HEREDITARY ENDOCRINE TUMOURS: CURRENT STATE-OF-THE-ART AND RESEARCH OPPORTUNITIES: New and future perspectives for parathyroid carcinoma

Perrier, Nancy D.; Arnold, Andrew; Costa-Guda, Jessica; Busaidy, Naifa L.; Nguyen, Ha H.; Chuang, Hubert H.; Brandi, Maria Luisa

doi:10.1530/erc-20-0018

Cited by 22 publications

(24 citation statements)

References 68 publications

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“…Application of next‐generation sequencing methods has significantly extended the landscape of genomic alterations in parathyroid carcinoma. Recent studies have suggested that genes from the PIK3CA/AKT/mTOR pathway were potentially targetable in parathyroid carcinoma 34,35 …”

Section: Discussionmentioning

confidence: 99%

Parathyroid carcinoma: Single centre experience

Cunha

Pinheiro²,

Donato

et al. 2022

Clinical Endocrinology

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Objective Parathyroid Carcinoma is a rare malignant neoplasm, accounting for less than 1% of primary hyperparathyroidism cases. Parathyroid carcinomas are characterized by markedly elevated levels of PTH, severe hypercalcemia and established target organ damage. The authors report the experience of a single centre regarding the management and outcome of patients with parathyroid carcinomas and revise relevant literature. Design Retrospective review of all patients with parathyroid carcinoma evaluated at a tertiary oncologic centre from 1991 until 2021. Results Seventeen patients were identified (10 males), with a mean age at diagnosis of 53 ± 16 years and a median follow‐up of 16.5 years. Most patients presented with hypercalcemia (n = 15), with a mean serum calcium concentration of 13.5 mg/dl (9.6–16.5) and mean PTH of 1173 pg/ml (276–2500). Hyperparathyroidism‐mediated organ damage was observed in most patients (n = 16), with predominant renal (n = 12) and skeletal (n = 9) complications. En bloc surgical resection was performed in nine patients. Three patients underwent adjuvant radiotherapy. Recurrence was observed in 8 cases (47.1%) after a median of 24 months following surgery and no independent predictors of recurrence were identified. The overall survival and disease specific survival at 5‐year was 88% and 94%, respectively. CDC73 mutations were present in 38.5% of analysed patients and one patient was diagnosed with MEN1. Conclusion Parathyroid carcinoma is associated with a significant rate of recurrence and limited effective treatment beyond initial complete surgical resection. Therefore, preoperatively high index of suspicion is paramount to optimize patient care. This is, to our knowledge, the largest Portuguese cohort published so far.

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Section: Discussionmentioning

confidence: 99%

Parathyroid carcinoma: Single centre experience

Cunha

Pinheiro²,

Donato

et al. 2022

Clinical Endocrinology

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“…PC is a radioresistant tumor, and there is no indication that radiotherapy is performed as a regular treatment ( 30 ). The PC analysis in the National Cancer Data Base (NCDB) showed that radiotherapy did not improve the OS rate of patients ( 31 ).…”

Section: Discussionmentioning

confidence: 99%

A Nomogram Predicting the Overall Survival and Cancer-Specific Survival in Patients with Parathyroid Cancer: A Retrospective Study

et al. 2022

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PurposeThis study aimed to explore a visual model for predicting the prognosis of patients with parathyroid carcinoma (PC) and analyze related biochemistries in different groups of stage.MethodsThe training dataset of 342 patients with PC was obtained from the Surveillance, Epidemiology, and End Results (SEER) database, and the validation dataset included 59 patients from The First Affiliated Hospital of Zhengzhou University. Univariate and multivariate Cox regression analyses were performed to evaluate significant independent prognostic factors. Based on those factors, nomograms and Web-based probability calculators were constructed to evaluate the overall survival (OS) and the cancer-specific survival (CSS) at 3, 5, and 8 years. The concordance index (C-index), receiver operating characteristic (ROC) curve, calibration curve, and decision curve analysis (DCA) were used to evaluate the nomogram in the training set and validation set. Moreover, biochemistries from the validation set were retrospectively analyzed in different groups of stage by Kruskal–Wallis test.ResultsAge, marital status, tumor size, stage, lymph node status, and radiation were identified as prognostic factors of OS. In contrast, only tumor size and stage were predictive for CSS. The nomogram was developed based on these independent factors. The C-index, ROC curve, calibration curve, and DCA of the nomogram in both training and validation sets showed that the nomogram had good predictive value, stability, and clinical benefit in predicting 3-, 5-, and 8-year OS and CSS in PC patients. Among the 59 PC patients from our hospital, lower albumin (ALB) levels and higher postoperative parathyroid hormone (PTH) levels were found in patients with distant metastasis (Distant vs. Regional ALB levels: p = 0.037; Distant vs. Local ALB levels: p = 0.046; Distant vs. Regional postoperative PTH levels: p = 0.002; Distant vs. Local postoperative PTH: p = 0.002).ConclusionThe established nomogram application can provide accurate prognostics for patients with PC in the Chinese population, but it must be validated on prospectively collected real-world data.

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“…Sorafenib was discontinued and a second more potent antiangiogenic inhibitor, lenvatinib, was initiated. After 20 months the disease was radiographically stable, and his calcium level was well controlled without any medication ( 30 ). When well-tolerated, TKIs ensure a radiographic and hormonal control of disease, associated with normalization of calcium, at least in the medium-short term.…”

Section: Discussionmentioning

confidence: 99%

Parathyroid Carcinoma All-In-One, a Rare Life-Threatening Case With Multiple Systemic Manifestations: Case Report and Review of the Literature

et al. 2022

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Parathyroid carcinoma (PC) is an extremely rare disease. Although it may occasionally occur in genetic syndromes, it is more often sporadic. It is usually associated with a consistent secretion of PTH, causing severe hypercalcemia and potentially all clinical conditions due to primary hyperparathyroidism. Management of PC can be challenging: some clinical, biochemical, and radiological features may be useful, but the final diagnosis of malignancy strictly relies on histological criteria. To date, radical surgery is the first-choice treatment and is the only effective therapy to control hypercalcemia and other clinical manifestations. On the other hand, chemo- or radiotherapy, local treatments, or novel drugs should be reserved for selected cases. We report an exceptionally unusual case of life-threatening PC, associated with several systemic manifestations: moderate pancreatitis, portal thrombosis, kidney stones, brown tumors, osteoporosis, hungry bone syndrome (HBS), chondrocalcinosis, neuropathy, and depression. The clinical case also represents an opportunity to provide a review of the recent literature, associated with a complete evaluation of the main diagnostic and therapeutic approaches.

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HEREDITARY ENDOCRINE TUMOURS: CURRENT STATE-OF-THE-ART AND RESEARCH OPPORTUNITIES: New and future perspectives for parathyroid carcinoma

Cited by 22 publications

References 68 publications

Parathyroid carcinoma: Single centre experience

Parathyroid carcinoma: Single centre experience

A Nomogram Predicting the Overall Survival and Cancer-Specific Survival in Patients with Parathyroid Cancer: A Retrospective Study

Parathyroid Carcinoma All-In-One, a Rare Life-Threatening Case With Multiple Systemic Manifestations: Case Report and Review of the Literature

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