Six-year follow-up of a characteristic osteolytic lesion in a patient with tumor-induced osteomalacia

Piemonte, Sara; Romagnoli, Elisabetta; Cipriani, Cristiana; Lucia, F. De; Pilotto, Roberta; Diacinti, Daniele; Pepe, Jessica; Minisola, Salvatore

doi:10.1530/eje-13-0581

Cited by 21 publications

(8 citation statements)

References 15 publications

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“…Our data are in accordance with the low BMD and T ‐score in the two TIO patients described by Piemonte et al 11 and Malabanan et al 12 who also had low phosphorus and history of fractures at the time of diagnosis, with one of the patients using a wheelchair. Other studies that reported lower BMD in individuals with other causes of hypophosphatemic osteomalacia included vitamin D deficiency, nutritional malabsorption and renal tubular acidosis.…”

Section: Discussionsupporting

confidence: 92%

Microarchitectural parameters and bone mineral density in patients with tumour‐induced osteomalacia by HR‐pQCT and DXA

Mendes

Coelho

Gehrke

et al. 2021

Clinical Endocrinology

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Introduction Tumour‐induced osteomalacia (TIO) is a rare paraneoplastic condition characterised by decreased tubular phosphate reabsorption. The purpose of this study is to evaluate bone mineral density (BMD) and microarchitecture in six TIO patients, compared with 18 healthy controls. Methods Volumetric BMD and microarchitecture were evaluated by high‐resolution peripheral quantitative computed tomography (HR‐pQCT), and areal BMD by dual‐energy X‐ray absorptiometry (DXA). Differences between groups were significant for p < .05. Results All TIO subjects were healthy until the development of diffuse bone pain and multiple skeletal fractures and deformities. At baseline, sPi and TmPi/GFR were low and patients were on vitamin D and phosphate replacement at the study. Compared with controls, TIO patients had lower aBMD at lumbar spine and hip, and lower vBMD at trabecular, cortical and entire bone, at distal radius (R) and distal tibia (T): trabecular vBMD (R = 118.3 × 177.1; T = 72.3 × 161.3 gHA/cm3); cortical vBMD (R = 782.3 × 866.5; T = 789.1 × 900.9 gHA/cm3); total region vBMD (R = 234.5 × 317; T = 167.1 × 295.8 gHA/cm3). Bone microarchitecture was very heterogeneous among patients and significantly different from controls: lower cortical thickness (R = 0.59 × 0.80; T = 0.90 × 1.31 mm), bone volume‐to‐total volume ratio (R = 0.09 × 0.14; T = 0.06 × 0.13) and Tb.N (R = 1.46 × 2.10; T = 0.93 × 1.96 mm−1) and also higher Tb.Sp (R = 0.70 × 0.41; T = 1.28 × 0.45 mm) and Tb.1/N.SD (R = 0.42 × 0.18; T = 0.87 × 0.20 mm). Conclusion In this original study of TIO patients, DXA and HR‐pQCT evaluation identified lower areal and volumetric BMD and severely impaired microarchitecture at cortical and trabecular bones, which probably contribute to bone fragility and fractures.

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Section: Discussionsupporting

confidence: 92%

Microarchitectural parameters and bone mineral density in patients with tumour‐induced osteomalacia by HR‐pQCT and DXA

Mendes

Coelho

Gehrke

et al. 2021

Clinical Endocrinology

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“…To our knowledge, most publications about surgical excision of phosphaturic mesenchymal tumors have been case reports [17][18][19][20][21][22][23][24][25][26][27][28][29][30][31][32] , and very few were case series 2,33,34 . Phosphaturic mesenchymal tumors seem to be commonly located in long bones, but we are not aware of any studies comparing the results of curettage and segmental resection with a minimum one-year follow-up.…”

Section: Discussionmentioning

confidence: 99%

Surgical Treatments of Tumor-Induced Osteomalacia Lesions in Long Bones

Wang

Zhong

Liu

et al. 2015

The Journal of Bone and Joint Surgery

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“…Finally, calcium was abnormally high in ∼9% of the patients with pancreatic ductal adenocarcinoma and these patients could have a paraneoplastic syndrome. 20 In pancreatic cancer patients, the results of a systemic review have shown that dietary vitamin D or circulating concentrations of 25-hydroxyvitamin D are not associated with the risk of pancreatic cancer. 21 Other authors have found that, in men, there is an increased risk of pancreatic cancer associated with currently recommended dietary vitamin D intake levels; no associations with vitamin D intake were observed among women.…”

Section: Discussionmentioning

confidence: 99%

Markers of Bone Metabolism in Patients With Chronic Pancreatitis and Pancreatic Ductal Adenocarcinoma

2015

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There are no studies comparing some of the most important markers, such as vitamin D, parathormone, osteocalcin, bone alkaline phosphatase, and calcium, in patients with chronic benign and malignant pancreatic diseases. Our objective was to comparatively evaluate serum markers of bone metabolism in patients with chronic pancreatitis and in those with ductal pancreatic adenocarcinoma. Sixty-three consecutive subjects were studied: 30 patients with a firm diagnosis of chronic pancreatitis and 33 having histologically confirmed pancreatic adenocarcinoma. Serum 25-hydroxyvitamin D, bone alkaline phosphatase, osteocalcin, parathormone, and calcium were determined using commercially available kits. Taking into consideration the clinical variables of all 63 patients studied, 25-hydroxyvitamin D was inversely correlated with only the body mass index (P = 0.007), whereas it was not correlated with age (P = 0.583) or fecal elastase-1 concentrations (P = 0.556). Regarding the other substances studied, parathormone was positively correlated with only the age of the patients (P = 0.015). Of the 5 substances studied, only bone alkaline phosphates were significantly different (P < 0.001) between patients with chronic pancreatitis and those with pancreatic ductal adenocarcinoma. Within the 2 groups of patients, the 23 patients with chronic pancreatitis without diabetes mellitus had serum concentrations of 25-hydroxyvitamin D significantly lower (P = 0.045) than those with chronic pancreatitis having diabetes mellitus, whereas smokers with pancreatic ductal adenocarcinoma had serum concentrations of calcium significantly higher (P < 0.001) as compared to nonsmokers. Altered bone metabolism seems to be associated with chronic diseases of the pancreas; however, the mechanism should be better elucidated.

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Six-year follow-up of a characteristic osteolytic lesion in a patient with tumor-induced osteomalacia

Cited by 21 publications

References 15 publications

Microarchitectural parameters and bone mineral density in patients with tumour‐induced osteomalacia by HR‐pQCT and DXA

Microarchitectural parameters and bone mineral density in patients with tumour‐induced osteomalacia by HR‐pQCT and DXA

Surgical Treatments of Tumor-Induced Osteomalacia Lesions in Long Bones

Markers of Bone Metabolism in Patients With Chronic Pancreatitis and Pancreatic Ductal Adenocarcinoma

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