Hyalinizing clear cell carcinoma of the sublingual gland: A case report and literature review.

RATIONALE: Hyalinizing clear cell carcinoma (HCCC) of the salivary glands is a rare low-grade malignant tumor. This type of tumor is particularly uncommon in the sublingual glands. PATIENT CONCERNS: A 57-year-old female with a mass on the left side of the floor of the mouth that had been present for 2 months. The computed tomography scan of the neck revealed a nodular abnormal density shadow in the left sublingual area, measuring approximately 2.6 cm × 1.9 cm. DIAGNOSES: Primary HCCC of the sublingual gland. INTERVENTIONS: The patient underwent surgical treatment and reconstruction using a left anterolateral femoral free flap, which showed immunohistochemical positivity for CK 5/6, CK 7, CK (AE1/AE3), and Ki-67 (<5%), but negative for SMA and S-100. OUTCOMES: No recurrence was observed during the 12-month postoperative follow-up period. LESSONS: The absence of characteristic clinical manifestations makes HCCC highly susceptible to misdiagnoses. This case presents a rare instance of HCCC in the sublingual gland, providing a reference for the clinical diagnosis and treatment of the disease.

Reconstructive strategies following surgical resection of malignant sublingual gland tumors: A single institution experience.

OBJECTIVE: To investigate the characteristics and treatment modalities of malignant tumors originating from the sublingual gland, as well as evaluate the therapeutic outcomes following free flap reconstruction. METHODS: A retrospective statistical analysis was conducted on the clinical data of nine patients diagnosed with malignant neoplasms tumor of the sublingual gland. RESULTS: Nine case of malignant tumors originated from the sublingual glandular tissue, encompassing eight adenoid cystic carcinoma (ACC) and a single case of bipartite differentiated carcinoma-a hybrid of epithelial-myoepithelial carcinoma and adenoid cystic carcinoma. Among the nine patients, four anterolateral thigh flaps were used (three of which were thin flaps), and five forearm flaps were also empoyed. The size of flaps varied, with the lengths ranging from 4 cm to 9 cm, and the widths ranging from 2.5 cm to 6 cm. The vessels chosen for anastomosis were the superior thyroid artery in seven cases, the facial artery in one case, and the lingual artery in one case. Among the eight patients who underwent dissection of cervical lymph nodes, metastasis were found in one case. Two patients underwent adjuvant radiotherapy. Upon postoperative follow-up, there was no recurrence in any of the nine patients . CONCLUSION: The anterolateral thigh perforator flap thinning technique can be employed for postoperative reconstruction of malignant sublingual gland tumors.

Sublingual Gland Carcinoma Revealed by Choroidal Metastasis.

A 65-year-old man presented with a 1-week history of left eye distortion. An elevated choroidal lesion covering 6 disc diameters was found in the posterior retina of the left eye. Systemic examination revealed sublingual gland carcinoma and multiple lung metastases, and the diagnosis was choroidal metastasis from sublingual gland carcinoma. Following chemotherapy and radiation therapy, the choroidal lesion shrunk and the patient's visual acuity improved. The patient died 23 months after his first visit. To the best of our knowledge, this is the first reported case of choroidal metastasis from sublingual gland carcinoma.

Carcinoma Showing Thymus-Like Differentiation (CASTLE) Arising in the Sublingual Gland.

Carcinoma showing thymic-like differentiation (CASTLE) is a rare tumor most commonly occurring in the thyroid and soft tissues of the neck. We report the first case of CASTLE occurring in the sublingual gland. The patient, a 35-year-old healthy man, presented with a submucosal lesion located in the anterior right floor of the oral cavity and an ipsilateral neck mass. The lesion had been previously investigated by neck computed tomography and ultrasound-guided fine needle aspiration cytology and diagnosed as metastatic squamous cell carcinoma. After oral cavity magnetic resonance imaging, positron emission tomography, and a non-diriment, fine needle aspiration cytology of the sublingual mass, the patient was treated as affected by a sublingual gland malignancy with removal of primary tumor and neck dissection. Morphological and immunohistochemical findings were diagnostic for primary sublingual gland CASTLE. The patient received adjuvant radiotherapy and is free of disease 2 years after treatment. We describe the pathological features of the lesion and discuss the possible differential diagnoses.

Malignant extrapleural solitary fibrous tumor arising in the sublingual gland: A case report and review of literature.

Solitary fibrous tumor is an uncommon neoplasm with unpredictable clinical behavior. Malignant solitary fibrous tumor is a rare morphological variant with more aggressive behavior and higher rates of local recurrences and distant metastasis, exceeding rare in oral cavity; our case occurred in the floor of the mouth in the sublingual gland.

The clinical features of epithelioid hemangioendothelioma in a Han Chinese population: A retrospective analysis.

Epithelioid hemangioendothelioma (EHE) is a rare indolent vascular tumor which occurs at liver, lung, bone, and so on. However, the etiology of EHE is evasive.These patients were enrolled at the First Affiliated Hospital of Xi'an Jiaotong University from January 2011 to December 2015. Retrospective analysis is done by demographic data of clinical manifestations, laboratory parameters, CT imaging, histological, and immunohistochemical features in 9 cases in Shaanxi.Of the patients, 8 were females (88.9%) and 1 were males (11.1%). The age ranged from 34 to 71 years (mean 49 years; median 49 years). Anatomical sites of primary lesions were as follows: liver (n = 6, 66.7%), upper extremities (n = 1, 11.1%), sublingual gland (n = 1, 11.1%), and spine (n = 1, 11.1%). Metastatic disease was diagnosed in 5 cases (55.6%) with occurrence in lung (n = 4, 44.4%), bone (n = 2, 22.2%), upper extremities (n = 1, 11.1%), pleura (n = 1, 11.1%), and spleen (n = 1, 11.1%). Tumor size ranged from 0.5 to 6.8 cm (mean 3 cm). The most tumors were composed of highly cellular areas with small and prominent nucleoli in vesicular nuclei, and ERG (100%) was the most frequently positive in these cases, followed by CD31 (88.9%) and CD34 (77.8%) via histology and immunohistochemistry techniques.EHE is a very rare in Shaanxi. It is significant to find its clinical, radiological, and pathological characters, helping for EHE early diagnosis and treatment, reducing misdiagnosis and improving life quality.

Predictors of outcomes in large cell undifferentiated carcinoma of the major salivary glands.

OBJECTIVE: Major salivary gland large-cell undifferentiated carcinoma (LCUC) is rare and has a poor prognosis. Characterization of patient demographics, tumor characteristics, and predictors of outcome have been limited by low case numbers, as well as grouped analysis with other salivary malignancies. The objective of this study was to address these issues using large-scale national data. STUDY DESIGN: Retrospective case series. METHODS: Data from the National Cancer Database, including cases diagnosed from 1998 to 2012, was analyzed, identifying 247 records of LCUC. Tumor, demographic, and survival information was extracted and analyzed retrospectively. RESULTS: Large-cell undifferentiated carcinoma comprised < 1% of all major salivary gland cancers. Seventy percent of patients presented with advanced-stage disease. The incidence of occult nodal disease was 39%. Surgery followed by radiation was the most common treatment. Five-year overall survival was 36%. Comorbidity, distant metastasis, and positive surgical margins were found to be predictors of overall survival. CONCLUSION: To our knowledge, this represents the largest reported case series of LCUC. The survival analysis demonstrates poorer survival in patients with positive surgical margins; therefore, efforts to complete resection are reasonable. Reported high rates of occult nodal disease also strongly support elective treatment of the neck. LEVEL OF EVIDENCE: 4. Laryngoscope, 2016 127:372-376, 2017.

Lymphoepithelial Carcinoma of the Sublingual Gland: Case Report and Review of the Literature.

Lymphoepithelial carcinoma represents only 0.4% of salivary gland neoplasms. Generally affecting the parotid gland, it has been reported only twice in the sublingual gland. Controversies concerning the treatment of lymphoepithelial carcinoma exist. Although the literature generally agrees that primary surgery with adjuvant radiotherapy is part of the treatment, the benefit of adjuvant chemotherapy is not well described. This report describes the case of a 55-year-old man diagnosed with lymphoepithelial carcinoma of the sublingual gland. The patient was admitted for progressive pain in the floor of the mouth associated with trismus. Biopsy examination confirmed the diagnosis of lymphoepithelial carcinoma of the sublingual gland and magnetic resonance imaging showed multiple left lymphadenopathies. Surgery consisted of a radical neck dissection type III, surgical resection of the floor of the mouth, and reconstruction with a left facial artery musculomucosal flap. The patient received adjuvant radiotherapy (60 Gy) and adjuvant chemotherapy (3 cycles of cisplatinum 100 mg/m(2)). The patient was disease free at 36 months of follow-up. The evidence base for administering adjuvant chemotherapy in this situation is discussed.

Tuberculous osteomyelitis of the mandible with diffuse swelling of the floor of the mouth: a case report.

Primary orofacial tuberculosis (TB) is uncommon, especially with regard to the jaw. We report an unusual case for which the final diagnosis was tuberculous osteomyelitis of the mandible with cervical tuberculous lymphadenitis. The follow-up examinations for our patient showed complete regression of the swelling and healing of the mandibular lesion after 4 months of TB antibiotic therapy. The purpose of the present study was to alert clinicians to our findings and encourage them to consider oral TB in the differential diagnosis for jaw lesions with multiple enlarged cervical lymph nodes.

Schwannoma derived from lingual nerve occurring in floor of mouth.

A schwannoma is a benign tumor composed of schwann cells which forms on the periphery of nerves. We report a case of a schwannoma derived from a lingual nerve occurring in the floor of the mouth. The patient was a 27-year-old woman who presented with the complaint of a swelling in the floor of the mouth. It is difficult to distinguish a swelling from a sublingual gland tumor, cyst, or malignant tumor by MRI alone. Therefore, a biopsy and cytological examination were performed one week prior to surgery to determine whether the growth represented a malignancy. The results revealed a class II growth which was suspected to be a schwannoma. Intraoperatively, it became clear that the tumor and lingual nerve were inseparable, making excision of the nerve unavoidable. On the other hand, there was a clear border between the tumor and the sublingual gland, so it was possible to preserve the sublingual gland. In the postoperative pathological diagnosis, a definitive diagnosis was difficult based solely on H-E staining. Therefore, immunohistochemical staining was performed, resulting in a diagnosis of schwannoma. Currently, the patient is still being followed up. The results of this case indicate that preoperative aspiration biopsy cytology is useful in deciding the operative method to be employed.

First description of a hybrid tumor of the sublingual gland.

BACKGROUND: Hybrid tumours of the salivary glands are rare neoplasms. They are composed of at least two different tumour entities located in the same topographic area and account for only 0.1% of all salivary gland tumours. The most common component is an adenoid cystic carcinoma. There are several possible forms of hybrid tumours, which are most commonly located in the parotid gland. CASE REPORT: We report on a 59-year-old female, who presented with a lesion of the caruncula of the left sublingual gland. The biopsy showed an adenoid cystic carcinoma in combination with a salivary duct carcinoma. Treatment consisted of tumour resection, bilateral selective neck dissection and adjuvant radiotherapy. Histopathologically, at least 30% of the tumour mass was composed of a salivary duct carcinoma and 70% of an adenoid cystic carcinoma. At 58 months after treatment, the patient is alive without evidence of recurrent disease. CONCLUSION: To our knowledge, the presented case is the first description of a hybrid tumour of the sublingual gland. Furthermore, the post-therapeutic course is encouraging, as hybrid tumours of the salivary glands usually have a poor prognosis.

Carcinoma adenoquístico de glándula sublingual: a propósito de un caso / Adenocystic carcinoma of the sublingual gland: on the purpose of a case

Los tumores de glándulas salivales son relativamente infrecuentes, constituyendo el 1 por ciento de los tumores de cabeza y cuello, siendo aún más raros los tumores de glándulas sublinguales, aunque frecuentemente su diagnóstico es maligno. El propósito de este trabajo es la presentación de un caso clínico que presentó un carcinoma adenoquístico de glándula sublingual en suelo de boca lado izquierdo. Clínicamente apareció una lesión alargada, sobre el trayecto del conducto de Wharton, con una coloración normal, lisa, brillante y asintomática. Se le realizó exéresis simple de la lesión y el Departamento de Anatomía Patológica confirmó el diagnóstico. Se procedió a revisar la bibliografía nacional e internacional sobre estas lesiones encontrándose escasas referencias debido a su rareza. La evolución de la paciente después de 16 meses ha sido satisfactoria(AU)

The salivary gland tumors are relatively infrequent, being only 1 per cent of the head and neck tumors; the sublingual gland tumors are even more unusual, although their diagnosis is frequently malignant. The purpose of the current work is presenting a clinical case carrying a sublingual gland adenocystic carcinoma on the left side of the mouth bottom. Clinically, there it was a long lesion, over the course of the Whartons duct, with an asymptomatic, normal, brilliant and smooth color. The lesion was simply removed and the Department of Pathologic Anatomy confirmed the diagnosis. We reviewed the national and international literature on these lesions, finding scarce references because of its unusualness. The patients evolution is satisfactory when 16 months have passed from the surgery(AU)

Pleomorphic adenoma of the sublingual salivary gland: an unusual diagnostic challenge.

OBJECTIVES: To describe a rare case of pleomorphic adenoma of the sublingual gland. CLINICAL PRESENTATION AND INTERVENTION: An 80-year-old patient presented with a painless solid submucosal mass of 4 months' duration in the anterior floor of the mouth, appearing as a distinct homogeneous, radiolucent lesion in CT imaging. Histologic findings after complete removal were suggestive of a pleomorphic adenoma almost completely surrounded by a fibrous capsule. No recurrence was observed after a 12-month follow-up. CONCLUSION: This case showed the possibility of sublingual salivary gland tumors and highlighted the need to consider them when making a differential diagnosis of masses located in the floor of the mouth.

Robotic-assisted transoral removal of a bilateral floor of mouth ranulas.

OBJECTIVE: To describe the management of bilateral oral ranulas with the use of the da Vinci Si Surgical System and discuss advantages and disadvantages over traditional transoral resection. STUDY DESIGN: Case Report and Review of Literature. RESULTS: A 47 year old woman presented to our service with an obvious right floor of mouth swelling. Clinical evaluation and computerized tomography scan confirmed a large floor of mouth ranula on the right and an incidental asymptomatic early ranula of the left sublingual gland. After obtaining an informed consent, the patient underwent a right transoral robotic-assisted transoral excision of the ranula and sublingual gland with identification and dissection of the submandibular duct and lingual nerve. The patient had an excellent outcome with no evidence of lingual nerve paresis and a return to oral intake on the first postoperative day. Subsequently, the patient underwent an elective transoral robotic-assisted excision of the incidental ranula on the left sublingual gland. CONCLUSION: We describe the first robotic-assisted excision of bilateral oral ranulas in current literature. The use of the da Vinci system provides excellent visualization, magnification, and dexterity for transoral surgical management of ranulas with preservation of the lingual nerve and Wharton's duct with good functional outcomes. However, the use of the robotic system for anterior floor of mouth surgery in terms of improved surgical outcomes as compared to traditional transoral surgery, long-term recurrence rates, and cost effectiveness needs further validation.

Clear cell carcinoma of the major salivary glands in an HIV-infected patient.

Clear cell carcinoma is a rare type of salivary gland carcinoma. It has a low degree of malignancy and long-term prognosis is favourable after surgical removal. The authors describe the case of a human immunodeficiency virus (HIV) infected 43-year-old woman who presented with a tumour on the floor of the mouth. After biopsy, left suprahyoid lymph node dissection and removal of the submandibular and sublingual glands was performed, followed by radiotherapy. Histologically, the tumour presented the characteristic features of hyalinizing clear cell carcinoma, defined as a variant of clear cell carcinoma by the latest World Health Organization classification. Hyalinizing clear cell carcinoma has a characteristic histological pattern and, to date, there is insufficient information to determine whether both forms behave similarly or differently. The present case illustrates a highly uncommon tumour variant occurring in a HIV-infected patient. To date, this association has not been described in the medical literature. The low grade of malignancy reported for this tumour demands a precise diagnosis and complete tumoral excision.

Sublingual gland tumors: clinical, pathologic, and therapeutic analysis of 13 patients treated in a single institution.

BACKGROUND: Sublingual gland tumors are rare, although frequently malignant. This study describes the clinicopathologic features and treatment results and reviews the literature. METHODS: Thirteen cases treated between 1996 and 2007 were reviewed with interest on clinical, pathologic, and therapeutic information. Survival data were calculated by the Kaplan-Meier method. RESULTS: Malignancies represented 92.3% of cases. Adenoid cystic carcinoma was the most common malignant type (66.7%). Most patients (83.3%) presented in advanced pathologic TNM stages (III or IV). All cases underwent surgical treatment. Neck dissection was performed in 69.2% with no metastases detected. Ten patients (83.3%) had adjuvant radiotherapy. Distant metastases occurred in 3 patients (25%). The 5-year overall and disease-free survival rates were 78.7% and 87.5%, respectively. CONCLUSIONS: Tumors of the sublingual gland are rare and are usually malignant. Radical surgery and adjuvant radiotherapy seems to offer adequate local and regional control. Unlike distant failure, local recurrence and regional metastases are not common.