小儿颞骨表面低级别骨肉瘤1例报告及文献复习。

IF 1.7 Q2 PEDIATRICS Clinical Medicine Insights-Pediatrics Pub Date : 2019-06-26 eCollection Date: 2019-01-01 DOI:10.1177/1179556519855381
Noémie Villemure-Poliquin, Mathieu Trudel, Sebastien Labonté, Valérie Blouin, Gaétan Fradet
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引用次数: 3

摘要

研究目的:原发性颞骨骨肉瘤是一种极为罕见的儿科疾病。到目前为止,在英国文献中只报道了3例。我们描述了一个16岁的女孩与乳突骨骨肉瘤的额外情况。本研究报告一例罕见的儿童颞骨低级别表面骨肉瘤病例。使用的材料和方法:进行文献综述,以更好地了解头颈部的儿童骨肉瘤,优化其研究,描述其组织病理学和放射学特征,并建立最佳的药物和手术治疗方式。使用PubMed和Embase库对之前发表的数据进行研究,关键词如下。结果:患者在3周的时间内表现为快速进展的左耳后病变。影像学检查表现为侵袭性和侵袭性特征。随后行开放性活组织检查,确诊为低级别表面骨肉瘤。根据多学科团队的意见,我们决定进行广泛手术切缘的完整手术切除。我们没有给予任何辅助治疗。术后26个月的对照计算机断层扫描(CT)仍未显示复发迹象。结论:骨肉瘤是发生在头颈部的侵袭性恶性肿瘤,发病率仅为6% ~ 10%。它们约占头颈癌的1%,通常为高级别病变。颞骨受累是罕见的,特别是对儿童患者的低级别病变。除了报告第4例原发性颞骨骨肉瘤的儿科病例外,本研究还描述了其具体的临床、组织病理学和放射学表现,以改善这种特殊临床实体患者的管理和预后。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

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Low-Grade Surface Osteosarcoma of the Temporal Bone in Paediatric Patients: A Case Report and Literature Review.

Purpose of the study: Primary osteosarcoma of the temporal bone is an exceedingly rare pathology in the paediatric population. As of now, only 3 cases have been reported in the English literature. We describe the additional case of a 16-year-old girl with an osteosarcoma of the mastoid bone. This study aims to report a rare paediatric case of low-grade surface osteosarcoma of the temporal bone.

Materials and methods used: A literature review was performed to better understand paediatric osteosarcomas of the head and neck region, to optimize their investigation, to describe their histopathological and radiological characteristics, and to establish the optimal modalities of medical and surgical treatments. The research of previous published data was done using PubMed and Embase library with the keywords mentioned below.

Results: The patient presented with a rapidly progressive left retroauricular lesion over a 3-week period. Radiological studies demonstrated aggressive and invasive features. An open biopsy followed and confirmed the diagnosis of a low-grade surface osteosarcoma. In accordance with the multidisciplinary team, we decided to perform a complete surgical resection with wide surgical margins. We did not administer any adjuvant therapies. A control computed tomography (CT) scan obtained 26 months postoperatively still showed no signs of recurrence.

Conclusion: Osteosarcomas are aggressive malignant neoplasms found in the head and neck region in only 6% to 10% of cases. They represent approximately 1% of head and neck cancers, and these are generally high-grade lesions. Temporal bone involvement is rare, particularly for low-grade lesions in paediatric patients. In addition to reporting the fourth paediatric case of primary temporal bone osteosarcoma, this study describes its specific clinical, histopathological, and radiological findings, to improve the management and the prognostic of patients affected with this particular clinical entity.

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