Carcinoma, Intraductal, Noninfiltrating ; Humans ; Salivary Gland Neoplasms/diagnosis* ; Salivary Glands

Objective: To study the clinicopathologic and genetic features of papillary cystic low-grade mucoepidermoid carcinoma (LG-MEC) and cystadenoma. Methods: A retrospective review was performed on salivary gland tumor patients with papillary cystic architecture who presented to department of oral pathology, Peking University School and Hospital of Stomatology between January 2010 and June 2022. Among this cohort, there were 17 males and 17 females with a range age of 23-82 years [(55.6±14.6) years]. Diagnosis was confirmed by histological, immunohistochemistry (IHC) and fluorescence in situ hybridization (FISH) analysis. Finally, 15 papillary cystic LG-MEC and 19 cystadenoma patients were included in the present study. All patients were followed clinically and radiologically, and the duration of follow-up ranged from 1 to 141 months. Results: All neoplasms showed papillary proliferation with multilocular or giant cystic tumors. Papillary cystic LG-MEC was characterized by epidermoid cells, intermediate cell and mucous cells with multiple lining-layers. Papillary cystic LG-MEC had mild cellular atypia and a pushing infiltration. Cystadenoma was characterized by cuboidal, columnar and ciliated pseudostratified columnar lining epithelium. Squamous metaplasia, mucinous metaplasia and acidophilic degeneration could also be observed focally in cystadenoma. For IHC staining, papillary cystic LG-MEC showed diffusely and strongly positive for mucin 4 (MUC4) (15/15) and mucin 5 Subtype AC (MUC5AC) (4/15) in the epidermoid cells, intermediate cell and mucous cells. The epidermoid cells and intermediate cells were diffusely positive for p40 and p63. The Ki-67 index was about 10%-15% in LG-MEC. As a contrast, p40 (17/19) and p63 (14/15) were only detected in the basal cells of cystadenoma. Cystadenoma showed focal MUC5AC (4/19)expression and MUC4 (19/19)diffuse expression. In addition, the Ki-67 index was 5%-10% in cystadenoma. The MAML2 gene translocation was detected in 11 LG-MEC patients, but none in cystadenoma. Conclusions: The differential diagnosis points between papillary cystic LG-MEC and cystadenoma included the specific epidermoid cells, intermediate cells and mucus cells in LG-MEC, cell atypia, the pushing-infiltration pattern, diffuse expression of p40 and p63 in the lining epithelium, and a MAML2 gene rearrangement. The molecular test of MAML2 should be recommended to reduce missed LG-MEC diagnoses.
Male ; Female ; Humans ; Carcinoma, Mucoepidermoid/pathology* ; In Situ Hybridization, Fluorescence ; Ki-67 Antigen/genetics* ; Biomarkers, Tumor/analysis* ; Salivary Gland Neoplasms/diagnosis* ; Transcription Factors/metabolism* ; Cystadenoma ; Metaplasia

Humans ; Salivary Gland Neoplasms/diagnosis* ; Salivary Glands

Salivary adenoid cystic carcinoma (SACC) is a common malignant tumor in the oral and maxillofacial region and accounts for approximately 3%-5% of all head and neck carcinomas. SACC always occurs in the palatal salivary gland and parotid gland. The tumor has the characteristics of strong invasion, perineural invasion, high hematogenous metastasis, and low lymph node metastasis rate. The biological characteristics of SACC determine the specificity of clinical treatment. Thus far, few clinical trials have investigated the efficacy of systemic therapy owing to the rarity of SACC with lung metastasis. Moreover, long-term results are poor, and no consensus on standard treatment has been reached yet. This systematic review aims to provide a retrospective analysis of treatment options and prognosis for SACC with lung metastasis and evidence for future clinical treatment.
Carcinoma, Adenoid Cystic ; diagnosis ; Cell Line, Tumor ; Humans ; Neoplasm Invasiveness ; Prognosis ; Retrospective Studies ; Salivary Gland Neoplasms ; diagnosis

BACKGROUND: Recent findings in molecular pathology suggest that genetic translocation and/or overexpression of oncoproteins is important in salivary gland tumorigenesis and diagnosis. We investigated PLAG1, SOX10, and Myb protein expression in various salivary gland neoplasm tissues. METHODS: A total of 113 cases of surgically resected salivary gland neoplasms at the National Cancer Center from January 2007 to March 2017 were identified. Immunohistochemical staining of PLAG1, SOX10, and Myb in tissue samples was performed using tissue microarrays. RESULTS: Among the 113 cases, 82 (72.6%) were benign and 31 (27.4%) were malignant. PLAG1 showed nuclear staining and normal parotid gland was not stained. Among 48 cases of pleomorphic adenoma, 29 (60.4%) were positive for PLAG1. All other benign and malignant salivary gland neoplasms were PLAG1-negative. SOX10 showed nuclear staining. In normal salivary gland tissues SOX10 was expressed in cells of acinus and intercalated ducts. In benign tumors, SOX10 expression was observed in all pleomorphic adenoma (48/48), and basal cell adenoma (3/3), but not in other benign tumors. SOX10 positivity was observed in nine of 31 (29.0%) malignant tumors. Myb showed nuclear staining but was not detected in normal parotid glands. Four of 31 (12.9%) malignant tumors showed Myb positivity: three adenoid cystic carcinomas (AdCC) and one myoepithelial carcinoma with focal AdCC-like histology. CONCLUSIONS: PLAG1 expression is specific to pleomorphic adenoma. SOX10 expression is helpful to rule out excretory duct origin tumor, but its diagnostic value is relatively low. Myb is useful for diagnosing AdCC when histology is unclear in the surgical specimen.
Adenoma ; Adenoma, Pleomorphic ; Antibody-Dependent Cell Cytotoxicity ; Carcinogenesis ; Carcinoma, Adenoid Cystic ; Diagnosis ; Immunohistochemistry ; Oncogene Proteins ; Oncogene Proteins v-myb ; Parotid Gland ; Pathology, Molecular ; Salivary Gland Neoplasms ; Salivary Glands ; SOX Transcription Factors ; Translocation, Genetic

Necrobiotic xanthogranulomatous reaction is a multiorgan, non-Langerhans cell histiocytosis with an unknown etiology. Occurrence in the salivary gland is extremely rare. We recently identified a case of necrobiotic xanthogranulomatous sialadenitis in a 73-year-old Korean woman who presented with a painless palpable lesion in the chin. There was no accompanying cutaneous lesion. Partial resection and subsequent wide excision with neck dissection were performed. Pathological examination showed a severe inflammatory lesion that included foamy macrophages centrally admixed with neutrophils, eosinophils, lymphocytes, plasma cells, and scattered giant cells, as well as necrobiosis. During the 12-month postoperative period, no grossly remarkable change in size was noted. Necrobiotic xanthogranulomatous inflammation may be preceded by or combined with hematologic malignancy. Although rare, clinicians and radiologists should be aware that an adhesive necrobiotic xanthogranuloma in the salivary gland may present with a mass-like lesion. Further evaluation for hematologic disease and close follow-up are needed when a pathologic diagnosis is made.
Adhesives ; Aged ; Chin ; Diagnosis ; Eosinophils ; Female ; Follow-Up Studies ; Giant Cells ; Hematologic Diseases ; Hematologic Neoplasms ; Histiocytosis ; Humans ; Inflammation ; Lymphocytes ; Macrophages ; Neck Dissection ; Necrobiotic Disorders ; Necrobiotic Xanthogranuloma ; Neutrophils ; Plasma Cells ; Postoperative Period ; Salivary Glands ; Sialadenitis ; Skin ; Submandibular Gland

Acinic cell carcinoma is a relatively rare salivary gland tumor predominantly occurring in the major glands. Therefore, acinic cell carcinoma rarely occurs in the mandible. In this study, a case of primary acinic cell carcinoma of the mandible was reported, and relevant literature was reviewed. The etiology, clinical symptom, image and histological features, diagnosis, treatment, and prognosis of acinic cell carcinoma in the mandible were discussed.
Carcinoma, Acinar Cell ; diagnosis ; Humans ; Mandible ; pathology ; Prognosis ; Salivary Gland Neoplasms ; diagnosis

PURPOSE: The purpose of the study was to investigate the usefulness of quantitative salivary single-photon emission computed tomography/computed tomography (SPECT/CT) using Tc-99m pertechnetate in Sjögren's syndrome (SS).METHODS: We retrospectively reviewed quantitative salivary SPECT/CT data from 95 xerostomic patients who were classified as either SS (n = 47, male:female = 0:47, age = 54.60 ± 13.16 y [mean ± SD]) or non-SS (n = 48, male:female = 5:43, age = 54.94 ± 14.04 y) by combination of anti-SSA/Ro antibody, labial salivary gland biopsy, unstimulated whole saliva flow rate, and Schirmer's test. Thyroid cancer patients (n = 43, male:female = 19:24, age = 46.37 ± 12.13 y) before radioactive iodine therapy served as negative controls. Quantitative SPECT/CT was performed pre-stimulatory 20 min and post-stimulatory 40 min after injection of Tc-99m pertechnetate (15 mCi). The %injected dose at 20 min and the %excretion between 20 and 40 min were calculated for parotid and submandibular glands, generating four quantitative parameters: %parotid uptake (%PU), %submandibular uptake (%SU), %parotid excretion (%PE), and %submandibular excretion (%SE). The most useful parameter for SS diagnosis was investigated.RESULTS: The uptake parameters (%PU and %SU) were significantly different among the SS, non-SS, and negative controls (p = 0.005 for %PU and p < 0.001 for %SU, respectively), but the excretion parameters (%PE and %SE) were not (p > 0.05 for both). The%PU and%SU were significantly lower in SS than in the negative controls and non-SS (p < 0.05 for all pair-wise comparisons). Additionally, the %SU was significantly lower in non-SS than in the negative controls (p < 0.05). Receiver-operating characteristic analysis revealed that the %SU had the greatest area-under-the curve of 0.720 (95% confidence interval = 0.618–0.807). Using the optimal cut-off value of %SU ≤ 0.07%, SS was identified with a sensitivity of 70.21% and a specificity of 70.83%.CONCLUSION: Reduced submandibular uptake of Tc-99m pertechnetate at 20 min (%SU) was proved useful for the diagnosis of SS. Quantitative salivary gland SPECT/CT holds promise as an objective imaging modality for assessment of salivary dysfunction and may facilitate accurate classification of SS.
Biopsy ; Classification ; Diagnosis ; Humans ; Iodine ; Retrospective Studies ; Saliva ; Salivary Glands ; Sensitivity and Specificity ; Sodium Pertechnetate Tc 99m ; Submandibular Gland ; Thyroid Neoplasms

BACKGROUND: Core needle biopsy is a relatively new technique used to diagnose salivary gland lesions, and its role in comparison with fine needle aspiration cytology needs to be refined. METHODS: We compared the results of 228 ultrasound-guided core needle biopsy and 371 fine needle aspiration procedures performed on major salivary gland tumors with their postoperative histological diagnoses. RESULTS: Core needle biopsy resulted in significantly higher sensitivity and more accurate tumor subtyping, especially for malignant tumors, than fine needle aspiration. No patient developed major complications after core needle biopsy. CONCLUSIONS: We recommend ultrasoundguided core needle biopsy as the primary diagnostic tool for the preoperative evaluation of patients with salivary gland lesions, especially when malignancy is suspected.
Biopsy, Fine-Needle* ; Biopsy, Large-Core Needle* ; Diagnosis ; Humans ; Parotid Gland ; Salivary Gland Neoplasms ; Salivary Glands* ; Submandibular Gland

Humans ; Salivary Gland Neoplasms ; diagnosis ; therapy