Pyostomatitis vegetans: report of three cases and review of the literature

Pyostomatitis vegetans is a rare condition of the mouth frequently associated with chronic inflammatory bowel disease. It is characterized by miliary abscesses and superficial erosions affecting a hyperplastic, soft and friable oral mucosa. Three cases are presented here and used as a basis for reviewing the literature and discussing the differential diagnosis, etiology and treatment of this unusual condition.     

Odontodysplasia. Report of two cases and review of the literature.

Odontodysplasia is a rare developmental anomaly affecting the tooth structures in both deciduous and permanent dentitions. The enamel is thin and uneven in thickness, and the detinal tissue surrounds very large pulp chambers. Denticles are present in the pulp organ. The maxilla is involved twice as frequently as the mandible. Most of the affected teeth are in the anterior segments; however, all other teeth can be affected. The cause is unknown. Because of the tendency of the affected teeth to develop abscesses, the most common treatment is extraction. Two additional cases are reported, and the literature is reviewed.     

Pyostomatitis vegetans. Clinical marker of ulcerative colitis

Pyodermatitis-pyostomatitis vegetans (PV), a rare disorder of the skin and oral mucosa, is considered a highly specific marker for inflammatory bowel disease, especially ulcerative colitis. We have presented the case of a patient with PV. This report emphasizes the relationship of PV to inflammatory bowel disease and the importance of the oral lesions as initial presenting signs of systemic disease or activity.     

Glandular odontogenic cyst. Report of two cases and review of the literature

In 1992, the World Heath Organization recognized a new type of cyst called glandular odontogenic cyst (GOC). Clinically, GOC usually presents as a painless, slow-growing swelling. The radiographic appearance is that of a large, multiloculated, well-defined radiolucency. GOC tends to affect the anterior mandible. The dental profession must be aware of the clinical significance of this lesion. The aggressive nature of the lesion has been reported and at least 25% to 55% recur following curettage. If not treated on time, extensive surgery and alteration of function may result. Two new cases are reported.     

Bilateral coronoid hyperplasia. Report of two cases and review of the literature

Two cases of bilateral hyperplasia of the coronoid processes of the mandible are presented. The patients were men and first noticed their symptoms at 14 and 10 years of age, respectively. They were treated successfully by intraoral bilateral coronoidectomies. A review of the literature revealed only forty-nine cases of the condition reported; 43 cases in western countries and 6 in Japan. As described previously, the majority of the cases were men and their symptoms occurred under the age of 19 years.     

Haemangioma of the Zygoma. Report of two cases with a review of the literature.

Zygomatic haemangioma is a rare benign neoplasm occurring most frequently in adult females. The authors, after reviewing the available literature, report two cases which presented with a swelling of the zygoma. Resection of the neoplasm, immediate reconstruction with cranial bone grafts and internal rigid fixation was the treatment of choice. Results two years after surgery are presented.     

Sinonasal undifferentiated carcinoma: Report of two cases and review of the literature

Sinonasal undifferentiated carcinoma is a rare, highly aggressive neoplasm of the paranasal sinuses. Approximately 40 cases of this neoplasm have been reported to date. Microscopically, this neoplasm is composed of medium-sized cohesive cells arranged in nests, ribbons, and trabeculae having hyperchromatic nuclei, often with prominent nucleoli. A high mitotic rate, tumor necrosis, and prominent vascular invasion are conspicuous features. Squamous or glandular differentiation is not observed at the light-microscopic level It appears to be a unique, distinctive clinicopathologic process that must be distinguished microscopically from other, less aggressive “round cell” sinonasal neoplasms. In this article, two additional cases of this neoplastic process are reviewed. Aggressive, multimodal treatment may offer the best chance for local control, palliation, and, ultimately, survival of the patient. The optimal treatment of SNUC, however, has yet to be determined.     

The melanotic neuroectodermal tumor of infancy. Report of two cases and review of the literature

The melanotic neuroectodermal tumor of infancy (MNTI) is a rare childhood neoplasm with an alarming but classical clinical presentation. It appears as a rapidly enlarging mass in the jaws of skull of infants and unless MNTI is considered in the differential diagnosis, the lesion can easily be mistaken for a malignant neoplasm. Although possessing an aggressive growth rate and radiographic appearance, the MNTI almost always behaves in a benign fashion and can be treated with local excision. However, recent reports of malignant behavior as well as of occasional recurrences make close follow-up important. Approximately 139 cases of the MNTI have been reviewed and tabulated with respect to age at discovery, sex, tumor location, length of follow-up, and whether recurrence occurred, 2 additional cases, 1 of which was in the mandible, are presented along with theories of origin and recommended therapy.     

Verrucous carcinoma of the oral cavity. Report of two cases and review of the Japanese literature

2 cases of verrucous carcinoma of the oral cavity are presented. In addition, 19 Japanese patients of verrucous carcinoma of the oral region from the literature are reviewed with respect to several clinical items.     

Melanotic neuroectodermal tumour of infancy: Report of two cases and review of the literature

Melanotic neuroectodermal tumour of infancy (MNTI) is an uncommon tumour affecting predominantly the craniofacial bones of the newborn infants. The neural crest origin of the tumour has been confirmed. MNTI is generally accepted as a benign tumour despite of its rapid and locally infiltrative growth. Recurrence rate varies between 10% and 60%, and malignant behaviour has been reported in 6.5% of MNTIs. Systematic review of the literature revealed 445 MNTIs published between 1918 and 2010. We present additional two cases of MNTI from our Department, typical in all terms, which equals a total number of 447 reported cases. One of our cases revealed histological features consistent with malignant behaviour, but at present, 18 months after the surgical excision, there is no evidence of recurrence. Biological behaviour of MNTI cannot be predicted by gross or histologic characteristics, thus early diagnosis and careful follow-up after the complete surgical excision is required.     

自体移植骨中复发性成釉细胞瘤2例报告及文献复习

报告2例因右下颌骨成釉细胞瘤行右侧下颌骨部分切除或半侧下颌骨切除术后即刻分别行自体髂骨或肋骨移植的病例,2例病例在行X线检查时分别于术后16年和14年在移植骨中发现成釉细胞瘤复发。例1行术中冷冻切片活检,病理学诊断为滤泡型成釉细胞瘤。结合本组2例病例,并对已发表的文献进行复习,认为成釉细胞瘤发生在肿瘤切除植骨后的骨中属于罕见病例,应对其复发机制进行深入研究。     

Ameloblastoma with varied sites of metastasis: Report of two cases and literature review

ObjectiveWe report two rare cases of lung metastasis from maxillary ameloblastoma, in order to review its risk and analyse the types of metastases that can present with this disease.MethodsA 40-year-old male with multiple recurrences and a 46-year-old female, who had undergone successful surgical treatment of a maxillary ameloblastoma, presented with metastatic lesions. The primary tumour and metastases were benign in both patients. We reviewed and analysed 20 cases of the same condition reported in recent years.ResultsOur initial treatment for the primary maxillary lesion was performed more than 10 years before the pulmonary lesions presented. Due to the aggressive nature of this tumour, metastases in the lungs and cervical lymph nodes (male patient) were confirmed.ConclusionThese cases presented a diagnostic challenge due to the multiple and varied sites of recurrence, which indicate the natural behaviour of this tumour. Different routes of metastasis can occur, including implanting, haematogenous, and lymphatic spread. CT-guided percutaneous transthoracic lung biopsy is an important method to confirm metastatic ameloblastoma.     

Rhinocerebral mucormycosis. Presentation of two cases and review of the literature

Mucormycosis is a serious infection with a fulminating course caused by opportunistic fungi. The territory most frequently affected is the maxillofacial region. The disease develops in patients with severe metabolic and immunological disorders. We present two cases that illustrate its severity and the need for early diagnosis and aggressive treatment.     

Pyostomatitis vegetans and its relation to inflammatory bowel disease, pyoderma gangrenosum, pyodermatitis vegetans, and pemphigus

J Oral Pathol Med (2012) 41: 584-588 Pyostomatitis vegetans (PSV) is an intraoral pustular eruption considered by most authors to represent the mucous analogous of cutaneous pyoderma gangrenosum and its vegetating presentations (pyodermatitis vegetans). A strong correlation of PSV with inflammatory bowel disease (IBD) is well documented. The histopathology of PSV lesions usually reveals acanthosis, and neutrophils and/or eosinophils infiltration with intraepithelial or subepithelial abscesses; acantholysis is present in some cases. We studied four patients with IBD that presented oral lesions suggestive of PSV. Two male and two female patients were included. The histopathology of oral lesions of two patients revealed findings typical for PSV. The other two patients showed findings typical for pemphigus vulgaris (PV), although the course of their symptoms paralleled that of the bowel disease. Our findings may suggest that pustular lesions in patients with IBD can be a presentation of both PSV and PV; adequate diagnosis is required because clinical presentation is very similar.