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1. Outcomes of allogeneic haematopoietic cell transplantation for myelofibrosis in children and adolescents: the retrospective study of the EBMT Paediatric Diseases WP

Author

Wachowiak, Jacek, Galimard, Jacques-Emmanuel, Dalissier, Arnaud, Rihani, Rawad, AlSaedi, Hawazen, Wynn, Robert F., Dalle, Jean-Hugues, Peffault de Latour, Régis, Sedlacek, Petr, Balduzzi, Adriana, Schroeder, Thomas, Bodova, Ivana, Gonzalez Vicent, Marta, Gruhn, Bernd, Hamladji, Rose-Marie, Krivan, Gergely, Patrick, Katharine, Sobkowiak-Sobierajska, Agnieszka, Stepensky, Polina, Unal, Ali, Amrolia, Persis, Perez Martinez, Antonio, Rialland, Fanny, Aljurf, Mahmoud, Isgro, Antonella, Toren, Amos, Bierings, Marc, Corbacioglu, Selim, and Kałwak, Krzysztof

Published
2024
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2. Cytogenetic abnormalities predict survival after allogeneic hematopoietic stem cell transplantation for pediatric acute myeloid leukemia: a PDWP/EBMT study

Author

Sharma, Akshay, Galimard, Jacques-Emmanuel, Pryce, Angharad, Bhoopalan, Senthil Velan, Dalissier, Arnaud, Dalle, Jean-Hugues, Locatelli, Franco, Jubert, Charlotte, Mirci-Danicar, Oana, Kitra-Roussou, Vassiliki, Bertrand, Yves, Fagioli, Franca, Rialland, Fanny, Biffi, Alessandra, Wynn, Robert F., Michel, Gérard, Tambaro, Francesco Paolo, Al-Ahmari, Ali, Tbakhi, Abdelghani, Furness, Caroline L., Diaz, Miguel Angel, Sedlacek, Petr, Bodova, Ivana, Faraci, Maura, Rao, Kanchan, Kleinschmidt, Katharina, Petit, Arnaud, Gibson, Brenda, Bhatt, Neel S., Kalwak, Krzysztof, and Corbacioglu, Selim

Published
2024
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3. Second allogeneic stem cell transplantation can rescue a significant proportion of patients with JMML relapsing after first allograft

Author

Vinci, Luca, Flotho, Christian, Noellke, Peter, Lebrecht, Dirk, Masetti, Riccardo, de Haas, Valerie, De Moerloose, Barbara, Dworzak, Michael, Hasle, Henrik, Güngör, Tayfun, Starý, Jan, Turkiewicz, Dominik, Ussowicz, Marek, de Heredia, Cristina Diaz, Buechner, Jochen, Jahnukainen, Kirsi, Kallay, Krisztian, Bodova, Ivana, Smith, Owen P., Zecca, Marco, Bresters, Dorine, Lang, Peter, Masmas, Tania Nicole, Meisel, Roland, Pichler, Herbert, Erlacher, Miriam, Göhring, Gudrun, Locatelli, Franco, Strahm, Brigitte, Niemeyer, Charlotte M., and Yoshimi, Ayami

Published
2023
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4. Hematopoietic stem cell transplantation for adolescents and adults with inborn errors of immunity: an EBMT IEWP study

Author

Aiuti, Alessandro, Maschan, Alexei, Aljurf, Mahmoud, Gedde-Dahl, Tobias, Gurman, Gunhan, Bordon, Victoria, Kriván, Gergely, Locatelli, Franco, Porta, Fulvio, Valcárcel, David, Beguin, Yves, Faraci, Maura, Kröger, Nicolaus, Kulagin, Aleksandr, Shaw, Peter J., Veelken, Joan Hendrik, Diaz de Heredia, Cristina, Fagioli, Franca, Felber, Matthias, Gruhn, Bernd, Holter, Wolfgang, Rössig, Claudia, Sedlacek, Petr, Apperley, Jane, Ayas, Mouhab, Bodova, Ivana, Choi, Goda, Cornelissen, J.J., Sirvent, Anne, Khan, Anjum, Kupesiz, Alphan, Lenhoff, Stig, Ozdogu, Hakan, von der Weid, Nicolas, Rovira, Montserrat, Schots, Rik, Vinh, Donald C., Albert, Michael H., Sirait, Tiarlan, Eikema, Dirk-Jan, Bakunina, Katerina, Wehr, Claudia, Suarez, Felipe, Fox, Maria Laura, Mahlaoui, Nizar, Gennery, Andrew R., Lankester, Arjan C., Beier, Rita, Bernardo, Maria Ester, Bigley, Venetia, Lindemans, Caroline A., Burns, Siobhan O., Carpenter, Ben, Dybko, Jaroslaw, Güngör, Tayfun, Hauck, Fabian, Lum, Su Han, Balashov, Dmitry, Meisel, Roland, Moshous, Despina, Schulz, Ansgar, Speckmann, Carsten, Slatter, Mary A., Strahm, Brigitte, Uckan-Cetinkaya, Duygu, Meyts, Isabelle, Vallée, Tanja C., Wynn, Robert, Neven, Bénédicte, and Morris, Emma C.

Published
2022
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5. Hematopoietic stem cell transplantation for Wiskott-Aldrich syndrome: an EBMT Inborn Errors Working Party analysis

Author

Albert, Michael H., Slatter, Mary A., Gennery, Andrew R., Güngör, Tayfun, Bakunina, Katerina, Markovitch, Benyamin, Hazelaar, Sheree, Sirait, Tiarlan, Courteille, Virginie, Aiuti, Alessandro, Aleinikova, Olga V., Balashov, Dmitry, Bernardo, Maria Ester, Bodova, Ivana, Bruno, Benedicte, Cavazzana, Marina, Chiesa, Robert, Fischer, Alain, Hauck, Fabian, Ifversen, Marianne, Kałwak, Krzysztof, Klein, Christoph, Kulagin, Alexander, Kupesiz, Alphan, Kuskonmaz, Baris, Lindemans, Caroline A., Locatelli, Franco, Lum, Su Han, Maschan, Alexey, Meisel, Roland, Moshous, Despina, Porta, Fulvio, Sauer, Martin G., Sedlacek, Petr, Schulz, Ansgar, Suarez, Felipe, Vallée, Tanja C., Winiarski, Jacek H., Zecca, Marco, Neven, Bénédicte, Veys, Paul, and Lankester, Arjan C.

Published
2022
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6. Comparison of outcomes of immunosuppressive therapy with rabbit versus horse antithymocyte globulin and cyclosporine a in children with acquired severe aplastic anemia.

Author

Yoshimi, Ayami, Noellke, Peter, Starý, Jan, Kállay, Krisztián, Smith, Owen, Locatelli, Franco, Buechner, Jochen, Bodova, Ivana, Sevilla, Julian, Schmugge, Markus, Bierings, Marc, Masmas, Tania, Dworzak, Michael, Labarque, Veerle, Pawelec, Katarzyna, Jahnukainen, Kirsi, Polychronopoulou, Sophia, Kjollerstrom, Paula, Kavcic, Marko, and Erlacher, Miriam

Published
2024
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7. P6 - NPM1 MUTATIONS IN CHILDREN WITH MYELODYSPLASTIC SYNDROME WITH EXCESS BLASTS

Author

Yoshimi, Ayami, Erlacher, Miriam, Noellke, Peter, Ramamoorthy, Senthilkumar, Göhring, Gudrun, Birenboim, Shlomit Barzilai –, Bodova, Ivana, Buechner, Jochen, Catala, Albert, Haas, Valérie De, Moerloose, Barbara De, Dworzak, Michael, Hasle, Henrik, Jahnukainen, Kirsi, Kallay, Krisztian, Kavcic, Marko, Kjollerstrom, Paula, Locatelli, Franco, Masetti, Riccardo, Polychronopoulou, Sophia, Schmugge, Markus, Smith, Owen, Stary, Jan, Turkiewicz, Dominik, Ussowicz, Marek, Rotari, Natalia, Wlodarski, Marcin, Strahm, Brigitte, and Niemeyer, Charlotte

Published
2023
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8. OC 20 - GENOTYPE/PHENOTYPE ASSOCIATIONS IN 174 INDIVIDUALS WITH GERMLINE GATA2 MUTATIONS

Author

Kotmayer, Lili, Kozyra, Emilia, Kaiser, Maximilian, Dworzak, Michael, Moerloose, Barbara De, Starý, Jan, Hasle, Henrik, Jahnukainen, Kirsi, Polychronopoulou, Sophia, Kállay, Krisztián, Smith, Owen, Barzilai, Shlomit, Masetti, Riccardo, Buechner, Jochen, Ussowicz, Marek, Kjollerstrom, Paula, Boďová, Ivana, Kavcic, Marko, Catala, Albert, Turkiewicz, Dominik, Schmugge, Markus, Haas, Valérie De, Voss, Rebecca, Bigas, Anna, Romero, Damia, Bödör, Csaba, Erlacher, Miriam, Giorgetti, Alessandra, Niemeyer, Charlotte, and Wlodarski, Marcin

Published
2023
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9. Specific Cytogenetic Abnormalities at Diagnosis Predict Survival after Hematopoietic Cell Transplant in Poor-Risk Pediatric Acute Myeloid Leukemia: A PDWP/EBMT Study

Author

Sharma, Akshay, Galimard, Jacques-Emmanuel, Pryce, Angharad, Bhoopalan, Senthil, Dalissier, Arnaud, Dalle, Jean-Hugues, Locatelli, Franco, Jubert, Charlotte, Mirci-Danicar, Oana, Kitra-Roussou, Vassiliki, Bertrand, Yves, Fagioli, Franca, Rialland, Fanny, Biffi, Alessandra, Wynn, Robert F, Michel, Gerard, Tambaro, Francesco Paolo P, Ahmari, Ali Abdallah, Tbakhi, Abdelghani, Furness, Caroline, Díaz Pérez, Miguel Ángel, Sedlacek, Petr, Bodova, Ivana, Faraci, Maura, Rao, Kanchan, Rubio, Marie Thérèse, Gibson, Brenda, Bhatt, Neel S., and Corbacioglu, Selim

Published
2022
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10. Publisher Correction : Clinical evolution, genetic landscape and trajectories of clonal hematopoiesis in SAMD9/SAMD9L syndromes (Nature Medicine, (2021), 27, 10, (1806-1817), )

Author

Sahoo, Sushree S., Pastor, Victor B., Goodings, Charnise, Voss, Rebecca K., Kozyra, Emilia J., Szvetnik, Amina, Noellke, Peter, Dworzak, Michael, Starý, Jan, Locatelli, Franco, Masetti, Riccardo, Schmugge, Markus, De Moerloose, Barbara, Catala, Albert, Kállay, Krisztián, Turkiewicz, Dominik, Hasle, Henrik, Buechner, Jochen, Jahnukainen, Kirsi, Ussowicz, Marek, Polychronopoulou, Sophia, Smith, Owen P., Fabri, Oksana, Barzilai, Shlomit, de Haas, Valerie, Baumann, Irith, Schwarz-Furlan, Stephan, Moerloose, Barbara De, Kallay, Krisztián, Smith, Owen, Haas, Valérie De, Gohring, Gudrun, Niemeyer, Charlotte, Nebral, Karin, Simonitsch-Kluppp, Ingrid, Paepe, Pascale De, Van Roy, Nadine, Campr, Vit, Zemanova, Zuzana, Clasen-Linde, Erik, Plesner, Tine, Schlegelberger, Brigitte, Rudelius, Martina, Manola, Kalliopi, Stefanaki, Kalliopi, Csomor, Judit, Andrikovics, Hajnalka, Betts, David, O’Sullivan, Maureen, Zohar, Yaniv, Jeison, Marta, Vito, Rita De, Pasquali, Francesco, Maldyk, Jadwiga, Haus, Olga, Alaiz, Helena, Kjollerstrom, Paula, Lemos, Luis Mascarenhas de, Bodova, Ivana, Čermák, Martin, Plank, Lukas, Gazic, Barbara, Kavcic, Marko, Podgornik, Helena, Ros, Margarita Llavador, Cervera, Jose, Gengler, Carole, Tchinda, Joelle, Beverloo, Berna, Leguit, Roos, Niewisch, Marena R., Sauer, Martin G., Burkhardt, Birgit, Lang, Peter, Bader, Peter, Beier, Rita, Müller, Ingo, Albert, Michael H., Meisel, Roland, Schulz, Ansgar, Cario, Gunnar, Panda, Pritam K., Wehrle, Julius, Hirabayashi, Shinsuke, Derecka, Marta, Durruthy-Durruthy, Robert, Göhring, Gudrun, Yoshimi-Noellke, Ayami, Ku, Manching, Lebrecht, Dirk, Erlacher, Miriam, Flotho, Christian, Strahm, Brigitte, Niemeyer, Charlotte M., and Wlodarski, Marcin W.

Subjects
Medizin
Abstract

Korrektur zu 10.1038/s41591-021-01511-6

Published
2021

11. Allogeneic Hematopoietic Stem Cell Transplantation for BCR/ABL1-Negative Myeloproliferative Neoplasms in Children - Retrospective Report on Behalf of I-BFM SCT Committee and EBMT Pediatric Diseases WP

Author

Patrick, Katharine, Gruhn, Bernd, Corbacioglu, Selim, Kupesiz, Alphan, Balduzzi, Adriana, Al-Seraihy, Amal, Wynn, Robert, Veys, Paul, Stepensky, Polina, Robinson, Stephen, Locatelli, Franco, Dalissier, Arnaud, Galimard, Jacques-Emmanuel, Wachowiak, Jacek, Tbakhi, Abdelghani, Diaz, Miguel Angel, Socie, Gerard, Sedlacek, Petr, Abboud, Miguel, Beelen, Dietrich, Krivan, Gergely, ÜNAL, ALİ, Bodova, Ivana, Freycon, Claire, Dalle, Jean-Hugues, Jubert, Charlotte, Ferster, Alina, Ifversen, Marianne, and Hamladji, Rose-Marie

Published
2020

13. Stem Cell Transplantation for Diamond–Blackfan Anemia. A Retrospective Study on Behalf of the Severe Aplastic Anemia Working Party of the European Blood and Marrow Transplantation Group (EBMT)

Author

Miano, Maurizio, Eikema, Dirk-Jan, de la Fuente, Josu, Bosman, Paul, Ghavamzadeh, Ardeshir, Smiers, Frans, Sengeløv, Henrik, Yesilipek, Akif, Formankova, Renata, Bader, Peter, Díaz Pérez, Miguel Ángel, Bertrand, Yves, Niemeyer, Charlotte, Diallo, Safiatou, Ansari, Marc, Bykova, Tatiana A, Faraci, Maura, Bonanomi, Sonia, Gozdzik, Jolanta, Satti, Tariq Mahmood, Bodova, Ivana, Wölfl, Matthias, Rocha, Vanderson G., Mellgren, Karin, Rascon, Jelena, Holter, Wolfgang, Lange, Andrzej, Meisel, Roland, Beguin, Yves, Mozo, Yasmina, Kriván, Gergely, Sirvent, Anne, Bruno, Benedicte, Dalle, Jean Hugues, Onofrillo, Daniela, Giardino, Stefano, Risitano, Antonio M., de Latour, Régis Peffault, and Dufour, Carlo

Published
2021
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14. Busulfan/Fludarabine- or Treosulfan/Fludarabine-Based Conditioning Regimen in Patients with Wiskott-Aldrich Syndrome Given Allogeneic Hematopoietic Cell Transplantation — an EBMT Inborn Errors Working Party and Scetide Retrospective Analysis

Author

Albert, Michael H., Slatter, Mary, Gennery, Andrew, Guengoer, Tayfun, Blok, Henric-Jan, Wang, Junfeng, Courteille, Virginie, Bernardo, Maria Ester, Bodova, Ivana, Bruno, Benedicte, Bykova, Tatjana A, Chiesa, Robert, Fischer, Alain, Formankova, Renata, Kalwak, Krzysztof, Klein, Christoph, Kozlovskaya, Svetlana, Kupesiz, O. Alphan, Locatelli, Franco, Mahlaoui, Nizar, Moshous, Despina, Neven, Benedicte, Porta, Fulvio, Schulz, Ansgar, Sykora, Karl-Walter, Unal, Ekrem, Winiarski, Jacek H., Zecca, Marco, Veys, Paul, and Lankester, Arjan

Published
2018
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15. Allogeneic stem cell transplantation for acquired pure red cell aplasia.

Author

Halkes, Constantijn, Wreede, Liesbeth C, Knol, Cora, Simand, Célestine, Aljurf, Mahmoud, Tbakhi, Abdelghani, Vazquez, Lourdes, Bloor, Adrian, Wagner‐Drouet, Eva, Vural, Filiz, Bodova, Ivana, Isaksson, Cecilia, Diaz, Miguel Ángel, Gruhn, Bernd, Snowden, John, Arat, Mutlu, Bazarbachi, Ali, Spilleboudt, Chloé, Kulagin, Alexander, and Marsh, Judith CW

Published
2019
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16. Busulfan/Fludarabine- or Treosulfan/Fludarabine-Based Conditioning Regimen for Patients with Wiskott-Aldrich Syndrome – an EBMT Inborn Errors Working Party and Scetide Study.

Author

Albert, Michael H., Slatter, Mary, Gennery, Andrew, Gungor, Tayfun, Blok, Henric-Jan, Hazelaar, Sheree, Wang, Junfeng, Courteille, Virginie, Mahlaoui, Nizar, Bernado, Maria Ester, Bodova, Ivana, Bruno, Bénédicte, Bykova, Tatiana, Chiesa, Robert, Fischer, Alain, Formankova, Renata, Kalwak, Krzysztof, Klein, Christoph, Kozlovskaya, Svetlana, and Kupesiz, Alphan

Subjects
*BUSULFAN, *WISKOTT-Aldrich syndrome, *INBORN errors of metabolism, *GENETIC disorder treatment, *HEMATOPOIETIC stem cell transplantation
Abstract

Introduction Excellent survival rates have been reported after allogeneic haematopoietic stem cell transplantation (HSCT) for Wiskott-Aldrich syndrome (WAS) patients. Recipient age >5 years in MUD HSCT as well as MMFD as donor were negative predictors for outcome. However, the vast majority of HSCTs in previously published studies were performed with (oral) busulfan/cyclophosphamide-based conditioning and in the early 2000 years or before. Objectives To compare OS and EFS after HSCT with either busulfan/fludarabine (BuFlu) ± thiotepa (TT) or treosulfan/fludarabine (TreoFlu) ± TT as recommended for primary immunodeficiencies since 2005 by the inborn errors working party (IEWP) of EBMT and ESID. Methods We performed a retrospective analysis via the EBMT and SCETIDE registries of WAS patients transplanted between 20006 and 2016 with these two regimens. At the time of this interim analysis, 174 patients were included, 92 (53%) with BuFlu±TT and 82 (47%) with TreoFlu±TT conditioning, with a median age of 1.6 years (0.2-30) at HSCT and a median follow-up of 32.9 months (1.5-128.9). Donors were MSD in 30, other MRD in 5, MUD (9/10 or 10/10) in 105, MMUD (<9/10) in 9 and MMFD in 25 (18 with ex-vivo T-cell depletion). Stem cell source was bone marrow in 93, peripheral blood in 62 and cord blood in 18. Results Two year overall survival (OS) of the entire cohort was 88.6% (95% c.i. 83.5%-93.6%). There was no significant difference in OS between BuFlu±TT or TreoFlu±TT conditioning (2-year OS 88.1% vs. 89.5%; p=0.7). Patients aged >5 years had a worse OS as compared to those 5 years or younger at HSCT (74.9% vs. 90.8%; p=0.005). The type of donor had no influence on OS: 96.4% for MSD/MFD, 86.8% for MUD/MMUD and 87.7% for MMFD (p=0.4). The rate of complete (≥90%) donor chimerism at last follow-up or before a secondary procedure (if a patient had one) was 41/42 (98%) in the BuFlu±TT group and 21/35 (60%) in the TreoFlu±TT group (p=0.0001). Twenty-six patients required a second procedure: stem cell boost in 4, donor lymphocyte infusion in 9, 2nd HSCT in 15 and splenectomy in 1. The 2-year cumulative incidence (CI) of second procedures was higher at 33.9% in the TreoFlu±TT versus 12.8% in the BuFlu±TT group (p=0.017), and 2-year EFS (events: second procedure or death) was 61.4% in the TreoFlu±TT and 75.0% in the BuFlu±TT group (p=0.2). Grade II-IV acute GVHD had the same incidence in both groups (24.4% vs. 26.3%; p=0.849) and chronic GVHD of any grade was borderline more frequent in the TreoFlu±TT group (17.2% vs 6.7%; p=0.054). Conclusion HSCT with either BuFlu±TT or TreoFlu±TT conditioning reliably cures almost 90% of patients with WAS regardless of donor type. Age >5 years at HSCT remains a negative risk factor. More patients were mixed chimeras and required second procedures after TreoFlu±TT than after BuFlu±TT conditioning. These data confirm the feasibility and efficacy of the regimens currently recommended by the IEWP. [ABSTRACT FROM AUTHOR]

Published
2019
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17. Matched unrelated donor transplantation versus haploidentical transplantation with post-transplant cyclophosphamide in children with acute myeloid leukemia: a PDWP-EBMT study.

Author

Ruggeri A, Santoro N, Galimard JE, Kalwak K, Algeri M, Zubarovskaya L, Czyzewski K, Skorobogatova E, Sedlacek P, Besley C, Balduzzi A, Bertrand Y, Peristeri J, Fagioli F, Ifversen M, Gozdzik J, Peters C, Versluijs B, Biffi A, Prete A, Faraci M, Ghemlas I, Bodova I, Aleinikova O, Dalissier A, Rocha V, and Corbacioglu S

Subjects
Humans, Child, Female, Male, Adolescent, Child, Preschool, Infant, Treatment Outcome, Histocompatibility Testing, Leukemia, Myeloid, Acute therapy, Leukemia, Myeloid, Acute mortality, Unrelated Donors, Cyclophosphamide therapeutic use, Cyclophosphamide administration & dosage, Hematopoietic Stem Cell Transplantation methods, Hematopoietic Stem Cell Transplantation adverse effects, Transplantation, Haploidentical methods, Graft vs Host Disease etiology, Graft vs Host Disease prevention & control, Transplantation Conditioning methods
Abstract

In children with acute myeloid leukemia (AML) who lack a human leukocyte antigen (HLA) identical sibling, the donor can be replaced with an HLA-matched unrelated donor (MUD) or a haploidentical donor (haplo). We compared outcomes of patients <18 years with AML in first and second complete remission (CR1 and CR2) undergoing a hematopoietic stem cell transplantation (HCT) either with a MUD with anti-thymocyte globulin (ATG) (N=420) or a haplo HCT with post-transplant cyclophosphamide (PT-CY) (N=96) after a myeloablative conditioning regimen (MAC) between 2011 and 2021, reported to the European Society for Blood and Marrow Transplantation. A matched pair analysis was performed to adjust for differences among groups. The final analysis was performed on 253 MUD and 95 haplo-HCT. In the matched cohort, median age at HCT was 11.2 and 10 years and median year of HCT was 2017 and 2018, in MUD and haplo-HCT recipients, respectively. The risk of grade III-IV acute graft-versus-host disease (aGVHD) was significantly higher in the haplo group (hazard ratio [HR]=2.33, 95% confidence interval [CI]: 1.18-4.58; P=0.01). No significant differences were found in 2 years overall survival (OS; 78.4% vs. 71.5%; HR=1.39, 95% CI: 0.84-2.31; P=0.19), leukemia-free survival (LFS; 72.7% vs. 69.5%; HR=1.22, 95% CI: 0.76-1.95; P=0.41), CI of relapse (RI; 19.3% vs. 19.5%; HR=1.14, 95% CI: 0.62-2.08; P=0.68) non-relapse-mortality (NRM; 8% vs. 11%; HR=1.39, 95% CI: 0.66-2.93; P=0.39) and graft-versus-host free relapse-free survival (GRFS; 60.7% vs. 54.5%, HR=1.38, 95% CI: 0.95-2.02; P=0.09) after MUD and haplo-HCT respectively. Our study suggests that haplo-HCT with PT-CY is a suitable option to transplant children with AML lacking a matched related donor.

Published
2024
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18. Hematopoietic stem cell transplantation for adolescents and adults with inborn errors of immunity: an EBMT IEWP study.

Author

Albert MH, Sirait T, Eikema DJ, Bakunina K, Wehr C, Suarez F, Fox ML, Mahlaoui N, Gennery AR, Lankester AC, Beier R, Bernardo ME, Bigley V, Lindemans CA, Burns SO, Carpenter B, Dybko J, Güngör T, Hauck F, Lum SH, Balashov D, Meisel R, Moshous D, Schulz A, Speckmann C, Slatter MA, Strahm B, Uckan-Cetinkaya D, Meyts I, Vallée TC, Wynn R, Neven B, Morris EC, Aiuti A, Maschan A, Aljurf M, Gedde-Dahl T, Gurman G, Bordon V, Kriván G, Locatelli F, Porta F, Valcárcel D, Beguin Y, Faraci M, Kröger N, Kulagin A, Shaw PJ, Veelken JH, Diaz de Heredia C, Fagioli F, Felber M, Gruhn B, Holter W, Rössig C, Sedlacek P, Apperley J, Ayas M, Bodova I, Choi G, Cornelissen JJ, Sirvent A, Khan A, Kupesiz A, Lenhoff S, Ozdogu H, von der Weid N, Rovira M, Schots R, and Vinh DC

Subjects
Adolescent, Adult, Child, Humans, Infant, Middle Aged, Retrospective Studies, Transplantation, Homologous, Young Adult, Bronchiectasis etiology, Graft vs Host Disease, Hematopoietic Stem Cell Transplantation adverse effects
Abstract

Allogeneic hematopoietic stem cell transplantation (HSCT) is the gold standard curative therapy for infants and children with many inborn errors of immunity (IEI), but adolescents and adults with IEI are rarely referred for transplant. Lack of published HSCT outcome data outside small, single-center studies and perceived high risk of transplant-related mortality have delayed the adoption of HSCT for IEI patients presenting or developing significant organ damage later in life. This large retrospective, multicenter HSCT outcome study reports on 329 IEI patients (age range, 15-62.5 years at HSCT). Patients underwent first HSCT between 2000 and 2019. Primary endpoints were overall survival (OS) and event-free survival (EFS). We also evaluated the influence of IEI-subgroup and IEI-specific risk factors at HSCT, including infections, bronchiectasis, colitis, malignancy, inflammatory lung disease, splenectomy, hepatic dysfunction, and systemic immunosuppression. At a median follow-up of 44.3 months, the estimated OS at 1 and 5 years post-HSCT for all patients was 78% and 71%, and EFS was 65% and 62%, respectively, with low rates of severe acute (8%) or extensive chronic (7%) graft-versus-host disease. On univariate analysis, OS and EFS were inferior in patients with primary antibody deficiency, bronchiectasis, prior splenectomy, hepatic comorbidity, and higher hematopoietic cell transplant comorbidity index scores. On multivariable analysis, EFS was inferior in those with a higher number of IEI-associated complications. Neither age nor donor had a significant effect on OS or EFS. We have identified age-independent risk factors for adverse outcome, providing much needed evidence to identify which patients are most likely to benefit from HSCT., (© 2022 by The American Society of Hematology.)

Published
2022
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19. A case of Exophiala dermatitidis infection in a child after allogeneic stem cell transplantation: case report and literature review of paediatric cases.

Author

Tanuskova D, Horakova J, Buzassyova D, Poczova M, Bodova I, Svec P, Chocholova A, Adamcakova J, Sykora T, Pozdechova M, Geczova L, and Kolenova A

Abstract

Introduction. Exophiala dermatitidis is a relatively common environmental black yeast with worldwide distribution and is a rare cause of fungal infection, mostly in patients with certain predisposing factors. Due to the rarity of the infection, little is known about the specific predisposing factors, way of infection or treatment. Case presentation. Here, we report what is to our knowledge the first case of E. dermatitidis infection in a child after allogeneic stem cell transplantation. We also review all paediatric cases reported in the literature since 1993. Conclusion. This is, to our knowledge, the first reported case of E. dermatitidis infection in a child after allogeneic stem cell transplantation. This report should increase the awareness of E. dermatitidis in immunocompromised paediatric patients, particularly after stem cell transplantation.

Published
2017
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