Tumor-Induced Hypophosphatemic Osteomalacia Diagnosed by the Combinatory Procedures of Magnetic Resonance Imaging and Venous Sampling for FGF23

Nasu, Toru; Kurisu, Seigo; Matsuno, Shohei; Tatsumi, Kunihiro; Kakimoto, Tetsuhiro; Kobayashi, Masato; Nakano, Yoshio; Wakasaki, Hisao; Furuta, Hiroto; Nishi, Masahiro; Sasaki, Hideyuki; Suzuki, Hisanori; Ito, Nobuaki; Fukumoto, Seiji; Nanjo, Kishio

doi:10.2169/internalmedicine.47.0745

Cited by 34 publications

(31 citation statements)

References 16 publications

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“…The important clinical issues for the patient are generalized, debilitating osteomalacia and rickets. 1,4 Hypophosphatemia in TIO results from an excessive renal loss of phosphate, with a low ratio of phosphorus tubule maximum (TmP) to glomerularˆltration rate (GFR) and a low percentage of tubular reabsorption of phosphate (TRP). Evidence shows that patients who undergo tumor removal-the primary treatment for TIO-enjoy complete relief from debilitating symptoms.…”

Section: Discussionmentioning

confidence: 99%

“…Evidence shows that patients who undergo tumor removal-the primary treatment for TIO-enjoy complete relief from debilitating symptoms. 1,17 Various modalities, such as CT, MR imaging, 5-7 FDG-PET, 9-11 111 In-pentetreotide, octreotide scin- Magnetic Resonance in Medical Sciences tigraphy 12,13 and systemic venous sampling 1,17,18 have been used to localize the tumor of TIO. In what we believe is the largest report from a single center, Jagtap and associates analyzed 9 patients with TIO, 11 using 18 F-FDG PET to localize the tumors.…”

Section: Discussionmentioning

confidence: 99%

“…Surgical removal of the responsible tumors is clinically essential. 1 In 1987, Weidner and Santa Cruz 2 reported that most cases of TIO are histologically distinctive and unlike other mesenchymal tumors. Yoshioka's group 3 coined the term``phosphaturic mesenchymal tumor mixed connective tissue variant'' (PMTMCT) to describe this entity.…”

Section: Introductionmentioning

confidence: 99%

“…Bone and muscle pain are generally theˆrst clinical manifestations of PMTMCT and precede identiˆcation of the responsible tumor, which is often di‹cult because the tumors are predominantly small, slow-growing benign neoplasms. 1,4 Recent evidence has shown thatˆbro-blast growth factor 23 (FGF23) from the tumor causes TIO, which induces renal phosphate wasting. 1 Skeletal survey by magnetic resonance (MR) imaging [5][6][7] is one radiologic tool for detecting PMTMCT.…”

Section: Introductionmentioning

confidence: 99%

“…1,4 Recent evidence has shown thatˆbro-blast growth factor 23 (FGF23) from the tumor causes TIO, which induces renal phosphate wasting. 1 Skeletal survey by magnetic resonance (MR) imaging [5][6][7] is one radiologic tool for detecting PMTMCT. [5][6][7][8][9][10][11][12][13] In theˆeld of oncology, whole-body MR imaging (WB-MRI) is being improved and widely used for detecting metastatic bone tumors 14,15 or lymph node metastases.…”

Section: Introductionmentioning

confidence: 99%

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Whole-body MR Imaging in Detecting Phosphaturic Mesenchymal Tumor (PMT) in Tumor-induced Hypophosphatemic Osteomalacia

et al. 2013

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We present 2 cases of tumor-induced osteomalacia (TIO). Both patients had histories of long-term bone and muscle pain. Laboratory data revealed hypophosphatemia. Wholebody magnetic resonance (MR) imaging (WB-MRI) clearly depicted a small subcutaneous mass in the left thigh of theˆrst patient and a right acetabular mass in the second patient. These lesions were pathologically proven to be hemangiopericytoma-phosphaturic mesenchymal tumors (PMT).

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

See 3 more Smart Citations

Whole-body MR Imaging in Detecting Phosphaturic Mesenchymal Tumor (PMT) in Tumor-induced Hypophosphatemic Osteomalacia

et al. 2013

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Tumor-induced osteomalacia: An important cause of adult-onset hypophosphatemic osteomalacia in China: Report of 39 cases and review of the literature

Jiang

Xia

Xing

et al. 2012

Journal of Bone and Mineral Research

194

190

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Tumor-induced osteomalacia (TIO) is an acquired form of hypophosphatemia. Tumor resection leads to cure. We investigated the clinical characteristics of TIO, diagnostic methods, and course after tumor resection in Beijing, China, and compared them with 269 previous published reports of TIO. A total of 94 patients with adult-onset hypophosphatemic osteomalacia were seen over a 6-year period (January, 2004 to May, 2010 in Peking Union Medical College Hospital. After physical examination (PE), all patients underwent technetium-99m octreotide scintigraphy ( 99 Tc m -OCT). Tumors were removed after localization. The results demonstrated that 46 of 94 hypophosphatemic osteomalacia patients had high uptake in 99 Tc m -OCT imaging. Forty of them underwent tumor resection with the TIO diagnosis established in 37 patients. In 2 patients, the tumor was discovered on PE but not by 99 Tc m -OCT. The gender distribution was equal (M/F ¼ 19/20). Average age was 42 AE 14 years. In 35 patients (90%), the serum phosphorus concentration returned to normal in 5.5 AE 3.0 days after tumor resection. Most of the tumors (85%) were classified as phosphaturic mesenchymal tumor (PMT) or mixed connective tissue variant (PMTMCT). Recurrence of disease was suggested in 3 patients (9%). When combined with the 269 cases reported in the literature, the mean age and sex distribution were similar. The tumors were of bone (40%) and soft tissue (55%) origins, with 42% of the tumors being found in the lower extremities. In summary, TIO is an important cause of adult-onset hypophosphatemia in China. 99 Tc m -OCT imaging successfully localized the tumor in the overwhelming majority of patients. Successful removal of tumors leads to cure in most cases, but recurrence should be sought by long-term follow-up. ß

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Tumor localization and biochemical response to cure in tumor-induced osteomalacia

Chong

Andreopoulou

Chen

et al. 2013

Journal of Bone and Mineral Research

116

108

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Tumor-induced osteomalacia (TIO) is a rare disorder of phosphate wasting due to fibroblast growth factor-23 (FGF23)-secreting tumors that are often difficult to locate. We present a systematic approach to tumor localization and postoperative biochemical changes in 31 subjects with TIO. All had failed either initial localization, or relocalization (in case of recurrence or metastases) at outside institutions. Functional imaging with 111 Indium-octreotide with single photon emission computed tomography (octreo-SPECT or SPECT/CT), and 18 fluorodeoxyglucose positron emission tomography/CT (FDG-PET/CT) were performed, followed by anatomic imaging (CT, MRI). Selective venous sampling (VS) was performed when multiple suspicious lesions were identified or high surgical risk was a concern. Tumors were localized in 20 of 31 subjects (64.5%). Nineteen of 20 subjects underwent octreo-SPECT imaging, and 16 of 20 FDG-PET/CT imaging. Eighteen of 19 (95%) were positive on octreo-SPECT, and 14 of 16 (88%) on FDG-PET/CT. Twelve of 20 subjects underwent VS; 10 of 12 (83%) were positive. Sensitivity, specificity, positive predictive value (PPV), and negative predictive value (NPV) were as follows: sensitivity ¼ 0.95, specificity ¼ 0.64, PPV ¼ 0.82, and NPV ¼ 0.88 for octreo-SPECT; sensitivity ¼ 0.88, specificity ¼ 0.36, PPV ¼ 0.62, and NPV ¼ 0.50 for FDG-PET/CT. Fifteen subjects had their tumor resected at our institution, and were disease-free at last follow-up. Serum phosphorus returned to normal in all subjects within 1 to 5 days. In 10 subjects who were followed for at least 7 days postoperatively, intact FGF23 (iFGF23) decreased to near undetectable within hours and returned to the normal range within 5 days. C-terminal FGF23 (cFGF23) decreased immediately but remained elevated, yielding a markedly elevated cFGF23/iFGF23 ratio. Serum 1, (1,25D) rose and exceeded the normal range. In this systematic approach to tumor localization in TIO, octreo-SPECT was more sensitive and specific, but in many cases FDG-PET/CT was complementary. VS can discriminate between multiple suspicious lesions and increase certainty prior to surgery. Sustained elevations in cFGF23 and 1,25D were observed, suggesting novel regulation of FGF23 processing and 1,25D generation.

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Tumor-Induced Hypophosphatemic Osteomalacia Diagnosed by the Combinatory Procedures of Magnetic Resonance Imaging and Venous Sampling for FGF23

Abstract: This report describes a 37-year-old man with tumor-induced osteomalacia (TIO

Cited by 34 publications

References 16 publications

Whole-body MR Imaging in Detecting Phosphaturic Mesenchymal Tumor (PMT) in Tumor-induced Hypophosphatemic Osteomalacia

Whole-body MR Imaging in Detecting Phosphaturic Mesenchymal Tumor (PMT) in Tumor-induced Hypophosphatemic Osteomalacia

Tumor-induced osteomalacia: An important cause of adult-onset hypophosphatemic osteomalacia in China: Report of 39 cases and review of the literature

Tumor localization and biochemical response to cure in tumor-induced osteomalacia

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