Leiomyomatous Hamartomas of the Oral Cavity: Clinicopathological and Immunohistochemical Features of 4 Cases and Literature Review.

Objective. To present 4 new cases of oral leiomyomatous hamartomas (OLH), describing its clinical, microscopical, and immunohistochemical features, and a literature review. Methods. The OLH cases were retrieved from the files of 2 Brazilian and 1 Peruvian oral pathology services. Clinical data were obtained from the pathology reports. Microscopical features were reviewed, and immunohistochemical reactions were performed. A review of the English-language literature about OLH was done. Results. The sample was composed of 3 females (4-, 10-, and 15-year-old) and 1 male (3-year-old). Three cases affected the dorsum of the tongue and one the incisive papilla, and all presented sessile or pedunculated smooth nodules. Microscopically, the lesions were composed of varying-size fascicles of smooth muscle fibers in a fibrous connective tissue background. Immunohistochemistry revealed positivity of the smooth muscle bundles for myogenic markers. The English-language literature review revealed 38 OLH cases reported, most were congenital or diagnosed up to 5 years old. The main locations were tongue and anterior maxilla. Conclusions. Although OLH is a rare lesion, it must be considered in the differential diagnosis of midline tongue or maxillary masses in newborns, infants, and youngsters.

Congenital epulides: a rare case report.

Congenital epulis (CE) or "Granular cell epulis" also previously termed as "Neumann's tumor" is a benign growth arising from the mucosa of the gingiva, typically seen as a mass protruding from the infant's mouth, often interfering with respiration and feeding. These tumors generally present as a single mass arising from the upper alveolus. We report a rare case of two congenital epulides arising from the maxillary and mandibular alveolus in a day old female infant, which was surgically excised, allowing for early initiation of breast feeding. The tumor cells stained negative for S100 protein hence differentiating from other granular cell tumors. The clinical presentation, differential diagnosis in regard to the various neonatal oral swellings and the management of CE has been discussed.

Leiomyomatous hamartoma of the midline maxillary gingival presenting as a congenital epulis: a case report with an immunohistochemical study.

An otherwise-healthy 2-year-old Japanese female presented with a polyp-like lesion on the palatal surface at the incisive papilla. The appearance of the lesion was similar to that of a congenital epulis. The histological findings showed proliferating mesenchymal components that contained mainly smooth muscle admixed with collagen fibres, nerve fibres, small vessels and mucous salivary glands. The immunohistochemical staining findings for α-smooth-muscle actin, desmin and S-100 protein were all positive. The histological diagnosis was therefore leiomyomatous hamartoma, based on clinical microscopic observations.

Management of serious isolated gingival synechia in a newborn: case report and review of the literature.

Congenital synechia of the gums is a rare malformation. The fusion may be partial or complete and may be associated with other facial dysmorphologic malformations. The synechia can be fibrous or bony and may develop between the upper and lower alveolar ridges. The authors report a newborn with partial, but broad and continuous, synechia of the gums and discuss its management during the first days of life.

Malformación vascular intraósea en el maxilar que se presenta en forma de sangrado gingival / Intra-osseous vascular malformation in the maxilla

Las malformaciones arterio-venosas intraóseas (MAVs) en la región maxilofacial son entidades clínicas poco frecuentes. Ofrecen una amplia gama de presentación clínica y no siempre se diagnostican sin sorpresa. El tratamiento de estas lesiones siempre ha representado un reto para el cirujano debido a su intensa vascularización y a la elevada incidencia de recurrencia. Se presenta el caso de una malformación arterio-venosa intraósea en el maxilar superior que se manifiesta como sangrado gingival, cuyos exámenes radiológicos de rutina no fueron concluyentes. Se llevó a cabo la excisión quirúrgica y el empaquetado del hueso con cera, y se realizó el seguimiento del paciente durante 1 año sin que se produjera recurrencia. Las malformaciones vasculares deben tenerse en consideración en el diagnóstico diferencial del sangrado gingival con radiografías dentales no concluyentes(AU)

Intraosseous arterio-venous malformations (AVM) in the maxillofacial region are rare clinical entities. They have a wide range of clinical presentation and are not always diagnosed without a surprise. Treatment of these lesions has always been a challenge to the surgeon due to extreme vascularity and the high recurrence rate. The case presented is that of an intraosseous arterio-venous malformation in the maxilla that started as gingival bleeding and routine radiographic examinations were inconclusive. Surgical excision and bone wax packing was performed there were no recurrences during 1-year of follow-up. Vascular malformations may be considered in differential diagnosis of gingival bleeding with inconclusive dental radiographs(AU)

Dental lamina cyst of newborn: a case report.

Dental lamina cyst, also known as gingival cyst of newborn, is a benign oral mucosal lesion of transient nature. These lesions are usually multiple but do not increase in size. Since the lesions are self-limiting and spontaneously shed a few weeks or months after birth no treatment is required. Clinical diagnoses of these conditions are important in order to avoid unnecessary therapeutic procedure and provide suitable information to parents about the nature of the lesion. In addition, it may be incorrectly diagnosed as natal teeth if present in mandibular anterior region. Here, we present a case of dental lamina cyst of newborn.

Leiomyomatous hamartoma presenting as a congenital epulis.

An otherwise-healthy 11-month-old white girl presented with a polyp-like lesion on the anteromedial part of the maxillary alveolar ridge. It looked like a congenital epulis, but histological examination showed fascicles of smooth muscle cells dispersed in collagenous stroma with a few peripheral nerve bundles that were intermingled with smooth muscle fibres. The muscle cells stained strongly for desmin and alpha-smooth-muscle actin. However, S-100 was found only in peripheral nerve bundles. It was therefore a leiomyomatous hamartoma.

[A rare tumor in a newborn: congenital epulis]. / Yenidoganda nadir bir tümör: dogustan epulis.

A seven-day-old female newborn was examined for a mass protruding from the mouth. Physical examination showed a smooth and pedicled mass with normal mucosal tissue, 3x2 cm in size, originating from the maxillary alveolar edge on the upper incisor and canine teeth. The mass was excised under general anesthesia. Histopathologic diagnosis was congenital epulis. No recurrence was observed during postoperative six months. Congenital epulis is a benign tumor. It should be dealt with surgically if it significantly affects respiratory and alimentary functions.

Congenital epulis.

Congenital epulis is a rare benign hamartoma of the alveolar ridge found in the newborn. The clinical characteristics of this lesion show some variability, and opinion of its pathogenesis lacks consensus. Congenital epulis can cause feeding or respiratory compromise. A case report and review of the literature is presented. The diagnosis, pathology, and treatment are reviewed.

Congenital gum synechiae as an isolated anomaly: a case report.

Congenital adhesions between different parts of the oral cavity rarely occur. These adhesions usually present difficulty in feeding and even respiration soon after birth. In the case presented here the mild form of gum synechia without associated congenital anomaly was treated by excision of the anterior and posterior alveolar bands. Potential jaw and temporomandibular joint development was not compromised and functional problems were eliminated with such a noninvasive procedure.

Congenital fibrous epulis in the infant.

Congenital fibrous epulis is an extremely rare tumor of infancy. It is a benign gingival tumor and generally seen in maxillary alveolar crest and its etiology remains the subject of debate. Congenital fibrous epulis could be considered a hamartomatous lesion. Histologically it does not show the closely packed large granular cells necessary for the diagnosis of an ordinary congenital epulis. Instead, it consists of irregular bundles of collagenous connective tissue, varying numbers of fusiform cells with oval or fusiform shaped nuclei and mild subepithelial inflammatory infiltration with tiny blood vessels and in this case a woven bone spicule in the deep area. Recommended treatment for this tumor is simple excision. We report upon a case of congenital fibrous epulis in a male infant and discuss the clinical features, histopathologic findings, and surgical treatment.

Congenital Fibrous Epulis in the Infant

Congenital fibrous epulis is an extremely rare tumor of infancy. It is a benign gingival tumor and generally seen in maxillary alveolar crest and its etiology remains the subject of debate. Congenital fibrous epulis could be considered a hamartomatous lesion. Histologically it does not show the closely packed large granular cells necessary for the diagnosis of an ordinary congenital epulis. Instead, it consists of irregular bundles of collagenous connective tissue, varying numbers of fusiform cells with oval or fusiform shaped nuclei and mild subepithelial inflammatory infiltration with tiny blood vessels and in this case a woven bone spicule in the deep area. Recommended treatment for this tumor is simple excision. We report upon a case of congenital fibrous epulis in a male infant and discuss the clinical features, histopathologic findings, and surgical treatment.

[Benign gingival granular cell tumor in the newborn: congenital epulis]. / Tumor gingival benigno de células granulares en el recién nacido: epulis congénito.

Gingival granular cell tumor, or congenital epulis, was first described by Neumman in 1871 and subsequently 201 cases have been published in 173 patients. It is an uncommon benign tumor that is present like a pedunculated, smooth surfaced, isolated lesion on the alveolar mucosa of the maxillar of the newborn child. Its firm consistency and variable size can occasionally cause problems in the child's breathing or feeding. This tumor is easily diagnosed clinically and, although spontaneous regression of the tumor mass has occasionally been reported, the current treatment is surgical removal. Two new cases of congenital epulis are reported and a literature review is included.