Case Of Odontogenic Keratocyst Research Articles

Alveolar Odontogenic Keratocyst: A Report of Two Cases

An odontogenic keratocyst （OKC） limited to the alveolar bone has never been reported. Herein, we report the first two cases of OKCs located in the mandibular alveolar bone in two adults. Radiographic examination revealed a solitary, mostly well-circumscribed radiolucency between the roots of the first and second premolars in patient 1 and between the roots of the canine and first premolar in patient 2. The histological appearance of the lesions supported the diagnosis of OKC. The rarity of this lesion and its resemblance to the more common peripheral OKC and lateral periodontal cyst may cause a diagnostic dilemma during the preoperative period.

Conservative Management of Odontogenic Keratocyst in a Tertiary Hospital.

Introduction:Odontogenic keratocysts (OKCs) are benign intraosseous odontogenic lesions that have a locally aggressive behavior and exhibit a high recurrence rate after the treatment. The most appropriate surgical approaches for the successful treatment of OKCs remain controversial.Aim:The aim of this study was to evaluate the conservative management of OKCs by enucleation along with peripheral ostectomy and chemical cauterization in terms of recurrence rates after the surgical procedure.Materials and Methods:A retrospective study on 36 cases of OKCs treated at the Oral and Maxillofacial Surgery Department of a tertiary hospital from 2010 to 2017 was done. The demographic, clinical, radiographic, and histologic data were collected for each patient. All cases were surgically treated by enucleation followed by peripheral ostectomy and chemical cauterization using Carnoy's solution. The teeth that were involved in the lesion were extracted. The diagnosis was confirmed with excisional biopsy and histopathology reports.Results:Most of the OKCs were found in the mandible, except three which were present in the maxilla. A significantly higher incidence was seen in males in the age group of 21–30 years. Most of the cases (30 out of 36 cases) were accessed intraorally. Patients were followed up for up to 5 years. Recurrence of the operated OKCs was observed in five cases which were managed by enucleation with peripheral ostectomy and chemical cauterization again with good results.Conclusion:The results suggest that proper enucleation followed by peripheral ostectomy and chemical cauterization using Carnoy's solution may be the best and optimal approach for the management of OKC.

An interesting case of odontogenic keratocyst mimicking traumatic bone cyst

An interesting case of odontogenic keratocyst mimicking traumatic bone cyst

The recurrence of odontogenic keratocysts in pediatric patients is associated with clinical findings of Gorlin-Goltz Syndrome.

Background Odontogenic keratocyst (OKC) is an odontogenic developmental cyst that presents distinct clinical behavior. This lesion has been described as dental cysts with keratinization since the 1930s, however the term "OKC" was established in 1956. This study aims to determine the frequency and features of OKC in children aged 0 to 14 years in an oral pathology service in Brazil. Material and Methods A retrospective study was performed to review cases of OKC in children diagnosed between 1986 and 2017. Clinical data were evaluated from medical records (gender, race, age, anatomical location, treatment, radiographic findings and follow-up). Results Ninety-seven cases of OKC were diagnosed in a 31-year-period in all age groups and 10 were found in children (10.3%). Age ranged from 2 to 14 years (mean age=10.5±3.5), with 8 males and 2 females. The most frequent location was the anterior region of the mandible (n=4). Patients were predominantly asymptomatic. Moreover, in two children, clinical findings of Gorlin-Goltz Syndrome were observed. The most commonly used treatment was enucleation followed by curettage. In all cases of Gorlin-Goltz Syndrome were observed recurrences and occurrence of new keratocysts. Conclusions Although uncommon in pediatric patients, OKC should be considered a differential diagnosis in cases of osteolytic lesions in gnathic bones. Thus, the periodic assessment of children by dentists and pediatricians is essential to get a correct diagnosis and early treatment to avoid greater mutilation of these patients. Key words:Odontogenic cysts, children, odontogenic keratocyst, Gorlin-Goltz Syndrome.

Factors influencing the recurrence of keratocysts: monocentric study

Introduction: The purpose of the study was to retrospectively analyse the recurrence rate of odontogenic keratocysts and to identify eventual features of the lesions that may influence recurrence. Material and methods: This was a retrospective study carried out for a period of 3 years. The medical records of patients treated in our institution were analysed to identify all the cases of odontogenic keratocysts. Results: A total of 16 odontogenic keratocysts were recorded. These lesions were treated with simple enucleation with or without adjuvant Carnoy's solution. The relapse occurred in 4 patients treated with simple enucleation and in none of the patients that underwent enucleation and Carnoy's solution application. The kind of treatment appeared not to influence recurrence rate at statistical analysis. Conclusions: Odontogenic keratocyst is a lesion with a locally aggressive behavior and a high tendency to relapse. This tendency of recurrence may be greater with syndromic presentation of odontogenic keratocyst, with soft tissue involvement, and with teeth proximity to the lesion. The application of Carnoy's solution may be useful to minimize recurrence rate in those odontogenic keratocysts with an aggressive clinical behavior and secondly may be used for all the other lesions treated with simple enucleation that experienced relapse.

The Advent of Studies on Jaw Cysts with Keratinization: A Review of Overlooked Papers on Odontogenic Keratocyst and Orthokeratinized Odontogenic Cyst.

Previous reports of odontogenic keratocyst (OKC) and orthokeratinized odontogenic cyst (OOC) with coverage of the old literature have tended to overlook the underlying theme of the first description. From a historical viewpoint, a French paper "kyste butyreux du sinus maxillaire simulant un cancer encéphaloid" published in 1855 by Maisonneuve popularized the notion of so-called "buttery cyst", which ultimately became known as jaw cyst with a keratinized lining. Soon after in 1856, Nélaton presented a case of OKC at the Anatomical Society of Paris, but his brief communication provided little information about its histopathology. It was Mikulicz who conducted, in 1876, a pioneering descriptive pathological study of OKC. In 1886, 10years after Mikulicz's German report, OOC was first described in detail by Jeannel of Toulouse. The mid to late decades of the nineteenth century saw notable European contributions on the topic.

COMPARISON OF CELLULAR PROLIFERATION AND MELANOCYTES IN NON-SYNDROMIC AND SYNDROMIC ODONTOGENIC KERATOCYSTS

Objective To compare cell proliferation and the presence of melanocytes in non-syndromic and syndromic odontogenic keratocyst (OK). Study Design We studied 20 cases of sporadic OK and 10 cases of OK associated with NBCC. To evaluate cell proliferation and the presence of melanocytes, immunohistochemistry was performed with antibodies against Ki-67 and Melan-A, respectively. Results The OK associated with NBCC presented 23.4% (range 8.6%-36.8%) and the sporadic OK 21.8% (range 7.6%-40.4%) of proliferating cells. This difference was not significant. Cells positive for Melan-A occurred exclusively in 4 of 9 cases of syndromic keratocyst. Conclusions No differences were found in the proliferation between OK associated with NBCC and sporadic. Only in the OK associated with the NBCC, the presence of melanocytes was detected.

Odontogenic keratocysts: a 22-year epidemiological study and case report

Odontogenic Keratocyst (OKC) is a cyst that presents local aggression and a high recurrence; thus, a better understanding of its clinical characteristics and treatment options is required. The aim of this study is to report a case and describe the epidemiology and characteristics of OKCs diagnosed at a referral service for patients with oral lesions. Patients with histopathological diagnosis of OKC were included in the sample over 22 years. The study highlights the demographic and clinical-radiological characteristics, as well as the treatment options. In addition, a case of an extensive OKC in the mandible is reported. 3064 medical records were analyzed and 16 cases of OKC (0.52%) were found, with two recurrences (12.5%). The mean age of the patients was 33.19 years, being the third decade of life more affected (31.25%), with equal prevalence for both sexes. The mandible was more affected (68.75%), mainly its body. The differential diagnosis included the dentigerous cyst (11.54%) and the ameloblastoma (11.54%). The most frequent treatment was the curettage enucleation (50%). In the reported case, enucleation associated with cryotherapy was performed, followed by rehabilitation with xenogen graft and osseointegrated implant. OKC presents a wide spectrum of differential diagnoses due to its non-specific clinical-radiographic characteristics. The case reported shows that treatments such as enucleation may be efficient when accompanied by adjuvant techniques.

CYSTIC AND TUMOR CHARACTERISTICS OF ODONTOGENIC KERATOCYST: CASE REPORT

In 2005, the World Health Organization classified the keratocyst as being a tumor due to its aggressiveness and recurrence. In 2017, it was reclassified as a cyst. Due to this dubious behavior, the present study aims to report a case of odontogenic keratocyst that illustrates its clinical and surgical characteristics. A 51-year-old patient presented with multilocular radiolucent lesions in the body and the right ramus of the mandible. In 2012, marsupialization was the first established approach with conservative surgery after 1 year. After 2 years of follow-up, there was recurrence in the mandibular ramus and surgical curettage was chosen. After another year, there was another recurrence in the mandibular body that was surgically treated again. The patient is healthy after 1 year. The 2 recurrences at different sites of the primary lesion demonstrated aggressive behavior; however, its reduction from marsupialization and the possibility of surgical curettage revealed the cystic nature of odontogenic keratocyst.

The potentialities of the Anatomage Table for head and neck pathology: medical education and informed consent.

The understanding of anatomy of head and neck region and of the relative pathological conditions may be challenging for most medical students. Furthermore, patients may encounter great difficulties to understand their pathology, the proposed surgical technique, as well as the anatomy of this body district. The purpose of the present study was to present and discuss the use of the Anatomage Table for the assessment of several benign and malignant conditions of head and neck district. The hospital database of Novara University Hospital, Novara, Italy, was searched for odontogenic keratocysts, thyroglossal duct cysts, and oral squamous cell carcinomas. A case for each pathology was randomly chosen, and preoperative computed tomography (CT) scans (in DICOM files) were retrieved. The DICOM data of preoperative CT examinations were uploaded in the Anatomage Table device, and an immediate 3-D reconstruction image was obtained from the Anatomage Table. Then, a workstation was used to obtain images of the body surface, "dissection cuts," or "vascular reconstructions," by the variations of the filters. The "dissection," "vascular," and "bony" images of three cases of odontogenic keratocyst, thyroglossal duct cyst, and mandibular oral squamous cell carcinoma were produced and presented. The examination of the different slices and the application of different filters represent a promising tool both for the education of students/residents and for an improved informed consent by the patients.

HIF-1α is Overexpressed in Odontogenic Keratocyst Suggesting Activation of HIF-1α and NOTCH1 Signaling Pathways.

Background: The odontogenic keratocyst (OKC) is an odontogenic cyst that shows aggressive and intriguing biological behavior. It is suggested that a hypoxic environment occurs in OKC, which led us to investigate the immunoexpression and location of hypoxia-inducible factor 1-alpha (HIF-1α) and other hypoxia-related proteins. Methods: Twenty cases of OKC were evaluated for the expression of Notch homolog 1 (NOTCH1), HIF-1α, disintegrin and metalloproteinase domain-containing protein 12 (ADAM-12), and heparin-binding epidermal growth factor-like growth factor (HBEGF) by immunohistochemistry and compared to eight control cases of calcifying odontogenic cystic (COC), orthokeratinized odontogenic cyst (OOC), and normal oral mucosa (OM) in basal and parabasal layers. Results: In OKC, all the proteins tested were expressed significantly higher in both basal (except for NOTCH1 and HBEGF in OOC) and suprabasal epithelial layers compared to controls. Looking at the epithelial layers within OKC, we observed an increased NOTCH1 and HIF-1α expression in parabasal layers. Conclusions: These results suggest that hypoxia occurs more intensively in OKC compared to COC, OM, and OOC. Hypoxia appeared to be stronger in parabasal layers as observed by higher HIF-1α expression in upper cells. Overexpression of NOTCH1, ADAM-12, and HBEGF in OKC was observed, which suggests that microenvironmental hypoxia could potentially regulate the expression of hypoxia-related proteins, and consequently, its clinical and biological behavior.

IMMUNOHISTOCHEMICAL DISTRIBUTION OF THE SIBLINGS IN AMELOBLASTOMA AND ODONTOGENIC KERATOCYST

Objective Ameloblastomas are benign but aggressive neoplasms of the jaw. The odontogenic keratocysts (OKC) are aggressive jaw cysts of odontogenic origin. A recent WHO classification designated OKC as a neoplasm but a current edition re-designated it as a cyst. The small integrin-binding ligand n-linked glycoproteins (SIBLINGs) are a family of molecules that some epithelial neoplasms use in furthering their progression. Members of the SIBLING family are bone sialoprotein (BSP), dentin matrix protein1 (DMP1), dentin sialophosphoprotein (DSPP), matrix extracellular phosphoglycoprotein (MEPE), and osteopontin (OPN). The objective of this study was to compare the expression of the SIBLINGs in ameloblastomas and odontogenic keratocysts. Findings Using immunohistochemistry, with appropriate controls, 49 cases of ameloblastomas and 35 cases of OKCs were screened for the expression of all five SIBLINGs in a retrospective study on archived paraffin sections. Immunoreactivity was scored as positive if > 10% of tumor/cyst cells stained for a SIBLING and negative if Conclusion While all members of the SIBLINGs are expressed in ameloblastomas and OKCs, DSPP expression in OKCs is significantly higher than in ameloblsatomas, whereas, MEPE expression in OKCs is considerably lower than in ameloblastomas. The differences in the expression of DSPP and MEPE between ameloblastomas and OKCs may indicate differences in the degree of aggressive behaviors.

The BRAF p.V600E mutation is a common event in ameloblastomas but is absent in odontogenic keratocysts.

Odontogenic keratocysts (OKCs) are jaw lesions with a tendency to recur. PTCH1 gene mutations are common events in most OKCs; however, other genetic alterations underlying OKC pathogenesis have not yet been elucidated. BRAF p.V600E mutations have recently been detected in some odontogenic tumors, such as ameloblastoma and ameloblastic fibroma, although their involvement in OKC is still unclear. In this study we aimed to clarify the presence and/or frequency of BRAF p.V600E mutations in OKCs. Thirty-five cases of OKCs, 13 of which were associated with Gorlin syndrome, were evaluated for BRAF p.V600E mutations by direct sequencing of the formalin-fixed, paraffin-embedded, and frozen tissue samples. Seventeen cases of ameloblastoma and six cases of dentinogenic ghost cell tumor were also included in this study for comparative purposes. BRAF p.V600E mutations were not detected in any of the OKCs or dentinogenic ghost cell tumors. In contrast, 14 of 17 cases of ameloblastoma (82.35%) were proven to harbor BRAF p.V600E mutations. BRAF p.V600E mutations were common in ameloblastomas, as previously reported, but were absent in OKCs and dentinogenic ghost cell tumors. These results further confirmed the noninvolvement of BRAF in OKCs and suggested different pathogenic mechanisms involved in various odontogenic lesions.

Investigation of Postoperative Complications of Intrabony Cystic Lesions in the Oral and Maxillofacial Region

The purpose of this study was to identify factors that can complicate the surgical removal of intrabony cysts and any relevant correlations between them. The medical records of 249 patients who underwent surgical removal of intrabony cysts were retrospectively reviewed. Outcome variables were postoperative complications, infection, and recurrence. Predictor variables were patient age, gender, comorbidities, anatomic location, pathologic diagnosis, preoperative infection, previous marsupialization, and bone graft methods. Logistic regression analysis was performed to identify risk factors of postoperative infection and recurrence. The cystic lesion was smallest in patients who did not receive a bone graft and increased steadily in those who received a xenogeneic bone graft and an autogenous bone graft, in that order. Paresthesia occurred after enucleation of the cystic lesion in 38 cases. Pathologic fractures were observed in 4 cases. There were 59 postoperative infections. The postoperative infection rate was as high as 63.6% in patients who underwent autogenous bone grafting. In contrast, infection rates were as low as 26.8 and 19.5% in those who underwent xenogeneic bone grafting and no bone grafting, respectively. Location of the cystic lesion in the maxilla or mandible affected the infection rate. When cysts were located in the anterior mandible, no postoperative infection occurred. In contrast, the infection rate was highest for cysts in the mandibular ramus, followed by those in the posterior mandible. Cystic lesion recurrence was observed in 7 cases: 5 cases of odontogenic keratocysts, 1 case of periapical cyst, and 1 case of dentigerous cyst. These findings suggest that cyst pathologic identity affects the recurrence rate. Cyst size was meaningfully correlated with recurrence rate. These results suggest that autogenous bone grafts increase the risk of postoperative infection compared with absence of a bone graft.

Odontogenic keratocyst with granular cell changes: A distinctive finding

Odontogenic keratocyst (OKC) originates from the dental lamina and is more commonly seen in the posterior mandible than in the maxilla. OKC is the most aggressive cyst of the oral cavity and is known for its rapid growth and its tendency to invade bone of the adjacent tissues. The recurrence rate of OKC is very high due to various reasons debated upon. Cases of OKC have shown the presence of calcifications, dentinoid formation and ossification. Here, we report the first case of OKC in a 27-year-old male showing granular cell changes.

Role of cell block technique as a novel diagnostic approach in odontogenic cysts and ameloblastomas of the jaw region.

Background:Fine-needle aspiration cytology (FNAC) involves puncturing a lesion for microscopic analysis. Smears are prepared using aspirated material for cytology and for cell block (CB) preparation. FNAC is a preoperative technique and does not provide sufficient information for precise diagnosis, and the risk of false diagnosis or intermittent diagnosis always exists. To overcome the deficiencies of FNAC, the CB technique (CBT) is considered a novel innovative technique for odontogenic lesions. The present study is carried out to evaluate the efficacy of CBT in diagnosing odontogenic cystic lesions and ameloblastoma of the jaw and comparing it with aspiration cytology and histopathology.Methods:In this prospective study, 17 patients with clinical diagnosis of odontogenic cysts and ameloblastoma are obtained. FNAC smears and CBs are prepared and compared with cytology and histopathology.Results:A total of 17 cases of odontogenic cysts and ameloblastoma are divided into seven odontogenic keratocyst (OKC), three dentigerous cysts, three radicular cysts and four ameloblastoma. In seven cases of OKC, five cases (71.4%) are showing positive features and two cases (28.6%) are showing negative features. Three dentigerous and three radicular cysts are showing three positive CB features (66.7%) and three negative features (33.3%). Among four cases of ameloblastoma, two cases (50%) are showing positive CB features and two cases (50%) are showing negative features.Conclusion:CB is a novel diagnostic technique in oral pathology for odontogenic cysts, tumors, metastatic tumors and bony lesions. Compared to FNAC, it gives accurate diagnostic architecture and apparent histopathological features. This technique can be used alternative to FNAC.

Recurrence of odontogenic keratocysts and possible prognostic factors: Review of 455 patients.

BackgroundTo describe epidemiological features of 565 Chinese patients with odontogenic keratocysts (OKC), to investigate possible prognostic factors related to recurrence, and to analyse features of recurrent OKC (rOKC).Material and MethodsA retrospective chart review of 565 cases of OKC treated between 2003 and 2015 was undertaken. The probability of recurrence related to prognostic factors including large size, cortical perforation combined with involved teeth in the lumen of the cyst, inflammation, sites of the involved lesion, sex, and daughter cyst variables were analysed. The subsequent relapse of each OKC was compared.ResultsPatients ranged in age from 7 to 81 years (mean age, 28.4 years) and, of those affected, 66.9% were male and 33.1% were female. Mandibular OKC occurred in 63.01% and 36.99% occurred in the maxilla, 80.53% of patients had non-rOKC, 10.44% rOKC, and 9.03% had multiple OKC lesions. Enucleation with preservation of the involved teeth in the cystic lesion combined with cortical perforation was statistically associated with high recurrence rate, as were daughter cysts, and multilocular lesions. The number of recurrences and the average time (in years) to relapse decreased from the first relapse of OKC to the third relapse, and the difference was significant (P<.05).ConclusionsPreservation of the involved teeth combined with cortical perforation appeared to be a potential prognostic factor associated with high recurrence. The follow-up evaluation period for rOKC with ≥ 2 previous treatments should be shorter than for first-time rOKC. The decreasing average duration (years postoperatively) to relapse was related to the number of rOKCs, timing of relapse, and rOKC type. Key words:Keratocystic odontogenic tumor, risk of recurrence, involved teeth and cortical perforation, relapse, epidemiological features.

Treatment evaluation of odontogenic keratocyst by using CBCT and fractal dimension analysis on panoramic radiograph

The odontogenic keratocyst (OKC) is a benign intraosseous lesion that derives from remnants of the dental lamina. General practitioners usually use panoramic radiograph and cone-beam computed tomography (CBCT) as radiography examination of a suspected OKC lesion in the mandible or maxilla. Evaluation of post marsupialization could be assessed by using fractal dimension (FD) analysis of trabecular bone in panoramic radiograph and CBCT. In the present case, we use panoramic radiograph and CBCT for the treatment evaluation of OKC. The aim of this case report is to describe a case of OKC and evaluation of treatment progress by using FD analysis on panoramic radiograph and CBCT. A 40-year-old female patient came with a complaint of slowly enlarged swelling on the left mandible for 10 years. Low-grade pain occasionally happened to that women. Panoramic radiographs revealed multi-locular radiolucency extending from anterior of the mandible body to ascending ramus. Coronoid and condyloid process, and inferior border of the mandible were intact. OKC was confirmed based on biopsy of the lesion from extraction site of left lower third molar tooth. We did marsupialization from tooth extraction socket and let it open as a window to apply obturator. The researchers did a serial panoramic radiograph to evaluate treatment, showing reduction of radiolucency size and increasing of FD in lesion peripheral, it means that there was an osseous healing process. One year after marsupialization, the researchers did CBCT examination to evaluate bone regeneration and density. The result of CBCT showed the size reduction of the lesion in sagittal, coronal, and axial planes. CBCT also revealed bone formation from the periphery of the lesion. CBCT assessment and FD analysis of trabecular bone on panoramic radiograph is a reliable imaging method for the treatment evaluation of OKC and determination of ossification as well.

Prosthetic Rehabilitation of a Case of Odontogenic Keratocyst in Anterior Maxilla: A Six-Year Follow-Up

Prosthetic Rehabilitation of a Case of Odontogenic Keratocyst in Anterior Maxilla: A Six-Year Follow-Up

Evaluation of mandibular odontogenic keratocyst and ameloblastoma by panoramic radiograph and computed tomography.

The purpose of this study was to describe and compare the main imagenological features of mandibular ameloblastomas and odontogenic keratocyst (OKC) using panoramic radiograph (PR) and CT. The sample consisted of nine cases of ameloblastomas and nine cases of OKC. PR and CT images were analyzed according to shape, internal structure, borders, associated unerupted tooth, root resorption, expansion and perforation of cortical bones. PR evaluation allowed the identification of the lesion's location, presence of sclerosis in the periphery, presence of associated non-erupted tooth and expansion of the mandible's lower border cortical bone. CT was more accurate than PR in the assessment of the lesion shape, presence of inner bone septa, root resorption, buccolingual expansion and rupture of cortical bone. Most cases of ameloblastoma and OKC presented buccolingual expansion and erosion of cortical bone. Only ameloblastomas showed tooth root resorption. Although PR is very helpful and widely used, CT provides more precise information on buccolingual expansion, calcification, bone septa, perforation of cortical bones and tooth resorption, features that are frequently underdescribed in the literature, particularly in OKC.