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Botryoid odontogenic cyst
Botryoid odontogeniccyst Report of a case with histologic evidence of multicentric origin R. S. Redman, B. W. Whitestone, C. E. Winne, M. W. Hudec, R. H. Patterson: Botryoid odontogenic cyst." report of a case with histologic evidence of multicentric origin. Int. J. Oral Maxillofac. Surg. 1990; 19: 144-146.
Robert S. Redman 1, Brian W. Whitestone la, Cynthia E. Winne la, Michael W. Hudec 1, Robert H. Patterson 3,4 1Dental and 3Laboratory Services, Department of Veterans Affairs Medical Center; 2Georgetown University School of Dentistry; and 4George Washington University School of Medicine, Washington, District of Columbia, USA
Abstract. Histologic examination of an excisionally biopsied botryoid odontogenic cyst (BOC) documented that it was composed of at least 2 separate cysts, the location of which suggested that both originated within the alveolar bone rather than from within the periodontal ligament. These observations provide evidence for a multicentric origin of this example of BOC, and are consistent with a previous suggestion that lateral periodontal cysts of non-inflammatory origin arise from remnants of the dental lamina.
Botryoid odontogenic cyst (BOC) is the term first employed by WEATHERS t~ WALDRON 8 for a multilocular variant of the lateral periodontal cyst. The BOC apparently is u n c o m m o n , and only recently has there been recognition that it has a tendency to recur 3-5'7. We present a case that was biopsied excisionally, and thus provided an unusual opportunity to c o m m e n t on the histogenesis and management of the BOC.
Case report A 67-year-old black male presented to the Dental Service of the Department of Veterans Affairs Medical Center, Washington, DC, with a complaint of a toothache. Examination revealed a periapical abscess of the mandibular right first molar to be the cause of the patient's discomfort. However, also seen on the radiographs was a multilocular radiolucency involving the roots of the mandibular right canine and first premolar (Fig. 1). There appeared to be 3 separate radiolucencies. One, ca. 0.7 cm in diameter, was between the roots at their middle third; a second, ca. 1.2 cm, was inferior to the first one; and a third, ca. 1.6 cm, was distal to the premolar root. The first 2 had faint radiopaque rims, but the distal one had less distinct boundaries. All 3 extended bnccally and lingually beyond the roots of the teeth. The width of the periodontal ligament space seemed normal. There was a slightly compressible swelling of the mandible buccal to the premolar and canine teeth, but no paresthesia, tenderness or other changes in sensation. The 2 teeth were asymptomatic, had
neither caries nor restorations, and responded within normal limits to electric and thermal pulp tests. There also were slight buccolingual mobility and 4-6 mm periodontal pockets that did not communicate with the radiolucent areas. The location and radiologic appearance of the abnormality and the age of the patient suggested a differential diagnosis of botryoid odontogenic cyst, odontogenic keratocyst and ameloblastoma, with other entities being less likely. During planning for access to and removal of the lesions, it became evident that the boney support would be severely (premolar) to moderately (canine) compromised, and that both teeth were likely to be devitalized because of the proximity of the second radiolucent area to both root apices. In addition, any of the most likely pathologic enti-
Key words: odontogenic cyst; botryoid odontogenic cyst. Accepted for publication 3 January 1990
ties of the differential diagnosis would have a tendency to recur, and, given the patient's past history of medical and dental care, he would be unreliable for follow up. Therefore an excisional biopsy was performed that included the canine and premolar teeth. The resulting defect later was prepared for a prosthesis with a vestibuloplasty, lowering of the muscle attachments and split thickness skin graft.
Histopathologic findings Microscopically, the portion of the multilocular radiolucency between the canine and premolar proved to be 2 cysts, separated by about 4 m m of bone at their closest approximation, as dem-
Fig. 1. Panoramic radiograph taken at the time of the patient's initial visit, showing the periapical radiolucencies of the 1st molar and the multilocular radiolucency (arrows) around the Ist premolar in the right mandible.
BOC with multieentric origin onstrated in serial sections prepared from this part of the block (Fig. 2). Each consisted of a cystic cavity with a relatively thin connective tissue wall lined by non-cornified stratified squamous epithelium. The wall of the superior cyst contacted the periodontal ligaments of both teeth; that of the inferior cyst, only the canine, above the apex. Inflammatory cells were scarce in both cyst walls, and epithelial rests of Malassez were not seen. The epithelium varied in thickness from 1-15 cells in the superior cyst (Fig. 3) to 2-4 cell layers in the inferior one, with one or more plaque-like focal epithelial thickenings occurring in both lesions. In several places the epithelium and subjacent connective tissue were artifactually separated. Though the periodic acid-Schiff stain with and without
145
Fig. 3. Photomicrograph of a portion of the wall of the superior of the two cysts shown in Fig. 2, in another section stained with H&E. The epithelium varies from three to 12 cells deep, and there is a luminal thickening or plaque. Much of the basal cell layer consists of flattened rather than cuboidal cells, and the connective tissue of the walls is collagenous and almost devoid of inflammatory cells (magnification, x 140).
prior incubation with diastase indicated that some of the cells contained glycogen, this was distributed in the same pattern as in the non-cornified areas of the gingival epithelium in the same sections; no clear cells were seen. The radiolucent area distal to the premolar apparently was lost when the teeth were removed to promote more rapid decalcification, and no evidence of a third cyst was found. The histopathologic diagnosis was
multiple lateral periodontal cysts [botryold variant], or botryoid odontogenic cyst, associated with the mandibular premolar and canine. Discussion
Fig. 2. Survey photomicrograph of a buccolingual section containing the 2 cysts (arrows) between the canine and premolar, originally stained with PAS and hematoxylin and subsequently re-stained with H&E. The interdental papilla is at the top and the buccal alveolar mucosa covers most of the right side of the section. The cysts were centered within the alveolar bone and are separated by about 4 mm of bone, the closest approximation of the cyst walls observed in serial sections through this region. The wall of the superior cyst is surrounded by bone except at the top, where it contacts the interdental periodontal fibers, and the lingual margin, which was trimmed to facilitate sectioning (magnification, x 5.0).
Cysts that have been curetted are likely to be divided and distorted, causing difficulty in determining if the specimen consists of multiple cysts or a single cyst with outpocketings. Although the resection in the present care provided one example of the BOC with a histologically demonstrated multicentric origin, other examples of the BOC may arise in other ways. Except for the lack of glycogen-rich clear cells, both cysts had characteristics which others have suggested indicate genesis from remnants of the dental lamina9. Such an origin offers an explanation for the presence of these same
features in a subset of non-inflammatory lateral periodontal cysts and in the gingival cyst of the adult 9. The present case provides additional support for this suggestion. Both cysts were centered in the alveolar bone, suggesting that they had originated there and expanded until encroaching upon the periodontal ligaments of the premolar and canine. Origin from rests of Malassez seems less likely, as the cysts then should have been centered in or very close to the periodontal ligament. In addition to the recently documented risk of recurrence, it might be noted that the spectrum of malignant changes reported to have occurred in lateral periodontal cysts is similar to that of other odontogenic cysts 1,2.6. Additional cases with adequate follow-up will be required before choices regarding the best surgical treatment for the BOC can be made with confidence. At present, an initial approach more aggressive than curettage may be difficult to justify, except in unusual circumstances such as those of the present case.
References
1. BAKERRD, D'ONOFRIOED, CORIORL, CRAWFORD BE, TERRY BC. Squamous cell carcinoma arising in a lateral periodontal cyst. Oral Surg 1979: 47: 495-9.
146
Redman et al.
2. EVERSOLELR, SABESWR, ROVlN S. Aggressive growth and neoplastic potential of odontogenic cysts. Cancer 1975: 35: 270-82. 3. GREER JR RO, JOHNSON M. Botryoid odontogenic cyst: clinicopathologic analysis of ten cases with three recurrences. J Oral Maxillofac Surg 1988: 46: 574-9. 4. HE1LK1NHEIMOK, HAPPONENRP, FORSELL K, KUUSILEHTOA, VIRTANENI. A botryoid odontogenic cyst with multiple recurrences. Int J Oral Maxillofac Surg 1989: 18: 10-13.
Botryoid odontogenic cyst. Oral Surg 1986: 62: 555-9. 6. PADAYACHEEA, VAN WYK CW. Two cystic lesions with features of both the botryoid odontogenic cyst and the central mucoepidermoid tumor: sialo-odontogenic cyst? J Oral Pathol 1987: 16: 499-504. 7. PHELAN JA, KRITCHMAN D, FuscO-RA5. KAUGARS QE.
MER M, FREEDMAN PD, LUMERMAN H.
Recurrent botryoid odontogenic cyst (lateral periodontal cyst). Oral Surg 1988: 66: 345-8. 8. WEATHERS DR, WALDRON CA. U n u s u a l multilocular cysts of the jaws (botyroid
odontogenic cysts). Oral Surg 1973: 36: 235~41. 9. WYSOCKI GP, BRANNON RB, GARDNER DG, SAt'P E Histogenesis of the lateral periodontal cyst and gingival cyst of the adult. Oral Surg 1980: 50: 327-34. Address: Dr. Robert S. Redman Oral Pathology Research Laboratory (151-I) VA Medical Center 50 Irving Street, N W Washington, DC 20422 USA
Robert S. Redman 1, Brian W. Whitestone la, Cynthia E. Winne la, Michael W. Hudec 1, Robert H. Patterson 3,4 1Dental and 3Laboratory Services, Department of Veterans Affairs Medical Center; 2Georgetown University School of Dentistry; and 4George Washington University School of Medicine, Washington, District of Columbia, USA
Abstract. Histologic examination of an excisionally biopsied botryoid odontogenic cyst (BOC) documented that it was composed of at least 2 separate cysts, the location of which suggested that both originated within the alveolar bone rather than from within the periodontal ligament. These observations provide evidence for a multicentric origin of this example of BOC, and are consistent with a previous suggestion that lateral periodontal cysts of non-inflammatory origin arise from remnants of the dental lamina.
Botryoid odontogenic cyst (BOC) is the term first employed by WEATHERS t~ WALDRON 8 for a multilocular variant of the lateral periodontal cyst. The BOC apparently is u n c o m m o n , and only recently has there been recognition that it has a tendency to recur 3-5'7. We present a case that was biopsied excisionally, and thus provided an unusual opportunity to c o m m e n t on the histogenesis and management of the BOC.
Case report A 67-year-old black male presented to the Dental Service of the Department of Veterans Affairs Medical Center, Washington, DC, with a complaint of a toothache. Examination revealed a periapical abscess of the mandibular right first molar to be the cause of the patient's discomfort. However, also seen on the radiographs was a multilocular radiolucency involving the roots of the mandibular right canine and first premolar (Fig. 1). There appeared to be 3 separate radiolucencies. One, ca. 0.7 cm in diameter, was between the roots at their middle third; a second, ca. 1.2 cm, was inferior to the first one; and a third, ca. 1.6 cm, was distal to the premolar root. The first 2 had faint radiopaque rims, but the distal one had less distinct boundaries. All 3 extended bnccally and lingually beyond the roots of the teeth. The width of the periodontal ligament space seemed normal. There was a slightly compressible swelling of the mandible buccal to the premolar and canine teeth, but no paresthesia, tenderness or other changes in sensation. The 2 teeth were asymptomatic, had
neither caries nor restorations, and responded within normal limits to electric and thermal pulp tests. There also were slight buccolingual mobility and 4-6 mm periodontal pockets that did not communicate with the radiolucent areas. The location and radiologic appearance of the abnormality and the age of the patient suggested a differential diagnosis of botryoid odontogenic cyst, odontogenic keratocyst and ameloblastoma, with other entities being less likely. During planning for access to and removal of the lesions, it became evident that the boney support would be severely (premolar) to moderately (canine) compromised, and that both teeth were likely to be devitalized because of the proximity of the second radiolucent area to both root apices. In addition, any of the most likely pathologic enti-
Key words: odontogenic cyst; botryoid odontogenic cyst. Accepted for publication 3 January 1990
ties of the differential diagnosis would have a tendency to recur, and, given the patient's past history of medical and dental care, he would be unreliable for follow up. Therefore an excisional biopsy was performed that included the canine and premolar teeth. The resulting defect later was prepared for a prosthesis with a vestibuloplasty, lowering of the muscle attachments and split thickness skin graft.
Histopathologic findings Microscopically, the portion of the multilocular radiolucency between the canine and premolar proved to be 2 cysts, separated by about 4 m m of bone at their closest approximation, as dem-
Fig. 1. Panoramic radiograph taken at the time of the patient's initial visit, showing the periapical radiolucencies of the 1st molar and the multilocular radiolucency (arrows) around the Ist premolar in the right mandible.
BOC with multieentric origin onstrated in serial sections prepared from this part of the block (Fig. 2). Each consisted of a cystic cavity with a relatively thin connective tissue wall lined by non-cornified stratified squamous epithelium. The wall of the superior cyst contacted the periodontal ligaments of both teeth; that of the inferior cyst, only the canine, above the apex. Inflammatory cells were scarce in both cyst walls, and epithelial rests of Malassez were not seen. The epithelium varied in thickness from 1-15 cells in the superior cyst (Fig. 3) to 2-4 cell layers in the inferior one, with one or more plaque-like focal epithelial thickenings occurring in both lesions. In several places the epithelium and subjacent connective tissue were artifactually separated. Though the periodic acid-Schiff stain with and without
145
Fig. 3. Photomicrograph of a portion of the wall of the superior of the two cysts shown in Fig. 2, in another section stained with H&E. The epithelium varies from three to 12 cells deep, and there is a luminal thickening or plaque. Much of the basal cell layer consists of flattened rather than cuboidal cells, and the connective tissue of the walls is collagenous and almost devoid of inflammatory cells (magnification, x 140).
prior incubation with diastase indicated that some of the cells contained glycogen, this was distributed in the same pattern as in the non-cornified areas of the gingival epithelium in the same sections; no clear cells were seen. The radiolucent area distal to the premolar apparently was lost when the teeth were removed to promote more rapid decalcification, and no evidence of a third cyst was found. The histopathologic diagnosis was
multiple lateral periodontal cysts [botryold variant], or botryoid odontogenic cyst, associated with the mandibular premolar and canine. Discussion
Fig. 2. Survey photomicrograph of a buccolingual section containing the 2 cysts (arrows) between the canine and premolar, originally stained with PAS and hematoxylin and subsequently re-stained with H&E. The interdental papilla is at the top and the buccal alveolar mucosa covers most of the right side of the section. The cysts were centered within the alveolar bone and are separated by about 4 mm of bone, the closest approximation of the cyst walls observed in serial sections through this region. The wall of the superior cyst is surrounded by bone except at the top, where it contacts the interdental periodontal fibers, and the lingual margin, which was trimmed to facilitate sectioning (magnification, x 5.0).
Cysts that have been curetted are likely to be divided and distorted, causing difficulty in determining if the specimen consists of multiple cysts or a single cyst with outpocketings. Although the resection in the present care provided one example of the BOC with a histologically demonstrated multicentric origin, other examples of the BOC may arise in other ways. Except for the lack of glycogen-rich clear cells, both cysts had characteristics which others have suggested indicate genesis from remnants of the dental lamina9. Such an origin offers an explanation for the presence of these same
features in a subset of non-inflammatory lateral periodontal cysts and in the gingival cyst of the adult 9. The present case provides additional support for this suggestion. Both cysts were centered in the alveolar bone, suggesting that they had originated there and expanded until encroaching upon the periodontal ligaments of the premolar and canine. Origin from rests of Malassez seems less likely, as the cysts then should have been centered in or very close to the periodontal ligament. In addition to the recently documented risk of recurrence, it might be noted that the spectrum of malignant changes reported to have occurred in lateral periodontal cysts is similar to that of other odontogenic cysts 1,2.6. Additional cases with adequate follow-up will be required before choices regarding the best surgical treatment for the BOC can be made with confidence. At present, an initial approach more aggressive than curettage may be difficult to justify, except in unusual circumstances such as those of the present case.
References
1. BAKERRD, D'ONOFRIOED, CORIORL, CRAWFORD BE, TERRY BC. Squamous cell carcinoma arising in a lateral periodontal cyst. Oral Surg 1979: 47: 495-9.
146
Redman et al.
2. EVERSOLELR, SABESWR, ROVlN S. Aggressive growth and neoplastic potential of odontogenic cysts. Cancer 1975: 35: 270-82. 3. GREER JR RO, JOHNSON M. Botryoid odontogenic cyst: clinicopathologic analysis of ten cases with three recurrences. J Oral Maxillofac Surg 1988: 46: 574-9. 4. HE1LK1NHEIMOK, HAPPONENRP, FORSELL K, KUUSILEHTOA, VIRTANENI. A botryoid odontogenic cyst with multiple recurrences. Int J Oral Maxillofac Surg 1989: 18: 10-13.
Botryoid odontogenic cyst. Oral Surg 1986: 62: 555-9. 6. PADAYACHEEA, VAN WYK CW. Two cystic lesions with features of both the botryoid odontogenic cyst and the central mucoepidermoid tumor: sialo-odontogenic cyst? J Oral Pathol 1987: 16: 499-504. 7. PHELAN JA, KRITCHMAN D, FuscO-RA5. KAUGARS QE.
MER M, FREEDMAN PD, LUMERMAN H.
Recurrent botryoid odontogenic cyst (lateral periodontal cyst). Oral Surg 1988: 66: 345-8. 8. WEATHERS DR, WALDRON CA. U n u s u a l multilocular cysts of the jaws (botyroid
odontogenic cysts). Oral Surg 1973: 36: 235~41. 9. WYSOCKI GP, BRANNON RB, GARDNER DG, SAt'P E Histogenesis of the lateral periodontal cyst and gingival cyst of the adult. Oral Surg 1980: 50: 327-34. Address: Dr. Robert S. Redman Oral Pathology Research Laboratory (151-I) VA Medical Center 50 Irving Street, N W Washington, DC 20422 USA