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Dairy consumption, bone turnover biomarkers, and osteo sono assessment index in Japanese adults: A cross-sectional analysis of data from the Iwaki Health Promotion Project 日本成年人的乳制品消费量、骨转换生物标志物和骨超声评估指数:磐城健康促进项目数据的横断面分析
IF 2.5
Bone Reports Pub Date : 2024-04-29 DOI: 10.1016/j.bonr.2024.101770
Ayatake Nakano , Hiroshi M. Ueno , Daisuke Kawata , Yota Tatara , Yoshinori Tamada , Tatsuya Mikami , Koichi Murashita , Shigeyuki Nakaji , Ken Itoh
{"title":"Dairy consumption, bone turnover biomarkers, and osteo sono assessment index in Japanese adults: A cross-sectional analysis of data from the Iwaki Health Promotion Project","authors":"Ayatake Nakano ,&nbsp;Hiroshi M. Ueno ,&nbsp;Daisuke Kawata ,&nbsp;Yota Tatara ,&nbsp;Yoshinori Tamada ,&nbsp;Tatsuya Mikami ,&nbsp;Koichi Murashita ,&nbsp;Shigeyuki Nakaji ,&nbsp;Ken Itoh","doi":"10.1016/j.bonr.2024.101770","DOIUrl":"https://doi.org/10.1016/j.bonr.2024.101770","url":null,"abstract":"<div><h3>Purpose</h3><p>Dairy foods are nutritional sources of calcium, phosphorus, protein, and other nutrients that improve bone health. However, the effects of dairy consumption on bone biomarkers in the Japanese population remain unclear. This study explored the association between dairy consumption and bone biomarkers in Japanese adults.</p></div><div><h3>Methods</h3><p>This cross-sectional study was conducted as part of the Iwaki Health Promotion Project in Aomori, Japan. In total, 1063 adults were included in the analysis. Bone turnover marker levels were measured in local citizens during their annual medical checkups. The calcaneus osteo sono assessment index (OSI) was calculated using a quantitative ultrasound technique. The dietary intake of foods and nutrients was estimated using a food frequency questionnaire. Linear regression models were established using dairy consumption and bone biomarkers with adjustments. Statistic significance was considered by <em>P</em> &lt; 0.05.</p></div><div><h3>Results</h3><p>In multivariate models, the tartrate-resistant acid phosphatase 5b and parathyroid hormone concentrations were inversely associated with dietary dairy consumption after adjusting for age and sex. The undercarboxylated osteocalcin, a procollagen type I N-terminal peptide to bone alkaline phosphatase ratio, and OSI were the directly associated with dairy consumption in multivariate models with adjustment.</p></div><div><h3>Conclusions</h3><p>Dairy consumption is partially associated with bone turnover biomarkers and OSI in adult Japanese participants. Habitual consumption of dairy foods may contribute to the nutritional supplementation for maintaining bone health, including turnover and structure.</p></div><div><h3>Clinical trial registry number and website where it was obtained</h3><p>The Japanese Clinical Trials Registry (UMIN000040459), <span>https://center6.umin.ac.jp/cgi-open-bin/ctr/ctr_view.cgi?recptno=R000046175</span><svg><path></path></svg>.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":null,"pages":null},"PeriodicalIF":2.5,"publicationDate":"2024-04-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2352187224000378/pdfft?md5=542f65543d0ab872c53d9afb70b8df7b&pid=1-s2.0-S2352187224000378-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140824179","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}
引用次数: 0
Amyloid-β neuropathology induces bone loss in male mice by suppressing bone formation and enhancing bone resorption 淀粉样蛋白-β神经病理学通过抑制骨形成和促进骨吸收诱导雄性小鼠骨质流失
IF 2.5
Bone Reports Pub Date : 2024-04-28 DOI: 10.1016/j.bonr.2024.101771
Younghun Jung , Birol Ay , Sajin M. Cyr , Christina M. Tognoni , Kaitlin Klovdahl , Julia Matthias , Qiuxia Cui , Daniel J. Brooks , Mary L. Bouxsein , Isabel Carreras , Alpaslan Dedeoglu , Murat Bastepe
{"title":"Amyloid-β neuropathology induces bone loss in male mice by suppressing bone formation and enhancing bone resorption","authors":"Younghun Jung ,&nbsp;Birol Ay ,&nbsp;Sajin M. Cyr ,&nbsp;Christina M. Tognoni ,&nbsp;Kaitlin Klovdahl ,&nbsp;Julia Matthias ,&nbsp;Qiuxia Cui ,&nbsp;Daniel J. Brooks ,&nbsp;Mary L. Bouxsein ,&nbsp;Isabel Carreras ,&nbsp;Alpaslan Dedeoglu ,&nbsp;Murat Bastepe","doi":"10.1016/j.bonr.2024.101771","DOIUrl":"https://doi.org/10.1016/j.bonr.2024.101771","url":null,"abstract":"<div><p>Alzheimer's disease (AD) and osteoporosis often coexist in the elderly. Although observational studies suggest an association between these two diseases, the pathophysiologic link between AD and skeletal health has been poorly defined. We examined the skeletal phenotype of 5xFAD mice, an AD model with accelerated neuron-specific amyloid-β accumulation causing full-blown AD phenotype by the age of 8 months. Micro-computed tomography indicated significantly lower trabecular and cortical bone parameters in 8-month-old male, but not female, 5xFAD mice than sex-matched wild-type littermates. Dynamic histomorphometry revealed reduced bone formation and increased bone resorption, and quantitative RT-PCR showed elevated skeletal RANKL gene expression in 5xFAD males. These mice also had diminished body fat percentage with unaltered lean mass, as determined by dual-energy X-ray absorptiometry (DXA), and elevated <em>Ucp1</em> mRNA levels in brown adipose tissue, consistent with increased sympathetic tone, which may contribute to the osteopenia observed in 5xFAD males. Nevertheless, no significant changes could be detected between male 5xFAD and wild-type littermates regarding the serum and skeletal concentrations of norepinephrine. Thus, brain-specific amyloid-β pathology is associated with osteopenia and appears to affect both bone formation and bone resorption. Our findings shed new light on the pathophysiologic link between Alzheimer's disease and osteoporosis.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":null,"pages":null},"PeriodicalIF":2.5,"publicationDate":"2024-04-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S235218722400038X/pdfft?md5=cee78fd4fdd3044d001499bf290c013b&pid=1-s2.0-S235218722400038X-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140824178","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}
引用次数: 0
Long-duration leptin transgene expression in dorsal vagal complex does not alter bone parameters in female Sprague Dawley rats 背迷走神经复合体中长期瘦素转基因表达不会改变雌性 Sprague Dawley 大鼠的骨骼参数
IF 2.5
Bone Reports Pub Date : 2024-04-24 DOI: 10.1016/j.bonr.2024.101769
Russell T. Turner , Adam J. Branscum , Urszula T. Iwaniec
{"title":"Long-duration leptin transgene expression in dorsal vagal complex does not alter bone parameters in female Sprague Dawley rats","authors":"Russell T. Turner ,&nbsp;Adam J. Branscum ,&nbsp;Urszula T. Iwaniec","doi":"10.1016/j.bonr.2024.101769","DOIUrl":"10.1016/j.bonr.2024.101769","url":null,"abstract":"<div><p>The hypothalamus and dorsal vagal complex (DVC) are both important for integration of signals that regulate energy balance. Increased leptin transgene expression in either the hypothalamus or DVC of female rats was shown to decrease white adipose tissue and circulating levels of leptin and adiponectin. However, in contrast to hypothalamus, leptin transgene expression in the DVC had no effect on food intake, circulating insulin, ghrelin and glucose, nor on thermogenic energy expenditure. These findings imply different roles for hypothalamus and DVC in leptin signaling. Leptin signaling is required for normal bone accrual and turnover. Leptin transgene expression in the hypothalamus normalized the skeletal phenotype of leptin-deficient <em>ob</em>/<em>ob</em> mice but had no long-duration (≥10 weeks) effects on the skeleton of leptin-replete rats. The goal of this investigation was to determine the long-duration effects of leptin transgene expression in the DVC on the skeleton of leptin-replete rats. To accomplish this goal, we analyzed bone from three-month-old female rats that were microinjected with recombinant adeno-associated virus encoding either rat leptin (rAAV-Leptin, <em>n</em> = 6) or green fluorescent protein (rAAV-GFP, control, <em>n</em> = 5) gene. Representative bones from the appendicular (femur) and axial (3rd lumbar vertebra) skeleton were evaluated following 10 weeks of treatment. Selectively increasing leptin transgene expression in the DVC had no effect on femur cortical or cancellous bone microarchitecture. Additionally, increasing leptin transgene expression had no effect on vertebral osteoblast-lined or osteoclast-lined bone perimeter or marrow adiposity. Taken together, the findings suggest that activation of leptin receptors in the DVC has minimal specific effects on the skeleton of leptin-replete female rats.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":null,"pages":null},"PeriodicalIF":2.5,"publicationDate":"2024-04-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2352187224000366/pdfft?md5=f2410a7092e4108b0036f33dd6456e03&pid=1-s2.0-S2352187224000366-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140761199","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}
引用次数: 0
Rare coexistence of hypopituitarism with osteogenesis imperfecta – A double-trouble for bone 垂体功能减退症与成骨不全症罕见并存--骨骼的双重难题
IF 2.5
Bone Reports Pub Date : 2024-04-23 DOI: 10.1016/j.bonr.2024.101768
Rajdeep Basu, Soumik Goswami, Nilanjan Sengupta, Arjun Baidya, Sunetra Mondal, Kumar Swapnil, Rajat Deb, Vibhu Ranjan Khare, Joydip Datta
{"title":"Rare coexistence of hypopituitarism with osteogenesis imperfecta – A double-trouble for bone","authors":"Rajdeep Basu,&nbsp;Soumik Goswami,&nbsp;Nilanjan Sengupta,&nbsp;Arjun Baidya,&nbsp;Sunetra Mondal,&nbsp;Kumar Swapnil,&nbsp;Rajat Deb,&nbsp;Vibhu Ranjan Khare,&nbsp;Joydip Datta","doi":"10.1016/j.bonr.2024.101768","DOIUrl":"https://doi.org/10.1016/j.bonr.2024.101768","url":null,"abstract":"<div><p>Osteogenesis imperfecta (OI) commonly involving defects in COL1A1 and COL1A2 is a rare hereditary disease of bone fragility affecting 6–7 per 100,000 population. On the other hand, hypopituitarism is a separate entity that manifests with reduced levels of pituitary hormones. The cooccurrence of these two is seldom reported previously in literature as a deviation from Occam's razor. Here, we reported a case of pathological fracture in a 31-year-old male who had blue sclera and secondary adrenal insufficiency, hypogonadotropic hypogonadism, and growth hormone deficiency along with primary autoimmune hypothyroidism. Diagnosis of OI was suggested by heterozygous missense variant in exon 40 of the COL1A2 gene (chr7: g.94423092G &gt; A; Depth: 99×) that resulted in the amino acid substitution of Serine for Glycine at codon 847. Replacement of glucocorticoid, levothyroxine, and testosterone was started along with antiresorptive therapy for better bone health outcomes.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":null,"pages":null},"PeriodicalIF":2.5,"publicationDate":"2024-04-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2352187224000354/pdfft?md5=e5bfc2857a237593bef2a7c7edf4ebd8&pid=1-s2.0-S2352187224000354-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140644316","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}
引用次数: 0
Do Paralympic athletes suffer from brittle bones? Prevalence and risk factors of low bone mineral density in Paralympic athletes 残奥会运动员是否患有脆骨症?残奥运动员骨质密度低的患病率和风险因素
IF 2.5
Bone Reports Pub Date : 2024-04-18 DOI: 10.1016/j.bonr.2024.101767
Vera C.R. Weijer , Jan-Willem van Dijk , Lotte van Dam , Linn Risvang , Judith Bons , Truls Raastad , Luc J.C. van Loon , Kristin L. Jonvik
{"title":"Do Paralympic athletes suffer from brittle bones? Prevalence and risk factors of low bone mineral density in Paralympic athletes","authors":"Vera C.R. Weijer ,&nbsp;Jan-Willem van Dijk ,&nbsp;Lotte van Dam ,&nbsp;Linn Risvang ,&nbsp;Judith Bons ,&nbsp;Truls Raastad ,&nbsp;Luc J.C. van Loon ,&nbsp;Kristin L. Jonvik","doi":"10.1016/j.bonr.2024.101767","DOIUrl":"https://doi.org/10.1016/j.bonr.2024.101767","url":null,"abstract":"<div><h3>Background</h3><p>Bone health may be a concern in Paralympic athletes, given the presence of multiple risk factors predisposing these athletes to low bone mineral density (BMD). Objective: We aimed to assess the prevalence of low BMD among Paralympic athletes participating in various sport disciplines, and to identify potential risk factors for low BMD.</p></div><div><h3>Methods</h3><p>Seventy Paralympic athletes, of whom 51 % were wheelchair-dependent, were included in this cross-sectional study. BMD of the whole-body, lumbar spine, total hip, and femoral neck were assessed by dual-energy x-ray absorptiometry. Comparisons between groups were conducted by one-way ANOVA, and regression analyses were conducted to identify potential risk factors for low BMD.</p></div><div><h3>Results</h3><p>The prevalence of low BMD (<em>Z</em>-score &lt; −1.0) was highest at femoral neck (34 %), followed by total hip (31 %), whole-body (21 %), and lumbar spine (18 %). Wheelchair-dependent athletes had significantly lower BMD Z-scores compared to the non-wheelchair-dependent athletes at whole-body level (−0.5 ± 1.4 vs 0.2 ± 1.3; <em>P</em> = 0.04), total hip (−1.1 ± 1.2 vs 0.0 ± 1.1; <em>P</em> &lt; 0.01), and femoral neck (−1.0 ± 1.3 vs −0.1 ± 1.2; <em>P</em> &lt; 0.01). At the lumbar spine, low BMD was completely absent in wheelchair basketball and tennis players. Regression analyses identified body mass, wheelchair dependence, and type of sport, as the main risk factors for low BMD.</p></div><div><h3>Conclusions</h3><p>In this cohort of Paralympic athletes, low BMD is mainly present at the hip, and to a lesser extent at the whole-body and lumbar spine. The most prominent risk factors for low BMD in Paralympic athletes are related to mechanical loading patterns, including wheelchair use, the type of sport, and body mass.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":null,"pages":null},"PeriodicalIF":2.5,"publicationDate":"2024-04-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2352187224000342/pdfft?md5=e7b9ed8799d8b76438001500ac5f0f86&pid=1-s2.0-S2352187224000342-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140632821","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}
引用次数: 0
Increase in lumbar spine but not distal radius bone mineral density in adults after pancreas kidney transplantation 胰肾移植后成人腰椎骨矿物质密度增加,但桡骨远端骨矿物质密度没有增加
IF 2.5
Bone Reports Pub Date : 2024-04-18 DOI: 10.1016/j.bonr.2024.101764
Simona Kratochvílová , Klara Maratova , Zdenek Sumnik , Jana Brunová , Zdeněk Hlávka , Peter Girman , František Saudek , Ondrej Soucek
{"title":"Increase in lumbar spine but not distal radius bone mineral density in adults after pancreas kidney transplantation","authors":"Simona Kratochvílová ,&nbsp;Klara Maratova ,&nbsp;Zdenek Sumnik ,&nbsp;Jana Brunová ,&nbsp;Zdeněk Hlávka ,&nbsp;Peter Girman ,&nbsp;František Saudek ,&nbsp;Ondrej Soucek","doi":"10.1016/j.bonr.2024.101764","DOIUrl":"https://doi.org/10.1016/j.bonr.2024.101764","url":null,"abstract":"<div><p>Osteoporosis occurs in every third individual after simultaneous pancreas kidney transplantation (SPKT). Currently used bone measures insufficiently predict their fracture risk. Lumbar spine Trabecular bone score (TBS) and distal radius areal and volumetric bone mineral density (BMD) were monitored for the first time in patients with type 1 diabetes and chronic renal failure after SPKT with steroid-sparing protocol. In 33 subjects (mean age 43.4 ± 9.8 years), dual-energy X-ray absorptiometry and peripheral quantitative computed tomography were performed just after SPKT (baseline) and one and three years later. While TBS <em>Z</em>-scores increased (−1.1 ± 1.2 and −0.3 ± 1.0; p˂0.001, at baseline and year three, respectively), trabecular volumetric BMD <em>Z</em>-scores at distal radius metaphysis did not change during the study (−1.3 ± 1.3 and −1.3 ± 1.0; <em>p</em> = 0.38). Similarly, areal BMD <em>Z</em>-scores increased at lumbar spine, total hip and femoral neck (all <em>p</em> &lt; 0.01), but not at the distal radius. SPKT induced bone measures' improvement at lumbar spine and hip but not at distal radius. Before suggesting changes in current clinical care, predictive value of individual bone measures or its combination for fracture risk assessment remains to be elucidated.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":null,"pages":null},"PeriodicalIF":2.5,"publicationDate":"2024-04-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2352187224000317/pdfft?md5=ea1669cb216b320a782fc48940361f42&pid=1-s2.0-S2352187224000317-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140620738","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}
引用次数: 0
A novel entity of massive multifocal osteolyses in the elderly 一种新型的老年人大面积多灶性骨溶解症
IF 2.5
Bone Reports Pub Date : 2024-04-17 DOI: 10.1016/j.bonr.2024.101765
Patrick Orth , Phillip Rolf Stahl , Wolfgang Tränkenschuh , Daniel Baumhoer , Tim Kehl , Hans-Peter Lehnhof , Günther Schneider , Eckart Meese , Henning Madry , Ulrike Fischer
{"title":"A novel entity of massive multifocal osteolyses in the elderly","authors":"Patrick Orth ,&nbsp;Phillip Rolf Stahl ,&nbsp;Wolfgang Tränkenschuh ,&nbsp;Daniel Baumhoer ,&nbsp;Tim Kehl ,&nbsp;Hans-Peter Lehnhof ,&nbsp;Günther Schneider ,&nbsp;Eckart Meese ,&nbsp;Henning Madry ,&nbsp;Ulrike Fischer","doi":"10.1016/j.bonr.2024.101765","DOIUrl":"https://doi.org/10.1016/j.bonr.2024.101765","url":null,"abstract":"<div><p>Osteolyses are common findings in elderly patients and most frequently represent malignant or locally aggressive bone tumors, infection, inflammatory and endocrine disorders, histiocytoses, and rare diseases such as Gorham-Stout syndrome. We here report on a novel entity of massive multifocal osteolyses in both shoulders, the right hip and left knee joint and the dens of an 83-year-old patient not relatable to any previously known etiopathology of bone disorders. The soft tissue mass is of myxoid stroma with an unspecific granulomatous inflammatory process, aggressively destroying extensive cortical and cancellous bone segments and encroaching on articulating bones in diarthrodial large joints. Radiological, nuclear medical, serological, histological, and immunohistochemical analyses were incapable of further classifying the disease pattern within the existing scheme of pathology. Quantitative polymerase chain reaction and next generation sequencing revealed that mutations are not suggestive of any known hereditary or acquired bone disease. Possible treatment options include radionuclide therapy for pain palliation and percutaneous radiation to arrest bone resorption while surgical treatment is inevitable for pathological fractures. This case study shall increase the awareness of the musculoskeletal community and motivate to collect further information on this rare but mutilating disorder.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":null,"pages":null},"PeriodicalIF":2.5,"publicationDate":"2024-04-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2352187224000329/pdfft?md5=2c37f5fe7d7f57bda56819ad12e905ff&pid=1-s2.0-S2352187224000329-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140617986","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}
引用次数: 0
IFITM5-related (type V) osteogenesis imperfecta with evidence of perinatal involvement: A case report 有围产期受累证据的 IFITM5 相关(V 型)成骨不全症:病例报告
IF 2.5
Bone Reports Pub Date : 2024-04-17 DOI: 10.1016/j.bonr.2024.101766
Valentina Martínez-Montoya , Miguel Angel Fonseca-Sánchez , Gerardo Fabian-Morales , Ramiro Vega-Gamas , Gloria Eugenia Queipo-García , Luis Felipe León-Madero , Luz María Sánchez-Sánchez
{"title":"IFITM5-related (type V) osteogenesis imperfecta with evidence of perinatal involvement: A case report","authors":"Valentina Martínez-Montoya ,&nbsp;Miguel Angel Fonseca-Sánchez ,&nbsp;Gerardo Fabian-Morales ,&nbsp;Ramiro Vega-Gamas ,&nbsp;Gloria Eugenia Queipo-García ,&nbsp;Luis Felipe León-Madero ,&nbsp;Luz María Sánchez-Sánchez","doi":"10.1016/j.bonr.2024.101766","DOIUrl":"https://doi.org/10.1016/j.bonr.2024.101766","url":null,"abstract":"<div><p>Osteogenesis imperfecta (OI) is a rare hereditary disorder characterized by bone fragility and frequent fractures. While most cases are attributed to variations in collagen-coding genes <em>COL1A1</em> and <em>COL1A2</em>, other genes such as <em>IFITM5</em> have also been associated with the disease, accounting for up to 5 % of cases. Here, we report a case of a 3-month-old female with a femur fracture and limb deformity. X-rays revealed evidence of osteopenia and previous fractures in the arms, clavicle, ribs, and left limb, alongside prenatal bone deformities detected by ultrasound. Initial clinical evaluation suggested progressively deforming (Sillence's type III) osteogenesis imperfecta (OI). Molecular testing led to the diagnosis of <em>IFITM5</em>-related OI, identifying the c.-14C&gt;T (rs587776916) variant. Although this variant has been previously reported in patients with <em>IFITM5</em>-related OI, prenatal involvement had not been associated with this variant.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":null,"pages":null},"PeriodicalIF":2.5,"publicationDate":"2024-04-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2352187224000330/pdfft?md5=cd27d189816350292a35c274b2872393&pid=1-s2.0-S2352187224000330-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140617987","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}
引用次数: 0
Chloride/proton antiporters ClC3 and ClC5 support bone formation in mice 氯离子/质子反转运体 ClC3 和 ClC5 支持小鼠骨骼形成
IF 2.5
Bone Reports Pub Date : 2024-04-16 DOI: 10.1016/j.bonr.2024.101763
Irina L. Tourkova , Quitterie C. Larrouture , Silvia Liu , Jianhua Luo , Katherine E. Shipman , Kelechi M. Onwuka , Ora A. Weisz , Vladimir Riazanski , Deborah J. Nelson , Matthew L. MacDonald , Paul H. Schlesinger , Harry C. Blair
{"title":"Chloride/proton antiporters ClC3 and ClC5 support bone formation in mice","authors":"Irina L. Tourkova ,&nbsp;Quitterie C. Larrouture ,&nbsp;Silvia Liu ,&nbsp;Jianhua Luo ,&nbsp;Katherine E. Shipman ,&nbsp;Kelechi M. Onwuka ,&nbsp;Ora A. Weisz ,&nbsp;Vladimir Riazanski ,&nbsp;Deborah J. Nelson ,&nbsp;Matthew L. MacDonald ,&nbsp;Paul H. Schlesinger ,&nbsp;Harry C. Blair","doi":"10.1016/j.bonr.2024.101763","DOIUrl":"https://doi.org/10.1016/j.bonr.2024.101763","url":null,"abstract":"<div><p>Acid transport is required for bone synthesis by osteoblasts. The osteoblast basolateral surface extrudes acid by Na<sup>+</sup>/H<sup>+</sup> exchange, but apical proton uptake is undefined. We found high expression of the Cl<sup>−</sup>/H<sup>+</sup> exchanger ClC3 at the bone apical surface. In mammals ClC3 functions in intracellular vesicular chloride transport, but when we found Cl<sup>−</sup> dependency of H<sup>+</sup> transport in osteoblast membranes, we queried whether ClC3 Cl<sup>−</sup>/H<sup>+</sup> exchange functions in bone formation. We used ClC3 knockout animals, and closely-related ClC5 knockout animals: <em>In vitro</em> studies suggested that both ClC3 and ClC5 might support bone formation. Genotypes were confirmed by total exon sequences. Expression of ClC3, and to a lesser extent of ClC5, at osteoblast apical membranes was demonstrated by fluorescent antibody labeling and electron microscopy with nanometer gold labeling. Animals with ClC3 or ClC5 knockouts were viable. In ClC3 or ClC5 knockouts, bone formation decreased ~40 % by calcein and xylenol orange labeling <em>in vivo</em>. In very sensitive micro-computed tomography, ClC5 knockout reduced bone relative to wild type, consistent with effects of ClC3 knockout, but varied with specific histological parameters. Regrettably, ClC5-ClC3 double knockouts are not viable, suggesting that ClC3 or ClC5 activity are essential to life. We conclude that ClC3 has a direct role in bone formation with overlapping but probably slightly smaller effects of ClC5. The mechanism in mineral formation might include ClC H<sup>+</sup> uptake, in contrast to ClC3 and ClC5 function in cell vesicles or other organs.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":null,"pages":null},"PeriodicalIF":2.5,"publicationDate":"2024-04-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2352187224000305/pdfft?md5=7596d372088c816194ecbc6906b7b353&pid=1-s2.0-S2352187224000305-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140604524","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}
引用次数: 0
Comparison of osteoanabolic agents (teriparatide and romosozumab) with bisphosphonates in prevention of subsequent vertebral fractures in patients treated for osteoporotic vertebral fracture for 12 months: An observational cohort study 比较骨合成代谢药物(特立帕肽和罗莫索单抗)与双磷酸盐类药物在预防骨质疏松性脊椎骨折患者 12 个月后发生脊椎骨折方面的作用:观察性队列研究
IF 2.5
Bone Reports Pub Date : 2024-04-12 DOI: 10.1016/j.bonr.2024.101762
Keishi Maruo , Tomoyuki Kusukawa , Masakazu Toi , Tetsuto Yamaura , Masaru Hatano , Hayato Oishi , Kazuma Nagao , Fumihiro Arizumi , Kazuya Kishima , Norichika Yoshie , Toshiya Tachibana
{"title":"Comparison of osteoanabolic agents (teriparatide and romosozumab) with bisphosphonates in prevention of subsequent vertebral fractures in patients treated for osteoporotic vertebral fracture for 12 months: An observational cohort study","authors":"Keishi Maruo ,&nbsp;Tomoyuki Kusukawa ,&nbsp;Masakazu Toi ,&nbsp;Tetsuto Yamaura ,&nbsp;Masaru Hatano ,&nbsp;Hayato Oishi ,&nbsp;Kazuma Nagao ,&nbsp;Fumihiro Arizumi ,&nbsp;Kazuya Kishima ,&nbsp;Norichika Yoshie ,&nbsp;Toshiya Tachibana","doi":"10.1016/j.bonr.2024.101762","DOIUrl":"https://doi.org/10.1016/j.bonr.2024.101762","url":null,"abstract":"<div><h3>Introduction</h3><p>Domino osteoporotic vertebral fracture (OVF) is as a subsequent fracture that develops within 3 months before the initial OVF heals. There is limited evidence regarding the efficacy of osteoanabolic agents on its treatment. This study evaluated the effects of bisphosphonates and anabolic agents teriparatide and romosozumab on subsequent domino OVF.</p></div><div><h3>Methods</h3><p>This was post hoc analysis of a prospective, multicenter, observational study conducted across 8 hospitals, enrolling 144 patients with conservatively treated OVF, grouped into patients receiving bisphosphonate (BP, <em>n</em> = 55), teriparatide (TPTD, <em>n</em> = 62), and romosozumab (Romo, <em>n</em> = 27). The primary outcome was the incidence of subsequent OVF at 3 and 12 months, whereas the secondary outcomes included the incidence of pseudoarthrosis and progression of vertebral collapse (VC). Pseudoarthrosis was classified as stable or unstable based on vertebral instability.</p></div><div><h3>Results</h3><p>The use of osteoanabolic agents did not reduce the incidence of subsequent OVF at 3 and 12 months. There were no significant differences in the background data or type of conservative treatment among the three groups. However, the TPTD and Romo groups had significantly lower rates of unstable pseudarthrosis (<em>p</em> = 0.03). Additionally, there were no significant differences in VC progression between groups, but it tended to be higher in the BP group than the TPTD and Romo group (<em>p</em> = 0.07).</p></div><div><h3>Conclusion</h3><p>Osteoanabolic agents were beneficial in reducing unstable pseudoarthrosis, but were not more effective than bisphosphonates in the development of subsequent domino OVF. A more comprehensive approach to the treatment of osteoporosis is needed to prevent domino OVFs.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":null,"pages":null},"PeriodicalIF":2.5,"publicationDate":"2024-04-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2352187224000299/pdfft?md5=7fd3d64d75be5559f6de66072120347c&pid=1-s2.0-S2352187224000299-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140554063","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}
引用次数: 0
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