Clinical Impact of Somatostatin Receptor Scintigraphy in the Management of Tumor-induced Osteomalacia

Duet, M.; Kerkeni, S.; Sfar, Raja; Bazille, Cécile; Lioté, Frédéric; Orcel, Philippe

doi:10.1097/rlu.0b013e31818866bf

Cited by 62 publications

(47 citation statements)

References 24 publications

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“…As a result of the presence of somatostatin receptors on these tumors, it has been suggested that octreotide, a synthetic somatostatin analogue, would be useful in decreasing FGF-23 secretion [20]. Although there is one case report regarding use of octreotide as mitigating therapy before definitive treatment [20], few clinicians have been successful in repeating the initial success with this technique [3,4,13,16]. One of our patients underwent a trial of octreotide chemotherapy, but this was unsuccessful.…”

Section: Discussionmentioning

confidence: 97%

The Phosphaturic Mesenchymal Tumor: Why is Definitive Diagnosis and Curative Surgery Often Delayed?

Ledford

Zelenski

Cardona

et al. 2013

Clinical Orthopaedics &Amp; Related Research

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Background Tumor-induced osteomalacia is a paraneoplastic syndrome resulting in renal phosphate wasting and decreased bone mineralization. Phosphaturic mesenchymal tumors represent a rare etiology of tumor-induced osteomalacia. Nonspecific symptoms of fatigue, bone pain, and musculoskeletal weakness make the diagnosis elusive and lead to a delay in surgical treatment. Questions/purposes In this case series, the following three questions were asked: (1) How do the clinical presentation and features of phosphaturic mesenchymal tumors delay the diagnosis? (2) What is the clinical course after surgical treatment of phosphaturic mesenchymal tumors? (3) How frequently do phosphaturic mesenchymal tumors recur and are there factors associated with recurrence?Methods This study retrospectively reviewed the cases of five adults diagnosed and treated for phosphaturic mesenchymal tumors. Patients were identified through an internal orthopaedic oncology database with clinical, surgical, and histologic data obtained through a systematic chart review. Results Five patients presented with a long-standing history of osteomalacia, generalized fatigue, pain, and weakness before the diagnosis was reached at an average of 7.2 years (range, 2-12 years) after initial symptom onset. The diagnosis appeared to be delayed owing to the cryptic medical presentation, difficulty in locating tumor by imaging, and confirming histologic appearance. Two patients treated with wide surgical resection did not experience recurrence compared with three patients who did show recurrent signs and symptoms after marginal excision. A postoperative increase in fibroblast-derived growth factor-23 was associated with recurrent disease. Conclusions Although uncommon, the diagnosis of phosphaturic mesenchymal tumor should be considered in any patient who presents with hypophosphaturic osteomalacia and no other physiologic cause. Definitive treatment is early, wide surgical resection. Level of Evidence Level IV, therapeutic study. See the Instructions for Authors for a complete description of levels of evidence.

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Section: Discussionmentioning

confidence: 97%

The Phosphaturic Mesenchymal Tumor: Why is Definitive Diagnosis and Curative Surgery Often Delayed?

Ledford

Zelenski

Cardona

et al. 2013

Clinical Orthopaedics &Amp; Related Research

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“…Somatostatin receptors have been found to be present in many TIO lesions, and 111-indium octreotide has a high affinity for the somatostatin receptor, especially subtype 2 and to a lesser extent subtype 5 [13,15]. As with FDG PET/CT, emphasis should be placed on making sure these imaging tests cover the entire body from head to toe, including the hands and feet.…”

Section: Discussionmentioning

confidence: 99%

“…It should be noted, however, that there are 'blind' spots in this functional imaging modality, for example, the brain uptake of FDG-PET tracer may obscure intracranial tumors, and uptake of octreotide in the liver and spleen may obscure potential lesions within these organs [3,4,13,15].…”

Section: Discussionmentioning

confidence: 99%

Ga68-DOTA Peptide PET/CT to Detect Occult Mesenchymal Tumor-Inducing Osteomalacia: A Case Series of Three Patients

2015

Nucl Med Mol Imaging

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Tumor-induced osteomalacia (TIO) is a rare disease that manifests with paraneoplasic syndrome and overproduction of fibroblast growth factor 23 (FGF23), leading to renal phosphate wasting and hyperphosphaturia, eventually leading to acquired hypophosphatemic osteomalacia. Diagnosis of this disease is often challenging because of the small size of the lesion, which can be localized in bone or soft tissue anywhere in the body. Detecting these occult mesenchymal tumors (OMT) is of great importance as they are potentially curable after tumor resection. The purpose of this case series is to provide some insight into the diagnosis and localization of OMT associated with osteomalacia, particularly using functional imaging with Ga68-DOTA peptide PET/CT scans.

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“…Since many tumors causing osteomalacia are small and difficult to locate, radiologic methods are often employed to localize the tumors. Several different methods have been used including magnetic resonance imaging (48-50), technetium 99m sestamibi scintigraphy (51-55), radiolabeled octreotide scanning (8,(56)(57)(58)(59)(60)(61), and F-18 fluorodeoxyglucose positron emission tomography with computed tomography (8,(62)(63)(64)(65)(66)(67). In some small series, authors have claimed sensitivities of 60-80% with radiolabeled octreotide scanning and PET/CT scanning.…”

Section: Radiological Investigationsmentioning

confidence: 99%

Tumor-induced Osteomalacia

Raj¹,

Chen²,

Sheldon³

et al. 2009

Encyclopedia of Molecular Mechanisms of Disease

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Clinical description and evaluation Disease phenotypeTumor-induced osteomalacia (TIO) is a rare syndrome (about 350 such cases have been described), characterized by hypophosphatemia, increased urinary losses of phosphate as a result of reduced tubular phosphate reabsorption, reduced or inappropriately normal concentrations of 1,25-dihydroxyvitamin D, normal parathyroid hormone concentrations, rickets or osteomalacia which are caused by substances (generally, fibroblast growth factor-23, FGF-23, and more rarely other peptides) elaborated by mesenchymal tumors (1-8). Patients manifest symptoms of osteomalacia or rickets with bone pain and proximal muscle weakness. Sometimes the causal tumors are readily visible or palpable on physical examination, but more often the tumors are small and very difficult to detect. Removal of the tumor results in reversal of the biochemical abnormalities, thereby demonstrating that the tumors produce a substance which alters phosphate homeostasis.Key biochemical abnormalities seen in the syndrome of TIO are shown in Table 1. As shown, these include hypophosphatemia, normal or low serum calcium concentrations, normal parathyroid hormone concentrations, normal 25-hydroxyvitamin D concentrations, inappropriately low 1,25-dihydroxyvitamin D concentrations, normal renal function, elevated fractional excretion of phosphorus or a low TMP/GFR and generally elevated serum FGF-23 concentrations. Most, but not all, patients with TIO have elevations in serum FGF-23 concentrations (9, 10). Patients generally have osteomalacia or rickets noted on skeletal radiography ( Figure 1A) although the findings can often be quite subtle or absent.

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Clinical Impact of Somatostatin Receptor Scintigraphy in the Management of Tumor-induced Osteomalacia

Abstract: We highlight the impact of somatostatin receptor evaluation in the management of TIO.

Cited by 62 publications

References 24 publications

The Phosphaturic Mesenchymal Tumor: Why is Definitive Diagnosis and Curative Surgery Often Delayed?

The Phosphaturic Mesenchymal Tumor: Why is Definitive Diagnosis and Curative Surgery Often Delayed?

Ga68-DOTA Peptide PET/CT to Detect Occult Mesenchymal Tumor-Inducing Osteomalacia: A Case Series of Three Patients

Tumor-induced Osteomalacia

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