Email Alert | RSS

Journal of Peking University(Health Sciences) ›› 2020, Vol. 52 ›› Issue (1): 35-42. doi: 10.19723/j.issn.1671-167X.2020.01.006

Previous Articles     Next Articles

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

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

CLC Number: 

  • R739.8

Figure 1

A case of nevoid basal cell carcinoma syndrome A, the bifid ribs (asterisk); B, calcification of the falx cerebri; C, multiple OKCs."

Table 1

The clinicopathologic characteristics and follow-up data of OKCs"

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)

Figure 2

The age and gender distribution of OKC patients A, the differential age distribution of sporadic OKC and syndromic OKC; B, the age and gender distribution of sporadic OKC."

Figure 3

The location distribution of sporadic OKC and syndromic OKC patients (the sporadic OKCs accounted in blue ellipses while syndromic OKCs in yellow ellipses, the percentage was in bracket)"

Figure 4

The histological characteristics of OKC A, the typical lining epithelium of OKC (HE ×400); B, daughter cysts (HE ×40); C, solid OKC (HE ×40); D, peripheral OKC (HE ×40)."

Figure 5

Various radiographic appearance of OKC A, B, and C, OKCs in the periapical regions mimic radicular cysts; D and E, typical radiographic features of OKCs (multilocular lesion with root apical absorption); F, multiple lesions of sporadic OKC."

Table 2

The quantities in the Cox regression equation with sporadic OKCs"

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] Fei WANG,Xiang ZHU,Bei HE,Hong ZHU,Ning SHEN. Spontaneous remission of follicular bronchiolitis with nonspecific interstitial pneumonia: A case report and literature review [J]. Journal of Peking University (Health Sciences), 2021, 53(6): 1196-1200.
[2] Wei-bo GAO,Mao-jing SHI,Hai-yan ZHANG,Chun-bo WU,Ji-hong ZHU. Relationship between marked hyperferritinemia and hemophagocytic lymphohistiocytosis [J]. Journal of Peking University (Health Sciences), 2021, 53(5): 921-927.
[3] Mei-xiang ZHANG,Wen-zhi SHI,Jian-xin LIU,Chun-jian WANG,Yan LI,Wei WANG,Bin JIANG. Clinical characteristics and prognosis of MLL-AF6 positive patients with acute myeloid leukemia [J]. Journal of Peking University (Health Sciences), 2021, 53(5): 915-920.
[4] Yan-fang JIANG,Jian WANG,Yong-jian WANG,Jia LIU,Yin PEI,Xiao-peng LIU,Ying-fang AO,Yong MA. Mid-to-long term clinical outcomes and predictors after anterior cruciate ligament revision [J]. Journal of Peking University (Health Sciences), 2021, 53(5): 857-863.
[5] XIAO Ruo-tao,LIU Cheng,XU Chu-xiao,HE Wei,MA Lu-lin. Prognostic value of preoperative platelet parameters in locally advanced renal cell carcinoma [J]. Journal of Peking University (Health Sciences), 2021, 53(4): 647-652.
[6] YU Yan-fei,HE Shi-ming,WU Yu-cai,XIONG Sheng-wei,SHEN Qi,LI Yan-yan,YANG Feng,HE Qun,LI Xue-song. Clinicopathological features and prognosis of fumarate hydratase deficient renal cell carcinoma [J]. Journal of Peking University (Health Sciences), 2021, 53(4): 640-646.
[7] ZHAO Xun,YAN Ye,HUANG Xiao-juan,DONG Jing-han,LIU Zhuo,ZHANG Hong-xian,LIU Cheng,MA Lu-lin. Influence of deep invasive tumor thrombus on the surgical treatment and prognosis of patients with non-metastatic renal cell carcinoma complicated with venous tumor thrombus [J]. Journal of Peking University (Health Sciences), 2021, 53(4): 665-670.
[8] CHEN Huai-an,LIU Shuo,LI Xiu-jun,WANG Zhe,ZHANG Chao,LI Feng-qi,MIAO Wen-long. Clinical value of inflammatory biomarkers in predicting prognosis of patients with ureteral urothelial carcinoma [J]. Journal of Peking University (Health Sciences), 2021, 53(2): 302-307.
[9] Shi-bo LIU,Hui GAO,Yuan-chun FENG,Jing LI,Tong ZHANG,Li WAN,Yan-ying LIU,Sheng-guang LI,Cheng-hua LUO,Xue-wu ZHANG. Clinical features of hydronephrosis induced by retroperitoneal fibrosis: 17 cases reports [J]. Journal of Peking University (Health Sciences), 2020, 52(6): 1069-1074.
[10] Wei-qian CHEN,Xiao-na DAI,Ye YU,Qin WANG,Jun-yu LIANG,Yi-ni KE,Cai-hong YI,Jin LIN. Analysis of clinical features and prognosis in patients with primary Sjögren’s syndrome and autoimmune liver disease [J]. Journal of Peking University (Health Sciences), 2020, 52(5): 886-891.
[11] Ru MA,Xin-bao LI,Feng-cai YAN,Yu-lin LIN,Yan LI. Clinical evaluation of tumor-stroma ratio in pseudomyxoma peritonei from the appendix [J]. Journal of Peking University (Health Sciences), 2020, 52(2): 240-246.
[12] Wen-peng WANG,Jie-fu WANG,Jun HU,Jun-feng WANG,Jia LIU,Da-lu KONG,Jian LI. Clinicopathological features and prognosis of colorectal stromal tumor [J]. Journal of Peking University (Health Sciences), 2020, 52(2): 353-361.
[13] Xiao WANG,Zhao-xing LI,Huan-fang FAN,Li-ying WEI,Xu-jin GUO,Na GUO,Tong WANG. A rare case of cystadenoma in the small intestine [J]. Journal of Peking University (Health Sciences), 2020, 52(2): 382-384.
[14] Yu-qing OUYANG,Lian-fang NI,Xin-min LIU. Prognosis factors analysis of patients with malignant solitary pulmonary nodules [J]. Journal of Peking University(Health Sciences), 2020, 52(1): 158-162.
[15] Jian-jun SUN,Jun YANG,Jing-cheng XIE,Qing CHANG,Chang-cheng MA,Mei ZHENG,Hung-I LIAO,Tao WANG,Xiao-dong CHEN,Yun-feng HAN,Guo-zhong LIN,Tao YU,Jia ZHANG,Yu SI. Comparative clinical study on seldom segment with multiple segment intramedullary primary spinal cord tumors [J]. Journal of Peking University(Health Sciences), 2019, 51(5): 840-850.
Viewed
Full text


Abstract

Cited
  Shared   
  Discussed   
[1] Author. English Title Test[J]. Journal of Peking University(Health Sciences), 2010, 42(1): 1 -10 .
[2] . [J]. Journal of Peking University(Health Sciences), 2009, 41(2): 188 -191 .
[3] . [J]. Journal of Peking University(Health Sciences), 2009, 41(3): 376 -379 .
[4] . [J]. Journal of Peking University(Health Sciences), 2009, 41(4): 459 -462 .
[5] . [J]. Journal of Peking University(Health Sciences), 2010, 42(1): 82 -84 .
[6] . [J]. Journal of Peking University(Health Sciences), 2007, 39(3): 319 -322 .
[7] . [J]. Journal of Peking University(Health Sciences), 2007, 39(3): 333 -336 .
[8] . [J]. Journal of Peking University(Health Sciences), 2007, 39(3): 337 -340 .
[9] . [J]. Journal of Peking University(Health Sciences), 2007, 39(3): 225 -328 .
[10] . [J]. Journal of Peking University(Health Sciences), 2007, 39(4): 346 -350 .
Copyright © Journal of Peking University (Health Sciences), All Rights Reserved.
Powered by Beijing Magtech Co. Ltd