ArticleNeurology(R) neuroimmunology & neuroinflammation2025
Single-Cell RNA Sequencing in Incontinentia Pigmenti With Neonatal Encephalopathy Reveals Broad Immune Activation Moderated by Steroids.
Article in Neurology(R) neuroimmunology & neuroinflammation, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 4 papers.
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Who cites it
4 citing papers in PubMed.
- Early brain-penetrant immunotherapy reverses interferon signature and improves motor outcome in a case of ADAR1-related Aicardi-Goutières syndrome.Clinical & translational immunology · 2026Article
- cGAS inhibitor IMSB301 modifies interferon signalling in peripheral mononuclear cells ofClinical & translational immunology · 2026Article
- IV Immunoglobulin Is Associated With Epigenetic, Ribosomal, and Immune Changes in Pediatric Acute-Onset Neuropsychiatric Syndrome.Neurology(R) neuroimmunology & neuroinflammation · 2025Article
- Single-Cell RNA Seq in Sydenham Chorea Shows B Cell HLA-DR/DQ Upregulation and Plasma Cell Proteasomal Activation.Annals of clinical and translational neurology · 2025Article
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Authors and funding
9 authors.
Funding
No grant is acknowledged in the PubMed record.
Abstract
BACKGROUND AND
objectivesDNA variations in the NF-kappa-B essential modulator (
methodsSingle-cell RNA (ribonucleic acid) sequencing (scRNAseq), using the HIVE single-cell system, was performed on a neonate with IP, before and after steroid treatment, compared with a sex-matched healthy control toddler.
resultsA total of 20,411 cells were sequenced and clustered into 10 cell types. In IP compared with control, upregulated significant gene set enrichment analysis gene ontology pathways (FDR <0.05) included defense response, complement activation, humoral immune response, and phagocytosis across all cell types. After steroid treatment, these pathways were predominantly downregulated in monocytes and neutrophils. The upregulated genes in IP that became downregulated after steroid treatment were interferon-related genes, oligoadenylate synthases, and immunoglobulin genes. DISCUSSION: IP-associated loss of
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