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儿童期发病的卡罗里病作为反复发热的病因:一例报告

Childhood-onset Caroli's disease as a cause of recurrent fever: A case report.

作者信息

Sun Jing, Wang Sheng, Chen Biquan

机构信息

Department of Infectious Diseases, Anhui Provincial Children's Hospital, Hefei, China.

出版信息

Front Pediatr. 2022 Aug 4;10:903285. doi: 10.3389/fped.2022.903285. eCollection 2022.

DOI:10.3389/fped.2022.903285
PMID:35989996
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC9386290/
Abstract

Caroli's disease is a rare congenital bile duct malformation characterized by intrahepatic bile duct dilatation. This kind of situation is seldom encountered in clinical work. We report such a case who presented to our emergency department with recurrent fever as initial symptom. According to the clinical manifestation and imaging examination, a 13-year-old boy was diagnosed with suppurative cholangitis and sepsis caused by Caroli's disease. The symptoms were got relieved after antibiotic therapy upgraded from cephalosporins to carbapenems. After 5 months of follow-up, he did not have fever, abdominal pain or any other discomfort. We believe the present report is of medical significance since it serves as a reminder that Caroli's disease may have atypical presentations and be masked by non-specific clinical findings. The report hopes to enlighten our pediatric colleagues by providing more knowledge on such rare congenital disease.

摘要

卡罗里病是一种罕见的先天性胆管畸形,其特征为肝内胆管扩张。这种情况在临床工作中很少遇到。我们报告这样一例,该患者以反复发热为首发症状就诊于我院急诊科。根据临床表现和影像学检查,一名13岁男孩被诊断为卡罗里病引起的化脓性胆管炎和脓毒症。在抗生素治疗从头孢菌素升级为碳青霉烯类后症状缓解。经过5个月的随访,他没有发热、腹痛或任何其他不适。我们认为本报告具有医学意义,因为它提醒人们卡罗里病可能有非典型表现,并被非特异性临床发现所掩盖。本报告希望通过提供更多关于这种罕见先天性疾病的知识,启发我们的儿科同事。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2ba4/9386290/dc400b352073/fped-10-903285-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2ba4/9386290/4d60d008c01e/fped-10-903285-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2ba4/9386290/4aa919a3e1fa/fped-10-903285-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2ba4/9386290/dc400b352073/fped-10-903285-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2ba4/9386290/4d60d008c01e/fped-10-903285-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2ba4/9386290/4aa919a3e1fa/fped-10-903285-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2ba4/9386290/dc400b352073/fped-10-903285-g003.jpg

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Childhood-onset Caroli's disease as a cause of recurrent fever: A case report.儿童期发病的卡罗里病作为反复发热的病因:一例报告
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引用本文的文献

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A Unique Case of Caroli Disease in Kenya's Medical Landscape.肯尼亚医学领域中的一例独特的 Caroli 病。
Am J Case Rep. 2024 Jan 12;25:e942019. doi: 10.12659/AJCR.942019.

本文引用的文献

1
Rare variants in PKHD1 associated with Caroli syndrome: Two case reports.PKHD1 中的罕见变异与 Caroli 综合征相关:两例病例报告。
Mol Genet Genomic Med. 2022 Aug;10(8):e1998. doi: 10.1002/mgg3.1998. Epub 2022 Jun 17.
2
Caroli's disease incidentally discovered in a 16-years-old female: a case report.偶然发现于 16 岁女性的 Caroli 病:病例报告。
Pan Afr Med J. 2022 Mar 14;41:204. doi: 10.11604/pamj.2022.41.204.34088. eCollection 2022.
3
Diffuse Versus Localized Caroli Disease: A Comparative MRCP Study.弥漫性与局限性肝内胆管扩张症:磁共振胰胆管成像的对比研究。
AJR Am J Roentgenol. 2021 Jun;216(6):1530-1538. doi: 10.2214/AJR.20.23522. Epub 2021 Apr 21.
4
[A cough in a patient with cholangitis in the course of caroli's disease. Case report].[卡罗利病病程中胆管炎患者的咳嗽。病例报告]
Wiad Lek. 2019;72(2):294-297.
5
Caroli Disease Revisited: A Case of a Kidney Transplant Patient With Autosomal Polycystic Kidney Disease and Recurrent Episodes of Cholangitis.再探卡罗里病:一例患有常染色体显性多囊肾病且反复发生胆管炎的肾移植患者
Transplant Proc. 2019 Mar;51(2):541-544. doi: 10.1016/j.transproceed.2018.12.025. Epub 2019 Jan 3.
6
Caroli's Disease as a Cause of Chronic Epigastric Abdominal Pain: Two Case Reports and a Brief Review of the Literature.卡罗里病作为慢性上腹部疼痛的病因:两例病例报告及文献简要综述
Cureus. 2017 Sep 20;9(9):e1701. doi: 10.7759/cureus.1701.
7
A Rare Congenital Disorder of Intrahepatic Bile Ducts.一种罕见的肝内胆管先天性疾病。
J Clin Exp Hepatol. 2016 Mar;6(1):65-7. doi: 10.1016/j.jceh.2016.02.004. Epub 2016 Mar 11.
8
Contrast-enhanced magnetic resonance cholangiography with gadoxetic-acid-disodium for the detection of biliary-cyst communication in Caroli disease.钆塞酸二钠增强磁共振胰胆管造影术用于检测卡罗里病中的胆管囊肿交通。
Gastroenterol Hepatol. 2016 Dec;39(10):669-670. doi: 10.1016/j.gastrohep.2015.07.012. Epub 2015 Nov 18.
9
Caroli's disease misdiagnosed as intraductal papillary neoplasm of the bile duct.被误诊为胆管内乳头状瘤的卡罗利病
Clin Mol Hepatol. 2015 Jun;21(2):175-9. doi: 10.3350/cmh.2015.21.2.175. Epub 2015 Jun 26.
10
Education and imaging. Hepatology: "central dot sign" of Caroli syndrome.
J Gastroenterol Hepatol. 2015 Feb;30(2):234. doi: 10.1111/jgh.12828.