高级别双表型鼻窦肉瘤1例报告。

IF 0.6 Q4 CLINICAL NEUROLOGY
Journal of Neurological Surgery Reports Pub Date : 2022-09-13 eCollection Date: 2022-07-01 DOI:10.1055/s-0042-1755599
Sukaina Hasnie, Chad Glenn, Jo E G Peterson, Edward T El Rassi, Kibwei A McKinney
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引用次数: 4

摘要

双表型鼻窦炎肉瘤(biphenotype sinonasal sarcoma, BSNS)是由Lewis等人首次描述的一种新发现的肿瘤。2012年,它被列入世界卫生组织(世卫组织)第四版头颈部肿瘤。BSNS已被描述为一种罕见的低级别肉瘤,起源于上鼻道。据信,在过去,BSNS以前可能被诊断为其他低级别或良性恶性肿瘤。纤维肉瘤、平滑肌肉瘤和周围神经鞘肿瘤都属于BSNS的鉴别诊断。然而,BSNS不同于其他间充质鼻窦肿瘤,因为它同时显示神经和肌源性分化。迄今为止,BSNS仅在大量病例报告中得到认可,所有这些病例报告都报道了中年人鼻腔或筛气细胞中发生的低级别软组织肿瘤的相似形态学特征,并累及神经。事实上,低级别肉瘤已成为该肿瘤的一个标志性特征,它甚至被称为具有神经和肌源性特征的低级别鼻窦肉瘤或LGSSNMF。然而,我们首次在一名健康的72岁女性中报道了BSNS的高度分化。患者从外耳鼻喉科(耳、鼻、喉)转介就诊,疑似息肉切除术病理结果为BSNS阳性。初步影像显示双侧纸莎草膜、前颅窝底和额窦后表糜烂。然后,她接受了联合内镜和双冠状开放入路切除颅底病变,发现该病变包括整个双侧鼻窦腔。术后,患者出现了明显的并发症,包括颅周皮瓣感染、脑气,最终死亡。由于BSNS是一个相当新的实体,目前只进行了四个病例系列,每个病例都确定了低级别肉瘤的特征,同时具有肌源性和神经分化。组织学上,BSNS为单核梭形细胞,细胞核均匀细长,在表面型呼吸上皮良性增生之间细胞质较少,有丝分裂率低。然而,我们的病例显示多形性高染细胞,有丝分裂活性高,坏死伴骨浸润,分期为高级别。免疫组织化学也不同于先前报道的标准。本病例描述了BSNS的一个新类别,它可能会改变目前用于这种颅底肿瘤的鉴别诊断、管理和手术建议。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

High-Grade Biphenotypic Sinonasal Sarcoma: A Case Report.

High-Grade Biphenotypic Sinonasal Sarcoma: A Case Report.

High-Grade Biphenotypic Sinonasal Sarcoma: A Case Report.

High-Grade Biphenotypic Sinonasal Sarcoma: A Case Report.

Introduction  Biphenotypic sinonasal sarcoma (BSNS) is a recently found entity that first described by Lewis et al. It was then added to the 4th edition of the World Health Organization (WHO) of head and neck tumors in 2012. BSNS has been described as a rare low-grade sarcoma arising in the upper sinonasal tract. It is believed that in the past, BSNS was, likely, previously diagnosed as other low-grade or benign malignancies. Fibrosarcoma, leiomyosarcoma, and peripheral nerve sheath tumors, all fall within the differential diagnosis of BSNS. However, BSNS is unlike other mesenchymal sinonasal tumors, as it displays both neural and myogenic differentiation. BSNS has thus far been recognized in only a hand full of case reports, all of which have reported similar morphologic features of a low-grade soft tissue tumor with neural involvement arising from the nasal cavity or ethmoid air cells in middle aged individuals. In fact, being low-grade sarcoma became such a hallmark characteristic of this tumor that it even received the name low-grade sinonasal sarcoma with neural and myogenic features or LGSSNMF. Case Presentation  We present, however, for the first time, a high-grade differentiation of BSNS in an otherwise healthy 72-year-old female. The patient was referred from an outside ENT (ear, nose, and throat) after pathology from a presumed polypectomy returned positive for a BSNS. Initial imaging revealed erosion through the bilateral lamina papyracea, anterior cranial fossa floor, and posterior table of the frontal sinus. She then underwent a combined endoscopic and bicoronal open approach for resection of the skull base lesion that was found to encompass the entirety of the sinonasal cavities bilaterally. Postoperatively, the patient underwent significant complications including infection of the pericranial flap, pneumocephalus, and eventually death. Discussion  As BSNS is a fairly new entity, currently there has only been four case series conducted, each identifying features of a low-grade sarcoma with both myogenic and neural differentiation. Histologically, BSNS has monophasic spindle cells with uniform, elongated nuclei with scant cytoplasm between benign proliferations of surface-type respiratory epithelium, with a low mitotic rate. Our case, however, revealed pleomorphic hyperchromatic cells with high mitotic activity and necrosis with invasion of bone, staging it as high grade. Immunohistochemistry also differed from the previously reported standards. This case describes a new category for BSNS which may change the differential diagnosis, management, and surgical recommendations that are currently utilized for this skull base neoplasm.

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