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

  

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] 欧俊永,倪坤明,马潞林,王国良,颜野,杨斌,李庚午,宋昊东,陆敏,叶剑飞,张树栋. 肌层浸润性膀胱癌合并中高危前列腺癌患者的预后因素[J]. 北京大学学报(医学版), 2024, 56(4): 582-588.
[2] 刘帅,刘磊,刘茁,张帆,马潞林,田晓军,侯小飞,王国良,赵磊,张树栋. 伴静脉癌栓的肾上腺皮质癌的临床治疗及预后[J]. 北京大学学报(医学版), 2024, 56(4): 624-630.
[3] 虞乐,邓绍晖,张帆,颜野,叶剑飞,张树栋. 具有低度恶性潜能的多房囊性肾肿瘤的临床病理特征及预后[J]. 北京大学学报(医学版), 2024, 56(4): 661-666.
[4] 周泽臻,邓绍晖,颜野,张帆,郝一昌,葛力源,张洪宪,王国良,张树栋. 非转移性T3a肾细胞癌患者3年肿瘤特异性生存期预测[J]. 北京大学学报(医学版), 2024, 56(4): 673-679.
[5] 方杨毅,李强,黄志高,陆敏,洪锴,张树栋. 睾丸鞘膜高分化乳头状间皮肿瘤1例[J]. 北京大学学报(医学版), 2024, 56(4): 741-744.
[6] 曾媛媛,谢云,陈道南,王瑞兰. 脓毒症患者发生正常甲状腺性病态综合征的相关因素[J]. 北京大学学报(医学版), 2024, 56(3): 526-532.
[7] 苏俊琪,王晓颖,孙志强. 舌鳞状细胞癌根治性切除术后患者预后预测列线图的构建与验证[J]. 北京大学学报(医学版), 2024, 56(1): 120-130.
[8] 李建斌,吕梦娜,池强,彭一琳,刘鹏程,吴锐. 干燥综合征患者发生重症新型冠状病毒肺炎的早期预测[J]. 北京大学学报(医学版), 2023, 55(6): 1007-1012.
[9] 刘欢锐,彭祥,李森林,苟欣. 基于HER-2相关基因构建风险模型用于膀胱癌生存预后评估[J]. 北京大学学报(医学版), 2023, 55(5): 793-801.
[10] 薛子璇,唐世英,邱敏,刘承,田晓军,陆敏,董靖晗,马潞林,张树栋. 青年肾肿瘤伴瘤栓的临床病理特征及预后分析[J]. 北京大学学报(医学版), 2023, 55(5): 802-811.
[11] 卢汉,张建运,杨榕,徐乐,李庆祥,郭玉兴,郭传瑸. 下颌牙龈鳞状细胞癌患者预后的影响因素[J]. 北京大学学报(医学版), 2023, 55(4): 702-707.
[12] 时云飞,王豪杰,刘卫平,米岚,龙孟平,刘雁飞,赖玉梅,周立新,刁新婷,李向红. 血管免疫母细胞性T细胞淋巴瘤临床与分子病理学特征分析[J]. 北京大学学报(医学版), 2023, 55(3): 521-529.
[13] 朱晓娟,张虹,张爽,李东,李鑫,徐玲,李挺. 人表皮生长因子受体2低表达乳腺癌的临床病理学特征及预后[J]. 北京大学学报(医学版), 2023, 55(2): 243-253.
[14] 赖玉梅,李忠武,李欢,吴艳,时云飞,周立新,楼雨彤,崔传亮. 68例肛管直肠黏膜黑色素瘤临床病理特征及预后[J]. 北京大学学报(医学版), 2023, 55(2): 262-269.
[15] 沈棋,刘亿骁,何群. 肾黏液样小管状和梭形细胞癌的临床病理特点及预后[J]. 北京大学学报(医学版), 2023, 55(2): 276-282.
Viewed
Full text


Abstract

Cited

  Shared   
  Discussed   
No Suggested Reading articles found!