The odontogenic keratocyst or keratocystic odontogenic tumour (KCOT) is a benign though locally aggressive tumour with a high recurrence rate. A wide range of treatment options have been reported ranging from enucleation alone, enucleation with adjunctive therapy to resection.
The aim of this review was to compare and rank different surgical treatments for odontogenic keratocsysts (OKC) with respect to recurrence rates.
Methods
The review protocol was registered with PROSPERO. Searches were conducted in the Cochrane Central, Medline, and Embase databases with no language restrictions. Any type of clinical comparative study (pro- or retrospective) with a minimum 10 cases and 1 year follow-up was considered. Two reviewers independently extracted data and assesses risk of bias using the Newcastle-Ottawa Scale (NOS). A network meta-analysis was performed.
Results
- 40 studies (2 prospective,1 ambispective, 34 retrospective and 2 case series.
- Patients age ranged for 10 to 89 years
- 29 of the 40 studies reported on sex with a male female ratio of 1.93:1.
- The mandible (third molar region & ascending ramus) was more commonly involved than the maxilla.
- Follow-up periods ranged from 12 to 252 months.
- 18 studies were considered to be high quality and 22 at moderate to high risk of bias.
Pairwise meta-analysis
- 3 retrospective studies (122 patients) compared MCS to 5FU showing a statistically significant reduction in recurrence rate in favour of 5FU, Odds ratio (OR) = 0.056 (95%CI; 0.010 to 0.314) [very low-quality evidence].
- 2 retrospective studies (157 patients) compared CS with MCS finding a statistically significant reduction in recurrence rate in favour of CS, OR = 0.395 (95%CI; 0.162 to 0.960 [very low-quality evidence].
- 5 retrospective studies (136 patients) compared marsupialisation alone to marsupialisation with PO with no significant difference between the two groups, OR = 1.455 (95%CI; 0.374 to 5.666) [very low-quality evidence].
Network meta-analysis (NMA)
- 40 studies contributed to the NMA the ranking for the most effective surgical treatment in reducing the recurrence rate of OKC is summarised in the table below.
| Treatment | Ranking | No. studies (patients) | Evidence quality |
| E+PO+5FU | 98.1%; | 3 (56) | very low |
| Resection | 83.5%; | 15 (128) | very low |
| E ± PO+CS | 63.8%; | 18 (464) | moderate |
| E + CRYO | 53.9%; | 6 (36) | very low |
| MARS + 2°E ± PO | 48.1%; | 14(206) | very low |
| E+PO/curettage | 36.7%; | 16 (7110 | low |
| E ± PO+MCS | 28.9%; | 6 (158) | very low |
| MARS alone | 25%; | 16 (143) | very low |
| Enucleation alone | 12%; | 24 (1087) | high |
Key
- CS = original Carnoy’s solution MCS = modified Carnoy’s solution (chloroform free)
- CRYO = cryotherapy PO = peripheral ostectomy/curettage
- E =enucleation 5FU = 5-Fluorouracil
- MARS = marsupialization/decompression alone
- MARS + 2°E ± PO = marsupialization with secondary cystectomy/enucleation with/without peripheral ostectomy
Conclusions
The authors concluded: –
the results of this network meta-analysis add a relevant weight of evidence to the concept that 5-fluorouracil appears to be an effective method with promising results that needs further exploration. Enucleation with peripheral ostectomy and Carnoy’s solution, as well as marsupialisation/decompression followed by secondary cystectomy/peripheral ostectomy remain the other previously applied options that can be used as the first line of management. Resection, no doubt successful in reducing the recurrence rate, should not be the preferred initial approach in non-syndromic cases. The efficacy of the use of MCS remains controversial, requiring further in vivo and in vitro studies to determine new protocols.
Comments
We have looked at a number of reviews of treatment for odontogenic keratocsysts (Dental Elf -Odontogenic keratocyst blogs). The most recent (Dental Elf – 1st Feb 2017) was authored by the same lead author of this new review providing an overall pooled recurrence rate 16.6% (95%CI; 12.3 – 22)% with resection having the lowest recurrence rate 8.4% (95%CI; 3.5% – 18.8%) and marsupialisation alone the highest rate of 32.3% (95%CI; 16.9% – 52.8%).
This new review has a protocol registered in PROSPERO, searches a good range of databases and includes 40 studies compared with 35 in the 2017 review. 34 of the included studies (85%) are retrospective and only 18 (45%) were considered to be of good quality. In addition, some of the treatment approaches were used on a small number of patients and there is considerable variation in the length of follow across the included studies. So, while the review provides a small number of direct comparisons and the NMA provides a potential ranking of the treatments in terms of their recurrence rates the data should be interpreted cautiously. High quality, well conducted and reported prospective studies or appropriate size and follow up intervals are needed to clarify the most effective treatment approached for odontogenic keratocsysts.
Links
Primary Paper
Al-Moraissi EA, Kaur A, Gomez RS, Ellis E 3rd. Effectiveness of different treatments for odontogenic keratocyst: a network meta-analysis. Int J Oral Maxillofac Surg. 2022 Sep 20:S0901-5027(22)00379-4. doi: 10.1016/j.ijom.2022.09.004. Epub ahead of print. PMID: 36150944.
https://pubmed.ncbi.nlm.nih.gov/36150944/
Review protocol on PROSPERO
https://www.crd.york.ac.uk/PROSPERO/display_record.php?RecordID=330880
Other references
Dental Elf – 1st Feb 2017
Dental Elf -Odontogenic keratocyst blogs
Picture Credits
Photo by JC Gellidon on Unsplash
