ADULT T-CELL LEUKEMIA/LYMPHOMA RELAPSED AFTER AUTOLOGOUS TRANSPLANT AND TREATED WITH HAPLOIDENTICAL STEM CELL TRANSPLANT

Introduction: Adult T-cell Leukemia/Lymphoma (ATL) is a highly aggressive T-cell malignancy that occurs in a proportion of individuals who are long-term carriers of Human T-cell Lymphotropic Virus type 1 (HTLV-1). Allogeneic hematopoietic stem cell transplantation has been suggested as a potentially...

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Main Authors: TFN Araújo, LPDS Rocha, CF Oliveira, BA Souza, CFM Ferreira, LS Abreu, RAF Machado, ML Puls, PHA Moraes, EL Rosa, CD Liz, RS Szor, VC Molla, C Arrais
Format: Article
Language:English
Published: Elsevier 2024-10-01
Series:Hematology, Transfusion and Cell Therapy
Online Access:http://www.sciencedirect.com/science/article/pii/S253113792402073X
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author TFN Araújo
LPDS Rocha
CF Oliveira
BA Souza
CFM Ferreira
LS Abreu
RAF Machado
ML Puls
PHA Moraes
EL Rosa
CD Liz
RS Szor
VC Molla
C Arrais
author_facet TFN Araújo
LPDS Rocha
CF Oliveira
BA Souza
CFM Ferreira
LS Abreu
RAF Machado
ML Puls
PHA Moraes
EL Rosa
CD Liz
RS Szor
VC Molla
C Arrais
author_sort TFN Araújo
collection DOAJ
description Introduction: Adult T-cell Leukemia/Lymphoma (ATL) is a highly aggressive T-cell malignancy that occurs in a proportion of individuals who are long-term carriers of Human T-cell Lymphotropic Virus type 1 (HTLV-1). Allogeneic hematopoietic stem cell transplantation has been suggested as a potentially curative strategy to eradicate HTLV-1 positive cells. Objective: To describe the case of a patient with ATLL initially treated as peripheral T lymphoma and presenting refractoriness to several lines of treatment who was successfully treated with an allogeneic haploidentical stem cell transplantation. Case description: A 60-year-old woman was diagnosed with peripheral T-cell non-Hodgkin lymphoma after a biopsy of skin lesions in December 2021. She did not present with lymphadenopathy. She was treated at another center with 6-cycles of Brentuximab Vedotin (BV), cyclophosphamide, doxorubicin and prednisone (BV-CHP) followed by autologous hematopoietic stem cell transplantation as consolidative therapy. Six months after transplant, she progressively presented worsening of her skin lesions. A new skin biopsy confirmed relapse, and she was referred to our center. A new skin biopsy revealed a diagnosis of T-cell lymphoma associated with a positive serology for the Human T-Lymphotropic virus 1 (HTLV-1). The combination of the biopsy findings and the association with HTLV-1 positivity supported the diagnosis of ATL. The patient achieved a complete response after 2-cycles of the association of BV, gemcitabine and oxaliplatin (Bv-Gemox), followed by a haploidentical allogeneic bone marrow transplant in July 2023. Her HCT-CI score was 1. The donor was her son, with a major ABO mismatch. Both were Cytomegalovirus (CMV) seropositive, and there were no HLA donor-specific antibodies. The reduced-intensity conditioning regimen consisted of fludarabine, cyclophosphamide, and Total Body Irradiation (TBI) 4 Gray, and the Graft-Versus-Host Disease (GVHD) prophylaxis was Post-transplant Cyclophosphamide (PtCy), sirolimus, and mycophenolate mofetil. Peripheral blood was the graft source with a 3.49×10̂6 CD34/Kg cell dose. Neutrophilic and platelets engraftment occurred at D+24 and D+41, respectively. She developed acute skin GVHD, which was successfully treated with topical steroids. She had CMV reactivation at low viral loads and did not require treatment. Sirolimus was withdrawn on D+70. Three months after transplant she presented disease relapse in an intraparotid lymph node, and BV was indicated. After 6 cycles, the patient achieved a complete response and BV was stopped. The patient developed chronic GVHD of the skin and mouth well controlled with topic steroids. In her last follow-up visit (13-months post-transplant), the patient remains in complete response and no systemic immunosuppression. Conclusion: This case highlights the potential of haploidentical HSCT as a viable and effective treatment for patients with refractory ATL. Successful management of associated complications and the use of post-transplant BV to achieve sustained remission highlights the importance of multidisciplinary care and targeted therapeutic strategies to improve outcomes of allogeneic HSCT as a curative approach for patients with this T-cell lymphomas.
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spelling doaj-art-12fd82e077ef401c801801b53da685a82025-08-20T02:17:41ZengElsevierHematology, Transfusion and Cell Therapy2531-13792024-10-0146S1021S102210.1016/j.htct.2024.09.1740ADULT T-CELL LEUKEMIA/LYMPHOMA RELAPSED AFTER AUTOLOGOUS TRANSPLANT AND TREATED WITH HAPLOIDENTICAL STEM CELL TRANSPLANTTFN Araújo0LPDS Rocha1CF Oliveira2BA Souza3CFM Ferreira4LS Abreu5RAF Machado6ML Puls7PHA Moraes8EL Rosa9CD Liz10RS Szor11VC Molla12C Arrais13Hospital Nove de Julho, Rede DASA, São Paulo, SP, BrazilHospital Nove de Julho, Rede DASA, São Paulo, SP, BrazilHospital Nove de Julho, Rede DASA, São Paulo, SP, BrazilHospital Nove de Julho, Rede DASA, São Paulo, SP, BrazilHospital Nove de Julho, Rede DASA, São Paulo, SP, BrazilHospital Nove de Julho, Rede DASA, São Paulo, SP, BrazilHospital Nove de Julho, Rede DASA, São Paulo, SP, BrazilHospital Nove de Julho, Rede DASA, São Paulo, SP, BrazilHospital Nove de Julho, Rede DASA, São Paulo, SP, Brazil; Universidade Federal de São Paulo (UNIFESP), São Paulo, SP, BrazilHospital Nove de Julho, Rede DASA, São Paulo, SP, Brazil; Universidade Federal de São Paulo (UNIFESP), São Paulo, SP, BrazilHospital Nove de Julho, Rede DASA, São Paulo, SP, BrazilHospital Nove de Julho, Rede DASA, São Paulo, SP, BrazilHospital Nove de Julho, Rede DASA, São Paulo, SP, Brazil; Universidade Federal de São Paulo (UNIFESP), São Paulo, SP, BrazilHospital Nove de Julho, Rede DASA, São Paulo, SP, Brazil; Universidade Federal de São Paulo (UNIFESP), São Paulo, SP, BrazilIntroduction: Adult T-cell Leukemia/Lymphoma (ATL) is a highly aggressive T-cell malignancy that occurs in a proportion of individuals who are long-term carriers of Human T-cell Lymphotropic Virus type 1 (HTLV-1). Allogeneic hematopoietic stem cell transplantation has been suggested as a potentially curative strategy to eradicate HTLV-1 positive cells. Objective: To describe the case of a patient with ATLL initially treated as peripheral T lymphoma and presenting refractoriness to several lines of treatment who was successfully treated with an allogeneic haploidentical stem cell transplantation. Case description: A 60-year-old woman was diagnosed with peripheral T-cell non-Hodgkin lymphoma after a biopsy of skin lesions in December 2021. She did not present with lymphadenopathy. She was treated at another center with 6-cycles of Brentuximab Vedotin (BV), cyclophosphamide, doxorubicin and prednisone (BV-CHP) followed by autologous hematopoietic stem cell transplantation as consolidative therapy. Six months after transplant, she progressively presented worsening of her skin lesions. A new skin biopsy confirmed relapse, and she was referred to our center. A new skin biopsy revealed a diagnosis of T-cell lymphoma associated with a positive serology for the Human T-Lymphotropic virus 1 (HTLV-1). The combination of the biopsy findings and the association with HTLV-1 positivity supported the diagnosis of ATL. The patient achieved a complete response after 2-cycles of the association of BV, gemcitabine and oxaliplatin (Bv-Gemox), followed by a haploidentical allogeneic bone marrow transplant in July 2023. Her HCT-CI score was 1. The donor was her son, with a major ABO mismatch. Both were Cytomegalovirus (CMV) seropositive, and there were no HLA donor-specific antibodies. The reduced-intensity conditioning regimen consisted of fludarabine, cyclophosphamide, and Total Body Irradiation (TBI) 4 Gray, and the Graft-Versus-Host Disease (GVHD) prophylaxis was Post-transplant Cyclophosphamide (PtCy), sirolimus, and mycophenolate mofetil. Peripheral blood was the graft source with a 3.49×10̂6 CD34/Kg cell dose. Neutrophilic and platelets engraftment occurred at D+24 and D+41, respectively. She developed acute skin GVHD, which was successfully treated with topical steroids. She had CMV reactivation at low viral loads and did not require treatment. Sirolimus was withdrawn on D+70. Three months after transplant she presented disease relapse in an intraparotid lymph node, and BV was indicated. After 6 cycles, the patient achieved a complete response and BV was stopped. The patient developed chronic GVHD of the skin and mouth well controlled with topic steroids. In her last follow-up visit (13-months post-transplant), the patient remains in complete response and no systemic immunosuppression. Conclusion: This case highlights the potential of haploidentical HSCT as a viable and effective treatment for patients with refractory ATL. Successful management of associated complications and the use of post-transplant BV to achieve sustained remission highlights the importance of multidisciplinary care and targeted therapeutic strategies to improve outcomes of allogeneic HSCT as a curative approach for patients with this T-cell lymphomas.http://www.sciencedirect.com/science/article/pii/S253113792402073X
spellingShingle TFN Araújo
LPDS Rocha
CF Oliveira
BA Souza
CFM Ferreira
LS Abreu
RAF Machado
ML Puls
PHA Moraes
EL Rosa
CD Liz
RS Szor
VC Molla
C Arrais
ADULT T-CELL LEUKEMIA/LYMPHOMA RELAPSED AFTER AUTOLOGOUS TRANSPLANT AND TREATED WITH HAPLOIDENTICAL STEM CELL TRANSPLANT
Hematology, Transfusion and Cell Therapy
title ADULT T-CELL LEUKEMIA/LYMPHOMA RELAPSED AFTER AUTOLOGOUS TRANSPLANT AND TREATED WITH HAPLOIDENTICAL STEM CELL TRANSPLANT
title_full ADULT T-CELL LEUKEMIA/LYMPHOMA RELAPSED AFTER AUTOLOGOUS TRANSPLANT AND TREATED WITH HAPLOIDENTICAL STEM CELL TRANSPLANT
title_fullStr ADULT T-CELL LEUKEMIA/LYMPHOMA RELAPSED AFTER AUTOLOGOUS TRANSPLANT AND TREATED WITH HAPLOIDENTICAL STEM CELL TRANSPLANT
title_full_unstemmed ADULT T-CELL LEUKEMIA/LYMPHOMA RELAPSED AFTER AUTOLOGOUS TRANSPLANT AND TREATED WITH HAPLOIDENTICAL STEM CELL TRANSPLANT
title_short ADULT T-CELL LEUKEMIA/LYMPHOMA RELAPSED AFTER AUTOLOGOUS TRANSPLANT AND TREATED WITH HAPLOIDENTICAL STEM CELL TRANSPLANT
title_sort adult t cell leukemia lymphoma relapsed after autologous transplant and treated with haploidentical stem cell transplant
url http://www.sciencedirect.com/science/article/pii/S253113792402073X
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