Submandibular Mass Research Articles

Kimura's disease sequentially involving multiple sites in the head and neck: A case report with a 13-year follow-up and literature review.

Kimura's disease is a rare, chronic inflammatory condition with a high recurrence rate, primarily affecting young to middle-aged Asian males. It typically manifests as masses in the head and neck, accompanied with regional lymphadenopathy. This report describes the case of a 50-year-old man initially diagnosed with Kimura's disease 13 years ago, following surgical removal of a left submandibular mass. Subsequent recurrences involved the parotid and contralateral submandibular regions, requiring radiotherapy and intermittent oral prednisone therapy. After 6 years, he developed progressive proptosis and visual impairment, revealing multiple orbital masses bilaterally. A puncture examination of the left submandibular mass was indicative of Kimura's disease. Throughout the follow-up period, the eosinophil levels correlated with prednisone use. This case highlights the disease's capacity for multisite recurrence within the head and neck over an extended duration. The extensive orbital involvement without renal manifestations is a rare presentation. Long-term follow-up is crucial in Kimura's disease management, and oral prednisone can effectively control disease progression.

Management of Diffuse Large B-Cell Lymphoma as Post-Transplant Lymphoproliferative Disorder in a Kidney Transplant Recipient: A Case Report

Background and Clinical Significance: Post-transplant lymphoproliferative disorder (PTLD) is a severe complication of solid organ transplantation, often associated with prolonged immunosuppression. Diffuse large B-cell lymphoma (DLBCL) is the most common subtype. Managing PTLD requires a balance between reducing immunosuppression and preventing graft rejection. Case Presentation: A 41-year-old female kidney transplant recipient developed PTLD eight years post-transplant, presenting with a right submandibular mass. Biopsy confirmed CD20-positive DLBCL. Initial treatment involved reducing immunosuppression and rituximab monotherapy, which failed to prevent disease progression. The patient underwent six cycles of R-CHOP chemotherapy, achieving complete metabolic remission. Relapse occurred twice, with disease progression in the cervical nodes and tonsils. Salvage therapies, including polatuzumab vedotin and rituximab, achieved remission. During a subsequent relapse, loncastuximab tesirine induced metabolic resolution. Compromised renal function limited treatment options and a second renal transplant was delayed, reducing the risk of PTLD recurrence. Conclusions: This case underscores the challenges of managing PTLD in transplant recipients, especially in relapsed/refractory cases. Single-agent rituximab was insufficient, but combination chemotherapy and novel agents like loncastuximab tesirine were effective. Balancing oncologic control and graft preservation remains critical. This case highlights the need for individualized approaches and novel therapies in managing PTLD while addressing the complexities of immunosuppression and organ preservation.

Epithelial-Myoepithelial Carcinoma of the Submandibular Gland Arising from Recurrent Pleomorphic Adenoma: A Rare Case Report.

A 36-year-old male with a history of smoking presented with recurrent right submandibular swelling. Initially diagnosed with pleomorphic adenoma, he underwent total submandibulectomy. After 2 years, he developed a recurrent submandibular mass. Clinical and imaging assessments revealed a 6.2 cm multinodular tumor without invasion of adjacent structures. Surgery under general anesthesia involved tumor resection and lymph node dissection. Pathology confirmed carcinoma ex pleomorphic adenoma, specifically epithelial-myoepithelial carcinoma (EMC). Despite focal necrosis and a tumor size of 10.8 cm, margins were clear, and no lymph node metastasis was detected (pT3N0). Postoperative imaging showed no recurrence or metastasis, and the patient remains disease-free at the 25th month. This case highlights the diagnostic and therapeutic complexities of EMC, a rare malignancy constituting <1% of salivary gland tumors. Multidisciplinary collaboration enabled accurate diagnosis and effective treatment. Early intervention in recurrent pleomorphic adenomas is crucial to mitigate malignant transformation risk. Long-term surveillance is essential due to EMC's potential for late recurrence.

Frozen Section Analysis in Submandibular Gland Tumors: Optimizing Intraoperative Decision-Making

Introduction: With around 25 different salivary gland tumor entities described by the World Health Organization, the correct preoperative identification of masses as benign or malignant remains a challenge. If preoperative needle biopsy is inconclusive, frozen section analysis is a possible alternative for accurate histological identification. The purpose of our study was to evaluate the diagnostic effectiveness of frozen section performed for primary submandibular gland masses. Methods: In addition to acquiring epidemiological data from patients who underwent submandibular gland excision over a 20-year period, we analyzed the diagnostic effectiveness of frozen section performed for submandibular gland masses. We also examined the impact of frozen section on overall survival. Furthermore, we investigated whether there was an impact on the surgical revision rate for malignant submandibular gland masses that required additional neck dissection within the submandibular triangle. Results: Frozen section was performed for 54 submandibular gland tumors, with a specificity of 100% and a sensitivity of 81.3%. Frozen section was conducted in 12 cases of primary salivary gland malignancies, of which 9 cases were identified correctly. In three cases, the frozen section results were inconclusive. We calculated a relative risk reduction of 27% for revision surgery by performing frozen section. There was no significant association between frozen section results and overall survival. Conclusions: Frozen section demonstrates a significant reduction in the need for revision surgery. With a specificity of 100%, frozen section is especially suited to identifying benign masses. It is a valid diagnostic tool when preoperative sampling is not possible or is inconclusive.

Non tutte le tumefazioni cervicali sono linfoadenopatie

Cervical masses are predominantly due to lymphadenopathy secondary to infectious or inflammatory processes, with congenital anomalies being less common. The paper describes the case of a 15-year-old girl presenting with a progressively enlarging, tender left submandibular mass, associated with night sweats and fatigue. Initial imaging and blood tests suggested lymphadenopathy, but further evaluation with MRI identified a second-branchial cleft cyst. Surgical excision resolved the condition. This case underscores the importance of considering congenital anomalies in cervical mass differential diagnosis and the utility of advanced imaging for accurate diagnosis.

Usage of Milan System in Risk Stratification of Submandibular Gland Fine Needle Aspiration Cytology.

The Milan System for Reporting Salivary Gland Cytopathology (MSRSGC) categorizes fine needle aspiration and cytology (FNAC) results into 6 tiers and assigns a risk of malignancy (ROM) to each category. Its utility in submandibular gland lesions remains unclear. Our article aims to study (1) the utility of the MSRSGC in classifying FNAC of submandibular gland masses, (2) describe the demographics and incidence of submandibular gland malignancy in our population, and (3) analyze the accuracy of FNAC in diagnosis of benign and malignant submandibular gland tumors. In this retrospective case series, the FNAC results of 92 patients who underwent pre-operative FNAC and excision of submandibular gland between May 2008 and December 2023 were reviewed and assigned to a MSRSGC category, and ROM was calculated based on histological diagnosis. 20/92 (21.7%) submandibular gland excisions with pre-operative FNAC were malignant. ROM is as follows: non-diagnostic 7.1%, non-neoplastic 12.5%, atypia of undetermined significance 35.3%, benign neoplasm 3.3%, salivary gland neoplasm of uncertain malignant potential 35%, suspicious for malignancy 100%, and malignant 100%. Our study validates the usage of the MSRSGC in the work up of submandibular gland tumors. 4 Laryngoscope, 2025.

Misleading Imaging Findings: Bilateral Mylohyoid Defect Presenting as a Submandibular Mass Due to Sublingual Gland Protrusion.

The muscular base of the oral cavity is formed of the mylohyoid muscle, which forms a sling inferior to the tongue. The muscle is often discontinuous, and defects may include salivary tissues, fat, and/or blood vessels. Hypertrophic sublingual glands located in mylohyoid defects can be herniated into bilateral submandibular spaces and present as palpable masses. The etiology of this condition may be congenital or acquired, and although such anatomical variations are common, they often go unrecognized in clinical practice. Sialoceles are cyst-like structures that result from chronic inflammation or ductal injury, indicating underlying problems with drainage efficiency. In this case series, we present two patients. The first patient is a 44-year-old female who presented with a slowly enlarging right submandibular mass for two years, while the second is a 70-year-old female who presented with nonspecific neck discomfort, lacking palpable masses. In both, initial imaging (ultrasound and CT) was inconclusive. MRI revealed right sublingual gland herniation through a mylohyoid defect (mylohyoid boutonniere) in both cases. This highlights the importance of comprehensive imaging in the diagnosis of submandibular masses and emphasizes the need for considering mylohyoid boutonniere in cases of bilateral submandibular masses. Further research is warranted into the sialoceles associated with salivary gland abnormalities.

Intranodal Hemangioma: Case Report and Systematic Review.

Intranodal hemangiomas are rare benign vascular tumors of the lymph nodes, often misdiagnosed as malignant lymphadenopathies due to their clinical and radiological features. This case report and systematic review aim to elucidate the epidemiology, clinical profile, and therapeutic interventions for intranodal hemangioma, enhancing diagnostic accuracy and management. A systematic review followed the Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines. Searches were conducted in PubMed, Scopus, Web of Science, and Google Scholar up to March 15, 2024, using keywords like "intranodal hemangioma," "primary intranodal hemangioma," "nodal hemangioma," "vascular tumor," and "lymph node hemangioma." Inclusion criteria included studies on clinical presentation, diagnostic strategies, and therapeutic interventions for intranodal hemangioma in patients of all ages. Excluded were reviews, opinion pieces, non-English studies, cases of hemangioendothelioma, sclerosing hemangioma, and animal studies. Key aspects analyzed included demographics, clinical presentation, management strategies, and histopathological findings. Our case involves a 3-year-old female with a submandibular mass, initially diagnosed with hemangioma. After no response to beta-blocker therapy and conflicting imaging results, surgical excision was performed. Histopathology confirmed intranodal hemangioma. From 233 records, 19 publications (36 patients, including our case) met the inclusion criteria. The review shows common clinical presentations as painless, slowly enlarging lymph nodes, primarily in the cervical and axillary regions. Diagnostic challenges often require imaging and biopsy to differentiate from malignancies. Recognizing intranodal hemangiomas, particularly in childhood, is crucial to prevent misdiagnosis and inappropriate treatment. The rarity of these tumors in childhood and their clinical presentation similar to malignancies in older patients make diagnosis challenging without widespread awareness. Surgical excision remains the mainstay of both diagnosis and treatment, often leading to favorable outcomes.

A rare case of a pleomorphic adenoma of the submandibular gland

We report the case of a 27-year-old female with a painless slowly growing mass that had developed over 2 years in the right submandibular region. Physical examination revealed a firm painless submandibular mass. Fine-needle aspiration and computed tomography (CT) scan concluded a pleomorphic adenoma of the submandibular gland. The patient underwent a right total submandibular sialoadenectomy. Histopathological examination confirmed the diagnosis of pleomorphic adenoma arising from the submandibular gland. Transient palsy of the marginal mandibular division of the facial nerve was noted post-operatively, which recovered with supportive management. Pleomorphic adenoma of the submandibular gland represents a diagnostic and therapeutic dilemma in young adults. Preoperative evaluation, including MRI/CT scan and fine-needle aspiration, is recommended in all cases. Total submandibular sialoadenectomy is the standard recommended treatment to minimise recurrences and avoid malignant transformation.

A case of primary small cell neuroendocrine carcinoma of the submandibular gland

Small cell neuroendocrine carcinoma (SmCC) is rarely found in the head and neck region. With most cases having been reported in the larynx, SmCC of the salivary glands remains a rare clinical entity. Similar to that in the lung and larynx, SmCC of the salivary glands has an aggressive nature associated with strong metastatic potential, although its prognosis is relatively better. The authors experienced a case of a 50-year-old male patient who presented with a submandibular mass. After surgical excision, histopathologic and immunohistochemical examination confirmed the mass to be primary small cell neuroendocrine carcinoma of the submandibular gland. The patient received postoperative chemoradiation and has remained disease-free during 36 months of follow-up.

Castleman's Disease Mimicking Malignant Submandibular Tumor: A Case Report.

A submandibular mass can be attributed to a variety of potential diagnoses. We report a case of a 71-year-old man presenting with left submandibular swelling associated with multiple lymph nodes.All investigations were negative except for a suspected malignant submandibular tumour.Unicentric Castleman's disease was diagnosed based on the final histological examination.

Follicular adenoma with a papillary architecture originating from an ectopic thyroid gland: a case report

BackgroundFollicular adenomas with papillary architecture are rare tumors of thyroid origin and are composed of completely encapsulated follicular cells with a papillary architecture lacking the nuclear characteristics of papillary carcinoma. Herein, we present a case of follicular adenoma with papillary architecture originating from an ectopic thyroid gland, diagnosed from a mass in the submandibular region.Case PresentationA 70-year-old woman was referred to our hospital with the chief complaint of a painless left submandibular mass that had been present for one year. The patient underwent left submandibular dissection for therapy and diagnosis. Microscopically, papillary lesions with fibrovascular cores were observed in the interior, and the epithelial cells were cylindrical in shape with eosinophilic cytoplasm, round or oval nuclei, with no pathological features, leading to a diagnosis of papillary carcinoma or follicular carcinoma. The mass was diagnosed as a follicular thyroid adenoma with papillary architecture. This is the first report of a follicular adenoma with a papillary architecture originating from an ectopic thyroid gland.ConclusionThis experience suggests that follicular adenoma should be included in the differential diagnosis of ectopic thyroid tumors.

Bilateral Submandibular Sialolithiasis: A Case Report

Introduction: Lithiasis is the most common salivary disease in adults. Most salivary stones (80% to 95%) produces in the submandibular gland, 5% to 20% in the parotid gland, while the sublingual gland and minor salivary glands account for no more than 2%. Isolated involvement of one gland is usual. Simultaneous lithiasis of both submandibular glands is less frequent. The objective of this study is to report our experience in the treatment of lithiasis of the bilateral submandibular gland. Observation: Mr. K. M, 68 years old, chronic smoking, consulted in emergency November 2023 at the Mohammed VI Oujda University Hospital, for bilateral submandibular swelling. This swelling has been developing for 10 years and increased in size during meals. The current history dates back to 1 month ago with the increase in the right submandibular mass associated with dysphagia. The clinical examination revealed a right and left submandibular swelling, presence of right inflammatory signs: warm mass, painful on palpation, fixed in the deep plane, with expression of pus from the Wharton orifice. Cervical CT scan injected with contrast product was in favor of bilateral sialadenitis on sialolithiasis complicated by submaxillary abscess on the right. Bilateral submandibulectomy with extraction the calculi is done via an external cervical. No post-operative complications were observed and the patient was discharged from the hospital after two days of hospitalization. Discussion: The family history of sialolithiasis suggests the search for a genetic or familial origin of bilateral lithiasis of the submandibular glands, if there is not an obvious or probable etiology. Conclusion: Bilateral submandibular lithiasis is a rare clinical event but one that must be researsh for even in cases of isolated submandibular lithiasis. Extraction of the calculation is made by radical submaxillectomy by cervical externally, and the results are satisfactory.

A Case of Follicular Dendritic Cell Sarcoma in Submandibular Region

Follicular dendritic cell sarcoma (FDCS) is rare lymphoid sarcoma occurs anywhere in body, mostly in lymph nodes. Sixty-two-year-old man presented left submandibular gland region mass for 5 months. Mass excision with submandibular gland resection was performed. Histopathology showed proliferation of spindle and ovoid cells with storiform arrangement which were positive for CD21, CD23, Vimentin, Ki-67, suggested FDCS in submandibular gland region lymph node. Tumor size was 3cm with no involvement of resection margin, nor cellular atypia and necrosis, so regular follow up was performed. After 4 years, new enhancing mass in left submandibular area was found. Wide excision of mass with neck dissection on left level I-III was performed. Histopathology confirmed recurrence of FDCS. The patient underwent radiation therapy from left mandible to hyoid area. After 2 years, new nodule was found in left lung upper lobe, and wedge resection confirmed metastasis of FDCS. The patient is on adjuvant chemotherapy.

Primary Lymphoepithelial Carcinoma of the Submandibular Gland in A Young Patient: A Case Report.

Lymphoepithelial carcinoma (LEC) is a rare entity of malignancy. It has been associated with Epstein-Barr virus (EBV), and in regions where it is endemic, such as Southeast Asia, EBV is typically associated with nasopharyngeal carcinoma. The incidence of such malignancies in the young population is unusual. Here we report a 17-year-old with a 5-year history of a painless right submandibular mass presenting with a sudden increase in size for one month before surgery. Neck examination revealed a firm right submandibular gland measuring five by five cm, with an irregular surface, non-tender with normal overlying skin. We performed a fine needle aspiration cytology (FNAC), and he underwent computed tomography (CT) scan of the neck. He underwent a right submandibulectomy. Lymph nodes from levels I and II were also removed. The final histopathology was reported as LEC. Following this tissue diagnosis, another follow-up surgery, a modified radical neck dissection, was carried out for locoregional clearance. Postoperatively, he completed chemoradiotherapy treatment and is now on regular follow-up.

Is the neutrophil-to-lymphocyte ratio a marker for differentiating between benign and malignant submandibular gland masses?

This study aimed to evaluate the effect of the neutrophil-to-lymphocyte ratio on the differentiation of benign and malignant masses in the submandibular triangle. We retrospectively evaluated 48 patients who underwent surgery for submandibular gland masses between January 2013 and February 2023. The patient's age, gender, preoperative complete blood count and imaging findings, postoperative histopathological diagnosis, and hemogram data were analysed. Patients were evaluated according to their postoperative histopathological diagnoses and categorised into four main groups: sialolithiasis, sialadenitis, benign tumours, and malignant tumours. Benign submandibular gland disease formations were evaluated under group B and malignant tumour formations under group M. A preoperative fine needle aspiration biopsy was performed on 19 patients due to sialadenitis, pleomorphic adenoma, and malignant diseases other than sialolithiasis. One patient died among the patients with malignant disease and the remaining 7 patients were compared with the benign group of 40 patients regarding preoperative and postoperative neutrophil-to-lymphocyte ratio. In the benign group, the neutrophil-to-lymphocyte ratio was 2.64 preoperatively and decreased to 2.34 in the first postoperative year. The preoperative neutrophil-to-lymphocyte ratio decreased from 4.79 to 1.77 postoperatively in the malignant group. A statistically significant difference was observed (p<0.05). This is the first study to demonstrate that the neutrophil-to-lymphocyte ratio can be used as a biomarker in submandibular gland masses and has prognostic significance in malignant masses. In addition to fine needle aspiration biopsy results, neutrophil-to-lymphocyte ratio can be used as a biomarker.

Pleomorphic Adenoma of Submandibular Gland in a Pediatric Case.

Neoplasms of the salivary glands are relatively uncommon, with varied histologic subtypes. Only 5% of these occur in the pediatric age group. The submandibular gland tumors account for just 11.5% of pediatric salivary gland tumors. Few studies have reported the occurrence of pleomorphic adenoma arising in submandibular gland in a child. We describe a case of pleomorphic adenoma affecting submandibular gland in a girl aged eleven years and provide a brief review of current literature. Thorough diagnostic evaluation is required, considering the diverse differential diagnoses of pediatric submandibular masses. Although benign, recurrence and malignant transformation are pertinent concerns in pleomorphic adenoma, especially in a child, considering long life expectancy.

Adenoid Cystic Carcinoma: A Case Report

A 52-year-old male patient attended on 13 July 2022 with the complaints of pain in right ear and voice change for 1 month and cough for 15 days, difficulty in swallowing along with weight loss for same duration of past 1 month. Examination revealed a swelling on the right side of neck over the submandibular region also with cervical lymphadenopathy (level IB). After all evaluation, all biochemical tests were normal, but Fiber Optic Laryngoscopy (FOL) revealed right vocal cord palsy, USG of neck revealed right submandibular mass and cervical lymphadenopathy, CT scan of Neck up to Mid-chest revealed no pathology and last but not the least pre-operative FNAC was done and revealed organizing abscess. After that patient underwent excision of the right sub mandibular mass and removal of few superficial group of lymph nodes. Post-operative histopathology revealed Adenoid Cystic Carcinoma (ACC). Patient presented to us with no complain about submandibular gland or fine needle aspiration cytology (FNAC) also doesn’t indicate about malignancy, but our clinical suspicion was submandibular gland malignancy, that’s why we have decided to publish this case report. After operation Patient was fine till his last follow-up.&#x0D; Eastern Med Coll J. Jan 2023; 8 (1) 28-30

Multimodality imaging evaluation of nasal sinus alveolar rhabdomyosarcoma: Two case reports

Rhabdomyosarcoma (RMS) is a common pleomorphic malignant soft tissue sarcoma in children and adolescents that originates from rhabdomyoblasts or mesenchymal precursor cells. Alveolar rhabdomyosarcoma (ARMS) mostly occurs in adolescents aged 10-15 years and is characterized by more aggressive behaviors and worse prognosis than other sarcomas, prone to lymphatic and hematogenous metastasis in the early stage as well as metastasizing to breast, testis, pancreas, and other parts. ARMS often occurs in the limbs and genitourinary system, however, head and neck ARMS are relatively rare when involving the nasal cavity or sinuses. The role of MRI and 18F-FDG positron emission tomography combined with computed tomography (PET/CT) remains to be established in ARMS. Case 1: An 18-year-old male was found with a left submandibular mass of approximately 1 cm in diameter 2 months ago, which gradually increased in size. CT showed multiple soft tissue masses in maxillofacial and neck regions and the lesions invaded the frontal lobe and the inner wall of the left orbital lobe. MRI showed the masses with hypointensity on T1WI, hyperintensity on T2WI, and diffusion-weighted imaging (DWI) with significant enhancement. 18F-FDG PET/CT showed multiple hypermetabolic lesions located in the maxillofacial, neck region, 3rd lumbar vertebra, and the right sacrum. A nasal endoscopic tumor biopsy and molecular testing finally helped to diagnose the ARMS. Case 2: A 14-year-old male presented with left maxillary pain with nasal congestion and left ocular swelling 15 days ago. CT demonstrated a soft tissue mass in the nasal cavity and sinuses with local protrusion into the left orbit. MRI showed the masses with a slightly low signal on T1WI, a high signal on T2WI, and DWI with significant heterogenous enhancement. 18F-FDG PET/CT showed hypermetabolic lesions in the left maxillofacial and neck regions. ARMS was finally diagnosed by a nasal endoscopic tumor biopsy and molecular testing. The patient had a recurrence of the lesion after chemotherapy and surgical resection and is currently undergoing radiation therapy. Nasal sinus ARMS is highly malignant with a poor prognosis. Accurate diagnosis relies not only on histopathology and immunohistochemistry examination but also on genetic detection of characteristic chromosomal translocations and fusion genes. Imaging methods, such as MRI and PET/CT can accurately assess the extent of the lesions and metastases, assist in the diagnosis of the disease and the selection of treatment regimens, provide precise localization for surgery, and help with treatment monitoring and follow-up.

CML-357 Jumping Out of the Frying Pan, Into the Fire: Case Report of Secondary Non-Hodgkin Lymphoma (NHL) in a Case of Chronic Myeloid Leukemia (CML)

CML-357 Jumping Out of the Frying Pan, Into the Fire: Case Report of Secondary Non-Hodgkin Lymphoma (NHL) in a Case of Chronic Myeloid Leukemia (CML)