Journal
SCIENTIFIC REPORTS
Volume 12, Issue 1, Pages -Publisher
NATURE PORTFOLIO
DOI: 10.1038/s41598-022-22405-8
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Funding
- Program for Intractable Disease Research Utilizing Disease-Specific iPS Cells from the Japan Science and Technology (JST) agency
- Japan Agency for Medical Research and Development (AMED) [JP15bm0404009, JP16bm0609002]
- Project for Technological Development of the Research Center Network for Realization of Regenerative Medicine (RCNRRM) - AMED [JP17bm0404018, JP20bm0404036]
- Acceleration Program for Intractable Diseases Research Utilizing Disease-Specific iPS Cells of RCNRRM - AMED [JP19bm0804011]
- Ministry of Education, Culture, Sports, Science, and Technology of Japan (MEXT) [JP17K09878, JP20K08859]
- Nagoya University Hospital Funding for Clinical Research [71004136]
- Fusion Oriented Research for disruptive Science and Technology (FOREST) by Japan Science and Technology Agency (JST)
- Hori Sciences and Arts Foundation
- Toyoaki Scholarship Foundation
- Daiko Foundation
- Nitto Foundation
- Suzuken Memorial Foundation
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In this study, a human in vitro model of familial neurohypophyseal diabetes insipidus (FNDI) was developed by converting human induced pluripotent stem cells (iPSCs) using a naive conversion kit. The converted iPSCs showed improved cell survival and differentiation into AVP neurons, allowing for the study of FNDI pathology.
Familial neurohypophyseal diabetes insipidus (FNDI) is a degenerative disease of vasopressin (AVP) neurons. Studies in mouse in vivo models indicate that accumulation of mutant AVP prehormone is associated with FNDI pathology. However, studying human FNDI pathology in vivo is technically challenging. Therefore, an in vitro human model needs to be developed. When exogenous signals are minimized in the early phase of differentiation in vitro, mouse embryonic stem cells (ESCs)/induced pluripotent stem cells (iPSCs) differentiate into AVP neurons, whereas human ESCs/iPSCs die. Human ESCs/iPSCs are generally more similar to mouse epiblast stem cells (mEpiSCs) compared to mouse ESCs. In this study, we converted human FNDI-specific iPSCs by the naive conversion kit. Although the conversion was partial, we found improved cell survival under minimal exogenous signals and differentiation into rostral hypothalamic organoids. Overall, this method provides a simple and straightforward differentiation direction, which may improve the efficiency of hypothalamic differentiation.
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