唾液腺成涎细胞瘤的临床及病理特征分析（附7例报告）

doi:10.3969/j.issn.1005-4979.2022.04.011

《口腔颌面外科杂志》 ›› 2022, Vol. 32 ›› Issue (4): 257-261. doi: 10.3969/j.issn.1005-4979.2022.04.011

唾液腺成涎细胞瘤的临床及病理特征分析（附7例报告）

吴一凡(), 卢浩, 朱云, 徐万林, 刘胜文(), 杨雯君

上海交通大学医学院附属第九人民医院口腔颌面-头颈肿瘤科，上海交通大学口腔医学院，国家口腔医学中心，国家口腔疾病临床医学研究中心，上海市口腔医学重点实验室，上海 200011

收稿日期:2021-04-25 修回日期:2022-07-08 出版日期:2022-08-28 发布日期:2022-08-29
通讯作者: 刘胜文，主治医师. E-mail: wubenzhimu21@163.com
作者简介:
吴一凡（1996—），男，江苏人，硕士，住院医师. E-mail: 122056213@qq.com
基金资助:
上海市青年科技英才扬帆计划资助(21YF1423500); 上海市青年科技英才扬帆计划资助(20YF1423300); 上海第九人民医院生物样本库项目(YBKB201920); 上海市卫生健康委员会项目（青年）(20204Y0456)

Clinical and pathologic features of sialoblastoma of the saliva gland: A report of 7 cases

WU Yifan(), LU Hao, ZHU Yun, XU Wanlin, LIU Shengwen(), YANG Wenjun

Department of Oral and Maxillofacial-Head and Neck Oncology, Shanghai Ninth People′s Hospital, Shanghai Jiao Tong University School of Medicine, College of Stomatology, Shanghai Jiao Tong University, National Center for Stomatology, National Clinical Research Center for Oral Diseases, Shanghai Key Laboratory of Stomatology, Shanghai 200011, China

Received:2021-04-25 Revised:2022-07-08 Online:2022-08-28 Published:2022-08-29

摘要/Abstract

摘要：

成涎细胞瘤是一种较为罕见的先天性恶性肿瘤，因其临床特征、影像学及病理学表现缺乏特异性，诊断相对困难。目前关于这一唾液腺肿瘤国内外文献报道相对较少，缺乏统一的治疗策略。本文回顾我院2012年1月—2020年12月期间收治的7例成涎细胞瘤，总结国内外相关文献，并结合病例的影像学表现特点、病理学特征、诊断、治疗及其预后进行分析。

关键词: 成涎细胞瘤, 唾液腺肿瘤, 临床特征, 病理特征

Abstract:

Sialoblastoma is a rare congenital malignant tumor, and its diagnosis is rather difficult due to lack of specificity in clinical characteristics, radiological and pathological features. To date, there are relatively few reports on this salivary gland tumor according to literatures in home and abroad, and there is also no standard treatment strategy. This article reports 7 cases of sialoblastoma in our hospital from January 2012 to December 2020, and reviewed relevant literature reports, and analyzed and discussed the radiological characteristics, pathologic features, diagnosis, treatment and prognosis of sialoblastoma of the salivary gland.

Key words: sialoblastoma, salivary gland tumor, clinical feature, pathological feature

中图分类号:

R782
R739.8

吴一凡, 卢浩, 朱云, 徐万林, 刘胜文, 杨雯君. 唾液腺成涎细胞瘤的临床及病理特征分析（附7例报告）[J]. 《口腔颌面外科杂志》, 2022, 32(4): 257-261.

WU Yifan, LU Hao, ZHU Yun, XU Wanlin, LIU Shengwen, YANG Wenjun. Clinical and pathologic features of sialoblastoma of the saliva gland: A report of 7 cases[J]. 《Journal of Oral and Maxillofacial Surgery》, 2022, 32(4): 257-261.

https://journal06.magtech.org.cn/Jweb_joms/CN/Y2022/V32/I4/257

图/表 4

表1 7例成涎细胞瘤病例资料汇总

Table 1 Summary of demographic data of 7 cases of sialoblastoma

编号	性别	年龄/岁	原发部位	病程/月	术式	预后
1	男	5	颌下腺	84	右颌下肿物切除术+右颌下腺摘除术	随访102个月，无复发
2	男	2	颊	24	右颊肿物扩大切除术+右颧弓部分切除术+右喙突切除术+人工补片植入术+碘仿纱条缝线打包	复发2次，患者死亡
3	男	6	腮腺	72	腮腺肿块切除术+面神经解剖术	随访64个月，无复发
4	女	0	颊	4	左颊肿物切除术	随访52个月，复发3次，肺转移
5	男	1	腮腺	7	左腮腺全切术+面神经修复术	随访39个月，肺转移
6	男	3	腮腺	6	腮腺全切术+颞肌瓣转移修复术+咬肌部分切除术+ 面神经切除术	随访6个月，无复发
7	女	9	颊	108	颊部恶性肿瘤局部扩大切除术+邻近瓣转移修复术	随访5个月，无复发

图1 病例2的MRI图像 A. 横断位示右侧上颌翼肌巨大软组织肿块影，边界不清；B. 冠状位示肿瘤累及整个右上颌窦腔及鼻腔。黄色箭头所示为肿瘤。

Figure 1 The MRI images of case No. 2

图2 病例3的 CT图像 A. 横断位；B. 冠状位。提示右侧腮腺肿大，呈弥漫性、结节样改变，局部可见软组织肿块影。黄色箭头所示为肿瘤。

Figure 2 The CT images of case No. 3

图3 病例1的HE染色镜下表现 A、B. 肿瘤组织由腺上皮和基底样细胞组成，呈巢状排列；C、D. 侵犯脂肪；E、F. 累及邻近神经组织。图3A、C、E放大倍数为100；图3B、D、F放大倍数为200。

Figure 3 Histologic features of case No.1

参考文献 31

[1]	Vawter GF, Tefft M. Congenital tumors of the parotid gland[J]. Arch Pathol, 1966, 82(3): 242-245.
[2]	Taylor GP. Congenital epithelial tumor of the parotid-sialoblastoma[J]. Pediatr Pathol, 1988, 8(4): 447-452. doi: 10.3109/15513818809041583 URL
[3]	Choudhary K, Panda S, Beena VT, et al. Sialoblastoma: A literature review from 1966-2011[J]. Natl J Maxillofac Surg, 2013, 4(1): 13-18. doi: 10.4103/0975-5950.117821 URL
[4]	Wang Q, Ma SN, Chen HH, et al. Sialoblastoma in chin and management of treatment[J]. Int J Pediatr Otorhinolaryngol, 2018, 109: 168-173. doi: 10.1016/j.ijporl.2018.03.023 URL
[5]	di Micco R, Prüfer F, Bruder E, et al. Sialoblastoma of the submandibular gland: A distinct entity?[J]. Eur J Pediatr, 2019, 178(8): 1301-1304. doi: 10.1007/s00431-019-03411-x pmid: 31257547
[6]	Ellis GL. What′s new in the AFIP fascicle on salivary gland tumors: A few highlights from the 4th Series Atlas[J]. Head Neck Pathol, 2009, 3(3): 225-230. doi: 10.1007/s12105-009-0128-z pmid: 20596976
[7]	Patil DT, Chou PM. Sialoblastoma: Utility of Ki-67 and p53 as a prognostic tool and review of literature[J]. Pediatr Dev Pathol, 2010, 13(1): 32-38. doi: 10.2350/09-05-0650-OA.1 URL
[8]	Prigent M, Teissier N, Peuchmaur M, et al. Sialoblastoma of salivary glands in children: Chemotherapy should be discussed as an alternative to mutilating surgery[J]. Int J Pediatr Otorhinolaryngol, 2010, 74(8): 942-945. doi: 10.1016/j.ijporl.2010.01.026 URL
[9]	Parag P. Congenital sialoblastoma of the accessory parotid gland[J]. S Afr J Radiol, 2012, 16(4): 134-135. doi: 10.4102/sajr.v16i4.258 URL
[10]	Sitthichaiyakul P, Somran J, Oilmungmool N, et al. Erratum: Sialoblastoma of the cheek: A case report and review of the literature[J]. Mol Clin Oncol, 2016, 5(6): 853. doi: 10.3892/mco.2016.1060 pmid: 28101362
[11]	Marucci DD, Lawson K, Harper J, et al. Sialoblastoma arising in ectopic salivary gland tissue[J]. J Plast Reconstr Aesthetic Surg, 2009, 62(8): e241-e246. doi: 10.1016/j.bjps.2007.09.047 URL
[12]	Saffari Y, Blei F, Warren SM, et al. Congenital minor salivary gland sialoblastoma: A case report and review of the literature[J]. Fetal Pediatr Pathol, 2011, 30(1): 32-39. doi: 10.3109/15513815.2010.502961 pmid: 21204664
[13]	Shet T, Ramadwar M, Sharma S, et al. An eyelid sialoblastoma-like tumor with a sarcomatoid myoepithelial component[J]. Pediatr Dev Pathol, 2007, 10(4): 309-314. doi: 10.2350/06-07-0135.1 URL
[14]	张银苹, 马艳英, 高歌, 等. 左颌下腺多发成涎细胞瘤1例并文献复习[J]. 临床与实验病理学杂志, 2020, 36(11): 1336-1338.
[15]	Alvarez-Mendoza A, Calderon-Elvir C, Carrasco-Daza D. Diagnostic and therapeutic approach to sialoblastoma: Report of a case[J]. J Pediatr Surg, 1999, 34(12): 1875-1877. pmid: 10626880
[16]	Simpson PR, Rutledge JC, Schaefer SD, et al. Congenital hybrid basal cell adenoma—adenoid cystic carcinoma of the salivary gland[J]. Pediatr Pathol, 1986, 6(2-3): 199-208. doi: 10.3109/15513818609037712 URL
[17]	Batsakis JG, MacKay B, Ryka AF, et al. Perinatal salivary gland tumours (embryomas)[J]. J Laryngol Otol, 1988, 102(11): 1007-1011. pmid: 3062115
[18]	Irace AL, Adil EA, Archer NM, et al. Pediatric sialoblastoma: Evaluation and management[J]. Int J Pediatr Otorhinolaryngol, 2016, 87: 44-49. doi: 10.1016/j.ijporl.2016.04.037 URL
[19]	Scott JX, Krishnan S, Bourne AJ, et al. Treatment of metastatic sialoblastoma with chemotherapy and surgery[J]. Pediatr Blood Cancer, 2008, 50(1): 134-137. pmid: 16514617
[20]	Green RS, Tunkel DE, Small D, et al. Sialoblastoma: Association with cutaneous hamartoma (organoid nevus)?[J]. Pediatr Dev Pathol, 2000, 3(5): 504-505. doi: 10.1007/s100240010099 URL
[21]	Siddiqi SH, Solomon MP, Haller JO. Sialoblastoma and hepatoblastoma in a neonate[J]. Pediatr Radiol, 2000, 30(5): 349-351. pmid: 10836603
[22]	Stones DK, Jansen JC, Griessel D. Sialoblastoma and hepatoblastoma in a newborn infant[J]. Pediatr Blood Cancer, 2009, 52(7): 883-885. doi: 10.1002/pbc.21945 pmid: 19156857
[23]	Cheng YKY, Chu WCW, Law LW, et al. A fetus with a huge neck mass and a large abdominal circumference—a rare case of sialoblastoma and hepatoblastoma[J]. Prenat Diagn, 2012, 32(9): 915-917. doi: 10.1002/pd.v32.9 URL
[24]	Fuchsmann C, Viremouneix L, Collardeau-Frachon S, et al. Management and treatment of a sialoblastoma of the submandibular gland in a neonate: Report of one case[J]. Int J Pediatr Otorhinolaryngol Extra, 2011, 6(4): 168-171. doi: 10.1016/j.pedex.2010.07.001 URL
[25]	Eken M, Altin G, Aydin S, et al. Sialoblastoma of the parotid gland[J]. Int J Pediatr Otorhinolaryngol Extra, 2010, 5(2): 63-65. doi: 10.1016/j.pedex.2009.02.003 URL
[26]	Sangwaiya A, Munghate A, Sharma J. Sialoblastoma of parotid gland: A rare case report and review of literature[J]. Clin Cancer Investig J, 2014, 3(5): 423. doi: 10.4103/2278-0513.138072 URL
[27]	Kataria SP, Kumar S, Singh G, et al. Sialoblastoma. diagnosis by FNAC: A case report[J]. Diagn Cytopathol, 2015, 43(11): 924-927. doi: 10.1002/dc.23324 pmid: 26214398
[28]	Hamdan A, Sichel JY, Kharenko O, et al. Sialoblastoma: A very rare cervical mass in neonates: A case report[J]. Int J Pediatr Otorhinolaryngol Extra, 2016, 13: 19-22. doi: 10.1016/j.pedex.2016.02.006 URL
[29]	Batsakis JG, Frankenthaler R. Embryoma (sialoblastoma) of salivary glands[J]. Ann Otol Rhinol Laryngol, 1992, 101(11): 958-960. doi: 10.1177/000348949210101115 URL
[30]	穆红, 盖俊芳, 吕翔. 涎腺成涎细胞瘤的临床病理学特点及鉴别诊断[J]. 诊断病理学杂志, 2013, 20(8): 504-505, 507.
[31]	Brandwein M, Said-Al-Naief N, Said Al-Naeif N, et al. Sialoblastoma: Clinicopathological/immunohistochemical study[J]. Am J Surg Pathol, 1999, 23(3): 342-348. pmid: 10078927

选择文件类型/文献管理软件名称

选择包含的内容

唾液腺成涎细胞瘤的临床及病理特征分析（附7例报告）

Clinical and pathologic features of sialoblastoma of the saliva gland: A report of 7 cases

RichHTML

PDF

可视化

被引次数

摘要/Abstract

引用本文

使用本文

图/表 4

参考文献 31

相关文章 2

编辑推荐

Metrics

本文评价

[1]	于天平, 王荣. 下颌第三磨牙位置与急性冠周炎发生的相关性研究[J]. 《口腔颌面外科杂志》, 2023, 33(3): 183-186.
[2]	马于斐1，徐珵珵2，赵志国1. 下唇黏液表皮样癌病例报道及文献复习[J]. 《口腔颌面外科杂志》, 2018, 28(4): 233-237.

模态框（Modal）标题

选择文件类型/文献管理软件名称

选择包含的内容

唾液腺成涎细胞瘤的临床及病理特征分析（附7例报告）

Clinical and pathologic features of sialoblastoma of the saliva gland: A report of 7 cases

RichHTML

PDF

可视化

被引次数

摘要/Abstract

引用本文

使用本文

图/表 4

参考文献 31

相关文章 2

编辑推荐

Metrics

本文评价