I
Liposarcoma
1
I
of the tongue: a case report and review of the literature
P. G. Guest Depurtment
of Oral Surgery,
John
Radcliffe Hospital,
Oxford,
OX3 9DlJ
SUMMARY. A case of the rare intraoral liposarcoma which presented as a lump on the tongue of 5 years duration is reported. The diffkulty of accurate clinical and histopathological diagnosis of this lesion is outlined.
INTRODUCTION
Liposarcomas of the oropharyngeal region are rare (O’Day et al., 1964; Pack & Pierson, 1954). Fewer than 100 cases have been reported. Liposarcomas may arise wherever there is adipose tissue normally present but the majority occur retroperitoneally and on the extremities (Enzinger & Winslow, 1962; Stout & Lattes, 1967). They account for 20% of all malignant soft tissue tumours (Stout & Lattes, 1967; Lattes, 1968). The head and neck region accounts for less than 4% of all sites affected (Enterline et al., 1960; Enzinger & Winslow, 1962; Enzinger, 1965). Unlike most sarcomas they are slow growing tumours with a broad spectrum of cellular differentiation other than the typical anaplasia and in the majority of cases they follow a relatively benign course. Liposarcomas are seen most commonly in the 5th and 7th decades of life and are slightly more common in males, in contrast with the benign lipomas which are common in females (Pack & Pierson. 1954; Enterline et al., 1960; Enzinger & Winslow, 1962; Spittle et al., 1970; Baden & Newman, 1977). Isolated examples of malignant transformation of a pre-existing lipoma have been reported (Correia, 1956; Kapur, 1968) and the lipoma has even been described as a potentially malignant lesion (Sawyer et al.. 1968). The head and neck region is relatively rarely affected and accounts for less than 5% of all sites (Pack & Pierson. 1954; Enterline et al., 1960; Enzinger & Winslow, 1962: Sawyer et al., 1968; Baden & Newman, 1977). Intraoral liposarcomas are even more rare (Pack & Pierson, 1954; ODay et al., 1964; Baden & Newman. 1977) but the benign intraoral lipoma is relatively much more common (Greer & Richardson, 1973).
Fig. I-Lesionat
lateral horderofthe tongue.
Fig. 2- Photomicrograph showing the features of a liposarcoma. (H & E. Original magnification~400).
sarcoma. It consisted of a mixture of adult fat cells and immature spindle shaped lipoblasts with a mucoid stroma (Fig. 2). It has been completely excised. The operative site healed uneventfully and at 2 years postoperatively there was no sign of recurrence of the tumour.
DISCUSSION
Case report
Liposarcoma is an extremely rare intraoral tumour and may present difficulty with diagnosis both clinically and histopathologically. The difficulty of distinguishing between a benign lipoma and a well differentiated liposarcoma has been commented upon by many authors (Kindblom rt ul.. 1~5; Hajdu. 1979: Evans c!t ~1.. 1979). Although the benign lipoma is said to have a characteristic clinical
A 71-year-old medical practitioner presented with a lump on his tongue which had been slowly increasing in size ovc‘r a S-year-period. It was not painful, not ulcerated and had never bled. Clinical examination revealed a firm yellowish lesion on the left lateral border of his tongue which was about 1 cm in diameter (Fig. I). The lesion was excised under local anaesthcsia and subsequent histological examination revealed it to be a well differentiated myxoid lipo26X
Liposarcoma
appearance, the only certain method of determining the exact nature of the tumour is by histopathological examination (Correll et al., 1982). On clinical examination liposarcomas tend to be more firm, less easily compressed and they may appear to be fixed to adjacent tissues (Westcott & Correll, 1984). They usually present as a painless mass or swelling and grow steadily but not rapidly. The silent, slow growth, especially in a submucosal or deep location, circumscription and firm texture often lead to the misdiagnosis of a cyst or a benign soft tissue neoplasm. At operation the liposarcoma is less yellow, less lobulated and less soft than normal adipose tissue or a lipoma (Hajdu, 1979). Histologically there is great diversity in the microscopic features and this has led in turn to various methods of classification and although outside the scope of this article a basic classification of myxoid, round ceil or plcomorphic types can be used. A review of the histological classifications is available elsewhere (Hajdu, 1979). The differential diagnosis must consider several lesions which may mimic the histological appearance of a liposarcoma, such as atypical lipoma, malignant fibrous xanthoma, myxoma, malignant mesenchymoma and embryonal rhabdomyosarcoma. Immediate good fixation and good biopsy sampling which includes all margins of the lesion are essential for the accurate evaluation of liposarcomas (Badcn & Newman, 1977). The most common intraoral sites of liposarcomas are the check and floor of the mouth (Badcn & Newman, 1977). The relatively few cases reported in the literature make any attempt to assess the biological behaviour of liposarcomas extremely difficult but there appears to be some correlation with the histological type (Baden 8r Newman., 1977). The well differentiated myxoid liposarcomas rarely metastasise, whereas the poorly differentiated tumours in adults metastasise in 40% of cases, primarily to lungs, pleura, liver and brain (Stout & Lattes, 1967). In contrast, the well differentiated liposarcomas do not appear to metastasise but may infiltrate locally (Das Gupta, 1973; Westcott & Correll, 1984). The correct treatment for liposarcomas is surgical excision with the line of excision 2 cm away from the tumour (Evans et al., 1979). Inadequate excision predisposes to local recurrences of the tumour (Das Gupta, 1973) and a case with multiple recurrences in the tongue during a 37-year period has been reported (Westcott & Correll, 1984). Any recurrence of a benign lipoma must be viewed with suspicion of the original diagnosis in view of the difficult histopathological differentiation between benign and malignant tumours. In addition. the possibility of malignant transformation of a lipoma makes it very important that patients with such lesions should be followed up closely and perhaps all should be excised. However the risk of malignant transformation is exceedingly small and the clinical course of the liposarcoma itself is relatively benign. It may well be that liposarcomata are more common than they are reported in view of
of the tongue
261)
the difficulty of diagnosis. In a review of cases designated Grade I liposarcoma it was determined that some of the neoplasms designated as well differentiated liposarcoma should have been called atypical lipoma because of their benign behaviour without any tendency to recur (Evans et al., 1979.) References Badcn.E.&Newman, oropharyngeal
R. (lY77). Liposarcomaofthe region. OralSurgrry, Oral Medicine, Oral
I’ulhology~ 44,889. Corrcia,P.D.(3956). Recurrent lipomaofthefloorofthe mouth. Revkra Puuksra DeMedicina(Sau Paulo),49,2XI. Correll, R. W., Wcstcott. W. B. &Pottcr. D. M. (1982). Non-ulcerated sessilc nodulcofthe lower lip./ourna/ofrhe
American DenralAxsociafion,
104,206.
DasGupta,‘l‘. (lY73). Managemcntofsoft tissuesarcomas. Surgery, ~ynaeculo~yat~dOh.steiric:s, 137,1012. Entcrline, H. T., Culberson, J. D.. Rochlin. 1). B.&Brady. L. W. (1960). I,iposarcoma: aclinicalandpathologicalstudy ofS3cascs. Cancer, 13,932. Enzinger, F. F.&Winslow. 1). J. (lY62). Liposarcoma. Astudy of 103cases. Virchow’s Archll’arholAtlal.], 335,367. Enzingcr, F. (1965). Soft tissue tumors. In: TumorsofRoneund So/r Tissue, ~~31-5332, Eighth AnnualClinicalConferencc onCancer, lY63,TheUnivcrsityofTcxasM.D. Anderson HospitalandTumor Institute, 1 louston,Texas. Chicago: YearBook Medical Publishers. Evans,ll. L..Soulc,E. H.& Winkelmann. R. K. (1979). Atypical lipoma, atypical intramuscular lipoma and well diffcrcntiatcdrctroperitonealliposarcoma. Cancer43.S74 Greer,R.O.&Richardson,J.F.(IY73).’Thcnatureoflipomas andtheirsignihcanccin thcoralcavity. Arevicwofcases. OralSurgeiy, Oral Medicine, Oral Parhology. 36,55 I, Haidu. S. 1. (1970). Patholo~vofSofi Ti.s.sueTurnours, nn. .. 225-229. Philadelphia:-Lca& Febigcr. Kapur.1.. R. (1968). Rccurrcnt lipomataofthclarynxand the pharynxwithlatemalignantchange. Joltrnaluf’Laryngolo~y,
82,761. Kindhlom. L. G., Angcrvall, L. & Svendscn. P. (1075). Liposarcoma. Aclinicopathological. radiographicand prognosticstudy. Actrr Pa~holo~icaandMicmhiolo~i~u
Scandivavica, Supplement 253. 1,attcs. R. (lY68).The
fatty tumours. JoL1rflcr/ofrheAmc,rican
MedicalAssoc~iarion. 205,597. O’Day, R. A..Soule, E. H. d Gores, R. J. (1964). Soft tissue tumoursofthcoralcavity. Proceedingsqffhe MayoClinic,
39,160. Pack.G.‘T.&Pierson.J.C.( 1954). Liposarcoma:AstudyoflOS cases. Surgery. 36,687. Sawyer. K. C..Sawycr, R. B., Lubchenco. A. E.. Brahlcy. II. F. &Fenton. W.C.( 196X).Thcunprcdictahlefattytumour.
Ar~hive~so/SurXery(C:hicago,Ii~),%,773. Snitt1c.M. F..Newton. K. A.& MacKcnzic. D. fl. (1970) ’ Liposarcoma: ArevicwofMcascs. Rrifi.shJ&nalo~
Chncer, 24,606. Stout.A.P.&Lattes.R.(lY(,7).Tumorsofthesofttissucs.In:
A//asoJTumorf’arholog~~.ppIl~l26.SectII.Fascl. Washington.DC: ArmcdForccslnstituteofPathology. Wcstcott,W. P.&Corrcll.R. W.(lY84).Multiplerecurrenccsof alesionat the bascofthe tongue. JournaloJrkeAmericrrn I)enialA.ssocialio,l, IO&23 I
TheAuthor I’.G. Guest MBChB,
BBS, FDSKCS, Registrar Dcpartmcnt of Oral Surgery John Radcliffe I lospital Oxford OX3YDU
FHCS
CorrcspondcnccandrequcstsforoffprintstoMrP. Papcrrcccivcd4Scptembcr AcccptcdjDeccmbcr IYYI
1091
G. Guest
Liposarcoma
1
I
of the tongue: a case report and review of the literature
P. G. Guest Depurtment
of Oral Surgery,
John
Radcliffe Hospital,
Oxford,
OX3 9DlJ
SUMMARY. A case of the rare intraoral liposarcoma which presented as a lump on the tongue of 5 years duration is reported. The diffkulty of accurate clinical and histopathological diagnosis of this lesion is outlined.
INTRODUCTION
Liposarcomas of the oropharyngeal region are rare (O’Day et al., 1964; Pack & Pierson, 1954). Fewer than 100 cases have been reported. Liposarcomas may arise wherever there is adipose tissue normally present but the majority occur retroperitoneally and on the extremities (Enzinger & Winslow, 1962; Stout & Lattes, 1967). They account for 20% of all malignant soft tissue tumours (Stout & Lattes, 1967; Lattes, 1968). The head and neck region accounts for less than 4% of all sites affected (Enterline et al., 1960; Enzinger & Winslow, 1962; Enzinger, 1965). Unlike most sarcomas they are slow growing tumours with a broad spectrum of cellular differentiation other than the typical anaplasia and in the majority of cases they follow a relatively benign course. Liposarcomas are seen most commonly in the 5th and 7th decades of life and are slightly more common in males, in contrast with the benign lipomas which are common in females (Pack & Pierson. 1954; Enterline et al., 1960; Enzinger & Winslow, 1962; Spittle et al., 1970; Baden & Newman, 1977). Isolated examples of malignant transformation of a pre-existing lipoma have been reported (Correia, 1956; Kapur, 1968) and the lipoma has even been described as a potentially malignant lesion (Sawyer et al.. 1968). The head and neck region is relatively rarely affected and accounts for less than 5% of all sites (Pack & Pierson. 1954; Enterline et al., 1960; Enzinger & Winslow, 1962: Sawyer et al., 1968; Baden & Newman, 1977). Intraoral liposarcomas are even more rare (Pack & Pierson, 1954; ODay et al., 1964; Baden & Newman. 1977) but the benign intraoral lipoma is relatively much more common (Greer & Richardson, 1973).
Fig. I-Lesionat
lateral horderofthe tongue.
Fig. 2- Photomicrograph showing the features of a liposarcoma. (H & E. Original magnification~400).
sarcoma. It consisted of a mixture of adult fat cells and immature spindle shaped lipoblasts with a mucoid stroma (Fig. 2). It has been completely excised. The operative site healed uneventfully and at 2 years postoperatively there was no sign of recurrence of the tumour.
DISCUSSION
Case report
Liposarcoma is an extremely rare intraoral tumour and may present difficulty with diagnosis both clinically and histopathologically. The difficulty of distinguishing between a benign lipoma and a well differentiated liposarcoma has been commented upon by many authors (Kindblom rt ul.. 1~5; Hajdu. 1979: Evans c!t ~1.. 1979). Although the benign lipoma is said to have a characteristic clinical
A 71-year-old medical practitioner presented with a lump on his tongue which had been slowly increasing in size ovc‘r a S-year-period. It was not painful, not ulcerated and had never bled. Clinical examination revealed a firm yellowish lesion on the left lateral border of his tongue which was about 1 cm in diameter (Fig. I). The lesion was excised under local anaesthcsia and subsequent histological examination revealed it to be a well differentiated myxoid lipo26X
Liposarcoma
appearance, the only certain method of determining the exact nature of the tumour is by histopathological examination (Correll et al., 1982). On clinical examination liposarcomas tend to be more firm, less easily compressed and they may appear to be fixed to adjacent tissues (Westcott & Correll, 1984). They usually present as a painless mass or swelling and grow steadily but not rapidly. The silent, slow growth, especially in a submucosal or deep location, circumscription and firm texture often lead to the misdiagnosis of a cyst or a benign soft tissue neoplasm. At operation the liposarcoma is less yellow, less lobulated and less soft than normal adipose tissue or a lipoma (Hajdu, 1979). Histologically there is great diversity in the microscopic features and this has led in turn to various methods of classification and although outside the scope of this article a basic classification of myxoid, round ceil or plcomorphic types can be used. A review of the histological classifications is available elsewhere (Hajdu, 1979). The differential diagnosis must consider several lesions which may mimic the histological appearance of a liposarcoma, such as atypical lipoma, malignant fibrous xanthoma, myxoma, malignant mesenchymoma and embryonal rhabdomyosarcoma. Immediate good fixation and good biopsy sampling which includes all margins of the lesion are essential for the accurate evaluation of liposarcomas (Badcn & Newman, 1977). The most common intraoral sites of liposarcomas are the check and floor of the mouth (Badcn & Newman, 1977). The relatively few cases reported in the literature make any attempt to assess the biological behaviour of liposarcomas extremely difficult but there appears to be some correlation with the histological type (Baden 8r Newman., 1977). The well differentiated myxoid liposarcomas rarely metastasise, whereas the poorly differentiated tumours in adults metastasise in 40% of cases, primarily to lungs, pleura, liver and brain (Stout & Lattes, 1967). In contrast, the well differentiated liposarcomas do not appear to metastasise but may infiltrate locally (Das Gupta, 1973; Westcott & Correll, 1984). The correct treatment for liposarcomas is surgical excision with the line of excision 2 cm away from the tumour (Evans et al., 1979). Inadequate excision predisposes to local recurrences of the tumour (Das Gupta, 1973) and a case with multiple recurrences in the tongue during a 37-year period has been reported (Westcott & Correll, 1984). Any recurrence of a benign lipoma must be viewed with suspicion of the original diagnosis in view of the difficult histopathological differentiation between benign and malignant tumours. In addition. the possibility of malignant transformation of a lipoma makes it very important that patients with such lesions should be followed up closely and perhaps all should be excised. However the risk of malignant transformation is exceedingly small and the clinical course of the liposarcoma itself is relatively benign. It may well be that liposarcomata are more common than they are reported in view of
of the tongue
261)
the difficulty of diagnosis. In a review of cases designated Grade I liposarcoma it was determined that some of the neoplasms designated as well differentiated liposarcoma should have been called atypical lipoma because of their benign behaviour without any tendency to recur (Evans et al., 1979.) References Badcn.E.&Newman, oropharyngeal
R. (lY77). Liposarcomaofthe region. OralSurgrry, Oral Medicine, Oral
I’ulhology~ 44,889. Corrcia,P.D.(3956). Recurrent lipomaofthefloorofthe mouth. Revkra Puuksra DeMedicina(Sau Paulo),49,2XI. Correll, R. W., Wcstcott. W. B. &Pottcr. D. M. (1982). Non-ulcerated sessilc nodulcofthe lower lip./ourna/ofrhe
American DenralAxsociafion,
104,206.
DasGupta,‘l‘. (lY73). Managemcntofsoft tissuesarcomas. Surgery, ~ynaeculo~yat~dOh.steiric:s, 137,1012. Entcrline, H. T., Culberson, J. D.. Rochlin. 1). B.&Brady. L. W. (1960). I,iposarcoma: aclinicalandpathologicalstudy ofS3cascs. Cancer, 13,932. Enzinger, F. F.&Winslow. 1). J. (lY62). Liposarcoma. Astudy of 103cases. Virchow’s Archll’arholAtlal.], 335,367. Enzingcr, F. (1965). Soft tissue tumors. In: TumorsofRoneund So/r Tissue, ~~31-5332, Eighth AnnualClinicalConferencc onCancer, lY63,TheUnivcrsityofTcxasM.D. Anderson HospitalandTumor Institute, 1 louston,Texas. Chicago: YearBook Medical Publishers. Evans,ll. L..Soulc,E. H.& Winkelmann. R. K. (1979). Atypical lipoma, atypical intramuscular lipoma and well diffcrcntiatcdrctroperitonealliposarcoma. Cancer43.S74 Greer,R.O.&Richardson,J.F.(IY73).’Thcnatureoflipomas andtheirsignihcanccin thcoralcavity. Arevicwofcases. OralSurgeiy, Oral Medicine, Oral Parhology. 36,55 I, Haidu. S. 1. (1970). Patholo~vofSofi Ti.s.sueTurnours, nn. .. 225-229. Philadelphia:-Lca& Febigcr. Kapur.1.. R. (1968). Rccurrcnt lipomataofthclarynxand the pharynxwithlatemalignantchange. Joltrnaluf’Laryngolo~y,
82,761. Kindhlom. L. G., Angcrvall, L. & Svendscn. P. (1075). Liposarcoma. Aclinicopathological. radiographicand prognosticstudy. Actrr Pa~holo~icaandMicmhiolo~i~u
Scandivavica, Supplement 253. 1,attcs. R. (lY68).The
fatty tumours. JoL1rflcr/ofrheAmc,rican
MedicalAssoc~iarion. 205,597. O’Day, R. A..Soule, E. H. d Gores, R. J. (1964). Soft tissue tumoursofthcoralcavity. Proceedingsqffhe MayoClinic,
39,160. Pack.G.‘T.&Pierson.J.C.( 1954). Liposarcoma:AstudyoflOS cases. Surgery. 36,687. Sawyer. K. C..Sawycr, R. B., Lubchenco. A. E.. Brahlcy. II. F. &Fenton. W.C.( 196X).Thcunprcdictahlefattytumour.
Ar~hive~so/SurXery(C:hicago,Ii~),%,773. Snitt1c.M. F..Newton. K. A.& MacKcnzic. D. fl. (1970) ’ Liposarcoma: ArevicwofMcascs. Rrifi.shJ&nalo~
Chncer, 24,606. Stout.A.P.&Lattes.R.(lY(,7).Tumorsofthesofttissucs.In:
A//asoJTumorf’arholog~~.ppIl~l26.SectII.Fascl. Washington.DC: ArmcdForccslnstituteofPathology. Wcstcott,W. P.&Corrcll.R. W.(lY84).Multiplerecurrenccsof alesionat the bascofthe tongue. JournaloJrkeAmericrrn I)enialA.ssocialio,l, IO&23 I
TheAuthor I’.G. Guest MBChB,
BBS, FDSKCS, Registrar Dcpartmcnt of Oral Surgery John Radcliffe I lospital Oxford OX3YDU
FHCS
CorrcspondcnccandrequcstsforoffprintstoMrP. Papcrrcccivcd4Scptembcr AcccptcdjDeccmbcr IYYI
1091
G. Guest
