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Keratocystic Odontogenic Tumour: Reclassification of the Odontogenic Keratocyst from Cyst to Tumour
Jonathan Madras, BSc (Hons), DDS; Henry Lapointe, DDS, PhD, FRCD(C)
Dr. Lapointe Email: firstname.lastname@example.org
The purpose of this paper is to review the features and behaviour of the odontogenic keratocyst (OKC), now officially known as the keratocystic odontogenic tumour (KCOT); to analyze a series of histologically confirmed KCOT cases; and to review and discuss the redesignation of KCOT and the implications for treatment. Based on a literature review, more aggressive treatment — either resection or enucleation supplemented with Carnoy’s solution with or without peripheral ostectomy — results in a lower recurrence rate than enucleation alone or marsupialization. However, the recurrence rate after marsupialization followed by enucleation is not significantly higher than that after aggressive modalities. In a case series of 21 patients (27 KCOTs), recurrence rate was 29%, consistent with published data; all recurrences occurred within 2 years after intervention. The size of most lesions was 0–15 cm2 (average 14 cm2) measured radiographically. WHO’s reclassification of this lesion from cyst to tumour underscores its aggressive nature and should motivate clinicians to manage the disease in a correspondingly aggressive manner. The most effective treatments are enucleation supplemented with Carnoy’s solution, or marsupialization with later cystectomy. Future treatment may involve molecular-based modalities, which may reduce or eliminate the need for aggressive surgical management.
For citation purposes, the electronic version is the definitive version of this article: www.cda-adc.ca/jcda/vol-74/issue-2/165.html
irst described by Philipsen in 1956,1 the odontogenic keratocyst (OKC) is now designated by the World Health Organization (WHO) as a keratocystic odontogenic tumour (KCOT) and is defined as “a benign uni- or multicystic, intraosseous tumour of odontogenic origin, with a characteristic lining of parakeratinized stratified squamous epithelium and potential for aggressive, infiltrative behaviour.”2 WHO “recommends the term keratocystic odontogenic tumour as it better reflects its neoplastic nature.”2 In light
of the reclassification, it is appropriate to review the salient features of this well-known lesion and to consider the implications for treatment. Case Series To assess the impact of treatment modality and lesion size on KCOT recurrence, 21 patient files on 27 histologically confirmed KCOTs were reviewed (Table 1). The 27 KCOTs included 5 recurrences of a lesion treated elsewhere, 16 de novo lesions and 6 recurrences
JCDA • www.cda-adc.ca/jcda • March 2008, Vol. 74, No. 2 •
5 6×4 5×2 Surface area.5 × 1.5 × 3 3 × 1.5 4.5 1 6.5 4×3 2. 74.5 × 2. 2 • .5 years Recurrence at 1 year Recurrence at 2 years 6 years 5 years 2 months 5.25 16.5 years 5 years 3.5 × 1.5 6.5 × 1.5 years 2.5 8 × 4.25 24 10 Treatment Curettage Marsupialization Curettage Curettage Curettage Marsupialization Curettage Curettage Curettage Curettage Resection Curettage Curettage Curettage Curettage Curettage Resection Curettage Curettage Marsupialization Curettage Curettage Curettage Curettage Curettage Curettage Curettage Follow-up period 1 year 1 year Recurrence at 1.75 12 3.5 5.5 × 3 2. Vol.75 19.5 years Recurrence at 8 months 5 years 1 year 6 years Recurrence at 9 months 7 years 1.5 × 3. left maxilla and sinus Right mandibular body and ramus Left posterior maxilla Left posterior maxilla Left mandibular body and ramus Right maxilla 1946-03-03 1949-01-13 1949-03-22 1957-08-28 1957-12-01 1960-07-18 1961-04-04 1966-09-23 1975-12-26 1986-05-06 a Initial presentation due to recurrence of previous tumour.5 × 2 9 × 2.5 45 18 8 21 8.75 20.5 × 1.5 5 22.25 36 12 5.5 years 5.5 years 2 years 2. cm2 24 19. at teeth 41–47 At teeth 44–46 Right mandibular body and ramus Right mandibular body and ramusa Anterior mandible Anterior mandible Left mandibular body and ramusa Left mandibular angle and ramus At tooth 23.5 × 4.5 × 3.25 1 1 1 6.ca/jcda • March 2008.5 6×3 4×2 7×3 2.5 6.5 1×1 4.––– Lapointe ––– Table 1 Description of keratocystic odontogenic tumours and treatment in a series of 21 patients Tumour Patient’s date of birth 1912-08-18 1918-01-13 1921-11-16 1922-07-17 1925-07-22 1925-11-27 1927-07-07 Location Anterior mandible Left mandibular body and ramusa Right posterior maxilla Right posterior maxilla Right mandibular body Left mandibular ramus a Size.cda-adc.75 19.5 years 16 months 18 months 1 year Recurrence at 1.5 10 × 4.5 4×3 3.5 years 2 years Right mandibular body and ramus At teeth 44 and 45 At teeth 44 and 45 At teeth 44 and 45 1929-01-19 1959-09-07 1933-05-15 1936-06-08 Left mandibular coronoid processa Right mandibular ramus Right mandibular body and ramus Right mandibular body.5 1×1 1×1 1×1 4. No. 165a JCDA • www.5 4. cm 8×3 6.
This included complete removal of the right mandible from the condyle to the bone distal to tooth 44. date of surgery: Dec. Most lesions were within the 0–15 cm 2 range and lesions in this range resulted in the greatest number and proportion of recurrences (Fig 3).5 years. date of surgery: Nov. Recurences Number of primary lesions Total lesions 7 6 5 4 3 2 1 0 10–19 20–29 30–39 40–49 50–59 60–69 70–79 80+ 16 14 12 10 8 6 4 2 0 0–15 > 15–30 > 30 Number of lesions KCOT size. Sample Cases Patient 1 (born 1949. Patient 3 (born 1949. The cyst was marsupialized and followed up for 3. Follow-up periods varied from 2 months to 7 years. 7). Figure 2 depicts the percentage of patients who remained free of recurrent KCOTs after the initial intervention at our clinic. Bone fill proceeded normally and there were no recurrences during that period. 17.––– Keratocystic Odontogenic Tumour ––– 20 18 16 14 12 10 8 6 4 2 0 % of disease-free patients Posterior maxilla Anterior maxilla Posterior mandible Anterior mandible 100 90 80 70 60 50 40 30 20 10 0 Number of lesions 1 2 3 4 5 6 KCOT location Figure 1: Keratocystic odontogenic tumour (KCOT) location among patients in our study group. All recurrences of lesions (previously recurrent or new lesions) treated at our clinic were within 2 years. measuring 18 cm 2 radiographically (Fig. in years Figure 4: KCOT distribution by age. No relation was found between age and number of primary lesions among our patient group (Fig. 6). Treatment consisted of enucleation and curettage for 22 of the lesions. 2001) This patient presented with a primary KCOT measuring 19 cm 2 radiographically and involving the left mandibular ramus (Fig. 3 in the anterior mandible and 6 in the posterior maxilla (Fig. Nine months later. 1993) This patient presented with a de novo KCOT of the anterior mandible. 22. date of surgery: Sept. No.cda-adc. years Figure 2: Percentage of disease-free patients over time. cm2 Figure 3: Relation between KCOT size and recurrence. 5). 4). 1999) This patient presented initially with recurrence of a KCOT (treated elsewhere 10 years earlier) of the right mandible. The tumour measured 45 cm 2 radiographically (Fig. 16. The tumour did not recur during the 6-year follow-up period. multilocularity and extent of soft tissue involvement in the lesion. 165b JCDA • www. 2 • . 74. Overall. There were 18 lesions in the posterior mandible. of lesions treated in our clinic. Because of the size.ca/jcda • March 2008. Time since surgery. resection for 2 and marsupialization for 3. Vol. The average surface area of the lesions measured radiographically was 14 cm 2. Patient age. It was treated by curettage. 1). resection was determined to be the most appropriate treatment method. Included are the 5 patients who presented with recurrence of a lesion treated elsewhere. as well as patients whose lesion recurred after initial treatment at our clinic. the recurrence rate was approximately 29%. Patient 2 (born 1925.
2. (b) 9 days.––– Lapointe ––– a b c Figure 5: Partial panoramic radiograph taken (a) pre-operatively and (b) 6 days and (c) 6 years after resection.15 The wide range in reported recurrence rates has been attributed to the variation in follow-up times used by examiners.16 Most JCDA • www. In addition to multiple KCOTs. 2 • . reportedly between 25% and 60%12 (when associated with NBCCS. calcification of the falx cerebri. a b c d Figure 6: Partial panoramic radiograph taken (a) pre-operatively.14 In 1976. Vol. This was curetted and followed up for 7 years.4. 74. and (c) 16 months and (d) 7 years after curettage of the recurring tumour. No. pain and discharge or may be asymptomatic.ca/jcda • March 2008. (c) 3 months and (d) 3.5 They almost always occur within bone. the surgical technique used and the number of cases incorporated into the studies. especially in the posterior body and ramus regions. 3 They occur most commonly in the mandible.cda-adc. (b) Recurrence at 9 months after curettage. recurrence was observed.5 years after marsupialization. multiple epidermoid cysts and medulloblastoma. Brannon5 proposed 3 mechanisms for KCOT recurrence: incomplete removal of the cyst lining.6–11 Patients may present with swelling. NBCCS is also characterized by nevoid basal cell carcinomas. a b c d Figure 7: (a) Pre-operative radiographic appearance of the lesion. KCOTs have a high 165c recurrence rate. especially when the lesion is associated with nevoid basal cell carcinoma syndrome (NBCCS) or Gorlin-Goltz syndrome. Distinctive clinical features include a potential for local destruction and a tendency for multiplicity. growth of a new KCOT from satellite cysts (or odontogenic rests left behind after surgery) and development of a new KCOT in an adjacent area that is interpreted as a recurrence. Clinical Features KCOTs comprise approximately 11% of all cysts of the jaws. frontal bossing. bifid ribs. although a small number of cases of peripheral KCOT have been reported. the recurrence rate is about 82%13).
2 • 165d . Vol.cda-adc. 74.ca/jcda • March 2008. 25 months 13–288 months 13–288 months 13–288 months 13–288 months 13–288 months 7–19 years > 16 months > 16 months 5–17 years 5–17 years 5–17 years 17–58 months 21–59 months 1–21 years 1–10 years 19 months to 10 years 19 months to 10 years ≥ 5 years > 5 years > 5 years > 5 years > 5 years Recurrence rate. No.8–4. % 24 10 35 0 11 29 38 0 50 0 9 0 14 55 18 0 50 0 18 25 23 18 56 60 33 38 14 3 18 0 51 31 0 0 0 el-Hajj and Anneroth Marker and others Pogrel and Jordan 25 23 10 3 26 Maurette and others Morgan and others 17 30 11 11 13 2 3 Brøndum and Jensen Browne 4 27 44 12 72 28 41 5 Forssell and others28 Jensen and others 29 12 13 30 Voorsmit and others Chuong and others31 52 40 22 1 32 Vedtofte and Praetorius Zachariades and others 57 13 1 1 1 33 JCDA • www.––– Keratocystic Odontogenic Tumour ––– Table 2 Review of literature relating treatment to recurrence rate Study Kondell and Wiberg 21 Chow22 Meara and others23 Bataineh and al Qudah16 Stoelinga 20 24 Cysts 29 70 49 31 82 63 16 1 2 3 Enucleation Treatment Enucleation + Carnoy’s + peripheral ostectomy Enucleation Resection Enucleation + Carnoy’s Enucleation Enucleation + cryosurgery Enucleation + surgical bur Enucleation + cryosurgery + surgical bur Resection Marsupialization + enucleation Marsupialization + later cystectomy Decompression then curettage Enucleation Peripheral ostectomy Peripheral ostectomy + Carnoy’s Enucleation + Carnoy’s Resection Decompression + later cystectomy Marsupialization Enucleation Enucleation in 1 piece Enucleation in > 1 piece Marsupialization Enucleation Enucleation + cryotherapy Enucleation Enucleation + Carnoy’s Enucleation Resection Enucleation Enucleation Resection Marsupialization Decompression + enucleation Follow-up period 1–8 years ≥ 5 years 1–15 years 2–8 years 1–25 years > 5 years > 5 years > 5 years > 5 years > 5 years 1–19 years 1.8 years Approx.
Aggressive modalities have generally been recommended for NBCCS cases. Finally. It has been asserted that a conservative approach is applicable not only to all age groups. marsupialization followed by cystectomy is likewise effective. with or without curettage. whereas more conservative methods tend to result in more recurrences (Tables 2 8 and 3). as this treatment does not result in a significantly higher rate of recurrence than enucleation plus Carnoy’s solution. thus. Ahlfors and others35 suggested that “if the OKC were recognized as a true. but large keratocysts near the base of the skull which have invaded soft tissue should be treated by radical excision. the KCOT is locally destructive and highly recurrent. However. a defined follow-up 33 period and a clear description of treatment. to minimize invasiveness and recurrence.18 Some authors advocate a site. 13 If a difference in recurrence rate between 2 modalities of ≥ 15% (arbitrarily chosen) is considered the threshold for clinical signifi0 cance. Conservative treatment is “cyst-oriented” and. enucleation plus Carnoy’s solution. 2 • . First. • Behaviour: As described earlier. the question of modified treatment schedules would be raised. 34 In 1984. Dammer and others19 suggest that “small keratocysts near the alveolar process a maximum of 1 cm in diameter should be treated by simple excision. results in a significantly lower rate of recurrence than enucleation alone. although some have been reported more than 10 years following initial intervention.and size-based approach to KCOT treatment planning. is not significantly better at eliminating recurrences than enucleation plus Carnoy’s solution or marsupialization plus cystectomy. benign cystic epithelial neoplasia. % 30 9 recurrence — have been found in the area where the KCOT was connected with the mucosa. or marsupialization. the most effective treatment option appears to be enucleation of the KCOT and subsequent application of Carnoy’s solution. Several factors form the basis of this decision.––– Lapointe ––– Table 3 Summary of treatment related to recurrence rate Treatment Enucleation Enucleation + Carnoy’s Enucleation + peripheral ostectomy Enucleation + Carnoy’s + peripheral ostectomy Enucleation + cryotherapy Marsupialization Marsupialization + cystectomy Resection Lesions 465 122 11 83 29 18 108 39 Recurrences 141 11 2 7 11 6 14 0 Recurrence rate. but also to patients with NBCCS. which is advocated because KCOTs commonly present in younger patients. as the latter option requires a protracted course of treatment. patient compliance must be considered. Its advantage is preservation of anatomical structures (including teeth). marsupialization as a definitive treatment is associated with a significantly higher recurrence rate than when the KCOT is subsequently enucleated. includes enucleation. the use of cryotherapy with enucleation appears to have no significant effect on the recurrence rate compared with enucleation alone. Second.18 Aggressive treatment addresses the “neoplastic nature” of the KCOT and includes peripheral ostectomy. Vol.” In the years since. large KCOTs and recurrent lesions.cda-adc. Toller suggested that the OKC may best be regarded as a benign neoplasm rather than a conventional cyst based on its clinical behaviour. Alternatively. despite a recurrence rate of 0. based on histologic evidence that clusters of epithelial islands and microcysts — presumably with the potential to cause 165e Recurrence A review of the literature suggests that recurrence rate is relatively low with aggressive 18 treatment.” This is presumably because of the potential for local invasion of the skull base. 74. Articles reviewed were required to meet the following inclusion criteria: histo38 logic diagnosis of OKC. resection. With surgical treatment. Third. • Histopathology: Studies such as that by Ahlfors and others35 show the basal layer of the KCOT budding JCDA • www. published reports have influenced WHO to reclassify the lesion as a tumour.17 These findings emphasize the importance of long-term follow-up as an essential aspect of the KCOT treatment plan. For example.20 recurrences take place within 5–7 years after treatment. removal of the mucosa overlying the lesion has been recommended. KCOT: The Neoplasm In 1967. chemical curettage with Carnoy’s solution or en bloc resection. with or without peripheral ostectomy. a few simple inferences are possible.ca/jcda • March 2008. lesions treated in this manner require several months of at-home irrigation by the patient as well as clinical observation before enucleation. Therefore. which can have catastrophic consequences. No. Common Treatment Modalities Morgan and colleagues17 categorize surgical treatment methods for KCOT as conservative or aggressive.
Forssell and others28 found that lesion size does not affect recurrence rate. In addition. in the case series all recurrences followed enucleation and curettage. WHO’s formal reclassification of it as a tumour underscores the fact that this lesion should not be managed as the simple cyst it was believed to be. in accordance with findings described above under “clinical features. squamous cell carcinomas and transitional cell carcinomas. SHH binding to PTCH releases this inhibition (Fig.” Likewise. it remains prudent to suggest at least 5 years follow-up for KCOTs for reasons stated earlier.8 years) and sample size (10 patients) were inadequate to draw definitive conclusions. a tumour suppressor gene involved in both NBCCS and sporadic KCOTs. PTCH forms a receptor complex with the oncogene SMO (“smoothened”) for the SHH (“sonic hedgehog”) ligand. Thus. our results regarding lesion size and associated recurrence are inconclusive. the literature makes little mention of recurrence of large versus small lesions. the recurrence rate we observed was 29%.13 Our follow-up interval ranged from 2 months to 7 years. 74.” with allelic loss at 9q22. Although all recurrences took place within 2 years post-intervention. This could influence the results. including basal cell carcinomas. Evidence has shown that the pathogenesis of NBCCS and sporadic KCOTs involves a “2-hit mechanism. No. as larger lesions should be inherently more difficult to excise in one piece and. occurs on chromosome 9q22. Figure 9: SHH releases PTCH from SMO. a plant-based steroidal alka165f JCDA • www. with the variability attributed to patient compliance and time since surgery.” Implications and the Future of KCOT Treatment The aggressive nature of the KCOT is universally acknowledged. “by definition a feature of tumorigenic tissue.43 The 2-hit mechanism refers to the process by which a tumour suppressor gene is inactivated.42. the followup period (≤ 4.––– Keratocystic Odontogenic Tumour ––– Figure 8: PTCH prevents the proliferation-inducing effect of SMO. the tumours presented primarily in the posterior mandible.cda-adc.44 The first hit is a mutation in one allele. they note that LOH is. According to Taipale and colleagues. should be more likely to recur. Although one study26 suggests treating with marsupialization alone and showed promising results (0 recurrences). smaller lesions were more often associated with recurrence. The second hit refers to loss of the other allele and is known as “loss of heterozygosity” (LOH). studies have hinted at possible new treatment methods for KCOT. Table 3 shows that an aggressive approach is more likely to reduce the risk of recurrence (and therefore the risk of trauma caused by repeated surgeries). as supported by the literature. this procedure is extreme. PTCH binding to SMO inhibits growth-signal transduction (Fig. the proliferation-stimulating effects of SMO are permitted to predominate. This contradicts our expectations.3-q31 region has been reported for many epithelial tumours. making small lesions appear to recur more often. therefore. Thus.43 Lench and others45 indicate that LOH in the 9q22. although it can be dominantly inherited. confirming earlier observations. has no phenotypic effect. Regarding a relation between treatment modality and recurrence. In our case series. in agreement with earlier research.46 cyclopamine.ca/jcda • March 2008.2 • Genetics: PTCH (“patched”). Vol. unless resection is deemed necessary. In recent years. Although some studies advocate more conservative treatment. this leads to the dysregulation of the oncoproteins cyclin D1 and p53. which. 8). allowing signal transduction. To date. this method of treatment was associated with the lowest rate of recurrence. into connective tissue.41 If normal functioning of PTCH is lost. In KCOTs. In our study. The largest of our lesions was resected and. 9). Despite the fact that resection of the jaw results in the lowest recurrence rate. 2 • . 36–40 Normally. the most appropriate action would be enucleation of the KCOT plus use of Carnoy’s solution or marsupialization followed by enucleation. WHO notes that mitotic figures are frequently found in the suprabasal layers.3-q31. Notably.
He is currently a general practice resident at Mount Sinai Hospital. Horie N. This article has been peer reviewed. Stoelinga PJ. and assistant director of postgraduate studies. 2004. London. preventing transduction. Comparative study of keratocysts. 13. 76(12):2312–5. 10). 4. Cunningham MJ. Morgan TA. Long-term follow-up on keratocysts treated according to a defined protocol. A retrospective clinical and histologic study. 6. el-Hajj G. Jorge J. Eveson JW. 60:963–71. Clausen PP. Bastian HL. Int J Oral Maxillofac Surg 2001. Gorlin RJ. 12. and its recent reclassification by WHO as a neoplasm should further motivate clinicians in this direction. Gorsky M. Chow HT. Contemporary oral and maxillofacial pathology. Part I. Madras is a graduate of the Schulich School of Medicine and Dentistry.cda-adc. Eversole LR. loid. 42(1):54–72. 73(9):1079–81. Nuebler-Moritz M. 128(5):225–31. Odontogenic keratocysts: a follow-up study of 29 cases. Oral Surg Oral Med Oral Pathol 1976. 35(1):46–8. Wysocki GP. 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