Ivica Lukšić

Salivary gland tumours: 25 years of experience from a single institution in Croatia

INTRODUCTION The aim of this study was to determine the types, frequency, distribution, and demographic characteristics of salivary gland tumours in a large representative sample. PATIENTS AND METHODS We retrospectively analysed the medical records of 779 patients with tumours of the salivary glands surgically treated from 1985 to 2009 at a single institution. RESULTS There were 500 benign and 279 malignant tumours. The average age of patients with benign tumours was 50 years and of malignant salivary gland tumours 56 years. No differences in age and incidence of tumours existed between males and females. The majority of the tumours occurred in the parotid gland (509), followed by the minor salivary glands (212), the submandibular gland (51) and lastly, the sublingual gland (7). Minor salivary gland tumours occurred most frequently on the palate, the pleomorphic adenoma being the most frequent benign tumour type and the adenoid cystic carcinoma being the commonest malignant tumour. Tumours of the sublingual gland were rare, but all were malignant. Malignant tumours were more common in the minor salivary glands and the submandibular gland. CONCLUSION This large study of salivary gland tumours in Croatia could improve our understanding of the significant differences in the global distribution of salivary gland tumours which have been reported.

Schwannoma of the tongue in a child

A schwannoma or neurilemmoma is a benign, slow growing, usually solitary and encapsulated tumour originating from Schwann cells of the nerve sheath. Approximately 25-40% of all schwannomas are seen in the soft tissues of the head and neck, often originate from the acoustic nerve. Intraoral schwannomas are rare and account for 1% of schwannomas of the head and neck region. We report the case of a 10-year-old boy diagnosed with a schwannoma of the tongue. The purpose of this report is to emphasize the possibility of diagnosing schwannoma among all other lingual lesions in children. The disease itself was diagnosed histologically after complete surgical excision. Five years after surgical treatment, the patient is without signs of recurrence. This paper highlights the importance of a multidisciplinary approach in the diagnosis and surgical treatment of this very rare entity.

Melanotic neuroectodermal tumour of infancy: Report of two cases and review of the literature

Melanotic neuroectodermal tumour of infancy (MNTI) is an uncommon tumour affecting predominantly the craniofacial bones of the newborn infants. The neural crest origin of the tumour has been confirmed. MNTI is generally accepted as a benign tumour despite of its rapid and locally infiltrative growth. Recurrence rate varies between 10% and 60%, and malignant behaviour has been reported in 6.5% of MNTIs. Systematic review of the literature revealed 445 MNTIs published between 1918 and 2010. We present additional two cases of MNTI from our Department, typical in all terms, which equals a total number of 447 reported cases. One of our cases revealed histological features consistent with malignant behaviour, but at present, 18 months after the surgical excision, there is no evidence of recurrence. Biological behaviour of MNTI cannot be predicted by gross or histologic characteristics, thus early diagnosis and careful follow-up after the complete surgical excision is required.

Lymphatic drainage patterns of head and neck cutaneous melanoma: does primary melanoma site correlate with anatomic distribution of pathologically involved lymph nodes?

The aim of this study was to analyse patterns of metastatic spread from cutaneous head and neck melanoma, which are said to be highly variable. The medical records of 145 patients with pathologically proven metastatic melanoma were reviewed retrospectively. The location of pathologically positive lymph nodes was compared with clinically predicted spread, and patients with metastatic disease in areas outside of predicted drainage patterns were considered aberrant. There were 33 curative and 73 elective neck dissections. 21 of 77 patients undergoing parotidectomy had positive results for metastases. Clinical prediction proved to be correct in 33 of 45 cases (73.3%). Two patients with lateralized melanomas were initially seen with contralateral metastases. Six of 45 patients (13.3%) developed contralateral metastases after neck dissection. Patients with clinical involvement of the parotid gland were at high risk of occult neck disease (40%). Patients undergoing neck dissection for primaries originating in face, forehead, coronal scalp, periauricular area, and upper neck should be considered for parotidectomy. Patients with posterior scalp and posterior neck primaries should be considered for selective neck dissection in conjunction with posterior lymphadenectomy. In patients with coronal scalp and periauricular primaries, a complete neck dissection including parotidectomy is the recommended approach.

Primary synovial sarcoma of the parotid gland in 15-year-old boy

Synovial sarcoma (SS) is a malignant mesenchymal tumour, predominantly found in the deep soft tissues of lower extremities, whereas only 3% occur in the head and neck region. Primary synovial sarcoma of the parotid gland is exceptionally uncommon. This is a report of a 15-year-old boy with a synovial sarcoma arising in the parotid gland, and, to the best of our knowledge, this is the youngest patient on record. The patient was treated primarily surgically, followed by chemotherapy and radiotherapy. Two years after this multimodal therapy, the patient is without signs of loco-regional recurrence or distant metastases. This paper highlights the importance of a multidisciplinary approach in the diagnosis and treatment of this very rare entity.

Intraoral adenoid cystic carcinoma: is the presence of perineural invasion associated with the size of the primary tumour, local extension, surgical margins, distant metastases, and outcome?

Adenoid cystic carcinoma is the most common malignancy of the minor salivary glands, and its biological behaviour is characterised by slow and indolent growth; rare involvement of regional lymph nodes; a high propensity for perineural invasion; multiple or delayed recurrences, or both; and a high incidence of distant metastases. Our aim was to find out the relation between the presence of perineural invasion and these factors. Between 1 January 1984 and 1 May 2008, 26 cases of adenoid cystic carcinoma of the intraoral salivary glands, which had initially been treated surgically, were reviewed retrospectively. The most common site was the palate, and perineural invasion was reported in 13 of the 26 resected specimens. There was no significant association between it and the size of the primary tumour (OR=1.0; p=1.00), invasion of the surgical margins (OR=2.08; p=0.4), the presence of distant metastases (OR=3.43; p=0.197), or local control (p=0.76). It was exclusively present in patients with local extension, and was significantly associated with outcome (p=0.04). Resection with clear margins is the gold standard of care for patients with intraoral adenoid cystic carcinoma, and the role of adjuvant irradiation remains controversial. Given its paradoxical and complex biological behaviour, large studies with long term follow-up are needed to define the clinicopathological and immunohistochemical variables associated with outcome, as well as the optimal treatment.

Oral inverted ductal papilloma

Anoralpapillomaisabenignsurfacetumourthatcan present anywhere in the oral cavity. Micro-scopically,itisanexophytic,finger-likegrowthofstratifiedsquamousepitheliumonathinfibrovas-cular core of loose connective tissue. Papillomaswithahistologicalendophyticorinvertedpatternof growth are rare. Benign papillary lesions thatseem to originate from the salivary ductal sys-temincludeintraductalpapilloma,invertedductalpapilloma, and sialadenoma papilliferum. Previ-ously,theyhavebeenincludedinthecategoriesofmonomorphicadenomaorductaladenoma.

Significance of myofibroblast appearance in squamous cell carcinoma of the oral cavity on the occurrence of occult regional metastases, distant metastases, and survival

The aim of the present study was to assess the frequency of appearance of stromal myofibroblasts in patients with oral squamous cell carcinoma (OSCC) and to further clarify whether myofibroblasts influence tumour suppression or progression. Surgical resection specimens from 152 patients with cT1-T3N0 OSCC were analysed. The frequency of myofibroblasts within the tumour stroma was assessed immunohistochemically and compared with other clinical and histopathological factors. The immunohistochemical reaction for alpha-smooth muscle actin showed positive cells in the stroma of 84.2% of OSCC (n=128). An increased presence of myofibroblasts in the tumour stroma was significantly correlated with T stage (P=0.019), the presence of occult neck metastasis (P<0.001), regional recurrence (P=0.037), and distant metastasis (P=0.008). There was also an association between the presence of myofibroblasts and patient survival (P=0.009). The presence of myofibroblasts was not associated with local recurrence, tumour cell differentiation, mode of invasion, or bone invasion. The results of this study suggest that myofibroblast proliferation facilitates tumour invasion, the occurrence of occult neck disease, and distant metastasis. The survival rate was poorer in patients with abundant myofibroblasts. Further investigations on tumour-associated stroma at the invasive front are needed in order to establish new diagnostic markers and therapeutic strategies.

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 Fishers 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.

Prognostic significance of extracapsular spread of lymph node metastasis from oral squamous cell carcinoma in the clinically negative neck

The presence of extracapsular spread (ECS) in patients with oral squamous cell carcinoma (OSCC) indicates a poor prognosis and is associated with a higher risk of regional recurrence and distant metastasis. The aim of this study was to analyse this important feature of cervical lymph nodes in the clinically node-negative setting. The study included 61 patients with clinically T1-T3N0 OSCC who underwent primary surgical treatment; 52 were male and nine were female, and their median age was 57 years. The 5-year disease-free survival, disease-specific survival, and overall survival rates were 30.6%, 28.3%, and 14.3%, respectively, in the ECS group compared to 61.9%, 61.9%, and 48.2%, respectively, in the pN+/ECS-negative group and 76.7%, 81.9%, and 47.0%, respectively, in the pN0 group. The differences between the survival curves were highly significant (P=0.023, P=0.003, and P=0.029, respectively). The incidence of local (50% vs. 14.9%, P=0.011) and regional (28.6% vs. 2.1%, P=0.008) recurrence was significantly greater in the ECS group compared to the other subgroups of patients. Furthermore, the time to recurrence was significantly shorter in the ECS subjects. The presence of ECS in patients with oral cancer indicates a poor prognosis. ECS is a frequent feature in clinically node-negative settings and may be more common in smaller lymph nodes than is generally appreciated.