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Oral Melanoacanthoma: A Report of Two Cases and a Review of the Literature
Poster Session TRAP-positive multinucleated cells formed in the culture, which were considered to be osteoclastic cells, were counted. Western blotting analysis and quantitative RTPCR was performed according to the ordinary protocols. To evaluate the effect of sudachitin on in vivo LPSinduced calvarial bone destruction, 8-week-old mice were injected with LPS subperiosteally in the calvarial bone daily for 5 days. After 6 days, mCT scanning of the calvariae was performed. The mean values of the groups were compared by unpaired Student’s t test or by oneway ANOVAs. P < 0.05 was considered significant. Results: When osteoclast precursors were treated with sudachitin in the presence of sRANKL and M-CSF, the number of TRAP-positive multinucleated osteoclasts formed in the culture was decreased compared with sudachitin-untreated culture in a dose-dependent manner [stachitin (10 mM) group vs. untreated group: 6.8 0.9 cells/well vs. 625.3 18.0 cells/well, p<0.05]. Since sudachitin had no effect on the proliferation and viability of osteoclast lineage cells, the inhibitory effect of sudachitin was attributed to the direct action in osteoclastogenesis. Consistent with the inhibition, sudachitin suppressed the activation of Erk signal in the osteoclast precursors. Furthermore, sudachitin decreased expression of c-fos and NFATc1 in mRNA and protein levels. Followed by the down-regulation of those transcription factors, the expressions of osteoclast differentiationrelated molecules such as TRAP, cathepsin K, DC-STAMP and Atp6v0d2 were also decreased. In addition, sudachitin suppressed the production of intracellular reactive oxygen species. Consistent with the in vitro experiments, when inflammatory bone destruction of calvariae was induced by LPS injection, simultaneous administration of sudachitin (50 mM) with LPS reduced the elevated bone resorption as well as the increased TRAP and cathepsin K expression in vivo [stachitin (10 mM) group vs. untreated group: relative amount of TRAP mRNA; 4.69 0.93 vs. 2.66 0.30 , p<0.05; relative amount of cathepsin K mRNA: 6.0 0.27 vs 2.52 0.37 , p<0.05]. Conclusion: Sudachitin, a polymethoxyflavone, inhibits osteoclast formation and suppresses LPS-induced inflammatory bone resorption in mice. Therefore, sudachitin could be an effective component for the prevention of inflammatory bone disorders. References: 1. Yuasa K, Tada K, Harita G, Fujimoto T, Tsukayama M, Tsuji A. Sudachitin, a polymethoxyflavone from Citrus sudachi, suppresses lipopolysaccharide-induced inflammatory responses in mouse macrophage-like RAW264 cells. Biosci Biotechnol Biochem, 76:598-600, 2012 2. Nakayachi M, Ito J, Hayashida C, Ohyama Y, Kakino A, Okayasu M, Sato T, Ogasawara T, Kaneda T, Suda N, Sawamura T, Hakeda Y. Lectin-like oxidized low-density lipoprotein receptor-1 abrogation causes resistance to inflammatory bone destruction in mice, despite promoting osteoclastogenesis in the steady state. Bone, 75:170-182, 2015
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POSTER 36 Oral Melanoacanthoma: A Report of Two Cases and a Review of the Literature P. G. Tolomeo: New York University Langone Medical Center/Bellevue Hospital Center, J. S. Lee, N. Zawada, A. R. Kerr, J. A. Phelan Oral melanoacanthoma (MA) is a rare, benign pigmented lesion that presents as a painless, rapidly growing, brown-black macular lesion that commonly affects the buccal mucosa in areas that are subject to chronic trauma/irritation.1,2 MA is commonly seen in the third and fourth decades of life and primarily affects blacks with a strong female predilection.3,4 Histopathologically, the lesions exhibit proliferation of keratinocytes and dendritic melanocytes.5 This report includes two cases of oral melanoacanthoma and a review of the literature. Case 1: A 43-year-old black female presented with a slowly enlarging pigmented lesion on the right buccal mucosa. The patient did not recall any known trauma to the area or previous infection and reported that the lesion was painless but had a gradually increased in size. Oral examination revealed a 2.0 x 2.0 cm. brown macule on the right buccal mucosa. A punch biopsy was taken of the pigmented area. The tissue was placed in 10% formalin and submitted for microscopic examination. The tissue was stained with hematoxylin and eosin and exhibited acanthotic, stratified squamous epithelium with dendritic melanocytes dispersed throughout the epithelium consistent with a diagnosis of melanoacanthoma. Case 2: A-35 year-old black female presented with a rapidly growing pigmented lesion on the left buccal mucosa. Two years prior to presentation the patient had noted a brown lesion on the buccal mucosa adjacent to a fractured tooth. The lesion remained unchanged and asymptomatic for approximately two years. One week prior to presentation, the patient noted that the lesion was enlarging, but remained painless. Oral examination revealed a 1.5 x 1.5 cm. brown macule surrounded by erythema on the left buccal mucosa adjacent to a fractured tooth. A punch biopsy was taken that included both the pigmented and erythematous areas. The tissue was placed in 10% formalin and submitted for microscopic examination. The tissue was stained with hematoxylin and eosin and exhibited similar histopathologic features to the previous case. Immunohistochemical staining with S-100 and Melan-A dramatically demonstrated the dendritic melanocytes. Review of the literature revealed a total of 50 cases of oral melanoacanthoma. These lesions were reported in black females on the buccal mucosa with subsequent resolution. The cases here demonstrate similar clinical AAOMS 2016
Poster Session features and age at presentation to previously reported cases. The pathogenesis of oral MA remains unclear, however, most studies suggest this is a reactive process due to chronic irritation.2 Oral MA may regress following biopsy and no surgical intervention is required due to its selfresolving quality.5 References: 1. Chandler K, Chaudhry Z, Kumar N, Barrett A, Porter S. Mela- noacanthoma: A rare cause of oral pigmentation. Oral Surg Oral Med Oral Pathol Oral Radiol Endod, 84:492-4, 1997 2. Fatahzadeh M, Sirois DA. Multiple intraoral melanoacanthomas: a case report with unusual findings. Oral Surg Oral Med Oral Pathol Oral Radiol Endod, 94(1):54, 2002 3. Fornatora ML, Reich RF, Haber S, Solomon F, Freedman PD: Oral melanoacanthoma – a report of 10 cases, review of the liter- ature, and immunohistochemical analysis for HMB-45 reac- tivity. Am J Dermatopathol, 25:12-15, 2003 4. Kauzman A, Pavone M, Blanas N, Bradley G: Pigmented lesions of the oral cavity: review, differential diagnosis and case presentations. J Can Dent Assoc, 70:682-683, 2004 5. Neville BW, Damm DD, Allen CM, Chi AC: Oral and Maxillofacial Pathology, ed 4, Elsevier, Inc.
POSTER 37 Solitary Fibrous Tumor of the Hard Palate: A Case Report and Review of the Literature J. S. Lee: New York University Langone Medical Center/ Bellevue Hospital Center, P. G. Tolomeo, E. J. A. Cappetta, J. A. Phelan, L. Alsabban Solitary fibrous tumors (SFTs) are a relatively rare group of mesenchymal neoplasms. Klemperer and Rabin first described a case in the pleura in 1931, but SFTs have also been reported in extrapleural sites, including the oral cavity. SFTs of the oral cavity most commonly affect the buccal mucosa and tongue of female patients in their sixth decade of life. To date, only seven cases of oral SFTs located in the palate (three soft palate, four hard palate) have been documented in the literature. We present a case of a solitary fibrous tumor of the hard palate with review of the literature. A 26-year-old female with a past medical history significant for tuberous sclerosis presented to NYU College of Dentistry reporting a several year history of a painless mass of the hard palate. The mass was biopsied, initially diagnosed as cellular angiofibroma, and referred to the Department of Oral & Maxillofacial Surgery at Bellevue Hospital Center for further management. Examination revealed a 3x3 cm exophytic lesion on the right hard palate extending past the midline. The mass was non-tender to palpation and the mucosa overlying the lesion was intact without evidence of ulceration or necrosis. A CTA of the lesion showed mild prominence of vasculature along the right lateral soft and hard palate, possibly demonstrating AAOMS 2016
supply from the ascending palatine artery. Postoperative surgical histopathology demonstrated a well-circumscribed, non-encapsulated lesion composed of spindle cells with admixed background slit-and-staghorn vessels in a patternless pattern. Immunohistochemical staining was diffusely positive for CD34 and Bcl-2 while negative for SMA, CD31, AE1/AE3, CAM5.2, S-100, EMA and demonstrated low KI-67 immunolabeling. SFTs constitute a heterogeneous group of rare spindlecell tumors that include benign and malignant neoplasms. Their cell of origin remains uncertain since CD34-positive spindle cells are also found in other mesenchymal neoplasms, such as giant cell angiofibromas and hemangiopericytoma, and share similar microscopic, immunohistochemical and biologic features. SFTs are usually well-demarcated and partially encapsulated neoplasms. Microscopically, SFTs show a wide range of morphological characteristics from predominantly fibrous lesions containing alternating fibrous areas and hyalinized thick-walled vessels to more cellular fibrous neoplasms with a ‘‘patternless pattern’’ and thinwalled branching vessels. Immunohistochemically, SFTs usually demonstrate CD34 and CD99, and vimentin positivity with variable Bcl-2, EMA and SMA positivity and are usually negative for CD68, desmin, pan-cytokeratins, and S-100 protein immunoreactivity. Malignant SFTs tend to demonstrate nuclear atypia, hypercellularity, loss of margin integrity, high mitotic rate (>4 per 10 high power fields) and lose CD34 immunoreactivity while overexpressing S-100 and p53. Our patient’s lesion demonstrated positivity for only CD34 and Bcl-2, which, along with its aforementioned histological characteristics, was more consistent with the diagnosis of benign SFT than for the original diagnosis of cellular angiofibroma. CT imaging typically demonstrates SFTs as well-circumscribed, hypervascular masses with varying degrees of enhancement, necrosis or cystic change, and may show occasional internal calcification. Our patient’s lesion demonstrated mild prominence of vasculature associated with the lesion with no invasion into the adjacent hard palate, consistent with a benign tumor. Due to its rare entity, SFTs are seldom considered in the differential diagnosis for submucosal masses of the oral cavity. However, reports suggest that SFTs may possess malignant characteristics and, thus, should be considered when evaluating well-circumscribed, solid masses in the oral cavity.
References: 1. Li XM, Yu JQ, Xu GH. Solitary fibrous tumor of the soft palate: A report of two cases. Oncol Lett, 7:1975-1977, 2014 2. Carlos R, de Andrade BAB, Canedo NHS, Abrah~ao AC, Agostini M, de Almeida OP, Roma~ nach MJ. Clinicopathologic and immunohistochemical features of five new cases of solitary fibrous tumor of the oral cavity. Oral Surg Oral Med Oral Pathol and Oral Radiol, 121:390-395, 2015
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POSTER 36 Oral Melanoacanthoma: A Report of Two Cases and a Review of the Literature P. G. Tolomeo: New York University Langone Medical Center/Bellevue Hospital Center, J. S. Lee, N. Zawada, A. R. Kerr, J. A. Phelan Oral melanoacanthoma (MA) is a rare, benign pigmented lesion that presents as a painless, rapidly growing, brown-black macular lesion that commonly affects the buccal mucosa in areas that are subject to chronic trauma/irritation.1,2 MA is commonly seen in the third and fourth decades of life and primarily affects blacks with a strong female predilection.3,4 Histopathologically, the lesions exhibit proliferation of keratinocytes and dendritic melanocytes.5 This report includes two cases of oral melanoacanthoma and a review of the literature. Case 1: A 43-year-old black female presented with a slowly enlarging pigmented lesion on the right buccal mucosa. The patient did not recall any known trauma to the area or previous infection and reported that the lesion was painless but had a gradually increased in size. Oral examination revealed a 2.0 x 2.0 cm. brown macule on the right buccal mucosa. A punch biopsy was taken of the pigmented area. The tissue was placed in 10% formalin and submitted for microscopic examination. The tissue was stained with hematoxylin and eosin and exhibited acanthotic, stratified squamous epithelium with dendritic melanocytes dispersed throughout the epithelium consistent with a diagnosis of melanoacanthoma. Case 2: A-35 year-old black female presented with a rapidly growing pigmented lesion on the left buccal mucosa. Two years prior to presentation the patient had noted a brown lesion on the buccal mucosa adjacent to a fractured tooth. The lesion remained unchanged and asymptomatic for approximately two years. One week prior to presentation, the patient noted that the lesion was enlarging, but remained painless. Oral examination revealed a 1.5 x 1.5 cm. brown macule surrounded by erythema on the left buccal mucosa adjacent to a fractured tooth. A punch biopsy was taken that included both the pigmented and erythematous areas. The tissue was placed in 10% formalin and submitted for microscopic examination. The tissue was stained with hematoxylin and eosin and exhibited similar histopathologic features to the previous case. Immunohistochemical staining with S-100 and Melan-A dramatically demonstrated the dendritic melanocytes. Review of the literature revealed a total of 50 cases of oral melanoacanthoma. These lesions were reported in black females on the buccal mucosa with subsequent resolution. The cases here demonstrate similar clinical AAOMS 2016
Poster Session features and age at presentation to previously reported cases. The pathogenesis of oral MA remains unclear, however, most studies suggest this is a reactive process due to chronic irritation.2 Oral MA may regress following biopsy and no surgical intervention is required due to its selfresolving quality.5 References: 1. Chandler K, Chaudhry Z, Kumar N, Barrett A, Porter S. Mela- noacanthoma: A rare cause of oral pigmentation. Oral Surg Oral Med Oral Pathol Oral Radiol Endod, 84:492-4, 1997 2. Fatahzadeh M, Sirois DA. Multiple intraoral melanoacanthomas: a case report with unusual findings. Oral Surg Oral Med Oral Pathol Oral Radiol Endod, 94(1):54, 2002 3. Fornatora ML, Reich RF, Haber S, Solomon F, Freedman PD: Oral melanoacanthoma – a report of 10 cases, review of the liter- ature, and immunohistochemical analysis for HMB-45 reac- tivity. Am J Dermatopathol, 25:12-15, 2003 4. Kauzman A, Pavone M, Blanas N, Bradley G: Pigmented lesions of the oral cavity: review, differential diagnosis and case presentations. J Can Dent Assoc, 70:682-683, 2004 5. Neville BW, Damm DD, Allen CM, Chi AC: Oral and Maxillofacial Pathology, ed 4, Elsevier, Inc.
POSTER 37 Solitary Fibrous Tumor of the Hard Palate: A Case Report and Review of the Literature J. S. Lee: New York University Langone Medical Center/ Bellevue Hospital Center, P. G. Tolomeo, E. J. A. Cappetta, J. A. Phelan, L. Alsabban Solitary fibrous tumors (SFTs) are a relatively rare group of mesenchymal neoplasms. Klemperer and Rabin first described a case in the pleura in 1931, but SFTs have also been reported in extrapleural sites, including the oral cavity. SFTs of the oral cavity most commonly affect the buccal mucosa and tongue of female patients in their sixth decade of life. To date, only seven cases of oral SFTs located in the palate (three soft palate, four hard palate) have been documented in the literature. We present a case of a solitary fibrous tumor of the hard palate with review of the literature. A 26-year-old female with a past medical history significant for tuberous sclerosis presented to NYU College of Dentistry reporting a several year history of a painless mass of the hard palate. The mass was biopsied, initially diagnosed as cellular angiofibroma, and referred to the Department of Oral & Maxillofacial Surgery at Bellevue Hospital Center for further management. Examination revealed a 3x3 cm exophytic lesion on the right hard palate extending past the midline. The mass was non-tender to palpation and the mucosa overlying the lesion was intact without evidence of ulceration or necrosis. A CTA of the lesion showed mild prominence of vasculature along the right lateral soft and hard palate, possibly demonstrating AAOMS 2016
supply from the ascending palatine artery. Postoperative surgical histopathology demonstrated a well-circumscribed, non-encapsulated lesion composed of spindle cells with admixed background slit-and-staghorn vessels in a patternless pattern. Immunohistochemical staining was diffusely positive for CD34 and Bcl-2 while negative for SMA, CD31, AE1/AE3, CAM5.2, S-100, EMA and demonstrated low KI-67 immunolabeling. SFTs constitute a heterogeneous group of rare spindlecell tumors that include benign and malignant neoplasms. Their cell of origin remains uncertain since CD34-positive spindle cells are also found in other mesenchymal neoplasms, such as giant cell angiofibromas and hemangiopericytoma, and share similar microscopic, immunohistochemical and biologic features. SFTs are usually well-demarcated and partially encapsulated neoplasms. Microscopically, SFTs show a wide range of morphological characteristics from predominantly fibrous lesions containing alternating fibrous areas and hyalinized thick-walled vessels to more cellular fibrous neoplasms with a ‘‘patternless pattern’’ and thinwalled branching vessels. Immunohistochemically, SFTs usually demonstrate CD34 and CD99, and vimentin positivity with variable Bcl-2, EMA and SMA positivity and are usually negative for CD68, desmin, pan-cytokeratins, and S-100 protein immunoreactivity. Malignant SFTs tend to demonstrate nuclear atypia, hypercellularity, loss of margin integrity, high mitotic rate (>4 per 10 high power fields) and lose CD34 immunoreactivity while overexpressing S-100 and p53. Our patient’s lesion demonstrated positivity for only CD34 and Bcl-2, which, along with its aforementioned histological characteristics, was more consistent with the diagnosis of benign SFT than for the original diagnosis of cellular angiofibroma. CT imaging typically demonstrates SFTs as well-circumscribed, hypervascular masses with varying degrees of enhancement, necrosis or cystic change, and may show occasional internal calcification. Our patient’s lesion demonstrated mild prominence of vasculature associated with the lesion with no invasion into the adjacent hard palate, consistent with a benign tumor. Due to its rare entity, SFTs are seldom considered in the differential diagnosis for submucosal masses of the oral cavity. However, reports suggest that SFTs may possess malignant characteristics and, thus, should be considered when evaluating well-circumscribed, solid masses in the oral cavity.
References: 1. Li XM, Yu JQ, Xu GH. Solitary fibrous tumor of the soft palate: A report of two cases. Oncol Lett, 7:1975-1977, 2014 2. Carlos R, de Andrade BAB, Canedo NHS, Abrah~ao AC, Agostini M, de Almeida OP, Roma~ nach MJ. Clinicopathologic and immunohistochemical features of five new cases of solitary fibrous tumor of the oral cavity. Oral Surg Oral Med Oral Pathol and Oral Radiol, 121:390-395, 2015
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