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51. Survival in primary hemophagocytic lymphohistiocytosis 2016-2021: etoposide is better than its reputation

Author

Böhm, Svea, primary, Wustrau, Katharina, additional, Pachlopnik Schmid, Jana, additional, Prader, Seraina, additional, Ahlman, Martina, additional, Yacobovich, Joanne, additional, Beier, Rita, additional, Speckmann, Carsten, additional, Behnisch, Wolfgang, additional, Ifversen, Marianne, additional, Jordan, Michael B., additional, Marsh, Rebecca A, additional, Naumann-Bartsch, Nora, additional, Mauz-Koerholz, Christine, additional, Honig, Manfred, additional, Schulz, Ansgar S, additional, Malinowska, Iwona, additional, Hines, Melissa Ruth, additional, Nichols, Kim E, additional, Gil-Herrera, Juana, additional, Talano, Julie-An, additional, Crooks, Bruce, additional, Formankova, Renata, additional, Jorch, Norbert, additional, Bakhtiar, Shahrzad, additional, Kühnle, Ingrid, additional, Streiter, Monika, additional, Nathrath, Michaela, additional, Russo, Alexandra, additional, Duerken, Matthias, additional, Lang, Peter, additional, Lindemans, Caroline A., additional, Henter, Jan-Inge, additional, Lehmberg, Kai, additional, and Ehl, Stephan, additional

Published
2023
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52. Efficacy of MSC for steroid-refractory acute GVHD associates with MSC donor age and a defined molecular profile

Author

van der Wagen, Lotte E., Miranda-Bedate, Alberto, Janssen, Anke, Fernando, Febilla, Appukudige, Nagesha, van Dooremalen, Sanne, Westinga, Kasper, Admiraal, Rick, Lorenowicz, Magdalena J., Huls, Gerwin, Janssen, Jeroen J. W. M., Broers, Annoek E. C., van der Velden, Walter J. F. M., van Marwijk Kooy, Rien, Hazenberg, Mette D., de Haar, Colin, Lindemans, Caroline, Jan Boelens, Jaap, and Kuball, Jürgen

Published
2020
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54. Health-Related Quality of Life Following Allogeneic Hematopoietic Cell Transplantation with Omidubicel versus Umbilical Cord Blood

Author

Lin, Chenyu, Sajeev, Gautam, Stiff, Patrick J., Brunstein, Claudio G., Cutler, Corey, Sanz, Guillermo, Lindemans, Caroline A., Rezvani, Andrew R., Hanna, Rabi, Koh, Liang Piu, Maziarz, Richard T., Hwang, William Y.K., Song, Yan, Liu, Qing, Manghani, Rocio, Sivaraman, Smitha, Signorovitch, James, Horwitz, Mitchell E., and Sung, Anthony D.

Published
2023
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56. Hematopoietic stem cell transplantation for CD40 ligand deficiency: Results from an EBMT/ESID-IEWP-SCETIDE-PIDTC study

Author

Ferrua, Francesca, Galimberti, Stefania, Courteille, Virginie, Slatter, Mary Anne, Booth, Claire, Moshous, Despina, Neven, Benedicte, Blanche, Stephane, Cavazzana, Marina, Laberko, Alexandra, Shcherbina, Anna, Balashov, Dmitry, Soncini, Elena, Porta, Fulvio, Al-Mousa, Hamoud, Al-Saud, Bandar, Al-Dhekri, Hasan, Arnaout, Rand, Formankova, Renata, Bertrand, Yves, Lange, Andrzej, Smart, Joanne, Wolska-Kusnierz, Beata, Aquino, Victor M., Dvorak, Christopher C., Fasth, Anders, Fouyssac, Fanny, Heilmann, Carsten, Hoenig, Manfred, Schuetz, Catharina, Kelečić, Jadranka, Bredius, Robbert G.M., Lankester, Arjan C., Lindemans, Caroline A., Suarez, Felipe, Sullivan, Kathleen E., Albert, Michael H., Kałwak, Krzysztof, Barlogis, Vincent, Bhatia, Monica, Bordon, Victoria, Czogala, Wojciech, Alonso, Laura, Dogu, Figen, Gozdzik, Jolanta, Ikinciogullari, Aydan, Kriván, Gergely, Ljungman, Per, Meyts, Isabelle, Mustillo, Peter, Smith, Angela R., Speckmann, Carsten, Sundin, Mikael, Keogh, Steven John, Shaw, Peter John, Boelens, Jaap Jan, Schulz, Ansgar S., Sedlacek, Petr, Veys, Paul, Mahlaoui, Nizar, Janda, Ales, Davies, E. Graham, Fischer, Alain, Cowan, Morton J., and Gennery, Andrew Richard

Published
2019
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57. CT-314 Multicenter Long-Term Follow Up of Allogeneic Hematopoietic Stem Cell Transplantation With Omidubicel: A Pooled Analysis of Five Prospective Clinical Trials

Author

Lin, Chenyu, Schwarzbach, Aurelie, Montesinos, Pau, Stiff, Patrick, Cutler, Corey, Parikh, Suhag, Brunstein, Claudio, Lindemans, Caroline A., Hanna, Rabi, Koh, Liang Piu, Maziarz, Richard T., Keating, Amy K., Huang, William Y.K., Rezvani, Andrew R., Valcarcel, David, Fernandes, Juliana F., Badell, Isabell S., Jagasia, Madan H., Frankfurt, Olga, Ram, Ron, McGuirk, Joseph P., Kurtzberg, Joanne, Sanz, Guillermo, Simantov, Ronit, and Horwitz, Mitchell E.

Published
2022
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58. CT-039 Health-Related Quality of Life Following Allogeneic Hematopoietic Stem Cell Transplantation With Omidubicel Versus Standard Umbilical Cord Blood

Author

Lin, Chenyu, Sajeev, Gautam, Stiff, Patrick, Brunstein, Claudio, Cutler, Corey, Sanz, Guillermo, Lindemans, Caroline, Rezvani, Andrew, Hanna, Rabi, Koh, Liang Piu, Maziarz, Richard, Hwang, William, Song, Yan, Liu, Qing, Manghani, Rocio, Sivaraman, Smitha, Signorovitch, James, Galamidi-Cohen, Einat, Horwitz, Mitchell, and Sung, Anthony

Published
2022
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61. Excellent leukemia control after second hematopoietic cell transplants with unrelated cord blood grafts for post-transplant relapse in pediatric patients

Author

Troullioud Lucas, Alexandre G., primary, Boelens, Jaap Jan, additional, Prockop, Susan E., additional, Curran, Kevin J., additional, Bresters, Dorine, additional, Kollen, Wouter, additional, Versluys, Birgitta, additional, Bierings, Marc B., additional, Archer, Anne, additional, Davis, Eric, additional, Klein, Elizabeth, additional, Kernan, Nancy A., additional, Lindemans, Caroline A., additional, and Scaradavou, Andromachi, additional

Published
2023
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63. P037: Design of a multi-center randomized phase 3 clinical trial (HURCULES) evaluating OTL-203 in MPS-IH vs allogeneic hematopoietic stem cell transplantation

Author

Harmatz, Paul, Wynn, Robert, Gupta, Ashish, Kharbanda, Sandhya, Lindemans, Caroline, Ahrens-Nicklas, Rebecca, van Hasselt, Peter, Lund, Troy, Olson, Timothy, Tucci, Francesca, Martin, Leonie, Boeglin, Nathalie, Brooks, Jean, Syonmez, Su, Campbell, Laura, Jones, Simon, Orchard, Paul, and Bernardo, Maria Ester

Published
2024
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64. Neurodegenerative disease after hematopoietic stem cell transplantation in metachromatic leukodystrophy

Author

Al‐Saady, Murtadha, primary, Beerepoot, Shanice, additional, Plug, Bonnie C., additional, Breur, Marjolein, additional, Galabova, Hristina, additional, Pouwels, Petra J. W., additional, Boelens, Jaap‐Jan, additional, Lindemans, Caroline, additional, van Hasselt, Peter M., additional, Matzner, Ulrich, additional, Vanderver, Adeline, additional, Bugiani, Marianna, additional, van der Knaap, Marjo S., additional, and Wolf, Nicole I., additional

Published
2023
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65. Multicenter Long-Term Follow-Up of Allogeneic Hematopoietic Cell Transplantation with Omidubicel: A Pooled Analysis of Five Prospective Clinical Trials

Author

Lin, Chenyu, primary, Schwarzbach, Aurelie, additional, Sanz, Jaime, additional, Montesinos, Pau, additional, Stiff, Patrick, additional, Parikh, Suhag, additional, Brunstein, Claudio, additional, Cutler, Corey, additional, Lindemans, Caroline A., additional, Hanna, Rabi, additional, Koh, Liang Piu, additional, Jagasia, Madan H., additional, Valcarcel, David, additional, Maziarz, Richard T., additional, Keating, Amy K., additional, Hwang, William Y.K., additional, Rezvani, Andrew R., additional, Karras, Nicole A., additional, Fernandes, Juliana F., additional, Rocha, Vanderson, additional, Badell, Isabel, additional, Ram, Ron, additional, Schiller, Gary J., additional, Volodin, Leonid, additional, Walters, Mark C., additional, Hamerschlak, Nelson, additional, Cilloni, Daniela, additional, Frankfurt, Olga, additional, McGuirk, Joseph P., additional, Kurtzberg, Joanne, additional, Sanz, Guillermo, additional, Simantov, Ronit, additional, and Horwitz, Mitchell E., additional

Published
2023
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68. Physical performance before and after hematopoietic stem cell transplantation in pediatric patients: a potential role for prehabilitation

Author

den Hartog, Emma, Lindemans, Caroline A., Fiocco, Marta, Tissing, Wim J. E., and Verwaaijen, Emma J.

Abstract

The trajectory of hematopoietic stem cell transplantation (HSCT) is often accompanied by physically disabling complications that impair physical performance of pediatric patients. However, knowledge about when impairments in physical performance arise and the factors contributing to these impairments is limited. Therefore, we conducted a retrospective analysis of physical performance 100 days post-HSCT in patients aged 3–18 years. Additionally, we aim to elucidate the relationship between pre- and post-HSCT physical performance and to unravel the impact of intensive HSCT procedures on post-HSCT physical performance. To explore associations between physical performance outcomes post-HSCT and covariates, linear regression models were estimated. Seventy-seven patients were included with a median age of 11.8 years (interquartile range: 5.9, 14.8). Patients had lower hip flexion muscle strength and appendicular skeletal muscle mass and a slower rising from the floor time 100 days post-HSCT compared to average values of the normal population. Pre-HSCT physical performance was positively associated with physical performance post-HSCT, independent of age, the cumulative glucocorticoids dosage administered and the total duration of hospitalization during the HSCT trajectory. This explorative study highlights the potential role of prehabilitation in enhancing physical performance of pediatric HSCT patients.

Published
2024
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71. Vaccination practices in pediatric transplantation: A survey among member centers of the European reference network TransplantChild.

Author

Donà, Daniele, Bravo‐Gallego, Luz Yadira, Remacha, Esteban Frauca, Cananzi, Mara, Gastaldi, Andrea, Canizalez, Juan Torres, Stephenne, Xavier, Lacaille, Florence, Lindemans, Caroline, Calore, Elisabetta, Galea, Nathalie, Benetti, Elisa, Nachbaur, Edith, Sandes, Ana Rita, Teixeira, Ana, Ferreira, Sandra, Klaudel‐Dreszler, Maja, Ackermann, Oanez, Boyer, Olivia, and Espinosa, Laura

Subjects
CHICKENPOX, VACCINATION, CHICKENPOX vaccines, VACCINATION coverage, MMR vaccines, HEMATOPOIETIC stem cell transplantation
Abstract

Background: There is considerable variation in vaccination practices between pediatric transplant centers. This study aims to evaluate active immunization attitudes and practices among ERN‐TransplantChild centers and identify potential areas of improvement that could be addressed by shared evidence‐based protocols. Methods: A cross‐sectional questionnaire of attitudes and practices toward immunization of pediatric SOT and HSCT candidates and recipients was sent to a representative member of multidisciplinary teams from 27 European centers belonging to the ERN‐TransplantChild. Results: A total of 28/62 SOT programs and 6/12 HSCT programs across 21 European centers participated. A quarter of centers did not have an on‐site protocol for the immunizations. At the time of transplantation, pediatric candidates were fully immunized (80%–100%) in 57% and 33% of the SOT and HSCT programs. Variations in the time between vaccine administration and admission to the waiting list were reported between the centers, with 2 weeks for inactivated vaccines and variable time (2–4 weeks) for live‐attenuated vaccines (LAVs). Almost all sites recommended immunization in the post‐transplant period, with a time window of 4–8 months for the inactivated vaccines and 16–24 months for MMR and Varicella vaccines. Only five sites administer LAVs after transplantation, with seroconversion evaluated in 80% of cases. Conclusions: The immunization coverage of European pediatric transplant recipients is still inconsistent and far from adequate. This survey is a starting point for developing shared evidence‐based immunization protocols for safe vaccination among pediatric transplant centers and generating new research studies. [ABSTRACT FROM AUTHOR]

Published
2023
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72. Chemotherapy-induced intestinal injury promotes Galectin-9-driven modulation of T cell function

Author

Jansen, Suze A., Cutilli, Alessandro, de Koning, Coco, van Hoesel, Marliek, Sierra, Leire Saiz, Nierkens, Stefan, Mokry, Michal, Nieuwenhuis, Edward E.S., Hanash, Alan M., Mocholi, Enric, Coffer, Paul J., and Lindemans, Caroline A.

Subjects
Article
Abstract

The intestine is vulnerable to chemotherapy-induced toxicity due to its high epithelial proliferative rate, making gut toxicity an off-target effect in several cancer treatments, including conditioning regimens for allogeneic hematopoietic cell transplantation (allo-HCT). In allo-HCT, intestinal damage is an important factor in the development of Graft-versus-Host Disease (GVHD), an immune complication in which donor immune cells attack the recipient’s tissues. Here, we developed a novel human intestinal organoid-based 3D model system to study the direct effect of chemotherapy-induced intestinal epithelial damage on T cell behavior. Chemotherapy treatment using busulfan, fludarabine, and clofarabine led to damage responses in organoids resulting in increased T cell migration, activation, and proliferation in ex-vivoco-culture assays. We identified galectin-9 (Gal-9), a beta-galactoside-binding lectin released by damaged organoids, as a key molecule mediating T cell responses to damage. Increased levels of Gal-9 were also found in the plasma of allo-HCT patients who later developed acute GVHD, supporting the predictive value of the model system in the clinical setting. This study highlights the potential contribution of chemotherapy-induced epithelial damage to the pathogenesis of intestinal GVHD through direct effects on T cell activation and trafficking promoted by galectin-9.

Published
2023

76. Atidarsagene Autotemcel, a European Post-Regulatory Approval Model for Delivery of Autologous Hematopoietic Stem Cell Gene Therapy Products Via a Network of Qualified Treatment Centers (QTCs)

Author

Wynn, Robert F., primary, Jones, Simon, additional, Fumagalli, Francesca, additional, Calbi, Valeria, additional, Aiuti, Alessandro, additional, Gröschel, Samuel, additional, Lang, Peter, additional, van Hasselt, Peter, additional, Lindemans, Caroline A., additional, Sevin, Caroline, additional, Dalle, Jean-Hugues, additional, and Snell, Katie, additional

Published
2023
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77. Atidarsagene autotemcel, a European post-regulatory approval model for delivery of autologous hematopoietic stem cell gene therapy products via a network of qualified treatment centers (QTCs)

Author

Jones, Simon, primary, Wynn, Robert, additional, Fumagalli, Francesca, additional, Calbi, Valeria, additional, Aiuti, Alessandro, additional, Groeschel, Samuel, additional, Lindemans, Caroline, additional, Sevin, Caroline, additional, Dalle, Jean-Hugues, additional, van Hasselt, Peter, additional, and Snell, Katie, additional

Published
2023
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79. Early Lymphocyte Immune Reconstitution As Predictor for Outcomes after Allogeneic Hematopoietic Cell Transplantation for Malignant Indications; A Tri-Institutional Analysis

Author

Troullioud Lucas, Alexandre G., primary, Lindemans, Caroline A., additional, Bhoopalan, Senthil Velan, additional, Dandis, Rana, additional, Prockop, Susan, additional, Keerthi, Dinesh, additional, de Koning, Coco, additional, Sharma, Akshay, additional, Nierkens, Stefan, additional, and Boelens, Jaap-Jan, additional

Published
2023
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81. Busulfan target exposure attainment in children undergoing allogeneic hematopoietic cell transplantation: a single day versus a multiday therapeutic drug monitoring regimen.

Author

Bognàr, T. (Tim), primary, Kingma, J. S. (Jurjen), additional, Smeijsters, E. H. (Erin), additional, Elst, K. C. M. (Kim) van der, additional, de Kanter, C. T. M. (Klaartje), additional, Lindemans, Caroline, additional, Egberts, A. C. G. (Toine), additional, Bartelink, I. H. (Imke), additional, and Lalmohammed, Arief, additional

Published
2023
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82. Early cytomegalovirus reactivation remains associated with increased transplant-related mortality in the current era: a CIBMTR analysis

Author

Teira, Pierre, Battiwalla, Minoo, Ramanathan, Muthalagu, Barrett, A. John, Ahn, Kwang Woo, Chen, Min, Green, Jaime S., Saad, Ayman, Antin, Joseph H., Savani, Bipin N., Lazarus, Hillard M., Seftel, Matthew, Saber, Wael, Marks, David, Aljurf, Mahmoud, Norkin, Maxim, Wingard, John R., Lindemans, Caroline A., Boeckh, Michael, Riches, Marcie L., and Auletta, Jeffery J.

Published
2016
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83. Additional file 1 of Meningococcal ACWY conjugate vaccine immunogenicity in adolescents with primary or secondary immune deficiencies, a prospective observational cohort study

Author

Ohm, Milou, van Straalen, Joeri W, de Joode-Smink, Gerrie, van Montfrans, Joris, Bartels, Marije, van Wildenbeest, Joanne G, Lindemans, Caroline A, Wennink, Roos AW, de Boer, Joke H, Sanders, Elisabeth AM, Verduyn-Lunel, Frans M, Berbers, Guy AM, Wulffraat, Nico M, and Jansen, Marc H.A.

Abstract

Supplementary Material 1

Published
2023
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84. Additional file 2 of Meningococcal ACWY conjugate vaccine immunogenicity in adolescents with primary or secondary immune deficiencies, a prospective observational cohort study

Author

Ohm, Milou, van Straalen, Joeri W, de Joode-Smink, Gerrie, van Montfrans, Joris, Bartels, Marije, van Wildenbeest, Joanne G, Lindemans, Caroline A, Wennink, Roos AW, de Boer, Joke H, Sanders, Elisabeth AM, Verduyn-Lunel, Frans M, Berbers, Guy AM, Wulffraat, Nico M, and Jansen, Marc H.A.

Abstract

Supplementary Material 2

Published
2023
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86. Multicenter experience in hematopoietic stem cell transplantation for serious complications of common variable immunodeficiency

Author

Wehr, Claudia, Gennery, Andrew R., Lindemans, Caroline, Schulz, Ansgar, Hoenig, Manfred, Marks, Reinhard, Recher, Mike, Gruhn, Bernd, Holbro, Andreas, Heijnen, Ingmar, Meyer, Deborah, Grigoleit, Goetz, Einsele, Hermann, Baumann, Ulrich, Witte, Thorsten, Sykora, Karl-Walter, Goldacker, Sigune, Regairaz, Lorena, Aksoylar, Serap, Ardeniz, Ömur, Zecca, Marco, Zdziarski, Przemyslaw, Meyts, Isabelle, Matthes-Martin, Susanne, Imai, Kohsuke, Kamae, Chikako, Fielding, Adele, Seneviratne, Suranjith, Mahlaoui, Nizar, Slatter, Mary A., Güngör, Tayfun, Arkwright, Peter D., van Montfrans, Joris, Sullivan, Kathleen E., Grimbacher, Bodo, Cant, Andrew, Peter, Hans-Hartmut, Finke, Juergen, Gaspar, H. Bobby, Warnatz, Klaus, and Rizzi, Marta

Published
2015
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88. Hematopoietic stem cell transplantation for adolescents and adults with inborn errors of immunity: an EBMT IEWP study

Author

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

Published
2022
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89. Poster: CT-039 Health-Related Quality of Life Following Allogeneic Hematopoietic Stem Cell Transplantation With Omidubicel Versus Standard Umbilical Cord Blood

Author

Lin, Chenyu, primary, Sajeev, Gautam, additional, Stiff, Patrick, additional, Brunstein, Claudio, additional, Cutler, Corey, additional, Sanz, Guillermo, additional, Lindemans, Caroline, additional, Rezvani, Andrew, additional, Hanna, Rabi, additional, Koh, Liang Piu, additional, Maziarz, Richard, additional, Hwang, William, additional, Song, Yan, additional, Liu, Qing, additional, Manghani, Rocio, additional, Sivaraman, Smitha, additional, Signorovitch, James, additional, Galamidi-Cohen, Einat, additional, Horwitz, Mitchell, additional, and Sung, Anthony, additional

Published
2022
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90. Poster: CT-314 Multicenter Long-Term Follow Up of Allogeneic Hematopoietic Stem Cell Transplantation With Omidubicel: A Pooled Analysis of Five Prospective Clinical Trials

Author

Lin, Chenyu, primary, Schwarzbach, Aurelie, additional, Montesinos, Pau, additional, Stiff, Patrick, additional, Cutler, Corey, additional, Parikh, Suhag, additional, Brunstein, Claudio, additional, Lindemans, Caroline A., additional, Hanna, Rabi, additional, Koh, Liang Piu, additional, Maziarz, Richard T., additional, Keating, Amy K., additional, Huang, William Y.K., additional, Rezvani, Andrew R., additional, Valcarcel, David, additional, Fernandes, Juliana F., additional, Badell, Isabell S., additional, Jagasia, Madan H., additional, Frankfurt, Olga, additional, Ram, Ron, additional, McGuirk, Joseph P., additional, Kurtzberg, Joanne, additional, Sanz, Guillermo, additional, Simantov, Ronit, additional, and Horwitz, Mitchell E., additional

Published
2022
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91. Abstract 8 Health-Related Quality of Life (HRQL) Following Transplantation with Omidubicel Versus Umbilical Cord Blood (UCB) in Patients with Hematologic Malignancies: Results from a Phase III Randomized, Multicenter Study

Author

Lin, Chenyu, primary, Sajeev, Gautam, additional, Sung, Anthony, additional, Stiff, Patrick, additional, Brunstein, Claudio, additional, Cutler, Corey, additional, Sanz, Guillermo, additional, Lindemans, Caroline, additional, Rezvani, Andrew, additional, Hanna, Rabi, additional, Koh, Liang, additional, Maziarz, Richard, additional, Hwang, William, additional, Song, Yan, additional, Liu, Qing, additional, Manghani, Rocio, additional, Sivaraman, Smitha, additional, Signorovitch, James, additional, and Horwitz, Mitchell, additional

Published
2022
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93. Interleukin-22 promotes intestinal-stem-cell-mediated epithelial regeneration

Author

Lindemans, Caroline A., Calafiore, Marco, Mertelsmann, Anna M., O'Connor, Margaret H., Dudakov, Jarrod A., Jenq, Robert R., Velardi, Enrico, Young, Lauren F., Smith, Odette M., Lawrence, Gillian, Ivanov, Juliet A., Fu, Ya-Yuan, Takashima, Shuichiro, Hua, Guoqiang, Martin, Maria L., O'Rourke, Kevin P., Lo, Yuan-Hung, Mokry, Michal, Romera-Hernandez, Monica, Cupedo, Tom, Dow, Lukas E., Nieuwenhuis, Edward E., Shroyer, Noah F., Liu, Chen, Kolesnick, Richard, van den Brink, Marcel R.M., and Hanash, Alan M.

Subjects
Regeneration (Biology) -- Health aspects, Interleukins -- Health aspects, Epithelial cells -- Physiological aspects, Environmental issues, Science and technology, Zoology and wildlife conservation
Abstract

Epithelial regeneration is critical for barrier maintenance and organ function after intestinal injury. The intestinal stem cell (ISC) niche provides Wnt, Notch and epidermal growth factor (EGF) signals supporting [Lgr5.sup.+] crypt base columnar ISCs for normal epithelial maintenance (1,2). However, little is known about the regulation of the ISC compartment after tissue damage. Using ex vivo organoid cultures, here we show that innate lymphoid cells (ILCs), potent producers of interleukin-22 (IL-22) after intestinal injury (3,4), increase the growth of mouse small intestine organoids in an IL-22-dependent fashion. Recombinant IL-22 directly targeted ISCs, augmenting the growth of both mouse and human intestinal organoids, increasing proliferation and promoting ISC expansion. IL-22 induced STAT3 phosphorylation in [Lgr5.sup.+] ISCs, and STAT3 was crucial for both organoid formation and IL-22-mediated regeneration. Treatment with IL-22 in vivo after mouse allogeneic bone marrow transplantation enhanced the recovery of ISCs, increased epithelial regeneration and reduced intestinal pathology and mortality from graft-versus-host disease. ATOH1deficient organoid culture demonstrated that IL-22 induced epithelial regeneration independently of the Paneth cell niche. Our findings reveal a fundamental mechanism by which the immune system is able to support the intestinal epithelium, activating ISCs to promote regeneration., The epithelial layer in the gastrointestinal tract represents a fundamental line of defence against potential enteric pathogens. Paneth cells contribute to this defence by producing antimicrobial molecules and by providing [...]

Published
2015

96. Hematopoietic cell transplantation in severe combined immunodeficiency: The SCETIDE 2006-2014 European cohort

Author

Lankester, Arjan C., primary, Neven, Benedicte, additional, Mahlaoui, Nizar, additional, von Asmuth, Erik G.J., additional, Courteille, Virginie, additional, Alligon, Mikael, additional, Albert, Michael H., additional, Serra, Isabelle Badell, additional, Bader, Peter, additional, Balashov, Dmitry, additional, Beier, Rita, additional, Bertrand, Yves, additional, Blanche, Stephane, additional, Bordon, Victoria, additional, Bredius, Robbert G., additional, Cant, Andrew, additional, Cavazzana, Marina, additional, Diaz-de-Heredia, Cristina, additional, Dogu, Figen, additional, Ehlert, Karoline, additional, Entz-Werle, Natacha, additional, Fasth, Anders, additional, Ferrua, Francesca, additional, Ferster, Alina, additional, Formankova, Renata, additional, Friedrich, Wilhelm, additional, Gonzalez-Vicent, Marta, additional, Gozdzik, Jolanta, additional, Güngör, Tayfun, additional, Hoenig, Manfred, additional, Ikinciogullari, Aydan, additional, Kalwak, Krzysztof, additional, Kansoy, Savas, additional, Kupesiz, Alphan, additional, Lanfranchi, Arnalda, additional, Lindemans, Caroline A., additional, Meisel, Roland, additional, Michel, Gerard, additional, Miranda, Nuno A.A., additional, Moraleda, Jose, additional, Moshous, Despina, additional, Pichler, Herbert, additional, Rao, Kanchan, additional, Sedlacek, Petr, additional, Slatter, Mary, additional, Soncini, Elena, additional, Speckmann, Carsten, additional, Sundin, Mikael, additional, Toren, Amos, additional, Vettenranta, Kim, additional, Worth, Austen, additional, Yeşilipek, Mehmet A., additional, Zecca, Marco, additional, Porta, Fulvio, additional, Schulz, Ansgar, additional, Veys, Paul, additional, Fischer, Alain, additional, and Gennery, Andrew R., additional

Published
2022
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97. Case Report: Lessons Learned From Subsequent Autologous and Allogeneic Hematopoietic Stem Cell Transplantations in a Pediatric Patient With Relapsing Polychondritis

Author

Veldkamp, Saskia R., Jansen, Marc H. A., Swart, Joost F., and Lindemans, Caroline A.

Subjects
Immunology, Immunology and Allergy
Abstract

BackgroundAutologous hematopoietic stem cell transplantation (autoHSCT) is increasingly being recognized as a treatment option for severe refractory autoimmune diseases (AD). However, efficacy is hampered by high relapse rates. In contrast, allogeneic HSCT (alloHSCT) has high potential to cure AD, but is associated with significant morbidity and mortality, and data in AD are limited. Experience with autoHSCT in relapsing polychondritis, a rare episodic inflammatory disorder characterized by destruction of cartilage, is scarce and alloHSCT has not been described before.Case PresentationHere, we present a case of a 9-year-old girl who was diagnosed with relapsing polychondritis, with severe airway involvement requiring a tracheostomy. The disease proved to be steroid-dependent and refractory to a wide array of disease-modifying anti-rheumatic drugs and biologicals. After an autoHSCT procedure, the disease became inactive for a short period of time, until the patient experienced a relapse after 31 days, accompanied by repopulation of effector/memory CD8+ T cells. Because of persistent inflammation and serious steroid toxicity, including severe osteoporosis, growth restriction, and excessive weight gain, the patient was offered an alloHSCT. She experienced transient antibody-mediated immune events post-alloHSCT, which subsided after rituximab. She ultimately developed a balanced immune reconstitution and is currently still in long-term disease remission, 8 years after alloHSCT.ConclusionThis case adds to the few existing reports on autoHSCT in relapsing polychondritis and gives new insights in its pathogenesis, with a possible role for CD8+ T cells. Moreover, it is the first report of successful alloHSCT as a treatment for children with this severe autoimmune disease.

Published
2022

98. Additional file 1 of Modified Delphi procedure-based expert consensus on endpoints for an international disease registry for Metachromatic Leukodystrophy: The European Metachromatic Leukodystrophy initiative (MLDi)

Author

Schoenmakers, Daphne H., Beerepoot, Shanice, van den Berg, Sibren, Adang, Laura, Bley, Annette, Boelens, Jaap-Jan, Fumagalli, Francesca, Goettsch, Wim G., Gr��nborg, Sabine, Groeschel, Samuel, van Hasselt, Peter M., Hollak, Carla E. M., Lindemans, Caroline, Mochel, Fanny, Mol, Peter G. M., Sevin, Caroline, Zerem, Ayelet, Sch��ls, Ludger, and Wolf, Nicole I.

Abstract

Additional file 1: Search strings. Additional information about literature review

Published
2022
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99. Additional file 2 of Modified Delphi procedure-based expert consensus on endpoints for an international disease registry for Metachromatic Leukodystrophy: The European Metachromatic Leukodystrophy initiative (MLDi)

Author

Schoenmakers, Daphne H., Beerepoot, Shanice, van den Berg, Sibren, Adang, Laura, Bley, Annette, Boelens, Jaap-Jan, Fumagalli, Francesca, Goettsch, Wim G., Gr��nborg, Sabine, Groeschel, Samuel, van Hasselt, Peter M., Hollak, Carla E. M., Lindemans, Caroline, Mochel, Fanny, Mol, Peter G. M., Sevin, Caroline, Zerem, Ayelet, Sch��ls, Ludger, and Wolf, Nicole I.

Abstract

Additional file 2: Flow chart. Additional information about literature review.

Published
2022
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100. Additional file 4 of Modified Delphi procedure-based expert consensus on endpoints for an international disease registry for Metachromatic Leukodystrophy: The European Metachromatic Leukodystrophy initiative (MLDi)

Author

Schoenmakers, Daphne H., Beerepoot, Shanice, van den Berg, Sibren, Adang, Laura, Bley, Annette, Boelens, Jaap-Jan, Fumagalli, Francesca, Goettsch, Wim G., Gr��nborg, Sabine, Groeschel, Samuel, van Hasselt, Peter M., Hollak, Carla E. M., Lindemans, Caroline, Mochel, Fanny, Mol, Peter G. M., Sevin, Caroline, Zerem, Ayelet, Sch��ls, Ludger, and Wolf, Nicole I.

Abstract

Additional file 4: Complete core and optional set of data elements. Complete lists of data elements.

Published
2022
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