Pembrolizumab with external radiation therapy effectively controlled TMB-high unresectable recurrent parathyroid cancer: a case report with review of literature
Parathyroid cancer (PC) is extremely resistant to chemotherapy and radiotherapy (RT), but hormonally functional by producing excessive parathyroid hormone (PTH), causing remarkable hypercalcemia even in biochemical disease recurrence. Accordingly, management of hypercalcemia by calcimimetics and bis...
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The Japan Endocrine Society
2024-11-01
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| Series: | Endocrine Journal |
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| Online Access: | https://www.jstage.jst.go.jp/article/endocrj/71/11/71_EJ24-0126/_html/-char/en |
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| author | Hiroshi Katoh Tomoya Mitsuma Riku Okamoto Kanako Naito Takaaki Tokito Mariko Kikuchi Takafumi Sangai |
| author_facet | Hiroshi Katoh Tomoya Mitsuma Riku Okamoto Kanako Naito Takaaki Tokito Mariko Kikuchi Takafumi Sangai |
| author_sort | Hiroshi Katoh |
| collection | DOAJ |
| description | Parathyroid cancer (PC) is extremely resistant to chemotherapy and radiotherapy (RT), but hormonally functional by producing excessive parathyroid hormone (PTH), causing remarkable hypercalcemia even in biochemical disease recurrence. Accordingly, management of hypercalcemia by calcimimetics and bisphosphonates has been main treatment for unresectable PC. Here, we report a case of unresectable tumor mutational burden (TMB)-high recurrent PC that has been effectively controlled by pembrolizumab (PEM) with RT. A 48-year-old male patient, with previous history of left single parathyroidectomy for primary hyperparathyroidism, underwent surgeries for recurrent hyperparathyroidism at 47 and 48 years of age, and was pathologically diagnosed with PC. He was referred to our hospital due to persistent hypercalcemia and elevated PTH. The recurrent tumors were identified in the superior mediastinum and radically resected, then the hyperparathyroidism was improved. A FoundationOne® CDx of the specimen called TMB-high. He demonstrated recurrent hyperparathyroidism at 49 years of age, and underwent a gross curative resection. However, hyperparathyroidism achieved only insufficient improvement, indicating biochemical residual cancer cells. PEM treatment was initiated in combination with RT to the left central-lateral neck and superior mediastinum. He successfully achieved evocalcet and zoledronate withdrawal, and the PTH level improvement was continuously observed for 8 months at present, with only grade 2 subclinical hypothyroidism. Interestingly, leukocyte fraction ratios were reversed corresponding to disease improvement. A combination of PEM and RT is a promising treatment of unresectable TMB-high PC. Recent evidence on the immunomodulatory effect of RT provides the rationale for the combination of RT and PEM. |
| format | Article |
| id | doaj-art-657c14e8ac0c499bbf8c479bfcd83a88 |
| institution | Kabale University |
| issn | 1348-4540 |
| language | English |
| publishDate | 2024-11-01 |
| publisher | The Japan Endocrine Society |
| record_format | Article |
| series | Endocrine Journal |
| spelling | doaj-art-657c14e8ac0c499bbf8c479bfcd83a882025-01-22T05:38:18ZengThe Japan Endocrine SocietyEndocrine Journal1348-45402024-11-0171111069107510.1507/endocrj.EJ24-0126endocrjPembrolizumab with external radiation therapy effectively controlled TMB-high unresectable recurrent parathyroid cancer: a case report with review of literatureHiroshi Katoh0Tomoya Mitsuma1Riku Okamoto2Kanako Naito3Takaaki Tokito4Mariko Kikuchi5Takafumi Sangai6Department of Breast and Thyroid Surgery, Kitasato University Hospital, Kanagawa 252-0374, JapanDepartment of Breast and Thyroid Surgery, Kitasato University Hospital, Kanagawa 252-0374, JapanDepartment of Breast and Thyroid Surgery, Kitasato University Hospital, Kanagawa 252-0374, JapanDepartment of Breast and Thyroid Surgery, Kitasato University Hospital, Kanagawa 252-0374, JapanDepartment of Breast and Thyroid Surgery, Kitasato University Hospital, Kanagawa 252-0374, JapanDepartment of Breast and Thyroid Surgery, Kitasato University Hospital, Kanagawa 252-0374, JapanDepartment of Breast and Thyroid Surgery, Kitasato University Hospital, Kanagawa 252-0374, JapanParathyroid cancer (PC) is extremely resistant to chemotherapy and radiotherapy (RT), but hormonally functional by producing excessive parathyroid hormone (PTH), causing remarkable hypercalcemia even in biochemical disease recurrence. Accordingly, management of hypercalcemia by calcimimetics and bisphosphonates has been main treatment for unresectable PC. Here, we report a case of unresectable tumor mutational burden (TMB)-high recurrent PC that has been effectively controlled by pembrolizumab (PEM) with RT. A 48-year-old male patient, with previous history of left single parathyroidectomy for primary hyperparathyroidism, underwent surgeries for recurrent hyperparathyroidism at 47 and 48 years of age, and was pathologically diagnosed with PC. He was referred to our hospital due to persistent hypercalcemia and elevated PTH. The recurrent tumors were identified in the superior mediastinum and radically resected, then the hyperparathyroidism was improved. A FoundationOne® CDx of the specimen called TMB-high. He demonstrated recurrent hyperparathyroidism at 49 years of age, and underwent a gross curative resection. However, hyperparathyroidism achieved only insufficient improvement, indicating biochemical residual cancer cells. PEM treatment was initiated in combination with RT to the left central-lateral neck and superior mediastinum. He successfully achieved evocalcet and zoledronate withdrawal, and the PTH level improvement was continuously observed for 8 months at present, with only grade 2 subclinical hypothyroidism. Interestingly, leukocyte fraction ratios were reversed corresponding to disease improvement. A combination of PEM and RT is a promising treatment of unresectable TMB-high PC. Recent evidence on the immunomodulatory effect of RT provides the rationale for the combination of RT and PEM.https://www.jstage.jst.go.jp/article/endocrj/71/11/71_EJ24-0126/_html/-char/enpembrolizumabradiation therapyparathyroid cancerunresectabletumor mutational burden (tmb) |
| spellingShingle | Hiroshi Katoh Tomoya Mitsuma Riku Okamoto Kanako Naito Takaaki Tokito Mariko Kikuchi Takafumi Sangai Pembrolizumab with external radiation therapy effectively controlled TMB-high unresectable recurrent parathyroid cancer: a case report with review of literature Endocrine Journal pembrolizumab radiation therapy parathyroid cancer unresectable tumor mutational burden (tmb) |
| title | Pembrolizumab with external radiation therapy effectively controlled TMB-high unresectable recurrent parathyroid cancer: a case report with review of literature |
| title_full | Pembrolizumab with external radiation therapy effectively controlled TMB-high unresectable recurrent parathyroid cancer: a case report with review of literature |
| title_fullStr | Pembrolizumab with external radiation therapy effectively controlled TMB-high unresectable recurrent parathyroid cancer: a case report with review of literature |
| title_full_unstemmed | Pembrolizumab with external radiation therapy effectively controlled TMB-high unresectable recurrent parathyroid cancer: a case report with review of literature |
| title_short | Pembrolizumab with external radiation therapy effectively controlled TMB-high unresectable recurrent parathyroid cancer: a case report with review of literature |
| title_sort | pembrolizumab with external radiation therapy effectively controlled tmb high unresectable recurrent parathyroid cancer a case report with review of literature |
| topic | pembrolizumab radiation therapy parathyroid cancer unresectable tumor mutational burden (tmb) |
| url | https://www.jstage.jst.go.jp/article/endocrj/71/11/71_EJ24-0126/_html/-char/en |
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