639
MAXYMIW AND WOOD
J Oral Maxillofac Surg 49:639-643.1991
Carcinoma Arising in a Dentigerous
Cyst:
A Case Report and Review of the Literature W.G. MAXYMIW,
DDS* AND R.E. WOOD, DDS, MSct
The dentigerous cyst is associated with the crown of a permanent tooth. The most common sites are the mandibular third molar and maxillary canine areas.’ A prominent radiographic feature of dentigerous cysts is a radiolucent area associated in some manner with an unerupted tooth. The tooth crown may be surrounded symmetrically by this radiolucency or the lucency may appear to project laterally from the tooth crown?
Received from the Department of Dentistry, Princess Margaret Hospital, Toronto, Canada. * Chief. t Staff Dentist. Address correspondence and reprint requests to Dr Maxymiw: The Ontario Cancer Institute Incorporating The Rrincess Margaret Hospital, 500 Sherboume St, Toronto, Ontario, Canada M4X lK9. 0 1991 American Association of Oral and Maxillofacial geons 0278-2391/91/4908-0013$3.00/O
FIGURE 1. Radiograph at time of initial treatment. Note horizontally impacted right mandibular third molar with associated well-corticated, coronal radiolucency. The mandibular canal is displaced inferiorly (arrow). A portion of the follicular space is visible distal to impacted molar and the lesion has a dentigerous relationship to the tooth.
Sur-
There are no characteristic microscopic features that reliably distinguish dentigerous cysts. They are composed of a connective tissue wall with a thin layer of stratified squamous epithelium lining the lumen. Rete peg formation is generally absent except in secondarily infected cases. Inflammatory cell infiltration of the connective tissue is common.3 Squamous cell carcinoma arising in an odontogenit cyst is rare. The purpose of this article is to report a histologically proven case of carcinoma arising in a dentigerous cyst, to detail its treatment, and to analyze the literature to determine how many cases of carcinoma arising in cysts fulfill criteria of histological proof of transition from normal cyst lining to carcinoma.4 Report of Case A 72-year-old man was referred with a diagnosis of squamous cell carcinoma of a right mandibular cyst. Panoramic radiographic examination (Fig 1) at the time of his original presentation (10/14/86) showed a horizontally im-
640
CARCINOMA ARISING IN A DENTIGEROUS
CYST
pacted, mandibular right third molar with an associated pericoronal radiolucency. The asymptomatic lesion had been surgically enucleated. Postoperatively, he experienced numbness affecting the right mandible and right lower lip. The patient gradually developed a stabbing pain that radiated to the right lower lip for which he was placed on oral analgesics. He had difficulty chewing, resorted to pureed foods, and subsequently lost 15 lb. He visited yet another oral surgeon (5/7/86), and a repeated panoramic radiograph of the mandible showed multiple areas of bone destruction. The overall appearance was thought to be consistent with osteomyelitis, but a contiiing biopsy was deferred because the patient developed atrial fibrillation. A follow-up panoramic radiograph (9/19/86) demonstrated progressive bone destruction, with irregular margins and infiltration into normal bone suggestive of malignancy. A computer-assisted tomographic examination revealed an adjacent soft-tissue mass as well as a pathologic fracture (Fig 2). A biopsy was eventually performed (9/26/86), and a diagnosis of well-differentiated squamous cell carcinoma was made. Although the original histologic diagnosis was dentigerous cyst, review of the histologic sections showed carcinoma arising in a dentigerous cyst (Fig 3). The patient was referred for surgical care, but because of serious medical considerations was offered radiation treatment. Examination prior to radiation showed a right mandibular swelling visible extraorally. The overlying mucosa in the right mandible was intact, with no evidence of any abnormality. Further investigations, including alkaline and acid phosphatase and a technetium 99m methylene diphosphonate nuclear scan failed to indicate evidence of metastatic disease. Radiotherapy consisted of right anterior and posterior oblique fields to the right face (a total dose of 5,750 cGy over 20 fractions). The contralateral parotid was spared. The treatment volume encompassed the right ascending ramus and body of the mandible extending to the anterior mandible. The right posterior maxilla was also within the treatment field. The maxilla and mandible received doses of 5,250 to 6,000 cGy. The patient’s congestive heart failure was controlled with medication, although his heart rate remained fast, ranging from 100 to 120 beats per minute. Following the completion of his radiation treatments, the patient was admitted to another hospital with increasing heart failure and atrial fibrillation. On March 20, 1987, he died of congestive cardiac failure complicated by end-stage renal failure. He was free of disease at the time of his death.
Discussion Before a diagnosis of carcinoma arising from a cyst lining can be established, a number of alternative possibilities must be excluded. It is possible that cyst and neoplasm may develop independently adjacent to one another and fuse. The oral mucosa
in this patient was perfectly normal. Alternately, cystic degeneration of an epithelial neoplasm may occur. This also did not happen in this patient because the original biopsy showed no evidence of a benign odontogenic tumor. The only incontrovertible proof that a carcinoma originated in an odontogenic cyst is the demonstration of a transition of
FIGURE 2. Computer-assisted tomographic scan shows a very large osteolytic lesion in the mandible (arrow) with an associated soft-tissue mass extending into the adjacent areas of the cheek. There is also a pathologic fracture.
the cells lining the cyst from a benign epithelium
to a carcinoma, which the current case demonstrates.4 A review of the English literature from 1947 shows 54 reported cases of carcinoma arising in a dentigenous cyst. 5-51 Of these, 19 cases satisfy Gardner’s criteria 5,9,16,21,24,29-32,34,37,38,43-47 These are reviewed, along with the present case, in Table 1. Carcinomatous change in cysts can proceed to an
MAXYMIW AND WOOD
641
FIGURE 3. A, Low-power photomicrograph showing transition of cyst lining epithehum to carcinoma (arrow) (hematoxylin-eosin stain, original magnification x25). B. Highpower photomicrograph showing microinvasion (arrow) in area of transition (hematoxylin-eosin stain, original magnification x63).
inoperable stage without ._ affecting the general condition of the patient. I\1An important clinical sign is rapid, firm, nontender enlargement of the jaw. Periodic symptoms of neuralgic pain and paresthesia may be experienced before the cancer becomes clinically evident. Lymph nodes are not commonly
involved. Extension to the oral cavity can follow, with exposure of tumor and rapid progression of the disease.3’ The tumor may extend perineurally along the mandibular cana1.28 It is often associated with failure of an extraction site to heal and displacement of teeth.”
642
CARCINOMA ARISING IN A DENTIGEROUS
Table 1.
CYST
Reported Cases With Transition of Normal to Malignant Epithelium in Cyst Wall
Site
Primary Treatment
Additional Treatment
Author(s)
Year
Age (yr)
Angelopoulos et al5 Banetjees Chretien et alI6 FrankI& Wiesne?’
1%6 1967 1970 1949
74 37 55 38
M M M F
Mandible Maxilla Mandible Maxilla
Resection Enucleation Enucleation Enucleation
Hardmanz4
1963
73
F
Mandible
Enucleation
Not stated Radiation Resection Resection Radiation Resection
Hardmanz4
1963
77
F
Mandible
Resection
Resection
Lapin et alz9
1973
55
M
Mandible
Enucleation
Resection
Lavery et a13’ Lee & Loke3i
1987 1%7
46 57
M M
Maxilla Maxilla
Enucleation Enucleation
Resection Resection
Macleod & Soames3’ Martensson
1988 1955
57 49
M M
Mandible Maxilla
Resection Enucleation
Not stated Radiation
Martensson Maxymiw & Wood
1955 1990
64 72
M M
Mandible Mandible
Enucleation Enucleation
Resection Radiation
Moos & Rennie3’ Nithianda3*
1987 1983
67 59
F M
Mandible Maxilla
Enucleation Resection
None None
Siar & Ng43 Stokke & Koppang45
1987 1968
40 90
M F
Maxilla Mandible
Enucleation Enucleation
Resection None
Van Der Waal et ale
1985
85
F
Mandible
?
Not stated
Sex
Radiographically odontogenic cysts undergoing malignant transformation may show margins that are jagged and have indentations and indistinct borders. Ultimately, erosion of the buccal, labial, and lingual plates occurs and roots of adjacent teeth are resorbed.38 If enlargement remains unchecked, pathologic fractures occur, as in the present case. Histologic review demonstrates malignant transformation accompanied by a chronic infiltrate of lymphocytes and plasma cells in the cyst wall connective tissue; this was seen in this instance. The cysts are lined in whole or in part by dyskeratotic squamous epithelium. The lining of stratified squamous epithelium shows the actual transition from normal squamous epithelium to invasive squamouscell carcinoma, which proliferates into the connective tissue wall of the cyst. Some regions of the cyst lining may contain intraepithelial carcinoma 11.30.32.37.52 Because the tumor is so rare, there is little accumulation of experience with regard to treatment. Treatment is based on experience with tumors arising elsewhere in the oral cavity. At the time of initial surgery, malignancy is usually not expected and the treatment is usually enucleation or marsupial-
Health Status Not stated Dead of disease Dead of disease Not stated Alive, no evidence of disease Alive, no evidence of disease Alive, no evidence of disease Not stated Alive, no evidence of disease Not stated Alive, no evidence of disease Not stated Dead of other causes Dead of disease Alive, no evidence of disease Not stated Dead of other causes Dead of other causes
Time From Diagnosis to Death Not stated 10 Months 13 Months
6 Months Not stated
4 Months 3 Weeks
ization. This was done in the present case, and the pathology was misinterpreted as normal. Because these tumours originate within bone, osseous erosion and invasion occur early. Following diagnosis, most patients are treated by wide resection of the affected jaw.47 The initial misdiagnosis of the patient’s condition in the present report underlines the importance of careful review of all histologic sections. References 1. Shafer WG, Hine MK, Levy BM: A Textbook of Oral Pathology (ed 4). Philadelphia, PA, Saunders, 1983, p 258 2. Worth HM: Principles and practices of Oral Radiographic interpretation (ed 2). Chicago, IL, Year Book Medical, 1972, p 420 3. Regezi JA, Sciubba JJ: Oral Pathology: Clinical Pathologic Conference. Philadelphia, PA, Saunders, 1989, p 301 4. Gardner AF: A survey of odontogenic cysts and their relationship to squamous cell carcinoma. J Can Dent Assoc 42: 161, 1975 5. Angelopoulos AP, Tilson HB, Stewart FW, et al: Malignant transformation of the epithelial lining of the odontogenic cysts. Oral Surg Oral Med Oral Path01 4:415. 1966 6. Arekn RG, McCaichey KD, Baker HL: Squamous cell carcinoma developing in an odontogenic keratocyst. Arch Otolaryngol 107:568, 1981
643
MAXYMIW AND WOOD
7. Baker RD, Donofrio ED, Corio RL, et al: Squamous cell carcinoma arising in a lateral periodontal cyst. Oral Surg Oral Med Oral Path01 47:495, 1979 8. Banejee SC: Squamous cell carcinoma in a maxillary cyst. Oral Surg Oral Med Oral Path01 23: 193, 1967 9. Berenholz L, Gottlieb RD, Cho SY, et al: Squamous cell carcinoma arising in a dentigerous cyst. Ear Nose Throat J 67:764, 1988 10. Bradfield WJD, Broadway ES: Malignant change in a dentigerous cyst. Br J Surg 45:657, 1958 I I. Bradley N, Thomas DM, Antoniades K. et al: Squamous cell carcinoma arising in an odontogenic cyst. Int J Oral Maxillofac Surg 17:260, 1988 12. Browne RM, Cough NC: Malignant change in the epithelium lining odontoeenic cvsts. Cancer 29: 1199. 1972 13. Brown: RM: Metaplasia and degeneration in odontogenic cysts in man. J Oral Pathol 1:145. 1972 14. Chongruk C: Cyst and carcinoma. Oral Surg Oral Med Oral Pathol 51:219, 1981 15. Choukas NC: Controversial lesion of the mandible: Report of case. J Oral Surg 29579. 1971 16. Chretien PB, Carpenter DF, White NS, et al: Squamous carcinoma arising in a dentigerous cyst. Oral Surg Oral Med Oral Pathol 30:809. 1970 17. Darlington CG, Ehrlich HE, Seldin HM: Malignant transformation of odontogenic cyst: Report of case. J Oral Surg 1164, 1953 18. Enriquez RE, Ciola B, Bahn SL: Verrucous carcinoma arising in an odontogenic cyst. Oral Surg Oral Med Oral Pathol 49: 151, 1980 19. Falkmer S. Herberts G, Olven S: Carcinoma arising in odontogenic cysts of the jaw. Odontol Tidskr 65:220,1957 20. Frank1 Z, Wiesner J: Cancer starting from a cyst. Dent Items Int 71564, 1949 2 1. Frank1 Z, Bouyssou M: Premalignant and malignant changes in the lining of odontogenic cysts and its therapeutic consequences. Dent Sci Res 9:93, 1976 22. Hampl PF, Harrigan WF: Squamous cell carcinoma possibly arising from a odontogenic cyst: Report of case. J Oral Surg 31:359, 1973 23. Hankey GT. Pedler JA: Primary squamous cell carcinoma of mandible arising from epithelial lining of dental cyst. Proc R Sot Med 50:680, 1957 24. Hardman FG: Carcinomata arising in dental cysts. Br J Oral Surg 1:124, 1963 25. High AS. Quirke P, Hume WJ: DNA-ploidy studies in a keratocyst undergoing subsequent malignant transformation. J Oral Path01 16:135. 1987 26. Meadow SR: Malignant change in a dental cyst. Oral Surg Oral Med Oral Pathol 21:282. 1%6 27. Kay LW, Kramer RH: Squamous-cell carcinoma arising in a dental cyst. Oral Surg Oral Med Oral Pathol 15:970, 1962 28. Kramer HS, Scribner JH: Squamous-cell carcinoma arising in a dentigerous cyst. Oral Surg Oral Med Oral Path01 19:555, 1965 29. Lapin R, Garfinkel AV, Catania AF, et al: Squamous cell carcinoma arising in a dentigerous cyst. J Oral Surg 31:354. 1973
30. Lavery K, Blomquist JE, Awty MD, et al: Squamous carcinoma arising in a dental cyst. Br Dent J 162:259, 1987 31. Lee KW, Loke SJ: Squamous cell carcinoma arising in a dentigerous cyst. Cancer 20:2241, 1%7 32. MacLeod RI, Soames JV: Squamous cell carcinoma arising in an odontogenic keratocyst. Br J Oral Maxillofac Surg 26:52, 1988 33. Martensson G: Cystor och cancer i kakama ett diagnostiskt problem. Sven Tandlak Tidskr 47:509, 1954 34. Martensson G: Cysts and carcinoma of the jaws. Oral Surg Oral Med Oral Pathol 8:673, 1955 35. Martinelli C, Melhado RM, Cahstini EA: Squamous cell carcinoma in a residual mandibular cyst. Oral Surg Oral Med Oral Pathol44:274, 1977 36. Meadow SR: Malignant change in a dental cyst. Oral Surg Oral Med Oral Path01 21:282, 1966 37. Moos KF, Rennie JS: Squamous cell carcinoma arising in a mandibular keratocyst in a patient with Gorlin’s syndrome. Br J Oral Maxillofac Surg 25:280, 1987 38. Nithiananda S: Squamous cell carcinoma arising in the lining of an odontogenic cyst. Br J Oral Surg 21:56, 1983 39. Norris LH, Baghaei-Rad M, Maloney PL, et al: Bilateral maxihary squamous odontogenic tumours and the mahgnant transformation of a mandibular radiolucent lesion. J Oral Maxillofac Surg 42:827, 1984 40. Pearcey RG: Squamous-cell carcinoma arising in dental cysts. Clin Radio1 36:387, 1985 41. Schneider LC: Incidence of epithelial atypia in radicular cysts: A preliminary investigation. J Oral Surg 35:370, 1977 42. Sealey VT: Epitheliomatous
43.
44.
45. 46.
47.
48. 49. 50.
51.
52.
development in dental cyst. Aust Dent J 51:83, 1947 Siar CH, Ng KH: Squamous cell carcinoma in an orthokeratinised odontogenic keratocyst. Int J Oral Maxillofac Surg 16:95. 1987 Stoelinga PJW: The incidence, multiple presentation and aggressive cysts of the jaws. J Craniomaxillofac Surg 16:184, 1988 Stokke T, Koppang HS: Squamous cell carcinoma arising in mandibular cyst. Acta Odontol Stand 26667, 1968 Van Der Waal I, Rauhamaa R, Van Der Kwast WAM. et al: Squamous cell carcinoma arising in the lining of odontogenie cysts. Int J Oral Surg 14:146, 1985 Waldron CA, Mustoe TA: Primary intraosseous carcinoma of the mandible with probable origin in an odontogenic cyst. Oral Surg Oral Med Oral Pathol 67:716, 1989 Ward TG, Cohen B: Squamous carcinoma in a mandibular cyst. Br J Oral Surg 1:8, 1963 Weisinger E, Doyle JL: Dental cyst with invasive carcinoma. N Y State Dent J 37:8, 1971 Whitlock RIH, Jones JH: Squamous cell carcinoma of the jaw arising in a simple cyst. Oral Surg Oral Med Oral Path01 24:530, 1967 Williams IE, Newman CW: Squamous-cell carcinoma associated with a dentigerous cyst of the maxilla. Oral Surg Oral Med Oral Pathol 16:1012, 1963 Gardner AF: The odontogenic cyst as a potential carcinoma: A clinicopathologic appraisal. J Am Dent Assoc 78:746, 1969
MAXYMIW AND WOOD
J Oral Maxillofac Surg 49:639-643.1991
Carcinoma Arising in a Dentigerous
Cyst:
A Case Report and Review of the Literature W.G. MAXYMIW,
DDS* AND R.E. WOOD, DDS, MSct
The dentigerous cyst is associated with the crown of a permanent tooth. The most common sites are the mandibular third molar and maxillary canine areas.’ A prominent radiographic feature of dentigerous cysts is a radiolucent area associated in some manner with an unerupted tooth. The tooth crown may be surrounded symmetrically by this radiolucency or the lucency may appear to project laterally from the tooth crown?
Received from the Department of Dentistry, Princess Margaret Hospital, Toronto, Canada. * Chief. t Staff Dentist. Address correspondence and reprint requests to Dr Maxymiw: The Ontario Cancer Institute Incorporating The Rrincess Margaret Hospital, 500 Sherboume St, Toronto, Ontario, Canada M4X lK9. 0 1991 American Association of Oral and Maxillofacial geons 0278-2391/91/4908-0013$3.00/O
FIGURE 1. Radiograph at time of initial treatment. Note horizontally impacted right mandibular third molar with associated well-corticated, coronal radiolucency. The mandibular canal is displaced inferiorly (arrow). A portion of the follicular space is visible distal to impacted molar and the lesion has a dentigerous relationship to the tooth.
Sur-
There are no characteristic microscopic features that reliably distinguish dentigerous cysts. They are composed of a connective tissue wall with a thin layer of stratified squamous epithelium lining the lumen. Rete peg formation is generally absent except in secondarily infected cases. Inflammatory cell infiltration of the connective tissue is common.3 Squamous cell carcinoma arising in an odontogenit cyst is rare. The purpose of this article is to report a histologically proven case of carcinoma arising in a dentigerous cyst, to detail its treatment, and to analyze the literature to determine how many cases of carcinoma arising in cysts fulfill criteria of histological proof of transition from normal cyst lining to carcinoma.4 Report of Case A 72-year-old man was referred with a diagnosis of squamous cell carcinoma of a right mandibular cyst. Panoramic radiographic examination (Fig 1) at the time of his original presentation (10/14/86) showed a horizontally im-
640
CARCINOMA ARISING IN A DENTIGEROUS
CYST
pacted, mandibular right third molar with an associated pericoronal radiolucency. The asymptomatic lesion had been surgically enucleated. Postoperatively, he experienced numbness affecting the right mandible and right lower lip. The patient gradually developed a stabbing pain that radiated to the right lower lip for which he was placed on oral analgesics. He had difficulty chewing, resorted to pureed foods, and subsequently lost 15 lb. He visited yet another oral surgeon (5/7/86), and a repeated panoramic radiograph of the mandible showed multiple areas of bone destruction. The overall appearance was thought to be consistent with osteomyelitis, but a contiiing biopsy was deferred because the patient developed atrial fibrillation. A follow-up panoramic radiograph (9/19/86) demonstrated progressive bone destruction, with irregular margins and infiltration into normal bone suggestive of malignancy. A computer-assisted tomographic examination revealed an adjacent soft-tissue mass as well as a pathologic fracture (Fig 2). A biopsy was eventually performed (9/26/86), and a diagnosis of well-differentiated squamous cell carcinoma was made. Although the original histologic diagnosis was dentigerous cyst, review of the histologic sections showed carcinoma arising in a dentigerous cyst (Fig 3). The patient was referred for surgical care, but because of serious medical considerations was offered radiation treatment. Examination prior to radiation showed a right mandibular swelling visible extraorally. The overlying mucosa in the right mandible was intact, with no evidence of any abnormality. Further investigations, including alkaline and acid phosphatase and a technetium 99m methylene diphosphonate nuclear scan failed to indicate evidence of metastatic disease. Radiotherapy consisted of right anterior and posterior oblique fields to the right face (a total dose of 5,750 cGy over 20 fractions). The contralateral parotid was spared. The treatment volume encompassed the right ascending ramus and body of the mandible extending to the anterior mandible. The right posterior maxilla was also within the treatment field. The maxilla and mandible received doses of 5,250 to 6,000 cGy. The patient’s congestive heart failure was controlled with medication, although his heart rate remained fast, ranging from 100 to 120 beats per minute. Following the completion of his radiation treatments, the patient was admitted to another hospital with increasing heart failure and atrial fibrillation. On March 20, 1987, he died of congestive cardiac failure complicated by end-stage renal failure. He was free of disease at the time of his death.
Discussion Before a diagnosis of carcinoma arising from a cyst lining can be established, a number of alternative possibilities must be excluded. It is possible that cyst and neoplasm may develop independently adjacent to one another and fuse. The oral mucosa
in this patient was perfectly normal. Alternately, cystic degeneration of an epithelial neoplasm may occur. This also did not happen in this patient because the original biopsy showed no evidence of a benign odontogenic tumor. The only incontrovertible proof that a carcinoma originated in an odontogenic cyst is the demonstration of a transition of
FIGURE 2. Computer-assisted tomographic scan shows a very large osteolytic lesion in the mandible (arrow) with an associated soft-tissue mass extending into the adjacent areas of the cheek. There is also a pathologic fracture.
the cells lining the cyst from a benign epithelium
to a carcinoma, which the current case demonstrates.4 A review of the English literature from 1947 shows 54 reported cases of carcinoma arising in a dentigenous cyst. 5-51 Of these, 19 cases satisfy Gardner’s criteria 5,9,16,21,24,29-32,34,37,38,43-47 These are reviewed, along with the present case, in Table 1. Carcinomatous change in cysts can proceed to an
MAXYMIW AND WOOD
641
FIGURE 3. A, Low-power photomicrograph showing transition of cyst lining epithehum to carcinoma (arrow) (hematoxylin-eosin stain, original magnification x25). B. Highpower photomicrograph showing microinvasion (arrow) in area of transition (hematoxylin-eosin stain, original magnification x63).
inoperable stage without ._ affecting the general condition of the patient. I\1An important clinical sign is rapid, firm, nontender enlargement of the jaw. Periodic symptoms of neuralgic pain and paresthesia may be experienced before the cancer becomes clinically evident. Lymph nodes are not commonly
involved. Extension to the oral cavity can follow, with exposure of tumor and rapid progression of the disease.3’ The tumor may extend perineurally along the mandibular cana1.28 It is often associated with failure of an extraction site to heal and displacement of teeth.”
642
CARCINOMA ARISING IN A DENTIGEROUS
Table 1.
CYST
Reported Cases With Transition of Normal to Malignant Epithelium in Cyst Wall
Site
Primary Treatment
Additional Treatment
Author(s)
Year
Age (yr)
Angelopoulos et al5 Banetjees Chretien et alI6 FrankI& Wiesne?’
1%6 1967 1970 1949
74 37 55 38
M M M F
Mandible Maxilla Mandible Maxilla
Resection Enucleation Enucleation Enucleation
Hardmanz4
1963
73
F
Mandible
Enucleation
Not stated Radiation Resection Resection Radiation Resection
Hardmanz4
1963
77
F
Mandible
Resection
Resection
Lapin et alz9
1973
55
M
Mandible
Enucleation
Resection
Lavery et a13’ Lee & Loke3i
1987 1%7
46 57
M M
Maxilla Maxilla
Enucleation Enucleation
Resection Resection
Macleod & Soames3’ Martensson
1988 1955
57 49
M M
Mandible Maxilla
Resection Enucleation
Not stated Radiation
Martensson Maxymiw & Wood
1955 1990
64 72
M M
Mandible Mandible
Enucleation Enucleation
Resection Radiation
Moos & Rennie3’ Nithianda3*
1987 1983
67 59
F M
Mandible Maxilla
Enucleation Resection
None None
Siar & Ng43 Stokke & Koppang45
1987 1968
40 90
M F
Maxilla Mandible
Enucleation Enucleation
Resection None
Van Der Waal et ale
1985
85
F
Mandible
?
Not stated
Sex
Radiographically odontogenic cysts undergoing malignant transformation may show margins that are jagged and have indentations and indistinct borders. Ultimately, erosion of the buccal, labial, and lingual plates occurs and roots of adjacent teeth are resorbed.38 If enlargement remains unchecked, pathologic fractures occur, as in the present case. Histologic review demonstrates malignant transformation accompanied by a chronic infiltrate of lymphocytes and plasma cells in the cyst wall connective tissue; this was seen in this instance. The cysts are lined in whole or in part by dyskeratotic squamous epithelium. The lining of stratified squamous epithelium shows the actual transition from normal squamous epithelium to invasive squamouscell carcinoma, which proliferates into the connective tissue wall of the cyst. Some regions of the cyst lining may contain intraepithelial carcinoma 11.30.32.37.52 Because the tumor is so rare, there is little accumulation of experience with regard to treatment. Treatment is based on experience with tumors arising elsewhere in the oral cavity. At the time of initial surgery, malignancy is usually not expected and the treatment is usually enucleation or marsupial-
Health Status Not stated Dead of disease Dead of disease Not stated Alive, no evidence of disease Alive, no evidence of disease Alive, no evidence of disease Not stated Alive, no evidence of disease Not stated Alive, no evidence of disease Not stated Dead of other causes Dead of disease Alive, no evidence of disease Not stated Dead of other causes Dead of other causes
Time From Diagnosis to Death Not stated 10 Months 13 Months
6 Months Not stated
4 Months 3 Weeks
ization. This was done in the present case, and the pathology was misinterpreted as normal. Because these tumours originate within bone, osseous erosion and invasion occur early. Following diagnosis, most patients are treated by wide resection of the affected jaw.47 The initial misdiagnosis of the patient’s condition in the present report underlines the importance of careful review of all histologic sections. References 1. Shafer WG, Hine MK, Levy BM: A Textbook of Oral Pathology (ed 4). Philadelphia, PA, Saunders, 1983, p 258 2. Worth HM: Principles and practices of Oral Radiographic interpretation (ed 2). Chicago, IL, Year Book Medical, 1972, p 420 3. Regezi JA, Sciubba JJ: Oral Pathology: Clinical Pathologic Conference. Philadelphia, PA, Saunders, 1989, p 301 4. Gardner AF: A survey of odontogenic cysts and their relationship to squamous cell carcinoma. J Can Dent Assoc 42: 161, 1975 5. Angelopoulos AP, Tilson HB, Stewart FW, et al: Malignant transformation of the epithelial lining of the odontogenic cysts. Oral Surg Oral Med Oral Path01 4:415. 1966 6. Arekn RG, McCaichey KD, Baker HL: Squamous cell carcinoma developing in an odontogenic keratocyst. Arch Otolaryngol 107:568, 1981
643
MAXYMIW AND WOOD
7. Baker RD, Donofrio ED, Corio RL, et al: Squamous cell carcinoma arising in a lateral periodontal cyst. Oral Surg Oral Med Oral Path01 47:495, 1979 8. Banejee SC: Squamous cell carcinoma in a maxillary cyst. Oral Surg Oral Med Oral Path01 23: 193, 1967 9. Berenholz L, Gottlieb RD, Cho SY, et al: Squamous cell carcinoma arising in a dentigerous cyst. Ear Nose Throat J 67:764, 1988 10. Bradfield WJD, Broadway ES: Malignant change in a dentigerous cyst. Br J Surg 45:657, 1958 I I. Bradley N, Thomas DM, Antoniades K. et al: Squamous cell carcinoma arising in an odontogenic cyst. Int J Oral Maxillofac Surg 17:260, 1988 12. Browne RM, Cough NC: Malignant change in the epithelium lining odontoeenic cvsts. Cancer 29: 1199. 1972 13. Brown: RM: Metaplasia and degeneration in odontogenic cysts in man. J Oral Pathol 1:145. 1972 14. Chongruk C: Cyst and carcinoma. Oral Surg Oral Med Oral Pathol 51:219, 1981 15. Choukas NC: Controversial lesion of the mandible: Report of case. J Oral Surg 29579. 1971 16. Chretien PB, Carpenter DF, White NS, et al: Squamous carcinoma arising in a dentigerous cyst. Oral Surg Oral Med Oral Pathol 30:809. 1970 17. Darlington CG, Ehrlich HE, Seldin HM: Malignant transformation of odontogenic cyst: Report of case. J Oral Surg 1164, 1953 18. Enriquez RE, Ciola B, Bahn SL: Verrucous carcinoma arising in an odontogenic cyst. Oral Surg Oral Med Oral Pathol 49: 151, 1980 19. Falkmer S. Herberts G, Olven S: Carcinoma arising in odontogenic cysts of the jaw. Odontol Tidskr 65:220,1957 20. Frank1 Z, Wiesner J: Cancer starting from a cyst. Dent Items Int 71564, 1949 2 1. Frank1 Z, Bouyssou M: Premalignant and malignant changes in the lining of odontogenic cysts and its therapeutic consequences. Dent Sci Res 9:93, 1976 22. Hampl PF, Harrigan WF: Squamous cell carcinoma possibly arising from a odontogenic cyst: Report of case. J Oral Surg 31:359, 1973 23. Hankey GT. Pedler JA: Primary squamous cell carcinoma of mandible arising from epithelial lining of dental cyst. Proc R Sot Med 50:680, 1957 24. Hardman FG: Carcinomata arising in dental cysts. Br J Oral Surg 1:124, 1963 25. High AS. Quirke P, Hume WJ: DNA-ploidy studies in a keratocyst undergoing subsequent malignant transformation. J Oral Path01 16:135. 1987 26. Meadow SR: Malignant change in a dental cyst. Oral Surg Oral Med Oral Pathol 21:282. 1%6 27. Kay LW, Kramer RH: Squamous-cell carcinoma arising in a dental cyst. Oral Surg Oral Med Oral Pathol 15:970, 1962 28. Kramer HS, Scribner JH: Squamous-cell carcinoma arising in a dentigerous cyst. Oral Surg Oral Med Oral Path01 19:555, 1965 29. Lapin R, Garfinkel AV, Catania AF, et al: Squamous cell carcinoma arising in a dentigerous cyst. J Oral Surg 31:354. 1973
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