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Abstract: Objective: The present study used single-cell RNA sequencing (scRNA-seq) to characterize the cellular composition of OCS and identify its molecular characteristics. Methods: scRNA-seq was performed in resected primary OCS for an in-depth analysis of tumor cells and the tumor microenvironment. Immunohistochemistry staining was used for validation. The scRNA-seq data of OCS was compared with that of HGSOC tumors and other OCS tumors. Results: Both malignant epithelial and malignant mesenchymal cells were observed in the OCS patient of this study. We identified four epithelial cell subclusters with different biological roles. Among them, epithelial subcluster 4 presented high levels of BRCA1 and TOP2A expression and was related to drug resistance and cell cycle. We analyzed the interaction between epithelial and mesenchymal cells and found that FGF and PTN signaling were the main pathways contributing to communication between these cells. Moreover, we compared the malignant epithelial and mesenchymal cells of this OCS tumor with our previous published high-grade serous ovarian carcinoma (HGSOC) scRNA-seq data and published OCS data. All the epithelial subclusters in the OCS tumor could be found in the HGSOC samples. Notably, the mesenchymal subcluster C14 exhibited specific expression patterns in the OCS tumor, characterized by elevated expression of CYP24A1, CYP23A1, CCK, BMP7, PTN, WIF1, and IGF2. Moreover, this subcluster showed distinct characteristics when compared to both another previously published OCS tumor and normal ovarian tissue. Conclusions: This study provides the single-cell transcriptomics signature of human OCS, which constitutes a new resource for elucidating OCS diversity.