BACKGROUND: 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.
METHODS: 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.
METHODS: Primary HCCC of the sublingual gland.
METHODS: 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.
RESULTS: No recurrence was observed during the 12-month postoperative follow-up period.
CONCLUSIONS: 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.

OBJECTIVE: Despite advancements in imaging techniques and cytological analysis, plunging ranula remains a challenging surgical, radiologic, and pathologic phenomenon. Of the 18 patients we evaluated at our institution, we highlight three cases that illustrate the high rate of misleading imaging and cytological results when assessing plunging ranula.
METHODS: Imaging results, biopsy findings, operative techniques, and pathological reports were reviewed from patients who had either a preoperative or postoperative diagnosis of ranula and underwent surgery by a single head and neck surgeon at a tertiary care center.
RESULTS: Of the 18 identified patients, computed tomography was correct on preoperative imaging 73% of the time and magnetic resonance imaging was correct on preoperative imaging 71% of the time. Two patients underwent preoperative ultrasound and their ultrasound reports did not accurately diagnose the presence of a ranula. Two patients underwent preoperative fine needle aspiration biopsy due to inconclusive preoperative imaging, in which results suggested either a ranula or epidermal cyst. Both ultimately did not match the final pathology. Three of eighteen patients (17%) underwent an inappropriate initial surgery due to incorrect imaging diagnoses and/or biopsy findings.
CONCLUSIONS: Despite use of preoperative modalities to distinguish plunging ranula from other cystic floor of mouth lesions, surgeons must be aware that no workup modality is fully precise. The potential for revision surgery must be included in all preoperative discussions for presumed plunging ranula.
METHODS: 4 Laryngoscope, 134:2689-2696, 2024.

Plunging ranula, a subtype of ranula, commonly presents as a submandibular or submental cystic mass without oral counterpart, and its clinical management remains challenging. Herein, the authors report an extremely rare case of 30-year-old female patient with plunging ranula involving the root of the left anterior neck.

Adamantinoma-like Ewing sarcoma (ALES) of the salivary glands is an exceedingly rare malignancy defined by the t(11,22) EWSR1::FLI1 fusion, with complex epithelial differentiation. To identify features that can allow for better recognition of this disease entity, we reviewed all published reports of molecularly confirmed ALES of the salivary glands and explored epidemiological, clinical, radiological, pathological, and therapeutic characteristics of a population of 21 patients including a single newly reported patient from our group. We searched the English-language literature indexed in PubMed, Medline, Scopus, and Web of Science using the keyword \'Adamantinoma-like Ewing sarcoma\' published up to June 2022. The median age at diagnosis was 46 years, and a slight female sex predilection was observed. Most tumors originated in the parotid gland (86%) and presented as a painless palpable mass with a median diameter of 3.6 cm. Metastatic dissemination was reported only in one patient (5%), and after a median follow-up of 13 months the 1-year overall survival rate was 92%. Salivary gland ALES were frequently misdiagnosed at presentation (62% of cases) and were pathologically characterized by the presence of highly monomorphic small round blue cells with infiltrative pattern and positive immunostaining for CD99 and high- and low-molecular weight cytokeratins. Epidemiological and clinical features of salivary gland ALES raise questions on the incorporation of this malignancy in the Ewing sarcoma family tumor group.

(1) Background: Extrapleural solitary fibrous tumors (ESFTs) are rare oncological entities occurring in the head and neck, and even more so in the salivary glands. The clinical presentation and histologic features are usually unspecific, resulting in frequent misclassification. As an unusual tumor, ESTFs have an unpredictable clinical behavior. (2) Methods: We present two clinical cases referred to our Maxillofacial Surgery Unit for the onset of a symptomless mass involving, in one case, the parotid gland, and in the other case, the sublingual gland. (3) Results: Solitary fibrous tumors could be considered as neoplasms with intermediate biological behavior that are not entirely predictable on the basis of morphological features, as these are mostly still unknown. However, a few histologic, immunohistochemical, and imaging features, such as a hypodense signal at the T1 sequence in an MRI, or positivity for CD34, bcl2, and CD99, and the NAB2-STATS6 fusion gene, could be useful for an early differential diagnosis of ESTFs. (4) Conclusions: All patients were alive at follow-up with no evidence of disease. Surgical management should always be considered as the first choice for oncological radicality, and clinical behavior should always be defined with the help of the study of radiological and anatomopathological features.

OBJECTIVE: Non-neoplastic salivary gland diseases are rare in children. This paper aims to present the spectrum of encountered non-neoplastic salivary gland diseases at a tertiary center by describing the demographics, clinical characteristics, and outcomes in this patient population.
METHODS: A review of electronic medical records was performed from 2010 until 2020. Relevant data were retrieved and charted according to the type of salivary gland disease. A comparison between diseases was made for demographics, presenting symptoms, treatment, and outcomes.
RESULTS: Fifty patients with 11 different non-neoplastic salivary gland diseases were identified. Sialolithiasis was the most prevalent condition (12/50), with 83% localized in the submandibular gland. In contrast to gender, age, and symptoms, the location of pathology was significantly associated with the diagnosis (p < 0.001). In patients with sialolithiasis, a hybrid procedure (combined endoscopy and lithotomy) resulted in 100% resolution of symptoms. For (plunging) ranula, marsupialisation had a relative risk of recurrence of 9.6 compared to (partial) extirpation of the sublingual gland.
CONCLUSIONS: Children with salivary gland diseases present with overlapping symptoms, making clinical diagnosis challenging. The present study may aid physicians and specialists in diagnosing the most prevalent conditions in children. Although no gold standards exist for their treatment, hybrid procedures (sialolithiasis) and subglingual gland extirpation (ranula) showed superior results over alternatives.

OBJECTIVE: In this article we review the literature on the malignant sublingual gland tumors from a surgical perspective.
BACKGROUND: Sublingual gland tumors occur with a very low incidence and most are malignant tumors. The extent of treatment, clinical outcomes and prognosis of malignant sublingual gland tumors have not been well defined, due to the rarity of this disease.
METHODS: A database search using Web of Science (https://webofknowledge.com/) and PubMed (https://pubmed.ncbi.nlm.nih.gov/) was conducted. The following keywords were used in the search: \"sublingual gland tumor\", AND \"malignancy\".
CONCLUSIONS: Although the sublingual glands are among the major salivary glands, they share common anatomical characteristics with minor salivary gland tumors. Therefore, the tumors from the sublingual gland may have different clinical behaviors from the other major salivary gland tumors. The sublingual glands are small without a true surrounding capsule of the glands; the extra-parenchymal extension is very common in sublingual gland tumors. Furthermore, the sublingual glands are located in close proximity to the lingual nerve, the submandibular gland-duct system and the mandible. Thus, the surgical approach to the malignant sublingual gland tumor should include the adequate management of neighboring structures. In addition, adjuvant radiation therapy provides a survival benefit for patients with malignant sublingual gland tumors, which have adverse features. This article summarizes the clinical characteristics and unique features of malignant sublingual gland tumors based on previous reports, and provides clinical information regarding the sublingual gland tumors to increase awareness of primary physicians as well as patients.

BACKGROUND: Adenoid cystic carcinoma (ACC) is a rare malignant tumor that primarily occurs in the salivary glands. Distant metastases can develop despite favorable local control. Moreover, distant metastasis of ACC can occur after a long time interval without local recurrence. We report the first case of ACC of the sublingual gland that developed lung metastasis 20 years after primary treatment.
METHODS: A 52-year-old man was referred to our department with a 1-year history of painful swelling on the right oral floor.
METHODS: An incisional biopsy was performed, and histopathological examination revealed malignancy.
METHODS: Surgical excision of the right oral floor and right supra-omohyoid neck dissection with postoperative chemoradiation therapy were performed, and ACC of the sublingual gland was diagnosed. Left pulmonary metastasis was detected 20 years after the primary treatment. Metastasectomy was performed; however, subsequently, skin and bone metastases developed.
RESULTS: After receiving palliative care, the patient died of multiple organ failure.
CONCLUSIONS: As late distant metastasis of salivary ACC can develop, patients who undergo primary treatment need a long-term, strict follow-up plan even if locoregional control is favorable.

OBJECTIVE: The aim of this article was to conduct a 25-year retrospective study within an oral surgery department, on the incidence of the ranula in children and its ideal treatment, associated with a literature review on the plunging ranula in pediatrics.
METHODS: A retrospective review of the medical records of pediatric patients (0-12 years) from 1995 to 2020 was performed in an oral surgery department in Maringá/Brazil. The examined data included age of the patients, sex, clinical signs, time of evolution and follow-up, complementary exams, type of ranula, diagnostic hypothesis, surgical procedures and accesses, complications and recurrence. In addition, a literature review was carried out on the plunging ranula in children, using the MEDLINE database, from 1995 to 2020, with the search terms: \"PLUNGING RANULA\" OR \"CERVICAL RANULA. A PICOS was established and PRISMA standards were followed.
RESULTS: In the retrospective study, of the 11 patients found, 10 were with simple ranulas and only 1 with plunging ranula. All patients were approached by intraoral access, and conservative treatments had higher recurrence rates. The case of plunging ranula was treated by intraoral resection of the sublingual gland and saliva drainage, and obtained good results with 15 years of follow-up. In the literature review, 372 articles were found, which 10 were qualitatively selected after inclusion and exclusion criteria. Excision of the sublingual gland was the most prevalent procedure, and intraoral and extraoral accesses had the same incidence, despite the fact that the last one had higher percentages of complications.
CONCLUSIONS: The treatment of ranulas is variable; however, it is proven that conservative methods have higher rates of recurrence. As for the plunging ranula, resection of the sublingual gland through intraoral access, associated with mucus leakage, is considered a safe and effective treatment.

BACKGROUND: Nodal metastasis in salivary gland malignancies has important clinical implications; a good understanding of their complex anatomy is paramount to the head and neck surgeon.
METHODS: A contemporary and comprehensive literature review was conducted of the lymphatic drainage of the salivary glands, with special emphasis on its surgical applications.
RESULTS: The parotid gland has extraglandular and intraglandular nodes acting as a single functional drainage unit. Intraglandular parotid notes are unique to the parotid gland, and consist of a larger superficial group and a smaller deep group. The presence of intraglandular submandibular nodes, as described by early anatomists, is much debated nowadays. The sublingual glands drain to the lingual lymph nodes, which are divided into median, intermediate and lateral groups.
CONCLUSIONS: This review highlights the complex arrangements of lymph nodes draining the salivary glands. It may provide a valid anatomical explanation for the nodal metastasis patterns commonly seen in salivary gland malignancy.