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 of the parotid gland associated with facial nerve paralysis.

INTRODUCTION: Necrotizing sialometaplasia is a benign inflammatory lesion involving most frequently the minor salivary gland of the hard palate. Involvement of the parotid gland is rare, involvement of the parotid gland associated with facial palsy is exceptional. CASE REPORT: A 56-year-old male patient with Marfan syndrome presented with swelling and inflammation of the left parotid gland associated with progressively complete facial nerve paralysis. CT scan and MRI showed a parotid collection with hyper signal of the nearest tissues associated with erosion of the styloid process. A malignant tumor was suspected. The histological examination of a biopsy showed a lobulocentric process with necrosis, squamous metaplasia, and inflammation. The immunohistochemical examination supported a final diagnosis of necrotizing sialometaplasia. DISCUSSION: Necrotizing sialometaplasia of the parotid gland associated with facial nerve paralysis presents like a malignant neoplasm, both clinically and histologically. Only advanced immunohistochemical examination can really confirm the diagnosis.

Bilateral necrotizing sialometaplasia of the hard palate in a patient with bulimia: a case report and review of the literature.

Necrotizing sialometaplasia (NS) is a rare and benign lesion that mostly affects the posterior hard palate. Its importance resides in its clinical and microscopic characteristics, which can closely mimic malignant neoplasias, in particular oral squamous cell carcinoma and mucoepidermoid carcinoma. Accurate histopathologic evaluation of an incisional biopsy is considered as the diagnostic gold standard. NS lesions heal spontaneously within weeks, and no further treatment is necessary. We report a case of a bilateral palatal NS in a 22-yearold woman with bulimia, where an incisional biopsy confirmed the clinical diagnosis. The different clinical stages of the lesions from onset to resolution and the possible etiologic factors are described in detail, as well as a discussion of the differential diagnoses of palatal ulcers. When taking a biopsy from suspicious oral lesions, care has to be taken that an appropriate tissue sample is harvested, and the histopathologic analysis is performed by an experienced pathologist to establish a correct diagnosis.

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.

HIF-1α, VEGF, and EGFR: contributing factors in the pathogenesis of necrotizing sialometaplasia.

OBJECTIVES: Necrotizing sialometaplasia (NS) is an uncommon reactive lesion involving the minor salivary glands. This study aimed to investigate the expression of hypoxia-inducible factor alpha (HIF-1α), vascular endothelial growth factor (VEGF), and epithelial growth factor receptor (EGFR) in the pathogenesis of NS. METHODS: Paraffin-embedded tissue sections from 10 cases of NS were immunohistochemically stained for HIF-1α, VEGF, and EGFR. A semiquantitative morphometric analysis was performed and compared with normal palatal salivary glands and traumatic ulcerations. RESULTS: Hypoxia-inducible factor alpha staining was observed in most elements of the affected area, the acini and ducts of the involved salivary glands as well as in the inflammatory infiltrate, the endothelial cells, and stromal cells. HIF-1α was almost absent in the control glands (P < 0.0001). VEGF staining was positive in the stromal capillaries and in the inflammatory infiltrate. The expression was higher in cases of NS compared with the normal salivary glands (P < 0.001). EGFR was expressed in the surface epithelium, the pseudo-epitheliomatous hyperplasia, and the islands of squamous metaplasia. VEGF expression in traumatic ulcerations was lower than that in cases of NS. CONCLUSION: This study provides molecular evidence to the role of hypoxia in NS; HIF-1α, the main regulator of hypoxia, was expressed in the infarcted salivary glands, EGFR in the metaplastic epithelium and VEGF in the stromal capillaries, all three components are the key factors induced by hypoxia.

Necrotizing sialometaplasia in an HIV positive cocaine user: a case report.

The aim of this paper was to present a case report of a male patient attending a Semiology and Stomatology Clinic with an erythematous ulcerated lesion on his palate. The patient reported that he was HIV positive as well as being addicted to cocaine. After a biopsy and a histopathological exam, he was diagnosed as having necrotizing sialometaplasia. The lesion diminished spontaneously in thirty days after the exam. Correct diagnosis as well as physical and complementary exams are paramount to avoid any incorrect therapy. As drug addiction and HIV infection have both been associated to necrotizing sialometaplasia, as in the present case, it is difficult to establish if the aetiological factor was drug usage or the HIV infection or even, the combination of these two factors. Although considering the influence of HIV infection on the oral health, we may assume that, at least, it favored the onset of this oral lesion.

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.

A case of necrotizing sialometaplasia involving bilateral parotid glands.

Necrotizing sialometaplasia of the parotid gland is infrequent and can be mistaken as a malignant disease. Its etiology is thought to be an insufficient blood supply. Bilateral involvement of this disease in parotid glands has been rarely reported in the English literature. We report a case of necrotizing sialometaplasia diagnosed after superficial parotidectomy due to bilateral parotid masses in a 69-year old heavy smoking female.

The clinical, histologic, and treatment spectrum in necrotizing sialometaplasia.

OBJECTIVES: The aim of this report was the clinical and histologic characterization of necrotizing sialometaplasia. STUDY DESIGN: We performed a retrospective case series analysis. RESULTS: The study included 4 women 29-71 years old. Possible contributing factors (drugs, alcohol abuse, bulimia, smoking, and pancreatic cancer) were identified. Patients presented with unilateral or bilateral rapidly progressing painful palatal ulcers. Necrotic salivary glands and inflammation were universal microscopic features; ductal metaplasia was present in only 1 case. Thrombosis and heavy fungal and bacterial overgrowth were observed in 1 case. In 3 of the cases the lesions healed within 4-6 weeks under conservative supportive care, whereas in 1 case persistent enlargement up to 25 mm diameter was observed. Surgical debridement combined with a palatal guard resulted in complete healing within 12 weeks. CONCLUSIONS: Significant variations may be observed in both clinical and microscopic manifestations of necrotizing sialometaplasia. Although this disease is considered to be self-limiting in the majority of cases, surgical intervention can be considered in unusually large cases.

Benign or malignant? Case report.

INTRODUCTION: Necrotizing sialometoplasia (NS) is a disease in which the clinical and sometimes even histological features can be confused with those of a malignant tumor, but which is in fact a self-limited inflammatory lesion of the salivary glands. CASE REPORT: The authors present a case study of a 43-year-old female patient who came to the ENT Emergency Department with a painful ulcerated lesion of the hard palate. The lesion was 8 days old and had appeared after an abdominoplasty. Given the clinical need to rule out a malignant neoplasm, a biopsy was performed: the histological result was NS. DISCUSSION/CONCLUSION: The authors make a literature review and stress the importance of differentiating between NS and salivary gland neoplasia. Histology is essential in case of ulcerated neoformation of the oral cavity, to adapt treatment, avoiding unsuitable approaches.

Necrotizing sialometaplasia: a practical approach to the diagnosis.

CONTEXT: Necrotizing sialometaplasia is a benign, self-limited lesion of both major and minor salivary glands, although more commonly the latter. It can represent a diagnostic dilemma and may be mistaken for a malignant neoplasm, such as mucoepidermoid carcinoma, as well as invasive squamous cell carcinoma. A major causal relationship has been ascribed to ischemia. Bulimia, an eating disorder with increasing prevalence in our society, may also be an underlying underreported cause. OBJECTIVE: To discuss the potential pathogenesis, diagnostic pitfalls, and the application of immunohistochemistry as an aid in the diagnosis of necrotizing sialometaplasia. DATA SOURCES: This report uses a previously published case history for illustrative purposes and a review of the current literature. CONCLUSIONS: The diagnosis of necrotizing sialometaplasia may be difficult and is reliant upon a well-oriented biopsy section and a complete clinical history. Diagnosis may be further supplemented via immunohistochemistry, demonstrating focal to absent immunoreactivity for p53, low immunoreactivity for MIB1 (Ki-67), and the presence of 4A4/p63- and calponin-positive myoepithelial cells. Interpreted in context collectively, these findings may be helpful adjuncts in the diagnosis of necrotizing sialometaplasia; nonetheless, to date, hematoxylin-eosin staining remains the gold standard.

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.

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.

Necrotizing sialometaplasia associated with bulimia: case report and literature review.

Necrotizing sialometaplasia (NSM) is a self-limiting disorder affecting mainly the minor salivary glands. The significance of NSM resides in its clinical and histopathological resemblance to carcinoma. Few cases of NSM associated with eating disorders have been reported to date. We present here the clinical features and histomorphology of an additional case of bulimia-associated NSM closely mimicking an invasive carcinoma. A high index of suspicion and good communication between clinician and pathologist are essential in recognizing this entity and preventing unnecessary surgical therapy.

Non-neoplastic salivary gland diseases.

A wide range of non neoplastic disorders can affect the salivary glands, although the more common are: mumps, acute suppurative sialadenitis, Sjögren's syndrome and drug-induced xerostomia. Salivary dysfunction is not a normal consequence of old age, and can be due to systemic diseases, medications or head and neck radiotherapy. Diagnosis of salivary disorders begins with a careful medical history, followed by a cautious examination. While complaints of xerostomia may be indicative of a salivary gland disorder, salivary diseases can present without symptoms. Therefore, routine examination of salivary function must be part of any head, neck, and oral examination. Health-care professionals can play a vital role in identifying patients at risk for developing salivary dysfunction, and should provide appropriate preventive and interventive techniques that will help to preserving a person's health, function, and quality of life. The present work provides an overview of most of the non neoplastic disorders of the salivary glands, in which the general presentation, pathology, and treatments are discussed.

Sialosis and necrotising sialometaplasia in bulimia; a case report.

Salivary gland involvement, particularly salivary gland enlargement (sialosis), is a recognised complication in bulimia. We report the rare association of sialosis and necrotising sialometaplasia with bulimia in the same patient. The association of sialosis and necrotising sialometaplasia in the same patient with bulimia has been reported previously in two patients and may be coincidental, but the appearance in this additional patient suggests it may be prudent to explore this further.