Tumor-induced osteomalacia (TIO) is a rare paraneoplastic syndrome caused by excessive fibroblast growth factor 23 (FGF23) secretion. However, TIO lesions located in the neurocranium (NC) and intracranial (IC) structures remain poorly characterized. To characterize the clinical features of TIO lesions in NC and IC structures and to compare them with those located in the sinuses. This retrospective study included patients diagnosed with TIO who underwent tumor resection at Peking Union Medical College Hospital between January 2016 and December 2023. Patients with lesions in NC and IC structures were defined as the study group, while those with sinus lesions served as the control group. Clinical characteristics, biochemical parameters, and surgical outcomes were systematically analyzed and compared between groups. TIO lesions in NC and IC structures were distributed across the skull base, temporal region, sellar region, frontal lobe, and occipital region. Compared with sinus lesions, patients with NC and IC lesions showed comparable clinical and biochemical profiles but exhibited significantly worse outcomes, including higher rates of non-remission and tumor recurrence. These findings suggest increased difficulty in complete tumor resection and disease control in this subgroup. TIO lesions in NC and IC structures are associated with poorer surgical outcomes and higher recurrence rates despite similar baseline characteristics. These results highlight the need for improved localization strategies and tailored management approaches for this challenging subgroup.

Eldecalcitol is an active vitamin D3 derivative approved for the treatment of osteoporosis in Japan and postmenopausal osteoporosis in China. However, the impact of concomitant calcium supplementation on its safety profile is unclear. This was a prospective, observational, non-interventional post-marketing drug intensive monitoring study in Chinese postmenopausal women with osteoporosis (NCT05433207). Data were collected using an electronic case report form at baseline, before initial eldecalcitol dosing, and during routine clinical visits over a 1-year follow-up period. One thousand patients were enrolled from 29 Chinese clinical sites; 958 received eldecalcitol (853 without/105 with calcium supplementation). The mean age (standard deviation [SD]) of patients was 65.6 (9.0) years and mean (SD) time since osteoporosis diagnosis was 2.7 (3.8) years. Adverse drug reactions (ADRs) affected 39.2

OBJECTIVE:Osteoporosis in men is a common but often neglected health problem. We aim to compare the efficacy and safety of denosumab, zoledronic acid and alendronate in men with osteoporosis. METHODS:In this randomized, comparative, open-label study, 390 men with osteoporosis or osteopenia were included. They were randomized to receive the treatment of denosumab, alendronate, or zoledronic acid for 12 months. The percentage changes in bone mineral density (BMD), trabecular bone score (TBS) and bone turnover biomarkers (BTMs) during the treatment were evaluated. Safety parameters were observed. RESULTS:The baseline characteristics were well balanced among the three groups. After 12 months of treatment, denosumab, alendronate and zoledronic acid significantly increased BMD by 4.83 ± 0.89%, 4.32 ± 0.77%, 5.18 ± 0.73% at lumbar spine, by 2.75 ± 0.51%, 2.50 ± 0.61% and 2.83 ± 0.59% at total hip, TBS was significantly increased by 2.44 ± 0.52%, 2.00 ± 0.64%, 2.29 ± 0.55%, respectively, without significant differences among the three groups. Serum levels of BTMs decreased significantly and similarly in all three groups (all P < .05 vs. baseline). Denosumab and alendronate group had fewer adverse events than zoledronic acid group. Denosumab had similar efficacy in patients with different gonadal functions. In patients previously receiving bone resorption inhibitors, denosumab continued to increase BMD and TBS, but the increments were reduced by approximately 30% in BMD at lumbar spine compared with treatment-naive patients. CONCLUSION:Denosumab, alendronate and zoledronic acid significantly and similarly reduced BTMs, increased BMD and TBS in men with osteoporosis, whether the gonadal function of patients was normal or decreased. Previous anti-bone resorption therapy may partially diminish the efficacy of denosumab.

ContextFibrous Dysplasia/McCune-Albright Syndrome (FD/MAS) is a rare skeletal disorder frequently manifesting in childhood, often leading to progressive bone lesions, pain and functional impairment. Denosumab as a monoclonal antibody targeting RANKL has emerged as a potential therapeutic option, while its safety and efficacy in pediatric population remains poorly defined.ObjectiveInvestigate the efficacy and safety of denosumab in pediatric FD/MAS population.Design12-month single-arm study.SettingSingle center study at Peking Union Medical College Hospital.PatientsFD/MAS patients under 18.InterventionsDenosumab 1mg/kg with a maximum dosage of 60mg every 3 months for 12-month follow-up.Main outcome measuresFD-related bone pain, bone turnover markers, 99mTc-MDP bone scintigraphy, bone mineral density.ResultsIn 5 pediatric FD/MAS patients treated with denosumab, significant clinical improvements were observed, including alleviation of FD-associated bone pain, reductions in bone turnover markers, and regression of FD lesions. Alkaline phosphatase levels dropped to an average of 41.8% of baseline, accompanied by concurrent reductions in C-terminal telopeptide and type 1 N-terminal pro-peptide levels. Adverse events particularly hypercalcemia following treatment cessation occurred in 2 of the 5 patients, indicating a relatively high incidence of rebound hypercalcemia and highlighting the need for careful monitoring and cautious use of denosumab in pediatric patients.ConclusionsDenosumab appears to be effective in pediatric FD/MAS patients, however safety concerns remain. Comprehensive pre-treatment evaluation and close monitoring throughout the treatment course are essential. Further prospective studies and randomized controlled trials are needed to determine optimal dosing strategies and establish long-term safety in this population.

Burosumab has shown benefits in treating X-linked hypophosphatemia (XLH), a rare genetic bone disorder that causes rickets among pediatric patients. However, data in Chinese children with XLH are lacking. Therefore, we evaluated the effectiveness, safety, and pharmacokinetics of burosumab among Chinese children (aged 1-12 yr) with XLH in this open-label, multi-center, single-arm, phase 4 study. Of the 28 patients enrolled (12 male, 16 female), 27 completed the study. All patients received s.c. injections of burosumab at a starting dose of 0.8 mg/kg every 2 wk for 64 wk. The mean ± SD patient age was 5.68 ± 3.07 yr. Burosumab treatment increased the serum phosphorus level by 0.96 ± 0.27 mg/dL from baseline during the burosumab dose cycles (average of week 2, 4, 8, 12, 16, 24, 32, 40, 52, and 64). The improvement from baseline (2.33 ± 0.24 mg/dL) to week 64 (3.29 ± 0.30 mg/dL) was significant (p < .05). The Radiograph Global Impression of Change was significantly improved at week 64 (1.90 ± 0.48; p < .0001 vs baseline), and the percentage predicted 6-min walking test among patients aged ≥5 yr with post-baseline values (n = 16) was significantly increased from 67.32% ± 8.27% at baseline to 71.91% ± 8.25% at week 64 (p < .05). All patients experienced treatment-emergent adverse events; most were mild to moderate. In conclusion, burosumab treatment in Chinese children with XLH corrected serum phosphorus levels and improved clinical parameters with tolerable safety profiles.

Objective: Pseudohypoparathyroidism (PHP) is a rare disorder characterized by hypocalcemia and elevated PTH. Although short stature is a key feature, especially in PHP type 1 (PHP1), growth data in Chinese patients remained limited. Methods: Clinical data, including height, age, and biochemical indices, were retrospectively collected from PHP1 patients at Peking Union Medical College Hospital. Molecular diagnosis was performed using MS-MLPA, Sanger sequencing, and WES. Growth charts for height, weight, and BMI in underage patients were constructed. Results: A total of 92 PHP1 patients (58 males, 34 females), including 32 pseudohypoparathyroidism type 1A (PHP-1A), 49 sporadic pseudohypoparathyroidism type 1B (PHP-S1B), and 11 autosomal dominant pseudohypoparathyroidism type 1B (PHP-AD1B), were recruited. Growth velocity peaked at 12 years in males and 5 years in females, with height plateau at 14 and 13 years, respectively. Adult height was 160.0 cm (SDS −1.04) in males and 155.0 cm (SDS −0.52) in females. PHP-1A patients had significantly shorter adult height than PHP-1B (male: 157.0 ± 6.9 vs 171.9 ± 8.9, P = 0.004; female: 146.8 ± 10.2 vs 156.2 ± 4.9, P = 0.007). PHP-1A showed early-onset weight gain from age 1, persisting into adulthood. Adult Wt-SDS was 1.67 vs 1.55 in males and 2.00 vs 3.10 in females (PHP-1A vs PHP-1B). Weight curves exceeded population P50 in most groups, with no significant differences in adult weight, Wt-SDS, BMI, or obesity prevalence between subtypes. Conclusion: Chinese PHP1 patients show early growth plateau and short stature, more severe in PHP-1A. Obesity varies by subtype and sex, with earlier and more persistent weight gain in PHP-1A. These growth charts may aid clinical management.

This study develops a machine learning model to predict osteoporosis in Chinese postmenopausal women. The model was trained using the largest nationwide cohort and externally validated. It showed AUCs of 0.744–0.798 and demonstrated prognostic value for 5-year fracture risk, supporting population-level screening where DXA resources are limited. Osteoporosis is highly prevalent among Chinese postmenopausal women, yet underdiagnosis remains due to limited DXA availability. We aimed to develop and validate a machine learning–based model for osteoporosis prediction using easily obtainable variables. The model was trained using data from the largest nationwide cohort (China Osteoporosis Prevalence Study, n = 6,574) and externally validated in two independent cohorts: the China Vertebral and Osteoporosis Study (n = 1,758) and the Peking Vertebral Fracture Study baseline (n = 1,439). Five predictors selected via LASSO regression were used to train the prediction model. Model performance was evaluated by area under the curve (AUC) and calibration plot. To evaluate its ability to predict future fractures, the model was evaluated in a 5-year follow-up cohort (n = 795). A web-based calculator was developed for public use. The model showed strong discrimination (AUC: 0.798 in the training cohort; 0.775 in the internal validation cohort; 0.750 and 0.744 in the external validation cohorts) and good calibration across all cohorts. A rule-out threshold (≥ 0.098) demonstrated high sensitivity (94.7

A unified consensus statement on medication-related osteonecrosis of the jaw (MRONJ) has not yet been established among the Asian member countries or regions of the Asian Federation of Osteoporosis Societies (AFOS). This study aimed to develop a consensus on MRONJ in patients with osteoporosis across these countries and regions. In this study, the term “Asia-Pacific” refers specifically to the Asian member countries and regions of AFOS. A structured survey consisting of nine MRONJ-related questions was distributed across 10 countries and regions to assess the level of agreement and summarize regional perspectives. In addition, a manual literature review and voting were conducted to evaluate the current evidence on MRONJ. The key aspects of MRONJ, including definition, staging, diagnosis, pathogenesis, risk factors, management, and prevention, were generally consistent among the AFOS countries and regions. The annual incidence and incidence rate of MRONJ associated with low-dose antiresorptive therapy in patients with osteoporosis ranged from 0.025% to 0.136% and 21 to 283 cases per 100,000 person-years, respectively. However, evidence regarding the benefits of drug discontinuation before dental surgery, such as tooth extraction, remains insufficient. Large-scale, multinational studies across AFOS countries and regions are warranted to determine the incidence of MRONJ better and evaluate the impact of antiresorptive drug discontinuation before dental procedures. These findings may contribute to the development of effective evidence-based strategies for preventing MRONJ in patients with osteoporosis.

Background Insights into basic bone biology and genetic disorders have led to advances in the treatment of osteoporosis. However, new drugs for the treatment of osteoporosis in men remain insufficient. We investigated the efficacy of a novel anti-Dkk1 monoclonal antibody (Dkk1-mAb) and its sequential alendronate treatment in a mouse model of osteoporosis induced by orchiectomy. Methods A specific Dkk1-mAb was generated using the hybridoma technology. A total of 40 male C57BL/6 mice aged 12 weeks underwent orchidectomy or sham surgery. At 8 weeks postoperatively, the orchiectomized mice were randomly divided into 4 groups (8 in each group) to receive treatment of Dkk1-mAb, alendronate, Dkk1-mAb followed by alendronate, or placebo. After 8 weeks of treatment, the mice were euthanized, and bone mineral density (BMD), skeletal mechanical properties, bone histology, bone turnover biomarkers, serum levels of Dkk1 and sclerostin, muscle properties, and activity of the Wnt signaling were evaluated. Results After 8 weeks of treatment, serum Dkk1 levels were lower in Dkk1-mAb and sequential groups than placebo group (P < 0.001, P < 0.05). L1-5 BMD was higher in Dkk1-mAb, alendronate, and sequential groups by 11.9 %, 22.8 %, and 23.1 % than placebo group (P < 0.05 or P < 0.001), with BMD at left femur increased by 12.8 % and 21.0 % in alendronate and sequential groups than placebo group (P < 0.05 or P < 0.001). The sequential group exhibited higher vertebral trabecular volume/total volume (39.9 %), bone surface/total volume (25.8 %), trabecular BMD (4.0 %), and trabecular thickness (13.1 %) than Dkk1-mAb group (P < 0.01 or P < 0.001), and higher cortical BMD, thickness than Dkk1-mAb (2.9 %, P < 0.01; 6.6 %, P < 0.05) and alendronate (2.2 %, 5.7 %, all P < 0.05) groups. All treatment groups showed greater bone strength, cross-sectional area of muscle, and grip strength than placebo group. The expression of Lef1 was significantly increased in bones of Dkk1-mAb and sequential groups. No pathological abnormalities in vital organs were found in all groups, suggesting that Dkk1-mAb had a good safety profile. Conclusions The novel Dkk1-mAb has therapeutic potential in osteoporosis, which can increase BMD and bone strength, improve bone microarchitecture and muscle performance of orchiectomy-induced osteoporotic mice through activating the WNT pathway, and its sequential treatment with alendronate achieves additive benefits. The Translational Potential of this Article This study found that the novel Dkk1-mAb can increase BMD and bone strength, improve bone microarchitecture and muscle performance of orchiectomy-induced osteoporosis mice, and sequential treatment with Dkk1-mAb and alendronate achieves additive benefits. Dkk1-mAb has translational potential for the treatment of osteoporosis.

Background:Pseudoachondroplasia (PSACH) and multiple epiphyseal dysplasia-1 (EDM1) are two rare skeletal diseases that represent distinct endpoints of a continuous phenotypic spectrum with substantial clinical overlap, caused by variants in the gene coding cartilage oligomeric matrix protein (COMP). Objectives:To summarize the clinical characteristics of PSACH/EDM1 and variants of COMP gene, as well as to explore the correlations between them. Methods:PubMed, China National Knowledge Infrastructure, and Wanfang were searched for case reports and case series of patients with genetic diagnosis of PSACH/EDM1 from the inception to 24 March 2025. The clinical characteristics and gene variants of enrolled patients were analyzed and compared to explore genotype-phenotype correlation. Results:A total of 830 PSACH/EDM1 patients (471probands) harboring 224 different variants of COMP gene were enrolled from 106 articles, with missense variants accounting for the majority (80.8%). Exon 13 (183 probands, 38.9%) and type III (T3) repeat domain (413 probands, 87.7%) were the most commonly affected regions, with c.1417_1419del (p.Asp473del) being the most common hotspot variant. Compared with EDM1, PSACH manifested earlier age of onset (p < 0.001), shorter stature (p < 0.001), higher rates of lower limb deformity (p < 0.001), joint laxity (p = 0.041), anterior beaking of the vertebra and irregular/flared metaphysis (p < 0.001), while lower rate of joint pain/osteoarthritis (p < 0.001) and abnormal femoral head (p = 0.008). Missense variants in T3-4 and T3-5 were more likely to cause EDM1 (all p < 0.001), while those in T3-1 and T3-6 to T3-8 were associated with a greater frequency of PSACH (p = 0.002 to 0.023). Majority of in-frame variants were found in PSACH, as c.1417_1419del (p.Asp473del) being PSACH specific. Conclusions:PSACH exhibits more severe phenotypes than EDM1, even with phenotypic overlap. In-frame variants are more strongly associated with PSACH, as the hotspot variant p.Asp473del exclusively identified in PSACH. In contrast, missense variants in T3-4 and T3-5 show a stronger association with EDM1.

To characterize the clinical and genetic features of Chinese FHH type 1 (FHH1) patients and evaluate biochemical discriminators between FHH1 and sporadic primary hyperparathyroidism (s-PHPT). This retrospective study included 11 genetically confirmed FHH1 and 55 s-PHPT patients. Their clinical, biochemical, and imaging data were compared. Genetic testing was conducted via targeted next-generation or whole-exome sequencing. The key discriminators were assessed by receiver operating characteristic (ROC) analysis. Among the 11 FHH1 patients, 63.6

Purpose Primary hypertrophic osteoarthropathy (PHO) is a rare hereditary disorder characterized by both skin and skeletal abnormalities and is classified into two subtypes: PHO autosomal recessive 1 (PHOAR1) and PHO autosomal recessive 2 (PHOAR2). Cyclooxygenase-2 (COX-2) inhibitor Etoricoxib is first-line medication for PHO which could alleviate digital clubbing and pachydermia. However, its effects on the skeletal abnormalities associated with PHO remain unclear. This study aims to comprehensively investigate changes in bone microarchitecture at the distal radius, tibia and interphalangeal bones in PHO patients after 12-month Etoricoxib treatment. Methods A total of 20 PHO patients were enrolled, including 9 PHOAR1 patients and 11 PHOAR2 patients. Bone microstructure was investigated by high-resolution peripheral quantitative computed tomography (HR-pQCT). Results After 12 months' treatment, periosteosis in long bones and osteolysis at the 3rd interphalangeal joint of PHO were visually reduced. HR-pQCT parameters revealed improvements in total volumetric bone mineral density (Tot.vBMD), cortical vBMD (Ct.vBMD) at distal radius and tibia, accompanied with decreased cortical porosity. Trabecular bone showed no significant improvement. Besides, bone stiffness and failure load were significantly enhanced at radius site. In subgroup analysis, PHOAR2 patients experienced decline in trabecular number at the distal tibia, a change not observed in PHOAR1 patients. Correlation analysis revealed inverse associations between disease duration and changes in vBMD and bone strength in PHOAR2 patients, whereas no significant associations were found in PHOAR1 patients. Conclusions Our findings indicated that 12 months of Etoricoxib treatment improved bone microstructure in PHO patients and highlighted differential treatment responses between different genotypes, which might optimize treatment strategies for PHO patients.

Introduction Osteoporosis (OP) is a systemic skeletal disorder that increases fragility and susceptibility to fractures. Despite the availability of teriparatide for the treatment of patients with acute fractures with better efficacy, its long-term daily injection and high cost limit its broader use among a wider patient population, especially for those living in low- and middle-income countries. This study aims to evaluate the efficacy of a novel sequential treatment with teriparatide daily for 6 months followed by denosumab every 6 months for another 18 months, in comparison with denosumab monotherapy every 6 months for 24 months, in reducing the risk of fractures in patients with newly diagnosed osteoporotic fractures. The study will also explore the possible difference between two sequential treatments (shifting to denosumab treatment at 6 or 12 months) in their effect on increasing bone mineral density (BMD).Methods and analysis This study is designed as a multicentre, open-label, randomised controlled trial among 2478 patients with newly diagnosed osteoporotic fractures from 58 hospitals across China. Participants will be randomly assigned in a 10:10:1 ratio to three treatment groups: 24 months of denosumab monotherapy, early sequential treatment (teriparatide for 6 months followed by denosumab for 18 months) and late sequential treatment (teriparatide for 12 months followed by denosumab for 12 months). The primary outcome is the incidence of vertebral fractures over 24 months of treatment. Secondary outcomes include changes in BMD at the lumbar spine, total hip and femoral neck, changes in bone turnover markers (β-carboxy-terminal telopeptide of type 1 collagen and procollagen type 1 N-terminal propeptide), treatment adherence and cost-effectiveness. Follow-up assessments are scheduled at 3, 6, 9, 12, 18 and 24 months post-randomisation for primary and secondary outcomes, and biannually afterwards for the primary outcome.Ethics and dissemination The study protocol has been registered on ClinicalTrials.gov and has received ethical approval from the Peking Union Medical College Hospital Medical Science Research Ethics Committee (1-22PJ939). The findings will be disseminated through peer-reviewed scientific journals.Trial registration number NCT05866029.

Purpose:Neural EGF-like 1 (Nell-1), originally implicated in craniosynostosis, has been identified as a key regulator in osteogenic processes. While preclinical data were encouraging, clinical studies correlating serum Nell-1 levels with osteoporosis remain scarce. This study aims to investigate the relationship between circulation Nell-1 level and bone turnover markers, bone mineral density (BMD), bone microstructure, muscle strength, fall risk, and fractures in Chinese postmenopausal women. Methods:Serum Nell-1 levels were measured in 123 Chinese postmenopausal women. Muscle function was evaluated through grip strength tests, the Short Physical Performance Battery (SPPB), and the Timed Up and Go (TUG) test. Dual-energy X-ray absorptiometry was used to assess areal bone mineral density (aBMD), lumbar trabecular bone score (TBS), and muscle mass. High-resolution peripheral quantitative computed tomography (HR-pQCT) was applied to determine volumetric bone mineral density (vBMD), analyze bone microarchitecture, and estimate bone strength. Result:Postmenopausal women with higher serum Nell-1 levels had higher aBMD and total volumetric bone mineral density (Tot.vBMD) at the distal tibia, larger cortical area (Ct.Ar) and thicker cortical thickness (Ct.Th) at the distal tibia, and higher bone strength. There was a significant negative association between serum Nell-1 levels and C-terminal cross-linking telopeptide of type I collagen (β-CTX), while no significant correlations were observed between serum Nell-1 levels and muscle mass or function. Conclusion:Postmenopausal women with higher serum Nell-1 levels exhibited higher BMD and bone strength, indicating its potential as a therapeutic invention for osteoporosis.

Context: Cutaneous-skeletal hypophosphatemia syndrome (CSHS) is a rare mosaic RASopathy characterized by epidermal or melanocytic nevi, skeletal dysplasia, and fibroblast growth factor 23 (FGF23)-mediated hypophosphatemic rickets. However, its bone microarchitectural features remain underdefined.Objectives: The study aimed to characterize the clinical, genetic, and skeletal microarchitectural features of CSHS.Methods: We summarized clinical findings of 6 CSHS patients with genetically confirmed somatic RAS mutations. Skeletal involvement was assessed by whole-body bone scintigraphy. High-resolution peripheral quantitative computed tomography parameters at the distal radius and tibia were compared with age- and sex-matched XLH controls (1:3). Literature review of 20 genetically confirmed CSHS patients was performed to assess genotype-phenotype correlation.Results: All patients were confirmed to carry somatic HRAS or NRAS mutation in affected skin. Hypophosphatemia, elevated alkaline phosphatase and FGF23 were found in all patients. Genotype-phenotype correlation analysis did not identify any significant biochemical or systemic burden differences across 3 genotypes. Compared with XLH controls, CSHS patients exhibited significantly reduced total and trabecular volumetric BMD (vBMD), with relatively preserved cortical vBMD. Bone cross-sectional geometry was less expanded than in XLH at the tibia. In addition, thinner trabeculae with greater separation were found at the radius, and the distal tibia showed thinner and decreased cortical porosity. Estimated bone stiffness and failure load were significantly lower at both the radius and tibia.Conclusions: CSHS is associated with pronounced trabecular microarchitectural impairment and reduced bone strength that exceed the expected effects of FGF23-mediated phosphate wasting alone, supporting a combined contribution of intrinsic skeletal dysplasia and chronic hypophosphatemia.

Pseudohypoparathyroidism type 1 (PHP1), caused by GNAS defects, is associated with metabolic syndrome components like obesity and insulin resistance, potentially involving adipokine dysregulation. This study characterized serum adipokine profiles in PHP1 and elucidated the independent and interactive effects of GNAS defects and obesity. This single-center, cross-sectional study included 60 PHP1 patients with molecularly confirmed GNAS defects (epigenetic or genetic). Controls (n = 60) were matched for body mass index (BMI), age, and sex. Serum adipokines were measured using multiplex enzyme-linked immunosorbent assay. Generalized linear models assessed independent/joint effects of GNAS defects and obesity on adipokines, adjusting for confounders. Overweight/obesity (OW/OB) prevalence was 58.3

ABSTRACT Background Duchenne muscular dystrophy (DMD) is an X‐linked recessive disorder that affects dystrophin production, characterized by progressive neuromuscular dysfunction, often accompanied by osteoporosis. We prospectively evaluate the effects of bisphosphonates on bone micro‐architecture reflected by trabecular bone score (TBS) of patients with DMD. Methods A total of 72 male children or adolescents with DMD were included, with a mean age of 9.5 ± 1.8 years. They were divided into bisphosphonate treatment groups and control group based on areal bone mineral density (aBMD) and history of fragility fractures. Patients in bisphosphonate treatment groups randomly received intravenous infusion of 5 mg zoledronic acid (ZOL) annually or oral 70 mg alendronate weekly for three years. All patients took calcium 600 mg plus 125 IU vitamin D daily and calcitriol 0.25 μg every other day. TBS at the lumbar spine (LS) and aBMD at the LS, femoral neck (FN) and total hip (TH) were measured annually by dual‐energy X‐ray absorptiometry. Serum levels of β‐isomerized carboxy‐telopeptide of type I collagen and alkaline phosphatase were measured annually during the follow‐up. Results A total of 25 (86.2%), 26 (92.9%) and 13 (86.7%) patients in the ZOL, alendronate and control groups completed the study. After 3 years, TBS Z‐score increased from baseline by 1.13 (p < 0.01), 0.68 (p < 0.01) and 0.26 (p > 0.05) in the ZOL, alendronate and control groups, respectively. The mean increase in TBS Z‐score from baseline was significantly greater in both bisphosphonate treatment groups compared to the control group (p < 0.05). No significant difference was found between the ZOL and alendronate groups. LS, FN and TH aBMD increased by 35.8%, 23.7% and 34.5% in the ZOL group (all p < 0.01 vs. baseline and control group) and by 21.5%, 29.3% and 25.0% in the alendronate group (all p < 0.05 vs. baseline and control group). LS and FN aBMD Z‐scores increased by 1.56 and 1.63 in the ZOL group (all p < 0.01 vs. baseline), by 1.32 and 1.48 in the alendronate group (all p < 0.05 vs. baseline). Bisphosphonates demonstrated a favourable safety profile during the study period. Conclusion This relatively long‐term study confirms that zoledronic acid and alendronate are beneficial to improve micro‐architecture reflected by TBS and aBMD of children or adolescents with DMD.

This study estimated FRAX®-based intervention thresholds for initiating osteoporosis treatment in Chinese postmenopausal women, using real-world data from the largest nationally representative osteoporosis survey in China and a validated Markov microsimulation model. Denosumab became cost-effective at a 10-year major osteoporotic fracture probability of 7

This is the first study to investigate bone microarchitecture in patients with syndrome of inappropriate secretion of thyrotropin (SITSH). Patients with SITSH had impaired bone microarchitecture compared with healthy controls, and the bone loss was associated with the degree of thyrotoxicosis. This study aims to evaluate bone microarchitecture through high-resolution peripheral quantitative computed tomography (HR-pQCT) in patients with syndrome of inappropriate secretion of thyrotropin (SITSH). This cross-sectional study enrolled 32 patients with SITSH. All patients underwent HR-pQCT at distal radius and tibia to quantify bone geometry, volumetric bone mineral density (vBMD) and bone microarchitecture. Of the patients with SITSH, 18 were surgically confirmed to have thyrotropin-secreting pituitary adenoma (TSHoma). An additional 3 patients were clinically diagnosed with TSHoma but did not undergo surgery. The remaining patients were clinically considered to have thyroid hormone resistance, despite the absence of pathogenic variants on genetic testing. Each patient with SITSH was matched by age and gender with one patient with primary hyperthyroidism and one healthy control to analyze the effect of thyroid function on bone microarchitecture. All patients with SITSH in this study presented with elevated thyroid hormone levels and concomitant clinical symptoms of hypermetabolism. Compared with age- and gender-matched healthy controls, patients with SITSH exhibited reduced vBMD, decreased trabecular number, increased trabecular separation, and thinner cortical thickness measured by HR-pQCT. At the non-weight-bearing distal radius, their bone microstructural deficits, including low bone volume, sparse trabeculae, and thin cortex, were comparable to those seen in primary hyperthyroidism. However, at the weight-bearing distal tibia, both trabecular and cortical microarchitecture were more severely compromised in SITSH than in primary hyperthyroidism. In the subgroup analysis of TSHoma, 18 patients were diagnosed as TSHoma after surgery, and they had lower vBMD and decreased cortical thickness compared with healthy controls. While radial microarchitectural impairment in TSHoma was similar to that in primary hyperthyroidism, tibial damage was markedly more severe. Moreover, patients with higher levels of thyroid hormones showed compromised bone microarchitecture. Compared with healthy controls, patients with SITSH exhibited impairment in bone microarchitecture. The degree of impairment at the non-weight-bearing distal radius was comparable to that in primary hyperthyroidism, but microarchitectural deterioration was more pronounced in SITSH at the weight-bearing distal tibia.