An unusual case of generalized severe gingival enlargement during pregnancy.

Increased hormone levels that are present during puberty and pregnancy are associated with localized or generalized gingival enlargement. This article reviews the gingival alterations that can occur during pregnancy and describes a case of generalized severe gingival enlargement associated with pregnancy and its management. A 36-year-old woman had severe bilateral gingival enlargement of short duration. The patient denied taking any medications. The laboratory report revealed no systemic abnormalities; however, the report disclosed that she was pregnant. Surgical therapy for the gingival enlargement included gingivectomy and gingivoplasty of all quadrants, which reduced the size of the enlarged gingiva. Postoperative visits demonstrated uneventful healing, with no recurrence seen at the one-year follow-up appointment. It appears that the English literature includes only one other case report that discusses generalized gingival enlargement during pregnancy. Pregnancy-related gingival enlargement should be included as a differential diagnosis in women who have non-drug-induced generalized gingival enlargement.

Extra Nodal Rosai-Dorfman Disease Originating in the Nasal and Paranasal Complex and Gnathic Bones: A Systematic Analysis of Seven Cases and Review of Literature.

Rosai-Dorfman disease (RDD) is a benign, self-limiting histiocytosis of unknown etiology. The classic form of the condition includes a painless cervical lymphaenopathy accompanied by fever, weight loss and an elevated ESR. Extra nodal RDD (ENRDD) is most frequent in the head and neck. Thirty-eight cases of ENRDD have been described. Seven cases of ENRDD were identified in our pathology biopsy services. The demographic and clinical information was tabulated logically on the basis of age, gender, location and presence or absence of symptoms, treatment and follow-up. Radiographic and histopathological features were also examined. The findings in these cases were correlated with those available from the previously reported cases. Six cases affected women and one case was diagnosed in a male. The age ranged from 22-55 years. Three cases presented as a nasal mass. One of these lesions extended into the paranasal sinuses. One case was located in the maxilla and extended to involve the maxillary sinus. Three cases were diagnosed in the mandible. The maxillary and one mandibular lesion (Case 2) resulted in significant painful irregular bone destruction with a non-healing socket and tooth mobility respectively. One mandibular lesion was asymptomatic (Case 6). The third case affecting the mandible presented as a rapidly expansile mass following a tooth extraction (Case 7). Nasal masses presented with symptoms of obstruction. Nasal masses were excised with no recurrence from up to 2-3 years of follow-up. The mandibular lesions were curetted aggressively. The oral mass in Case 7 was excised synchronously. No recurrence up to 2 years was recorded in Case 2. Follow-up information is not available for Cases 6 and 7. The maxillary lesion was not intervened surgically. The patient has persistent but stable disease for a follow-up period of 2 years. ENRDD is rarely considered in the differential diagnosis in the absence of lymph node involvement. Lesions of ENRDD resemble many other histiocytic and histiocyte-rich lesions of the head and neck. This makes the diagnosis of ENRDD challenging with the potential for under diagnosis or misdiagnosis and delay in treatment.

Intraoral cellular blue nevus: report of a unique histopathologic entity and review of the literature.

The blue nevus is found most frequently on the skin; however, in rare instances, it has been reported on oral mucous membranes. Intramucosal nevi make up more than one half of all reported intraoral melanocytic nevi. The common blue nevus is the second most common variant. Among the 3 variants of blue nevi, the cellular variant occurs less frequently than the common and combined variants. We present a rare case of intraoral cellular blue nevus that occurred on the oral mucosa of the hard palate. Because of the clinical and microscopic resemblance of the cellular blue nevus to melanoma and the rarity of this lesion in the oral cavity, recognition and accurate diagnosis are critical.

Gingival involvement in Crohn disease.

BACKGROUND: Although the oral manifestations of Crohn disease are well-established, there is little specific documentation of the gingival involvement. CASE DESCRIPTION: The authors describe four patients with significant gingival involvement and identify clinical signs and symptoms of the disease involving the gingivae, along with other oral manifestations. Patients had persistent gingival lesions manifesting as pustular ulcerations, erythema, swelling and cobblestoning. The authors also discuss the differential diagnosis, treatment options and prognostic factors. CLINICAL IMPLICATIONS: Patients with gingival and/or other oral lesions with or without other constitutional symptoms should be evaluated for Crohn disease. Dentists can play a critical role in the early diagnosis, and they can help prevent complications and improve the prognosis.

Effects of simvastatin gels on murine calvarial bone.

BACKGROUND: The cholesterol-lowering drug simvastatin has been shown to stimulate murine calvarial bone growth after multiple injections. The purpose of this study was to test if similar bone stimulation could be induced by 2 single-dose drug delivery systems appropriate to periodontal therapy. METHODS: ICR Swiss mice were treated with the following protocols: 1) injection of methylcellulose gel alone, subcutaneously over the calvarium (INJ-GEL; n = 8); 2) injection of gel with simvastatin (INJ-SIM; 2.2 mg, n = 16); 3) polylactide membrane (PLA) containing gel alone implanted over calvarium (MEM-GEL; n = 10); 4) implanted PLA membrane containing gel and simvastatin (MEM-SIM; n = 10); and 5) untreated mice (n = 12). Animals were sacrificed after 22 or 44 days, calvaria decalcified and stained with hematoxylin and eosin, and images digitized and measured for bone thickness and area. Data were compared using analysis of variance. RESULTS: INJ-SIM stimulated a 53% (P = 0.02) increase at the thickest point of calvarial bone, while MEM-SIM caused a highly significant (P < or = 0.0005) increase in bone thickness (159% to 172%) and bone area (144% to 180%) compared to gel controls. Simvastatin gels caused soft tissue inflammation, which appeared to be related to bone increases. If INJ-SIM animals showing leakage of gel and/or no inflammation were excluded from analysis, INJ-SIM resulted in more bone (58% to 83%) than gel controls. An insignificant amount of SIM-stimulated bone was lost over the long term (44 days). CONCLUSIONS: A single, high dose of simvastatin gel can stimulate murine cranial bone apposition, particularly when delivered under an occlusive membrane. Both approaches should be investigated further for possible development for periodontal therapy.

Lymphoepithelioma-like carcinoma of head and neck skin: a systematic analysis of 11 cases and review of literature.

Lymphoepithelioma-like carcinoma of the skin (LELCS) is a rare tumor of unknown etiology, low malignant potential, and microscopic resemblance to undifferentiated nasopharyngeal carcinoma. Clinically, it presents as a flesh-colored firm nodule or plaque on the face, scalp, or shoulder of middle-aged to elderly individuals. Histologically, LELCS is composed of islands of enlarged epithelial cells with large vesicular nuclei surrounded and permeated by a dense lymphoplasmacytic infiltrate. LELCS exhibits immunoreactivity with high-molecular-weight cytokeratins and epithelial membrane antigen, indicating the epithelial origin. The differential diagnosis includes basal cell carcinoma, squamous cell carcinoma, lymphoma, pseudolymphoma, and Merkel cell carcinoma. We report 11 cases of LELCS of the head and neck region with discussion of the clinical, histopathologic, immunohistochemical, and therapeutic aspects of this rare cutaneous neoplasm. In addition, we systematically review and compare the findings with the previously published cases of LELCS. This study is the largest case series of LELCS reported in the English-language literature. It attempts to more clearly define the diagnostic criteria for LELCS. Its histomorphologic and immunophenotypic features help distinguish this tumor from similar-appearing malignancies, including metastatic nasopharyngeal carcinoma.

Hereditary paraganglioma of the nasopharynx.

Head and neck paragangliomas are rare neuroendocrine tumors derived from neural crest cells of parasympathetic ganglia or the widely dispersed neuroendocrine cells of the head and neck region. Paragangliomas of the sinonasal tract and nasopharynx are rare. The clinicopathologic features of this unique example of a hereditary, nasopharyngeal paraganglioma, and selective entities that are included in its differential diagnosis are presented.

Xerostomia and lichenoid reaction in a hepatitis C patient treated with interferon-alpha: a case report.

Hepatitis C virus (HCV) infection is a liver disease characterized by the development of necrosis, inflammatory changes, and progressive liver fibrosis, leading to complications including cirrhosis, end-stage liver disease, and hepatocellular carcinoma. The clinical features resemble those of other forms of acute viral hepatitis, namely, malaise, nausea, abdominal discomfort, pale stools, dark urine, and jaundice. The most frequently reported extrahepatic manifestations of HCV are lichen planus, sialadenitis, and cutaneous lesions. Sjogren's syndrome-like symptoms and lichenoid reactions have been previously reported in association with hepatitis C. This article describes a case of sicca-like syndrome and oral lichenoid reaction associated with interferon-alpha therapy for HCV infection. In this unique case, significant oral symptoms arose right after initiation of interferon-alpha treatment and resolved completely within days upon completion of treatment with interferon-alpha. Physicians and oral health care specialists should be aware of the association among HCV infection, interferon-alpha therapy, and development of possible oral signs and symptoms including lichenoid lesions and xerostomia.

Colloid milium of the oral cavity: a rare presentation.

Colloid milium (CM) is a rare condition characterized by the presence of multiple dome-shaped amber- or flesh-colored papules developing on sun-exposed skin. It is a degenerative condition linked to excessive sun exposure and possibly exposure to petroleum products and hydroquinone. The origin of the colloid deposition in the dermis is uncertain, although it is thought to be due to degeneration of elastic fibers. The condition is rare, with only 100 case reports documented in the world literature. The most commonly involved sites are the face, periorbital region, backs of the hands, back and sides of the neck, and ears. Involvement of the oral cavity is extremely rare. The purpose of this article is to report an extremely rare first case of CM of the oral cavity and to discuss the clinical, pathologic and therapeutic spectra of CM.

Post-transplant lymphoproliferative disorders of oral cavity.

Post-transplant lymphoproliferative disorders (PTLD) are long-term complications of immunosuppression after solid organ/bone marrow transplantation. In most cases, PTLD arises as a result of primary or reactivated Epstein-Barr virus infection in a host with impaired cellular immunity. PTLD is most often seen in the gastrointestinal tract, although it has also been reported in other organ systems, including the central nervous system and, rarely, in the head and neck. It is characterized histologically by abnormal lymphoid cell proliferation. Although many forms of PTLD do not meet all of the histologic criteria of lymphoma, they often behave clinically in a malignant fashion if left untreated. We present 3 rare cases of PTLD manifesting in the oral cavity as mucosal masses after solid organ transplantation. There are only 8 published reports of PTLD in the literature presenting as oral lesions. The clinical, pathologic, and therapeutic spectra of PTLD are discussed.

Labial salivary gland biopsies in Sjögren's syndrome: still the gold standard?

OBJECTIVES: The accuracy and diagnostic benefits of the labial salivary gland (LSG) biopsy for Sjögren's syndrome (SS) have received mixed reviews. This study was conducted to assess (1) the inter-rater agreement among 5 pathologists, and (2) the relationship between biopsy findings and clinical disease parameters. STUDY DESIGN: Three oral pathologists (OP) and two surgical pathologists (SP) provided independent diagnoses, focus scores, and plasma cell characterizations for 37 LSG biopsies. Inter-rater reliability was assessed using percentage of overall agreement and intraclass correlation coefficients. Relationships between diagnoses and clinical parameters were assessed by nonparametric correlations. RESULTS: Overall agreement among the pathologists was poor, although the intra-specialty agreement was good. The ratings of OP were most highly correlated with serological measures, while those of SP were correlated with salivary flow rate and disease damage. CONCLUSION: Since the LSG biopsy can be the determining factor in SS diagnoses, these demonstrated inconsistencies merit further consideration.

Cicatricial pemphigoid with severe gingival and laryngeal involvement in an 18-year-old female.

Cicatricial pemphigoid (CP), also known as mucous membrane pemphigoid, is an autoimmune vesiculobullous disease occurring mostly in elderly people and seldom occurring in individuals under the age of 20 years. It predominantly affects the mucosal surfaces, primarily the oral and conjunctival mucosa. Uncommonly, the upper aerodigestive tract is involved, which can lead to life-threatening complications. We present the case of an 18-year-old girl with desquamative gingivitis and severe laryngeal webbing and stenosis, caused by cicatricial pemphigoid. Airway management necessitated a tracheostomy and additional surgical procedure. The clinical, diagnostic, and therapeutic features of this disease are discussed, focusing on the oral and unique laryngeal manifestations.