Parathyroid carcinoma in a child: an unusual case of an ectopically located malignant parathyroid gland with tumor invading the thymus

Fiedler, Amy G.; Rossi, Christopher T.; Gingalewski, Cynthia

doi:10.1016/j.jpedsurg.2009.04.024

Cited by 22 publications

(8 citation statements)

References 22 publications

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“…A review of the literature identified only 12 other pediatric cases (Supplementary Table S1). Demographic details appear to match those of our patient with the median age of presentation reported to be 13 years of age and a near 1:1 male‐to‐female ratio. Of the eight cases in the literature with follow‐up and relapse data, 3 patients appear to have relapsed with pulmonary metastases within a timeframe of 6 months to 5 years.…”

Section: Discussionsupporting

confidence: 81%

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Severe Primary Hyperparathyroidism Caused by Parathyroid Carcinoma in a 13‐Year‐Old Child; Novel Findings From HRpQCT

et al. 2020

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Primary hyperparathyroidism is a condition that occurs infrequently in children. Parathyroid carcinoma, as the underlying cause of hyperparathyroidism in this age group, is extraordinarily rare, with only a few cases reported in the literature. We present a 13‐year‐old boy with musculoskeletal pain who was found to have brown tumors from primary hyperparathyroidism caused by parafibromin‐immunodeficient parathyroid carcinoma. Our patient had no clinical, biochemical, or radiographic evidence of pituitary adenomas, pancreatic tumors, thyroid tumors, pheochromocytoma, jaw tumors, renal abnormalities, or testicular lesions. Germline testing for AP2S1, CASR, CDC73/HRPT2, CDKN1B, GNA11, MEN1, PTH1R, RET, and the GCM2 gene showed no pathological variants, and a microarray of CDC73/HRPT2 did not reveal deletion or duplication. He was managed with i.v. fluids, calcitonin, pamidronate, and denosumab prior to surgery to stabilize hypercalcemia. After removal of a single parathyroid tumor, he developed severe hungry bone syndrome and required 3 weeks of continuous i.v. calcium infusion, in addition to oral calcium and activated vitamin D. Histopathological examination identified an angioinvasive parathyroid carcinoma with global loss of parafibromin (protein encoded by CDC73/HRPT2).HRpQCT and DXA studies were obtained prior to surgery and 18‐months postsurgery. HRpQCT showed a resolution of osteolytic lesions combined with structural improvement of cortical porosity and an increase in both cortical thickness and density compared with levels prior to treatment. These findings highlight the added value of HRpQCT in primary hyperparathyroidism. In addition to our case, we have provided a review of the published cases of parathyroid cancer in children. © 2019 The Authors. JBMR Plus published by Wiley Periodicals, Inc. on behalf of American Society for Bone and Mineral Research.

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Section: Discussionsupporting

confidence: 81%

“…The tumor cells were also variably positive for PGP9.5 (C) and galectin-3 (D), and showed reduced expression for p27 (E). While the tumor displayed intratumoral proliferative heterogeneity, the MIB-1 labeling index was as high as25.45% in hot spots (F).…”

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confidence: 99%

Severe Primary Hyperparathyroidism Caused by Parathyroid Carcinoma in a 13‐Year‐Old Child; Novel Findings From HRpQCT

et al. 2020

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“…We identified 17 cases of PC in children under 16 years old in the literature [1][2][3][4][5][6][7][8][9][10][11][12][13][14][15][16][17] (Table 1). Very high levels of serum calcium and i-PTH, a palpable neck mass, and severe clinical symptoms are the specific clinical features of PC [18] and seem to be characteristic of PC in children as well.…”

Section: Discussionmentioning

confidence: 99%

“…PC appearing in children often suggests the involvement of hereditary diseases, such as hyperparathyroidism-jaw tumor syndrome. Indeed, three previous pediatric patients had a family history of PHPT [4,6,7], and two had a confirmed germline CDC73 mutation [12,15].…”

Section: Discussionmentioning

confidence: 99%

Parathyroid carcinoma in a 13-year-old girl with a long-term survival

et al. 2020

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Background: Parathyroid carcinoma as a cause of primary hyperparathyroidism in children is extremely rare. We report a case of parathyroid carcinoma which occurred in a 13-year-old girl who survived for more than 45 years after the first operation. Case presentation: A woman was admitted to our hospital for the treatment of recurrent parathyroid carcinoma in the neck. She had been diagnosed with primary hyperparathyroidism from a fibula fracture and underwent parathyroidectomy at 13 years old. She had no family history of multiple endocrine neoplasia or jaw tumor syndrome. Genetic testing was not performed, and the histopathological diagnosis of the tumor had been parathyroid adenoma at the time. At 22 years old, she showed hypercalcemia after a femur fracture. Pulmonary metastases of parathyroid carcinoma in the bilateral lungs were found and surgically removed. Regarding the clinical course, her diagnosis was corrected from parathyroid adenoma to parathyroid carcinoma. At 33 years old, re-resection of the lung metastases was performed. For 10 years, her serum calcium level stayed within the normal range. However, her serum calcium level and intact parathyroid hormone eventually began to increase. Two masses suspected of being parathyroid carcinoma recurrence were found in the neck when she was 57 years old. En bloc resection was performed. Pathologically, the tumors were diagnosed as parathyroid adenoma. The serum calcium level and intact parathyroid hormone did not decrease after the operation. A 99m Tc-methoxy-isobutyl-isonitrile-and 18 F-fluorodeoxyglucose-negative, 11 C-methionine-positive tumor was detected at the right side of the trachea in the neck. The tumor was removed, along with the thyroid, muscle, and trachea that were involved. The pathological diagnosis was parathyroid carcinoma recurrence. The serum calcium level and intact parathyroid hormone decreased temporarily but had increased again 8 months later. Methionine-positive tumors were found at the right side of the trachea and suspected of being a recurrence. Denosumab reduced her serum calcium level, and radiation successfully suppressed the growth of the recurrent tumors. Conclusion: We have reported a rare case of parathyroid carcinoma in a child who has survived for over 40 years. Positron emission tomography of 11 C-methionine was useful for detecting local recurrence. This patient's long-term survival has been attributed to multimodality treatment including repeated surgery, medication, and radiation.

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“…It accounts for less than 1% of cases of HPT in adults and it is probably even rarer in children, although the precise incidence of parathyroid cancer in the pediatric population is unknown (6)(7)(8). Adenocarcinoma of the parathyroid is defined by gross or histologic invasion of blood vessels, perineural tissue, thyroid gland, or other surrounding tissues, or by the presence of distant metastases.…”

Section: Hyperparathyroidismmentioning

confidence: 99%