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PubMedDate not supplied

Liposarcoma of the oral cavity--case reports of the pleomorphic and the dedifferentiated variants and a review of the literature.

Liposarcoma is one of the commonest soft-tissue sarcomas, but very rare in the oral cavity. We present two cases of liposarcoma of the oral cavity, together with the related clinical, histopathological and immunohistochemical findings: one affecting the cheek of a 62-year-old man and the other the gingival maxillary tuber of a 41-year-old woman. At histological examination a diagnosis of liposarcoma was made in both cases. In the first case, immunohistochemical analysis revealed intense positivity for p53, MIB-1, MDM2, and focal positivity for S100 protein and CD34, but was negative for alpharsmooth muscle actin, desmin and CD68. The second case it was intensely positive for p53, MIB-1, S-100, and focal positive for MDM2, but negative for alpha smooth muscle actin, CD34, CD68 and desmin. Histological examination and immunohistochemical profiles in the first case were consistent with pleomorphic liposarcoma, whilst that in the second case with dedifferentiated liposarcoma. Both patients were subjected to surgical treatment with wide surgical margins, without adjuvant radio- or chemotherapy. The first case was lost at follow-up one year after surgery, while the second case has not undergone relapse after seven years. We discuss differential diagnosis, examining the histopathological and immunohistochemical features that are potentially useful for distinguishing this tumor from other malignant adipose tissue tumors.

Adult↗
PubMed1990

[A case of primary mediastinal liposarcoma which presents a singular appearance].

An operated case of primary mediastinal liposarcoma is reported. A 71-year-old male who was asymptomatic was unexpectedly pointed out a huge abnormal mass shadow in the mediastinum on the chest roentgenogram, when he caught a common cold. The CT scan showed the singular horseshoe-shaped tumor in the posterior mediastinum which was adjacent to the antero-lateral phase of the vertebrae. MRI was useful to our recognition of its whole shape and localization. Extirpation of the tumor via right postero-lateral thoracotomy was performed successfully. The pathological diagnosis was liposarcoma; well differentiated, lipoma-like type. Primary mediastinal liposarcoma is a very rare mediastinal tumor (0.2%). Thirty-six cases of primary mediastinal liposarcoma including our case have been reported at present in Japan, so far as we surveyed. It is generally known that liposarcoma has the low sensitivity to the radiotherapy and chemotherapy and has high incidence of recurrence. We think that this case needs the strict postoperative follow-up.

Aged↗
PubMed1986

[Problems of the differential diagnosis and classification of liposarcomas (electron microscopy study)].

Ultrastructure of both liposarcomas of different histological types and those with non-verified diagnosis are studied. Liposarcomas are shown to contain a basic complex of ultrastructural features of lipid differentiation and different histological types of these tumours have, in principle, identical features varying in the degree of their expression. The questions of a differential diagnosis of liposarcomas and other tumours can be successfully solved on the basis of the above findings, and even more, a group of liposarcomas with a severe cataplasia that does not have histological analogues may be distinguished. The results allowed to work out an ultrastructural classification of liposarcomas in which the degree of cataplasia is directly taken into account.

Diagnosis, Differential↗
PubMed1984

Liposarcomas. A histogenetic approach to the classification of adipose tissue neoplasms.

A series of atypical lipomas, well-differentiated/dedifferentiated liposarcomas, and myxoid/round cell liposarcomas were examined by light and electron microscopy in order to understand better the marked variation in histologic growth patterns exhibited by adipose tissue neoplasms. We found that they were composed of a mixture of adipocytes and nonfat-storing mesenchymal cells or were predominantly made up of only one of these cell types. From these and other published observations, we feel that 1) myxoid/round cell liposarcomas are composed of adipocytes; 2) spindle cell lipomas, atypical lipomas, and well-differentiated liposarcomas are composed of a mixture of the two cell types; and 3) the dedifferentiated regions of well-differentiated liposarcomas are composed of nonfat-storing mesenchymal cells. The basic structural differences of the two cell types and the quantitative variations when they occur in mixtures seem to account for the marked variations in neoplastic growth patterns.

Adipose Tissue↗
PubMed1994

[Liposarcoma of the head].

Cephalic localization of liposarcoma is exceptional. We report three cases, in an 87-year-old patient with liposarcoma of the tongue, a 78-year-old patient with multiple recurrent liposarcoma of the orbit and a 42-year-old patient with ethmoido-orbital liposarcoma of metastasis origin. These three cases and a review of the literature were the basis of a summary of the incidence, the anatomic classification and therapeutic options for this type of liposarcomas.

Adult↗
PubMed1996

Liposarcoma of the base of tongue and tonsillar fossa: A possibly underdiagnosed neoplasm.

Liposarcomas of the head and neck are exceedingly rare, and fewer than 90 cases have been reported in the literature. Liposarcoma of the oral cavity is an even less common entity, and to our knowledge only nine cases have been reported to date. We report the clinical and pathologic findings of a case of well-differentiated liposarcoma of the base of tongue and tonsillar fossa. The patient is a 76-year-old white man with a long-standing history of a mass in the oral cavity and hypopharynx. The mass had been resected several times over the span of 23 years, and diagnoses of lipoma, neurofibroma, mesenchymoma, and angiofibrolipoma have been rendered on different occasions. At the last admission, a polypoid mass of the left tonsillar fossa and base of tongue was resected. The tumor was multinodular and measured 2.5 cm in greatest diameter. Histologically the tumor was ill-defined with infiltrating borders and was composed predominantly of mature adipose tissue with occasional lipoblasts. A small proportion of the tumor consisted of clusters of spindle cells and pleomorphic lipoblasts. Mitotic activity was not seen. The pleomorphic cells were positive for S100 protein and negative for muscle-specific markers. Ultrastructural analysis confirmed the nature of the lipoblasts. Our case depicts the typical natural history and histologic features of liposarcoma of the oral cavity. This tumor is usually well differentiated and has a high recurrence rate and almost no tendency for metastasis. Based on our case and review of the literature, it appears that well-differentiated liposarcoma of the oral cavity can occasionally be underdiagnosed because of the low mitotic activity and long latent period between the original diagnosis and first recurrence.

Aged↗
PubMed1996

Focal accumulation of iodine-123-BMIPP in liposarcoma of the thigh.

Findings for focal accumulation of 123I-15-(p-iodophenyl)-3-R,S-methylpentadecanoic acid ([123I]BMIPP) in a patient with liposarcoma of the thigh are presented. Iodine-123-BMIPP accumulated heterogeneously in the liposarcoma. The region with marked accumulation of [123I]BMIPP was diagnosed as mixoid liposarcoma. The region with little accumulation of [123I]BMIPP was diagnosed as well-differentiated liposarcoma. Differences in the accumulation of [123I]BMIPP may reflect differences in fatty acid metabolism between histopathological types of liposarcoma.

Adult↗
PubMed1996

Myxoid liposarcoma with cartilaginous differentiation.

Common histologic variants of liposarcoma are readily recognized. Histologic classification might be difficult, however, when liposarcoma demonstrates cartilaginous differentiation. Although this phenomenon has previously been noted, it has not, to our knowledge, been reported as a specific pathologic entity. All three patients in the present study were men, ages 37, 42, and 63 years. Each presented with a solitary, enlarging mass of the thigh that was surgically excised. The tumors ranged in size from 8 to 13 cm. Microscopically, each lesion displayed characteristic features of myxoid liposarcoma; in addition, all possessed discrete, rounded foci of mature-appearing hyaline cartilage. One case displayed small foci showing chondrocyte atypia. No other patterns of mesenchymal differentiation were present. All patients received postoperative radiation therapy. No recurrences or metastases have been identified (mean duration, 39 mo). Myxoid liposarcomas with cartilaginous differentiation are of importance because they might be misdiagnosed as malignant mesenchymoma. The latter, if not qualified as to histologic grade, might be presumed to be a biologically more aggressive lesion. In addition, these lesions must be distinguished from two benign processes: chondroid lipoma and extraskeletal chondroma with lipoblast-like cells. Additional studies of this uncommon variant of liposarcoma will be necessary to document further its status as a low-grade sarcoma.

Adult↗
PubMed2002

TLS-CHOP target gene DOL54 expression in liposarcomas and malignant fibrous histiocytomas.

Downstream of the gene for the liposarcoma-associated fusion oncoprotein 54 (DOL54) is a target gene of the myxoid liposarcoma and round cell liposarcoma (M-LPS/RC-LPS) oncogene, TLS/FUS-CHOP. The DOL54 gene product is closely associated with adipogenic differentiation. DOL54 overexpression resulted in tumorigenicity when Chinese Hamster Ovary (CHO) cells were injected subcutaneously into nude mice. The biological significance of DOL54 expression for human malignant soft tissue tumors, however, has not yet been investigated. We examined TLS-CHOP and DOL54 expression in M-LPS/RC-LPS, well-differentiated liposarcoma and malignant fibrous histiocytoma (MFH), a tumor whose cellular origin has not been determined. We observed DOL54 expression in 50% of M-LPS/RC-LPS cases (in which TLS-CHOP was also expressed) and 33% of MFH cases, suggesting that a portion of MFH lesions may either derive from adipocytic precursor cells or have the potential to undergo adipogenic differentiation. In this manner, M-LPS/RC-LPS and MFH lesions may share tumorigenic characteristics, resulting from the unscheduled expression of DOL54.

Blotting, Northern↗
PubMed2003

Radiologically and histologically mixed liposarcoma: a report of two biphasic cases.

We report two cases of liposarcomas showing biphasic patterns, radiologically and histologically. The first case was a 52-year-old man with a 17 x 12 cm intramuscular tumor in the right thigh. MR imaging revealed a mass composed of two components: a fat component and another soft tissue component. Histological diagnosis revealed mixed-type liposarcoma consisting of well-differentiated and myxoid liposarcoma. The second case was a 62-year-old man with a 22 x 15 cm intramuscular tumor in the left calf. MR imaging showed a mass composed of fat and non-fat components. The histological diagnosis was well-differentiated and pleomorphic liposarcoma.

Diagnosis, Differential↗
PubMed1995

Liposarcomas of the oral tissues: a clinicopathologic study of four tumors.

Liposarcomas of the oral tissues are rare. There have been 28 previously reported in the literature. A clinicopathologic study of 4 patients with liposarcoma involving the oral tissues is presented--one in the tongue, one in the palate, and two in the submandibular space. Two of the tumors were myxoid, one was of well-differentiated type and one was of pleomorphic type. Three of the lesions were diagnosed clinically as benign. The most important lesson to be learned from the histopathology is the positive identification of atypical lipoblasts. Follow-up data indicate that prognosis generally corresponds to the tumor type. The patients with myxoid and well-differentiated liposarcoma remain alive and with no evidence of disease. The patient who died of tumor had a pleomorphic liposarcoma. Treatment of these lesions should consist of wide surgical excision and consideration of adjunctive or palliative radiotherapy.

Adult↗
PubMedDate not supplied

Limited role of TP53 and TP53-related genes in myxoid liposarcoma.

AIMS: Circumstantial evidence suggests that genetic changes may lead to tumor progression within the myxoid liposarcoma tumors (MLTs) carrying non-random chromosomal translocation t(12;16). METHODS: To address this subject an immunophenotypic analysis, applying antibodies against proteins encoded by TP53, MDM2 and CDK4 genes, complemented by molecular analysis of eight suitable cases, was performed on 104 consecutive cases. Chromosomal translocations were assessed either by cytogenetic analysis or by RT-PCR in 9 suitable cases and chimeric transcripts were found in all cases but two pleomorphic liposarcomas. RESULTS: Based on immunophenotyping and tumor site, the case material consisted of three groups. The first one was made up of 92 non-retroperitoneal cases carrying a null p53, mdm2, cdk4 immunophenotype, which remained unchanged over the time of recurrences and along the gamut of histologic subtypes. The second group was represented by five p53+, mdm2-, cdk4- non-retroperitoneal cases, 4 of which were further analysed by PCR-SSCP for p53 mutation. The immunophenotypic profile of these cases, complemented by the molecular findings, supported a role of TP53 in tumor progression in three high-grade MLTs. The third group, consisting of 7 retroperitoneal cases, showed a heterogeneous immunophenotype, sharing immunophenotypic and molecular features with the well-differentiated/evoluted (dedifferentiated) liposarcoma group. CONCLUSIONS: TP53 mutations seem to play a role in tumor progression in a few cases of MLTs (2.8%) showing more aggressive histologic characteristics. The unexpected finding that a number of retroperitoneal LMTs display the immunophenotypic profile of the well differentiated/evoluted (dedifferentiated) liposarcomas, deserves further investigation.

DNA Mutational Analysis↗
PubMedDate not supplied

Recurrent liposarcomas of the abdomen and retroperitoneum: three case reports.

We report three cases of patients with liposarcomas of the abdomen who had been treated during the last 13 years (1980-1993). Two patients were men, aged 29 and 51 years, with tumors of the retroperitoneal space and the third patient was a woman aged 64 years with a tumor in the peritoneal cavity. Therapeutic treatment was as aggressive as possible excision of the tumor. In the case of the first male patient, the histological examination revealed a retroperitoneal myxoid liposarcoma which recurred 5 times within 13 years. In the second male patient, it revealed a well differentiated retroperitoneal liposarcoma of the sclerosing type which recurred 5 times within 5 years since the first treatment. Finally, the one female patient had 2 recurrences of myxoid liposarcoma of the abdomen 9 years after the first operation, presented with an infected mass and has been well since then.

Adult↗
PubMed1997

Molecular aberrations of the G1-S checkpoint in myxoid and round cell liposarcoma.

Myxoid and round cell liposarcoma represents a morphological spectrum in which tumor progression from low-grade myxoid to high-grade round cell areas is frequently observed. A distinctive t(12;16)(q13;p11) reciprocal translocation rearranges the CHOP gene localized to 12q13 in most cases. Data concerning the occurrence of cell cycle aberrations in this subset of mesenchymal malignancies are very limited. Therefore, we analyzed a histologically homogeneous series of 21 cases of myxoid and round cell liposarcoma. The p53 pathway was studied by investigating the TP53 gene and protein, mdm2 protein, and p21Waf1 protein. The Rb-cyclin D pathway was analyzed by studying the pRb protein, the p16MTS1 gene, cyclin D1, cyclin D3, p27Kip1, cdk4, and cdk6 proteins. In contrast with the rare involvement of the TP53 gene in well differentiated liposarcoma, aberrations of the TP53 gene were observed in approximately 30% of cases of myxoid and round cell liposarcoma. Notably, mdm2 overexpression was seen in 56% of cases and correlated with histological grade, therefore indicating a possible role in tumor progression. Abnormalities involving the Rb-cyclin D pathway were observed in more than 90% of cases. pRb loss was present in one-third of cases and, at variance with that observed in other subsets of sarcoma, overexpression of cyclin Ds represented a rare event. Interestingly, upregulation of either cdk4 or cdk6 was demonstrated in 85% of cases.

Chromosome Aberrations↗
PubMed1999

Telomeric lengths and telomerase activity in liposarcomas.

To assess the role of telomerase in the development of liposarcomas, we measured telomerase activity in 36 malignant and seven benign lipomatous neoplasias from 34 patients. A sensitive polymerase chain reaction-based telomerase assay (the telomeric repeat amplification protocol) was applied. Shortening or elongation of telomeric repeat fragment lengths, as measured by using hybridization with a telomere-specific oligonucleotide probe, was correlated with the presence of telomerase activity. The latter was demonstrable in 69% of malignant tumors. Benign tumors can be distinguished from malignant neoplasias on the basis of telomerase activity. However, telomerase expression seems to be characteristic of poorly differentiated liposarcomas. Myxoid/round cell liposarcomas exhibited a higher telomerase activity level than the classical low-grade variants. Telomerase activity was not correlated with age at the time of diagnosis or with sex. In most cases, telomerase-positive tumors showed higher proliferation indices than did neoplasias lacking telomerase. All eight recurrences expressed telomerase activity, reflecting a close association of telomerase with the biological behavior of liposarcomas. Our findings suggest that telomerase may play a key role in the establishment and progression of malignant lipomatous tumors.

Adult↗
PubMed1981

Primary liposarcoma of bone: light and electron microscopic study.

The case history of a patient with primary liposarcoma of the humerus associated with a schwannoma of the radial nerve in the arm is described. Ultrastructurally, the neoplasm showed multiple and varied lipid droplets, dilated RER, mitochondria with electron-dense matrix, and microfilaments as the most prominent features. It is concluded, based on these ultrastructural observations, that liposarcoma of the bone is similar to pleomorphic liposarcoma arising in the soft tissues. This is the first ultrastructural study of a primary liposarcoma of the bone and the first reported association of this tumor with a schwannoma. The possible pathogenetic relationship between both tumors remains unknown.

Adult↗
PubMed1982

Variability of histologic pattern in recurrent soft tissue sarcomas originally diagnosed as liposarcoma.

The histopathologic classification of liposarcoma and its clinicopathologic significance are well delineated in previous studies, but the literature is inexplicably mute on recurrent liposarcomas. Supposition and experience may have led to the assumption that the microscopic features remained static over a period of years and multiple recurrences. Thirteen sarcomas originally classified as liposarcoma were reviewed to assess patterns of histologic change with recurrence. A total of 52 recurrences was studied. In 7 of the 13 cases, multiple morphologic sarcomatous patterns were identified, either as a change with recurrence or as multiple discrete patterns within a tumor. All changes except in one case occurred before irradiation or chemotherapy. The variations included patterns such as malignant fibrous histiocytoma, hemangiopericytoma, malignant schwannoma, and unclassified spindle-cell sarcoma as well as the various liposarcoma subtypes. This variability of patterns can cause significant diagnostic difficulties and can cause one to question the validity of using monomorphic diagnoses in all cases. In addition, the polymorphic differentiation of these tumors supports the concept that soft tissue sarcomas are derived from a pluripotential mesenchymal stem cell.

Adult↗
PubMed1984

Childhood liposarcoma. Report of a case and review of the literature.

A case of liposarcoma presenting in an adolescent black male is described. Features unique to this case include a mediastinal primary site (the fourth to be documented in a pediatric patient), a demonstrable response to radiotherapy and chemotherapy allowing complete surgical excision of an initially inoperable tumor, and the apparent histologic maturation of the tumor following multimodal therapy. A review of previously published accounts of pediatric liposarcoma revealed the following: (1) peak incidences during infancy and in early adolescence; (2) the extremities to be the most common site of origin (51%); (3) a predominance of myxoid histology (76%); and (4) a lower overall recurrence rate when compared with adult cases (37% versus 72%, respectively). The influence of histology, location of the tumor, and completeness of surgical excision on the prognosis of adult liposarcoma was confirmed in this limited pediatric experience. While the use of radiotherapy and chemotherapy cannot be advocated in all instances of liposarcoma, our experience and that from other reports would support the incorporation of these modalities of treatment in selected patients where the tumor is surgically inaccessible due to size.

Adolescent↗