Searchable abstracts of presentations at key conferences in endocrinology
Endocrine Abstracts (2016) 41 EP638 | DOI: 10.1530/endoabs.41.EP638

1Third Department of Internal Medicine, Division of Diabetes, Endocrinology and Metabolism, Kyorin University School of Medicine, Tokyo, Japan; 2Department of Orthopaedic Surgery, Faculty of Medicine, Kyorin University, Tokyo, Japan; 3Department of Pathology, Kyorin University School of Medicine, Tokyo, Japan.


A 32-year-old woman with a nine-year history of right hip pain was admitted after she became unable to walk. Based on marked hypophosphatemia as indicated by serum phosphate of 1.4 mg/dl, intactPTH;29 pg/ml, TmP/GFR;1.14 mg/dl, and fibroblast growth factor 23 (FGF23) increased to 713 pg/ml, tumor-induced osteomalacia (TIO) was diagnosed. Systemic venous sampling of FGF23 performed for locating the tumor revealed an elevated level of 1600 pg/ml in the right femoral vein, and MRI of the right leg showed a tumor 26mm in size on the dorsal side of the right knee. In June 2011, the tumor in the right knee was surgically resected. Hypophosphatemia promptly improved following surgery, and FGF23 also returned to normal. The patient became able to walk independently and was able to reintegrate into society. The pathological diagnosis was phosphaturic mesenchymal tumor. However, serum phosphate again decreased in April 2013 to 2.6 mg/dl. FGF23 also increased to 221 pg/ml. Leg MRI was performed, and a tumor 25 mm in size was again detected on the dorsal side of the right knee. FDG PET/CT revealed significant uptake that was consistent with the tumor on the dorsal side of the right knee, as well as multiple nodules seen in both lung fields on CT. The tumor in the right knee was surgically resected in September 2013. However, FGF23, which was elevated to 1500 pg/ml preoperatively, remained high postoperatively at 950 pg/ml. Hypophosphatemia also did not improve, and phosphate treatment is being continued. Because she tested positive for somatostatin receptor 2 on RT-PCR using a resected sample, treatment using a somatostatin analog is being considered.

Summary: Malignant TIO is extremely rare, and no effective treatment exists for this condition. We herein report our experience with a case of malignant TIO in which recurrence and metastases to both lungs were observed two years after tumor resection.

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