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中耳和乳突的复发性血管球瘤(“真性”血管球瘤)

Recurrent glomangioma ("true" glomus tumor) of the middle ear and mastoid.

作者信息

Kaufman Adam C, Brant Jason A, Luu Neil N, LiVolsi Virginia A, Bigelow Douglas C

机构信息

Department of Otorhinolaryngology Head and Neck Surgery, Hospital of the University of Pennsylvania, Philadelphia, PA, USA.

Perelman School of Medicine at the University of Pennsylvania, Philadelphia, PA, USA.

出版信息

World J Otorhinolaryngol Head Neck Surg. 2019 Dec 13;5(4):175-179. doi: 10.1016/j.wjorl.2019.01.003. eCollection 2019 Dec.

Abstract

OBJECTIVE

To review current literature and experience with glomangiomas, or true glomus tumors of the middle ear and mastoid as well asto report on the exceptionally rare case of a glomangiomastemming from the middle ear space with multiple recurrences.

METHODS

Review of existing world literature and description of personal experience with rare cases of a glomangioma of the middle ear and mastoid.

RESULTS

Review of existing literature revealed two cases of patients presenting with tinnitus and hearing loss refractory to medical management. Both patients were ultimately diagnosed with glomangioma on histopathology. Complete surgical excision is thought to be curative.

PATIENT

A 36-year-old woman presented with a rare case of a glomangioma of the middle ear presenting with unilateral hearing loss. She was noted to have a mass behind the tympanic membrane. Imaging revealed a diffuse mass filling the mastoid air cells. Imaging characteristics and histology were consistent with a glomangioma.

INTERVENTION

Initial resection via mastoidectomy using a postauricular approach. The tympanic membrane was reconstructed with temporalis tissue. Follow-up revision tympanomastoidectomy was performed upon recurrence of disease. The chorda tympani were sacrificed due to tumor involvement. The incus and head of the malleus were removed to gain better access to the tumor. The ossicular chain was reconstructed with a Goldenberg Total Ossicular Prosthesis.

MAIN OUTCOME MEASURE

Recurrence of disease.

FOLLOW-UP: In the 67 months since her most recent surgery, there has been no evidence of recurrence by CT or physical exam.

CONCLUSION

Glomangioma of the middle ear represents an exceptionally rare entity that can present in a similar fashion to a paraganglioma.

摘要

目的

回顾有关中耳和乳突血管球瘤(即真性血管球瘤)的当前文献及经验,并报告一例源于中耳腔且多次复发的极为罕见的血管球瘤病例。

方法

回顾世界现有文献并描述中耳和乳突血管球瘤罕见病例的个人经验。

结果

回顾现有文献发现两例患者出现耳鸣和听力损失,药物治疗无效。两名患者最终经组织病理学诊断为血管球瘤。完整手术切除被认为可治愈。

患者

一名36岁女性,患有罕见的中耳血管球瘤,表现为单侧听力损失。发现其鼓膜后有肿物。影像学检查显示弥漫性肿物充满乳突气房。影像学特征和组织学与血管球瘤一致。

干预措施

最初采用耳后入路经乳突切除术进行切除。用颞肌组织重建鼓膜。疾病复发后进行了二次鼓室乳突切除术。因肿瘤侵犯牺牲了鼓索神经。切除砧骨和锤骨头以更好地暴露肿瘤。用戈德堡全听骨假体重建听骨链。

主要观察指标

疾病复发情况。

随访

自她最近一次手术以来的67个月里,CT检查或体格检查均未发现复发迹象。

结论

中耳血管球瘤是一种极为罕见的疾病,其表现可能与副神经节瘤相似。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0564/7015857/a3acb1aa3a1e/gr1.jpg

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