Oncogenic osteomalacia: Diagnostic importance of fibroblast growth factor 23 and F-18 fluorodeoxyglucose PET/CT SCAN for the diagnosis and follow-up in one case

Dupond, J.L.; Mahammedi, Hakim; Prié, Dominique; Collin, Françoise; Gil, H.; Blagosklonov, Oleg; Ricbourg, B; Méaux-Ruault, N.; Kantélip, B.

doi:10.1016/j.bone.2005.01.001

Cited by 99 publications

(60 citation statements)

References 19 publications

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“…High levels of FGF-23 cause phosphate diuresis known as phosphaturia and inhibit renal phosphate reabsorption which results in hypophosphatemia. This will stimulate mobilization of phosphate and calcium from the bone into the blood as a compensatory mechanism causing the bone to become weak and fracture [16,17]. Microscopically, the tumor is a spindle cell mesenchymal neoplasm with variable cellularity.…”

Section: Introductionmentioning

confidence: 99%

Phosphaturic Mesenchymal Tumor: 2 New Oral Cases and Review of 53 Cases in the Head and Neck

Qari

Hamao-Sakamoto

Fuselier

et al. 2015

Head and Neck Pathol

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Phosphaturic mesenchymal tumor (PMT) is a rare neoplasm that secretes fibroblast growth factor-23 (FGF-23) and causes oncogenic osteomalacia. It occurs in adults with equal gender distribution and the most common location is the lower extremities, followed by the head and neck. Besides osteomalacia, the clinical presentation includes bone pain and multiple bone fractures. Microscopic features consist of spindle cells, multinucleated giant cells, and calcifications embedded in a chondromyxoid matrix. Laboratory findings indicate normal calcium and parathyroid levels, hypophosphatemia, and increased levels of FGF-23 that usually revert to normal after surgical removal. Due to its rarity, the purpose of the study was to report 2 new oral cases of PMT and to review the literature in the head and neck. The first case occurred in the gingiva and had been present for 6 years. The second case was a recurrence of a previously diagnosed PMT in the right mandible that metastasized to the lung and soft tissue. The literature review included 53 cases in the head and neck. There was a predilection for extra-oral sites (76 %) compared to intra-oral sites (24 %) with paranasal sinuses considered the most common location (38 %) followed by the mandible (15 %). There were 9 recurrences that included 3 malignant cases indicating a potentially aggressive tumor. Due to the indeterminate biological behavior of PMT and its rarity, a comprehensive evaluation of medical, laboratory, radiographic, and histological findings are crucial for a definitive diagnosis and treatment.

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Section: Introductionmentioning

confidence: 99%

Phosphaturic Mesenchymal Tumor: 2 New Oral Cases and Review of 53 Cases in the Head and Neck

Qari

Hamao-Sakamoto

Fuselier

et al. 2015

Head and Neck Pathol

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“…In many cases, CT and MRI are not contributory in diagnosing oncogenic osteomalacia [3] because this tumor is often small, grows slowly, and develops in unpredictable locations such as feet [17], mandibular ramus [18], and frontal bone [19]. Previous case reports suggested that 18 F-FDG PET/CT is a useful modality in detection of oncogenic tumors causing oncogenic osteomalacia [11][12][13][14][15]. In the present report, the mesenchymal tumor causing oncogenic osteomalacia was easily detected by 18 F-FDG PET/CT.…”

Section: Discussionmentioning

confidence: 99%

“…Somatostatin receptor expression is elevated in mesenchymal tumors, and it has been reported that 111 In-pentetreotide or octreotide scintigraphy was useful in some cases [8][9][10]. However, somatostatin receptor imaging was not contributory in another report [11].…”

Section: Introductionmentioning

confidence: 99%

“…To the best of our knowledge, only a few case reports have evaluated the feasibility of 18 F-fluorodeoxyglucose positron emission tomography ( 18 F-FDG PET)/CT in detecting occult mesenchymal tumors in oncogenic osteomalacia [11][12][13][14][15]. Despite the benign nature of mesenchymal tumors, each case report indicated that the mesenchymal tumor associated with oncogenic osteomalacia exhibited relatively high 18 F-FDG uptake and was easily detected by 18 F-FDG PET/CT.…”

Section: Introductionmentioning

confidence: 99%

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Using 18F-FDG PET/CT to Detect an Occult Mesenchymal Tumor Causing Oncogenic Osteomalacia

Seo

Choi

Jeong

et al. 2011

Nucl Med Mol Imaging

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Oncogenic osteomalacia is a rare paraneoplastic syndrome characterized by renal phosphate excretion, hypophosphatemia, and osteomalacia. This syndrome is often caused by tumors of mesenchymal origin. Patients with oncogenic osteomalacia have abnormal bone mineralization, resulting in a high frequency of fractures. Tumor resection is the treatment of choice, as it will often correct the metabolic imbalance. Although oncogenic osteomalacia is a potentially curable disease, diagnosis is difficult and often delayed because of the small size and sporadic location of the tumor. Bone scintigraphy and radiography best characterize osteomalacia; magnetic resonance imaging findings are nonspecific. Here, we report a case of oncogenic osteomalacia secondary to a phosphaturic mesenchymal tumor that was successfully detected by 18 F-fluorodeoxyglucose positron emission tomography/computed tomography ( 18 F-FDG PET/CT). This case illustrates the advantages of 18 F-FDG PET/CT in detecting the occult mesenchymal tumor that causes oncogenic osteomalacia.

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“…Tumours usually bearing the ability to over-secrete FGF23 are generally of mesenchymal origin, but there are cases of an adenocarcinoma of the colon and prostate (Ramon et al 2011, Mak et al 2012, Leaf et al 2013. FGF23 measurements combined with 18 FDG PET/CT SCAN can be a decisive tool in cases of TIO, and is likely going to be of considerable importance for facilitating early diagnosis and follow-up (Dupond et al 2005). Current management of TIO involves tumour resection.…”

Section: Less Common Endocrine Pns Secondary To the Secretion Of Peptmentioning

confidence: 99%

Paraneoplastic endocrine syndromes

Dimitriadis

Angelousi

Weickert

et al. 2017

Endocrine-Related Cancer

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The majority of neoplasms are responsible for symptoms caused by mass effects to surrounding tissues and/or through the development of metastases. However, occasionally neoplasms, with or without endocrine differentiation, acquire the ability to secrete a variety of bioactive substances or induce immune cross-reactivity with the normal tissues that can lead to the development of characteristic clinical syndromes. These syndromes are named endocrine paraneoplastic syndromes when the specific secretory components (hormones, peptides or cytokines) are unrelated to the anticipated tissue or organ of origin. Endocrine paraneoplastic syndromes can complicate the patient's clinical course, response to treatment, impact prognosis and even be confused as metastatic spread. These syndromes can precede, occur concomitantly or present at a later stage of tumour development, and along with the secreted substances constitute the biological 'fingerprint' of the tumour. Their detection can facilitate early diagnosis of the underlying neoplasia, monitor response to treatment and/or detect early recurrences following successful initial management. Although when associated with tumours of low malignant potential they usually do not affect long-term outcome, in cases of highly malignant tumours, endocrine paraneoplastic syndromes are usually associated with poorer survival outcomes. Recent medical advances have not only improved our understanding of paraneoplastic syndrome pathogenesis in general but also enhanced their diagnosis and treatment. Yet, given the rarity of endocrine paraneoplastic syndromes, there is a paucity of prospective clinical trials to guide management. The development of well-designed prospective multicentre trials remains a priority in the field in order to fully characterise these syndromes and provide evidence-based diagnostic and therapeutic protocols.

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Oncogenic osteomalacia: Diagnostic importance of fibroblast growth factor 23 and F-18 fluorodeoxyglucose PET/CT SCAN for the diagnosis and follow-up in one case

Cited by 99 publications

References 19 publications

Phosphaturic Mesenchymal Tumor: 2 New Oral Cases and Review of 53 Cases in the Head and Neck

Phosphaturic Mesenchymal Tumor: 2 New Oral Cases and Review of 53 Cases in the Head and Neck

Using 18F-FDG PET/CT to Detect an Occult Mesenchymal Tumor Causing Oncogenic Osteomalacia

Paraneoplastic endocrine syndromes

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