Primary epithelial salivary gland tumours in children and adolescents

Primary epithelial tumours of the salivary glands are very rare in paediatric patients. The aim of this study was to evaluate the clinical course, treatment, and outcomes of these uncommon neoplasms based on the authors’ experience and the recent literature. The medical charts of 12 female patients and seven male patients with primary epithelial salivary gland tumours were reviewed. All were under 19 years of age and underwent surgical treatment between 1994 and 2016. The results of this group of paediatric patients were compared with those of 621 adult patients. The two most common tumours in the paediatric patients were pleomorphic adenoma and mucoepidermoid carcinoma (89.4%; P = 0.004). The incidence of facial nerve palsy following surgery of the parotid tumours was similar in the two groups (P = 1.000). The most common primary cancer in the paediatric group was mucoepidermoid carcinoma (77.8%), while in the adult group, adenoid cystic carcinoma was most common (P < 0.001). The paediatric group had only low-grade cancers in early stages (P < 0.001), with an overall 5-year survival rate of 100%. These results show that the incidence of malignant salivary gland tumours is higher in paediatric patients than in adult patients. This should be taken into account during diagnosis and therapy.     

Salvage surgery for a locally persistent or recurrent tumour in maxillary cancer patients who have undergone radiotherapy and concomitant intra-arterial cisplatin: implications for surgical margin assessment

Limited information about salvage surgery is available for locally persistent and recurrent maxillary sinus cancers after the completion of chemoradiation therapy. Seventy-six maxillary sinus cancer patients who had undergone chemoradioselection using initial radiotherapy and concomitant intra-arterial cisplatin were screened retrospectively. Twenty-four of these patients who had a locally persistent or recurrent tumour were investigated. The 2-year overall survival rate of patients with maxillary sinus cancer of all types was 39.0% for those who underwent salvage surgery and 10.0% for those who did not. The 2-year overall survival rate of patients with maxillary sinus squamous cell carcinoma was 45.8% for those who underwent salvage surgery and 11.1% for those who did not. Furthermore, the 2-year local control and overall survival rates of patients with positive and negative surgical margins were 14.3% and 83.3% and 14.3% and 66.7%, respectively. There were significant differences in local control (P = 0.004) and overall survival (P = 0.005) regarding surgical margin status. Although salvage surgery for a locally persistent or recurrent maxillary sinus cancer is a feasible treatment, patients with positive surgical margins are more prone to local relapse. Therefore, surgical safety margins should be assessed thoroughly.     

Margins and survival in oral cancer

In the surgical management of oral squamous cell carcinoma (SCC) we aim to resect the tumour with clear margins in all planes. The aim of this study was to identify and compare overall survival in a group of 591 patients who had resections, and to relate this to the clearance of margins at the tumour bed. We used life tables to calculate survival at one, two, three, five, and 10 years after diagnosis by margin (clear = 5 mm or more; close = 2–5 mm; and involved = less than 2 mm). Kaplan–Meier curves were produced for the margins alone, which were defined as clear in 480 patients (81%), close in 63 (11%), and involved in 48 (8%). Five-year survival was 81%, 75%, and 54% for clear, close, and involved margins, respectively, which highlights the importance of clear margins for survival. There is a significant prognostic implication associated with close, and particularly with involved, margins.     

Risk factors and prognosis for the primary intraosseous carcinoma of the jaw

Primary intraosseous carcinoma (PIOC) is a rare but aggressive type of odontogenic tumour arising within the jawbone. Diagnosis criteria and treatment strategy remain difficult and controversial. The present study aimed to clarify the clinicopathological features and determine prognostic factors in management of PIOC. A retrospective study of 30 patients with PIOC, treated at the Hospital of Stomatology of Sun Yat-sen University between 2009 and 2017, was conducted. Clinical, histopathological and treatment modality data were collected. Follow-up data were recorded to determine prognostic factors. There were 19 males and 11 females with a mean age of 52.3 years. The most common location of the tumour was the mandible (90%). Having a history of tooth extraction or tooth mobility was the major characteristic symptom (63.3%), jaw swelling coming in second (53.3%). Half of the patients underwent surgery alone. The estimated 2-year overall survival rate (OS) and recurrence-free survival rate (RFS) were 61.3% and 40.1%, respectively. Higher histological grade was an independent risk factor for poor OS (hazard ratio (HR) 0.233 [0.059–0.915], P = 0.037), while at pN₊ stage for RFS, HR = 5.627 [1.199–26.409], P = 0.029. Because of its rarity and intrabony site, the classification, staging and treatment guidelines for PIOC should be further studied and established.     

Prognostic factors in primary salivary gland mucoepidermoid carcinoma: an analysis of 376 cases in an Eastern Chinese population

Mucoepidermoid carcinoma (MEC) is an infrequent malignant neoplasm that originates most commonly in the salivary glands. The present study aimed to provide new information on prognostic factors in patients with salivary gland MEC. A retrospective analysis of the medical records of patients diagnosed with primary salivary gland MEC between 2003 and 2010 was conducted. The incidence of MEC in the minor salivary glands (62.2%) was almost twice that in the major salivary glands (37.8%). The most frequently affected sites were the parotid gland and palate. Lymph node metastasis was reported more frequently in male than female patients (P = 0.02), in high-grade than low/intermediate grade lesions (P < 0.001), and in lesions involving the submandibular gland (P < 0.001). The disease-free survival (DFS) at 5 years was 80.47%, with rates of 98.0%, 86.5%, and 38.5% for low-, intermediate-, and high-grade tumours, respectively. Among various clinicopathological factors, the only independent prognostic factor was histological grade (P < 0.001). Primary tumour site and histological grade are two important factors affecting cervical lymph node metastasis. Histological grade is the only independent factor affecting survival beyond tumor lymph node metastasis (TNM) staging in salivary gland MEC. Further advances in therapy are needed to improve the outcomes for patients with high-grade lesions.     

Warthin’s tumour in oral and maxillofacial regions: an 18-year retrospective study of 1084 cases in an eastern-Chinese population

There is little information in the English-language literature regarding Warthin’s tumour (WT) in the eastern-Chinese population. A large retrospective study (1084 primary tumours over a period of 18 years) was carried out to investigate the clinicopathological features (patients’ gender, age and tumour location) of these tumours in this population. A total of 994 (91.7%) patients were male and 90 (8.3%) were female, with a male/female ratio of 11:1. The mean age was 56.48 years (range 20–89 years), with a peak incidence in the fifth to seventh decade (82.1%). The favorite primary site of the tumour was the parotid gland (n = 1055), followed by intra-/peri-parotid lymph nodes (n = 13), upper neck (n = 10), submandibular gland (n = 4) and upper lip (n = 1). Multifocal WTs arose in 9.5% (103 patients) of cases whereas bilateral multifocal WTs were found in 0.65% (seven patients). In 24 (2.2%) patients, WT were found to coexist with other different types of neoplasm synchronously. The most common subtype of metaplasia was the squamous metaplasia (166/250, 66.4%). The usual treatment measure is (bilateral) superficial parotidectomy and the patients should be followed long term, in view of possible metachronous WT, even after prolonged time intervals.     

Tumor PD-L1 expression is associated with improved survival and lower recurrence risk in young women with oral cavity squamous cell carcinoma

Young patients with oral cavity squamous cell carcinoma (OCSCC) are often recognized as a distinct epidemiological cohort. In this study, genomic and immune-based metrics were correlated with long-term outcomes for a young patient population treated at a single institution. A fully clinically annotated, retrospective cohort of 81 patients aged ≤45 years with OCSCC is described, and the impact of clinicopathological features on long-term survival outcomes is reported. Genomic and immune parameters were integrated utilizing a whole-exome sequencing and immunohistochemical approach among females in the cohort. It was found that young OCSCC patients had favorable outcomes (10-year disease-free survival 79.1%, overall survival 80.0%) regardless of sex, particularly if they presented with oral tongue primaries and early stage disease. While mutational analysis appeared similar to that of older patients with OCSCC who lack a smoking history, a comparatively high degree of PD-L1 expression and PD-1/L1 concordance (P = 0.001) was found among young female OCSCC patients. Subjects with greater membranous PD-L1 positivity and the presence of tumor-infiltrating lymphocytes had a decreased risk of recurrence (P = 0.01 and P = 0.01, respectively) and improved survival (P = 0.04 and P = 0.03, respectively). These findings warrant further validation and support the investigation of immunotherapeutic approaches targeting PD-1/L1 interactions in young OCSCC patients.     

Determinants of level Ib involvement in oral squamous cell carcinoma and implications for submandibular gland-sparing neck dissection

Traditional neck dissection for oral squamous cell carcinoma (OSCC) involves removal of the submandibular salivary gland. Several studies have cited the low incidence of direct gland invasion by tumours and have recommended gland-sparing neck dissection. In this study, a detailed audit of level Ib involvement in OSCC was performed in order to assess the feasibility of submandibular gland-sparing in neck dissection; the rate of direct involvement by the primary tumours, the involvement of periglandular level Ib nodes, and their determinants were investigated. A total of 586 neck dissection specimens obtained between 2005 and 2014 from patients operated on at the study institution for floor of mouth, tongue, and buccal primaries, were evaluated for direct invasion of the gland and periglandular lymphadenopathy. Of 226 node-positive patients, 21 (9.3%) had direct gland invasion by tumour. Risk factors were tumour diameter >4 cm (P = 0.002) and depth of invasion >10 mm (P = 0.003). Determinants of periglandular lymphadenopathy were depth of invasion >10 mm (P < 0.001), perineural invasion (P = 0.02), lymphovascular invasion (P = 0.014), and moderate/poor differentiation (P < 0.0001). Gland-sparing neck dissection is safe in early tumours (pT1pN0–1), with a good chance of minimizing xerostomia without radiotherapy. Larger tumours without clear evidence of submandibular gland invasion or suspicious level Ib lymphadenopathy may be considered for gland preservation, however the oncological safety is unclear.     

Primary squamous cell carcinoma of the parotid gland: clinicopathological characteristics,treatment, and prognosis

Squamous cell carcinoma (SCC) of the parotid gland is a rare aggressive malignancy with a poor prognosis. The clinical behaviour, histopathological characteristics, and treatment strategies for parotid SCC still need to be comprehensively demonstrated. In this study, a retrospective review of patients diagnosed with parotid SCC was performed, covering the past two decades. Twenty-nine patients with primary parotid SCC and 10 patients with recurrent parotid SCC were identified. The clinicopathological characteristics of parotid SCC were summarized. Imaging records were used to determine the extent of invasion of the parotid SCC. Histopathological alterations in the parotid resulting from the infiltration of SCC were demonstrated. A set of treatment strategies was developed, involving parotidectomy, neck dissection, facial nerve treatment, defect repair, adjuvant radiotherapy, and chemotherapy/targeted therapy. The median patient survival was 24 months for those with primary parotid SCC and 14.5 months for those with recurrent parotid SCC. Comparatively, patients with a larger tumour size of primary parotid SCC experienced poorer overall survival (hazard ratio 8.986; P = 0.013). Great efforts have been made over the past two decades to identify and treat parotid SCC. Consensus regarding therapeutic options for parotid SCC has not been widely achieved and there is still a great need for well-designed prospective studies.     

Prognostic value of elective neck dissection in adenoid cystic carcinoma of head and neck: a meta-analysis: A call for randomized trials and international consensus

Adenoid cystic carcinoma of head and neck (AdCCHN) is an uncommon salivary gland cancer characterized for infrequent neck metastases, and high rate of local and distant recurrence. The aim of this meta-analysis was to analyse the significance of elective neck dissection (END) in terms of overall survival (OS) in patients with AdCCHN. A systematic literature search and meta-analysis was performed. Endpoint assessed by this meta-analysis included 5-year OS (death from any cause). Statistical heterogeneity was assessed using the Cochrane Q test and I² statistic. A pooled odds ratio (OR) was reported with 95% confidence interval (CI). There were 1934 patients in the END arm and 3083 in the observation group. The pooled OR, calculated for END vs. observation, was 0.94. Patients receiving END had similar risk for death compared to observation cohort (P = 0.76). No significant difference in final outcome after patient stratification based on T stage was identified (OR for T1/T2 1.27, P = 0.39; OR for T3/T4 0.95, P = 0.90). Observation for cN0 neck is a reasonable option in AdCCHN. These findings suggest the need for prospective trials on indications and extent of END in AdCCHN.     

Clinical and oncological outcomes after surgical excision of parotid gland tumours in patients aged over 80 years

The objective of this study was to evaluate the surgical and long-term outcomes of a series of patients aged over 80 years, operated on for parotid neoplasms. Among 614 parotidectomies for neoplasms performed between 1998 and 2008, 34 patients (5.5%) aged over 80 years were identified retrospectively. Pathological examination showed a malignant tumour in 24 and a benign tumour in 10 cases. Overall survival (OS) and disease-free survival (DFS) were determined by Kaplan–Meier analysis. A search for parameters that could influence the postoperative complication rate and long-term outcomes was carried out by univariate analysis. There was no postoperative death. Eight patients (24%) had postoperative complications. Malignant histopathology (P = 0.05) and radical resection (P = 0.033) were found to have a significant negative impact on the postoperative course. Focusing on malignant tumours, only histopathological type (metastasis vs primary tumour) was found to have a negative impact on OS. The 2- and 5-year OS rates were 86% and 86%, respectively, for primary tumours, and 67% and 29%, respectively, for metastasis (P = 0.05). Malignant or benign histopathology had no impact on OS. Our results showed acceptable clinical and long-term oncological outcomes in very elderly patients operated on for parotid tumours, including malignant tumours.     

Retention of lipiodol after parotid gland sialography

There is limited information about the retention of lipiodol in the parotid gland after parotid gland sialography. This study assesses the prevalence of lipiodol retention after parotid sialography and determines if retention of lipiodol is related to the sialography technique or the underlying salivary gland pathology. Using the electronic hospital database (1996–2006), 66 out of 565 patients were identified who had additional maxillofacial radiographic examinations after the initial sialography. Additional radiographs up to October 2007 were included; these were orthopantomographic radiographs in all cases. In 28 patients (42%) signs of lipiodol retention were observed (mean radiographic follow-up: 15 ± 13 months). Retention was characterized by small radiopaque spots in the periphery of the gland. Lipiodol retention was predominantly associated with a fausse route (n = 8) or the presence of salivary gland disease (sialectasia; n = 17). In 9 patients with signs of lipiodol retention, a series of radiographs was available. Lipiodol radiodensities decreased in size during 28 months, and could disappear gradually (follow-up 14–57 months). Despite the high frequency of retention of small depots of lipiodol for years after sialography in patients subjected to additional radiographic examinations, no clinically adverse effects were observed.     

Costochondral grafting for paediatric temporomandibular joint reconstruction: 10-year outcomes in 55 cases

Costochondral grafting (CCG) can be used for the reconstruction of ankylotic, hypoplastic, and resected temporomandibular joint (TMJ) defects. CCGs have previously been considered the gold standard in children due to their growth potential and autogenous origin, but the disadvantages are unpredictable growth and joint ankylosis. This was a retrospective study of all children who received CCGs for TMJ reconstruction from 1985 to 2004, to allow a 10-year follow-up. Fifty-five patients were included in this study, with 74 grafts being placed; their mean age was 7.9 ± 4.2 years. Infection-related ankylosis (18.2%) and craniofacial microsomia (16.4%) were the most common diagnoses. Overall, 58.2% of patients suffered one or more complications over the follow-up period, with ankylosis (32.7%) and overgrowth (16.4%) being most common. There was a significant correlation between those with infection-related ankylosis and subsequent complications (χ² = 8.8, df = 1, P < 0.005), while ankylotic patients in general exhibited greater overall complication rates (χ² = 9.0, df = 1, P < 0.005). Patients with congenital TMJ defects were more likely to be complication-free than those with acquired defects (χ² = 4.0, df = 1, P < 0.05). Caution is advised when placing CCGs in paediatric patients with ankylosed TMJs, especially those with infection-related ankylosis.     

Comparison of the eighth edition of the TNM and Brigham Women’s Hospital staging systems for cutaneous squamous cell carcinoma of the head and neck: a six-year review

This study was undertaken with the aim to compare the T stages in a series of cutaneous squamous cell carcinoma (cSCC) patients using both the eighth edition of the Union for International Cancer Control (UICC) TNM Classification of Malignant Tumours (TNM8) and Brigham and Women’s Hospital (BWH) staging systems. This would allow comparison of the two to determine suitability with regards to T stage and the effect on local recurrence and nodal disease. This was a six-year retrospective cohort study of patients with primary invasive cSCC of the head and neck who were diagnosed and treated at Western Sussex Hospitals Trust in the United Kingdom between 2007 and 2012. The TNM8 and BWH staging systems were applied to these primary cSCCs. A total of 695 invasive cSCCs treated in 604 patients over six years were identified. Most patients were male (76%), with a mean (range) age of 81 (50-103) years. The most common location for local recurrence was the scalp (n = 26, 58%). Regional metastasis occurred most commonly in the parotid gland (n = 20, 63%). All tumours were classified using both staging systems. Specifically, 432 tumours remained in the same T stage (61%), and 192 were downstaged (27%) and 71 upstaged using the BWH (10%). The median (SD) follow-up time was 23 (28) months (range 1-123). The BWH alternative staging system overlapped with the TNM8 in high-stage and low-stage tumour assignment. The highest percentage of local recurrence and regional metastasis occurred in T2b tumours.     

Long-term outcomes of craniofacial implants for the restoration of facial defects

The aim of this study was to evaluate the long-term survival of craniofacial implants and prostheses and to identify factors associated with failure in a cohort of patients. A 25-year retrospective analysis was conducted at Royal Melbourne Hospital. Data included demographic characteristics, age, site and cause of the deformity, and number and survival of implants. Odds ratios were calculated and event-to-time Kaplan–Meier analyses performed. One hundred and ten patients were included (341 implants); their mean age was 46.2 years. The overall implant survival rate was 79.5% (mean follow-up 10.6 years). Temporal implants had the highest success rate (97.0%), followed by nasal implants (87.5%) and orbital implants (63.3%); differences were statistically significant (P < 0.0001 and P = 0.033, respectively). Kaplan–Meier analyses to determine long-term implant and prosthesis survival found temporal implants had the highest prosthetic (P < 0.0001) and implant survival (P < 0.0001). Patients with congenital deformities demonstrated the highest success rate. Radiotherapy was found to increase the risk of implant failure (P = 0.02). Craniofacial implant-retained prostheses are a reliable and effective option for the restoration of facial defects, with good long-term success rates. Orbital implants and those placed post oncological surgery have a higher failure rate.     

Prognosis of oral cancer: a comparison of the staging systems given in the 7th and 8th editions of the American Joint Committee on Cancer Staging Manual

The 8th edition of the American Joint Committee on Cancer (AJCC) Staging Manual introduces “depth of invasion” and “extranodal extension” into the head and neck section, and our aim was to find out if these changes have an impact on prognosis. We evaluated 174 patients who had had oral squamous cell carcinomas (SCC) resected between 2003 and 2012. The clinical records were reviewed, the patients’ tumours restaged according to the 8th edition of the AJCC, and we analysed five-year survival to verify whether different correlations were made between the T and N stages and disease-specific survival using the 7th and 8th editions. We excluded seven cases because information was incomplete, and the final sample was 167 patients. The five-year overall survival was 68% and the five-year disease-specific survival was 78%. The variable pT was upstaged in 51 patients (31%), and no tumour was downstaged. When we used the 7th edition, the pT category did not correlate with survival (p = 0.055), but when we used the 8th edition, there was a significant association between increased pT categories and disease-specific survival (p = 0.01). In the pN category 23 cases were upstaged (14%) and this affected disease-specific survival using both the 7th and the 8th editions (p = 0.001). When patients were restaged, there was an improvement in discrimination between T categories in relation to disease-specific survival, and confirmation of the prognostic impact of the variable pN. T stage and depth of invasion are complementary predictors of disease-specific survival, and their combination results in the new AJCC staging system giving a better prognosis.     

Risk factors associated with post-loading implant loss of removable and fixed implant-supported prostheses in edentulous jaws

Purpose

This study analyzed risk factors for post-loading implant loss in cases of implant-supported prostheses applied to edentulous jaws of Japanese patients.

Prognostic value of lymph node count from selective neck dissection in oral squamous cell carcinoma

Unlike the levels of anatomical exploration, there is no consensus on the extent of lymph node dissection, or lymph node count (LNC), during selective neck dissection (SND). The aim of this study was to validate the prognostic impact of LNC on survival and to determine an optimal LNC cut-off value for SND. A retrospective investigation identified 78 patients with a diagnosis of oral squamous cell carcinoma (OSCC) who underwent SND (levels I–III or levels I–IV). LNC and clinicopathological variables were analyzed for any association with survival in Cox proportional hazards models. Based on the receiver operating characteristic curve, a cut-off value of 19 lymph nodes was found to predict overall survival (OS) (area under the curve 0.732, sensitivity 67.8%, specificity 75.0%; P = 0.026) and disease-specific survival (DSS) (area under the curve 0.762, sensitivity 68.1%, specificity 77.8%; P = 0.011). On Cox regression, LNC (≥19 vs. <19) was the only independent predictor of OS (hazard ratio 5.29, 95% confidence interval 1.39–20.05; P = 0.014) and DSS (hazard ratio 6.76, 95% confidence interval 1.40–32.77; P = 0.018). Similar results were obtained in the pathologically lymph node-negative subgroup (n = 66). Based on the study findings, SND should include 19 or more lymph nodes for a survival benefit.     

Multiple calcifications within the parotid gland of patients with Sjögren's syndrome

PurposeThe purpose of this study was to investigate computed tomography (CT) and clinical features relating to calcifications within the parotid gland of patients with Sjögren's syndrome (SS).MethodsData from 30 patients with SS who had been examined by CT were extracted from our radiological information database accumulated from 2001 to 2011, and their CT images were reread carefully. Of these patients, 14 (all female; age range 20–95 years; mean age 61.4 years) with calcifications within the parotid gland were retrospectively investigated with CT findings. The relationship between calcification occurrence and clinical symptoms including parotid swelling and/or saliva colic was investigated. The degree of destruction of the parotid gland on CT images was also evaluated.ResultsAll calcifications of 14 patients were located within the parotid gland, not in the parotid duct. CT images of all calcifications showed small and regular round shapes. Multiple occurrences of calcifications were recognized in 10 patients, and a solitary occurrence was seen in 4 patients. Seven patients had bilateral calcifications. There was little relationship between the occurrence of calcifications and clinical symptoms, and the severity of destruction of the parotid gland.ConclusionThe presented CT and clinical features would be peculiar to SS because too many patients lacked the typical features of sialoliths within the parotid gland.     

Significance of clinical stage,extent of surgery and outcome in cutaneous squamous cell carcinoma of the head and neck

The authors analyzed a new clinical staging system and its correlation with pathologic findings and patient survival. Patients were eligible for inclusion in this longitudinal retrospective cohort study if they had cutaneous squamous cell carcinoma on the head or neck, underwent surgery and had a minimum 3 year follow-up. The primary study variable was using a new clinical staging system. Secondary variables included the parotid as a predictor of metastatic spread to the lymphatic nodes in the neck and primary lesion histopathologic traits. The outcome variable was patient survival. Associations between variables were assessed using Fisher's exact test, Mann–Whitney test, Kaplan–Meier method and Mantel log-rank test. p < 0.05 was considered significant. The sample comprised 103 patients. Regional metastatic disease was found in 24 patients. Histopathological analysis showed a higher frequency of neck metastatic disease if the parotid was positive for metastases (p = 0.022). An extended staging system showed significant correlation between survival rate and substages (p = 0.0105). Perineural invasion was a negative prognostic factor (p = 0.0151). The results of this study suggest that combining curative parotidectomy and elective neck dissection could be beneficial in high risk patients. Both neck and parotid metastases should be included in the clinical and histological N classification.