Tumor-induced osteomalacia – Current imaging modalities and a systematic approach for tumor localization

Rayamajhi, Sampanna Jung; Yeh, Randy; Wong, Tony T.; Dumeer, Shifali; Mittal, Bhagwant Rai; Remotti, Fabrizio; Chikeka, Ijeuru; Reddy, Arun

doi:10.1016/j.clinimag.2019.04.007

Cited by 26 publications

(27 citation statements)

References 84 publications

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“…Even if false negative findings are possible, in about 10% of cases, it should be underlined that SSTR-PET/CT has allowed the detection of culprit tumors which remained occult with conventional imaging methods in most of the cases [16][17][18][19][20][21][22][23][24][25][26][27][28][29]. The main explanation for false negative results at conventional imaging methods is the reduced size of most culprit tumors causing osteomalacia [1,3,4]. False positive findings of SSTR-PET/CT in this setting are also described [20,25], in particular caused by inflammatory lesions due to the overexpression of SSTR by activated inflammatory cells [30,31].…”

Section: Discussionmentioning

confidence: 99%

“…Regarding imaging methods, several techniques can be used for the localization of culprit tumors in patients with TIO, including anatomic imaging modalities such as plain radiographs, computed tomography (CT), and magnetic resonance imaging (MRI), nuclear medicine imaging techniques or hybrid functional and morphological imaging modalities such as single-photon emission computed tomography/CT (SPECT/CT), or positron emission tomography/CT (PET/CT) using different radiopharmaceuticals [4].…”

Section: Introductionmentioning

confidence: 99%

“…SSTR-PET/CT is extensively used for the diagnosis of neuroendocrine tumors, due to the overexpression of SSTRs in neuroendocrine tumor cells [6][7][8]. However, SSTRs are also expressed in non-neuroendocrine tumors [9], such as mesenchymal tumors [4,5]; therefore, as most of culprit lesions in patients with TIO are mesenchymal tumors, they can be detected using SSTR-PET/CT [4,5].…”

Section: Introductionmentioning

confidence: 99%

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Detection Rate of Culprit Tumors Causing Osteomalacia Using Somatostatin Receptor PET/CT: Systematic Review and Meta-Analysis

et al. 2019

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Background: Tumor-induced or oncogenic osteomalacia (TIO) is a rare paraneoplastic syndrome in which osteomalacia is a consequence of fibroblast growth factor 23 (FGF23) secretion by a mesenchymal tumor. The localization of the culprit lesion in patients with TIO is often challenging. Several studies have evaluated the detection rate (DR) of these tumors using somatostatin receptor positron emission tomography (SSTR-PET/CT). We aimed to summarize literature findings on this topic providing pooled estimates of DR. Methods: A comprehensive literature search by screening PubMed, Embase and Cochrane library electronic databases through August 2019 was performed. The pooled DR of culprit tumors using SSTR-PET/CT in patients with TIO was calculated using a random-effects statistical model. Results: Fourteen studies on the use of SSTR-PET/CT in detecting the culprit tumor in patients with TIO were included in the qualitative analysis. The pooled DR of SSTR-PET/CT on a per-patient-based analysis calculated using eleven studies (166 patients) was 87.6% (95% confidence interval (95% CI) 80.2–95.1%). Statistical heterogeneity among studies was detected (I-square = 63%), likely due to the use of different radiolabeled somatostatin analogues, as demonstrated by a subgroup analysis. Conclusions: Despite limited literature data due to the rarity of the disease, SSTR-PET/CT demonstrated a very high DR of culprit tumors in patients with TIO and it could be used as first-line imaging method for this indication.

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Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Detection Rate of Culprit Tumors Causing Osteomalacia Using Somatostatin Receptor PET/CT: Systematic Review and Meta-Analysis

et al. 2019

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“…PMTs are typically benign, slowly growing, and therefore often small and without local symptoms. Furthermore, they do not have a predilection site, but may occur anywhere in the body, including unexpected locations, such as hands and soles of the feet [5]. In our case, the tumor itself had been pictured months before the final diagnosis, but was not identified as such, probably because the MRI scan was performed in order to detect skeletal changes and a soft tissue tumor was not expected.…”

Section: Discussionmentioning

confidence: 77%

Fibroblast Growth Factor 23-Producing Phosphaturic Mesenchymal Tumor with Extraordinary Morphology Causing Oncogenic Osteomalacia

Then¹,

Asbach²,

Bartsch

et al. 2020

Medicina

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A possible cause of hypophosphatemia is paraneoplastic secretion of fibroblast growth factor 23 (FGF-23). Tumors secreting FGF-23 are rare, mostly of mesenchymal origin, usually benign, and may be located anywhere in the body, including hands and feet, which are often not represented in conventional imaging. A 50-year-old woman presented with diffuse musculoskeletal pain and several fractures. Secondary causes of osteoporosis were excluded. Laboratory analysis revealed hypophosphatemia and elevated alkaline phosphatase, parathyroid hormone, and FGF-23. Thus, oncogenic osteomalacia due to neoplastic FGF-23 secretion was suspected. FDG-PET-CT and DOTATATE-PET-CT imaging demonstrated no tumor. Cranial MRI revealed a tumorous mass in the left cellulae ethmoidales. The tumor was resected and histopathological examination showed a cell-rich tumor with round to ovoid nuclei, sparse cytoplasm, and sparse matrix, resembling an olfactory neuroblastoma. Immunohistochemical analysis first led to diagnosis of olfactory neuroblastoma, which was later revised to phosphaturic mesenchymal tumor. Following the resection, FGF-23 and phosphate levels normalized. In conclusion, we here describe a patient with an FGF-23-secreting phosphaturic mesenchymal tumor with an unusual morphology. Furthermore, we emphasize diagnostic pitfalls when dealing with FGF-23-induced hypophosphatemia.

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“…The most common type of inborn rickets and osteomalacia (approximately 1:20 000 individuals) is due to X-linked hypophosphatemia (XLH) mutation of PHEX (phosphate-regulating gene with homologies to endopeptidases on the X chromosome), that causes increase in FGF23 levels [4]. X-linked hypophosphatemia and tumor-induced osteomalacia (TIO) are the most common causes of hypophosphatemic vitamin D-resistant rickets/osteomalacia [5].…”

Section: Introductionmentioning

confidence: 99%

The Difficult Diagnosis of Hypophosphatemic Rickets-A Review of 8 Clinical Cases

Borissovа¹,

Vlahov²,

Fukumoto³

et al. 2020

CMR

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Tumor-induced osteomalacia – Current imaging modalities and a systematic approach for tumor localization

Cited by 26 publications

References 84 publications

Detection Rate of Culprit Tumors Causing Osteomalacia Using Somatostatin Receptor PET/CT: Systematic Review and Meta-Analysis

Detection Rate of Culprit Tumors Causing Osteomalacia Using Somatostatin Receptor PET/CT: Systematic Review and Meta-Analysis

Fibroblast Growth Factor 23-Producing Phosphaturic Mesenchymal Tumor with Extraordinary Morphology Causing Oncogenic Osteomalacia

The Difficult Diagnosis of Hypophosphatemic Rickets-A Review of 8 Clinical Cases

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