原发性筛窦尤文氏肉瘤颅内累及一例罕见病例并文献复习。

Ameni Amri, Rim Braham, Salma Kamoun, Omar Feki, Alia Methnani, Mohamed Dhaha, Souheil Jbali, Sawssen Dhambri, Skander Kedous
{"title":"原发性筛窦尤文氏肉瘤颅内累及一例罕见病例并文献复习。","authors":"Ameni Amri, Rim Braham, Salma Kamoun, Omar Feki, Alia Methnani, Mohamed Dhaha, Souheil Jbali, Sawssen Dhambri, Skander Kedous","doi":"10.1177/01455613251333184","DOIUrl":null,"url":null,"abstract":"<p><p>Ewing sarcoma (ES) is a rare and highly-aggressive tumor. This malignancy derives from primitive neuroectodermal cells. The most frequently-affected population is adolescents and young adults, and the tumor typically grows in the bones. ES of the sinonasal tract is an exceedingly-rare situation. Particularly, primary occurrence in the ethmoid sinus is extremely scarce, with only 27 cases documented worldwide. Extension to the anterior cranial fossa is even thinner on the ground with limited cases in literature. We report the case of a 20-year-old female with history of Hodgkin disease presenting with facial pain, anosmia, and visual impairment on the right side. Imaging revealed a large invasive tumor originating from the right ethmoid sinus, with extension into the orbit and anterior cranial fossa. On biopsy, the tumor showed focal expression of Epithelial membrane antigen (EMA) and CD99 and EWSR1 rearrangement. The diagnosis of ES was confirmed. The patient underwent neoadjuvant chemotherapy with etoposide. Given the stability of the tumor, the patient was deemed inoperable. The decision was to proceed with chemoradiotherapy. Unfortunately, the patient passed away during treatment due to intracranial hypertension with seizures 12 months after diagnosis. This case underscores the rarity of sinonasal ES, particularly with primary involvement in the ethmoid sinus with anterior cranial fossa invasion. We conducted an exhaustive review of literature on similar cases to highlight the importance of a multidisciplinary approach combining advanced imaging, molecular diagnostics, chemotherapy, surgical intervention, and radiotherapy to optimize patients' survival. Sinonasal ES, especially in the ethmoid sinus with cranial extension, is a rare and challenging situation. Detection at an early stage through meticulous imaging, immunohistochemistry, and molecular biology tools is crucial to initiate a multimodal treatment approach. Through this case report and literature review, we aimed to contribute to the growing literature on rare sinonasal malignancies.</p>","PeriodicalId":93984,"journal":{"name":"Ear, nose, & throat journal","volume":" ","pages":"1455613251333184"},"PeriodicalIF":0.0000,"publicationDate":"2025-04-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Primary Ewing Sarcoma of the Ethmoid Sinus With Intracranial Involvement: A Rare Case and Literature Review.\",\"authors\":\"Ameni Amri, Rim Braham, Salma Kamoun, Omar Feki, Alia Methnani, Mohamed Dhaha, Souheil Jbali, Sawssen Dhambri, Skander Kedous\",\"doi\":\"10.1177/01455613251333184\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Ewing sarcoma (ES) is a rare and highly-aggressive tumor. This malignancy derives from primitive neuroectodermal cells. The most frequently-affected population is adolescents and young adults, and the tumor typically grows in the bones. ES of the sinonasal tract is an exceedingly-rare situation. Particularly, primary occurrence in the ethmoid sinus is extremely scarce, with only 27 cases documented worldwide. Extension to the anterior cranial fossa is even thinner on the ground with limited cases in literature. We report the case of a 20-year-old female with history of Hodgkin disease presenting with facial pain, anosmia, and visual impairment on the right side. Imaging revealed a large invasive tumor originating from the right ethmoid sinus, with extension into the orbit and anterior cranial fossa. On biopsy, the tumor showed focal expression of Epithelial membrane antigen (EMA) and CD99 and EWSR1 rearrangement. The diagnosis of ES was confirmed. The patient underwent neoadjuvant chemotherapy with etoposide. Given the stability of the tumor, the patient was deemed inoperable. The decision was to proceed with chemoradiotherapy. Unfortunately, the patient passed away during treatment due to intracranial hypertension with seizures 12 months after diagnosis. This case underscores the rarity of sinonasal ES, particularly with primary involvement in the ethmoid sinus with anterior cranial fossa invasion. We conducted an exhaustive review of literature on similar cases to highlight the importance of a multidisciplinary approach combining advanced imaging, molecular diagnostics, chemotherapy, surgical intervention, and radiotherapy to optimize patients' survival. Sinonasal ES, especially in the ethmoid sinus with cranial extension, is a rare and challenging situation. Detection at an early stage through meticulous imaging, immunohistochemistry, and molecular biology tools is crucial to initiate a multimodal treatment approach. Through this case report and literature review, we aimed to contribute to the growing literature on rare sinonasal malignancies.</p>\",\"PeriodicalId\":93984,\"journal\":{\"name\":\"Ear, nose, & throat journal\",\"volume\":\" \",\"pages\":\"1455613251333184\"},\"PeriodicalIF\":0.0000,\"publicationDate\":\"2025-04-23\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Ear, nose, & throat journal\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.1177/01455613251333184\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"\",\"JCRName\":\"\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Ear, nose, & throat journal","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.1177/01455613251333184","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"","JCRName":"","Score":null,"Total":0}
引用次数: 0

摘要

尤文氏肉瘤(ES)是一种罕见的高侵袭性肿瘤。这种恶性肿瘤起源于原始神经外胚层细胞。最常受影响的人群是青少年和年轻人,肿瘤通常生长在骨骼中。鼻窦炎是一种极为罕见的情况。特别是,主要发生在筛窦的病例极为罕见,全世界只有27例。前颅窝的延伸在地面上甚至更薄,文献中有有限的病例。我们报告一个20岁的女性霍奇金病病史的情况下,表现为面部疼痛,嗅觉丧失,并在右侧的视觉障碍。影像显示一个大的侵袭性肿瘤起源于右筛窦,并延伸到眼眶和颅前窝。活检显示肿瘤局灶性表达上皮膜抗原(EMA)、CD99和EWSR1重排。确诊为ES。患者接受依托泊苷新辅助化疗。鉴于肿瘤的稳定性,该患者被认为不适合手术。决定继续进行放化疗。不幸的是,患者在诊断后12个月因颅内高压和癫痫发作而在治疗期间去世。本病例强调了窦鼻性ES的罕见性,特别是原发性累及筛窦并侵犯前颅窝的病例。我们对类似病例的文献进行了详尽的回顾,以强调将先进的影像学、分子诊断、化疗、手术干预和放疗相结合的多学科方法对优化患者生存的重要性。窦鼻ES是一种罕见且具有挑战性的情况,特别是在颅延伸的筛窦。通过精细的成像、免疫组织化学和分子生物学工具进行早期检测对于启动多模式治疗方法至关重要。通过这一病例报告和文献回顾,我们旨在为越来越多的关于罕见鼻窦恶性肿瘤的文献做出贡献。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Primary Ewing Sarcoma of the Ethmoid Sinus With Intracranial Involvement: A Rare Case and Literature Review.

Ewing sarcoma (ES) is a rare and highly-aggressive tumor. This malignancy derives from primitive neuroectodermal cells. The most frequently-affected population is adolescents and young adults, and the tumor typically grows in the bones. ES of the sinonasal tract is an exceedingly-rare situation. Particularly, primary occurrence in the ethmoid sinus is extremely scarce, with only 27 cases documented worldwide. Extension to the anterior cranial fossa is even thinner on the ground with limited cases in literature. We report the case of a 20-year-old female with history of Hodgkin disease presenting with facial pain, anosmia, and visual impairment on the right side. Imaging revealed a large invasive tumor originating from the right ethmoid sinus, with extension into the orbit and anterior cranial fossa. On biopsy, the tumor showed focal expression of Epithelial membrane antigen (EMA) and CD99 and EWSR1 rearrangement. The diagnosis of ES was confirmed. The patient underwent neoadjuvant chemotherapy with etoposide. Given the stability of the tumor, the patient was deemed inoperable. The decision was to proceed with chemoradiotherapy. Unfortunately, the patient passed away during treatment due to intracranial hypertension with seizures 12 months after diagnosis. This case underscores the rarity of sinonasal ES, particularly with primary involvement in the ethmoid sinus with anterior cranial fossa invasion. We conducted an exhaustive review of literature on similar cases to highlight the importance of a multidisciplinary approach combining advanced imaging, molecular diagnostics, chemotherapy, surgical intervention, and radiotherapy to optimize patients' survival. Sinonasal ES, especially in the ethmoid sinus with cranial extension, is a rare and challenging situation. Detection at an early stage through meticulous imaging, immunohistochemistry, and molecular biology tools is crucial to initiate a multimodal treatment approach. Through this case report and literature review, we aimed to contribute to the growing literature on rare sinonasal malignancies.

求助全文
通过发布文献求助,成功后即可免费获取论文全文。 去求助
来源期刊
自引率
0.00%
发文量
0
×
引用
GB/T 7714-2015
复制
MLA
复制
APA
复制
导出至
BibTeX EndNote RefMan NoteFirst NoteExpress
×
提示
您的信息不完整,为了账户安全,请先补充。
现在去补充
×
提示
您因"违规操作"
具体请查看互助需知
我知道了
×
提示
确定
请完成安全验证×
copy
已复制链接
快去分享给好友吧!
我知道了
右上角分享
点击右上角分享
0
联系我们:info@booksci.cn Book学术提供免费学术资源搜索服务,方便国内外学者检索中英文文献。致力于提供最便捷和优质的服务体验。 Copyright © 2023 布克学术 All rights reserved.
京ICP备2023020795号-1
ghs 京公网安备 11010802042870号
Book学术文献互助
Book学术文献互助群
群 号:481959085
Book学术官方微信