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1. SMCHD1 has separable roles in chromatin architecture and gene silencing that could be targeted in disease

2. SMCHD1’s ubiquitin-like domain is required for N-terminal dimerization and chromatin localization.

3. Relating SMCHD1 structure to its function in epigenetic silencing.

4. Crystal structure of the hinge domain of Smchd1 reveals its dimerization mode and nucleic acid–binding residues.

5. SMCHD1 is involved in de novo methylation of the DUX4-encoding D4Z4 macrosatellite.

6. FSHD2- and BAMS-associated mutations confer opposing effects on SMCHD1 function.

7. SMCHD1 is involved in de novo methylation of the DUX4-encoding D4Z4 macrosatellite

8. FSHD2- and BAMS-associated mutations confer opposing effects on SMCHD1 function

9. MORC2 phosphorylation fine tunes its DNA compaction activity.

10. De novo mutations in SMCHD1 cause Bosma arhinia microphthalmia syndrome and abrogate nasal development.

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