Wei Abraham E, Garrett Matthew R, Gupta Ankur
AACE Clin Case Rep. 2019 Aug 14;5(6):e384-e387. doi: 10.4158/ACCR-2019-0225. eCollection 2019 Nov-Dec.
To present a rare case of parathyromatosis.
We present the clinical, laboratory, and imaging findings, along with a review of the literature.
A 33-year-old man with a history of right upper parathyroid adenoma removal 5 years prior due to hyperparathyroidism was admitted for severe hypercalcemia (15.6 mg/dL; normal, 8.5 to 10.5 mg/dL) with elevated plasma parathyroid hormone (PTH) (882 pg/mL; normal, 15 to 65 pg/mL). Ultrasound, computed tomography (CT), sestamibi, and positron emission tomography scans were unremarkable; however, a four-dimensional CT (4DCT) of the neck showed an area of increased signal enhancement and hypervascularity without discrete nodule in the posterior right thyroid region. The patient underwent parathyroid surgical exploration with right hemithyroidectomy and compartment neck dissection to remove the affected tissue. PTH levels dropped to 208 pg/mL postoperatively; calcium decreased but remained elevated at 12.7 mg/dL. Pathology revealed the presence of several small nodular foci of atypical hyperplastic parathyroid tissue in the right thyroid and soft tissue in the left central neck compartment consistent with parathyromatosis.
This case report represents the first-time use of 4DCT to localize parathyromatosis. Parathyromatosis is a rare but problematic cause of recurrent hyperparathyroidism. Ultrasound and 4DCT may represent the best imaging modalities for identification and perioperative management to remove all affected tissue without reseeding.
报告一例罕见的甲状旁腺瘤病病例。
我们展示了该病例的临床、实验室及影像学检查结果,并对相关文献进行了回顾。
一名33岁男性,5年前因甲状旁腺功能亢进接受了右上甲状旁腺腺瘤切除术,此次因严重高钙血症(15.6mg/dL;正常范围8.5至10.5mg/dL)及血浆甲状旁腺激素(PTH)升高(882pg/mL;正常范围15至65pg/mL)入院。超声、计算机断层扫描(CT)、甲氧基异丁基异腈(sestamibi)及正电子发射断层扫描均未见异常;然而,颈部四维CT(4DCT)显示右甲状腺后方区域信号增强及血管增多,但无离散结节。患者接受了甲状旁腺手术探查,行右半甲状腺切除术及颈部区域淋巴结清扫以切除受累组织。术后PTH水平降至208pg/mL;血钙降低但仍高于正常,为12.7mg/dL。病理检查显示右甲状腺及左中央颈部软组织中有多个非典型增生性甲状旁腺组织的小结节灶,符合甲状旁腺瘤病表现。
本病例报告首次使用4DCT定位甲状旁腺瘤病。甲状旁腺瘤病是复发性甲状旁腺功能亢进的罕见但棘手的病因。超声和4DCT可能是识别及围手术期管理以切除所有受累组织且避免种植转移的最佳影像学检查方法。