Impaired bone microarchitecture and strength in patients with tumor-induced osteomalacia

Zanchetta, María Belén; Jerkovich, Fernando; Nuñez, Selva; Mocarbel, Yamile; Pignatta, Analía; Elias, Natália; Díaz, Alcira Ofélia; Roganovich, Juan Manuel; Vigovich, Carlos; Balonga, María Celeste; Cohen, Ana Carolina; Mumbach, Giselle; González, Sofía; Plantalech, L.; Fradinger, Erich; Zanchetta, José R.

doi:10.1002/jbmr.4325

Cited by 18 publications

(25 citation statements)

References 29 publications

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“…This finding provides evidence for the trabecularization of the cortex in patients with TIO, which was speculated above. However, different from recent study that revealed more correlation between iPTH levels and HR-pQCT parameters at the tibia, (7) we only found a correlation between iPTH levels and HR-pQCT parameters at the radius. In addition, none of the biochemical indices were associated with TBS.…”

Section: Discussioncontrasting

confidence: 99%

“…Consistent with the findings of Zanchetta and colleagues, (7) we found that the tibia showed greater defects than the radius in most HR-pQCT parameters. As previously reported, mobility impairment and fracture were common in patients with TIO, with most patients requiring crutches or wheelchairs.…”

Section: Discussionsupporting

confidence: 92%

“…In this study, both trabecular and cortical bone was extensively deteriorated at the radius and tibia, leading to reduced bone strength in TIO patients compared with that of healthy controls, which was consistent with the findings in recent studies by Zanchetta and colleagues and Mendes and colleagues. (7,8) However, the percentage differences in HR-pQCT parameters and bone strength between patients and controls were smaller in our study than those in a recent study by Zanchetta and colleagues. (7) On the one hand, it might be due to the shorter disease duration observed in our patients.…”

Section: Discussioncontrasting

confidence: 68%

“…(7,8) However, the percentage differences in HR-pQCT parameters and bone strength between patients and controls were smaller in our study than those in a recent study by Zanchetta and colleagues. (7) On the one hand, it might be due to the shorter disease duration observed in our patients. On the other hand, previous studies have revealed that physical activity improves bone density and microarchitecture, (17) while lack of activity has a negative effect on bone.…”

Section: Discussioncontrasting

confidence: 68%

“…In addition, bone strength of the peripheral skeleton could be estimated by micro-finite element analysis (μFEA). Although two recent studies reported impaired microarchitecture and bone strength assessed by HR-pQCT in a small number of patients with a clinical diagnosis of TIO, (7,8) these findings need to be further validated in a large cohort of patients with a confirmed diagnosis of TIO. In addition, trabecular bone score (TBS), a textural index that evaluates trabecular microarchitecture by calculating pixel gray-level variations using DXA images of the lumbar spine, (9) is easier to access than HR-pQCT.…”

Section: Introductionmentioning

confidence: 99%

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Bone Impairment in a Large Cohort of Chinese Patients With Tumor-Induced Osteomalacia Assessed by HR-pQCT and TBS

Feng

Guan

et al. 2020

Journal of Bone and Mineral Research

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Tumor-induced osteomalacia (TIO) is a rare paraneoplastic syndrome caused by excessive production of fibroblast growth factor 23 (FGF23) by a tumor. Previous studies have revealed generalized mineralization defects and low areal bone mineral density (aBMD) in TIO. However, data on the bone microarchitecture in TIO are limited. In this study, we evaluated the microarchitecture in the peripheral (distal radius and tibia) and axial (lumbar spine) skeleton using high-resolution peripheral quantitative computed tomography (HR-pQCT) and trabecular bone score (TBS) and investigated related factors in a large cohort of Chinese patients with TIO. A total of 186 patients with TIO who had undergone dual-energy X-ray absorptiometry (DXA) or HR-pQCT scans were enrolled. Compared with age-, sex-, and body mass index (BMI)-matched healthy controls, TIO patients (n = 113) had lower volumetric BMD, damaged microstructure, and reduced bone strength in the peripheral skeleton, especially at the tibia. The average TBS obtained from 173 patients was 1.15 AE 0.16. The proportion of patients with abnormal TBS (<1.35) was higher than that with low L 1 to L 4 aBMD Z-score (Z ≤ À2) (43.9% versus 89.6%, p < 0.001). Higher intact fibroblast growth factor 23 (iFGF23), intact parathyroid hormone (iPTH), alkaline phosphatase, and β-isomerized C-terminal telopeptide of type I collagen (β-CTx) levels, more severe mobility impairment, and a history of fracture were associated with poorer HR-pQCT parameters but not with lower TBS. However, greater height loss and longer disease duration were correlated with worse HR-pQCT parameters and TBS. Moreover, TBS was correlated with both trabecular and cortical HR-pQCT parameters in TIO. In conclusion, we revealed impaired bone microarchitecture in the axial and peripheral skeleton in a large cohort of Chinese TIO patients. HR-pQCT parameters and TBS showed promising advantages over aBMD for assessing bone impairment in patients with TIO. A longer follow-up period is needed to observe changes in bone microarchitecture after tumor resection.

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Section: Discussioncontrasting

confidence: 99%

Section: Discussionsupporting

confidence: 92%

Section: Discussioncontrasting

confidence: 68%

Section: Discussioncontrasting

confidence: 68%

Section: Introductionmentioning

confidence: 99%

See 3 more Smart Citations

Bone Impairment in a Large Cohort of Chinese Patients With Tumor-Induced Osteomalacia Assessed by HR-pQCT and TBS

Feng

Guan

et al. 2020

Journal of Bone and Mineral Research

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Unusual presentation of tumor‐induced osteomalacia mismanaged due to misdiagnosis: A literature review based on a case report

Fatollahzadeh,

Meybodi,

Pajavand

et al. 2024

Clinical Case Reports

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Key Clinical MessageTumor‐induced osteomalacia is a rare but potentially serious disease with nonspecific misguiding manifestations that can result in a wrong diagnosis and being treated for rheumatologic or other similar diseases. In patients with unexpected fractures, resistant musculoskeletal pains, and hypophosphatemia, this diagnosis should be considered by the physicians and approached through a complete history taking, physical exam laboratory, and radiologic evaluation to give the opportunity of on‐time treatment to the patient.AbstractTumor‐induced osteomalacia (TIO) is an uncommon mesenchymal tumor that results in disproportionate phosphorus excretion, primarily leading to bone‐related symptoms. Laboratory, imaging, and histopathological evaluation can confirm this pathologic condition. In this case, we present the history and subsequent clinical parts of a 50‐year‐old woman who presented with an unusual presentation of generalized musculoskeletal pains and a right ankle mass. Her disease was diagnosed with multidisciplinary evaluation and was approached by a surgical treatment. The patient was treated with total resection of the tumor, which led to complete resolution of musculoskeletal and metabolic abnormalities, which were resolved following total tumor resection. TIO is a paraneoplastic disease that results in abnormal secretion of phosphatonins, particularly fibroblast growth factor 23 (FGF23). This can cause hypophosphatemia, hyperparathyroidism, lower bone density, and increased risk of pathologic fractures. These tumors are mostly cured by surgical ± radiotherapy. The present study aims to provide insight into the fact that a TIO diagnosis is not always straightforward. However, in suspicious cases such as unexplained hypophosphatemia, it should be considered to prevent delayed diagnosis of the progressive pathology. The earlier treatment can prevent several complications and reduce the risk of mortality.

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Tumor-Induced Osteomalacia: A Systematic Clinical Review of 895 Cases

et al. 2022

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Tumor-induced osteomalacia (TIO) is a rare and largely underdiagnosed paraneoplastic condition. Previous reviews often reported incomplete data on clinical aspects, diagnosis or prognosis. The aim of this study was to present a systematic clinical review of all published cases of TIO. A search was conducted in Pubmed, Embase, Web of Science from inception until April 23rd, 2020. We selected case reports and case series of patients diagnosed with TIO, with information on tumor localization and serum phosphate concentration. Two reviewers independently extracted data on biochemical and clinical characteristics including bone involvement, tumor localization and treatment. 468 articles with 895 unique TIO cases were included. Median age was 46 years (range 9 months–90 years) and 58.3% were males. Hypophosphatemia and inappropriately low or normal 1,25-dihydroxyvitamin D levels, characteristic for TIO, were present in 98% of cases. Median tumor size was 2.7 cm (range 0.5 to 25.0 cm). Serum fibroblast growth factor 23 was related to tumor size (r = 0.344, P < 0.001). In 32% of the cases the tumor was detected by physical examination. Data on bone phenotype confirmed skeletal involvement: 62% of cases with BMD data had a T-score of the lumbar spine ≤ − 2.5 (n = 61/99) and a fracture was reported in at least 39% of all cases (n = 346/895). Diagnostic delay was longer than 2 years in more than 80% of cases. 10% were reported to be malignant at histology. In conclusion, TIO is a debilitating disease characterized by a long diagnostic delay leading to metabolic disturbances and skeletal impairment. Increasing awareness of TIO should decrease its diagnostic delay and the clinical consequences.

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Impaired bone microarchitecture and strength in patients with tumor-induced osteomalacia

Cited by 18 publications

References 29 publications

Bone Impairment in a Large Cohort of Chinese Patients With Tumor-Induced Osteomalacia Assessed by HR-pQCT and TBS

Bone Impairment in a Large Cohort of Chinese Patients With Tumor-Induced Osteomalacia Assessed by HR-pQCT and TBS

Unusual presentation of tumor‐induced osteomalacia mismanaged due to misdiagnosis: A literature review based on a case report

Tumor-Induced Osteomalacia: A Systematic Clinical Review of 895 Cases

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