Home > Publications Database > X-linked deletion of Crossfirre, Firre, and Dxz4 in vivo uncovers diverse phenotypes and combinatorial effects on autosomes. > print

001     273928
005     20250120102327.0
024 7 _ |a 10.1038/s41467-024-54673-5
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037 _ _ |a DZNE-2024-01402
041 _ _ |a English
082 _ _ |a 500
100 1 _ |a Hasenbein, Tim P
|0 0000-0001-7028-6559
|b 0
245 _ _ |a X-linked deletion of Crossfirre, Firre, and Dxz4 in vivo uncovers diverse phenotypes and combinatorial effects on autosomes.
260 _ _ |a [London]
|c 2024
|b Nature Publishing Group UK
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520 _ _ |a The lncRNA Crossfirre was identified as an imprinted X-linked gene, and is transcribed antisense to the trans-acting lncRNA Firre. The Firre locus forms an inactive-X-specific interaction with Dxz4, both loci providing the platform for the largest conserved chromatin structures. Here, we characterize the epigenetic profile of these loci, revealing them as the most female-specific accessible regions genome-wide. To address their in vivo role, we perform one of the largest X-linked knockout studies by deleting Crossfirre, Firre, and Dxz4 individually and in combination. Despite their distinct epigenetic features observed on the X chromosome, our allele-specific analysis uncovers these loci as dispensable for imprinted and random X chromosome inactivation. However, we provide evidence that Crossfirre affects autosomal gene regulation but only in combination with Firre. To shed light on the functional role of these sex-specific loci, we perform an extensive standardized phenotyping pipeline and uncover diverse knockout and sex-specific phenotypes. Collectively, our study provides the foundation for exploring the intricate interplay of conserved X-linked loci in vivo.
536 _ _ |a 352 - Disease Mechanisms (POF4-352)
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588 _ _ |a Dataset connected to CrossRef, PubMed, , Journals: pub.dzne.de
650 _ 7 |a RNA, Long Noncoding
|2 NLM Chemicals
650 _ 2 |a Animals
|2 MeSH
650 _ 2 |a RNA, Long Noncoding: genetics
|2 MeSH
650 _ 2 |a RNA, Long Noncoding: metabolism
|2 MeSH
650 _ 2 |a Female
|2 MeSH
650 _ 2 |a Male
|2 MeSH
650 _ 2 |a Phenotype
|2 MeSH
650 _ 2 |a X Chromosome Inactivation: genetics
|2 MeSH
650 _ 2 |a Genes, X-Linked
|2 MeSH
650 _ 2 |a X Chromosome: genetics
|2 MeSH
650 _ 2 |a Genomic Imprinting
|2 MeSH
650 _ 2 |a Mice
|2 MeSH
650 _ 2 |a Mice, Knockout
|2 MeSH
650 _ 2 |a Epigenesis, Genetic
|2 MeSH
650 _ 2 |a Alleles
|2 MeSH
650 _ 2 |a Mice, Inbred C57BL
|2 MeSH
700 1 _ |a Hoelzl, Sarah
|0 0000-0003-0308-8522
|b 1
700 1 _ |a Smith, Zachary D
|0 0000-0002-9283-1957
|b 2
700 1 _ |a Gerhardinger, Chiara
|0 0000-0002-6113-0381
|b 3
700 1 _ |a Gonner, Marion O C
|b 4
700 1 _ |a Aguilar-Pimentel, Antonio
|b 5
700 1 _ |a Amarie, Oana V
|0 0000-0003-1705-6812
|b 6
700 1 _ |a Becker, Lore
|0 0000-0002-6890-4984
|b 7
700 1 _ |a Calzada-Wack, Julia
|b 8
700 1 _ |a Dragano, Nathalia R V
|b 9
700 1 _ |a da Silva-Buttkus, Patricia
|b 10
700 1 _ |a Garrett, Lillian
|b 11
700 1 _ |a Hölter, Sabine M
|0 0000-0003-4878-5241
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700 1 _ |a Kraiger, Markus
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700 1 _ |a Östereicher, Manuela A
|b 14
700 1 _ |a Rathkolb, Birgit
|0 0000-0003-1239-0547
|b 15
700 1 _ |a Sanz-Moreno, Adrián
|0 0000-0001-9478-5432
|b 16
700 1 _ |a Spielmann, Nadine
|b 17
700 1 _ |a Wurst, Wolfgang
|0 P:(DE-2719)2000028
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700 1 _ |a Gailus-Durner, Valerie
|0 0000-0002-6076-0111
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700 1 _ |a Fuchs, Helmut
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700 1 _ |a Hrabě de Angelis, Martin
|b 21
700 1 _ |a Meissner, Alexander
|0 0000-0001-8646-7469
|b 22
700 1 _ |a Engelhardt, Stefan
|0 0000-0001-5378-8661
|b 23
700 1 _ |a Rinn, John L
|0 0000-0002-7231-7539
|b 24
700 1 _ |a Andergassen, Daniel
|0 0000-0003-1196-4289
|b 25
773 _ _ |a 10.1038/s41467-024-54673-5
|g Vol. 15, no. 1, p. 10631
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856 4 _ |y OpenAccess
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910 1 _ |a Deutsches Zentrum für Neurodegenerative Erkrankungen
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