Isogenic hiPSC models of Turner syndrome development reveal shared roles of inactive X and Y in the human cranial neural crest network.

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Tác giả: Darcy T Ahern, Erin C Banda, Prakhar Bansal, Owen M Chambers, Isaac V Faustino, Heather R Glatt-Deeley, Yuvabharath Kondaveeti, Rachael E Massey, Stefan F Pinter

Ngôn ngữ: eng

Ký hiệu phân loại: 623.822 *Modern wind-driven ships

Thông tin xuất bản: United States : American journal of human genetics , 2025

Mô tả vật lý:

Bộ sưu tập: NCBI

ID: 91532

Viable human aneuploidy can be challenging to model in rodents due to syntenic boundaries or primate-specific biology. Human monosomy-X (45,X) causes Turner syndrome (TS), altering craniofacial, skeletal, endocrine, and cardiovascular development, which in contrast remain unaffected in X-monosomic mice. To learn how monosomy-X may impact embryonic development, we turned to 45,X and isogenic euploid human induced pluripotent stem cells (hiPSCs) from male and female mosaic donors. Because the neural crest (NC) is hypothesized to give rise to craniofacial and cardiovascular changes in TS, we assessed differential expression of hiPSC-derived anterior NC cells (NCCs). Across three independent isogenic panels, 45,X NCCs show impaired acquisition of PAX7
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