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Related Experiment Video

Updated: Jul 3, 2026

Genome-wide Analysis of HDAC Inhibitor-mediated Modulation of microRNAs and mRNAs in B Cells Induced to Undergo Class-switch DNA Recombination and Plasma Cell Differentiation
11:06

Genome-wide Analysis of HDAC Inhibitor-mediated Modulation of microRNAs and mRNAs in B Cells Induced to Undergo Class-switch DNA Recombination and Plasma Cell Differentiation

Published on: September 20, 2017

Longitudinal Single-Cell Transcriptomic Profiling Reveals Dynamic Immune Cell Alterations During Burosumab Therapy in

Yue Xie1,2, Li Li2,3, Rong Li2,3

  • 1Chongqing Key Laboratory of Child Neurodevelopment and Cognitive Disorders Pediatric Research Institute, Children's Hospital of Chongqing Medical University Chongqing China.

Pediatric Discovery
|July 2, 2026
PubMed
Summary

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Burosumab treatment for X-linked hypophosphatemia (XLH) alters immune cell dynamics, normalizing elevated T helper 2 and regulatory T cells while impacting natural killer cells. This study reveals key immunological changes during XLH therapy.

Area of Science:

  • Immunology
  • Genetics
  • Pediatrics

Background:

  • X-linked hypophosphatemia (XLH) is a rare genetic disorder impacting bone mineralization due to PHEX gene mutations and elevated FGF23.
  • Burosumab, an anti-FGF23 antibody, is effective but its immunological effects are unknown.

Purpose of the Study:

  • To investigate peripheral immune cell alterations during burosumab therapy in pediatric XLH patients.
  • To characterize the immunological dynamics associated with treatment response in XLH.

Main Methods:

  • Longitudinal single-cell RNA sequencing (scRNA-seq) of peripheral blood mononuclear cells from XLH patients and healthy controls across five time points.
  • Bioinformatic analysis including clustering, pseudotime trajectory, gene expression, and cell-cell communication analysis.
Keywords:
BurosumabX‐linked hypophosphatemialongitudinal studyosteoclast differentiationsingle‐cell RNA sequencing

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Single-cell Analysis of Immunophenotype and Cytokine Production in Peripheral Whole Blood via Mass Cytometry
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Single-cell Analysis of Immunophenotype and Cytokine Production in Peripheral Whole Blood via Mass Cytometry

Published on: June 26, 2018

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Last Updated: Jul 3, 2026

Genome-wide Analysis of HDAC Inhibitor-mediated Modulation of microRNAs and mRNAs in B Cells Induced to Undergo Class-switch DNA Recombination and Plasma Cell Differentiation
11:06

Genome-wide Analysis of HDAC Inhibitor-mediated Modulation of microRNAs and mRNAs in B Cells Induced to Undergo Class-switch DNA Recombination and Plasma Cell Differentiation

Published on: September 20, 2017

Single-cell Analysis of Immunophenotype and Cytokine Production in Peripheral Whole Blood via Mass Cytometry
12:36

Single-cell Analysis of Immunophenotype and Cytokine Production in Peripheral Whole Blood via Mass Cytometry

Published on: June 26, 2018

  • Analysis of 93,112 cells across pretreatment and burosumab therapy phases.

    • Main Results:

    • Dynamic changes were observed in T cell and natural killer (NK) cell subtypes.
    • T helper 2 (Th2) and regulatory T (Treg) cells decreased, while T helper 17 (Th17) cells increased during treatment.
    • Upregulated genes in Treg cells and NK cells were linked to osteoclast differentiation pathways.
    • Specific ligand-receptor pairs (KLRB1-CLEC2D, SELL-SELPLG) mediated cell-cell communication.

    Conclusions:

    • This study provides the first comprehensive longitudinal transcriptomic analysis of immune cell dynamics during burosumab therapy in pediatric XLH.
    • Findings offer novel insights into the immunological mechanisms underlying burosumab treatment response in XLH.
    • The observed immune cell alterations may contribute to the therapeutic efficacy of burosumab in XLH.