Rutledge Stephanie, Harrison Michele, O'Connell Martin, O'Dwyer Tadhg, Byrne Maria M
Department of Endocrinology, Mater Misericordiae University Hospital, Eccles Street, Dublin 7, Ireland.
Department of Pathology, Mater Misericordiae University Hospital, Eccles Street, Dublin 7, Ireland.
J Med Case Rep. 2016 Oct 19;10(1):286. doi: 10.1186/s13256-016-1078-1.
We report the case of a giant intrathyroidal parathyroid adenoma weighing 59 g in a young woman presenting acutely with severe hypercalcemia requiring correction and adequate preoperative management prior to surgery. Parathyroid adenomas account for 85 % of cases of primary hyperparathyroidism. Those weighing more than 3.5g are classified as giant parathyroid adenomas. There are only 25 cases of parathyroid adenomas weighing over 30g reported in the literature. With the wide availability of biochemical screening tests in Western countries, mildly elevated calcium levels are often discovered incidentally. Our case is unusual for the extreme level of hypercalcemia, the patient's young age, and the weight of the adenoma, particularly in a developed country.
A 21-year-old Irish woman presented with a 3-week history of an enlarging right-sided neck mass. There was no dysphagia, stridor, or symptoms of hyperthyroidism or hypercalcemia. On examination, there was a firm painless swelling in the right lobe of her thyroid. Her thyroid function tests were normal. Corrected serum calcium was markedly elevated at 3.96 mmol/L with hypophosphatemia of 0.35 mmol/L. She was treated with bisphosphonates and fluids administered intravenously. Her parathyroid hormone level was over 20 times the upper limit of normal. Ultrasound revealed a solid and cystic nodule in the lower pole of the right lobe of her thyroid. Parathyroid scintigraphy demonstrated a 5×4 cm lesion which concentrated tracer. A right-sided parathyroidectomy, right thyroid lobectomy, and level VI neck dissection were performed. An encapsulated multiloculated solid cystic mass weighing 59 g was removed. There was no definite infiltration of the capsule and MIB1 count was low at 1 % thus the specimen lacked the diagnostic features of carcinoma. On the third postoperative day, hungry bone syndrome developed and calcium replacement administered intravenously was required. At 1-year postoperative, she was weaned off calcium and alfacalcidol. A follow-up ultrasound showed unremarkable residual thyroid.
Any patient with an isolated hypercalcemia warrants a thorough work-up. Hungry bone syndrome is a potentially avoidable condition; thus the clinician should be highly attuned to the risk of hungry bone syndrome post-parathyroidectomy, which correlates with the weight of the adenoma resected.
我们报告了一例年轻女性巨大甲状腺内甲状旁腺腺瘤病例,腺瘤重达59克,患者急性出现严重高钙血症,术前需要纠正并进行充分管理。甲状旁腺腺瘤占原发性甲状旁腺功能亢进病例的85%。重量超过3.5克的被归类为巨大甲状旁腺腺瘤。文献中仅报道了25例重量超过30克的甲状旁腺腺瘤病例。在西方国家,由于生化筛查测试广泛可用,轻度升高的钙水平常常被偶然发现。我们的病例不同寻常之处在于高钙血症的严重程度、患者的年轻年龄以及腺瘤的重量,尤其是在一个发达国家。
一名21岁的爱尔兰女性,右侧颈部肿物增大3周前来就诊。无吞咽困难、喘鸣,也无甲状腺功能亢进或高钙血症症状。检查发现甲状腺右叶有一个质地硬、无痛的肿物。甲状腺功能检查正常。校正血清钙显著升高至3.96 mmol/L,血磷降低至0.35 mmol/L。给予双膦酸盐和静脉补液治疗。甲状旁腺激素水平超过正常上限20倍以上。超声显示甲状腺右叶下极有一个实性和囊性结节。甲状旁腺闪烁显像显示一个5×4厘米的病变摄取示踪剂。进行了右侧甲状旁腺切除术、右侧甲状腺叶切除术和VI区颈部淋巴结清扫术。切除了一个包膜完整、多房的实性囊性肿物,重59克。包膜无明确浸润,MIB1计数低,为1%,因此标本缺乏癌的诊断特征。术后第三天,出现饥饿骨综合征,需要静脉补钙。术后1年,她停用了钙剂和阿法骨化醇。随访超声显示残余甲状腺无异常。
任何孤立性高钙血症患者都需要进行全面检查。饥饿骨综合征是一种可能避免的情况;因此临床医生应高度警惕甲状旁腺切除术后饥饿骨综合征的风险,其与切除腺瘤的重量相关。