Bone Reports最新文献

{"title":"Clinical and functional characterization of COL2A1 p.Gly444Ser variant: From a fetal phenotype to a previously undisclosed postnatal phenotype","authors":"Enrica Marchionni ,&nbsp;Maria Rosaria D'Apice ,&nbsp;Viviana Lupo ,&nbsp;Giovanna Lattanzi ,&nbsp;Elisabetta Mattioli ,&nbsp;Gina Lisignoli ,&nbsp;Elena Gabusi ,&nbsp;Gerardo Pepe ,&nbsp;Manuela Helmer Citterich ,&nbsp;Elena Campione ,&nbsp;Anna Maria Nardone ,&nbsp;Paola Spitalieri ,&nbsp;Noemi Pucci ,&nbsp;Dario Cocciadiferro ,&nbsp;Eliseo Picchi ,&nbsp;Francesco Garaci ,&nbsp;Antonio Novelli ,&nbsp;Giuseppe Novelli","doi":"10.1016/j.bonr.2023.101728","DOIUrl":"https://doi.org/10.1016/j.bonr.2023.101728","url":null,"abstract":"<div><p><em>COL2A1</em> gene encodes the alpha-1 chain of type-II procollagen. Heterozygous pathogenic variants are associated with the broad clinical spectrum of genetic diseases known as type-II collagenopathies. We aimed to characterize the NM_001844.5:c.1330G&gt;A;p.Gly444Ser variant detected in the <em>COL2A1</em> gene through trio-based prenatal exome sequencing in a fetus presenting a severe skeletal phenotype at 31 Gestational Weeks and in his previously undisclosed mild-affected father. Functional studies on father's cutaneous fibroblasts, along with <em>in silico</em> protein modeling and <em>in vitro</em> chondrocytes differentiation, showed intracellular accumulation of collagen-II, its localization in external Golgi vesicles and nuclear morphological alterations. Extracellular matrix showed a disorganized fibronectin network. These results showed that p.Gly444Ser variant alters procollagen molecules processing and the assembly of mature type-II collagen fibrils, according to COL2A1-chain disorganization, displayed by protein modeling. Clinical assessment at 38 y.o., through a reverse-phenotyping approach, revealed limp gait, short and stocky appearance. X-Ray and MRI showed pelvis asymmetry with severe morpho-structural alterations of the femoral heads bilaterally, consistent with a mild form of type-II collagenopathy. This study shows how the fusion of genomics and clinical expertise can drive a diagnosis supported by cellular and bioinformatics studies to effectively establish variants pathogenicity.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":null,"pages":null},"PeriodicalIF":2.5,"publicationDate":"2023-11-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2352187223000748/pdfft?md5=d25a97e41fa8d592e4d1e3014942921d&pid=1-s2.0-S2352187223000748-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"138448702","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":"For older individuals there is greater variance in low mean Bone Material Strength Index values obtained with the OsteoProbe","authors":"Kara B. Anderson ,&nbsp;Pamela Rufus-Membere ,&nbsp;Jacob W. Harland ,&nbsp;Julie A. Pasco ,&nbsp;Adolfo Diez-Perez ,&nbsp;Mark A. Kotowicz ,&nbsp;Kara L. Holloway-Kew","doi":"10.1016/j.bonr.2023.101727","DOIUrl":"10.1016/j.bonr.2023.101727","url":null,"abstract":"<div><h3>Purpose</h3><p>Bone Material Strength Index (BMSi) quantifies the resistance of bone to a specified force in vivo at the mid tibia using impact microindentation (IMI). Anecdotal evidence suggests that within-participant variance in BMSi may be associated with the individual's mean BMSi. This study aimed to investigate associations between mean and variance of IMI measures in a population-based study.</p></div><div><h3>Methods</h3><p>Participants were men (<em>n</em> = 420) and women (<em>n</em> = 55) from the Geelong Osteoporosis Study who underwent BMSi measurement using the OsteoProbe at recent follow-up phases (men 2016–2022; women 2022–2023). Median age was 63.7 yr (IQR 53.0–71.8). BMSi standard deviation was skewed and therefore natural log transformed (referred to as ln-SD). Linear regression models were developed with ln-SD as the dependent variable and mean BMSi as the independent variable adjusting for sex, age, height and weight.</p></div><div><h3>Results</h3><p>In unadjusted models, greater BMSi was associated with lower ln-SD (β = −1.58, <em>p</em> = 0.042). This association was sustained after adjustment (<em>p</em> = 0.013), and an interaction between BMSi and age was observed (<em>p</em> = 0.004). In those aged 63.7 yr and over (median age), mean BMSi was inversely associated with ln-SD (β = −3.22, <em>p</em> = 0.002). Sex was not identified as an effect modifier. In younger participants, no BMSi*ln-SD association was observed.</p></div><div><h3>Conclusion</h3><p>In older men and women, there was greater variance in low BMSi values. This suggests that standard deviation of the BMSi measure may provide additional information in the assessment of bone health and is worthy of further investigation.</p></div><div><h3>Mini abstract</h3><p>In older men and women, greater variance is observed when BMSi values are low, reflecting potential variation in the bone surface.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":null,"pages":null},"PeriodicalIF":2.5,"publicationDate":"2023-11-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2352187223000736/pdfft?md5=328b8b1ccf43cac50640b1aa38bfbf64&pid=1-s2.0-S2352187223000736-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"135763609","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":"Novel approaches to correlate computerized tomography imaging of bone fracture callus to callus structural mechanics","authors":"Yazan Kadkoy ,&nbsp;Sangeeta Abraham ,&nbsp;Peter Michael ,&nbsp;Tasmima Tazin ,&nbsp;Charlene Wetterstrand ,&nbsp;J. Patrick O'Connor","doi":"10.1016/j.bonr.2023.101726","DOIUrl":"https://doi.org/10.1016/j.bonr.2023.101726","url":null,"abstract":"<div><p>Estimating the mechanical properties of bone in vivo without destructive testing would be useful for research and clinical orthopedic applications. Micro-computerized tomography (μCT) imaging can provide quantitative, high-resolution 3D representations of bone morphology and is generally the basis from which bone mechanical properties are non-destructively estimated. The goal of this study was to develop metrics using qualitative and quantitative aspects of bone microarchitecture derived from μCT imaging to estimate the mechanical integrity of bone fracture calluses. Mechanical testing data (peak torque) and μCT image data from 12 rat femur fractures were collected at 4 weeks after fracture. MATLAB was used to analyze the callus μCT imaging data which were then correlated to the empirically determined peak torque of the callus. One metric correlated <em>Z</em>-rays, linear contiguities of voxels running parallel to the neutral axis of the femur and through the fracture callus, to peak torque. Other metrics were based on voxel linkage values (LVs), which is a novel measurement defined by the number of voxels surrounding a given voxel (ranging from 1 to 27) that are all above a specified threshold. Linkage values were utilized to segment the callus and compute healing scores (termed eRUST) based on the modified Radiographic Union Score for Tibial fractures (mRUST). Linkage values were also used to calculate linked bone areas (LBAs). All metrics positively correlated with peak torque, yielding correlations of determination (R²) of 0.863 for eRUST, 0.792 for <em>Z</em>-ray scoring, and 0.764 for a normalized Linked Bone Area metric. These novel metrics appear to be promising approaches for extrapolating fracture callus structural properties from bone microarchitecture using objective analytical methods and without resorting to computationally complex finite element analyses.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":null,"pages":null},"PeriodicalIF":2.5,"publicationDate":"2023-11-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2352187223000724/pdfft?md5=117778e5bbe8a97f91e3e7b2718b8100&pid=1-s2.0-S2352187223000724-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"134652936","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":"Serum levels of bone turnover markers including calculation of Z-scores: Data from a Dutch healthy reference cohort","authors":"Mark Siderius ,&nbsp;Suzanne Arends ,&nbsp;Anneke Muller Kobold ,&nbsp;Lucie Wagenmakers ,&nbsp;Karin Koerts ,&nbsp;Anneke Spoorenberg ,&nbsp;Eveline van der Veer","doi":"10.1016/j.bonr.2023.101724","DOIUrl":"https://doi.org/10.1016/j.bonr.2023.101724","url":null,"abstract":"<div><h3>Introduction</h3><p>Bone turnover markers (BTM) are biochemical compounds reflecting different stages of bone metabolism. Their levels change with age and differ between males and females. This makes clinical interpretation and comparison more difficult. Therefore, our aim was to establish BTM reference values which can be used to calculate <em>Z</em>-scores for use in daily clinical practice.</p></div><div><h3>Methods</h3><p>Serum markers of collagen resorption, bone formation/regulation, collagen formation and bone mineralization (sCTX, OC, PINP and BALP, respectively) were measured in non-fasting volunteers without bone-related abnormalities. Raw data was plotted and gender-specific age cohorts were established with their respective means and standard deviations (SD). <em>Z</em>-scores can be calculated using these reference values to correct for the influence of age and gender on BTM.</p></div><div><h3>Results</h3><p>In total, 856 individuals were included of which 486 (57 %) were female. Individuals were aged between 7 and 70 years. Highest serum levels of BTM were found in childhood and puberty. Peak levels are higher in boys than girls and prevail at later ages. In adults, BTM levels decrease before reaching stable nadir levels. In adults, 10-year reference cohorts with means and SD were provided to calculate <em>Z</em>-scores.</p></div><div><h3>Conclusion</h3><p>With our data, <em>Z</em>-scores of sCTX, OC, PINP and BALP can be calculated using reference categories (for age and gender) of Caucasian healthy volunteers. Clinicians can use BTM <em>Z</em>-scores to determine whether there are changes in bone turnover physiology beyond those expected during aging. BTM Z-scores facilitate harmonization of data interpretation in daily clinical practice and research.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":null,"pages":null},"PeriodicalIF":2.5,"publicationDate":"2023-11-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2352187223000700/pdfft?md5=922ae86d5d117aa83333d8d032174088&pid=1-s2.0-S2352187223000700-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"134652937","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":"Burosumab for the treatment of cutaneous-skeletal hypophosphatemia syndrome","authors":"Lillian Abebe ,&nbsp;Kim Phung ,&nbsp;Marie-Eve Robinson ,&nbsp;Richelle Waldner ,&nbsp;Sasha Carsen ,&nbsp;Kevin Smit ,&nbsp;Andrew Tice ,&nbsp;Joanna Lazier ,&nbsp;Christine Armour ,&nbsp;Marika Page ,&nbsp;Saunya Dover ,&nbsp;Frank Rauch ,&nbsp;Khaldoun Koujok ,&nbsp;Leanne M. Ward","doi":"10.1016/j.bonr.2023.101725","DOIUrl":"10.1016/j.bonr.2023.101725","url":null,"abstract":"<div><p>Cutaneous-skeletal hypophosphatemia syndrome (CSHS) is a rare bone disorder featuring fibroblast growth factor-23 (FGF23)-mediated hypophosphatemic rickets. We report a 2-year, 10-month-old girl with CSHS treated with burosumab, a novel human monoclonal antibody targeting FGF23. This approach was associated with rickets healing, improvement in growth and lower limb deformity, and clinically significant benefit to her functional mobility and motor development. This case report provides evidence for the effective use of FGF23-neutralizing antibody therapy beyond the classic FGF23-mediated disorders of X-linked hypophosphatemia and tumor-induced osteomalacia.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":null,"pages":null},"PeriodicalIF":2.5,"publicationDate":"2023-11-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2352187223000712/pdfft?md5=e68530551553c2e4963f5398bff995df&pid=1-s2.0-S2352187223000712-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"135664947","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"The importance of BMPs and TGF-βs for endochondral bone repair – A longitudinal study in hip arthroplasty patients","authors":"Jean Cassuto ,&nbsp;Agnetha Folestad ,&nbsp;Jan Göthlin ,&nbsp;Henrik Malchau ,&nbsp;Johan Kärrholm","doi":"10.1016/j.bonr.2023.101723","DOIUrl":"https://doi.org/10.1016/j.bonr.2023.101723","url":null,"abstract":"<div><h3>Introduction</h3><p>Osseointegration of hip implants, although a decade-long process, shows striking similarities with the four major phases of endochondral bone repair. In the current study we investigated the spatiotemporal involvement of bone morphogenic proteins (BMPs) and transforming growth factor betas (TGF-βs) throughout the process of bone repair leading to successfully osseointegrated hip implants.</p></div><div><h3>Materials and methods</h3><p>Twenty-four patients that had undergone primary total hip arthroplasty (THA) due to one-sided osteoarthritis (OA) were investigated during a period of 18 years (Y) with repeated measurements of plasma biomarkers as well as clinical and radiological variables. All implants were clinically and radiographically well anchored throughout the follow-up. Eighty-one healthy donors divided in three gender- and age-matched groups and twenty OA patients awaiting THA, served as controls. Plasma was analyzed for BMP-1, -2, -3, -4, -6, -7 -9 and TGF-β1, -β2, -β3 by use of a high-sensitivity and wide dynamic range electrochemiluminescence technique allowing for detection of minor changes.</p></div><div><h3>Results</h3><p>Spatiotemporal changes during the follow-up are presented in the context of the four phases of endochondral bone repair shown in earlier studies and transposed to the current study based on similarities in biomarker responses. Phase 1: <em>Primary proinflammatory phase</em> lasting from surgery until day 7, Phase 2: <em>Chondrogenic phase</em> from day 7 until 18 months postsurgery, Phase 3: <em>Secondary proinflammatory and cartilage remodeling phase</em> lasting from 18 months until 7Y, Phase 4: <em>coupled bone remodeling</em> from 7Y until 18Y postsurgery. BMP-1 increased sharply shortly after surgery and remained significantly above healthy during the chondrocyte recruitment, proliferation, and hypertrophy phases with a subsequent return to control level at 5Y postsurgery. BMP-2 was above healthy controls before surgery and 1 day after surgery before decreasing to control level and remaining there throughout the follow-up. BMP-3 was at control level from presurgery until 6M after surgery when it increased to a peak at 2Y during the cartilage hypertrophy phase followed by a gradual decrease to control level at 10Y during the phase of bone formation. In the following, BMP-3 decreased below controls to a nadir 15Y postsurgery during coupled bone remodeling. BMP-4 was at control level from presurgery until 10Y postsurgery when it increased to a sharp peak at 15Y after surgery followed by a return to the level of healthy at 18Y. BMP-6 did not differ from healthy during the follow-up. BMP-7 was at control level from presurgery until 1Y postsurgery before gradually increasing to a peak at 10Y during the early phase of osteogenesis with a gradual return to control level at 18Y during the phase of coupled bone remodeling. BMP-9 was above OA before surgery followed by a decrease to basal le","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":null,"pages":null},"PeriodicalIF":2.5,"publicationDate":"2023-11-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2352187223000694/pdfft?md5=3c682b8d3d61fed52d8375815f726e3e&pid=1-s2.0-S2352187223000694-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"134831624","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":"Frailty syndrome in women with osteoporosis, should physicians consider screening? A cross-sectional study","authors":"Shiva Rahimipour Anaraki ,&nbsp;Ali Mohammadian ,&nbsp;Samaneh Saghafian Larijani ,&nbsp;Maryam Niksolat ,&nbsp;Vahid Rashedi ,&nbsp;Milad Gholizadeh Mesgarha","doi":"10.1016/j.bonr.2023.101722","DOIUrl":"10.1016/j.bonr.2023.101722","url":null,"abstract":"<div><p>Despite its high prevalence and profound impact, frailty syndrome often goes undiagnosed. The study revealed a significant correlation between osteoporosis and frailty syndrome, with predictive accuracy exceeding 75 %. Given these findings and the existing recommendation for osteoporosis screening in older women, we underscore the importance of concurrently screening osteoporotic women for frailty.</p></div><div><h3>Introduction</h3><p>Frailty syndrome, a prevalent and significant geriatric condition, impacts healthcare costs and quality of life. Previous reviews have associated frailty syndrome with osteoporosis, but original research on this link is limited and has produced conflicting results. This study aims to investigate the relationship between frailty syndrome, osteoporosis, bone mineral densitometry T-score, and other influencing factors.</p></div><div><h3>Methods</h3><p>In this cross-sectional study, post-menopausal women underwent screening for osteoporosis and frailty syndrome using bone mineral densitometry and the Fried phenotype. Exclusion criteria included a history of diseases related to bone loss or medications affecting bone metabolism. Bivariate and multivariable tests were used to examine the correlation between frailty syndrome and various covariates, including the diagnosis of osteoporosis.</p></div><div><h3>Results</h3><p>A total of 272 women aged 60 to 89 years (mean age 68.57 ± 6.22) were evaluated. Osteoporosis was prevalent in 44.9 % of participants, and frailty syndrome was identified in 36.4 %. The regression model identified age, menopausal age, and the diagnosis of osteoporosis as variables significantly and independently associated with frailty syndrome. A T-score lower than −2.5 in the femur neck or lumbar spine exhibited a sensitivity of 86.6 % and specificity of 76.5 % in predicting frailty syndrome.</p></div><div><h3>Conclusion</h3><p>Older adults with osteoporosis face an increased risk of frailty syndrome. Therefore, we recommend that primary care providers screen osteoporotic women for frailty syndrome and, when appropriate, refer this group to geriatric specialists for further evaluation.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":null,"pages":null},"PeriodicalIF":2.5,"publicationDate":"2023-10-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10624968/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"71477816","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":"Evaluation of cortical bone remodeling in canines treated with daily and weekly administrations of teriparatide by establishing AI-driven morphometric analyses and GIS-based spatial mapping","authors":"Marie Hoshi-Numahata ,&nbsp;Aya Takakura ,&nbsp;Atsuko Nakanishi-Kimura ,&nbsp;Haruhisa Watanabe ,&nbsp;Kentaro Takada ,&nbsp;Mai Nishiura ,&nbsp;Yoshiaki Sato ,&nbsp;Ryoko Takao-Kawabata ,&nbsp;Tadahiro Iimura","doi":"10.1016/j.bonr.2023.101720","DOIUrl":"https://doi.org/10.1016/j.bonr.2023.101720","url":null,"abstract":"<div><p>Larger animal models with a well-developed Haversian system, as observed in humans, are ideal to analyze cortical bone remodeling in pharmacological studies of anti-osteoporosis drugs, although they have some limitations in controlling individual variability in size, weight, age, and number. This study aimed to morphometrically analyze cortical bone remodeling focusing on Haversian canals in dogs using four regimens of TPTD with daily and weekly administrations at lower and higher weekly doses (4.9 μg/kg/week and 19.8 μg/kg/week, respectively) for 9 months. A micro-computed tomography-based analysis showed no significant differences among regimen groups. By establishing artificial intelligence (AI)-driven morphometric analyses and geographical information system (GIS)-based spatial mapping of Haversian canals that does not require confocal microscopy but is possible with more commonly used wide field microscopes, we successfully observed significant morphometric distinctions among regimens applied even in dogs. Our analytical results suggested that the daily higher regimen specifically increased the number of eroded pores creating spaces between existing canals, thus stimulating cortical bone remodeling.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":null,"pages":null},"PeriodicalIF":2.5,"publicationDate":"2023-10-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"49855082","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"An unusually aggressive multiple non-ossifying fibroma of the distal tibia and fibula: A case report","authors":"Walid Bouaicha ,&nbsp;Mohamed Jlidi ,&nbsp;Salwa Nechi ,&nbsp;Mouldi Lammouchi ,&nbsp;Siwar Sbaihi ,&nbsp;Selim Daas","doi":"10.1016/j.bonr.2023.101721","DOIUrl":"10.1016/j.bonr.2023.101721","url":null,"abstract":"<div><h3>Background and purpose</h3><p>Non-ossifying fibroma (NOF) is one of the most commonly seen benign bone tumours. Although renowned for their benign behaviour and tendency for spontaneous healing, these tumours can occasionally exhibit an aggressive course. Few published papers have focused on the treatment options of symptomatic NOFs.</p><p>The aim of this case report is to discuss the clinical presentation of a painful and unusually aggressive multiple NOF of the distal tibia in a female adolescent patient.</p></div><div><h3>Case presentation</h3><p>The case of a 17-year-old female patient who was complaining of a painful swollen right lower leg for the past few months. The symptoms became gradually worse, preventing her from sporting activities and becoming more and more debilitating. The patient was diagnosed with a particularly aggressive multiple non-ossifying fibroma of the distal tibia and fibula. She was treated with lesion curettage, bone grafting and external fixation with good clinical and radiological outcomes.</p></div><div><h3>Conclusion</h3><p>Non-ossifying fibroma is a benign lesion that only requires observation in most of the cases. However, symptomatic lesions with aggressive behaviour or complicated with pathologic fracture may warrant surgical intervention.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":null,"pages":null},"PeriodicalIF":2.5,"publicationDate":"2023-10-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/1e/9c/main.PMC10582832.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"49674087","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":"Utility of iliac crest tetracycline-labelled bone biopsy in osteoporosis and metabolic bone disease: An evaluation of 95 cases over a period of 25 years","authors":"Juan M. Colazo ,&nbsp;Julia Quirion ,&nbsp;Anthony D. Judice ,&nbsp;Jennifer Halpern ,&nbsp;Herbert S. Schwartz ,&nbsp;S. Bobo Tanner ,&nbsp;Joshua M. Lawrenz ,&nbsp;Kathryn M. Dahir ,&nbsp;Ginger E. Holt","doi":"10.1016/j.bonr.2023.101715","DOIUrl":"10.1016/j.bonr.2023.101715","url":null,"abstract":"<div><h3>Background</h3><p>Metabolic bone diseases (MBD) are typically diagnosed by non-invasive imaging and clinical biomarkers. However, imaging does not provide structural information, and biomarkers can be transiently affected by many systemic factors. Bone biopsy and pathologic evaluation is the gold standard for diagnosis of MBD, however, it is rarely utilized. We describe our technique for iliac crest tetracycline-labelled bone using a cannulated drill and assess the utility of bone biopsies to provide diagnostic and therapeutic guidance.</p></div><div><h3>Methods</h3><p>In the 25-year period between March 1998 and January 2023, a total of 95 bone biopsies were performed on 94 patients for an osteological indication at Vanderbilt University Medical Center (VUMC). Patient demographics, bone biopsy indications, complications, diagnostic utility, and subsequent therapeutic guidance were retrospectively reviewed and analyzed.</p></div><div><h3>Results</h3><p>The procedure had minimal complications and was well tolerated by patients. This technique provided good quality specimens for pathology, which helped establish a diagnosis and treatment change in most patients. Patients that had biopsy-guided treatment alterations showed significant increases in Dual-Energy X-ray Absorptiometry (DEXA) bone mineral density (BMD) scores post-biopsy and subsequent treatment.</p></div><div><h3>Conclusion</h3><p>Despite scientific and technological progress in non-invasive diagnostic imaging, clinical biomarkers, and procedures for MBD, there remains a small but significant subset of patients who may benefit from inclusion of tetracycline-labelled bone biopsy into the diagnostic and therapeutic picture. Future prospective comparison studies are warranted.</p></div><div><h3>Mini abstract</h3><p>Tetracycline-labelled bone biopsies are under-utilized. Biopsy led to a histological diagnosis and ensuing treatment alteration in most patients with significant increases in bone mineral density. The biopsy procedure used herein provided good specimens with low pain/adverse events. Bone biopsy remains a valuable tool in a small, though significant, subset of patients.</p></div>","PeriodicalId":9043,"journal":{"name":"Bone Reports","volume":null,"pages":null},"PeriodicalIF":2.5,"publicationDate":"2023-09-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10558706/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"41097064","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}