Abstract

Odontogenic keratocyst was first mentioned in the literature by Mikulicz in 1876, and described by Philipsen in 1956. These cysts are developmental odontogenic cysts arising from dental laminate remnants. Generally, these cysts can be seen in the second or third decade of life, in both mandibular and maxillary jaws; but most often they are localized in the mandibular jaw. A 23-year-old male patient who came to our clinic complaining of swelling in the lower jaw and did not have a systemic disease had a radiolucent lesion measuring about 4 * 2 cm in the mandible and lateral tooth area in the maxilla. Enucleation of radiolucent lesions which are localized in maxillary and mandibular jaws was performed. Both lesions were sent to the pathology laboratory for detailed examination. Extraction of the impacted right mandibular canine was made. According to the pathology report, a diagnosis of odontogenic keratocyst was made to both lesions. In this case keratocysts were seen as multiocular, and it was suspected that there could be Gorlin Goltz syndrome. According to the medical genetic report of

Keywords

Gorlin-Goltz; Gorlin Syndrome; Odontogenic Keratocyst; Multilocular Keratocyst

Introduction

Odontogenic keratocyst (OKC) was first mentioned in the literature by Mikulicz in 1876 and described by Philipsen in 1956 [1,2]. Odontogenic Keratocyst is a developmental epithelial cyst that commonly occurs in the jawbones. It accounts for approximately 11% of all cysts observed in the jaw. Generally, these cysts can be seen in the second or third decade of life, on both mandibular and maxillary jaws; but most often they are localized on the mandibular jaw [3]. WASOKC is a benign intraosseous cyst that can occur either as a unilocular or multilocular lesion within the bones [4]. High recurrence rate, distinctive histopathological features, and aggressive biological behaviours are seen as basic principles which differentiate the OKC from other types of cysts [5]. OKC can be seen as a well-defined radiolucent area with smooth and usually cortical borders, larger lesions may appear as multiocular [6]. OKC constitute approximately 10-12% of odontogenic cysts. The aim of surgery is the minimize both recurrence risk and surgical morbidity. Conservative treatments that can be applied in OKC are simple enucleation with or without curettage and marsupialisation [7]. Gorlin-Goltz is characterized by multiple odontogenic keratocysts. About 60-70% of patients have odontogenic keratocyst, which is considered the primary finding of Gorlin-Goltz syndrome [8]. Gorlin-Goltz Syndrome is a rare disease in the population and requires the coexistence of complex clinical findings [9].

Case

A systemically healthy 23-year-old male patient applied to the Oral and Maxillofacial Surgery Clinic of Istanbul Kent University Faculty of Dentistry with a complaint of pain and swelling in the mandible. In the extra-oral examination, slight swelling and pain were observed in the anterior region of the mandible. There was no redness or fistula. There was no lymphadenopathy or any restriction on jaw movements. In the orthopantomography radiography a single-chambered lesion with well-defined boundaries, measuring 5 × 2 × 1 cm, was seen between the teeth from the left mandibular second premolar to the right mandibular first molar (Figure 1). As a result of the vitality test, it was observed that the mandibular left teeth numbered 1-2-3 were non-vital. As a result of the Cone Beam Computed Tomography (CBCT) examination, significant destruction was not observed in the buccal and lingual cortex associated with the lesion. Under local anaesthetics, fine needle aspiration biopsy and incisional biopsy were performed to make a diagnosis of cystic structure which is related to impacted canine. According to the biopsy report; 0.5 cc of grey-coloured aspirate and two pieces of tissue in the form of a wall, measuring 0.8 × 0.7 × 0.6 cm and 0.8 × 0.6 × 0.2 cm, were taken and sent. Histologically examined sections showed a thin wall consisting of fibrocytes, fibroblasts, and collagen fibres. The inner surface of the wall was lined with thin, parakeratotic stratified squamous epithelium consisting of several layers. The cells in the basal layer of the epithelium had hyperchromatic nuclei arranged parallel to each other. The epithelial-connective tissue boundary was smooth. The lumen-facing surface of the epithelium showed undulations. The epithelium was seen to be detached from the fibrotic wall in some areas. In a few areas, the epithelium was hyperplastic with intracytoplasmic birefringent material accumulation. Numerous microcysts (Satellite cysts) and odontogenic epithelial islands were observed in the wall. The fibrotic wall showed focal, dense infiltration of neutrophils, lymphocytes, plasma cells, and histiocytes. Before the surgical operation, root canal treatment was performed on the NonVital incisors in the mandibular left region. Then, under general anaesthesia, the multilocular cysts were enucleated and curetted by opening a mucoperiosteal flap from the mandibular left first premolar to the mandibular right first molar, preserving healthy bone tissue. In the same session, the impacted canine tooth in the mandibular right region was extracted. An excisional biopsy was taken between the left maxillary lateral and left maxillary first premolar by opening a mucoperiosteal flap. The enucleation and curettage of the cyst were performed while preserving healthy bone tissue. During the operation, the cystic tissue piece and impacted canine tooth were removed from the mandible, and the excisional biopsy specimen measuring 3x1.5x1 cm in grey-white colour removed from the maxilla was sent to pathology for examination. After irrigating the surgical areas with saline, the flaps were primarily closed with 3-0 Vicryl suture material. Postoperatively, the patient was prescribed oral antibiotics (Augmentin BID 1g tb), oral and I.M. anti-inflammatory (Dikloron 75mg/3 amp, Arveles 25mg tb), and chlorhexidine mouthwash (Kloroben mouth washing). The patient was discharged post-surgery. No complications were observed post-operation. The patient experienced no issues with functional jaw movements or sensory functions. Contact information was obtained for long-term follow-up. According to the pathology report, the cyst epithelium was seen in focal small areas due to the dense lymphoplasmacytic infiltration rich in polymorphonuclear leukocytes on the inner surface of the cyst wall, and the result was reported as an infected odontogenic keratocyst. Since both pathology reports of the patient were diagnosed with keratocyst, Gorlin-Goltz syndrome was suspected, and the patient was referred to medical genetics. According to the medical genetics report, no clinically significant mutation was detected in the analysed regions (Figures 2-4).

Figure 1: Panaromical radiographic image of the patient.

Figure 2: Pathological report of the cyst which has been taken from the maxilla of the patient.

Figure 3: Pathological report of the cyst which has been taken from the mandible of the patient.

Figure 4: Medical genetic report of the patient.

Discussion

OKC is known for its high recurrence rate and aggressive behaviour. Therefore, the treatment approach should aim to eliminate the pathological presence and reduce the potential for recurrence [10,11]. In many studies, it has been reported that odontogenic keratocysts (OKC) are more commonly seen in the mandible compared to the maxilla, with the most common localization being the posterior mandible [12,13]. There are several differential diagnoses for unilocular odontogenic keratocysts (OKC). When seen in the periapical region, it can be confused with a periapical cyst; when seen around the crown of an unerupted tooth, with a dentigerous cyst; in the inter radicular region, with a lateral periodontal cyst; and when seen in the midline of the maxilla, with a nasopalatine canal cyst [14]. OKC (odontogenic keratocysts) are often associated with unerupted teeth, most commonly with impacted mandibular third molars. A dentigerous cyst encases the crown of an unerupted tooth, whereas OKC can encompass both the crown and the root of the tooth. OKC in the mandible is associated with an unerupted tooth, while OKC in the maxilla is not typically associated with any unerupted tooth. Smaller OKC may be asymptomatic and painless, while larger ones can present with pain, swelling, and fistula formation. Despite significant size in the mandible, only mild swelling and tenderness on palpation were observed. When multiple OKCs are present, suspicion of an underlying syndrome should be considered until proven otherwise. Additionally, patients with multiple OKC should be regularly monitored to evaluate the potential course of other systemic symptoms. Upon discovering multiple OKC in the jaws, investigation into a possible association with Gorlin-Goltz syndrome should be prioritized. In this case, consultation was sought from the medical genetics department, and despite the presence of multiple odontogenic keratocysts in the jaws, no association with Gorlin-Goltz syndrome was identified according to the patient’s report.

Conclusion

Multiple odontogenic keratocysts seen in the jaws can appear independently, as in this case, without being associated with syndromes such as Gorlin-Goltz. These cysts should be thoroughly evaluated by experienced oral surgeons and genetic specialists due to their characteristics that can complicate treatment, and detailed follow-up of the patient should be ensured after appropriate treatment. In this case, correct diagnosis should be made with accurate biopsy and genetic tests, but we believe that longer follow-up of the patient and more studies like this will be more enlightening in determining an effective treatment protocol.

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Article Information

Article Type: CASE REPORT

Citation: Nuhoglu GEK, Nuhoglu C, Ceylan EK, Dayıoglu H, Kaya HM (2024) Odontogenıc Keratocyst: A Case Report. Int J Dent Oral Health 10(3): dx.doi.org/10.16966/2378-7090.425

Copyright: © 2024 Nuhoglu GEK, et al. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.

Publication history: 

Received date: 16 Aug, 2024

Accepted date: 07 Sep, 2024

Published date: 19 Sep, 2024