异基因造血干细胞移植后糖皮质激素难治性多种自身免疫性疾病:病例报告及文献复习。

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

机构信息

Department of Hematology, The First Hospital of Putian City, Putian, China.

The School of Clinical Medicine, Fujian Medical University, Fuzhou, China.

出版信息

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.

摘要

我们在此报告一例 50 岁男性患者,他最初被诊断为伴多系病态造血的骨髓增生异常综合征-2 级(MDS-EB-2),并于 2019 年接受异基因造血干细胞移植(allo-HSCT),达到完全缓解。然而,他在 2021 年被诊断出患有几种自身免疫性疾病,包括自身免疫性肝炎(AIH)、桥本甲状腺炎(HT)和自身免疫性溶血性贫血(AIHA)。这被称为多种自身免疫综合征(MAS),这是 allo-HSCT 后罕见的情况,正如文献中所指出的那样。尽管接受了糖皮质激素、环孢素 A 和其他药物治疗,但患者并未完全康复。为了解决糖皮质激素难治性 MAS,给予患者为期四周、每周剂量为 100mg 的利妥昔单抗(RTX)治疗,患者病情显著改善。因此,本病例报告强调了在移植后自身免疫性疾病患者中,对于糖皮质激素难治或依赖的患者,采用替代治疗的重要性,并突出了 RTX 作为二线治疗的有效性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0289/11066190/0dc506d0581c/fimmu-15-1366101-g001.jpg

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