Department of Oral Pathology, Medicine and Radiology Works

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Oral Lichen Planus: Clinical Presentation and Management
(2002-09) Edwards, Paul C.; Kelsch, Robert
C-kit expression in the salivary gland neoplasms adenoid cystic carcinoma, polymorphous low-grade adenocarcinoma, and monomorphic adenoma
(2003-05) Edwards, Paul C.; Bhuiya, Tawfigul; Kelsch, Robert D
Objective. Differentiating between adenoid cystic carcinomas (ACCs), polymorphous low-grade adenocarcinomas (PLGAs), and the monomorphic adenomas (including canalicular adenomas, trabecular adenomas, and basal cell adenomas) can present a diagnostic challenge, especially when examining tissue obtained from small incisional or fragmented biopsies. Recent studies have revealed that overexpression of the tyrosine kinase receptor protein c-kit occurs in a narrow subset of malignant neoplasms, including gastrointestinal stromal tumors, myeloid leukemias, seminomas, and ACCs. C-kit reportedly is not expressed in PLGAs. We compared the expression of the c-kit antigen in the malignant salivary gland neoplasms ACC and PLGA with its expression in salivary gland monomorphic adenoma (including canalicular adenoma and basal cell adenoma). Study design. Formalin-fixed paraffin-embedded sections of 49 salivary gland neoplasms (17 monomorphic adenomas, 17 PLGAs, and 15 ACCs) accessioned between 1989 and 2002 were retrieved from the files of the Department of Pathology, Long Island Jewish Medical Center, and were stained with an anti-c-kit polyclonal antibody. Results. C-kit reactivity was uniformly positive in the cytoplasm of luminal neoplastic cells in ACCs (15/15, 100%). Positive reactivity was also identified in the majority of PLGAs (16/17, 94%), with at least 25% of the tumor cells being positive. Similar reactivity was seen in monomorphic adenomas (16/17, 94%). Conclusions. In contrast to previous reports, we find that c-kit expression was not restricted to ACC but was expressed in all 3 tumor types evaluated (ACC, PLGA, and monomorphic adenoma). Therefore, c-kit does not appear to be a useful marker for distinguishing between either ACC and PLGA in equivocal cases, or in benign and malignant salivary gland neoplasms.
Dermoid cysts of the tongue: report of five cases and review of the literature
(2003-11) Edwards, Paul C.; Lustrin, Liz; Valderrama, Elsa
Dermoid cysts of the tongue are uncommon. To date, there have been nine reported cases in the English language literature. In this article, we describe five cases accessioned at our institution over a 12-year period, two of which have previously been reported. The prevalence of dermoid cysts at our institution over this period was quite low. Of 324,042 surgical cases, 0.24% (765 cases) were dermoid cysts. Of these, five were from the tongue, representing only 0.7% of the dermoid cysts accessioned and 0.0015% of the total surgical specimens. The literature is reviewed and the possible origin of these lesions is discussed.
Assessment of p63 expression in the salivary gland neoplasms adenoid cystic carcinoma, polymorphous low-grade adenocarcinoma, and basal cell and canalicular adenomas
(2004-05) Edwards, Paul C.; Bhuiya, Tawfiqul; Kelsch, Robert D
Purpose The purpose of this study was to determine the extent of p63 immunoreactivity in the malignant salivary gland neoplasms adenoid cystic carcinoma (ACC) and polymorphous low-grade adenocarcinoma (PLGA) and to compare this to the expression of this marker in the benign salivary gland tumors canalicular adenoma and basal cell adenoma. Few studies on the expression of p63 in head and neck salivary gland tumors have been published to date. P63, a selective immunohistochemical marker of basal/stem cells of stratified epithelium and of myoepithelial cells, is a p53 homologue that plays an essential role in both morphogenesis of epidermis and limb development. P63 immunoreactivity has been demonstrated in squamous cell and urothelial carcinomas. It is generally absent in most nonsquamous cell carcinomas. Study design Formalin-fixed paraffin-embedded sections from 49 salivary gland neoplasms, representing 6 canalicular adenomas, 11 basal cell adenomas, 17 PLGA and 15 ACC accessioned from 1989 to 2002 by the Department of Pathology, Long Island Jewish Medical Center, New Hyde Park, NY, were stained with an anti-p63 monoclonal antibody. Results Nuclear p63 reactivity was uniformly positive in PLGA (17/17, 100%). Positive reactivity was also identified in the majority of cases of ACC (13/15, 87%), primarily in the nonluminal myoepithelial-like cells surrounding luminal cells. Canalicular adenoma did not exhibit any p63 immunoreactivity. All basal cell adenomas of parotid origin stained strongly for p63, with staining localized to the peripheral tumor cells situated adjacent to the connective tissue stroma. None of the basal cell adenomas originating in the upper lip stained with p63. In native adjacent salivary gland tissue, p63 reactivity was identified focally in the nuclei of myoepithelial and basal duct cells. Conclusions P63 is strongly expressed in basal cell adenoma of parotid origin, and in ACC and PLGA. Canalicular adenoma did not demonstrate p63 staining, consistent with this tumor's putative luminal ductal cell differentiation. Our results suggest that the neoplastic cells in PLGA may represent either a population of p63-positive epithelial stem/reserve cells similar to the basal cells of stratified epithelium, or modified myoepithelial cells. Given the staining pattern of the tumors examined, p63 does not appear to be an ideal marker for distinguishing between ACC, PLGA, and basal cell adenoma.
Sonic hedgehog gene-enhanced tissue engineering for bone regeneration
(2004-10) Edwards, Paul C.; Ruggiero, Salvatore; Fantasia, John; Burakoff, Ronald; Moorji, Sameer; Razzano, Pasquale; Grande, Daniel A; Mason, James M
Improved methods of bone regeneration are needed in the craniofacial rehabilitation of patients with significant bone deficits secondary to tumor resection, congenital deformities, and prior to prosthetic dental reconstruction. In this study, a gene-enhanced tissue-engineering approach was used to assess bone regenerative capacity of Sonic hedgehog (Shh)-transduced gingival fibroblasts, mesenchymal stem cells, and fat-derived cells delivered to rabbit cranial bone defects in an alginate/collagen matrix. Human Shh cDNA isolated from fetal lung tissue was cloned into the replication-incompetent retroviral expression vector LNCX, in which the murine leukemia virus retroviral LTR drives expression of the neomycin-resistance gene. The rat beta-actin enhancer/promoter complex was engineered to drive expression of Shh. Reverse transcriptase-polymerase chain reaction analysis demonstrated that the transduced primary rabbit cell populations expressed Shh RNA. Shh protein secretion was confirmed by enzyme-linked immunosorbent assay (ELISA). Alginate/ type I collagen constructs containing 2 times 106 Shh-transduced cells were introduced into male New Zealand White rabbit calvarial defects (8 mm). A total of eight groups (N=6) were examined: unrestored empty defects, matrix alone, matrix plus the three cell populations transduced with both control and Shh expression vectors. The bone regenerative capacity of Shh gene enhanced cells was assessed grossly, radiographically and histologically at 6 and 12 weeks postimplantation. After 6 weeks, new full thickness bone was seen emanating directly from the alginate/collagen matrix in the Shh-transduced groups. Quantitative two-dimensional digital analysis of histological sections confirmed statistically significant (P<0.05) amounts of bone regeneration in all three Shh-enhanced groups compared to controls. Necropsy failed to demonstrate any evidence of treatment-related side effects. This is the first study to demonstrate that Shh delivery to bone defects, in this case through a novel gene-enhanced tissue-engineering approach, results in significant bone regeneration. This encourages further development of the Shh gene-enhanced tissue-engineering approach for bone regeneration.
Bilateral Central Giant Cell Granulomas of the Mandible in An Eight Year-Old Girl with Noonan Syndrome (Noonan-Like/Multiple Giant Cell Lesions Syndrome)
(2005-03) Edwards, Paul C.; Fox, Joyce; Fantasia, John E; Goldberg, Jeff; Kelsch, Robert D
A number of conditions can present with lesions that histologically are indistinguishable from the central giant cell granuloma (CGCG) of bone, including brown tumors of hyperparathyroidism, cherubism, and, less commonly, a number of inherited syndromes. We report a case of an eight-year girl who presented with bilateral CGCGs of the posterior mandible. Characteristic facial features, reported increased post-operative bleeding and history of pulmonary stenosis led us to suspect a diagnosis of Noonan syndrome. A medical geneticist confirmed this on further evaluation. This case report will discuss the salient features of this diagnosis.
Salivary Heterotopia Of The Parathyroid Gland: A Report of Two Cases and Review of the Literature
(2005-05) Edwards, Paul C.; Bhuiya, Taqfiq; Kahn, Leonard B; Fantasia, John E
Two cases of periparathyroid salivary gland heterotopia are described. A review of the records of the Department of Pathology, Long Island Jewish Medical Center, over a 4-year period, identified 759 surgical specimens containing parathyroid gland tissue. Of these, 2 (0.26%) contained foci of ectopic salivary gland tissue. Both cases were associated with cyst formation. To date, 9 additional cases of heterotopic salivary gland tissue associated with the parathyroid gland have been described in the literature.
Evaluation of Cystic Salivary Gland Lesions by Fine-Needle Aspiration: An Analysis of 21 Cases
(2005-09) Edwards, Paul C.; Wasserman, Patricia
Objective: To analyze the potential sources of diagnostic errors and overall accuracy rate of the fine needle aspiration biopsy (FNAB) diagnosis of cystic salivary gland neoplasms. Study Design: A 10-year (1993-2002) retrospective review of the cytopathology slides from the Department of Pathology, Division of Cytopathology at Long Island Jewish Medical Center, New Hyde Park, NY, identified a total of 97 consecutive salivary gland FNAB cases that microscopically were interpreted as representing cystic lesions. Of these, 21 cases had histologic follow up at our institution. Results: A correct diagnosis was rendered by FNAB in 15/21 (72%) cases. This included 9 Warthin’s tumors, 2 mucoepidermoid carcinomas, 2 simple cysts, 1 cystadenoma and 1 abscess. Clinically insignificant discrepancies were identified in 3 of 21 (14%) FNABs. Clinically significant misdiagnoses were identified in a further 3 of 21 (14%) cases. Conclusions: A systematic approach to the diagnosis of cystic salivary gland lesions by FNAB can result in a correct diagnosis in greater than 70% of cases. Careful attention should be directed at identifying the extracellular fluid component(s) present (mucoid vs. watery proteinaceous) as well as the predominant cellular component (e.g. lymphocytes, histiocytes, epithelial cells and oncocytes). It is important to recognize, however, that occasionally epithelial cells may not be detected on FNAB of cystic salivary gland lesions, either as a result of cellular dilution by cyst fluid or due to inadequate sampling. However, with all FNABs tentatively diagnosed as a mucinous cystic lesion, the referring clinician should be informed that a low-grade mucoepidermoid cannot be ruled out.
External cervical root resorption involving multiple maxillary teeth in a patient with hereditary hemorrhagic telangiectasia
(2005-11) Edwards, Paul C.; McVaney, Tim
Hereditary hemorrhagic telangiectasia (HHT) is an inherited syndrome characterized by mucocutaneous telangiectases that commonly involve the tongue, lips, fingers, and conjunctiva. While root resorption has been reported in association with central hemangiomas of bone, the association of HHT with external cervical root resorption has not been described to date. We report a case of a 57-year-old female with HTT who presented with advanced cervical root resorption involving multiple maxillary anterior teeth. Histologic examination of the gingival tissue adjacent to the area of root resorption demonstrated multiple thin-walled vascular elements as well as larger vascular channels surrounded by a thickened muscular layer. We hypothesize that the external root resorption seen in this case is the result of the HHT-related vascular process in the adjacent gingival tissue.
Sinonasal undifferentiated carcinoma of the maxillary sinus
(2006) Edwards, Paul C.; Hess, Stephen J.; Saini, Tarnjit
We describe the case of a 43-year-old man who was referred to our dental school’s acute care clinic with pain and swelling of presumed dental origin in the left maxillary quadrant. Radiographic examination revealed extensive dental decay and periodontal disease. On questioning, the patient admitted to paresthesia of recent onset. Paresthesia associated with pain or swelling of the jaws is an ominous sign that should alert the clinician to the possibility of an underlying aggressive neoplasm. In this case, biopsy of the lesion confirmed that the patient had a rare malignant tumour of maxillary sinus origin,a sinonasal undifferentiated carcinoma.

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