Department of General Surgery, Uludag University Faculty of Medicine, Bursa, Türkiye.
Front Endocrinol (Lausanne). 2024 Oct 8;15:1371098. doi: 10.3389/fendo.2024.1371098. eCollection 2024.
Primary hyperparathyroidism (PHPT) due to ectopic parathyroid adenoma is a rare case of hypercalcemia in the pediatric population. Herein, a rare case of PHPT due to ectopic intrathymic parathyroid adenoma was described in an asymptomatic 15-year-old girl who had incidental diagnosis based on laboratory abnormalities but experienced a 3-month postoperative course of persistently elevated parathyroid hormone (PTH) and hypercalcemia following the initial unsuccessful parathyroidectomy operation carried out in a non-parathyroid expert center. The curative surgical treatment was accomplished only after the patient was reoperated with video-assisted thoracoscopic surgery (VATS) thymectomy by the surgeon experienced in parathyroid surgery with implementation of the combined imaging modalities for accurate localization of ectopic adenoma including 99mTc sestamibi (MIBI) plus neck and thoracic computed tomography (CT) and the appropriate surgical strategies including intraoperative intact PTH monitoring and frozen section diagnosis. Before the reoperation (VATS thymectomy), laboratory findings showed elevated PTH (1,171 ng/L; reference range: 21.80 ng/L-87.5 ng/L) and hypercalcemia (13.4 mg/dL; reference range: 8.4 mg/dL-10.2 mg/dL). The preoperative PTH levels were 94 ng/L at 5 min after thymectomy and 78 ng/L at 10 min. The PTH and calcium levels were 54.3 ng/L and 8.47 mg/dL, respectively, on postoperative day 1 and were 34.2 ng/L and 8.1 mg/dL on postoperative day 2. The patient was discharged on postoperative day 2 without any complications. In conclusion, our findings indicate the likelihood of isolated primary hyperparathyroidism to be incidentally diagnosed based solely on laboratory abnormalities with no specific clinical manifestations in the pediatric age. In addition, using combined imaging modalities (such as MIBI and CT) in accurate localization of ectopic parathyroid adenoma and implementation of surgery by experienced surgeons along with intraoperative intact PTH monitoring and frozen section diagnosis seem crucial to ensure the curative surgical treatment.
原发性甲状旁腺功能亢进症（PHPT）由异位甲状旁腺腺瘤引起，在儿科人群中是一种罕见的高钙血症病例。在此，描述了一例罕见的由胸腺内异位甲状旁腺腺瘤引起的 PHPT 病例，患者为无症状的 15 岁女孩，在非甲状旁腺专家中心进行初次不成功的甲状旁腺切除术手术后，因实验室异常而偶然诊断出该疾病，但经历了 3 个月的甲状旁腺激素（PTH）持续升高和高钙血症的术后过程。只有在有经验的甲状旁腺外科医生进行了电视辅助胸腔镜手术（VATS）胸腺切除术，并结合 99mTc sestamibi（MIBI）联合颈部和胸部计算机断层扫描（CT）等准确定位异位腺瘤的联合成像方式，以及术中完整的 PTH 监测和冰冻切片诊断等适当的手术策略后，才能完成治愈性手术治疗。在再次手术（VATS 胸腺切除术）之前，实验室检查结果显示 PTH 升高（1171ng/L；参考范围：21.80ng/L-87.5ng/L）和高钙血症（13.4mg/dL；参考范围：8.4mg/dL-10.2mg/dL）。胸腺切除术后 5 分钟时的术前 PTH 水平为 94ng/L，10 分钟时为 78ng/L。术后第 1 天的 PTH 和钙水平分别为 54.3ng/L 和 8.47mg/dL，术后第 2 天分别为 34.2ng/L 和 8.1mg/dL。患者术后第 2 天无任何并发症出院。总之，我们的研究结果表明，儿科人群中仅基于实验室异常而无特定临床表现，可能偶然诊断出孤立性原发性甲状旁腺功能亢进症。此外，使用联合成像方式（如 MIBI 和 CT）准确定位异位甲状旁腺腺瘤，并由经验丰富的外科医生进行手术，以及术中完整的 PTH 监测和冰冻切片诊断，对于确保治愈性手术治疗似乎至关重要。
