Recurrence of β-thalassemia major more than 20 years after HLA-identical sibling BMT treated successfully with donor lymphocyte infusion

Inamoto, Yoshihiro; Yannucci, J; Storb, Rainer; Flowers, Mary E.D.

doi:10.1038/bmt.2011.37

Cited by 3 publications

(3 citation statements)

References 10 publications

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“…Although patients developing MC after 6 months post-transplantation were less likely to undergo graft rejection, 7 late recurrences of thalassemia due to decreased donor chimerism have been reported. 36,37 We questioned whether there were some treatments that were effective and had few side effect for patients developing late-onset MC or those developing early-onset MC, but DLIs were not available. It has been reported that an ultra-low dose (1 × 10 5 U/ m 2 -2 × 10 5 U/m 2 ) of IL-2 increases circulating Tregs without expanding conventional effector CD4 + or CD8 + T cells and may be used to induce immune tolerance between donor and host cells.…”

Section: Discussionmentioning

confidence: 99%

“…Although patients developing MC after 6 months post‐transplantation were less likely to undergo graft rejection, 7 late recurrences of thalassemia due to decreased donor chimerism have been reported 36,37 . We questioned whether there were some treatments that were effective and had few side effect for patients developing late‐onset MC or those developing early‐onset MC, but DLIs were not available.…”

Section: Discussionmentioning

confidence: 99%

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Prediction, management, and prognosis of mixed chimerism after hematopoietic stem cell transplantation in transfusion‐dependent pediatric thalassemia patients

Chen

Zhan

et al. 2020

Pediatric Transplantation

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Background: Early-onset mixed chimerism (MC) with a high proportion of residual host cells is considered a signal of graft rejection in patients undergoing allogenic hematopoietic stem cell transplantation for transfusion-dependent thalassemia. In order to prevent graft rejection and minimize the risk of treatment-related graft-versus-host disease (GVHD), we established a hierarchical management system based on chimerism analysis. Method: This retrospective study provides a comprehensive review of the characteristics, interventions, and outcomes of the 38 patients who developed MC after transplantation among the 144 pediatric thalassemia patients between July 2007 and January 2019 at our center. Results: A sibling donor, a blood type-matched donor, conditioning regimens without fludarabine, and transplants containing <10 × 10 8 total nucleated cells/kg were identified to be associated with the development of MC. Among the 38 patients developing MC, only four patients rejected the grafts. The response rate to donor lymphocyte infusion (DLI, only for patients receiving sibling donor transplantation) and cytokine immunomodulation without DLI was 70.6% and 42.9%, respectively. Patients that developed GVHD after DLI or cytokine therapy had a more significant increase in donor cell chimerism (16%, range 0%-35%) than those without (8.5%, range −21% to 40%, P = .049). However, even when treatment-related GVHD was included, patients with MC had a lower cumulative incidence of total acute GVHD than patients with complete donor chimerism (29.2% vs 48.0%, P = .030). Conclusions: Interventions based on chimerism analysis were effective in preventing graft rejection and did not increase treatment-related GVHD in thalassemia patients with MC.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Prediction, management, and prognosis of mixed chimerism after hematopoietic stem cell transplantation in transfusion‐dependent pediatric thalassemia patients

Chen

Zhan

et al. 2020

Pediatric Transplantation

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“…One patient was reported with late unstable mixed chimerism 22 years after matched sibling allo‐HSCT for thalassemia, despite early full donor chimerism. This patient recovered full donor chimerism after DLI, though suffered both acute and chronic GVHD .…”

mentioning

confidence: 81%

Late complications of mixed chimerism following allogeneic bone marrow transplantation for thalassemia major

Spitzer

Giardina

O’Reilly

et al. 2015

Pediatric Blood & Cancer

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Hematopoietic Stem Cell Transplantation in Patients with Hemoglobinopathies

Yeşilipek

2020

Hemoglobin

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Recurrence of β-thalassemia major more than 20 years after HLA-identical sibling BMT treated successfully with donor lymphocyte infusion

Cited by 3 publications

References 10 publications

Prediction, management, and prognosis of mixed chimerism after hematopoietic stem cell transplantation in transfusion‐dependent pediatric thalassemia patients

Prediction, management, and prognosis of mixed chimerism after hematopoietic stem cell transplantation in transfusion‐dependent pediatric thalassemia patients

Late complications of mixed chimerism following allogeneic bone marrow transplantation for thalassemia major

Hematopoietic Stem Cell Transplantation in Patients with Hemoglobinopathies

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