- Email: [email protected]
Rapidly growing mass in mandibular gingiva
CLINICOPATHOLOGIC CONFERENCE J Oral Maxillofac Surg 62:214-217, 2004
Rapidly Growing Mass in Mandibular Gingiva Chieh-Yuan Cheng, DDS,* Kuo-Ming Chang, MD,† Kuo-Wei Chang, DDS, PhD,‡ Chia-Hoa Chang, DDS,§ Chung-Ji Liu, DDS, MS,¶ and George M. Kushner, DMD, MD㛳 Case Presentation
old girl. A previous biopsy revealed fibrous granulation tissue, which is a rather nondescript diagnosis and gives us very limited useful knowledge. One must wonder if a representative sample of the lesion was not obtained or there was a possible misinterpretation or misdiagnosis of the tissue that was sent for pathologic examination. On my review of the provided radiograph, there appears to be bony cratering of the mandible distal to the right mandibular first molar with displacement of the right mandibular second molar posteriorly. There is no mention of paresthesia or alterations in sensation of the mandible. This mass is firm with a rubbery feel and mildly tender to palpation. In establishing a differential diagnosis, we need to look at the broad categories of congenital, developmental, infectious, reactive, and neoplastic processes. I believe we can essentially rule out a congenital or developmental problem in this patient. Under the category of infectious etiology, we still must consider a granulomatous etiology such as histoplasmosis. The granulomatous diseases are known as the “great masqueraders” in the head and neck region because they often present with atypical signs and symptoms. However, this patient has no lymphadenopathy, and this diagnosis would clearly be much lower on my list of possible etiologies. Under the category of “reactive lesions,” one must consider a pyogenic granuloma. The orthodontic appliance or luting cement can easily be an irritant in the oral cavity but the presence of this orthodontic appliance may be totally unrelated to the final diagnosis. The diagnosis of pyogenic granuloma would certainly go along with the fibrous granulation tissue seen on the prior pathology report. However, pyogenic granulomas are usually soft and bleed easily. This mass is firm and has a rubbery feel to palpation. One must also consider a peripheral ossifying fibroma and a peripheral giant cell granuloma as other reactive lesions that could mimic this disease entity. Both of these tumors would certainly feel more solid than a pyogenic granuloma. Under the category of “neoplastic diseases,” at the top of the list is the broad category of fibromatosis.
A 9-year-old girl, with the chief complaint for the painful swelling on the right mandibular gingiva for about 4 weeks, was referred to our Oral and Maxillofacial Surgery Department in December 2000. She had undergone treatment with the lingual holding arch, which was retained by the lower first molar bilaterally as a space maintainer due to the early loss of the lower deciduous molars. After wearing the appliance for a few weeks, an easily bleeding tumor was found just behind the right mandibular first molar. At that time, the clinical impression was irritation-induced pyogenic granuloma so local debridement was performed by the patient’s family dentist. The pathology report was fibrous granulation tissue. One week later, the lesion grew larger with associated pain. She was referred to our Oral and Maxillofacial Surgery Department for further treatment. Physical examination revealed a rubbery tumor on right mandibular gingiva that measured about 4 ⫻ 3 cm with mild tenderness (Fig 1A). No obvious cervical lymphadenopathy was observed. Panoramic film showed a bony resorptive lesion behind the right mandibular first molar with a displaced second molar (Fig 1B).
Differential Diagnosis George M. Kushner, DMD, MD In summary, we are basically dealing with a rather rapidly growing lesion in an otherwise healthy 9-year*Attending Physician, Oral and Maxillofacial Surgery Department, Mackay Memorial Hospital, Taipei, Taiwan. †Attending Physician, Department of Pathology, Mackay Memorial Hospital, Taipei, Taiwan. ‡Professor, Institute of Oral Biology, Yang Ming University, Taipei, Taiwan. §Attending Physician, Pedodontics Department, Mackay Memorial Hospital, Taipei, Taiwan. ¶Attending Physician, Oral and Maxillofacial Surgery Department, Mackay Memorial Hospital, Taipei, Taiwan. 㛳Associate Professor of Oral and Maxillofacial Surgery, Department of Surgical and Hospital Dentistry, School of Dentistry, University of Louisville, Louisville, KY. Address correspondence and reprint requests to Dr Liu: No 92, Sec 2, Chungshan North Road, 104, Taipei, Taiwan; e-mail: [email protected] © 2004 American Association of Oral and Maxillofacial Surgeons
0278-2391/04/6202-0013$30.00/0 doi:10.1016/j.joms.2002.12.004
214
215
CHENG ET AL
try to gather several likely or common lesions within each category to provide a broad base of possible pathologic conditions. At this point, I recommend a generous incisional biopsy with adequate tissue to definitively establish the clinical diagnosis. I certainly prefer that this tissue be read by an oral and maxillofacial pathologist. Final treatment recommendations would be based on the correct clinical diagnosis.
Subsequent Course
FIGURE 1. A, Photograph showing ulcerated tumor (arrow) located in the right mandibular gingiva and measuring about 4 ⫻ 3 cm. B, Panoramic radiograph showing ill-defined radiolucent lesion with mandibular osteolytic change and the second molar pushed away (arrow).
These lesions generally occur in children and can grow rapidly, which would fit our history rather nicely. Juvenile fibromatosis, desmoplastic fibroma, and myofibroma are all potential specific diagnoses within the fibromatosis category. Any one of these diagnoses could fit the clinical scenario as presented. In addition, one should keep the peripheral odontogenic fibroma on the list as a potential neoplasm, although this is a rather less common entity. Unfortunately, the clinician must consider a rapidly growing tumor that is resorbing bone in a young patient as possibly being malignant. The very broad category of sarcomas is a relatively common pediatric malignancy. Sarcomas, including rhabdomyosarcoma, fibrosarcoma, and osteogenic sarcoma could easily have the clinical presentation that has been described. Any one of these lesions could be a rapidly growing lesion that resorbs bone in an otherwise healthy young patient. Paresthesia is a later symptom of these diseases as the inferior alveolar nerve becomes affected with the growth of the lesion. Last, a diagnosis of Langerhans cell/histiocytosis X should be considered. These lesions typically resorb bone, can have exophytic growth and certainly occur in the pediatric population. This clinical entity could certainly fit quite nicely with the clinical presentation as described. In establishing a differential diagnosis, I
Although the previous pathology report was granulation tissue, malignant lesion was highly suspected and surgical excision was suggested. After admission, the surgery was performed under general anesthesia. The frozen section was made at first and reported as spindle-shaped tumor with minimal nuclear atypia and occasional mitoses. The tumor was compositely excised with 1-cm soft tissue margin and marginal mandibular bone. Microscopically, the tumor, composed of spindle cells, was arranged in whorling bundles. Some fibrosis was observed. These fusiform cells exhibited spindle-shaped nuclei with blunted ends. Mitotic figures were common (Fig 2A). The immunostains for smooth muscle actin were all positive on tumor cells and normal smooth muscle of blood vessel wall (Fig 2B). Cytokeratin, factor VIII, S-100, and desmin showed all negative immunoreactivity (Fig 2C).
Pathologic Diagnosis The final diagnosis was low-grade leiomyosarcoma of right mandibular gingiva. The patient has been closely followed up for 18 months and remains disease free, with no evidence of local recurrence or distant metastasis (Fig 3).
Discussion Leiomyosarcoma (LMS) is a smooth muscle neoplasm that rarely occurs in the oral cavity and confirmed only with ultrastructural and immunohistochemical findings. To our knowledge, fewer than 50 cases of oral LMS are reported in the English literature to date.1 Only 0.064% of smooth muscle tumors have an intraoral location.2 Oral LMS usually presents as a mass that may or may not exhibit ulceration. Tooth mobility and pericoronal soft tissue swelling may be the initial presenting features. The possible intraoral sites of origin are included: 1) the circumvallate papillae of the tongue, 2) pluripotential undifferentiated mesenchymal cells, 3) smooth muscle elements in blood vessel walls, 4) neurovascular bundle, 5) erector pilis muscle in aberrant hair follicles in the cheek, and 6) myoepithelial cells of mucous glands.3-6 In our case, gingival LMS likely arises from smooth muscle cells, which invest gingival blood vessels, smooth muscular elements surrounding adjacent glandular ex-
216
GROWING MASS IN MANDIBULAR GINGIVA
FIGURE 2. A, Photomicrograph showing fusiform tumor cells with blunt-ended nuclei with occasional mitotic changes (arrow) (hematoxylin and eosin stain, original magnification ⫻400). B, Photomicrograph showing strong immunoreaction to smooth muscle actin in tumor cells and muscle elements of normal vessel wall (immunostaining for smooth muscle actin, original magnification ⫻200). C, Photomicrograph showing negative immunoreaction to desmin in tumor cells (immunostaining for desmin, original magnification ⫻100).
cretory duct, or perhaps from pluripotential remnants of embryonal mesenchyme. The differential diagnosis of oral LMS should consist of odontogenic tumor, spindle cell carcinoma, other sarcoma, myoepithelioma, melanoma, schwannoma, myofibroma, myofibromatosis, angioleiomyoma, fibrous histocytoma, central giant cell granuloma, and lymphoma. Histopathologic diagnosis of mesenchymal tumors, and those with mesenchymal-like features, can be difficult. Spindle cell carcinoma is often strongly positive for cytokeratin. Myoepithelioma has the shorter and often more slender nuclei with a more regular nuclear contour. Melanoma stains negative for myogenic marker and cytokeratin and positive for S-100 protein. Fibrosarcoma cells tend to be tapered, whereas schwannoma cells are wavy, buckled, and distinctly asymmetric. Typical LMS with interlacing bundles of spindle cells have blunt-ended nuclei, hyperchromatism, cellular pleomorphism, and mitotic activity. LMS stains positive for muscle-specific actin (HHF35) and smooth muscle actin and negative for S-100 protein, cytokeratin, and epithelial membrane antigen. Myofilament and deeply clefted nuclei may be shown on electron microscopy. With cytochemical, immunocytochemical, and ultrastructural evaluation, one can make a specific diagnosis for LMS from other similar lesions.1,3,7-11 The location of LMS is important in determining the prognosis. There are 3 geographic (or regional) groups according to certain clinical and biological differences: 1) LMS of deep soft tissue, 2) LMS of cutaneous and subcutaneous tissue, and 3) LMS of vascular origin.7 The more superficial location of LMS
FIGURE 3. Postoperative clinical appearance (A) and panoramic radiograph (B) showed healthy gingival mucosa with no evidence of recurrence.
CHENG ET AL
results in a better prognosis. The prognosis for patients with oral LMS is guarded, with a recurrence rate that approaches 50% after wide extirpation. Oral LMS tends to metastasize to the cervical nodes and lung, whereas when the tumor originates in other locations, it usually metastasizes to the lung and liver. In general, surgical resection of early lesions is the initial treatment for superficial and peripheral soft tissue LMS. The best surgical treatment for oral LMS, because of its high local recurrence rate and propensity for metastasis, is complete surgical excision (early wide surgical excision) including a composite adequate margin of uninvolved tissue. Long-term follow-up is mandatory.12,13
References 1. Dry SM, Jorgensen JL, Fletcher DMS: Leiomyosarcoma of the oral cavity: An unusual topographic subset easily mistaken for nonmesenchymal tumors. Histopathogy 36:210, 2000 2. Wertheimer-Hatch L, Hatch GF III, Hatch KF, et al: Tumors of the oral cavity and pharynx. World J Surg 24:395, 2000
217 3. Canter LC, Aguirre A, Boyd B, et al: Primary leiomyosarcoma in a 7-year-old girl. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 87:477, 1999 4. Goldschmidt PR, Goldschmidt JD, Eisenberg E: Leiomyosarcoma presenting as a mandibular gingival swelling: A case report. J Periodontol 70:84, 1999 5. Aydin H, Dreyer T: Leiomyosarcoma of the base of the tongue. Treated with radiotherapy: A case report. Oral Oncol 5:351, 1994 6. Christopher RE, Whyte AM: Leiomyosarcoma of the cheek: A case report. Br J Oral Surg 16:100, 1977 7. Enzinger FM, Weiss SW: Soft Tissue Tumor (ed 3). St Louis, MO, Mosby, 1995, pp 491-510 8. Allan GF, Kay S, Province C: Oral leiomyosarcoma—report of a case and review of the literature pertaining to smooth-muscle tumors of the oral cavity. J Oral Surg 143:402, 1977 9. Cawson RA, Binnie WH, Speight P, et al: Tumors of smooth muscle, in Lucas’s Pathology of Tumors of the Oral Tissues (ed 5). London, England, Churchill Livingstone, 1998, pp 319-321 10. Poon CK, Kwan PC, Yin NT, et al: Leiomyosarcoma of gingiva. J Oral Maxillofac Surg 45:888, 1987 11. Das DK, Grover RK, Anand VJ, et al: Oral leiomyosarcoma in child. Acta Cytol 43:1150, 1999 12. Izumi K, Maede T, Cheng J, et al: Primary leiomyosarcoma of maxilla with regional lymph node metastasis. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 80:310, 1995 13. Weitzner S: Leiomyosarcoma of the anterior maxillary alveolar ridge. J Oral Surg 50:62, 1980
Rapidly Growing Mass in Mandibular Gingiva Chieh-Yuan Cheng, DDS,* Kuo-Ming Chang, MD,† Kuo-Wei Chang, DDS, PhD,‡ Chia-Hoa Chang, DDS,§ Chung-Ji Liu, DDS, MS,¶ and George M. Kushner, DMD, MD㛳 Case Presentation
old girl. A previous biopsy revealed fibrous granulation tissue, which is a rather nondescript diagnosis and gives us very limited useful knowledge. One must wonder if a representative sample of the lesion was not obtained or there was a possible misinterpretation or misdiagnosis of the tissue that was sent for pathologic examination. On my review of the provided radiograph, there appears to be bony cratering of the mandible distal to the right mandibular first molar with displacement of the right mandibular second molar posteriorly. There is no mention of paresthesia or alterations in sensation of the mandible. This mass is firm with a rubbery feel and mildly tender to palpation. In establishing a differential diagnosis, we need to look at the broad categories of congenital, developmental, infectious, reactive, and neoplastic processes. I believe we can essentially rule out a congenital or developmental problem in this patient. Under the category of infectious etiology, we still must consider a granulomatous etiology such as histoplasmosis. The granulomatous diseases are known as the “great masqueraders” in the head and neck region because they often present with atypical signs and symptoms. However, this patient has no lymphadenopathy, and this diagnosis would clearly be much lower on my list of possible etiologies. Under the category of “reactive lesions,” one must consider a pyogenic granuloma. The orthodontic appliance or luting cement can easily be an irritant in the oral cavity but the presence of this orthodontic appliance may be totally unrelated to the final diagnosis. The diagnosis of pyogenic granuloma would certainly go along with the fibrous granulation tissue seen on the prior pathology report. However, pyogenic granulomas are usually soft and bleed easily. This mass is firm and has a rubbery feel to palpation. One must also consider a peripheral ossifying fibroma and a peripheral giant cell granuloma as other reactive lesions that could mimic this disease entity. Both of these tumors would certainly feel more solid than a pyogenic granuloma. Under the category of “neoplastic diseases,” at the top of the list is the broad category of fibromatosis.
A 9-year-old girl, with the chief complaint for the painful swelling on the right mandibular gingiva for about 4 weeks, was referred to our Oral and Maxillofacial Surgery Department in December 2000. She had undergone treatment with the lingual holding arch, which was retained by the lower first molar bilaterally as a space maintainer due to the early loss of the lower deciduous molars. After wearing the appliance for a few weeks, an easily bleeding tumor was found just behind the right mandibular first molar. At that time, the clinical impression was irritation-induced pyogenic granuloma so local debridement was performed by the patient’s family dentist. The pathology report was fibrous granulation tissue. One week later, the lesion grew larger with associated pain. She was referred to our Oral and Maxillofacial Surgery Department for further treatment. Physical examination revealed a rubbery tumor on right mandibular gingiva that measured about 4 ⫻ 3 cm with mild tenderness (Fig 1A). No obvious cervical lymphadenopathy was observed. Panoramic film showed a bony resorptive lesion behind the right mandibular first molar with a displaced second molar (Fig 1B).
Differential Diagnosis George M. Kushner, DMD, MD In summary, we are basically dealing with a rather rapidly growing lesion in an otherwise healthy 9-year*Attending Physician, Oral and Maxillofacial Surgery Department, Mackay Memorial Hospital, Taipei, Taiwan. †Attending Physician, Department of Pathology, Mackay Memorial Hospital, Taipei, Taiwan. ‡Professor, Institute of Oral Biology, Yang Ming University, Taipei, Taiwan. §Attending Physician, Pedodontics Department, Mackay Memorial Hospital, Taipei, Taiwan. ¶Attending Physician, Oral and Maxillofacial Surgery Department, Mackay Memorial Hospital, Taipei, Taiwan. 㛳Associate Professor of Oral and Maxillofacial Surgery, Department of Surgical and Hospital Dentistry, School of Dentistry, University of Louisville, Louisville, KY. Address correspondence and reprint requests to Dr Liu: No 92, Sec 2, Chungshan North Road, 104, Taipei, Taiwan; e-mail: [email protected] © 2004 American Association of Oral and Maxillofacial Surgeons
0278-2391/04/6202-0013$30.00/0 doi:10.1016/j.joms.2002.12.004
214
215
CHENG ET AL
try to gather several likely or common lesions within each category to provide a broad base of possible pathologic conditions. At this point, I recommend a generous incisional biopsy with adequate tissue to definitively establish the clinical diagnosis. I certainly prefer that this tissue be read by an oral and maxillofacial pathologist. Final treatment recommendations would be based on the correct clinical diagnosis.
Subsequent Course
FIGURE 1. A, Photograph showing ulcerated tumor (arrow) located in the right mandibular gingiva and measuring about 4 ⫻ 3 cm. B, Panoramic radiograph showing ill-defined radiolucent lesion with mandibular osteolytic change and the second molar pushed away (arrow).
These lesions generally occur in children and can grow rapidly, which would fit our history rather nicely. Juvenile fibromatosis, desmoplastic fibroma, and myofibroma are all potential specific diagnoses within the fibromatosis category. Any one of these diagnoses could fit the clinical scenario as presented. In addition, one should keep the peripheral odontogenic fibroma on the list as a potential neoplasm, although this is a rather less common entity. Unfortunately, the clinician must consider a rapidly growing tumor that is resorbing bone in a young patient as possibly being malignant. The very broad category of sarcomas is a relatively common pediatric malignancy. Sarcomas, including rhabdomyosarcoma, fibrosarcoma, and osteogenic sarcoma could easily have the clinical presentation that has been described. Any one of these lesions could be a rapidly growing lesion that resorbs bone in an otherwise healthy young patient. Paresthesia is a later symptom of these diseases as the inferior alveolar nerve becomes affected with the growth of the lesion. Last, a diagnosis of Langerhans cell/histiocytosis X should be considered. These lesions typically resorb bone, can have exophytic growth and certainly occur in the pediatric population. This clinical entity could certainly fit quite nicely with the clinical presentation as described. In establishing a differential diagnosis, I
Although the previous pathology report was granulation tissue, malignant lesion was highly suspected and surgical excision was suggested. After admission, the surgery was performed under general anesthesia. The frozen section was made at first and reported as spindle-shaped tumor with minimal nuclear atypia and occasional mitoses. The tumor was compositely excised with 1-cm soft tissue margin and marginal mandibular bone. Microscopically, the tumor, composed of spindle cells, was arranged in whorling bundles. Some fibrosis was observed. These fusiform cells exhibited spindle-shaped nuclei with blunted ends. Mitotic figures were common (Fig 2A). The immunostains for smooth muscle actin were all positive on tumor cells and normal smooth muscle of blood vessel wall (Fig 2B). Cytokeratin, factor VIII, S-100, and desmin showed all negative immunoreactivity (Fig 2C).
Pathologic Diagnosis The final diagnosis was low-grade leiomyosarcoma of right mandibular gingiva. The patient has been closely followed up for 18 months and remains disease free, with no evidence of local recurrence or distant metastasis (Fig 3).
Discussion Leiomyosarcoma (LMS) is a smooth muscle neoplasm that rarely occurs in the oral cavity and confirmed only with ultrastructural and immunohistochemical findings. To our knowledge, fewer than 50 cases of oral LMS are reported in the English literature to date.1 Only 0.064% of smooth muscle tumors have an intraoral location.2 Oral LMS usually presents as a mass that may or may not exhibit ulceration. Tooth mobility and pericoronal soft tissue swelling may be the initial presenting features. The possible intraoral sites of origin are included: 1) the circumvallate papillae of the tongue, 2) pluripotential undifferentiated mesenchymal cells, 3) smooth muscle elements in blood vessel walls, 4) neurovascular bundle, 5) erector pilis muscle in aberrant hair follicles in the cheek, and 6) myoepithelial cells of mucous glands.3-6 In our case, gingival LMS likely arises from smooth muscle cells, which invest gingival blood vessels, smooth muscular elements surrounding adjacent glandular ex-
216
GROWING MASS IN MANDIBULAR GINGIVA
FIGURE 2. A, Photomicrograph showing fusiform tumor cells with blunt-ended nuclei with occasional mitotic changes (arrow) (hematoxylin and eosin stain, original magnification ⫻400). B, Photomicrograph showing strong immunoreaction to smooth muscle actin in tumor cells and muscle elements of normal vessel wall (immunostaining for smooth muscle actin, original magnification ⫻200). C, Photomicrograph showing negative immunoreaction to desmin in tumor cells (immunostaining for desmin, original magnification ⫻100).
cretory duct, or perhaps from pluripotential remnants of embryonal mesenchyme. The differential diagnosis of oral LMS should consist of odontogenic tumor, spindle cell carcinoma, other sarcoma, myoepithelioma, melanoma, schwannoma, myofibroma, myofibromatosis, angioleiomyoma, fibrous histocytoma, central giant cell granuloma, and lymphoma. Histopathologic diagnosis of mesenchymal tumors, and those with mesenchymal-like features, can be difficult. Spindle cell carcinoma is often strongly positive for cytokeratin. Myoepithelioma has the shorter and often more slender nuclei with a more regular nuclear contour. Melanoma stains negative for myogenic marker and cytokeratin and positive for S-100 protein. Fibrosarcoma cells tend to be tapered, whereas schwannoma cells are wavy, buckled, and distinctly asymmetric. Typical LMS with interlacing bundles of spindle cells have blunt-ended nuclei, hyperchromatism, cellular pleomorphism, and mitotic activity. LMS stains positive for muscle-specific actin (HHF35) and smooth muscle actin and negative for S-100 protein, cytokeratin, and epithelial membrane antigen. Myofilament and deeply clefted nuclei may be shown on electron microscopy. With cytochemical, immunocytochemical, and ultrastructural evaluation, one can make a specific diagnosis for LMS from other similar lesions.1,3,7-11 The location of LMS is important in determining the prognosis. There are 3 geographic (or regional) groups according to certain clinical and biological differences: 1) LMS of deep soft tissue, 2) LMS of cutaneous and subcutaneous tissue, and 3) LMS of vascular origin.7 The more superficial location of LMS
FIGURE 3. Postoperative clinical appearance (A) and panoramic radiograph (B) showed healthy gingival mucosa with no evidence of recurrence.
CHENG ET AL
results in a better prognosis. The prognosis for patients with oral LMS is guarded, with a recurrence rate that approaches 50% after wide extirpation. Oral LMS tends to metastasize to the cervical nodes and lung, whereas when the tumor originates in other locations, it usually metastasizes to the lung and liver. In general, surgical resection of early lesions is the initial treatment for superficial and peripheral soft tissue LMS. The best surgical treatment for oral LMS, because of its high local recurrence rate and propensity for metastasis, is complete surgical excision (early wide surgical excision) including a composite adequate margin of uninvolved tissue. Long-term follow-up is mandatory.12,13
References 1. Dry SM, Jorgensen JL, Fletcher DMS: Leiomyosarcoma of the oral cavity: An unusual topographic subset easily mistaken for nonmesenchymal tumors. Histopathogy 36:210, 2000 2. Wertheimer-Hatch L, Hatch GF III, Hatch KF, et al: Tumors of the oral cavity and pharynx. World J Surg 24:395, 2000
217 3. Canter LC, Aguirre A, Boyd B, et al: Primary leiomyosarcoma in a 7-year-old girl. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 87:477, 1999 4. Goldschmidt PR, Goldschmidt JD, Eisenberg E: Leiomyosarcoma presenting as a mandibular gingival swelling: A case report. J Periodontol 70:84, 1999 5. Aydin H, Dreyer T: Leiomyosarcoma of the base of the tongue. Treated with radiotherapy: A case report. Oral Oncol 5:351, 1994 6. Christopher RE, Whyte AM: Leiomyosarcoma of the cheek: A case report. Br J Oral Surg 16:100, 1977 7. Enzinger FM, Weiss SW: Soft Tissue Tumor (ed 3). St Louis, MO, Mosby, 1995, pp 491-510 8. Allan GF, Kay S, Province C: Oral leiomyosarcoma—report of a case and review of the literature pertaining to smooth-muscle tumors of the oral cavity. J Oral Surg 143:402, 1977 9. Cawson RA, Binnie WH, Speight P, et al: Tumors of smooth muscle, in Lucas’s Pathology of Tumors of the Oral Tissues (ed 5). London, England, Churchill Livingstone, 1998, pp 319-321 10. Poon CK, Kwan PC, Yin NT, et al: Leiomyosarcoma of gingiva. J Oral Maxillofac Surg 45:888, 1987 11. Das DK, Grover RK, Anand VJ, et al: Oral leiomyosarcoma in child. Acta Cytol 43:1150, 1999 12. Izumi K, Maede T, Cheng J, et al: Primary leiomyosarcoma of maxilla with regional lymph node metastasis. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 80:310, 1995 13. Weitzner S: Leiomyosarcoma of the anterior maxillary alveolar ridge. J Oral Surg 50:62, 1980