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Gingival liposarcoma: An unusual polyp
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British Journal of Oral and Maxillofacial Surgery 46 (2008) 150–151
Short communication
Gingival liposarcoma: An unusual polyp W.Y. Chan a , I.D.S. McHenry a , L.M. Carter a,∗ , G. Reall b , C.J. Wales a a b
Department of Oral & Maxillofacial Surgery, York District Hospital, Wiggington Road, York YO31 8HE, United Kingdom Histopathology Department, York District Hospital, Wiggington Road, York YO31 8HE, United Kingdom
Accepted 20 December 2006 Available online 5 February 2007
Abstract Liposarcomas account for up to 20% of all soft tissue tumours. They are uncommon in the head and neck. Sarcomas of the oral region (excluding lymphoma) account for 5% of all oral cancers, of which 10% are liposarcomas. Fewer than 80 cases have been reported in the world literature, most of which presented as expansile masses. The prognosis of liposarcoma is dependant on the histopathologic type, location, and adequacy of surgical treatment. Well-differentiated types have a good prognosis and minimal metastatic potential. We report a case of well-differentiated liposarcoma (lipoma-like subtype), that presented as a gingival polyp. © 2007 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Keywords: Liposarcoma; Gingiva; Polyp; Oral
Case report A 58-year-old man presented with an asymptomatic soft polyp 1 cm in diameter on the anterior mandibular alveolus at the level of the mucogingival line adjacent to the lower right central incisor. He had had it for nine months, and it looked benign. Histological examination of an excisional biopsy suggested a well-differentiated liposarcoma. The tumour was composed of adipocytes of varying sizes, with intervening atypical stromal cells and hyperchromatic, pleomorphic nuclei (Fig. 1). Some of the adipocytes showed focal multivacuolation and nuclear scalloping, both features of which are typical of lipoblasts (Fig. 2). Lateral and deep margins had been invaded and radiographs showed no bony erosion. The Regional Sarcoma Panel confirmed the diagnosis of a well-differentiated liposarcoma, lipoma-like subtype. After a multidisciplinary discussion, we did a further wide local excision, which entailed resection of the anterior rim of the mandible en-bloc including the lower right canine to the lower left central incisor. He recovered well, and no ∗
Corresponding author. Tel.: +44 7795 438086. E-mail address: [email protected] (L.M. Carter).
evidence of liposarcoma was found in the specimen. No loco-regional recurrence has been detected one-year after resection. Discussion We know of no previous case of an oral liposarcoma that presented as a gingival polyp. Liposarcoma is a malignant growth of fat cells and was first described by Virchow in 1857. It most commonly presents in the deep soft tissues of the retroperitoneum, lower limb, and shoulder,1 and fewer than 1% present intraorally.2–4 Of these the most common is in the buccal mucosa, and other sites include floor of the mouth, tongue, and maxillary gingival mucosa. They usually present as an expanding mass with or without compression or invasion of adjacent structures.2,5,6 Most liposarcomas present between the ages of 40 and 60 years, and they rarely occur before the age of 30. There is a slight male predilection.2–5 The World Health Organization (WHO) divided liposarcomas into five main types: well-differentiated, myxoid, round cell, pleomorphic, and dedifferentiated.7 Histopathological diagnosis is dependent on the presence of lipoblasts that are distinct from other cells with vacuolar degeneration, tumours containing mucin or
0266-4356/$ – see front matter © 2007 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
doi:10.1016/j.bjoms.2006.12.006
W.Y. Chan et al. / British Journal of Oral and Maxillofacial Surgery 46 (2008) 150–151
Fig. 1. Adipocytes of varying sizes (haematoxylin and eosin, original magnification ×20).
151
the most common types and have favourable prognoses.2 They tend to be poorly-circumscribed and recur locally after incomplete excision. Metastases are rare, but repeated local recurrences may cause tumours to dedifferentiate in about 10% of cases, which results in a worse prognosis and a higher risk of metastases.5,6 Accurate histopathological diagnosis is essential for appropriate treatment. The differential diagnoses must include intramuscular myxoma, spindle cell or pleomorphic lipoma, lipogranuloma, and glioblastoma. Wide local excision with or without postoperative radiotherapy results in the best survival rate,3,6,8 but this may be challenging within the oral and maxillofacial region if one considers the aesthetic and functional implications. We suggest that all cases of liposarcoma should be discussed with a multidisciplinary team and the Regional Sarcoma Panel.
References
Fig. 2. Multivacuolation and nuclear scalloping of lipoblasts (haematoxylin and eosin, original magnification ×40).
glycogen, variation in the size of cells and atypical nuclei. There is considerable microscopic variability, and the biological behaviour is dependent on the exact histopathological appearance.3,5,6 Our patient had a well-differentiated liposarcoma (lipomalike subtype), which together with myxoid liposarcomas are
1. Piattelli A, Di Alberti L, Favia GF. Liposarcoma involving the periodontal tissues. A case report. J Periodontol 2000;71:322–4. 2. Nascimento AF, McMenamin ME, Fletcher CD. Liposarcoma/atypical lipomatous tumors of the oral cavity: a clinicopathologic study of 23 cases. Ann Diagn Pathol 2002;6:83–93. 3. Capodiferro S, Scully C, Maiorano E, Lo Muzio L, Favia G. Liposarcoma circumscriptum (lipoma-like) of the tongue: report of a case. Oral Dis 2004;10:398–400. 4. Gagari E, Kabani S, Gallagher GT. Intraoral liposarcoma: case report and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2000;89:66–72. 5. Eidinger G, Katsikeris N, Gullane P. Liposarcoma: report of a case and review of the literature. J Oral Maxillofac Surg 1990;48:984–8. 6. Nikitakis NG, Lopes MA, Pazoki AE, Ord RA, Sauk JJ. MDM2+/CDK4+/p53+ oral liposarcoma: case report and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2001;92:194–201. 7. Fletcher CD, Unni KK, Mertens F. Adipocytic tumours. In: World Health Organization classification of tumors: pathology and genetics of soft tissue and bone. Lyon: ARC Press; 2002. p. 35–46. 8. Zheng JW, Wang Y. Liposarcoma in the oral and maxillofacial region: an analysis of 10 consecutive patients. J Oral Maxillofac Surg 1994;52:595–8.
British Journal of Oral and Maxillofacial Surgery 46 (2008) 150–151
Short communication
Gingival liposarcoma: An unusual polyp W.Y. Chan a , I.D.S. McHenry a , L.M. Carter a,∗ , G. Reall b , C.J. Wales a a b
Department of Oral & Maxillofacial Surgery, York District Hospital, Wiggington Road, York YO31 8HE, United Kingdom Histopathology Department, York District Hospital, Wiggington Road, York YO31 8HE, United Kingdom
Accepted 20 December 2006 Available online 5 February 2007
Abstract Liposarcomas account for up to 20% of all soft tissue tumours. They are uncommon in the head and neck. Sarcomas of the oral region (excluding lymphoma) account for 5% of all oral cancers, of which 10% are liposarcomas. Fewer than 80 cases have been reported in the world literature, most of which presented as expansile masses. The prognosis of liposarcoma is dependant on the histopathologic type, location, and adequacy of surgical treatment. Well-differentiated types have a good prognosis and minimal metastatic potential. We report a case of well-differentiated liposarcoma (lipoma-like subtype), that presented as a gingival polyp. © 2007 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Keywords: Liposarcoma; Gingiva; Polyp; Oral
Case report A 58-year-old man presented with an asymptomatic soft polyp 1 cm in diameter on the anterior mandibular alveolus at the level of the mucogingival line adjacent to the lower right central incisor. He had had it for nine months, and it looked benign. Histological examination of an excisional biopsy suggested a well-differentiated liposarcoma. The tumour was composed of adipocytes of varying sizes, with intervening atypical stromal cells and hyperchromatic, pleomorphic nuclei (Fig. 1). Some of the adipocytes showed focal multivacuolation and nuclear scalloping, both features of which are typical of lipoblasts (Fig. 2). Lateral and deep margins had been invaded and radiographs showed no bony erosion. The Regional Sarcoma Panel confirmed the diagnosis of a well-differentiated liposarcoma, lipoma-like subtype. After a multidisciplinary discussion, we did a further wide local excision, which entailed resection of the anterior rim of the mandible en-bloc including the lower right canine to the lower left central incisor. He recovered well, and no ∗
Corresponding author. Tel.: +44 7795 438086. E-mail address: [email protected] (L.M. Carter).
evidence of liposarcoma was found in the specimen. No loco-regional recurrence has been detected one-year after resection. Discussion We know of no previous case of an oral liposarcoma that presented as a gingival polyp. Liposarcoma is a malignant growth of fat cells and was first described by Virchow in 1857. It most commonly presents in the deep soft tissues of the retroperitoneum, lower limb, and shoulder,1 and fewer than 1% present intraorally.2–4 Of these the most common is in the buccal mucosa, and other sites include floor of the mouth, tongue, and maxillary gingival mucosa. They usually present as an expanding mass with or without compression or invasion of adjacent structures.2,5,6 Most liposarcomas present between the ages of 40 and 60 years, and they rarely occur before the age of 30. There is a slight male predilection.2–5 The World Health Organization (WHO) divided liposarcomas into five main types: well-differentiated, myxoid, round cell, pleomorphic, and dedifferentiated.7 Histopathological diagnosis is dependent on the presence of lipoblasts that are distinct from other cells with vacuolar degeneration, tumours containing mucin or
0266-4356/$ – see front matter © 2007 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
doi:10.1016/j.bjoms.2006.12.006
W.Y. Chan et al. / British Journal of Oral and Maxillofacial Surgery 46 (2008) 150–151
Fig. 1. Adipocytes of varying sizes (haematoxylin and eosin, original magnification ×20).
151
the most common types and have favourable prognoses.2 They tend to be poorly-circumscribed and recur locally after incomplete excision. Metastases are rare, but repeated local recurrences may cause tumours to dedifferentiate in about 10% of cases, which results in a worse prognosis and a higher risk of metastases.5,6 Accurate histopathological diagnosis is essential for appropriate treatment. The differential diagnoses must include intramuscular myxoma, spindle cell or pleomorphic lipoma, lipogranuloma, and glioblastoma. Wide local excision with or without postoperative radiotherapy results in the best survival rate,3,6,8 but this may be challenging within the oral and maxillofacial region if one considers the aesthetic and functional implications. We suggest that all cases of liposarcoma should be discussed with a multidisciplinary team and the Regional Sarcoma Panel.
References
Fig. 2. Multivacuolation and nuclear scalloping of lipoblasts (haematoxylin and eosin, original magnification ×40).
glycogen, variation in the size of cells and atypical nuclei. There is considerable microscopic variability, and the biological behaviour is dependent on the exact histopathological appearance.3,5,6 Our patient had a well-differentiated liposarcoma (lipomalike subtype), which together with myxoid liposarcomas are
1. Piattelli A, Di Alberti L, Favia GF. Liposarcoma involving the periodontal tissues. A case report. J Periodontol 2000;71:322–4. 2. Nascimento AF, McMenamin ME, Fletcher CD. Liposarcoma/atypical lipomatous tumors of the oral cavity: a clinicopathologic study of 23 cases. Ann Diagn Pathol 2002;6:83–93. 3. Capodiferro S, Scully C, Maiorano E, Lo Muzio L, Favia G. Liposarcoma circumscriptum (lipoma-like) of the tongue: report of a case. Oral Dis 2004;10:398–400. 4. Gagari E, Kabani S, Gallagher GT. Intraoral liposarcoma: case report and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2000;89:66–72. 5. Eidinger G, Katsikeris N, Gullane P. Liposarcoma: report of a case and review of the literature. J Oral Maxillofac Surg 1990;48:984–8. 6. Nikitakis NG, Lopes MA, Pazoki AE, Ord RA, Sauk JJ. MDM2+/CDK4+/p53+ oral liposarcoma: case report and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2001;92:194–201. 7. Fletcher CD, Unni KK, Mertens F. Adipocytic tumours. In: World Health Organization classification of tumors: pathology and genetics of soft tissue and bone. Lyon: ARC Press; 2002. p. 35–46. 8. Zheng JW, Wang Y. Liposarcoma in the oral and maxillofacial region: an analysis of 10 consecutive patients. J Oral Maxillofac Surg 1994;52:595–8.