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Peer-reviewedCC BY-NC-NDInternal Medicine

Late-onset Human Herpesvirus 6 Encephalitis with Secondary Hemophagocytic Lymphohistiocytosis after CAR T-cell Therapy

7 more authors

Abstract

Human herpesvirus 6 (HHV-6) encephalitis after chimeric antigen receptor (CAR) T-cell therapy is rare and it usually occurs early after infusion. We report a case of a 76-year-old man with diffuse large B-cell lymphoma who developed late-onset HHV-6 encephalitis with secondary hemophagocytic lymphohistiocytosis (HLH) 14 months after the infusion of lisocabtagene maraleucel. A cerebrospinal fluid (CSF) analysis revealed elevated HHV-6B DNA levels, and hemophagocytosis was identified in both the bone marrow and CSF. Despite ganciclovir and HLH-directed therapy, the patient died of a bacterial infection. HHV-6-associated disease should be considered even long after CAR T-cell therapy in patients with persistent fever, hyperinflammation, cytopenia, or neurological symptoms.

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BibTeX
@article{masuda2026lateonset,
  title = {{Late-onset Human Herpesvirus 6 Encephalitis with Secondary Hemophagocytic Lymphohistiocytosis after CAR T-cell Therapy}},
  author = {Kyoko Masuda and Mana Sota and Aika Kitamura and Tsutomu Yoshida and Tomohiko Tanigawa and Kaito Takikawa and Kiho Tozuka and Miki Sakamoto and Takahide Shindo and Takayuki Fujii and Kentaro Yamaguchi and Yuriko Nakamura and Ryo Inaba and Kei Ishizuchi and Hiromichi Matsushita and Rie Yamazaki and Yuya Koda and Masatoshi Sakurai and Keisuke Kataoka},
  journal = {Internal Medicine},
  year = {2026},
  month = aug,
  doi = {10.2169/internalmedicine.7887-26},
  url = {https://www.jstage.jst.go.jp/article/internalmedicine/advpub/0/advpub_7887-26/_article},
  note = {CC BY-NC-ND},
}
RIS
TY  - JOUR
TI  - Late-onset Human Herpesvirus 6 Encephalitis with Secondary Hemophagocytic Lymphohistiocytosis after CAR T-cell Therapy
AU  - Kyoko Masuda
AU  - Mana Sota
AU  - Aika Kitamura
AU  - Tsutomu Yoshida
AU  - Tomohiko Tanigawa
AU  - Kaito Takikawa
AU  - Kiho Tozuka
AU  - Miki Sakamoto
AU  - Takahide Shindo
AU  - Takayuki Fujii
AU  - Kentaro Yamaguchi
AU  - Yuriko Nakamura
AU  - Ryo Inaba
AU  - Kei Ishizuchi
AU  - Hiromichi Matsushita
AU  - Rie Yamazaki
AU  - Yuya Koda
AU  - Masatoshi Sakurai
AU  - Keisuke Kataoka
PY  - 2026
DA  - 2026/08/19
JO  - Internal Medicine
DO  - 10.2169/internalmedicine.7887-26
UR  - https://www.jstage.jst.go.jp/article/internalmedicine/advpub/0/advpub_7887-26/_article
AB  - Human herpesvirus 6 (HHV-6) encephalitis after chimeric antigen receptor (CAR) T-cell therapy is rare and it usually occurs early after infusion. We report a case of a 76-year-old man with diffuse large B-cell lymphoma who developed late-onset HHV-6 encephalitis with secondary hemophagocytic lymphohistiocytosis (HLH) 14 months after the infusion of lisocabtagene maraleucel. A cerebrospinal fluid (CSF) analysis revealed elevated HHV-6B DNA levels, and hemophagocytosis was identified in both the bone marrow and CSF. Despite ganciclovir and HLH-directed therapy, the patient died of a bacterial infection. HHV-6-associated disease should be considered even long after CAR T-cell therapy in patients with persistent fever, hyperinflammation, cytopenia, or neurological symptoms.
SN  - 1349-7235
SN  - 0918-2918
ER  -