Difficult Diagnostic Cases

Auethavekiat, Paranee; Roberts, Jefferson; Biega, Timothy J.; Toney, Morakinyo O.; Christensen, Reed S.; Belnap, Christina; Berenberg, Jeffrey L.

doi:10.1200/jco.2005.05.043

Cited by 16 publications

(3 citation statements)

References 3 publications

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“…Recent literature suggests that they variably express SSTRs. [ 11 ] These characteristics have aroused interest in SSTR imaging in these patients. Agrawal et al .…”

Section: Discussionmentioning

confidence: 99%

Diagnostic importance of contrast enhanced ¹⁸ F-fluorodeoxyglucose positron emission computed tomography in patients with tumor induced osteomalacia: Our experience

et al. 2016

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Aims and Objectives:To assess the diagnostic utility of contrast-enhanced 18F-fluorodeoxyglucose positron emission tomography-computed tomography (FDG PET-ceCT) in localization of tumors in patients with clinical diagnosis of tumor-induced osteomalacia (TIO), in correlation with histopathological results.Materials and Methods:Eight patients (five male and three female) aged 24–60 (mean 42) years with a clinical diagnosis of TIO were included in this prospective study. They underwent whole body (head to toe) FDG PET-ceCT following a standard protocol on Philips GEMINI TF PET-CT scanner. The FDG PET-ceCT results were correlated with postoperative histology findings and clinical follow-up.Results:All the patients had an abnormal PET-ceCT study. The sensitivity of PET-ceCT was 87.5%, and positive predictive value was 100%. The tumor was located in the craniofacial region in 6/8 patients and in bone in 2/8 patients. Hemangiopericytoma was the most common reported histology. All patients underwent surgery, following which they demonstrated clinical improvement. However, one patient with atypical findings on histology did not show any clinical improvement, hence, underwent 68Gallium-DOTANOC PET-ceCT scan for relocalization of the site of the tumor.Conclusion:The tumors causing TIO are small in size and usually located in obscure sites in the body. Hence, head to toe protocol should be followed for FDG PET-ceCT scans with the inclusion of upper limbs. Once the tumor is localized, regional magnetic resonance imaging can be performed for better characterization of soft tissue lesion. Imaging with FDG PET-ceCT plays an important role in detecting the site of the tumor and thereby facilitating timely management.

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“…Recent literature suggests that they variably express SSTRs. [ 11 ] These characteristics have aroused interest in SSTR imaging in these patients. Agrawal et al .…”

Section: Discussionmentioning

confidence: 99%

Diagnostic importance of contrast enhanced ¹⁸ F-fluorodeoxyglucose positron emission computed tomography in patients with tumor induced osteomalacia: Our experience

et al. 2016

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“…Способностью секретировать ФРФ23 обладают остеоциты и их предшественники, по сути дела, это мезенхимальные стволовые клетки на различных этапах дифференцировки в сторону остеобласта/остеоцита, которые и составляют секреторную часть опухоли. Гиперсекреция ФРФ23 описана для ряда доброкачественных опухолей мезенхимального происхождения: гемангиоперицитомы, липомы, опухоли околоушной железы [2,10,11]. Концепцию объединения под термином «фосфатурических мезенхимальных опухолей» гетерогенных опухолей костей и мягких тканей, секретирующих фосфатурические гормоны, предложил в 1987 г. Weidner N. и соавт.…”

Section: Discussionunclassified

A clinical case of phosphopenic osteomalacia due to paraneoplastic secretion of metastatic prostate cance

et al. 2023

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Phosphaturic mesenchymal tumor can cause osteomalacia due to excessive secretion of fibroblast growth factor 23 (FGF23), which disrupts the metabolism of phosphate and vitamin D. These tumors are predominantly benign, but less than 5% of them are malignant forms. This article presents the first clinical case in the Russian Federation of a 69-year-old patient with severe hypophosphatemia due to metastatic prostate cancer. Increased secretion of FGF23 are described in the androgen-resistent prostate cancer, which led to pronounced disorders of mineral metabolism, accompanied by a clinical symptom of weakness, pain in the bones, immobilization of the patient. The condition was regarded as worsening against the background of the progression of the disease. However, symptomatic therapy aimed at increasing the level of phosphate significantly improved the patient’s general condition. The medical community should be aware of the possibility of developing hypophosphatemia in patients with weakness and bone pain, which are not always associated with the progression of metastatic prostate cancer.

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“…Гиперсекреция ФРФ23 описана для ряда доброкачественных (атерома, гемангиоперицитома и др.) [6][7][8][9][10] и, реже, злокачественных (рак предстательной железы, рак молочной железы, анапластический рак щитовидной железы, рак толстой кишки, мелкоклеточный рак легкого) опухолей [1, [11][12][13][14][15][16][17][18].…”

Section: Introductionunclassified

Clinical features, diagnostics and treatment of FGF23 secreting tumors: series of 40 clinical cases

Gronskaia,

Belaya,

Rozhinskaya

et al. 2023

Probl Endokrinol (Mosk)

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Introduction: Tumor-induced osteomalacia is an acquired rare disease manifested by hypophosphatemic osteomalacia due to excessive secretion of fibroblast growth factor 23 (FGF23). FGF 23 is a non-classical hormone secreted by bone tissue (osteocytes) and regulates phosphorus metabolism.The aim of this work is to present clinical experience in the diagnosis, treatment and rehabilitation of patients with tumor-induced osteomalacia.Materials and methods: 40 patients with clinically-confirmed tumor-induced osteomalacia were included in the study, 34 of whom had the tumor localized, 27 underwent surgical treatment and 21 achieved stable remission.Results: The median age was 48 [41; 63] years, 43% were men, the time left from the the onset of the disease was 8 [4; 10] years. Biochemical findings were hypophosphatemia 0.47 [0.4; 0.53] mmol/l, a decrease in the tubular reabsorption phosphate 62 [52; 67]%, and an increase in alkaline phosphatase of 183 [112; 294] units/l. At the time of diagnosis, 100% had multiple pathological fractures, only 10% could move independently, and 77.5% classified the pain as unbearable (8–10 points according to the 10-point pain syndrome scale ). Among the methods used to detect tumors, the most sensitive were scintigraphy with tectrotide with SPECT/CT 71.4% (20/28) and MRI 90% (18/20). In 35% of cases, the tumor was localized in soft tissues and in 65% in bone tissue; The tumor was most often detected in the lower extremities, followed by the head in frequency of localization. 18 patients currently have no remission and they receive conservative treatment (phosphorus and alfacalcidol n=15 and burosumab n=3). In case of achieving remission (n=21), regression of clinical symptoms and restoration of bone and muscle mass was observed. Extensive excision of the tumor without prior biopsy resulted in the best percentage of remission — 87%.Conclusion: Tumor-induced osteomalacia is characterized by severe damage to bone and muscle tissue with the development of multiple fractures, muscle weakness and severe pain syndrome. In laboratory diagnostics, attention should be paid to hypophosphatemia, a decrease in the tubular reabsorption phosphate index and increased alkaline phosphatase. The use of functional diagnostic methods with a labeled somatostatin analogue to the subtype 2 receptor and MRI with contrast enhancement are the most accurate methods of topical diagnostics. In case of localization of the tumor, a wide excision without a preliminary biopsy is recommended.

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Difficult Diagnostic Cases

Cited by 16 publications

References 3 publications

Diagnostic importance of contrast enhanced ¹⁸ F-fluorodeoxyglucose positron emission computed tomography in patients with tumor induced osteomalacia: Our experience

Diagnostic importance of contrast enhanced ¹⁸ F-fluorodeoxyglucose positron emission computed tomography in patients with tumor induced osteomalacia: Our experience

A clinical case of phosphopenic osteomalacia due to paraneoplastic secretion of metastatic prostate cance

Clinical features, diagnostics and treatment of FGF23 secreting tumors: series of 40 clinical cases

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Difficult Diagnostic Cases

Cited by 16 publications

References 3 publications

Diagnostic importance of contrast enhanced 18 F-fluorodeoxyglucose positron emission computed tomography in patients with tumor induced osteomalacia: Our experience

Diagnostic importance of contrast enhanced 18 F-fluorodeoxyglucose positron emission computed tomography in patients with tumor induced osteomalacia: Our experience

A clinical case of phosphopenic osteomalacia due to paraneoplastic secretion of metastatic prostate cance

Clinical features, diagnostics and treatment of FGF23 secreting tumors: series of 40 clinical cases

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Diagnostic importance of contrast enhanced ¹⁸ F-fluorodeoxyglucose positron emission computed tomography in patients with tumor induced osteomalacia: Our experience

Diagnostic importance of contrast enhanced ¹⁸ F-fluorodeoxyglucose positron emission computed tomography in patients with tumor induced osteomalacia: Our experience