Base editing-derived models of human WDR34 and WDR60 disease alleles replicate retrograde intraflagellar transport (IFT) and hedgehog signaling defects
Abstract Cytoplasmic Dynein-2 / IFT-dynein is the only known retrograde motor for intraflagellar transport. Dysfunction of the two intermediate dynein chains WDR34 and WDR60 causes Short Rib Thoracic Dystrophy (SRTD), a human skeletal chondrodysplasias with high lethality. However, individual protei...
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| Main Authors: | , , , , , , , , , , |
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| Format: | Artigo |
| Language: | Inglês |
| Published: |
Nature Portfolio
2026-07-01
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| Series: | Communications Biology |
| Online Access: | https://doi.org/10.1038/s42003-026-10507-2 |
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