Ann Dtol Rhinol LaryngollOO:1991
PATHOLOGY CONSULTATION
SALIVARY NEOPLASMS OF THE JAW BONES WITH PARTICULAR REFERENCE TO CENTRAL MUCOEPIDERMOID CARCINOMAS JANET
M. BRUNER, MD
JOHN
G. BATSAKIS, MD
HOUSTON, TEXAS
Intragnathic salivary-type neoplasms are few in number, are of debated histogenesis, and are most often classified as mucoepidermoid carcinomas. Two generating tissues have been proposed for the origin of these central (intraosseous) tumors: enclaves of salivary tissue and a metaplasia of the linings of odontogenic cysts. They are not mutually exclusive. Although most mucoepidermoid carcinomas have been low grade, these neoplasms nonetheless have ~ very respectable recurrence rate and can be fatal.
Primary nonodontogenic epithelial neoplasms of the gnathic bones are rare lesions and should always be approached with considerable diagnostic circumspection. When metastases to the jaw bones and direct extension from mucosal or cutaneous primaries are excluded, there remain central (intraosseous) tumors of presumed salivary origin. Mucoepidermoid carcinomas are the class of tumor most often reported. Much fewer in number are salivary adenomas (pleomorphic and monomorphic adenomas) and adenoid cystic carcinomas.
mandibular and sublingual glands closely apposed in bony defects or cavities in the lingual cortex of the mandible, or 3) from parts of these latter glands that have been embryologically evaginated. A third speculative source has also been proposed for the beginnings of central mucoepidermoid carcinomas: metaplasia in an odontogenic cyst or epithelial rests of Malassez.v" The commonly accepted criteria for the diagnosis of a central mucoepidermoid carcinoma are also applicable to other histologic types of salivary gland tumors of the jaw bones. They include 1) intact cortical plates, 2) radiographic evidence of bone destruction, 3) exclusion of another primary that in its metastasis could histologically mimic the central tumor, 4) exclusion of an odontogenic tumor, and 5) histopathologic confirmation. A detectable intracellular mucin is often added as confirming evidence of a mucoepidermoid carcinoma. In the appropriate context, this finding is helpful, but it is
Presumed origin from a maternal salivary gland tissue is predicated on the presence of the tissue within the jaw bones. Evidence in support for centrally placed salivary tissue is strongest for the mandible. Salivary gland inclusions mostly occur in bone spaces located near the angle of the mandible, where they have a rather consistent position and radiographic appearance. 1 Only a few inclusions have been found in the anterior mandible (in theIncisor region or between the canines and first molars). 1 No single theory of histogenesis is applicable to all cases.v" True examples of central pleomorphic or monomorphic adenoma and adenoid cystic carcinoma do not arise from a metaplasia of odontogenic cysts and must therefore take origin 1) from enclaved retromolar mucous glands during embryologic development of the jaw bones, 2) from subCENTRAL (INTRAOSSEOUS) MUCOEPIDERMOID CARCINOMA (N = 66) Gnathic bone involved Age of patients Sex of patients
Recurrences Metastases
48 in mandible, 18 in maxilla 1 to 85 y (mean, 51 y) 36 female, 28 male, 2 not indicated in report Varied and nondiagnostic; unilocular or multilocular radiolucencies 17/66 (25.80/0) 8/66 (12.1 %)
Death due to neoplasm
6/66 (9.1 %)
Radiographic findings
Fig 1. Low-grade mucoepidermoid carcinoma of mandible with histologic appearance like that seen in mucoepidermoid carcinomas of major and minor salivary glands (H & E, original x140).
From data presented by Waldron and Koh."
From the University of Texas M. D. Anderson Cancer Center, Houston, Texas. REPRINTS - John G. Batsakis, MD, Dept of Pathology, University of Texas M. D. Anderson Cancer Center, 1515 Holcombe BIvd, Houston, TX 77030.
954
Downloaded from aor.sagepub.com at MCMASTER UNIV LIBRARY on July 25, 2015
Bruner & Batsakis, Pathology Consultation
955
Fig 2. Intraosseous, low-grade mucoepidermoid carcinoma of mandible (H & E). It is this appearance that suggests origin from odontogenic cyst rather than from salivary tissue and raises differential diagnosis of sialodontogenic cyst. A) Original x40. B) Original x200.
not diagnostic. Mucinous metaplasia is known to occur in odontogenic cysts, and intracellular mucin is not restricted to only mucoepidermoid neoplasms. Intact cortical plates of the involved bone are certainly strong evidence for an intraosseous origin of a salivary-type neoplasm, but may not be a completely reasonable requirement, especially for tumors of the thinner maxilla as opposed to the mandible. Waldron and Koh" accept cortical plate defects in a mandible overlying the radiolucent lesion if it is unassociated with an obvious soft tissue tumor and if the periosteum is intact. Only central mucoepidermoid carcinomas have sufficient reported numbers to allow clinicopathologic analysis." Other salivary neoplasms are presented in single case reports or small series with insufficient documentation.t" Waldron and Koh" indicate that at least 66 cases of central mucoepidermoid carcinoma of the jaws have been reported (see Table"), with the mandible involved almost three times as often as the maxilla. The great majority of the carcinomas have been low grade (Fig 1) and preponderantly cystic." Treatment has ranged from enucleation or curettage to en bloc or radical excision. Several patients
have also received radiotherapy. 4 Enucleation or curettage, with or without electrocoagulation, is associated with the highest recurrence rate (45%), but no method of treatment is completely free of that event. As recorded in the review by Waldron and Koh, metastases and death were highest in patients undergoing hemimaxillectomy or hemimandibulectomy." This finding likely reflects an advanced stage of disease at the time of initial treatment. The principal differential diagnoses for gnathic mucoepidermoid carcinomas are metastases from other primary carcinomas and cystic odontogenic lesions, particularly the sialodontogenic cyst (glandular odontogenic cyst, mucoepidermoid cyst).4.7.8 These lesions enter the differential diagnosis in instances in which the carcinoma manifests sufficient histologic differences from the prototypes of extraosseous mucoepidermoid carcinomas (Fig 2). The sialodontogenic cyst is relatively unknown to pathologists, and its features overlap with those of some low-grade mucoepidermoid carcinomas.v':" For that reason, Waldron and Koh" wonder if the sialodontogenic cyst is not just part of a spectrum of central mucoepidermoid carcinoma.
REFERENCES 1. Anneroth G, Berglund G, Kahnberg K-E. Intraosseous salivary gland tissue of the mandible mimicking a periapical lesion. Int J Oral Maxillofac Surg 1990'19,74-5 ,. · 2. Gingell JC, Beckerman T, Levy BA, Snider LA. Central mucoepidermoid carcinoma. Review of the literature and report of a case associated with an apical periodontal cyst. Oral Surg 1984;57:436-40.
1990;48:871-7. . . . 5. Drinkard DW, Schow CEo Benign mixed tumor of the mandible 17 years after the occurrence of a similar lesion in the parotid gland. Oral Surg Oral Med Oral Path 01 1986;62:381-4. 6. Johnson PA, Millar BG, Leopard PJ. Intraosseous adenoid cystic carcinoma of the mandible. Br J Oral Maxillofac Surg 1989; 27:501-5.
3. Browand BC, Waldron CA. Central mucoepidermoid tumors of the jaws. Report of nine cases and review of the literature. Oral Surg 1975;40:631-43.
7. Gardner DC, Kessler HP, Morency R, Schaffner DL. The glandular odontogenic cyst: an apparent entity. J Oral Pathol 1988;17:359-66.
4. Waldron CA, Koh ML. Central mucoepidermoid carcino-. rna of the jaws: report of four cases with analysis of the literature and discussion of the relationship to mucoepidermoid, sialodontogenic, and glandular odontogenic cysts. J Oral Maxillofac Surg
8. Padayachee A, van Wyk CWo Two cystic lesions with features of both botryoid odontogenic cyst and the central mucoepidermoid tumor: sialo-odontogenic cyst? J Oral Pathol 1987;16: 499-502.
Downloaded from aor.sagepub.com at MCMASTER UNIV LIBRARY on July 25, 2015
PATHOLOGY CONSULTATION
SALIVARY NEOPLASMS OF THE JAW BONES WITH PARTICULAR REFERENCE TO CENTRAL MUCOEPIDERMOID CARCINOMAS JANET
M. BRUNER, MD
JOHN
G. BATSAKIS, MD
HOUSTON, TEXAS
Intragnathic salivary-type neoplasms are few in number, are of debated histogenesis, and are most often classified as mucoepidermoid carcinomas. Two generating tissues have been proposed for the origin of these central (intraosseous) tumors: enclaves of salivary tissue and a metaplasia of the linings of odontogenic cysts. They are not mutually exclusive. Although most mucoepidermoid carcinomas have been low grade, these neoplasms nonetheless have ~ very respectable recurrence rate and can be fatal.
Primary nonodontogenic epithelial neoplasms of the gnathic bones are rare lesions and should always be approached with considerable diagnostic circumspection. When metastases to the jaw bones and direct extension from mucosal or cutaneous primaries are excluded, there remain central (intraosseous) tumors of presumed salivary origin. Mucoepidermoid carcinomas are the class of tumor most often reported. Much fewer in number are salivary adenomas (pleomorphic and monomorphic adenomas) and adenoid cystic carcinomas.
mandibular and sublingual glands closely apposed in bony defects or cavities in the lingual cortex of the mandible, or 3) from parts of these latter glands that have been embryologically evaginated. A third speculative source has also been proposed for the beginnings of central mucoepidermoid carcinomas: metaplasia in an odontogenic cyst or epithelial rests of Malassez.v" The commonly accepted criteria for the diagnosis of a central mucoepidermoid carcinoma are also applicable to other histologic types of salivary gland tumors of the jaw bones. They include 1) intact cortical plates, 2) radiographic evidence of bone destruction, 3) exclusion of another primary that in its metastasis could histologically mimic the central tumor, 4) exclusion of an odontogenic tumor, and 5) histopathologic confirmation. A detectable intracellular mucin is often added as confirming evidence of a mucoepidermoid carcinoma. In the appropriate context, this finding is helpful, but it is
Presumed origin from a maternal salivary gland tissue is predicated on the presence of the tissue within the jaw bones. Evidence in support for centrally placed salivary tissue is strongest for the mandible. Salivary gland inclusions mostly occur in bone spaces located near the angle of the mandible, where they have a rather consistent position and radiographic appearance. 1 Only a few inclusions have been found in the anterior mandible (in theIncisor region or between the canines and first molars). 1 No single theory of histogenesis is applicable to all cases.v" True examples of central pleomorphic or monomorphic adenoma and adenoid cystic carcinoma do not arise from a metaplasia of odontogenic cysts and must therefore take origin 1) from enclaved retromolar mucous glands during embryologic development of the jaw bones, 2) from subCENTRAL (INTRAOSSEOUS) MUCOEPIDERMOID CARCINOMA (N = 66) Gnathic bone involved Age of patients Sex of patients
Recurrences Metastases
48 in mandible, 18 in maxilla 1 to 85 y (mean, 51 y) 36 female, 28 male, 2 not indicated in report Varied and nondiagnostic; unilocular or multilocular radiolucencies 17/66 (25.80/0) 8/66 (12.1 %)
Death due to neoplasm
6/66 (9.1 %)
Radiographic findings
Fig 1. Low-grade mucoepidermoid carcinoma of mandible with histologic appearance like that seen in mucoepidermoid carcinomas of major and minor salivary glands (H & E, original x140).
From data presented by Waldron and Koh."
From the University of Texas M. D. Anderson Cancer Center, Houston, Texas. REPRINTS - John G. Batsakis, MD, Dept of Pathology, University of Texas M. D. Anderson Cancer Center, 1515 Holcombe BIvd, Houston, TX 77030.
954
Downloaded from aor.sagepub.com at MCMASTER UNIV LIBRARY on July 25, 2015
Bruner & Batsakis, Pathology Consultation
955
Fig 2. Intraosseous, low-grade mucoepidermoid carcinoma of mandible (H & E). It is this appearance that suggests origin from odontogenic cyst rather than from salivary tissue and raises differential diagnosis of sialodontogenic cyst. A) Original x40. B) Original x200.
not diagnostic. Mucinous metaplasia is known to occur in odontogenic cysts, and intracellular mucin is not restricted to only mucoepidermoid neoplasms. Intact cortical plates of the involved bone are certainly strong evidence for an intraosseous origin of a salivary-type neoplasm, but may not be a completely reasonable requirement, especially for tumors of the thinner maxilla as opposed to the mandible. Waldron and Koh" accept cortical plate defects in a mandible overlying the radiolucent lesion if it is unassociated with an obvious soft tissue tumor and if the periosteum is intact. Only central mucoepidermoid carcinomas have sufficient reported numbers to allow clinicopathologic analysis." Other salivary neoplasms are presented in single case reports or small series with insufficient documentation.t" Waldron and Koh" indicate that at least 66 cases of central mucoepidermoid carcinoma of the jaws have been reported (see Table"), with the mandible involved almost three times as often as the maxilla. The great majority of the carcinomas have been low grade (Fig 1) and preponderantly cystic." Treatment has ranged from enucleation or curettage to en bloc or radical excision. Several patients
have also received radiotherapy. 4 Enucleation or curettage, with or without electrocoagulation, is associated with the highest recurrence rate (45%), but no method of treatment is completely free of that event. As recorded in the review by Waldron and Koh, metastases and death were highest in patients undergoing hemimaxillectomy or hemimandibulectomy." This finding likely reflects an advanced stage of disease at the time of initial treatment. The principal differential diagnoses for gnathic mucoepidermoid carcinomas are metastases from other primary carcinomas and cystic odontogenic lesions, particularly the sialodontogenic cyst (glandular odontogenic cyst, mucoepidermoid cyst).4.7.8 These lesions enter the differential diagnosis in instances in which the carcinoma manifests sufficient histologic differences from the prototypes of extraosseous mucoepidermoid carcinomas (Fig 2). The sialodontogenic cyst is relatively unknown to pathologists, and its features overlap with those of some low-grade mucoepidermoid carcinomas.v':" For that reason, Waldron and Koh" wonder if the sialodontogenic cyst is not just part of a spectrum of central mucoepidermoid carcinoma.
REFERENCES 1. Anneroth G, Berglund G, Kahnberg K-E. Intraosseous salivary gland tissue of the mandible mimicking a periapical lesion. Int J Oral Maxillofac Surg 1990'19,74-5 ,. · 2. Gingell JC, Beckerman T, Levy BA, Snider LA. Central mucoepidermoid carcinoma. Review of the literature and report of a case associated with an apical periodontal cyst. Oral Surg 1984;57:436-40.
1990;48:871-7. . . . 5. Drinkard DW, Schow CEo Benign mixed tumor of the mandible 17 years after the occurrence of a similar lesion in the parotid gland. Oral Surg Oral Med Oral Path 01 1986;62:381-4. 6. Johnson PA, Millar BG, Leopard PJ. Intraosseous adenoid cystic carcinoma of the mandible. Br J Oral Maxillofac Surg 1989; 27:501-5.
3. Browand BC, Waldron CA. Central mucoepidermoid tumors of the jaws. Report of nine cases and review of the literature. Oral Surg 1975;40:631-43.
7. Gardner DC, Kessler HP, Morency R, Schaffner DL. The glandular odontogenic cyst: an apparent entity. J Oral Pathol 1988;17:359-66.
4. Waldron CA, Koh ML. Central mucoepidermoid carcino-. rna of the jaws: report of four cases with analysis of the literature and discussion of the relationship to mucoepidermoid, sialodontogenic, and glandular odontogenic cysts. J Oral Maxillofac Surg
8. Padayachee A, van Wyk CWo Two cystic lesions with features of both botryoid odontogenic cyst and the central mucoepidermoid tumor: sialo-odontogenic cyst? J Oral Pathol 1987;16: 499-502.
Downloaded from aor.sagepub.com at MCMASTER UNIV LIBRARY on July 25, 2015
