鼻腔鼻窦腺样囊性癌15例临床分析

doi:10.6040/j.issn.1673-3770.0.2018.545

摘要/Abstract

摘要： 目的

探讨鼻腔鼻窦腺样囊性癌的临床特点、诊断、治疗方式及预后。

方法

回顾性分析2008年1至10月南京医科大学第一附属医院收治的15例经病理证实为鼻腔鼻窦腺样囊性癌患者的临床资料，并对该病预后相关因素进行统计学分析。

结果

15例患者首诊症状：鼻塞5例（33％），鼻出血5例（33％），眼胀痛、溢泪3例（20％），局部隆起3例（20％），面部麻木2例（13％），头痛1例（7％），无症状1例（7％）。15例患者均行手术切除，其中4例行鼻内镜手术治疗（26.7％），9例行鼻侧切手术+术后放疗（60％），2例行鼻侧切手术+术后放化疗（13.3％）。术后病理学检查结果显示，高分化（管状型、筛状型）10例（66.7％），低分化（实体型）5例（33.3％）。15例患者均获随访，随访时间6个月～10年。截至2018年10月，15例患者存活8例（53％），死亡7例（47％）；早期患者（Ⅰ、Ⅱ期）4例，死亡1例，晚期患者（Ⅲ、Ⅳ期）11例，死亡6例；总体1年、3年、5年、10年生存率分别为93％、87％、73％、53％。手术模式与生存年限间呈正相关（t=4.406 P=0.003）；年龄、分化程度与生存年限间呈负相关（t分别为-2.390、-6.126， P分别为0.048、＜0.001）；性别、手术方式、临床分期与生存年限无相关性（P分别为0.154、0.262、0.052）。

结论

鼻腔鼻窦腺样囊性癌临床表现多样且无特异性，早期诊断困难。外科手术是治疗鼻腔鼻窦腺样囊性癌的主要治疗手段，鼻内镜手术或鼻内镜辅助开放手术有较好的临床效果。高龄、分化程度与预后相关，对于低分化及晚期肿瘤患者应采取综合治疗模式，并缩短定期复查时间，终身随访。

关键词: 鼻腔鼻窦, 腺样囊性癌, 预后因素分析

Abstract: Objective

To explore the clinical features, diagnosis, treatment, and prognosis of adenoid cystic carcinoma of the nasal cavity and paranasal sinuses.

Method

We performed a retrospective analysis of the clinical data of 15 patients with adenoid cystic carcinoma of the nasal cavity and paranasal sinuses admitted to the First Affiliated Hospital of Nanjing Medical University in the period from January to October 2008. The prognostic factors of the disease were statistically analyzed.

Result

The initial diagnoses in the 15 patients included: nasal congestion in 5 cases (33%), nose bleeding in 5 cases (33%), eye pain and tearing in 3 cases (20%), local uplift in 3 cases (20%), facial numbness in 2 cases (13%), headache in 1 case (7%), and 1 case (7%) without symptoms. All 15 patients underwent surgical resection; 4 underwent endoscopic sinus surgery (26.7%), 9 underwent nasal resection and postoperative radiotherapy (60%), and 2 underwent nasal excision and postoperative radiochemotherapy (13.3%). Postoperative pathological examination revealed highly differentiated (tubular type, sieve type) adenoid cystic carcinoma in 10 cases (66.7%) and poorly differentiated (solid type) in 5 cases (33.3%). All 15 patients were followed up for a period of 6 months to 10 years. As of October 2018, 8 of the 15 patients survived (53%), and 7 had died (47%). Of the 4 patients at an early stage of the disease (I and II), one died, and among the 11 patients that were in the advanced stage of the disease (III, IV), there were 6 deaths. The overall 1-, 3-, 5-, and 10-year survival rates were 93%, 87%, 73%, and 53%, respectively. There was a positive correlation between the surgical mode and the life span (t=4.406， P=0.003), and a negative correlation between age, differentiation, and life span (t= -2.390, -6.126 P=0.048, ＜0.001, respectively). Gender, surgical procedure, clinical stage, and life span were not correlated (P=0.154, P=0.262, P=0.052, respectively).

Conclusion

The clinical manifestations of adenoid cystic carcinoma of the nasal cavity and paranasal sinuses are diverse and non-specific, and early diagnosis is difficult. Surgery is the main treatment for this type of carcinoma. Endoscopic surgery or endoscopic-assisted open surgery has good clinical application value. The age and degree of pathological differentiation are related to the prognosis. For patients with poorly differentiated and advanced tumors, comprehensive treatment mode should be adopted, and regular follow-up time should be shortened for increasing the lifespan.

Key words: Nasal sinus, Adenoid cystic carcinoma, Prognostic factor analysis

中图分类号:

R739.62

冯剑,周涵,宋圣花,赵青,张佳程,刘雅琴,沈宇杰,董伟达. 鼻腔鼻窦腺样囊性癌15例临床分析[J]. 山东大学耳鼻喉眼学报, 2019, 33(5): 87-91.

Jian FENG,Han ZHOU,Shenghua SONG,Qing ZHAO,Jiacheng ZHANG,Yaqin LIU,Yujie SHEN,Weida DONG. Clinical analysis of 15 cases of adenoid cystic carcinoma in the paranasal sinuses and nasal cavity[J]. Journal of Otolaryngology and Ophthalmology of Shandong University, 2019, 33(5): 87-91.

图/表 2

表1

不同指标人群生存情况[例（%）]"

指标	例数	生存	死亡	Fisher/t	P
性别				-	0.619
男	8（53.33）	5(62.50)	3(37.50)
女	7（46.67）	3(42.86)	4(57.14)
年龄（岁, $x ˉ$ $± s ?$ ）	52.73±12.49	48.25±10.22	57.86±13.58	-1.561	0.143
＜60	11（73.33）	8(72.73)	3(27.27)		0.026
≥60	4（26.67）	0(0.00)	4(100.00)
治疗模式				0.434	>0.999
手术	4（26.67）	2(50.00)	2(50.00)
手术联合放疗	9（60.00）	5(55.56)	4(44.44)
手术联合放化疗	2（13.33）	1(50.00)	1(50.00)
临床分期				-	0.569
Ⅰ～Ⅱ	4（26.67）	3(75.00)	1(25.00)
Ⅲ～Ⅳ	11（73.33）	5(45.45)	6(54.55)
手术方式				-	0.077
鼻侧切	11（73.33）	4(36.36)	7(63.64)
鼻内镜	4（26.67）	4(100.00)	0(0.00)
分化程度				-	0.007
高分化	10（66.67）	8(80.00)	2(20.00)
低分化	5（33.33）	0(0.00)	5(100.00)

表1

表2

参考文献 26

1	Martinez-Rodriguez N , Leco-Berrocal I , Rubio-Alonso L , et al . Epidemiology and treatment of adenoid cystic carcinoma of the minor salivary glands: A meta-analytic study[J]. Med Oral, 2011: e884-e889. doi:10.4317/medoral.17200 . doi: 10.4317/medoral.17200
2	Doescher J , Veit JA , Hoffmann TK .The 8th edition of the AJCC cancer staging manual : Updates in otorhinolaryngology, head and neck surgery[J].HNO, 2017,65(12):956-961.
3	汤钊猷．鼻腔鼻窦恶性肿瘤[Ｍ].３版．现代肿瘤学上海：复旦大学出版社，2014：1294-1300．
4	Stell PM . Adenoid cystic carcinoma[J]. Clin Otolaryngol, 1986, 11(4): 267-291. doi:10.1111/j.1365-2273. 1986. tb01928.x . doi: 10.1111/j.1365-2273. 1986. tb01928.x
5	Wardas P , Tymowski M , Piotrowska-Seweryn A , et al . Endoscopic approach to the resection of adenoid cystic carcinoma of paranasal sinuses and nasal cavity: case report and own experience[J]. Eur J Med Res, 2015, 20: 97. doi:10.1186/s40001-015-0189-2 . doi: 10.1186/s40001-015-0189-2
6	Liu W , Chen XH . Adenoid cystic carcinoma of the larynx: a report of six cases with review of the literature[J]. Acta Oto-Laryngologica, 2015, 135(5): 489-493. doi:10.3109/00016489.2014.990583 . doi: 10.3109/00016489.2014.990583
7	Michel G , Joubert M , Delemazure AS , et al . Adenoid cystic carcinoma of the paranasal sinuses: retrospective series and review of the literature[J]. Eur Ann Otorhinolaryngol Head Neck Dis, 2013,130(5):257-262.doi:10.1016/j.anorl.2012.09.010 doi: 10.1016/j.anorl.2012.09.010
8	Coca-Pelaz A , Rodrigo JP , Bradley PJ , et al . Adenoid cystic carcinoma of the head and neck-An update[J]. Oral Oncology, 2015, 51(7): 652-661. doi:10.1016/j.oraloncology. 2015. 04. 005 . doi: 10.1016/j.oraloncology. 2015. 04. 005
9	Ruhlmann V , Poeppel TD , Veit J , et al . Diagnostic accuracy of 18F-FDG PET/CT and MR imaging in patients with adenoid cystic carcinoma[J]. BMC Cancer, 2017,17(1):887. doi: doi:10.1186/s12885-017-3890-4 . doi: doi:10.1186/s12885-017-3890-4
10	Seong SY , Hyun DW , Kim YS , et al . Treatment outcomes of sinonasal adenoid cystic carcinoma: 30 cases from a single institution[J]. J Craniomaxillofac Surg，2014, 42(5): e171-e175. doi:10.1016/j.jcms. 2013. 08.002 . doi: 10.1016/j.jcms. 2013. 08.002
11	Lupinetti AD , Roberts DB , Williams MD , et al . Sinonasal adenoid cystic carcinoma[J]. Cancer, 2007, 110(12): 2726-2731. doi:10.1002/cncr.23096 . doi: 10.1002/cncr.23096
12	Wang HG , Shen CX , Chen F , et al .Clinical features of advanced adenoid cystic carcinoma in the nasal cavity and paranasal sinuses: analysis of 21 cases[J].Nan Fang Yi Ke Da Xue Xue Bao， 2017， 37(6):847-852.
13	Leafstedt SW , Gaeta JF , Sako K , et al . Adenoid cystic carcinoma of major and minor salivary glands[J]. Am J Surg, 1971,122(6):756-762.doi:10.1016/0002-9610(71)90440-5 doi: 10.1016/0002-9610(71)90440-5
14	Bell D , Roberts D , Kies M , et al . Cell type-dependent biomarker expression in adenoid cystic carcinoma[J]. Cancer, 2010, 116(24): 5749-5756. doi:10.1002/cncr. 25541 . doi: 10.1002/cncr. 25541
15	陈琦, 巴云鹏 . 67例鼻腔鼻窦腺样囊性癌的临床特征及预后分析[J]. 临床耳鼻咽喉头颈外科杂志, 2017, 31(22): 1740-1744. doi:10.13201/j.issn.1001-1781.2017.22.009 . doi: 10.13201/j.issn.1001-1781.2017.22.009
	CHEN Qi , BA Yunpeng . The clinical features and prognosis analysis of adenoid cystic carcinoma in nasal cavity and paranasal sinus[J]. Journal of Clinical Otorhinolaryngology Head and Neck Surgery, 2017, 31(22): 1740-1744. doi:10.13201/j.issn.1001-1781.2017.22.009 . doi: 10.13201/j.issn.1001-1781.2017.22.009
16	Feichtinger M , Pau M , Zemann W , et al . Intraoperative control of resection margins in advanced head and neck cancer using a 3D-navigation system based on PET/CT image fusion[J]. Journal of Cranio-Maxillofacial Surgery, 2010, 38(8): 589-594. doi:10.1016/j.jcms. 2010. 02.004 . doi: 10.1016/j.jcms. 2010. 02.004
17	房居高, 何宁 . 鼻内镜技术在鼻腔鼻窦前颅底恶性肿瘤手术中的应用[J]. 山东大学耳鼻喉眼学报, 2017, 31(2): 7-11. doi:10.6040/j.issn.1673-3770.0.2017.085 . doi: 10.6040/j.issn. 1673-3770.0.2017.085
	FANG Jugao , HE Ning . The clinical use of nasal endoscopic surgical technique in treatment of rhinosinusal malignant tumor involving in the anterior skull base[J]. Journal of Otolaryngology and Ophthalmology of Shandong University, 2017, 31(2): 7-11. doi:10.6040/j.issn. 1673-3770.0.2017.085 . doi: 10.6040/j.issn. 1673-3770.0.2017.085
18	祝康, 何莹, 闫静, 等 . 鼻内镜下低温等离子射频手术治疗鼻腔鼻窦良性肿瘤[J]. 山东大学耳鼻喉眼学报, 2015, 29(2): 62-64.doi:10.6040/j.issn.1673-3770.0.2014.337 . doi: 10.6040/j.issn.1673-3770.0.2014.337
	ZHU Kang , HE Ying , YAN Jing , et al . Nasal endoscopy by low-temperature plasma radiofrequency for benign tumor resection[J]. Journal of Otolaryngology and Ophthalmology of Shandong University, 2015, 29(2): 62-64.doi:10.6040/j.issn.1673-3770.0.2014.337 . doi: 10.6040/j.issn.1673-3770.0.2014.337
19	Miglianico L , Eschwege F , Marandas P , et al . Cervico-facial adenoid cystic carcinoma: Study of 102 cases. Influence of radiation therapy[J]. Inter J Radiation Oncol Biol Physics, 1987, 13(5): 673-678. doi:10.1016/0360-3016(87)90284-7 . doi: 10.1016/0360-3016(87)90284-7
20	Amit M , Binenbaum Y , Sharma K , et al . Adenoid cystic carcinoma of the nasal cavity and paranasal sinuses: a meta-analysis[J]. J Neurol Surg B, 2013, 74(3): 118-125. doi:10.1055/s-0033-1347358 . doi: 10.1055/s-0033-1347358
21	Laurie SA , Ho AL , Fury MG , et al . Systemic therapy in the management of metastatic or locally recurrent adenoid cystic carcinoma of the salivary glands: a systematic review[J]. The Lancet Oncology, 2011, 12(8): 815-824. doi:10.1016/s1470-2045(10)70245-x . doi: 10.1016/s1470-2045(10)70245-x
22	Airoldi M , Pedani F , Succo G , et al . Phase II randomized trial comparing vinorelbine versus vinorelbine plus cisplatin in patients with recurrent salivary gland malignancies[J]. Cancer, 2001, 91(3): 541-547.doi:10. 1002/1097-0142(20010201)91:3<541:aid-cncr1032>3.0.co;2-y . doi: 10. 1002/1097-0142(20010201)91:3<541:aid-cncr1032>3.0.co;2-y
23	Roxbury CR , Ishii M , Richmon JD , et al .Endonasal endoscopic surgery in the management of sinonasal and anterior skull base malignancies[J].Head Neck Pathol，2016， 10(1):13-22.doi: 10.1007/s12105-016-0687-8 . doi: 10.1007/s12105-016-0687-8
24	Castello A , Olivari L , Lopci E , et al . Adenoid cystic carcinoma: focus on heavy ion therapy and molecular imaging[J]. Am J Nucl Med Mol Imaging, 2018, 8(1):1-14.
25	Chae YK , Chung SY , Davis AA , et al . Adenoid cystic carcinoma: current therapy and potential therapeutic advances based on genomic profiling[J]. Oncotarget, 2015, 6(35): 37117-37134. doi:10.18632/oncotarget. 5076 . doi: 10.18632/oncotarget. 5076
26	Jakob JA , Kies MS , Glisson BS , et al . Phase II study of gefitinib in patients with advanced salivary gland cancers[J].Head Neck， 2015,37(5):644-649. doi: 10.1002/hed.23647 . doi: 10.1002/hed.23647

多维度评价

Viewed

Full text

Abstract

Cited

Shared

Discussed

指标	B	SE	t	CI	P
(常量)	3.710	0.560	6.628	2.387 ~5.034	0.000
性别	0.351	0.219	1.599	-0.168 ~0.870	0.154
年龄	-0.662	0.277	-2.390	-1.317 ~-0.007	0.048
手术方式	-0.357	0.292	-1.221	-1.047 ~0.334	0.262
临床分期	-0.797	0.341	-2.340	-1.603 ~0.008	0.052
手术模式	1.039	0.236	4.406	0.481 ~1.597	0.003
分化程度	-1.900	0.310	-6.126	-2.633 ~-1.166	0.000

[1]	何健,钱立庭,高劲. 外耳道腺样囊性癌侵犯中耳、颞叶且伴颈部淋巴结转移一例报告[J]. 山东大学耳鼻喉眼学报, 2018, 32(6): 109-111.
[2]	韩阳,杨小健,唐力行,王蓬鹏,张杰,葛文彤. 鼻腔鼻窦青少年型骨化纤维瘤六例临床分析[J]. 山东大学耳鼻喉眼学报, 2018, 32(6): 79-83.
[3]	徐谦,古庆家,樊建刚,李静娴. 鼻腔鼻窦低度恶性肌纤维母细胞肉瘤一例[J]. 山东大学耳鼻喉眼学报, 2018, 32(2): 110-112.
[4]	王梦君综述, 李慧,马士崟审校. 鼻腔鼻窦恶性黑色素瘤的临床诊疗进展[J]. 山东大学耳鼻喉眼学报, 2017, 31(2): 116-120.
[5]	房居高,何宁. 鼻内镜技术在鼻腔鼻窦前颅底恶性肿瘤手术中的应用[J]. 山东大学耳鼻喉眼学报, 2017, 31(2): 7-11.
[6]	胡廷保,梁健刚. 舌根腺样囊性癌大出血救治1例[J]. 山东大学耳鼻喉眼学报, 2016, 30(2): 106-108.
[7]	金鹏1，李春炜2，李莹滢2，赵莉1，王德云2，史丽3. EMP1在鼻腔鼻窦内翻性乳头状瘤中表达及其与上皮化生的相关性[J]. 山东大学耳鼻喉眼学报, 2014, 28(2): 72-75.
[8]	唐智，王继群，李玉兰，陈勇明，涂智峰. 鳞状细胞癌抗原在鼻腔鼻窦鳞癌患者血清中的表达及临床意义[J]. 山东大学耳鼻喉眼学报, 2013, 27(2): 24-25.
[9]	王小婷，时光刚，刘亦青，纪宏志，何明强，李建峰，王海波. 少年和老人鼻腔、鼻窦肿瘤[J]. 山东大学耳鼻喉眼学报, 2011, 25(6): 19-22.
[10]	敬尚林. 鼻内镜手术治疗鼻腔鼻窦真菌病12例[J]. 山东大学耳鼻喉眼学报, 2010, 24(01): 41-42.
[11]	侯东明曹荣萍孟国珍杨军. 儿童鼻腔鼻窦肿瘤在内镜辅助下的诊断及手术治疗[J]. 山东大学耳鼻喉眼学报, 2008, 22(6): 515-516.