Unicentric Castleman's disease in the parotid gland associated with psoriasis: a case report.

BACKGROUND: Castleman's disease is a rare lymphoproliferative disorder that is often misdiagnosed because of its untypical clinical or imaging features except for a painless mass. Besides, it is also difficult to cure Castleman's disease due to its unclear pathogenesis. CASE PRESENTATION: We present a Castleman's disease case with diagnostic significance regarding a 54-year-old Chinese male who has a painless mass in his left parotid gland for 18 months with a 30-years history of autoimmune disease psoriasis. Computed tomography scan showed a high-density nodule with clear boundaries in the left parotid and multiple enlarged lymph nodes in the left submandibular and neck region. General checkup, the extremely elevated serum interleukin-6 and lymph node biopsy in the left submandibular region gave us an initial suspicion of Castleman's disease. Then the patient underwent a left superficial parotidectomy. Based on histopathologic analysis, we made a certain diagnosis of Castleman's disease and gave corresponding treatments. In 18 months of follow-up, the patient showed no evidence of recurrence, with the level of serum interleukin-6 decreased. CONCLUSIONS: Clinicians should be aware of the possibility of Castleman's disease when faced with masses or enlarged lymph nodes in the parotid gland to avoid misdiagnosis, especially in patients with autoimmune diseases and elevated serum interleukin-6.

Metastasizing pleomorphic adenoma of the parotid gland: A common tumour with a very unusual presentation.

Metastasizing pleomorphic adenoma is recognized as a subtype of pleomorphic adenoma in WHO classification 5th edition of salivary glands. The controversy pertaining to the entity is the benign features of the disease even at a metastatic site. We present a rare case of left recurrent pre-auricular swelling in a young male reported as metastasizing pleomorphic adenoma. A nineteen-year-old male presented with left preauricular swelling seven years ago which was diagnosed as pleomorphic adenoma and underwent complete excision of tumour. The tumour recurred twice - two and five years after the surgery. At the second recurrence, the level II neck dissection showed multiple encapsulated deposits of pleomorphic adenoma having similar morphology in the cervical soft tissue with no features of high-grade transformation.

[Diffuse thickening of bronchial walls with multiple nodular mucosal protrusions].

A 58-year-old woman presented with a six-month history of nasal congestion, sore throat and cough, and a five-month history of dyspnea. She had a history of xerostomia for one year. On examination, the bilateral submandibular gland and parotid glands were enlarged. Parotid and anterior cervical lymph nodes were palpable. There were rales in both lungs. The rest of the physical examination was unremarkable. Sialographic analysis showed normal caliber in the main duct, stenosis in secondary ducts, and dilation in the proximal ducts. Minor salivary gland biopsy demonstrated periductal lymphocytic infiltration. Chest computed tomography (CT) showed diffuse thickening of the tracheal and bilateral bronchial walls. Bronchoscopy revealed macroscopic multiple nodules mainly in the trachea and bilateral main bronchus. Endobronchial biopsy showed lymphocytic infiltration in the bronchial submucosa. She was diagnosed with Sjögren's syndrome and treated with glucocorticoids. The dose of prednisone was started at 30 mg/d and tapered gradually. Following treatment, the patient's clinical condition improved dramatically, with shrinkage of the enlarged lymph nodes, bilateral submandibular and parotid glands. A repeated chest CT scan revealed improvement of the tracheal and bilateral bronchial thickening. Multiple nodules in the airway regressed, as evidenced by repeated bronchoscopic examination. The final diagnosis was a large-airway disease associated with Sjögren's syndrome.Among airway diseases in Sjögren's syndrome, peripheral airway diseases including bronchiolitis and bronchiectasis are common; however, central airway lesions in Sjögren's syndrome, especially with macroscopic nodules, are rare. In this case, we demonstrated tracheal and endobronchial nodules in Sjögren's syndrome as determined by clinical features, CT scan, bronchoscopy, and response to therapy.

Chinese surgical robot-assisted surgery for parotid tumor: a case report.

Robotic surgery is known as the "third technological revolution" in the field of surgery, and is an important milestone in the development of modern surgery. However, our country's innovative surgical robot industry is still in its early stages, and it is only being utilized in certain surgical fields. To explore the effectiveness of the application of domestic surgical robot in oral and maxillofacial surgery, the author successfully completed a case of benign parotid tumor resection with the assistance of a domestic autonomous robot. The operation was successful, facial nerve function was preserved, and postoperative wound healing was good.

Clinical decision making when cytology indicates a Warthin tumor.

Warthin tumor (WT) is a benign tumor usually affecting the parotid gland. The main diagnostic tool remains ultrasound combined with fine-needle aspiration cytology (FNAC). This study aims to examine how reliably FNAC indicates WT for clinical decision making regarding surgical versus conservative management. We included all patients who underwent FNAC from a parotid gland lesion between 2016 and 2018 at our institution, and whose FNAC revealed WT suspicion. The FNACs were divided into three groups based on the cytology report: certain, likely, and possible WT. The patients were divided into two groups based on having had either surgery or follow-up. We sent a questionnaire to patients who had not undergone surgery in order to obtain follow-up for a minimum of four years. Altogether, 135 FNAC samples, from 133 tumors and 125 patients, showed signs of WT. Of the 125 patients, 44 (35%) underwent surgery, and 81 (65%) were managed conservatively. Preoperative misdiagnosis in FNAC occurred in three (7%) surgically treated tumors. Their FNACs were reported as possible WTs, but histopathology revealed another benign lesion. In the conservatively treated group, two patients underwent surgery later during the follow-up. Cytological statements of WT were seldom false, and none were malignant. The majority of the patients were only followed-up and rarely required further treatment. A certain or likely diagnosis of WT in the FNAC report by an experienced head and neck pathologist is highly reliable in selecting patients for conservative surveillance.

Basal cell adenoma and basal cell adenocarcinoma of the parotid gland: clinical findings and surgical outcomes in a single-institution study.

BACKGROUND: Basal cell adenoma (BCA) is a rare benign tumor within the salivary glands. Basal cell adenocarcinoma (BCAC), the malignant counterpart of BCA, is also an exceedingly rare tumor with very limited clinical studies conducted. This study aims to investigate the clinical characteristics, demographics, and surgical outcomes of patients diagnosed with BCA and BCAC within the parotid gland. METHODS: A retrospective analysis from May 2003 to August 2023 was performed for all patients undergoing parotidectomy for masses. Retrospective data on gender, age, tumor characteristics, and outcomes were collected. Surgical approaches, including negative margin attainment, capsule removal, and histological diagnosis, were also detailed. RESULTS: The study included 1268 patients who underwent parotidectomy, resulting in 81 cases of BCA and 7 cases of BCAC. BCA patients, with a mean age of 55.1 years, showed diverse age distribution and predominantly presented in the 50s. In BCAC cases, seven female patients exhibited a predominant location in the deep lobes. FNA revealed BCAC in three out of seven cases, and subsequent parotidectomy was performed, resulting in no observed recurrences or metastases. CONCLUSION: This study reports the largest number of BCA cases from a single institution and provides comprehensive insights into the demographics, tumor characteristics, and clinical outcomes of both BCA and BCAC. Although further research should be conducted, based on clinical follow-up results, appropriately including the capsule in the tumor excision indicates favorable outcomes, especially when the tumor size is not large.

Persistent bleeding following biopsy of an epidermoid cyst within the parotid gland.

Epidermoid cysts are benign lesions most commonly found in the skin but which can arise in many other locations including, very rarely the salivary glands. This rarity often leaves them off standard differential lists and can create a diagnostic dilemma. A patient with an incidentally detected parotid mass on MRI underwent core biopsy, which was unfortunately complicated by formation of a pseudoaneurysm and persistent arterial bleeding requiring coil embolisation. The histology showed only keratinous material and, in retrospect, the signal characteristics of the mass were entirely typical of an epidermoid cyst. Recognition of this common, benign entity in a very rare location can obviate the need for invasive tests and potential complications and direct management to more appropriate imaging follow-up.

Carcinoma showing thymus-like elements (CASTLE) with amyloid deposition in the parotid gland.

Carcinoma showing thymus-like elements (CASTLE) is a rare tumor that commonly occurs in the thyroid gland. Extrathyroidal CASTLE is rarer, and only 11 cases of CASTLE of major salivary glands have been reported to date. We report the first case of amyloid deposition in parotid CASTLE. A 63-year-old man presented with a slowly growing mass in the left parotid region. Computed tomography revealed an approximately 28 × 23 mm mass lesion in the left parotid gland, and squamous cell carcinoma was suspected on biopsy. The patient underwent a parotidectomy with neck dissection. Morphologically, the tumor cells were squamoid and formed nests with lymphoid infiltration. Immunohistochemically, the tumor cells exhibited immunoreactivity for CD5, CD117/c-kit and Bcl-2, p40, and CK5 but not for p16. We diagnosed the tumor as parotid CASTLE. Amyloid deposition was also observed in the primary tumor and metastatic lymph node lesions, which were immunoreactive for cytokeratin 5. Tumor cytokeratin-derived amyloid deposition may be one of characteristics of parotid CASTLE.

Differential expression of TRKB tyrosine kinase in the two histological types of parotid salivary duct carcinoma with cancer aggressiveness.

Parotid salivary duct carcinoma (SDC) is a rare and aggressive parotid gland carcinoma (PGC). SDC has two origins: de novo and ex pleomorphic adenoma (SDC ex PA); however, because of its rarity, the clinical and molecular features of the two types of SDC are not sufficiently understood. Here, we studied the differences in their clinicopathological and molecular features using clinical specimens while comparing them to those of adenoid cystic carcinoma (AdCC), an intermediate-grade PGC. Clinicopathological analysis of tissues from patients with PGC revealed significant associations between histological types and malignant phenotypes, including nodal metastasis, recurrence, vascular invasion, and neural invasion, and revealed more malignant phenotypes of de novo SDC than of SDC ex PA. The de novo SDC showed a significantly higher frequency of intra-neural invasion (intra-NI) and vascular invasion than AdCC and SDC ex PA. PGCs with high intra-NI were significantly correlated with malignant phenotypes and survival rates. Recently, we observed the overexpression of tropomyosin receptor kinase B (TRKB), a receptor tyrosine kinase, in PGC cells. Here, immunohistochemical and clinicopathological analyses showed that TRKB was highly expressed in SDC cells, particularly de novo SDC cells, and was significantly associated with poor survival and highly malignant phenotypes, including intra-NI and vascular invasion. Collectively, these data show that TRKB expression is significantly elevated in PGC, particularly in de novo SDC, and can be one of the biomarkers of their aggressiveness.

Case of clear-cell oncocytoma of parotid gland and literature review.

Oncocytoma is a benign tumor of the salivary gland. Its incidence is very low and very seldom documen-ted in literature. Clear-cell dominant oncocytoma is even less common. The tumor's clinical symptoms and imaging results are nonspecific, so distinguishing other salivary gland tumors (such as oncocytic carcinoma) from clear-cell renal carcinoma is difficult, possibly leading to misdiagnosis and maltreatment. Here, a case of clear-cell dominant oncocytoma was presented, and the relevant literature was evaluated to investigate the diagnosis and management of clear-cell dominant oncocytoma.

Mucosa-associated lymphoid tissue lymphoma at the parotid gland.

MALT (mucosa-associated lymphoid tissue) lymphomas are low-grade extra-nodal B-cell lymphomas that may involve various sites in the head and neck including the thyroid, salivary, and lacrimal glands. Development of MALT lymphoma in the head and neck is often associated with auto-immune diseases such as Sjögren syndrome or Hashimoto thyroiditis. Here, we report a case of a MALT lymphoma of the left buucal mucosa that likely arose in the parotid gland. The patient was successfully treated with surgical excision with chemotherapy and remained disease-free at the 10-year follow-up. Since it was rare in the head and neck region, we present this case.

Diagnostic Challenges and Imaging Considerations for Intraparotid Facial Nerve Schwannoma: A Case Report and Literature Review.

BACKGROUND A mass in the parotid gland usually indicates parotid gland neoplasia. Warthin tumors or pleomorphic adenomas are common differential diagnoses. Less frequently, other differential diagnoses and sites of origin are considered. Schwannomas are rare, benign tumors in the head and neck region. Even more rarely, these tumors occur in the intraparotid course of the facial nerve. In the following, we report about 2 patients in whom a mass in the right parotid gland was found incidentally during magnetic resonance imaging (MRI). CASE REPORT We reviewed data from the literature on intraparotid facial nerve schwannomas (IPFNS) and compared them with those from our cases. The focus was on data such as clinical history, clinical symptoms, electroneurography, and various imaging modalities, such as ultrasonography and MRI combined with diffusion-weighted imaging. CONCLUSIONS It is challenging to distinguish facial nerve schwannomas from other neoplasms. Patient's history, clinical symptoms, MRI examination with diffusion-weighted imaging, and high-resolution ultrasound imaging are decisive factors for diagnosis and should be performed when IPFNS is suspected. Diagnosis and therapy for IPFNS remain challenging. A wait-and-scan approach could be an option for patients with small tumors and good facial nerve function. On the other hand, patients with advanced tumors associated with limited facial nerve function can benefit from surgical approaches or stereotactic radiosurgery.

Correlation between MRI (DWI and DCE) and cellularity of parotid gland pleomorphic adenomas.

PURPOSE: Parotid pleomorphic adenomas present a risk of recurrence, higher when the tumour is a hypocellular subtype. The aim of the study was to determine whether it is possible to characterize this histological subtype with diffusion and perfusion sequences of the preoperative MRI. METHODS: This retrospective study included 97 patients operated between 2010 and 2020. Histologic slides review was performed to classify tumours into three histologic subtypes: hypocellular, classical and hypercellular. Univariate and multivariate analyses studied the correlation between histology and diffusion and perfusion MRI parameters obtained with OleaSphere® software. RESULTS: The hypocellular subtype had higher apparent diffusion coefficient values than the other two subtypes: 2.13 ± 0.23, 1.83 ± 0.42, and 1.61 ± 0.4 × 10-3 mm2/s for hypocellular, classical and hypercellular subtype respectively (p < 0.0001). Multivariate analysis showed that an ADCmean > 1.88 × 10-3 mm2/s was suggestive of a hypocellular pleomorphic adenoma in 79% of the cases, with a specificity and PPV of 94 and 96% (p < 0.001), respectively. CONCLUSION: The histological subtype of a pleomorphic adenoma can be predicted preoperatively with ADC values. A prospective and multicentric study on a larger cohort is needed to confirm our results.

Parotid Gland Metastases of Cutaneous Squamous Cell Carcinoma of the Head: Occult Metastases Occurrence and Their Late Manifestation.

Cutaneous squamous cell carcinomas (cSCC) are malignant tumours with excellent prognosis unless nodal metastases develop. The aim of our study is to determine the prognostic significance of the clinical stage of parotid gland metastases and the incidence of occult cervical lymph node involvement in cSCC of the head. Our retrospective analysis includes 39 patients with cSCC parotid gland metastases, 15 of whom had concurrent cervical node involvement. In 32 patients, the lymph nodes manifested at stage N3b. A total of 26 patients were treated with parotidectomy, 9 patients received radiotherapy alone, and 4 received symptomatic therapy. The surgical treatment included either total conservative (21 cases) or superficial parotidectomy (5 cases) and neck dissection (therapeutic neck dissections in 11 cases and elective in 14 cases). In all cases, surgery was performed with sufficient tumour-free resection margins. Adjuvant radiotherapy was administered postoperatively in 16 patients. Occult metastases were present in 21% of cases after an elective neck dissection, but not in any case in the deep lobe of the parotid gland. The five-year overall survival and recurrence-free interval were 52% and 55%, respectively. Patients with the cN3b stage and G3 histological grade tend to have a worse prognosis, but not at a statistically significant level. The prognosis was not worse in patients with concurrent parotid and cervical metastases compared to those with metastases limited to the parotid gland only. The addition of adjuvant irradiation, in comparison to a single modality surgical treatment, was the only statistically significant prognostic factor that reduced the risk of death from this diagnosis (p=0.013). The extent of parotidectomy (partial vs. total) had no impact on either the risk of recurrence or patient prognosis. The combination of surgery with irradiation provides the best results and should be applied to all patients who tolerate the treatment. A partial superficial parotidectomy should be sufficient, with a minimum risk of occult metastasis in the deep lobe. Conversely, the relatively high incidence of occult neck metastases indicates that patients could likely benefit from elective neck dissection.

Development of Presumptive Sialadenosis Following a Chronic Oropharyngeal Stick Injury in a Dog.

A 3 yr old female spayed Labrador retriever was referred for the treatment of a chronic oropharyngeal stick injury. After computed tomography scan evaluation, the cervical area was explored surgically and a right-sided cervical abscess that contained a wooden stick was identified adjacent to the vagosympathetic trunk and carotid artery. The ipsilateral mandibular salivary gland was resected concurrently given its abnormal appearance, and histology confirmed inflammation and necrosis of the gland, which was suspected to be due to direct trauma from the foreign body. The clinical signs initially improved but then recurred, and a follow-up computed tomography scan was suggestive of sialadenosis or sialadenitis in the right parotid, zygomatic, and molar salivary glands. A presumptive diagnosis of sialadenosis was made and a course of phenobarbital was initiated. The clinical signs resolved completely within a few days, and there was no recurrence several months after termination of the phenobarbital treatment. This is the first case report of presumptive sialadenosis in a dog as a suspected complication of an oropharyngeal stick injury. Informed consent was obtained from the owner of the dog and the patient was managed according to contemporary standards of care.

Disease and toxicity outcomes for a modern cohort of patients with squamous cell carcinoma of cutaneous origin involving the parotid gland: Comparison of volumetric modulated arc therapy and pencil beam scanning proton therapy.

OBJECTIVES: We sought to describe outcomes for locally advanced cutaneous squamous cell carcinoma (SCC) involving the parotid treated with volumetric modulated arc therapy (VMAT) versus pencil beam scanning proton beam therapy (PBT). MATERIALS AND METHODS: Patients were gathered from 2016 to 2022 from 5 sites of a large academic RT department; included patients were treated with RT and had parotid involvement by: direct extension of a cutaneous primary, parotid regional spread from a previously or contemporaneously resected but geographically separate cutaneous primary, or else primary parotid SCC (with a cutaneous primary ostensibly occult). Acute toxicities were provider-reported (CTCAE v5.0) and graded at each on treatment visit. Statistical analyses were conducted. RESULTS: Median follow-up was 12.9 months (1.3 - 72.8); 67 patients were included. Positive margins/extranodal extension were present in 34 cases; gross disease in 17. RT types: 39 (58.2 %) VMAT and 28 (41.8 %) PBT. Concurrent systemic therapy was delivered in 10 (14.9 %) patients. There were 17 treatment failures (25.4 %), median time of 168 days. Pathologically positive neck nodes were associated with locoregional recurrence (p = 0.015). Oral cavity, pharyngeal constrictor, and contralateral parotid doses were all significantly lower for PBT. Median weight change was -3.8 kg (-14.1 - 5.1) for VMAT and -3 kg (-16.8 - 3) for PBT (p = 0.013). Lower rates of ≥ grade 1 xerostomia (p = 0.002) and ≥ grade 1 dysguesia (p < 0.001) were demonstrated with PBT. CONCLUSIONS: Cutaneous SCC involving the parotid can be an aggressive clinical entity despite modern multimodal therapy. PBT offers significantly lower dose to organs at risk compared to VMAT, which seemingly yields diminished acute toxicities.

Dysgenetic Polycystic Disease of the Salivary Glands: A Case Report of This Rare Entity Occurring for the First Time in the Minor Salivary Glands of the Tongue, and a Review of the Literature.

Dysgenetic polycystic disease, also known just as polycystic disease, is a very rare developmental abnormality affecting the salivary gland duct system. This entity has been reported in only 21 patients previously, although a careful review suggests only 16 patients have histological evidence of the disease. In previously reported cases, this lesion most commonly presents as either an incidental finding or as a swelling affecting the parotid glands bilaterally, or rarely the submandibular glands bilaterally. This case report details the first time dysgenetic polycystic disease is found affecting the minor salivary glands of the tongue in a 55-year-old male. Histochemical and immunohistochemical stains are presented and include positivity for AE1/AE3 and p63, and negativity for progesterone receptor, androgen receptor, mammaglobin, S100 and BRAF V600E. PAS-D and Congo Red highlight special microamyloid spheroliths structures intraluminally.

Differentiation of parotid pleomorphic adenoma from Warthin tumor using signal intensity ratios on fat-suppressed T2-weighted magnetic resonance imaging.

OBJECTIVE: To investigate the value of magnetic resonance imaging (MRI) signal intensity ratios (SIRs) based on fat-suppressed T2-weighted imaging (FS-T2WI), together with demographic features, MRI anatomical characteristics, and SIRs of histopathological patterns of the tumors, in the differentiation of parotid pleomorphic adenoma (PA) from Warthin tumor (WT). STUDY DESIGN: In total, 90 patients with PA and 56 patients with WT were enrolled in the study. SIRs of tumor to normal parotid gland (SIR-T/P), spinal cord (SIR-T/S), and muscle (SIR-T/M) were calculated. Demographic and radiological features of the 2-patient groups were compared with univariate analysis and multivariate logistic regression analysis. The area under the receiver operating characteristic curve (AUC), sensitivity, and specificity were analyzed to evaluate the utility of SIRs in distinguishing between PA and WT. RESULTS: SIR-T/P exhibited outstanding discriminating ability (AUC = 0.934), SIR-T/S had excellent discrimination (AUC = 0.839), and SIR-T/M showed acceptable discrimination (AUC = 0.728). When SIR-T/P of 1.96 was selected as the cutoff value, sensitivity and specificity were 0.756 and 0.982, respectively. SIR-T/P, age, sex, and number of lesions were identified as independent predictors by multivariate logistic regression analysis. Differences in SIRs between histopathological patterns were significant. CONCLUSION: SIR-T/P based on FS-T2WI is an effective discriminator in the differential diagnosis between PA and WT. Age, sex, and number of lesions provided additional value in differentiation.

Trichilemmal carcinoma metastatic to parotid gland: First report of fine needle aspiration cytology features.

Trichilemmal carcinoma is a rare malignant cutaneous adnexal neoplasm arising from the outer root sheath of the hair follicle. Majority of cases occur on sun-exposed sites such as the face, scalp and neck, making them easily amenable to being biopsied and subjected to histological examination for definitive diagnosis. Thus, cytological features of trichilemmal carcinoma have not been described till date. Trichilemmal carcinoma is a low-grade malignancy, albeit with potential to metastasize to regional lymph nodes and distant sites. We report the case of trichilemmal carcinoma of scalp that metastasized to cervical lymph nodes and parotid gland and underwent fine-needle aspiration cytology (FNAC) from the parotid lesion. The aspirate showed tightly cohesive cell clusters with sharp borders. Tumour cells ranged from basaloid with scant cytoplasm to those with abundant cytoplasm. Nuclei were vesicular, with inconspicuous to prominent nuclei. Intercellular bridges, masses of keratin, and fragments of desmoplastic stroma were present, closely recapitulating histological features of trichilemmal carcinoma, which enabled diagnosis as metastasis. Cell block showed similar tumour fragments with evidence of differentiation towards outer root sheath. FNAC is the first-line investigation to obtain a tissue diagnosis of masses in the head and neck region. Although rarely encountered, the lack of knowledge of cytological features of trichilemmal carcinoma may hamper its FNAC diagnosis at metastatic sites. When intraparotid metastases occur, they may be mistaken as primary salivary gland carcinoma. Thus, awareness of the cytological features of this tumour must be raised among cytopathologists to enable accurate diagnosis and further management.

Carcinosarcoma of the parotid gland: a case report and review of the literature.

BACKGROUND: Carcinosarcoma of the parotid gland is an extremely rare malignancy comprising of 0.04-0.16% of all salivary gland tumors. This is the first case of an adenoid cystic carcinoma with chondrosarcoma to the best of our knowledge. They consist of distinct carcinomatous and sarcomatous components and may arise de novo or from a preexisting pleomorphic adenoma. CASE PRESENTATION: Herein we present a case of an 80-year-old white female who presented with progressively increasing left facial swelling over 6 weeks. Magnetic Resonance Imagining revealed a mass (3.4 cm) in the parotid gland with a predominant cystic/necrotic component. The cytology was atypical (Milan3) and a total parotidectomy and selective lymph node dissection was done. The resection showed extensive necrosis with high grade sarcomatous (chondrosarcoma) areas. The epithelial component was adenoid cystic carcinoma with perineural invasion. The patient is currently undergoing radiotherapy of the tumor bed and skull base due to propensity of perineural invasion of the adenoid cystic component. The most common carcinomas in carcinosarcomas of salivary glands are adenocarcinoma and squamous cell carcinoma. CONCLUSION: Carcinosarcoma is a high-grade aggressive lesion with a poor prognosis and should be treated aggressively. More studies are needed to understand the origin of these tumors.