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Clinicopathological spectrum, management, and outcome of ectopic parathyroid carcinoma: Experience with 24 cases

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Abstract

Purpose

Ectopic parathyroid carcinoma (EPC) is a rare clinical entity with multiple diagnostic pitfalls, making surgical cures challenging. We assessed the clinicopathological spectrum and outcome of EPCs.

Methods

In this retrospective cohort study, 24 EPCs were identified from 133 PC patients treated at a tertiary referral center. The relationship between clinicopathological findings and locations was analyzed.

Results

The locations of EPCs were predominantly intrathyroidal (62.5%), followed by 16.7% in the mediastinum, 8.3% in the retropharyngeal space, 8.3% in the carotid sheath, and 4.2% in the upper neck. Intrathyroidal EPC patients experienced higher serum calcium (p = 0.020), a higher rate of vascular invasion (p = 0.040), and a slightly higher incidence of non-R0 initial resection (p = 0.092) than those in other ectopic locations. Intrathyroidal EPC patients also suffered a trend of higher upper aerodigestive tract (UAT) invasion rate (p = 0.070) and higher risks of distant metastasis (p = 0.037) than the other PC patients. The 5-year disease-free survival rate after surgery was slightly compromised at 41.5% in intrathyroidal EPC patients compared with 77.8% among those in other ectopic locations (p = 0.143) and 59.7% among the other PC patients (log-rank = 3.194; p = 0.074), though without statistical significance.

Conclusion

Intrathyroidal EPC might cause a more biochemically and invasively distinct PC picture compared with other PCs. Special caution should be exercised in the preoperative diagnosis and management of such cases.

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References

  1. E. Shane, Clinical review 122: Parathyroid carcinoma. J. Clin. Endocrinol. Metab. 86(2), 485–493 (2001). https://doi.org/10.1210/jcem.86.2.7207

    Article  CAS  PubMed  Google Scholar 

  2. A.S. Salcuni, F. Cetani, V. Guarnieri, V. Nicastro, E. Romagnoli, D. de Martino et al. Parathyroid carcinoma. Best Pract. Res. Clin. Endocrinol. Metab. 32(6), 877–889 (2018). https://doi.org/10.1016/j.beem.2018.11.002

    Article  CAS  PubMed  Google Scholar 

  3. S. Xue, H. Chen, C. Lv, X. Shen, J. Ding, J. Liu et al. Preoperative diagnosis and prognosis in 40 parathyroid carcinoma patients. Clin. Endocrinol. 85(1), 29–36 (2016). https://doi.org/10.1111/cen.13055

    Article  Google Scholar 

  4. N. Talat, K.M. Schulte, Clinical presentation, staging and long-term evolution of parathyroid cancer. Ann. Surg. Oncol. 17(8), 2156–2174 (2010). https://doi.org/10.1245/s10434-010-1003-6

    Article  PubMed  Google Scholar 

  5. A. Harari, A. Waring, G. Fernandez-Ranvier, J. Hwang, I. Suh, E. Mitmaker et al. Parathyroid carcinoma: a 43-year outcome and survival analysis. J. Clin. Endocrinol. Metab. 96(12), 3679–3686 (2011). https://doi.org/10.1210/jc.2011-1571

    Article  CAS  PubMed  Google Scholar 

  6. C.H. Wei, A. Harari, Parathyroid carcinoma: update and guidelines for management. Curr. Treat. Options Oncol. 13(1), 11–23 (2012). https://doi.org/10.1007/s11864-011-0171-3

    Article  PubMed  Google Scholar 

  7. A. Al-Kurd, M. Mekel, H. Mazeh, Parathyroid carcinoma. Surg. Oncol. 23(2), 107–114 (2014). https://doi.org/10.1016/j.suronc.2014.03.005

    Article  PubMed  Google Scholar 

  8. M. Roy, H. Mazeh, H. Chen, R.S. Sippel, Incidence and localization of ectopic parathyroid adenomas in previously unexplored patients. World J. Surg. 37(1), 102–106 (2013). https://doi.org/10.1007/s00268-012-1773-z

    Article  PubMed  Google Scholar 

  9. R.D. Gaz, P.B. Doubler, C.A. Wang, The management of 50 unusual hyperfunctioning parathyroid glands. Surgery. 102(6), 949–957 (1987)

    CAS  PubMed  Google Scholar 

  10. N. Poortmans, G. Verfaillie, D. Unuane, S. Raeymaeckers, J. Lamote, Intrathyroidal parathyroid carcinoma presenting as an asymptomatic hypercalcemia: a case report. Acta Chir. Belg. 120(6), 433–436 (2020). https://doi.org/10.1080/00015458.2019.1631626

    Article  PubMed  Google Scholar 

  11. M.I. Cook, Y.A. Qureshi, C.E. Todd, R.S. Cummins, An unusual ectopic location of a parathyroid carcinoma arising within the thyroid gland. J. Clin. Endocrinol. Metab. 97(6), 1829–1833 (2012). https://doi.org/10.1210/jc.2011-3115

    Article  CAS  PubMed  Google Scholar 

  12. Y. **n, T. Zhao, B. Wei, H. Gu, M. **, H. Shen et al. Intrapericardial parathyroid carcinoma: a case report. Endocrine 69(2), 456–460 (2020). https://doi.org/10.1007/s12020-020-02283-8

    Article  CAS  PubMed  Google Scholar 

  13. S.V. Gowrishankar, R. Bidaye, T. Das, V. Majcher, B. Fish, R. Casey et al. Intrathyroidal parathyroid adenomas: Sco** review on clinical presentation, preoperative localization, and surgical treatment. Head Neck 45(3), 706–720 (2023). https://doi.org/10.1002/hed.27287

    Article  PubMed  Google Scholar 

  14. W. Zhao, R. Lu, L. Yin, B. Wei, M. **, C. Zhang et al. The value of preoperative and intraoperative ultrasound in the localization of intrathyroidal parathyroid adenomas. J. Invest. Surg. 35(4), 752–757 (2022). https://doi.org/10.1080/08941939.2021.1933273

    Article  PubMed  Google Scholar 

  15. B. Wei, T. Zhao, H. Shen, M. **, Q. Zhou, X. Liu et al. Extended en bloc reoperation for recurrent or persistent parathyroid carcinoma: Analysis of 31 cases in a single institute experience. Ann. Surg. Oncol. 29(2), 1208–1215 (2022). https://doi.org/10.1245/s10434-021-10962-7

    Article  PubMed  Google Scholar 

  16. A.M. Parikh, J.W. Suliburk, F.E. Morón, Imaging localization and surgical approach in the management of ectopic parathyroid adenomas. Endocr. Pract. 24(6), 589–598 (2018). https://doi.org/10.4158/EP-2018-0003

    Article  PubMed  Google Scholar 

  17. G. Noussios, P. Anagnostis, K. Natsis, Ectopic parathyroid glands and their anatomical, clinical and surgical implications. Exp. Clin. Endocrinol. Diabetes. 120(10), 604–610 (2012). https://doi.org/10.1055/s-0032-1327628

    Article  CAS  PubMed  Google Scholar 

  18. M.B. Albuja-Cruz, B.J. Allan, P.P. Parikh, J.I. Lew, Efficacy of localization studies and intraoperative parathormone monitoring in the surgical management of hyperfunctioning ectopic parathyroid glands. Surgery 154(3), 453–460 (2013). https://doi.org/10.1016/j.surg.2013.05.034

    Article  PubMed  Google Scholar 

  19. S.G. Haciyanli, S. Karaisli, N. Erdogan, B. Turgut, E.O. Gur, M. Haciyanli, Intrathyroidal parathyroid adenomas in primary hyperparathyroidism: Clinical and imaging findings. Sisli Etfal Hastan. Tip Bul. 56(2), 256–261 (2022). https://doi.org/10.14744/SEMB.2022.82584

    Article  PubMed  PubMed Central  Google Scholar 

  20. J.S. Andrade, J.P. Mangussi-Gomes, L.A. Rocha, M.N. Ohe, M. Rosano, M.C. das Neves et al. Localization of ectopic and supernumerary parathyroid glands in patients with secondary and tertiary hyperparathyroidism: surgical description and correlation with preoperative ultrasonography and Tc99m-Sestamibi scintigraphy. Braz. J. Otorhinolaryngol. 80(1), 29–34 (2014). https://doi.org/10.5935/1808-8694.20140008

    Article  PubMed  PubMed Central  Google Scholar 

  21. C. Vulpio, M. Bossola, A. De Gaetano, G. Maresca, I. Bruno, G. Fadda et al. Usefulness of the combination of ultrasonography and 99mTc-sestamibi scintigraphy in the preoperative evaluation of uremic secondary hyperparathyroidism. Head Neck 32(9), 1226–1235 (2010). https://doi.org/10.1002/hed.21320

    Article  PubMed  Google Scholar 

  22. S. Tseleni-Balafouta, H. Gakiopoulou, N. Kavantzas, G. Agrogiannis, N. Givalos, E. Patsouris, Parathyroid proliferations: a source of diagnostic pitfalls in FNA of thyroid. Cancer 111(2), 130–136 (2007). https://doi.org/10.1002/cncr.22576

    Article  PubMed  Google Scholar 

  23. H. Ketha, M.A. Lasho, A. Algeciras-Schimnich, Analytical and clinical validation of parathyroid hormone (PTH) measurement in fine-needle aspiration biopsy (FNAB) washings. Clin. Biochem. 49(1-2), 16–21 (2016). https://doi.org/10.1016/j.clinbiochem.2015.09.006

    Article  CAS  PubMed  Google Scholar 

  24. C. Glasgow, E.Y.C. Lau, L. Aloj, I. Harper, H. Cheow, T. Das et al. An approach to a patient with primary hyperparathyroidism and a suspected ectopic parathyroid adenoma. J. Clin. Endocrinol. Metab. 107(6), 1706–1713 (2022). https://doi.org/10.1210/clinem/dgac024

    Article  PubMed  Google Scholar 

  25. Y. Hu, Y. Bi, M. Cui, X. Zhang, Z. Su, M. Wang et al. The influence of surgical extent and parafibromin staining on the outcome of parathyroid carcinoma: 20-year experience from a single institute. Endocr. Pract. 25(7), 634–641 (2019). https://doi.org/10.4158/EP-2018-0538

    Article  PubMed  Google Scholar 

  26. Y. Chen, A. Song, M. Nie, Y. Jiang, M. Li, W. **a et al. Clinical and genetic analysis of atypical parathyroid adenoma compared with parathyroid carcinoma and benign lesions in a Chinese cohort. Front. Endocrinol. 14, 1027598 (2023). https://doi.org/10.3389/fendo.2023.1027598

    Article  Google Scholar 

  27. T. Zhao, W. Yang, R. Shen, Q. Chen, M. **, H. Gu et al. Risk factors of distant metastatic parathyroid carcinoma and insights into therapeutic perspectives. Ann. Surg. Oncol. (2024). https://doi.org/10.1245/s10434-024-15611-3

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Acknowledgements

We would like to thank Prof. Bin Hu for invaluable technical guidance. This study was supported by the Bei**g Municipal Administration of Hospitals Incubating Program (PX2023009) and the National Natural Science Foundation of China (82200872).

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Authors

Contributions

T. Zhao, J. H., Y. W., and B. W. contributed to the study conception and design. T. Zhao, J. H., Y. W., H. S., X. L., D. F., J. W., and Q. W. collected all data. T. Zhao, D. Z., T. Zhang, R. L., H. G., M. J., and B. W. analyzed and interpreted all data. The main manuscript was written by T. Zhao and J. H. All authors read and approved the final manuscript.

Corresponding author

Correspondence to Bojun Wei.

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The authors declare no competing interests.

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This is a retrospective cohort study. The study was approved by the ethics committee of Bei**g Chaoyang Hospital.

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Written informed consent was obtained from all patients included in the study.

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Zhao, T., Huang, J., Wang, Y. et al. Clinicopathological spectrum, management, and outcome of ectopic parathyroid carcinoma: Experience with 24 cases. Endocrine (2024). https://doi.org/10.1007/s12020-024-03964-4

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