Management regulations for odontogenic keratocyst: a case report and review of the literature.

BACKGROUND: Reconstruction of the entire dentition with odontogenic keratocyst is a very challenging quandary. Most cases of odontogenic keratocyst are often reported to be benign, resulting in severe occlusal discrepancies with the maxillary and mandibular dentition. Dental radiographs occasionally reveal an uncommon, locally aggressive developing cyst termed as odontogenic keratocyst, which is typically located in the posterior jaw. When this cyst occurs in the anterior region, it is often misdiagnosed with other periapical lesions due to its lack of response to pulp vitality tests. CASE PRESENTATION: This clinical case scenario demarcates the endodontic management of a patient diagnosed with odontogenic keratocyst. A 37-year-old Indian male patient reported to the department with throbbing pain in the lower left posterior tooth requiring endodontic therapy. This patient also presented with odontogenic keratocyst in the anterior region of the jaw, for which he had undergone surgical rehabilitation. This case report highlights the clinical protocol for the endodontic therapy in patient diagnosed with ododntogenic keratocyst. Masticatory impairment was not visible after the follow-up period and the treatment outcome was successful. CONCLUSION: This case report details the presentation, characteristic radiographic findings, and endodontic management of a patient with an extremely rare condition of odontogenic keratocyst. The management involves multidisciplinary approach for the rehabilitation.

Glandular odontogenic cyst: Review of literature and report of two cases.

Glandular odontogenic cyst (GOC) is a rare developmental odontogenic cyst from the cell rests of Serres. GOC is locally aggressive with a tendency toward recurrence. The most common site of occurrence is the anterior mandible with an asymptomatic presentation. Radiographically, it presents as unilocular or multilocular radiolucency. It bears histopathological resemblance to low-grade mucoepidermoid carcinoma. We report two cases of GOC occurring in a 16-year-old and a 33-year-old male patient with a review of the clinical presentation, histopathological features, and diagnostic aspects of GOC reported so far in literature.

Long-term investigation of minimally invasive alcohol-based therapy as the treatment of odontogenic keratocyst:A retrospective cohort study.

The aim of this study was to evaluate the clinical efficacy of alcohol-based therapy for patients with large odontogenic keratocysts (OKCs). The study was implemented as a retrospective, single-center study. Patients treated with ethanol-based therapy for odontogenic keratocyst were retrospectively evaluated for baseline and postoperative data. The pre- and postoperative clinical situation and the extent of radiographic shrinkage were compared. The event is defined as the achievement of >50% reduction in cyst volume. The cyst reduction rate calculated on panoramic radiographs ranged from 7.4% to 99.9% (mean [standard deviation] 55.3% [27.9%]) and was statistically significant (P < 0.05). Specifically, it has been found that, radiographically, 47.6% of patients achieved >50% reduction in cyst volume within 12 months. The continuous cortical bone was rebuilt, and the cyst cavity was filled with regenerated trabecular bone. The 22 included patients presented with nonclinical problems, had no need for further intervention, and exhibited persistent impaction of the teeth. The results of this study demonstrated that ethanol-based therapy triggered marked radiographic reductions of large OKC, indicating that using this technique is efficient.

Transformation of an odontogenic keratocyst into a solid variant of odontogenic keratocyst/keratoameloblastoma during long­term follow­up: A case report.

Keratoameloblastoma (KA) and solid variant of odontogenic keratocyst (SOKC) are rare odontogenic lesions, and their relationship and differences are unclear. The present study described a case that started as an odontogenic keratocyst (OKC) and transformed to SOKC/KA upon recurrence. Briefly, a 26­year­old man presented with swelling in the right cheek and was referred to the Department of Oral and Maxillofacial surgery, Hiroshima University Hospital (Hiroshima, Japan). At the initial visit, unicystic bone permeation was observed extending from the right canine to the molar, maxillary sinus and nasal cavity. After the biopsy, the patient underwent excisional surgery and was diagnosed with OKC. Thereafter, the lesion recurred six times over a period of 13 years and showed different histopathological features from those of the primary lesion, all consisting of numerous cysts with keratinization, which were diagnosed as SOKC/KA. The Ki­67 positivity rate was ~10%, which was higher than that of the primary lesion, but there was no atypia. Genetic analysis of the recurrent lesion revealed mutations in adenomatous polyposis coli and Kirsten rat sarcoma viral oncogene homolog. This case originated from OKC, and the morphological features of OKC and KA were mixed upon recurrence, supporting the commonality and association between the two. However, multiple mutations different from those of OKC and ameloblastoma were detected, suggesting an association of SOKC/KA with increased proliferative activity and a high recurrence rate.

Mandibular primary intraosseous carcinoma arising from long-standing odontogenic keratocyst.

Primary intraosseous carcinoma (PIOC) of the jaw is a rare neoplasm arising from the lining epithelium of odontogenic cysts or de novo from odontogenic epithelial rests that has no communication with the surrounding mucosa of the upper aerodigestive tract. We present a case of PIOC ex-odontogenic keratocyst (PIOC ex-OKC) in a 35-year-old male. Histopathologic examination revealed a cystic lesion with a fibrous capsule lined by corrugated parakeratinized stratified squamous epithelium resting on a basal cell layer composed of columnar cells exhibiting palisaded hyperchromatic nuclei, features consistent with OKC. Surgical treatment consisted of bilateral crestal and crevicular incision, a reflection of the flap, breaking of all OKC locules, creation of a continuous cavity, and fitting of a decompression mold around the mandibular teeth. This case highlights the importance of knowing the features of PIOC and considering PIOC in the differential diagnosis of malignant tumors of odontogenic epithelium for timely surgical treatment.

[Surgical removal of an atypically large extensive radicular cyst in the mandible: a case report.] / Atypisch grosse radikuläre Zyste. Operative Entfernung einer atypisch grossen ausgedehnten radikulären Zyste im Unterkiefer, ein Fallbericht.

The radicular cyst is the most common odontogenic cyst and is caused by inflammation. It can become atypically large, although the size of the radiographic osteolysis says nothing about the entity of the lesion. This case shows an unusually large multilocular radicular cyst expanding buccally from tooth 46 in a patient with severe autism who can only be treated under general anesthesia. The clinical and radiological picture as well as the intraoperative situation was more indicative of an aggressive cyst or benign tumor. The lesion was surgically completely removed and the teeth 46, 47 and 48 were extracted because of poor compliance and prognosis. Histopathology revealed a radicular cyst. There were no postoperative complications. After eight months, the lesions had almost completely reossified.

[Nasal obstruction: Odontogenic cysts in 4 brachycephalic dogs]. / Nasale Obstruktion: Odontogene Zysten bei 4 brachyzephalen Hunden.

OBJECTIVE: Odontogenic cysts may be of developmental or inflammatory origin. They are frequently observed in brachycephalic dogs. Due to their expansive growth, cysts in the maxilla may extend into the nasal cavity, obstruct the nasal airway and cause nasal discharge. Epithelial cysts may lead to a comparable clinical picture. A new endonasal endoscopic intervention for the removal of these cysts is described. MATERIAL AND METHODS: Four brachycephalic dogs (Boxer, Chihuahua, French bulldog, Pug) with unilateral cysts obstructing the nasal cavity were included in the study. The animals underwent clinical examination, computed tomography (CT) imaging and rhinoscopy examination. Histopathologic evaluation of the cyst wall was performed. In addition to dental surgery and conventional extraction of the involved tooth, the cyst wall was removed by endoscopic intervention via the physiologic nasal opening. Clinical course following treatment was assessed by rhinoscopy and CT. RESULTS: The main clinical signs were unilateral or bilateral serous to hemorrhagic nasal discharge. Nasal airflow was restricted in all patients. Removal of the cysts and involved teeth did not lead to any intra- and postoperative complications. No recurrence of the cysts was observed. Histologic diagnosis consisted of an epidermoid cyst, a follicular cyst, a radicular cyst, and a canine odontogenic parakeratinized cyst (COPC). Neoplastic transformation was excluded in all cases. CONCLUSIONS: Removal of the cyst wall may be performed by means of minimally invasive endoscopic intervention via the physiologic nasal entrance. The procedure is curative and carries the risk of only few complications. In cases in which treatment is restricted to extraction of the affected tooth, the nasal airway may remain obstructed due to a calcified cyst wall. CLINICAL RELEVANCE: In brachycephalic dogs, oronasal defects due to dental pathology are the most common reason for chronic nasal discharge, whereas in normocephalic dogs nasal neoplasia are. Odontogenic cysts carry importance as differential diagnosis of nasal discharge.

Multiple odontogenic keratocysts in a patient with Lowe syndrome: a first case report and literature review.

Lowe syndrome (LS) is a rare disease (1:500,000) with X-linked recessive inheritance involving the kidneys, eyes, and nervous system. A Mexican 25-year-old male patient presented for diagnosis of multiple radiolucent lesions observed on routine radiographic examination. General aspects revealed cognitive delay, eye alterations, and kidney involvement, which support the diagnosis of LS. Radiolucent well-delimited lesions were observed in both mandibular angle and symphysis. Under general anesthesia, incisional biopsy and decompression were performed. Histological aspects led to diagnosing odontogenic keratocyst (OKC) for all lesions. The lesions in the right and left mandibular angles were decompressed, and the symphyseal lesion was enucleated. A 2-month follow-up shows the bone healing process. There are few reports detailing oral findings in LS. Here, we reported the first case of multiple OKC in a patient with LS. In addition, we performed a literature review on odontogenic lesions in patients affected by LS.

Volumetric reduction in large cystic jaw lesions postoperative enucleation: a longitudinal clinical study.

BACKGROUND: Enucleation, a surgical procedure, is commonly used to treat large jaw cysts, unicystic ameloblastomas and keratocysts. However, it remains unclear to what extent the jaw bone regenerates after enucleation. We aimed to evaluate the percentage and the survival analysis of jaw bone regeneration, in terms of cavity volume residual (CVR), in patients who underwent enucleation of large jaw cysts, unicystic ameloblastomas and keratocysts. METHODS: We collected data longitudinally from 75 patients who underwent jaw cystic lesions enucleation at the Stomatological Hospital of Xi'an Jiaotong University, between January 2015 and June 2021. All patients had both preoperative and postoperative cone-beam computed tomography (CBCT) imaging data. CBCT images were analyzed using Image J. Changes in the CVR were assessed at various follow-up time points, and the Kaplan-Meier method was utilized to evaluate the CVR over time. RESULTS: The patients had a mean age of 31.7 years (range: 5.5-72 years) with 58.66% of them being male. The postoperative CVR was 32.20% at three months, 21.10% at six months, 15.90% at 12 months, and 5.60% at 24 months. The percentage of CVR during follow-up periods for the initial size Quartile (Q)1 (212.54-1569.60 mm3) was substantially lower than those of Q2 and Q3 at and after seven months of follow-up and became statistically significant at the 12-month mark. CONCLUSION: This study demonstrates that spontaneous bone regeneration can occur after enucleation of large jaw cysts, unicystic ameloblastomas and keratocysts, even without the use of filler materials. The initial size of the lesion had a significant impact on the outcome of cystic lesion enucleation over time. To minimize the risks associated with radiation exposure and expenses, we recommend reducing the frequency of CT imaging follow-ups for patients with small initial cavity sizes (ranging from 212.54 to 1569.60 mm3).

Idiopathic bone cavity mimicking a botryoid odontogenic cyst: A rare radiographic presentation in an older adult.

OBJECTIVES: This report describes an unusual case of a multilocular idiopathic bone cavity (IBC) that presented as a botryoid odontogenic cyst situated between the mandibular lateral incisor and canine in an older adult. BACKGROUND: The IBC represents an intraosseous concavity that appears radiographically as a unilocular or multilocular radiolucent lesion found in various skeletal sites, including the jaw. Atypical cases of gnathic IBC have not been appreciated in the gerodontologic literature. MATERIALS AND METHODS: The teeth adjacent to the bony lesion had normal pulpal responses to cold. A full-thickness flap was elevated and provided a direct entry into a bony concavity, which was devoid of an epithelial lining and fluid. RESULTS: The lack of a cystic lining within the empty osseous lesion following surgical entry, concomitant with the vital pulpal status of the proximal teeth, led to a diagnosis of an IBC. The bony walls underwent curettage and copious irrigation prior to primary closure. A 10-month follow-up revealed partial evidence of osseous repair. The patient will continue to be monitored. CONCLUSION: Timely surgical intervention of central lesions of the jaws may improve clinical outcomes. Variants of the IBC should be included in the differential diagnosis of multilocular lesions, particularly in the geriatric population.

Mandibular reconstruction after excision of recurrent odontogenic keratocyst using a novel mandibular distraction osteogenesis method- a case report.

BACKGROUND: Odontogenic keratocyst is one of the most common benign odontogenic neoplasms with a high recurrence rate. Its resection has the potential to lead to mandibular segmental defects. In this case report, we describe a patient with odontogenic keratocyst who underwent radical resection using a novel distraction osteogenesis (DO) method to reconstruct mandibular segmental defect. CASE PRESENTATION: This case report describes a 19-year-old woman with odontogenic keratocyst of the mandible that recurred after multiple curettages and eventually necessitated radical resection. Mandibular segmental defect after radical resection was reconstructed using a novel DO method that involved directly contacting the segment ends of the defect without the transport disk. However, the distractor broke during the retention period, and a molding titanium plate was used for fixation. This novel distraction method achieved mandibular reconstruction and restored mandibular function and contour.

[Treatment tactics for patients with retention cysts, odontogenic cysts, mucocele and other pathologies of the maxillary sinuses as preparation for a sinus lift]. / Lechebnaya taktika v otnoshenii retentsionnykh kist, odontogennykh kist, mukotsele i drugikh zabolevanii verkhnechelyustnoi pazukhi pri podgotovke k sinus-liftingu.

OBJECTIVE: The aim the studi. Differential diagnosis of retention cysts of the maxillary sinus with odontogenic cysts, acute and chronic sinusitis, aspergillosis, and mucocele in preparation for sinus-lifting surgery. MATERIAL AND METHODS: The analysis of the case histories of 265 patients aged 18-65 years of both sexes who were treated at the dental clinic «Rudenta Family¼ from 2016 to 2021 was carried out. On the basis of clinical symptoms and data of cone-beam computed tomography (CBCT), a description of the pathologies of HPV is given for the purpose of differential diagnosis and correct interdisciplinary interaction with ENT doctors for the purpose of dental implantation in the lateral parts of the upper jaw. RESULTS: In 90 (out of 265) patients (34%), a change in the condition of the maxillary sinus mucosa was detected. 18 patients (7%) for preoperative preparation were sent to the ENT department of the FSBI «CCB with polyclinic¼ of the UDP of the Russian Federation with diagnoses: chronic maxillary sinusitis of various etiologies and mucocele. In this group of patients, sinus lifting followed by dental implantation was performed 6 months after endoscopic maxillofacial surgery under the control of CBCT. Retention cyst Retention cysts of the maxillary sinus were of different sizes in 62 (23.4%) patients and, depending on the size and localization of the sinus-lifting was performed with simultaneous removal of the cyst, or without removal of the cyst. CONCLUSION: Retention cysts do not need to be removed as a preoperative preparation for sinus lifting. In the case of large sizes and difficulty in peeling the Schneider membrane, retention cysts are removed by a dental surgeon during antral augmentation as one of the stages of the operation. In such pathologies as odontogenic cyst, acute and chronic sinusitis, aspergillosis, mucocele, interdisciplinary interaction of ENT doctor and dentist is necessary. Differential diagnosis of maxillary snus pathology is carried out on the basis of clinical data and a picture of cone-beam computed tomography.

Quiste odontogénico: no todo son flemones / Odontogenic cyst: not all are phlegmons

Los quistes odontogénicos aparecen en el maxilar o la mandíbula y se generan por la proliferación de restos epiteliales estimulados habitualmente por una periodontitis. Son infrecuentes en niños. No suelen dar síntomas y el diagnóstico es radiográfico casual. La evolución del quiste odontogénico o periapical suele ser lenta, pero tiende a destruir el hueso adyacente, por lo que requiere tratamiento. Describimos un caso clínico que tuvo una presentación no habitual (AU)

Odontogenic cysts appear in the maxilla or mandible and are generated by the proliferation of epithelial remains stimulated by periodontitis. They are uncommon in children. They do not usually give symptoms and the diagnosis is casual radiographic. The evolution of the periapical cyst is usually slow, but tends to destroy the adjacent bone, so it usually requires endodontic treatment. We describe a clinical case that had an unusual presentation. (AU)

[Clinical analysis of the treatment of maxillary odontogenic cyst by nasal endoscope fenestration through nasal base].

Objective:To investigate the feasibility and clinical effect of the surgical approach and method of transnasal fenestration under nasal endoscope for the treatment of maxillary odontogenic cyst. Methods:The clinical data of 23 cases with maxillary odontogenic cysts treated by nasal endoscopy through nasal fenestration were retrospectively analyzed. All cases underwent nasal endoscopy and CT examination before the operation. The mucosal membrane of the parietal wall of the cyst was excised through fenestration of the nasal base. The cyst fluid was removed by decompression, and the bony opening of the nasal base was trimmed and enlarged to the edge of the cyst. The intraoperative and postoperative effects were observed. Results:All cases were well exposed under the direct vision of nasal endoscope. The top wall of the cyst was removed to maximize the communication between the cyst cavity and the nasal floor. There were no complications such as nasolacrimal duct injury, turbinate atrophy, necrosis, and facial numbness. All patients were followed up for 6-12 months, and the clinical symptoms gradually disappeared after surgery. The inferior turbinate was in good shape, the cyst cavity was smooth, the cyst wall was determined, and no cyst recurrence was observed. Conclusion:The treatment of odontogenic cyst of maxilla under nasal endoscope through nasal fenestration is convenient. It has less trauma, fewer complications and a satisfactory curative effect, which is worthy of clinical promotion.

Safety and efficacy of adjunctive therapy in the treatment of odontogenic keratocyst: a systematic review.

The odontogenic keratocyst (OKC) is a common cystic lesion in the jaw. Its management, however, is highly debated with no consensus on the best treatment option. Clinicians base their approach on treatment efficacy and associated morbidity. Management often consists of enucleation with peripheral ostectomy and adjunctive therapy to prevent recurrence. The aim of our systematic review was to evaluate the safety and efficacy of these different modalities. Embase, Medline, and Cochrane were searched according to the PRISMA guidelines for articles that presented non-syndromic patients with histopathologically confirmed OKC treated with 5-fluorouracil (5FU), Carnoy's solution (CS), or modified Carnoy's solution (MCS) as adjunctive therapy after enucleation and peripheral ostectomy. The outcomes of interest were safety (measured as adverse events) and efficacy (expressed as recurrence). Risk of bias was evaluated using the Newcastle-Ottawa scale. Four studies were included and 62 patients were evaluated. The results show that recurrence occurred only in patients treated with MCS. Reported adverse events were mostly limited to paraesthesia that could be permanent (in the CS and MCS treatment groups) or transient (across all adjunctive therapies). With the prohibition of CS, both MCS and 5FU are promising replacement adjunctive therapies. From a safety and efficacy perspective we consider 5FU, which was associated with the lowest recurrence and fewest adverse events, to be the most viable option. More high-evidence prospective studies, such as randomised controlled trials, with a longer follow-up period are necessary to draw definite conclusions.

A retrospective study of the malignant change of odontogenic keratocyst.

The odontogenic keratocyst (OKC) is a common cystic lesion of the maxilla and mandible. Squamous cell carcinoma (SCC) arising from OKC or dysplasia occurring in OKC is rare. This study aimed to explore the incidence and clinical features of the dysplasia and malignant transformation of OKC. In this study, 544 patients diagnosed with OKC were collected. Among them, 3 patients were diagnosed as SCC arising from OKC, and 12 patients were diagnosed as OKC with dysplasia. The incidence was calculated. Clinical features were analyzed by chi-square test. In addition, a representative case reconstructing mandible with vascularized fibula flap under general anesthesia was reported. And cases reported before were reviewed. The incidence of the dysplasia and malignant transformation of OKC, which are highly associated with the clinical features of swelling and chronic inflammation, is about 2.76%. But the relevance between the dysplasia and malignant transformation and age, gender together with pain is not statistically high. All in all, the clinical features of swelling and chronic inflammation can be considered as characteristics of the dysplasia and malignant transformation of OKC. Although the pain isn't statistically relevant, it may be a dangerous clew. Also, combined with earlier literatures, the dysplasia and malignant transformation of OKC shows unique features of radiographs and histopathology.

Volumetric changes in the size of odontogenic keratocysts after decompression followed by enucleation, peripheral ostectomy, and Carnoy's solution: A retrospective study.

The study aimed to retrospectively analyze the reduction pattern of odontogenic keratocysts (OKCs) after decompression, followed by enucleation (EN), peripheral ostectomy (PO), and Carnoy's solution (CS) to establish the appropriate time for inserting implants, along with assessing the long-term success of conservative treatment with adjunctive therapy. The predictable variables were the reduction pattern and the study's treatment option. The outcome variable was the volumetric changes in the size of bony defects. These changes were determined using a percentage difference and a reduction rate. They were recorded after decompression and one, three, six, twelve, and eighteen months after EN. P-values of .05 were considered significant. The study included 66 patients with 71 OKCs. Males, younger ages, and mandibular OKCs significantly predominated. The decompression significantly changed the initial volume from 135.40 ± 1.2 cm3 to 101.55 ± 0.1 cm3 with 28.6 percentage difference and 25% reduction rate. At the end of the first and third months after EN, the reduction pattern is 50.0%-75.5% of the initial volume, with no significant prediction for the direction of the reduction pattern. After 18 months, all bony defects disappeared, with no recurrences for the next 18 years. In conclusion, the reduction pattern is 75.5% of its initial volume at the end of the third month after OKC management. Therefore, within the limitations of the study, its treatment approach seems to be an option amongst other protocols that includes a view to early implant based dental rehabilitation.

Squamous Cell Carcinoma Arising From Odontogenic Cyst Epithelium or Oral Mucosa Epithelium After Marsupialization of Odontogenic Radicular Cyst in Posterior Mandible?

Squamous cell carcinomas (SCCs) are common malignant tumors in the oral and maxillofacial region. However, SCCs secondary to marsupialization of odontogenic radicular cysts are extremely rare. The authors report an unusual case of a 43-year-old male presented with dull pain in the right molar region of the mandible without numbness of lower lips, who had a long history of smoking, alcohol consumption, and betel nut chewing. Computerized tomography revealed a round well-defined unilocular radiolucent at the apex of lower right premolars, 2 nonvital teeth. The clinical diagnosis was the radicular cyst of the right mandible. The patient was initially treated with root canal therapy of the teeth followed by marsupialization with a mandibular vestibular groove incision. While the patient did not follow the instruction of irrigation of the cyst and had no regular follow-up. The reexamination of computerized tomography indicated a round well-defined unilocular radiolucent at the apex of lower right premolars and filled with a soft tissue without clear boundary with buccal muscles at 31 months follow-up. There were no masses or ulcer around the mandibular vestibular groove incision and the patient had no sign of numbness of lower lips. The clinical diagnosis was the radicular cyst of right mandible with infection. A curettage was performed. However, the pathologic diagnosis was well-differentiated SCC. An extended radical surgical resection including segmental resection of the right mandible was performed. The histopathology was well-differentiated SCC without the cyst epithelium and invasion of bone, which can be distinguished from primary intraosseous SCC. The case indicates that marsupialization performed in patients with a history of smoking, alcohol consumption, and betel nut chewing, has a risk of suffering from oral SCC.

An evidence-based surgical algorithm for management of odontogenic keratocyst.

The effective management of odontogenic keratocyst (OKC) remains a subject of interest and confusion in the oral and maxillofacial surgery literature. Currently, there is a lack of consensus regarding the most appropriate treatment for patients with OKC. Of the various treatment options available, no modality to date has been shown to demonstrate a zero or near-zero recurrence rates except wide resection with clear margins. With the prevailing dearth of evidence based surgical protocols for the management of patients with OKC in the literature, this study aims to present a surgical algorithm, based on meta-analysis results, that hopefully will be beneficial in enhancing treatment of patients with this condition. Also, new meta-analysis was done to compare between modified Carnoy's solution (MCS) and 5-fluorouracil (5-FU) in respect of recurrence rate of OKC. Using parameters like size, lesion type (primary or secondary), syndromic or solitary nature of the lesion, presence of cortical perforations, and locularity; we present a decision tree, to aid treatment planning and help attain the least chance of recurrence in the management of the OKC. There was very low-quality evidence indicating that application of 5-FU, after enucleation and peripheral ostectomy of OKCs, significantly lowered recurrence rate when compared to MCS (RR = 0.087, CI: 0.017 to 0.436, P value = 0.003).