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肝门部异位甲状腺合并胆道闭锁:一例报告及文献复习

Biliary atresia with ectopic thyroid in the porta hepatis: a case report and literature review.

作者信息

Zhao Li, Zhao Linsheng, Hu Xiaoli, Wang Zhiru, Zhan Jianghua

机构信息

Department of Pathology, Tianjin Children's Hospital (Children's Hospital, Tianjin University), Tianjin, China.

Department of General Surgery, Tianjin Children's Hospital (Children's Hospital, Tianjin University), Tianjin, China.

出版信息

Transl Pediatr. 2024 Nov 30;13(11):2059-2066. doi: 10.21037/tp-24-307. Epub 2024 Nov 26.

DOI:10.21037/tp-24-307
PMID:39649640
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11621884/
Abstract

BACKGROUND

Ectopic thyroid is a rare disease, with ectopic thyroid in the porta hepatis being extremely rare. We present a rare case involving an infant with biliary atresia (BA) wherein ectopic thyroid tissue was incidentally discovered in the porta hepatis after the Kasai operation.

CASE DESCRIPTION

A 2-month-old boy initially presented with jaundice, manifesting as yellow skin and sclera, on the first day following birth. After receiving treatment at a local healthcare facility, jaundice partially subsided, but the color of his stool became lighter 15 days before admission to Tianjin Children's Hospital. Upon B-ultrasound examination at our outpatient clinic, the patient showed bilateral oblique hernia, bilateral testicular hydrocele, and umbilical hernia, and he was admitted to the Department of Gastroenterology at Tianjin Children's Hospital for further treatment of unexplained jaundice. Laparotomy was performed after a comprehensive evaluation, during which we observed the gallbladder to be small and poorly developed. Cholangiography was then performed, revealing visualization of the gallbladder and common bile duct but not the common hepatic duct or right and left hepatic ducts. Intraoperative frozen section examination of wedge-shaped liver tissue indicated findings consistent with extrahepatic biliary obstruction. The Kasai operation was subsequently performed, and an examination of the gallbladder and a fibrous plaque from the porta hepatis after the operation revealed the presence of ectopic thyroid tissues.

CONCLUSIONS

Ectopic thyroid in the porta hepatis is extremely rare. We reviewed literature from 1960 to 2022 and identified six relevant case reports of ectopic thyroid in the porta hepatis. To our knowledge, this report constitutes the first report of BA complicated by ectopic thyroid in the porta hepatis.

摘要

背景

异位甲状腺是一种罕见疾病,肝门部异位甲状腺极为罕见。我们报告一例罕见病例,一名患有胆道闭锁(BA)的婴儿在进行葛西手术后,偶然在肝门部发现异位甲状腺组织。

病例描述

一名2个月大男孩出生后第一天最初出现黄疸,表现为皮肤和巩膜发黄。在当地医疗机构接受治疗后,黄疸部分消退,但入院前15天其大便颜色变浅。在我院门诊进行B超检查时,患者显示双侧腹股沟斜疝、双侧睾丸鞘膜积液和脐疝,遂入住天津市儿童医院消化内科进一步治疗不明原因的黄疸。综合评估后进行剖腹手术,术中观察到胆囊小且发育不良。随后进行胆管造影,显示胆囊和胆总管显影,但肝总管及左右肝管未显影。术中对楔形肝组织进行冰冻切片检查,结果与肝外胆管梗阻相符。随后进行了葛西手术,术后对胆囊和肝门部的纤维斑块检查发现存在异位甲状腺组织。

结论

肝门部异位甲状腺极为罕见。我们回顾了1960年至2022年的文献,确定了6例肝门部异位甲状腺的相关病例报告。据我们所知,本报告是首例BA合并肝门部异位甲状腺的报告。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/009c/11621884/c62d167daac9/tp-13-11-2059-f7.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/009c/11621884/3443d25a7a6e/tp-13-11-2059-f1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/009c/11621884/eb0324b0f950/tp-13-11-2059-f2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/009c/11621884/bdaed1f14c29/tp-13-11-2059-f3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/009c/11621884/85b68a674cf8/tp-13-11-2059-f4.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/009c/11621884/5eba6483fea5/tp-13-11-2059-f5.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/009c/11621884/e1cbe4a16337/tp-13-11-2059-f6.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/009c/11621884/c62d167daac9/tp-13-11-2059-f7.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/009c/11621884/3443d25a7a6e/tp-13-11-2059-f1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/009c/11621884/eb0324b0f950/tp-13-11-2059-f2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/009c/11621884/bdaed1f14c29/tp-13-11-2059-f3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/009c/11621884/85b68a674cf8/tp-13-11-2059-f4.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/009c/11621884/5eba6483fea5/tp-13-11-2059-f5.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/009c/11621884/e1cbe4a16337/tp-13-11-2059-f6.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/009c/11621884/c62d167daac9/tp-13-11-2059-f7.jpg

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本文引用的文献

1
The clinicopathological features, treatment outcomes and follow-up results of 47 ectopic thyroid gland cases: a single-center retrospective study.47 例异位甲状腺临床病理特征、治疗效果及随访结果:单中心回顾性研究。
Front Endocrinol (Lausanne). 2023 Nov 24;14:1278734. doi: 10.3389/fendo.2023.1278734. eCollection 2023.
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A rare presentation of ectopic thyroid gland at right axilla.
右腋窝异位甲状腺的罕见表现。
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Radiological insights of ectopic thyroid in the porta hepatis: A case report and review of the literature.肝门部异位甲状腺的影像学见解:一例报告并文献复习
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Ectopic Thyroid Tissue in Duodenum-A Rare Case in a Young Woman.十二指肠异位甲状腺组织——一名年轻女性的罕见病例
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Rare Case of an Intrapericardial Ectopic Thyroid (Struma Cordis) on the Ascending Aorta.升主动脉上的心包内异位甲状腺(心内甲状腺肿)罕见病例。
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