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颅底软骨-骨呼吸上皮腺样错构瘤:1例报告及文献复习

Chondro-Osseous Respiratory Epithelial Adenomatoid Hamartoma of the Skull Base: Report of a Case and Literature Review.

作者信息

Trent Monica S, Wang Beverly, Kuan Edward C

机构信息

Department of Otolaryngology - Head and Neck Surgery, University of California, Irvine Medical Center, Orange, California, United States.

Department of Anatomic and Clinical Pathology, Cytopathology, University of California, Irvine Medical Center, Orange, California, United States.

出版信息

J Neurol Surg Rep. 2025 Apr 3;86(2):e50-e56. doi: 10.1055/a-2556-0732. eCollection 2025 Apr.

Abstract

OBJECTIVE

Chondro-osseous respiratory epithelial adenomatoid hamartoma (COREAH) is a rare benign growth within the nasal cavity or paranasal sinuses. We aim to highlight the pathogenesis and clinical presentation of an extremely rare benign mass within the nasal cavity and demonstrate the workup, diagnosis, and management of this rare lesion, as well as review the literature.

METHODS

Retrospective review of a patient presenting with COREAH of the sinonasal cavity treated at a single tertiary academic medical center. The MEDLINE database was additionally searched for all case series or reports of sinonasal or skull base COREAH.

RESULTS

A total of sixteen articles were identified for review. In addition to the current case, a total of nineteen patients were identified in literature found to have sinonasal or skull base COREAH. The most common primary sites of attachment were the lateral nasal wall and posterior septum. Only two cases were identified with skull base attachment. Computed topography (CT) was employed in 84.2% of patients, and CT with MRI was utilized in 26.3% of patients. Endoscopic resection was performed in 100% of patients, with one recurrence reported in the literature and one incomplete resection requiring revision surgery.

CONCLUSION

COREAH is an extremely rare hamartoma consisting of glandular proliferation with cartilaginous and osseous stroma. COREAH is symptomatic in 100% of reported cases in the literature, and complete surgical resection is often curative. Our case represents the longest follow-up reported with 4 years postoperatively revealing no growth or recurrence of COREAH.

摘要

目的

软骨-骨化性呼吸上皮腺样错构瘤(COREAH)是鼻腔或鼻窦内一种罕见的良性肿物。我们旨在强调鼻腔内一种极其罕见的良性肿物的发病机制和临床表现,展示对该罕见病变的检查、诊断和处理方法,并对相关文献进行综述。

方法

对在一家三级学术医学中心接受治疗的鼻窦COREAH患者进行回顾性研究。另外检索MEDLINE数据库,查找所有关于鼻窦或颅底COREAH的病例系列或报告。

结果

共确定16篇文章进行综述。除了当前病例外,在文献中总共确定了19例患有鼻窦或颅底COREAH的患者。最常见的附着原发部位是鼻侧壁和后鼻中隔。仅2例确定有颅底附着。84.2%的患者采用了计算机断层扫描(CT),26.3%的患者采用了CT联合磁共振成像(MRI)。100%的患者进行了内镜下切除,文献报道有1例复发,1例不完全切除需要再次手术。

结论

COREAH是一种极其罕见的错构瘤,由腺性增生伴软骨和骨基质组成。文献报道的所有病例中COREAH均有症状,完整手术切除通常可治愈。我们的病例是报道中随访时间最长的,术后4年显示COREAH无生长或复发。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c3de/11968135/6f7115f41e7c/10-1055-a-2556-0732-i24sep0053-1.jpg

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本文引用的文献

1
Unveiling the Rarity: A Fascinating Case of Chondro-Osseous Respiratory Epithelial Adenomatoid Hamartoma (COREAH) in the Sinonasal Tract.
Indian J Otolaryngol Head Neck Surg. 2024 Jun;76(3):2809-2812. doi: 10.1007/s12070-024-04500-6. Epub 2024 Feb 1.
3
Not just another nasal polyp: Chondro-osseous respiratory epithelial adenomatoid hamartomas of the sinonasal tract.
Laryngoscope Investig Otolaryngol. 2021 May 22;6(3):376-385. doi: 10.1002/lio2.580. eCollection 2021 Jun.
4
Chondro-Osseous Respiratory Epithelial Adenomatoid Hamartoma (COREAH): Case Report and Literature Review.
Case Rep Otolaryngol. 2019 Jul 25;2019:5247091. doi: 10.1155/2019/5247091. eCollection 2019.
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Outcomes and imaging findings of respiratory epithelial adenomatoid hamartoma: a systematic review.
Int Forum Allergy Rhinol. 2019 Jun;9(6):674-680. doi: 10.1002/alr.22298. Epub 2019 Jan 18.
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Bizarre stromal cells mimicking malignancy in chondro-osseous respiratory epithelial adenomatoid hamartoma.
Pathol Int. 2017 May;67(5):276-277. doi: 10.1111/pin.12523. Epub 2017 Mar 8.
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Nasal Chondromesenchymal Hamartoma (NCMH): a systematic review of the literature with a new case report.
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