Parathyroid carcinoma. The experience of treatment of 15 patients and a review
https://doi.org/10.17650/2222-1468-2020-10-3-19-26
Abstract
Background. Parathyroid cancer (PC) is extremely rare, usually diagnosed after surgery, there are no clinical recommendations for the treatment of metastatic PC.
The study objective is to present the results of a clinical examination and treatment of patients with PC.
Materials and methods. Retrospective analysis of 15 PC patients treated from 2001 to 2019. The results of laboratory tests, the surgical approach, the results of treatment, the rate of metastasis and relapse within 19 months (3 months – 11.6 years) after surgery are presented.
Results. Tumor size is 35 mm (20–45 mm). Lymph node metastases – 1 (6.7 %), distant metastases – 2 (13 %). The median initial parathyroid hormone (PTH) is 735 pg/mL, calcium 3.22 mmol/L. Severe hyperparathyroidism with osteitis fibrosa cystica – in 6 (40 %). The surgical approach was parathyroidectomy in 10 (67 %), en bloc resection in 5 (33 %). Normalization of PTH and calcium after surgery – 13 (87 %). In two patients with distant metastases, the PTH and calcium remained high. A patient with Th6 vertebral metastasis was successfully operated on, with normalization of PTH and calcium, and 1.5 years was without relapse. A patient with lung and liver metastases received sorafenib after surgery, with decrease in calcium level. She died of progression 12 months after operation. Among patients with normalization of postoperative PTH, one had local relapse after 4 years. The patient was re-operated and 1 year after the second operation without relapse. The remaining patients are without relapse/progression.
Conclusion. At the time of diagnosis, lymph node metastases are in 6.7 %, distant metastases – 13 %. Normalization of PTH and calcium after surgery suggests a good prognosis, but does not exclude the recurrence in the future, which requires long-term follow-up. Repeated surgery for local recurrence or solitary distant metastases can provide stable remission. Sorafenib in metastatic PC has managed to control hypercalcemia in the short term, however, antitumor efficacy requires further study.
About the Authors
N. V. SeverskayaRussian Federation
4 Koroleva St., Obninsk 249036
A. A. Ilyin
Russian Federation
4 Koroleva St., Obninsk 249036
I. V. Chebotareva
Russian Federation
4 Koroleva St., Obninsk 249036
N. V. Zhelonkina
Russian Federation
4 Koroleva St., Obninsk 249036
V. V. Polkin
Russian Federation
4 Koroleva St., Obninsk 249036
P. A. Isaev
Russian Federation
4 Koroleva St., Obninsk 249036
D. N. Derbugov
Russian Federation
4 Koroleva St., Obninsk 249036
S. O. Podvyaznikov
Russian Federation
Bld. 1, 2/1 Barrikadnaya St., Moscow 125993
References
1. Cetani F., Pardi E., Marcocci C. Update on parathyroid carcinoma. J Endocrinol Invest 2016;39(6):595–606. DOI: 10.1007/s40618-016-0447-3.
2. Givi B., Shah J.P. Parathyroid carcinoma. Clin Oncol (R Coll Radiol) 2010;22(6):498–507. DOI: 10.1016/j.clon.2010.04.007.
3. Cinque L., Sparaneo A., Salcuni A.S. et al. MEN1 gene mutation with parathyroid carcinoma: first report of a familial case. Endocr Connect 2017;6(8):886–91. DOI: 10.1530/ec-17-0207.
4. Cardoso L., Stevenson M., Thakker R.V. Molecular genetics of syndromic and non-syndromic forms of parathyroid carcinoma. Hum Mutat 2017;38(12): 1621–48. DOI: 10.1002/humu.23337.
5. Howell V.M., Haven C.J., Kahnoski K. et al. HRPT2 mutations are associated with malignancy in sporadic parathyroid tumours. J Med Genet 2003;40(9):657–63. DOI: 10.1136/jmg.40.9.657.
6. Guarnieri V., Battista C., Muscarella L.A. et al. CDC73 mutations and parafibromin immunohistochemistry in parathyroid tumors: clinical correlations in a singlecentre patient cohort. Cell Oncol (Dordr) 2012;35(6):411–22. DOI: 10.1007/s13402-012-0100-x.
7. Wilkins B.J., Lewis J.S. Jr. Non-functional parathyroid carcinoma: a review of the literature and report of a case requiring extensive surgery. Head Neck Pathol 2009;3(2):140–9. DOI: 10.1007/s12105-009-0115-4.
8. Cetani F., Frustaci G., Torregrossa L. et al. A nonfunctioning parathyroid carcinoma misdiagnosed as a follicular thyroid nodule. World J Surg Oncol 2015;13:270. DOI: 10.1186/s12957-015-0672-9.
9. Ferraro V., Sgaramella L.I., Di Meo G. et al. Current concepts in parathyroid carcinoma: a single centre experience. BMC Endocr Disord 2019;19(Suppl 1):46. DOI: 10.1186/s12902-019-0368-1.
10. Cetani F., Pardi E., Marcocci C. Parathyroid carcinoma. Front Horm Res 2019;51:63–76. DOI: 10.1159/000491039.
11. Artinyan A., Guzman E., Maghami E. et al. Metastatic parathyroid carcinoma to the liver treated with radiofrequency ablation and transcatheter arterial embolization. J Clin Oncol 2008;26(24):4039–41. DOI: 10.1200/jco.2007.15.9038.
12. Tochio M., Takaki H., Yamakado K. et al. A case report of 20 lung radiofrequency ablation sessions for 50 lung metastases from parathyroid carcinoma causing hyperparathyroidism. Cardiovasc Intervent Radiol 2010;33(3):657–9. DOI: 10.1007/s00270-009-9730-4.
13. DasGupta R., Shetty S., Keshava S.N. et al. Metastatic parathyroid carcinoma treated with radiofrequency ablation: a novel therapeutic modality. Australas Med J 2014;7(9):372–5. DOI: 10.4066/amj.2014.2084.
14. Rozhinskaya L., Pigarova E., Sabanova E. et al. Diagnosis and treatment challenges of parathyroid carcinoma in a 27-year-old woman with multiple lung metastases. Endocrinol Diabetes Metab Case Rep 2017;2017:16-0113. DOI: 10.1530/edm-16-0113.
15. Talat N., Schulte K.M. Clinical presentation, staging and long-term evolution of parathyroid cancer. Ann Surg Oncol 2010;17(8):2156–74. DOI: 10.1245/s10434-010-1003-6.
16. Ryhanen E.M., Leijon H., Metso S. et al. A nationwide study on parathyroid carcinoma. Acta Oncol 2017;56(7):991–1003. DOI: 10.1080/0284186x.2017.1306103.
17. Baloch Z.W., Livolsi V.A. Parathyroids morphology and pathology. In: Parathyroids: basic and clinical concepts. Ed. by J.P. Bilezikian, R. Marcus, M.A. Levine et al. 3rd edn. Academic Press, 2015. Pp. 23–36. DOI: 10.1016/b978-0-12-397166-1.00001-1.
18. Lee P.K., Jarosek S.L., Virnig B.A. et al. Trends in the incidence and treatment of parathyroid cancer in the United States. Cancer 2007;109(9):1736–41. DOI: 10.1002/cncr.22599.
19. Favia G., Lumachi F., Polistina F., D’Amico D.F. Parathyroid carcinoma: sixteen new cases and suggestions for correct management. World J Surg 1998;22(12):1225–30. DOI: 10.1007/s002689900549.
20. Shane E. Clinical review 122: parathyroid carcinoma. J Clin Endocrinol Metab 2001;86(2):485–93. DOI: 10.1210/jcem.86.2.7207.
21. Deandreis D., Terroir M., Al Ghuzlan A. et al. 18Fluorocholine PET/CT in parathyroid carcinoma: a new tool for disease staging? Eur J Nucl Med Mol Imaging 2015;42(12):1941–2. DOI: 10.1007/s00259-015-3130-6.
22. Andersen K.F., Albrecht-Beste E. Brown tumors due to primary hyperparathyroidism in a patient with parathyroid carcinoma mimicking skeletal metastases on (18)F-FDG PET/CT. Diagnostics(Basel) 2015;5(3):290–3. DOI: 10.3390/diagnostics5030290.
23. Tsushima Y., Sun S., Via M.A. Brown tumors secondary to parathyroid carcinoma masquerading as skeletal metastases on 18F-FDG PET/CT: a case report. AACE Clin Case Rep 2019;5(4):e230–2. DOI: 10.4158/accr-2018-0633.
24. Agarwal G., Mishra S.K., Kar D.K. et al. Recovery pattern of patients with osteitis fibrosa cystica in primary hyperparathyroidism after successful parathyroidectomy. Surgery 2002;132(6):1075–83. DOI: 10.1067/msy.2002.128484.
25. Montenegro F.L., Chammas M.C., Juliano A.G. et al. Ethanol injection under ultrasound guidance to palliate unresectable parathyroid carcinoma. Arq Bras Endocrinol Metabol 2008;52(4):707–11. DOI: 10.1590/s0004-27302008000400019.
26. Mauz P.S., Stiegler M., Holderried M., Brosch S. Complications of ultrasound guided percutaneous ethanol injection therapy of the thyroid and parathyroid glands. Ultraschall Med 2005;26(2):142–5. DOI: 10.1055/s-2005-857867.
27. Qiu Z.L., Wu C.G., Zhu R.S. et al. Unusual case of solitary functioning bone metastasis from a “parathyroid adenoma”: imagiologic diagnosis and treatment with percutaneous vertebroplasty – case report and literature review. J Clin Endocrinol Metab 2013;98(9):3555–61. DOI: 10.1210/jc.2013-2014.
28. Munson N.D., Foote R.L., Northcutt R.C. et al. Parathyroid carcinoma: is there a role for adjuvant radiation therapy? Cancer 2003;98(11):2378–84. DOI: 10.1002/cncr.11819.
29. Storvall S., Ryhanen E., Bensch F.V. et al. Recurrent metastasized parathyroid carcinoma-long-term remission after combined treatments with surgery, radiotherapy, cinacalcet, zoledronic acid, and temozolomide. JBMR Plus 2019;3(4):e10114. DOI: 10.1002/jbm4.10114.
Review
For citations:
Severskaya N.V., Ilyin A.A., Chebotareva I.V., Zhelonkina N.V., Polkin V.V., Isaev P.A., Derbugov D.N., Podvyaznikov S.O. Parathyroid carcinoma. The experience of treatment of 15 patients and a review. Head and Neck Tumors (HNT). 2020;10(3):19-26. (In Russ.) https://doi.org/10.17650/2222-1468-2020-10-3-19-26