J Oral
Maxillofac
50:904-906.
Surg
1992
Synovial Sarcoma of the Tongue: Case Report and Review of the Literature ROSARIO CARRILLO, MD,* ADEL K. EL-NAGGAR, MD,t JOSE L. RODRIGUEZ-PERALTO, MD,* AND JOHN G. BATSAKIS, Synovial sarcoma is a malignant soft-tissue neoplasm that rarely occurs in the head and neck region.‘” In this location, the cervical and the parapharyngeal areas are most frequently affected; only a few examples of intraoral synovial sarcoma have been reported.3a We report a case of synovial sarcoma of the tongue, and add it to a review of the six other reported synovial sarcomas of the tongue.
MD§
Discussion Synovial sarcoma is a malignant soft-tissue neoplasm that accounts for 7% to 10% of all soft-tissue sarcomas.’
Report of a Case A 13-year-old boy presented with a swelling in the left lateral aspect of the tongue. On clinical examination, a solitary, round, well-delineated nodule was noted in the lingual musculature. Complete magnetic resonance imaging (MRI) and computed tomography (CT) scans were negative. The tumor was conservatively excised with negative surgical margins. Grossly, a 1.1 X 0.5 X OS-cm, light-tan, firm nodule was noted in lingual muscle. Although the neoplasm appeared circumscribed, there was microscopic evidence of invasion of skeletal muscle. Biphasic features consisting of glandlike structure and intertwining spindle cell components were observed (Fig 1). The glandlike component consisted of polygonal cells that were strongly reactive to keratin antibodies (Fig 2). The spindle component was composed of fusiform cells with scant cytoplasm and was strongly stained with antibodies against vimentin. Mitoses were infrequently noted. Flow cytometric DNA analysis performed on paraffin blockretrieved tissue sections, revealed a diploid pattern with low S-phase (4%). The patient is alive and well 1 year following excision.
Received from the Department of Pathology, The University of Texas, M.D. Anderson Cancer Center, Houston. * Visiting Fellow. 7 Associate Professor. $ Visiting Fellow. $ Chairman. Address correspondence and reprint requests to Dr El-Naggar: Department of Pathology, The University of Texas, M.D. Anderson Cancer Center, 15 15 Holcombe Blvd, Box 85, Houston, TX 77030.
FIGURE 1. A, Low-power photomicrograph showing a well-circumscribed portion of the neoplasm and adjacent skeletal muscle. B, Biphasic cellular pattern of synovial sarcoma with a prominent epithelioid component (hematoxylin-eosin stain, original magnification X250).
0 1992 AmericanAssociationof Oral and MaxillofacialSurgeons 0279-2391/92/5006-0023$3.00/0
904
905
CARRILLO ET AL
Table 1.
Synovial Sarcomas
of the Tongue
Reference
(yr)/Sex
Size (cm)
Histology
Location
Management
Follow-up/yr
Mir-Abedy’ Novotny & Fort’ Moussavi & Ghodsi” Guzman-Patraca et al” Bridge et aI6 Shmookler et al5 Present case
23/F 21/M 40/M 21/M 31/M 16/M 13/M
5.0 3.5 6.0 1.0 2.5 1.2 1.0
Biphasic Biphasic Biphasic Biphasic Biphasic Biphasic Biphasic
Base of tongue Base of tongue Base of tongue Base of tongue Base of tongue Mobile tongue Mobile tongue
SX RTX SX, RTX SX sx sx sx
A&W/3 A&W/2 A & W/l.3 A with lung metastases/l NK A&W/8 A&W/L
Age
Abbreviations: SX, surgical excision; RTX, irradiation therapy; A, alive; A & W. alive and well: NK. not known.
The neoplasm occurs most frequently in extremities of young adults, with a clear predilection to juxta-artitular locations. It may less frequently occur, however, in areas with no apparent relationship to synovial structures. ‘,2 Jernstrom7 was first to report the occurrence of this tumor in the head and neck region. Since then, more than 50 cases in the head and neck have been reported.2-” The majority of these neoplasms was located in retropharyngeal or parapharyngeal spaces. Intraoral synovial sarcoma is very rare. Only six other lingual synovial sarcomas have been The clinicopathologic charpreviously reported. 5~6,8-‘1 acteristics of these neoplasms, along with our case, are presented in Table 1. Five of the sarcomas were located in the base of the tongue, near the hypopharynx. Only two, including our case, were in the mobile part of the tongue. Five patients were treated with excision only, one with excision and radiotherapy, and one with radio-
therapy alone. Although the follow-up periods are short, it is interesting that all patients are alive without evidence of recurrence, except one who is living with lung metastasis. This generally less aggressive course is in accordance with observations relating to synovial sarcomas in other head and neck sites.4*5It may be that early detection, small size, and the relatively young age of the patients with head and neck tumors, in comparison with synovial sarcomas of the extremities, play a role in their less aggressive clinical behavior. The flow cytometric DNA content analysis of the neoplasm manifested a diploid pattern and a low proliferative activity. These features have been associated with synovial sarcomas displaying epithelioid features and an improved clinical co~rse.‘~-‘~ The flow cytometric DNA content also correlates with recent cytogenetic analyses of synovial sarcomas in which diploid or hypodiploid chromosomal numbers and nonrandom chromosomal translocations were found.6,‘4,‘5 The differential diagnosis of a biphasic synovial sarcoma is rather limited; its monophasic variant poses a greater diagnostic problem. Sarcomatoid carcinoma, spindle-cell melanoma, and leiomyosarcoma are the principal entities to be excluded. Immunohistochemical studies for intermediate filaments that phenotype these neoplasms are useful in such instances. I6 References
FIGURE 2. Strong immunoreactivity against keratin antibodies is displayed in the epithehoid elements of the synovial sarcoma (original magnification X250).
1. Enzinger FM, Weiss SW: Soft Tissue Tumors. St Louis, MO, Mosby, 1988, p 659 2. Roth JA, Enzinger FM, Tannenbaum M: Synovial sarcoma of the neck: A follow-up study of 24 cases. Cancer 35: 1243,1975 3. Moore DM, Berke GS: Synovial sarcoma of the head and neck. Arch Otolaryngol Head Neck Surg 113:311, 1987 4. Mamelle G, Richard J, Luboinski B, et al: Synovial sarcoma of the head and neck: An account of four cases and review of the literature. Eur J Surg Oncol 12:347, 1986 5. Shmookler BM, Usar MC, Enzinger FM, et al: Orofacial synovial sarcoma A clinicopathologic study of 11 new cases and review of the literature. Cancer 50~269, 1982 6. Bridge JA, Bridge RS, Borek DA, et al: Translocation t(x; 18) in orofacial synovial sarcoma. Cancer 62:935, 1988 7. Jemstrom P: Synovial sarcoma of the pharynx. Report of a case. Am J Clin Path01 24957, 1954
906
CLEAR-CELL VARIANT OF MUCOEPIDERMOID
8. Novotny GM, Fort TC: Synovial sarcoma of the tongue. Arch Otolarvneol94:77. 1971 9. Mir-Abedy M: Considerations of the base of the tongue and its tumors. Ann d’Otolaryngo1 Chirurg Cervico-Fat 79:547, 1962 10. Moussavi M, Ghodsi S: Synovial sarcoma of the tongue: Report of a case. J Laryngol Otol88:795, 1974 11. Guzman-Patraca C, Torres HO, Garza RG: Synovial sarcoma of the oral cavity (foramen caecum): Report of a case and review of the literature. Prensa Med Mex 40:2 15, 1975 12. El-Naggar AK, Ayala AG, Abdul-Karim FW, et ah Synovial sarcoma. A DNA Bow cytometric study. Cancer 65:2295, 1990
J Oral Maxillofac 50:906-910,
CARCINOMA
13. Kreicbergs A, Tribukait B, Willems J, et al: DNA flow analysis of soft tissue tumors. Cancer 59: 128. 1987 14. Griffin CG, Emanuel BS: Translocation (X;18) in a synovial sarcoma. Cancer Genet Cytognet 26: 181, 1987 15. Turc-Care1 C, Dal Cin P, Limon J, et al: Translocation X;18 in synovial sarcoma. Cancer Genet Cytogenet 22:93, 1986 16. Ordoiiez NG, Mahfouz SM, McKay B: Synovial sarcoma: An immunohistochemical and ultrastructural study. Hum Path01 21:733, 1990
Surg
1992
Clear-Cell Variant of Mucoepidermoid Carcinoma: Report of a Case With lmmunohistochemical and Ultrastructural Observations IKUKO OGAWA, DDS, PHD,* HIROMASA NIKAI, DDS, PtdD,t TAKASHI TAKATA, DDS, PHD,* AND RYOICHI YASUI, DDS, PHD,!$
Mucoepidermoid carcinoma, first described as a distinct entity by Stewart et al in 1945,’ is relatively common among major and minor salivary gland mahgnancies. Histologically, this tumor is composed of mucus-secreting cells, epidermoid cells, and intermediate cells in varying proportions. In addition to these essential cell types, clear cells have also been described as an occasional component of the tumor parenchyma. 1-4Less commonly, clear cells may predominate, which causes difficulty in diagnosis.5,6 This article reports a rare case of this type of mucoepidermoid carcinoma arising from minor salivary glands of the retromolar region. Pertinent histochemical, immunohistochemical, and ultrastructural findings that can aid
in the differentiation of this tumor from other clear cell neoplasms are discussed. Report of a Case A 6 1-year-old Japanese woman complained of a large mass in the right retromolar region, which had been present for one year and gradually enlarging, causing asymmetry of the
Received from Hiroshima University Dental Hospital and Hiroshima University School of Dentistry, Hiroshima, Japan. * Research Fellow in Oral Pathology, Clinical Laboratory. 7 Professor, Department of Oral Pathology. $ Assistant Professor, Clinical Laboratory. 0 Research Associate, Department of Oral and Maxillofacial Surgery II. Address correspondence and reprint requests to Dr Ogaw~ Clinical Laboratory, Hiroshima University Dental Hospital, l-2-3 Kasumi Minami-ku, Hiroshima, 734, Japan. 0 1992 AmericanAssociationof Oral and MaxillofacialSurgeons 0276-2391/92/5006-0024$3.00/O
FIGURE 1. Clinical view of tumor at initial examination.
Maxillofac
50:904-906.
Surg
1992
Synovial Sarcoma of the Tongue: Case Report and Review of the Literature ROSARIO CARRILLO, MD,* ADEL K. EL-NAGGAR, MD,t JOSE L. RODRIGUEZ-PERALTO, MD,* AND JOHN G. BATSAKIS, Synovial sarcoma is a malignant soft-tissue neoplasm that rarely occurs in the head and neck region.‘” In this location, the cervical and the parapharyngeal areas are most frequently affected; only a few examples of intraoral synovial sarcoma have been reported.3a We report a case of synovial sarcoma of the tongue, and add it to a review of the six other reported synovial sarcomas of the tongue.
MD§
Discussion Synovial sarcoma is a malignant soft-tissue neoplasm that accounts for 7% to 10% of all soft-tissue sarcomas.’
Report of a Case A 13-year-old boy presented with a swelling in the left lateral aspect of the tongue. On clinical examination, a solitary, round, well-delineated nodule was noted in the lingual musculature. Complete magnetic resonance imaging (MRI) and computed tomography (CT) scans were negative. The tumor was conservatively excised with negative surgical margins. Grossly, a 1.1 X 0.5 X OS-cm, light-tan, firm nodule was noted in lingual muscle. Although the neoplasm appeared circumscribed, there was microscopic evidence of invasion of skeletal muscle. Biphasic features consisting of glandlike structure and intertwining spindle cell components were observed (Fig 1). The glandlike component consisted of polygonal cells that were strongly reactive to keratin antibodies (Fig 2). The spindle component was composed of fusiform cells with scant cytoplasm and was strongly stained with antibodies against vimentin. Mitoses were infrequently noted. Flow cytometric DNA analysis performed on paraffin blockretrieved tissue sections, revealed a diploid pattern with low S-phase (4%). The patient is alive and well 1 year following excision.
Received from the Department of Pathology, The University of Texas, M.D. Anderson Cancer Center, Houston. * Visiting Fellow. 7 Associate Professor. $ Visiting Fellow. $ Chairman. Address correspondence and reprint requests to Dr El-Naggar: Department of Pathology, The University of Texas, M.D. Anderson Cancer Center, 15 15 Holcombe Blvd, Box 85, Houston, TX 77030.
FIGURE 1. A, Low-power photomicrograph showing a well-circumscribed portion of the neoplasm and adjacent skeletal muscle. B, Biphasic cellular pattern of synovial sarcoma with a prominent epithelioid component (hematoxylin-eosin stain, original magnification X250).
0 1992 AmericanAssociationof Oral and MaxillofacialSurgeons 0279-2391/92/5006-0023$3.00/0
904
905
CARRILLO ET AL
Table 1.
Synovial Sarcomas
of the Tongue
Reference
(yr)/Sex
Size (cm)
Histology
Location
Management
Follow-up/yr
Mir-Abedy’ Novotny & Fort’ Moussavi & Ghodsi” Guzman-Patraca et al” Bridge et aI6 Shmookler et al5 Present case
23/F 21/M 40/M 21/M 31/M 16/M 13/M
5.0 3.5 6.0 1.0 2.5 1.2 1.0
Biphasic Biphasic Biphasic Biphasic Biphasic Biphasic Biphasic
Base of tongue Base of tongue Base of tongue Base of tongue Base of tongue Mobile tongue Mobile tongue
SX RTX SX, RTX SX sx sx sx
A&W/3 A&W/2 A & W/l.3 A with lung metastases/l NK A&W/8 A&W/L
Age
Abbreviations: SX, surgical excision; RTX, irradiation therapy; A, alive; A & W. alive and well: NK. not known.
The neoplasm occurs most frequently in extremities of young adults, with a clear predilection to juxta-artitular locations. It may less frequently occur, however, in areas with no apparent relationship to synovial structures. ‘,2 Jernstrom7 was first to report the occurrence of this tumor in the head and neck region. Since then, more than 50 cases in the head and neck have been reported.2-” The majority of these neoplasms was located in retropharyngeal or parapharyngeal spaces. Intraoral synovial sarcoma is very rare. Only six other lingual synovial sarcomas have been The clinicopathologic charpreviously reported. 5~6,8-‘1 acteristics of these neoplasms, along with our case, are presented in Table 1. Five of the sarcomas were located in the base of the tongue, near the hypopharynx. Only two, including our case, were in the mobile part of the tongue. Five patients were treated with excision only, one with excision and radiotherapy, and one with radio-
therapy alone. Although the follow-up periods are short, it is interesting that all patients are alive without evidence of recurrence, except one who is living with lung metastasis. This generally less aggressive course is in accordance with observations relating to synovial sarcomas in other head and neck sites.4*5It may be that early detection, small size, and the relatively young age of the patients with head and neck tumors, in comparison with synovial sarcomas of the extremities, play a role in their less aggressive clinical behavior. The flow cytometric DNA content analysis of the neoplasm manifested a diploid pattern and a low proliferative activity. These features have been associated with synovial sarcomas displaying epithelioid features and an improved clinical co~rse.‘~-‘~ The flow cytometric DNA content also correlates with recent cytogenetic analyses of synovial sarcomas in which diploid or hypodiploid chromosomal numbers and nonrandom chromosomal translocations were found.6,‘4,‘5 The differential diagnosis of a biphasic synovial sarcoma is rather limited; its monophasic variant poses a greater diagnostic problem. Sarcomatoid carcinoma, spindle-cell melanoma, and leiomyosarcoma are the principal entities to be excluded. Immunohistochemical studies for intermediate filaments that phenotype these neoplasms are useful in such instances. I6 References
FIGURE 2. Strong immunoreactivity against keratin antibodies is displayed in the epithehoid elements of the synovial sarcoma (original magnification X250).
1. Enzinger FM, Weiss SW: Soft Tissue Tumors. St Louis, MO, Mosby, 1988, p 659 2. Roth JA, Enzinger FM, Tannenbaum M: Synovial sarcoma of the neck: A follow-up study of 24 cases. Cancer 35: 1243,1975 3. Moore DM, Berke GS: Synovial sarcoma of the head and neck. Arch Otolaryngol Head Neck Surg 113:311, 1987 4. Mamelle G, Richard J, Luboinski B, et al: Synovial sarcoma of the head and neck: An account of four cases and review of the literature. Eur J Surg Oncol 12:347, 1986 5. Shmookler BM, Usar MC, Enzinger FM, et al: Orofacial synovial sarcoma A clinicopathologic study of 11 new cases and review of the literature. Cancer 50~269, 1982 6. Bridge JA, Bridge RS, Borek DA, et al: Translocation t(x; 18) in orofacial synovial sarcoma. Cancer 62:935, 1988 7. Jemstrom P: Synovial sarcoma of the pharynx. Report of a case. Am J Clin Path01 24957, 1954
906
CLEAR-CELL VARIANT OF MUCOEPIDERMOID
8. Novotny GM, Fort TC: Synovial sarcoma of the tongue. Arch Otolarvneol94:77. 1971 9. Mir-Abedy M: Considerations of the base of the tongue and its tumors. Ann d’Otolaryngo1 Chirurg Cervico-Fat 79:547, 1962 10. Moussavi M, Ghodsi S: Synovial sarcoma of the tongue: Report of a case. J Laryngol Otol88:795, 1974 11. Guzman-Patraca C, Torres HO, Garza RG: Synovial sarcoma of the oral cavity (foramen caecum): Report of a case and review of the literature. Prensa Med Mex 40:2 15, 1975 12. El-Naggar AK, Ayala AG, Abdul-Karim FW, et ah Synovial sarcoma. A DNA Bow cytometric study. Cancer 65:2295, 1990
J Oral Maxillofac 50:906-910,
CARCINOMA
13. Kreicbergs A, Tribukait B, Willems J, et al: DNA flow analysis of soft tissue tumors. Cancer 59: 128. 1987 14. Griffin CG, Emanuel BS: Translocation (X;18) in a synovial sarcoma. Cancer Genet Cytognet 26: 181, 1987 15. Turc-Care1 C, Dal Cin P, Limon J, et al: Translocation X;18 in synovial sarcoma. Cancer Genet Cytogenet 22:93, 1986 16. Ordoiiez NG, Mahfouz SM, McKay B: Synovial sarcoma: An immunohistochemical and ultrastructural study. Hum Path01 21:733, 1990
Surg
1992
Clear-Cell Variant of Mucoepidermoid Carcinoma: Report of a Case With lmmunohistochemical and Ultrastructural Observations IKUKO OGAWA, DDS, PHD,* HIROMASA NIKAI, DDS, PtdD,t TAKASHI TAKATA, DDS, PHD,* AND RYOICHI YASUI, DDS, PHD,!$
Mucoepidermoid carcinoma, first described as a distinct entity by Stewart et al in 1945,’ is relatively common among major and minor salivary gland mahgnancies. Histologically, this tumor is composed of mucus-secreting cells, epidermoid cells, and intermediate cells in varying proportions. In addition to these essential cell types, clear cells have also been described as an occasional component of the tumor parenchyma. 1-4Less commonly, clear cells may predominate, which causes difficulty in diagnosis.5,6 This article reports a rare case of this type of mucoepidermoid carcinoma arising from minor salivary glands of the retromolar region. Pertinent histochemical, immunohistochemical, and ultrastructural findings that can aid
in the differentiation of this tumor from other clear cell neoplasms are discussed. Report of a Case A 6 1-year-old Japanese woman complained of a large mass in the right retromolar region, which had been present for one year and gradually enlarging, causing asymmetry of the
Received from Hiroshima University Dental Hospital and Hiroshima University School of Dentistry, Hiroshima, Japan. * Research Fellow in Oral Pathology, Clinical Laboratory. 7 Professor, Department of Oral Pathology. $ Assistant Professor, Clinical Laboratory. 0 Research Associate, Department of Oral and Maxillofacial Surgery II. Address correspondence and reprint requests to Dr Ogaw~ Clinical Laboratory, Hiroshima University Dental Hospital, l-2-3 Kasumi Minami-ku, Hiroshima, 734, Japan. 0 1992 AmericanAssociationof Oral and MaxillofacialSurgeons 0276-2391/92/5006-0024$3.00/O
FIGURE 1. Clinical view of tumor at initial examination.
