984

5. 6.

7. 8.

9.

10.

LIPOSARCOMA:

lymphadenopathies and lymphomas with primary salivary gland presentation. Lab Invest 56:33A, 1987 Ioachim HL, Ryan JR, Blaugrund SM: Salivary gland lymph nodes. Arch Path01 Lab Med 112: 1224, 1988 Ioachim HL, Cooper MC, Hellman GC: Lymphomas in men at high risk for acquired immune deficiency syndrome (AIDS). Cancer 56~2831, 1985 Puterman M, Goldstein J: Primary lymph nodal Kaposi’s sarcoma of the parotid gland. Head Neck Surg 5:535, 1983 Yeh C-K, Fox PC, Fox CH, et al: Kaposi’s sarcoma of the parotid gland in acquired immunodeficiency syndrome. Oral Sure Oral Med Oral Path01 67:308. 1989 Holliday R& Cohen WA, Schinella RA, et al: Benign lymphoepithelial parotid cysts and hyperplastic cervical adenopathy in AIDS-risk patients: A new CT appearance. Radiology 168:439, 1988 Poletti A, Manconi R, Volpe R, et al: Study of AIDS-related

11.

12. 13.

14.

15. 16.

CASE REPORT AND LITERATURE

REVIEW

lymphadenopathy in the intraparotid and perisubmaxillary gland lymph nodes. J Oral Path01 17:164, 1988 Smith F, Rajdeo H, Panesar N, et al: Benign lymphoepithelial lesion of parotid gland in intravenous drug users. Lab Invest 56:74a, 1987 Persistent generalized lymphadenopathy among homosexual males. MMWR 31:249, 1979 Fujibayash T, Hoh H: Lymphoepithelial (so-called branchial) cyst within the parotid gland: Report of a case and review of the literature. Int J Oral Surg 10:283, 1981 Morris MR, Moore DW, Shearer GL: Bilateral multiple benign lymphoepithelial cysts of the parotid gland. Otolaryngol Head Neck Surg 97:87, 1987 Bemier JR, Bhaskar SN: Lymphoepithelial lesions of the salivary glands. Cancer 11:1156, 1958 Cohen, MN, Rao Y, Shedd DP: Benign cysts of the parotid gland. J Surg Oncol 27:85, 1984

J Oral Maxillofac Surg 4s:9s4-9tls.1990

Liposarcoma: Report of a Case and Review of the Literature GINNY EIDINGER, DMD,* NICK KATSIKERIS, DDS, DR DENT,t PATRICK GULLANE, MD, FRCS(C)S

Liposarcoma is one of the most common malignant mesenchymal neoplasms, comprising approximately 15% of all soft-tissue sarcomas.“’ First described by Virchow in 1857, it has been extensively reported in the literature, although its incidence remains exceedingly rare in the head and neck region. 3 The purpose of this article is to report an additional case of liposarcoma of the buccal mucosa and to review the literature. Report of a Case In November to a peripheral

1983, an 80-year-old treatment

center

white man presented with a complaint of a

Received from Toronto General Hospital, Toronto, Ontario, Canada. * Chief Resident, Department of Oral and Maxillofacial Surgery. t Assistant Professor, Faculty of Dentistry, University of Toronto; Staff Oral Surgeon, Toronto General Hospital. $ Associate Professor, Department of Otolaryngology, University of Toronto; Otolaryngologist-in-Chief, Toronto General Hospital. Address correspondence and reprint requests to Dr Eidinger: Department of Oral and Maxillofacial Surgery, Toronto General Hospital, 124 Edward St, Toronto, Ontario MSG IG6, Canada. 0 1990 geons.

American

Association

0278-2391/9014809-0014$3.0010

of Oral

and Maxillofacial

Sur-

AND

rapidly growing mass of the left cheek of 2 months’ duration. Microscopic examination of the mass following excision revealed a pleomorphic liposarcoma. One month later the patient returned for additional treatment necessitated by recurrence of the mass, which at this point measured 8 x 8 x 5 cm. Subsequently, five excisions were performed but each was followed by recurrence. A Zl-day course of radiotherapy totaling 2,400 rad was then administered. The mass remained the same size for 8 months until September 1984, when further growth was noted. Radioactive gold implants delivering an estimated 6,000 rad were then inserted. After minimal response to radiation, the patient was referred to Toronto General Hospital in December 1984 for further assessment and treatment. The patient’s past medical history included insulindependent diabetes mellitus and controlled hypertension. Clinical examination showed an elderly-appearing man with a disfiguring mass of the left cheek measuring approximately 8 x 8 x 5 cm (Fig 1). Cervical or supraclavicular lymph nodes could not be palpated. Routine preoperative laboratory studies, electrocardiogram, and chest radiographs were within the range of normal. Serial axial computerized tomography (CT) of the neck revealed a superficial soft-tissue mass extending from the level of the midmaxillary sinus to the inferior border of the mandible. Soft-tissue extension into the left parotid gland, and encroachment into the left infratemporal fossa was observed (Fig 2). There was no evidence of bony destruction on the CT scan. On December 13, 1984, a tracheostomy and composite resection of the mass and parotid gland were performed, with immediate reconstruction of the soft-tissue defect using a pectoralis major myocutaneous flap and an amni-

EIDINGER ET AL

985

FIGURE 1. Preoperative frontal view of lesion of left cheek.

otic membrane graft for closure of the oral defect (Figs 3 and 4) A bilateral suprahyoid neck dissection was also performed. Gross examination of the surgical specimen revealed a well-circumscribed multilobulated yellow mass, with multiple areas of hemorrhage and necrosis. The overlying skin was not ulcerated, although the buccal

FIGURE 2. Preoperative CT scan showing tumor of the left parotid gland with left infratemporal fossa extension.

mucosa on the medial aspect of the specimen was adherent to the underlying mass. Microscopic examination confirmed the diagnosis of a poorly differentiated pleomorphic liposarcoma (Fig 5). High-power light microscopy demonstrated a haphazard mixture of neoplastic fat cells at various stages of differentiation, ranging in size from small preadipocytes to multinucleated cells. Varying quantities of intracytoplasmic lipid material was observed adjacent to cells displaying an eosinophilic cytoplasm. Mitotic figures were frequently observed, another feature typical of pleomorphic liposarcoma. A vascular connective tissue stroma with sparse cellularity was present. Re-

FIGURE 3. Intraoperative view of surgically created defect.

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LIPOSARCOMA:

CASE REPORT AND LITERATURE

REVIEW

FIGURE 4. Intraoperative lateral view of soft tissue reconstruction with pectoralis major myocutaneous flap.

g&d

lymph nodes, muscle, parotid gland, and surgical resection margins were negative for malignancy. One day postoperatively, the patient developed a large subpedicle hematoma that required exploration and evacuation under general anesthesia. In January 1985, the pa-

tient was brought to the operating room for debridement of the pectoralis major flap which had undergone partial necrosis, and creation of a controlled orocutaneous fistula which was closed with a left deltopectoral flap that was divided in March 1985. The patient was discharged from hospital in April 1985in excellent condition. Persis-

Factors influencing differentiation are not entirely clear, but quantity and distribution of adipose tissue may be related to hormonal, nutritional, and genetic factors. Hajdu suggests that “adipose cells may arise from undifferentiated fibroblasts and histiocytes, as well as from primitive mesenchymal cells

tent drooling and ptosis of the left comer of the mouth were secondary to sacrifice of branches of the facial nerve during parotidectomy. At follow-up since 1985, the patient has been free of recurrent disease (Figs 6 and 7).

Discussion Adipose tissue constitutes approximately 15% to 25%of body weight. It may be divided functionally and morphologically into brown and white types.

FIGURE 5. Photomicrograph of tumor specimen demonstrating features of pleomorphic liposarcoma. (Hematoxylin-eosin stain. Original magnification X lOa.)

FIGURE 6.

Postoperative

frontal view.

EIDINGER

987

ET AL

symptoms vary according to the area involved and size of tumor, and may include compression of nerves and vessels manifested by paresthesia and ischemia. l5 Rate of growth may vary from slow and indolent to rapid and aggressive, with local invasion and widespread dissemination.5 Liposarcoma rarely occurs before the age of 30, with the median age of presentation in the sixth decade of life.‘,3,9*14 The literature suggests a slight male predilection 2,6,9,12,16

FIGURE 7. deficit.

Postoperative

frontal view illustrating facial nerve

that are abundant around blood vessels.“4 The prolipoblast is the first stem cell identified from this cell line, followed by the lipoblast, preadipocyte, and adipocyte. Each of these cell types may be predominate within the subgroups of liposarcoma. Prolipoblasts are predominant in fibroblastic liposarcoma, whereas the round cell or lipoblastic variant is composed mostly of lipoblasts. Preadipocytes predominate in myxoid liposarcomas, and adipocytes may be identified in well-differentiated tumors. Ceils from all four cell lines may be identified in pleomorphic liposarcomas. Hajdu also suggests that as the proportion of intracytoplasmic fat globules diminishes and the pleomorphism of tumor cells and number of abnormal mitoses increases, so does the grade of malignancy.4 Liposarcoma occurs most frequently in the lower extremities, including the medial thigh, popliteal fossa, buttocks, and retroperitoneum, although it may occur wherever fat tissue is present.3,5-‘2 In 1979, Saunders et al’ reviewed 25 cases of liposarcoma of the head and neck while reporting an additional 4 cases. To date, approximately 40 cases of liposarcoma of the head and neck have been reported. The orbit, scalp, and phamyx are the most common extraoral sites, while the buccal mucosa and floor of mouth are the principal intraoral sites.‘.3,5.1’,‘3,14 Deep tissues give rise to malignant lesions, wheres lipomas generally arise superficially within subcutaneous tissue.5Y12 Liposarcoma is thought to arise de novo, not from sarmatous degeneration of a lipoma.2*5*7,9*‘4 The clinical course of liposarcoma is extremely variable. It generally begins as an inconspicuous, progressively enlarging, painless, mass. Signs and

Microscopic classification of liposarcoma has undergone several revisions since the tumor was first described. Robinson was the first to classify liposarcoma into two subgroups in 1916,17 and in 1926, Jaffe separated them into three categoties.18 Stout’s classification followed in 1944 and remains the most widely used system. 13*19Pack and Pierson modified Stout’s classification in 1954,12 and Enterline et al provided their own modifications in 1960.7 Enzinger and Winslow and the World Health Organization then divided liposarcoma into five subgroups,15 and recently, Kindblom et al proposed a six-subtype grading system.20 In 1977, Russell and coworkers proposed a grading and TMN staging system that has been adopted by the International Union Against Cancer.” The AFIP (Armed Forces Institute of Pathology) classification, used at our institution, includes four categories: 1) myxoid liposarcoma; 2) round cell liposarcoma; 3) well-differentiated liposarcoma, subclassified into lipoma-like, sclerosing, inflammatory, and dedifferentiated; and 4) pleomorphic liposarcoma.22 Treatment of liposarcoma varies among centers. Wide surgical excision with postoperative radiotherapy remains the treatment of choice as evidenced by improved 5- and lo-year survival rates when both treatment modalities are combined.6,12*‘6,23 According to Pack and Pierson, 5year survival rates are improved from 50% to 87.5% with combined therapy.12 Spittle et al reported a 64% 5-year survival rate in their series,16 and Evans stated that surgical excision combined with 6,000 to 7,000 rad of radiation significantly decreases recurrence when compared with excision alone.23 Excision of the pseudoencapsulated tumor leads to increased recurrence; therefore, it is critical that wide excision with tumor-free margins be obtained. l2 Enterline and others have suggested that recurrence is directly related to tumor size as larger tumors have an increased tendency to be invasive.7*23 Muscle compartment resections are recommended when feasible, and amputation is advocated in cases where neither wide surgical excision or radiotherapy are considered appropriate treatment options.8*‘2*‘3 The myxoid variant is the most common micro-

988

LIPOSARCOMA:

scopic form involving the head and neck, followed by the round cell, and pleomorphic cell variants.13 According to Enzinger and Enterline, myxoid and well-differentiated liposarcomas have a better prognosis than the pleomorphic or round cell variants.‘,15 This is not surprising as the more undifferentiated the tumor, with increased mitoses, the more aggressive the biologic behavior. A combination of these factors leads to a poorer prognosis for some patients. l6 Retroperitoneal tumors are associated with a poorer prognosis than tumors of the extremities. l6 Prognosis is unaffected by gender, although overall prognosis is improved if affected individuals are under the age of 50.* Chemotherapy has yet to be established as an effective adjuvant treatment.8’20 Metastasis of liposarcoma occurs most frequently to the lungs.23 The myxoid variant comprises approximately 70% of liposarcomas in children and is also associated with the best longterm prognosis.24-27 Lipoblastomatosis in the pediatric population must be distinguished from myxoid liposarcoma, as they may have a similar microscopic appearance. 1624 Microscopic differential diagnosis should also include myxoma, myxosarcoma, benign fatty tumors, including lipoma and hibernoma, angiolipoma, fibrolipoma, pseudosarcomatous fasciitis, and malignant fibrous histiocytoma.7,10,25

8. 9.

10. 11.

12. 13. 14. 15. 16. 17. 18.

19.

Summary

20.

Liposarcoma of the head and neck region is extremely rare. An additional case of pleomorphic liposarcoma of the buccal mucosa is reported, representing an even rarer combination. The importance of combined radiation and radical surgical therapy as a means of improving survival is emphasized.

21.

References

24.

22. 23.

25. 1. Saunders JR, Darrell AJ, Casterline PF, et al: Liposarcoma of the head and neck: A review of the literature and addition of four cases. Cancer 43:162, 1979 2. Dahl EC, Hammond HL, Sequeira E: Liposarcoma of the head and neck. J Oral Maxillofac Surg 40:674, 1982 3. Baden E, Newman R: Liposarcoma of the oropharyngeal

26. 27.

CASE REPORT AND LITERATURE

REVIEW

region: Review of the literature and report of two cases. Oral Sure. Oral Med Oral Path01 44:889. 1977 Hajdu SI: Pathology of Soft Tissue Turnours. Philadelphia, PA, Lea & Febiger, 1979 Amariit S. Sineh A. Naeoal BL. et al: Lioosarcoma of the cheek. J Or2 Surg 36311, 1978 A Ramon Y, Horowitz Y, Oberman M, et al: Liposarcoma of the buccal mucosa. Int J Oral Surg 6:226, 1977 Enterline HT, Culberson JD, Rochlin DB, et al: Liposarcoma: A clinical and pathologic study of 53 cases. Cancer 13:932, 1960 Reitan JB, Kaalhas 0, Brennhovd IO, et al: Prognostic factors in liposarcoma. Cancer 55:2482, 1985 Hudson C, Cove P, Adekeye EO: Liposarcoma of the head and neck: Report of a case and review of the literature. J Oral Surg 36:380, 1978 Jones JK, Baker HW: Liposarcoma of the parotid gland. Arch Otolaryngol 106:497, 1980 Kindblom LG. Annervall L. Jarlstedt J: Lioosarcoma of the neck: A clinicop~thologi~ study of 4 cases. Cancer 42~774, 1978 Pack GT, Pierson JC: Liposarcoma: A study of 105 cases. Surgery 36:687, 1954 Arlen M, Marcove RC: Surgical Management of Soft Tissue Sarcomas. St Louis, MO, Mosby, 1987 Suzuki H, Nagayama M, Kaneda T, et al: Liposarcoma of the cheek in an infant. J Oral Maxillofac Surg 42: 180, 1984 Enzinger FM, Winslow DJ: Liposarcoma: A-study of 103 cases. Virch Arch Path01 Anat 335:367. 1962 Spittle MF, Newton KA, Mackenzie DHI Liposarcoma: A review of 60 cases. Br J Cancer 24:696, 1970 Robinson HE: Lipoma myxomatodes. J Med Res 35: 131, 1916 Jaffe RH: Recurrent lipomatous tumour of the groin. Liposarcoma and lipoma pseudomyxomatodes. Arch Path01 1:381, 1926 Stout AP: Liposarcoma-Malignant tumour of lipoblasts. Ann Surg 119:86, 1944 Kindblom LG, Angervall L, Svendson P: Liposarcoma: A clinical, radiographic, and prognostic study. Acta Path01 Microbial Stand 253:3, 1975 (suppl A) Russell WO, Cohen J, Ehzinger F, et al: A clinical and pathological staging system for soft tissue sarcomas. Cancer 40: 1562, 1977 Weiss S, Enzinger FM: Soft Tissue Tumours. St Louis, MO, Mosby, 1983 Evans HL: Liposarcoma: A study of 55 cases with a reassessment of its classification. Am J Surg Path01 3:507, 1979 Chung EB, Enzinger FM: Benign lipoblastomatosis: An analysis of 35 cases. Cancer 32~482, 1973 Schmookler BM, Enzinger FM: Liposarcoma occurring in children: An analysis of 17 cases and review of the literature. Cancer 52:567, 1983 Kaufman SL, Stout AP: Lipoblastic tumours of children. Cancer 12:912, 1959 William JP, Hazra T: Retroperitoneal liposarcoma in a child. Urology 7:89, 1976

Liposarcoma: report of a case and review of the literature.

Liposarcoma of the head and neck region is extremely rare. An additional case of pleomorphic liposarcoma of the buccal mucosa is reported, representin...
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