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半侧颜面短小畸形合并椎动脉血管畸形:一例报告

Hemifacial microsomia associated with vascular malformation of vertebral: A case report.

作者信息

Prasetyo Arif Tri, Putri Indri Lakhsmi, Wardani Anggun Esti

机构信息

Division of Plastic Reconstructive and Aesthetic Surgery, Department of Surgery, Faculty of Medicine, Universitas Padjadjaran, Bandung, Indonesia.

Department of Plastic Reconstructive and Aesthetic Surgery, Faculty of Medicine, Universitas Airlangga, Surabaya, Indonesia; Universitas Airlangga Hospital, Surabaya, Indonesia; Airlangga Health Science Institute, Indonesia.

出版信息

Int J Surg Case Rep. 2023 Nov;112:108906. doi: 10.1016/j.ijscr.2023.108906. Epub 2023 Oct 3.

DOI:10.1016/j.ijscr.2023.108906
PMID:37837667
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10667787/
Abstract

INTRODUCTION AND IMPORTANCE

Hemifacial microsomia (HFM) is a complex congenital facial anomaly characterized by a wide spectrum of clinical features, which encompass the facial skeleton and other organ systems. Currently, there is no evidence to suggest an association between Hemifacial Microsomia and vascular malformations, whether of the vertebral or any other kind.

CASE PRESENTATION

Reporting a case of a 12-year-old male diagnosed with Hemifacial Microsomia (HFM) and left Microtia. The patient had previously undergone left auricle reconstruction; however, unfortunately, the flap resulted in necrosis. In our next step, we intend to proceed with further reconstruction. Before this, we plan to perform CT angiography to identify viable flap options for effectively closing the auricular defect. During this evaluation, we identified an anomaly structure in the vertebral vascularization.

CLINICAL DISCUSSION

During the CT angiography, we found a vascular malformation in the vertebral region. This anomaly manifested as tortuosity in the left vertebral vein, with the diameter on the left side being larger than that on the right. Additionally, the diameter of the left internal jugular artery was found to be smaller than its counterpart on the right. The maxillary artery of the left side was larger than the right. Notably, there was an absence of a submental artery on the left side, and a hypoplasia of the left angularis artery was observed.

CONCLUSION

Hemifacial microsomia could be associated with other malformations. Despite the fact that vertebral artery anomaly is not considered common anomaly in HFM, it is mandatory to perform CT angiography before reconstructive surgery, considering the possibility of massive bleeding during the operation.

摘要

引言与重要性

半侧颜面短小畸形(HFM)是一种复杂的先天性面部畸形,其临床特征范围广泛,涵盖面部骨骼和其他器官系统。目前,尚无证据表明半侧颜面短小畸形与血管畸形之间存在关联,无论是椎动脉还是其他类型的血管畸形。

病例报告

报告一例12岁男性,诊断为半侧颜面短小畸形(HFM)和左侧小耳畸形。该患者此前已接受左耳廓重建;然而,不幸的是,皮瓣发生坏死。在下一步,我们打算继续进行进一步的重建。在此之前,我们计划进行CT血管造影,以确定有效的皮瓣选择,用于有效闭合耳廓缺损。在此次评估过程中,我们在椎动脉血管化中发现了一个异常结构。

临床讨论

在CT血管造影过程中,我们在椎骨区域发现了一个血管畸形。这种异常表现为左侧椎动脉迂曲,左侧直径大于右侧。此外,发现左侧颈内动脉直径小于右侧。左侧上颌动脉大于右侧。值得注意的是,左侧颏下动脉缺如,左侧口角动脉发育不全。

结论

半侧颜面短小畸形可能与其他畸形相关。尽管椎动脉异常在HFM中不被认为是常见异常,但考虑到手术过程中可能出现大出血,在重建手术前进行CT血管造影是必要的。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2fe6/10667787/9bad99c971aa/gr6.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2fe6/10667787/d18371c2b88b/gr1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2fe6/10667787/da2c09c86105/gr2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2fe6/10667787/8df0090f4968/gr3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2fe6/10667787/c6a8300fbff2/gr4.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2fe6/10667787/d76310b161ca/gr5.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2fe6/10667787/9bad99c971aa/gr6.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2fe6/10667787/d18371c2b88b/gr1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2fe6/10667787/da2c09c86105/gr2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2fe6/10667787/8df0090f4968/gr3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2fe6/10667787/c6a8300fbff2/gr4.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2fe6/10667787/d76310b161ca/gr5.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2fe6/10667787/9bad99c971aa/gr6.jpg

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