal decline), representing a significantly higher risk than in patients with eGFR ≥ 60 mL/min (p < 0.01). Pathological fractures occurred in 21 patients, six with eGFR < 60 mL/min (p = 0.035). Eight patients required surgery for skeletal-related events, four with eGFR < 60 mL/min (p = 0.012).
Conclusion
Cancer patients with CKD receiving denosumab have an increased risk of severe hypocalcaemia and skeletal complications. Close monitoring of calcium and renal function is essential, and clinicians should carefully balance the benefits of denosumab against these risks in this vulnerable population.
{"title":"Chronic kidney disease and denosumab in metastatic bone disease: A multicenter Turkish cohort study on severe hypocalcemia, skeletal events, and survival","authors":"Halil İbrahim Ellez , Hüseyin Salih Semiz , Ferhat Ekinci , Atike Pınar Erdoğan , Fatih Kuş , Fatih Karataş , Ertuğrul Bayram , Kubilay Karaboyun , Havva Yeşil Çınkır , Nilgün Yıldırım , Melek Karakurt Eryılmaz , Esma Türkmen Bekmez , Özkan Alan , Melike Özçelik , Yakup Düzköprü , Teoman Şakalar , Naziye Ak , Yusuf İlhan , Tuğba Yavuzşen , Nazım Serdar Turhal","doi":"10.1016/j.jbo.2025.100730","DOIUrl":"10.1016/j.jbo.2025.100730","url":null,"abstract":"<div><h3>Background</h3><div>Denosumab, a monoclonal antibody against receptor activator of nuclear factor kappa-B ligand (RANKL), is widely used to prevent skeletal-related events (SREs) in patients with bone metastases from solid tumours. However, its safety in individuals with advanced chronic kidney disease (CKD), particularly regarding severe hypocalcaemia and skeletal complications, is not well defined.</div></div><div><h3>Methods</h3><div>We conducted a retrospective, multicentre study within the Turkish Oncology Group including patients with breast, prostate, or lung cancer who received denosumab between January 2011 and December 2022. Demographic and clinical data, CKD stage, prior fractures, serum calcium levels, episodes of hypocalcaemia, concomitant medications, and adverse events were recorded. Primary endpoints were the incidences of grade ≥ 3 hypocalcaemia and other grade ≥ 3 toxicities; secondary endpoints included skeletal-related events and overall survival.</div></div><div><h3>Results</h3><div>We analysed 264 patients from 17 oncology centres. Overall, 18 patients (6.8 %) experienced grade ≥ 3 toxicity, including 16 cases of severe hypocalcaemia and two of renal function decline. Among 42 patients with baseline estimated glomerular filtration rate (eGFR) < 60 mL/min, 13 (31.0 %) developed grade ≥ 3 toxicity (11 hypocalcaemia, two renal decline), representing a significantly higher risk than in patients with eGFR ≥ 60 mL/min (p < 0.01). Pathological fractures occurred in 21 patients, six with eGFR < 60 mL/min (p = 0.035). Eight patients required surgery for skeletal-related events, four with eGFR < 60 mL/min (p = 0.012).</div></div><div><h3>Conclusion</h3><div>Cancer patients with CKD receiving denosumab have an increased risk of severe hypocalcaemia and skeletal complications. Close monitoring of calcium and renal function is essential, and clinicians should carefully balance the benefits of denosumab against these risks in this vulnerable population.</div></div>","PeriodicalId":48806,"journal":{"name":"Journal of Bone Oncology","volume":"55 ","pages":"Article 100730"},"PeriodicalIF":3.5,"publicationDate":"2025-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145614431","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}
Pub Date : 2025-11-12DOI: 10.1016/j.jbo.2025.100726
Jin Qi , Gang Xue , Baomin Wu , Peng Zhu , Yapeng Wang
Background
Osteosarcoma (OS) is a life-threatening malignancy in children and adolescents, with limited treatment options for resistant or metastatic disease. Exosomes derived from hMSCs regulate tumor immunity by transporting molecules such as long non-coding RNA (lncRNA). The lncRNA MIR17HG is known to promote OS progression, yet its role in regulating T follicular helper (Tfh) cell differentiation in OS is unclear. This study is the first to systematically explore how MIR17HG influences Tfh cell differentiation, activation, and OS cell proliferation via the miR-372-3p/BCL6 signaling cascade.
Methods
Exosomes were extracted from hMSCs using differential centrifugation. Characterization of hMSCs-derived exosomes was conducted by TEM, NTA and western blotting. The expression of MIR17HG, BCL6 and PD-1 was determined by qRT‒PCR or western blotting. The differentiation and activation of Tfh cells, as well as OS apoptosis, were assessed through flow cytometry. OS cell viability was determined by a CCK-8 assay. The effect of hMSCs-Exo-MIR17HG on tumors was assessed in an MG63-derived xenograft mouse model. The expression of Ki67 was examined via IHC. RNA immunoprecipitation was performed to validate the interaction between MIR17HG and miR-372-3p. A dual-luciferase reporter assay was performed to explore the correlation between miR-372-3p and BCL6.
Results
The lncRNA MIR17HG was expressed in hMSCs-derived exosomes and upregulated by overexpression. hMSCs-Exo or hMSCs-Exo-MIR17HG promoted the differentiation and activation of Tfh cells, accompanied by increased PD-1 and BCL6 expression in CD4+ T cells, which enhanced OS cell proliferation. Furthermore, hMSCs-Exo-MIR17HG exerted a tumor-promoting effect in a CDX mouse model. Mechanistically, the effects driven by MIR17HG were abolished by miR-372-3p overexpression, and BCL6 was identified as a direct functional target of miR-372-3p.
Conclusion
These findings demonstrate that exosomal MIR17HG derived from hMSCs drives the differentiation of follicular helper T cells and the progression of OS via the miR-372-3p/BCL6 axis.
{"title":"hMSCs-derived exosomal MIR17HG promotes follicular helper T cell differentiation and osteosarcoma progression via the miR-372-3p/BCL6 axis","authors":"Jin Qi , Gang Xue , Baomin Wu , Peng Zhu , Yapeng Wang","doi":"10.1016/j.jbo.2025.100726","DOIUrl":"10.1016/j.jbo.2025.100726","url":null,"abstract":"<div><h3>Background</h3><div>Osteosarcoma (OS) is a life-threatening malignancy in children and adolescents, with limited treatment options for resistant or metastatic disease. Exosomes derived from hMSCs regulate tumor immunity by transporting molecules such as long non-coding RNA (lncRNA). The lncRNA MIR17HG is known to promote OS progression, yet its role in regulating T follicular helper (Tfh) cell differentiation in OS is unclear. This study is the first to systematically explore how MIR17HG influences Tfh cell differentiation, activation, and OS cell proliferation via the miR-372-3p/BCL6 signaling cascade.</div></div><div><h3>Methods</h3><div>Exosomes were extracted from hMSCs using differential centrifugation. Characterization of hMSCs-derived exosomes was conducted by TEM, NTA and western blotting. The expression of MIR17HG, BCL6 and PD-1 was determined by qRT‒PCR or western blotting. The differentiation and activation of Tfh cells, as well as OS apoptosis, were assessed through flow cytometry. OS cell viability was determined by a CCK-8 assay. The effect of hMSCs-Exo-MIR17HG on tumors was assessed in an MG63-derived xenograft mouse model. The expression of Ki67 was examined via IHC. RNA immunoprecipitation was performed to validate the interaction between MIR17HG and miR-372-3p. A dual-luciferase reporter assay was performed to explore the correlation between miR-372-3p and BCL6.</div></div><div><h3>Results</h3><div>The lncRNA MIR17HG was expressed in hMSCs-derived exosomes and upregulated by overexpression. hMSCs-Exo or hMSCs-Exo-MIR17HG promoted the differentiation and activation of Tfh cells, accompanied by increased PD-1 and BCL6 expression in CD4<sup>+</sup> T cells, which enhanced OS cell proliferation. Furthermore, hMSCs-Exo-MIR17HG exerted a tumor-promoting effect in a CDX mouse model. Mechanistically, the effects driven by MIR17HG were abolished by miR-372-3p overexpression, and BCL6 was identified as a direct functional target of miR-372-3p.</div></div><div><h3>Conclusion</h3><div>These findings demonstrate that exosomal MIR17HG derived from hMSCs drives the differentiation of follicular helper T cells and the progression of OS via the miR-372-3p/BCL6 axis.</div></div>","PeriodicalId":48806,"journal":{"name":"Journal of Bone Oncology","volume":"55 ","pages":"Article 100726"},"PeriodicalIF":3.5,"publicationDate":"2025-11-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145568330","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}
Pub Date : 2025-11-12DOI: 10.1016/j.jbo.2025.100727
Lea Hatoum , Rita El Murr , Ahmad Assi , Rami Mohanna , Amer Sebaaly , Hampig-Raphaël Kourie
Background
Colorectal cancer ranks as the third most prevalent cancer globally, with over 1.9 million new cases and 930,000 deaths in 2020. While the liver and lungs are the most common site of colorectal cancer metastases, bone metastasis is relatively rare but clinically significant. The rarity of bone metastasis in colorectal cancer, early diagnosis challenges, and the poorly understood mechanisms underlying bone metastasis have resulted in lesser research compared to other metastatic sites of colorectal cancer.
Objective
The goal of this review is to summarize pathophysiology, clinical presentation, risk and prognostic factors, survival outcomes, and treatment options for bone metastasis in colorectal cancer. Results: Bone metastasis occurred in 6 to 10 % of colorectal cancer patients, with the spine and pelvis being the most common sites. Bone metastasis from colorectal cancer may result from hematogenous dissemination and retrograde venous flow through Batson’s plexus. Early diagnosis can be challenging, and Positron Emission Tomography / Computed Tomography (PET/CT) provided the best accuracy for diagnosing bone metastasis in colorectal cancer. Isolated bone metastasis, low neutrophil-to-lymphocyte ratio (NLR) (high NLR with HR: 1.54), low alkaline phosphatase levels, N0 stage colorectal cancer were associated with a better prognosis and survival. High carcinoembryonic antigen (OR: 2.368, HR: 3.300), alkaline phosphatase (OR: 6.89, HR: 2.12), and CA 19–9 levels (HR: 1.5), Lactate dehydrogenase (HR: 1.961), perineural invasion (HR: 3.457), right sided location (HR: 1.84), multiples bone metastasis sites (HR: 1.452), hypercalcemia (HR: 3.75), pathologic fracture (HR:1.91) and more than two extra-bone metastatic organs (HR: 2.357) were linked to worse prognosis. The therapeutic approach for colorectal cancer patients with bone metastasis consists of a multidisciplinary strategy including systemic chemotherapy, palliative surgery, radiotherapy and bisphosphonate.
Conclusion
Early diagnosis and management of bone metastasis in colorectal cancer patients is essential to improve quality of life and survival. Further research is needed to identify methods for early detection and develop tailored treatment strategies.
{"title":"Bone metastasis in colorectal cancer: Pathophysiology, prognostic factors, survival outcomes, and treatment strategies – A comprehensive review","authors":"Lea Hatoum , Rita El Murr , Ahmad Assi , Rami Mohanna , Amer Sebaaly , Hampig-Raphaël Kourie","doi":"10.1016/j.jbo.2025.100727","DOIUrl":"10.1016/j.jbo.2025.100727","url":null,"abstract":"<div><h3>Background</h3><div>Colorectal cancer ranks as the third most prevalent cancer globally, with over 1.9 million new cases and 930,000 deaths in 2020. While the liver and lungs are the most common site of colorectal cancer metastases, bone metastasis is relatively rare but clinically significant. The rarity of bone metastasis in colorectal cancer, early diagnosis challenges, and the poorly understood mechanisms underlying bone metastasis have resulted in lesser research compared to other metastatic sites of colorectal cancer.</div></div><div><h3>Objective</h3><div>The goal of this review is to summarize pathophysiology, clinical presentation, risk and prognostic factors, survival outcomes, and treatment options for bone metastasis in colorectal cancer. Results: Bone metastasis occurred in 6 to 10 % of colorectal cancer patients, with the spine and pelvis being the most common sites. Bone metastasis from colorectal cancer may result from hematogenous dissemination and retrograde venous flow through Batson’s plexus. Early diagnosis can be challenging, and Positron Emission Tomography / Computed Tomography (PET/CT) provided the best accuracy for diagnosing bone metastasis in colorectal cancer. Isolated bone metastasis, low neutrophil-to-lymphocyte ratio (NLR) (high NLR with HR: 1.54), low alkaline phosphatase levels, N0 stage colorectal cancer were associated with a better prognosis and survival. High carcinoembryonic antigen (OR: 2.368, HR: 3.300), alkaline phosphatase (OR: 6.89, HR: 2.12), and CA 19–9 levels (HR: 1.5), Lactate dehydrogenase (HR: 1.961), perineural invasion (HR: 3.457), right sided location (HR: 1.84), multiples bone metastasis sites (HR: 1.452), hypercalcemia (HR: 3.75), pathologic fracture (HR:1.91) and more than two extra-bone metastatic organs (HR: 2.357) were linked to worse prognosis. The therapeutic approach for colorectal cancer patients with bone metastasis consists of a multidisciplinary strategy including systemic chemotherapy, palliative surgery, radiotherapy and bisphosphonate.</div></div><div><h3>Conclusion</h3><div>Early diagnosis and management of bone metastasis in colorectal cancer patients is essential to improve quality of life and survival. Further research is needed to identify methods for early detection and develop tailored treatment strategies.</div></div>","PeriodicalId":48806,"journal":{"name":"Journal of Bone Oncology","volume":"55 ","pages":"Article 100727"},"PeriodicalIF":3.5,"publicationDate":"2025-11-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145520108","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}
Giant cell tumor of bone (GCTB) is a benign but locally aggressive neoplasm with a high risk of recurrence. Among its patterns of relapse, soft-tissue recurrence (STR) is an uncommon but clinically significant entity, often presenting as ossified or non-ossified perilesional nodules. Despite its rarity, STR poses diagnostic and therapeutic challenges that require clarification.
Methods
A comprehensive literature review was performed across PubMed, Embase, and Google Scholar from 1980 through January 2025, focusing on the epidemiology, pathogenesis, imaging features, histopathology, management, and outcomes of STR in GCTB. Case reports, series, and retrospective studies explicitly distinguishing STR from intraosseous recurrence were included, and findings were synthesized narratively.
Results
STR occurs in approximately 2–3 % of GCTB cases, typically within 6–12 months after surgery. Major risk factors include curettage procedures, pathological fractures, cortical breaches, and unrecognized microscopic soft-tissue extension. Imaging reveals three distinct patterns: peripheral “eggshell” ossification, central nodular calcification, and purely soft-tissue lesions. Histology mirrors primary GCTB, often with osteogenic metaplasia, while molecular testing confirms retention of H3F3A mutations. Surgical excision with clear margins remains the mainstay of treatment, yielding excellent functional outcomes. However, up to 60 % of patients experience multiple recurrences, highlighting the need for vigilant surveillance. Systemic agents such as denosumab or bisphosphonates remain investigational, and radiotherapy is generally contraindicated due to malignant transformation risk.
Conclusion
STR represents a rare but distinct subset of GCTB recurrences. Awareness of risk factors, early imaging-based detection, and complete surgical excision are critical for optimal outcomes. Further multicenter studies are required to define surveillance protocols, validate molecular predictors, and clarify the role of systemic therapy in this challenging condition.
{"title":"Soft tissue recurrence in giant cell tumor of Bone: A comprehensive review of pathogenesis, imaging features, and clinical management","authors":"Khodamorad Jamshidi , Hamed Naghizadeh , Sadegh Saberi , Farshad Zand Rahimi , Aidin Arabzadeh , Seyyed Saeed Khabiri","doi":"10.1016/j.jbo.2025.100725","DOIUrl":"10.1016/j.jbo.2025.100725","url":null,"abstract":"<div><h3>Background</h3><div>Giant cell tumor of bone (GCTB) is a benign but locally aggressive neoplasm with a high risk of recurrence. Among its patterns of relapse, soft-tissue recurrence (STR) is an uncommon but clinically significant entity, often presenting as ossified or non-ossified perilesional nodules. Despite its rarity, STR poses diagnostic and therapeutic challenges that require clarification.</div></div><div><h3>Methods</h3><div>A comprehensive literature review was performed across PubMed, Embase, and Google Scholar from 1980 through January 2025, focusing on the epidemiology, pathogenesis, imaging features, histopathology, management, and outcomes of STR in GCTB. Case reports, series, and retrospective studies explicitly distinguishing STR from intraosseous recurrence were included, and findings were synthesized narratively.</div></div><div><h3>Results</h3><div>STR occurs in approximately 2–3 % of GCTB cases, typically within 6–12 months after surgery. Major risk factors include curettage procedures, pathological fractures, cortical breaches, and unrecognized microscopic soft-tissue extension. Imaging reveals three distinct patterns: peripheral “eggshell” ossification, central nodular calcification, and purely soft-tissue lesions. Histology mirrors primary GCTB, often with osteogenic metaplasia, while molecular testing confirms retention of H3F3A mutations. Surgical excision with clear margins remains the mainstay of treatment, yielding excellent functional outcomes. However, up to 60 % of patients experience multiple recurrences, highlighting the need for vigilant surveillance. Systemic agents such as denosumab or bisphosphonates remain investigational, and radiotherapy is generally contraindicated due to malignant transformation risk.</div></div><div><h3>Conclusion</h3><div>STR represents a rare but distinct subset of GCTB recurrences. Awareness of risk factors, early imaging-based detection, and complete surgical excision are critical for optimal outcomes. Further multicenter studies are required to define surveillance protocols, validate molecular predictors, and clarify the role of systemic therapy in this challenging condition.</div></div>","PeriodicalId":48806,"journal":{"name":"Journal of Bone Oncology","volume":"55 ","pages":"Article 100725"},"PeriodicalIF":3.5,"publicationDate":"2025-11-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145520107","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}
Pub Date : 2025-11-02DOI: 10.1016/j.jbo.2025.100721
Kang-Wen Xiao , Zhenyi Chen , Chong Zhang , Zhiqiang Yang , Liangyu Guo , Yuanlong Xie , Jun Lei , Lin Cai
Osteosarcoma (OS) is a malignant bone tumor with high incidence of metastasis. However, the molecular landscape of osteosarcoma remains incompletely understood. Weighted gene co-expression network analysis (WGCNA), differential expressed genes (DEGs), Cox regression, gene set enrichment analysis (GSEA), receiver operating characteristic curve (ROC) and survival analysis were conducted to screen potential targets and molecular mechanism for OS. Seven modules were considered to be closely related to the prognosis of OS. Subsequent immunohistochemistry (IHC), survival and ROC analysis indicated that high expression of COL13A1 was seen in OS tissue and was significantly associated with poor clinical outcome. COL13A1 expression may correlate with inhibition of M1 polarization and could serve as a predictor for immunotherapy response. Further cellular experiments showed that the expression of COL13A1 promoted the proliferation, migration and invasion. Besides, high expression of COL13A1 enhanced TGF-β signaling through β1 integrin and upregulated MMP9 and cyclin D1 expression. Finally, the low expression of COL13A1 limited the weight and lung metastasis of tumor, and reduced bone destruction in the orthotopic tumor-bearing model. COL13A1 was identified as a novel regulator of OS progression via TGF-β signaling, suggesting its potential as a therapeutic target pending further validation.
{"title":"WGCNA-identified COL13A1 drives osteosarcoma metastasis and progression via TGF-β signaling","authors":"Kang-Wen Xiao , Zhenyi Chen , Chong Zhang , Zhiqiang Yang , Liangyu Guo , Yuanlong Xie , Jun Lei , Lin Cai","doi":"10.1016/j.jbo.2025.100721","DOIUrl":"10.1016/j.jbo.2025.100721","url":null,"abstract":"<div><div>Osteosarcoma (OS) is a malignant bone tumor with high incidence of metastasis. However, the molecular landscape of osteosarcoma remains incompletely understood. Weighted gene co-expression network analysis (WGCNA), differential expressed genes (DEGs), Cox regression, gene set enrichment analysis (GSEA), receiver operating characteristic curve (ROC) and survival analysis were conducted to screen potential targets and molecular mechanism for OS. Seven modules were considered to be closely related to the prognosis of OS. Subsequent immunohistochemistry (IHC), survival and ROC analysis indicated that high expression of COL13A1 was seen in OS tissue and was significantly associated with poor clinical outcome. COL13A1 expression may correlate with inhibition of M1 polarization and could serve as a predictor for immunotherapy response. Further cellular experiments showed that the expression of COL13A1 promoted the proliferation, migration and invasion. Besides, high expression of COL13A1 enhanced TGF-β signaling through β1 integrin and upregulated MMP9 and cyclin D1 expression. Finally, the low expression of COL13A1 limited the weight and lung metastasis of tumor, and reduced bone destruction in the orthotopic tumor-bearing model. COL13A1 was identified as a novel regulator of OS progression via TGF-β signaling, suggesting its potential as a therapeutic target pending further validation.</div></div>","PeriodicalId":48806,"journal":{"name":"Journal of Bone Oncology","volume":"55 ","pages":"Article 100721"},"PeriodicalIF":3.5,"publicationDate":"2025-11-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145520105","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}
Pub Date : 2025-11-02DOI: 10.1016/j.jbo.2025.100722
Christina Enciso Holm, Jesper Peter Bömers, Allan Villadsen, Michael Mørk Petersen
Background
Modular knee mega-prostheses are considered the method of choice for reconstruction after resection of malignant bone lesions around the knee. The available literature evaluating Zimmer® Segmental mega-prostheses is sparse. The purpose of the present study is to evaluate the use of Zimmer® Segmental mega-prosthesis for reconstruction of the distal femur, following the resection of bone malignancies and aggressive benign bone tumors.
Material and Methods
A retrospective study including 59 consecutive patients (F/M = 35/24), mean age 58 (range 17–86) who underwent reconstructions of the distal femur due to malignant bone lesions (n = 51) or aggressive benign bone tumors (n = 8) from 2017 to 2022. All reconstructions were performed with the most recent Zimmer® Segmental System with the XT Distal Femoral Component. Kaplan-Meier survival analysis was used for evaluation of overall survival. Competing risk analysis was used for assessing cumulative incidence of revision and amputation. Patients were followed until death or end of study (December 31st, 2024). Functional outcome and quality of life was evaluated with Musculoskeletal Tumor Society Score (MSTS), European quality of life − 5 Dimensions score (EQ 5D) and Oxford Knee Score (OKS).
Results
Twenty-eight (n = 28) patients were alive at follow-up. Overall survival after 5-year was 44 % (CI95%: 30–58 %). The risk of implant failure after 1 and 5 years was 8 % (95 %CI 1–16 %) and 12 % (95 %CI 4–20 %) respectively. One patient (2 %) underwent amputation. Mean MSTS score was 17 (57 %) (range 3–30). Mean EQ 5D index score was 0.88 and mean EQ-5D VAS score was 68. OKS demonstrated a mean score of 31.
Conclusion
The Zimmer® Segmental mega-prosthesis in distal femur reconstruction with the improved XT femoral component, demonstrated a relatively low risk of implant failure, and a low risk of amputation. The risk of implant failure was higher among patients who underwent radiotherapy.
{"title":"Evaluation of Zimmer® segmental distal femur mega-prostheses: Patient survival, surgical outcomes and functional outcome","authors":"Christina Enciso Holm, Jesper Peter Bömers, Allan Villadsen, Michael Mørk Petersen","doi":"10.1016/j.jbo.2025.100722","DOIUrl":"10.1016/j.jbo.2025.100722","url":null,"abstract":"<div><h3>Background</h3><div>Modular knee mega-prostheses are considered the method of choice for reconstruction after resection of malignant bone lesions around the knee. The available literature evaluating Zimmer® Segmental mega-prostheses is sparse. The purpose of the present study is to evaluate the use of Zimmer® Segmental mega-prosthesis for reconstruction of the distal femur, following the resection of bone malignancies and aggressive benign bone tumors.</div></div><div><h3>Material and Methods</h3><div>A retrospective study including 59 consecutive patients (F/M = 35/24), mean age 58 (range 17–86) who underwent reconstructions of the distal femur due to malignant bone lesions (n = 51) or aggressive benign bone tumors (n = 8) from 2017 to 2022. All reconstructions were performed with the most recent Zimmer® Segmental System with the XT Distal Femoral Component. Kaplan-Meier survival analysis was used for evaluation of overall survival. Competing risk analysis was used for assessing cumulative incidence of revision and amputation. Patients were followed until death or end of study (December 31st, 2024). Functional outcome and quality of life was evaluated with Musculoskeletal Tumor Society Score (MSTS), European quality of life − 5 Dimensions score (EQ 5D) and Oxford Knee Score (OKS).</div></div><div><h3>Results</h3><div>Twenty-eight (n = 28) patients were alive at follow-up. Overall survival after 5-year was 44 % (CI95%: 30–58 %). The risk of implant failure after 1 and 5 years was 8 % (95 %CI 1–16 %) and 12 % (95 %CI 4–20 %) respectively. One patient (2 %) underwent amputation. Mean MSTS score was 17 (57 %) (range 3–30). Mean EQ 5D index score was 0.88 and mean EQ-5D VAS score was 68. OKS demonstrated a mean score of 31.</div></div><div><h3>Conclusion</h3><div>The Zimmer® Segmental mega-prosthesis in distal femur reconstruction with the improved XT femoral component, demonstrated a relatively low risk of implant failure, and a low risk of amputation. The risk of implant failure was higher among patients who underwent radiotherapy.</div></div>","PeriodicalId":48806,"journal":{"name":"Journal of Bone Oncology","volume":"55 ","pages":"Article 100722"},"PeriodicalIF":3.5,"publicationDate":"2025-11-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145466328","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}
Pub Date : 2025-11-01DOI: 10.1016/j.jbo.2025.100724
Deanna N. Edwards , Shreya Patel , Gabriel M. Pagnotti
The 2024 Annual Conference of the Cancer and Bone Society (CABS), held jointly with the Bone Research Society at the University of Sheffield, highlighted the Society’s enduring mission to advance understanding of cancer–bone interactions through collaboration, mentorship, and scientific excellence. Established from the early Cancer Induced Bone Disease meetings and re-emerging as an independent body in 2016, CABS continues to serve as a global community bridging basic, translational, and clinical research. This year’s meeting featured a comprehensive scientific program spanning tumor–bone biology, therapeutic innovation, and microenvironmental regulation. Clinical sessions emphasized the evolution of bone-targeted treatments such as bisphosphonates, denosumab, and radiopharmaceuticals, alongside predictive biomarkers to guide personalized care. Presentations on renal cell carcinoma bone metastases, multiple myeloma, and prostate cancer underscored persistent clinical challenges and opportunities for integration of immunotherapies and precision radiotherapy. Molecular sessions revealed how non-coding RNAs, TGF-β/Gli2 signaling, metabolic dependencies, and stromal mediators—including osteomodulin, IL-1β, and adiponectin—govern tumor colonization, dormancy, and resistance. Advances in immuno-oncology demonstrated the potential of γδ CAR-T cells, oncolytic viruses, and immune-metabolic targeting to enhance therapeutic efficacy in the bone microenvironment. Spatial and single-cell profiling provided unprecedented resolution of the metastatic niche, identifying novel osteoblast-derived stromal subsets and neurovascular remodeling as key determinants of disease progression. Complementary preclinical models and computational simulations, including 3D engineered bone marrow constructs and micro–finite element analysis, offered new frameworks to study therapeutic responses and fracture risk. The meeting also celebrated the growing leadership of Early-Stage Investigators (ESIs), whose contributions extended beyond research presentations to creative networking and mentorship events. These efforts reflected CABS’ commitment to inclusivity, community building, and sustaining the next generation of leaders in cancer and bone research. Collectively, the 2024 CABS Annual Conference captured a transformative moment in the field—where mechanistic discoveries, advanced modeling, and translational collaboration converge to improve outcomes for patients with skeletal malignancies. As CABS looks ahead to the 2026 meeting in Tampa, Florida, the Society remains steadfast in fostering global partnerships and advancing integrative approaches to conquer cancer in bone.
癌症与骨骼学会(CABS)与谢菲尔德大学骨骼研究学会(Bone Research Society)联合举办的2024年年会强调了该学会的持久使命,即通过合作、指导和卓越的科学成就来促进对癌症与骨骼相互作用的理解。CABS成立于早期癌症诱导骨病会议,并于2016年重新成为一个独立的机构,CABS继续作为一个全球性的社区,连接基础、转化和临床研究。今年的会议以全面的科学项目为特色,涵盖肿瘤骨生物学、治疗创新和微环境调节。临床会议强调骨靶向治疗的发展,如双膦酸盐、地诺单抗和放射性药物,以及预测性生物标志物来指导个性化护理。关于肾细胞癌、骨转移、多发性骨髓瘤和前列腺癌的报告强调了免疫治疗和精确放疗结合的持续临床挑战和机遇。分子会议揭示了非编码rna、TGF-β/Gli2信号、代谢依赖性和基质介质(包括骨调节素、IL-1β和脂联素)如何控制肿瘤定植、休眠和耐药性。免疫肿瘤学的进展证明了γδ CAR-T细胞、溶瘤病毒和免疫代谢靶向在骨微环境中增强治疗效果的潜力。空间和单细胞分析提供了转移生态位的前所未有的分辨率,确定了新的成骨细胞来源的基质亚群和神经血管重塑是疾病进展的关键决定因素。互补的临床前模型和计算模拟,包括3D工程骨髓构建和微有限元分析,为研究治疗反应和骨折风险提供了新的框架。会议还赞扬了早期研究人员日益增长的领导力,他们的贡献从研究报告扩展到创造性的网络和指导活动。这些努力反映了CABS对包容性、社区建设和支持下一代癌症和骨骼研究领导者的承诺。总的来说,2024年CABS年会抓住了该领域的一个变革时刻——机制发现、先进建模和转化合作汇集在一起,以改善骨骼恶性肿瘤患者的预后。随着CABS展望2026年在佛罗里达州坦帕市举行的会议,该协会将继续坚定地促进全球合作伙伴关系,推进攻克骨癌的综合方法。
{"title":"Cutting edge developments in bone oncology – Highlights from the Cancer and Bone Society 2024 annual meeting","authors":"Deanna N. Edwards , Shreya Patel , Gabriel M. Pagnotti","doi":"10.1016/j.jbo.2025.100724","DOIUrl":"10.1016/j.jbo.2025.100724","url":null,"abstract":"<div><div>The 2024 Annual Conference of the Cancer and Bone Society (CABS), held jointly with the Bone Research Society at the University of Sheffield, highlighted the Society’s enduring mission to advance understanding of cancer–bone interactions through collaboration, mentorship, and scientific excellence. Established from the early Cancer Induced Bone Disease meetings and re-emerging as an independent body in 2016, CABS continues to serve as a global community bridging basic, translational, and clinical research. This year’s meeting featured a comprehensive scientific program spanning tumor–bone biology, therapeutic innovation, and microenvironmental regulation. Clinical sessions emphasized the evolution of bone-targeted treatments such as bisphosphonates, denosumab, and radiopharmaceuticals, alongside predictive biomarkers to guide personalized care. Presentations on renal cell carcinoma bone metastases, multiple myeloma, and prostate cancer underscored persistent clinical challenges and opportunities for integration of immunotherapies and precision radiotherapy. Molecular sessions revealed how non-coding RNAs, TGF-β/Gli2 signaling, metabolic dependencies, and stromal mediators—including osteomodulin, IL-1β, and adiponectin—govern tumor colonization, dormancy, and resistance. Advances in immuno-oncology demonstrated the potential of γδ CAR-T cells, oncolytic viruses, and immune-metabolic targeting to enhance therapeutic efficacy in the bone microenvironment. Spatial and single-cell profiling provided unprecedented resolution of the metastatic niche, identifying novel osteoblast-derived stromal subsets and neurovascular remodeling as key determinants of disease progression. Complementary preclinical models and computational simulations, including 3D engineered bone marrow constructs and micro–finite element analysis, offered new frameworks to study therapeutic responses and fracture risk. The meeting also celebrated the growing leadership of Early-Stage Investigators (ESIs), whose contributions extended beyond research presentations to creative networking and mentorship events. These efforts reflected CABS’ commitment to inclusivity, community building, and sustaining the next generation of leaders in cancer and bone research. Collectively, the 2024 CABS Annual Conference captured a transformative moment in the field—where mechanistic discoveries, advanced modeling, and translational collaboration converge to improve outcomes for patients with skeletal malignancies. As CABS looks ahead to the 2026 meeting in Tampa, Florida, the Society remains steadfast in fostering global partnerships and advancing integrative approaches to conquer cancer in bone.</div></div>","PeriodicalId":48806,"journal":{"name":"Journal of Bone Oncology","volume":"55 ","pages":"Article 100724"},"PeriodicalIF":3.5,"publicationDate":"2025-11-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145466329","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}
Pub Date : 2025-10-31DOI: 10.1016/j.jbo.2025.100723
Weiming Xie , Xiaozhou Bai , Miao Liu , Haonan Shangguan , Ying Zhan , Xiaodan Wu , Yingxin Dai , Yusong Pei , Guoxu Zhang , Zhiguo Wang , Zhaomin Yao
Purpose
This study aims to improve the diagnostic accuracy of SPECT/CT imaging in distinguishing benign from malignant bone lesions by integrating self-supervised deep learning and radiomics, reducing subjectivity in traditional image interpretation for more reliable clinical decision-making.
Methods
We developed a multi-scale, multi-modal framework combining radiomics with self-supervised learning. The novel SPECT-guided model, SPARC-Net, uses functional SPECT data as semantic priors to extract discriminative features from CT scans without manual annotations. Deep features from SPARC-Net were fused with radiomics to form a unified representation. The model was trained and validated on 741 confirmed bone lesion cases using five-fold cross-validation, with interpretability assessed via Grad-CAM.
Results
The fused model achieved 82.3 % accuracy, 0.890 AUC, 72.3 % F1 score, 79.3 % precision, 66.6 % sensitivity, and 90.7 % specificity, outperforming single-modality models. Grad-CAM confirmed the model focused on metabolically active regions identified by SPECT.
Conclusion
SPARC-Net, integrating SPECT-guided self-supervised learning with CT-based radiomics, improves classification of benign and malignant lesions, enhancing the accuracy, robustness, and interpretability of SPECT/CT imaging for bone tumor diagnosis.
{"title":"Benign and malignant bone lesion diagnosis based on self-supervised and radiomics fusion using SPECT/CT images","authors":"Weiming Xie , Xiaozhou Bai , Miao Liu , Haonan Shangguan , Ying Zhan , Xiaodan Wu , Yingxin Dai , Yusong Pei , Guoxu Zhang , Zhiguo Wang , Zhaomin Yao","doi":"10.1016/j.jbo.2025.100723","DOIUrl":"10.1016/j.jbo.2025.100723","url":null,"abstract":"<div><h3>Purpose</h3><div>This study aims to improve the diagnostic accuracy of SPECT/CT imaging in distinguishing benign from malignant bone lesions by integrating self-supervised deep learning and radiomics, reducing subjectivity in traditional image interpretation for more reliable clinical decision-making.</div></div><div><h3>Methods</h3><div>We developed a multi-scale, multi-modal framework combining radiomics with self-supervised learning. The novel SPECT-guided model, SPARC-Net, uses functional SPECT data as semantic priors to extract discriminative features from CT scans without manual annotations. Deep features from SPARC-Net were fused with radiomics to form a unified representation. The model was trained and validated on 741 confirmed bone lesion cases using five-fold cross-validation, with interpretability assessed via Grad-CAM.</div></div><div><h3>Results</h3><div>The fused model achieved 82.3 % accuracy, 0.890 AUC, 72.3 % F1 score, 79.3 % precision, 66.6 % sensitivity, and 90.7 % specificity, outperforming single-modality models. Grad-CAM confirmed the model focused on metabolically active regions identified by SPECT.</div></div><div><h3>Conclusion</h3><div>SPARC-Net, integrating SPECT-guided self-supervised learning with CT-based radiomics, improves classification of benign and malignant lesions, enhancing the accuracy, robustness, and interpretability of SPECT/CT imaging for bone tumor diagnosis.</div></div>","PeriodicalId":48806,"journal":{"name":"Journal of Bone Oncology","volume":"55 ","pages":"Article 100723"},"PeriodicalIF":3.5,"publicationDate":"2025-10-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145466325","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}
Multiple myeloma (MM) frequently presents with myeloma bone disease (MBD), manifesting as osteolytic lesions and skeletal-related events (SREs), significantly impairing quality of life and increasing morbidity. Whole-body low-dose computed tomography (WBLDCT) has become the standard for assessing bone involvement at diagnosis, but its prognostic significance remains unclear. The aim of this study was to evaluate the burden of MBD in newly diagnosed MM patients using WBLDCT and examined associations between imaging characteristics and survival outcomes.
Methods
In this retrospective, single center, analysis of 119 MM patients, WBLDCT was performed at diagnosis prior to treatment initiation. Imaging findings, including vertebral compression fractures (VCFs), lesion number, cortical destruction, and appendicular skeleton medullary cavity (ASMC) patterns, were recorded. Progression-free survival (PFS) and overall survival (OS) were analyzed using Kaplan-Meier curves and Cox regression models.
Results
VCFs were significantly associated with inferior PFS (18.1 vs. 33.6 months; p = 0.013) and OS (51.5 months vs. not reached; p = 0.023) in univariate analyses. However, in multivariable models, no imaging parameter, including VCFs, retained independent prognostic significance. Other imaging variables (lesion count, ASMC subtype, cortical destruction) were not predictive of outcomes.
Conclusions
While VCFs identified on WBLDCT correlate with poor outcomes in univariate analysis, they do not serve as independent prognostic markers when adjusting for established clinical factors. These findings suggest that in the era of novel anti-myeloma therapeutics, the bone disease burden at diagnosis may not impact prognosis significantly.
背景:多发性骨髓瘤(MM)经常表现为骨髓瘤骨病(MBD),表现为溶骨病变和骨骼相关事件(SREs),显著降低生活质量,增加发病率。全身低剂量计算机断层扫描(WBLDCT)已成为诊断时评估骨受累的标准,但其预后意义尚不清楚。本研究的目的是利用WBLDCT评估新诊断MM患者的MBD负担,并检查影像学特征与生存结果之间的关系。方法回顾性、单中心分析119例MM患者,在诊断前进行WBLDCT检查。记录影像学表现,包括椎体压缩性骨折(VCFs)、病变数量、皮质破坏和阑尾骨髓腔(ASMC)模式。采用Kaplan-Meier曲线和Cox回归模型分析无进展生存期(PFS)和总生存期(OS)。结果单因素分析中,vcf与不良PFS (18.1 vs. 33.6 个月;p = 0.013)和OS(51.5 个月vs.未达到;p = 0.023)显著相关。然而,在多变量模型中,没有影像学参数(包括vcf)保留独立的预后意义。其他影像学变量(病变计数、ASMC亚型、皮质破坏)不能预测预后。结论:虽然在单因素分析中,在WBLDCT上发现的vcf与不良预后相关,但在调整已确定的临床因素时,它们不能作为独立的预后指标。这些发现表明,在新型抗骨髓瘤疗法的时代,诊断时的骨病负担可能不会显著影响预后。
{"title":"Bone disease burden does not impact overall survival in newly diagnosed patients with multiple myeloma − a single center, retrospective imaging analysis on 119 patients","authors":"Evangelos Terpos , Vassilis Koutoulidis , Ioannis Ntanasis-Stathopoulos , Stylianos Mavropoulos-Papoudas , Maria Douka , Maria Gavriatopoulou , Panagiotis Malandrakis , Vasiliki Spiliopoulou , Foteini Theodorakakou , Despina Fotiou , Magdalini Migkou , Nikolaos Kanellias , Evangelos Eleutherakis-Papaiakovou , Efstathios Kastritis , Lia-Angela Moulopoulos , Meletios A Dimopoulos","doi":"10.1016/j.jbo.2025.100720","DOIUrl":"10.1016/j.jbo.2025.100720","url":null,"abstract":"<div><h3>Background</h3><div>Multiple myeloma (MM) frequently presents with myeloma bone disease (MBD), manifesting as osteolytic lesions and skeletal-related events (SREs), significantly impairing quality of life and increasing morbidity. Whole-body low-dose computed tomography (WBLDCT) has become the standard for assessing bone involvement at diagnosis, but its prognostic significance remains unclear. The aim of this study was to evaluate the burden of MBD in newly diagnosed MM patients using WBLDCT and examined associations between imaging characteristics and survival outcomes.</div></div><div><h3>Methods</h3><div>In this retrospective, single center, analysis of 119 MM patients, WBLDCT was performed at diagnosis prior to treatment initiation. Imaging findings, including vertebral compression fractures (VCFs), lesion number, cortical destruction, and appendicular skeleton medullary cavity (ASMC) patterns, were recorded. Progression-free survival (PFS) and overall survival (OS) were analyzed using Kaplan-Meier curves and Cox regression models.</div></div><div><h3>Results</h3><div>VCFs were significantly associated with inferior PFS (18.1 vs. 33.6 months; p = 0.013) and OS (51.5 months vs. not reached; p = 0.023) in univariate analyses. However, in multivariable models, no imaging parameter, including VCFs, retained independent prognostic significance. Other imaging variables (lesion count, ASMC subtype, cortical destruction) were not predictive of outcomes.</div></div><div><h3>Conclusions</h3><div>While VCFs identified on WBLDCT correlate with poor outcomes in univariate analysis, they do not serve as independent prognostic markers when adjusting for established clinical factors. These findings suggest that in the era of novel anti-myeloma therapeutics, the bone disease burden at diagnosis may not impact prognosis significantly.</div></div>","PeriodicalId":48806,"journal":{"name":"Journal of Bone Oncology","volume":"55 ","pages":"Article 100720"},"PeriodicalIF":3.5,"publicationDate":"2025-10-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145417510","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}