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英夫利昔单抗治疗活动性克罗恩病和高钙血症:病例报告及文献综述

Active crohn disease and hypercalcemia treated with infliximab: case report and literature review.

作者信息

Ioachimescu Adriana G, Bauer Thomas W, Licata Angelo

机构信息

Department of Endocrinology, Diabetes, and Metabolism, Cleveland Clinic, A53, 9500 Euclid Avenue, Cleveland, OH 44195, USA.

出版信息

Endocr Pract. 2008 Jan-Feb;14(1):87-92. doi: 10.4158/EP.14.1.87.

Abstract

OBJECTIVE

To report a case of 1,25-dihydroxyvitamin D-mediated hypercalcemia caused by active Crohn disease that improved with infliximab therapy.

METHODS

We present the clinical and laboratory findings and describe the clinical course of a patient who had hypercalcemia during Crohn disease exacerbations. The literature is reviewed regarding 1,25-dihydroxyvitamin D production in Crohn disease, and the 3 cases of hypercalcemia in individuals with Crohn disease reported in the literature are described.

RESULTS

A 50-year-old man with long-standing Crohn disease treated with multiple bowel resections presented for take-down ileostomy. He was hypercalcemic and had suppressed parathyroid hormone and parathyroid hormone-related peptide levels. Histopathology of the resected ileostomy site and adjacent small bowel indicated active Crohn disease. Hypercalcemia promptly resolved after a few days of treatment with intravenous glucocorticoids. One month later, hypercalcemia recurred in the presence of an inappropriately high 1,25-dihydroxyvitamin D level and increased urinary calcium and serum angiotensin-converting enzyme levels. The serum and urinary calcium levels became normal with infliximab therapy. Three previous reports of hypercalcemia caused by active Crohn disease describe effective treatment with glucocorticoids. This is the first report of successful response to infliximab in this setting.

CONCLUSION

Hypercalcemia mediated by 1,25-dihydroxyvitamin D in the setting of Crohn disease may respond to glucocorticoid-sparing immunomodulators.

摘要

目的

报告一例由活动性克罗恩病引起的1,25 - 二羟维生素D介导的高钙血症,该患者经英夫利昔单抗治疗后病情改善。

方法

我们呈现该患者的临床和实验室检查结果,并描述其在克罗恩病加重期间出现高钙血症的临床病程。回顾了关于克罗恩病中1,25 - 二羟维生素D产生的文献,并描述了文献中报道的3例克罗恩病患者高钙血症的病例。

结果

一名50岁患有长期克罗恩病且接受过多次肠道切除术的男性因回肠造口还纳术前来就诊。他存在高钙血症,甲状旁腺激素和甲状旁腺激素相关肽水平受到抑制。切除的回肠造口部位及相邻小肠的组织病理学检查显示为活动性克罗恩病。静脉注射糖皮质激素治疗几天后,高钙血症迅速缓解。1个月后,在1,25 - 二羟维生素D水平异常升高、尿钙增加和血清血管紧张素转换酶水平升高的情况下,高钙血症复发。英夫利昔单抗治疗后,血清和尿钙水平恢复正常。之前有3篇关于活动性克罗恩病引起高钙血症的报道描述了糖皮质激素治疗有效。这是在这种情况下英夫利昔单抗治疗成功的首例报道。

结论

在克罗恩病背景下,由1,25 - 二羟维生素D介导的高钙血症可能对糖皮质激素节省型免疫调节剂有反应。

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