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Purpose <p>To describe the effectiveness and safety of fostamatinib off-label use in a pediatric patient with autoimmune cytopenia and autoimmune hepatitis following allogeneic hematopoietic stem cell transplantation (HSCT).</p> Methods <p>We report the detailed changes in clinical, histopathological, and cytokine profiling in a pediatric HSCT recipient during treatment with fostamatinib.</p> Results <p>Fostamatinib administration was well-tolerated with no adverse events observed, leading to sustained hematological recovery and a significant improvement in liver enzyme levels. Histological features confirmed the complete remission of autoimmune hepatitis. Cytokine profiling demonstrated substantial normalization of pro-inflammatory markers, downregulated at onset.</p> Conclusion <p>Fostamatinib demonstrated a dual benefit in resolving autoimmune cytopenia and immune-mediated liver inflammation, supporting its potential as a therapeutic option for immune dysregulation following allogeneic HSCT.</p>

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Fostamatinib Dual Immunomodulation in Post-Haploidentical HSCT Autoimmune Cytopenia and Autoimmune Hepatitis: A Case Report and Review of Literature

  • 期刊论文

  • 出版模式：

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  • 发表日期：

    2026年3月21日

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  • Antimo Tessitore,
  • Debora Curci,
  • Aurelio Sonzogni,
  • Martina Franzin,
  • Giulia Schillani,
  • Anna Flamigni,
  • Gilda Paternuosto,
  • Natalia Maximova

摘要

Purpose

To describe the effectiveness and safety of fostamatinib off-label use in a pediatric patient with autoimmune cytopenia and autoimmune hepatitis following allogeneic hematopoietic stem cell transplantation (HSCT).

Methods

We report the detailed changes in clinical, histopathological, and cytokine profiling in a pediatric HSCT recipient during treatment with fostamatinib.

Results

Fostamatinib administration was well-tolerated with no adverse events observed, leading to sustained hematological recovery and a significant improvement in liver enzyme levels. Histological features confirmed the complete remission of autoimmune hepatitis. Cytokine profiling demonstrated substantial normalization of pro-inflammatory markers, downregulated at onset.

Conclusion

Fostamatinib demonstrated a dual benefit in resolving autoimmune cytopenia and immune-mediated liver inflammation, supporting its potential as a therapeutic option for immune dysregulation following allogeneic HSCT.