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一名常染色体显性多囊肾病患者在使用托伐普坦治疗期间发生急性肺血栓栓塞。

Acute pulmonary thromboembolism occurring during treatment with tolvaptan in a patient with autosomal-dominant polycystic kidney disease.

作者信息

Morimoto Katsuhiko, Akai Yasuhiro, Matsui Masaru, Yano Hiroki, Tagawa Miho, Samejima Ken-Ichi, Saito Yoshihiko

机构信息

First Department of Internal Medicine, Nara Medical University, Kashihara, Japan.

Department of Cardiology and Nephrology, Nara Prefecture General Medical Center, Nara, Japan.

出版信息

CEN Case Rep. 2017 May;6(1):61-65. doi: 10.1007/s13730-016-0245-y. Epub 2017 Jan 24.

Abstract

Autosomal-dominant polycystic kidney disease (ADPKD) is the most prevalent cystic kidney disease, with approximately half of the patients reaching end-stage renal disease by the age of 60. Tolvaptan prevents renal cyst growth by inhibiting intracellular cyclic AMP and is recommended for patients with ADPKD. Reports of thrombotic complications with tolvaptan have been limited. We report a case of a 60-year-old man who developed thromboembolisms during tolvaptan treatment for ADPKD. The patient started tolvaptan in July 2014. He was brought to our hospital in February 2015 with a sudden onset of dyspnea and chest pain after 6 days of persistent watery diarrhea. Blood tests revealed enhanced coagulation and fibrinolysis, and contrast-enhanced computed tomography confirmed the presence of multiple thromboembolisms. Venous thromboembolism (VTE) with acute pulmonary and lower extremity thrombi was diagnosed, and the patient was immediately admitted. Tolvaptan was discontinued on admission, and intravenous fluid loading and monteplase were started. Subsequently, chest pain and dyspnea resolved, with thrombi resolution occurring by day 14; the patient was discharged on day 18 in stable condition. VTE was attributed to continued tolvaptan during diarrhea and dehydration; tolvaptan itself was not associated with enhanced coagulability. Dehydrated patients with ADPKD, such as the patient in this case, are at an increased risk for thrombus formation. Proper education should be provided to maintain appropriate fluid status and discontinue tolvaptan upon volume depletion.

摘要

常染色体显性遗传性多囊肾病(ADPKD)是最常见的囊性肾病,约半数患者在60岁时发展为终末期肾病。托伐普坦通过抑制细胞内环磷酸腺苷来阻止肾囊肿生长,被推荐用于ADPKD患者。关于托伐普坦血栓形成并发症的报道有限。我们报告一例60岁男性患者,在接受托伐普坦治疗ADPKD期间发生血栓栓塞。该患者于2014年7月开始使用托伐普坦。2015年2月,在持续水样腹泻6天后,他突然出现呼吸困难和胸痛,被送至我院。血液检查显示凝血和纤溶增强,增强CT证实存在多处血栓栓塞。诊断为伴有急性肺和下肢血栓的静脉血栓栓塞(VTE),患者立即入院。入院后停用托伐普坦,开始静脉补液并使用门冬酰胺酶。随后,胸痛和呼吸困难缓解,第14天时血栓溶解;患者于第18天病情稳定出院。VTE归因于腹泻和脱水期间继续使用托伐普坦;托伐普坦本身与凝血性增强无关。像本例患者这样的ADPKD脱水患者,血栓形成风险增加。应提供适当的教育,以维持适当的液体状态,并在出现容量耗竭时停用托伐普坦。

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