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DESMOPLASTIC FIBROMA OF THE MANDIBLE: A DIAGNOSTIC CHALLENGE
ABSTRACTS
e100
margins, anteroposterior growth in mandibular body to the right up to the ascending branch on the same side, and involvement of the inferior alveolar nerve canal, suggesting an ameloblastoma diagnosis. After an incisional biopsy, the histopathologic report revealed an odontogenic keratocyst. Decompressive treatment with rigid latex device was used to allow reduction of the cystic cavity to facilitate removal, decrease the rate of recurrence, and not compromise the sensitive innervation of the affected region. However, periodic radiographic examination is necessary to follow up and define the surgical moment for enucleation.
REPORT OF DENTAL ALTERATIONS IN AN ARNOLD-CHIARI TYPE 2 SYNDROME PATIENT. ARTHUR CASTILHOS VIDOR, ARTHUR REGINATO CORBELLINI, LARISSA CASTILHOS VIDOR, SULIANE RECH, THIAGO DE OLIVEIRA GAMBA, FELLIPE CORA and, MARINA CURRA Arnold-Chiari syndrome is characterized by enlargement of the cerebellar tonsils going through the foramen magnum, reaching the spinal canal. The syndrome has 4 ratings: type I (asymptomatic in childhood), types II and III (diagnosed in the second week life), and type IV (incompatible with life). Oral alterations are rarely described, tooth agenesis being the most common. This study aims to report the case of a 3-year-old patient who is an Arnold-Chiari syndrome type II carrier with dental alterations. The case was evaluated by the hospital dentistry team because of the dental changes. Based on clinical finds, a skull x-ray (posterior-anterior projection, oblique lateral x-ray) where dental anomaly of number (supranumerary teeth) and enamel hypoplasia was observed. Topic fluor applications and monitoring are being made. The consistent clinical presentation provides further evidence of the dental characteristics of Arnold-Chiari type II syndrome patients.
DISSEMINATED FUSARIOSIS WITH ORAL AND NASAL FIRST MANIFESTATION: A CASE REPORT. STEFANIE THIEME, CAROLINA DUMMEL, SHANICE MARTIN, JULIA TURRA RIBEIRO, JESSICA RODRIGUEZ STREY, VINICIUS COELHO CARRARD and, MANOELA DOMINGUES MARTINS Fusariosis is a fungal infection that affects immunocompromised hosts and is mainly fatal in its disseminated form. A 49year-old male patient with acute myeloid leukemia, hepatic and splenic infiltration, thrombocytopenia, odynophagia, and abdominal pain was referred for evaluation. Clinical examination revealed nasal and small oral ulcers with erythematous halo on the soft palate, and diagnostic hypotheses were bacterial or viral infection. Treatment with cefepime and vancomycin, local hygiene, and low-level laser therapy were adopted, and lesions presented remission. The clinical condition improved, and chemotherapy with cytarabine was initiated; however, new lesions emerged concomitantly as well as pain in the sinuses and nasal obstruction. Biopsy and surgical debridement were performed, and the final diagnosis was fusariosis based on histopathologic and culture examination. Treatment with amphotericin B and vancomycin was performed, but there was worsening of immunosuppression with the dissemination of infection evolving to death.
OOOO January 2020 TERATOID CYST IN THE FLOOR OF THE MOUTH WITH SQUAMOUS, INTESTINAL, AND RESPIRATORY EPITHELIA. GUSTAVO ALCANTARA DA TRINDADE, HUGO COSTA NETO, TATYANNE SANTOS DE FREITAS, GLORIA MARIA DE FRANCA, ¸ LUIZ CARLOS MOREIRA JUNIOR, GERMANO DE LELIS BEZERRA JUNIOR and, HEBEL CAVALCANTI e GALVAO Disontogenic cysts are an embryonic developmental error, classified histopathologically as dermoid, epidermoid, or teratoid. Teratoid cyst, a cystic form of teratoma, is the least common presentation among these cysts. This work describes the clinical case of a tumor in the floor of the mouth, characterized by presence of squamous, intestinal, and respiratory tract epithelium. A 1-year-old girl was referred to service of oral surgery for examination of a swelling in the sublingual region without painful symptomatology. Intraoral examination revealed a swelling lesion on the floor of the mouth with soft consistency, measuring about 4 £ 5 cm. Surgery was performed under general anesthesia with complete enucleation of the lesion. Microscopic examination revealed fragments of developmental cystic lesion containing orthokeratinized stratified squamous epithelium with skin adnexa, intestinal and respiratory epithelia, and connective tissue elements. Based on morphologic findings, diagnosis of teratoid cyst was confirmed. The patient was followed up for 1 year without sign of recurrence.
ORAL MELANOCYTIC NEVI: A CASE REPORT. RAFAELA AMARAL MOREIRA, BARBARA MARTINS ANDRADE, RICARDO SANTIAGO GOMEZ, OSLEI PAES DE ALMEIDA, CIRO DANTAS SOARES, RICARDO ALVES MESQUITA and, FELIPE PAIVA FONSECA Melanocytic nevi are benign proliferations of nevus cells in the epithelial layer, the submucosal/subepidermal layer, or both. Although melanocytic nevi are frequently found on the skin, oral lesions can also be found. We describe a new case affecting a 13-year-old boy who was referred to our department because of an asymptomatic macule on the retromolar trigone observed several years before. The patient’s past medical history was noncontributory, and the intraoral examination revealed a black to brown pigmented lesion measuring approximately 4 cm in size. An incisional biopsy was done, and microscopic analysis showed the presence of ovoid-shaped nevus cells with melanin granules in the connective tissue and overlying epithelium. Some atypical nuclei were also found. Immunohistochemistry was positive for S100, and Ki67 demonstrated a low proliferative index, confirming the diagnosis of oral melanocytic nevi. The patient is under follow-up with no evidence of additional lesion growth. Support: FAPEMIG.
DESMOPLASTIC FIBROMA OF THE MANDIBLE: A DIAGNOSTIC CHALLENGE. PATRICIA GUERRA PEIXE GONCALVES, ¸ RODRIGO PORPINO MAFRA, HELLEN BANDEIRA DE PONTES SANTOS, LARISSA SANTOS AMARAL ROLIM, PATRICIA DARVIN GOMES PARENTE, LELIA BATISTA e PEREIRA PINTO DE SOUZA and, LEAO
OOOO Volume 129, Number 1 Desmoplastic fibroma (DF) is a rare benign myofibroblastic tumor with a locally aggressive behavior and high recurrence rate. We describe a case of DF in a 27-year-old woman presenting a painful, soft exophytic lesion on the mandible with approximately 7 months of evolution. Panoramic radiograph showed a radiolucent unilocular lesion with sclerotic borders involving teeth 44, 45, and 47. Clinical diagnosis was ossifying fibroma, and incisional biopsy was performed. Histopathologic analysis revealed a neoplasm characterized by proliferation of round, ovoid, and spindle-shaped mesenchymal cells with indistinct borders and no atypia. Intense deposition of collagen fibers, interspersed with areas of low-to-moderate cellularity, were also found. Neoplastic cells showed immunopositivity for a smooth muscle actin, S-100, synaptophysin, neuron-specific enolase, and Ki-67 but no immunoexpression of desmin. The patient was referred for tumor excision. This case reinforces the importance of associating clinical, radiographic, histopathologic, and immunohistochemical findings for the correct diagnosis and management of DF.
PRIMARY ORAL SOFT TISSUE ANGIOSARCOMA OF ORAL CAVITY IN A YOUNG PATIENT: CASE REPORT. FILIPE NOBRE CHAVES, MARCELO BONIFACIO DA SILVA SAMPIERI, DENISE HELEN IMACULADA PEREIRA DE OLIVEIRA, FRANCISCO SAMUEL RODRIGUES CARVALHO, MARIO ROGERIO LIMA MOTA, ANA PAULA NEGREIROS NUNES ALVES and, KARUZA MARIA ALVES PEREIRA Angiosarcoma is a rare and aggressive malignant tumor that has a poor prognosis and occurs predominantly in elderly patients, usually in the trunk and limb region. It represents less than 1% of all malignant tumors occurring in the oral cavity. A 31-year-old man presented an extensive sessile nodule, which was asymptomatic and erythematous in the alveolar ridge extending to hard palate. Histologically, the tumor consisted of spindle and polygonal cells with hyperchromatic nuclei and prominent nucleoli. These cells are sometimes arranged in a stent-shaped pattern or form numerous vascular structures of varying calibers. The tumor had invasion in the surrounding tissue, and lymphovascular permeation was noted. Grocott staining was negative. Immunohistochemically, tumor cells were positive for CD34 in vessels and scattered cells, with Friend leukemia integration site 1 in spindle cells and Ki-67 labeling greater than 15%, concluding the pathologic diagnosis of angiosarcoma. Metastatic lesions are found in upper limbs and thorax. Radical surgery is now planned associated with chemotherapy.
CHORISTOMAS OF THE TONGUE: A SERIES OF 5 NEW CASES. MARIENE DA SILVA MONTEIRO, THAMYRES CAMPOS ^ CA, THAMIRIS DE CASTRO ABRANTES, FONSE MICHELLE AGOSTINI, BRUNO AUGUSTO BENEVENUTO DE ANDRADE, MARIO JOSE e e ^ A ABRAHAO ROMANACH and, ALINE CORRE Choristoma is a mass of normal tissue in an abnormal location that occasionally may affect the oral cavity, particularly as tumor-like proliferation of osseous and cartilaginous tissues in the tongue. We contribute with 5 new cases of choristomas of the tongue. There were 2 female patients and 3 male patients, with mean age of 34 years. Lesions appeared as small, smooth-
ABSTRACTS
e101
surfaced, and normal-colored nodules; were hard-to-soft in consistency; and were mainly located in the dorsum (3 cases), base (1 case), and lateral border (1 case) of the tongue. Conservative surgical excision was performed in all cases. Microscopically, 4 cases were classified as osseous choristomas and 1 case as sebaceous choristoma of the tongue. Recurrence was not observed after follow-up from 0.5 to 4 years. Choristomas of the tongue are uncommon, being usually characterized by the occurrence of osseous tissues as a hardened small nodule in the dorsal tongue of young adults.
DIAGNOSIS AND TREATMENT OF AN UNUSUAL CICATRICIAL PEMPHIGOID: CASE REPORT. ELOA BORGES LUNA, e THAYLLA NUNEZ AMIN DICK, BRUNA LAVINAS SAYED PICCIANI, ARLEY SILVA JUNIOR, ELIANE PEDRA DIAS, DANIELLE CASTEX CONDE and, RAFAELA ELVIRA ROZZA-DE-MENEZES Cicatricial pemphigoid is a chronic autoimmune blistering disease in which autoantibodies are produced against the basement membrane zone (BMZ). A 65-year-old black man was referred by the dermatology clinic to our oral diagnostic clinic, complaining of a lip painful ulcer with 2 years of evolution. There was a previous biopsy, but the histopathologic analysis was inconclusive. Oral examination revealed an ulcer covered by a pseudomembrane on the lower lip measuring 2 cm. He had no other lesions. The hypotheses were pemphigus vulgaris, squamous cell carcinoma associated with actinic cheilitis, and actinic prurigo. An incisional biopsy was performed, and the histopathologic analysis revealed cicatricial pemphigoid. Indirect immunofluorescence showed linear deposits of IgG, IgA, IgM, and C3 along the BMZ. The use of topical and systemic corticoids associated with low-level laser therapy provided a complete remission of the lesion in 3 months. The patient is being followed-up for 1 month without recurrence.
EPIDERMOID CYST OF THE BUCCAL REGION OF THE FACE. PAULA HELENA DE ^ NIO ARANTES, ACCIOLY COSTA, EUGE ^ CA, MARCIA THAMYRES CAMPOS FONSE GRILLO e CABRAL, MARIO JOSE ROMANACH, BRUNO AUGUSTO BENEVENUTO DE ANDRADE and, ALINE e ^ A ABRAHAO CORRE Epidermoid cyst is a common cutaneous cyst that occasionally affects the oral cavity. A 52-year-old black woman presented a subcutaneous well-defined swelling in the right cheek, which caused considerable facial asymmetry with duration of 15 years. The lesion was asymptomatic, was firm in consistency, and measured 5 cm. Intraoral examination showed a submucosal nodule in the buccal mucosa. Intraoral conservative surgical removal was performed and grossly, a brownish fragment of irregular cut surface was observed. Microscopic evaluation showed a cystic cavity filled by keratin and lined by orthokeratinized stratified squamous epithelium with highly pigmented basal cells, suggesting origin from the entrapment of the superficial pigmented epithelium of the skin face. The final diagnosis was epidermal cyst of the face with extension to the buccal mucosa. Exuberant epidermal cyst may cause facial asymmetry, and intraoral surgical approach may be required to avoid postsurgical esthetic concerns.
e100
margins, anteroposterior growth in mandibular body to the right up to the ascending branch on the same side, and involvement of the inferior alveolar nerve canal, suggesting an ameloblastoma diagnosis. After an incisional biopsy, the histopathologic report revealed an odontogenic keratocyst. Decompressive treatment with rigid latex device was used to allow reduction of the cystic cavity to facilitate removal, decrease the rate of recurrence, and not compromise the sensitive innervation of the affected region. However, periodic radiographic examination is necessary to follow up and define the surgical moment for enucleation.
REPORT OF DENTAL ALTERATIONS IN AN ARNOLD-CHIARI TYPE 2 SYNDROME PATIENT. ARTHUR CASTILHOS VIDOR, ARTHUR REGINATO CORBELLINI, LARISSA CASTILHOS VIDOR, SULIANE RECH, THIAGO DE OLIVEIRA GAMBA, FELLIPE CORA and, MARINA CURRA Arnold-Chiari syndrome is characterized by enlargement of the cerebellar tonsils going through the foramen magnum, reaching the spinal canal. The syndrome has 4 ratings: type I (asymptomatic in childhood), types II and III (diagnosed in the second week life), and type IV (incompatible with life). Oral alterations are rarely described, tooth agenesis being the most common. This study aims to report the case of a 3-year-old patient who is an Arnold-Chiari syndrome type II carrier with dental alterations. The case was evaluated by the hospital dentistry team because of the dental changes. Based on clinical finds, a skull x-ray (posterior-anterior projection, oblique lateral x-ray) where dental anomaly of number (supranumerary teeth) and enamel hypoplasia was observed. Topic fluor applications and monitoring are being made. The consistent clinical presentation provides further evidence of the dental characteristics of Arnold-Chiari type II syndrome patients.
DISSEMINATED FUSARIOSIS WITH ORAL AND NASAL FIRST MANIFESTATION: A CASE REPORT. STEFANIE THIEME, CAROLINA DUMMEL, SHANICE MARTIN, JULIA TURRA RIBEIRO, JESSICA RODRIGUEZ STREY, VINICIUS COELHO CARRARD and, MANOELA DOMINGUES MARTINS Fusariosis is a fungal infection that affects immunocompromised hosts and is mainly fatal in its disseminated form. A 49year-old male patient with acute myeloid leukemia, hepatic and splenic infiltration, thrombocytopenia, odynophagia, and abdominal pain was referred for evaluation. Clinical examination revealed nasal and small oral ulcers with erythematous halo on the soft palate, and diagnostic hypotheses were bacterial or viral infection. Treatment with cefepime and vancomycin, local hygiene, and low-level laser therapy were adopted, and lesions presented remission. The clinical condition improved, and chemotherapy with cytarabine was initiated; however, new lesions emerged concomitantly as well as pain in the sinuses and nasal obstruction. Biopsy and surgical debridement were performed, and the final diagnosis was fusariosis based on histopathologic and culture examination. Treatment with amphotericin B and vancomycin was performed, but there was worsening of immunosuppression with the dissemination of infection evolving to death.
OOOO January 2020 TERATOID CYST IN THE FLOOR OF THE MOUTH WITH SQUAMOUS, INTESTINAL, AND RESPIRATORY EPITHELIA. GUSTAVO ALCANTARA DA TRINDADE, HUGO COSTA NETO, TATYANNE SANTOS DE FREITAS, GLORIA MARIA DE FRANCA, ¸ LUIZ CARLOS MOREIRA JUNIOR, GERMANO DE LELIS BEZERRA JUNIOR and, HEBEL CAVALCANTI e GALVAO Disontogenic cysts are an embryonic developmental error, classified histopathologically as dermoid, epidermoid, or teratoid. Teratoid cyst, a cystic form of teratoma, is the least common presentation among these cysts. This work describes the clinical case of a tumor in the floor of the mouth, characterized by presence of squamous, intestinal, and respiratory tract epithelium. A 1-year-old girl was referred to service of oral surgery for examination of a swelling in the sublingual region without painful symptomatology. Intraoral examination revealed a swelling lesion on the floor of the mouth with soft consistency, measuring about 4 £ 5 cm. Surgery was performed under general anesthesia with complete enucleation of the lesion. Microscopic examination revealed fragments of developmental cystic lesion containing orthokeratinized stratified squamous epithelium with skin adnexa, intestinal and respiratory epithelia, and connective tissue elements. Based on morphologic findings, diagnosis of teratoid cyst was confirmed. The patient was followed up for 1 year without sign of recurrence.
ORAL MELANOCYTIC NEVI: A CASE REPORT. RAFAELA AMARAL MOREIRA, BARBARA MARTINS ANDRADE, RICARDO SANTIAGO GOMEZ, OSLEI PAES DE ALMEIDA, CIRO DANTAS SOARES, RICARDO ALVES MESQUITA and, FELIPE PAIVA FONSECA Melanocytic nevi are benign proliferations of nevus cells in the epithelial layer, the submucosal/subepidermal layer, or both. Although melanocytic nevi are frequently found on the skin, oral lesions can also be found. We describe a new case affecting a 13-year-old boy who was referred to our department because of an asymptomatic macule on the retromolar trigone observed several years before. The patient’s past medical history was noncontributory, and the intraoral examination revealed a black to brown pigmented lesion measuring approximately 4 cm in size. An incisional biopsy was done, and microscopic analysis showed the presence of ovoid-shaped nevus cells with melanin granules in the connective tissue and overlying epithelium. Some atypical nuclei were also found. Immunohistochemistry was positive for S100, and Ki67 demonstrated a low proliferative index, confirming the diagnosis of oral melanocytic nevi. The patient is under follow-up with no evidence of additional lesion growth. Support: FAPEMIG.
DESMOPLASTIC FIBROMA OF THE MANDIBLE: A DIAGNOSTIC CHALLENGE. PATRICIA GUERRA PEIXE GONCALVES, ¸ RODRIGO PORPINO MAFRA, HELLEN BANDEIRA DE PONTES SANTOS, LARISSA SANTOS AMARAL ROLIM, PATRICIA DARVIN GOMES PARENTE, LELIA BATISTA e PEREIRA PINTO DE SOUZA and, LEAO
OOOO Volume 129, Number 1 Desmoplastic fibroma (DF) is a rare benign myofibroblastic tumor with a locally aggressive behavior and high recurrence rate. We describe a case of DF in a 27-year-old woman presenting a painful, soft exophytic lesion on the mandible with approximately 7 months of evolution. Panoramic radiograph showed a radiolucent unilocular lesion with sclerotic borders involving teeth 44, 45, and 47. Clinical diagnosis was ossifying fibroma, and incisional biopsy was performed. Histopathologic analysis revealed a neoplasm characterized by proliferation of round, ovoid, and spindle-shaped mesenchymal cells with indistinct borders and no atypia. Intense deposition of collagen fibers, interspersed with areas of low-to-moderate cellularity, were also found. Neoplastic cells showed immunopositivity for a smooth muscle actin, S-100, synaptophysin, neuron-specific enolase, and Ki-67 but no immunoexpression of desmin. The patient was referred for tumor excision. This case reinforces the importance of associating clinical, radiographic, histopathologic, and immunohistochemical findings for the correct diagnosis and management of DF.
PRIMARY ORAL SOFT TISSUE ANGIOSARCOMA OF ORAL CAVITY IN A YOUNG PATIENT: CASE REPORT. FILIPE NOBRE CHAVES, MARCELO BONIFACIO DA SILVA SAMPIERI, DENISE HELEN IMACULADA PEREIRA DE OLIVEIRA, FRANCISCO SAMUEL RODRIGUES CARVALHO, MARIO ROGERIO LIMA MOTA, ANA PAULA NEGREIROS NUNES ALVES and, KARUZA MARIA ALVES PEREIRA Angiosarcoma is a rare and aggressive malignant tumor that has a poor prognosis and occurs predominantly in elderly patients, usually in the trunk and limb region. It represents less than 1% of all malignant tumors occurring in the oral cavity. A 31-year-old man presented an extensive sessile nodule, which was asymptomatic and erythematous in the alveolar ridge extending to hard palate. Histologically, the tumor consisted of spindle and polygonal cells with hyperchromatic nuclei and prominent nucleoli. These cells are sometimes arranged in a stent-shaped pattern or form numerous vascular structures of varying calibers. The tumor had invasion in the surrounding tissue, and lymphovascular permeation was noted. Grocott staining was negative. Immunohistochemically, tumor cells were positive for CD34 in vessels and scattered cells, with Friend leukemia integration site 1 in spindle cells and Ki-67 labeling greater than 15%, concluding the pathologic diagnosis of angiosarcoma. Metastatic lesions are found in upper limbs and thorax. Radical surgery is now planned associated with chemotherapy.
CHORISTOMAS OF THE TONGUE: A SERIES OF 5 NEW CASES. MARIENE DA SILVA MONTEIRO, THAMYRES CAMPOS ^ CA, THAMIRIS DE CASTRO ABRANTES, FONSE MICHELLE AGOSTINI, BRUNO AUGUSTO BENEVENUTO DE ANDRADE, MARIO JOSE e e ^ A ABRAHAO ROMANACH and, ALINE CORRE Choristoma is a mass of normal tissue in an abnormal location that occasionally may affect the oral cavity, particularly as tumor-like proliferation of osseous and cartilaginous tissues in the tongue. We contribute with 5 new cases of choristomas of the tongue. There were 2 female patients and 3 male patients, with mean age of 34 years. Lesions appeared as small, smooth-
ABSTRACTS
e101
surfaced, and normal-colored nodules; were hard-to-soft in consistency; and were mainly located in the dorsum (3 cases), base (1 case), and lateral border (1 case) of the tongue. Conservative surgical excision was performed in all cases. Microscopically, 4 cases were classified as osseous choristomas and 1 case as sebaceous choristoma of the tongue. Recurrence was not observed after follow-up from 0.5 to 4 years. Choristomas of the tongue are uncommon, being usually characterized by the occurrence of osseous tissues as a hardened small nodule in the dorsal tongue of young adults.
DIAGNOSIS AND TREATMENT OF AN UNUSUAL CICATRICIAL PEMPHIGOID: CASE REPORT. ELOA BORGES LUNA, e THAYLLA NUNEZ AMIN DICK, BRUNA LAVINAS SAYED PICCIANI, ARLEY SILVA JUNIOR, ELIANE PEDRA DIAS, DANIELLE CASTEX CONDE and, RAFAELA ELVIRA ROZZA-DE-MENEZES Cicatricial pemphigoid is a chronic autoimmune blistering disease in which autoantibodies are produced against the basement membrane zone (BMZ). A 65-year-old black man was referred by the dermatology clinic to our oral diagnostic clinic, complaining of a lip painful ulcer with 2 years of evolution. There was a previous biopsy, but the histopathologic analysis was inconclusive. Oral examination revealed an ulcer covered by a pseudomembrane on the lower lip measuring 2 cm. He had no other lesions. The hypotheses were pemphigus vulgaris, squamous cell carcinoma associated with actinic cheilitis, and actinic prurigo. An incisional biopsy was performed, and the histopathologic analysis revealed cicatricial pemphigoid. Indirect immunofluorescence showed linear deposits of IgG, IgA, IgM, and C3 along the BMZ. The use of topical and systemic corticoids associated with low-level laser therapy provided a complete remission of the lesion in 3 months. The patient is being followed-up for 1 month without recurrence.
EPIDERMOID CYST OF THE BUCCAL REGION OF THE FACE. PAULA HELENA DE ^ NIO ARANTES, ACCIOLY COSTA, EUGE ^ CA, MARCIA THAMYRES CAMPOS FONSE GRILLO e CABRAL, MARIO JOSE ROMANACH, BRUNO AUGUSTO BENEVENUTO DE ANDRADE and, ALINE e ^ A ABRAHAO CORRE Epidermoid cyst is a common cutaneous cyst that occasionally affects the oral cavity. A 52-year-old black woman presented a subcutaneous well-defined swelling in the right cheek, which caused considerable facial asymmetry with duration of 15 years. The lesion was asymptomatic, was firm in consistency, and measured 5 cm. Intraoral examination showed a submucosal nodule in the buccal mucosa. Intraoral conservative surgical removal was performed and grossly, a brownish fragment of irregular cut surface was observed. Microscopic evaluation showed a cystic cavity filled by keratin and lined by orthokeratinized stratified squamous epithelium with highly pigmented basal cells, suggesting origin from the entrapment of the superficial pigmented epithelium of the skin face. The final diagnosis was epidermal cyst of the face with extension to the buccal mucosa. Exuberant epidermal cyst may cause facial asymmetry, and intraoral surgical approach may be required to avoid postsurgical esthetic concerns.