68Ga DOTA-TATE PET/CT allows tumor localization in patients with tumor-induced osteomalacia but negative 111In-octreotide SPECT/CT

Breer, S.; Brunkhorst, Thomas; Beil, Frank Timo; Peldschus, Kersten; Heiland, Max; Klutmann, Susanne; Barvencik, Florian; Zustin, Jozef; Gratz, K. F.; Amling, Michael

doi:10.1016/j.bone.2014.04.016

Cited by 77 publications

(103 citation statements)

References 36 publications

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“…8, 15 Imaging methods such as gallium-68 ( 68 Ga) DOTA-NOC, 68 Ga DOTA-TOC, and 68 Ga DOTA-TATE positron emission tomography/CT show promising first results in patients with TIO and negative octreotide scans. 16 Our challenge was on one hand the rapidly growing CGCG and on the other hand the consequences of FGF23 overproduction. The most common treatment of CGCG has been surgical curettage, with high recurrence rates.…”

Section: Figurementioning

confidence: 99%

Tumor-Induced Rickets in a Child With a Central Giant Cell Granuloma: A Case Report

et al. 2015

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Tumor-induced osteomalacia/rickets is a rare paraneoplastic disorder associated with a tumor-producing fibroblast growth factor 23 (FGF23). We present a child with symptoms of rickets as the first clinical sign of a central giant cell granuloma (CGCG) with high serum levels of FGF23, a hormone associated with decreased phosphate resorption. A 3-year-old boy presented with a limp and 6 months later with painless growth of the jaw. On examination gingival hypertrophy and genu varum were observed. Investigations revealed hypophosphatemia, normal 1,25 and 25 (OH) vitamin D, and high alkaline phosphatase. An MRI showed an osteolytic lesion of the maxilla. Radiographs revealed typical rachitic findings. Incisional biopsy of the tumor revealed a CGCG with mesenchymal matrix. The CGCG was initially treated with calcitonin, but the lesions continued to grow, making it necessary to perform tracheostomy and gastrostomy. One year after onset the hyperphosphaturia worsened, necessitating increasing oral phosphate supplements up to 100 mg/kg per day of elemental phosphorus. FGF23 levels were extremely high. Total removal of the tumor was impossible, and partial reduction was achieved after percutaneous computed tomography-guided radiofrequency, local instillation of triamcinolone, and oral propranolol. Compassionate use of cinacalcet was unsuccessful in preventing phosphaturia. The tumor slowly regressed after the third year of disease; phosphaturia improved, allowing the tapering of phosphate supplements, and FGF23 levels normalized. Tumor-induced osteomalacia/rickets is uncommon in children and is challenging for physicians to diagnose. It should be suspected in patients with intractable osteomalacia or rickets. A tumor should be ruled out if FGF23 levels are high.Tumor-induced osteomalacia/rickets (TIO) is a rare paraneoplastic disorder caused by overproduction of fibroblast growth factor 23 (FGF23) from the responsible tumors. 1,2 FGF23 is a hormone associated with decreased resorption of phosphate that causes hyperphosphaturia, leading to osteomalacia or rickets. [3][4][5] Tumors are typically benign, small, and with mesenchymal origin. 2,[6][7][8] We report the first pediatric case of TIO in association with a central giant cell granuloma (CGCG). 9 Our case is unique because the child presented with clinical signs of rickets as the first sign of CGCG with high FGF23 serum levels. PATIENT PRESENTATIONA previously healthy 3-year-old boy with no history of trauma or other joint problems presented with an increasing limp. Initial radiographs and basic laboratory studies, including calcium and phosphate, were normal. He initially received a diagnosis of

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Section: Figurementioning

confidence: 99%

Tumor-Induced Rickets in a Child With a Central Giant Cell Granuloma: A Case Report

et al. 2015

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“…In a different study involving 5 patients who had both 68 Ga DOTATATE PET/CT scan and In111 octreoscan, 68 Ga DOTATATE PET/CT was significant more sensitive in the detection of culprit tumors. 21 However, direct comparison between PETbased and gamma camera-based tracer in the same patient population in future study is necessary to determine which somatostatin-receptor imaging modality has the highest accuracy in this clinical setting.…”

Section: Discussionmentioning

confidence: 99%

“…20,21,[27][28][29] However, because of the lack of outcome of patients with negative studies and small number of the patients involved, a diagnostic accuracy of 68 Ga DOTATATE or 68 Ga DOTANOC PET/CT could not be derived from these case series. The results from our investigation indicated that 68 Ga DOTATATE PET/CT is very effective in the evaluation of hypophosphatemic patients who had typical symptoms of TIO.…”

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confidence: 81%

68Ga DOTATATE PET/CT is an Accurate Imaging Modality in the Detection of Culprit Tumors Causing Osteomalacia

Zhang

Zhu

Zhong³

et al. 2015

Clinical Nuclear Medicine

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“…Magnetic resonance imaging, indium-labeled octreotide scan and PET-FDG are the additional imaging that has been recommended for patients without obvious tumor (5). Because of limitation in conventional imaging for detection of small tumors, somatostatin-receptor imaging such as octreotide scintigraphy or octreotide SPECT/CT are recommended in patients with hypophosphatemic osteomalacia and normal primary imaging studies (6). However a large number of tumors remained undetected by octreotide imaging.…”

Section: Introductionmentioning

confidence: 99%

“…Consequently studies focused on different SSTR imaging methods such as 68Ga DOTA-TATE PET/CT. It is an effective and promising diagnostic imaging in undetectable small tumors (6). Various factors including PTH and vitamin D regulate phosphate absorption in the intestine and phosphate reabsorption in kidney.…”

Section: Introductionmentioning

confidence: 99%

Glomus tumor as a cause of oncogenic osteomalacia

Dehghani

Dabbaghmanesh

Khalafi-Nezhad

et al. 2017

ccmbm

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SummaryMany tumors that occasionally are benign in origin causes hypophosphatemic osteomalacia. Here we present a case of glomus tumor in a 59-year-old man with oncogenic osteomalacia. Diagnosis was made after observation of abnormal increase activity in octreotide scan. The magnetic resonance imaging showed a round lesion in left ankle joint. Surgical excision of tumor was curative and all symptoms and intractable hypophosphatemia improved after few weeks.

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68Ga DOTA-TATE PET/CT allows tumor localization in patients with tumor-induced osteomalacia but negative 111In-octreotide SPECT/CT

Cited by 77 publications

References 36 publications

Tumor-Induced Rickets in a Child With a Central Giant Cell Granuloma: A Case Report

Tumor-Induced Rickets in a Child With a Central Giant Cell Granuloma: A Case Report

68Ga DOTATATE PET/CT is an Accurate Imaging Modality in the Detection of Culprit Tumors Causing Osteomalacia

Glomus tumor as a cause of oncogenic osteomalacia

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