case study J. Stomat. Occ. Med. (2013) 6:140–143 DOI 10.1007/s12548-013-0092-9
Single tooth prosthetic rehabilitation of a patient with recurrent keratocystic odontogenic tumor Yusuf Emes · Bora Öncü · Itır S¸ebnem Arpınar · Buket Aybar · Vakur Olgaç · Serhat Yalçın
Received: 26 February 2013 / Accepted: 5 August 2013 / Published online: 29 October 2013 © Springer-Verlag Wien 2013
Abstract Background Keratocystic odontogenic tumor (KCOT) is one of the most common odontogenic neoplasms. There are many kinds of treatment modalities which can be a conservative method, such as enucleation or an aggressive method, such as enucleation followed by cryosurgery and radical surgical tactics with bone resection. In the literature a relatively high recurrence rate reported for KCOT with a range of 5-70 % depending on the method of treatment. Case presentation The aim of this case report is to present the prosthetic rehabilitation of an 18-year-old male patient who had previously been operated on for a KCOT. Enucleation was preferred due to the size of the lesion. Even though a recurrence was observed 1 year postoperatively, a less extensive procedure was sufficient for the treatment. The patient was successfully rehabilitated with an implant after a follow up period of 19 months following the second operation. Conclusion Prosthetic rehabilitation of a single tooth implant after enucleation of KCOT is an optimal treatment option. No temporization was needed due to the patient preferences. After waiting for a 3 month period of healing phase, the gingiva former was fixed. After 1 week an impression was made and the crown was cemented. The restoration was clinically successful in the 3 month, 6 month and 1 year of follow-up.
I. S¸. Arpınar, DDS () · Y. Emes, DDS, PhD · B. Öncü, DDS · B. Aybar, DDS, PhD · S. Yalçın, DDS, PhD Department of Oral and Maxillofacial Surgery, Faculty of Dentistry, Istanbul University, 34104 Istanbul, Turkey e-mail:
[email protected] V. Olgaç, MD Department of Tumor Pathology and Oncological Cytology, Institute of Oncology, Istanbul University, Istanbul, Turkey
Keywords: Keratocystic odontogenic tumor · Recurrence · Dental implants · Single-tooth implant · Prosthetic rehabilitation
Introduction The term “odontogenic keratocyst” was introduced in 1956 by Philipsen [10]. Although being previously defined by the World Health Organization (WHO) as a cyst arising in the teeth-bearing areas of the jaws, its rapid growth potential and the propensity to recur gave rise to a controversy about its true pathological nature and terminology. In 2005 it was defined by the WHO as “keratocystic odontogenic tumor” (KCOT) and was described as a benign unicystic or multicystic intraosseous tumor originating from odontogenic epithelial remnants [3, 8, 17] with a characteristic lining of parakeratinized stratified squamous epithelium and an aggressive behavior with propensity to recur. It can be seen as a solitary lesion or multiple lesions which can be a symptom of inherited naevoid basal cell carcinoma syndrome (NBCCS). The orthokeratinizing variant of this tumor is not classified as a KCOT [17] and KCOT is one of the most common odontogenic neoplasms of the jaw. In a recent analysis, Gaitán-Cepeda et al. [7] pointed out that reclassification of parakeratotic keratocystic odontogenic tumor (P-OKC) into a KCOT significantly changed the frequency distribution of odontogenic tumors making KCOTs the most frequent odontogenic tumor. There are many kinds of treatment modalities which can be a conservative method, such as enucleation, decompression and marsupialization or an aggressive method, such as enucleation followed by cryosurgery, chemical destruction of the residual cavity with Carnoy’s solution and radical surgical tactics with bone resection. A relatively high recurrence rate reported for KCOT with a range of 5–70 % depending on the method of treatment [8]. Considering non-syndromic cases, region classified recurrence is most common in the mandibular molar area [5, 11, 13, 20].
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The aim of this case report is to present the prosthetic rehabilitation of an 18-years-old male patient who had previously been operated on for a KCOT.
Case report The then 18-year-old male patient was initially referred to Istanbul University Faculty of Dentistry Department of Oral and Maxillofacial Surgery clinic for a unilocular radioluscent lesion observed in the panoramic examination between the roots of teeth numbered 36 and 37, both which were found to be vital. There were no systemic diseases. There were no intraoral findings about the lesion which was first thought to have been a cyst. The lesion was enucleated with the patient under local anesthesia and the impacted third molar was removed. The histopathological examination revealed a KCOT (Figs. 1 and 2). In the microscopic examination parakeratinization of the epithelial layer and palisade structures of basal layer cells were seen. Also primitive odontogenic epithelial islands in the cystic membrane could be observed. The patient was started to be followed up from that time onwards. In the 1 year follow-up visit, although the intraoral examination was normal, a recurrence was observed in the form of two separate lesions in the same region and in the extraction site (Fig. 3) in the control radiograph. The patient was operated on again under local anesthesia and the second molar was extracted. The pathology report showed that the lesions were KCOTs again. The control radiographic examinations at 4, 9 and 19 months, which were taken at the time intervals when the patient had the opportunity to attend the faculty, showed no recurrences and 19 months after the second operation the site was rehabilitated with a dental implant (4.1–12 mm, Straumann, Basel, Switzerland) by a standard implant operation. After raising the flap, the bone was prepared with implant drills and the implant was placed in the newly regenerated bone. Then the flaps were attached and the wound was closed with 3–0 silk suture. The patient was instructed not to eat hard food with that region and prescribed augmentin 1000 mg and cataflam tablets two times daily and andorex oral rinse solution three times daily. After the implantation procedure, no temporization was needed in the healing phase as the patient was able to use tooth 36 due to the information of first molars as being the center of chewing in all people. The patient also preferred not to have a temporary crown. After waiting for a 3-month period of healing phase, cover screws of the implant were removed and replaced with the gingiva former. After 1 week an impression was made and the crown was cemented. The crown was prepared to have the same occlusion with the other molars in the mouth. The restoration was clinically successful in the 3 month, 6 month and 1 year of follow up. After 17 months the radiograph showed a 2–3 mm bone resorption around the neck of the implant but it was still accepted to be successful as this minimal resorption was within normal limits.
Fig. 1 Parakeratinization of epithelial layer with palisade structures of basal layer cells (H&EX200)
Fig. 2 Primitive odontogenic epithelial islands in the cystic membrane (H&EX200)
Fig. 3 Radiograph showing the recurrent lesion
Discussion It is known that KCOT has an aggressive behavior and a high recurrence rate which varies between 25–56 % depending on the treatment method used [20]. A wide range of treatment methods are available, such as marsupialization or block resection [8]. Resection is accepted to cause no recurrence at all but it produces
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morbidity and unacceptable esthetic results [18] while conservative methods have the disadvantage of the probable recurrence. Application of Carnoy’s solution in the residual bony defect is a common method used after many lesions, such as unicystic ameloblastoma and KCOT. Its success is due to the penetration into the cancellous bone trabeculae and fixates the possible remaining tumor cells [11]. It should not be used for places near to the inferior alveolar nerve or maxillary sinus to avoid neural damage or sinus wall necrosis [20]. The reformulated Carnoy’s solution without chloroform (ethanol, glacial acetic acid and ferric chloride) is currently generally accepted because exposure to chloroform has been associated with cancer and reproductive toxicity [6]. Cryosurgery is an additive technique used after enucleation like applying Carnoy’s solution in the cavity. Its unique ability to devitalize bone while protecting the inorganic components makes it useful in avoiding recurrences. This technique is also recommended for large mandibular lesions in which the vital structures are nearby and with noncooperative patients [2]. Cryosurgery is better than application of Carnoy’s solution or peripheral osteotomy as it protects the osseous structure and only damages the epithelial remnants and/ or satellite cysts [4, 16, 18]. In the case series of Schmidt and Pogrel [18] 26 patients were treated with cryosurgery and a recurrence rate of 11.5 % was observed. In another study by Morgan et al. [12] a recurrence rate of 18.18 % was found after using peripheral osteoctomy alone and Kolokythas et al. [9] found no recurrences after the patients were treated by the same techniques. Application of Carnoy’s solution after peripheral osteotomy resulted in no recurrences in the same case series of Morgan et al. [12]. These results show that the use of cryotherapy alone after enucleation resulted in a lower recurrence rate than when using Carnoy’s solution or peripheral osteoctomy. It seems that preserving the bony structure and preventing bony necrosis or neuronal damage is only possible with the application of cryotherapy after enucleation. The disadvantage of the cryotherapy technique is the risk of pathological fractures that occurred in a case in the report of Schmidt and Pogrel [18]. There is also a report in the literature about immediately bone grafting the area that is treated with cryosurgery [14]. Peripheral ostectomy is an alternative option when resection can be avoided. A combination of peripheral ostectomy and an application of Carnoy’s solution resulted in no recurrence. These two methods together eliminate the epithelial remnants and this combination seems to be better than radical bone resection in protecting the bone integrity. The disadvantage of marsupialization which is accepted as the least invasive treatment method [16] is that the patient must be cooperative and compliant to regularly irrigate the cyst cavity. Decompression is very similar to marsupialization and is performed by maintaining an open hole in the lesion cavity with an obturator or drain. After the lesion
diminishes it can be enucleated without injuring the anatomical structures that are nearby [15]. In this case enucleation was preferred due to the size of the lesion. Even though a recurrence was observed 1 year postoperatively, a less extensive procedure was sufficient for the treatment. Various factors must be considered in the selection of the treatment modality. These may be size and extent, location, presence of perforation or soft tissue involvement, age of the individual and primary or the recurrence tendency. Tumor dimension is one of the most important ones. Larger tumors require more aggressive approaches so recurrences occur more often in the presence of smaller lesions. For tumors which have not yet perforated the cortical plate conservative treatment is recommended. Tumors that are not restricted to cancellous bone and if the lesion is a recurrence must be treated aggressively [13, 21]. There is no evidence in the literature to determine the exact period of time after KCOT elimination as follow-up. The general idea is that the patient must be followedup for at least 5 years. Long-term follow up is suggested because KCOTs have been shown to have late recurrences [19]. In the case described here the missing tooth was replaced using an implant 18 months later. Further delay of tooth replacement was not considered due to the young age of the patient. The implant was considered as normal due to implant success criteria of Albrektsson et al. [1].
Conclusions Prosthetic rehabilitation of a single-tooth implant after enucleation of a keratocystic odontogenic tumor is an optimal treatment option. Larger studies must be performed for determination of the exact follow-up time after enucleation of keratocystic odontogenic tumors. Acknowledgement The authors thank Dr. Olgaç for interpreting the histopathology slides. Ethical adherence An informed consent was signed by the patient. Conflict of interest The authors declare that there are no actual or potential conflicts of interest in relation to this article.
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