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Parathyroid carcinoma presenting as normocalcemic hyperparathyroidism

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Abstract

Parathyroid carcinoma (PC) is a rare malignancy, with an indolent but progressive course. Long-term survival is largely dependent on the extent of the primary surgical resection. Hence, pre- or intraoperative suspicion of malignancy is of great importance. We describe the case of a 62-year-old woman with a 2-year history of asthenia and mental depression. Her past medical history was significant for osteoporosis. A diagnosis of primary normocalcemic hyperparathyroidism was established and the patient underwent surgery. PC was suspected intraoperatively because of the size and appearance of the parathyroid mass (a grayish, lobulated 3.5 cm mass). Thus, aggressive surgery (en bloc resection) was performed, along with bilateral neck exploration. Pathological examination of the specimens confirmed the suspicion of PC, demonstrating vascular invasion and extracapsular infiltration into adjacent soft tissue. Immunohistochemical staining revealed an elevated Ki-67 score (8.43%; cut-off value 5%). The mean area of silver-stained nucleolar organizer regions (AgNOR) was high (4.972 μm2), indicating an elevated proliferation rate. Serum calcium and parathyroid hormone levels normalized postoperatively, and the patient’s 5-year outcome was good. The present case provides evidence that parathyroid malignancy cannot be excluded a priori based on normocalcemic hyperparathyroidism, emphasizing the variability in clinical presentation. Moreover, Ki-67 expression and AgNOR analysis confirmed their additional value in complementing the histological evaluation of a parathyroid malignant mass.

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References

  1. Shane E (2001) Clinical review 122: parathyroid carcinoma. J Clin Endocrinol Metab 86:485–493

    Article  PubMed  CAS  Google Scholar 

  2. Mittendorf EA, McHenry CR (2005) Parathyroid carcinoma. J Surg Oncol 89:136–142

    Article  PubMed  CAS  Google Scholar 

  3. Marcocci C, Cetani F, Rubin MR, Silverberg SJ, Pinchera A, Bilezikian JP (2008) Parathyroid carcinoma. J Bone Min Res 23:1868–1880

    Article  Google Scholar 

  4. Hundahl SA, Fleming ID, Fremgen AM, Menck HR (1999) Two hundred eighty-six cases of parathyroid carcinoma treated in the U.S. between 1985–1995: a National Cancer Data Base Report. The American College of Surgeons Commission on Cancer and the American Cancer Society. Cancer 86:538–544

    Article  PubMed  CAS  Google Scholar 

  5. Witteveen JE, Haak HR, Kievit J, Morreau H, Romijn JA, Hamdy NA (2010) Challenges and pitfalls in the management of parathyroid carcinoma: 17-year follow-up of a case and review of the literature. Horm Cancer 1:205–214

    Article  PubMed  Google Scholar 

  6. Tuccari G, Abbona GC, Giuffrè G, Papotti M, Gasparri G, Barresi G, Bussolati G (2000) AgNOR quantity as a prognostic tool in hyperplastic and neoplastic parathyroid glands. Virchows Arch 437:298–303

    Article  PubMed  CAS  Google Scholar 

  7. Koea JB, Shaw JH (1999) Parathyroid cancer: biology and management. Surg Onc 8:155–165

    Article  CAS  Google Scholar 

  8. Fernandez-Ranvier GG, Jensen K, Khanafshar E, Quivey JM, Glastonbury C, Kebebew E, Duh QY, Clark OH (2007) Nonfunctioning parathyroid carcinoma: case report and review of literature. Endocr Pract 13:750–757

    PubMed  Google Scholar 

  9. Wilkins BJ, Lewis JS Jr (2009) Non-functional parathyroid carcinoma: a review of the literature and report of a case requiring extensive surgery. Head Neck Pathol 3:140–149

    Article  PubMed  Google Scholar 

  10. Silverberg SJ, Lewiecki EM, Mosekilde L, Peacock M, Rubin MR (2009) Presentation of asymptomatic primary hyperparathyroidism: proceedings of the Third International Workshop. J Clin Endocrinol Metab 94:351–365

    Article  PubMed  CAS  Google Scholar 

  11. Messerer CL, Bugis SP, Baliski C, Wiseman SM (2006) Normocalcemic parathyroid carcinoma: an unusual clinical presentation. World J Surg Oncol 4:1–5

    Article  Google Scholar 

  12. Lowe H, McMahon DJ, Rubin MR, Bilezikina JP, Silverberg J (2007) Normocalcemic primary hyperparathyroidism: further characterization of a new clinical phenotype. J Clin Endocrinol Metab 92:3001–3005

    Article  PubMed  CAS  Google Scholar 

  13. Sundelin K, Tullgren O, Farnebo LO (1994) Clinical course of metastatic parathyroid cancer. World J Surg 18:594–598

    Article  Google Scholar 

  14. Schantz A, Castleman B (1973) Parathyroid carcinoma: a study of 70 cases. Cancer 31:600–605

    Article  PubMed  CAS  Google Scholar 

  15. Bondeson L, Sandelin K, Grimelius L (1993) Histopathological variables and DNA cytometry in parathyroid carcinoma. Am J Surgical Pathol 17:820–829

    Article  CAS  Google Scholar 

  16. DeLellis RA (2006) “Parathyroid carcinoma,” in World Health Organization Classification of Tumours. In: DeLellis RA, Lloyd RV, Heitz PU, Eng C (eds) Pathology and genetics of tumours of endocrine organs. IARC Press, Lyon

  17. Juhlin CC, Villablanca A, Sandelin K, Haglund F, Nordenström J, Forsberg L, Bränström R, Obara T, Arnold A, Larsson C, Höög A (2007) Parafibromin immunoreactivity: its use as an additional diagnostic marker for parathyroid tumor classification. Endocr Relat Cancer 14:501–512

    Article  PubMed  CAS  Google Scholar 

  18. Juhlin C, Larsson C, Yakoleva T, Leibiger I, Leibiger B, Alimov A, Weber G, Höög A, Villablanca A (2006) Loss of parafibromin expression in a subset of parathyroid adenomas. Endocr Relat Cancer 13:509–523

    Article  PubMed  CAS  Google Scholar 

  19. Kayath MJ, Martin LC, Vieira JGH, Roman LM, Nośe-Alberti V (1998) A comparative study of p53 immunoexpression in parathyroid hyperplasias secondary to uremia, primary hyperplasias, adenomas and carcinomas. Eur J Endocrinol 139:78–83

    Article  PubMed  CAS  Google Scholar 

  20. Vasef MA, Brynes RK, Sturm M, Bromley C, Robinson RA (1999) Expression of cyclin D1 in parathyroid carcinomas, adenomas, and hyperplasias: a paraffin immunohistochemical study. Mod Pathol 12:412–416

    PubMed  CAS  Google Scholar 

  21. Lloyd RV, Carney JA, Ferreiro JA, Jin L, Thompson GB, Van Heerden JA, Grant CS, Wollan PC (1995) Immunohistochemical analysis of the cell cycle-associated antigens Ki-67 and retinoblastoma protein in parathyroid carcinomas and adenomas. Endocr Pathol 6:279–287

    Article  PubMed  Google Scholar 

  22. Farnebo F, Auer G, Farnebo LO, Teh BT, Twigg S, Aspenblad U, Thompson NW, Grimelius L, Larsson C, Sandelin K (1999) Evaluation of retinoblastoma and Ki-67 immunostaining as diagnostic markers of benign and malignant parathyroid disease. World J Surg 23:68–74

    Article  PubMed  CAS  Google Scholar 

  23. Abbona GC, Papotti M, Gasparri G, Bussolati G (1995) Proliferative activity in parathyroid tumors as detected by Ki-67 immunostaining. Hum Pathol 26:135–138

    Article  PubMed  CAS  Google Scholar 

  24. Boquist LL (1990) Nucleolar organizer regions in normal, hyperplastic and neoplastic parathyroid glands. Virchows Arch A Pathol Anat Histopathol 417:237–241

    Article  PubMed  CAS  Google Scholar 

  25. Kanematsu E, Matsui H, Deguchi T, Yamamoto O, Korematsu M, Kobayashi A, Nezasa SI, Yamamoto N, Takeuchi T, Tanaka T, Kawada Y (1997) Significance of AgNOR counts for distinguishing carcinoma from adenoma and hyperplasia in parathyroid gland. Hum Pathol 28:421–427

    Article  PubMed  CAS  Google Scholar 

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Conflict of interest

None of the authors had any personal or financial conflicts of interest.

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Authors and Affiliations

  1. Unità di Medicina Nucleare, Dipartimento di Scienze Radiologiche, Padiglione E, piano terra, Policlinico Universitario “G. Martino”, University of Messina, v. Consolare Valeria, 98125, Messina, Italy

    Alfredo Campennì, Alessandro Sindoni & Sergio Baldari

  2. Department of Clinical and Experimental Medicine and Pharmacology, Section of Endocrinology, University of Messina, Messina, Italy

    Rosaria M. Ruggeri, Salvatore Giovinazzo & Salvatore Benvenga

  3. Department of Human Pathology, Section of Surgery, University of Messina, Messina, Italy

    Enrico Calbo & Letterio Calbo

  4. Department of Human Pathology, Section of Anatomic Pathology, University of Messina, Messina, Italy

    Antonio Ieni & Giovanni Tuccari

Authors
  1. Alfredo Campennì
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  2. Rosaria M. Ruggeri
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  3. Alessandro Sindoni
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  4. Salvatore Giovinazzo
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  5. Enrico Calbo
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  6. Antonio Ieni
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  7. Letterio Calbo
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  8. Giovanni Tuccari
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  9. Sergio Baldari
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  10. Salvatore Benvenga
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Correspondence to Alfredo Campennì.

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Campennì, A., Ruggeri, R.M., Sindoni, A. et al. Parathyroid carcinoma presenting as normocalcemic hyperparathyroidism. J Bone Miner Metab 30, 367–372 (2012). https://doi.org/10.1007/s00774-011-0344-y

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  • DOI: https://doi.org/10.1007/s00774-011-0344-y

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