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颞下颌关节和下颌骨骨内动静脉畸形1例罕见病例——病例报告及文献复习

A rare case of a intraosseous arteriovenous malformation of the temporomandibular joint and mandible - Case report and literature review.

作者信息

Blackhall Kristian K, Ling Eugenie, Kunjur Jayanth

机构信息

Dept. Oral and Maxillofacial Surgery, Salisbury District Hospital, Salisbury, Wiltshire, SP2 8BJ, UK.

Dept. Oral and Maxillofacial Surgery, Salisbury District Hospital, Salisbury, Wiltshire, SP2 8BJ, UK.

出版信息

Int J Surg Case Rep. 2020;76:394-398. doi: 10.1016/j.ijscr.2020.10.011. Epub 2020 Oct 7.

Abstract

INTRODUCTION

This case report looks at a 47 year-old patient, presenting with a rapid onset mass of the left face. This report examines the case, the investigations undertaken, its management and undertakes a literature review of this uncommon condition and rare presentation.

PRESENTATION OF CASE

This patient had experienced functional deterioration, restriction and an evident swelling of the left face. Having identified the mass to arise from the mandibular condyle, an incisional biopsy was undertaken. The histology was suggestive of an arteriovenous malformation. Surgery was undertaken to excise the entire left mandibular condyle along with the lesion and then replace the temporomandibular joint with a custom made prosthesis.

DISCUSSION

Arteriovenous malformations, or Haemiangiomas, can arise in any part of the body and can often grow over time. Although benign, lesions can be locally invasive and distort anatomy, resulting in functional issues. Their risk for hemorrhage and potentially significant blood losses is also a risk factor in their presence. Rarely arteriovenous malformations can occur in hard tissue, as in this case distorting and obliterating the temporomandibular joint. One's only recourse is to remove and eliminate the lesion, however in cases such as this significant morbidity could ensue due to functional deficit. Hence consideration must be given to reconstruction.

CONCLUSION

Having excised the mass and reconstructed the temporomandibular joint with a titanium prosthesis, the patient experienced a good recovery with a return to normality.

摘要

引言

本病例报告关注一名47岁患者,其左脸迅速出现肿物。本报告对该病例、所进行的检查、治疗方法进行了分析,并对这种罕见病症及罕见表现进行了文献综述。

病例介绍

该患者出现功能恶化、受限以及左脸明显肿胀。在确定肿物起源于下颌髁突后,进行了切开活检。组织学检查提示为动静脉畸形。手术切除了整个左下颌髁突及病变组织,然后用定制假体替换颞下颌关节。

讨论

动静脉畸形,即血管瘤,可发生于身体任何部位,且通常会随时间生长。虽然是良性病变,但可局部侵袭并扭曲解剖结构,导致功能问题。其出血风险及潜在的大量失血也是其存在的危险因素。动静脉畸形很少发生于硬组织,如此例中就扭曲并破坏了颞下颌关节。唯一的解决办法是切除病变组织,然而在此类病例中,由于功能缺陷可能会导致严重的并发症。因此必须考虑进行重建。

结论

切除肿物并用钛假体重建颞下颌关节后,患者恢复良好,回归正常生活。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7724/7575646/b430f7e67cae/gr1.jpg

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