Salvage HLA-haploidentical Peripheral Blood Stem Cell Transplantation Using Post-transplant Cyclophosphamide for Recurrent Hemophagocytic Lymphohistiocytosis-associated Graft Failure after Cord Blood Transplantations: A Case Report

Keijiro Sato; Wataru Komaba; Saika Oka; Shintaro Kazama; Ryuto Ishikawa; Takahiro Morikawa; Mari Kitahara; Hiroko Kazumoto; Hiroko Kaiume; Toshimitsu Ueki; Yuki Hiroshima; Masahiko Sumi; Hikaru Kobayashi

doi:10.2169/internalmedicine.5159-24

Keijiro Sato, Wataru Komaba, Saika Oka, Shintaro Kazama, Ryuto Ishikawa, Takahiro Morikawa, Mari Kitahara, Hiroko Kazumoto, Hiroko Kaiume, Toshimitsu Ueki, Yuki Hiroshima, Masahiko Sumi, Hikaru Kobayashi

Author information

Keijiro Sato
Department of Hematology, Nagano Red Cross Hospital, Japan
Wataru Komaba
Department of Hematology, Nagano Red Cross Hospital, Japan
Saika Oka
Department of Hematology, Nagano Red Cross Hospital, Japan
Shintaro Kazama
Department of Hematology, Nagano Red Cross Hospital, Japan
Ryuto Ishikawa
Department of Hematology, Nagano Red Cross Hospital, Japan
Takahiro Morikawa
Department of Hematology, Nagano Red Cross Hospital, Japan
Mari Kitahara
Department of Hematology, Nagano Red Cross Hospital, Japan
Hiroko Kazumoto
Department of Hematology, Nagano Red Cross Hospital, Japan
Hiroko Kaiume
Department of Hematology, Nagano Red Cross Hospital, Japan
Toshimitsu Ueki
Department of Hematology, Nagano Red Cross Hospital, Japan
Yuki Hiroshima
Department of Hematology, Nagano Red Cross Hospital, Japan
Masahiko Sumi
Department of Hematology, Nagano Red Cross Hospital, Japan
Hikaru Kobayashi
Department of Hematology, Nagano Red Cross Hospital, Japan

Keywords: Hemophagocytic lymphohistiocytosis, HLA-haploidentical transplantation, Cord blood transplantation, Post-transplant cyclophosphamide, Graft failure

JOURNAL OPEN ACCESS Advance online publication

Article ID: 5159-24

DOI https://doi.org/10.2169/internalmedicine.5159-24

Details

Abstract

We describe a case of immunological rejection occurring twice after cord blood transplantation (CBT) for mixed phenotype blast phase chronic myeloid leukemia that was successfully salvaged by haploidentical peripheral blood stem cell transplantation (haplo-PBSCT) with post-transplant cyclophosphamide (PT-Cy). Pre-engraftment immune reaction (PIR) and subsequent hemophagocytic lymphohistiocytosis (HLH), likely due to HLA mismatch in the graft-versus-host (GVH) direction, lead to poor graft function (PGF) and graft failure (GF). This case highlights the pathophysiology of PIR, HLH, PGF, and GF, collectively termed "post-transplant cytokine syndrome." PT-Cy haplo-PBSCT, with wide donor availability and reduced infection risk leading to HLH via rapid engraftment, may be a suitable salvage option for post-CBT cytokine syndrome-related GF.

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