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• W3048694567 abstract "An 83-year-old man was referred to our hospital for a hard nontender mass on the right side of the abdomen. Apart from a higher CA19-9 level of 49.50 U/mL (normal range, 0-37.0 U/mL), the remainder of the laboratory tests were unremarkable. The abdominal computerized tomography (CT) revealed a gallbladder mass (3.2 × 4.3 × 6.0 cm), with irregular enhancement and areas of fat density (Figure 1A). Cholecystectomy with partial hepatectomy was performed on the patient (Figure 1B). Gross examination revealed an enlarged and asymmetric mass (Figure 1C,D). On microscopic examination, there were tiny tissue fragments with infiltrating atypical cells having pleomorphic nuclei and focal lipogenic differentiation (Figure 1E). Immunohistochemical analysis was positive for cyclin dependent kinase 4 (CDK4) and murine double minute 2 (MDM2) (Figure 1F,G). Therefore, the diagnosis for this sarcoma was dedifferentiated liposarcoma (DDLS) of the gallbladder. The patient was discharged 11 days after surgery and has been undergoing regular follow-up for 4 years until now. Liposarcoma is a malignant mesenchymal tumor that typically occurs in the lower extremities and retroperitoneum. Liposarcoma arising in the gastrointestinal tract is rare, with one report indicating that the incidence at autopsy is about 0.1 to 5.8%.1 Primary gallbladder liposarcoma is even rarer, with only six cases being reported in literature to date and only two cases of DDLS of the gallbladder in the world. The clinical presentation of gallbladder liposarcoma is nonspecific. DDLS of the gallbladder usually requires several years to grow and is often detected incidentally.2 Making a preoperative diagnosis can be extremely difficult due to normal laboratory tests and only an enhancing nodule, which resembles adipose tissue on CT imaging. Surgery remains the mainstay of treatment for DDLS, same as other subtypes of liposarcoma. The tumor is removed with 1-cm margins of normal tissue.3 Regarding the first case of DDLS of the gallbladder, da Costa et al performed a cholecystectomy with partial hepatectomy and the patient remained alive without signs of recurrence 8 months after surgery. Though the DDLS is a high grade and aggressive tumor, chemotherapy and radiotherapy do not improve the course of the disease, except in cases of myxoid liposarcoma, which is more sensitive to chemoradiotherapy.4 The DDLS is characterized by an atypical lipomatous neoplasm/well-differentiated liposarcoma (ALN/WDL) juxtaposed to a high-grade nonlipogenic sarcomatous area and consistently overexpressed MDM2 genes. The dedifferentiated nonlipogenic sarcomatous area frequently resembles an undifferentiated pleomorphic sarcoma or a high-grade fibrosarcoma. Although infrequent, some DDLS can demonstrate features of a low-grade sarcoma or mimic fibromatosis, characterized by uniform fibroblastic spindle cells with mild nuclear atypia. Approximately 5% of DDLS possess a heterologous component, and most of them show myogenic differentiation. Similar to ALN/WDL, DDLS expresses a giant marker and ring chromosomes, which include the amplified sequence of 12q13-15.5 MDM2 and CDK4 genes, usually located in this region, are consistently amplified in DDLS. The immunohistochemical study of these two markers can serve as the first-line screening tool whenever DDLS is suspected, as the positive results can be confirmed using fluorescence in-situ hybridization (FISH) of MDM2. The prognosis of patients with primary gallbladder liposarcoma is expected to be poor as these are aggressive-behaving tumors that generally present with high rates of recurrence and metastasis. The authors declare no potential conflict of interest." @default.
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• W3048694567 date "2020-08-12" @default.
• W3048694567 modified "2023-09-26" @default.
• W3048694567 title "Dedifferentiated liposarcoma of the gallbladder" @default.
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• W3048694567 doi "https://doi.org/10.1002/kjm2.12295" @default.
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