Multiple autoimmune disorders refractory to glucocorticoids after allogeneic hematopoietic stem cell transplantation: a case report and review of the literature

Front Immunol. 2024 Apr 19:15:1366101. doi: 10.3389/fimmu.2024.1366101. eCollection 2024.

Abstract

We report here the case of a 50-year-old man who was first diagnosed with myelodysplastic syndrome with excess blasts-2 (MDS-EB-2) and underwent allogeneic hematopoietic stem cell transplantation (allo-HSCT) in 2019, resulting in complete remission. However, he was diagnosed in 2021 with several autoimmune disorders, including autoimmune hepatitis (AIH), Hashimoto's thyroiditis (HT), and autoimmune hemolytic anemia (AIHA). This is referred as multiple autoimmune syndrome (MAS), which is a rare occurrence after allo-HSCT, as previously noted in the literature. Despite being treated with glucocorticoids, cyclosporine A, and other medications, the patient did not fully recover. To address the glucocorticoid-refractory MAS, a four-week course of rituximab (RTX) at a weekly dose of 100mg was administered, which significantly improved the patient's condition. Thus, this case report underscores the importance of implementing alternative treatments in patients with post-transplant autoimmune diseases, who are glucocorticoid-refractory or glucocorticoid-dependent, and highlights the effectiveness of RTX as second-line therapy.

Keywords: allogeneic hematopoietic stem cell transplantation; autoimmune hemolytic anemia; autoimmune hepatitis; multiple autoimmune syndrome; rituximab.

Publication types

  • Case Reports
  • Review
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Anemia, Hemolytic, Autoimmune / drug therapy
  • Anemia, Hemolytic, Autoimmune / etiology
  • Anemia, Hemolytic, Autoimmune / therapy
  • Autoimmune Diseases* / etiology
  • Autoimmune Diseases* / therapy
  • Drug Resistance
  • Glucocorticoids* / therapeutic use
  • Hematopoietic Stem Cell Transplantation* / adverse effects
  • Humans
  • Male
  • Middle Aged
  • Rituximab / therapeutic use
  • Transplantation, Homologous*

Substances

  • Glucocorticoids
  • Rituximab

Grants and funding

The author(s) declare financial support was received for the research, authorship, and/or publication of this article. This work was supported by grants from the project of Putian science and technology department (No. 2023SY005). The funding body had no role in the design of the study and collection, analysis, and interpretation of data and in writing the manuscript.