Bone Reports最新文献_第7页

Intake of eggshell membrane enhances bone mass and suppresses bone marrow adiposity in normal growing rats 摄取蛋壳膜可增加正常生长大鼠的骨量，抑制骨髓脂肪

IF 2.1 Q3 ENDOCRINOLOGY & METABOLISM

Bone Reports

Pub Date : 2025-04-01 DOI: 10.1016/j.bonr.2025.101840

Nao Yashima , Kaoru Fujikawa , Wataru Minamizono , Hiroya Matsunaga , Jiazheng Lyu , Hirai Suito , Takumi Okunuki , Shingo Nakai , Masafumi Ohsako

Eggshell membrane intake is considered to have beneficial effects on bone health; however, relevant evidence remains scant. Therefore, we aimed to explore the direct effects of eggshell membrane intake on osteogenic function in normal growing rats. Six-week-old male Wistar rats were divided into control (CO) and eggshell membrane (EM) groups. The experiment was conducted over 8 weeks. Visual observation and micro-computed tomography analysis revealed a significant increase in bone mass in the EM group compared with that in the CO group. Histological analysis showed thick and long trabeculae in the EM group, accompanied by an increase in the number of osteoblasts and suppression of adipocyte accumulation. Furthermore, Col1a1 expression was significantly higher in the EM group than in the CO group, although no significant differences were found in the number of TRAP-positive osteoclasts or Ctsk expression. Immunohistochemical analysis demonstrated a notable increase in the number of Col1-positive osteoblasts but a significant decrease in the number of Dlk1-positive adipocytes in the EM group. Gene expression analysis revealed no difference in the expression of Runx2 (the master regulator of osteoblast differentiation) between the groups. However, the expression of Sp7, which functions downstream of Runx2, was significantly upregulated, whereas that of Pparg, the master regulator of adipocyte differentiation, was significantly downregulated in the EM group compared with those in the CO group. Overall, the intake of eggshell membranes may enhance osteogenic function and suppress bone marrow adiposity. These findings support the beneficial effects of eggshell membrane intake on bone health.

蛋壳膜的摄入被认为对骨骼健康有益；然而，相关证据仍然不足。因此，我们旨在探讨蛋壳膜摄取量对正常生长大鼠成骨功能的直接影响。6周龄雄性Wistar大鼠分为对照组（CO）和蛋壳膜组（EM）。实验进行了8周。目视观察和显微计算机断层扫描分析显示，与CO组相比，EM组骨量明显增加。组织学分析显示EM组骨小梁粗长，成骨细胞数量增加，脂肪细胞积聚受到抑制。此外，尽管在trap阳性破骨细胞数量或Ctsk表达方面没有发现显著差异，但EM组Col1a1的表达明显高于CO组。免疫组织化学分析显示，EM组中col1阳性的成骨细胞数量显著增加，而dlk1阳性的脂肪细胞数量显著减少。基因表达分析显示，各组间Runx2（成骨细胞分化的主要调控因子）的表达无差异。然而，与CO组相比，EM组Runx2下游功能的Sp7表达显著上调，而脂肪细胞分化的主要调控因子Pparg表达显著下调。综上所述，摄取蛋壳膜可增强成骨功能，抑制骨髓脂肪。这些发现支持蛋壳膜摄入对骨骼健康的有益作用。

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引用次数: 0

Can AI reveal the next generation of high-impact bone genomics targets? 人工智能能否揭示下一代高影响骨基因组学目标？

IF 2.1 Q3 ENDOCRINOLOGY & METABOLISM

Bone Reports

Pub Date : 2025-03-24 DOI: 10.1016/j.bonr.2025.101839

Casey S. Greene , Christopher R. Gignoux , Marc Subirana-Granés , Milton Pividori , Stephanie C. Hicks , Cheryl L. Ackert-Bicknell

Genetic studies have revealed hundreds of loci associated with bone-related phenotypes, including bone mineral density (BMD) and fracture risk. However, translating discovered loci into effective new therapies remains challenging. We review success stories including PCSK9-related drugs in cardiovascular disease and evidence supporting the use of human genetics to guide drug discovery, while highlighting advances in artificial intelligence and machine learning with the potential to improve target discovery in skeletal biology. These strategies are poised to improve how we integrate diverse data types, from genetic and electronic health records data to single-cell profiles and knowledge graphs. Such emerging computational methods can position bone genomics for a future of more precise, effective treatments, ultimately improving the outcomes for patients with common and rare skeletal disorders.

遗传学研究已经揭示了数百个与骨相关表型相关的基因座，包括骨矿物质密度（BMD）和骨折风险。然而，将发现的基因座转化为有效的新疗法仍然具有挑战性。我们回顾了包括pcsk9相关药物在心血管疾病中的成功案例，以及支持使用人类遗传学指导药物发现的证据，同时强调了人工智能和机器学习的进展，这些进展有可能改善骨骼生物学中的靶点发现。这些策略将改善我们如何整合各种数据类型，从遗传和电子健康记录数据到单细胞概况和知识图谱。这种新兴的计算方法可以为未来更精确、更有效的治疗定位骨基因组学，最终改善患有常见和罕见骨骼疾病的患者的治疗效果。

引用次数: 0

Bone mineral density and microarchitecture improvement in a young patient with Hajdu-Cheney syndrome and autosomal dominant polycystic kidney disease treated with alendronate 阿仑膦酸钠治疗Hajdu-Cheney综合征合并常染色体显性多囊肾病年轻患者的骨密度和微结构改善

IF 2.1 Q3 ENDOCRINOLOGY & METABOLISM

Bone Reports

Pub Date : 2025-03-24 DOI: 10.1016/j.bonr.2025.101838

André Silva Franco , Valeria de Falco Caparbo , Elieser Hitoshi Watanabe , Rosa Maria Rodrigues Pereira , Luiz Fernando Onuchic

Introduction

Osteoporosis, typically seen in postmenopausal women, can also affect younger individuals, a condition known as Early-Onset Osteoporosis (EOOP). EOOP may be secondary to various conditions or arise from rare genetic disorders such as Hajdu-Cheney Syndrome (HCS), characterized by systemic bone involvement and fragility fractures.

Case Report

A 14-year-old male presented with a distal left femur fragility fracture. His medical history included spina bifida and bilateral tarsal coalition, with no family history of osteoporosis, and polycystic kidneys associated with a positive family history of autosomal dominant polycystic kidney disease (ADPKD). Laboratory tests were unremarkable, but dual X-ray absorptiometry (DXA) revealed low bone mineral density (BMD), and high resolution peripheral quantitative computed tomography (HR-pQCT) showed decreased volumetric bone density (vBMD), particularly in the cortical bone. At age 17, his kidneys were cystic and mildly enlarged. Whole exome sequencing revealed a pathogenic variant in NOTCH2, confirming the diagnosis of HCS, and a very likely causative variant in PKD1, supporting the diagnosis of ADPKD.

The treatment regimen included weekly alendronate, impact exercise, a calcium-rich diet, and vitamin D supplementation. After 3 years, follow-up DXA and HR-pQCT demonstrated significant improvements in BMD and vBMD, mainly in the cortical bone.

Discussion

This case highlights the effectiveness of alendronate in managing osteoporosis in a patient with HCS and ADPKD, despite the current lack of strong supportive evidence. Long-term monitoring revealed substantial improvements in bone density and microarchitecture, underscoring the importance of early diagnosis and intervention for genetic causes of osteoporosis to prevent fracture-related morbidity.

骨质疏松症，通常见于绝经后妇女，也可以影响年轻人，这种情况被称为早发性骨质疏松症（EOOP）。EOOP可能继发于各种疾病，或由Hajdu-Cheney综合征（HCS）等罕见遗传疾病引起，其特征是全身骨骼受累和脆性骨折。病例报告：一名14岁男性表现为左股骨远端脆性骨折。病史包括脊柱裂和双侧跗骨联合，无骨质疏松家族史，多囊肾伴常染色体显性多囊肾病（ADPKD）家族史阳性。实验室检查无明显异常，但双x线吸收仪（DXA）显示低骨密度（BMD），高分辨率外周定量计算机断层扫描（HR-pQCT）显示体积骨密度（vBMD）下降，尤其是皮质骨。17岁时，他的肾脏呈囊状并轻度增大。全外显子组测序显示NOTCH2的致病变异，证实了HCS的诊断，PKD1的致病变异非常可能，支持ADPKD的诊断。治疗方案包括每周阿仑膦酸钠、冲击性运动、富含钙的饮食和补充维生素D。3年后，随访DXA和HR-pQCT显示BMD和vBMD显著改善，主要在皮质骨。本病例强调了阿仑膦酸钠治疗HCS和ADPKD患者骨质疏松症的有效性，尽管目前缺乏强有力的支持证据。长期监测显示骨密度和微结构的显著改善，强调了早期诊断和干预骨质疏松症遗传原因以预防骨折相关发病率的重要性。

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引用次数: 0

Mild antiresorptive activity of an anti-vascular endothelial growth factor A antibody and sunitinib in a rat model of bone resorption 抗血管内皮生长因子A抗体和舒尼替尼在骨吸收大鼠模型中的轻度抗骨吸收活性

IF 2.1 Q3 ENDOCRINOLOGY & METABOLISM

Bone Reports

Pub Date : 2025-03-17 DOI: 10.1016/j.bonr.2025.101837

J.I. Aguirre, S.M. Croft, E.J. Castillo, C.J. Cruz-Camacho, D.B. Kimmel

Medication-Related-Osteonecrosis-of-the-Jaw (MRONJ) is an adverse event linked to antiresorptives such as bisphosphonates and denosumab. While MRONJ predominantly affects cancer patients treated with these agents, it has been less frequently reported in cancer patients receiving angiogenesis inhibitors (AgIs) like bevacizumab and sunitinib, even without concurrent use of antiresorptives. We hypothesized that certain AgIs exhibit antiresorptive activity in addition to their antiangiogenic effects, potentially influencing the pathophysiology of MRONJ.

52 five-week-old SD rats were randomized to receive vehicle (VEH), an oncologic dose of zoledronic acid (ZOL), or low (LD) and high doses (HD) of either an anti-VEGFA antibody or sunitinib (SU) for 10 days. We used the Schenk assay to assess the in vivo antiresorptive properties of these drugs/agents. We evaluated serum biomarkers of bone resorption (TRACP 5b) and formation (P1NP), pQCT variables of the femurs/tibias, and bone resorption/formation variables by bone histomorphometry at the distal femur metaphysis.

ZOL reduced TRACP-5b levels, osteoclast number, and BFR while increasing vBMD, mineralized tissue volume, calcified cartilage volume, and bone volume. Both anti-VEGFA and SU decreased osteoclast number and increased calcified cartilage volume relative to total mineralized tissue volume, though to a lesser extent than ZOL. Anti-VEGFA (HD) also reduced TRACP-5b levels. Furthermore, both AgIs decreased P1NP levels, MAR, and bone elongation rate but increased growth cartilage thickness and induced physeal dysplasia.

In conclusion, AgIs, particularly anti-VEGFA, exhibit significant yet milder antiresorptive activity compared to ZOL. They also affect bone formation, suggesting a complex mechanism that may play a role in the pathophysiology of MRONJ.

药物相关性颌骨坏死（MRONJ）是一种与双膦酸盐和地诺单抗等抗吸收药物相关的不良事件。虽然MRONJ主要影响接受这些药物治疗的癌症患者，但在接受血管生成抑制剂（AgIs）如贝伐单抗和舒尼替尼的癌症患者中，即使没有同时使用抗吸收药物，也很少报道。我们假设某些AgIs除了具有抗血管生成作用外还具有抗吸收活性，可能影响mron2的病理生理。52只5周大的SD大鼠随机接受载药（VEH）、肿瘤剂量唑来来酸（ZOL）、低（LD）和高剂量（HD）抗vegfa抗体或舒尼替尼（SU）治疗10天。我们使用申克试验来评估这些药物/制剂的体内抗吸收特性。我们评估了骨吸收（TRACP 5b）和形成（P1NP）的血清生物标志物，股骨/胫骨的pQCT变量，以及股骨远端骺端骨组织形态学测量的骨吸收/形成变量。ZOL降低了TRACP-5b水平、破骨细胞数量和BFR，同时增加了vBMD、矿化组织体积、钙化软骨体积和骨体积。抗vegfa和SU均能降低破骨细胞数量，增加钙化软骨体积（相对于矿化组织总体积），但程度低于ZOL。抗vegfa （HD）也降低了TRACP-5b水平。此外，两种AgIs均降低了P1NP水平、MAR和骨伸长率，但增加了生长软骨厚度并诱导了骨性发育不良。总之，与ZOL相比，AgIs，特别是抗vegfa，表现出显著但较温和的抗吸收活性。它们也会影响骨形成，这表明在MRONJ的病理生理中可能起作用的复杂机制。

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引用次数: 0

Chronic heavy alcohol consumption impairs the ability of demineralized allogenic bone matrix to support osteoinduction in alcohol-naïve rats 慢性重度饮酒损害alcohol-naïve大鼠脱矿异体骨基质支持骨诱导的能力

IF 2.1 Q3 ENDOCRINOLOGY & METABOLISM

Bone Reports

Pub Date : 2025-03-12 DOI: 10.1016/j.bonr.2025.101836

Russell T. Turner , Amida F. Kuah , Cynthia H. Trevisiol , Kathy S. Howe , Adam J. Branscum , Urszula T. Iwaniec

Allografts play an important role in treatment of complex bone fractures and deformities. The purpose of this study was to test the hypothesis that alcohol consumption impairs graft incorporation and bone healing by two mechanisms: (1) by lowering osteoinductive capacity and (2) by suppressing bone formation. We performed experiments using a demineralized allogeneic bone matrix (DBM) model in which DBM harvested from donor rats fed control or ethanol diet was implanted subcutaneously into recipient rats fed control or ethanol diet. We also evaluated the efficacy of intermittent parathyroid hormone (PTH) on bone graft incorporation (DBM from donor rats fed alcohol or control diet) using a critical size defect model. Bone formed during osteoinduction was measured by micro-computed tomography. Experiment 1: Bone volume was lower in DBM harvested from ethanol-consuming donors 6 weeks following implantation into recipients fed control diet, indicating that exposure of the donor rats to ethanol lowered osteoinductive capacity. Experiment 2: Bone volume was lower in DBM harvested 3 weeks following implantation from ethanol-consuming donors into ethanol-consuming recipients compared to DBM harvested from control donors implanted into control recipients or DBM harvested from control donors implanted into ethanol-consuming recipients. Experiment 3: Ethanol consumption by donors resulted in a tendency for lower DBM bone volume (p = 0.085) whereas PTH treatment resulted in higher DBM bone volume in the critical size defect model. Our results suggest that chronic heavy alcohol consumption by allograft donors may impair osteoinduction and this negative outcome may be worsened by alcohol intake during bone healing. Additionally, PTH has the potential to increase osteoinduction in DBM harvested from both abstinent and alcohol-consuming donors.

同种异体骨移植在复杂骨折和畸形的治疗中发挥着重要作用。本研究的目的是验证饮酒通过两种机制损害移植物结合和骨愈合的假设：(1)降低骨诱导能力；(2)抑制骨形成。我们使用脱矿异体骨基质（DBM）模型进行了实验，在该模型中，从喂食对照或乙醇饮食的供体大鼠身上采集的DBM皮下植入喂食对照或乙醇饮食的受体大鼠。我们还使用临界尺寸缺陷模型评估了间歇性甲状旁腺激素（PTH）对骨移植结合（来自喂食酒精或对照饮食的供体大鼠的DBM）的功效。骨诱导过程中形成的骨通过微型计算机断层扫描测量。实验1：将乙醇供体的DBM植入对照组小鼠6周后，骨体积降低，表明乙醇供体大鼠的骨诱导能力降低。实验2：与将对照供体骨骨移植到对照受者或将对照供体骨骨移植到摄入乙醇受者的骨骨骨骨相比，将摄入乙醇的供体骨骨骨骨移植3周后，从摄入乙醇的供体骨骨骨移植到摄入乙醇的受者的骨骨骨体积更小。实验3：在临界尺寸缺陷模型中，供体消耗乙醇导致DBM骨体积减小（p = 0.085），而PTH治疗导致DBM骨体积增大。我们的研究结果表明，同种异体移植供体长期大量饮酒可能会损害骨诱导，并且在骨愈合期间饮酒可能会使这种负面结果恶化。此外，甲状旁腺激素有可能增加来自戒酒和饮酒供体的DBM的骨诱导。

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引用次数: 0

The biological function of integrin-linked kinase on bone formation 整合素连接激酶在骨形成中的生物学功能

IF 2.1 Q3 ENDOCRINOLOGY & METABOLISM

Bone Reports

Pub Date : 2025-03-10 DOI: 10.1016/j.bonr.2025.101834

Yu-ling Liu , Yue-ming Mei , Jing-qiong Xun , Zhuo-yue Lv , Qian He , Zhou-bo-ran Liu , Lin Li , Fen Xie , Ru-chun Dai

Bone remodeling process is the basis for maintaining normal bone microstructure and promoting fracture repair. Recent studies have proven that integrins can promote bone formation and fracture repair. Integrin-linked kinase (ILK), as the proximal effector of the integrin receptor, is a key protein factor linking integrin and cytoskeleton. It is involved in crucial cellular processes including proliferation, survival, differentiation, migration, invasion, and angiogenesis reflects on systemic changes in the kidney, heart, muscle, skin, and vascular system. At present, the regulation effect of ILK in bone formation attracts the attention of researchers. This review emphasizes that ILK as a key molecule affects the functions of bone marrow stromal cells (BMSCs) and osteoblasts, and regulates bone formation. Additionally, ILK plays a key role in the process of“angiogenic–osteogenic coupling ”. The present role of ILK in the pathogenesis of osteoporosis is also described. Strategies that target ILK may as a new prospective treatment for osteoporosis (OP).

骨重塑过程是维持骨结构正常、促进骨折修复的基础。最近的研究证明整合素可以促进骨形成和骨折修复。整合素连接激酶（integrin -linked kinase， ILK）作为整合素受体的近端效应因子，是连接整合素与细胞骨架的关键蛋白因子。它参与了关键的细胞过程，包括增殖、存活、分化、迁移、侵袭和血管生成，反映了肾脏、心脏、肌肉、皮肤和血管系统的系统性变化。目前，ILK在骨形成中的调控作用引起了研究者的关注。本文综述了ILK作为影响骨髓基质细胞（BMSCs）和成骨细胞功能，调控骨形成的关键分子。此外，ILK在“血管生成-成骨耦合”过程中起着关键作用。本文还描述了ILK在骨质疏松症发病机制中的作用。针对ILK的策略可能成为骨质疏松症（OP）的一种新的前瞻性治疗方法。

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引用次数: 0

Corrigendum to “SERPINF1 gene variants causing late-onset progressive deforming osteogenesis imperfecta – A study of 18 patients from India” [Bone Rep. 2023 May 26;18:101690. doi: 10.1016/j.bonr.2023.101690] “serinf1基因变异导致迟发性进行性变形性成骨不全-一项来自印度的18例患者的研究”[骨杂志2023年5月26日；18:10 . 1690]的勘误。doi: 10.1016 / j.bonr.2023.101690]

IF 2.1 Q3 ENDOCRINOLOGY & METABOLISM

Bone Reports

Pub Date : 2025-03-01 DOI: 10.1016/j.bonr.2025.101832

Agnes Selina , Madhavi Kandagaddala , Vignesh Kumar , Suneetha Susan Cleave Abraham , Sumita Danda , Vrisha Madhuri

引用次数: 0

A novel SGMS2 mutation associated with high bone mass; description of an affected family with recurrent fragility fractures 与高骨量有关的新型 SGMS2 基因突变；描述一个反复发生脆性骨折的受影响家族

IF 2.1 Q3 ENDOCRINOLOGY & METABOLISM

Bone Reports

Pub Date : 2025-03-01 DOI: 10.1016/j.bonr.2025.101833

Shinjan Patra , Sweekruti Jena , Ketki Kedar , Minal Pande , Kishore K Katam , Ashka Prajapti , Udhaya Kotecha , Parin Vyas

SGMS2 mutation can present with childhood-onset low bone mass and recurrent fragility fractures. We report a 25-year-old man with a three-generation family history of recurrent fragility fractures and diffuse high bone mass. He was found to have a heterozygous frameshift variant c.1052_1074dup in the SGMS2 gene.

Our case highlights a novel genetic mutation in the SGMS2 gene and reports the first family of SGMS2 mutation with high bone mass.

SGMS2突变可表现为儿童期低骨量和复发性脆性骨折。我们报告一位25岁的男性，有三代复发性脆性骨折和弥漫性高骨量的家族史。在SGMS2基因中发现一个杂合移码变异c.1052_1074dup。我们的病例强调了SGMS2基因的一个新的基因突变，并报道了第一个具有高骨量的SGMS2突变家族。

引用次数: 0

Biomechanical influence of numerical variants of lumbosacral transitional vertebra with Castellvi type I on adjacent discs and facet joints based on 3D finite element analysis 基于三维有限元分析的Castellvi型腰骶过渡椎数值变异体对相邻椎间盘和关节突的生物力学影响

IF 2.1 Q3 ENDOCRINOLOGY & METABOLISM

Bone Reports

Pub Date : 2025-02-20 DOI: 10.1016/j.bonr.2025.101831

Tongxin Zhu, Zhangyan Xu, Dan Liu, Wei Zeng, Yongliang Pu, Haitao Yang

Objectives

To investigate the effect of lumbosacral transitional vertebra (LSTV) on the biomechanical properties of adjacent discs and facet joints based on geometrically 3D personalized FEA.

Methods

A total of 45 individuals who underwent low dose whole body CT scans were retrospectively included and equally divided into 23, 24, and 25 presacral vertebrae (PSV) groups. Three-dimensional Finite Element computational models of normal and number-variant sub-types of LSTV were created. The biomechanical parameters, including the range of motion (ROM), the intervertebral disc pressure (IDP), and facet joint forces (FJF), were all evaluated to determine the biomechanical effects. IDP was equally divided into anterior (AIDP), middle (MIDP) and posterior (PIDP) parts along the short axis of the intervertebral disc.

Results

During extension, the 23 PSV group exhibited significantly higher von Meiss stress in the upper intervertebral disc compared to the 24 and 25 PSV groups (P = 0.003), indicating concentrated stress in the upper lumbar region and an increased the likelihood of localized disc degeneration over time. Furthermore, the 23 PSV group exhibited a larger ROM (3.28°) than the 25 PSV group (1.40°) (P = 0.011), implying greater segmental mobility and possible instability in the transitional segment. During flexion, the 25 PSV group showed higher stress in the lower intervertebral disc and a larger ROM than the 23 and 24 PSV groups; however, the differences were not significant (P > 0.05).

Conclusions

The increased stress distribution and ROM in the upper disc of the transitional segment were only found in the 23 PSV sub-type of Castellvi type I LSTVs during extension, but not in the 25 PSV sub-type, which may help to further understand the impact of LSTV on the surrounding structures.

目的基于几何三维个性化有限元分析探讨腰骶过渡椎体（LSTV）对相邻椎间盘和关节突关节生物力学特性的影响。方法回顾性分析45例接受低剂量全身CT扫描的患者，平均分为23、24、25个骶前椎（PSV）组。建立了LSTV正常子型和变数子型的三维有限元计算模型。生物力学参数，包括活动范围（ROM）、椎间盘压力（IDP）和关节突关节力（FJF），均被评估以确定生物力学效应。沿椎间盘短轴将IDP平均分为前（AIDP）、中（MIDP）和后（PIDP）部分。结果在伸展过程中，与24和25 PSV组相比，23 PSV组在上椎间盘表现出更高的von Meiss应力（P = 0.003），表明应力集中在上腰椎区域，随着时间的推移，椎间盘局部退变的可能性增加。此外，23 PSV组的ROM（3.28°）比25 PSV组（1.40°）更大（P = 0.011），这意味着更大的节段流动性和过渡节段可能的不稳定性。屈曲时，与23和24 PSV组相比，25 PSV组下椎间盘的应力更高，ROM更大；但差异不显著(P >；0.05)。结论Castellvi I型LSTV在伸展过程中，仅23 PSV亚型出现过渡段上盘应力分布和ROM增加，而25 PSV亚型无此现象，这有助于进一步了解LSTV对周围结构的影响。

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引用次数: 0

Total talectomy and reconstruction using unrestricted 3D printed prosthesis for pediatric talus hemangioendothelioma 儿童距骨血管内皮瘤的全距骨切除术和3D打印假体重建

IF 2.1 Q3 ENDOCRINOLOGY & METABOLISM

Bone Reports

Pub Date : 2025-02-13 DOI: 10.1016/j.bonr.2025.101830

Yunlong Zhang, Zhichang Zhang

Background

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.

Case presentation

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.

Conclusion

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.

深上皮样血管内皮瘤（EHE）是一种极其罕见的血管肉瘤，发病率和患病率极低，尤其是在儿童中。我们报告的情况下，9岁的女孩诊断为EHE。距骨血管内皮瘤全距骨切除术后的重建方法有限，在儿童病例中使用3d打印距骨假体以前没有记录。病例介绍：一名9岁女孩因脚踝肿胀、疼痛和活动受限一个月无明显原因而来我科就诊。x线及CT显示距骨溶骨性病变，确定为低级别恶性肿瘤，几乎完全侵入距骨，周围为未成熟骨。美国足踝外科协会（AOFAS）评分为75/100。我们进行了距骨全切除术，然后进行了无限制距骨置换。手术后三个月，孩子可以独立行走了。术后6个月和13个月对踝关节功能进行评估，AOFAS评分从75分提高到91分，表明患者的日常生活功能需求基本得到满足。结论在完全切除病变后，在踝关节重建过程中使用不受限制的3d打印假体成功地保留了距骨周围的未成熟骨。在6个月的随访中，患者表现出满意的踝关节功能。这种方法既安全又稳定，产生了有希望的结果，特别是对青少年患者。

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引用次数: 0