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支气管内血管球瘤二例并文献复习

Bronchial glomus tumor: report of 2 cases and review of the literature

摘要:

目的 探讨支气管内血管球瘤的临床表现、病理特征、诊断及鉴别诊断.方法 对2例支气管内血管球瘤行组织病理学检查及免疫组织化学染色,1例通过支气管镜活检证实,1例通过外科手术后病理证实.以“支气管血管球瘤”和“bronchial glomus tumor”为关键词,由万方数据库、维普网、中国知网和PubMed数据库进行文献检索,对2例病例结合相关文献进行复习.结果 2例均为男性,年龄为48岁和54岁.气管镜下发现支气管内新生物,例1荧光镜下可见明显粉染,气管镜活检组织病理检查可见间质内细胞巢,细胞呈圆形,胞质淡染,边界不清,细胞核轻度不规则.免疫组织化学染色示细胞角蛋白(CK)阴性,波形蛋白阳性,平滑肌肌动蛋白(SMA)弱阳性,突触素部分阳性,CD34血管内皮细胞阳性.病理诊断:右上肺内侧段血管球瘤.例2术后标本病理结果提示右主支气管间叶来源肿瘤,侵及黏膜下层,未侵犯气管软骨,肿瘤细胞呈巢状分布,间质血管丰富,细胞异形性不明显,细胞中等大小,核圆形,未见核分裂象.免疫组织化学染色波形蛋白和SMA阳性,CD99阳性.病理诊断:右主支气管血管球瘤(恶性潜能未定性).在数据库中共检索出16篇文献,15篇可用,共有15例,男12例,女3例,发病年龄20 ~ 79岁.其中发生于左主支气管8例,右主支气管2例,右中间段支气管3例,右上叶支气管2例.肿瘤直径为0.7~6.5 cm,临床表现为咳嗽、呼吸困难伴或不伴发热7例,痰中带血7例,肺不张4例.免疫组织化染色CK阴性,SMA阳性.结论 发生于支气管内的血管球瘤罕见,临床表现缺乏特异性,容易与支气管哮喘相混淆.组织病理学检查分为实体性球瘤、球血管瘤、球血管平滑肌瘤,免疫组织化学表现为波形蛋白和SMA阳性,CK阴性,不表达上皮标志物.临床应与硬化性血管瘤、血管外皮细胞瘤、类癌及上皮性肿瘤相鉴别.

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abstracts:

Objective Glomus tumor is a small,predominantly benign tumor,and typically occurs in the soft tissues,rarely in bronchus.The aim of this study was to discuss the clinical manifestation,histology,diagnosis and differential diagnosis of bronchial glomus tumor.Methods We studied the histopathological and immunohistochemical results of 2 cases with bronchial glomus tumor.One case was diagnosed by bronchoscopic biopsy,and another by surgery.We searched Wanfang,VIP,CNKI and PubMed database for related articles with key word "bronchial glomus tumor" both in English and in Chinese for literature review.Results Fiberoptic bronchoscopy demonstrated a bronchial neoplasm in both 2 cases.For Case one,the tumor was pink under fluorescence bronchoscopy,and was histologically composed of groups of nuclear-irregular round cells in interstices,with pale staining plasma and unclear boundary.Immunohistochemically,cytokeratin (CK) was negative,while vimentin and CD34 vascular endothelial cell were positive,smooth muscle actin (SMA) weakly positive,and Synaptophysin partially positive in tumor cells.These results led to the diagnosis of bronchial glomus tumor of right upper lobe bronchus.The tumor of Case two was histologically from the right main bronchus mesechyma,and it invaded into submucosa,but not involving the tracheal cartilage.Histological examination showed groups of medium sized tumor cells with round nuclei,and abundance of interstitial vasculature.No cellular atypia or mitoses were observed.Immunohistochemical staining demonstrated positive reactivity for vimentin,SMA and CD99.Pathological diagnosis was right main bronchus glomus tumor (malignant potential indeterminacy).We identified 16 studies from databases,of which 15 studies including 15 cases (12 males,3 females) were applicable.Age of onset ranged from 20 to 79 years.The lesion was in the left main bronchus in 8 cases,the right main broncus in 2,the right middle lobe bronchus in 3,and the right upper lobe bronchus in 2 cases.The tumor size ranged from 0.7 to 6.5 cm.Cough,dyspnea with or without fever were observed in 7 patients.Seven cases had blood in phlegm,and 4 patients showed pulmonary atelectasis.All cases showed negative CK staining and positive SMA staining.Conclusion Bronchial glomus tumor usually lacks clinical manifestations,and is often misdiagnosed as bronchial asthma.It can be classified as solid glomus tumor,ball hemangioma,ball vascular leiomyoma by histopathology.Glomus tumors show positive immunohistochemical stainings for vimentin and smooth muscle actin (SMA),and are usually negative for cytokeratin (CK) and epithelial markers.Clinical differential diagnoses such as sclerosing hemangioma,hemangiopericytoma,carcinoid tumor and epithelial tumor should be considered.

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作者: 张新军 [1] 朱亚玲 [2] 王晓娟 [2] 苗劲柏 [3] 李辉 [3] 童朝晖 [2]
期刊: 《中华结核和呼吸杂志》2014年37卷10期 758-763页 MEDLINEISTICPKUCSCD
栏目名称: 论著
DOI: 10.3760/cma.j.issn.1001-0939.2014.10.014
发布时间: 2014-10-31
基金项目:
北京卫生系统高层卫生技术人才-学科带头人
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