Email Alert | RSS

北京大学学报（医学版） ›› 2020, Vol. 52 ›› Issue (1): 35-42. doi: 10.19723/j.issn.1671-167X.2020.01.006

• 论著 • 上一篇    下一篇

844例牙源性角化囊肿的临床病理学分析

王彦瑾1,谢晓艳2,洪瑛瑛3,白嘉英1,张建运1,△(),李铁军1,△()   

  1. 1. 北京大学口腔医学院·口腔医院, 病理科, 北京　100081
    2. 北京大学口腔医学院·口腔医院,口腔颌面医学影像科, 北京　100081
    3. 北京大学口腔医学院·口腔医院,门诊部　国家口腔疾病临床医学研究中心　口腔数字化医疗技术和材料国家工程实验室　口腔数字医学北京市重点实验室,北京　100081
  • 收稿日期:2019-10-08 出版日期:2020-02-18 发布日期:2020-02-20
  • 通讯作者: 张建运,李铁军 E-mail:jianyunz0509@aliyun.com;litiejun22@vip.sina.com

Clinicopathological analysis of 844 cases of odontogenic keratocysts

Yan-jin WANG1,Xiao-yan XIE2,Ying-ying HONG3,Jia-ying BAI1,Jian-yun ZHANG1,△(),Tie-jun LI1,△()   

  1. 1. Peking University School and Hospital of Stomatology & Department of Oral Pathology, Beijing 100081, China
    2. Peking University School and Hospital of Stomatology & Department of Oral and Maxillofacial Radiology, Beijing 100081, China
    3. First Clinical Division, Peking University School and Hospital of Stomatology & National Clinical Research Center for Oral Diseases & National Engineering Laboratory for Digital and Material Technology of Stomatology & Beijing Key Laboratory of Digital Stomatology, Beijing 100081, China
  • Received:2019-10-08 Online:2020-02-18 Published:2020-02-20
  • Contact: Jian-yun ZHANG,Tie-jun LI E-mail:jianyunz0509@aliyun.com;litiejun22@vip.sina.com

RICH HTML

 36   

PDF (PC)

343

摘要：

目的:分析牙源性角化囊肿(odontogenic keratocyst, OKC)的临床病理特点及预后相关因素,为临床诊治及预后判断提供依据。方法:收集2000—2018年北京大学口腔医院病理诊断为OKC或与其相关疾病的病例844例,分析其临床资料、影像学及病理特点,分别归类为散发型OKC[发生于颌骨内,不伴痣样基底细胞癌综合征(nevoid basal cell carcinoma syndrome, NBCCS)]、综合征相关OKC、实性型OKC以及外周型OKC四组,并分析患者的治疗及随访资料。结果:844例患者中,散发型OKC患者805例(95.4%),综合征相关OKC患者32例(3.8%),实性型OKC患者3例,外周型OKC患者4例。散发型OKC患者平均年龄36.03岁(11~30岁为发病高峰),男女性别比1.27 ∶1,好发于下颌磨牙升支部(56.2%),经影像学检查,其中428例(71.2%)为单房性病损,多房性病损占28.8%。588例散发型OKC患者行刮治术后有随访资料,其中118例复发(20.1%),复发高峰发生于术后1~3年,影像学呈多房表现者易复发(39.0%),开窗术后刮治术(43例)和单纯刮治术(545例)这两种保守治疗的复发率差异无统计学意义(P>0.05)。综合征相关OKC患者的发病年龄(平均20.97岁)小于散发型OKC,易多发(30/32,93.7%), 好发于下颌磨牙升支部(41.7%),综合征相关OKC组织学上囊壁中含子囊者较散发型OKC多见(分别为56.3%和17.9%),且更易复发(13/29,44.9%)。实性型和外周型OKC患者的年龄较大(平均年龄分别为45.00岁和65.75岁),4例外周型OKC摘除后无复发。结论:散发型OKC行刮治术后的复发率为20.1%,影像学表现多房者更易复发,开窗术后刮治术或单纯刮治术的复发率无明显差异,临床上应注意适应证;综合征相关OKC的发病年龄小、更易多发、刮治后易复发;实性型OKC和外周型OKC少见,患者年龄较大。

关键词: 牙源性囊肿, 临床病理, 预后

Abstract:

Objective: To investigate the clinicopathologic features and prognostic factors in odontoge-nic keratocyst (OKC), and to provide new reference for clinic treatment and management of these patients. Methods: Clinicopathological data of 844 cases initially diagnosed as or associated with OKC at Department of Oral Pathology, Peking University Hospital of Stomatology from 2000 to 2018 were collec-ted. The cases were divided into 4 groups: sporadic OKCs (intraosseous, cystic lesion irrelevant to nevoid basal cell carcinoma syndrome), syndromic OKCs, solid OKCs and peripheral OKCs. The patients were follow-up for 6 to 216 months and the factors that might relate to recurrence were analyzed. Results: There were 805 cases (95.4%) of sporadic OKCs, 32 cases (3.8%) of syndromic OKCs, 3 cases of solid OKCs and 4 cases of peripheral OKCs. The main age of sporadic OKCs was 36.03 years with the peak at the second and third decades. Ratio of male and female was 1.27 ∶1. The predilection site was the molar and ramus area of mandibular (56.2%). In the study, 428 cases (71.2%) were unilocular in radiography while 28.8% were multilocular. The recurrent rate of enucleation with the follow-up was 20.1% (118/588) while most of them occurred in 1-3 years after surgery. The recurrent rate of multilocular patients (39.0%) was significantly higher than that of the unilocular. Enucleation after marsu-pialization (43 cases) or enucleation only (545 cases) showed no difference in recurrence (P>0.05). The syndromic OKCs was younger (main 20.97) and preferred to be multiple compared with sporadic OKCs (30/32, 93.7%). The predilection site was also molar and ramus area of mandibular (41.7%). Age and gender distribution of multiple cases had no significant difference with those in sporadic OKCs. More daughter cysts and epithelial islands were seen (56.3% and 17.9%). Furthermore, the recurrent rate was significantly higher than that of the sporadic OKCs (13/29, 44.9%). But there was no evidence of recurrent-related factors. The age of solid and peripheral OKCs, averaged at 45.00 and 65.75 years, were older than others. Four of peripheral OKCs showed no recurrence after enucleation. Conclusion: The recurrence rate of sporadic OKCs after enucleation is 20.1%. The multilocular lesions prefer to be recurrent. There is no significant difference of recurrence with enucleation only or enucleation after marsupialization. Compared with sporadic OKCs, the syndromic patients are younger and easier to be multiple. It tends to be recurrent frequently and rapidly. There are no related factors about recurrence of syndromic patients. The clinicians should considerate comprehensively and make an individual management of therapy and follow-up. Solid and peripheral OKCs are rare and older.

Key words: Odontogenic cysts, Clinicopathology, Prognosis

中图分类号: 

  • R739.8

图1

痣样基底细胞癌综合征1例"

表1

OKC的临床病理及随访资料"

Characteristics Syndromic (n=32) Sporadic (n=805) Solid (n=3) Peripheral (n=4) Total (n=844)
Age/years, n (%)
≤10 6 (18.8) 10 (1.3) 0 0 16 (1.9)
11-20 14 (43.8) 146 (18.1) 0 0 160 (19.0)
21-30 7 (21.8) 213 (26.5) 0 0 220 (26.1)
31-40 0 161 (20.0) 1 (33.3) 0 162 (19.2)
41-50 3 (9.4) 113 (14.0) 2 (67.7) 0 118 (14.0)
51-60 2 (6.2) 70 (8.7) 0 1 (25.0) 73 (8.6)
61-70 0 66 (8.2) 0 1 (25.0) 67 (7.9)
71-80 0 21 (2.6) 0 2 (50.0) 23 (2.7)
>80 0 5 (0.6) 0 0 5 (0.6)
Gender, n (%)
Male 20 (62.5) 450 (55.9) 2 (67.7) 3 (75.0) 475 (56.3)
Female 12 (37.5) 355 (44.1) 1 (33.3) 1 (25.0) 369 (43.7)
Quantity, n (%)
Single 2 (6.3) 791 (98.3) 3 (100) 4 (100.0) 800 (94.8)
Multiple 30 (93.7) 14 (1.7) 0 0 44 (5.2)
Location*, n (%) n=103 n=824 n=930
Maxillary anterior 7 (6.8) 32 (3.9) 0 NA 39 (4.2)
Maxillary premolar 4 (3.9) 48 (5.8) 1 (33.3) NA 53 (5.7)
Maxillary molar 27 (26.2) 127 (15.4) 0 NA 154 (16.6)
Mandibular anterior 7 (6.8) 37 (4.5) 0 NA 44 (4.7)
Mandibular premolar 8 (7.8) 99 (12.0) 0 NA 107 (11.5)
Mandibular molar-ramus 43 (41.7) 463 (56.2) 2 (67.7) NA 508 (54.6)
Cross midline 7 (6.8) 18 (2.2) 0 NA 25 (2.7)
Histological features, n (%)
Inflammation 26 (81.3) 681 (84.6) 2 (67.7) 4 (100.0) 713 (84.5)
Daughter cyst & epithelium island 18 (56.3) 144 (17.9) 1 (33.3) 0 163 (19.4)
Active proliferation 0 19 (2.4) 1 (33.3) 0 19 (2.2)
Orthokeratinized area 0 17 (2.1) 0 0 17 (2.0)
Budding 1 (0.3) 35 (4.3) 0 0 36 (4.3)
Radiographic features, n (%) n=80 n=601 n=2 n=684
Root absorption 40 (50.0) 331 (55.1) 2 (100.0) NA 373 (54.5)
Impacted teeth 31 (38.8) 232 (38.6) 0 NA 263 (38.5)
Unilocular 68 (85.0) 428 (71.2) 0 NA 496 (72.5)
Multilocular 12 (15.0) 173 (28.8) 2 (100.0) NA 188 (27.5)
Treatment, n (%) n=103 n=824 n=934
Enucleation 99 (96.1) 725 (88.0) 2 (67.7) 4 (100.0) 830 (88.8)
Marsupialization 4 (3.8) 63 (7.6) 0 0 67 (7.2)
Osteotomy 0 36 (4.4) 1 (33.3) 0 37 (4.0)
Recurrence#, n (%) n=29 n=613 n=2 n=4 n=663
<1 year 0 0 0 0 0
1-3 years 11 (37.9) 71 (11.5) 1 (50.0) 0 83 (12.7)
4-6 years 2 (6.9) 30 (4.9) 0 0 32 (4.8)
7-9 years 0 13 (2.1) 0 0 13 (2.0)
Over 10 years 0 4 (0.6) 0 0 4 (0.6)

图2

OKC患者的年龄与部位分布"

图3

散发型OKC及综合征相关OKC的发病部位分布(蓝色为散发型OKC,黄色为综合征相关OKC,括号内为百分比)"

图4

OKC的组织病理特点"

图5

OKC的多种影像学表现"

表2

散发型OKC的Cox回归分析"

Items B P Exp (B) 95%CI for Exp (B)
Lower Upper
Step 1
Treatment 0.241 0.608 1.273 0.506 3.205
Unilocular/multilocular 1.099 <0.001 3.001 1.952 4.613
Age -0.038 0.580 0.962 0.840 1.102
Step 2
Unilocular/multilocular 1.100 <0.001 3.005 1.955 4.619
Age -0.044 0.523 0.957 0.837 1.095
Step 3
Unilocular/multilocular 1.103 <0.001 3.015 1.961 4.633
[1] El-Naggar A, Chan J, Grandis JR , et al. WHO classification of head and neck tumors[M]. 4th ed. Lyon, France: IARC, 2017: 235-236.
[2] Stoelinga PJW . Keratocystic odontogenic tumour (KCOT) has again been renamed odontogenic keratocyst (OKC)[J]. Int J Oral Maxillofac Surg, 2019,48(3):415-416.
[3] Li TJ . The odontogenic keratocyst: a cyst, or a cystic neoplasm?[J]. J Dent Res, 2011,90(2):133-142.
[4] Bresler SC, Padwa BL, Granter SR . Nevoid basal cell carcinoma syndrome (Gorlin syndrome)[J]. Head Neck Pathol, 2016,10(2):119-124.
[5] Jawa DS, Sircar K, Somani R , et al. Gorlin-Goltz syndrome[J]. J Oral Maxillofac Pathol, 2009,13(2):89-92.
[6] Kawano K, Okamura K, Kashima K , et al. Solid variant of keratocystic odontogenic tumor of the mandible: report of a case with a clear cell component and review of the literature[J]. Oral Surg Oral Med Oral Pathol Oral Radiol, 2013,116(5):e393-398.
[7] Chi AC, Owings JR Jr, Muller S . Peripheral odontogenic keratocyst: report of two cases and review of the literature[J]. Oral Surg Oral Med Oral Pathol Oral Radiol Endod, 2005,99(1):71-78.
[8] Luo HY, Li TJ . Odontogenic tumors: a study of 1309 cases in a Chinese population[J]. Oral Oncol, 2009,45(8):706-711.
[9] Gorlin RJ . Nevoid basal cell carcinoma syndrome[J]. Dermatol Clin, 1995,13(1):113-125.
[10] Slusarenko da Silva Y, Stoelinga PJW, Naclério-Homem MDG . Recurrence of nonsyndromic odontogenic keratocyst after marsu-pialization and delayed enucleation vs. enucleation alone: a systematic review and meta-analysis[J]. Oral Maxillofac Surg, 2019,23(1):1-11.
[11] Fidele NB, Yueyu Z, Zhao Y , et al. Recurrence of odontogenic keratocysts and possible prognostic factors: Review of 455 patients[J]. Med Oral Patol Oral Cir Bucal, 2019,24(4):e491-e501.
[12] Stoelinga PJ . Long-term follow-up on keratocysts treated according to a defined protocol[J]. Int J Oral Maxillofac Surg, 2001,30(1):14-25.
[13] Cunha JF, Gomes CC, de Mesquita RA , et al. Clinicopathologic features associated with recurrence of the odontogenic keratocyst: a cohort retrospective analysis[J]. Oral Surg Oral Med Oral Pathol Oral Radiol, 2016,121(6):629-635.
[14] 李铁军 . 牙源性角化囊肿的生长与行为[J]. 中华口腔医学杂志, 2000, ( 04):66-68.
[15] Sigua-Rodriguez EA, Goulart DR, Sverzut A , et al. Is surgical treatment based on a 1-step or 2-step protocol effective in managing the odontogenic keratocyst? [J]. J Oral Maxillofac Surg, 2019, 77(6): 1210. e1-1210. e7.
[16] 陶谦, 兰天俊 . 开窗治疗颌骨囊性病变的临床思考与循证[J]. 口腔疾病防治, 2018,26(12):759-765.
[17] Al-Moraissi EA, Dahan AA, Alwadeai MS , et al. What surgical treatment has the lowest recurrence rate following the management of keratocystic odontogenic tumor?: A large systematic review and meta-analysis[J]. J Craniomaxillofac Surg, 2017,45(1):131-144.
[18] Vazquez-Romero MD, Serrera-Figallo ML, Alberdi-Navarro J , et al. Maxillary peripheral keratocystic odontogenic tumor. A clinical case report[J]. J Clin Exp Dent, 2017,9(1):e167-e171.
[1] 王飞,朱翔,贺蓓,朱红,沈宁. 自发缓解的滤泡性细支气管炎伴非特异性间质性肺炎1例报道并文献复习[J]. 北京大学学报（医学版）, 2021, 53(6): 1196-1200.
[2] 高伟波,石茂静,张海燕,吴春波,朱继红. 显著高铁蛋白血症与噬血细胞性淋巴组织细胞增多症的相互关系[J]. 北京大学学报（医学版）, 2021, 53(5): 921-927.
[3] 张梅香,史文芝,刘建新,王春键,李燕,王蔚,江滨. MLL-AF6融合基因阳性急性髓系白血病的临床特征及预后[J]. 北京大学学报（医学版）, 2021, 53(5): 915-920.
[4] 蒋艳芳,王健,王永健,刘佳,裴殷,刘晓鹏,敖英芳,马勇. 前交叉韧带翻修重建术后中长期临床疗效及影响因素[J]. 北京大学学报（医学版）, 2021, 53(5): 857-863.
[5] 肖若陶,刘承,徐楚潇,何为,马潞林. 术前血小板参数与局部进展期肾细胞癌预后[J]. 北京大学学报（医学版）, 2021, 53(4): 647-652.
[6] 于妍斐,何世明,吴宇财,熊盛炜,沈棋,李妍妍,杨风,何群,李学松. 延胡索酸水合酶缺陷型肾细胞癌的临床病理特征及预后[J]. 北京大学学报（医学版）, 2021, 53(4): 640-646.
[7] 赵勋,颜野,黄晓娟,董靖晗,刘茁,张洪宪,刘承,马潞林. 癌栓粘连血管壁对非转移性肾细胞癌合并下腔静脉癌栓患者手术及预后的影响[J]. 北京大学学报（医学版）, 2021, 53(4): 665-670.
[8] 陈怀安,刘硕,李秀君,王哲,张潮,李凤岐,苗文隆. 炎症生物标志物对输尿管尿路上皮癌患者预后预测的临床价值[J]. 北京大学学报（医学版）, 2021, 53(2): 302-307.
[9] 刘世博,高辉,冯元春,李静,张彤,万利,刘燕鹰,李胜光,罗成华,张学武. 腹膜后纤维化致肾盂积水的临床分析:附17例报道[J]. 北京大学学报（医学版）, 2020, 52(6): 1069-1074.
[10] 陈伟钱,戴小娜,余叶,王沁,梁钧昱,柯旖旎,易彩虹,林进. 原发性干燥综合征合并自身免疫性肝病的临床特点及预后分析[J]. 北京大学学报（医学版）, 2020, 52(5): 886-891.
[11] 姜妮,乔国梁,王小利,周心娜,周蕾,宋雨光,赵艳杰,任军. 中性粒细胞与淋巴细胞比例对评估接受过继性细胞免疫治疗的晚期胰腺癌患者预后的临床意义[J]. 北京大学学报（医学版）, 2020, 52(3): 597-602.
[12] 马茹,李鑫宝,闫风彩,林育林,李雁. 肿瘤间质比评估阑尾来源腹膜假黏液瘤的临床价值[J]. 北京大学学报（医学版）, 2020, 52(2): 240-246.
[13] 王文鹏,王捷夫,胡均,王俊锋,刘嘉,孔大陆,李健. 结直肠间质瘤临床病理特征及预后分析[J]. 北京大学学报（医学版）, 2020, 52(2): 353-361.
[14] 王骁,李兆星,范焕芳,魏莉瑛,郭旭瑾,郭娜,王彤. 罕见小肠囊腺瘤1例报道[J]. 北京大学学报（医学版）, 2020, 52(2): 382-384.
[15] 欧阳雨晴,倪莲芳,刘新民. 恶性孤立性肺结节患者预后因素分析[J]. 北京大学学报（医学版）, 2020, 52(1): 158-162.
Viewed
Full text


Abstract

Cited
  Shared   
  Discussed   
[1] 张三. 中文标题测试[J]. 北京大学学报（医学版）, 2010, 42(1): 1 -10 .
[2] 赵磊, 王天龙 . 右心室舒张末期容量监测用于肝移植术中容量管理的临床研究[J]. 北京大学学报（医学版）, 2009, 41(2): 188 -191 .
[3] 万有, , 韩济生, John E. Pintar. 孤啡肽基因敲除小鼠电针镇痛作用增强[J]. 北京大学学报（医学版）, 2009, 41(3): 376 -379 .
[4] 张燕, 韩志慧, 钟延丰, 王盛兰, 李玲玲, 郑丹枫. 骨骼肌活组织检查病理诊断技术的改进及应用[J]. 北京大学学报（医学版）, 2009, 41(4): 459 -462 .
[5] 赵奇, 薛世华, 刘志勇, 吴凌云. 同向施压测定自酸蚀与全酸蚀粘接系统粘接强度[J]. 北京大学学报（医学版）, 2010, 42(1): 82 -84 .
[6] 林红, 王玉凤, 吴野平. 学校生活技能教育对小学三年级学生行为问题影响的对照研究[J]. 北京大学学报（医学版）, 2007, 39(3): 319 -322 .
[7] 丰雷, 程嘉, 王玉凤. 注意缺陷多动障碍儿童的运动协调功能[J]. 北京大学学报（医学版）, 2007, 39(3): 333 -336 .
[8] 李岳玲, 钱秋瑾, 王玉凤. 儿童注意缺陷多动障碍成人期预后及其预测因素[J]. 北京大学学报（医学版）, 2007, 39(3): 337 -340 .
[9] . 书讯[J]. 北京大学学报（医学版）, 2007, 39(3): 225 -328 .
[10] 牟向东, 王广发, 刁小莉, 阙呈立. 肺黏膜相关淋巴组织型边缘区B细胞淋巴瘤一例[J]. 北京大学学报（医学版）, 2007, 39(4): 346 -350 .
ISSN 1671-167X
CN 11-4691/R
主管单位：中华人民共和国教育部
主办单位：北京大学
地址： 北京市海淀区学院路38号 北京大学医学部 《北京大学学报（医学版）》
邮编：100191
电话：010-82801551
Email： xbbjb2@bjmu.edu.cn

《北京大学学报（医学版）》
微信公众号
版权所有 © 2018 《北京大学学报（医学版）》编辑部