Osteoporosis international : a journal established as result of cooperation between the European Foundation for Osteoporosis and the National Osteoporosis Foundation of the USA最新文献
Kristine E Ensrud, Sheena Patel, Lisa Langsetmo, Peggy M Cawthon, Howard A Fink, John T Schousboe, Deborah Kado, Eric S Orwoll
{"title":"Circumstances of fall-related fracture events among men in late life.","authors":"Kristine E Ensrud, Sheena Patel, Lisa Langsetmo, Peggy M Cawthon, Howard A Fink, John T Schousboe, Deborah Kado, Eric S Orwoll","doi":"10.1007/s00198-026-08265-2","DOIUrl":"https://doi.org/10.1007/s00198-026-08265-2","url":null,"abstract":"<p><p>Among older men, most fractures occur in late life and are fall-related. We followed 1469 late-life (average age 90 years) men for 3 years to characterize the circumstances of 160 incident fall-related fracture events. Most were due to minimal trauma and occurred indoors when the activity level was normal or less.</p><p><strong>Introduction: </strong>Of fractures in older men, most are fall-related and occur in men 80 years and older. To better guide the development of fracture prevention approaches in late-life men, detailed information on fall-related fracture events was collected in a cohort of 1469 active surviving men (mean age 90 years) in the Osteoporotic Fractures in Men study from March 2021 until November 2024.</p><p><strong>Methods: </strong>Participants were contacted every 4 months to identify fractures confirmed by radiographic report. Fractures are classified as fall- or non-fall-related and characterized by the degree of trauma. Participants further queried about where, when and how fractures occurred.</p><p><strong>Results: </strong>During a mean follow-up of 3 years, there were 169 confirmed clinical fracture events not due to malignancy or severe external trauma; of these, 160 (94.7%) were fall-related. Most fall-related events were due to falls from ≤standing height (88.1%), occurred indoors (70.6%), and when self-reported activity level was ≤normal (82.5%). Regular use of a walking aid was commonly reported (56.3%); however, of the interviews reporting regular use, 42% indicated that the aid was not in use at the time the event occurred. Fall-related events resulted in fractures across multiple skeletal locations; all fracture types most often occurred indoors, except upper limb fractures that most often occurred outdoors (p = 0.001).</p><p><strong>Conclusion: </strong>Most clinical fracture events in late-life men are fall-related, due to minimal trauma, occur indoors and when activity level is normal or less. Findings suggest that fracture prevention research in this patient group should prioritize evaluation of interventions that may reduce the risk of indoor falls.</p>","PeriodicalId":520737,"journal":{"name":"Osteoporosis international : a journal established as result of cooperation between the European Foundation for Osteoporosis and the National Osteoporosis Foundation of the USA","volume":" ","pages":""},"PeriodicalIF":5.4,"publicationDate":"2026-10-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149225798","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":"Osteopenia in adolescent idiopathic scoliosis.","authors":"Wenjie Zhong, Kai Cao, Fangqian Wu","doi":"10.1007/s00198-026-08273-2","DOIUrl":"https://doi.org/10.1007/s00198-026-08273-2","url":null,"abstract":"<p><strong>Background: </strong>Adolescent idiopathic scoliosis (AIS) is a common type of scoliosis, which occurs in 10 to 18-year-old adolescents and is often accompanied by osteopenia, but the etiology of osteopenia is still unknown. Exploring the etiology may play an important role in fully understanding the pathogenesis of AIS and drawing new treatment ideas.</p><p><strong>Purpose: </strong>Understanding the etiology of osteopenia in AIS.</p><p><strong>Methods: </strong>This article conducted a comprehensive search across PubMed, Web of Science (WOS), and CNKI for literature concerning osteopenia in adolescent idiopathic scoliosis. The primary search strategy employed \"(adolescent idiopathic scoliosis) AND (osteopenia)\", supplemented by additional keywords encompassing \"bone mineralization disorders\", \"hormones\", \"leptin\", \"calcitonin\", \"vitamin K\", \"treatment\", etc. The review prioritized recent, high-quality literature.</p><p><strong>Results: </strong>This review disclosed the research process of osteopenia in AIS and expounded its possible pathogenesis resulting in osteogenic and bone metabolic abnormalities. Meanwhile, this review summarized some other possible pathogenesis, like gut microbiota, nutritional factors resulting in osteopenia in AIS. Furtherly, the article revealed the relationship between osteopenia and AIS treatment outcome.</p><p><strong>Conclusions: </strong>Osteopenia in AIS patients is caused by multiple factors. During the treatment of AIS, it may be necessary to intervene in osteopenia.</p>","PeriodicalId":520737,"journal":{"name":"Osteoporosis international : a journal established as result of cooperation between the European Foundation for Osteoporosis and the National Osteoporosis Foundation of the USA","volume":" ","pages":""},"PeriodicalIF":5.4,"publicationDate":"2026-10-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149225799","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}
Felix N von Brackel, Mikolaj Bartosik, Oskar Windels, Michael Amling, Ralf Oheim
{"title":"Response to the Letter to the Editor on \"Interpreting romosozumab-associated BMD changes in osteogenesis imperfecta: design, densitometry, and multiplicity\".","authors":"Felix N von Brackel, Mikolaj Bartosik, Oskar Windels, Michael Amling, Ralf Oheim","doi":"10.1007/s00198-026-08260-7","DOIUrl":"https://doi.org/10.1007/s00198-026-08260-7","url":null,"abstract":"<p><p>We respond to methodological questions raised in a \"Letter to the Editor\" regarding our retrospective case series on romosozumab treatment in osteogenesis imperfecta (OI). We clarify that the retrospective study design, the rarity of the disease, the small number of cases, and the exploratory nature of the analyses limit the methodological approaches as already acknowledged in our original manuscript. Thus, the primary concern is an inherent limitation of an exploratory study in a rare disease. Nevertheless, these data are helpful, as they generate new hypotheses given the currently very limited clinical data available.</p>","PeriodicalId":520737,"journal":{"name":"Osteoporosis international : a journal established as result of cooperation between the European Foundation for Osteoporosis and the National Osteoporosis Foundation of the USA","volume":" ","pages":""},"PeriodicalIF":5.4,"publicationDate":"2026-10-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149187682","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}
Alyssa M Tondat, Benoit R Lafleur, Marina Mourtzakis, Andrew C Laing
{"title":"Sex-specific predictive equations for estimating trochanteric soft tissue thickness across body positions and imaging modalities.","authors":"Alyssa M Tondat, Benoit R Lafleur, Marina Mourtzakis, Andrew C Laing","doi":"10.1007/s00198-026-08261-6","DOIUrl":"https://doi.org/10.1007/s00198-026-08261-6","url":null,"abstract":"<p><p>Soft tissue thickness is an important factor in fall-related hip fractures. However, methods used to measure soft tissue thickness are inconsistent. We developed sex-specific equations for estimating soft tissue thickness between body positions and imaging modalities. The equations support across-study comparisons and refinement of sex-specific hip fracture risk prediction tools.</p><p><strong>Introduction: </strong>Trochanteric soft tissue thickness (TSTT), which is the amount of soft tissues overlying the greater trochanter, may improve hip fracture risk assessments by improving estimates of the impact force applied to the hip during a fall. Accordingly, TSTT is an important variable to consider in hip fracture risk assessment. However, measurement of TSTT is inconsistent, with different body positions (supine, side-lying, standing) and imaging modalities (ultrasound, dual-energy X-ray absorptiometry (DXA)) being used.</p><p><strong>Purpose: </strong>The objectives of this study were to develop sex-specific regression equations to allow for estimation of 1) ultrasound-measured TSTT across three common body positions, and 2) ultrasound-based TSTT from DXA-derived TSTT.</p><p><strong>Methods: </strong>Measurements of TSTT were obtained in standing, supine, and side-lying positions using ultrasound, and from a whole-body DXA scan in 45 community-dwelling older adults. Sex-specific linear regression equations were developed for estimating ultrasound-measured TSTT across body positions, and for estimating ultrasound-measured TSTT from DXA-derived TSTT.</p><p><strong>Results: </strong>Linear regression models revealed significant positive relationships between ultrasound-measured TSTT across all body positions and by DXA and ultrasound-measured TSTT in all body positions (r<sup>2</sup> = 0.589-0.927, all p < 0.01). Association strengths were reduced in males compared to females (male: r<sup>2</sup> = 0.589-0.83; female: r<sup>2</sup> = 0.8-0.902).</p><p><strong>Conclusions: </strong>The regression equations from this study can be used to estimate TSTT between body positions and imaging modalities. The data provide valuable tools to support across-study comparisons and further refinement of sex-specific hip fracture risk prediction tools.</p>","PeriodicalId":520737,"journal":{"name":"Osteoporosis international : a journal established as result of cooperation between the European Foundation for Osteoporosis and the National Osteoporosis Foundation of the USA","volume":" ","pages":""},"PeriodicalIF":5.4,"publicationDate":"2026-10-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149187709","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}
Polyzois Makras, Marina Kouveletsou, Ioanna Siska, Dimitrios Kalkanis, Maria P Yavropoulou, Athanasios D Anastasilakis, Socrates Papapoulos
{"title":"Building bone in the jaw with denosumab in adult Langerhans cell histiocytosis.","authors":"Polyzois Makras, Marina Kouveletsou, Ioanna Siska, Dimitrios Kalkanis, Maria P Yavropoulou, Athanasios D Anastasilakis, Socrates Papapoulos","doi":"10.1007/s00198-026-08252-7","DOIUrl":"https://doi.org/10.1007/s00198-026-08252-7","url":null,"abstract":"<p><p>A patient with multisystem Langerhans cell histiocytosis and extensive jaw osteolysis experienced remarkable mandibular bone regeneration following short-term denosumab therapy. This unique observation challenges current concepts of denosumab action in the jaw and provides novel in vivo evidence of structural repair through RANKL inhibition.</p><p><strong>Background: </strong>Denosumab is an established inhibitor of receptor activator of nuclear factor-κB ligand (RANKL) that effectively suppresses osteoclastic bone resorption but has also been associated with medication-related osteonecrosis of the jaw (MRONJ).</p><p><strong>Case presentation: </strong>We report a 41-year-old man with multisystem Langerhans cell histiocytosis (LCH) presenting with extensive osteolytic destruction of the maxilla and mandible following multiple dental extractions, clinically resembling MRONJ. Molecular analysis identified a MAP2K1 mutation, and staging confirmed multisystem disease involving the jaw, clavicle, and lungs. The patient received denosumab (120 mg every 2 months for 6 months), resulting in rapid pain relief, marked suppression of serum C-terminal telopeptide of type I collagen and procollagen type I N-terminal propeptide, and substantial reduction in metabolic activity on FDG-PET/CT. Most remarkably, serial imaging demonstrated extensive new bone formation with restoration of mandibular cortical integrity and alveolar architecture despite profound suppression of bone turnover. This striking regenerative response contrasts with the established association between denosumab and MRONJ and suggests that inhibition of disease-driven RANKL signaling in LCH may permit reversal of severe jaw destruction.</p><p><strong>Conclusion: </strong>These observations provide novel in vivo evidence that denosumab may promote structural repair of LCH-associated jaw lesions and raise important mechanistic questions regarding the role of RANKL in pathological jaw bone remodeling.</p>","PeriodicalId":520737,"journal":{"name":"Osteoporosis international : a journal established as result of cooperation between the European Foundation for Osteoporosis and the National Osteoporosis Foundation of the USA","volume":" ","pages":""},"PeriodicalIF":5.4,"publicationDate":"2026-10-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149187722","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}
Shuquan Zhang, Zhixiang Wang, Ruotong Zhou, Wei Yu, Qianqian Pang, Ruizhi Jiajue, Lijia Cui, Wei Liu, Yan Jiang, Mei Li, Xiaoping Xing, Ou Wang, Yue Chi, Weibo Xia
{"title":"Impact of bisphosphonate drug holidays on bone mineral density and osteoporotic fracture risk: real-world evidence from a retrospective cohort study.","authors":"Shuquan Zhang, Zhixiang Wang, Ruotong Zhou, Wei Yu, Qianqian Pang, Ruizhi Jiajue, Lijia Cui, Wei Liu, Yan Jiang, Mei Li, Xiaoping Xing, Ou Wang, Yue Chi, Weibo Xia","doi":"10.1007/s00198-026-08239-4","DOIUrl":"https://doi.org/10.1007/s00198-026-08239-4","url":null,"abstract":"<p><p>Bisphosphonate drug holidays can reduce rare adverse effects, but their skeletal impact is unclear. In this retrospective cohort study, ALN holiday caused modest BMD loss and BTMs rise without increased fracture risk; effects of ZOL holiday were minimal. This study suggests bisphosphonate drug holidays are safe for selected patients with regular monitoring.</p><p><strong>Purpose: </strong>Bisphosphonate (BP) drug holiday prevents rare adverse effects of BPs in osteoporotic patients, but its effect on bone mineral density (BMD), bone turnover markers (BTMs), and fracture risk is still unclear. This study aimed to investigate BMD/BTM variations and clinical fracture risk for patients undergoing BP drug holidays.</p><p><strong>Methods: </strong>In this single-center retrospective cohort study, patients who received ≥4.5 years of alendronate (ALN) or ≥3 doses of zoledronate (ZOL) were included. BMD at the lumbar spine (LS), femoral neck (FN), and total hip (TH), BTMs, and 3-year fracture incidence were assessed between drug holiday (ALN: n = 71; ZOL: n = 20) and ongoing treatment groups (ALN: n = 59; ZOL: n = 47).</p><p><strong>Results: </strong>Compared with ongoing ALN therapy, initiating drug holiday resulted in moderate declines in BMD at LS (-2.23%, 95% CI: -3.70% to -0.76%), TH (-1.62%, 95% CI: -2.68% to -0.56%), and FN (-1.89%, 95% CI: -3.49% to -0.28%) within 1 year. BTMs all rose significantly over 1 year, yet remained below pretreatment levels for at least 3 years. In contrast, there were no significant differences in BMD (1-year LS: -0.47%, 95% CI: -2.38% to 1.43%, TH: -1.07%, 95% CI: -2.77% to 0.64%) and BTM changes between ZOL groups at 1- and 2-year follow-up. No significant differences in clinical fragility fractures were observed between ALN groups (9.9% vs. 6.8%; HR 1.45) and ZOL groups (5% vs. 6.4%).</p><p><strong>Conclusion: </strong>Our findings support that BP drug holidays are feasible for selected patients with adequate BMD. ZOL conferred more sustained skeletal protection than ALN, particularly at the LS, whereas ALN discontinuation was associated with earlier and more pronounced BMD decline and BTM elevation; annual monitoring of BMD and BTMs appears particularly important during the ALN drug holiday period.</p>","PeriodicalId":520737,"journal":{"name":"Osteoporosis international : a journal established as result of cooperation between the European Foundation for Osteoporosis and the National Osteoporosis Foundation of the USA","volume":" ","pages":""},"PeriodicalIF":5.4,"publicationDate":"2026-10-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149170828","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}
Jinmin Liu, Hongwei Zhan, Zhi Yi, Cong Tian, Bin Geng
{"title":"Metabolic dysfunction-associated steatotic liver disease is associated with higher bone mineral density: results from NHANES.","authors":"Jinmin Liu, Hongwei Zhan, Zhi Yi, Cong Tian, Bin Geng","doi":"10.1007/s00198-026-08226-9","DOIUrl":"https://doi.org/10.1007/s00198-026-08226-9","url":null,"abstract":"<p><p>We used data from the National Health and Nutrition Examination Survey (NHANES) to investigate the association between metabolic dysfunction-associated steatotic liver disease (MASLD) and bone mineral density (BMD) among U.S. adults. Our findings indicated that participants with MASLD had higher BMD than those without MASLD.</p><p><strong>Introduction: </strong>Given the stigma associated with the term \"NAFLD,\" experts participating in a Delphi consensus proposed renaming the condition \"MASLD\". Nevertheless, the association between MASLD and BMD remains unclear. This study aimed to investigate the relationship between MASLD and BMD.</p><p><strong>Methods: </strong>A cross-sectional study was conducted among 6470 participants aged 18 years or older using data from the 2005 to 2010, 2013 to 2014, and 2017 to 2018 cycles of the NHANES. MASLD was defined according to diagnostic criteria established through a Delphi consensus. Multivariable linear regression analyses were performed to evaluate the associations of MASLD with femoral neck and total femur BMD, while logistic regression analysis was used to investigate the association between MASLD and osteoporosis.</p><p><strong>Results: </strong>Among the 6470 participants, the mean age was 53.16 ± 17.27 years, 49.54% were male, and 23.52% met the criteria for MASLD. After multivariable adjustment, participants with MASLD had higher femoral neck BMD (β = 0.0238; 95% CI, 0.0154-0.0321) and total femur BMD (β = 0.0332; 95% CI, 0.0244-0.0419) than those without MASLD. In subgroup analyses, participants with individual cardiometabolic components of MASLD, including BMI ≥ 25 kg/m², diabetes, hypertension, and hyperlipidemia, also exhibited higher femoral BMD after multivariable adjustment. Moreover, multivariable logistic regression analysis indicated that MASLD was significantly associated with lower odds of osteoporosis.</p><p><strong>Conclusions: </strong>Our findings suggest that participants with MASLD have higher BMD compared to those without MASLD.</p>","PeriodicalId":520737,"journal":{"name":"Osteoporosis international : a journal established as result of cooperation between the European Foundation for Osteoporosis and the National Osteoporosis Foundation of the USA","volume":" ","pages":""},"PeriodicalIF":5.4,"publicationDate":"2026-10-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149169724","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}
Michaela Rippl, Sarikia Moewes, Daniel Arian Kraus, Michael Drey, Ralf Schmidmaier, Peyman Hadji
{"title":"Age-related bone mineral density patterns in a large German clinical DXA cohort: device-specific comparisons with international reference populations.","authors":"Michaela Rippl, Sarikia Moewes, Daniel Arian Kraus, Michael Drey, Ralf Schmidmaier, Peyman Hadji","doi":"10.1007/s00198-026-08245-6","DOIUrl":"https://doi.org/10.1007/s00198-026-08245-6","url":null,"abstract":"<p><p>In a large German clinical DXA cohort, comparisons with international reference populations were strongly device-dependent. iDXA values were consistently lower, whereas Prodigy Pro values showed closer but age- and cohort-dependent agreement. The data should not be interpreted as scanner-independent German normative reference values.</p><p><strong>Purpose: </strong>To describe age-related bone mineral density patterns in a German DXA cohort and to compare device-specific femoral neck (FN) and total hip (TH) standardized BMD values with international reference cohorts.</p><p><strong>Methods: </strong>We retrospectively analyzed data from 6,086 women using GE Lunar iDXA or Prodigy Pro systems. Device-related differences were evaluated separately by age-group and measurement site using absolute and percentage differences, Hedges' g, Welch tests, and multiple comparison adjustment. For external comparisons, FN and TH BMD data for women aged 20-79 years were converted to standardized BMD and compared separately by device with NHANES III, the Irish DXA-HIP study, and the Italian DINS study. Hedges' g with 95% confidence intervals, two-sided Welch tests, and Holm-adjusted p values were calculated from summary statistics.</p><p><strong>Results: </strong>Prodigy Pro BMD values were higher than iDXA across most age groups and measurement sites, with moderate effect sizes in younger and middle-aged women and smaller differences at older ages. In external comparisons, iDXA sBMD was lower than all reference cohorts in all 36 comparisons (age/site/reference), remaining significant after Holm adjustment. Prodigy Pro showed smaller and age-dependent differences, with 18 of 36 comparisons remaining Holm-significant. Across the reference cohorts, Prodigy Pro percentage differences ranged from -8.9% to 1.3%, whereas iDXA ranged from -19.0% to -1.8%.</p><p><strong>Conclusion: </strong>Comparisons with international reference cohorts were strongly dependent on the DXA device. The present clinical cohort should not be interpreted as a scanner-independent German normative reference population. Device-stratified results provide a more appropriate description of concordance with international datasets than pooled analyses.</p>","PeriodicalId":520737,"journal":{"name":"Osteoporosis international : a journal established as result of cooperation between the European Foundation for Osteoporosis and the National Osteoporosis Foundation of the USA","volume":" ","pages":""},"PeriodicalIF":5.4,"publicationDate":"2026-10-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149130177","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}
Valentin S Schäfer, Jessica Hinman, Katie Moss, Julia F Charles, Jonathan D Adachi, Andrea J Singer, Vrinda Saraff, Asma Deeb, Fatih Süheyl Ezgü, Fumio Otsuka, Liliana-Georgiana Wegmann, Shona Fang, Carolina Tornero
{"title":"Prevalence of hypophosphatasia in general and selected patient populations: a systematic review.","authors":"Valentin S Schäfer, Jessica Hinman, Katie Moss, Julia F Charles, Jonathan D Adachi, Andrea J Singer, Vrinda Saraff, Asma Deeb, Fatih Süheyl Ezgü, Fumio Otsuka, Liliana-Georgiana Wegmann, Shona Fang, Carolina Tornero","doi":"10.1007/s00198-026-08227-8","DOIUrl":"https://doi.org/10.1007/s00198-026-08227-8","url":null,"abstract":"<p><p>Hypophosphatasia (HPP) has diverse manifestations overlapping other skeletal and rheumatic disorders. This systematic review assessed frequency of HPP in select and general clinical populations with persistently low alkaline phosphatase (ALP) activity. Undiagnosed HPP was frequently identified. Clinicians should consider HPP for patients with persistently low ALP and hallmark manifestations.</p><p><strong>Background: </strong>Diagnosis of hypophosphatasia (HPP) can be missed owing to its rare disease status and heterogeneous manifestations that can overlap with other skeletal or rheumatic disorders. The objective of this systematic review was to assess the frequency of HPP among patients with persistently low alkaline phosphatase (ALP) activity and determine overall HPP prevalence in the general population.</p><p><strong>Methods: </strong>English language articles published from January 1, 2015, to September 29, 2025, were identified in PubMed and Embase and through manual searching. Observational studies conducted in patients of any age from general and select clinical populations (e.g., rheumatology, endocrinology) were included if the frequency of HPP or potential HPP was reported or could be enumerated from results. Risk of bias was assessed based on 4 predefined criteria. HPP prevalence was determined from general clinical population studies with the lowest risk of bias.</p><p><strong>Results: </strong>A total of 710 articles were retrieved; 27 met the inclusion criteria. The frequency of genetically confirmed HPP among patients with persistently low ALP activity in studies with low risk of bias (≥3 predefined criteria) was 10.4-26.2% in general clinical populations, 46.4-56.5% in rheumatology populations, and 57.1-87.5% in osteoporosis populations. The total prevalence of HPP in the general population may be 17.6-36.0 per 100,000 population based on 3 studies meeting all 4 criteria.</p><p><strong>Conclusion: </strong>Undiagnosed patients with HPP were frequently identified in studies of ALP screening, highlighting the importance of screening. Clinicians should consider a diagnosis of HPP for patients with persistently low ALP and hallmark manifestations.</p>","PeriodicalId":520737,"journal":{"name":"Osteoporosis international : a journal established as result of cooperation between the European Foundation for Osteoporosis and the National Osteoporosis Foundation of the USA","volume":" ","pages":""},"PeriodicalIF":5.4,"publicationDate":"2026-10-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149130160","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}
Lütfiye Parlak, Nusret Seher, Funda Levendoğlu, Ezgi A Tezcan, Esra Öz, Mehmet S Durmaz, İlknur A Gezer
{"title":"Does 25(OH)D deficiency affect muscle elasticity, physical function, pain, and quality of life? A shear-wave elastography-based cross-sectional study.","authors":"Lütfiye Parlak, Nusret Seher, Funda Levendoğlu, Ezgi A Tezcan, Esra Öz, Mehmet S Durmaz, İlknur A Gezer","doi":"10.1007/s00198-026-08231-y","DOIUrl":"https://doi.org/10.1007/s00198-026-08231-y","url":null,"abstract":"<p><p>In this cross-sectional study of 80 young adults, 25(OH)D deficiency (< 20 ng/mL) was associated with lower SWE-measured muscle elasticity (FCR, BB, and TA), and reduced fingertip pinch strength. When women and men were evaluated separately, muscle elasticity values were lower in the low 25(OH)D group in both sexes. Muscle elasticity showed strong correlations with 25(OH)D and accurately discriminated deficiency (AUC 0.935-0.957) in this young cohort.</p><p><strong>Background: </strong>Vitamin D is a key biological regulator in maintaining bone mineralization and muscle function by regulating calcium and phosphorus metabolism, promoting muscle protein synthesis, and supporting muscle cell growth and differentiation.</p><p><strong>Aim: </strong>To assess muscle elasticity using shear-wave elastography (SWE) in young adults with low and adequate 25-hydroxyvitamin D [25(OH)D] and explore the diagnostic usefulness of SWE in distinguishing 25(OH)D deficiency.</p><p><strong>Design: </strong>A cross-sectional study.</p><p><strong>Setting: </strong>Physical Medicine and Rehabilitation and Radiology Departments in the Selcuk University Faculty of Medicine Hospital.</p><p><strong>Population: </strong>Eighty participants (18-45 years) were included in two groups regarding 25(OH)D levels.</p><p><strong>Methods: </strong>Participants with serum 25(OH)D levels( < 20 ng/mL and those with ≥ 20 ng/mL) were classified as Groups I(n = 40) and II(n = 40). Muscle elasticity of flexor carpi radialis (FCR), short head of biceps brachii (BB), and tibialis anterior (TA) was measured using SWE (kPa).</p><p><strong>Results: </strong>Group I consisted of 21 women and 19 men, while Group II consisted of 22 women and 18 men. The groups were similar in demographic characteristics, physical activity, and SF-36 scores. FCR-kPa, BB-kPa, and TA-kPa were lower in Group I (p < 0.001). When women and men were evaluated separately, FCR, BB, and TA muscle elasticity values were significantly lower in both sexes in the low 25(OH)D group (all p < 0.001). Fingertip pinch strength was lower in the dominant and non-dominant hands in Group I (p = 0.001; p = 0.023). AUC values for predicting 25(OH)D deficiency were FCR: 0.94 (cut-off 23.5 kPa), BB: 0.957 (26.5 kPa), and TA: 0.935 (23.75 kPa). Serum 25(OH)D showed a strong positive correlation with elasticity measurements (r = 0.870-0.916; p < 0.001).</p><p><strong>Conclusion: </strong>Low 25(OH)D levels were found to be associated with lower elasticity values, as measured by SWE, in FCR, BB, and TA muscles in women and men.</p>","PeriodicalId":520737,"journal":{"name":"Osteoporosis international : a journal established as result of cooperation between the European Foundation for Osteoporosis and the National Osteoporosis Foundation of the USA","volume":" ","pages":""},"PeriodicalIF":5.4,"publicationDate":"2026-10-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149122906","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}