Donor cell myelodysplastic syndrome after allogeneic stem cell transplantation responding to donor lymphocyte infusion: Case report and literature review

Abstract

Allogeneic hematopoietic stem cell transplantation (SCT) is a potentially curative treatment for patients with myelodysplastic syndrome (MDS). Relapses after transplantation however, are not uncommon and are usually due to re-emergence of a recipient derived, neoplastic, stem cell clone. We report a unique case of MDS recurring 5 months after non-myeloablative, sibling, allogeneic SCT. Interestingly, chimerism analysis at relapse showed hematopoiesis to be entirely of donor origin confirming donor cell MDS. Donor lymphocyte infusion (DLI) produced a hematological response lasting several months. Our review of the literature shows donor-derived MDS to be very rare, with only four such cases described previously. In this report, we describe the details of our case and discuss putative mechanisms underlying the genesis of donor cell MDS and the observed response to DLI. Am.

Therapy-related, donor-derived AML responding to a second allogeneic BMT

“…We chose this 'semi-autologous' graft because graft rejection is unlikely, but GVL reactions may occur. 8 This treatment option has the additional advantage that the 'autograft' from the unaffected donor is not contaminated with leukemic cells. In conclusion, we demonstrate that a second BMT from the same donor may be a curative treatment option for these patients, as our patient is in ongoing complete remission for 4 years.…”

Donor-cell MDS in a 12-year-old girl 3 years after allogeneic hematopoietic SCT for MDS, both with a t(3;3)(q21;q26) cytogenetic aberration

“…One patient had normal cytogenetics in the primary, as well as in the donor-derived MDS. 10 The other two patients had abnormal cytogenetics that differed between the primary and the secondary MDS. 8,9 Our case is unique in that the primary MDS as well as the donor cell MDS contained the relatively rare t(3;3)(q21;q26) cytogenetic abnormality, which is of particular interest regarding the pathogenesis of donor cell MDS/leukemia.…”

“…A literature search revealed only three such cases, two of which had a sibling donor as in our case (Table 1). [8][9][10] In all cases, the second malignancy was MDS. One patient had normal cytogenetics in the primary, as well as in the donor-derived MDS.…”

“…Successful reduced-intensity BMT from an alternative, unrelated donor after donor-derived MDS has been described, 9 as well as the response of donor cell MDS to donor lymphocyte infusion. 10 …”

“…8,9 Our case is unique in that the primary MDS as well as the donor cell MDS contained the relatively rare t(3;3)(q21;q26) cytogenetic abnormality, which is of particular interest regarding the pathogenesis of donor cell MDS/leukemia. Komrokji et al 10 proposed several hypotheses and/or contributing factors for the development of donor cell leukemia: (1) abnormal or defective stroma and/or microenvironment, (2) genetic susceptibility, (3) immune mediated, (4) treatment related, (5) infection and (6) transfection of oncogenic material from degenerating recipient malignant cells to donor cells. Further evidence for the role of the hematopoietic microenvironment in the development of MDS was recently provided by Raaijmakers et al 11 Microenvironmental abnormalities together with genetic susceptibility might have been the main contributing factors in our patient.…”