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Shi C, Lu N, Yong YJ, Chu HD, Xia AJ. Parathyroid carcinoma: Three case reports. World J Clin Cases 2023; 11:5934-5940. [PMID: 37727485 PMCID: PMC10506032 DOI: 10.12998/wjcc.v11.i25.5934] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/12/2023] [Revised: 07/14/2023] [Accepted: 08/01/2023] [Indexed: 09/01/2023] Open
Abstract
BACKGROUND Parathyroid carcinoma (PC) is a rare, slow-growing malignant tumor and a rare cause of primary hyperfunctioning of the parathyroid, with a highly variable clinical course, depending on the aggressiveness of the individual tumor and the degree of hypercalcemia. CASE SUMMARY The aim of this report is to summarize the diagnosis and treatment of three cases of PC and to review and conclude aspects regarding the three collected cases with reference to other relevant cases to explore the value of ultrasound in the diagnosis of PC. All three patients had hypercalcemia, consisting of a high serum calcium level and a high level of parathyroid hormone that was > 2-fold (even > 30-fold) of the normal upper limit. The ultrasonographic findings of the parathyroid gland showed that the glands were all > 30 mm, and the internal echo was uneven. All patients underwent surgery. PC in three cases was confirmed by routine histopathology and immunohistochemistry. CONCLUSION As clinical signs and laboratory results are nonspecific, it is difficult to diagnose PC preoperatively, so imaging examinations are often needed.
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Affiliation(s)
- Ce Shi
- Department of Ultrasound, Yantaishan Hospital, Yantai 264003, Shandong Province, China
| | - Ning Lu
- Department of Pathology, Yantaishan Hospital, Yantai 264003, Shandong Province, China
| | - Yan-Jie Yong
- Department of Ultrasound, Yantaishan Hospital, Yantai 264003, Shandong Province, China
| | - Hai-Di Chu
- Department of Thyroid Surgery, Yantaishan Hospital, Yantai 264003, Shandong Province, China
| | - Ai-Jun Xia
- Department of Ultrasound, Yantaishan Hospital, Yantai 264003, Shandong Province, China
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Abstract
INTRODUCTION Parathyroid carcinoma (PC) is a rare endocrine disorder, commonly causing severe primary hyperparathyroidism (PHPT). PC is mainly a sporadic disease, but it may occur in familial PHPT. Patients with PC usually present markedly elevated serum calcium and PTH. The clinical features are mostly due to the effects of the excessive secretion of PTH rather than to the spread of tumor. At times, the diagnosis can be difficult. PURPOSE The aim of this work is to review the available data on PC, and focus its molecular pathogenesis and the clinical utility of CDC73 genetic testing and immunostaining of its product, parafibromin. The pathological diagnosis of PC is restricted to lesions showing unequivocal growth into adjacent tissues or metastasis. Inactivating mutations of the cell division cycle 73 (CDC73) gene have been identified in up to 70 % of apparently sporadic PC and in one-third are germline. Loss of parafibromin immunostaining has been shown in most PC. The association of CDC73 mutations and loss of parafibromin predicts a worse clinical outcome and a lower overall 5- and 10-year survival. CONCLUSIONS The treatment of choice is the en bloc resection of the tumor. The course of PC is variable; most patients have local recurrences or distant metastases and die from unmanageable hypercalcemia.
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Affiliation(s)
- F Cetani
- University Hospital of Pisa, Endocrine Unit 2, Via Paradisa, 2, 56124, Pisa, Italy.
| | - E Pardi
- Department of Clinical and Experimental Medicine, University of Pisa, Endocrine Unit 2, Pisa, Italy
| | - C Marcocci
- Department of Clinical and Experimental Medicine, University of Pisa, Endocrine Unit 2, Pisa, Italy
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Yang Z, Guo M, Wu B, Zheng Q, Fan Y. Focused parathyroidectomy through an open-lateral approach for treating solitary parathyroid adenoma. SURGICAL PRACTICE 2015. [DOI: 10.1111/1744-1633.12149] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/29/2022]
Affiliation(s)
- Zhili Yang
- Center of Thyroid and Parathyroid; Department of General Surgery; Shanghai Jiao Tong University Affiliated Sixth People's Hospital; Shanghai China
| | - Minggao Guo
- Center of Thyroid and Parathyroid; Department of General Surgery; Shanghai Jiao Tong University Affiliated Sixth People's Hospital; Shanghai China
| | - Bo Wu
- Center of Thyroid and Parathyroid; Department of General Surgery; Shanghai Jiao Tong University Affiliated Sixth People's Hospital; Shanghai China
| | - Qi Zheng
- Center of Thyroid and Parathyroid; Department of General Surgery; Shanghai Jiao Tong University Affiliated Sixth People's Hospital; Shanghai China
| | - Youben Fan
- Center of Thyroid and Parathyroid; Department of General Surgery; Shanghai Jiao Tong University Affiliated Sixth People's Hospital; Shanghai China
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Cetani F, Frustaci G, Torregrossa L, Magno S, Basolo F, Campomori A, Miccoli P, Marcocci C. A nonfunctioning parathyroid carcinoma misdiagnosed as a follicular thyroid nodule. World J Surg Oncol 2015; 13:270. [PMID: 26350418 PMCID: PMC4563849 DOI: 10.1186/s12957-015-0672-9] [Citation(s) in RCA: 15] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/15/2015] [Accepted: 08/03/2015] [Indexed: 01/26/2023] Open
Abstract
Parathyroid carcinoma (PC) is a rare endocrine malignancy. The tumor is mostly functioning, causing severe primary hyperparathyroidism, with high serum calcium and parathyroid hormone (PTH) levels. Nonfunctioning PC is extremely rare. We report a 50-year-old male patient who was referred to our Department for a right thyroid nodule, incidentally detected on carotid Doppler ultrasound scan, with a fine-needle aspiration cytology showing a follicular lesion. At the time of our evaluation, neck ultrasound showed a 1.3 cm right hypoechoic thyroid nodule with irregular margins and the absence of enlarged bilateral cervical lymph nodes. Thyroid function tests were normal. Serum calcium was normal and plasma PTH slightly above the upper limit of the normal range. The patients underwent right lobectomy. The intraoperative frozen-section pathological examination raised the suspicion of a PC. Definitive histology showed a markedly irregular infiltrative growth of the tumor with invasion of the thyroid tissue and cervical soft tissues. Immunostaining for thyroglobulin was negative, whereas staining for chromogranin A and PTH showed a strong reactivity. Based on the microscopic findings and the immunohistochemical profile, the tumor was diagnosed as a PC. Postoperative serum calcium and phosphate levels were in the normal range. One month after surgery, serum calcium and PTH were normal. Neck ultrasound and total body computed tomography scan were negative for local and metastatic disease. Eight months later, serum calcium was normal and plasma PTH level remained around the upper limit of normal range. Neck ultrasound did not show any pathological lesions. This is the first case of a nonfunctioning sporadic PC misdiagnosed prior of surgery as a follicular thyroid nodule. The parathyroid nature of the neck lesion could not be suspected before surgery. Fine-needle aspiration cytology (FNAC) may fail to distinguish a parathyroid tumor from a benign thyroid nodule because at FNAC, parathyroid and thyroid lesions have some morphological similarities. Histological criteria are not always sufficient for the differential diagnosis, which can definitely be established using immunohistochemistry.
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Affiliation(s)
- Filomena Cetani
- Endocrine Unit 2, University Hospital of Pisa, Via Paradisa 2, 56124, Pisa, Italy.
| | - Gianluca Frustaci
- Department of Surgical, Medical and Molecular Pathology and Critical Area, University of Pisa, Pisa, Italy.
| | - Liborio Torregrossa
- Department of Surgical, Medical and Molecular Pathology and Critical Area, University of Pisa, Pisa, Italy.
| | - Silvia Magno
- Department of Clinical and Experimental Medicine, University of Pisa, Pisa, Italy.
| | - Fulvio Basolo
- Department of Surgical, Medical and Molecular Pathology and Critical Area, University of Pisa, Pisa, Italy.
| | - Alberto Campomori
- Endocrine Unit 2, University Hospital of Pisa, Via Paradisa 2, 56124, Pisa, Italy.
| | - Paolo Miccoli
- Department of Surgical, Medical and Molecular Pathology and Critical Area, University of Pisa, Pisa, Italy.
| | - Claudio Marcocci
- Department of Clinical and Experimental Medicine, University of Pisa, Pisa, Italy.
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Kim DDW, Que L, Raizis AM, Florkowski CM. Novel inactivating mutation of the calcium-sensing receptor in a young woman with mild hypercalcaemia. Intern Med J 2014; 44:413-6. [PMID: 24754691 DOI: 10.1111/imj.12383] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/06/2013] [Accepted: 11/24/2013] [Indexed: 11/30/2022]
Abstract
A young woman with mild hypercalcaemia and an inappropriately normal serum parathyroid hormone had parathyroid scintigraphy suggestive of an active ectopic parathyroid tissue in the superior mediastinum. Urinary calcium to creatinine clearance ratio was low, and a subsequent genetic analysis confirmed a novel mutation (Q164K) in the calcium sensing receptor gene, consistent with familial hypocalciuric hypercalcaemia. We propose that this mutation accounts for her clinical and investigational findings, although a double pathology of Q164K and an ectopic parathyroid adenoma is also conceivable.
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Affiliation(s)
- D D W Kim
- Department of Endocrinology and Diabetes, Waitemata District Health Board, Auckland, New Zealand
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Four ultrasound and clinical pictures of parathyroid carcinoma. Case Rep Endocrinol 2012; 2012:363690. [PMID: 23213580 PMCID: PMC3505635 DOI: 10.1155/2012/363690] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/18/2012] [Accepted: 10/24/2012] [Indexed: 11/18/2022] Open
Abstract
Parathyroid carcinoma is a rare cause of primary hyperparathyroidism. It may be suspected based on severe clinical signs, significant laboratory findings, and the tumor size. High-resolution ultrasonography with Doppler imaging has become the principal imaging method in the preoperative diagnosis of primary hyperparathyroidism. The ultrasound finding is not specific, but three of the described pictures are different from the typical appearance of benign adenoma of the thyroid gland and were suspicious in the context of clinical findings. According to the ultrasound criteria, one finding was benign and only the histological examination revealed carcinoma.
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Cruz RP, Padoin AV, Vilhordo DW, Hoffmann A, Mottin CC. Use of a gamma probe to identify and guide resection of recurrent parathyroid carcinoma: report of a case. Surg Today 2011; 41:237-41. [PMID: 21264760 DOI: 10.1007/s00595-009-4233-0] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/15/2009] [Accepted: 10/22/2009] [Indexed: 11/25/2022]
Abstract
Parathyroid carcinoma (PC) accounts for less than 0.005% of all cancers and less than 5% of causes of hyperparathyroidism. This tumor is difficult to identify during surgery, which is detrimental to the oncologic results. Surgery is still the main treatment for the primary tumor and to control parathyroid hormone levels after recurrence. We report a case of recurrent parathyroid carcinoma in a 30-year-old man, identified and managed with the use of a gamma probe during surgery. To our knowledge, this is only the second report of a gamma probe being used to guide resection of a recurrent PC. We discuss the diagnosis and treatment, analyzing the current evidence-based literature.
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Affiliation(s)
- Ricardo Pedrini Cruz
- Serviço de Cirurgia Geral, Ambulatório de Cirurgia Endocrinológica, Hospital São Lucas da Pontifícia Universidade Católica do Rio Grande do Sul, Porto Alegre, RS, Brazil
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Gassmann P, Senninger N, Colombo-Benkmann M. Intraoperative calcium monitoring is insufficient to predict the surgical success of parathyroidectomy for primary hyperparathyroidism. Surg Today 2010; 40:1123-8. [PMID: 21110154 DOI: 10.1007/s00595-009-4191-6] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/02/2009] [Accepted: 11/12/2009] [Indexed: 10/18/2022]
Abstract
PURPOSE Serum calcium has been suggested to be a surrogate marker for intraoperative monitoring during a parathyroidectomy (PTX) for primary hyperparathyroidism (pHPT). The objective of the present prospective study was to investigate whether serum calcium can be used as an alternative for parathyroid hormone intraoperative monitoring. METHODS Intact parathyroid hormone (iPTH), total and ionized serum calcium, total protein (tP), and pH were monitored in parallel in 25 patients during a successful directed PTX for pHPT. RESULTS All patients had at least a 50% decrease of iPTH 10 min after PTX. Simultaneously measured total serum calcium (tCa) and ionized serum calcium (iCa) did not correlate with iPTH or pH. However, tCa and iCa levels correlated with changes in tP (r = 0.859; P < 0.001; r = 0.483; P = 0.014). In addition, intraoperative iCa changes correlated with preoperative iCa levels (r = -0.475, P = 0.016). A linear regression analysis for parameters potentially influencing intraoperative tCa and iCa dynamics changes identified tP changes as the only parameter influencing tCa and iCa changes (P < 0.001). CONCLUSION Individual short-term intraoperative changes of tCa or iCa are not predictive for successful PTX, as they more likely reflect changes in intraoperative fluid management rather than decreased iPTH.
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Affiliation(s)
- Peter Gassmann
- Department of General and Visceral Surgery, University Hospital Muenster, Waldeyerstrasse 1, 48149, Muenster, Germany
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McMullen TPW, Learoyd DL, Williams DC, Sywak MS, Sidhu SB, Delbridge LW. Hyperparathyroidism in pregnancy: options for localization and surgical therapy. World J Surg 2010; 34:1811-6. [PMID: 20386905 DOI: 10.1007/s00268-010-0569-2] [Citation(s) in RCA: 68] [Impact Index Per Article: 4.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/30/2022]
Abstract
BACKGROUND Hyperparathyroidism in pregnancy is a threat to the health of both mother and fetus. The mothers suffer commonly from nephrolithiasis, hyperemesis, or even hypercalcemic crisis. Untreated disease will commonly complicate fetal development and fetal death is a significant risk. Treatment options, including medical and surgical therapy, are debated in the literature. METHODS This is a case series comprising seven patients with primary hyperparathyroidism in pregnancy. Data collected included symptoms at diagnosis, biochemical abnormalities, pathologic findings, treatment regimes, and subsequent maternal and fetal outcomes. RESULTS Seven women, aged 20 to 39 years, presented with hyperparathyroidism during pregnancy. The earliest presented at 8 weeks and the latest at 38 weeks. Four of seven patients experienced renal calculi. Calcium levels were 2.7-3.5 mmol/l. All were found to have solitary parathyroid adenomas, of which two were in ectopic locations. Fetal complications included three preterm deliveries and one fetal death with no cases of neonatal tetany. Maternal and fetal complications could not be predicted based on duration or severity of hypercalcemia. Three patients were treated during pregnancy with surgery, and two of these had ectopic glands that required reoperations with a novel approach using Tc-99m sestamibi scanning during pregnancy to assist in localizing the abnormal gland. Four cases were treated postpartum with a combination of open and minimally invasive approaches after localization. No operative complications or fetal loss related to surgery were observed in this cohort. CONCLUSIONS Primary hyperparathyroidism in pregnancy represents a significant risk for maternal and fetal complications that cannot be predicted by duration of symptoms or serum calcium levels. Surgical treatment should be considered early, and a minimally invasive approach with ultrasound is best suited to mitigating risk to mother and fetus. Equally important, Tc-99m sestamibi imaging may be used safely for localization of the parathyroids after negative cervical explorations.
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Affiliation(s)
- Todd P W McMullen
- Department of Surgery 2D, Walter C. Mackenzie Health Science Centre, 8440-112 Street, Edmonton, AB, T6G 2B7, Canada.
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Sendt W, Spieker K, Michael G, Jandt K, Altendorf-Hofmann A. Radio-guided parathyroidectomy in patients with primary hyperparathyroidism and concomitant multinodular goiter. Surg Today 2010; 40:825-30. [DOI: 10.1007/s00595-009-4144-0] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/26/2009] [Accepted: 07/21/2009] [Indexed: 11/24/2022]
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[Hypercalcemic crisis as first manifestation of primary hyperparathyroidism in adolescence]. Cir Esp 2010; 88:418-9. [PMID: 20362280 DOI: 10.1016/j.ciresp.2010.01.005] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/02/2009] [Revised: 01/08/2010] [Accepted: 01/17/2010] [Indexed: 11/22/2022]
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Graff-Baker A, Roman SA, Boffa D, Aslanian H, Sosa JA. Diagnosis of Ectopic Middle Mediastinal Parathyroid Adenoma Using Endoscopic Ultrasonography-Guided Fine-Needle Aspiration with Real-Time Rapid Parathyroid Hormone Assay. J Am Coll Surg 2009; 209:e1-4. [DOI: 10.1016/j.jamcollsurg.2009.05.023] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/03/2009] [Revised: 05/18/2009] [Accepted: 05/18/2009] [Indexed: 11/16/2022]
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Abstract
PURPOSE OF REVIEW Imaging is a critical component of both neuroendocrine lesion identification and anatomic delineation for treatment planning. Cross-sectional and isotopic-based physiologic imaging techniques have, to date, been the radiological modalities of choice. This review focuses on recent advances in imaging that are related to the evaluation of neuroendocrine abnormalities, in particular advances in MRI. RECENT FINDINGS Magnetic-resonance perfusion examination of tissue characteristics in the pituitary, adrenal and thyroid glands indicates that, in many cases, adenomas of these glands have distinguishable hemodynamic characteristics relative to the parenchyma of the gland as a whole and to other lesions. Moreover, perfusion metrics might provide a basis for evaluating response to therapy (in the pituitary) and delineation of lesions in the adrenal and thyroid glands. Anisotropy-based imaging techniques show promise in providing direct, relevant information about pituitary tumor involvement of the adjacent cavernous sinuses. SUMMARY The most recent methodological advances in the imaging of neuroendocrine disorder involves the continued development and application of MRI, in particular using pulse sequences, which provide a greater insight into the internal structure and physiology of the tissues interrogated, relative to standard sequences.
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Affiliation(s)
- Bart P Keogh
- Radia Inc. P.S. & Swedish Neuroscience Institute, Seattle, Washington 98122, USA.
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