Treatment Options for Keratocyst Odontogenic Tumour (KCOT): a Systematic Review G
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Oral Surgery ISSN 1752-2471 ORIGINAL ARTICLE Treatment options for keratocyst odontogenic tumour (KCOT): a systematic review G. Dias1, T. Marques2 & P. Coelho1 1Oral Surgery Department, School of Dentistry, University of Lisbon, Lisbon, Portugal 2Improvement in Teaching Methods in Conservative Dentistry, School of Dentistry, University of Lisbon, Lisbon, Portugal Key words: Abstract keratocystic odontogenic tumour, odontogenic keratocyst, odontogenic Background: The keratocystic odontogenic tumour (KCOT) is a benign tumours, recurrence, treatment intraosseous odontogenic lesion relatively frequent in the oral cavity. It has a locally aggressive behaviour and exhibits a high propensity to Correspondence to: recur after treatment. All the singular characteristics of this pathology G Dias have originated controversy in the scientific community regarding the Oral Surgery Department most appropriate surgical approaches for the successful treatment of this School of Dentistry University of Lisbon tumour. Rua Duque de Palmela Objectives: To analyse the optimal treatment choice for this tumour, No. 6, loja 11 ensuring high success rates of treatment, preventing future recurrences 1250-098 Lisbon and allowing the maintenance of the patient’s quality of life. Portugal Materials and methods: A search was conducted in Cochrane – 1 result – Tel.: +351213158086 and in PubMed – 756 results. The selection of articles was based on email: goncalosegurodias@gsd-dentalclinics. abstracts and inclusion and exclusion criteria. Three research studies com were considered for the final analysis. Accepted: 12 September 2016 Results: One hundred and nineteen lesions were identified (73 males and 46 females). Twenty-nine tumours were found in the maxilla and doi:10.1111/ors.12250 91 in the mandible; the applied therapeutic methods were: marsupialization/decompression, marsupialization followed by enucleation and adjunctive therapies (peripheral ostectomy and Carnoy’s solution), solely enucleation, enucleation and Carnoy’s solution, enucleation followed by peripheral ostectomy and Carnoy’s solution and resection. Discussion and conclusions: Treatment by enucleation, in combination with adjunctive measures, is associated with minor recurrence rates when compared with enucleation alone. The small number of KCOT that were treated with en bloc resection did not obtain statistically relevant results. Therefore, more studies with well-established criteria are necessary to enable an adequate analysis of recurrence rates associated with each treatment modality. 1–7 Introduction potentially infiltrative behaviour . The KCOT mor- phological code in the International Classification of In 2005, WHO reclassified the intraosseous parakera- Diseases for Oncology (ICD-O) is 9270/01. tinised variant of the odontogenic keratocyst. The This reclassification reflected the necessity to dis- keratocystic odontogenic tumour (KCOT) is actually tinguish this lesion from all other keratinised odon- defined as a benign intraosseous odontogenic togenic cysts due to its particular characteristics: tumour, uni or multicystic, with a regular parakera- aggressive nature, increased epithelial proliferative tinised stratified squamous epithelial lining, and a capacity and propensity to recur after Oral Surgery 10 (2017) 193--209. 193 © 2016 The British Association of Oral Surgeons and John Wiley & Sons Ltd An evidence-based approach for KCOT treatment Dias et al. treatment2,3,5,7–9. A difference in genetic and molec- adjacent bone cavity. It aims to remove tumour ular mechanisms, when compared to other cystic epithelial islands and/or microcysts21,27,28. lesions of the jaws, also suggests a distinct biological Enucleation and Carnoy’s solution consists of a origin of this entity7,10. chemical cauterisation agent with rapid local fixation The KCOT may be associated with Gorlin–Goltz and haemostatic action and a penetration capacity of syndrome patients2,3,5,6. Keratinised lesions, both 1.54 mm in the cystic locus, after enucleation of the orthokeratinised and parakeratinised, peripheral and KCOT5,15,29,30. The original composition of the solu- solid variants, do not form part of a KCOT’s spec- tion consisted of 3 ml of chloroform, 6 ml of abso- trum4,7,11. lute alcohol (95%), 1 ml of glacial acetic acid and 1 g of ferric chloride. Chloroform is currently not a component of this solution, due to the fact of being Treatment modalities a carcinogenic agent5,15,30,31. Carnoy’s solution must A wide variety of surgical approaches are presently be applied to the bone defect for 3 min after the suggested for the treatment of KCOT, ranging tumour’s enucleation, preventing any axonal dam- according to the size, extension and clinical and/or ages and optimising the elimination of any possible – radiographic appearance10,12 14. remaining tumour cells5,26,32. The main purpose of Marsupialization and decompression are consid- the application of this solution is to eliminate possi- ered distinct surgical techniques, despite having the ble viable tumour epithelial cells remaining in the same purpose15. Both methods allow a decrease of bone cavity2,11,15,33–35. the intraluminal lumen15. The majority of authors Enucleation and cryotherapy with liquid nitrogen currently reviews them as inadequate and non-defi- is a surgical method highly recommended by some nitive therapies in KCOT treatment, given the fact authors8,15,27,36–40. Liquid nitrogen presents the abil- that the odontogenic epithelium remains in situ in ity to eliminate the organic component in the lesion the KCOT cavity16, allowing the continuity of locus, keeping the inorganic architecture of the bone epithelial proliferation and enabling future recur- intact8,19,38,39,41. The cryotherapy technique should rences15,17,18. be applied after enucleation of KCOT and consists in Marsupialization was first mentioned by Partsch in the vaporisation of the bone defect with liquid nitro- 1892 and is described as the surgical removal of a gen for 1 min, one or two times, with an interval of wall in the KCOT’s body followed by the suture of 5 min between applications8,15,40. the tumour’s boundaries to the adjacent mucosa. A Enucleation and peripheral ostectomy are similar surgical window that communicates with the oral to a simple enucleation. However, it is followed by cavity is created, allowing for regular irrigation by the removal of 1.5–2 mm of bone with a handpiece – the patient5,8,19 21. Decompression involves any in KCOT margins8,20. method that allows a decrease in intracystic pressure, Enucleation, peripheral ostectomy and Carnoy’s based on the fact that this pressure is responsible for solution consist of the surgical excision of the the KCOT expansion14,15. This surgical approach tumour followed by peripheral ostectomy with a may be conducted through an opening made in the handpiece and posterior application of Carnoy’s solu- lesion cavity. It is then kept in touch with the oral tion20. The combination of adjunctive techniques is cavity through a drain15,22,23. Two-stage surgical pro- used due to the fact that simple enucleation presents cedures (marsupialization and/or decompression pre- high recurrence rates (25–50%), whereby the use of cede tumour enucleation) are the primary indication adjuvant therapies will reduce these recurrences in for these two methods, by reducing volume and size 10%, optimising the treatment8,26,30,34,42–45. in extensive KCOT, enabling the preservation of vital En bloc resection may be realised by two different structures, such as teeth16,24,25. methods: marginal or segmental resection. The surgi- Enucleation or simple enucleation is composed by cal en bloc excision of KCOT is made together with the complete removal of KCOT from the bone cavity the extraction of 1 cm of healthy bone beyond the without any macroscopic remnants of the tumour’s margins15,42. lesion20,21,26. The KCOT excision with or without Marginal resection is based on the surgical bone perforation in one surgical piece is difficult due removal of KCOT, leaving a portion of the non- to the thin and friable epithelial lining12,15,21,27. involved bone with the conservation of its continu- Radical enucleation involves the excision of KCOT ity. Segmental resection involves mandibular or together with the removal of the overlaying mucosa maxilary section removal without maintenance of followed by extensive curettage and reduction of the bone continuity20,21,26. 194 Oral Surgery 10 (2017) 193--209. © 2016 The British Association of Oral Surgeons and John Wiley & Sons Ltd Dias et al. An evidence-based approach for KCOT treatment Materials and methods in the anterior region (between the upper canines) and 20 in the posterior region (between the first Following the PICO model (Problem, Intervention, upper premolar and the third upper molar). Ninety- Comparison, Outcome), the research was structured one lesions were found in the mandible: 8 in the with the aim of answering the following question: anterior region (between the lower canines), 61 in “In the treatment of keratocystic odontogenic the posterior region (between the first lower premo- tumours, what is the best suited therapeutic method lar and the third lower molar) and 48 in the for minimising the short- and long-term recurrence mandibular ramus region (posterior to the third rates associated with this lesion?”. lower molar). The radiographic appearance was only Primary and secondary databases (Cochrane; referred in one study, with the unilocular aspect EMBASE e MEDLINE) were searched with the kera- (sevencases)