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Donor cell-derived transient abnormal myelopoiesis as a specific complication of umbilical cord blood transplantation

Bone Marrow Transplantation volume 53, pages 225–227 (2018)Cite this article

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Umbilical cord blood (UCB) has been increasingly used as an alternative stem cell source for patients without suitable HLA-matched related or unrelated donors. An estimated 730 000 UCB units have been donated and stored to date, and more than 35 000 UCB transplantations (UCBTs) have been performed worldwide.1 Transient abnormal myelopoiesis (TAM) is a unique disorder that presents from fetal life to the first few days after birth and occurs in ~10% of Down syndrome (DS) neonates.2, 3 TAM presents with clinical and morphological findings indistinguishable from acute megakaryoblastic leukemia (AMKL), but resolves spontaneously at a high rate within a few weeks or months of birth.4 By contrast, myeloid leukemia associated with DS (ML-DS) as non-transient AMKL develops about 1–5 years later in ~20% of these neonates.5 TAM develops in the presence of constitutive trisomy 21 and a single GATA1 mutation.6 Subsequent ML-DS evolves from a pre-existing TAM clone through the acquisition of additional gene mutations in cohesin or epigenetic regulators and in RAS or signal-transducing molecules.6, 7 TAM uncommonly occurs in phenotypically normal neonates with trisomy 21 mosaicism8, 9 or without trisomy 21.10, 11

In this report, we describe a patient who developed TAM derived from donor cells about 4 months after UCBT for refractory angioimmunoblastic T-cell lymphoma (AITL). To our knowledge, this is the first report of donor cell-derived TAM (DCD TAM) as a specific complication of UCBT, which cannot occur after allogeneic stem cell transplantation using bone marrow (BM) or peripheral blood (PB).

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Acknowledgements

We thank Ms Eiko Yamashita, Mr Kazuyuki Ueno and Ms Kyoko Tanaka from the Department of Clinical Laboratory, Kobe City Medical Center General Hospital for specimen management and technical assistance.

Author contributions

NH, TI, NY, YS and YO designed the study; HM and NH performed the immunophenotyping; JT, YO, KY, HT, KC, YS and SO performed sequencing data analyses; KU and YI performed pathological analyses; NH, JT, KY and YO integrated and interpreted the results; NH, SY, JT, YO and TI wrote the manuscript; and all authors revised and approved the manuscript.

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Authors and Affiliations

  1. Department of Cell Therapy, Institute of Biomedical Research and Innovation, Kobe, Japan

    N Hiramoto

  2. Department of Pathology and Tumor Biology, Graduate School of Medicine, Kyoto University, Kyoto, Japan

    J Takeda, K Yoshida & S Ogawa

  3. Department of Hematology, Kobe City Medical Center General Hospital, Kobe, Japan

    Y Ono, S Yoshioka, N Yamauchi, A Fujimoto & T Ishikawa

  4. Department of Clinical Laboratory, Kobe City Medical Center General Hospital, Kobe, Japan

    H Maruoka

  5. Laboratory of DNA Information Analysis, Human Genome Center, Institute of Medical Science, The University of Tokyo, Tokyo, Japan

    Y Shiraishi, H Tanaka, K Chiba & S Miyano

  6. Department of Clinical Pathology, Kobe City Medical Center General Hospital, Kobe, Japan

    Y Imai

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  1. N Hiramoto
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Corresponding author

Correspondence to K Yoshida.

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The authors declare no conflict of interest.

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Hiramoto, N., Takeda, J., Yoshida, K. et al. Donor cell-derived transient abnormal myelopoiesis as a specific complication of umbilical cord blood transplantation. Bone Marrow Transplant 53, 225–227 (2018). https://doi.org/10.1038/bmt.2017.226

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