Neonatal alveolar lymphangioma: A rare benign oral disease case report and literature review.

BACKGROUND: Neonatal alveolar lymphangioma (NAL) is a rare benign condition most often reported in the oral cavity of neonates of African descent. CASE DESCRIPTION: The authors present a case report of bilateral NAL, including follow-up images at 6 months and 17 months. The clinical differential diagnosis includes dental lamina cyst, hemangioma, congenital epulis of the neonate, and melanotic neuroectodermal tumor of infancy. Key differences to assist in making the distinction are described. PRACTICAL IMPLICATIONS: The distinguishing characteristics of NAL make a clinical diagnosis possible. Accurate clinical recognition of NAL is essential because biopsy is not typically indicated. Because this is a clinical diagnosis, follow-up for 1 year or until complete resolution; biopsy is suggested if clinically indicated for residual or recurrent lesion.

Infantile Hemangioma: A Common Lesion in a Vulnerable Population.

Infantile hemangioma (IH) is important to all dentists, particularly dentists providing services to pediatric populations, because significant morbidity and mortality can occur from vascular lesions in children. Specialists of the oral cavity have the responsibility to identify patients with IH, a lesion that could be potentially life threatening. We present a case of IH and will provide a narrative review of the most recent literature. We discuss the diagnosis, risk stratification, treatment, complications, and impact on routine dental procedures. A proper diagnosis is crucial as oral and perioral IH are considered high-risk due to the increased risk of ulceration and feeding impairment. Referral to a hemangioma specialist for comprehensive team treatment is optimal. The natural history of IH consists of a long proliferative phase, which manifests as clinically visible growth. As a result of early encounters with patients, the pediatric dentist may often be considered the primary care provider.

Selection of mucocele management technique: considerations in pediatric patients.

The objectives of this article are to describe the surgical excision of an oral mucocele in a 9-year-old boy and to examine the literature on mucocele management approaches, highlighting their advantages and disadvantages in terms of a patient's ability to cooperate in the dental chair. In the present case, a 9-year-old boy who was known to be a cooperative patient underwent conventional surgical excision and tolerated the procedure well. A review of the literature from 2010 to 2018 revealed 20 relevant studies outlining a variety of approaches to mucocele management, including surgical excision, intralesional corticosteroid injection, laser removal, micromarsupialization, cryosurgery, and withholding treatment for cases in which a lesion is small and allowed to spontaneously regress. While conventional surgical excision of mucoceles is suitable for cooperative patients, alternative approaches may offer reduced chair time, minimal intraoperative bleeding, and enhanced postoperative recovery. For pediatric patients who demonstrate maladaptive behavior, certain treatment modalities may prove superior to surgical excision for the management of mucoceles.

Vascularized ameloblastoma: A case report and clinicopathologic review of 18 cases from the literature.

The notable features of ameloblastoma do not typically include prominent vascularity. However, on rare occasions, vascular ameloblastoma has been described under a variety of names. We present a case of vascularized ameloblastoma that had a bloody return on fine-needle aspiration. The English language literature contains a total of 16 reports (18 cases) of vascular or hemangioma-like ameloblastoma. We reviewed the clinical, pathologic, and radiographic features of the 19 cases, but further study and more cases are needed. The recognition of this variation of ameloblastoma is important for clinicians to note that fine-needle aspiration with a bloody return does not exclude ameloblastoma from diagnostic consideration. We suggest the term vascularized ameloblastoma to avoid any suggestion of a vascular neoplasm.

Giant peripheral ossifying fibroma: a case report and clinicopathologic review of 10 cases from the literature.

Peripheral ossifying fibroma (POF) is most often a self-limiting, sessile or pedunculated, gingival nodule that is believed to be a reactive rather than neoplastic pathologic process. The lesion is typically <2cm, however it has been recognized that some examples may grow quite large and may displace teeth. The mass-like clinical presentation and radiographic appearance of soft tissue calcification may lead to misclassification; however the histologic appearance is diagnostic. Giant POFs (GPOF) have been referred to in the literature by several other names (large, atypical, huge, gigantiform). The distinguishing characteristics of GPOFs and the factors that contribute to their growth have primarily been explored through case reports. We present a new case of POF that was giant and review 10 previously reported giant lesions, with focus on the clinical presentation, radiographic features, and outcome to explore the possibility that this represents a distinct clinical subset of lesion, with a unique set of features that warrant recognition for accurate diagnosis.

Congenital epulis of the newborn: 10 new cases of a rare oral tumor.

Congenital epulis of the newborn (CEN) is a rare benign lesion that exclusively occurs in the oral and maxillofacial regions of newborns. The clinicopathologic features of CEN were examined and reviewed from the files of the Armed Forces Institute of Pathology from 1970 to 2000. Ten cases were included. Patient lesions were all present at birth but were surgically excised between 2 days and 6 weeks (median, 5.5 days). Nine lesions were in females; 1 case did not designate patient sex. Locations included 6 on the maxilla, 2 on the mandible, 1 on the designated maxillary lip, and 1 unknown. The cases included a patient with 2 lesions: 1 on mandibular and 1 on maxillary alveolar ridges, respectively. All other lesions were solitary and polypoid. Microscopically, these were pedunculated and nodular, composed of sheets to grouped clusters of medium-sized, ovoid-to-polygonal cells with abundant granular cytoplasm, distinct cell membranes, vascular-rich stroma, and attenuated overlying mucosa. Two cases also demonstrated spindled cells. The nuclei were vesicular and focally stippled, with distinct and slightly convoluted nuclear membranes; nucleoli were visible but not prominent. Mitotic activity was not observed. The vascular channels ranged from capillary-sized to venous, some staghorn-like with rare perivascular long-term inflammation. The venules exhibited a perivascular pericytic proliferation. Odontogenic epithelial rests were present in 2 cases. No cases demonstrated cytoplasmic hyaline globules. The lesional cells in all cases were negative for S-100 protein, CD68, CD34, CD31, keratins, desmin, calponin, and smooth muscle actin. Perivenular pericytes were positive for smooth muscle actin. Congenital epulis of the newborn is a rare oral entity with characteristic clinicopathologic features. It predominately affects girls, mainly on the maxillary alveolar ridge. It may be separated from "granular cell tumor" by location, patient age, absence of cytoplasmic hyaline globules, solid growth pattern, pericytic proliferation, attenuated overlying epithelium, and negativity for S-100 protein.

Lipoma of the oral and maxillofacial region: Site and subclassification of 125 cases.

OBJECTIVE: Lipomas and lipoma variants are common soft tissue tumors, but occur infrequently in the oral and maxillofacial region. In this study, we reviewed 125 lipomas in specific oral and maxillofacial locations. We wanted to examine and compare the clinicopathologic features of these tumors. Study design The records from the Oral and Maxillofacial Pathology Registry of the Armed Forces Institute of Pathology from 1970 to the present were searched for cases coded as "lipoma." This study included 125 cases based on location within the oral and maxillofacial region, benign histology, and available clinical information. Subcutaneous and intraosseous lipomas were excluded. The tumors were classified according to the most recent World Heath Organization classification for soft tissue tumors. RESULTS: Of 125 lipomas, 91 tumors occurred in males, 33 in female patients, and 1 of unknown gender. The mean age was 51.9 years, range 9-92 years. Four tumors occurred in pediatric patients (age <18 years). Specific anatomic sites within the oral and maxillofacial region included the parotid region (n=30); buccal mucosa (n=29); lip (n=21); submandibular region (n=17); tongue (n=15); palate (n=6); floor of mouth (n=5); and vestibule (n=2). The mean size of tumors was 2.2 centimeters, range 0.5 to 8.0 centimeters. The mean duration of the tumors prior to excision was 3.2 years, range 6 weeks to 15 years. Most patients presented with an asymptomatic, circumscribed mass. Grossly, most tumors were described as pink and smooth, occasionally mucoid. Histologically, the tumors were subclassified as classic lipomas (n=62); spindle cell/pleomorphic lipomas (n=59); fibrolipoma (n=2), and chondroid lipoma (n=2). Fourteen tumors exhibited secondary changes, such as fat necrosis, atrophy, and prominent hyalinization; 23 tumors were histologically confirmed to be intramuscular. CONCLUSIONS: Lipomas of the oral and maxillofacial region occur most commonly in adult males in the parotid region, followed closely by the buccal mucosa. These tumors are uncommon in children. Interestingly, spindle cell lipomas are common in this region and comprise the majority of our parotid and lip tumors. Angiolipomas were absent in this anatomic region in this study. Secondary changes and atrophy should not be confused with the malignant histologic features of a liposarcoma.

Oral and salivary gland angiosarcoma: a clinicopathologic study of 29 cases.

Angiosarcomas of the oral and salivary gland area are extremely rare, mostly presented as case reports. We wanted to study the clinicopathologic features of a series of oral and salivary gland angiosarcomas. Cases coded as "angiosarcoma" were retrieved from the Oral and Maxillofacial Pathology Department of the Armed Forces Institute of Pathology. Patient folders and pathology were reviewed and recorded; immunohistochemistry and follow-up were obtained. Inclusion required oral or salivary gland location, vasoformative growth, cytologic atypia, mitoses, and vascular markers. Skin, bone, and subcutaneous angiosarcomas were excluded. Primary and secondary (metastatic) oral angiosarcomas were included. The 22 primary angiosarcomas involved tongue (n = 9), parotid (n = 4), lip (n = 4), submandibular gland (n = 3), and 1 each of soft and hard palate. The 7 secondary angiosarcomas involved the gingiva (n = 4) and parotid gland (n = 3). Overall, patient ages ranged from 6-90 years (mean, 55 years). There were 15 males and 14 females. Symptoms included a mass with recent enlargement and bleeding. Tumor sizes ranged from 0.8-7.0 cm (mean, 2.6 cm). Histologically, all tumors were vasoformative; 86% had solid and 17% had distinctive papillary areas. Eight (28%) were classified as the epithelioid subtype. Immunohistochemical stains showed that the tumor cells were positive for Factor VIIIrag in 19/21, CD31 in 16/19, CD34 in 7/12, and Ulex in 1/1. Primary tumors were classified as low grade (n = 7, in all locations except salivary gland), intermediate (n = 7), and high grade (n = 8); all secondary tumors were high grade. Follow-up was available on 14/22 primary and 7/7 secondary angiosarcomas. Of primary tumors, two tongue angiosarcoma patients died at 1 and 9 years, but 4 were alive without disease over a mean of 7.3 years (range, 1-13 years). Four primary salivary gland angiosarcoma patients were alive without disease over a mean of 5.8 years (range, 1-14 years), and 1 had only a late (15 years) metastasis and death (at 20 years). Three primary lip angiosarcoma patients were without disease over a mean of 14.3 years (range, 13-16 years). Of secondary tumors, three salivary gland angiosarcoma patients died within 1 year, and all four secondary gingival angiosarcoma patients died of disease within 3 years. Assessing follow-up of primary oral and salivary gland angiosarcoma patients by grade, 5 patients with high-grade tumors had no evidence of disease over a mean of 7.6 years (range, 1-16 years), 3 patients with intermediate-grade tumors had no evidence of disease over a mean of 12.7 years (range, 11-14 years), 2 patients with intermediate-grade tumors died of disease at 9 and 20 years, 3 patients with low-grade tumors had no evidence of disease over a mean of 6.3 years (range, 1-14 years), and 1 patient with low-grade tumor died of disease at 1 year. Primary oral and salivary gland angiosarcomas, albeit rare, mostly involve the tongue, parotid gland, and lip of adults, often with relatively good outcome. Although the most common angiosarcoma morphology in this area is spindled vasoformative and solid, almost one third of oral and salivary gland angiosarcomas are the rare epithelioid angiosarcoma variant. Most gingival and few parotid angiosarcomas appear to be metastases from other locations, with many patients succumbing to death within 3 years. Despite predominantly high- or intermediate-grade morphology, patients with primary angiosarcoma of the tongue, salivary gland, and lip have a better prognosis than do patients with primary cutaneous or deep soft tissue angiosarcoma, including those patients with secondary oral and salivary gland involvement.

Hemangioma of the salivary gland: a study of ten cases of a rarely biopsied/excised lesion.

Hemangioma is a common soft tissue tumor that frequently occurs in the oral and maxillofacial region including salivary glands, but is rarely biopsied and is therefore often unfamiliar to the surgical pathologist. Our study examined the subclassification and histologic features of salivary gland hemangioma (SGH). Consultative cases coded as hemangioma and located in salivary gland from 1970 to 2000 were retrieved from the Registry of Oral and Maxillofacial Pathology of the Armed Forces Institute of Pathology (Washington, DC). Only cases with histologic evidence of salivary gland involvement were included. Slide material and patient history for all cases were reviewed, subclassification assigned, and histologic features were noted. Ten cases coded as hemangioma with slides and history met our inclusion criteria. Seven cases were the "juvenile hemangioma" subtype in the parotid of infants, ranging in age from 3 to 10 months (mean age, 5.3 months) with a male predominance. These SGH had a distinctive histologic appearance of a cellular proliferation of capillary sized vessels around retained salivary gland ducts. Mitoses were easily identified. Three additional cases in females included an arteriovenous hemangioma of a lip minor salivary gland since birth in a 15-month-old infant and two parotid gland lesions: a lobular capillary hemangioma of a 10-year-old and a cavernous hemangioma in a 51-year-old. The latter three cases grew as replacing masses and lacked retained salivary gland ducts within the lesion, despite glandular tissue at the periphery of the tumor. No SGH cases in our series were identified in the submandibular or sublingual glands. Despite its common occurrence, SGH is relatively rare in our surgical pathology files. The parotid gland is the most common location (90%). Salivary gland hemangioma includes usual hemangioma subtypes, mainly in females, and a distinctive infantile subtype of capillary hemangioma (juvenile hemangioma), displaying distinctive histology and found predominately in males. The cellularity, mitotic activity, and retained salivary gland ducts in the latter lesion should not make one consider malignancy.

Liposarcoma of the oral and salivary gland region: a clinicopathologic study of 18 cases with emphasis on specific sites, morphologic subtypes, and clinical outcome.

Liposarcoma is rare in the oral and salivary gland region (OSG), previously described in only case reports and two small series. Clinicopathologic features of a large series of these tumors were studied. Cases coded as "liposarcoma or lipoma" from 1970 to 2000 were searched for in our files. Inclusion required an OSG location and diagnosis by established soft tissue criteria. Dermal, other soft tissue, and intraosseous liposarcomas were excluded. Clinical and pathologic material was reviewed and follow-up obtained. Eighteen liposarcomas were included: 10 from males and 8 from females. The median patient age was 51 years (range, 30-70 years). Specific anatomic locations included buccal mucosa (n = 7), tongue (n = 4), parotid gland (n = 3), soft tissue overlying the mandible (n = 2), and one each of palate and submandibular gland. The average tumor size was 4.2 cm (range, 1.5 to 6.0 cm). Histologically, most tumors were well differentiated, including one atypical lipoma (n = 10), followed by myxoid (n = 5) and dedifferentiated (n = 3). OSG liposarcomas of all subtypes had increased numbers of lipoblasts. All patients were treated with surgical excision alone. Follow-up on 15 patients (83%) over a mean of 16.5 years (range, 2 to 53 years) revealed that three patients had between one and six local recurrences over periods of 18 months to 6 years. Twelve patients were without recurrence, with a mean follow-up of 12.8 years (range, 2-23 years). No patients, including those with dedifferentiated liposarcoma, had metastases or died of disease. OSG liposarcomas are rare tumors of adults, occurring most commonly in the buccal mucosa, tongue, and then parotid gland. There were no pleomorphic liposarcomas in this series; well-differentiated liposarcoma was the most common subtype, which can locally recur but, even with high-grade dedifferentiation, does not necessarily predict poor outcome. Therefore, OSG liposarcomas have better prognosis than liposarcoma in other soft-tissue locations, perhaps based on smaller size at presentation. Complete local excision and careful patient follow-up, without adjuvant therapy, appears to be the best treatment for OSG liposarcoma.