Sandra Nay, Nora Möhn, Lea Grote-Levi, Agnes Bonifacius, Mieke L Saßmann, Kevin Karacondi, Sabine Tischer-Zimmermann, Henning Pöter, Nima Mahmoudi, Mike P Wattjes, Britta Maecker-Kolhoff, Günter Höglinger, Britta Eiz-Vesper, Thomas Skripuletz
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We describe a combined therapy approach using directly isolated allogeneic Human Polyomavirus 1 (also known as BKV) and Epstein-Barr virus (EBV) specific cytotoxic T-cells for the treatment of PML in conjunction with identified EBV in the cerebrospinal fluid (CSF) of a male patient infected with human immunodeficiency virus (HIV). A 53-year-old HIV-positive male, recently diagnosed with PML, presented with rapidly worsening symptoms, including ataxia, tetraparesis, dysarthria, and dysphagia, leading to respiratory failure. The patient developed PML even after commencing highly active antiretroviral therapy (HAART) 3 months prior. Brain magnetic resonance imaging (MRI) revealed multifocal demyelination lesions involving the posterior fossa and right thalamus suggestive of PML. In addition to the detection of human polyomavirus 2 (also known as JCV), analysis of CSF showed positive results for EBV deoxyribonucleic acid (DNA). His neurological condition markedly deteriorated over the following 2 months. Based on MRI, there was no evidence of Immune Reconstitution Inflammatory Syndrome contributing to this decline. The patient did not have endogenous virus-specific T-cells. We initiated an allogeneic, partially human leukocyte antigen-matched transfer of EBV and utilizing the cross-reactivity between BKV and JCV-BKV specific T-cells. This intervention led to notable neurological improvement and partial resolution of the MRI lesions within 6 weeks. Our case of a patient with acquired immune deficiency syndrome demonstrates that PML and concurrent EBV co-infection can still occur despite undergoing HAART treatment. This innovative experimental therapy, involving a combination of virus-specific T-cells, was demonstrated to be an effective treatment option in this patient.</p>","PeriodicalId":22980,"journal":{"name":"Therapeutic Advances in Neurological Disorders","volume":"17 ","pages":"17562864241253917"},"PeriodicalIF":4.7000,"publicationDate":"2024-05-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11135084/pdf/","citationCount":"0","resultStr":"{\"title\":\"Combined treatment with allogeneic Epstein-Barr- and human polyomavirus 1 specific T-cells in progressive multifocal leukoencephalopathy and EBV infection: a case report.\",\"authors\":\"Sandra Nay, Nora Möhn, Lea Grote-Levi, Agnes Bonifacius, Mieke L Saßmann, Kevin Karacondi, Sabine Tischer-Zimmermann, Henning Pöter, Nima Mahmoudi, Mike P Wattjes, Britta Maecker-Kolhoff, Günter Höglinger, Britta Eiz-Vesper, Thomas Skripuletz\",\"doi\":\"10.1177/17562864241253917\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Opportunistic viral infections in individuals with severe immunodeficiency can lead to fatal conditions such as progressive multifocal leukoencephalopathy (PML), for which treatment options are limited. 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引用次数: 0
摘要
严重免疫缺陷患者的机会性病毒感染可导致进行性多灶性白质脑病(PML)等致命病症,但治疗方案却很有限。这些感染会带来巨大风险,尤其是当合并感染其他病毒时。我们描述了一种联合治疗方法,使用直接分离的异体人类多瘤病毒 1(又称 BKV)和 Epstein-Barr 病毒(EBV)特异性细胞毒性 T 细胞治疗 PML,同时在一名感染人类免疫缺陷病毒(HIV)的男性患者的脑脊液(CSF)中发现了 EBV。一名 53 岁的 HIV 阳性男性患者最近被诊断为 PML,其症状迅速恶化,包括共济失调、四肢瘫痪、构音障碍和吞咽困难,最终导致呼吸衰竭。患者在 3 个月前开始接受高效抗逆转录病毒疗法(HAART)后仍出现了 PML。脑磁共振成像(MRI)显示,后窝和右侧丘脑出现多灶性脱髓鞘病变,提示为 PML。除了检测到人类多瘤病毒2(又称JCV)外,CSF分析还显示EB病毒脱氧核糖核酸(DNA)阳性。在随后的两个月里,他的神经状况明显恶化。根据核磁共振成像,没有证据表明免疫重建炎症综合征是导致病情恶化的原因。患者没有内源性病毒特异性 T 细胞。我们利用 BKV 和 JCV-BKV 特异性 T 细胞之间的交叉反应,启动了异体、部分人类白细胞抗原匹配的 EBV 转移。这一干预措施在 6 周内使患者的神经功能明显改善,核磁共振成像病灶部分消退。我们这例获得性免疫缺陷综合征患者的病例表明,尽管接受了 HAART 治疗,PML 和 EBV 合并感染仍有可能发生。这种创新的实验性疗法涉及病毒特异性 T 细胞的组合,在该患者身上被证明是一种有效的治疗方案。
Combined treatment with allogeneic Epstein-Barr- and human polyomavirus 1 specific T-cells in progressive multifocal leukoencephalopathy and EBV infection: a case report.
Opportunistic viral infections in individuals with severe immunodeficiency can lead to fatal conditions such as progressive multifocal leukoencephalopathy (PML), for which treatment options are limited. These infections pose significant risks, especially when co-infections with other viruses occur. We describe a combined therapy approach using directly isolated allogeneic Human Polyomavirus 1 (also known as BKV) and Epstein-Barr virus (EBV) specific cytotoxic T-cells for the treatment of PML in conjunction with identified EBV in the cerebrospinal fluid (CSF) of a male patient infected with human immunodeficiency virus (HIV). A 53-year-old HIV-positive male, recently diagnosed with PML, presented with rapidly worsening symptoms, including ataxia, tetraparesis, dysarthria, and dysphagia, leading to respiratory failure. The patient developed PML even after commencing highly active antiretroviral therapy (HAART) 3 months prior. Brain magnetic resonance imaging (MRI) revealed multifocal demyelination lesions involving the posterior fossa and right thalamus suggestive of PML. In addition to the detection of human polyomavirus 2 (also known as JCV), analysis of CSF showed positive results for EBV deoxyribonucleic acid (DNA). His neurological condition markedly deteriorated over the following 2 months. Based on MRI, there was no evidence of Immune Reconstitution Inflammatory Syndrome contributing to this decline. The patient did not have endogenous virus-specific T-cells. We initiated an allogeneic, partially human leukocyte antigen-matched transfer of EBV and utilizing the cross-reactivity between BKV and JCV-BKV specific T-cells. This intervention led to notable neurological improvement and partial resolution of the MRI lesions within 6 weeks. Our case of a patient with acquired immune deficiency syndrome demonstrates that PML and concurrent EBV co-infection can still occur despite undergoing HAART treatment. This innovative experimental therapy, involving a combination of virus-specific T-cells, was demonstrated to be an effective treatment option in this patient.
期刊介绍:
Therapeutic Advances in Neurological Disorders is a peer-reviewed, open access journal delivering the highest quality articles, reviews, and scholarly comment on pioneering efforts and innovative studies across all areas of neurology. The journal has a strong clinical and pharmacological focus and is aimed at clinicians and researchers in neurology, providing a forum in print and online for publishing the highest quality articles in this area.