Intellectual disability‐associated dBRWD 3 regulates gene expression through inhibition of HIRA / YEM ‐mediated chromatin deposition of histone H3.3
| العنوان: | Intellectual disability‐associated |
|---|---|
| المؤلفون: | Li-Kai Chen, June-Tai Wu, Mei-Ju May Chen, Benjamin Loppin, Hsuan Liu, Bertrand Chin-Ming Tan, Wei-Yu Chen, Hsiu-Hsiang Lee, Kuei‐Yan Liu, Hsueh-Tzu Shih, Tsung‐Han Tsai, Chien-Yu Chen, Ounissa Aït-Ahmed, Zong-Siou Shih |
| المصدر: | EMBO reports. 16:528-538 |
| بيانات النشر: | EMBO, 2015. |
| سنة النشر: | 2015 |
| مصطلحات موضوعية: | 0106 biological sciences, Genetics, 0303 health sciences, Mutant, Biology, 010603 evolutionary biology, 01 natural sciences, Biochemistry, Chromatin, 03 medical and health sciences, Histone H3, 0302 clinical medicine, Histone, Gene expression, biology.protein, Nucleosome, Epigenetics, Molecular Biology, Gene, 030217 neurology & neurosurgery, 030304 developmental biology |
| الوصف: | Many causal mutations of intellectual disability have been found in genes involved in epigenetic regulations. Replication-independent deposition of the histone H3.3 variant by the HIRA complex is a prominent nucleosome replacement mechanism affecting gene transcription, especially in postmitotic neurons. However, how HIRA-mediated H3.3 deposition is regulated in these cells remains unclear. Here, we report that dBRWD3, the Drosophila ortholog of the intellectual disability gene BRWD3, regulates gene expression through H3.3, HIRA, and its associated chaperone Yemanuclein (YEM), the fly ortholog of mammalian Ubinuclein1. In dBRWD3 mutants, increased H3.3 levels disrupt gene expression, dendritic morphogenesis, and sensory organ differentiation. Inactivation of yem or H3.3 remarkably suppresses the global transcriptome changes and various developmental defects caused by dBRWD3 mutations. Our work thus establishes a previously unknown negative regulation of H3.3 and advances our understanding of BRWD3-dependent intellectual disability. |
| تدمد: | 1469-3178 1469-221X |
| الوصول الحر: | https://explore.openaire.eu/search/publication?articleId=doi_dedup___::fb4a1ae9105e6a08a654a44c25b76dcaTest https://doi.org/10.15252/embr.201439092Test |
| حقوق: | OPEN |
| رقم الانضمام: | edsair.doi.dedup.....fb4a1ae9105e6a08a654a44c25b76dca |
| قاعدة البيانات: | OpenAIRE |
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