JBMR Plus最新文献

{"title":"Correction to: Fracture discrimination capability of ulnar flexural rigidity measured via Cortical Bone Mechanics Technology: study protocol for The STRONGER Study.","authors":"","doi":"10.1093/jbmrpl/ziaf095","DOIUrl":"https://doi.org/10.1093/jbmrpl/ziaf095","url":null,"abstract":"<p><p>[This corrects the article DOI: 10.1093/jbmrpl/ziad002.].</p>","PeriodicalId":14611,"journal":{"name":"JBMR Plus","volume":"9 6","pages":"ziaf095"},"PeriodicalIF":3.4,"publicationDate":"2025-05-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12123522/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144199146","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":"Reduced orthodontic tooth movement in <i>Ank</i> knockout mice.","authors":"Marta Rizk, Emily Yin Chu, Rogerio Bastos Craveiro, Merve Elmas, Sihem Brenji, Christian Niederau, Nikolaus Marx, Brian Lee Foster, Martha Joan Somerman, Michael Wolf","doi":"10.1093/jbmrpl/ziaf064","DOIUrl":"10.1093/jbmrpl/ziaf064","url":null,"abstract":"<p><p>Hypercementosis has been previously reported in mice lacking progressive ankylosis protein (<i>Ank</i> KO mice, or <i>Ank, KO - knockout, WT - wildtype</i>) due to decreased levels of the mineralization inhibitor inorganic pyrophosphate. However, the impact of hypercementosis on alveolar bone remodeling and periodontal ligament (PDL) maintenance from orthodontic forces during orthodontic tooth movement (OTM) remains unclear. To investigate the roles of ANK protein on tooth movement, PDL maintenance, alveolar bone remodeling, and tooth root resorption, we performed a split-mouth model of OTM induced by a closed-coil spring stretched between the maxillary first molar and maxillary incisors in <i>Ank</i> KO and WT mice (including both males and females). Micro-computed tomographic analysis revealed a 36.6% reduction in OTM in <i>Ank</i> KO mice compared with WT mice, although OTM-induced thickening of PDL was found to be similar in both groups. While reduced tissue mineral density (TMD) of the alveolar bone was observed in WT mice, TMD in <i>Ank</i> KO mice was maintained. Loss of <i>Ank</i> leads to wider roots with thicker cementum on the untreated, contralateral side, whereas a significant increase in OTM-induced root resorption was observed on the lateral tension side. Histologic analysis of root resorption confirmed these data and showed increased resorption lacunae located prevalently in the OTM tooth root cementum of <i>Ank</i> KO mice. Using a quantitative PCR array of bone-associated markers to interrogate total RNA harvested from PDL tissues along the root surface, we found alterations in gene expression from OTM in both WT and <i>Ank</i> KO mice, which included genes involved in bone remodeling, calciotropic hormones and receptors, cytokines, growth factors, and receptors. Our findings advance the understanding of the role of <i>Ank</i> in regulating mineralization in the periodontium as well as factors involved in root resorption.</p>","PeriodicalId":14611,"journal":{"name":"JBMR Plus","volume":"9 6","pages":"ziaf064"},"PeriodicalIF":3.4,"publicationDate":"2025-04-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12087959/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144101108","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":"Expression of the aryl hydrocarbon receptor in Osterix-lineage cells regulates adult skeletal homeostasis in a compartment-specific manner.","authors":"Jennifer Dorn, Dima W Alhamad, Husam Bensreti, Christopher L Yearwood, Tate J Allen, Michaela Cushing, Joseph C Shaver, Colby Gross, William C Whichard, Caihong Dai, Kanglun Yu, Roger Zhong, Marion A Cooley, Maribeth H Johnson, Wendy B Bollag, Sadanand Fulzele, Carlos M Isales, Mark W Hamrick, William D Hill, Meghan E McGee-Lawrence","doi":"10.1093/jbmrpl/ziaf067","DOIUrl":"10.1093/jbmrpl/ziaf067","url":null,"abstract":"<p><p>Kynurenine (KYN), a tryptophan metabolite that increases with age, impairs osteoblast function. The aryl hydrocarbon receptor (AhR) has been proposed to mediate KYN's actions in bone. To test whether deletion of AhR in osteoblasts is beneficial for bone, we established an adult-onset AhR conditional knockout (CKO) model using Osx-Cre and examined the effects of AhR CKO at 4.5 and 6 mo of age (representing ~6 and 12 wk of CKO). While BMSC-derived osteoblasts from WT mice demonstrated reduced matrix formation from KYN treatment, AhR CKO osteoblasts were unaffected by KYN. Kynurenine's harmful effects were most pronounced in the middle of an osteoblastic differentiation time course, and these effects could be rescued via the AhR antagonist BAY2416964. In vivo, AhR deletion in Osx-expressing cells promoted sex- and compartment-specific skeletal phenotypes. Trabecular bone was increased in the distal femur of male and female AhR CKO mice at both 4.5 and 6 mo of age, potentially driven by a net decrease in the ratio of trabecular osteoclasts to osteoblasts despite a reduction in mineral apposition rate at 6 mo of age. In contrast, cortical bone phenotypes induced by AhR deletion depended on age and sex. In males, cortical bone volume fraction (Ct.BV/TV) was elevated in AhR CKO mice vs WT littermates at 4.5 mo of age, but differences resolved by 6 mo of age. In contrast, cortical bone was reduced in female AhR CKO as compared to WT littermates at 6 mo of age. These results underscore the complexity of AhR signaling in skeletal biology that must be considered while exploring AhR as a therapeutic target for conditions like osteoporosis and musculoskeletal frailty. Future studies will be needed to test the effects of osteoblastic AhR deletion at advanced ages, when the endogenous AhR ligand KYN is elevated in the circulation and skeletal niche.</p>","PeriodicalId":14611,"journal":{"name":"JBMR Plus","volume":"9 6","pages":"ziaf067"},"PeriodicalIF":3.4,"publicationDate":"2025-04-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12105101/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144150492","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":"How does bone recover in patients with tumor-induced osteomalacia? Long-term follow-up in a national cohort study.","authors":"María Belén Zanchetta, Fernando Jerkovich, Florencia Scioscia, Yamile Mocarbel, Analía Pignatta, Natalia Elías, Juan Manuel Roganovich, Carlos Vigovich, María Celeste Balonga, Ana Carolina Cohen, Giselle Mumbach, José Luis Mansur, Carolina Fux Otta, Walter Guillermo Douthat, Pilar Tartaglia, Griselda Cecchi, María Bastianello, Luisa Plantalech, Erich Fradinger, José Rubén Zanchetta","doi":"10.1093/jbmrpl/ziaf041","DOIUrl":"https://doi.org/10.1093/jbmrpl/ziaf041","url":null,"abstract":"<p><p>Tumor-induced osteomalacia (TIO) is a rare disorder characterized by impaired bone mineralization due to phosphate wasting. Long-term changes in BMD and microarchitecture after surgical cure or medical therapy in TIO are not well understood. This study describes changes in BMD, microarchitecture, and bone strength in patients with TIO following surgical cure or medical therapy. A prospective cohort study included adults diagnosed with TIO from May 2018 to 2024, categorized into those with surgical cure and those on medical therapy. Follow-up assessments were classified as early (median 8 mo), intermediate (median 17 mo), and long-term (median 26 mo). Fifteen patients were included: seven achieved surgical cure, and eight remained on medical therapy. Lumbar spine BMD increased by +19% at early, +27% at intermediate, and +15% at long-term follow-up. Total hip BMD increased by +31%, +36%, and +31% at early, intermediate, and long-term assessments, respectively. All patients achieved a normal lumbar spine BMD, while 91% attained a normal total hip BMD. At the distal tibia, substantial increases in bone microarchitecture parameters-cortical area (Ct.Ar), cortical volumetric density (Ct.vBMD), and cortical thickness (Ct.Th)-were observed. Notably, Ct.Th improved to levels comparable to healthy controls. Bone strength improved by 13% but was not statistically significant, probably due to the small sample size. At the distal radius, most parameters remained stable. Patients with surgical cure showed more rapid and substantial improvements in BMD and cortical microarchitecture than non-cured patients, but these differences did not reach statistical significance. Overall, bone recovery in TIO is gradual, with gains in spine and hip BMD and significant improvements in tibial cortical parameters. However, some aspects of bone microarchitecture remained below control levels, underscoring the need for ongoing monitoring and individualized management strategies.</p>","PeriodicalId":14611,"journal":{"name":"JBMR Plus","volume":"9 5","pages":"ziaf041"},"PeriodicalIF":3.4,"publicationDate":"2025-04-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12010155/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144001245","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":"Real-world differences in denosumab persistence, reinitiation, and switching among cohorts of older adults in Canada and the United States.","authors":"Kaleen N Hayes, Selvam R Sendhil, Sulbh Aggarwal, Andrew R Zullo, Sarah D Berry, Arman Oganisian, Michael Adegboye, Suzanne M Cadarette","doi":"10.1093/jbmrpl/ziaf061","DOIUrl":"10.1093/jbmrpl/ziaf061","url":null,"abstract":"<p><p>Denosumab is an injectable osteoporosis medication administered twice per year. Discontinuation of denosumab can result in rapid rebound fractures, but the evidence is limited on real-world persistence with denosumab. We conducted 2 parallel, population-based cohort studies leveraging (1) healthcare administrative data from Ontario, Canada (ON; 100% population) and (2) a 20% random sample of US Medicare beneficiaries (US). The first denosumab claim (US: 1/2010-12/2019; ON: 1/2012-12/2021) was identified using pharmacy claims (ON) and Medicare Parts D and B claims (US). Patients aged <66 yr, residing in long-term care (LTC), or with implausible data (eg, death before first claim) were excluded. We developed and applied an algorithm that used dosing and days between dispensations to clean denosumab claims. We assumed a days supply of 183 d for each dispensation and defined discontinuation as a 60-d gap in coverage. We estimated initial persistence, reinitiation, and switching to other osteoporosis medications using Kaplan-Meier estimators, censoring on death, disenrollment (US only), LTC admission, or study end (12/31/2022 [ON], 12/31/2020 [US]). We also estimated the monthly proportion of patients with an on-time denosumab dose to explore time trends. We identified 168 339 eligible individuals in ON (mean age = 78 yr; 90% female) and 97 595 in the US (mean age = 77 yr; 90% female). In ON, the median time to denosumab discontinuation was longer (median 2.3 yr [ON] vs 1.7 yr [US]; 3-yr persistence: 44% [ON] vs 31% [US]), and time to reinitiation was shorter (median = 0.5 yr [ON] vs 1.9 yr [US]). In both populations, around 10% switched to another osteoporosis medication. Women and those with prior oral bisphosphonate use had longer durations of denosumab treatment in ON but not in the US. The proportion persisting with on-time doses did not increase over time in the US or ON. Research to improve persistence with denosumab and optimize post-denosumab treatment is critical.</p>","PeriodicalId":14611,"journal":{"name":"JBMR Plus","volume":"9 6","pages":"ziaf061"},"PeriodicalIF":3.4,"publicationDate":"2025-04-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12087952/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144101885","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":"Osseous morphology differences in the foot and ankle associated with Charcot-Marie-Tooth disease.","authors":"Melissa R Requist, Andrew C Peterson, Timothy C Beals, Bopha Chrea, Amy L Lenz","doi":"10.1093/jbmrpl/ziaf058","DOIUrl":"10.1093/jbmrpl/ziaf058","url":null,"abstract":"<p><p>Charcot-Marie-Tooth (CMT) disease is a genetic, progressive peripheral nerve disease that commonly manifests in a cavovarus foot deformity. Previously, this foot deformity has been believed to be an alignment change in the foot, but recent research has shown that there are bone morphology differences in individuals with CMT. Differences in bone morphology have been identified in the calcaneus, talus, and medial cuneiform, but have not been consistently analyzed throughout the foot or studied in relation to different genetic subtypes of CMT. This study is a retrospective, cross-sectional analysis of bone morphology in CMT using weight-bearing computed tomography and statistical shape modeling. This analysis identified bone morphology differences between CMT and control groups throughout the hindfoot, midfoot, and forefoot. Bone morphology differences were also present between the 2 primary disease subtypes throughout the foot. Key morphologic findings include the altered shape of the subtalar articular surfaces on the talus, bending of the metatarsals, variation in navicular process morphology, and differences between subtypes in the talus, medial cuneiform, and medial metatarsals. There are several possible theoretical mechanisms for this osseous deformation, including bone remodeling in response to altered loading from alignment change or from decreased musculotendinous forces, but the patterns of morphological variation seen in these data cannot be fully explained by these mechanisms, suggesting that there may be an interaction between the neuronal disease and bone remodeling. Future work is necessary to characterize the progression of bony deformity throughout development and to correlate bone shape with function, gait, muscle morphology and strength to elucidate the mechanism of osseous morphology change in varying subtypes of CMT.</p>","PeriodicalId":14611,"journal":{"name":"JBMR Plus","volume":"9 6","pages":"ziaf058"},"PeriodicalIF":3.4,"publicationDate":"2025-04-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12087961/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144101881","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":"Concerns about the paper, \"Benefits of targeted vibration for bone strength and bone density in postmenopausal women with osteopenia: a randomized, sham-controlled trial\".","authors":"Douglas P Kiel, Theresa A Guise, Maya Styner, Janet Rubin","doi":"10.1093/jbmrpl/ziaf051","DOIUrl":"https://doi.org/10.1093/jbmrpl/ziaf051","url":null,"abstract":"","PeriodicalId":14611,"journal":{"name":"JBMR Plus","volume":"9 5","pages":"ziaf051"},"PeriodicalIF":3.4,"publicationDate":"2025-04-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12035689/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143996447","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":"AGN1 local osteo-enhancement procedure increases proximal femur volumetric bone mineral density of women with post-menopausal osteoporosis as assessed by quantitative computed tomography analysis.","authors":"Michelle Chin, Ronald Hill, Bryan Huber, James Howe, Klaus Engelke","doi":"10.1093/jbmrpl/ziaf036","DOIUrl":"https://doi.org/10.1093/jbmrpl/ziaf036","url":null,"abstract":"<p><p>In this study, QCT was used to analyze the AGN1 Local Osteo-Enhancement Procedure (LOEP) as a treatment to form bone in the proximal femurs of patients with osteoporosis. Using this minimally invasive procedure, a resorbable triphasic AGN1 implant material was injected into the left femurs of 12 women with post-menopausal osteoporosis. Computed tomography scans were taken before treatment (baseline) and at 12 wk, 24 wk, and 5-7 yr after treatment. Quantitative computed tomography was used to investigate the resorption of AGN1 within the treated proximal femurs and to analyze the treatment's impact on integral, trabecular, and cortical bone. The untreated right femurs were used as controls. Data illustrated an increase in trabecular volumetric BMD (trab vBMD) of treated hips at all timepoints (baseline: 22 ± 21 mg/cm³ vs 217 ± 56 mg/cm³, 161 ± 18 mg/cm³, and 121 ± 37 mg/cm³ at 12-wk, 24-wk, and 5- to 7-yr timepoints, respectively), and an increase in integral vBMD of 65% at the 12-wk timepoint and 34% at the 5- to 7-yr timepoint. The increase in trab vBMD was observed in the location where the AGN1 implant material bolus was injected, and at the 5- to 7-yr timepoint, no significant BMD change was observed in the trabecular regions surrounding the original implantation zone (treated: 32 ± 16 mg/cm³, control: 31 ± 16 mg/cm³). This QCT study provides a more detailed understanding of the resorption and transformation of the AGN1 implant material into bone and supports, with some limitations, that the AGN1 LOEP treatment can locally increase trabecular bone density in weakened areas of the proximal femur where strength increase is most needed to reduce the risk of hip fragility fracture.</p>","PeriodicalId":14611,"journal":{"name":"JBMR Plus","volume":"9 5","pages":"ziaf036"},"PeriodicalIF":3.4,"publicationDate":"2025-04-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12035697/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144035282","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":"Bone disease and osteoporosis associated with Pompe disease.","authors":"Lucas Maxey, Hannah Freibert, Auremil Quinonez, Hartmut Malluche, Madhumathi Rao","doi":"10.1093/jbmrpl/ziaf045","DOIUrl":"https://doi.org/10.1093/jbmrpl/ziaf045","url":null,"abstract":"<p><p>Pompe disease is a lysosomal storage disorder defined by a mutation in the GAA gene encoding alpha-1,4-glucosidase alpha (acid maltase). Pompe disease encompasses a range of clinical presentations that are broadly characterized as either classic infantile Pompe disease or late-onset Pompe disease (LOPD). LOPD is a milder manifestation of the disease that presents after the first year of life and is typically characterized by mild proximal muscle weakness and lack of cardiac involvement compared to the classic infantile form. The mainstay of treatment is enzyme replacement therapy (EnRT). Decreased bone mineral density (BMD) is frequently encountered in LOPD. While bone loss is thought to be due to mechanical unloading secondary to the progressive muscle weakness associated with the disease, there is a lack of tissue-level data in support of this mechanism. We describe a 60-yr-old female with LOPD managed with EnRT who presented with proximal muscle weakness and decreased BMD on dual-energy X-ray absorptiometry. Undecalcified bone histology showed low turnover osteoporosis, and treatment was initiated with romosozumab. Romosozumab specifically may provide a promising osteoporosis therapy for LOPD-associated osteoporosis. As a sclerostin inhibitor, it both inhibits bone resorption and promotes new bone formation. We additionally emphasize that bone biopsy should be considered as a useful diagnostic tool in the evaluation of osteoporosis associated with uncommon pathologies, since bone histology provides more specific tissue-level information over clinical and laboratory evaluation as well as substantive guidance for treatment.</p>","PeriodicalId":14611,"journal":{"name":"JBMR Plus","volume":"9 5","pages":"ziaf045"},"PeriodicalIF":3.4,"publicationDate":"2025-04-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11994030/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143967481","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":"Response to Letter to the Editor for \"Benefits of targeted vibration for bone strength and bone density in postmenopausal women with osteopenia: a randomized, sham-controlled trial\".","authors":"Laura D Bilek, Michael J Jaasma","doi":"10.1093/jbmrpl/ziaf050","DOIUrl":"https://doi.org/10.1093/jbmrpl/ziaf050","url":null,"abstract":"","PeriodicalId":14611,"journal":{"name":"JBMR Plus","volume":"9 5","pages":"ziaf050"},"PeriodicalIF":3.4,"publicationDate":"2025-04-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12010160/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143965895","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}