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一名患有克朗凯特-加拿大综合征患者的深静脉血栓形成:一例复杂病例报告。

Deep vein thrombosis in a patient with Cronkhite-Canada syndrome: a complex case report.

作者信息

Feng Xiao-Kai, Chen Xiao-Fen, Wang Bei-Bei, Zeng Zhi-Gang, Liu Chao, Sha Wei-Hong, Ma Juan

机构信息

Department of Respiratory and Critical Care Medicine, Beijing Institute of Respiratory Medicine and Beijing Chao-Yang Hospital, Capital Medical University, Beijing, China.

Department of Gastroenterology and Hepatology, Guangdong Provincial People's Hospital (Guangdong Academy of Medical Sciences), Southern Medical University, Guangzhou, China.

出版信息

Thromb J. 2023 Mar 15;21(1):29. doi: 10.1186/s12959-023-00473-8.

Abstract

BACKGROUND

Cronkhite-Canada syndrome (CCS) is a rare disease characterized by generalized gastrointestinal polyps, ectodermal abnormalities and variable gastrointestinal symptoms. Few cases to date have described complications with deep vein thrombosis (DVT). Here we reported a rare case of CCS concomitant with DVT. The patient's clinical details, endoscopic findings, safety, and efficacy are reported.

CASE PRESENTATION

A 58-year-old patient was admitted to our hospital with recurrent diarrhea, overall abnormal appearance, including hyperpigmentation, hair loss and onychodystrophy, and multiple polyps distributed along the gastrointestinal tract except the esophagus. After considerable assessment, the patient was diagnosed with CCS. She was also diagnosed with concurrent DVT, nephrotic syndrome, and infectious enteritis during the course of disease. After treatment with a combination of methylprednisolone, mesalazine, antibiotics, rivaroxaban, and nutritional support during the 24 months of following the patient in this case, the clinical manifestations and endoscopic findings reached complete remission two years after the diagnosis.

CONCLUSION

To our knowledge, this study is the first case of CCS complicated with DVT reported in China. Although rare, it is important to consider that DVT may occur after CCS and that it is vital to conduct careful follow-up.

摘要

背景

克朗凯特-加拿大综合征(CCS)是一种罕见疾病,其特征为广泛性胃肠道息肉、外胚层异常及多样的胃肠道症状。迄今为止,鲜有病例描述其合并深静脉血栓形成(DVT)的并发症。在此,我们报告一例罕见的CCS合并DVT病例。报告了该患者的临床细节、内镜检查结果、安全性及疗效。

病例介绍

一名58岁患者因反复腹泻、全身外观异常(包括色素沉着、脱发和甲营养不良)以及除食管外沿胃肠道分布的多个息肉入住我院。经过全面评估,该患者被诊断为CCS。在病程中,她还被诊断并发DVT、肾病综合征和感染性肠炎。在对该病例患者随访的24个月期间,联合使用甲泼尼龙、美沙拉嗪、抗生素、利伐沙班及营养支持进行治疗后,诊断两年后临床表现和内镜检查结果达到完全缓解。

结论

据我们所知,本研究是中国首例报道的CCS合并DVT病例。尽管罕见,但需考虑CCS后可能发生DVT,且进行仔细随访至关重要。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a1e0/10018956/3368eb55d9cd/12959_2023_473_Fig1_HTML.jpg

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