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胆道闭锁婴儿肌间血肿致桡神经麻痹:1 例报告。

Radial nerve palsy caused by a rapidly growing intramuscular hematoma in an infant with biliary atresia: a case report.

机构信息

Department of Pediatrics, Aichi Medical University School of Medicine, 1-1, Yazakokarimata, Nagakute, Aichi, 480-1195, Japan.

Postgraduate Clinical Training Center, Aichi Medical University School of Medicine, Nagakute, Aichi, Japan.

出版信息

BMC Pediatr. 2023 May 19;23(1):247. doi: 10.1186/s12887-023-04071-5.

DOI:10.1186/s12887-023-04071-5
PMID:37208637
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10197374/
Abstract

BACKGROUND

Biliary atresia (BA) is a rare cause of persistent jaundice in infants that can result in vitamin K malabsorption and vitamin K deficiency bleeding (VKDB). We present an infant with BA who developed a rapidly growing intramuscular hematoma in her upper arm after a vaccination which caused a radial nerve palsy.

CASE PRESENTATION

An 82-day-old girl was referred to our hospital because of a rapidly growing left upper arm mass. She had received three doses of oral vitamin K before age 1 month. At age 66 days, she received a pneumococcal vaccination in her left upper arm. On presentation, she showed no left wrist or finger extension. Blood examination revealed direct hyperbilirubinemia, liver dysfunction, and coagulation abnormalities, indicating obstructive jaundice. Magnetic resonance imaging showed a hematoma in the left triceps brachii. Abdominal ultrasonography revealed an atrophic gallbladder and the triangular cord sign anterior to the portal vein bifurcation. BA was confirmed on cholangiography. VKDB resulting from BA in conjunction with vaccination in the left upper arm were considered the cause of the hematoma. The hematoma was considered the cause of her radial nerve palsy. Although she underwent Kasai hepatic portoenterostomy at age 82 days, the obstructive jaundice did not sufficiently improve. She then underwent living-related liver transplantation at age 8 months. The wrist drop was still present at age 1 year despite hematoma resolution.

CONCLUSIONS

Delayed detection of BA and inadequate prevention of VKDB can result in permanent peripheral neuropathy.

摘要

背景

胆道闭锁(BA)是一种导致婴儿持续性黄疸的罕见病因,可导致维生素 K 吸收不良和维生素 K 缺乏性出血(VKDB)。我们报告了一例 BA 婴儿,在左上臂接种疫苗后发生快速生长的肌内血肿,导致桡神经麻痹。

病例介绍

一名 82 天大的女孩因左上臂迅速生长的肿块被转至我院。她在 1 个月前接受了 3 剂口服维生素 K。在 66 天大时,她在左上臂接种了肺炎球菌疫苗。就诊时,她的左手腕和手指无法伸展。血液检查显示直接高胆红素血症、肝功能异常和凝血异常,提示梗阻性黄疸。磁共振成像显示左肱三头肌血肿。腹部超声显示胆囊萎缩和门静脉分叉前三角索征。胆管造影证实为 BA。BA 合并左上臂疫苗接种导致的 VKDB 被认为是血肿的原因。血肿被认为是导致其桡神经麻痹的原因。尽管她在 82 天大时接受了 Kasai 肝门肠吻合术,但梗阻性黄疸并未得到充分改善。随后她在 8 个月大时接受了活体肝移植。尽管血肿已消退,但 1 岁时手腕下垂仍存在。

结论

BA 的延迟发现和 VKDB 的预防不足可导致永久性周围神经病。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c6b1/10197374/8c209f00b25b/12887_2023_4071_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c6b1/10197374/7e8019caed01/12887_2023_4071_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c6b1/10197374/79e6a9c8fc46/12887_2023_4071_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c6b1/10197374/8c209f00b25b/12887_2023_4071_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c6b1/10197374/7e8019caed01/12887_2023_4071_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c6b1/10197374/79e6a9c8fc46/12887_2023_4071_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c6b1/10197374/8c209f00b25b/12887_2023_4071_Fig3_HTML.jpg

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本文引用的文献

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Population-based screening methods in biliary atresia: a systematic review and meta-analysis.基于人群的先天性胆道闭锁筛查方法:系统评价和荟萃分析。
Arch Dis Child. 2023 Jun;108(6):468-473. doi: 10.1136/archdischild-2022-324946. Epub 2023 Feb 16.
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Vaccine safety surveillance informs public health policy beyond immunization: A case-series on bleeding following vaccination, Nepal, 2016-2018.
疫苗安全监测对公共卫生政策具有重要意义:尼泊尔 2016-2018 年疫苗接种后出血病例系列研究
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Nutrients. 2020 Mar 16;12(3):780. doi: 10.3390/nu12030780.
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Temporal association between serious bleeding and immunization: vitamin K deficiency as main causative factor.严重出血与免疫接种之间的时间关联:维生素 K 缺乏为主要致病因素。
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Pediatrics. 2016 May;137(5). doi: 10.1542/peds.2015-4222.
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Unilateral radial nerve palsy in a newborn.新生儿单侧桡神经麻痹
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