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一名四岁女童的双脑膨出:病例报告并文献复习

Double encephalocele in a four-year-old girl: A case report with literature review.

作者信息

Khizar Ahtesham, Zahid Hassaan, Khan Manal, Farooq Abdul Rahim, Natt Muhammad Aqeel

机构信息

Ahtesham Khizar, MBBS, FCPS (Neurosurgery), Department of Neurosurgery Unit-I, Punjab Institute of Neurosciences, Lahore, Pakistan.

Hassaan Zahid, MBBS, FCPS (Neurosurgery), MS (Pediatric Neurosurgery), Department of Neurosurgery Unit-I, Punjab Institute of Neurosciences, Lahore, Pakistan.

出版信息

Pak J Med Sci. 2024 Dec;40(12PINS Suppl):S80-S86. doi: 10.12669/pjms.40.12(PINS).10964.

DOI:10.12669/pjms.40.12(PINS).10964
PMID:39703960
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11654668/
Abstract

Encephalocele is a congenital neural tube defect (NTD). The pathophysiology of the NTDs is exceedingly complex. Numerous explanations have been proposed to explain it. Double encephaloceles are highly unusual. There have only been fifteen previously reported cases of double encephalocele in the medical literature, with this index case being the oldest and first from Pakistan. A four-year-old girl presented with two occipital scalp swellings from infancy. The occipital swelling measured about 7x5x3 cm, while the suboccipital swelling measured about 7x9x5 cm. The skin over both the swellings was intact. Following a thorough history, physical examination, and radiological investigations, surgical excision and repair was performed. Postoperative recovery was uneventful. She did not develop hydrocephalus until the three month follow-up.Double encephalocele is a rare entity. The multisite closure theory appears to be the most plausible explanation for the development of multiple NTDs. The management of double encephalocele requires a case based approach.

摘要

脑膨出是一种先天性神经管缺陷(NTD)。NTD的病理生理学极其复杂。人们提出了许多解释。双脑膨出非常罕见。医学文献中此前仅报道过15例双脑膨出病例,本索引病例是年龄最大且首例来自巴基斯坦的。一名4岁女孩自婴儿期起就出现两个枕部头皮肿胀。枕部肿胀大小约为7×5×3厘米,而枕下肿胀大小约为7×9×5厘米。两个肿胀部位的皮肤均完整。经过全面的病史询问、体格检查和影像学检查后,进行了手术切除和修复。术后恢复顺利。直到三个月随访时她才出现脑积水。双脑膨出是一种罕见的病症。多部位闭合理论似乎是多个NTD发生的最合理的解释。双脑膨出的治疗需要基于病例的方法。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/765e/11654668/f925fe79100e/PJMS-40-S80-g005.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/765e/11654668/74d4e365d0e3/PJMS-40-S80-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/765e/11654668/79a9a0de8257/PJMS-40-S80-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/765e/11654668/126bd4f9cbdf/PJMS-40-S80-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/765e/11654668/f52ce08b43bf/PJMS-40-S80-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/765e/11654668/f925fe79100e/PJMS-40-S80-g005.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/765e/11654668/74d4e365d0e3/PJMS-40-S80-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/765e/11654668/79a9a0de8257/PJMS-40-S80-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/765e/11654668/126bd4f9cbdf/PJMS-40-S80-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/765e/11654668/f52ce08b43bf/PJMS-40-S80-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/765e/11654668/f925fe79100e/PJMS-40-S80-g005.jpg

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本文引用的文献

1
Double encephalocele with an excellent outcome postoperatively: A case report from Iraq.双脑膨出术后效果良好:来自伊拉克的一例病例报告
eNeurologicalSci. 2023 Feb 8;30:100449. doi: 10.1016/j.ensci.2023.100449. eCollection 2023 Mar.
2
In-utero Diagnosis of Double Encephalocele - Imaging Features and Review of Literature.子宫内诊断双脑膨出 - 影像学特征及文献复习。
J Radiol Case Rep. 2021 Dec 1;15(12):1-9. doi: 10.3941/jrcr.v15i12.4230. eCollection 2021 Dec.
3
Amniotic band syndrome with double encephalocele: A case report.
合并双脑膨出的羊膜带综合征:一例报告。
Surg Neurol Int. 2020 Dec 22;11:448. doi: 10.25259/SNI_454_2020. eCollection 2020.
4
A case report of triple neural tube defect: revisiting the multisite closure theory.一例三重神经管缺陷病例报告:重新审视多部位闭合理论。
BMC Surg. 2019 Nov 6;19(1):164. doi: 10.1186/s12893-019-0633-2.
5
Double Encephalocele: An Unusual Presentation.双脑膨出:一种罕见的表现。
J Pediatr Neurosci. 2018 Apr-Jun;13(2):264-266. doi: 10.4103/jpn.JPN_22_18.
6
CARE guidelines for case reports: explanation and elaboration document.病例报告的CARE指南:解释与阐述文件。
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Giant Parietal Encephalocele with Massive Brain Herniation and Suboccipital Encephalocele in a Neonate: An Unusual Form of Double Encephalocele.新生儿巨大顶叶脑膨出伴大量脑疝形成及枕下脑膨出:一种罕见的双脑膨出形式
World Neurosurg. 2017 Feb;98:867.e9-867.e11. doi: 10.1016/j.wneu.2016.11.030. Epub 2016 Nov 17.
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Parietal and occipital encephalocele in same child: A rarest variety of double encephalocele.同一患儿出现顶枕部脑膨出:一种极为罕见的双脑膨出类型。
Eur J Paediatr Neurol. 2016 May;20(3):493-6. doi: 10.1016/j.ejpn.2015.12.008. Epub 2016 Feb 4.
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Supra- and infra-torcular double occipital encephalocele.枕骨大孔上和枕骨大孔下双枕部脑膨出。
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Double suboccipital meningoencephalocele: a unique case report.双枕下脑膜脑膨出:一例独特病例报告。
Pediatr Neurosurg. 2012;48(5):331-2. doi: 10.1159/000348886. Epub 2013 Jul 6.