Uterine leiomyosarcomas with osteoclast-like giant cells associated with high expression of RUNX2 and RANKL

Terasaki, Mika; Terasaki, Yasuhiro; Wakamatsu, Kyoko; Kuwahara, Naomi; Yoneyama, Kihei; Kawase, Rieko; Kurose, Kouichi; Toda, Etsuko; Endo, Yoko; Κunugi, Shinobu; Terasaki, Yasuhiro; Shimizu, Akira

doi:10.1007/s00428-020-02996-1

Cited by 3 publications

(8 citation statements)

References 41 publications

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“…Some studies have reported that malignant tumors in other organs with osteoclast-like multinucleated giant cells showed hypervascularity and/or hemorrhage in tumor stroma ( 6 , 24 , 25 ). In our case, the brownish appearance of the gallbladder carcinosarcoma was likely associated with extensive hemorrhage in the stroma.…”

Section: Discussionmentioning

confidence: 99%

“…In our case, the brownish appearance of the gallbladder carcinosarcoma was likely associated with extensive hemorrhage in the stroma. In addition, in certain malignancies (e.g., differentiated hepatocellular carcinoma, uterine leiomyosarcoma, and breast carcinoma) the presence of osteoclast-like multinucleated giant cells has been associated with an aggressive clinical course ( 6 , 26 , 27 ). Hatano et al ( 28 ) reported that osteoclast-like multinucleated giant cells produce vascular endothelial growth factor (VEGF)-C, which promotes tumor growth and lymphangiogenesis.…”

Section: Discussionmentioning

confidence: 99%

“…Malignant tumors with osteoclast-like multinucleated giant cells are also found in various tissues, and RANKL-positive tumor cells have been noted in several neoplasms that develop in the soft tissue, uterus, bladder, and breast (e.g., leiomyosarcoma), as well as sarcomatoid hepatocellular carcinoma ( 6 , 8 , 10 , 25 ). In this case, the expression of RANKL was recognized in tumor cells, especially those around osteoclast-like multinucleated giant cells.…”

Section: Discussionmentioning

confidence: 99%

“…It is known that the differentiation and activation of osteoclastic multinucleated giant cells are induced via interaction with receptor activator of nuclear factor-κB (RANK) and RANK-Ligand (RANKL), which are known to be contained in the tumor necrosis factor (TNF) and TNF receptor (TNFR) superfamilies. In bone tissue, osteoblasts secrete RANKL, and RANKL binds to RANK on monocyte/macrophage lineage progenitor cells for osteoclastic differentiation ( 6 ). Recently, certain carcinomas were reported to express RANK, suggesting an association between RANK-RANKL signaling and tumor development ( 7 ).…”

Section: Introductionmentioning

confidence: 99%

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Case Report: A Case of Gallbladder Carcinosarcoma With Osteoclast-like Multinucleated Giant Cells that Was Associated With RANK‐RANKL Signaling

Niwa¹,

Tomita²,

Watanabe³

et al. 2022

Pathol. Oncol. Res.

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Introduction: Gallbladder carcinosarcoma with osteoclast-like multinucleated giant cells is known to be most uncommon form of gallbladder cancer. Owing to its rarity, the pathogenesis of gallbladder carcinosarcoma with osteoclast-like multinucleated giant cells is largely unknown.Case Presentation: We present a case of carcinosarcoma with osteoclast-like multinucleated giant cells in the gallbladder. A 57-year-old woman visited our hospital due to jaundice. An examination revealed calculous cholecystitis and gallbladder carcinoma. After cholecystectomy, macroscopic examination disclosed one whitish mass and another distinct brown and pendulous mass in the body of the gallbladder. A pathological examination revealed that each mass had a different histological type: adenosquamous carcinoma and carcinosarcoma with osteoclast-like multinucleated giant cells. Immunohistochemistry revealed that these osteoclast-like multinucleated giant cells are CD68(+), CD163(−), and MIB-1(−). In addition, the osteoclast-like multinucleated giant cells showed the strong expression of RANK and sarcoma cells around the osteoclast-like multinucleated giant cells, were positive for RANKL. Furthermore, RUNX2 was positive for some sarcoma cells. The result indicated that osteoclastic and osteoblast-like differentiation occurred in our case.Conclusion: To our knowledge, this is the first case to show the interaction of RANK-RANKL signaling in gallbladder carcinosarcoma with osteoclast-like multinucleated giant cells.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Case Report: A Case of Gallbladder Carcinosarcoma With Osteoclast-like Multinucleated Giant Cells that Was Associated With RANK‐RANKL Signaling

Niwa¹,

Tomita²,

Watanabe³

et al. 2022

Pathol. Oncol. Res.

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“…Leiomyosarcoma, one of the most common sarcomas arising in the uterus, is pathologically classified as conventional, epithelioid, or myxoid [1]. They sometimes contain unusual cellular components, such as rhabdoid cells or osteoclast-like giant cells [2]. However, their clinical characteristics are not well known because the number of reported cases is too low.…”

Section: Introductionmentioning

confidence: 99%

Clinically Aggressive Uterine Epithelioid Leiomyosarcoma with Rhabdomyoblastic Differentiation and High Proliferation Rate: A Case Report

Kim

2023

Am J Case Rep

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Objective:Rare disease Background:Leiomyosarcoma is the most common uterine sarcoma. Leiomyosarcoma is classified into conventional leiomyosarcoma, epithelioid leiomyosarcoma, and myxoid leiomyosarcoma. Leiomyosarcomas with rhabdoid features have been rarely reported. Herein, we report a case of uterine leiomyosarcoma with rhabdoid features. Case Report:A 58-year-old Korean woman presented with acute abdominal pain. Computed tomography and magnetic resonance imaging of the pelvis revealed a large solid mass in the posterior wall of the uterus that extended to the uterine cervix. The patient underwent total hysterectomy with bilateral salpingo-oophorectomy and tumorectomy. Microscopic and immunohistochemical examination of the tumor revealed leiomyosarcoma with rhabdoid features and high proliferation rate. Next-generation sequencing showed PI3K amplification and ERBB2 amplification. Postoperative abdominal and pelvic computed tomography performed 3 weeks after the operation showed a mass at the vaginal stump that was attached to the urinary bladder and rectum. The patient underwent pelvic exenteration of remnant vaginal stump, rectum, and urinary bladder with loop ileostomy, and was diagnosed with recurrent leiomyosarcoma. One month later, after the second operation, a 13-cm recurrent mass was noted on the computed tomography. Chemotherapy was not done and the patient died during supportive treatment 7 months after diagnosis. Conclusions:This case, which is a uterine leiomyosarcoma with rhabdoid features and high proliferation rate, recurred very fast, within 1 month, and showed an aggressive clinical course. The molecular classification and postoperative therapy are not well established in uterine leiomyosarcomas. Further studies are required to clarify the clinical and pathological characteristics of leiomyosarcomas.

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Uterine Leiomyosarcoma With Osteoclast-like Giant Cells: Report of 2 Cases and Review of Literature

Chen

Yung

et al. 2023

International Journal of Gynecological Pathology

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Leiomyosarcoma (LMS) with osteoclast-like giant cells (OLGCs) is a rare entity with only 18 reported cases thus far. It is not known whether these OLGCs are a reactive or malignant component of LMS. Herein we describe the clinical, histologic, and molecular characteristics of 2 cases of LMS with OLGCs and perform a brief literature review. In 2 of our cases, the OLGCs, marked with CD68, had a low proliferation index with Ki67 and did not show diffuse positivity for smooth muscle markers by immunohistochemistry. By next-generation sequencing, one case harbored a clinically significant TP53 mutation, which has been reported in a significant subset of conventional LMSs. In this case, based on immunohistochemistry, OLGCs showed different molecular alterations as compared with LMS. Although we did not show a distinct immunophenotype or molecular profile for LMS with OLGCs, this study provides additional data on this rare entity.

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Uterine leiomyosarcomas with osteoclast-like giant cells associated with high expression of RUNX2 and RANKL

Cited by 3 publications

References 41 publications

Case Report: A Case of Gallbladder Carcinosarcoma With Osteoclast-like Multinucleated Giant Cells that Was Associated With RANK‐RANKL Signaling

Case Report: A Case of Gallbladder Carcinosarcoma With Osteoclast-like Multinucleated Giant Cells that Was Associated With RANK‐RANKL Signaling

Clinically Aggressive Uterine Epithelioid Leiomyosarcoma with Rhabdomyoblastic Differentiation and High Proliferation Rate: A Case Report

Uterine Leiomyosarcoma With Osteoclast-like Giant Cells: Report of 2 Cases and Review of Literature

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