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Aggressive odontogenic epithelial neoplasm mimicking metastatic breast carcinoma: sclerosing odontogenic carcinoma?
206
Abstracts
CYTOMEGALOVIRUS INCLUSIONS IN HIV ASSOCIATED ORAL
KAPOSI'S SARCOMA. W. Yih, C. Meshul, F. Kratochvil. Oregon Health Sciences U., Portland Cytomegalovirus (CMV) has been implicated as a possible initiating factor in the development of Kaposi' s sarcoma (KS). The relationship between C M V and KS has been very controversial and most investigators admit that a causative association has not been proven. A number of studies have demonstrated the presence of C M V D N A and/or antigens in both extraoral and intraoral KS. Several investigators have reported the presence of CMV-type inclusions, by light microscopy, in cases of KS but there is only a rare intraoral example. We reviewed 12 cases of intraoral KS and noted two of those cases demonstrated CMV-type inclusions by light microscopy. Immunohistochemical staining with antibodies against C M V were positive in those two cases. Electron microscopy demonstrated herpes-type viral particles c/w CMV. These light findings m a y be a manifestation of generalized C M V infection rather than a sole causative factor. O D O N T O G E N I C KERATOCYSTS OF THE MIDLINE MAXILLARY
REGION. B. Neville, D. Damm, and T. Brock. Medical U. of South Carolina, Charleston, and U. of Kentucky, Lexington. The odontogenic keratocyst (OKC) is a distinctive histopathologic lesion that is estimated to represent 10% to 12% of all developmental odontogenic cysts. Approximately 65% of OKCs occur in the mandible with a predilection for the molarh'amus area. They often clinically mimic other odontogenic lesions, such as the dentigerous cyst, lateral periodontal cyst, and periapical cyst. The purpose of this paper is to report a series of 18 O K C s of the anterior maxillary region that crossed the midline and clinically mimicked nasopalatine duct cysts or periapical cysts. The patients' ages ranged from 41 to 87 years, with a mean age o f 69.9 years. Twelve of the 18 patients were in the seventh and eighth decades of life, and 16 of the patients were past the age of 60. These figures vary dramatically from O K C s in general, where the reported m e a n age is from 37 to 40 years. Thirteen of the cases were in males and five were in females. The most c o m m o n clinical diagnoses were periapical cyst (8 cases) and nasopalatine duct cyst (7 cases). The clinical diagnosis of O K C was suggested in only two cases, one of which occurred in a patient with the nevoid basal cell carcinoma syndrome. Recognition of the O K C is important because the lesion has a high recurrence rate, which has been estimated at around 30%. In addition, the O K C is the specific type of odontogenic cyst that m a y sometimes be associated with the nevoid basal cell carcinoma syndrome. Because of these features, it is important for the clinician to consider the diagnosis of O K C for lesions of the midline maxillary region, especially when they occur in an older patient. AGGRESSIVE O D O N T O G E N I C EPITHELIAL NEOPLASM M I M ICKING METASTATIC BREAST CARCINOMA:SCLEROSING O D -
ONTOGENIC CARCINOMA? D. Landwehr, C. Allen. The Ohio State University, Columbus. While most odontogenic tumors fall into relatively well-known categories, occasionally a lesion will present that defies classification. Report of case: A 46-year-old white w o m a n presented initially to her dentist with pain in her right mandible. Radiographically, axial and coronal images revealed a poorly defined osteolytic lesion at the right angle of the mandible. The buccal cortical plate was perforated while the lingual cortical plate was markedly thinned. An oral surgeon submitted an initial biopsy specimen consisting of dense fibrous connective tissue admixed with small islands of neoplastic epithelial cells that were moder-
O R A L SURGERY O R A L MEDICINE O R A L P A T H O L O G Y August 1996 ately pleomorphic with enlarged nuclei and clear to eosinophilic cytoplasm. No peripheral palisading of the nuclei was seen, and a c o m m e n t to rule out metastatic breast disease was appended. Follow-up breast biopsy and m a m m o g r a m proved negative, as were CT and bone scans. At this point, the oral surgeon submitted a second portion of the original biopsy, which he had retained, to a general pathologist who rendered a diagnosis of ameloblastoma. This tissue revealed larger epithelial islands; however, nuclear palisading was still not present. Consultations were obtained, and a consensus diagnosis of odontogenic carcinoma was reached. The patient had a resection with 1 cm margins that grossly appeared normal, however, lesional tissue was present at both the anterior and posterior margins microscopically. Follow-up surgery with frozen sections resulted in final margins at the contralateral canine and the neck of the condyle. Because of its unique histopathologic features and aggressive clinical behavior, we propose the term "sclerosing odontogenic carcinoma" for this lesion. CALIBER-PERSISTENT LABIAL ARTERY: A C O M M O N ANAT O M I C VARIANT OF NORMAL. J. Lovas, B. Rodu, H. Hammond,
C. Allen and G. Wysocki Dalhousie U, Halifax; U. of Alabama, Birmingham; U. of Iowa, Iowa City; Ohio State U, Columbus; and U. of Western Ontario, London. Only 16 cases of caliber-persistent labial artery (CPLA) of the lip have been reported to date in the English-language literature. Six of these were clinically misdiagnosed and treated with wedge resection as squamous cell carcinoma. In 1993, Lovas and Goodday reported the first two cases of C P L A to be diagnosed clinically since the original description of the condition by Howell and Freeman in 1973 under the name "prominent inferior labial artery." To date, the present authors have seen 99 cases clinically and 26 cases through our biopsy services. The senior author (J.L.) sees several cases daily, as incidental findings, in his Clinical O M F Pathology practice. The purpose of this paper is to familiarize OMF pathologists with the clinical and histologic features of this seldom-reported but very c o m m o n anatomic variant of normal. Careful clinical observation usually reveals a soft bluish linear or papular elevation above the labial mucosal surface. The unique feature is pulsation, not simply toward and away from the observer, which is usually caused by an underlying artery, but also laterally, which only an artery can exhibit. All but 2 of our 125 cases were asymptomatic; the upper to lower lip ratio among the clinical cases was 2:1, yet three times as m a n y lower than upper lip lesions were biopsied. Men and w o m e n were equally affected. None of the clinical histories from the biopsied cases mentioned pulsation. A vascular term (artery, hemangioma, phlebolith, varix, vascular malformation) was mentioned in half of the clinical differential diagnoses. Unlike the Miko et al. (1980, 1983, 1988) cases, none of our cases presented as a clinical ulcer, nor was carcinoma ever mentioned in the clinical differential diagnosis. Clinicians should carefully look for lateral pulsation in lip mucosal nodules so as to avoid unnecessary surgery and intraoperative arterial bleeding. Pathologists should realize that relatively large caliber superficial arteries in lip biopsies are not coincidental findings but represent the presumed " l e s i o n s " for which the biopsies were obtained. , INTRAORAL MANIFESTATION OF CUTANEOUS T-CELL LYM-
PHOMA, MYCOSIS FUNGOIDES. A. Manganaro, J. Startzell. Brooke Army Medical Center, Fort Sam Houston, Tex. A 61-year-old w o m a n presented with swelling of the right mucobuccal fold which at the time of her appointment was of 24-hours duration. The lesion was painless with hypesthesia of the
Abstracts
CYTOMEGALOVIRUS INCLUSIONS IN HIV ASSOCIATED ORAL
KAPOSI'S SARCOMA. W. Yih, C. Meshul, F. Kratochvil. Oregon Health Sciences U., Portland Cytomegalovirus (CMV) has been implicated as a possible initiating factor in the development of Kaposi' s sarcoma (KS). The relationship between C M V and KS has been very controversial and most investigators admit that a causative association has not been proven. A number of studies have demonstrated the presence of C M V D N A and/or antigens in both extraoral and intraoral KS. Several investigators have reported the presence of CMV-type inclusions, by light microscopy, in cases of KS but there is only a rare intraoral example. We reviewed 12 cases of intraoral KS and noted two of those cases demonstrated CMV-type inclusions by light microscopy. Immunohistochemical staining with antibodies against C M V were positive in those two cases. Electron microscopy demonstrated herpes-type viral particles c/w CMV. These light findings m a y be a manifestation of generalized C M V infection rather than a sole causative factor. O D O N T O G E N I C KERATOCYSTS OF THE MIDLINE MAXILLARY
REGION. B. Neville, D. Damm, and T. Brock. Medical U. of South Carolina, Charleston, and U. of Kentucky, Lexington. The odontogenic keratocyst (OKC) is a distinctive histopathologic lesion that is estimated to represent 10% to 12% of all developmental odontogenic cysts. Approximately 65% of OKCs occur in the mandible with a predilection for the molarh'amus area. They often clinically mimic other odontogenic lesions, such as the dentigerous cyst, lateral periodontal cyst, and periapical cyst. The purpose of this paper is to report a series of 18 O K C s of the anterior maxillary region that crossed the midline and clinically mimicked nasopalatine duct cysts or periapical cysts. The patients' ages ranged from 41 to 87 years, with a mean age o f 69.9 years. Twelve of the 18 patients were in the seventh and eighth decades of life, and 16 of the patients were past the age of 60. These figures vary dramatically from O K C s in general, where the reported m e a n age is from 37 to 40 years. Thirteen of the cases were in males and five were in females. The most c o m m o n clinical diagnoses were periapical cyst (8 cases) and nasopalatine duct cyst (7 cases). The clinical diagnosis of O K C was suggested in only two cases, one of which occurred in a patient with the nevoid basal cell carcinoma syndrome. Recognition of the O K C is important because the lesion has a high recurrence rate, which has been estimated at around 30%. In addition, the O K C is the specific type of odontogenic cyst that m a y sometimes be associated with the nevoid basal cell carcinoma syndrome. Because of these features, it is important for the clinician to consider the diagnosis of O K C for lesions of the midline maxillary region, especially when they occur in an older patient. AGGRESSIVE O D O N T O G E N I C EPITHELIAL NEOPLASM M I M ICKING METASTATIC BREAST CARCINOMA:SCLEROSING O D -
ONTOGENIC CARCINOMA? D. Landwehr, C. Allen. The Ohio State University, Columbus. While most odontogenic tumors fall into relatively well-known categories, occasionally a lesion will present that defies classification. Report of case: A 46-year-old white w o m a n presented initially to her dentist with pain in her right mandible. Radiographically, axial and coronal images revealed a poorly defined osteolytic lesion at the right angle of the mandible. The buccal cortical plate was perforated while the lingual cortical plate was markedly thinned. An oral surgeon submitted an initial biopsy specimen consisting of dense fibrous connective tissue admixed with small islands of neoplastic epithelial cells that were moder-
O R A L SURGERY O R A L MEDICINE O R A L P A T H O L O G Y August 1996 ately pleomorphic with enlarged nuclei and clear to eosinophilic cytoplasm. No peripheral palisading of the nuclei was seen, and a c o m m e n t to rule out metastatic breast disease was appended. Follow-up breast biopsy and m a m m o g r a m proved negative, as were CT and bone scans. At this point, the oral surgeon submitted a second portion of the original biopsy, which he had retained, to a general pathologist who rendered a diagnosis of ameloblastoma. This tissue revealed larger epithelial islands; however, nuclear palisading was still not present. Consultations were obtained, and a consensus diagnosis of odontogenic carcinoma was reached. The patient had a resection with 1 cm margins that grossly appeared normal, however, lesional tissue was present at both the anterior and posterior margins microscopically. Follow-up surgery with frozen sections resulted in final margins at the contralateral canine and the neck of the condyle. Because of its unique histopathologic features and aggressive clinical behavior, we propose the term "sclerosing odontogenic carcinoma" for this lesion. CALIBER-PERSISTENT LABIAL ARTERY: A C O M M O N ANAT O M I C VARIANT OF NORMAL. J. Lovas, B. Rodu, H. Hammond,
C. Allen and G. Wysocki Dalhousie U, Halifax; U. of Alabama, Birmingham; U. of Iowa, Iowa City; Ohio State U, Columbus; and U. of Western Ontario, London. Only 16 cases of caliber-persistent labial artery (CPLA) of the lip have been reported to date in the English-language literature. Six of these were clinically misdiagnosed and treated with wedge resection as squamous cell carcinoma. In 1993, Lovas and Goodday reported the first two cases of C P L A to be diagnosed clinically since the original description of the condition by Howell and Freeman in 1973 under the name "prominent inferior labial artery." To date, the present authors have seen 99 cases clinically and 26 cases through our biopsy services. The senior author (J.L.) sees several cases daily, as incidental findings, in his Clinical O M F Pathology practice. The purpose of this paper is to familiarize OMF pathologists with the clinical and histologic features of this seldom-reported but very c o m m o n anatomic variant of normal. Careful clinical observation usually reveals a soft bluish linear or papular elevation above the labial mucosal surface. The unique feature is pulsation, not simply toward and away from the observer, which is usually caused by an underlying artery, but also laterally, which only an artery can exhibit. All but 2 of our 125 cases were asymptomatic; the upper to lower lip ratio among the clinical cases was 2:1, yet three times as m a n y lower than upper lip lesions were biopsied. Men and w o m e n were equally affected. None of the clinical histories from the biopsied cases mentioned pulsation. A vascular term (artery, hemangioma, phlebolith, varix, vascular malformation) was mentioned in half of the clinical differential diagnoses. Unlike the Miko et al. (1980, 1983, 1988) cases, none of our cases presented as a clinical ulcer, nor was carcinoma ever mentioned in the clinical differential diagnosis. Clinicians should carefully look for lateral pulsation in lip mucosal nodules so as to avoid unnecessary surgery and intraoperative arterial bleeding. Pathologists should realize that relatively large caliber superficial arteries in lip biopsies are not coincidental findings but represent the presumed " l e s i o n s " for which the biopsies were obtained. , INTRAORAL MANIFESTATION OF CUTANEOUS T-CELL LYM-
PHOMA, MYCOSIS FUNGOIDES. A. Manganaro, J. Startzell. Brooke Army Medical Center, Fort Sam Houston, Tex. A 61-year-old w o m a n presented with swelling of the right mucobuccal fold which at the time of her appointment was of 24-hours duration. The lesion was painless with hypesthesia of the