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病例报告
鼻腔鼻窦型血管外皮细胞瘤合并海绵状血管瘤一例
王俊华 黄宏亮 王肖 翟晓静 张皓

Cite this article as: Wang JH, Huang HL, Wang X, et al. One case of sinonasal-type haemangiopericytma combined with cavernous hemangioma[J]. Chin J Magn Reson Imaging, 2022, 13(3): 93-94.本文引用格式:王俊华, 黄宏亮, 王肖, 等. 鼻腔鼻窦型血管外皮细胞瘤合并海绵状血管瘤一例[J]. 磁共振成像, 2022, 13(3): 93-94. DOI:10.12015/issn.1674-8034.2022.03.020.


[摘要] 本研究经兰州大学第一医院伦理委员会批准,免除受试者知情同意,批准文号:LDYYLL2021-365。患者女,68岁,13年前发现右侧鼻翼一黄豆大小肿物,质韧,活动度可,无特殊不适,未予诊治,此后肿物一直存在。于9年前鼻外伤后就诊于当地医院,检查提示右侧鼻腔肿物,近一年患者自觉鼻腔肿物逐渐增大,蚕豆大小,活动度可,质韧,伴双侧交替性鼻塞,嗅觉减退,流涕,偶有鼻出血。遂至兰州大学第一医院就诊。实验室检查无特殊。专科检查:右侧鼻翼可见一约5 mm×5 mm隆起,质软,右侧鼻腔堵满红色新生物,右侧鼻腔结构显示不清,左侧鼻腔黏膜略充血,左侧下鼻甲充血肿胀。CT增强示:右侧鼻前庭可见一大小约25 mm×14 mm的不均匀明显强化的软组织肿物影,形态欠规则,与鼻中隔及鼻腔外侧壁关系密切,动脉期CT值约42 HU,静脉期CT值约96 HU。右侧鼻翼皮下见大小约9 mm×8 mm的环形强化软组织结节影,边界清,形态规则,中央部分动脉期CT值约为48 HU,静脉期CT值约为56 HU,环形强化部分动脉期CT值约为68 HU,静脉期CT值约为66 HU (图1A~1B)。浅表肿物彩超示:右侧近鼻翼皮下实质性包块。MRI增强示:右侧鼻腔见大小约26 mm×14 mm的T1等低信号、T2高信号、DWI高信号、ADC低信号肿物,边界清,形态欠规则,内可见粗大流空血管;右侧鼻翼皮下软组织内见大小约为9 mm×8 mm的T1等信号、T2高信号、DWI高信号、ADC低信号结节,边界清,形态规则(图1C~1F)。患者于全麻下行鼻背肿物切除术及鼻内镜下等离子鼻腔肿物切除术,术中鼻翼皮下摘除约2.0 cm×1.0 cm×0.5 cm的软组织肿物,表面光滑,与周围组织无粘连。鼻内镜下右侧下鼻甲前端见直径约1.5 cm×1.0 cm×0.4 cm的软组织肿物,质韧,形态欠规则,等离子沿肿物表面切除。免疫组化:Vimentin (+),CD31 (血管+),D2-40 (-),CKP (被覆上皮+),SMA (梭形细胞2+),Desmin (-),Ki-67 (5%)。病理诊断(图1G~1H):鼻内病损:血管外皮细胞瘤;鼻外病损:海绵状血管瘤。患者术后恢复良好,无复发。
[关键词] 血管外皮细胞瘤;鼻腔;鼻窦;血管瘤;磁共振成像
[Keywords] haemangiopericytma;nasal cavity;paranasal sinus;hemangioma;magnetic resonance imaging

王俊华 1   黄宏亮 2   王肖 1   翟晓静 1   张皓 2*  

1 兰州大学第一临床医学院,兰州 730000

2 兰州大学第一医院放射科,兰州 730000

张皓,E-mail:zhanghao@lzu.edu.cn

作者利益冲突声明:全部作者均声明无利益冲突。


收稿日期:2021-11-29
接受日期:2022-03-01
中图分类号:R445.2  R730.262  R732.2 
文献标识码:B
DOI: 10.12015/issn.1674-8034.2022.03.020
本文引用格式:王俊华, 黄宏亮, 王肖, 等. 鼻腔鼻窦型血管外皮细胞瘤合并海绵状血管瘤一例[J]. 磁共振成像, 2022, 13(3): 93-94. DOI:10.12015/issn.1674-8034.2022.03.020

       本研究经兰州大学第一医院伦理委员会批准,免除受试者知情同意,批准文号:LDYYLL2021-365。患者女,68岁,13年前发现右侧鼻翼一黄豆大小肿物,质韧,活动度可,无特殊不适,未予诊治,此后肿物一直存在。于9年前鼻外伤后就诊于当地医院,检查提示右侧鼻腔肿物,近一年患者自觉鼻腔肿物逐渐增大,蚕豆大小,活动度可,质韧,伴双侧交替性鼻塞,嗅觉减退,流涕,偶有鼻出血。遂至兰州大学第一医院就诊。实验室检查无特殊。专科检查:右侧鼻翼可见一约5 mm×5 mm隆起,质软,右侧鼻腔堵满红色新生物,右侧鼻腔结构显示不清,左侧鼻腔黏膜略充血,左侧下鼻甲充血肿胀。CT增强示:右侧鼻前庭可见一大小约25 mm×14 mm的不均匀明显强化的软组织肿物影,形态欠规则,与鼻中隔及鼻腔外侧壁关系密切,动脉期CT值约42 HU,静脉期CT值约96 HU。右侧鼻翼皮下见大小约9 mm×8 mm的环形强化软组织结节影,边界清,形态规则,中央部分动脉期CT值约为48 HU,静脉期CT值约为56 HU,环形强化部分动脉期CT值约为68 HU,静脉期CT值约为66 HU (图1A1B)。浅表肿物彩超示:右侧近鼻翼皮下实质性包块。MRI增强示:右侧鼻腔见大小约26 mm×14 mm的T1等低信号、T2高信号、DWI高信号、ADC低信号肿物,边界清,形态欠规则,内可见粗大流空血管;右侧鼻翼皮下软组织内见大小约为9 mm×8 mm的T1等信号、T2高信号、DWI高信号、ADC低信号结节,边界清,形态规则(图1C1F)。患者于全麻下行鼻背肿物切除术及鼻内镜下等离子鼻腔肿物切除术,术中鼻翼皮下摘除约2.0 cm×1.0 cm×0.5 cm的软组织肿物,表面光滑,与周围组织无粘连。鼻内镜下右侧下鼻甲前端见直径约1.5 cm×1.0 cm×0.4 cm的软组织肿物,质韧,形态欠规则,等离子沿肿物表面切除。免疫组化:Vimentin (+),CD31 (血管+),D2-40 (-),CKP (被覆上皮+),SMA (梭形细胞2+),Desmin (-),Ki-67 (5%)。病理诊断(图1G1H):鼻内病损:血管外皮细胞瘤;鼻外病损:海绵状血管瘤。患者术后恢复良好,无复发。

图1  女,68岁,鼻腔鼻窦型血管外皮细胞瘤合并鼻翼海绵状血管瘤。1A~1B:CT副鼻窦+鼻骨增强扫描。1A:动脉期,右侧鼻前庭见一边缘强化的软组织肿物影,形态欠规则,与鼻中隔及鼻腔外侧壁关系密切,CT值约42 HU;右侧鼻翼皮下见大小约9 mm×8 mm的环形强化软组织结节影,边界清,形态规则,中央部分CT值约为48 HU,环形强化部分CT值约为68 HU。1B:静脉期,右侧鼻前庭肿物CT值约96 HU,肿物呈渐进性明显不均匀强化;右侧鼻翼皮下结节中央部分CT值约为56 HU,环形强化部分CT值约为66 HU。1C~1F:MRI鼻窦平扫。1C:T1WI,右侧鼻腔T1等、稍低信号肿物,右侧鼻翼皮下T1等信号结节;1D:T2WI,右侧鼻腔T2高信号肿物,右侧鼻翼皮下T2高信号结节;1E:DWI,右侧鼻腔DWI高信号肿物,右侧鼻翼皮下DWI高信号结节;1F:ADC,右侧鼻腔ADC低信号肿物,右侧鼻翼皮下ADC低信号结节。1G~1H (HE ×200):鼻内病损(血管外皮细胞瘤)组织表面被覆假复层纤毛柱状上皮,上皮下梭形细胞增生,呈旋涡状排列,间质血管丰富,组织充血水肿,慢性炎细胞浸润。
Fig. 1  Female, 68-year-old, sinonasal-type haemangiopericytma combined with cavernous hemangioma. 1A-1B: Paranasal sinus+nasal bone enhancement CT. 1A: Arterial phase, a marginal enhanced soft tissue mass in the right nasal vestibule, with irregular shape, which was closely related to the nasal septum and the lateral wall of the nasal cavity, the CT value was about 42 HU. In the subcutaneous area of the right alar of the nose, there were 9 mm×8 mm ring enhanced soft tissue nodules with clear boundary and regular shape, the CT value of the central part was about 48 HU, and the CT value of the ring enhanced part was about 68 HU. 1B: Venous phase, the CT value of the right nasal vestibular mass was about 96 HU, and the mass showed progressive and obvious uneven enhancement. The CT value of the central part of the subcutaneous nodule on the right alar nose was about 56 HU, and the CT value of the ring enhancement part was about 66 HU. 1C-1F: MRI plain sinus scan. 1C: T1WI, equal, slightly lower signal mass in the right nasal cavity, subcutaneous equal signal nodule in the right nasal alar; 1D: T2WI, higher signal mass in right nasal cavity, higher signal nodule subcutaneously in right alar nose; 1E: DWI, higher signal mass of right nasal cavity, and higher signal nodule in subcutaneous of right alar nose; 1F: ADC, lower signal mass in the right nasal cavity, and lower signal nodule in the subcutaneous area of the right alar nose. 1G-1H (HE ×200): The surface of the nasal lesion (hemangiopericytoma) was covered with pseudolayered ciliated columnar epithelium. Subepithelial spindle cells were proliferous and arranged in a spiral pattern. Interstitial blood vessels were abundant, and the tissue was hyperemic and edema, with chronic inflammatory cell infiltration.

讨论

       血管外皮细胞瘤是起源于间叶组织的低度恶性肿瘤,好发于腹膜后及下肢,头颈部少见,鼻腔鼻窦罕见[1]。本病例为鼻腔鼻窦型血管外皮细胞瘤(sinonasal-type haemangiopericytma,SNTHPC)合并鼻部海绵状血管瘤,因二者影像学表现相似且发生部位相近,易将二者误诊为同一疾病加以诊治。SNTHPC在所有鼻窦肿瘤中所占比例小于0.5%~1%[2],占鼻窦血管源性肿瘤的2.5%[3],60~70岁女性发病率较高。以单侧鼻息肉、鼻出血(78%)、鼻塞(52%)及头痛(17%)为主要临床症状[4]。鼻腔鼻窦肿瘤的诊断首选内镜检查,其次为CT和MRI。在MRI上,典型的SNTHPC表现为T1等信号,T2等低信号的不规则形肿块影,边界常较清晰,增强呈明显均匀或不均匀强化。其内粗大“流空血管影”,具有重要的鉴别诊断价值[5],本病例除T2高信号外,其余表现与其相符。CT可更清晰地显示病变对邻近骨质的破坏情况,肿瘤多呈等、稍高密度,钙化极少,增强后均匀或不均匀显著强化。另有学者认为血管造影对病变显示及降低术中肿瘤出血风险有一定价值[6]。文献报道,SNTHPC免疫组化Vimentin和Actin通常表现为强阳性,Desmin、CD117、CD34、CD99、S-100以及 Bcl-2常为阴性[7],本例与文献相符。SNTHPC以手术治疗为主,其中内镜下完全切除肿瘤,对降低复发率及术后监测意义重大[8]。SNTHPC需与鼻咽血管纤维瘤、内翻性乳头状瘤、血管瘤等进行鉴别[9]。血管瘤在CT平扫上呈等或稍高密度,海绵状血管瘤偶见静脉石,毛细胞血管瘤强化迅速且均匀,海绵状血管瘤不均匀、渐进性强化,由于这一影像特点,导致了本例患者的误诊。

       综上所述,SNTHPC较为罕见,同时合并鼻部海绵状血管瘤者较易误诊,因此,需将影像与病理及免疫组化相结合,最终可以确诊。

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