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PubMed2006

A case representing coexistence of acute myeloblastic leukemia and dedifferentiated liposarcoma: the possible role of chemotherapy in triggering dedifferentiation.

Acute myelogenous leukemia (AML) is a hematological disorder that is characterized by an abnormal proliferation of immature myeloid cells. Dedifferentiated and well-differentiated liposarcomas are the two pathological subtypes of liposarcoma, based on the WHO classification. Transition from well-differentiated to dedifferentiated liposarcoma is a well-recognized phenomenon. Well-differentiated tumors are known to have low malignancy grade. However, when dedifferentiation occurs, the tumor acquires the aggressive features of a fully malignant lesion. This process largely is believed to progress in a time-dependant manner; however, time is not the only factor of importance. The potential roles of other factors in this transition are still unclear. To date, the coexistence of AML and liposarcoma has not been reported in the literature. In this paper, we report on a case of coexistence of AML and liposarcoma, and on the unusual behavior of a well-differentiated tumor after dedifferentiation occurs.

Abdominal Neoplasms↗
PubMed1996

Focal rhabdomyosarcomatous differentiation in primary liposarcoma.

A unique case of primary myxoid liposarcoma of the thigh, in which focal pleomorphic areas were present containing rhabdomyoblasts, is described. Focal rhabdomyosarcoma in liposarcoma has only rarely been reported previously and only in dedifferentiated liposarcomas of the retroperitoneum. All but one have been recurrences with rhabdomyoblasts being absent in the primary liposarcoma. As rhabdomyoblasts were only focally present, the present case is regarded as liposarcoma with focal divergent rhabdomyoblastic differentiation rather than malignant mesenchymoma.

Aged↗
PubMed2002

Imaging of fatty tumors: distinction of lipoma and well-differentiated liposarcoma.

PURPOSE: To review the reliability of computed tomographic (CT) and magnetic resonance (MR) imaging features in distinguishing lipoma and well-differentiated liposarcoma. MATERIALS AND METHODS: CT (n= 29) and MR (n = 40) images and radiographs (n = 28) of 60 patients with histologically verified fatty tumors (35 lipomas and 25 well-differentiated liposarcomas) were retrospectively reviewed in 31 females and 29 males (mean age, 56 years; age range, 1-88 years). Images were assessed for adipose tissue content, and non-fatty component was classified (thin and/or thick septa and nodular and/or globular components) as absent, mild, moderate, or pronounced. Also assessed were signal intensity and tissue attenuation of the fatty components and non-adipose elements. RESULTS: Statistically significant imaging features favoring a diagnosis of liposarcoma included lesion larger than 10 cm (P <.001), presence of thick septa (P =.001), presence of globular and/or nodular non-adipose areas (P =.003) or masses (P =.001), and lesion less than 75% fat (P <.001). The most statistically significant radiologic predictors of malignancy were male sex, presence of thick septa, and associated non-adipose masses, which increased the likelihood of malignancy by 13-, nine-, and 32-fold, respectively. Both lipoma and liposarcoma demonstrated thin septa and regions of increased signal intensity on fluid-sensitive MR images. CONCLUSION: A significant number of lipomas will have prominent non-adipose areas and will demonstrate an imaging appearance traditionally ascribed to well-differentiated liposarcoma. Features that suggest malignancy include increased patient age, large lesion size, presence of thick septa, presence of nodular and/or globular or non-adipose mass-like areas, and decreased percentage of fat composition.

Adipose Tissue↗
PubMed2006

Telomere maintenance mechanisms in liposarcomas: association with histologic subtypes and disease progression.

Human cancer cells maintain telomeres by telomerase activity (TA) or by alternative lengthening of telomeres (ALT). We proposed to define the prevalence of the two telomere maintenance mechanisms (TMM), to assess their association with histology, and to compare their prognostic relevance in a series of 93 patients with liposarcoma. ALT was detected by assaying ALT-associated promyelocytic leukemia nuclear bodies and TA was assayed using the telomeric repeat amplification protocol. ALT or TA was found in 25.9% or 26.6% of 139 tested liposarcoma lesions, respectively. Three lesions were ALT+/TA+ whereas approximately 50% of lesions did not show any known TMM. TMM phenotype was consistent during disease progression. ALT was prevalent in dedifferentiated and in grade 3 liposarcomas whereas TA prevailed in most round-cell myxoid and in grade 2 liposarcomas. ALT and TA incidence was similar in primary and recurrent lesions whereas metastases were more frequently TA+ than ALT+ (59% versus 18%; P = 0.04). TMM presence negatively affected patient prognosis (P = 0.001): increased mortality was associated with positivity for TA (P = 0.038) or ALT (P < 0.0001) compared with TMM absence. ALT proved to be a stronger prognostic discriminant of increased mortality than TA even when adjusted for tumor location, grade, and histology (hazard ratio for cause-specific death, 3.58 versus 1.15). Our results indicate that ALT can support fully malignant liposarcomas and is associated with unfavorable disease outcome.

Adult↗
PubMedDate not supplied

Fine needle aspiration cytology of a dedifferentiated liposarcoma: report of a case with histologic and immunohistochemical follow-up.

BACKGROUND: Dedifferentiation is a histologic progression of a neoplasm from low grade to high grade histology. It occurs in tumors of the retroperitoneum and in those undergoing treatment. This usually occurs in the setting of radiation or chemotherapy or as a spontaneous process over a long period. The features of dedifferentiation can be toward any mesenchymal element of the underlying neoplastic process. CASE: We report the cytologic features of a dedifferentiated liposarcoma arising in a 76-year-old man who had a history of well-differentiated liposarcoma. Papanicolaou- and Diff-Quik-stained smears from a radiologically guided fine needle aspiration biopsy showed a hypercellular sample. The smears showed a mixed population of cells. There were multinucleated, pleomorphic giant cells with abundant cytoplasm, smaller clusters of cells with a high nuclear/cytoplasmic ratio and cells with spindled and elongated nuclear features. The follow-up surgical resection specimen showed a dedifferentiated liposarcoma with strong and diffuse immunoreactivity to vimentin, desmin and CD68 in the large, pleomorphic cells; focal and weak immunoreactivity to smooth muscle actin and S-100 in these cells; and strong and focal immunoreactivity to desmin, smooth muscle actin and muscle-specific actin in the spindle cells. This supports the dedifferentiated components of this tumor to be of fibrohistiocytic and leiomyosarcomatous differentiation. CONCLUSION: Dedifferentiation of a well-differentiated liposarcoma should be entertained in the setting of a mass lesion in the retroperitoneum in patients with prior histories of well-differentiated liposarcoma. The radiologic features of a particular neoplastic process can be very helpful in determining the nature of this process.

Aged↗
PubMed2000

Liposarcoma of the tongue: case report and literature update.

Liposarcoma is an exceedingly rare tumor in the oral cavity. We report a case of a 70-year-old man with liposarcoma that presented as a lump on the tongue. The excised tumor was diagnosed as a well-differentiated lipoma-like liposarcoma. The majority of cases of well-differentiated liposarcoma follow a relatively benign course, but the disease has a high recurrence rate. It appears that accurate clinical and histopathologic diagnosis of this lesion is difficult. The prognosis seems to depend on the histologic type, size, and location of the lesion. Wide surgical excision is important for successful management of these liposarcomas.

Aged↗
PubMed2001

Immunoreactivity of p53, mdm2, and p21WAF1 in dedifferentiated liposarcoma: special emphasis on the distinct immunophenotype of the well-differentiated component.

Alteration of the p53/mdm2 pathway has been reported in the well-differentiated liposarcoma (WDLS)/dedifferentiated liposarcoma (DDLS) group. We investigated the immunoreactivity of p53, mdm2, and p21WAF1, along with the MIB-1-labeling index (MIB-1-LI) in 21 WDLS and 21 DDLS cases, to clarify the association of these markers with the morphologic changes and the biological factors responsible for the aggressiveness of DDLS. Within DDLS, p53 and p21WAF1 expression and mdm2 overexpression were significantly more prevalent in the dedifferentiated (DD) components than in the well-differentiated (WD) components. The mdm2 overexpression and p21WAF1 expression was significantly associated with sclerosing liposarcomas in both WDLS and the WD components of DDLS. There was no significant difference in the immunoreactivity of p53, mdm2, or p21WAF1 or MIB-1-LI between WDLS and the WD components of DDLS. An association was found between p53 expression and mdm2 overexpression in the WD group (comprising WDLS and WD components of DDLS) and in the DD group, significantly so in the WD group. Notably, this correlation was found in the subtype of sclerosing liposarcoma but not in that of lipoma-like liposarcoma. Within DDLS, the clinical outcome of the nonaccessible soft tissue (non-AST: comprising retroperitoneum and mediastinum) group was significantly worse than that of the accessible soft tissue (AST: comprising extremities, buttocks, axilla, and scrotum) group; however, the immunophenotypes of p53, mdm2, and p21WAF1 and the MIB-1-LI showed no correlation with survival in the AST group alone, in the non-AST group alone, or in the 2 together. This study suggests that the immunoreactivity of p53, mdm2, and p21WAF1 is associated with the morphologic changes, but not with the biological factors responsible for the aggressiveness of DDLS.

Adult↗
PubMed2000

Influence of biologic factors and anatomic site in completely resected liposarcoma.

PURPOSE: Soft tissue sarcoma (STS) encompasses a group of neoplasms that are anatomically and biologically diverse. Retroperitoneal/visceral (RP/V) tumors have a poorer prognosis than extremity/trunk (E/T) lesions, and this has been attributed to frequent presentation with tumors of large size and multiorgan involvement that precludes complete resection. The worse prognosis that is associated with RP/V tumors has also been thought to be histopathologically dependent and not necessarily related to anatomic site. The aim of this study was to determine the role of anatomic site and biologic features in prognosis and outcome in patients after complete resection by examining a large cohort of STS patients with a single histopathology, ie, liposarcoma. METHODS: All patients who were treated for liposarcoma from July 1, 1982, through July 1, 1998, were included. Univariate analyses were performed using log-rank test and Kaplan-Meier estimates, and multivariate analyses were performed using Cox regression. The three end points examined were local recurrence (LR), distant recurrence, and disease-specific survival (DSS). RESULTS: Seven hundred twenty patients with liposarcoma were evaluated, and of these, 460 had completely resected primary or completely resected locally recurrent disease. Breakdown of anatomic site was 65% E/T (n = 301) and 35% RP/V (n = 159). The median follow-up period for patients who underwent complete resection was 42 months (range, 1 to 194 months). We found that RP/V site is a poor prognosticator that is independent of patient sex and age; tumor size, grade, and margin; and recurrent presentation. Sixty-nine percent of patients with RP/V tumors who died had local disease only and no distant metastasis at the time of death. CONCLUSION: In liposarcoma, tumor location exerts as strong an influence on prognosis as biology. In contrast to extremity liposarcoma, LR without distant metastasis often results in death for patients with RP/V tumors. For these patients, local control accomplished by complete surgical resection +/- adjuvant radiation therapy should impact strongly on DSS.

Disease-Free Survival↗
PubMed1996

CT and MR imaging of abdominal liposarcoma.

OBJECTIVE: CT and MR images were reviewed to correlate the histologic subtypes of abdominal liposarcoma with the radiologic findings. SUBJECTS AND METHODS: Ten patients with liposarcoma who underwent CT or MR imaging before surgery were included in this study. CT and MR imaging findings for these patients were compared retrospectively with histologic findings. RESULTS: Major histologic subtypes found in our group of patients were five well-differentiated, three myxoid, one pleomorphic, and one round-cell liposarcomas. The well-differentiated subtype consisted of lipoma-like and/or sclerosing components. The predominant attenuation and signal intensity characteristics of the lipoma-like components on CT and MR images resembled those of fat, whereas the predominant attenuation and signal intensity characteristics of the sclerosing components resembled those of muscle. The myxoid subtype showed, on unenhanced images, predominant attenuation and signal intensity characteristics that resembled those of water; on contrast-enhanced images, this subtype showed gradual reticular enhancement. The appearance of the round-cell and pleomorphic subtypes was that of heterogeneous, nonfatty tumors. Their characteristics were indistinguishable from those of other malignant soft-tissue masses. CONCLUSION: Each histologic subtype of abdominal liposarcoma showed different CT attenuation or MR imaging signal intensity characteristics. A clear understanding of these findings should prove helpful in the diagnosis of liposarcoma.

Abdominal Neoplasms↗
PubMed2002

CT differentiation of large exophytic renal angiomyolipomas and perirenal liposarcomas.

OBJECTIVE: The purpose of our study was to describe the imaging findings and CT characteristics that lead to accurate distinction of large exophytic renal angiomyolipomas from retroperitoneal perirenal liposarcomas, which at times can be confused on imaging studies and even at pathologic examination. MATERIALS AND METHODS: We retrospectively analyzed CT images of 15 large exophytic renal angiomyolipomas and 12 well-differentiated perirenal liposarcomas. Pathologic correlation was available for six of 15 angiomyolipomas and all of the liposarcomas. All examinations were evaluated for lesion size, renal parenchymal defect, enlarged vessels, kidney displacement, lesion encapsulation or margination, associated hemorrhage, and additional angiomyolipomas. The records of patients with tuberous sclerosis or the forme fruste of that condition were excluded from the study. RESULTS: The average size of the angiomyolipomas was 14 x 10 cm. They showed a renal parenchymal defect (n = 15), enlarged vessels (n = 12), renal displacement (n = 14), good margination without a distinct capsule (n = 14), hemorrhage (n = 1), and additional (one or two) angiomyolipomas (n = 4). The average size of the liposarcomas was 18 x 11.6 cm. They showed enlarged vessels (n = 3), renal displacement (n = 11), and encapsulation (n = 4); none showed a renal parenchymal defect, hemorrhage, or associated angiomyolipomas. CONCLUSION: Although large exophytic angiomyolipomas and well-differentiated retroperitoneal liposarcomas may have similar appearances on imaging, careful evaluation for a defect in the renal parenchyma combined with the presence of enlarged vessels in angiomyolipomas should enable accurate differentiation in almost all cases. Achieving an accurate diagnosis can have a significant impact on patient treatment.

Adult↗
PubMed2006

A case of huge primary liposarcoma in the liver.

Primary liver liposarcoma is a rare disease. Because of its rarity, the knowledge of the clinical course, management, and prognosis of primary liver liposarcoma are all limited for clinicians. A 61-year-old female patient who suffered from a huge primary liposarcoma in the central portion of the liver had the clinical presentations of fever, nausea, vomiting, jaundice, and body weight loss. The huge tumor was resected successfully. However, the tumor recurred repeatedly and she had repeated hepatectomies to remove the tumor. The tumor became aggravating after repeated surgeries. Eventually, the patient had cervical spinal metastasis of liposarcoma and she survived for 26 months after liver liposarcoma was diagnosed. Although the tumor may become aggravating after repeated surgeries, repeated hepatectomies are still the best policy to achieve a long-term survival for the patients.

Female↗
PubMedDate not supplied

Long-term results of aggressive surgical treatment of primary and recurrent retroperitoneal liposarcomas.

The aim of this study was to establish the role of surgery in the treatment of retroperitoneal liposarcomas. Data concerning 28 patients submitted to surgery for retroperitoneal liposarcoma in our department over the period from 1972 to 1999 were reviewed retrospectively and analysed. Seventy-four operations were performed; in 54% of the operations it was necessary to resect contiguous organs (kidney 60%, colon 50%, adrenal gland 35%). In 89%, grossly curative resection was achieved at the first operation; 20 patients had at least one local recurrence after first operation (median time interval: 22 months). The mean follow-up was 80 months; median survival time was 51 months and 5-year actuarial survival time 51%. Patients with low-grade liposarcoma showed a statistically significant improvement (P < 0.001) in median survival (153 months) versus those with medium- (37 months) and high-grade sarcomas (8 months). At present surgery is still the treatment of choice in the treatment of primary and recurrent liposarcoma; in the case of low-grade liposarcomas especially, an aggressive surgical approach can result in long-term survival.

Adolescent↗
PubMed2001

[Scrotal liposarcoma].

OBJECTIVE: To describe an additional case of scrotal liposarcoma and its treatment, which was distinct from conventional treatment. METHODS: The literature on paratesticular masses is reviewed with special reference to the liposarcomas. An additional case of liposarcoma is described which was treated by tumor excision without involvement of the testis. The diagnosis, classification and treatment of this condition are discussed. RESULTS: Liposarcoma is a very uncommon tumor and its diagnosis is based on the anatomopathological findings. Treatment is by orchidectomy with high ligation of the spermatic cord. However, the case described herein was treated only by resection of the tumor. Radiotherapy may be indicated in some cases. Chemotherapy does not appear to be useful in this type of tumor. CONCLUSION: In some cases of scrotal liposarcoma, the tumor can be resected without performing orchidectomy.

Aged↗
PubMed2001

Dedifferentiated liposarcoma.

Dedifferentiated liposarcoma is a high-grade nonlipogenic sarcoma that arises in a background of a preexisting well-differentiated liposarcoma. The phenomenon of dedifferentiation is time dependent, and primary or de novo tumors exceed secondary neoplasms in a ratio of 9:1. The tumor occurs most frequently in adults beyond the 6th decade of life, slightly predominates in men, and involves the abdominal cavity most often. Pleomorphic malignant fibrous histiocytoma-like histologic features are the most commonly observed phenotype, although other sarcomatous phenotypes have been described less frequently. Surgical treatment is the main form of therapy for dedifferentiated liposarcoma, which is associated with a reported local recurrence rate of 41% to 52%, 15% distant metastatic rate, and 30% disease-related mortality rate. On a chromosomal level, dedifferentiated liposarcoma frequently displays the same chromosomal abnormality associated with well-differentiated liposarcomas--ie, the presence of a supernumerary ring or giant chromosome derived from the 12q(13-15) region.

Chromosome Aberrations↗
PubMed2003

Establishment of a novel human dedifferentiated liposarcoma cell line, FU-DDLS-1: conventional and molecular cytogenetic characterization.

A number of human cell lines derived from well-differentiated, myxoid/round cell, or pleomorphic liposarcoma have been described. To our knowledge, however, no human cell line established from dedifferentiated liposarcoma has been reported. In this study, we established a new human cell line, FU-DDLS-1, which originated from a dedifferentiated liposarcoma arising in the retroperitoneum of a 61-year-old man. This cell line was characterized by immunocytochemistry, conventional banding analysis, fluorescence in situ hybridization with chromosome painting probe, and comparative genomic hybridization (CGH). FU-DDLS-1 cells were spindle or polygonal shaped and possessed oval nuclei and slender cytoplasmic processes. The cultured cells were successfully maintained in vitro for over 90 passages over more than 30 months. The histologic features of heterotransplanted tumors in severe combined immunodeficiency mice were essentially the same as those of the original nonlipogenic sarcoma resembling a malignant fibrous histiocytoma. Both in vitro and in vivo, the cells exhibited immunopositive reaction for mdm2 and p53 proteins. Cytogenetically, FU-DDLS-1 displayed a hypertetraploid karyotype with giant marker chromosomes composed partly of chromosome 12 material. In addition, CGH analysis demonstrated that DNA sequence copy number changes including a gain of 12q12-q21 detected in FU-DDLS-1 were essentially the same as those in the original sarcoma. The FU-DDLS-1 cell line, which exhibits the unique conventional and molecular cytogenetic characteristics of dedifferentiated liposarcoma, should be a particularly useful model for studying the molecular pathogenesis of human dedifferentiated liposarcoma.

Aneuploidy↗
PubMedDate not supplied

Management of liposarcoma occurring in pregnant women.

BACKGROUND: The coexistence of pregnancy and liposarcoma is rare. Only 12 cases of pregnancy-associated liposarcoma have previously been reported in the English-language literature. MATERIALS AND METHODS: We present two cases of liposarcoma occurring in pregnant patients: one myxoid liposarcoma in a 29-year-old woman at 29 weeks' gestation; another well-differentiated liposarcoma in a 44-year-old woman at 22 weeks' gestation. RESULTS: The first patient's infant was delivered by cesarean section after the confirmation of pulmonary maturity of the fetus. The patient was subsequently treated with chemotherapy and radiotherapy. The second patient awaited delivery until 32 weeks' gestation with no treatment of the tumor because she had a low-grade sarcoma. After cesarean section, surgical treatment followed. CONCLUSION: Treatment of sarcomas occurring during pregnancy is difficult. Both mother and fetus should be appropriately managed, depending on the trimester at the time of diagnosis and the histological type and grade of the tumor.

Adult↗
PubMed2006

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

Liposarcoma is the most common soft-tissue malignancy in adults, but the appearance of a liposarcoma in the head and neck region is distinctly unusual. Intraoral liposarcomas represent a particularly interesting subset of this tumor in that (1) they are exceedingly rare and (2) affected patients tend to have a better prognosis than do patients with a similar lesion located elsewhere in the head and neck. An understanding of the histologic subtypes and corresponding clinical behavior of liposarcomas will assist physicians in appropriately managing these patients. Most of these tumors can be effectively treated with conservative surgery. We report the rare case of a well-differentiated liposarcoma arising in the tongue of a 55-year-old man. We also discuss the typical pathologic findings in these malignancies and review the diagnosis, associated controversies, management, and prognosis.

Humans↗
PubMed2006

[Unique massive recurrence liposarcoma of the retroperitoneum].

Liposarcoma constitutes a heterogeneous group of tumors which rarely arise from the retroperitoneal area. They are the single most common tumor in all sarcoma of the retroperitoneum space. Surgery plays a dominant role in the treatment of retroperitoneal liposarcoma. The histologic subtype and margin of resection are prognostic for survival in primary tumors. Local recurrences are the most frequent cause of failure of the surgery. We report the case of a patient with massive recurrences ofretroperitoneal liposarcoma. A 45 year-old male had low-grade liposarcoma of the retroperitoneum which recurred 6 times within 10 years. The next retroperitoneal recurrence tumor excised weighted 25 kg in total. Seven months after resection the patient feels well. We conclude that aggressive surgery in recurrent tumors can produce prolonged survival in patients with low-grade retroperitoneal liposarcoma.

Follow-Up Studies↗