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Oncogenic osteomalacia: two case reports with surprisingly different outcomes

  • Osteoporotic Fracture Management
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Abstract

Oncogenic osteomalacia is a rare paraneoplastic syndrome of acquired hypophosphatemic osteomalacia, resulting from a deficit in renal tubular phosphate reabsorption, in which fibroblast growth factor 23 (FGF23) seems to be implicated. This condition is usually associated with a phosphaturic mesenchymal tumor of mixed connective tissue located in the bone or soft tissue. The clinical and the radiologic findings are the same as those seen in osteomalacia, and the biochemical features include renal phosphate loss, low serum phosphate and 1,25-(OH)2 vitD3 levels, increased alkaline phosphatase, and normal calcium, PTH, calcitonin, 25-OH-vitD3 and 25,25-(OH)2 vitD3. We present two cases of oncogenic osteomalacia associated with phosphaturic mesenchymal tumors, which were histologically similar, but presented a completely different evolution. In the first patient, the tumor developed on the sole of the foot. Following removal of the mass, the symptoms resolved and biochemical and radiological parameters returned to normal. However, in the second patient, a liver tumor developed and resection did not resolve the disease. Multiple lesions appeared in several locations during follow-up. This disease usually remits with complete tumor resection. Nevertheless, if this is not possible, oral treatment with phosphate, calcium and calcitriol can improve the symptoms. If scintigraphy of the tumor shows octreotide receptors, patients may respond partially to therapy with somatostatin analogs, with stabilization of the lesion.

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References

  1. Braunwald E, Fauci AS, Dennis LK, Hauser SL, Longo DL, Jameson JL (2002) Principios de medicina interna, 15th edn. McGraw-Hill, Madrid, p 2568, pp 2574–2576

  2. Carpenter TO (2003) Oncogenic osteomalacia: a complex dance of factors [editorial]. N Engl J Med 348(17):1705–1708

    Article  PubMed  Google Scholar 

  3. Cheung FM, Ma L, Wu WC, Siu TH, Choi PT, Tai YP (2006) Oncogenic osteomalacia associated with an occult phosphaturic mesenchymal tumour: clinico-radiologico-pathological correlation and ultrastructural studies. Hong Kong Med J 12(4):319–321

    PubMed  CAS  Google Scholar 

  4. Felig P, Frohman LA (2001) Endocrinology and metabolism, 4th edn. McGraw-Hill, New York, pp 1409–1423

    Google Scholar 

  5. Folpe AL, Fanburg-Smith JC, Billings SD, Bisceglia M, Bertoni F, Cho JY, Econs MJ, Inwards CY, Jan de Beur SM, Mentzel T, Montgomery E, Michal M et al (2004) Most osteomalacia-associated mesenchymal tumors are a single histopathologic entity: an analysis of 32 cases and a comprehensive review of the literature. Am J Surg Pathol 28(1):1–30

    Article  PubMed  Google Scholar 

  6. Huang QL, Feig DS, Blackstein ME (2000) Development of tertiary hyperparathyroidism after phosphate supplementation in oncogenic osteomalacia. J Endocrinol Invest 23(4):263

    PubMed  CAS  Google Scholar 

  7. Jonsson KB, Zahradnik R, Larsson T, White KE, Sugimoto T, Imanishi Y, Yamamoto T, Hampson G, Koshiyama H, Ljunggren O, Oba K et al (2003) Fibroblast growth factor 23 in oncogenic osteomalacia and X-linked hypophosphatemia. N Engl J Med 348(17):1656–1663

    Article  PubMed  CAS  Google Scholar 

  8. Larsen PR, Kronenberg HM, Melmed S, Polonsky KS (2003) Endocrinology, 10th edn. Saunders, Philadelphia, pp 1349–1351, 1393–1395 and 1847

  9. Park YK, Unni KK, Beabout JW, Hodgson SF (1994) Oncogenic osteomalacia: a clinicopathologic study of 17 bone lesions. J Korean Med Sci 9(4):289–298

    PubMed  CAS  Google Scholar 

  10. Seufert J, Ebert K, Müller J et al (2001) Octreotide therapy for tumor-induced osteomalacia. N Engl J Med 345:1883–1888

    Article  PubMed  CAS  Google Scholar 

  11. Shane E, Parisien M, Henderson JE, Dempster DW, Feldman F, Hardy MA, Tohme JF, Karaplis AC, Clemens TL (1997) Tumor-induced osteomalacia: clinical and basic studies. J Bone Miner Res 12(9):1502–1511

    Article  PubMed  CAS  Google Scholar 

  12. Stewart I, Roddie C, Gill A, Clarkson A, Mirams M, Coyle L, Ward C, Clifton-Bligh P, Robinson BG, Mason RS, Clifton-Bligh RJ (2006) Elevated serum FGF23 concentrations in plasma cell dyscrasias. Bone 39(2):369–376

    Article  PubMed  CAS  Google Scholar 

  13. Tartaglia F, Minisola S, Sgueglia M, Blasi S, Brunelli D, Degli Effetti E, Maturo A, Cola A, Custureri F, Campana FP (2006) Tumor-induced hypophosphatemic osteomalacia associated with tertiary hyperparathyroidism: a case report. G Chir 27(1–2):9–13

    PubMed  CAS  Google Scholar 

  14. Weidner N, Bar RS, Weiss D, Strottmann P (1985) Neoplastic pathology of oncogenic osteomalacia/rickets. Cancer 55:1691–1705

    Article  PubMed  CAS  Google Scholar 

  15. Weidner N, Santa Cruz D (1987) Phosphaturic mesenchymal tumors: a polymorphous group causing osteomalacia or rickets. Cancer 59:1442–1454

    Article  PubMed  CAS  Google Scholar 

  16. Yeung SJ, McCutcheon IE, Schultz P, Gagel RF (2000) Use of long-term intravenous phosphate infusion in the palliative treatment of tumor-induced osteomalacia. J Clin Endocrinol Metab 85(2):549–555

    Article  PubMed  CAS  Google Scholar 

  17. Zimering MB, Caldarella FA, White KE, Econs MJ (2005) Persistent tumor-induced osteomalacia confirmed by elevated postoperative levels of serum fibroblast growth factor-23 and 5-year follow-up of bone density changes. Endocr Pract 11(2):108–114

    PubMed  Google Scholar 

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Correspondence to Roberto Seijas.

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Seijas, R., Ares, O., Sierra, J. et al. Oncogenic osteomalacia: two case reports with surprisingly different outcomes. Arch Orthop Trauma Surg 129, 533–539 (2009). https://doi.org/10.1007/s00402-008-0808-2

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  • DOI: https://doi.org/10.1007/s00402-008-0808-2

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