眼眶血管平滑肌瘤伴GJA4突变似海绵状静脉畸形1例报告及综合复习。

IF 0.8 Q4 OPHTHALMOLOGY
Marie Callet, Arnault Tauziede-Espariat, Augustin Lecler, Sorin Aldea, Pierre Vincent Jacomet, Olivier Galatoire, Mathieu Veyrat
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引用次数: 0

摘要

摘要眼眶血管平滑肌瘤是一种罕见的良性肿瘤,其影像学和病理组织学特征重叠,与眼眶海绵状静脉畸形相似。我们提出的情况下,一个42岁的妇女与顶端眼眶血管平滑肌瘤偶然发现在评估偏头痛。磁共振成像显示一界限分明、钆增强的腔内肿块,初步提示OCVM。手术切除通过鼻内窥镜入路进行,完全切除无复发。组织病理学分析和分子检测证实了诊断,鉴定出体细胞GJA4 (p.Gly41Cys)突变。本病例强调了区分眼眶血管平滑肌瘤和OCVM的诊断挑战,并强调了免疫组织化学和分子分析的重要性,特别是GJA4突变筛查,以实现诊断准确性。GJA4突变的鉴定可以帮助临床医生和病理学家更好地表征真正的血管平滑肌瘤,特别是在OCVM和血管平滑肌瘤海绵状亚型之间的区别在组织学上不明确的情况下。血管平滑肌瘤可能在眼眶病理中未被充分认识,并且可能代表以前归类为OCVM或颅内脑膜瘤的病变的一个子集。一个全面的,多模式的诊断方法是必要的适当分类和管理这些肿瘤。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Orbital angioleiomyoma with GJA4 mutation mimicking cavernous venous malformation: a case report and comprehensive review.

Orbital angioleiomyomas are rare benign tumors that can mimic orbital cavernous venous malformations (OCVM) due to overlapping radiological and histopathological features. We present the case of a 42-year-old woman with an apical orbital angioleiomyoma incidentally discovered during evaluation for migraines. Magnetic resonance imaging revealed a well-defined, gadolinium-enhancing intraconal mass, initially suggestive of OCVM. Surgical excision was performed via an endonasal endoscopic approach, achieving complete removal without recurrence. Histopathological analysis and molecular testing confirmed the diagnosis, identifying a somatic GJA4 (p.Gly41Cys) mutation.This case highlights the diagnostic challenge of distinguishing orbital angioleiomyomas from OCVM and emphasizes the importance of immunohistochemistry and molecular analysis, particularly GJA4 mutation screening, in achieving diagnostic accuracy. The identification of GJA4 mutations may assist clinicians and pathologists in better characterizing true angioleiomyomas, especially in cases where the distinction between OCVM and the cavernous subtype of angioleiomyoma is histologically ambiguous. Angioleiomyomas may be underrecognized in orbital pathology and could represent a subset of lesions previously classified as OCVM or intracranial meningiomas. A comprehensive, multimodal diagnostic approach is essential for appropriate classification and management of these tumors.

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来源期刊
CiteScore
2.40
自引率
9.10%
发文量
136
期刊介绍: Orbit is the international medium covering developments and results from the variety of medical disciplines that overlap and converge in the field of orbital disorders: ophthalmology, otolaryngology, reconstructive and maxillofacial surgery, medicine and endocrinology, radiology, radiotherapy and oncology, neurology, neuroophthalmology and neurosurgery, pathology and immunology, haematology.
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