Calcified Tissue International最新文献

{"title":"Correction to: Dual Osteoprotective Actions of the Kefir Peptide KFP-1: Enhancement of Bone Formation and Suppression of Bone Resorption in Cells and Murine Models.","authors":"Min-Che Tung, Gary Ro-Lin Chang, Min-Yu Tu, Hueng-Chuen Fan, Chih-Ching Yen, Abdulkadir Cidem, I-Chien Chen, Chuan-Mu Chen","doi":"10.1007/s00223-026-01589-5","DOIUrl":"10.1007/s00223-026-01589-5","url":null,"abstract":"","PeriodicalId":9601,"journal":{"name":"Calcified Tissue International","volume":"117 1","pages":""},"PeriodicalIF":3.5,"publicationDate":"2026-09-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13538149/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148879057","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Evaluation of the Role of Interleukin 6 in Paget's Disease of Bone.","authors":"Kathryn Berg, Katya Johnson, James F Wilson, Jonathan C Y Tang, Stuart H Ralston","doi":"10.1007/s00223-026-01595-7","DOIUrl":"10.1007/s00223-026-01595-7","url":null,"abstract":"<p><p>Raised levels of serum interleukin 6 (IL-6) have been found in some studies of Paget's disease of bone (PDB), and this cytokine has been suggested to act as a mediator of increased osteoclast activity in the disease. Here weevaluated serum IL-6 in two cohorts of patients with PDB and controls and related serum IL-6 levels in PDB patients to clinical features of the disease. Serum IL-6 was measured by ELISA in 553 individuals with PDB from the PIP study (n =  165), the PRISM study (n = 388) and 533 population-based controls. In the PDB cases, we studied associations between IL-6 and disease extent, pain, previous bisphosphonate therapy, and biochemical markers of bone turnover. Serum IL-6 was correlated with age (r = 0.256, p < 0.001) and serum creatinine (rho = 0.148, p < 0.001) but values did not differ in males and females. Following adjustment for age and serum creatinine, there was no significant difference in IL-6 between PIP cases and controls (p = 0.268) or PRISM cases and controls (p = 0.399) nor was there a significant correlation between serum IL-6 and number of affected bones in PDB or bodily pain. Serum IL-6 was correlated with total alkaline phosphatase (rho = 0.174, p < 0.001), and PINP (rho = 0.095, p < 0.05) but not with the bone resorption marker CTX. In summary, we found no difference between circulating IL-6 values in PDB and controls but serum IL-6 was related to alkaline phosphatase and PINP. These observations do not support a role for raised circulating IL-6 values as an important driver of osteoclast activity in PDB.</p>","PeriodicalId":9601,"journal":{"name":"Calcified Tissue International","volume":"117 1","pages":""},"PeriodicalIF":3.5,"publicationDate":"2026-09-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13538248/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148879009","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Estimating the Extent of Missed Opportunities for Osteoporosis Treatment Before Hip Fracture Occurrence.","authors":"Gaetano Paride Arcidiacono, Marco Onofrio Torres, Chiara Ceolin, Mattia Giantin, Giovanni Tripepi, Valentina Camozzi, Anna Bertocco, Mor Peleg Falb, Martin Diogo, Francesca Guidolin, Giacomo Voltan, Maria Grazia Rodà, Elisa Pala, Andrea Angelini, Deris Gianni Boemo, Maria Vittoria Nesoti, Elena Campello, Pietro Ruggieri, Maria Fusaro, Paolo Simioni, Giuseppe Sergi, Sandro Giannini, Marina De Rui, Stefania Sella","doi":"10.1007/s00223-026-01594-8","DOIUrl":"10.1007/s00223-026-01594-8","url":null,"abstract":"<p><p>Hip fractures (HF) are among the most serious consequences of osteoporosis. Many individuals who subsequently sustain a HF may already fulfill recognized criteria for fracture risk assessment, bone mineral density (BMD) evaluation, or anti-osteoporosis treatment before the event occurs, yet remain unidentified and untreated. We aimed to quantify these missed opportunities for osteoporosis management prior to fragility HF occurrence. We performed a cross-sectional analysis of 1,103 patients aged ≥ 50 years hospitalized for fragility HF and evaluated within a Fracture Liaison Service. Pre-fracture fracture risk was retrospectively estimated using FRAX^® based on clinical risk factors only, excluding the index HF. Eligibility for anti-osteoporosis treatment and BMD assessment was evaluated according to international recommendations and national reimbursement criteria. Only 82 patients (7.4%) had received anti-osteoporosis therapy before the index HF. Female sex, rheumatologic diseases, chronic glucocorticoid therapy, and previous fractures were independently associated with prior treatment, whereas diabetes mellitus was negatively associated. Among patients aged 50-90 years, 69.6% would have been classified as being at high or very high fracture risk before fracture occurrence, yet only 10.2% had received anti-osteoporosis therapy. Among the remaining patients, 90.3% already met indications for BMD assessment. Most patients who sustain a HF already meet recognized criteria for osteoporosis assessment or treatment but remain untreated. These findings highlight substantial missed opportunities for fracture prevention and support earlier identification and management of individuals at increased fracture risk.</p>","PeriodicalId":9601,"journal":{"name":"Calcified Tissue International","volume":"117 1","pages":""},"PeriodicalIF":3.5,"publicationDate":"2026-08-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13525934/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148856966","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Improvement of Bone Healing by Combined Treatment with Selective Androgen and Estrogen Receptor Modulators in Female and Male Rats.","authors":"Marina Komrakova, Fabian Nicolai Brunnert, Christian Anders, Kai Oliver Böker, Arndt Friedrich Schilling, Paul Jonathan Roch, Daniel Bernd Hoffmann, Wolfgang Lehmann, Stephan Sehmisch","doi":"10.1007/s00223-026-01585-9","DOIUrl":"10.1007/s00223-026-01585-9","url":null,"abstract":"<p><p>With age, both men and women experience a decline of sex-hormones and impaired bone repair. This study examined whether bone healing could be improved by combined treatment with the selective androgen and estrogen receptor modulators ostarine (Ost) and raloxifene (Ral), and compared its efficacy with monotherapy in female and male rat models. Three‑month‑old female (Experiment 1, n = 15/group) and 8‑month‑old male (Experiment 2, n = 15/group) Sprague-Dawley rats were either intact (healthy control) or ovariectomized/orchiectomized (Ovx/Orx). Ovx/Orx rats received daily either no treatment, Ost, Ral, or combined Ost + Ral at the respective monotherapy doses. Eight weeks after Ovx or 12 weeks after Orx, rats underwent bilateral metaphyseal osteotomy of the tibia with plate fixation. Healing was assessed five in females or 6 weeks in males later by micro‑CT, biomechanics, and histology. Increased total, callus, and cortical densities as well as bone volume fraction were observed in the Ost + Ral group, accompanied by reduced callus volume and surface area, indicating advanced healing. The effect of Ost + Ral was stronger than that of Ral, whereas Ost alone did not alter bone parameters. Biomechanical properties were improved in the Ral and Ost + Ral groups relative to Ovx/Orx controls. Uterus weight was lowest in Ovx and Ovx + Ral groups and enhanced by Ost and Ost + Ral. Prostate weight was lowest in Orx and Orx + Ral rats and increased by Ost. The combination with Ral reduced the androgenic effect of Ost on prostate. Combined treatment may represent a promising approach for osteoporotic bone repair in both sexes, though potential side effects of Ost require consideration.</p>","PeriodicalId":9601,"journal":{"name":"Calcified Tissue International","volume":"117 1","pages":""},"PeriodicalIF":3.5,"publicationDate":"2026-08-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13506596/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148817571","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Paraspeckle-Mediated Integration of Nuclear RNA Regulation in Skeletal Homeostasis and Pathological Disorder.","authors":"Liangliang Wei, Yan Zhang, Dageng Huang","doi":"10.1007/s00223-026-01593-9","DOIUrl":"https://doi.org/10.1007/s00223-026-01593-9","url":null,"abstract":"<p><p>Paraspeckles are NEAT1_2-dependent nuclear ribonucleoprotein condensates that regulate gene expression through RNA retention, protein sequestration, transcriptional control, and RNA processing. This review systematically summarizes paraspeckle organization and examines the roles of NEAT1 isoforms and paraspeckle-associated proteins in diverse skeletal diseases. By integrating evidence from osteoporosis, osteoarthritis, osteosarcoma, and intervertebral disc degeneration, we outline how paraspeckle-related pathways influence skeletal cell differentiation, stress responses, and disease progression. The strongest direct evidence comes from osteoblasts, in which mechanically induced NEAT1_2-dependent paraspeckles retain Smurf1 mRNA in the nucleus, reduce SMURF1 translation, stabilize RUNX2, and promote osteoblast function. In other skeletal contexts, however, many studies have examined total NEAT1 without directly verifying intact paraspeckle function. By distinguishing these levels of evidence, this review clarifies current mechanistic understanding and highlights the therapeutic potential of paraspeckle-related pathways in skeletal disorders.</p>","PeriodicalId":9601,"journal":{"name":"Calcified Tissue International","volume":"117 1","pages":""},"PeriodicalIF":3.5,"publicationDate":"2026-08-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148808325","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Effects of Dapagliflozin on the Bone of Patients with CKD on Dialysis.","authors":"Lauter E Pelepenko, Joaquim Barreto, Marília P Martins, Pedro B Chiteculo, Jessica M Camargo, Mariana C de Oliveira, Eric A Corrêa, Mauro Pascoa, Gil Guerra-Junior, Wagner Vasques Dominguez, Cinthia E M Carbonara, Loïc Louvet, Rosa M A Moyses, Makoto Kuro-O, Andrei C Sposito, Rodrigo Bueno de Oliveira","doi":"10.1007/s00223-026-01587-7","DOIUrl":"10.1007/s00223-026-01587-7","url":null,"abstract":"<p><p>The effects of gliflozins on bones of CKD patients on dialysis are unknown. Recently, SGLT2 expression in bone tissue of patients with CKD has been reported. We hypothesized that dapagliflozin may act in bone cells and modulate the Klotho-FGF23 axis. This study analyzed the effects of dapagliflozin on serum bone biomarkers and related outcomes. In this predefined post-hoc analysis of a previous RCT, patients on dialysis received (1:1) dapagliflozin 10 mg daily or standard care for 24 weeks. The primary endpoint was the change from baseline in serum Klotho and FGF23 levels, compared by ranked analysis of covariance, adjusted by baseline. Exploratory analyses evaluated calcium, phosphate, PTH, bone-ALP, TRAP-5b, DKK1, sclerostin, and bone proteins. Bone fractures, osteopenia, and osteoporosis were outcomes of interest. Seventy-nine patients were included in this analysis (Control N=40 and Dapagliflozin N=39). At baseline, no significant differences in biomarkers were observed. The prevalence of osteopenia and osteoporosis was 73% and 43% (p = 0.60 and p = 1.00), respectively. After 24 weeks, the between-group analysis of the change from baseline (delta), adjusted by baseline, revealed a significant difference in serum Klotho [19.1 (- 38.9-86.3) pg/mL, Control group; - 29.1 (- 94.1-43.9) pg/mL, Dapagliflozin group (F:4.946, p = 0.03)], but not on FGF23 levels, nor other biomarkers. The follow-up prevalences of osteopenia and osteoporosis were 70% and 48% (p = 0.27 and p = 0.61, respectively). No bone fractures were observed. Dapagliflozin use over 24 weeks in patients on dialysis was independently associated with lowered serum Klotho levels, without effects in other bone biomarkers or related outcomes.</p>","PeriodicalId":9601,"journal":{"name":"Calcified Tissue International","volume":"117 1","pages":""},"PeriodicalIF":3.5,"publicationDate":"2026-08-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13473233/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148760875","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Spatial Regulation of Preameloblast Development in Response to Distal Mouse Incisor Injury.","authors":"Alexia Campbell, Kevin Lin, Jake Ngu, Cierra Rose Ahlstrand, Terumi Kohwi-Shigematsu, Yan Zhang","doi":"10.1007/s00223-026-01571-1","DOIUrl":"https://doi.org/10.1007/s00223-026-01571-1","url":null,"abstract":"<p><p>Enamel, the outermost mineralized tissue of the tooth, is produced by specialized dental epithelial cells called ameloblasts. Unlike human enamel, which lacks regenerative capacity, the mouse incisor grows throughout life, driven by adult stem cells residing in the labial cervical loop (LaCl). To maintain tissue homeostasis, dental epithelial stem cells produce transit-amplifying cells (TACs) that commit to preameloblasts (PABs), migrate distally, and differentiate into enamel-forming ameloblasts. The full dental epithelial differentiation trajectory coexists within a single mouse incisor, making it an accessible model for studying adult tissue repair and regeneration. We have shown that the genome organizer SATB1 is enriched in PABs and is required for their differentiation into ameloblasts. Here, we investigated the injury response of PABs following mouse incisor tip trimming. Injured wild-type (wt) incisors exhibited an expanded PAB zone with intensive proliferation, reduced SATB1 in the ameloblast lineage, associated with a spatial delay in the deposition of the dentin/enamel matrix compared to uninjured controls. Trimming of Satb1 cKO mouse incisor failed to elicit this response, highlighting SATB1's role in PAB's response to injury. Compared with wt controls, injured wt incisors and both Satb1 cKO groups showed increased Ki67 immunoreactivity in LaCl mesenchymal and epithelial compartments, along with reduced Col1a1 expression in PAB microenvironment. In vitro, SATB1-transduced ameloblast lineage cells (ALCs) cultured on increasing concentrations of type I collagen exhibited reduced Ki67 but elevated Amelx/Ambn expression. There findings indicate that SATB1 is required for epithelial TACs to exit the cell cycle and transition toward PABs. Incisor tip injury delays PAB differentiation by stimulating LaCL mesenchymal proliferation and altering ECM remodeling within the PAB niche.</p>","PeriodicalId":9601,"journal":{"name":"Calcified Tissue International","volume":"117 1","pages":""},"PeriodicalIF":3.5,"publicationDate":"2026-08-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13473234/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148765947","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"The Association Between Type 2 Diabetes and Bone Microarchitecture Assessed by HR-pQCT.","authors":"Alexandre Mercier-Guery, Blandine Merle, Maxime Auroux, Feriel Benzema, Elisabeth Sornay-Rendu, Emmanuelle Vignot, Pawel Szulc, Eric Lespessailles, Roland Chapurlat","doi":"10.1007/s00223-026-01590-y","DOIUrl":"https://doi.org/10.1007/s00223-026-01590-y","url":null,"abstract":"<p><p>Type 2 diabetic patients generally have higher BMD but greater fracture risk. Exploring their bone microarchitecture might help to explain their bone fragility. We conducted a cross-sectional matched analysis within three prospective cohorts including 477 men and women. Each patient with T2DM was matched for age, sex and BMI with two controls, yielding 159 cases and 318 controls. We used high-resolution peripheral quantitative computed tomography (HR-pQCT) to characterize cortical and trabecular bone microarchitecture. In predefined subgroup analyses within T2DM, we compared bone parameters according to glucose-lowering treatment, including metformin-containing regimens versus non-metformin oral regimens and metformin versus insulin-treated patients. In men, femoral neck aBMD was higher in T2DM (0.83 ± 0.13 vs. 0.79 ± 0.14 g/cm²; p < 0.05). Bone size was ~ 5% lower in T2DM-at the tibia in women (623.94 ± 92.42 vs. 656.65 ± 107.09 mm²; p < 0.05) and at the radius in men (386.66 ± 66.71 vs. 407.25 ± 62.53 mm²; p < 0.05). Among women, radial cortical area (44.38 ± 9.55 vs. 41.25 ± 9.94 mm²; p < 0.05) and cortical thickness (0.67 ± 0.16 vs. 0.62 ± 0.17 mm; p < 0.05) were higher in T2DM. Metformin use was associated with higher femoral neck/hip T-scores, and improved cortical microarchitecture-lower cortical porosity at the radius and tibia (p < 0.001 and p = 0.012) and greater tibial cortical thickness/area (p = 0.025/0.009)-compared with other oral therapies. T2DM was associated with lower tibial total and trabecular area in women, and lower radial total and trabecular area in men. Differences in bone microarchitecture between T2DM participants and controls were modest, heterogeneous, and site- and sex-specific. Exploratory subgroup analyses suggested that metformin use was associated with a more favorable cortical profile, but these findings require cautious interpretation.</p>","PeriodicalId":9601,"journal":{"name":"Calcified Tissue International","volume":"117 1","pages":""},"PeriodicalIF":3.5,"publicationDate":"2026-08-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148757735","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Optimal Resistance Exercise Strategies for Improving Bone Mineral Density in Middle-Aged and Older Adults: A Network Meta-Analysis Based on Exercise Intensity and Frequency.","authors":"Wenhua Zhang, Jingqi Mei, Xun Li, Keliang Chen, Dianbo Zhang, Jun Liu","doi":"10.1007/s00223-026-01578-8","DOIUrl":"https://doi.org/10.1007/s00223-026-01578-8","url":null,"abstract":"<p><p>This study used a network meta-analysis to evaluate the effects of different resistance training intensities and frequencies on bone mineral density (BMD) in middle-aged and older adults, aiming to identify the optimal exercise strategy for each skeletal site. PubMed, Embase, Cochrane, Web of Science, Google Scholar, and Scopus databases were searched for randomized controlled trials (RCTs) meeting the inclusion criteria. The Cochrane RoB 2 tool was used for literature quality assessment, and Stata 17.0 was used for network meta-analysis. The standardized mean difference (SMD) and 95% confidence interval (CI) were used for effect size pooling. Interventions were ranked using the surface under the cumulative ranking (SUCRA). Thirty-four RCTs (n = 1746 participants) were included. For BMD at the lumbar spine (LS), femoral neck (FN), total hip (TH), trochanter (Troch), and Ward's triangle (WT), moderate-intensity resistance training three times per week (3MI) ranked highest in SUCRA probability, with SUCRA values of 80.3%, 75.2%, 68.2%, 86.0%, and 80.5%, respectively. For total body (TB) BMD, high-intensity resistance exercise twice weekly (2HI) ranked highest in SUCRA probability, with a SUCRA value of 88.9%. Compared with the control group, 3MI significantly increased BMD at the FN (SMD = 0.26, 95% CI 0.06, 0.46) and Troch (SMD = 0.34, 95% CI 0.10, 0.58). Compared with the control group, both 2HI (SMD = 0.49, 95% CI 0.07, 0.91) and 3MI (SMD = 0.30, 95% CI 0.01, 0.59) significantly increased TB BMD. Subgroup analyses showed that exercise frequency, intensity, duration, session length, study population sex, bone health status, and age significantly influenced BMD at different sites.  Based on SUCRA probability rankings, 3MI had the highest-ranking probability for improving BMD at the LS, FN, TH, Troch, and WT, while 2HI had the highest-ranking probability for improving TB BMD. However, no statistically significant differences were observed between interventions at the LS, TH, or WT, and these ranking results should be interpreted with caution.</p>","PeriodicalId":9601,"journal":{"name":"Calcified Tissue International","volume":"117 1","pages":""},"PeriodicalIF":3.5,"publicationDate":"2026-08-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148700797","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Burden of Disease in Pediatric Tumor-Induced Osteomalacia: A Literature Review.","authors":"Salvatore Minisola, Suzanne M Jan de Beur, Thomas O Carpenter, Kathryn M Dahir, María Belén Zanchetta, Leanne Ward, Ben Johnson, Neil Webb, Cody Broadhurst, Victoria Ward, Erik A Imel","doi":"10.1007/s00223-026-01580-0","DOIUrl":"10.1007/s00223-026-01580-0","url":null,"abstract":"<p><p>Tumor-induced osteomalacia (TIO) is an ultra-rare, paraneoplastic syndrome caused by tumors secreting fibroblast growth factor 23 (FGF23). In children, TIO may be mistaken for more common causes of rickets and osteomalacia, including monogenic forms, leading to long diagnostic delays. This review aimed to identify evidence on the diagnostic journey and burden of TIO in pediatric patients. A literature review was conducted to identify publications reporting disease characteristics, investigations, treatments, and clinical outcomes in pediatric patients diagnosed with TIO. In total, 41 studies were included in the review, reporting on 46 pediatric patients. Mean age at presentation was 11.2 years (standard deviation [SD]: 4.6). The majority of individuals (60.9%) were male. The most commonly reported symptoms at presentation were pain (65.2%), weakness (47.8%), and impaired physical function (43.5%). Rickets was reported in 45.7% of patients and fractures in 34.8% of patients. Mean time from onset of symptoms to diagnosis was 4.3 years (SD: 2.6) and mean number of imaging procedures per patient was 5.4 (SD: 2.7). Low serum phosphate concentration for age was reported in 93.5% of patients at presentation. Attempted surgical resection was reported in the majority of patients (89.1%) and was successful in 60.9%. Pediatric-onset TIO is associated with a substantial symptomatic and healthcare burden. Increasing awareness of TIO in children may prevent delays in diagnosis, reduce the need for radiation-conferring tests, and lower morbidity due to effective management. Further research in this area is needed to address the scarcity of data available in pediatric patients.</p>","PeriodicalId":9601,"journal":{"name":"Calcified Tissue International","volume":"117 1","pages":""},"PeriodicalIF":3.5,"publicationDate":"2026-08-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13457281/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148700745","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}