Bone Reports最新文献

{"title":"Total talectomy and reconstruction using unrestricted 3D printed prosthesis for pediatric talus hemangioendothelioma","authors":"Yunlong Zhang,&nbsp;Zhichang Zhang","doi":"10.1016/j.bonr.2025.101830","DOIUrl":"10.1016/j.bonr.2025.101830","url":null,"abstract":"<div><h3>Background</h3><div>Epithelioid hemangioendothelioma (EHE) is an ultra-rare vascular sarcoma with an extremely low incidence and prevalence, particularly in children. We report the case of a 9-year-old girl diagnosed with EHE. There are limited reconstruction methods available following total talus resection for vascular endothelioma of the talus, and the use of a 3D-printed talus prosthesis in pediatric cases has not been previously documented.</div></div><div><h3>Case presentation</h3><div>A 9-year-old girl presented to our unit with swelling, pain, and limited mobility of the ankle for one month without an obvious cause. X-ray and CT imaging revealed osteolytic lesions in the talus, which was identified as a low-grade malignant tumor that had nearly completely invaded the talus and was surrounded by immature bone. The American Foot and Ankle Surgery Association (AOFAS) score was 75/100. We performed a total resection of the talus followed by unrestricted talus replacement. Three months post-operation, the child was able to walk unaided. Ankle function was assessed at 6 and 13 months post-surgery, with the AOFAS score improving from 75 to 91, indicating that her functional needs for daily life were largely met.</div></div><div><h3>Conclusion</h3><div>Following complete excision of the lesion, the immature bone surrounding the talus was successfully preserved using an unrestricted 3D-printed prosthesis during ankle reconstruction. Our patient demonstrated satisfactory ankle function during the 6-month follow-up. This method is both safe and stable, yielding promising results, particularly for juvenile patients.</div></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":"24 ","pages":"Article 101830"},"PeriodicalIF":2.1,"publicationDate":"2025-02-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143422503","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Elevated sclerostin levels contribute to reduced bone mineral density in non-ambulatory stroke patients","authors":"Hye Kyoung Lee ,&nbsp;Geneva Rose Notario ,&nbsp;Sun Young Won ,&nbsp;Jung Hwan Kim ,&nbsp;Su Min Lee ,&nbsp;Ha Seong Kim ,&nbsp;Sung-Rae Cho","doi":"10.1016/j.bonr.2025.101829","DOIUrl":"10.1016/j.bonr.2025.101829","url":null,"abstract":"<div><div>Osteoporosis following stroke is a significant impediment to patient recovery. Decreased mechanical loading and locomotion following the onset of paralysis in stroke patients, especially those who are non-ambulatory, contributes greatly to bone loss. Sclerostin, a protein encoded by the SOST gene, accumulates as a result of reduced mechanical loading and inhibits bone formation. This study explores the relationship between mechanical unloading, sclerostin levels, and bone mineral density (BMD) in stroke patients, utilizing three cohorts. Analysis of Cohort 1, consisting of patients with available sclerostin level measurements, found significantly elevated sclerostin levels in non-ambulatory patients compared to ambulatory patients, indicating the influence of ambulatory status on sclerostin regulation. Cohort 2, consisting of patients with BMD measurements, demonstrated that prolonged mechanical unloading in non-ambulatory patients resulted in a greater decline in BMD over time. Analysis in Cohort 3 patients, who had bilateral BMD measurements available, revealed that hemiplegic sides subjected to reduced mechanical loading exhibited lower BMD compared to non-hemiplegic sides. These findings collectively confirm the hypothesis that reduced mechanical loading elevates sclerostin levels and accelerates bone loss. By integrating data across the three cohorts, this study underscores the critical impact of mechanical unloading on bone health, particularly in chronic stroke patients with limited mobility. Our study provides clinical insights for treatments integrating ambulatory status, sclerostin levels, and BMD in chronic stroke patients and highlights an increased need for therapeutics targeting mechanical loading pathways and sclerostin accumulation which can be administered to treat chronic osteoporosis following stroke.</div></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":"25 ","pages":"Article 101829"},"PeriodicalIF":2.1,"publicationDate":"2025-02-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143685344","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Heterotopic ossification (HO) prophylaxis in total hip arthroplasty (THA): A systematic review of level I and level II evidence since 2000","authors":"Troy B. Puga ,&nbsp;McKenna W. Box ,&nbsp;Vincent M. Dieu ,&nbsp;Charles R. Marchese ,&nbsp;John T. Riehl","doi":"10.1016/j.bonr.2025.101828","DOIUrl":"10.1016/j.bonr.2025.101828","url":null,"abstract":"<div><h3>Introduction</h3><div>Heterotopic ossification (HO) is a somewhat common occurrence after total hip arthroplasty (THA), particularly with certain approaches. This complication can be detrimental to the success of the surgical outcome. Indomethacin and radiotherapy remain common treatment modalities; however, no true gold-standard treatment is universally agreed upon. This study aims to evaluate Level I and Level II evidence for treatment practices of HO prophylaxis since 2000.</div></div><div><h3>Methods</h3><div>To evaluate HO prophylaxis in total hip arthroplasty, a search was conducted across MEDLINE/Pubmed, Cochrane, and Embase databases using keywords and Medical Subject Heading (MeSH) terms. Titles and abstracts were screened for eligibility for inclusion criteria. Full texts were screened and included if they met eligibility criteria.</div></div><div><h3>Results</h3><div>HO chemical prophylaxis was more effective than no HO prophylaxis, except for aspirin. Multiple NSAIDs showed equivalence and better side effect profiles than indomethacin. No one superior NSAID was found, and numerous modalities showed efficacy. The most effective dosages of radiation therapy and combination therapy remain unclear. Additionally, both etidronate and salmon calcitonin showed benefit in preventing HO in one study each.</div></div><div><h3>Conclusion</h3><div>Radiation, NSAIDs, and combination therapy all showed efficacy as HO prophylaxis modalities. HO prophylaxis treatment and modalities should be guided upon patient and surgical factors such as surgical approach, side effects and tolerability of modalities, comorbidities, and available facility resources to optimize the prevention of HO.</div><div>Level of evidence: Level IV Therapeutic.</div></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":"24 ","pages":"Article 101828"},"PeriodicalIF":2.1,"publicationDate":"2025-01-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143158952","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Long-term effects of infrapatellar fat pad SVF infiltration in knee osteoarthritis management: A prospective cohort study","authors":"Klaus Werner Labarre,&nbsp;Gerald Zimmermann","doi":"10.1016/j.bonr.2025.101827","DOIUrl":"10.1016/j.bonr.2025.101827","url":null,"abstract":"&lt;div&gt;&lt;h3&gt;Background&lt;/h3&gt;&lt;div&gt;Knee osteoarthritis (OA) is a prevalent and debilitating condition that significantly impacts patients' quality of life and poses a substantial socioeconomic burden. Current treatments, including nonsteroidal anti-inflammatory drugs (NSAIDs) and physical therapy, often provide only temporary relief and fail to halt disease progression, particularly in advanced stages where knee replacement surgery becomes the primary option. Regenerative cell therapies, particularly those utilizing mesenchymal stem cells (MSCs), have emerged as promising alternatives due to their anti-inflammatory and regenerative properties. This study investigates the efficacy of stromal vascular fraction (SVF) derived from autologous adipose tissue when injected into the infrapatellar (Hoffa's) fat pad, an approach that leverages the rich vascular and stem cell environment of the fat pad to potentially modulate inflammation and promote tissue repair.&lt;/div&gt;&lt;/div&gt;&lt;div&gt;&lt;h3&gt;Methods&lt;/h3&gt;&lt;div&gt;Patients receiving therapy with SVF were invited to participate in the study. Inclusion criteria encompassed male and female patients aged 18 years or older with a Kellgren-Lawrence score up to 4, while exclusion criteria included malignant tumors, sepsis, or skin lesions at the site of collection or injection. A total of 25 patients were included in the study cohort, with two patients receiving bilateral treatment, resulting in 27 knees analyzed.&lt;/div&gt;&lt;div&gt;For the correlation analysis, an additional four patients who had only completed the six-month follow-up were included, one of whom underwent bilateral treatment. This extended the correlation analysis cohort to 29 patients and 32 knees. However, these four patients were excluded from the final study analysis as they had not completed the two-year follow-up. Consequently, the final analysis focused exclusively on the 25 patients (27 knees) who completed the full two-year follow-up.&lt;/div&gt;&lt;/div&gt;&lt;div&gt;&lt;h3&gt;Results&lt;/h3&gt;&lt;div&gt;Significant improvements were observed in VAS pain scores and KOOS subscales for pain, activities of daily living (ADL), and quality of life (QOL) at 6 and 24 months (&lt;em&gt;p&lt;/em&gt; &lt; 0.05). The correlation between the number of injected cells and functional improvements was significant for ADL at 6 months (Spearman's rho = 0.31, &lt;em&gt;p&lt;/em&gt; = 0.044). This time point was prioritized to evaluate early therapeutic responses, as it represents a critical window when cellular activity and therapeutic effects are believed to peak. Focusing on the six-month follow-up allowed for a detailed assessment of these early impacts while minimizing potential confounding factors observed in later stages. No major complications were reported.&lt;/div&gt;&lt;/div&gt;&lt;div&gt;&lt;h3&gt;Conclusion&lt;/h3&gt;&lt;div&gt;SVF infiltration into the infrapatellar fat pad shows promising long-term benefits in pain relief and functional improvement for knee OA patients. Despite the lack of blinding and a control group, these findings suggest that SVF therapy coul","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":"24 ","pages":"Article 101827"},"PeriodicalIF":2.1,"publicationDate":"2025-01-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143158119","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"A simple and user-friendly machine learning model to detect osteoporosis in health examination populations in Southern Taiwan","authors":"Wei-Chin Huang ,&nbsp;I-Shu Chen ,&nbsp;Hsien-Chung Yu ,&nbsp;Chi-Shen Chen ,&nbsp;Fu-Zong Wu ,&nbsp;Chiao-Lin Hsu ,&nbsp;Pin-Chieh Wu","doi":"10.1016/j.bonr.2025.101826","DOIUrl":"10.1016/j.bonr.2025.101826","url":null,"abstract":"<div><h3>Background</h3><div>Osteoporosis is a growing public health concern in aging populations such as Taiwan, where limited utilization of dual-energy X-ray absorptiometry (DXA) often leads to underdiagnosis and even delayed treatment. Therefore, we leveraged machine learning (ML) and aimed to develop a simple and easily accessible model that effectively identifies individuals at high risk of osteoporosis.</div></div><div><h3>Methods</h3><div>This retrospective analysis enrolled 5510 men aged ≥50 years and 4720 postmenopausal women who underwent DXA at the Kaohsiung Veterans General Hospital, with another cohort of 610 men and 523 women for validation. We developed separate models for men and women using decision trees, random forests, support vector machines, k-nearest neighbors, extreme gradient boosting, and artificial neural networks (ANNs) to predict osteoporosis. Furthermore, we compared each model with the traditional Osteoporosis Self-Assessment Tool for Asians (OSTA) model.</div></div><div><h3>Results</h3><div>We identified age, height, weight, and BMI as variables for our prediction model and evaluated the model's performance using the area under the receiver operating characteristic curve (AUC). The ANN model significantly outperformed the OSTA model and all the other ML models for both men and women (AUC: 0.67 for men; 0.77 for women). The validation data for the ANN model showed similar AUCs for both men and women.</div></div><div><h3>Conclusion</h3><div>This study developed ML models to help identify individuals at high risk of osteoporosis in postmenopausal women and men aged ≥50 years in southern Taiwan. Our ML models, especially the ANN model, surpassed the OSTA model and consistently performed well across different populations.</div></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":"24 ","pages":"Article 101826"},"PeriodicalIF":2.1,"publicationDate":"2025-01-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11783436/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143078586","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Ano5Cys360Tyr mutation leads to bone dysfunction of gnathodiaphyseal dysplasia via disturbing Akt signaling","authors":"Hongyu Li,&nbsp;Shengnan Wang,&nbsp;Shuai Zhang,&nbsp;Rui Dong,&nbsp;Congcong Miao,&nbsp;Zhenchuan Tian,&nbsp;Ying Hu","doi":"10.1016/j.bonr.2025.101825","DOIUrl":"10.1016/j.bonr.2025.101825","url":null,"abstract":"<div><h3>Background</h3><div>Gnathodiaphyseal dysplasia (GDD) is a rare autosomal dominant genetic disease characterized by osteosclerosis of the tubular bones and cemento-osseous lesions of the mandibles. <em>Anoctamin 5</em> (<em>ANO5</em>) is the pathogenic gene, however, the specific molecular mechanism of GDD remains unclear. Herein, a knockin (<em>Ano5</em>^{<em>KI/KI</em>}) mouse model expressing the human mutation p.Cys360Tyr was used to investigate the role of Akt signaling in enhanced osteogenesis and decreased osteoclastogenesis in GDD.</div></div><div><h3>Methods</h3><div>Bone marrow-derived macrophages (BMMs) and mouse calvarial osteoblasts (mCOBs) were isolated from homozygous <em>Ano5</em>^{<em>KI/KI</em>} mice and treated with SC79, a specific Akt activator. The differentiation and F-actin ring formation of osteoclasts were examined by TRAP and phalloidin staining, respectively. Osteoblast differentiation and mineralization were examined by ALP and alizarin red staining. The expression of bone remodeling-related factors was measured by qRT-PCR.</div></div><div><h3>Results</h3><div>Akt activation promoted the generation of TRAP-positive multinucleated osteoclasts and the formation of actin rings in <em>Ano5</em>^{<em>KI/KI</em>} BMMs cultures, accompanied by increased expression of <em>Nfatc1</em>, <em>Trap</em>, <em>Dc-stamp</em>, <em>Mmp9</em>, <em>Ctsk</em>, and <em>Atp6v0d2</em>. Additionally, <em>Ano5</em>^{<em>Cys360Tyr</em>} mutation down-regulated the Akt phosphorylation level in osteoblast. ALP activity and matrix mineralization capacity in <em>Ano5</em>^{<em>KI/KI</em>} osteoblast cultures were inhibited after SC79 stimulation, with reduced expression of <em>Runx2, Opn, Col1a1</em>, <em>and Ocn</em>.</div></div><div><h3>Conclusion</h3><div>Akt activation by SC79 stimulation can obviously rescue abnormal increased osteogenesis and decreased osteoclastogenesis in <em>Ano5</em>^{<em>KI/KI</em>} mouse model, which demonstrated that disturbed Akt signaling pathway may play a pivotal role in the pathogenesis of GDD, and an Akt activator is probable a therapeutic target for GDD.</div></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":"24 ","pages":"Article 101825"},"PeriodicalIF":2.1,"publicationDate":"2025-01-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11763220/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143045635","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Ischemic stroke reduces bone perfusion and alters osteovascular structure","authors":"Nicholas J. Hanne ,&nbsp;Andrew J. Steward ,&nbsp;Carla Geeroms ,&nbsp;Elizabeth D. Easter ,&nbsp;Hannah T. Gensch ,&nbsp;Greet Kerckhofs ,&nbsp;Tatjana N. Parac-Vogt ,&nbsp;Huaxin Sheng ,&nbsp;Jacqueline H. Cole","doi":"10.1016/j.bonr.2025.101824","DOIUrl":"10.1016/j.bonr.2025.101824","url":null,"abstract":"<div><div>Stroke patients lose bone mass and experience fracture at an elevated rate. Although functional intraosseous vasculature is necessary for skeletal maintenance, the effect of stroke on osteovasculature is unknown. In this study we characterized changes to osteovascular perfusion, structure, and composition following mild-to-moderate stroke severity in mice, both with and without exercise therapy. Twelve-week-old male mice (<em>n</em> = 27) received either an ischemic stroke (middle cerebral artery occlusion) or sham procedure, followed by a four-week recovery with either moderate daily treadmill or sedentary activity. Intraosseous perfusion, measured weekly in the proximal tibial metaphysis with laser Doppler flowmetry, was reduced for two weeks in the stroke group relative to the sham group. After four weeks, osteovascular structure was assessed in the distal femoral metaphysis with contrast-enhanced computed tomography. Increased osteovascular volume and branching, decreased number of smaller vessels (6–22 μm), and increased number of larger vessels (&gt;66 μm) were observed in the stroke groups compared to sham groups, which may be a compensatory response to early perfusion deficits. Although moderate exercise mitigated the impact of stroke on osteovascular perfusion and volume, it tended to reduce the amount of osteogenic type H vasculature quantified with immunofluorescence microscopy and, exacerbated by stroke effects, produced fewer vessels in close proximity to bone and thus may have detrimental effects on bone remodeling during early stroke recovery. Since results were similar in both limbs, the effects of ischemic stroke on osteovascular perfusion and structure were primarily systemic, rather than resulting from paresis or disuse, providing new insight for future studies on the pathogenesis and treatment of skeletal fragility in stroke patients.</div></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":"24 ","pages":"Article 101824"},"PeriodicalIF":2.1,"publicationDate":"2025-01-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11782850/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143078625","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"The association between dietary inflammatory index and bone health in US adolescents: Analysis of the NHANES data","authors":"Yuanyuan Zhang,&nbsp;Xuejing Wang,&nbsp;Shiguang Huo,&nbsp;Li Hong,&nbsp;Feifei Li","doi":"10.1016/j.bonr.2024.101823","DOIUrl":"10.1016/j.bonr.2024.101823","url":null,"abstract":"<div><h3>Introduction</h3><div>Adolescents with a lower peak bone mineral density (BMD) and bone mineral content (BMC) have an elevated risk of osteoporosis in adulthood. The impact of diet on bone health, particularly its role in managing inflammation, which is a key factor in bone health, is gaining wider recognition. Despite evidence that anti-inflammatory diets can enhance bone health, the link between the dietary inflammatory index (DII) and bone health among US adolescents has not been thoroughly investigated. This study aimed to evaluate the correlation between DII score and bone health in this population.</div></div><div><h3>Methods</h3><div>This cross-sectional study used data from the National Health and Nutrition Examination Survey (NHANES) of US adolescents aged 12–18 years, spanning surveys from 2001 to 2018. The DII was derived from dietary recall data obtained through questionnaire interviews. Bone health was assessed through total body less head (TBLH) BMD and BMC z-scores and lumbar spine bone mineral apparent density for age (BMAD_a).</div></div><div><h3>Results</h3><div>The study comprised 8773 adolescents with a mean age of 14.94 ± 1.97 years, 52.2 % were male. Multivariate linear regression analysis revealed a negative correlation between DII and lumbar spine BMAD_a (β = −0.000003, 95 % confidence interval [CI], −0.000005 to −0.000001; <em>P</em> = 0.001).This significant association remained robust when DII was treated as a categorical variable. Compared with individuals in quartile 1(Q1) DII scores (−3.71 to 1.04), those in Q4 (3.37 to 5.04) had lower BMAD_a, with a regression coefficient of −0.00002 (95 % CI, −0.00003 to −0.000007, <em>P</em> &lt; 0.001). DII was negatively correlated with TBLH BMC z-scores; however, the difference was not statistically significant. Subgroup analyses showed that DII was associated with lumbar spine BMAD_a and TBLH BMC z-scores in participants who were male, non-black, with a higher educational level, with a high family income, and underweight to normal weight. We found no significant association between DII and TBLH BMD z-scores.</div></div><div><h3>Conclusion</h3><div>The findings from this cross-sectional analysis indicate a significant association between the DII and bone health among adolescents in the US, with a notable impact in males and non-black. These insights underscore the importance of adopting dietary patterns to mitigate inflammation and to support optimal bone health and metabolism.</div></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":"24 ","pages":"Article 101823"},"PeriodicalIF":2.1,"publicationDate":"2025-01-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11758120/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143045638","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Clinical features and treatment of hypophosphatemia and associated complications induced by Phosphaturic mesenchymal tumors: A case series of six patients","authors":"Guoqiang Lai ,&nbsp;Wangsheng Zuo ,&nbsp;Runmin Tang ,&nbsp;Zengbo Lu ,&nbsp;Dehai Shi","doi":"10.1016/j.bonr.2024.101822","DOIUrl":"10.1016/j.bonr.2024.101822","url":null,"abstract":"<div><div>Phosphaturic mesenchymal tumor (PMT) is a rare benign mesenchymal tumor characterized by excessive secretion of fibroblast growth factor 23 (FGF23), leading to phosphate loss and systemic osteomalacia. Despite recent progress in PMT research, no consensus on diagnosis and treatment guidelines has been established. This case series describes the clinical and pathological features of six pathologically confirmed PMT patients treated at the Third Affiliated Hospital of Sun Yat-sen University from 2010 to 2024, aiming to provide new insights for the management of this condition. The patients, consisting of three males and three females with an average age of 44 years and follow-up periods of 0.5 to 4.5 years, presented primarily with muscle pain and lower limb weakness. One patient experienced loose teeth, and two had palpable, painless masses. One case developed hyperphosphatemia, tertiary hyperparathyroidism, and renal impairment after prolonged phosphate supplementation. Tumor localization was achieved using 18F-FDG or 68Ga-DOTATATE Positron Emission Tomography-Computed Tomography(PET/CT) and MRI, followed by complete surgical resection. Pathological examination confirmed PMT, and postoperative recovery was marked by significant symptom relief and normalization of serum phosphate levels. Two patients experienced recurrence within three years but showed no further recurrence following repeat surgery by the last follow-up. The diagnosis of PMT is challenging and may take years, potentially leading to complications due to inadequate treatment. Complete tumor resection remains the primary treatment, generally resulting in a favorable prognosis; however, long-term monitoring is essential to detect potential recurrences and initiate timely interventions.</div></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":"24 ","pages":"Article 101822"},"PeriodicalIF":2.1,"publicationDate":"2024-12-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11760829/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143045636","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Integrating deep learning and machine learning for improved CKD-related cortical bone assessment in HRpQCT images: A pilot study","authors":"Youngjun Lee ,&nbsp;Wikum R. Bandara ,&nbsp;Sangjun Park ,&nbsp;Miran Lee ,&nbsp;Choongboem Seo ,&nbsp;Sunwoo Yang ,&nbsp;Kenneth J. Lim ,&nbsp;Sharon M. Moe ,&nbsp;Stuart J. Warden ,&nbsp;Rachel K. Surowiec","doi":"10.1016/j.bonr.2024.101821","DOIUrl":"10.1016/j.bonr.2024.101821","url":null,"abstract":"<div><div>High resolution peripheral quantitative computed tomography (HRpQCT) offers detailed bone geometry and microarchitecture assessment, including cortical porosity, but assessing chronic kidney disease (CKD) bone images remains challenging. This proof-of-concept study merges deep learning and machine learning to 1) improve automatic segmentation, particularly in cases with severe cortical porosity and trabeculated endosteal surfaces, and 2) maximize image information using machine learning feature extraction to classify CKD-related skeletal abnormalities, surpassing conventional DXA and CT measures.</div><div>We included 30 individuals (20 non-CKD, 10 stage 3 to 5D CKD) who underwent HRpQCT of the distal and diaphyseal radius and tibia and contributed data to develop and validate four different AI models for each anatomical site. Manually annotated cortical bone was used to train each segmentation deep-learning model. Textural features were extracted via Gray-Level Co-occurrence Matrix (GLCM) and classified as CKD or non-CKD using XGBoost with each segmentation model. For comparison, manufacturer-supplied segmentation was used to extract cortical geometry, microarchitecture, and finite element analysis (FEA) outcomes. Model performance was confirmed using the test dataset and a separate independent validation cohort which included HRpQCT imaging from 42 additional individuals (18 non-CKD, 24 CKD stage 5D).</div><div>For segmentation, the diaphyseal location showed strong performance on test datasets, with Mean IoUs of 0.96 and 0.95, and accuracies of 0.97 for both radius and tibia sites in CKD. Model 4 developed from the diaphyseal tibia region excelled in classifying test and independent validation datasets, achieving F1 scores of 0.99 and 0.96, AUCs of 0.99 and 0.94, sensitivities of 0.99, and specificities of 0.99 and 0.92. No single parameter, including BMD and cortical porosity, among conventional CT outcomes consistently differentiated CKD from non-CKD across all anatomical sites.</div><div>Integrating HRpQCT with deep and machine learning, this innovative approach enables precise automatic segmentation of severely deteriorated endocortical surfaces and enhances sensitivity to CKD-related cortical bone changes compared to standard DXA and HRpQCT outcomes.</div></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":"24 ","pages":"Article 101821"},"PeriodicalIF":2.1,"publicationDate":"2024-12-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11763521/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143045637","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}