Sandakly Nicolas, Zreika Bachir, Haddad Fady, Ghorra Claude, Abboud Bassam
Faculty of Medical Sciences, Lebanese University, Hadath Campus, Lebanon; Department of Internal Medicine, Lebanese Hospital Geitaoui University Medical Center, Lebanon.
Faculty of Medical Sciences, Lebanese University, Hadath Campus, Lebanon; Department of General Surgery, Lebanese Hospital Geitaoui University Medical Center, Lebanon.
Int J Surg Case Rep. 2025 Apr 21;131:111326. doi: 10.1016/j.ijscr.2025.111326.
Primary hyperparathyroidism (PHPT) is the most common cause of hypercalcemia and has shifted from a rare condition to a common endocrine disorder. Advances in diagnostic methods have led to more incidental diagnoses, particularly in asymptomatic patients. Ectopic parathyroid adenomas are a rare cause of PHPT, with the vast majority of cases involving parathyroid glands in typical anatomical locations. However, the occurrence of a fifth parathyroid adenoma is exceptionally uncommon, especially when located in an ectopic position.
We report an uncommon case of primary hyperparathyroidism in a 60-year-old male with longstanding hypercalcemia attributed to a fifth ectopic parathyroid adenoma localized in the aortopulmonary window. Initial imaging modalities failed to localize the adenoma, which was detected by 4D-CT but not on SPECT/CT, and remission was achieved through surgical resection, with intraoperative PTH monitoring confirming total resection.
Delayed diagnosis of ectopic parathyroid adenomas can lead to complications, as in our patient with nephrolithiasis and hypercalcemia. Advanced imaging techniques like 4D-CT are crucial when conventional methods fail. Embryological anomalies can result in ectopic or supernumerary parathyroid glands, complicating localization. Intraoperative PTH monitoring ensures complete resection and lowers reoperation rates.
This case highlights the challenges of diagnosing ectopic parathyroid adenomas and the importance of considering rare ectopic adenomas in patients with PHPT, particularly when standard imaging doesn't yield conclusive results. We also discuss the role of advanced imaging modalities and intraoperative PTH monitoring in guiding surgical decisions and determining the appropriate point to conclude the procedure, ensuring successful outcomes.
原发性甲状旁腺功能亢进症（PHPT）是高钙血症最常见的病因，已从一种罕见疾病转变为常见的内分泌疾病。诊断方法的进步导致更多的偶然诊断，尤其是在无症状患者中。异位甲状旁腺腺瘤是PHPT的罕见病因，绝大多数病例涉及典型解剖位置的甲状旁腺。然而，第五个甲状旁腺腺瘤的发生极为罕见，尤其是位于异位位置时。
我们报告了一例罕见的原发性甲状旁腺功能亢进症病例，患者为一名60岁男性，长期高钙血症归因于位于主肺动脉窗的第五个异位甲状旁腺腺瘤。最初的影像学检查未能定位腺瘤，4D-CT检测到了腺瘤，但SPECT/CT未检测到，通过手术切除实现了缓解，术中甲状旁腺激素监测证实为完全切除。
异位甲状旁腺腺瘤的延迟诊断可导致并发症，如我们的患者出现肾结石和高钙血症。当传统方法失败时，像4D-CT这样的先进成像技术至关重要。胚胎学异常可导致异位或额外的甲状旁腺，使定位复杂化。术中甲状旁腺激素监测可确保完全切除并降低再次手术率。
本病例突出了诊断异位甲状旁腺腺瘤的挑战，以及在PHPT患者中考虑罕见异位腺瘤的重要性，特别是当标准影像学检查未得出确凿结果时。我们还讨论了先进成像模式和术中甲状旁腺激素监测在指导手术决策和确定合适的手术结束点以确保成功结果方面的作用。
