TGF-ß3 from fibroblasts promotes necrotising sialometaplasia by suppressing salivary gland cell proliferation and inducing squamous metaplasia.

Necrotising sialometaplasia (NSM) is a non-neoplastic lesion mainly arising in the minor salivary glands of the oral cavity. In the clinical features, NSM shows swelling with or without ulceration, and can mimic a malignant disease such as squamous cell carcinoma. Histopathologically, NSM usually shows the lobular architecture that is observed in the salivary glands. Additionally, acinar infarction and squamous metaplasia of salivary ducts and acini are observable. The aetiology of this lesion remains unknown, although it has a characteristic feature that sometimes requires clinical and histopathological differentiation from malignancy. In this study, we investigated upregulated genes in NSM compared with normal salivary glands, and focused on the TGF-ß3 (TGFB3) gene. The results of the histopathological studies clarified that fibroblasts surrounding the lesion express TGF-ß3. Moreover, in vitro studies using mouse salivary gland organoids revealed that TGF-ß3 suppressed salivary gland cell proliferation and induced squamous metaplasia. We demonstrated a possible aetiology of NSM by concluding that increased TGF-ß3 expression during wound healing or tissue regeneration played a critical role in cell proliferation and metaplasia. © 2024 The Pathological Society of Great Britain and Ireland.

Necrotizing sialometaplasia: a case report of a non-ulcerated histopathological presentation.

A 27-year-old woman presented with the chief complaint of severe pain in the palate region, which had been present for two months. Upon examination, she was found to have a firm, non-ulcerated nodule measuring about 2.5cm at the palatal junction. Incisional biopsy was recommended because the clinical differential diagnosis was mucoepidermoid carcinoma or squamous cell carcinoma. Anatomopathological examination revealed squamous metaplasia of the salivary gland ducts with preservation of the lobular architecture. Immunohistochemistry showed metaplastic ducts with low reactivity for p53 and Ki67, as well as positivity for CK AE1/AE3, CK7, p63, S-100, and SMA. The final diagnosis was necrotizing sialometaplasia. No treatment is required for this disease. Thirty-nine days after biopsy, total remission was observed with no signs of relapse after two years.

Necrotizing Sialometaplasia and Bulimia: A Case Report.

Bulimia is an eating disorder with a great prevalence in young women. Due to its multifactor ethiology, bulimia has systemic consequences. In the literature, necrotising sialometaplasia is seldom associated with bulimia. Its etiopathogenesis is discussed by several authors; nevertheless, the consensus does not consider the relevance of local trauma associated with induced vomiting. A case of necrotising sialomethaplasia, presented with a single hard palatal ulcer in a bulimic woman is described in the present report. The patient did not present significant systemic laboratorial values, nor physical weight variations, which highlights the relevance of performing a complete medical clinical history when diagnosing this rare pathology.

Necrotizing sialometaplasia can hide the presence of salivary gland tumors: A case series.

OBJECTIVE: Necrotizing sialometaplasia (NS) is an uncommon benign process which affects minor and, more rarely, major salivary glands. While self-limiting, the condition might be clinically and histologically mistaken for malignancy. Furthermore, NS may accompany neoplasms. The aim of this paper was to report a series of Italian patients affected by NS associated with an unusual high presence of neoplasms of minor and major salivary glands. STUDY DESIGN: Clinical and histological features of twelve patients with NS were retrospectively evaluated. RESULTS: Eight patients presented NS of the minor salivary glands of the palate, and two of them had associated neoplasms (pleomorphic adenoma and adenoid cystic carcinoma) at the same site. Four patients had NS of the parotid gland associated with a history of fine-needle aspiration biopsy performed to diagnose parotid neoplasms. These were epithelial-myoepithelial carcinoma, adenoid cystic carcinoma, Warthin's tumor, and oncocytoma, respectively. CONCLUSION: Tumors of minor and major salivary glands might be associated with NS more frequently than previously reported. While NS of the parotid is generally the result of invasive diagnostic procedure and is detected after the excision of the tumor, NS of the minor salivary glands may obscure an underlying neoplasm, resulting in delays in referral or treatment.

Necrotising sialometaplasia: a diagnostic perplexity? An innocent entity to malignant masquerade.

OBJECTIVE: Necrotising Sialometaplasia is a benign self limiting reactive condition of major and minor salivary glands, which can arouse suspicion for malignancy, clinically and histopathologically. Here, we report a case of 38-year-old female with a painful ulcer on the palate. The case enlightens the importance of clinicopathologic correlation and diligent follow up in diagnosis and management of the case.

Necrotizing sialometaplasia of the palatal mucosa in patient with history of anorexia: review and case report.

Necrotizing sialometaplasia is a self-limiting disorder affecting minor salivary glands resembling a malignant process both clinically and histopathologically. The etiology of this rare inflammatory` disorder is related to an ischemic event. Identified risk factors include alcohol abuse, smoking, drugs, denture wearing, injury and systemic diseases. Also reported are cases of necrotizing sialometaplasia associated with bulimia. This paper identifies the whole body incitement among additional risk factors by presenting a case of necrotizing sialometaplasia in an 18 year old patient with the history of anorexia nervosa. Furthermore it describes the effects of extreme cooling of palatal mucosa with ice chips resulting in constriction of blood vessels as the direct cause, reinforcing ischemic etiology of necrotizing sialometaplasia.

Two rare entities in the same palate lesion: hyalinizing-type clear cell carcinoma and necrotizing sialometaplasia.

Hyalinizing clear cell carcinoma is a low-grade malignant epithelial neoplasm of the salivary glands. The tumor has epithelial cells and lacks myoepithelial cells. Necrotizing sialometaplasia is a benign, self-limiting lesion of the salivary glands. The clinical and histologic features mimic those of mucoepidermoid carcinoma or squamous cell carcinoma. The importance of these entities are the rarity of both of them and their potential to be misdiagnosed as other lesions. Pathologists and clinicians should be aware of these entities to prevent misdiagnosis. This is the first clinical report of 2 rare and consecutive different entities of the same location on the hard palate to our knowledge.

[Necrotizing sialometaplasia--diagnostic difficulties--literature review]. / Necrotizing sialometaplasia--trudnosci diagnostyczne--przeglad pismiennictwa.

Necrotizing sialometaplasia is a benign, self-limiting inflammatory process, which concerns small salivary glands. This kind of lesion is very rare, with difficulties in clinical and histopathological diagnosis. Ulceration is the dominating symptom, which may suggest that it is a malignancy. Lesions usually heal spontaneously.

[Necrotizing sialometaplasia]. / Nekrotisoiva sialometaplasia--suun tuumori ei ollutkaan syöpä.

Necrotizing sialometaplasia is a rare inflammatory condition clinically and histologically resembling a malignant tumor. The lesion is usually present on the palatal mucosa, and heals up without treatment during a couple of months. Differential diagnosis is essential in order to avoid excessive therapy. We describe a case where an oral tumor was not a cancer after all.

Necrotizing sialometaplasia in a patient with an eating disorder: palatal ulcer accompanied by dental erosion due to binge-purging.

This report describes a case of necrotizing sialometaplasia (NS) accompanied by significant dental erosion of the maxillary teeth of the palatal surfaces owing to chronic self-induced vomiting. This observation contributed to the determination of an immediate and appropriate provisional diagnosis of NS in a patient with an eating disorder, which subsequently was confirmed histopathologically as NS. The diagnostic challenges presented by NS associated with eating disorders and its management are discussed.

Diagnostic dilemma: our experience with necrotising sialometaplasia.

A man in his 40s presented to our outpatient department with a painful ulcer in the oral cavity for 1 week. After intraoral examination, a single hard palate ulcer, which was non-tender on palpation, was noted. Baseline blood investigations such as haemogram and serological evaluation were within normal limits. Under local anaesthesia, an excisional biopsy was performed. The histopathological examination revealed a reactive necrotising inflammatory process involving minor salivary glands with no cytological atypia. Weekly follow-up was performed and at the end of 4 weeks, complete healing of the lesion had occurred without any further intervention.

Necrotizing sialometaplasia: A report of two cases and review of the literature.

Necrotizing sialometaplasia (NS) affects salivary glands, and despite being a benign condition, its clinical and histopathological features sometimes mimic other malignant pathologies of epithelial origin. This article presents two cases of NS and discusses clinicopathological features and the differential diagnosis of this condition. The first case, a 76-year-old woman with a 6-month history of painful oral thrush. Intraoral examination showed an ulcerative lesion located on the hard palate. The clinical hypothesis was squamous cell carcinoma. Second, a 26-year-old man with a 40-days ulcerative lesion on the soft palate. Intraoral examination revealed a reddish ulcer measuring 0.5 cm. Clinical hypothesis was traumatic ulcer. In both cases, a biopsy was performed, and a histopathological diagnosis of NS was established. NS cause is poorly understood, and its clinical features resemble other oral lesions with ulcerative aspects. Thus, dentists must be aware of the clinical features of oral ulcers with more than a 2-week duration without defined etiology.

Update from the 5th Edition of the World Health Organization Classification of Head and Neck Tumors: Tumours of the Oral Cavity and Mobile Tongue.

The fifth chapter of the upcoming fifth edition of the 2022 World Health Organization Classification of Tumours of the Head and Neck titled Tumours of the oral cavity and mobile tongue, has had some modifications from the 2017 fourth edition. A new section "Non-neoplastic Lesions", introduces two new entries: necrotizing sialometaplasia and melanoacanthoma. The combined Oral potentially malignant disorders and Oral epithelial dysplasia section in the 2015 WHO has now been separated and submucous fibrosis and HPV-associated dysplasia are also discussed in separate sections. Carcinoma cuniculatum and verrucous carcinoma are described in dedicated sections, reflecting that the oral cavity is the most common location in the head and neck for both these entities which have distinct clinical and histologic features from conventional squamous cell carcinoma. This review summarizes the changes in Chapter 5 with special reference to new additions, deletions, and sections that reflect current clinical, histological, and molecular advances.

Case report of necrotizing sialometaplasia.

INTRODUCTION: Necrotizing sialometaplasia is a rare inflammatory necrotizing reactive process that can involve minor and major salivary glands. Gland tissue ischaemia has been proposed as the aetiology. CASE REPORT: A 21-year-old woman was referred with a 1-week history of bilateral painful swelling of the palate, with the development of two deep ulcers after the first 3 days. Ulcer edges were elevated but not indurated, and the bases had a necrotic appearance. An incisional biopsy of the margin of one ulcer confirmed the diagnosis of necrotizing sialometaplasia. The ulcers spontaneously healed within 8 weeks without treatment. DISCUSSION: The clinical and histological similarity between this entity and a malignant lesion implies a risk of unnecessary or inadequate treatment. This case illustrates the need for an incisional biopsy to be analyzed by an experienced pathologist to establish a correct diagnosis.

Necrotizing sialometaplasia of the parotid gland associated with angiocentric T-cell lymphoma: a case report and review of the literature.

A very rare case of necrotizing sialometaplasia of the parotid gland associated with angiocentric T-cell lymphoma was described. A 66-year-old male had left neck and pharyngeal masses and biopsy specimen showed a monotonous proliferation of atypical lymphoid cells with massive necrosis in the parotid gland. Angiocentric pattern or vascular invasion by the lymphoid cells was observed and the involved parotid gland exhibited squamous metaplasia of the ducts and acini; necrotizing sialometaplasia. Immunohistochemical analysis revealed a cytotoxic T-cell phenotype of the lymphoid cells (CD3+, CD4-, CD5+, CD8+, CD56-, Granzyme B+, TIA-1+, Perforin-) but in situ hybridization showed no relation to Epstein-Barr virus. Although necrotizing sialometaplasia is relatively rare in the parotid gland, angiocentric T-cell lymphoma should be considered for a causative condition of necrotizing sialometaplasia.

Necrotizing sialometaplasia of the parotid gland in a dog.

Necrotizing sialometaplasia (NS) is a self-limiting, benign, ischemic, inflammatory disease that is most often described in the submandibular glands of dogs, with clinical and histologic features that resemble malignancy. Unilateral swelling of the parotid salivary gland in a 7-year-old Cocker Spaniel dog was diagnosed as NS. The dog also had otitis externa on the same side as the parotid gland lesions. The main histologic features were included lobular necrosis of salivary tissue; fibrinoid necrosis of some arteries; marked squamous metaplasia of duct and/or acinar epithelium, with intercellular bridge formation; preservation of salivary lobular morphology; and variable inflammation and fibrosis. Etiologic factors for NS in both humans and animals remain obscure.

Necrotizing sialometaplasia of the palate associated with angiocentric T-cell lymphoma.

In this article we present 2 cases of necrotizing sialometaplasia (NS) associated with angiocentric lymphoma of the midline. Immunohistochemical analysis confirmed a T-cell origin, and in situ hybridization in one case revealed its relationship to Epstein-Barr virus. These findings suggest that vascular occlusion by the neoplastic cells produces ischemia, which leads to local infarction contributing to the salivary gland lesion. To our knowledge, the association between angiocentric lymphoma and NS has been previously reported in only one instance, and we suggest that this particular type of lymphoma should be added to the list of related conditions for NS.

Necrotizing Sialometaplasia of the Hypopharynx.

Necrotizing sialometaplasia (NSM) is a benign, reactive metaplastic condition of the minor salivary gland tissue typically seen in the setting of injury, chemical or traumatic, and is nonneoplastic and self-limited. The diagnosis may be challenging as it may clinically mimic malignancy. We present the case of a 74-year-old male with a 1 pack per day smoking history for 60 years who presented with a reported 20-pound weight loss, dysphagia, and dysphonia progressing over the course of 6 months and found to have a 3.5-cm hypopharyngeal mass on computed tomography imaging and fiberoptic laryngoscopy. Initial frozen section of the mass was concerning for squamous cell carcinoma in situ, but permanent specimens returned as nondiagnostic. Repeat biopsy established a diagnosis of NSM. Two-month follow-up showed complete resolution of the mass. Clinicians should be aware that NSM may present in unusual locations when considering differential diagnoses for laryngeal masses and evaluating for malignancy.

[Necrotizing sialometaplasia: diagnosis of a rare entity]. / Die nekrotisierende Sialometaplasie: Diagnostik einer seltenen Entität.

A differential diagnosis of an ulcer on the hard palate must consider a wide variety of diseases and conditions, among them rare entities such as necrotizing sialometaplasia. We report the case of a patient who presented with a painful ulcer on the hard palate. A biopsy taken at the initial presentation of the patient revealed a diagnosis of necrotizing sialometaplasia. Histology showed foci of eosinophilic granulocytes with lobular infarction or necrosis, bland-appearing nuclear morphology of squamous cells, simultaneous metaplasia of ducts and mucous acini. The ulcer resolved spontaneously within seven weeks and required no specific treatment. The case presented here shows that early diagnosis of necrotizing sialometaplasia is important in order to prevent unnecessary treatment.

Sialometaplasia of the soft palate in a 2-year-old girl.

A case of sialometaplasia of the soft palate is reported in a 2-year-old girl 3 months after she had an adenoidectomy. Dental practitioners should be aware of the possible causes of intraoral swellings in both children and adults. The appearance of some conditions in children may differ from their characteristic appearance in adults. Necrotizing sialometaplasia may appear as an aggressive-looking lesion in an adult, possibly resembling squamous cell carcinoma. In the young patient reported here, frank necrosis was not evident from clinical or histological examination. Necrosis may not be part of the presentation of sialometaplasia in such young children.