Keratocystic odontogenic tumors (KOT) are locally aggressive tumors of odontogenic origin that were formally known as odontogenic keratocysts (OKC). They have a high recurrence and a range of treatment approaches have used and their treatment remains controversial.

The aim of this review was to compare the recurrence rate of KOTs in patients who underwent enucleation with or without adjuvant therapies to patients who underwent decompression with or without residual cystectomy.

Methods

Searches were conducted in the PubMed/Medline, and Cochrane Central databases. Prospective randomised controlled clinical trials (RCTs), controlled clinical studies (CCS) either prospective or retrospective, retrospective reviews, and case series comparing enucleation of non-syndromic, para-keratinised odontogenic tumors (virgin or recurrent) with or without adjuvant therapy to decompression with or without secondary cystectomy with regard to recurrence rate with an adequate follow-up period.

Two reviewers independently selected studies, abstracted data and assessed risk of bias using the Newcastle-Ottawa Scale (NOS). Recurrence rates were pooled and reported as recurrence event rate, odds ratio (OR) and 95% confidence intervals (CI).

Results

• 14 studies (13 retrospective, 1 case series) were included.
• All of the studies were considered to be of high quality scoring 6 or more on the NOS.

	Enucleation with or without adjuvant therapy	Decompression/marsupialization with or without secondary cystectomy
Number of KOTs	843	154
Mean follow-up period	1-25 years	1-25 years
Weighted recurrence rate	14.3% – 22.8%	18% – 38.5%
Overall pooled recurrence rate random	18.2% (95%CI; 14.3%-22.8%)	27.1% (95%CI; 18% -38.5%)

• There was a significant advantage for the enucleation ± adjuvant therapy group in recurrence prevention OR= 0.514 (95%CI; 0.302 – 0.875). That is, the use of enucleation ± adjuvant therapy in the treatment of KOTs decreases the incidence of recurrences by 48.6% compared with decompression ± secondary cystectomy.

Conclusions

The authors concluded: –

initial cystectomy, with or without adjuvant therapy, is associated with the least chance of recurrence. A prospective, randomized, blinded, multicenter study with a long follow-up period to compare various treatments for patients with KOTs in regard to recurrence are strongly recommended.

Comments

This is a topic that generates a significant level of interest and debate and has been the subject of a number of reviews since 2000 (see other references). The current review has only searched two major databases and identified 14 studies. Almost all of these were retrospective with the exception of a single case series. While the studies were assessed for quality, and all considered to be of high quality, retrospective studies are always more likely then prospective studies to suffer from selection bias and missing data. Consequently it is important for this to be taken into consideration when assessing the findings.

A Cochrane review of treatments for KCOT was originally published in 2010 and revisited again in 2015. Unfortunately no RCTs comparing different treatment modalities for KCOT have been carried out. Consequently it is easy to agree with the conclusion of the authors that prospective randomised controlled trials should be conducted to clarify the best treatment approach. These need to be high quality developed in in line with the SPIRIT statement and reported following the CONSORT guideline

Links

Primary paper

Al-Moraissi EA, Pogrel MA, Ellis E 3rd. Enucleation with or without adjuvant therapy versus marsupialization with or without secondary enucleation in the treatment of keratocystic odontogenic tumors: A systematic review and meta-analysis. J Craniomaxillofac Surg. 2016 Sep;44(9):1395-403. doi: 10.1016/j.jcms.2016.05.020. Epub 2016 May 31. PubMed PMID: 27452613.

Other references

Dental Elf -2^nd Jul 2014

Another review finds limited evidence for making treatment recommendations for keratocystic odontogenic tumors

Díaz-Belenguer Á, Sánchez-Torres A, Gay-Escoda C. Role of carnoy’s solution in the treatment of keratocystic odontogenic tumor: A systematic review. Med Oral Patol Oral Cir Bucal. 2016 Jul 31:0. [Epub ahead of print] PubMed PMID: 27475699.

Al-Moraissi EA, Pogrel MA, Ellis E 3rd. Does the Excision of Overlying Oral Mucosa Reduce the Recurrence Rate in the Treatment of the Keratocystic Odontogenic Tumor? A Systematic Review and Meta-Analysis. J Oral Maxillofac Surg. 2016 Apr 7. pii: S0278-2391(16)30033-7. doi: 10.1016/j.joms.2016.03.043. [Epub ahead of print] Review. PubMed PMID: 27134155.

Sharif FN, Oliver R, Sweet C, Sharif MO. Interventions for the treatment of keratocystic odontogenic tumours. Cochrane Database Syst Rev. 2015 Nov 5;(11):CD008464. doi: 10.1002/14651858.CD008464.pub3. Review. PubMed PMID: 26545201.

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