Literature DB >> 28181822

Oncogenic osteomalacia: role of Ga-68 DOTANOC PET/CT scan in identifying the culprit lesion and its management.

Deepa Singh1, Aditi Chopra2, Mudalsha Ravina1, Srikant Kongara2,3, Eesh Bhatia2, Narvesh Kumar1, Sushil Gupta2, Subhash Yadav2, Preeti Dabadghao2, Rajnikant Yadav4, Veeresh Dube1, Utham Kumar1, Manish Dixit1, Sanjay Gambhir1.   

Abstract

OBJECTIVE: The aim of this study was to evaluate the role of 68Ga-DOTANOC positron emission tomography (PET)/CT scan in localization of culprit lesion for biopsy and required intervention [surgical excision/radiofrequency ablation (RFA)] in patients with long-standing oncogenic osteomalacia (OOM)/tumour-induced osteomalacia.
METHODS: 17 patients (8 males and 9 females) underwent 68Ga-DOTANOC PET/CT scan. The patients referred with clinical and biochemical evidence of hypophosphatemia and raised fibroblast growth factor-23. Qualitative and semi-quantitative parameters were used to identify culprit lesions.
RESULTS: 68Ga-DOTANOC PET/CT scan revealed 52 lesions in 17 patients, and 37/52 of these lesions were tracer avid. 26/37 lesions were non-specific focal tracer-avid skeletal lesions (fractures or degenerative changes). 11/37 tracer-avid skeletal lesions present in 9 patients (3 lesions in 1 patient and 1 each in rest of the 8 patients) were highly suspicious for culprit lesions in view of high maximum standardized uptake value (SUVmax) (range 1.5-15.4; mean 7.0 ± 4.6), lesion size (0.9-5.0 cm; mean 3.3 ± 1.5) and associated soft-tissue component. During subsequent imaging with CT/MRI, 7/9 patients showed concordant lesions which were excised or biopsied and histopathologically verified as phosphaturic mesenchymal tumours. Surgical excision was resorted to in most of the detected lesions, and RFA was performed in one patient.
CONCLUSION: There is some overlap in SUVmax between fracture-/bone-associated lesions and culprit lesions with a tendency of most non-culprit lesions to have lower SUVmax and no associated soft-tissue component. In such scenario, intensely tracer-avid, larger non-fracture lesions with soft-tissue component may lead to identification of culprit lesion among multiple lesions. Following detection of culprit lesion, surgical removal is the best treatment. RFA is alternative to surgery in cases where surgery is not possible owing to osteopenia/poor bone health. Advances in knowledge: The main challenge in patients of long-standing OOM is the presence of multiple skeletal lesions (both tumour- or tracer-avid fractures), and it is confusing to identify culprit lesion. This was noted in our study with 68Ga-DOTANOC and has not been mentioned in studies performed with 68Ga-DOTATATE/TOC PET/CT. In such scenario, 68Ga-DOTANOC PET/CT needs to be reviewed and read thoroughly to localize the culprit lesion out of the multiple tracer-avid lesions.

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Year:  2017        PMID: 28181822      PMCID: PMC5605070          DOI: 10.1259/bjr.20160811

Source DB:  PubMed          Journal:  Br J Radiol        ISSN: 0007-1285            Impact factor:   3.039


  29 in total

1.  Usefulness of 201Tl and 99mTc MIBI scintigraphy in a case of oncogenic osteomalacia.

Authors:  Takao Kimizuka; Yutaka Ozaki; Yukiharu Sumi
Journal:  Ann Nucl Med       Date:  2004-02       Impact factor: 2.668

2.  Tumor-induced osteomalacia.

Authors:  Suzanne M Jan de Beur
Journal:  JAMA       Date:  2005-09-14       Impact factor: 56.272

3.  Oncogenic osteomalacia: exact tumor localization by co-registration of positron emission and computed tomography.

Authors:  Eric Hesse; Eckart Moessinger; Herbert Rosenthal; Florian Laenger; Georg Brabant; Thorsten Petrich; Klaus F Gratz; Leonard Bastian
Journal:  J Bone Miner Res       Date:  2007-01       Impact factor: 6.741

4.  Localisation of mesenchymal tumours by somatostatin receptor imaging.

Authors:  Suzanne M Jan de Beur; Elizabeth A Streeten; A Cahid Civelek; Edward F McCarthy; Liliana Uribe; Stephen J Marx; Olufunmilayo Onobrakpeya; Lawrence G Raisz; Nelson B Watts; Michael Sharon; Michael A Levine
Journal:  Lancet       Date:  2002-03-02       Impact factor: 79.321

5.  Functional imaging in primary tumour-induced osteomalacia: relative performance of FDG PET/CT vs somatostatin receptor-based functional scans: a series of nine patients.

Authors:  Swati Jadhav; Rajeev Kasaliwal; Vikram Lele; Venkatesh Rangarajan; Piyush Chandra; Hina Shah; Gaurav Malhotra; Varsha S Jagtap; Sweta Budyal; Anurag R Lila; Tushar Bandgar; Nalini S Shah
Journal:  Clin Endocrinol (Oxf)       Date:  2014-03-12       Impact factor: 3.478

6.  DOTA-NOC, a high-affinity ligand of somatostatin receptor subtypes 2, 3 and 5 for labelling with various radiometals.

Authors:  Damian Wild; Jörg S Schmitt; Mihaela Ginj; Helmut R Mäcke; Bert F Bernard; Eric Krenning; Marion De Jong; Sandra Wenger; Jean-Claude Reubi
Journal:  Eur J Nucl Med Mol Imaging       Date:  2003-08-21       Impact factor: 9.236

7.  Phosphaturic mesenchymal tumors. A polymorphous group causing osteomalacia or rickets.

Authors:  N Weidner; D Santa Cruz
Journal:  Cancer       Date:  1987-04-15       Impact factor: 6.860

8.  Effective detection of the tumors causing osteomalacia using [Tc-99m]-HYNIC-octreotide (99mTc-HYNIC-TOC) whole body scan.

Authors:  Hongli Jing; Fang Li; Hongming Zhuang; Zhenghua Wang; Jian Tian; Xiaoping Xing; Jin Jin; Dingrong Zhong; Jingjing Zhang
Journal:  Eur J Radiol       Date:  2013-05-27       Impact factor: 3.528

9.  Oncogenic osteomalacia: localization of underlying peripheral mesenchymal tumors with use of Tc 99m sestamibi scintigraphy.

Authors:  Stephen F Hodgson; Bart L Clarke; Peter J Tebben; Brian P Mullan; William P Cooney; Thomas C Shives
Journal:  Endocr Pract       Date:  2006 Jan-Feb       Impact factor: 3.443

Review 10.  Tumor-induced osteomalacia with elevated fibroblast growth factor 23: a case of phosphaturic mesenchymal tumor mixed with connective tissue variants and review of the literature.

Authors:  Fang-Ke Hu; Fang Yuan; Cheng-Ying Jiang; Da-Wei Lv; Bei-Bei Mao; Qiang Zhang; Zeng-Qiang Yuan; Yan Wang
Journal:  Chin J Cancer       Date:  2011-11
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  13 in total

1.  Successful Localization Using 68Ga-DOTATOC PET/CT of a Phosphaturic Mesenchymal Tumor Causing Osteomalacia in a Patient with Concurrent Follicular Lymphoma.

Authors:  Sejin Ha; Sujin Park; Hyunji Kim; Heounjeong Go; Seung Hun Lee; Ji Yoon Choi; Jung Yong Hong; Jin-Sook Ryu
Journal:  Nucl Med Mol Imaging       Date:  2018-09-12

2.  Diagnostic performance and impact on patient management of 68Ga-DOTA-TOC PET/CT for detecting osteomalacia-associated tumours.

Authors:  Marie Paquet; Mathieu Gauthé; Jules Zhang Yin; Valérie Nataf; Ophélie Bélissant; Philippe Orcel; Christian Roux; Jean-Noël Talbot; Françoise Montravers
Journal:  Eur J Nucl Med Mol Imaging       Date:  2018-03-12       Impact factor: 9.236

Review 3.  Phosphaturic mesenchymal tumors: what an endocrinologist should know.

Authors:  J M Boland; P J Tebben; A L Folpe
Journal:  J Endocrinol Invest       Date:  2018-02-14       Impact factor: 4.256

4.  Extended Whole-body Ga-68 DOTATATE PET-CT in evaluating Tumour-Induced Osteomalacia: Case report and review of literature.

Authors:  Teik Hin Tan; Ew-Jun Chen; Ming Tsuey Chew; Ping Ching Chye; Ming Wong
Journal:  Nucl Med Mol Imaging       Date:  2021-04-19

5.  Usefulness of 68Ga-DOTATOC PET/CT to localize the culprit tumor inducing osteomalacia.

Authors:  Dong Yun Lee; Seung Hun Lee; Beom-Jun Kim; Wanlim Kim; Pil Whan Yoon; Sang Ju Lee; Seung Jun Oh; Jung-Min Koh; Jin-Sook Ryu
Journal:  Sci Rep       Date:  2021-01-19       Impact factor: 4.379

Review 6.  Ameloblastic Fibroodontoma of Mandible Causing Tumor Induced Osteomalacia: A Case Report with Review of 88 Phosphaturic Oral Neoplasms.

Authors:  Deepika Mishra; Harpreet Kaur; Ashu Seith Bhalla; Smita Manchanda; Jithin Sasikumar; Bhaskar Agarwal; Ajoy Roychoudhury
Journal:  Head Neck Pathol       Date:  2021-01-04

7.  CT and MR imaging features in phosphaturic mesenchymal tumor-mixed connective tissue: A case report.

Authors:  Zhenshan Shi; Yiqiong Deng; Xiumei Li; Yueming Li; Dairong Cao; Vikash Sahadeo Coossa
Journal:  Oncol Lett       Date:  2018-02-05       Impact factor: 2.967

8.  Phosphaturic mesenchymal tumors among elderly patients: a case report and review of literature.

Authors:  Zaina Adnan; David Nikomarov; Michal Weiler-Sagie; Noga Roguin Maor
Journal:  Endocrinol Diabetes Metab Case Rep       Date:  2019-05-03

9.  Tumor induced osteomalacia in head and neck region: single center experience and systematic review.

Authors:  Ravikumar Shah; Anurag R Lila; Ramteke-Swati Jadhav; Virendra Patil; Abhishek Mahajan; Sushil Sonawane; Puja Thadani; Anil Dcruz; Prathamesh Pai; Munita Bal; Subhada Kane; Nalini Shah; Tushar Bandgar
Journal:  Endocr Connect       Date:  2019-10       Impact factor: 3.335

Review 10.  Detection Rate of Culprit Tumors Causing Osteomalacia Using Somatostatin Receptor PET/CT: Systematic Review and Meta-Analysis.

Authors:  Marie Meyer; Marie Nicod Lalonde; Nathalie Testart; Mario Jreige; Christel Kamani; Sarah Boughdad; Barbara Muoio; Fabio Becce; Niklaus Schaefer; Christian Candrian; Luca Giovanella; John O Prior; Giorgio Treglia; Martin Riegger
Journal:  Diagnostics (Basel)       Date:  2019-12-18
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