Case report: Long-term survival with anlotinib in a metastatic parathyroid carcinoma patient harboring EGFR-sensitizing mutation

Abstract

Abstract Parathyroid carcinoma (PC) is a rare and aggressive endocrine malignancy with limited treatment options. Despite the lack of standard management approaches to prevent recurrence and metastasis, current treatments such as chemotherapy and radiotherapy have demonstrated limited efficacy. Here, we report the case of a 54-year-old male patient who presented with a range of symptoms including polydipsia, polyuria, and joint pain. Further examination revealed a neck lump, hypercalcemia, and hyperparathyroidism, leading to a diagnosis of PC after en bloc surgery. Seven months following initial treatment, the patient was found to have local recurrence and distant metastases in the lung, which were subsequently resected via left lateral neck dissection and thoracoscopic wedge resection. Given the rapid progression of the patient's tumors, a 422-gene panel test was conducted, revealing the presence of epidermal growth factor receptor (EGFR) p.L858R (c. T2573G) mutation, which may sensitize the EGFR-TKI response, and PIK3CA p. E545KV(c. G1633A) mutation, which can potentially be targeted by everolimus. Despite treatment with gefitinib and everolimus, neither therapy yielded a response. After multidisciplinary treatment discussions, the patient was advised to undergo a multi-target tyrosine kinase inhibitor, anlotinib, which resulted in continuous survival benefits for 19 months. This case highlights the potential of targeted therapy to achieve long-term survival in cases of PC with distant metastases. It also underscores the importance of precision therapy guided by genome sequencing to identify potential targets for treatment. Further multicenter studies are needed to standardize the treatment of recurrent or metastatic PC.