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类固醇反应性特发性骨化三醇诱导高钙血症:病例报告及文献复习。

Steroid responsive idiopathic calcitriol induced hypercalcemia: a case report and review of the literature.

机构信息

Sidney Kimmel Medical College, Thomas Jefferson University, Philadelphia, United States.

Department of Internal Medicine, Division of Nephrology, Thomas Jefferson University, Philadelphia, United States.

出版信息

BMC Nephrol. 2023 Jun 6;24(1):159. doi: 10.1186/s12882-023-03203-4.

Abstract

BACKGROUND

Idiopathic Calcitriol Induced Hypercalcemia is a rare cause of a common condition of hypercalcemia. Hypercalcemia is most commonly the result of hyperparathyroidism and together with hypercalcemia of malignancy accounts for over 95% of cases. Idiopathic Calcitriol Induced Hypercalcemia can mimic hypercalcemia secondary to granulomatous diseases like sarcoidosis, but with apparent absences of both imaging and physical exam findings consistent with the disease. We report here a 51-year-old man who presented with recurrent nephrolithiasis, hypercalcemia, and acute kidney injury.

CASE PRESENTATION

A 51-year-old man presented with severe back pain and mild hematuria. He had a history of recurrent nephrolithiasis over the course of a 15-year period. On presentation his calcium was elevated at 13.4 mg/dL, creatinine was 3.1 mg/dL (from baseline of 1.2), and his PTH was reduced at 5 pg/mL. CT abdomen and pelvis showed acute nephrolithiasis which was managed medically. Work up for the hypercalcemia included an SPEP which was normal, Vit D,1,25 (OH)2 was elevated at 80.4 pg/mL, CT chest showed no evidence of sarcoidosis. Management with 10 mg prednisone showed marked improvement in the hypercalcemia and he no longer had any symptoms of hypercalcemia.

CONCLUSION

Idiopathic Calcitriol Induced Hypercalcemia is a rare cause of hypercalcemia. All reported cases benefit from more intensive long-term immunosuppression. This report helps consolidate the diagnosis of Idiopathic Calcitriol Induced Hypercalcemia and encourages researchers to better investigate its underlying pathogenesis.

摘要

背景

特发性钙化醇诱导高钙血症是一种罕见的高钙血症常见病因。高钙血症最常见于甲状旁腺功能亢进症,与恶性肿瘤高钙血症一起占 95%以上的病例。特发性钙化醇诱导的高钙血症可模拟结节病等肉芽肿性疾病引起的高钙血症,但影像学和体格检查均无明显异常,与疾病表现不一致。我们在此报告一例 51 岁男性,表现为复发性肾结石、高钙血症和急性肾损伤。

病例介绍

一名 51 岁男性因严重腰痛和轻度血尿就诊。他在过去 15 年中有反复发作肾结石病史。就诊时血钙升高至 13.4mg/dL,肌酐为 3.1mg/dL(基线值为 1.2),甲状旁腺激素(PTH)降低至 5pg/mL。腹部和骨盆 CT 显示急性肾结石,采用药物治疗。高钙血症的检查包括 SPEP 正常,维生素 D1,25(OH)2升高至 80.4pg/mL,胸部 CT 未见结节病证据。采用 10mg 泼尼松治疗,高钙血症明显改善,且不再有高钙血症的任何症状。

结论

特发性钙化醇诱导的高钙血症是高钙血症的罕见病因。所有报告病例均受益于更强化的长期免疫抑制治疗。本报告有助于巩固特发性钙化醇诱导的高钙血症的诊断,并鼓励研究人员更好地研究其潜在发病机制。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/eeae/10245421/db282d109ff3/12882_2023_3203_Fig1_HTML.jpg

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