JBMR Plus最新文献

{"title":"Real-world differences in denosumab persistence, reinitiation, and switching among cohorts of older adults in Canada and the United States.","authors":"Kaleen N Hayes, Selvam R Sendhil, Sulbh Aggarwal, Andrew R Zullo, Sarah D Berry, Arman Oganisian, Michael Adegboye, Suzanne M Cadarette","doi":"10.1093/jbmrpl/ziaf061","DOIUrl":"10.1093/jbmrpl/ziaf061","url":null,"abstract":"<p><p>Denosumab is an injectable osteoporosis medication administered twice per year. Discontinuation of denosumab can result in rapid rebound fractures, but the evidence is limited on real-world persistence with denosumab. We conducted 2 parallel, population-based cohort studies leveraging (1) healthcare administrative data from Ontario, Canada (ON; 100% population) and (2) a 20% random sample of US Medicare beneficiaries (US). The first denosumab claim (US: 1/2010-12/2019; ON: 1/2012-12/2021) was identified using pharmacy claims (ON) and Medicare Parts D and B claims (US). Patients aged <66 yr, residing in long-term care (LTC), or with implausible data (eg, death before first claim) were excluded. We developed and applied an algorithm that used dosing and days between dispensations to clean denosumab claims. We assumed a days supply of 183 d for each dispensation and defined discontinuation as a 60-d gap in coverage. We estimated initial persistence, reinitiation, and switching to other osteoporosis medications using Kaplan-Meier estimators, censoring on death, disenrollment (US only), LTC admission, or study end (12/31/2022 [ON], 12/31/2020 [US]). We also estimated the monthly proportion of patients with an on-time denosumab dose to explore time trends. We identified 168 339 eligible individuals in ON (mean age = 78 yr; 90% female) and 97 595 in the US (mean age = 77 yr; 90% female). In ON, the median time to denosumab discontinuation was longer (median 2.3 yr [ON] vs 1.7 yr [US]; 3-yr persistence: 44% [ON] vs 31% [US]), and time to reinitiation was shorter (median = 0.5 yr [ON] vs 1.9 yr [US]). In both populations, around 10% switched to another osteoporosis medication. Women and those with prior oral bisphosphonate use had longer durations of denosumab treatment in ON but not in the US. The proportion persisting with on-time doses did not increase over time in the US or ON. Research to improve persistence with denosumab and optimize post-denosumab treatment is critical.</p>","PeriodicalId":14611,"journal":{"name":"JBMR Plus","volume":"9 6","pages":"ziaf061"},"PeriodicalIF":3.4,"publicationDate":"2025-04-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12087952/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144101885","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":"Osseous morphology differences in the foot and ankle associated with Charcot-Marie-Tooth disease.","authors":"Melissa R Requist, Andrew C Peterson, Timothy C Beals, Bopha Chrea, Amy L Lenz","doi":"10.1093/jbmrpl/ziaf058","DOIUrl":"10.1093/jbmrpl/ziaf058","url":null,"abstract":"<p><p>Charcot-Marie-Tooth (CMT) disease is a genetic, progressive peripheral nerve disease that commonly manifests in a cavovarus foot deformity. Previously, this foot deformity has been believed to be an alignment change in the foot, but recent research has shown that there are bone morphology differences in individuals with CMT. Differences in bone morphology have been identified in the calcaneus, talus, and medial cuneiform, but have not been consistently analyzed throughout the foot or studied in relation to different genetic subtypes of CMT. This study is a retrospective, cross-sectional analysis of bone morphology in CMT using weight-bearing computed tomography and statistical shape modeling. This analysis identified bone morphology differences between CMT and control groups throughout the hindfoot, midfoot, and forefoot. Bone morphology differences were also present between the 2 primary disease subtypes throughout the foot. Key morphologic findings include the altered shape of the subtalar articular surfaces on the talus, bending of the metatarsals, variation in navicular process morphology, and differences between subtypes in the talus, medial cuneiform, and medial metatarsals. There are several possible theoretical mechanisms for this osseous deformation, including bone remodeling in response to altered loading from alignment change or from decreased musculotendinous forces, but the patterns of morphological variation seen in these data cannot be fully explained by these mechanisms, suggesting that there may be an interaction between the neuronal disease and bone remodeling. Future work is necessary to characterize the progression of bony deformity throughout development and to correlate bone shape with function, gait, muscle morphology and strength to elucidate the mechanism of osseous morphology change in varying subtypes of CMT.</p>","PeriodicalId":14611,"journal":{"name":"JBMR Plus","volume":"9 6","pages":"ziaf058"},"PeriodicalIF":3.4,"publicationDate":"2025-04-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12087961/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144101881","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":"Concerns about the paper, \"Benefits of targeted vibration for bone strength and bone density in postmenopausal women with osteopenia: a randomized, sham-controlled trial\".","authors":"Douglas P Kiel, Theresa A Guise, Maya Styner, Janet Rubin","doi":"10.1093/jbmrpl/ziaf051","DOIUrl":"https://doi.org/10.1093/jbmrpl/ziaf051","url":null,"abstract":"","PeriodicalId":14611,"journal":{"name":"JBMR Plus","volume":"9 5","pages":"ziaf051"},"PeriodicalIF":3.4,"publicationDate":"2025-04-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12035689/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143996447","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":"AGN1 local osteo-enhancement procedure increases proximal femur volumetric bone mineral density of women with post-menopausal osteoporosis as assessed by quantitative computed tomography analysis.","authors":"Michelle Chin, Ronald Hill, Bryan Huber, James Howe, Klaus Engelke","doi":"10.1093/jbmrpl/ziaf036","DOIUrl":"https://doi.org/10.1093/jbmrpl/ziaf036","url":null,"abstract":"<p><p>In this study, QCT was used to analyze the AGN1 Local Osteo-Enhancement Procedure (LOEP) as a treatment to form bone in the proximal femurs of patients with osteoporosis. Using this minimally invasive procedure, a resorbable triphasic AGN1 implant material was injected into the left femurs of 12 women with post-menopausal osteoporosis. Computed tomography scans were taken before treatment (baseline) and at 12 wk, 24 wk, and 5-7 yr after treatment. Quantitative computed tomography was used to investigate the resorption of AGN1 within the treated proximal femurs and to analyze the treatment's impact on integral, trabecular, and cortical bone. The untreated right femurs were used as controls. Data illustrated an increase in trabecular volumetric BMD (trab vBMD) of treated hips at all timepoints (baseline: 22 ± 21 mg/cm³ vs 217 ± 56 mg/cm³, 161 ± 18 mg/cm³, and 121 ± 37 mg/cm³ at 12-wk, 24-wk, and 5- to 7-yr timepoints, respectively), and an increase in integral vBMD of 65% at the 12-wk timepoint and 34% at the 5- to 7-yr timepoint. The increase in trab vBMD was observed in the location where the AGN1 implant material bolus was injected, and at the 5- to 7-yr timepoint, no significant BMD change was observed in the trabecular regions surrounding the original implantation zone (treated: 32 ± 16 mg/cm³, control: 31 ± 16 mg/cm³). This QCT study provides a more detailed understanding of the resorption and transformation of the AGN1 implant material into bone and supports, with some limitations, that the AGN1 LOEP treatment can locally increase trabecular bone density in weakened areas of the proximal femur where strength increase is most needed to reduce the risk of hip fragility fracture.</p>","PeriodicalId":14611,"journal":{"name":"JBMR Plus","volume":"9 5","pages":"ziaf036"},"PeriodicalIF":3.4,"publicationDate":"2025-04-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12035697/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144035282","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":"Bone disease and osteoporosis associated with Pompe disease.","authors":"Lucas Maxey, Hannah Freibert, Auremil Quinonez, Hartmut Malluche, Madhumathi Rao","doi":"10.1093/jbmrpl/ziaf045","DOIUrl":"https://doi.org/10.1093/jbmrpl/ziaf045","url":null,"abstract":"<p><p>Pompe disease is a lysosomal storage disorder defined by a mutation in the GAA gene encoding alpha-1,4-glucosidase alpha (acid maltase). Pompe disease encompasses a range of clinical presentations that are broadly characterized as either classic infantile Pompe disease or late-onset Pompe disease (LOPD). LOPD is a milder manifestation of the disease that presents after the first year of life and is typically characterized by mild proximal muscle weakness and lack of cardiac involvement compared to the classic infantile form. The mainstay of treatment is enzyme replacement therapy (EnRT). Decreased bone mineral density (BMD) is frequently encountered in LOPD. While bone loss is thought to be due to mechanical unloading secondary to the progressive muscle weakness associated with the disease, there is a lack of tissue-level data in support of this mechanism. We describe a 60-yr-old female with LOPD managed with EnRT who presented with proximal muscle weakness and decreased BMD on dual-energy X-ray absorptiometry. Undecalcified bone histology showed low turnover osteoporosis, and treatment was initiated with romosozumab. Romosozumab specifically may provide a promising osteoporosis therapy for LOPD-associated osteoporosis. As a sclerostin inhibitor, it both inhibits bone resorption and promotes new bone formation. We additionally emphasize that bone biopsy should be considered as a useful diagnostic tool in the evaluation of osteoporosis associated with uncommon pathologies, since bone histology provides more specific tissue-level information over clinical and laboratory evaluation as well as substantive guidance for treatment.</p>","PeriodicalId":14611,"journal":{"name":"JBMR Plus","volume":"9 5","pages":"ziaf045"},"PeriodicalIF":3.4,"publicationDate":"2025-04-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11994030/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143967481","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":"Response to Letter to the Editor for \"Benefits of targeted vibration for bone strength and bone density in postmenopausal women with osteopenia: a randomized, sham-controlled trial\".","authors":"Laura D Bilek, Michael J Jaasma","doi":"10.1093/jbmrpl/ziaf050","DOIUrl":"https://doi.org/10.1093/jbmrpl/ziaf050","url":null,"abstract":"","PeriodicalId":14611,"journal":{"name":"JBMR Plus","volume":"9 5","pages":"ziaf050"},"PeriodicalIF":3.4,"publicationDate":"2025-04-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12010160/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143965895","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":"Obese-diabetic female <i>Ksr2</i> knockout mice develop brittle bones near end of life.","authors":"Gustavo A Gomez, Sasidhar Uppuganti, Sheila Pourteymoor, Jillian Bray, Jeffry S Nyman, Subburaman Mohan","doi":"10.1093/jbmrpl/ziaf052","DOIUrl":"https://doi.org/10.1093/jbmrpl/ziaf052","url":null,"abstract":"<p><p>The influence of obesity and type 2 diabetes (T2D) on the skeleton is complex, with affected individuals having higher fracture risk despite having higher BMD. To evaluate how obesity and T2D affect skeletal health, we studied mice with disruption of a gene that regulates energy intake and expenditure, <i>Ksr2</i>, which results in reduced metabolic rate and severe insulin resistance in both mice and a subpopulation of humans. Relative to 28-wk-old littermate sibling controls, <i>Ksr2</i> mutants weighed more than double the body and fat weight. Moreover, leptin and insulin were elevated by 20- and 10-fold in <i>Ksr2</i> mutant serum, consistent with prior reports of a T2D state. Micro-computed tomography analysis revealed increased trabecular bone volume (BV) per total volume (TV) in the mutant's distal femur, proximal tibia, and vertebrae. While the bone size (cortical (Ct) cross-sectional bone area) was increased by 7%-11% at the mid-diaphysis of femurs and tibiae, Ct BV adjusted for TV was unaffected. Three-point bending tests revealed increased ultimate force to failure and ultimate bending stress at the mid-diaphysis of femurs by 13% and 8%, respectively in <i>Ksr2</i> mutants. However, bone toughness, a measure of bone quality that assesses how well Ct bone resists fracture, was reduced by 25%. To determine the cause of reduced bone quality in <i>Ksr2</i> mutants, we evaluated femurs for bone hydration by nuclear magnetic resonance relaxometry and found reduced pore water (20%) in <i>Ksr2</i> mutant femurs relative to controls. Moreover, analysis of hydrolysates from femurs for advanced glycation end products revealed a 14% increase in <i>Ksr2</i> mutants. Based on our data, we conclude that while bone density and strength are increased in mice with obesity-induced insulin resistance, bone toughness is compromised due to reduced bone tissue quality, thus suggesting therapeutics focused on improving bone tissue are needed to reduce fracture risk in obese patients.</p>","PeriodicalId":14611,"journal":{"name":"JBMR Plus","volume":"9 5","pages":"ziaf052"},"PeriodicalIF":3.4,"publicationDate":"2025-03-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12035690/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143967438","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":"Calcium isotope ratio in patients with monogenic bone diseases: a prospective, cross-sectional, single-center pilot study.","authors":"Robert Munzinger, Felix N von Brackel, Mikolaj Bartosik, Florian Barvencik, Michael Amling, Ralf Oheim","doi":"10.1093/jbmrpl/ziaf032","DOIUrl":"https://doi.org/10.1093/jbmrpl/ziaf032","url":null,"abstract":"<p><p>Stable calcium isotope fractions have long been related to the calcium metabolism in living organisms. The blood and urine proportions of calcium isotopes ⁴⁴Ca and ⁴²Ca (δ^44/42Ca) have recently again attracted attention as a potential diagnostic tool in metabolic bone diseases, in particular osteoporosis. The hypothesis is that the lighter isotopes (Ca⁴²) get incorporated into bone more quickly; hence, δ^44/42Ca ratios in urine and serum are higher for bone formation and lower for resorption phases. Therefore, δ^44/42Ca in blood and urine may serve as an indicator of bone metabolism, potentially reflecting bone density in general. We have conducted clinical characterization by means of laboratory assessment, bone densitometry, HRpQCT, and isotope analysis to test for the hypothesis in patients with monogenic bone diseases. We included 40 adult subjects with hereditary bone diseases, such as early-onset osteoporosis (<i>n</i> = 7), osteogenesis imperfecta (<i>n</i> = 12), hypophosphatasia (<i>n</i> = 12), and X-linked hypophosphatemia (XLH, <i>n</i> = 9), and controls (<i>n</i> = 17). Regression analyses revealed significant correlations of δ^44/42Ca with Ca/creatinine_urine (<i>R</i> ² = 0.6200, <i>p</i> < .0001), and bone densitometric parameters were significantly correlated with δ^44/42Ca (BMD: δ^44/42Ca_serum  <i>R</i> ² = 0.2685, <i>p</i> ≤ .001; δ^44/42Ca_urine  <i>R</i> ² = 0.3554; <i>p</i> < .0002). XLH differed significantly from the other diseases and controls by means of higher δ^44/42Ca_urine. Our results suggest that δ^44/42Ca is strongly coupled to urinary calcium excretion in patients with hereditary bone diseases. Significant correlations with BMD suggest an interaction of δ^44/42Ca and bone mass though it lacks discriminative power. Further studies are needed to evaluate the utility of δ^44/42Ca in clinical practice.</p>","PeriodicalId":14611,"journal":{"name":"JBMR Plus","volume":"9 5","pages":"ziaf032"},"PeriodicalIF":3.4,"publicationDate":"2025-03-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12009546/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143990150","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":"Fracture risk by cortisol excess status in patients with adrenal incidentalomas: a population-based cohort study.","authors":"Annette L Adams, In-Lu Amy Liu, Iris Anne C Reyes, Hina Chowdhry, Richard Contreras, Yuqian M Gu, Mackenzie Crawford, Bennett McDonald, Joshua I Barzilay, Tish Villanueva, David A Katz, Frank S Czerwiec, Wansu Chen","doi":"10.1093/jbmrpl/ziaf043","DOIUrl":"https://doi.org/10.1093/jbmrpl/ziaf043","url":null,"abstract":"<p><p>Adrenal incidentalomas (AIs) may secrete excess cortisol, representing an elevated endogenous exposure to glucocorticoids, which could decrease bone mineral density and increase fracture risk. However, measurement of cortisol excess is not routinely done in patients with AI; thus, those with hormonally active AI at increased risk for fracture are under-identified. We sought to examine the association between excess cortisol levels and the incidence of fragility fracture in people with AI. This retrospective cohort study, conducted within two Kaiser Permanente regions (Southern California and Georgia), comprised women and men aged ≥50 yr with identified AI in the study period January 1, 2015-August 31, 2022. Patients' cortisol excess status was categorized by the type of test conducted (if any) and the test result. Fractures and relevant covariates were ascertained via International Classification of Diseases (ICD)-9/10 codes. Hazard ratios (HR) were estimated using Cox proportional hazard models with mortality as a competing risk. Among the cohort of 14 886 patients with AI, 273 (1.8%) had autonomous cortisol secretion (ACS) confirmed by dexamethasone suppression test (DST) results >1.8 μg/dL (>50 nmol/L), and another 201 (1.4%), tested with urine free or random cortisol tests, had results suggestive of excess cortisol production. Most of the cohort (<i>n</i> = 9353, 62.8%) were untested around AI diagnosis or during follow-up. Compared to patients with normal DST results (and adjusted for age, sex, race/ethnicity, and several other clinical characteristics), the estimated HR of fracture risk for patients with ACS (HR 1.42, CI 0.86-2.32), evidence of cortisol excess (1.41, 0.85-2.32), and untested patients (1.28, 0.88-1.87) were suggestive of elevated risk. However, none of the elevated hazard rates were statistically significant at the 95% significance level. The apparent elevated risk in the untested patients suggests that many untested patients may have hormonally active AI that puts them at risk for fracture from secondary osteoporosis.</p>","PeriodicalId":14611,"journal":{"name":"JBMR Plus","volume":"9 5","pages":"ziaf043"},"PeriodicalIF":3.4,"publicationDate":"2025-03-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12036655/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144024168","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 treatment of a child with McCune-Albright syndrome/polyostotic fibrous dysplasia: challenges and benefits.","authors":"Sophia D Sakka, Danai Georgakopoulou, Artemis Doulgeraki, Andreas H Krieg, John Anastasopoulos, Gabor Szinnai, Christina Kanaka-Gantenbein","doi":"10.1093/jbmrpl/ziaf042","DOIUrl":"https://doi.org/10.1093/jbmrpl/ziaf042","url":null,"abstract":"<p><p>Fibrous dysplasia/McCune-Albright syndrome (FD/MAS) is a rare condition caused by a mutation in the GNAS locus. Apart from endocrinopathies, some cases are characterized by excessive fibroblast growth factor 23 (FGF23) production from abnormal fibro-osseous tissue in FD lesions, resulting in increased renal phosphate excretion. We present a girl with FD/MAS and severe skeletal burden, evidenced by the presence of polyostotic fibrous dysplasia, which was complicated with bone fractures. She also had hyperthyroidism and GnRH-independent precocious puberty. She received zoledronic acid infusions in preparation for hip surgery. Despite optimal conventional management with oral phosphate and alphacalcidol, which was poorly tolerated, she presented persistent hypophosphatemia. To control hypophosphatemia and its deleterious effects on bone health, treatment with burosumab off-label at a dose of 0.66 mg/kg (20 mg) every 2 wk was initiated. Serum phosphate levels normalized within 2 wk of treatment. Laboratory results showed improvement in serum alkaline phosphatase (ALP) and PTH levels. After the second injection of burosumab, phosphate and PTH rose above the normal range with normal vitamin D levels; therefore, the interval between doses was increased to 3 wk, and calcium 500 mg daily was added. However, phosphate levels dropped again below normal range, so she had to return to 2-weekly injections of 20 mg. After 11 mo on burosumab, she remains with high normal phosphate levels and normal PTH and ALP values. Burosumab is well tolerated, with no adverse events to date. Burosumab is a human monoclonal antibody against FGF23 that reduces the risk of developing FGF23-mediated hypophosphatemia and its associated complications. Burosumab should be considered as an effective and safe alternative strategy for FGF23-mediated hypophosphatemia in FD/MAS for those who either cannot tolerate or do not respond to conventional therapy. To our knowledge, this is the fourth published case worldwide describing successful treatment with burosumab in FD/MAS.</p>","PeriodicalId":14611,"journal":{"name":"JBMR Plus","volume":"9 5","pages":"ziaf042"},"PeriodicalIF":3.4,"publicationDate":"2025-03-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12035695/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143967501","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}