A case of primary hyperparathyroidism due to an intrathymic ectopic parathyroid adenoma in a 15-year-old boy
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Hypercalcemia due to primary hyperparathyroidism (PHPT) is uncommon in children. PHPT is typically caused by a single parathyroid adenoma. Ectopic parathyroid adenomas account for 6%–16% of all parathyroid adenomas and are rare in children but should be considered in cases that present with hypercalcemia. We report the case of a 15-year-old boy with PHPT due to an intrathymic ectopic parathyroid adenoma. Neck ultrasonography and Tc-99m-sestamibi (MIBI) scanning with single-photon emission computed tomography/computed tomography (SPECT/CT) revealed ectopic parathyroid adenoma in the thymus. Video-assisted thoracoscopic surgery was performed to remove the ectopic parathyroid adenoma. Pathology showed intrathymic ectopic parathyroid adenoma. After surgery, the patient’s serum calcium level immediately normalized. Intact parathyroid hormone (iPTH) and alkaline phosphatase levels returned to normal ranges within 3 months. Delayed diagnosis of PHPT can cause end-organ damage; a timely diagnosis is especially critical to preserve bone and renal function. If ectopic parathyroid adenomas are well localized in preoperative imaging evaluation and intraoperative iPTH level decreases after resection, ectopic parathyroidectomy without bilateral neck exploration may be performed to avoid unnecessary morbidity.
Abstract
Hypercalcemia due to primary hyperparathyroidism (PHPT) is uncommon in children. PHPT is typically caused by a single parathyroid adenoma. Ectopic parathyroid adenomas account for 6%–16% of all parathyroid adenomas and are rare in children but should be considered in cases that present with hypercalcemia. We report the case of a 15-year-old boy with PHPT due to an intrathymic ectopic parathyroid adenoma. Neck ultrasonography and Tc-99m-sestamibi (MIBI) scanning with single-photon emission computed tomography/computed tomography (SPECT/CT) revealed ectopic parathyroid adenoma in the thymus. Video-assisted thoracoscopic surgery was performed to remove the ectopic parathyroid adenoma. Pathology showed intrathymic ectopic parathyroid adenoma. After surgery, the patient’s serum calcium level immediately normalized. Intact parathyroid hormone (iPTH) and alkaline phosphatase levels returned to normal ranges within 3 months. Delayed diagnosis of PHPT can cause end-organ damage; a timely diagnosis is especially critical to preserve bone and renal function. If ectopic parathyroid adenomas are well localized in preoperative imaging evaluation and intraoperative iPTH level decreases after resection, ectopic parathyroidectomy without bilateral neck exploration may be performed to avoid unnecessary morbidity.
참고문헌 (19)
- McNeilly JD, "Frequency and aetiology of hypercalcaemia", Arch Dis Child, 101, pp.344~347 (2016)
- Stokes VJ, "Hypercalcemic disorders in children", J Bone Miner Res, 32, pp.2157~2170 (2017)
- Bilezikian JP, "Primary hyperparathyroidism", J Clin Endocrinol Metabol, 103, pp.3993~4004 (2018)
- Cormier C, "Genetic hypercalcemia", Joint Bone Spine, 86, pp.459~466 (2019)
- Walker MD, "Primary hyperparathyroidism", Nat Rev Endocrinol, 14, pp.115~125 (2018)
- Kollars J, "Primary hyperparathyroidism in pediatric patients", Pediatrics, 115, pp.974~980 (2005)
- Nicholson KJ, "Comparative characteristics of primary hyperparathyroidism in pediatric and young adult patients", Surgery, 160, pp.1008~1016 (2016)
- Roy M, "Incidence and localization of ectopic parathyroid adenomas in previously unexplored patients", World J Surg, 37, pp.102~106 (2013)
- Li CC, "A 10-year retrospective study of primary hyperparathyroidism in children", Exp Clin Endocrinol Diabetes, 120, pp.229~233 (2012)
- Liu X, "Primary hyperparathyroidism due to ectopic parathyroid adenoma in an adolescent : a case report and review of the literature", Endocrine, 64, pp.38~42 (2019)
- Arambewela MH, "Case report : rare skeletal manifestations in a child with primary hyperparathyroidism", BMC Endocr Disord, 17, pp.45~ (2017)
- Udelsman R, "The surgical management of asymptomatic primary hyperparathyroidism : proceedings of the Fourth International Workshop", J Clin Endocrinol Metab, 99, pp.3595~3606 (2014)
- Khan AA, "Primary hyperparathyroidism : review and recommendations on evaluation, diagnosis, and management. A Canadian and international consensus", Osteoporos Int, 28, pp.1~19 (2017)
- Cheung K, "A meta-analysis of preoperative localization techniques for patients with primary hyperparathyroidism", Ann Surg Oncol, 19, pp.577~583 (2012)
- Iihara M, "Thoracoscopic removal of mediastinal parathyroid lesions : selection of surgical approach and pitfalls of preoperative and intraoperative localization", World J Surg, 36, pp.1327~1334 (2012)
- Kaplan EL, "Primary hyperparathyroidism in the 1990s. Choice of surgical procedures for this disease", Ann Surg, 215, pp.300~317 (1992)
- Chou PL, "Minimally invasive removal of mediastinal ectopic parathyroid glands : a single-center experience", Formos J Surg, 52, pp.6~10 (2019)
- Said SM, "Minimally invasive resection for mediastinal ectopic parathyroid glands", Ann Thorac Surg, 96, pp.1229~1233 (2013)
- Thakker RV, "Clinical practice guidelines for multiple endocrine neoplasia type 1 (MEN1)", J Clin Endocrinol Metabol, 97, pp.2990~3011 (2012)