Immuno-oncologic signature of malignant transformation in oral squamous cell carcinoma.

OBJECTIVE: The purpose of this study is to identify the immuno-oncologic (IO) signature at the surgical tumor margin (TM) of oral squamous cell carcinoma (OSCC) that is involved in the process of malignant transformation. STUDY DESIGN: Under institutional review board approval, TM of 73 OSCC were investigated using immunohistochemistry for the immune biomarker, programmed death ligand-1 (PD-L1). NanoString 770 IO-focused gene set was analyzed in 5 pairs of TM and invasive tumor (T). PD-L1 regulation in response to interferon-gamma (IFN-γ) was investigated in an oral potentially malignant cell line (OPMC). RESULTS: Programmed death ligand-1 expression in the epithelial margin directly correlated with its expression in the underlying immune cells (P = .0082). Differential gene expression showed downregulation of PD-L1 and IFN-γ 6 gene signature in the TM relative to T pair.CD8 and macrophages were higher in TM. CNTFR, LYZ, C7, RORC, and FGF13 downregulation in T relative to TM. TDO2, ADAM12, MMP1, LAMC2, MB21D1, TYMP, OASL, COL5A1, exhausted_CD8, Tregs,and NK_CD56dim were upregulated in T relative to TM. Finally, IFN-γ induced upregulation of PD-L1 in the OPMC. CONCLUSIONS: Our work suggests a role for IFN-γ in PD-L1 upregulation in OPMC and presents novel IO transcriptional signatures for frankly invasive OSCC relative to TM.

Extracorporealization of the Mandibular Condyle: Effects on Viability and Function.

Study Design: For certain condylar fractures, extracorporealization of the condylar segment may be performed via extra-oral vertical ramus osteotomy (EVRO) to facilitate reduction and fixation. This approach can similarly be used for condyle-sparing resection of osteochondromas of the condyle. Due to controversy regarding long-term health of the condyle after extracorporealization, we conducted a retrospective analysis of surgical outcomes. Objective: For certain condylar fractures, extracorporealization of the condylar segment may be performed via extra-oral vertical ramus osteotomy (EVRO) to facilitate reduction and fixation. This approach can similarly be used for condyle-sparing resection of osteochondromas of the condyle. Due to controversy regarding long-term health of the condyle after extracorporealization, we investigated the viability of this technique through a retrospective analysis of outcomes. Methods: Twenty-six patients were treated using EVRO with extracorporealization of the condyle for both condylar fractures (18 patients) and osteochondroma (8 patients). Of the 18 trauma patients, 4 were excluded due to limited follow-up. Clinical outcomes were measured, including occlusion, maximum interincisal opening (MIO), facial asymmetry, incidence of infection, and temporomandibular joint (TMJ) pain. Radiographic signs of condylar resorption were investigated, quantified, and categorized using panoramic imaging. Results: Average follow-up was 15.9 months. Average maximum interincisal opening was 36.8 mm. Four patients demonstrated mild resorption and one patient demonstrated moderate resorption. Two cases of malocclusion were attributed to failed repairs of other concurrent facial fractures. Three patients reported TMJ pain. Conclusions: Extracorporealization of the condylar segment with EVRO to facilitate open treatment of condylar fractures is a viable treatment option when more conventional approaches prove unsuccessful.

Cytokine profiling in plasma distinguishes the histological inflammatory subtype of head and neck squamous cell carcinoma and a novel regulatory role of osteopontin.

Head and neck squamous cell carcinoma (HNSCC) can be classified according to the histological inflammatory subtype (HIS) into inflamed (HIS-INF) or immune excluded (HIS-IE). HIS-IE was previously associated with higher levels of soluble Semaphorin 4D (HsS4D) in plasma, and higher transcriptional levels of osteopontin (OPN) in the tumor tissue, compared to HIS-INF. The goal of the current study is to investigate whether the HIS inflammatory subtype can be distinguished by a differential cytokine panel in peripheral blood. Retrospectively collected five HIS-INF and five HIS-IE tumor tissue with paired plasma were included in the study. Five healthy donors (HD) and five autoimmune/chronic inflammatory conditions (AI/CI) were controls. The ELISA-Luminex™ system was used to detect 40 traditional cytokines in plasma. Human cytokine array (104 cytokines) was used for the conditioned medium (CM) of the HNSCC HN6 cell line. Semaphorin 4D (Sema4D) siRNA and recombinant human osteopontin (rh-OPN) were used to investigate the effect of OPN on Sema4D expression. The HIS-IE cytokine profile was higher than HIS-INF but comparable to AI/CI. HIS-INF had the lowest cytokine levels. HIS-IE was differentially higher in IP-10 and IL8 compared to HD, while HIS-INF was higher in IL-10. Sema4D inhibition in HN6 resulted in a decrease of OPN in the CM of HN6, and treatment with rh-OPN rescued Sema4D in HN6 cell lysate and associated CM. In conclusion, the current work demonstrates a novel association between the HIS subtypes and a differential pattern of cytokine expression in plasma. These findings can open new avenues for HNSCC patient stratification and hence provide better personalized treatment.

Juvenile trabecular ossifying fibroma: Immunohistochemical expression of MDM2, CDK4 and p53 compared to conventional ossifying fibroma.

BACKGROUND: Juvenile ossifying fibroma (JOF) is an uncommon benign fibro-osseous lesion of the craniofacial skeleton; compared to conventional ossifying fibroma (OF), JOF is characterized by local aggressiveness and propensity for recurrence. The biologic basis for this different biologic behavior between JOF and OF remains elusive. The aim of this study was to evaluate the immunohistochemical expression of MDM2, CDK4 and p53, molecules associated with bone oncogenesis, in the trabecular variant of JOF. MATERIAL AND METHODS: The study material consisted of five cases of trabecular JOF, affecting three male and two female patients with a mean age of 11.8 years. Three cases arose in the maxilla and two in the mandible. All cases were initially treated by enucleation; two cases recurred necessitating more aggressive treatment. Immunohistochemical study of MDM2, CDK4 and p53 was performed in all cases, as well as in five control cases of conventional OF. RESULTS: CDK4 positivity was noted in all JOF cases; the staining pattern was diffuse and strong in 4 cases and focal and weak in one case. In contrast, 4 out of 5 cases of OF were weakly and focally CDK4 positive, the remaining one being negative. Immunostaining for MDM2 was observed in 3 JOF cases; all OF were MDM2 negative. All cases of OF and JOF were negative for p53, except for one focally positive JOF case. CONCLUSIONS: CDK4 and MDM2 expression in the trabecular variant of JOF is higher compared to conventional OF. In contrast, p53 expression is almost universally negative in JOF and OF. Despite some overlapping features, differential expression patterns of proteins involved in bone oncogenesis can elucidate the pathogenesis and may facilitate accurate diagnosis and prediction of behavior of bone tumors in the craniofacial region. Key words:Juvenile ossifying fibroma, trabecular variant, conventional ossifying fibroma, MDM2, CDK4, p53.

Oral squamous cell carcinoma in patients aged 45 and younger: Prognosis, survival, and quality of life.

OBJECTIVE: To perform a detailed analysis of the epidemiology, tumor biology, treatment, overall survival, and quality of life in a young patient (age ≤45 years) cohort with oral squamous cell carcinoma (OSCC). STUDY DESIGN: A retrospective cohort study between 1992 and 2017 at an academic tertiary care center. RESULTS: In total, 80 patients were included (36 female and 44 male) with stage I (American Joint Committee on Cancer eighth ed.) disease and lateral tongue was most common presentation. Mean follow-up was 6.28 years. The overall disease recurrence rate was 28.7% (23 of 80). Human papillomavirus was positive in 22% of patients tested. Free flap reconstruction was not associated with improved margin status (P = .62) but significant for recurrent disease (P < .04). Overall 2-year survival was significantly poorer in patients with close/positive margin status and free flap reconstruction. Patients with early-stage disease (stage II) requiring adjuvant radiotherapy, chemotherapy (all stages), or flap reconstruction (Stage III patients) had significantly worse 5-year survival rates. CONCLUSIONS: OSCC in young patients (age ≤45 years) is an increasingly more common disease that occurs in patients without known risk factors. Despite their earlier presentation of disease pathology, constant vigilance and standard aggressive treatment similar to other age groups will result in similar and improved outcomes and survival.

Impact of the Novel Coronavirus 2019 (COVID-19) Pandemic on Head and Neck Cancer Care.

OBJECTIVE: The study aimed to assess the impact of the coronavirus disease 2019 (COVID-19) pandemic on head and neck oncologic care at a tertiary care facility. STUDY DESIGN: This was a cross-sectional study conducted between March 18, 2020, and May 20, 2020. The primary planned outcome was the rate of treatment modifications during the study period. Secondary outcome measures were tumor conference volume, operative volume, and outpatient patient procedure and clinic volumes. SETTING: This single-center study was conducted at a tertiary care academic hospital in a large metropolitan area. METHODS: The study included a consecutive sample of adult subjects who were presented at a head and neck interdepartmental tumor conference during the study period. Patients were compared to historical controls based on review of operative data, outpatient procedures, and clinic volumes. RESULTS: In total, 117 patients were presented during the review period in 2020, compared to 69 in 2019. There was an 8.4% treatment modification rate among cases presented at the tumor conference. There was a 61.3% (347 from 898) reduction in outpatient clinic visits and a 63.4% (84 from 230) reduction in procedural volume compared to the prior year. Similarly, the operative volume decreased by 27.0% (224 from 307) compared to the previous year. CONCLUSION: Restrictions related to the COVID-19 pandemic resulted in limited treatment modifications. Transition to virtual tumor board format observed an increase in case presentations. While there were reductions in operative volume, there was a larger proportion of surgical cases for malignancy, reflecting the prioritization of oncologic care during the pandemic.

Soluble Sema4D in Plasma of Head and Neck Squamous Cell Carcinoma Patients Is Associated With Underlying Non-Inflamed Tumor Profile.

Semaphorin 4D (Sema4D) is a glycoprotein that is expressed by several tumors and immune cells. It can function as a membrane bound protein or as a cleaved soluble protein (sSema4D). We sought to investigate the translational potential of plasma sSema4D as an immune marker in plasma of patients with head and neck squamous cell carcinoma (HNSCC). Paired peripheral blood and tumor tissue samples of 104 patients with HNSCC were collected at the same time point to allow for real time analysis. Scoring of the histological inflammatory subtype (HIS) was carried out using Sema4D immunohistochemistry on the tumor tissue. sSema4D was detected in plasma using direct ELISA assay. Defining elevated sSema4D as values above the 95th percentile in healthy controls, our data showed that sSema4D levels in plasma were elevated in 25.0% (95% CI, 16.7-34.9%) of the patients with HNSCC and showed significant association with HIS immune excluded (HIS-IE) (p = 0.007), Sema4D+ve tumor cells (TCs) (p = 0.018) and PD-L1+ve immune cells (ICs) (p = 0.038). A multi-variable logistic regression analysis showed that HIS was significantly (P = 0.004) associated with elevated sSema4D, an association not explained by available patient-level factors. Using the IO-360 nanoString platform, differential gene expression (DGE) analysis of 10 HNSCC tumor tissues showed that patients with high sSema4D in plasma (HsS4D) clustered as IFN-γ negative tumor immune signature and were mostly HIS-IE. The IC type in the HsS4D paired tumor tissue was predominantly myeloid, while the lymphoid compartment was higher in the low sSema4D (LsS4D). The Wnt signaling pathway was upregulated in the HsS4D group. Further analysis using the IO-360, 770 gene set, showed significant non-inflamed profile of the HsS4D tumors compared to the LsS4D. In conclusion, our data reveals an association between sSema4D and the histological inflammatory subtype.

Factor V Leiden thrombophilia dental considerations.

Factor V Leiden thrombophilia, a relatively common inherited type of hypercoagulability resulting from a mutation in the gene for factor V, has received minimal attention in the dental literature. This review examines related demographic information, risk factors, comorbidities, the thrombotic mechanism, clinical features, diagnostic measures, and medical management strategies. In addition, oral and maxillofacial sequelae and management guidelines are provided. If a patient is known to have the mutation, the clinician should review the patient's potential risk factors for development of thrombosis and ascertain whether any coagulation agents are currently being administered. The practitioner should be prepared to manage instances of prolonged bleeding. The dentist also should be aware of an overall increased risk of systemic thromboembolic events, particularly following head and neck trauma. Rarely, the factor V Leiden mutation has been associated with osteonecrosis of the jaw, usually concurrent with intake of sex hormones.

Is Long-Term Follow-Up Mandatory for Stage I Oral Tongue Cancer?

PURPOSE: The objective of this study was to analyze the outcomes and possible risk factors for late recurrence of pathologic stage I oral tongue squamous cell carcinomas (SCCs) in patients considered disease free at 3 years. MATERIALS AND METHODS: This retrospective study evaluated all patients with pathologic stage I oral tongue cancer within a tertiary care center from 2003 through 2013 who had been followed for a minimum of 36 months. RESULTS: One hundred twelve patients met inclusion criteria for long-term analysis. Despite the high overall survival of 92.2% for true pT1N0M0 disease, initial surgery failed in 25 of 112 patients (22.3%) who developed late disease recurrence (>36-month follow-up) locally (19.6%; n = 22), regionally (4.4%; n = 5), or as second primary disease (11.6%; n = 13). Eleven patients (50%) who had local recurrence could be salvaged with a second surgery, requiring no further treatment (mean, 48.7 months). Projected 10-year disease-free survival and overall survival were 61 and 89%, respectively. Thirty-three percent (n = 3 of 9) of deaths occurred in long-term patients considered disease free at 36 months. CONCLUSION: Stage I tongue SCC is more common in women and is associated with pre-existing leukoplakia. Although overall survival is excellent, a high failure rate from local recurrence or a new second primary is seen over an extended period. Long-term follow-up is mandatory because local salvage rates are excellent if SCC is diagnosed early. Regional failure carries a poor prognosis.

Margin Analysis: Malignant Salivary Gland Neoplasms of the Head and Neck.

There are no established protocols for the optimum surgical margin required for salivary gland malignancies. Factors including histologic diagnosis and TNM stage have been shown to be important in prognosis and survival outcome and mandate special consideration of margin size. Salivary cancers are treated differently at different anatomic sites, and different histologic types show a propensity for major or minor glands. Low-grade malignancies are treated with soft tissue margins of 1 cm or less. The facial nerve is preserved unless infiltrated and encased. Adenoid cystic carcinoma and carcinoma ex pleomorphic adenoma require more complex planning to obtain negative margins.

The role of perineural invasion in treatment decisions for oral cancer patients: A review of the literature.

PURPOSE: The role of perineural invasion (PNI) in the management of patients with oral squamous cell carcinoma (OSSC) is still controversial, and there is no consensus regarding the most appropriate therapeutic approach. The purpose of this study is to review the findings in the literature describing OSCC as a neurotropic malignancy, with the aim of correlating perineural invasion with treatment decisions and disease prognosis. MATERIALS AND METHODS: A literature search was conducted of references based on the MEDLINE and Cochrane Database of Systematic Reviews, with subject keywords including four main categories: perineural invasion, perineural spread, oral squamous cell cancinoma, neurotropic carcinoma. RESULTS: In this systematic review and analysis, more than 350 publications met the eligibility criteria of the authors. CONCLUSION: Perineural invasion (PNI) is a widely recognized indicator of poor prognosis in oral cancer patients, strongly correlating with aggressive tumor behavior, disease recurrence, and increased morbidity and mortality. Elective neck dissection could be an indicator in improving neck control in PNI-positive patients, while the addition of adjuvant postoperative radiotherapy may not significantly improve survival rates. Various molecular markers have been correlated with perineural tumor spread, but further investigations are required before targeting PNI as part of advanced cancer therapies.

Extensive Regional Metastasis of High-Grade Mucoepidermoid Carcinoma of an Unknown Primary Tumor.

PURPOSE: Mucoepidermoid carcinoma (MEC) is the most common salivary carcinoma. It arises most frequently in the major salivary glands, but can also arise in minor glands or intraosseous sites. MEC of an unknown primary occurs very rarely. The present report documents only the third case reported in medical studies. METHODS: A 66-year-old man with previous carcinoma in situ (CIS) of the left posterior oral tongue that had been excised in 2004 and again in 2010 presented with a hard lymph node, 3 × 2 cm at level II of the right neck in July 2015. Positron emission tomography-computed tomography (PET-CT) revealed multiple, bilateral cervical lymphadenopathy, with no primary site identified. Fine needle aspiration biopsy and cytologic examination from the right neck was positive for malignancy, suggestive of metastatic squamous cell carcinoma. Panendoscopy and biopsy revealed CIS at the tongue bases and tonsils bilaterally (p16-negative). The patient's case was presented to a tumor board, and definitive concurrent cispl.atin-based chemoradiation was recommended for TisN2cM0, stage IVA oropharyngeal CIS, which was completed in November 2015. PET-CT in January 2015 showed complex interval changes, with some areas demonstrating improvement (ie, no uptake in the left neck) and worsening in others (ie, increased metabolic activity in the right neck), suggestive of residual disease. Repeat PET-CT in March 2016 showed increased nodal involvement and increasing standardized uptake value. Bilateral modified radical neck dissection was undertaken, and histologic examination showed high-grade MEC in 51 of 61 lymph nodes with extracapsular spread and soft tissue involvement. RESULTS: The patient died in May 2016 at 2 months after surgery. CONCLUSIONS: Metastatic MEC of an unknown primary is a diagnostic challenge. PET-CT might not be the most reliable diagnostic investigation to identify the primary or metastatic foci, such as was demonstrated in the present case.

Analysis of speech and tongue motion in normal and post-glossectomy speaker using cine MRI.

OBJECTIVE: Since the tongue is the oral structure responsible for mastication, pronunciation, and swallowing functions, patients who undergo glossectomy can be affected in various aspects of these functions. The vowel /i/ uses the tongue shape, whereas /u/ uses tongue and lip shapes. The purpose of this study is to investigate the morphological changes of the tongue and the adaptation of pronunciation using cine MRI for speech of patients who undergo glossectomy. MATERIAL AND METHODS: Twenty-three controls (11 males and 12 females) and 13 patients (eight males and five females) volunteered to participate in the experiment. The patients underwent glossectomy surgery for T1 or T2 lateral lingual tumors. The speech tasks "a souk" and "a geese" were spoken by all subjects providing data for the vowels /u/ and /i/. Cine MRI and speech acoustics were recorded and measured to compare the changes in the tongue with vowel acoustics after surgery. 2D measurements were made of the interlip distance, tongue-palate distance, tongue position (anterior-posterior and superior-inferior), tongue height on the left and right sides, and pharynx size. Vowel formants Fl, F2, and F3 were measured. RESULTS: The patients had significantly lower F2/Fl ratios (F=5.911, p=0.018), and lower F3/F1 ratios that approached significance. This was seen primarily in the /u/ data. Patients had flatter tongue shapes than controls with a greater effect seen in /u/ than /i/. CONCLUSION: The patients showed complex adaptation motion in order to preserve the acoustic integrity of the vowels, and the tongue modified cavity size relationships to maintain the value of the formant frequencies.

Analysis of speech and tongue motion in normal and post-glossectomy speaker using cine MRI

ABSTRACT Objective Since the tongue is the oral structure responsible for mastication, pronunciation, and swallowing functions, patients who undergo glossectomy can be affected in various aspects of these functions. The vowel /i/ uses the tongue shape, whereas /u/ uses tongue and lip shapes. The purpose of this study is to investigate the morphological changes of the tongue and the adaptation of pronunciation using cine MRI for speech of patients who undergo glossectomy. Material and Methods Twenty-three controls (11 males and 12 females) and 13 patients (eight males and five females) volunteered to participate in the experiment. The patients underwent glossectomy surgery for T1 or T2 lateral lingual tumors. The speech tasks “a souk” and “a geese” were spoken by all subjects providing data for the vowels /u/ and /i/. Cine MRI and speech acoustics were recorded and measured to compare the changes in the tongue with vowel acoustics after surgery. 2D measurements were made of the interlip distance, tongue-palate distance, tongue position (anterior-posterior and superior-inferior), tongue height on the left and right sides, and pharynx size. Vowel formants Fl, F2, and F3 were measured. Results The patients had significantly lower F2/Fl ratios (F=5.911, p=0.018), and lower F3/F1 ratios that approached significance. This was seen primarily in the /u/ data. Patients had flatter tongue shapes than controls with a greater effect seen in /u/ than /i/. Conclusion The patients showed complex adaptation motion in order to preserve the acoustic integrity of the vowels, and the tongue modified cavity size relationships to maintain the value of the formant frequencies.

Adenoid cystic carcinoma of the minor salivary glands: a retrospective series of 29 cases and review of the literature.

OBJECTIVE: Approximately 78% of minor salivary gland tumors are malignant, of which adenoid cystic carcinoma (ACC) represents 15% in our series. ACC is an uncommon tumor characterized by slow growth and a high potential for recurrence. This series of 29 consecutive patients examines clincopathologic features, management, and survival outcomes. MATERIALS AND METHODS: This study is a retrospective chart review of 29 patients with ACC of the minor salivary glands in a period of 23 years (1989 and 2012). RESULTS: The mean age was 61.2 years (16-89 years), with no gender predilection. The majority occurred in the palate/maxilla (66%) and initial presentation was stage IV. Mean follow-up was 42.6 months. Recurrence rate was 10% local, 14% distant over the observation period. CONCLUSIONS: The palate/maxilla is the preferred location for occurrence, and initial presentation at stage IV is common. Postoperative radiation remains a common strategy to prevent local recurrence in lesions with adverse features.

Benign Pediatric Salivary Gland Lesions.

Salivary gland lesions are rare in pediatric patients. In addition, the types of salivary gland tumors are different in their distribution in specific sites in the major and minor salivary glands in children compared with adults. This article reviews benign neoplastic and nonneoplastic salivary gland disorders in pediatric patients to help clinicians to develop an orderly differential diagnosis that will lead to expedient treatment of pediatric patients with salivary gland lesions.

Pediatric Salivary Gland Malignancies.

Pediatric malignant salivary gland tumors are extremely rare. The percentage of malignant tumors is higher than that seen in adults, although the outcomes in terms of survival are better in pediatric patients. The mainstay of treatment is surgical excision with negative margins. This article reviews current concepts in demographics, etiology, management, and outcomes of malignant salivary tumors in children.

Oropharyngeal cancer as a driver of racial outcome disparities in squamous cell carcinoma of the head and neck: 10-year experience at the University of Maryland Greenebaum Cancer Center.

BACKGROUND: Racial outcome disparities have been observed in head and neck squamous cell carcinoma (HNSCC) with diminished survival for black patients compared with white patients. METHODS: We retrospectively analyzed 1318 patients with primary HNSCC treated at the University of Maryland Greenebaum Cancer Center (UMGCC) from 2000 to 2010. RESULTS: Of all the patients, 65.9% were white, 30.7% were black, and 3.3% were of other races. Black patients were less likely to present with oral cavity cancer, and more likely to present with laryngeal or hypopharyngeal cancers. White patients were more likely to have early stage disease, especially in the oral cavity. Black race was independently associated with worse overall survival (OS) in the entire cohort. Black patients had a significantly worse OS among oral cavity and oropharyngeal cancers, with the largest disparity in oropharyngeal cancer. However, in multivariate analysis, race was only still significant in oropharyngeal cancer. CONCLUSION: We observed differences by race in distribution of disease site, stage, and OS. Survival disparity in the entire cohort was driven mostly by differences among oropharyngeal cancer.

Cystic Lesions of the Jaw in Children: A 15-Year Experience.

IMPORTANCE: Pediatric jaw cysts represent a rarely symptomatic clinical entity and are not well addressed in the otolaryngology literature. It is important that otolaryngologists should be familiar with these lesions, which can manifest as jaw swelling or as paranasal sinus abnormalities. OBJECTIVE: To review the clinical presentation, radiologic features, management, and outcomes of jaw cysts in children treated at a single academic institution. DESIGN, SETTING, AND PARTICIPANTS: Retrospective medical record review at a tertiary care children's hospital among patients 16 years and younger who were seen with a cystic jaw lesion. MAIN OUTCOMES AND MEASURES: Medical records were reviewed for data on symptoms, physical examination findings, imaging, pathology, interventions, and outcomes among children who were seen at the Department of Oral and Maxillofacial Surgery or the Department of Otorhinolaryngology-Head and Neck Surgery at the University of Maryland Medical Center between January 1997 and December 2012 and were diagnosed as having a jaw cyst. RESULTS: Fifty-seven patients were identified who were diagnosed as having a true cystic jaw lesion and whose complete medical records were available for review. The most common cystic lesions were keratocystic odontogenic tumors (n = 19) and dentigerous cysts (n = 17). Fifty-six percent (32 of 57) of all cystic lesions were asymptomatic on presentation and were identified by imaging. The second most common presentation was local swelling (n = 15), followed by dental irregularities (n = 6). All patients, except for 1 with an eruption cyst, required surgical intervention, including biopsy, enucleation, curettage, or ostectomy, with reconstruction as indicated. Keratocystic odontogenic tumors tended to require more treatment (median, 2 procedures) for metachronous lesions or recurrence. CONCLUSIONS AND RELEVANCE: Pediatric jaw cysts are unusual, and data are scarce regarding their presentation and management. Many of these cysts are asymptomatic and are identified incidentally on orthopantomography. Keratocystic odontogenic tumors were the most common lesion seen in our series, followed by dentigerous cysts. Surgical intervention is required in most patients with a cystic lesion of the jaw.

Keratocystic Odontogenic Tumor (KCOT/OKC)-Clinical Guidelines for Resection.

PURPOSE: There is no clinical consensus for the treatment of Keratocystic Odontogenic Tumor (KCOT). KCOTs are regarded as benign aggressive tumors and resection is usually considered as a last option. We review the clinical indications for resection based on a case series. METHODS: This is a retrospective study of patients with KCOT treated in a single unit over 17 years. Eighty patients were identified, of which 12 (15 %) underwent resection. The remaining 68 patients were managed by enucleation and curettage, enucleation with peripheral ostectomy, or decompression with secondary enucleation. Data extracted includes gender, age, race, location, previous treatment for the lesion, surgery and outcome/follow up. RESULTS: Twelve patients treated by resection were identified. The location in the ten benign resected KCOTs was either the mandibular ramus or the posterior maxilla. All mandibular KCOTs exhibited perforation of the lingual plate and involvement of the pterygoid musculature. Seven of the ten cases were recurrent KCOTs and three had no prior treatment. Two had malignant changes in the KCOT and were also resected. CONCLUSION: The primary reason for resection of KCOT was involvement of the pterygoid muscles. The presence of malignant change was a separate indication for resection.