Intrathyroidal parathyroid carcinoma mimicking a thyroid nodule in a MEN type 1 patient

Lee, Kyung Mi; Kim, Eui Jong; Choi, Woo Suk; Park, Won Seo; Kim, Sung Won

doi:10.1002/jcu.22090

Cited by 27 publications

(25 citation statements)

References 13 publications

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“…In our case, the left upper parathyroid gland was ectopically situated in the thyroid. This anatomical variation made it more difficult to diagnose PC prior to surgery and similar to a case reported by Lee et al [15]. Tamiya et al reported the positive correlation between i-PTH and total calculated volume of parathyroid glands [19].…”

supporting

confidence: 68%

“…Endocrine Journal Advance Publication Discussion PHPT of MEN1 is mostly caused by parathyroid hyperplasia, and PC is quite rare as the causasive lesion with a prevalence reported as 0.28 to 2% [3,5,6]. There were 14 cases of MEN1-associated PC previously reported (Table 2) [5][6][7][8][9][10][11][12][13][14][15][16]. Very high levels of serum Ca and i-PTH, a palpable neck mass, and severe bone disease such as osteitis fibrosa cystica are the specific clinical features of PC [17].…”

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confidence: 99%

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Parathyroid carcinoma occurred in two glands in multiple endocrine neoplasia 1: a report on a rare case

et al. 2018

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Abstract. Primary hyperparathyroidism is the most common hormonal manifestation associated with multiple endocrine neoplasia 1 (MEN1). It is generally caused by parathyroid hyperplasia, and parathyroid carcinoma is rare. Here, we report a case of MEN1 with parathyroid carcinoma in two parathyroid glands causing primary hyperparathyroidism. A 40-year-old man with primary hyperparathyroidism due to MEN1 underwent a total parathyroidectomy. His corrected calcium and intact PTH (i-PTH) serum levels were 10.8 mg/dL and 203 pg/mL, respectively. Although three glands were successfully removed, the left upper parathyroid gland could not be detected. Since the right lower parathyroid lesion had invaded into the thyroid, right lobectomy was performed. A portion of the left lower parathyroid tissue was transplanted into his forearm. The histological findings of the left lower and the right upper parathyroid glands were consistent with hyperplasia while that of the right lower parathyroid gland was parathyroid carcinoma. Since the post-surgical i-PTH levels remained high, the intrathyroidal lesion of the left lobe, which was initally diagnosed as an adenomatous nodule, was suspected to contain parathyroid tumor. A fine needle aspiration of the tumor revealed a high concentration of i-PTH. One week after the first surgery, a left thyroid lobectomy was performed. The pathological diagnosis of the tumor was parathyroid carcinoma. After the surgery, calcium and i-PTH levels were normal. Although it is rare, parathyroid carcinoma should be considered as a cause of hyperparathyroidism in MEN1 patients. Since it is difficult to diagnose parathyroid carcinoma before surgery, intraoperative findings are important for the appropriate treatment. nancy that is found in less than 5% of PHPT patients [2]. PC occurs only in 2% of MEN1 patients that have received surgery for PHPT [3] in contrast to the 15% of PC found in hyperparathyroidism-jaw tumor syndrome patients [4]. Here, we report a case of MEN1 with PC causing primary hyperparathyroidism. Case presentationA 40-year-old man was referred for follow up of MEN1. His mother was diagnosed as PHPT due to MEN1, 3 years before by his previous doctor but she had not yet been treated. She also had non-functioning pancreatic tumor, lung cartinoid and was suspected as having prolactinoma as her serum PRL level elavated and pituitary gland swelled. At that time, the previous doctor examined his serum calcium (Ca) and intact-PTH (i-PTH) level and detected hypercalcemia and elevation of

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confidence: 68%

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confidence: 99%

Parathyroid carcinoma occurred in two glands in multiple endocrine neoplasia 1: a report on a rare case

et al. 2018

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“…In a review of the literature, we found 10 case reports of patients with PC and MEN1 . In our cohort, the prevalence of PC in patients with MEN1 was found to be 0·28% (95% CI, 0–1·4%).…”

Section: Discussionmentioning

confidence: 64%

“…5 In a review of the literature, we found 10 case reports of patients with PC and MEN1. [9][10][11][12][13][14][15][16][17] In our cohort, the prevalence of PC in patients with MEN1 was found to be 0Á28% (95% CI, 0-1Á4%). Our point estimate for prevalence is lower than the reported prevalence of PC in patients with PHP of 0Á74 (95% CI, 0Á53-0Á95), which is based on a systematic review of more than 20 000 patients; however, the confidence intervals overlap.…”

Section: Discussionmentioning

confidence: 99%

Prevalence of parathyroid carcinoma in 348 patients with multiple endocrine neoplasia type 1 – case report and review of the literature

Ospina

Sebo

Thompson

et al. 2015

Clinical Endocrinology

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“…Anecdotally, parathyroid carcinomas are described in MEN1, but this remains rare (del Pozo et al 2011, Lee et al 2014, Singh Ospina et al 2016.…”

Section: Primary Hyperparathyroidismmentioning

confidence: 99%

The future: medical advances in MEN1 therapeutic approaches and management strategies

Leeuwaarde

Laat

Pieterman

et al. 2017

Endocrine-Related Cancer

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Multiple endocrine neoplasia type 1 is a rare autosomal inherited disorder associated with a high risk for patients to simultaneously develop tumors of the parathyroid glands, duodenopancreatic neuroendocrine tumors and tumors of the anterior pituitary gland. Early identification of MEN1 in patients enables presymptomatic screening of manifestations, which makes timely interventions possible with the intention to prevent morbidity and mortality. Causes of death nowadays have shifted toward local or metastatic progression of malignant neuroendocrine tumors. In early cohorts, complications like peptic ulcers in gastrinoma, renal failure in hyperparathyroidism, hypoglycemia and acute hypercalcemia were the primary causes of early mortality. Improved medical treatments of these complications led to a significantly improved life expectancy. The MEN1 landscape is still evolving, considering the finding of breast cancer as a new MEN1-related manifestation and ongoing publications on follow-up and medical care for patients with MEN1. This review aims at summarizing the most recent insights into the follow-up and medical care for patients with MEN1 and identifying the gaps for future research.

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Intrathyroidal parathyroid carcinoma mimicking a thyroid nodule in a MEN type 1 patient

Cited by 27 publications

References 13 publications

Parathyroid carcinoma occurred in two glands in multiple endocrine neoplasia 1: a report on a rare case

Parathyroid carcinoma occurred in two glands in multiple endocrine neoplasia 1: a report on a rare case

Prevalence of parathyroid carcinoma in 348 patients with multiple endocrine neoplasia type 1 – case report and review of the literature

The future: medical advances in MEN1 therapeutic approaches and management strategies

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