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Persistent phosphaturic mesenchymal tumor causing tumor-induced osteomalacia treated with image-guided ablation

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Abstract

Phosphaturic mesenchymal tumors (PMTs) can present with vague symptoms of diffuse bone pain with pathologic fractures that often lead to a delayed diagnosis. We present a 60-year-old patient with a PMT that was persistently hypophosphatemic after resection, who was then successfully treated with cryoablation of the tumor. Tumor-induced osteomalacia (TIO) is a rare cause of hypophosphatemia characterized by vague symptoms of gradual muscle weakness and diffuse bone pain with pathologic fractures that often lead to a delayed diagnosis. This condition is usually caused by benign phosphaturic mesenchymal tumors (PMTs). Here, we present a case of persistent PMT after surgical resection treated with image-guided ablation. We present the patient’s clinical examinations and laboratory findings (phosphorus, 1,25 (OH)2d, FGF-23, Intact PTH). Representative histologic images of a PMT are also presented. A 61-year-old male was evaluated for persistent hypophosphatemia and presumed osteomalacia. Six years earlier, he underwent surgical excision of a left ischial mass after presenting with TIO. The pathology was consistent with a PMT; however, hypophosphatemia persisted suggesting incomplete resection. He was treated with calcitriol and phosphate salts. A PET Ga68 dotatate scan of the patient revealed an avid left ischial mixed lytic and sclerotic lesions with marked amount of radiotracer uptake, suggesting persistent tumor. The patient was resistant to re-excision of the tumor due to the extended recovery period from his prior surgery and was treated instead with cryoablation of the tumor. His biochemical findings of hypophosphatemia and elevated FGF23 resolved after the ablation and have remained normal for 5 months after surgery. In patients with TIO, wide surgical excision is the treatment of choice. When this is not possible, image-guided ablation is an alternative therapeutic option.

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Funding

This research was funded by Medical College of Wisconsin’s Department of Orthopedic Surgery.

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Authors and Affiliations

  1. Department of Orthopaedic Surgery, Medical College of Wisconsin, Wauwatosa, WI, 53213, USA

    J. C. Horng, E. Van Eperen & A. N. Wooldridge

  2. Department of Vascular and Interventional Radiology, Medical College of Wisconsin, Wauwatosa, WI, 53213, USA

    S. Tutton

  3. Department of Pathology, Medical College of Wisconsin, Milwaukee, WI, USA

    R. Singh

  4. Department of Medicine (Endocrinology), Medical College of Wisconsin, Milwaukee, WI, USA

    J. L. Shaker

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  1. J. C. Horng
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  2. E. Van Eperen
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  3. S. Tutton
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  4. R. Singh
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  5. J. L. Shaker
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  6. A. N. Wooldridge
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Contributions

All authors provided substantial contributions to conception and design, or acquisition of data, or analysis and interpretation of data in preparing this manuscript.

Corresponding author

Correspondence to A. N. Wooldridge.

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Conflict of interest

Dr Sean Tutton is a consultant for Galil Medical, consultant for Stryker, consultant for IZI Medical, and provides research support for Siemens. Dr Joseph Shaker receives authorship royalties from McGraw-Hill and is a consultant for Shire, Alexion, and Ultragenyx. Jonathan Horng, Erik Van Eperen, Reena Singh, and Adam Wooldridge declare that they have no conflicts of interest.

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Supplementary information

Supplementary Figure 1

MRI demonstrates a heterogeneous lesion at the posterior medial aspect of the acetabulum extending into the ischium with a convex margin at the medial aspect of the acetabulum which likely corresponds with the area of cortical deficiency on the prior CT. There is enhancement in this location as well as at the inferior aspect of the lesion corresponding with the more lytic areas on the CT performed a few hours prior. (PNG 307 kb)

Supplementary Figure 2

CT multi-planar reformat obtained in real-time during biopsy and ablation demonstrating the coaxial bone trochar and cryoablation probe positioned in the center of the lesion. Navigational lines are not shown so as not to obscure the lesion. (PNG 181 kb)

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Horng, J.C., Van Eperen, E., Tutton, S. et al. Persistent phosphaturic mesenchymal tumor causing tumor-induced osteomalacia treated with image-guided ablation. Osteoporos Int 32, 1895–1898 (2021). https://doi.org/10.1007/s00198-020-05795-1

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  • DOI: https://doi.org/10.1007/s00198-020-05795-1

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