518
CENTRAL MUCOEPIDERMOID CARCINOMA
J Oral Maxillofac 48:518-522,
Surg
1990
Central Mucoepidermoid Carcinoma the Jaw With Distant Metastasis:
of
A Case Report and Review of the Literature JON P. LEBSACK,
DDS,* A.J. MARROGI,
Criteria for the diagnosis of mucoepidermoid carcinoma of the jaw bones are well established,’ and more than 60 cases have been reported in the world literature. Although the tumor is considered to be a carcinoma of low malignant potential, several instances of metastasis to the regional lymph nodes have been reported.’ We report a patient with central mucoepidermoid carcinoma of the mandible with distant metastasis but without regional lymph node metastasis. Report of a Case A 7%year-old woman was referred to an oral surgeon in February 1988 for the evaluation and treatment of a painful swelling of the right posterior mandible that had been present for approximately 3 years. At the time of referral, the lesion appeared as a multicystic radiolucency with a large area measuring 2 x 3 cm and adjacent smaller radiolucencies (Fig 1). On physical examination, the lesion could be palpated at the apices of the premolar teeth, and it extended to a point 1.5 cm distally. The patient reported no other symptoms; there was no history of lip parasthesia. The initial clinical and radiographic impressions included metastatic carcinoma and ameloblastoma. The medical history included aortic stenosis, hypertension, and mitral valve prolapse. There was no previous history of tobacco use. The evaluation included a chest radiograph,
mammograms,
and
abdominal
MD,t AND SCOTT A. MARTIN, MD*
computed tomography scans, all of which failed to reveal a primary tumor. The patient underwent primary excision of the mandibular tumor in June 1988. At surgery, after elevation of a
mucoperiosteal flap over the crest of the alveolar ridge, the lesion was found to have perforated the buccal cortical plate, but the periosteum had remained intact and there was no evidence of communication between the tumor and the oral mucosa. To facilitate enucleation of the tumor, the mandibular premolar teeth were removed. The inferior alveolar neurovascular bundle was closely associated with the lesion in the region of the mental foramen and was removed with the tumor. Hemostasis was achieved and the flap was closed with 000 chromic suture in a mattress fashion. The postoperative recovery was uneventful. Routine hematoxylin-eosin-stained sections of formalin-fixed, paraffin-embedded tissue showed an intraosseous carcinoma in which the cells were arranged in loosely cohesive sheets (Fig 2A), and focally displayed a
and pelvic
Received from St John’s Mercy Medical Center, St Louis, MO. * Resident, Department of Oral and Maxillofacial Surgery. t Fellow, Department of Pathology/Section of Surgical Pathology. $ Director of Surgical Pathology, Department of Pathology. Address correspondence and reprint requests to Dr Marrogi: Department of Pathology, St John’s Mercy Medical Center, 615 S New Ballas Rd, St Louis, MO 63141-8221. 0 1990 American Association of Oral and Maxillofacial geons 0278-2391190148050017$3.00/O
Sur-
FIGURE 1. Central mucoepidermoid carcinoma of the right mandible. The tumor appears as a radiolucent, multilocular lesion (arrows).
LEBSACK, MARROGI, AND MARTIN
FIGURE 2. Central mucoepidermoid carcinoma. A, Loosely cohesive sheets of intraosseous tumor. (Hematoxylin-eosin
stain. Orig-
inal magnification.
x90.)
x45.) B, Areas of tumor cells with clear cytoplasm. (Hematoxylin-eosin
rudimentary abortive acinar arrangement. The architectural features were variable throughout the tumor. Some cellular nests were surrounded by a hemorrhagic vascular stroma, and in these areas the tumor was reminiscent of paraganglioma. In other sections the malignant cells inliltrated the bone marrow spaces and were separated by widened bony trabeculae. Some tumor cells had a conspicuous. clear cytoplasm (Fig 2B). The immunocytochemical stain for carcinoembryonic antigen (Dako, Santa Barbara, CA) demonstrated focal intracytoplasmic staining in these cells. Histochemically, the tumor displayed intracytoplasmic PAS-positive diastase-resistant material. In areas of acinar arrangement there was extracellular mucicarminophilic material in the abortive lumina. The histologic diagnosis was central mucopidermoid carcinoma of the mandible. Approximately 2 months after the surgery, the patient began to experience dull pain in the right clavicle. A bone scan showed increased activity in that location (Fig 3). The patient subsequently underwent a biopsy from the area. The histologic features of the clavicular lesion were similar to those of the mandibular tumor (Fig 4). Following further evaluation which demonstrated no other metastatic lesions, the patient received radiation therapy after the extraction of the remaining mandibular teeth. She received a total dose of 6,000 rads to the mandible and 5,000 rads to the clavicle. Radiographic examination of both the mandible and the clavicle in November 1988 showed no evidence of recurrence, and the patient had no symptoms or complaints.
stain. Original magnification,
Discussion The criteria as proposed by Alexander et al’ and modified by Browand and Waldron3 for the diagno-
FIGURE 3. Tc99 bone scan showing lesion of the right mandible and metastasis to the right clavicle.
520
CENTRAL MUCOEPIDERMOID
CARCINOMA
FIGURE 4. Metastatic central mucoepidermoid carcinoma to the clavicle. The histoiogic appearance is similar to that of the primary tumor (Fig 2A). (Hematoxylin-eosin stain. Original magnitication, x 135.)
sis of central mucoepidermal carcinoma of the jaw bones include 1) the presence of an intact cortical bone plate and/or periosteum, 2) radiographic evidence of bone destruction, 3) histologic features of mucoepidermoid carcinoma and evidence of mucin production, 4) absence of a primary tumor elsewhere with potential histologic similarities to central mucoepidermoid carcinoma of the jaw, and 5) absence of a primary odontogenic cyst. The world literature contains reports on 63 cases of central mucoepidermoid carcinoma of the jaw Table 1. Case Source
Central Mucoepidermoid Carcinoma of the Jaws With Metastasis Agel Sex
Site
Hertz”
49/F
Mandible
Chaudhry et al4
16/F
Maxilla
Brown et al’
50/M
Mandible
Weinstein et al6
bones. l-3’ Of those patients, only 7 (9%) have been reported to develop metastasis, mainly to the regional lymph nodes (Table 1). One patient reported in the series of Dhawan et al* had metastatic tumor in the lung in the absence of nodal metastasis. This particular case may not represent an example of central mucoepidermoid carcinoma because this patient had tumors arising simultaneously both in the mandible and the adjacent parotid salivary gland. Of the 7 patients with metastatic tumor in the lymph nodes, 4 had lymph node involvement at the
50/F
Maxilla
Symptoms, Duration
Radiographic Appearance
Initial Treatment
Neuralgic pain and swelling Toothache and swelling, a few weeks Enlarged mass, nonpainful, 20 yr
Bone destruction
Res/Irr
Impacted L 3rd molar tooth
Infraorbital swelling, unknown duration
Time and Site of Metastasis
Follow-Up 20 yr, no tumor
Partial max/ LND
1 yr/ cervical node Simultaneous cervical node
Multiloculated cystic lesion
Cur
14 yrl
NA
Diffuse radiolucency
Hmax/LND
(Continued on hollowing page)
submaxillary, & jugular nodes Simultaneous/ jugular nodes
NA
34 mo, no tumor
521
LEBSACK, MARROGI, AND MARTIN
Table 1.
Central Mucoepidermoid Carcinoma of the Jaws With Metastasis (Cont’d)
Case Source
Agel Sex
Site
Smith et al’
49/F
Mandible
Dhawan et al”
5QlF
Mandible
Ganz et al9
18/M
Mandible
Radiographic Appearance
Initial Treatment
Intermittant swelling, adenopathy, several years Swelling and pain, 5 mos’ duration
Cystic lesion (ameloblastoma)
Enucleatiom LND
Simultaneous/ submaxillary nodes
32 yr, no tumor
NA
Mand
1 yr
4 yr, died with tumor
Swelling, nontender,
Multiloculated radiolucency Multicystic radiolucency
Partial Mandl LND
ipslateral 3 yr contr/ cervical nodes Simultaneous/ cervical nodes
1 yr Lebsack et al (this report)
?SiF
Mandible
Time and Site of Metastasis
Symptoms, Duration
duration Swelling, pain, 3 yrs’
Cur/IlT
Abbreviations: Hmax, hemimaxillectomy; Max, maxillectomy; Mand, mandibulectomy; available; Cur. curretage; Irr, irradiation; Res, resection; contr, contralateral.
time of resection of the primary tumor. The remaining 3 patients developed regional lymph node metastasis from 1 to 24 years after the time of initial management. The patient reported herein is another example of central mucoepidermoid carcinoma of the jaw bone. Even though the cortical bone plate was found to be perforated, there was no attachment of the tumor mass to the epithelial lining of the oral mucosa, and there was no involvement of the periosteum at the time of the surgical procedure. The patient is without recurrence 19 months after the treatment of her primary mandibular tumor. The reported case is unique in that the patient developed an osseous metastasis in the absence of regional lymph node involvement. Most patients with central mucoepidermoid carcinoma do not suffer from metastatic disease. Acknowledgment The authors thank W.E. Bisch, DDS, oral surgeon, for the clinical information provided.
References 1. Alexander RW. Dupuis RH, Holtn H: Central mucoepidermoid tumor (carcinoma) of the mandible. J Oral Surg 32:541. 1974 2. Eversole LR, Sabes WR, Sheldon R: Aggressive growth and neoplastic potential of odontogenic cysts. Cancer 35:270, 1975
2 ma/R clavicle
Follow-Up
30 mo, no tumor
9 mo, no tumor
LND, lymph node dissection;
NA, not
3. Browand BC, Waldron CA: Central mucoepidermoid tumors of the jaws. Oral Surg 40:63 1, 1975 4. Chaudhry AP, Dedolph T, Vickers RA: Mucoepidermoid tumor arising from ectopic salivary gland in the maxilla. J Oral Surg Anesth Hosp Dent Serv 19:521, 1961 5. Brown AM, Lucchesi FJ: Central mucoepidermoid tumor of the mandible-Report of case. J Oral Surg 24:356, 1966 6. Weinstein IR, Nagai I, Yamanaka H: Mucoepidermoid tumor of the maxilla. Report of a case. Oral Surg Oral Med Oral Path01 23: 1, 1967 7. Smith RL, Dahlin DC, Waite DE: Mucoepidermoid carcinoma of the jawbones. J Oral Surg 16:387, 1968 8. Dhawan IK, Bhargava S, Nayak NC, et al: Central salivary gland tumors of jaws. Cancer 26:211, 1970 9. Ganz SD, Toth BB: Central mucoenidermoid carcinoma simulating an odontogenic tumor. J bra1 Med 40: 179, 1985 10. Gingell JC, Beckerman T, Levy BA, et al: Central mucoepidermoid carcinoma. Oral Surg 5:436, 1984 1I. Grubka JM, Wesley RK, Monaco F: Primary intraosseous mucoepidermoid carcinoma of the anterior part of the mandible. J Oral Maxillofac Surg 41:389, 1983 12. Bhaskar SN, Bemier JL: Mucoepidermoid tumors of major and minor salivary glands. Clinical features, histology, variations, natural history, and results of treatment for 144 cases. Cancer 15:801, 1962 13. Tamai C: Mucoepidermoid tumor of the mandible. Am J Surg 92~872, 1956 14. Stoll HC, Marchetta FC, Schobinger R: Malignant epithelial tumors of the mandible and maxilla. Arch Path01 64:239, 1957 15. Bhaskar SN: Central mucoepidermoid tumors of the mandible: Report of 2 cases. Cancer 16:721, 1963 16. Auriol M, Chomette G, Delcourt A, et al: Tumeur mucoepidermoide mandibulaire. Rev Stomatol Chir Maxillofac 86:414, 1985 17. Ito H, Soda T, Nakajima T, et al: Central mucoepidermoid tumor of the mandible: Report of case. Bull Tokyo Med Dent Univ 16:251, 1969 18. Silverglade LB, Alvares OF, Olech E: Central mucoepidermoid tumors of the jaws. Cancer 22:650, 1%X
522
PERIOSTEAL
19. Schultz W, Whitten JB: Mucoepidermoid carcinoma in the mandible: Report of a case. J Oral Surg 27:337, 1969 20. Frederickson C, Cherrick HM: Central mucoepidermoid carcinoma of the jaws. J Oral Med 33:80, 1978 21. Unal T, Zeytinoglu B: Central mucoepidermoid carcinoma of the jaws. J Oral Med 40:202, 1985 22. Pincock JL, El-Mofty SK: Recurrence of cystic central mucoepidermoid tumor of the mandible. Int J Oral Surg 14:81, 1985 23. Stoch RB, Smith I: Mucoepidermoid carcinoma of the mandible: Report of case. J Oral Surg 3856, 1980 24. Lepp H, cited in Hertz J: Mucus secreting tumors of the jaws. Acta Chir Stand 103:276, 1952 25. Hertz J: Mucus secreting tumors of the jaws. Acta Chir Stand 103:276, 1952
J Oral Maxillofac 46:522-526,
OSTEOSARCOMA
OF THE MAXILLA
26. Sonesson A: Mucus-secreting and cystic epidermoid carcinoma of the jaws. Acta Radio1 34:25, 1950 27. Szerlip L: Cystic mucoepidermoid tumor. Report of case. Oral Sure 9:584. 1956 28. Rzucidlo i?B, Rzeszotko-Adamiczka D: Uber einen mucoepidermoid tumors eines Unterkiefers. Zentralbl Allg Pathol 105:409, 1964 29. Attenson M, Kaufman M, Brown S: Mucoepidermoid carcinoma of the mandible: Report of a case. J Oral Surg 22:350, 1964 30. Lacour M, Reynaud J, Grappin G, et al: A case of mucoepidermoid tumors of the mandible. Bull Sot Med Afr Lang Fr 11:73, 1966 31. Karmarkar SR: Central mucoepidermoid tumors of the mandible. Indian J Surg 31: 154, 1969
Surg
1990
Perios teal Osteosarcoma
of the Maxilla:
A Case Report and Review of Literature LTC ADRIAN PATTERSON, DDS, DC, USA,* ROBERT 0. GREER JR, DDS, ScD,t AND DANIEL HOWARD, DDSS
Juxtacortical osteosarcoma was first fully described by Jaffee and Selin’ in 1951, although Geschickter and Copeland had described a similar lesion under the heading “parosteal osteoma” in 1949.*s3 Since these initial reports, juxtacortical osteosarcoma has been described using various names, including ossifying parosteal osteogenic sarcoma, and parosteal and periosteal ossifying fibrosarcoma.4T5 Juxtacortical osteogenic sarcoma occurs much less commonly than central medullary osteosarcoma, which has an incidence rate of 0.07 per 1,000 population. The clinicopathology of fewer than a dozen cases of juxtacortical osteosarcoma has been
* Chief, Surgery Branch, United States Institute of Dental Research, Department of the Army, Walter Reed Army Medical Center, Washington, DC. t Professor and Chairman, Division of Oral Pathology, University of Colorado School of Dentistry; Professor of Pathology, University of Colorado School of Medicine, Denver. $ In private practice, Washington, DC. The opinions or assertations contained herein are the private views of the authors and are not to be construed as official or as reflecting the view of the Department of the Army. Address correspondence and reprint requests to LTC Patterson: Surgery Branch, US Institute of Dental Research, Department of the Army, Walter Reed Army Medical Center, Washington, DC 20307-5300. 0 1990 American
Association
geons 0278-2391/90/4805-0018$3.00/O
of Oral
and Maxillofacial
Sur-
well documented in the English literature (Table l)? Recently, Zarbo et al’* suggested that juxtacortical osteosarcoma can be further subdivided into two clinicopathologic variants: parosteal osteosarcoma and periosteal osteosarcoma. The purpose of this article is to report a rare example of maxillary juxtacortical osteosarcoma of the periosteal type and to discuss the clinicopathologic nature of that disease process. Report of a Case A 65year-old black woman was referred by her general dentist for evaluation and treatment of a mass in the right maxillary vestibule and alveolus. The lesion had been present and slowly enlarging for approximately 1 year. It had been asymptomatic until about 3 weeks before her visit, when the patient began to have mild pain in the maxillary right quadrant. Physical examination revealed a 2.1 x 2.5 x 2 cm hard mass in the right maxillary vestibule (Fig 1). The lesion was lobular in appearance. The gingiva overlying the lesion showed increased vascularity, but no ulceration. The buccal vestibule was nearly obliterated by the lesion in the region of the premolar and canine teeth. There was neither paresthesia or anesthesia of the infraorbital nerve. A panoramic radiograph showed a slightly radiopaque shadow approximately 0.5 x 0.5 cm distal to the premolar (Fig 2), as well as a longitudinal fracture of this tooth. There was also a loss of lamina dura around the involved teeth. Neck examination was negative for tenderness and adenopathy. An excisional biopsy was performed under local anes-
CENTRAL MUCOEPIDERMOID CARCINOMA
J Oral Maxillofac 48:518-522,
Surg
1990
Central Mucoepidermoid Carcinoma the Jaw With Distant Metastasis:
of
A Case Report and Review of the Literature JON P. LEBSACK,
DDS,* A.J. MARROGI,
Criteria for the diagnosis of mucoepidermoid carcinoma of the jaw bones are well established,’ and more than 60 cases have been reported in the world literature. Although the tumor is considered to be a carcinoma of low malignant potential, several instances of metastasis to the regional lymph nodes have been reported.’ We report a patient with central mucoepidermoid carcinoma of the mandible with distant metastasis but without regional lymph node metastasis. Report of a Case A 7%year-old woman was referred to an oral surgeon in February 1988 for the evaluation and treatment of a painful swelling of the right posterior mandible that had been present for approximately 3 years. At the time of referral, the lesion appeared as a multicystic radiolucency with a large area measuring 2 x 3 cm and adjacent smaller radiolucencies (Fig 1). On physical examination, the lesion could be palpated at the apices of the premolar teeth, and it extended to a point 1.5 cm distally. The patient reported no other symptoms; there was no history of lip parasthesia. The initial clinical and radiographic impressions included metastatic carcinoma and ameloblastoma. The medical history included aortic stenosis, hypertension, and mitral valve prolapse. There was no previous history of tobacco use. The evaluation included a chest radiograph,
mammograms,
and
abdominal
MD,t AND SCOTT A. MARTIN, MD*
computed tomography scans, all of which failed to reveal a primary tumor. The patient underwent primary excision of the mandibular tumor in June 1988. At surgery, after elevation of a
mucoperiosteal flap over the crest of the alveolar ridge, the lesion was found to have perforated the buccal cortical plate, but the periosteum had remained intact and there was no evidence of communication between the tumor and the oral mucosa. To facilitate enucleation of the tumor, the mandibular premolar teeth were removed. The inferior alveolar neurovascular bundle was closely associated with the lesion in the region of the mental foramen and was removed with the tumor. Hemostasis was achieved and the flap was closed with 000 chromic suture in a mattress fashion. The postoperative recovery was uneventful. Routine hematoxylin-eosin-stained sections of formalin-fixed, paraffin-embedded tissue showed an intraosseous carcinoma in which the cells were arranged in loosely cohesive sheets (Fig 2A), and focally displayed a
and pelvic
Received from St John’s Mercy Medical Center, St Louis, MO. * Resident, Department of Oral and Maxillofacial Surgery. t Fellow, Department of Pathology/Section of Surgical Pathology. $ Director of Surgical Pathology, Department of Pathology. Address correspondence and reprint requests to Dr Marrogi: Department of Pathology, St John’s Mercy Medical Center, 615 S New Ballas Rd, St Louis, MO 63141-8221. 0 1990 American Association of Oral and Maxillofacial geons 0278-2391190148050017$3.00/O
Sur-
FIGURE 1. Central mucoepidermoid carcinoma of the right mandible. The tumor appears as a radiolucent, multilocular lesion (arrows).
LEBSACK, MARROGI, AND MARTIN
FIGURE 2. Central mucoepidermoid carcinoma. A, Loosely cohesive sheets of intraosseous tumor. (Hematoxylin-eosin
stain. Orig-
inal magnification.
x90.)
x45.) B, Areas of tumor cells with clear cytoplasm. (Hematoxylin-eosin
rudimentary abortive acinar arrangement. The architectural features were variable throughout the tumor. Some cellular nests were surrounded by a hemorrhagic vascular stroma, and in these areas the tumor was reminiscent of paraganglioma. In other sections the malignant cells inliltrated the bone marrow spaces and were separated by widened bony trabeculae. Some tumor cells had a conspicuous. clear cytoplasm (Fig 2B). The immunocytochemical stain for carcinoembryonic antigen (Dako, Santa Barbara, CA) demonstrated focal intracytoplasmic staining in these cells. Histochemically, the tumor displayed intracytoplasmic PAS-positive diastase-resistant material. In areas of acinar arrangement there was extracellular mucicarminophilic material in the abortive lumina. The histologic diagnosis was central mucopidermoid carcinoma of the mandible. Approximately 2 months after the surgery, the patient began to experience dull pain in the right clavicle. A bone scan showed increased activity in that location (Fig 3). The patient subsequently underwent a biopsy from the area. The histologic features of the clavicular lesion were similar to those of the mandibular tumor (Fig 4). Following further evaluation which demonstrated no other metastatic lesions, the patient received radiation therapy after the extraction of the remaining mandibular teeth. She received a total dose of 6,000 rads to the mandible and 5,000 rads to the clavicle. Radiographic examination of both the mandible and the clavicle in November 1988 showed no evidence of recurrence, and the patient had no symptoms or complaints.
stain. Original magnification,
Discussion The criteria as proposed by Alexander et al’ and modified by Browand and Waldron3 for the diagno-
FIGURE 3. Tc99 bone scan showing lesion of the right mandible and metastasis to the right clavicle.
520
CENTRAL MUCOEPIDERMOID
CARCINOMA
FIGURE 4. Metastatic central mucoepidermoid carcinoma to the clavicle. The histoiogic appearance is similar to that of the primary tumor (Fig 2A). (Hematoxylin-eosin stain. Original magnitication, x 135.)
sis of central mucoepidermal carcinoma of the jaw bones include 1) the presence of an intact cortical bone plate and/or periosteum, 2) radiographic evidence of bone destruction, 3) histologic features of mucoepidermoid carcinoma and evidence of mucin production, 4) absence of a primary tumor elsewhere with potential histologic similarities to central mucoepidermoid carcinoma of the jaw, and 5) absence of a primary odontogenic cyst. The world literature contains reports on 63 cases of central mucoepidermoid carcinoma of the jaw Table 1. Case Source
Central Mucoepidermoid Carcinoma of the Jaws With Metastasis Agel Sex
Site
Hertz”
49/F
Mandible
Chaudhry et al4
16/F
Maxilla
Brown et al’
50/M
Mandible
Weinstein et al6
bones. l-3’ Of those patients, only 7 (9%) have been reported to develop metastasis, mainly to the regional lymph nodes (Table 1). One patient reported in the series of Dhawan et al* had metastatic tumor in the lung in the absence of nodal metastasis. This particular case may not represent an example of central mucoepidermoid carcinoma because this patient had tumors arising simultaneously both in the mandible and the adjacent parotid salivary gland. Of the 7 patients with metastatic tumor in the lymph nodes, 4 had lymph node involvement at the
50/F
Maxilla
Symptoms, Duration
Radiographic Appearance
Initial Treatment
Neuralgic pain and swelling Toothache and swelling, a few weeks Enlarged mass, nonpainful, 20 yr
Bone destruction
Res/Irr
Impacted L 3rd molar tooth
Infraorbital swelling, unknown duration
Time and Site of Metastasis
Follow-Up 20 yr, no tumor
Partial max/ LND
1 yr/ cervical node Simultaneous cervical node
Multiloculated cystic lesion
Cur
14 yrl
NA
Diffuse radiolucency
Hmax/LND
(Continued on hollowing page)
submaxillary, & jugular nodes Simultaneous/ jugular nodes
NA
34 mo, no tumor
521
LEBSACK, MARROGI, AND MARTIN
Table 1.
Central Mucoepidermoid Carcinoma of the Jaws With Metastasis (Cont’d)
Case Source
Agel Sex
Site
Smith et al’
49/F
Mandible
Dhawan et al”
5QlF
Mandible
Ganz et al9
18/M
Mandible
Radiographic Appearance
Initial Treatment
Intermittant swelling, adenopathy, several years Swelling and pain, 5 mos’ duration
Cystic lesion (ameloblastoma)
Enucleatiom LND
Simultaneous/ submaxillary nodes
32 yr, no tumor
NA
Mand
1 yr
4 yr, died with tumor
Swelling, nontender,
Multiloculated radiolucency Multicystic radiolucency
Partial Mandl LND
ipslateral 3 yr contr/ cervical nodes Simultaneous/ cervical nodes
1 yr Lebsack et al (this report)
?SiF
Mandible
Time and Site of Metastasis
Symptoms, Duration
duration Swelling, pain, 3 yrs’
Cur/IlT
Abbreviations: Hmax, hemimaxillectomy; Max, maxillectomy; Mand, mandibulectomy; available; Cur. curretage; Irr, irradiation; Res, resection; contr, contralateral.
time of resection of the primary tumor. The remaining 3 patients developed regional lymph node metastasis from 1 to 24 years after the time of initial management. The patient reported herein is another example of central mucoepidermoid carcinoma of the jaw bone. Even though the cortical bone plate was found to be perforated, there was no attachment of the tumor mass to the epithelial lining of the oral mucosa, and there was no involvement of the periosteum at the time of the surgical procedure. The patient is without recurrence 19 months after the treatment of her primary mandibular tumor. The reported case is unique in that the patient developed an osseous metastasis in the absence of regional lymph node involvement. Most patients with central mucoepidermoid carcinoma do not suffer from metastatic disease. Acknowledgment The authors thank W.E. Bisch, DDS, oral surgeon, for the clinical information provided.
References 1. Alexander RW. Dupuis RH, Holtn H: Central mucoepidermoid tumor (carcinoma) of the mandible. J Oral Surg 32:541. 1974 2. Eversole LR, Sabes WR, Sheldon R: Aggressive growth and neoplastic potential of odontogenic cysts. Cancer 35:270, 1975
2 ma/R clavicle
Follow-Up
30 mo, no tumor
9 mo, no tumor
LND, lymph node dissection;
NA, not
3. Browand BC, Waldron CA: Central mucoepidermoid tumors of the jaws. Oral Surg 40:63 1, 1975 4. Chaudhry AP, Dedolph T, Vickers RA: Mucoepidermoid tumor arising from ectopic salivary gland in the maxilla. J Oral Surg Anesth Hosp Dent Serv 19:521, 1961 5. Brown AM, Lucchesi FJ: Central mucoepidermoid tumor of the mandible-Report of case. J Oral Surg 24:356, 1966 6. Weinstein IR, Nagai I, Yamanaka H: Mucoepidermoid tumor of the maxilla. Report of a case. Oral Surg Oral Med Oral Path01 23: 1, 1967 7. Smith RL, Dahlin DC, Waite DE: Mucoepidermoid carcinoma of the jawbones. J Oral Surg 16:387, 1968 8. Dhawan IK, Bhargava S, Nayak NC, et al: Central salivary gland tumors of jaws. Cancer 26:211, 1970 9. Ganz SD, Toth BB: Central mucoenidermoid carcinoma simulating an odontogenic tumor. J bra1 Med 40: 179, 1985 10. Gingell JC, Beckerman T, Levy BA, et al: Central mucoepidermoid carcinoma. Oral Surg 5:436, 1984 1I. Grubka JM, Wesley RK, Monaco F: Primary intraosseous mucoepidermoid carcinoma of the anterior part of the mandible. J Oral Maxillofac Surg 41:389, 1983 12. Bhaskar SN, Bemier JL: Mucoepidermoid tumors of major and minor salivary glands. Clinical features, histology, variations, natural history, and results of treatment for 144 cases. Cancer 15:801, 1962 13. Tamai C: Mucoepidermoid tumor of the mandible. Am J Surg 92~872, 1956 14. Stoll HC, Marchetta FC, Schobinger R: Malignant epithelial tumors of the mandible and maxilla. Arch Path01 64:239, 1957 15. Bhaskar SN: Central mucoepidermoid tumors of the mandible: Report of 2 cases. Cancer 16:721, 1963 16. Auriol M, Chomette G, Delcourt A, et al: Tumeur mucoepidermoide mandibulaire. Rev Stomatol Chir Maxillofac 86:414, 1985 17. Ito H, Soda T, Nakajima T, et al: Central mucoepidermoid tumor of the mandible: Report of case. Bull Tokyo Med Dent Univ 16:251, 1969 18. Silverglade LB, Alvares OF, Olech E: Central mucoepidermoid tumors of the jaws. Cancer 22:650, 1%X
522
PERIOSTEAL
19. Schultz W, Whitten JB: Mucoepidermoid carcinoma in the mandible: Report of a case. J Oral Surg 27:337, 1969 20. Frederickson C, Cherrick HM: Central mucoepidermoid carcinoma of the jaws. J Oral Med 33:80, 1978 21. Unal T, Zeytinoglu B: Central mucoepidermoid carcinoma of the jaws. J Oral Med 40:202, 1985 22. Pincock JL, El-Mofty SK: Recurrence of cystic central mucoepidermoid tumor of the mandible. Int J Oral Surg 14:81, 1985 23. Stoch RB, Smith I: Mucoepidermoid carcinoma of the mandible: Report of case. J Oral Surg 3856, 1980 24. Lepp H, cited in Hertz J: Mucus secreting tumors of the jaws. Acta Chir Stand 103:276, 1952 25. Hertz J: Mucus secreting tumors of the jaws. Acta Chir Stand 103:276, 1952
J Oral Maxillofac 46:522-526,
OSTEOSARCOMA
OF THE MAXILLA
26. Sonesson A: Mucus-secreting and cystic epidermoid carcinoma of the jaws. Acta Radio1 34:25, 1950 27. Szerlip L: Cystic mucoepidermoid tumor. Report of case. Oral Sure 9:584. 1956 28. Rzucidlo i?B, Rzeszotko-Adamiczka D: Uber einen mucoepidermoid tumors eines Unterkiefers. Zentralbl Allg Pathol 105:409, 1964 29. Attenson M, Kaufman M, Brown S: Mucoepidermoid carcinoma of the mandible: Report of a case. J Oral Surg 22:350, 1964 30. Lacour M, Reynaud J, Grappin G, et al: A case of mucoepidermoid tumors of the mandible. Bull Sot Med Afr Lang Fr 11:73, 1966 31. Karmarkar SR: Central mucoepidermoid tumors of the mandible. Indian J Surg 31: 154, 1969
Surg
1990
Perios teal Osteosarcoma
of the Maxilla:
A Case Report and Review of Literature LTC ADRIAN PATTERSON, DDS, DC, USA,* ROBERT 0. GREER JR, DDS, ScD,t AND DANIEL HOWARD, DDSS
Juxtacortical osteosarcoma was first fully described by Jaffee and Selin’ in 1951, although Geschickter and Copeland had described a similar lesion under the heading “parosteal osteoma” in 1949.*s3 Since these initial reports, juxtacortical osteosarcoma has been described using various names, including ossifying parosteal osteogenic sarcoma, and parosteal and periosteal ossifying fibrosarcoma.4T5 Juxtacortical osteogenic sarcoma occurs much less commonly than central medullary osteosarcoma, which has an incidence rate of 0.07 per 1,000 population. The clinicopathology of fewer than a dozen cases of juxtacortical osteosarcoma has been
* Chief, Surgery Branch, United States Institute of Dental Research, Department of the Army, Walter Reed Army Medical Center, Washington, DC. t Professor and Chairman, Division of Oral Pathology, University of Colorado School of Dentistry; Professor of Pathology, University of Colorado School of Medicine, Denver. $ In private practice, Washington, DC. The opinions or assertations contained herein are the private views of the authors and are not to be construed as official or as reflecting the view of the Department of the Army. Address correspondence and reprint requests to LTC Patterson: Surgery Branch, US Institute of Dental Research, Department of the Army, Walter Reed Army Medical Center, Washington, DC 20307-5300. 0 1990 American
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well documented in the English literature (Table l)? Recently, Zarbo et al’* suggested that juxtacortical osteosarcoma can be further subdivided into two clinicopathologic variants: parosteal osteosarcoma and periosteal osteosarcoma. The purpose of this article is to report a rare example of maxillary juxtacortical osteosarcoma of the periosteal type and to discuss the clinicopathologic nature of that disease process. Report of a Case A 65year-old black woman was referred by her general dentist for evaluation and treatment of a mass in the right maxillary vestibule and alveolus. The lesion had been present and slowly enlarging for approximately 1 year. It had been asymptomatic until about 3 weeks before her visit, when the patient began to have mild pain in the maxillary right quadrant. Physical examination revealed a 2.1 x 2.5 x 2 cm hard mass in the right maxillary vestibule (Fig 1). The lesion was lobular in appearance. The gingiva overlying the lesion showed increased vascularity, but no ulceration. The buccal vestibule was nearly obliterated by the lesion in the region of the premolar and canine teeth. There was neither paresthesia or anesthesia of the infraorbital nerve. A panoramic radiograph showed a slightly radiopaque shadow approximately 0.5 x 0.5 cm distal to the premolar (Fig 2), as well as a longitudinal fracture of this tooth. There was also a loss of lamina dura around the involved teeth. Neck examination was negative for tenderness and adenopathy. An excisional biopsy was performed under local anes-
