Pleomorphic Liposarcoma Arising in a Lipoleiomyosarcoma of the Uterus: Report of a Case With Genetic Profiling by a Next Generation Sequencing Panel

Schoolmeester, J. Kenneth; Stamatakos, Michael D.; Moyer, Ann M.; Park, Kay J.; Fairbairn, M. Gerardi; Fader, Amanda N.

doi:10.1097/pgp.0000000000000241

Cited by 12 publications

(4 citation statements)

References 15 publications

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“…Since the 1977 report by Fujii et al [1], 19 cases of uterine liposarcoma [1][2][3][4][5][6][7][8][9][10][11][12][13][14][15][16], including this report, have been confirmed (Table 1). To summarize the reported cases, uterine liposarcoma generally occurs during or after menopause.…”

Section: Discussionsupporting

confidence: 62%

“…Fadare and Khabele [12] chose a chemotherapy regimen of gemcitabine and docetaxel at recurrence. Schoolmeester et al [14] opted for a chemotherapy regimen of gemcitabine and docetaxel at recurrence.…”

Section: Discussionmentioning

confidence: 99%

“…After surgical removal, the lesion was histopathologically diagnosed as liposarcoma. The case has been reported with a literature review [1][2][3][4][5][6][7][8][9][10][11][12][13][14][15][16].…”

Section: Introductionmentioning

confidence: 99%

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Well-Differentiated Liposarcoma That Increased in Size after Menopause: A Case Report and a Review of the Literature

Hoshino,

Takada,

Sugihara

et al. 2024

Case Reports in Obstetrics and Gynecology

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This study reports a case of uterine liposarcoma together with a literature review. At 52 years old, our patient was diagnosed with lipoleiomyoma by MRI. A mass (39×32×41 mm3) protruding from the anterior wall of the uterine body was observed. When the patient was 58, her previous doctor found that the tumor had grown, and she was referred to the gynecology department of our hospital. On MRI, the major diameter was 1.23-fold longer and the volume was 1.85-fold higher compared with the prior imaging findings. Diffusion-weighted images revealed no significant anomalous signals. Thus, malignant tumors were included in the differential diagnosis. The patient consented to total abdominal hysterectomy and bilateral salpingo-oophorectomy. The mass on the anterior wall remained completely in the myometrium. No implantation was found in the abdominal cavity, and ascites was not detected. No bleeding or necrosis was observed on the cut surface. Histopathologically, differences in the sizes of adipocytes and stromal cells were identified. There were irregularities in the nuclear findings. The immunohistochemical findings were as follows: CDK4 (+), desmin (+), S100p (−), and Ki−67=1%. Therefore, a diagnosis of well-differentiated liposarcoma was rendered. The lesion was localized in the uterus, and it was completely removed during surgery. Well-differentiated liposarcoma of uterine primary has no possibility of recurrence following complete resection, and thus, the patient underwent follow-up without additional treatment. No metastasis or recurrence has been observed for 10 months after surgery.

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Section: Discussionsupporting

confidence: 62%

Section: Discussionmentioning

confidence: 99%

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Well-Differentiated Liposarcoma That Increased in Size after Menopause: A Case Report and a Review of the Literature

Hoshino,

Takada,

Sugihara

et al. 2024

Case Reports in Obstetrics and Gynecology

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“…Many reported patients with liposarcoma have a neoplasm originating from the uterine cervix rather than the corpus. To the best of our knowledge, there are only 9 patients, including ours, reported in the English-language literature with a liposarcoma occurring in the uterine corpus( Bapat & Brustein, 1989 ; Sosnik et al, 2006 ; Hong et al, 2008 ; McDonald et al, 2011 ; Schoolmeester et al, 2016 ; Fadare & Khabele, 2011 ) ( Table 1 ). Patients with lipoleiomyosarcoma or mesenchymal malignant tumors having only a small part of liposarcomatous differentiation are excluded from this review.…”

Section: Discussionmentioning

confidence: 88%

Liposarcoma of the uterine corpus: A case report and literature review

Kiuchi

Hasegawa

Ochiai

et al. 2018

Gynecologic Oncology Reports

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Liposarcoma of the uterine corpus is extremely rare. We performed a laparotomy on a 55-year-old woman with the complaints of abdominal distension and genital bleeding who was found to have a uterine tumor, 17 × 16 cm in diameter. The preoperative diagnosis was a lipoma or lipoleiomyoma of the uterine corpus. However, pathological examination revealed proliferation of mature adipocytes and lipoblast-like atypical cells with small, weakly pleomorphic nuclei and foamy or vacuolated cytoplasm present within a fibrous septum. Immunohistochemistry showed that the tumor cells were focally positive for mouse double minute 2 homolog (MDM2). The final pathological diagnosis was a well-differentiated liposarcoma of International Federation of Gynecology and Obstetrics (FIGO) stage IB (pT1bNxM0). On magnetic resonance imaging (MRI), T1 -weighted and fat-saturated images showed high and low intensity in the tumor, respectively, suggesting that this tumor contained a fat component. The septum inside the tumor had a contrast enhancement on T1-weighted, gadolinium-enhanced imaging. The septum was nonuniformly thickened and partially nodular. In hindsight, these findings may have suggested a well-differentiated liposarcoma in the uterine corpus rather than a lipoma or lipoleiomyoma. Clinicians should be aware of the possibility of a liposarcoma of the uterine corpus when a neoplasm contains adipose tissue and a nonuniformly thickened or partially nodular septum on MRI.

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PLAG1‐rearrangement in a uterine leiomyosarcoma with myxoid stroma and heterologous differentiation

Thiryayi

Turashvili

Latta

et al. 2021

Genes Chromosomes & Cancer

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A variety of molecular alterations have been reported in uterine leiomyosarcomas, but most are considered nondiagnostic. There are, however, rare exceptions including PLAG1 rearrangement which has recently been identified in a subset of myxoid leiomyosarcomas. A 41-year-old woman presented with symptoms of a fibroid. She underwent a myomectomy which revealed a high-grade uterine sarcoma with areas of myxoid stroma and heterologous elements. The tumor expressed desmin, smooth muscle actin, H-caldesmon, and estrogen and progesterone receptors. RNA sequencing revealed a novel TRIM13-PLAG1 fusion gene which was subsequently independently confirmed by fluorescence in situ hybridization. On further evaluation the patient was found to have multiple pulmonary metastases and died due to disease progression shortly after diagnosis. This report describes a novel fusion partner of PLAG1 in a uterine leiomyosarcoma with myxoid leiomyosarcoma and heterologous elements, thereby broadening the spectrum of morphologic and genetic findings within this rare group of neoplasms.

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Pleomorphic Liposarcoma Arising in a Lipoleiomyosarcoma of the Uterus: Report of a Case With Genetic Profiling by a Next Generation Sequencing Panel

Cited by 12 publications

References 15 publications

Well-Differentiated Liposarcoma That Increased in Size after Menopause: A Case Report and a Review of the Literature

Well-Differentiated Liposarcoma That Increased in Size after Menopause: A Case Report and a Review of the Literature

Liposarcoma of the uterine corpus: A case report and literature review

PLAG1‐rearrangement in a uterine leiomyosarcoma with myxoid stroma and heterologous differentiation

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