Curative surgical treatment after inefficient long-acting somatostatin analogues therapy of a tumor-induced osteomalacia

Mékinian, A.; Ladsous, M.; Balavoine, Anne-Sophie; Carnaille, B.; Aubert, Sébastien; Soudan, Benoı̂t; Wémeau, Jean-Louis

doi:10.1016/j.lpm.2010.10.011

Cited by 11 publications

(17 citation statements)

References 20 publications

(21 reference statements)

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“…Here we present a cohort of subjects with TIO in whom a 3‐day regimen of octreotide was prescribed in an attempt to reduce FGF23 levels. Consistent with most previous case‐reports on the same topic, octreotide was ineffective and did not modify FGF23 secretion or phosphate status in any subject.…”

Section: Discussionsupporting

confidence: 90%

“…One could argue that the lack of effects observed in our cohort compared to the patient reported by Seufert and colleagues could be secondary to shorter treatment duration (3 days in our cohort versus 13 days), but this is unlikely because biochemical improvement was evident during the first days in Seufert and colleagues’ case despite a lower initial octreotide dose than the one we employed. Additional reports have shown that octreotide therapy given for ≥2 months in two TIO patients was ineffective, with no observable changes in phosphate homeostasis . It is possible that the patient studied by Seufert and colleagues expressed an unusual array of SSTRs because mRNA analysis of the PMT showed expression for both SSTR2 and SSTR5, albeit much weaker for the latter.…”

Section: Discussionmentioning

confidence: 98%

“…Thus, the impressive biochemical response presented in the report of Seufert and colleagues, and the demonstration of SSTRs on PMTs, suggest that octreotide may be a viable treatment option for TIO. There have been additional case reports evaluating different regimens of octreotide to determine its potential as a treatment for TIO with negative results (Supporting Table 1) …”

Section: Introductionmentioning

confidence: 99%

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Octreotide Is Ineffective in Treating Tumor-Induced Osteomalacia: Results of a Short-Term Therapy

Ovejero

El‐Maouche

Brillante

et al. 2017

Journal of Bone and Mineral Research

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Tumor-induced osteomalacia (TIO) is a rare paraneoplastic syndrome in which unregulated hypersecretion of fibroblast growth factor 23 (FGF23) by phosphaturic mesenchymal tumors (PMT) causes renal phosphate wasting, hypophosphatemia, and osteomalacia. The resulting mineral homeostasis abnormalities and skeletal manifestations can be reversed with surgical resection of the tumor. Unfortunately, PMTs are often difficult to locate, and medical treatment with oral phosphate and vitamin D analogues is either insufficient to manage the disease or not tolerated. Octreotide has been proposed as a potential treatment for TIO due to the presence of somatostatin receptors (SSTR) on PMTs; however, the role of somatostatin signaling in PMTs and the efficacy of treatment of TIOs with somatostatin analogues is not clear. In an effort to evaluate the efficacy of octreotide therapy in TIO, five subjects with TIO were treated with octreotide for 3 days. Blood intact FGF23, phosphate, and 1,25(OH) D , and tubular reabsorption of phosphate (TRP) were measured at frequent time points during treatment. Octreotide's effects were assessed by comparing group means of the biochemical parameters at each time-point to mean baseline values. There were no significant changes in blood phosphate, FGF23, 1,25(OH) D , or TRP during octreotide treatment, consistent with a lack of efficacy of octreotide in treating TIO. © 2017 American Society for Bone and Mineral Research.

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Section: Discussionsupporting

confidence: 90%

Section: Discussionmentioning

confidence: 98%

Section: Introductionmentioning

confidence: 99%

See 1 more Smart Citation

Octreotide Is Ineffective in Treating Tumor-Induced Osteomalacia: Results of a Short-Term Therapy

Ovejero

El‐Maouche

Brillante

et al. 2017

Journal of Bone and Mineral Research

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“…Basing on high Octreotide uptake in tumor, we decided to use the long acting analogue of Somatostatine, followed by short acting Octreotide in order to amplify the effect of medication on the phosphorus blood levels. We found similar cases in the literature; unfortunately most authors described that Sandostatin analogues allowed to decrease FGF-levels but failed to restore normal phosphorus level [10–12]. In our patient phosphorus level after the Octreotide injection was also still low; however it increased from extremely low values to values near normal ranges with evident improvement of clinical outcomes.…”

Section: Discussionsupporting

confidence: 77%

FGF23 Producing Mesenchymal Tumor

Papierska

Ćwikła

Misiorowski

et al. 2014

Case Reports in Endocrinology

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A 40-year-old patient was referred to Clinic of Endocrinology due to hypophosphatemia causing pain, cramps, and weakness of muscles. Moreover, his bone mineral density was very low. The previous treatment with phosphorus and active vitamin D metabolites was ineffective. In lab tests the hypophosphatemia, hyperphosphaturia, and elevated FGF23 levels were found. Somatostatin receptor scintigraphy (SRS) showed increased radiotracer uptake in the right maxillary sinus and CT scans confirmed presence of tumor in this localization. Biopsy and cytological examination created suspicion of mesenchymal tumor—glomangiopericytoma. Waiting for surgery the patient was treated with long acting Somatostatine analogue, and directly before operation short acting Octreotide and intravenous phosphorus were used. Histology confirmed the cytological diagnosis and the phosphatemia return to normal values in 10 days after the tumor removal.

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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: 98%

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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Curative surgical treatment after inefficient long-acting somatostatin analogues therapy of a tumor-induced osteomalacia

Cited by 11 publications

References 20 publications

Octreotide Is Ineffective in Treating Tumor-Induced Osteomalacia: Results of a Short-Term Therapy

Octreotide Is Ineffective in Treating Tumor-Induced Osteomalacia: Results of a Short-Term Therapy

FGF23 Producing Mesenchymal Tumor

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

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