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

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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

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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
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