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. 2025 Jul 24;18:47. doi: 10.1186/s13072-025-00612-7

Table 2.

Developmental disorders with affected iDMRs

Disorder Gene(s) involved Category Patients number Affected iDMRs
Cerebellar ataxia, deafness, and narcolepsy, autosomal dominant (ADCADN) DNMT1 DNA methyltransferase 5 IGF2:Ex9
Coffin–Siris syndrome 4 (CSS4_c.2650) SMARCA4 Chromatin remodeler 3 WDR27:Int13
Down Syndrome (DS)

HMGN1

MIS18A

AIRE

USP16

PRMT2

DNMT3L

RBM11

CHAF1B

BRWD1

Chromatin remodeler

Chromatin remodeler

Transcription factor

Histone ubiquitination

Histone methyltransferase

DNA methyltransferase

Alternative splicing

Chromatin remodeler

Histone reader

40 IGF1R:Int2 NNAT:TSS
Floating-Harbour syndrome (FLHS) SRCAP Chromatin remodeler 21 IGF1R:Int2 NNAT:TSS
Genitopatellar syndrome (GTPTS) KAT6B Histone acetyltransferase 4 VTRNA2-1
Helsmoortel–Van der Aa syndrome (ADNP syndrome (Central) (HVDAS_C) ADNP Transcription factor 14 PPIEL:Ex1
Helsmoortel–Van der Aa syndrome (ADNP syndrome (Terminal) (HVDAS_T) ADNP Transcription factor 21 PPIEL:Ex1
Hunter–McAlpine craniosynostosis syndrome (HMA)

NPM1

UIMC

NSD1

Histone chaperone

Histone reader

Histone methyltransferase

8 SNRPN:Int1-DMR2
Immunodeficiency, centromeric instability, facial anomalies syndrome 1 (ICF1) DNMT3B DNA methyltransferase 8 DIRAS3:Ex2 HTR5A:TSS IGF1R:Int2 MAGEL2:TSS MKRN3:TSS NDN:TSS SNRPN:alt-TSS SNRPN:Int1-DMR2 ZNF597:TSS
Immunodeficiency, centromeric instability, facial anomalies syndrome 2,3,4 (ICF2-3-4)

ZBTB24,

CDCA7, HELLS

Transcription factor and chromatin remodelers 7

MKRN3:TSS NDN:TSS SNRPN:alt-TSS SNRPN:Int1-DMR1

SNRPN:Int1-DMR2

Intellectual developmental disorder, X-linked, syndromic, Snyder–Robinson type (MRXSSR) SMS Enzyme 17 VTRNA2-1
Luscan–Lumish syndrome (LLS) SETD2 Histone methyltransferase 4 INPP5F:Int2 NNAT:TSS PPIEL:Ex1
Rahman syndrome (RMNS) HIST1H1E Linker histone 9 NNAT:TSS
Tatton–Brown–Rahman syndrome (TBRS) DNMT3A DNA methyltransferase 30 NNAT:TSS
Williams–Beuren deletion syndrome (Williams)

BAZ1B

GTF2I

Histone phosphorylation

Transcription factor

22 IGF1R:Int2