Skip to main content

Thank you for visiting nature.com. You are using a browser version with limited support for CSS. To obtain the best experience, we recommend you use a more up to date browser (or turn off compatibility mode in Internet Explorer). In the meantime, to ensure continued support, we are displaying the site without styles and JavaScript.

  • Letter to the Editor
  • Published:

Therapy-related myelodysplastic syndrome of recipient origin in a juvenile myelomonocytic leukemia patient 17 years after allogeneic BMT

Bone Marrow Transplantation volume 46, pages 1023–1025 (2011)Cite this article

Subjects

Therapy-related myelodysplastic syndrome (t-MDS)/AML of recipient origin is rarely reported after allogeneic BMT. Here, we describe the numerous chromosomal aberrations arising in various recipient cells of a patient who developed t-MDS 17 years after allogeneic BMT. Further, we review the relevant literature on t-MDS after allogeneic BMT.

A 4-month-old boy was diagnosed with juvenile myelomonocytic leukemia on the basis of a typical blood-film examination and hypersensitivity to G-CSF and spontaneous colony formation. Cytogenetic analysis revealed that all the BM cells had a 46,XY karyotype. Around 7 months after diagnosis, the patient underwent an unrelated donor BMT from a male donor fully matched for HLA-A, B and DRB1. Conditioning consisted of 12-Gy-fractionated TBI and administration of high-dose etoposide (50 mg/kg × 1) and Ara-c (3 g/m2 × 8); GVHD prophylaxis included the administration of CsA and short-term MTX. Engraftment was uneventful, but the patient developed grade II gastrointestinal GVHD, which was successfully treated with steroids. The patient did not develop chronic GVHD, and his blood cell counts had been normal for 17 years. However, the patient developed pancytopenia at the age of 18 years. A BM aspirate, containing 1.4% leukemia cells, showed hypoplastic marrow with trilineage dysplasia. Chimerism by STR analysis of BM mononuclear cells was 94.9% recipient type. Karyotypic analysis of the BM cells showed a complex clonal chromosomal aberration, 46,XY,t(1;11)(p22;q22),add(1)(p13),del(11)(q23), and three non-clonal abnormal cells (Table 1). Rearrangement of the mixed-lineage leukemia gene was not observed. Peripheral blood cultured with PHA for 72 h showed 15 random non-clonal chromosomal aberrations and a normal male karyotype (Table 1). Furthermore, karyotypic analysis of BM fibroblasts and skin fibroblasts revealed random, non-clonal chromosomal aberrations of 100% recipient origin (Table 1). Diepoxybutane treatment did not cause chromosomal breakage in the peripheral lymphocytes; this finding suggested that he did not have chromosomal instability syndromes, such as Fanconi anemia. He was diagnosed with t-MDS, and a second BMT from another HLA-matched unrelated donor was performed; 45 months after BMT, the patient is well with 100% second donor-type chimerism.

Table 1 Structural cytogenetic aberrations in the BM, PB, BM fibroblast and skin fibroblast
Full size table

This is a preview of subscription content, access via your institution

Access options

Buy this article

  • Purchase on Springer Link
  • Instant access to full article PDF
Buy now

Prices may be subject to local taxes which are calculated during checkout

References

  1. Lowe T, Bhatia S, Somlo G . Second malignancies after allogeneic hematopoietic cell transplantation. Biol Blood Marrow Transplant 2007; 13: 1121–1134.

    Article  Google Scholar 

  2. Au WY, Lie AK, Ma SK, Leung YH, Siu LL, Kwong YL . Therapy-related myelodysplastic syndrome of recipient origin after allogeneic bone marrow transplantation for acute lymphoblastic leukemia. Br J Haematol 2001; 112: 424–426.

    Article  CAS  Google Scholar 

  3. Muroi K, Kawano C, Yokote T, Otsuki T, Kirito K, Komatsu N et al. Early myelodysplastic syndrome after allogeneic bone marrow transplantation for acute myeloid leukemia. Lukemia Lymphoma 2002; 43: 1493–1496.

    Article  Google Scholar 

  4. Yamamoto K, Yakushijin K, Kawamomori Y, Minagawa K, Katayama Y, Matsui T . Translocation (7;9)(q22;q34) in therapy-related myelodysplastic syndrome after allogeneic bone marrow transplantation for acute myeloblastic leukemia. Cancer Genet Cytogen 2007; 16: 61–66.

    Article  Google Scholar 

  5. Roos-Weil D, Nguyen S, Uzunov M, Bories D, Chapiro E, Nguyen-Khac F et al. Therapy-related myelodysplastic syndrome after allogeneic BMT: successful treatment by donor lymphocyte infusions. Bone Marrow Transplant 2010; 45: 1471–1473.

    Article  CAS  Google Scholar 

  6. Pedersen-Bjergaard J, Anderson AK, Christiansen DH . Therapy-related acute myeloid leukemia and myelodysplasia after high-dose chemotherapy and autologus stem cell transplantation. Blood 2000; 95: 3273–3279.

    CAS  PubMed  Google Scholar 

  7. Nakanishi M, Tanaka K, Shintani T, Takahashi T, Kamada N . Chromosomal instability in acute myeloid leukemia and myelodysplastic syndrome patients among atomic bomb survivors. J Radiat Res 1999; 40: 159–167.

    Article  CAS  Google Scholar 

  8. Lin YW, Hamahata K, Watanabe K, Adachi S, Akiyama Y, Kubota M et al. Repetitious appearance and disappearance of different kinds of clonal cytogenetic abnormalities after allogeneic bone marrow transplantation. Int J Hematol 2001; 74: 86–89.

    Article  CAS  Google Scholar 

  9. Yoshihara T, Hibi S, Yamane Y, Morimoto A, Hashida T, Iwami H et al. Numerous nonclonal chromosomal aberrations arising in residual recipient hematopoietic cells following allogeneic bone marrow transplantation. Bone Marrow Transplant 2005; 35: 587–589.

    Article  CAS  Google Scholar 

  10. Litzow MR, Tarima S, Pérez WS, Bolwell BJ, Cairo MS, Camitta BM et al. Allogeneic transplantation for therapy-related myelodysplastic syndrome and acute myeloid leukemia. Blood 2010; 115: 1850–1857.

    Article  CAS  Google Scholar 

Download references

Author information

Authors and Affiliations

  1. Department of Cell Transplantation, Tokai University Hospital, Isehara, Kanagawa, Japan

    M Yabe, S Kato & H Yabe

  2. Department of Pediatrics, Tokai University Hospital, Isehara, Kanagawa, Japan

    T Morimoto, T Shimizu, T Koike & H Takakura

  3. Central Laboratory, Tokai University Hospital, Isehara, Kanagawa, Japan

    S Arakawa

Authors
  1. M Yabe
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. T Morimoto
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. T Shimizu
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. T Koike
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. H Takakura
    View author publications

    You can also search for this author in PubMed Google Scholar

  6. S Arakawa
    View author publications

    You can also search for this author in PubMed Google Scholar

  7. S Kato
    View author publications

    You can also search for this author in PubMed Google Scholar

  8. H Yabe
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to M Yabe.

Ethics declarations

Competing interests

The authors declare no conflict of interest.

Rights and permissions

Reprints and permissions

About this article

Cite this article

Yabe, M., Morimoto, T., Shimizu, T. et al. Therapy-related myelodysplastic syndrome of recipient origin in a juvenile myelomonocytic leukemia patient 17 years after allogeneic BMT. Bone Marrow Transplant 46, 1023–1025 (2011). https://doi.org/10.1038/bmt.2010.224

Download citation

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1038/bmt.2010.224

This article is cited by

Search

Advanced search

Quick links