Bone Reports最新文献

{"title":"Trabecular bone score assessed by dual-energy X ray absorption predicts vertebral fractures in HIV infected young adults","authors":"Teresa Mannarino ,&nbsp;Adriana D'Antonio ,&nbsp;Simona Mercinelli ,&nbsp;Maria Falzarano ,&nbsp;Federica Volpicelli ,&nbsp;Ciro Gabriele Mainolfi ,&nbsp;Emanuela Zappulo ,&nbsp;Giovanni Di Filippo ,&nbsp;Maria Rosaria Cotugno ,&nbsp;Ivan Gentile ,&nbsp;Alberto Cuocolo","doi":"10.1016/j.bonr.2024.101797","DOIUrl":"10.1016/j.bonr.2024.101797","url":null,"abstract":"<div><h3>Introduction</h3><p>Bone mineral density (BMD) is reduced in patients with human immunodeficiency virus (HIV) infection. Trabecular bone score (TBS) is an additional feature calculated by dual-energy X ray absorption (DXA) that measures texture inhomogeneity at lumbar spine level, providing an index of bone microarchitecture. However, its clinical value still needs to be fully addressed. Aims of the study were to assess BMD and TBS in a cohort of patients with HIV compared to a population of healthy subjects and to investigate the prognostic value of TBS in HIV infected patients.</p></div><div><h3>Method</h3><p>Bone health was assessed by DXA in 165 patients with HIV infection (120 men, mean age 40 ± 7 years) and in 164 healthy subjects (53 male, mean age 37 ± 10 years). BMD was measured at level of lumbar spine (L1-L4), femoral neck and total hip. TBS was computed from the images of lumbar spine using machine proprietary software.</p></div><div><h3>Results</h3><p>BMD at femoral neck level was similar in HIV infected patients and healthy subjects (<em>p</em> = 0.57), whereas BMD measured in total femur was lower in HIV infected patients compared to healthy subjects (<em>p</em> &lt; 0.05). Although mean BMD in lumbar spine was similar between HIV infected patients and healthy subjects (<em>p</em> = 0.90), mean lumbar TBS was lower in patients with HIV infection compared to healthy subjects (<em>p</em> &lt; 0.05). Age, sex and HIV infection resulted independent predictors of reduced TBS. In HIV infected patients age, sex and protease inhibitor duration resulted independent predictors of reduced TBS. TBS was a significant predictor of vertebral fractures during follow-up (<em>p</em> &lt; 0.05).</p></div><div><h3>Conclusion</h3><p>Patients with HIV infection have a significant reduction of TBS, a texture parameter related to bone microarchitecture that may provide skeletal information that is not captured from the standard BMD measurement.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":"22 ","pages":"Article 101797"},"PeriodicalIF":2.1,"publicationDate":"2024-08-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2352187224000640/pdfft?md5=77cc61c34ab5a365ce5712922ea486e0&pid=1-s2.0-S2352187224000640-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141979017","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 effectiveness and safety of combined calcium 600 mg and cholecalciferol 2000 IU for treating vitamin d deficiency: Results from a nationwide study with focus in osteoporosis","authors":"Rosa Pinto-Bonilla ,&nbsp;José Baeza-Noci ,&nbsp;Clara Casado Blanco ,&nbsp;Guillermo Javier Valls Gumbau ,&nbsp;Rubén Juarez Fernández ,&nbsp;María Pascual-Pastor ,&nbsp;Blanca García Magallón ,&nbsp;Blanca Panero Lamothe ,&nbsp;Carmen Moragues Pastor ,&nbsp;Rafael Izquierdo Aviñó ,&nbsp;Eva García Aguilar ,&nbsp;Paula Saz-Leal ,&nbsp;On behalf of the DOSTEO Research Group","doi":"10.1016/j.bonr.2024.101796","DOIUrl":"10.1016/j.bonr.2024.101796","url":null,"abstract":"<div><h3>Introduction</h3><p>Treatment of calcium (Ca) and vitamin D (VD) deficiency (VDD) is crucial for health, especially in bone conditions, such as low bone mineral density (BMD) and osteoporosis. Despite updates in clinical guideline recommendations, no studies have evaluated the efficacy and safety of administering 2000 IU of cholecalciferol combined with calcium. Thus, the main objective of this study was to evaluate VD levels following treatment with Ca 600 mg/ cholecalciferol 2000 IU in real-life clinical practice.</p></div><div><h3>Methods</h3><p>This multicenter, retrospective, observational study included 302 adult patients receiving Ca 600 mg/D3 2000 IU orodispersible tablets, daily for ≥24 weeks. The primary outcome was 25-hydroxivitamin D [25(OH)D] serum levels following treatment. Key secondary outcomes included changes in serum 25(OH)D levels and other bone metabolism (BM) parameters, safety and tolerability. The protocol was approved by a Research Ethics Committee.</p></div><div><h3>Results</h3><p>285 patients were evaluated (mean age [SD]: 67.4 [12.6] years old; 88.4 % women; basal serum 25(OH)D: 20.0 [8.6] ng/mL); 80.7 % reported previous history of osteoporosis/low BMD (osteopenia) and 37.2 % had received other Ca/VD prior to start study treatment. Median treatment duration was 38.5 weeks [range 24.0–82.4]. Overall, 94.4 % of patients increased serum 25(OH)D following treatment to a mean of 36.3 [11.8] ng/mL (<em>p</em> &lt; 0.001 <em>vs.</em> baseline). Patients with basal VDD, significantly increased serum 25(OH)D to a mean over 30 ng/mL; no significant change found in repleted patients (basal 25(OH)D level ≥ 30 ng/mL). PTH was significantly reduced after treatment, with no clinically relevant effect on serum Ca or phosphate. Three non-serious treatment-emergent adverse events were reported. A post-hoc analysis on osteoporotic patients revealed virtually identical results in this population.</p></div><div><h3>Conclusion</h3><p>Treatment with Ca 600 mg/cholecalciferol 2000 IU for at least 24 weeks is effective and safe, especially in osteoporosis. Patients with VDD significantly increase plasma 25(OH)D to optimal range for bone health, with no clinically relevant changes on other bone metabolism parameters other than reducing secondary hyperparathyroidism. The magnitude of 25(OH)D increase directly correlates with the severity of VDD, with no effect in basally repleted patients.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":"22 ","pages":"Article 101796"},"PeriodicalIF":2.1,"publicationDate":"2024-07-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2352187224000639/pdfft?md5=bdc917db9b4ba637f30ac64242fedcd3&pid=1-s2.0-S2352187224000639-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141853586","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":"Losartan alters osteoblast differentiation and increases bone mass through inhibition of TGFB signalling in vitro and in an OIM mouse model","authors":"Mai Morita ,&nbsp;Fawaz Arshad ,&nbsp;Lewis A. Quayle ,&nbsp;Christopher N. George ,&nbsp;Diane V. Lefley ,&nbsp;Ivo Kalajzic ,&nbsp;Meena Balsubramanian ,&nbsp;Tugba Cebe ,&nbsp;Gwen Reilly ,&nbsp;Nicolas J. Bishop ,&nbsp;Penelope D. Ottewell","doi":"10.1016/j.bonr.2024.101795","DOIUrl":"10.1016/j.bonr.2024.101795","url":null,"abstract":"<div><p>Excessive production of Transforming Growth Factor β (TGFβ) is commonly associated with dominant and recessive forms of OI. Previous reports have indicated that administration of TGFβ-targeted antibodies maybe of potential therapeutic benefit to OI patients. However, direct targeting of TGFβ is likely to cause multiple adverse effects including simulation of autoimmunity. In the current study we use patient-derived normal and OI fibroblasts, osteoblasts and OIM mouse models to determine the effects of Losartan, an angiotensin II receptor type 1 (AT1) antagonist, on TGFβ signalling and bone morphology in OI. In OIM mice bred on a mixed background administration of 0.6 g/L losartan for 4 weeks was associated with a significant reduction in TGFβ from 79.2 g/L in the control to 60.0 ng/ml following losartan (<em>p</em> &lt; 0.05), reduced osteoclast activity as measured by CTX from 275.9 ng/ml in the control to 157.2 ng/ml following 0.6 g/L of losartan (p &lt; 0.05) and increased cortical bone thickness (<em>P</em> &lt; 0.001). Furthermore in OIM mice bred on a C57BL/6 background 0.6 g/L losartan increased trabecular bone volume in the tibiae (<em>P</em> &lt; 0.05) and the vertebrae (<em>P</em> &lt; 0.01), increased cortical bone thickness (<em>P</em> &lt; 0.001) reduced the trabecular pattern factor (<em>P</em> &lt; 0.01 and P &lt; 0.001 for the tibiae and vertebrae respectively), reduced osteoclast (<em>P</em> &lt; 0.05) and osteoblast (P &lt; 0.01) numbers as well as reducing the area of bone covered by these cell types. Interestingly, losartan did not affect immune cells infiltrating into bone, nor did this drug alter TGFβ signalling in normal or OI fibroblasts. Instead, losartan reduced SMAD2 phosphorylation in osteoblasts, inhibiting their ability to differentiate. Our data suggest that losartan may be an effective treatment for the bone-associated dysmorphia displayed in OI whilst minimising potential adverse immune cell-related effects.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":"22 ","pages":"Article 101795"},"PeriodicalIF":2.1,"publicationDate":"2024-07-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2352187224000627/pdfft?md5=5ea325f3ca19b7323c47fed2d8141ed1&pid=1-s2.0-S2352187224000627-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141849885","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":"Vitamin D supplementation and falls in residential aged care: A longitudinal multisite cohort study","authors":"Nasir Wabe ,&nbsp;Isabelle Meulenbroeks ,&nbsp;Desiree C. Firempong ,&nbsp;Magdalena Z. Raban ,&nbsp;Amy D. Nguyen ,&nbsp;Jacqueline T. Close ,&nbsp;Stephen R. Lord ,&nbsp;Johanna I. Westbrook","doi":"10.1016/j.bonr.2024.101791","DOIUrl":"10.1016/j.bonr.2024.101791","url":null,"abstract":"<div><h3>Background</h3><p>Vitamin D is vital for musculoskeletal health, and supplementation may lower risk of falls. Past research in residential aged care (RAC) settings on the effects of vitamin D on falls have reported inconclusive findings, partly due to study design limitations. We utilised a longitudinal study design to assess the association between the use of vitamin D and falls over 36 months in RAC.</p></div><div><h3>Method</h3><p>A longitudinal cohort study was conducted using routinely collected electronic data spanning 9 years from 27 RAC facilities in Sydney, New South Wales, Australia. The study included 4520 permanent residents aged 65 years or older who were admitted for the first time from 1 July 2014 and stayed for a minimum of one month. We identified daily vitamin D usage over 36 months, and measured adherence using the Proportion of Days Covered (PDC) metric. A PDC value of ≥80 % signifies optimal adherence. Primary outcomes were the number of all falls and injurious falls. A rolling time-varying predictor-outcome approach and Generalized Estimating Equations (GEE) were applied to determine the longitudinal link between vitamin D supplement use and subsequent risk of falls.</p></div><div><h3>Results</h3><p>Over two-thirds of residents (67.8 %; <em>n</em> = 3063) received vitamin D supplements during their stay, with a median PDC of 74.8 % among users, and 44.6 % (<em>n</em> = 1365) achieving optimal adherence. Increasing age, osteoporosis or fracture history, and dementia were associated with a greater likelihood of achieving optimal adherence. Crude fall incident rates were 8.05 and 2.92 incidents per 1000 resident days for all falls and injurious falls respectively. After accounting for relevant demographic and clinical factors, no significant links were observed between vitamin D supplement usage and fall outcomes: all falls (Incident Rate Ratio [IRR] 1.01; 95 % CI 1.00–1.02; <em>P</em> = 0.237) and injurious falls (IRR 1.01; 95 % CI 1.00–1.02; <em>P</em> = 0.091).</p></div><div><h3>Conclusion</h3><p>Vitamin D supplementation was not associated with a reduced risk of falls, suggesting it is not an effective intervention for preventing falls in RAC. While clinicians should ensure adequate vitamin D intake for residents' nutritional and bone health, it should not be a standalone falls prevention intervention in RAC populations.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":"22 ","pages":"Article 101791"},"PeriodicalIF":2.1,"publicationDate":"2024-07-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2352187224000585/pdfft?md5=011a8360a1936a146ead5cb414181812&pid=1-s2.0-S2352187224000585-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141950521","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":"Are bone targeted agents still useful in times of immunotherapy? The SAKK 80/19 BTA pilot study","authors":"Michael Mark ,&nbsp;Alfonso Rojas Mora ,&nbsp;Thomas Winder ,&nbsp;Anastasios Stathis ,&nbsp;Andreas Jakob ,&nbsp;Gisela Müller ,&nbsp;Stefanie Hayoz ,&nbsp;Patrick Reimann ,&nbsp;Ulf Petrausch ,&nbsp;Roger von Moos ,&nbsp;the Swiss Group for Clinical Cancer Research (SAKK)","doi":"10.1016/j.bonr.2024.101794","DOIUrl":"10.1016/j.bonr.2024.101794","url":null,"abstract":"<div><h3>Background</h3><p>Patients with bone metastases from solid tumors often have additional treatment with bone targeted agents (BTAs) to avoid symptomatic skeletal events (SSEs) such as clinically significant pathological fracture leading toradiation therapy or surgery to the bone, spinal cord compression, or hypercalcemia. The absolute value of BTA treatment in the era of immunotherapy (IO) is unknown.</p></div><div><h3>Methods</h3><p>Patients with bone metastases treated with immunotherapy within the Alpine Tumor Immunology Registry were compared based on whether they received an additional BTA such as denosumab or zoledronic acid. The primary endpoint was time to first SSE. Continuous data were summarized as median and range, categorical data using frequency counts and percentages. Kaplan-Meier estimates were used to describe and visualize the effect of categorical variables.</p></div><div><h3>Results</h3><p>One hundred and ninety-seven patients with bone metastases and treatment with immunotherapy such as nivolumab (48 %), pembrolizumab (40 %), atezolizumab (12 %), ipilimumab (9 %) and other immunotherapy (5 %) were included. The most frequent tumor types were lung cancer (50 %), malignant melanoma (11 %), renal cell cancer (10 %) and bladder cancer (9 %), respectively. One hundred and twenty-two patients (62 %) received a BTA treatment (91 % denosumab). The median treatment duration of a BTA was 178 days (min: 1 day, max: 2010 days). Out of the 197 patients, 47 (24 %) experienced at least one SSE, 100 (51 %) had bone pain. Ten of the 122 patients (8 %) receiving a BTA developed osteonecrosis of the jaw (ONJ). The percentage of patients without an SSE at fixed time points was higher if treated with a BTA (e.g., at 6 months, 92 % [95 % CI: 84 % - 96 %] versus 88 % [95 % CI: 77 % - 94 %]), but no significant difference in time to first SSE (HR 0.69; 95 % CI 0.34–1.39, log-rank <em>p</em> = 0.29) or time to first bone pain (HR: 0.85; 95 % CI: 0.51–1.43, <em>p</em> = 0.54) between these two groups could be detected. There were differences in OS between patients treated with a BTA and patients not treated with a BTA (HR: 1.46; 95 % CI: 1.01–2.10, <em>p</em> = 0.043).</p></div><div><h3>Conclusion</h3><p>No significant difference in time to first SSE or bone pain was observed between patients who have received a BTA or not when treated with immunotherapy. Based on these retrospective results the indication of BTAs to reduce SSEs in cancer patients under treatment with immunotherapy needs further evaluation.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":"22 ","pages":"Article 101794"},"PeriodicalIF":2.1,"publicationDate":"2024-07-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2352187224000615/pdfft?md5=05a28ca252251e065c67a70caec2e446&pid=1-s2.0-S2352187224000615-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141951432","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 Kielin/chordin-like protein is regulated by BMP-2 in osteoblasts","authors":"Kazuki Toba ,&nbsp;Atsushi Yamada ,&nbsp;Kiyohito Sasa ,&nbsp;Tatsuo Shirota ,&nbsp;Ryutaro Kamijo","doi":"10.1016/j.bonr.2024.101793","DOIUrl":"10.1016/j.bonr.2024.101793","url":null,"abstract":"<div><p>Bone morphogenetic protein (BMP), an osteoinductive factor, is a cytokine that induces osteoblast differentiation and mineralization, and expected to be applicable for hard tissue reconstruction. Kielin/chordin-like protein (Kcp), a member of the family of cysteine-rich proteins, enhances BMP signaling. The present study found that expression of <em>Kcp</em> in osteoblasts was induced by BMP-2 in a concentration- and time-dependent manner. Up-regulation of <em>Kcp</em> by BMP-2 was inhibited by Dorsomorphin, a SMAD signaling inhibitor. The involvement of up-regulation of <em>Kcp</em> by BMP-2 in induction of osteoblast differentiation by BMP-2 was also examined, which showed that suppression of <em>Kcp</em> expression by si <em>Kcp</em> partially inhibited induction of osteoblast differentiation and mineralization by BMP-2. Together, these results suggest that <em>Kcp</em> induced by BMP-2 functions to provide positive feedback for promotion of osteoblastic differentiation and mineralization by BMP-2 in osteoblasts.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":"22 ","pages":"Article 101793"},"PeriodicalIF":2.1,"publicationDate":"2024-07-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2352187224000603/pdfft?md5=0071f0dadb2e48b4153e24bc6b7a6a5b&pid=1-s2.0-S2352187224000603-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141950522","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":"Vasorin-deficient mice display disturbed vitamin D and mineral homeostasis in combination with a low bone mass phenotype","authors":"Marco Eijken ,&nbsp;A. Michaela Krautzberger ,&nbsp;Manuela Scholze-Wittler ,&nbsp;Bianca Boers-Sijmons ,&nbsp;Marijke Koedam ,&nbsp;Barbara Kosiol ,&nbsp;Heinrich Schrewe ,&nbsp;Johannes P. van Leeuwen ,&nbsp;Bram C. van der Eerden","doi":"10.1016/j.bonr.2024.101792","DOIUrl":"10.1016/j.bonr.2024.101792","url":null,"abstract":"<div><p>Vasorin (Vasn) is a pleiotropic molecule involved in various physiological and pathological conditions, including cancer. Vasn has also been detected in bone cells of developing skeletal tissues but no function for Vasn in bone metabolism has been implicated yet. Therefore, this study aimed to investigate if Vasn plays a significant role in bone biology. First, we investigated tissue distribution of <em>Vasn</em> expression, using lacZ knock-in reporter mice. We detected clear Vasn expression in skeletal elements of postnatal mice. In particular, osteocytes and bone forming osteoblasts showed high expression of Vasn, while the bone marrow was devoid of signal. Vasn knockout mice (<em>Vasn</em>^{<em>−/−</em>}) displayed postnatal growth retardation and died after four weeks. MicroCT analysis of femurs from 22- to 25-day-old <em>Vasn</em>^{<em>−/−</em>} <em>mice</em> demonstrated reduced trabecular and cortical bone volume corresponding to a low bone mass phenotype. <em>Ex vivo</em> bone marrow cultures demonstrated that osteoclast differentiation and activity were not affected by <em>Vasn</em> deficiency. However, osteogenesis of <em>Vasn</em>^−/− bone marrow cultures was disturbed, resulting in lower numbers of alkaline phosphate positive colonies, impaired mineralization and lower expression of osteoblast marker genes. In addition to the bone phenotype, these mice developed a vitamin D₃-related phenotype with a strongly reduced circulating 25-hydroxyvitamin D₃ and 1,25-dihydroxyvitamin D3 and urinary loss of vitamin D binding protein. In conclusion, Vasn-deficient mice suffer from severe disturbances in bone metabolism and mineral homeostasis.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":"22 ","pages":"Article 101792"},"PeriodicalIF":2.1,"publicationDate":"2024-07-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2352187224000597/pdfft?md5=7a597eed62295540e98b2527843458a5&pid=1-s2.0-S2352187224000597-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141841019","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 clinical and molecular characterization of a Pakistani family with multicentric osteolysis, nodulosis and arthropathy (MONA) syndrome","authors":"Safeer Ahmad ,&nbsp;Mari Muurinen ,&nbsp;Petra Loid ,&nbsp;Muhammad Zeeshan Ali ,&nbsp;Muhammad Muzammal ,&nbsp;Sana Fatima ,&nbsp;Jabbar Khan ,&nbsp;Muzammil Ahmad Khan ,&nbsp;Outi Mäkitie","doi":"10.1016/j.bonr.2024.101789","DOIUrl":"10.1016/j.bonr.2024.101789","url":null,"abstract":"<div><p>Multicentric osteolysis nodulosis and arthropathy (MONA) is a rare skeletal dysplasia characterized primarily by progressive osteolysis, particularly affecting the carpal and tarsal bones, accompanied by osteoporosis. In addition, it features subcutaneous nodules on the palms and soles, along with the progressive onset of arthropathy, encompassing joint contractures, pain, swelling and stiffness. It is caused by a deficiency of the Matrix Metalloproteinase-2 (MMP2). In the current study we present a comprehensive clinical, radiological, genetic and <em>in silico</em> analysis of MONA in a consanguineous Pakistani family. Clinical and radiological examinations of the three severely affected siblings demonstrated a progressive MONA syndrome with phenotypic variability. The patients presented unusual facial appearance, thickened skin, severe short stature, short hands and feet. Radiographs revealed extensive bone deformities affecting upper and lower arms, legs, vertebrae and hip. Genetic analysis revealed a homozygous missense variant [c.539 A &gt; T p.(Asp180Val)] in the <em>MMP2</em> gene. <em>In silico</em> findings suggested a mutant MMP2 protein with a decreased stability and an altered pattern of interactions. Our findings add to the existing literature on the skeletal phenotype of MONA syndrome, including the specific clinical and radiological patterns observed. Moreover, the study will aid in genetic counseling and accurate diagnosis of families affected by the same disorder within the Pakistani population.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":"22 ","pages":"Article 101789"},"PeriodicalIF":2.1,"publicationDate":"2024-07-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2352187224000561/pdfft?md5=96cda048ba4b842ca9fd3802eb1eae21&pid=1-s2.0-S2352187224000561-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141622340","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":"Combination treatment with whole body vibration and simvastatin improves the early osseointegration in aged rats","authors":"Zheng-Bo Qiao ,&nbsp;Ming-Zhong Gu ,&nbsp;Yu-Wu Wang ,&nbsp;Bin-Bin Ma ,&nbsp;Shan-Shan Pang","doi":"10.1016/j.bonr.2024.101790","DOIUrl":"10.1016/j.bonr.2024.101790","url":null,"abstract":"<div><h3>Background</h3><p>Current research has demonstrated that Simvastatin (SIM) and Whole Body Vibration (WBV) actively contributes to the repair of osteoporotic bones. However, there is still limited knowledge regarding the impact of this combined therapy on osseointegration in elderly individuals. Objective: The objective of this study was to verify the influence of WBV and SIM combination treatment on Titanium implants' fixation strength in aged rats.</p></div><div><h3>Methods</h3><p>Male Sprague-Dawley rats at 24 months old were utilized for this investigation. Titanium rods were surgically inserted into the distal femoral canal on their left side. Subsequently, all animals were randomly assigned to one of four groups: Control group; WBV group; SIM group; and WBV + SIM group. Each group received Saline, Whole Body Vibration, Simvastatin, or a combination of Whole Body Vibration plus Simvastatin treatment until they reached their natural death after 12 weeks. The bilateral femurs and serum samples from these rats were collected for evaluation purposes.</p></div><div><h3>Results</h3><p>Both WBV and SIM treatments exhibited an increase in bone mass, osseointegration, and push-out force compared to the Control group (all, <em>P</em> &lt; 0.05). Additionally, levels of oxidative stress and inflammatory factors decreased with both treatments when compared to the Control group alone (all, <em>P</em> &lt; 0.05). Notably, the WBV + SIM group displayed superior effects on new bone formation, biomechanical strength, BMP2 expression in bone tissue as well as SOD2 expression regulation related to bone repair genes when compared to other groups involved in this study (all, <em>P</em> &lt; 0.05).</p></div><div><h3>Conclusion</h3><p>These findings suggest that combining physiotherapy (WBV) with drug therapy (SIM) proves beneficial for enhancing implant fixation in aged rats.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":"22 ","pages":"Article 101790"},"PeriodicalIF":2.1,"publicationDate":"2024-07-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2352187224000573/pdfft?md5=e7816163a7e80395bf3b74970a44bf72&pid=1-s2.0-S2352187224000573-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141622338","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":"Amyloid-β peptide treatment reverses bone loss in the mandibular condyle via Wnt signalling pathway","authors":"Qiang Chen ,&nbsp;Shaoqin Tu ,&nbsp;Dongxiao Tang ,&nbsp;Jiaming Wei ,&nbsp;Nan Wei ,&nbsp;Hong Ai ,&nbsp;Bu Yang ,&nbsp;Zheng Chen","doi":"10.1016/j.bonr.2024.101788","DOIUrl":"https://doi.org/10.1016/j.bonr.2024.101788","url":null,"abstract":"<div><h3>Objective</h3><p>To explore the effect of amyloid-β peptide (Aβ) on mandibular condyle to develop a new treatment for postmenopausal women with Temporomandibular joint osteoarthritis.</p></div><div><h3>Methods</h3><p>A murine bone loss model was established by ovariectomy. Microstructure parameters of the condyle were measured by microcomputed tomography before and after intraperitoneal injection with Aβ. Flow cytometry, Alizarin red staining, RT-qPCR assays, FITC/PI staining, Oil Red O staining and western blotting were used to evaluate the effect of Aβ on the osteogenic differentiation of mouse bone marrow stromal stem cells (mBMSCs).</p></div><div><h3>Results</h3><p>In vivo, condylar microstructure parameters increased. Serum osteoprotegerin and procollagen type 1 N propeptide increased in a dose-dependent manner after the injection of Aβ, which were opposite the changes observed in c-terminal telopeptides of type I collagen, tumor necrosis factor-α and the high serum level of leptin. In vitro, Aβ promoted calcium nodule formation in the cells. The expression of ALP, Runx2, osteorix and osteocalcin increased significantly. The expression of mRNAs related to the Wnt signaling pathway was significantly upregulated, which could be blocked by DKK1.</p></div><div><h3>Conclusion</h3><p>Aβ can reverse bone loss in the mandibular condyle in ovariectomized mice through promoting the osteogenic differentiation of mBMSCs via the Wnt pathway.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":"22 ","pages":"Article 101788"},"PeriodicalIF":2.1,"publicationDate":"2024-07-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S235218722400055X/pdfft?md5=66ad14a98e70bbc6d64ae7aba01cdd70&pid=1-s2.0-S235218722400055X-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141605921","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}