Skip to main content

Advertisement

SpringerLink
Log in

Parathyroid-hormone-related protein-mediated hypercalcemia in benign congenital mesoblastic nephroma

  • Brief Report
  • Published:
Pediatric Nephrology Aims and scope Submit manuscript

Abstract

Parathyroid hormone-related protein (PTHrP) mediated hypercalcemia of malignancy is rare in children, and even more so in the setting of a benign tumor. We report two infants with PTHrP-mediated hypercalcemia secondary to congenital mesoblastic nephroma and their outcome after removal of the benign tumor. Pre-operatively hypercalcemia was corrected with saline hydration, furosemide, calcitonin and/ or pamidronate. Following resection of the tumor serum PTHrP normalized. Immunohistochemical staining of tumor cells was positive for PTHrP. Post-operatively the infants developed elevated serum parathyroid hormone with low- normal serum Ca and P, and undetectable urinary Ca and P, probably due to their movement into bone. Children needed treatment with calcitriol, Ca and P supplementation for 6-12 weeks until PTH normalized and urinary Ca and P were detected, suggesting bone replenishment. We conclude that benign congenital mesoblastic nephroma can secrete PTHrP that can cause severe hypercalcemia; and following excision one should anticipate the development of a transient modified “hungry bone”-like condition requiring Ca, P and calcitriol therapy for several weeks accompanied by careful monitoring of mineral homeostasis.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Log in via an institution

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Fig. 1

References

  1. Moseley JM, Kubota M, Diefenbach-Jagger H, Wettenhall RE, Kemp BE, Suva LJ, Rodda CP, Ebeling PR, Hudson PJ, Zajac JD, Martin TJ (1987) Parathyroid hormone-related protein purified from a human lung cancer cell line. Proc Natl Acad Sci USA 84:5048–5052

    Article  PubMed  CAS  Google Scholar 

  2. Mangin M, Ikeda K, Dreyer BE, Broadus AE (1989) Isolation and characterization of the human parathyroid hormone-like peptide gene. Proc Natl Acad Sci USA 86:2408–2412

    Article  PubMed  CAS  Google Scholar 

  3. Suva LJ, Winslow GA, Wettenhall RE, Hammonds RG, Moseley JM, Diefenbach-Jagger H, Rodda CP, Kemp BE, Rodriguez H, Chen EY, Hudson PJ, Martin TJ, Wood WI (1987) A parathyroid hormone-related protein implicated in malignant hypercalcemia: cloning and expression. Science 237:893–896

    Article  PubMed  CAS  Google Scholar 

  4. Strewler GJ (2000) The physiology of parathyroid-hormone related protein. N Engl J Med 342:177–185

    Article  PubMed  CAS  Google Scholar 

  5. Knecht T, Behling C, Burton D, Glass C, Deftos L (1996) The humoral hypercalcemia of benignancy. A newly appreciated syndrome. Am J Clin Pathol 105:487–492

    PubMed  CAS  Google Scholar 

  6. Dagdelen S, Kalan I, Gurlek A (2008) Humoral hypercalcemia of benignancy secondary to parathyroid hormone-related protein secreting uterine leiomyoma. Am J Med Sci 335:407–408

    Article  PubMed  Google Scholar 

  7. Herring R, Laji K (2008) Humoral hypercalcaemia of benignancy. A case report. QJM 101:329–330

    Article  PubMed  CAS  Google Scholar 

  8. Ravakhah K, Gover A, Mukunda BN (1999) Humoral hypercalcaemia associated with uterine fibroid. Ann Intern Med 130:702

    PubMed  CAS  Google Scholar 

  9. Bilici A, Doventas A, Karadag B, Hekim N, Tezcan V (2004) Hypercalcemia associated with a uterine leiomyoma: a case report and review of the literature. Gynecol Oncol 93:269–271

    Article  PubMed  Google Scholar 

  10. Rathaus M, Bernheim JL, Griffel B, Bernheim J, Taragan R, Gutman A (1979) Leiomyoma of the small bowel with hypercalcaemia: presence of a substance with parathormone activity (author's transl). Nouv Presse Méd 8:3245–3246

    PubMed  CAS  Google Scholar 

  11. Kimura S, Nishimura Y, Yamaguchi K, Nagasaki K, Shimada K, Uchida H (1990) A case of pheochromocytoma producing parathyroid hormone-related protein and presenting with hypercalcemia. J Clin Endocrinol Metab 70:1559–1573

    Article  PubMed  CAS  Google Scholar 

  12. Mune T, Katakami H, Kato Y, Yasuda K, Matsukura MKS (1993) Production and secretion of parathyroid hormone-related protein in pheochromocytoma: a participation of an alpha-adrenergic mechanism. J Clin Endocrinol Metab 76:757–762

    Article  PubMed  CAS  Google Scholar 

  13. Kutluk MT, Hazar V, Akyuz C, Varan A, Buyukpamukcu M (1997) Childhood cancer and hypercalcemia: report of a case treated with pamidronate. J Pediatr 130:828–831

    Article  PubMed  CAS  Google Scholar 

  14. Mahoney C, Cassady C, Weinberger E, Winters W, Benjamin D (1997) Humoral hypercalcemia due to an occult renal adenoma. Pediatr Nephrol 11:339–342

    Article  PubMed  CAS  Google Scholar 

  15. Danks JA, Ebeling PR, Hayman J, Chou ST, Moseley JM, Dunlop J, Kemp BE, Martin TJ (1989) Parathyroid hormone-related protein: immunohistochemical localization in cancers and in normal skin. J Bone Miner Res 4:273–278

    Article  PubMed  CAS  Google Scholar 

  16. McKay C, Furman WL (1993) Hypercalcemia complicating childhood malignancies. Cancer 72:256–260

    Article  PubMed  CAS  Google Scholar 

  17. Jayabose S, Iqbal K, Newman L, San Filippo JA, Davidian MM, Noto R, Sagel I (1998) Hypercalcemia in childhood renal tumors. Cancer 61:788–791

    Article  Google Scholar 

  18. Brasier AR, Nussbaum SR (1998) Hungry bone syndrome: clinical and biochemical predictors of its occurrence after parathyroid surgery. Am J Med 84:654–660

    Article  Google Scholar 

  19. Malloy PJ, Wang J, Srivastava T, Feldman D (2010) Hereditary 1, 25-dihydroxyvitamin D-resistant rickets with alopecia resulting from a novel missense mutation in the DNA-binding domain of the vitamin D receptor. Mol Genet Metab 99:72–79

    Article  PubMed  CAS  Google Scholar 

Download references

Acknowledgement

The study was supported by the Sam and Helen Kaplan Research Fund in Pediatric Nephrology and Eric McClure Research Fund in Pediatric Bone and Mineral Disorders.

Author information

Authors and Affiliations

  1. Section of Nephrology, Children’s Mercy Hospital and University of Missouri at Kansas City, 2401 Gillham Road, Kansas City, MO, 64108, USA

    Tarak Srivastava & Uri S. Alon

  2. Department of Pathology and Laboratory Medicine, Children’s Mercy Hospital and University of Missouri at Kansas City, Kansas City, MO, USA

    Alexander Kats

  3. Bone Cell Biology and Disease Unit, St. Vincent’s Institute, The University of Melbourne, Fitzroy, VIC, Australia

    T. John Martin & Suelli Pompolo

Authors
  1. Tarak Srivastava
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Alexander Kats
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. T. John Martin
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Suelli Pompolo
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. Uri S. Alon
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to Tarak Srivastava.

Additional information

Tarak Srivastava, MD and Alexander Kats, MD contributed equally to this work.

Rights and permissions

Reprints and permissions

About this article

Cite this article

Srivastava, T., Kats, A., Martin, T.J. et al. Parathyroid-hormone-related protein-mediated hypercalcemia in benign congenital mesoblastic nephroma. Pediatr Nephrol 26, 799–803 (2011). https://doi.org/10.1007/s00467-010-1728-2

Download citation

  • Received:

  • Revised:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00467-010-1728-2

Keywords

Search

Navigation