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Long-term bone mineral density changes after surgical cure of patients with tumor-induced osteomalacia

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Abstract

Summary

This paper reports our personal experience filling the gap regarding changes of bone mineral density after surgical treatment in patient suffering from tumor-induced osteomalacia.

Introduction

No systematic data are available regarding long-term bone mineral density (BMD) changes after surgical cure of patients with tumor-induced osteomalacia.

Methods

From October 2001 through April 2018, we studied 10 consecutive patients (mean age ± SD, 45.5 ± 13.8 years; 5 males and 5 females) with tumor-induced osteomalacia. We evaluated BMD when initially presented at our Center and after surgical removal of the tumor.

Results

Basal BMD and corresponding Z-score values (mean values ± SD) measured by DXA were as follows: L1–L4 = 0.692 ± 0.15 g/cm2, Z-score = − 2.80 ± 1.60; femur neck 0.447 ± 0.10 g/cm2, Z-score = − 2.66 ± 0.93; total femur = 0.450 ± 0.08 g/cm2, Z-score = −3.04 ± 0.85). Furthermore, Trabecular Bone Score (TBS) was evaluated in three patients (basal values, 0.990 ± 0.32). Seven patients were intermittently followed after surgical excision of the tumor while supplemented with cholecalciferol and calcium salts; the remaining three were lost to follow-up. There was a striking increase of BMD values that peaked at 26.7 ± 6.50 months: L1–L4 = 1.289 ± 0.247 g/cm2, p < 0.001, Z-score + 1.75 ± 1.42; femur neck = 0.890 ± 0.235 g/cm2, p = 0.028, Z-score = + 0.50 ± 1.40; total femur = 0.834 ± 0.150 g/cm2, p = 0.005, Z-score = − 0.74 ± 1.14. In patients with the greatest bone involvement at lumbar site, there was a striking increase of an average 1.5% (p < 0.01) in respect to baseline Z-score value for each additional month of observation during the first 2–3 years post-surgery. An improvement of trabecular microarchitecture was also documented (TBS, 1.255 ± 0.16).

Conclusion

This is the first case series documenting an impressive increase of BMD at both lumbar and femoral sites, together with an improvement of trabecular microarchitecture as documented by TBS. This is the consequence of huge mineralization of the large amount of osteoid tissue after resolution of the disease.

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References

  1. Minisola S, Peacock M, Fukumoto S, Cipriani C, Pepe J, Tella SH, Collins MT (2017) Tumour-induced osteomalacia. Nat Rev Dis Primers 3:17044

    Article  Google Scholar 

  2. Boland JM, Tebben PJ, Folpe AL (2018) Phosphaturic mesenchymal tumors: what an endocrinologist should know. J Endocrinol Investig 41:1173–1184

    Article  CAS  Google Scholar 

  3. Lee JC, Jeng YM, Su SY, Wu CT, Tsai KS, Lee CH, Lin CY, Carter JM, Huang JW, Chen SH, Shih SR, Mariño-Enríquez A, Chen CC, Folpe AL, Chang YL, Liang CW (2015) Identification of a novel FN1-FGFR1 genetic fusion as a frequent event in phosphaturic mesenchymal tumour. J Pathol 235:539–545

    Article  CAS  Google Scholar 

  4. Sapir-Koren R, Livshits G (2014) Bone mineralization is regulated by signaling cross talk between molecular factors of local and systemic origin: the role of fibroblast growth factor 23. Biofactors 40:555–568

    Article  CAS  Google Scholar 

  5. Piemonte S, Romagnoli E, Cipriani C, De Lucia F, Pilotto R, Diacinti D, Pepe J, Minisola S (2014) Six-year follow-up of a characteristic osteolytic lesion in a patient with tumor-induced osteomalacia. Eur J Endocrinol 170:K1–K4

    Article  CAS  Google Scholar 

  6. Umphrey LG, Whitaker MD, Bosch EP, Cook CB (2007) Clinical and bone density outcomes of tumor-induced osteomalacia after treatment. Endocr Pract 13:458–462

    Article  Google Scholar 

  7. 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 11:108–114

    Article  Google Scholar 

  8. Walton RJ, Bijvoet OL (1975) Nomogram for derivation of renal threshold phosphate concentration. Lancet 2:309–310

    Article  CAS  Google Scholar 

  9. Ferrone F, Pepe J, Danese VC et al (2019) The relative influence of serum ionized calcium and 25-hydroxyvitamin D in regulating PTH secretion in healthy subjects. Bone 125:200–206

    Article  CAS  Google Scholar 

  10. Pedrazzoni M, Girasole G, Bertoldo F, Bianchi G, Cepollaro C, del Puente A, Giannini S, Gonnelli S, Maggio D, Marcocci C, Minisola S, Palummeri E, Rossini M, Sartori L, Sinigaglia L (2003) Definition of a population-specific DXA reference standard in Italian women: the Densitometric Italian normative study (DINS). Osteoporos Int 14:978–982

    Article  CAS  Google Scholar 

  11. Cipriani C, Pepe J, Silva BC et al (2018) Comparative effect of rhPTH(1-84) on bone mineral density and trabecular bone score in hypoparathyroidism and postmenopausal osteoporosis. J Bone Miner Res 33:2132–2139

    Article  CAS  Google Scholar 

  12. Colangelo L, Cipriani C, Pepe J, Corsi A, Sonato C, Follacchio G, Cilli M, Gianni W, Ferrone F, Moreschini O, Fitzpatrick LA, Minisola S (2018) A challenging case of tumor-induced osteomalacia: pathophysiological and clinical implications. Calcif Tissue Int 103:465–468

    Article  CAS  Google Scholar 

  13. Spence HM (1984) The life and death of captain Charles Martell and kidney stone disease. J Urol 132:1204–1207

    Article  CAS  Google Scholar 

  14. Beck L, Karaplis AC, Amizuka N, Hewson AS, Ozawa H, Tenenhouse HS (1998) Targeted inactivation of Npt2 in mice leads to severe renal phosphate wasting, hypercalciuria, and skeletal abnormalities. Proc Natl Acad Sci U S A 95:5372–5377

    Article  CAS  Google Scholar 

  15. Compston JE, McClung MR, Leslie WD (2019) Osteoporosis. Lancet 393:364–376

    Article  CAS  Google Scholar 

  16. Minisola S, Cipriani C, Occhiuto M, Pepe J (2017) New anabolic therapies for osteoporosis. Intern Emerg Med 12:915–921

    Article  Google Scholar 

  17. Kendler DL, Marin F, Zerbini CAF, Russo LA, Greenspan SL, Zikan V, Bagur A, Malouf-Sierra J, Lakatos P, Fahrleitner-Pammer A, Lespessailles E, Minisola S, Body JJ, Geusens P, Möricke R, López-Romero P (2018) Effects of teriparatide and risedronate on new fractures in post-menopausal women with severe osteoporosis (VERO): a multicentre, double-blind, double-dummy, randomised controlled trial. Lancet 391:230–240

    Article  CAS  Google Scholar 

  18. Bhambri R, Naik V, Malhotra N, Taneja S, Rastogi S, Ravishanker U, Mithal A (2006) Changes in bone mineral density following treatment of osteomalacia. J Clin Densitom 9:120–127

    Article  Google Scholar 

  19. Carpenter TO, Whyte MP, Imel EA et al (2018) Burosumab therapy in children with X-linked hypophosphatemia. N Engl J Med 378:1987–1998

    Article  Google Scholar 

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Correspondence to L. Colangelo.

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Colangelo, L., Pepe, J., Nieddu, L. et al. Long-term bone mineral density changes after surgical cure of patients with tumor-induced osteomalacia. Osteoporos Int 31, 1383–1387 (2020). https://doi.org/10.1007/s00198-020-05369-1

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  • DOI: https://doi.org/10.1007/s00198-020-05369-1

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