Long-term outcome of progressive multifocal leukoencephalopathy with recombinant interleukin-2 treatment and an associated increase in the number of HPyV-2-specific T-cells: a case report.

IF 3.4 3区 医学 Q2 HEMATOLOGY Therapeutic Advances in Hematology Pub Date : 2023-10-09 eCollection Date: 2023-01-01 DOI:10.1177/20406207231201721
Fieke W Hoff, John Rolwes, Paula A Hardeman, Molly Perkins, Eugene O Major, Daniel Douek, Robert H Collins, Benjamin M Greenberg
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Abstract

Progressive multifocal leukoencephalopathy (PML) is a demyelinating disease caused by reactivation of the human polyomavirus 2 (HPyV-2). PML is associated with a high morbidity and mortality rate and there is currently no standard curative therapy. We report short-term immunologic response and long-term clinical outcomes in a patient diagnosed with follicular lymphoma (FL) who developed PML. Diagnosis of PML was established conclusively based on findings from a brain biopsy. The patient was treated with recombinant interleukin 2 (IL-2) and showed rapid clinical improvement. HPyV-2-specific T-cells were tracked longitudinally and correlation with clinical status, viral load, and radiographic imaging was documented. After the progression of the patient's FL, which required an allogeneic bone marrow transplant, the patient prophylactically received human leukocyte antigen-matched donor-derived HPyV-2 T-cells to prevent the recurrence of the PML as part of a clinical trial. Twelve years after the initial diagnosis of PML, he did not develop a relapse of his PML, supporting data that therapies that increase HPyV-2-specific T-cells, including IL-2, may be effective in the management of PML.

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重组白细胞介素-2治疗进行性多灶性白质脑病的长期结果及HPyV-2特异性T细胞数量的相关增加:一例报告。
进行性多灶性白质脑病(PML)是一种由人类多瘤病毒2型(HPyV-2)再激活引起的脱髓鞘疾病。PML与高发病率和死亡率有关,目前还没有标准的治疗方法。我们报告了一名诊断为滤泡性淋巴瘤(FL)并发展为PML的患者的短期免疫反应和长期临床结果。PML的诊断是根据脑活检的结果最终确定的。该患者接受了重组白细胞介素2(IL-2)治疗,并显示出快速的临床改善。对HPyV-2特异性T细胞进行纵向追踪,并记录其与临床状态、病毒载量和放射学成像的相关性。在需要异基因骨髓移植的患者FL进展后,作为临床试验的一部分,患者预防性地接受人类白细胞抗原匹配的供体来源的HPyV-2 T细胞,以防止PML复发。在最初诊断为PML 12年后,他没有出现PML复发,这支持了增加HPyV-2特异性T细胞(包括IL-2)的治疗可能对PML的管理有效的数据。
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来源期刊
CiteScore
4.30
自引率
0.00%
发文量
54
审稿时长
7 weeks
期刊介绍: Therapeutic Advances in Hematology delivers the highest quality peer-reviewed articles, reviews, and scholarly comment on pioneering efforts and innovative studies across all areas of hematology. The journal has a strong clinical and pharmacological focus and is aimed at clinicians and researchers in hematology, providing a forum in print and online for publishing the highest quality articles in this area.
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