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Published in: Endocrine 1/2020

01-01-2020 | Parathyroid Cancer | Original Article

Long-term remission of disseminated parathyroid cancer following immunotherapy

Authors: Marta Sarquis, Stephen J. Marx, Albert Beckers, Arthur R. Bradwell, William F. Simonds, Maria Aparecida C. Bicalho, Adrian F. Daly, Daniela Betea, Eitan Friedman, Luiz De Marco

Published in: Endocrine | Issue 1/2020

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Abstract

Purpose

Parathyroid cancer is a rare tumor associated with poor prognosis particularly when disseminated. While chemotherapy and/or radiotherapy are of no clinical value in disseminated disease, immunotherapy should be considered.

Subject and results

A patient with CDC73-associated metastatic parathyroid carcinoma was treated with combined anti-hPTH immunotherapy and surgery.

Conclusions

Following five courses of anti-hPTH immunotherapy and subsequent surgery, a 12-year long remission of disseminated parathyroid cancer is reported. This case further supports the ever-expanding spectrum of cancers that may benefit from immunotherapy.
Literature
1.
go back to reference C. Sadler, C. Sadler, K.W. Gow, E.A. Beierle, J.J. Doski, M. Langer, J.G. Nuchtern, S.A. Vasudevan, M. Goldfarb, Parathyroid carcinoma in more than 1,000 patients: a population-level analysis. Surgery 156, 1622–1629 (2014)CrossRef C. Sadler, C. Sadler, K.W. Gow, E.A. Beierle, J.J. Doski, M. Langer, J.G. Nuchtern, S.A. Vasudevan, M. Goldfarb, Parathyroid carcinoma in more than 1,000 patients: a population-level analysis. Surgery 156, 1622–1629 (2014)CrossRef
2.
go back to reference D. Betea, I. Potorac, A. Beckers, Parathyroid carcinoma: challenges in diagnosis and treatment. Ann. Endocrinol. 76, 169–177 (2015)CrossRef D. Betea, I. Potorac, A. Beckers, Parathyroid carcinoma: challenges in diagnosis and treatment. Ann. Endocrinol. 76, 169–177 (2015)CrossRef
3.
go back to reference A.S. Salcuni, F. Cetani, V. Guarnieri, V. Nicastro, E. Romagnoli, D. de Martino, A. Scillitani, D.E.C. Cole, Parathyroid carcinoma. Best Pract. Res. Clin. Endocrinol. Metab. 32, 877–889 (2018)CrossRef A.S. Salcuni, F. Cetani, V. Guarnieri, V. Nicastro, E. Romagnoli, D. de Martino, A. Scillitani, D.E.C. Cole, Parathyroid carcinoma. Best Pract. Res. Clin. Endocrinol. Metab. 32, 877–889 (2018)CrossRef
4.
go back to reference K. Brewer., J. Costa-Guda, A. Arnold, Molecular genetic insights into sporadic primary hyperparathyroidism. Endocr. Relat. Cancer 26, R53–R72 (2019)CrossRef K. Brewer., J. Costa-Guda, A. Arnold, Molecular genetic insights into sporadic primary hyperparathyroidism. Endocr. Relat. Cancer 26, R53–R72 (2019)CrossRef
5.
go back to reference E. Cavalier, A.F. Daly, D. Betea, P.N. Pruteanu-Apetrii, P. Delanaye, P. Stubbs, A.R. Bradwell, J.P. Chapelle, A. Beckers, The ratio of parathyroid hormone as measured by third- and second-generation assays as a marker of parathyroid carcinoma. J. Clin. Endocrinol. Metab. 95, 3745–3749 (2010)CrossRef E. Cavalier, A.F. Daly, D. Betea, P.N. Pruteanu-Apetrii, P. Delanaye, P. Stubbs, A.R. Bradwell, J.P. Chapelle, A. Beckers, The ratio of parathyroid hormone as measured by third- and second-generation assays as a marker of parathyroid carcinoma. J. Clin. Endocrinol. Metab. 95, 3745–3749 (2010)CrossRef
6.
go back to reference A. Harari, A. Waring, G. Fernandez-Ranvier, J. Hwang, I. Suh, E. Mitmaker, W. Shen, J. Gosnell, Q.Y. Duh, O. Clark, Parathyroid carcinoma: a 43-year outcome and survival analysis. J. Clin. Endocrinol. Metab. 96, 3679–3686 (2011)CrossRef A. Harari, A. Waring, G. Fernandez-Ranvier, J. Hwang, I. Suh, E. Mitmaker, W. Shen, J. Gosnell, Q.Y. Duh, O. Clark, Parathyroid carcinoma: a 43-year outcome and survival analysis. J. Clin. Endocrinol. Metab. 96, 3679–3686 (2011)CrossRef
7.
go back to reference E.A. Asare, C. Sturgeon, D.J. Winchester, L. Liu, B. Palis, N.D. Perrier, D.B. Evans, D.P. Winchester, T.S. Wang, Parathyroid carcinoma: an update on treatment outcomes and prognostic factors from the National Cancer Data Base (NCDB). Ann. Surg. Oncol. 22, 3990–3995 (2015)CrossRef E.A. Asare, C. Sturgeon, D.J. Winchester, L. Liu, B. Palis, N.D. Perrier, D.B. Evans, D.P. Winchester, T.S. Wang, Parathyroid carcinoma: an update on treatment outcomes and prognostic factors from the National Cancer Data Base (NCDB). Ann. Surg. Oncol. 22, 3990–3995 (2015)CrossRef
8.
go back to reference Y. Takeuchi, S. Takahashi, D. Miura, M. Katagiri, N. Nakashima, H. Ohishi, R. Shimazaki, Y. Tominaga, Cinacalcet hydrochloride relieves hypercalcemia in Japanese patients with parathyroid cancer and intractable primary hyperparathyroidism. J. Bone Min. Metab. 35, 616–622 (2017)CrossRef Y. Takeuchi, S. Takahashi, D. Miura, M. Katagiri, N. Nakashima, H. Ohishi, R. Shimazaki, Y. Tominaga, Cinacalcet hydrochloride relieves hypercalcemia in Japanese patients with parathyroid cancer and intractable primary hyperparathyroidism. J. Bone Min. Metab. 35, 616–622 (2017)CrossRef
9.
go back to reference A.R. Bradwell, T.C. Harvey, Control of hypercalcaemia of parathyroid carcinoma by immunisation. Lancet 353, 370–373 (2019)CrossRef A.R. Bradwell, T.C. Harvey, Control of hypercalcaemia of parathyroid carcinoma by immunisation. Lancet 353, 370–373 (2019)CrossRef
10.
go back to reference D. Betea, A.R. Bradwell, T.C. Harvey, G.P. Mead, H. Schmidt-Gayk, B. Ghaye, A.F. Daly, A. Beckers, Hormonal and biochemical normalization and tumor shrinkage induced by anti-parathyroid hormone immunotherapy in a patient with metastatic parathyroid carcinoma. J. Clin. Endocrinol. Metab. 89, 3413–3420 (2014)CrossRef D. Betea, A.R. Bradwell, T.C. Harvey, G.P. Mead, H. Schmidt-Gayk, B. Ghaye, A.F. Daly, A. Beckers, Hormonal and biochemical normalization and tumor shrinkage induced by anti-parathyroid hormone immunotherapy in a patient with metastatic parathyroid carcinoma. J. Clin. Endocrinol. Metab. 89, 3413–3420 (2014)CrossRef
11.
go back to reference I. Horie, T. Ando, N. Inokuchi, Y. Mihara, S. Miura, M. Imaizumi, T. Usa, N. Kinoshita, I. Sekine, S. Kamihara, K. Eguchi, First Japanese patient treated with parathyroid hormone peptide immunization for refractory hypercalcemia caused by metastatic parathyroid carcinoma. Endocr. J. 57, 287–292 (2010)CrossRef I. Horie, T. Ando, N. Inokuchi, Y. Mihara, S. Miura, M. Imaizumi, T. Usa, N. Kinoshita, I. Sekine, S. Kamihara, K. Eguchi, First Japanese patient treated with parathyroid hormone peptide immunization for refractory hypercalcemia caused by metastatic parathyroid carcinoma. Endocr. J. 57, 287–292 (2010)CrossRef
12.
go back to reference V.M. Howell, C.J. Haven, K. Kahnoski, S.K. Khoo, D. Petillo, J. Chen, G.J. Fleuren, B.G. Robinson, L.W. Delbridge, J. Philips, A.E. Nelson, U. Krause, K. Hammje, H. Dralle, C. Hoang-Vu, O. Gimm, D.J. Marsh, H. Morreau, B.T. The, HRPT2 mutations are associated with malignancy in sporadic parathyroid tumours. J. Med Genet. 40, 657–663 (2003)CrossRef V.M. Howell, C.J. Haven, K. Kahnoski, S.K. Khoo, D. Petillo, J. Chen, G.J. Fleuren, B.G. Robinson, L.W. Delbridge, J. Philips, A.E. Nelson, U. Krause, K. Hammje, H. Dralle, C. Hoang-Vu, O. Gimm, D.J. Marsh, H. Morreau, B.T. The, HRPT2 mutations are associated with malignancy in sporadic parathyroid tumours. J. Med Genet. 40, 657–663 (2003)CrossRef
13.
go back to reference M.S. Sarquis, L.G. Silveira, F.J. Pimenta, E.P. Dias, B.T. Teh, E. Friedman, R.S. Gomez, G.C. Tavares, C. Eng, L. De Marco, Familial hyperparathyroidism: surgical outcome after 30 years of follow-up in three families with germline HRPT2 mutations. Surgery 43, 630–640 (2008)CrossRef M.S. Sarquis, L.G. Silveira, F.J. Pimenta, E.P. Dias, B.T. Teh, E. Friedman, R.S. Gomez, G.C. Tavares, C. Eng, L. De Marco, Familial hyperparathyroidism: surgical outcome after 30 years of follow-up in three families with germline HRPT2 mutations. Surgery 43, 630–640 (2008)CrossRef
14.
go back to reference L. Rozhinskaya, E. Pigarova, E. Sabanova, E. Mamedova, I. Voronkova, J. Krupinova, L. Dzeranova, A. Tiulpakov, V. Gorbunova, N. Orel, A. Zalian, G. Melnichenko, I. Dedov, Diagnosis and treatment challenges of parathyroid carcinoma in a 27-year-old woman with multiple lung metastases. Endocrinol. Diabetes Metab. Case Rep. 6, 0113 (2017) L. Rozhinskaya, E. Pigarova, E. Sabanova, E. Mamedova, I. Voronkova, J. Krupinova, L. Dzeranova, A. Tiulpakov, V. Gorbunova, N. Orel, A. Zalian, G. Melnichenko, I. Dedov, Diagnosis and treatment challenges of parathyroid carcinoma in a 27-year-old woman with multiple lung metastases. Endocrinol. Diabetes Metab. Case Rep. 6, 0113 (2017)
15.
go back to reference C.N. Clarke, P. Katsonis, T.K. Hsu, A.M. Koire, A. Silva-Figueroa, I. Christakis, M.D. Williams, M. Kutahyalioglu, L. Kwatampora, Y. Xi, J.E. Lee, E.S. Koptez, N.L. Busaidy, N.D. Perrier, O. Lichtarge, Comprehensive genomic characterization of parathyroid cancer identifies novel candidate driver mutations and core pathways. J. Endocr. Soc. 3, 544–559 (2018)CrossRef C.N. Clarke, P. Katsonis, T.K. Hsu, A.M. Koire, A. Silva-Figueroa, I. Christakis, M.D. Williams, M. Kutahyalioglu, L. Kwatampora, Y. Xi, J.E. Lee, E.S. Koptez, N.L. Busaidy, N.D. Perrier, O. Lichtarge, Comprehensive genomic characterization of parathyroid cancer identifies novel candidate driver mutations and core pathways. J. Endocr. Soc. 3, 544–559 (2018)CrossRef
16.
go back to reference P. Piranavan, Y. Li, E. Brown, E.H. Kemp, N. Trivedi, Immune checkpoint inhibitor-induced hypoparathyroidism associated with Calcium-Sensing Receptor-Activating autoantibodies. J. Clin. Endocrinol. Metab. 104, 550–556 (2019)CrossRef P. Piranavan, Y. Li, E. Brown, E.H. Kemp, N. Trivedi, Immune checkpoint inhibitor-induced hypoparathyroidism associated with Calcium-Sensing Receptor-Activating autoantibodies. J. Clin. Endocrinol. Metab. 104, 550–556 (2019)CrossRef
Metadata
Title
Long-term remission of disseminated parathyroid cancer following immunotherapy
Authors
Marta Sarquis
Stephen J. Marx
Albert Beckers
Arthur R. Bradwell
William F. Simonds
Maria Aparecida C. Bicalho
Adrian F. Daly
Daniela Betea
Eitan Friedman
Luiz De Marco
Publication date
01-01-2020
Publisher
Springer US
Published in
Endocrine / Issue 1/2020
Print ISSN: 1355-008X
Electronic ISSN: 1559-0100
DOI
https://doi.org/10.1007/s12020-019-02136-z

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