妊娠合并原发性甲状旁腺功能亢进致高钙危象及急性胰腺炎 1 例

Primary hyperparathyroidism in pregnancy resulting in hypercalcemic crisis and acute pancreatitis：A case report

摘要

原发性甲状旁腺功能亢进症（primary hyperparathyroidism，PHPT）是由于甲状旁腺产生过多的甲状旁腺激素所致，妊娠期间首次发现的 PHPT 十分罕见，且对母体和胎儿有严重不良影响。本文报告了一例妊娠合并PHPT、高钙危象、急性胰腺炎的病例，患者行急诊剖宫产术，产后第47天行甲状旁腺腺瘤切除术，所娩出胎儿在分娩后曾有数次低钙抽搐。产后 9 个月随访时患者恢复良好，婴儿生长发育良好。本例患者为 30 岁妊娠期女性，孕32周时因腹痛、呕吐就诊，实验室检查提示血钙 3.56mmol/L 、甲状旁腺激素（parathyroid hormone，PTH） 388.6ng/L，影像学检查证实急性胰腺炎及甲状旁腺占位。值得注意的是，患者分娩后 PTH 水平较产前明显下降（48.91~53.08ng/L），结合术后病理所见腺瘤内出血及含铁血黄素沉积，分析可能与急性胰腺炎期间有效循环血容量减少及瘤体部分缺血坏死有关。临床医师应提高对于妊娠合并 PHPT 的警惕性，特别是孕前检查及孕早期应当注意血钙水平，并尽早采取预防措施以避免并发症及不良妊娠结局的发生。

Abstract

SUMMARY Primary hyperparathyroidism（PHPT）is caused by excessive production of parathyroid hor- mone by the parathyroid gland.PHPT discovered for the first time during pregnancy is extremely rare and has serious adverse effects on both the mother and the fetus.This article reports a case of pregnancy com- plicated by PHPT，hypercalcemic crisis，and acute pancreatitis.The patient was a 30 -year-old woman at 32 weeks of gestation who presented with a four-day history of progressive abdominal pain and vomiting. Laboratory evaluation on admission revealed a markedly elevated serum total calcium of 3.56 mmol/L and a parathyroid hormone（PTH）level of 388.6 ng/L，with the normal reference range being 15 to 65 ng/ L.Imaging studies confirmed the presence of acute pancreatitis，left nephrolithiasis，right hydronephro- sis，and a suspicious cystic-solid mass at the lower pole of the right thyroid lobe measuring 2.1 cm× 2.0 cm×1.2 cm.Due to rapid clinical deterioration and the development of a hypercalcemic crisis，an emergency cesarean section was performed，resulting in the delivery of a live premature male infant with a birth weight of 2400 g and a length of 45 cm.The neonate＇s initial serum calcium was 3.08 mmol/L， and several episodes of hypocalcemic seizures occurred shortly after birth，necessitating admission to the neonatal intensive care unit.Postpartum，the mother was managed with aggressive intravenous fluid re- suscitation，subcutaneous calcitonin，and intravenous bisphosphonate therapy.Notably，her serum PTH level decreased substantially after delivery，ranging from 48.91 to 53.08ng/L.This spontaneous decline was likely attributable to reduced effective circulating blood volume during the episode of acute pancreati- tis and partial ischemic necrosis of the parathyroid adenoma.At 47 days postpartum，the patient under- went successful surgical resection of a right inferior parathyroid adenoma measuring approximately 3 cm in diameter.Pathological examination of the resected specimen confirmed the diagnosis and revealed focal vascular proliferation and congestion，interstitial fibrosis with hyaline degeneration，and hemosiderin deposition.Following parathyroidectomy，her serum calcium and PTH levels normalized rapidly and remained stable.At the 9 -month follow-up visit，the mother was completely asymptomatic with normal biochemical parameters，and the infant demonstrated appropriate growth and development.Clinicians should be vigilant about the presence of PHPT during pregnancy，especially during preconception and early pregnancy，monitoring blood calcium levels and taking preventive measures early to avoid complica-tions and adverse pregnancy outcomes.

关键词

原发性甲状旁腺功能亢进症 / 妊娠 / 急性胰腺炎 / 甲状旁腺腺瘤 / 出血

Key words

Primary hyperparathyroidism / Pregnancy / Acute pancreatitis / Parathyroid adenoma / Hemorrhage

参考文献

[1] MacKenzie-Fdeder J, Sirrs S, Anderson D, et al. Primary hyperparathyroidism:An overview[J]. Int J Endocrinol, 2011, 2011: 251410.

[2] Wermers RA, Khosla S, Atkinson EJ, et al. Incidence of primary hyperparathyroidism in Rochester,Minnesota,1993-2001:An update on the changing epidemiology of the disease[J]. J Bone Miner Res, 2006, 21 (1):171-177.

[3] Hirsch D, Kopel V, Nadler V, et al. Pregnancy outcomes in women with primary hyperparathyroidism[J]. J Clin Endocrinol Metab, 2015, 100(5):2115-2122.

[4] DiMarco A, Christakis I, Constantinides V, et al. Undiagnosed primary hyperparathyroidism and recurrent miscarriage:The first prospective pilot study[J]. World J Surg, 2018, 42 (3): 639 - 645.

[5] Horjus C, Groot I, Telting D, et al. Cinacalcet for hyperparathy- roidism in pregnancy and puerperium[J]. J Pediatr Endocrinol Metab, 2009, 22(8):741-749.

[6] Schnatz PF, Curry SL. Primary hyperparathyroidism in pregnancy: Evidence-based management[J]. Obstet Gynecol Surv, 2002, 57 (6):365-376.

[7] Kort KC, Schiller HJ, Numann PJ. Hyperparathyroidism and pregnancy[J]. Am J Surg, 1999, 177(1):66-68.

[8] García MA, Acosta Feria M, Soto Moreno A, et al. Primary hy- perparathyroidism in pregnancy[J]. Gynecol Endocrinol, 2004, 19(2):111-114.

[9] Hong MK, Hsieh CT, Chen BH, et al. Primary hyperpara- thyroidism and acute pancreatitis during the third trimester of preg- nancy[J]. J Matern Fetal Neonatal Med, 2001, 10(3): 214 - 218.

[10] Iqbal N, Aldasouqi S, Peacock M, et al. Life-threatening hyper- calcemia associated with primary hyperparathyroidism during preg- nancy:Case report and review of literature[J]. Endocr Pract, 1999, 5(6):337-342.

[11] Cherry TAD, Kauffman RP, Myles TD. Primary hyperpara-thyroidism,hypercalcemic crisis and subsequent seizures occurring during pregnancy:A case report[J]. J Matern Fetal Neonatal Med, 2002, 12(5):349-352.

[12] Frick TW. The role of calcium in acute pancreatitis[J]. Surgery, 2012, 152(3Suppl 1): S157-S163.

[13] Hacker FM, Whalen PS, Lee VR, et al. Maternal and fetal out- comes of pancreatitis in pregnancy[J]. Am J Obstet Gynecol, 2015, 213(4):568.e1-568.e5.

[14] Kondo Y, Nagai H, Kasahara K, et al. Primary hyperpara- thyroidism and acute pancreatitis during pregnancy[J]. Int J Pancreatol, 1998, 24(1):43-47.

[15] Yang J, Dong MJ, Chen F. A rare lethal case of severe acute ne- crotizing pancreatitis due to a parathyroid adenoma in a third- trimester pregnant woman[J]. BMC Endocr Disord, 2019, 19 (1): 82.

[16] Diaz-Soto G, Linglart A, Sénat MV, et al. Primary hyperparathy- roidism in pregnancy[J]. Endocrine, 2013, 44(3):591-597.

[17] Richa CG, Saad KJ, Chaaban AK, et al. A rare case of hypercal- cemia-induced pancreatitis in a first trimester pregnant woman[J]. Endocrinol Diabetes Metab Case Rep, 2018, 2018:17-0175.

[18] Horton JW, Burnweit CA. Hemodynamic function in acute pancreatitis[J]. Surgery, 1988, 103(5):538-546.

[19] Pereira FA, Brandão DF, Elias J, et al. Parathyroid adenoma apoplexy as a temporary solution of primary hyperparathyroidism: A case report[J]. J Med Case Rep, 2007, 1(1): 139.

[20] Liu Y, Li J, Liu H, et al. Spontaneous remission after a hypercal- cemic crisis caused by an intracystic hemorrhage of bilateral para- thyroid adenomas:A case report and literature review[J]. Front Endocrinol, 2021, 12: 766234.