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

Author

Vinci, L., Flotho, C., Noellke, P., Lebrecht, D., Masetti, R., de Haas, V., De Moerloose, B., Dworzak, M., Hasle, H., Gungor, T., Stary, J., Turkiewicz, D., Ussowicz, M., de Heredia, C. D., Buechner, J., Jahnukainen, K., Kallay, K., Bodova, I., Smith, O. P., Zecca, M., Bresters, D., Lang, P., Masmas, T. N., Meisel, R., Pichler, H., Erlacher, M., Gohring, G., Locatelli, Franco, Strahm, B., Niemeyer, C. M., Yoshimi, A., Locatelli F. (ORCID:0000-0002-7976-3654), Vinci, L., Flotho, C., Noellke, P., Lebrecht, D., Masetti, R., de Haas, V., De Moerloose, B., Dworzak, M., Hasle, H., Gungor, T., Stary, J., Turkiewicz, D., Ussowicz, M., de Heredia, C. D., Buechner, J., Jahnukainen, K., Kallay, K., Bodova, I., Smith, O. P., Zecca, M., Bresters, D., Lang, P., Masmas, T. N., Meisel, R., Pichler, H., Erlacher, M., Gohring, G., Locatelli, Franco, Strahm, B., Niemeyer, C. M., Yoshimi, A., and Locatelli F. (ORCID:0000-0002-7976-3654)

Abstract

NO ABSTRACT

Published
2023

5. The protective effects of hormonal suppression by a gonadotropin-releasing hormone agonist or an oral contraceptive on the decreased ovarian reserve in female rats exposed to isotretinoin.

Author

BAS, S., CETINKAYA, N., OZGU, E., KORKMAZ, E., OZ, M., ISIKALAN, M., CAYDERE, M., HUCUMENOGLU, S., GUNGOR, T., and BUYUKKAGNICI, U.

Abstract

OBJECTIVE: The objective of our study was to evaluate whether ovarian suppression by two different hormonal methods may spare the ovary the cytotoxic effects of isotretinoin in a rat model. MATERIALS AND METHODS: Four groups (n=8 Sprague-Dawley albino rats per group) were studied: control (Group I), 7.5 mg/kg/day isotretinoin (Group II), isotretinoin plus the combination of 0.030 mg ethinyl estradiol/0.15 mg levonorgestrel (combined oral contraceptive, COC), and isotretinoin plus 100 µg (microgram) leuprolide acetate (GnRHa) (Group III and IV, respectively). Four rats from each group were decapitated on the 30th day of treatment, and the remaining rats were decapitated on the 30th day of untreated follow-up. Serum anti-Mullerian hormone (AMH) concentrations, healthy and atretic follicle numbers, and apoptotic activity of follicles in oophorectomy specimens were compared between the groups. RESULTS: There were no significant differences in AMH levels among the study groups before, immediately after (first month), and one month after their last medication (second month) (p=0.08, 0.47, and 0.08, respectively). At the end of the first month, the control group had a higher median count of healthy primordial follicles compared to the study groups: 13.5 (8-22), 5.5 (3-11), 6 (2-13), and 1 (0-1) in control, isotretinoin, isotretinoin+ COC, and isotretinoin+GnRHa groups, respectively (p=0.02). However, there was no statistically significant difference in the number of healthy primordial follicles between the groups one month after the last medication (p=0.33). The median atretic antral follicle counts in the first month were 2 (1-4), 3.5 (1-4), 0 (0-2), and 0 (0-0) in the control, isotretinoin, isotretinoin+COC, and isotretinoin+GnRHa groups, respectively (p=0.02). Otherwise, there were no significant differences in other types of follicles among the control and treated groups (p>0.05). There was also no statistical difference between the groups regarding immunostaining intensity for active caspase-3 evaluated in the first or second month of treatment (p=0.8 and 0.2, respectively). CONCLUSIONS: Our results show that GnRH agonists or COC have no protective effects on ovarian reserve when co-administered with isotretinoin in the rat model. [ABSTRACT FROM AUTHOR]

Published
2023

6. Supportive Care During Pediatric Hematopoietic Stem Cell Transplantation: Prevention of Infections. A Report From Workshops on Supportive Care of the Paediatric Diseases Working Party (PDWP) of the European Society for Blood and Marrow Transplantation (EBMT)

Author

Ifversen, M, Meisel, R, Sedlacek, P, Kalwak, K, Sisinni, L, Hutt, D, Lehrnbecher, T, Balduzzi, A, Diesch, T, Jarisch, A, Gungor, T, Stein, J, Yaniv, I, Bonig, H, Kuhlen, M, Ansari, M, Nava, T, Dalle, J, Diaz-de-Heredia, C, Trigoso, E, Falkenberg, U, Hartmann, M, Deiana, M, Canesi, M, Broggi, C, Bertaina, A, Gibson, B, Krivan, G, Vettenranta, K, Matic, T, Buechner, J, Lawitschka, A, Peters, C, Yesilipek, A, Yalcin, K, Lucchini, G, Bakhtiar, S, Turkiewicz, D, Niinimaki, R, Wachowiak, J, Cesaro, S, Dalissier, A, Corbacioglu, S, Willasch, A, Bader, P, Ifversen M., Meisel R., Sedlacek P., Kalwak K., Sisinni L., Hutt D., Lehrnbecher T., Balduzzi A., Diesch T., Jarisch A., Gungor T., Stein J., Yaniv I., Bonig H., Kuhlen M., Ansari M., Nava T., Dalle J. -H., Diaz-de-Heredia C., Trigoso E., Falkenberg U., Hartmann M., Deiana M., Canesi M., Broggi C., Bertaina A., Gibson B., Krivan G., Vettenranta K., Matic T., Buechner J., Lawitschka A., Peters C., Yesilipek A., Yalcin K., Lucchini G., Bakhtiar S., Turkiewicz D., Niinimaki R., Wachowiak J., Cesaro S., Dalissier A., Corbacioglu S., Willasch A. M., Bader P., Ifversen, M, Meisel, R, Sedlacek, P, Kalwak, K, Sisinni, L, Hutt, D, Lehrnbecher, T, Balduzzi, A, Diesch, T, Jarisch, A, Gungor, T, Stein, J, Yaniv, I, Bonig, H, Kuhlen, M, Ansari, M, Nava, T, Dalle, J, Diaz-de-Heredia, C, Trigoso, E, Falkenberg, U, Hartmann, M, Deiana, M, Canesi, M, Broggi, C, Bertaina, A, Gibson, B, Krivan, G, Vettenranta, K, Matic, T, Buechner, J, Lawitschka, A, Peters, C, Yesilipek, A, Yalcin, K, Lucchini, G, Bakhtiar, S, Turkiewicz, D, Niinimaki, R, Wachowiak, J, Cesaro, S, Dalissier, A, Corbacioglu, S, Willasch, A, Bader, P, Ifversen M., Meisel R., Sedlacek P., Kalwak K., Sisinni L., Hutt D., Lehrnbecher T., Balduzzi A., Diesch T., Jarisch A., Gungor T., Stein J., Yaniv I., Bonig H., Kuhlen M., Ansari M., Nava T., Dalle J. -H., Diaz-de-Heredia C., Trigoso E., Falkenberg U., Hartmann M., Deiana M., Canesi M., Broggi C., Bertaina A., Gibson B., Krivan G., Vettenranta K., Matic T., Buechner J., Lawitschka A., Peters C., Yesilipek A., Yalcin K., Lucchini G., Bakhtiar S., Turkiewicz D., Niinimaki R., Wachowiak J., Cesaro S., Dalissier A., Corbacioglu S., Willasch A. M., and Bader P.

Abstract

Specific protocols define eligibility, conditioning, donor selection, graft composition and prophylaxis of graft vs. host disease for children and young adults undergoing hematopoietic stem cell transplant (HSCT). However, international protocols rarely, if ever, detail supportive care, including pharmaceutical infection prophylaxis, physical protection with face masks and cohort isolation or food restrictions. Supportive care suffers from a lack of scientific evidence and implementation of practices in the transplant centers brings extensive restrictions to the child's and family's daily life after HSCT. Therefore, the Board of the Pediatric Diseases Working Party (PDWP) of the European Society for Blood and Marrow Transplantation (EBMT) held a series of dedicated workshops since 2017 with the aim of initiating the production of a set of minimal recommendations. The present paper describes the consensus reached within the field of infection prophylaxis.

Published
2021

7. The impact of donor type on the outcome of pediatric patients with very high risk acute lymphoblastic leukemia. A study of the ALL SCT 2003 BFM-SG and 2007-BFM-International SG

Author

Dalle, J, Balduzzi, A, Bader, P, Pieczonka, A, Yaniv, I, Lankester, A, Bierings, M, Yesilipek, A, Sedlacek, P, Ifversen, M, Svec, P, Toporski, J, Gungor, T, Wachowiak, J, Glogova, E, Poetschger, U, Peters, C, Dalle J. -H., Balduzzi A., Bader P., Pieczonka A., Yaniv I., Lankester A., Bierings M., Yesilipek A., Sedlacek P., Ifversen M., Svec P., Toporski J., Gungor T., Wachowiak J., Glogova E., Poetschger U., Peters C., Dalle, J, Balduzzi, A, Bader, P, Pieczonka, A, Yaniv, I, Lankester, A, Bierings, M, Yesilipek, A, Sedlacek, P, Ifversen, M, Svec, P, Toporski, J, Gungor, T, Wachowiak, J, Glogova, E, Poetschger, U, Peters, C, Dalle J. -H., Balduzzi A., Bader P., Pieczonka A., Yaniv I., Lankester A., Bierings M., Yesilipek A., Sedlacek P., Ifversen M., Svec P., Toporski J., Gungor T., Wachowiak J., Glogova E., Poetschger U., and Peters C.

Abstract

Allogeneic HSCT represents the only potentially curative treatment for very high risk (VHR) ALL. Two consecutive international prospective studies, ALL-SCT-(I)BFM 2003 and 2007 were conducted in 1150 pediatric patients. 569 presented with VHR disease leading to any kind of HSCT. All patients >2 year old were transplanted after TBI-based MAC. The median follow-up was 5 years. 463 patients were transplanted from matched donor (MD) and 106 from mismatched donor (MMD). 214 were in CR1. Stem cell source was unmanipulated BM for 330 patients, unmanipulated PBSC for 135, ex vivo T-cell depleted PBSC for 62 and cord-blood for 26. There were more advanced disease, more ex vivo T-cell depletion, and more chemotherapy based conditioning regimen for patients transplanted from MMD as compared to those transplanted from MSD or MD. Median follow up (reversed Kaplan Meier estimator) was 4.99 years, median follow up of survivals was 4.88, range (0.01–11.72) years. The 4-year CI of extensive cGvHD was 13 ± 2% and 17 ± 4% (p = NS) for the patients transplanted from MD and MMD, respectively. 4-year EFS was statistically better for patients transplanted from MD (60 ± 2% vs. 42 ± 5%, p < 0.001) for the whole cohort. This difference does not exist if considering separately patients treated in the most recent study. There was no difference in 4-year CI of relapse. The 4-year NRM was lower for patients transplanted from MD (9 ± 1% vs. 23 ± 4%, p < 0.001). In multivariate analysis, donor-type appears as a negative risk-factor for OS, EFS, and NRM. This paper demonstrates the impact of donor type on overall results of allogeneic stem cell transplantation for very-high risk pediatric acute lymphoblastic leukemia with worse results when using MMD stem cell source.

Published
2021

8. Correction: Myeloablative conditioning for allo-HSCT in pediatric ALL: FTBI or chemotherapy?—A multicenter EBMT-PDWP study (Bone Marrow Transplantation, (2020), 55, 8, (1540-1551), 10.1038/s41409-020-0854-0)

Author

Willasch A. M., Willasch, A, Peters, C, Sedlacek, P, Dalle, J, Kitra-Roussou, V, Yesilipek, A, Wachowiak, J, Lankester, A, Prete, A, Hamidieh, A, Ifversen, M, Buechner, J, Krivan, G, Hamladji, R, Diaz-de-Heredia, C, Skorobogatova, E, Michel, G, Locatelli, F, Bertaina, A, Veys, P, Dupont, S, Or, R, Gungor, T, Aleinikova, O, Sufliarska, S, Sundin, M, Rascon, J, Kaare, A, Nemet, D, Fagioli, F, Klingebiel, T, Styczynski, J, Bierings, M, Nagy, K, Abecasis, M, Afanasyev, B, Ansari, M, Vettenranta, K, Alseraihy, A, Chybicka, A, Robinson, S, Bertrand, Y, Kupesiz, A, Ghavamzadeh, A, Campos, A, Pichler, H, Dalissier, A, Labopin, M, Corbacioglu, S, Balduzzi, A, Galimard, J, Bader, P, Willasch A. M., Peters C., Sedlacek P., Dalle J. -H., Kitra-Roussou V., Yesilipek A., Wachowiak J., Lankester A., Prete A., Hamidieh A. A., Ifversen M., Buechner J., Krivan G., Hamladji R. -M., Diaz-de-Heredia C., Skorobogatova E., Michel G., Locatelli F., Bertaina A., Veys P., Dupont S., Or R., Gungor T., Aleinikova O., Sufliarska S., Sundin M., Rascon J., Kaare A., Nemet D., Fagioli F., Klingebiel T. E., Styczynski J., Bierings M., Nagy K., Abecasis M., Afanasyev B., Ansari M., Vettenranta K., Alseraihy A., Chybicka A., Robinson S., Bertrand Y., Kupesiz A., Ghavamzadeh A., Campos A., Pichler H., Dalissier A., Labopin M., Corbacioglu S., Balduzzi A., Galimard J. -E., Bader P., Willasch A. M., Willasch, A, Peters, C, Sedlacek, P, Dalle, J, Kitra-Roussou, V, Yesilipek, A, Wachowiak, J, Lankester, A, Prete, A, Hamidieh, A, Ifversen, M, Buechner, J, Krivan, G, Hamladji, R, Diaz-de-Heredia, C, Skorobogatova, E, Michel, G, Locatelli, F, Bertaina, A, Veys, P, Dupont, S, Or, R, Gungor, T, Aleinikova, O, Sufliarska, S, Sundin, M, Rascon, J, Kaare, A, Nemet, D, Fagioli, F, Klingebiel, T, Styczynski, J, Bierings, M, Nagy, K, Abecasis, M, Afanasyev, B, Ansari, M, Vettenranta, K, Alseraihy, A, Chybicka, A, Robinson, S, Bertrand, Y, Kupesiz, A, Ghavamzadeh, A, Campos, A, Pichler, H, Dalissier, A, Labopin, M, Corbacioglu, S, Balduzzi, A, Galimard, J, Bader, P, Willasch A. M., Peters C., Sedlacek P., Dalle J. -H., Kitra-Roussou V., Yesilipek A., Wachowiak J., Lankester A., Prete A., Hamidieh A. A., Ifversen M., Buechner J., Krivan G., Hamladji R. -M., Diaz-de-Heredia C., Skorobogatova E., Michel G., Locatelli F., Bertaina A., Veys P., Dupont S., Or R., Gungor T., Aleinikova O., Sufliarska S., Sundin M., Rascon J., Kaare A., Nemet D., Fagioli F., Klingebiel T. E., Styczynski J., Bierings M., Nagy K., Abecasis M., Afanasyev B., Ansari M., Vettenranta K., Alseraihy A., Chybicka A., Robinson S., Bertrand Y., Kupesiz A., Ghavamzadeh A., Campos A., Pichler H., Dalissier A., Labopin M., Corbacioglu S., Balduzzi A., Galimard J. -E., and Bader P.

Abstract

The article “Myeloablative conditioning for allo-HSCT in pediatric ALL: FTBI or chemotherapy?—A multicenter EBMT-PDWP study,” written by Andre Manfred Willasch, Christina Peters, Petr Sedlácek, Jean-Hugues Dalle, Vassiliki Kitra-Roussou, Akif Yesilipek, Jacek Wachowiak, Arjan Lankester, Arcangelo Prete, Amir Ali Hamidieh, Marianne Ifversen, Jochen Buechner, Gergely Kriván, Rose-Marie Hamladji, Cristina Diaz-de-Heredia, Elena Skorobogatova, Gérard Michel, Franco Locatelli, Alice Bertaina, Paul Veys, Sophie Dupont, Reuven Or, Tayfun Güngör, Olga Aleinikova, Sabina Sufliarska, Mikael Sundin, Jelena Rascon, Ain Kaare, Damir Nemet, Franca Fagioli, Thomas Erich Klingebiel, Jan Styczynski, Marc Bierings, Kálmán Nagy, Manuel Abecasis, Boris Afanasyev, Marc Ansari, Kim Vettenranta, Amal Alseraihy, Alicja Chybicka, Stephen Robinson, Yves Bertrand, Alphan Kupesiz, Ardeshir Ghavamzadeh, Antonio Campos, Herbert Pichler, Arnaud Dalissier, Myriam Labopin, Selim Corbacioglu, Adriana Balduzzi, Jacques-Emmanuel Galimard, Peter Bader, on behalf of the EBMT Paediatric Diseases Working Party, was originally published online first without Open Access. After publication in volume 55, issue 8, page 1540–1551, the author decided to opt for Open Choice and to make the article an Open Access publication. Therefore, the copyright of the article has been changed to © The Author(s) 2020 and the article is forthwith distributed under the terms of the Creative Commons Attribution 4.0 InternationalS License, which permits use, sharing, adaptation, distribution, and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The images or other third-party material in this article are included in the article’s Creative Commons license, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Comm

Published
2021

9. Hematopoietic Cell Transplantation Cures Adenosine Deaminase 2 Deficiency: Report on 30 Patients (vol 41, pg 1633, 2021)

Author

Hashem, H., Bucciol, G., Ozen, S., Unal, S., Bozkaya, I.O., Akarsu, N., Taskinen, M., Koskenvuo, M., Saarela, J., Dimitrova, D., Hickstein, D.D., Hsu, A.P., Holland, S.M., Krance, R., Sasa, G., Kumar, A.R., Muller, I., Sousa, M.A. de, Delafontaine, S., Moens, L., Babor, F., Barzaghi, F., Cicalese, M.P., Bredius, R., Montfrans, J. van, Baretta, V., Cesaro, S., Stepensky, P., Benedicte, N., Moshous, D., Guenno, G. le, Boutboul, D., Dalal, J., Brooks, J.P., Dokmeci, E., Dara, J., Lucas, C.L., Hambleton, S., Wilson, K., Jolles, S., Koc, Y., Gungor, T., Schnider, C., Candotti, F., Steinmann, S., Schulz, A., Chambers, C., Hershfield, M., Ombrello, A., Kanakry, J.A., and Meyts, I.

Published
2022
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10. Hematopoietic stem cell transplantation for Wiskott-Aldrich syndrome

Author

Albert, M.H., Slatter, M.A., Gennery, A.R., Gungor, T., Bakunina, K., Markovitch, B., Hazelaar, S., Sirait, T., Courteille, V., Aiuti, A., Aleinikova, O.V., Balashov, D., Bernardo, M.E., Bodova, I., Bruno, B., Cavazzana, M., Chiesa, R., Fischer, A., Hauck, F., Ifversen, M., Kalwak, K., Klein, C., Kulagin, A., Kupesiz, A., Kuskonmaz, B., Lindemans, C.A., Locatelli, F., Lum, S.H., Maschan, A., Meisel, R., Moshous, D., Porta, F., Sauer, M.G., Sedlacek, P., Schulz, A., Suarez, F., Vallee, T.C., Winiarski, J.H., Zecca, M., Neven, B., Veys, P., Lankester, A.C., EBMT, European Soc Immunodeficiencies ES, and Stem CELL Transplant Primary Immun

Subjects
Transplantation Conditioning, Immunology, Hematopoietic Stem Cell Transplantation, Graft vs Host Disease, Cell Biology, Hematology, Biochemistry, Tissue Donors, Wiskott-Aldrich Syndrome, Treatment Outcome, surgical procedures, operative, Settore MED/38 - PEDIATRIA GENERALE E SPECIALISTICA, immune system diseases, Child, Preschool, Humans, Busulfan, Retrospective Studies
Abstract

Allogeneic hematopoietic stem cell transplantation (HSCT) is a potentially curative treatment for patients affected by Wiskott-Aldrich syndrome (WAS). Reported HSCT outcomes have improved over time with respect to overall survival, but some studies have identified older age and HSCT from alternative donors as risk factors predicting poorer outcome. We analyzed 197 patients undergoing transplant at European Society for Blood and Marrow Transplantation centers between 2006 and 2017 who received conditioning as recommended by the Inborn Errors Working Party (IEWP): either busulfan (n = 103) or treosulfan (n = 94) combined with fludarabine ± thiotepa. After a median follow-up post-HSCT of 44.9 months, 176 patients were alive, resulting in a 3-year overall survival of 88.7% and chronic graft-versus-host disease (GVHD)-free survival (events include death, graft failure, and severe chronic GVHD) of 81.7%. Overall survival and chronic GVHD-free survival were not significantly affected by conditioning regimen (busulfan- vs treosulfan-based), donor type (matched sibling donor/matched family donor vs matched unrelated donor/mismatched unrelated donor vs mismatched family donor), or period of HSCT (2006-2013 vs 2014-2017). Patients aged

Published
2022

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

Author

Albert, M. H., Slatter, M. A., Gennery, A. R., Gungor, T., Bakunina, K., Markovitch, B., Hazelaar, S., Sirait, T., Courteille, V., Aiuti, A., Aleinikova, O. V., Balashov, D., Bernardo, M. E., Bodova, I., Bruno, B., Cavazzana, M., Chiesa, R., Fischer, A., Hauck, F., Ifversen, M., Kalwak, K., Klein, C., Kulagin, A., Kupesiz, A., Kuskonmaz, B., Lindemans, C. A., Locatelli, Franco, Lum, S. H., Maschan, A., Meisel, R., Moshous, D., Porta, F., Sauer, M. G., Sedlacek, P., Schulz, A., Suarez, F., Vallee, T. C., Winiarski, J. H., Zecca, M., Neven, B., Veys, P., Lankester, A. C., Locatelli F. (ORCID:0000-0002-7976-3654), Albert, M. H., Slatter, M. A., Gennery, A. R., Gungor, T., Bakunina, K., Markovitch, B., Hazelaar, S., Sirait, T., Courteille, V., Aiuti, A., Aleinikova, O. V., Balashov, D., Bernardo, M. E., Bodova, I., Bruno, B., Cavazzana, M., Chiesa, R., Fischer, A., Hauck, F., Ifversen, M., Kalwak, K., Klein, C., Kulagin, A., Kupesiz, A., Kuskonmaz, B., Lindemans, C. A., Locatelli, Franco, Lum, S. H., Maschan, A., Meisel, R., Moshous, D., Porta, F., Sauer, M. G., Sedlacek, P., Schulz, A., Suarez, F., Vallee, T. C., Winiarski, J. H., Zecca, M., Neven, B., Veys, P., Lankester, A. C., and Locatelli F. (ORCID:0000-0002-7976-3654)

Abstract

Allogeneic hematopoietic stem cell transplantation (HSCT) is a potentially curative treatment for patients affected by Wiskott-Aldrich syndrome (WAS). Reported HSCT outcomes have improved over time with respect to overall survival, but some studies have identified older age and HSCT from alternative donors as risk factors predicting poorer outcome. We analyzed 197 patients undergoing transplant at European Society for Blood and Marrow Transplantation centers between 2006 and 2017 who received conditioning as recommended by the Inborn Errors Working Party (IEWP): either busulfan (n = 103) or treosulfan (n = 94) combined with fludarabine ± thiotepa. After a median follow-up post-HSCT of 44.9 months, 176 patients were alive, resulting in a 3-year overall survival of 88.7% and chronic graft-versus-host disease (GVHD)-free survival (events include death, graft failure, and severe chronic GVHD) of 81.7%. Overall survival and chronic GVHD-free survival were not significantly affected by conditioning regimen (busulfan- vs treosulfan-based), donor type (matched sibling donor/matched family donor vs matched unrelated donor/mismatched unrelated donor vs mismatched family donor), or period of HSCT (2006-2013 vs 2014-2017). Patients aged <5 years at HSCT had a significantly better overall survival. The overall cumulative incidences of grade III to IV acute GVHD and extensive/moderate/severe chronic GVHD were 6.6% and 2.1%, respectively. Patients receiving treosulfan-based conditioning had a higher incidence of graft failure and mixed donor chimerism and more frequently underwent secondary procedures (second HSCT, unconditioned stem cell boost, donor lymphocyte infusion, or splenectomy). In summary, HSCT for WAS with conditioning regimens currently recommended by IEWP results in excellent survival and low rates of GVHD, regardless of donor or stem cell source, but age ≥5 years remains a risk factor for overall survival.

Published
2022

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

Author

Albert, M. H., Sirait, T., Eikema, D. -J., Bakunina, K., Wehr, C., Suarez, F., Fox, M. L., Mahlaoui, N., Gennery, A. R., Lankester, A. C., Beier, R., Bernardo, M. E., Bigley, V., Lindemans, C. A., Burns, S. O., Carpenter, B., Dybko, J., Gungor, T., Hauck, F., Lum, S. H., Balashov, D., Meisel, R., Moshous, D., Schulz, A., Speckmann, C., Slatter, M. A., Strahm, B., Uckan-Cetinkaya, D., Meyts, I., Vallee, T. C., Wynn, R., Neven, B., Morris, E. C., Aiuti, A., Maschan, A., Aljurf, M., Gedde-Dahl, T., Gurman, G., Bordon, V., Krivan, G., Locatelli, Franco, Porta, F., Valcarcel, D., Beguin, Y., Faraci, M., Kroger, N., Kulagin, A., Shaw, P. J., Veelken, J. H., Diaz de Heredia, C., Fagioli, F., Felber, M., Gruhn, B., Holter, W., Rossig, C., Sedlacek, P., Apperley, J., Ayas, M., Bodova, I., Choi, G., Cornelissen, J. J., Sirvent, A., Khan, A., Kupesiz, A., Lenhoff, S., Ozdogu, H., von der Weid, N., Rovira, M., Schots, R., Vinh, D. C., Locatelli F. (ORCID:0000-0002-7976-3654), Albert, M. H., Sirait, T., Eikema, D. -J., Bakunina, K., Wehr, C., Suarez, F., Fox, M. L., Mahlaoui, N., Gennery, A. R., Lankester, A. C., Beier, R., Bernardo, M. E., Bigley, V., Lindemans, C. A., Burns, S. O., Carpenter, B., Dybko, J., Gungor, T., Hauck, F., Lum, S. H., Balashov, D., Meisel, R., Moshous, D., Schulz, A., Speckmann, C., Slatter, M. A., Strahm, B., Uckan-Cetinkaya, D., Meyts, I., Vallee, T. C., Wynn, R., Neven, B., Morris, E. C., Aiuti, A., Maschan, A., Aljurf, M., Gedde-Dahl, T., Gurman, G., Bordon, V., Krivan, G., Locatelli, Franco, Porta, F., Valcarcel, D., Beguin, Y., Faraci, M., Kroger, N., Kulagin, A., Shaw, P. J., Veelken, J. H., Diaz de Heredia, C., Fagioli, F., Felber, M., Gruhn, B., Holter, W., Rossig, C., Sedlacek, P., Apperley, J., Ayas, M., Bodova, I., Choi, G., Cornelissen, J. J., Sirvent, A., Khan, A., Kupesiz, A., Lenhoff, S., Ozdogu, H., von der Weid, N., Rovira, M., Schots, R., Vinh, D. C., and Locatelli F. (ORCID:0000-0002-7976-3654)

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.

Published
2022

13. 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 AJ, Beier R, Bernardo ME, Bigley V, Lindemans CA, OBurns S, Carpenter B, Dybko J, Gungor T, Hauck F, Lum SH, Balashov D, Meisel R, Moshous D, Schulz A, Speckmann C, Slatter MA, Strahm B, Uckan-Cetinkaya D, Meyts I, Vallee TC, Wynn R, Neven B, Morris EC, EBMT IEWP, ESID

Published
2022
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14. Correction: Supportive care during pediatric hematopoietic stem cell transplantation: beyond infectious diseases. A report from workshops on supportive care of the Pediatric Diseases Working Party (PDWP) of the European Society for Blood and Marrow Transplantation (EBMT) (Bone Marrow Transplantation, (2020), 55, 6, (1126-1136), 10.1038/s41409-020-0818-4)

Author

Nava T., Nava, T, Ansari, M, Dalle, J, de Heredia, C, Gungor, T, Trigoso, E, Falkenberg, U, Bertaina, A, Gibson, B, Jarisch, A, Balduzzi, A, Boenig, H, Krivan, G, Vettenranta, K, Matic, T, Buechner, J, Kalwak, K, Lawitschka, A, Yesilipek, A, Lucchini, G, Peters, C, Turkiewicz, D, Niinimaki, R, Diesch, T, Lehrnbecher, T, Sedlacek, P, Hutt, D, Dalissier, A, Wachowiak, J, Yaniv, I, Stein, J, Yalcin, K, Sisinni, L, Deiana, M, Ifversen, M, Kuhlen, M, Meisel, R, Bakhtiar, S, Cesaro, S, Willasch, A, Corbacioglu, S, Bader, P, Nava T., Ansari M., Dalle J. -H., de Heredia C. D., Gungor T., Trigoso E., Falkenberg U., Bertaina A., Gibson B., Jarisch A., Balduzzi A., Boenig H., Krivan G., Vettenranta K., Matic T., Buechner J., Kalwak K., Lawitschka A., Yesilipek A., Lucchini G., Peters C., Turkiewicz D., Niinimaki R., Diesch T., Lehrnbecher T., Sedlacek P., Hutt D., Dalissier A., Wachowiak J., Yaniv I., Stein J., Yalcin K., Sisinni L., Deiana M., Ifversen M., Kuhlen M., Meisel R., Bakhtiar S., Cesaro S., Willasch A., Corbacioglu S., Bader P., Nava T., Nava, T, Ansari, M, Dalle, J, de Heredia, C, Gungor, T, Trigoso, E, Falkenberg, U, Bertaina, A, Gibson, B, Jarisch, A, Balduzzi, A, Boenig, H, Krivan, G, Vettenranta, K, Matic, T, Buechner, J, Kalwak, K, Lawitschka, A, Yesilipek, A, Lucchini, G, Peters, C, Turkiewicz, D, Niinimaki, R, Diesch, T, Lehrnbecher, T, Sedlacek, P, Hutt, D, Dalissier, A, Wachowiak, J, Yaniv, I, Stein, J, Yalcin, K, Sisinni, L, Deiana, M, Ifversen, M, Kuhlen, M, Meisel, R, Bakhtiar, S, Cesaro, S, Willasch, A, Corbacioglu, S, Bader, P, Nava T., Ansari M., Dalle J. -H., de Heredia C. D., Gungor T., Trigoso E., Falkenberg U., Bertaina A., Gibson B., Jarisch A., Balduzzi A., Boenig H., Krivan G., Vettenranta K., Matic T., Buechner J., Kalwak K., Lawitschka A., Yesilipek A., Lucchini G., Peters C., Turkiewicz D., Niinimaki R., Diesch T., Lehrnbecher T., Sedlacek P., Hutt D., Dalissier A., Wachowiak J., Yaniv I., Stein J., Yalcin K., Sisinni L., Deiana M., Ifversen M., Kuhlen M., Meisel R., Bakhtiar S., Cesaro S., Willasch A., Corbacioglu S., and Bader P.

Abstract

An amendment to this paper has been published and can be accessed via a link at the top of the paper.

Published
2020

15. Correction: Myeloablative conditioning for allo-HSCT in pediatric ALL: FTBI or chemotherapy?—A multicenter EBMT-PDWP study (Bone Marrow Transplantation, (2020), 55, 8, (1540-1551), 10.1038/s41409-020-0854-0)

Author

Willasch, A. M., Peters, C., Sedlacek, P., Dalle, J. -H., Kitra-Roussou, V., Yesilipek, A., Wachowiak, J., Lankester, A., Prete, A., Hamidieh, A. A., Ifversen, M., Buechner, J., Krivan, G., Hamladji, R. -M., Diaz-de-Heredia, C., Skorobogatova, E., Michel, G., Locatelli, F., Bertaina, A., Veys, P., Dupont, S., Or, R., Gungor, T., Aleinikova, O., Sufliarska, S., Sundin, M., Rascon, J., Kaare, A., Nemet, D., Fagioli, F., Klingebiel, T. E., Styczynski, J., Bierings, M., Nagy, K., Abecasis, M., Afanasyev, B., Ansari, M., Vettenranta, K., Alseraihy, A., Chybicka, A., Robinson, S., Bertrand, Y., Kupesiz, A., Ghavamzadeh, A., Campos, A., Pichler, H., Dalissier, A., Labopin, M., Corbacioglu, S., Balduzzi, A., Galimard, J. -E., and Bader, P.

Subjects
Settore MED/38 - PEDIATRIA GENERALE E SPECIALISTICA, conditioning regimens
Published
2021

16. Targeted busulfan-based reduced-intensity conditioning and HLA-matched HSCT cure hemophagocytic lymphohistiocytosis

Author

Felber, M., Steward, C.G., Kentouche, K., Fasth, A., Wynn, R.F., Zeilhofer, U., Haunerdinger, V., Volkmer, B., Prader, S., Gruhn, B., Ehl, S., Lehmberg, K., Muller, D., Gennery, A.R., Albert, M.H., Hauck, F., Rao, K., Veys, P., Hassan, M., Lankester, A.C., Schmid, J.P., Hauri-Hohl, M.M., Gungor, T., Inborn Errors Working Party IEWP E, European Soc Immunodeficiencies ES, and University of Zurich

Subjects
medicine.medical_specialty, Hemophagocytic lymphohistiocytosis, business.industry, medicine.medical_treatment, 2720 Hematology, 610 Medicine & health, Hematology, Hematopoietic stem cell transplantation, Defibrotide, medicine.disease, Gastroenterology, Fludarabine, medicine.anatomical_structure, 10036 Medical Clinic, Cord blood, Internal medicine, 540 Chemistry, Medicine, Alemtuzumab, Bone marrow, business, Busulfan, 10038 Institute of Clinical Chemistry, medicine.drug
Abstract

Reduced-intensity/reduced-toxicity conditioning and allogeneic T-cell replete hematopoietic stem cell transplantation are curative in patients with hemophagocytic lymphohistiocytosis (HLH). Unstable donor chimerism (DC) and relapses are clinical challenges . We examined the effect of a reduced-intensity conditioning regimen based on targeted busulfan to enhance myeloid DC in HLH. The European Society for Bone and Marrow Transplantation–approved reduced-intensity conditioning protocol comprised targeted submyeloablative IV busulfan, IV fludarabine, and serotherapy comprising IV alemtuzumab (0.5-0.8 mg/kg) for unrelated-donor and IV rabbit anti–T-cell globulin for related-donor transplants. We assessed toxicity, engraftment, graft-versus-host disease (GHVD), DC in blood cell subtypes, and overall survival/event-free survival. Twenty-five patients from 7 centers were treated (median age, 0.68 year). The median total dose and cumulative area under the curve of busulfan was 13.1 mg/kg (6.4-26.4) and 63.1 mg/L × h (48-77), respectively. Bone marrow, peripheral blood stem cell, or cord blood transplants from HLA-matched related (n = 7) or unrelated (n = 18) donors were administered. Donor cells engrafted in all patients (median: neutrophils d+20/platelets d+28). At last follow-up (median, 36 months; range, 8-111 months), the median DC of CD15+ neutrophils, CD3+ T cells, and CD16+56+ natural killer cells was 99.5% (10-100), 97% (30-100), and 97.5% (30-100), respectively. Eight patients (32%) developed sinusoidal obstruction syndrome, resolving after defibrotide treatment. The 3-year overall survival and event-free survival rates were both 100%. None of the patients developed acute grade III to IV GHVD. Limited chronic GVHD was encountered in 4%. This regimen achieves excellent results with stable DC in patients with HLH.

Published
2020

17. Total body irradiation or chemotherapy conditioning in childhood all: A multinational, randomized, noninferiority phase III study

Author

Peters, C., Dalle, J. -H., Locatelli, Franco, Poetschger, U., Sedlacek, P., Buechner, J., Shaw, P. J., Staciuk, R., Ifversen, M., Pichler, H., Vettenranta, K., Svec, P., Aleinikova, O., Stein, J., Gungor, T., Toporski, J., Truong, T. H., Diaz-De-Heredia, C., Bierings, M., Ariffin, H., Essa, M., Burkhardt, B., Schultz, K., Meisel, R., Lankester, A., Ansari, M., Schrappe, M., von Stackelberg, A., Balduzzi, A., Corbacioglu, S., Bader, P., Locatelli F. (ORCID:0000-0002-7976-3654), Peters, C., Dalle, J. -H., Locatelli, Franco, Poetschger, U., Sedlacek, P., Buechner, J., Shaw, P. J., Staciuk, R., Ifversen, M., Pichler, H., Vettenranta, K., Svec, P., Aleinikova, O., Stein, J., Gungor, T., Toporski, J., Truong, T. H., Diaz-De-Heredia, C., Bierings, M., Ariffin, H., Essa, M., Burkhardt, B., Schultz, K., Meisel, R., Lankester, A., Ansari, M., Schrappe, M., von Stackelberg, A., Balduzzi, A., Corbacioglu, S., Bader, P., and Locatelli F. (ORCID:0000-0002-7976-3654)

Abstract

PURPOSE Total body irradiation (TBI) before allogeneic hematopoietic stem cell transplantation (HSCT) in pediatric patients with acute lymphoblastic leukemia (ALL) is efficacious, but long-term side effects are concerning. We investigated whether preparative combination chemotherapy could replace TBI in such patients. PATIENTS AND METHODS FORUM is a randomized, controlled, open-label, international, multicenter, phase III, noninferiority study. Patients # 18 years at diagnosis, 4-21 years at HSCT, in complete remission pre-HSCT, and with an HLA-compatible related or unrelated donor were randomly assigned to myeloablative conditioning with fractionated 12 Gy TBI and etoposide versus fludarabine, thiotepa, and either busulfan or treosulfan. The noninferiority margin was 8%. With 1,000 patients randomly assigned in 5 years, 2-year minimum follow-up, and one-sided alpha of 5%, 80% power was calculated. A futility stopping rule would halt random assignment if chemoconditioning was significantly inferior to TBI (EudraCT: 2012-003032-22; ClinicalTrials.gov: NCT01949129). RESULTS Between April 2013 and December 2018, 543 patients were screened, 417 were randomly assigned, 212 received TBI, and 201 received chemoconditioning. The stopping rule was applied on March 31, 2019. The median follow-up was 2.1 years. In the intention-to-treat population, 2-year overall survival (OS) was significantly higher following TBI (0.91; 95% CI, 0.86 to 0.95; P,.0001) versus chemoconditioning (0.75; 95% CI, 0.67 to 0.81). Two-year cumulative incidence of relapse and treatment-related mortality were 0.12 (95% CI, 0.08 to 0.17; P,.0001) and 0.02 (95% CI,, 0.01 to 0.05; P 5.0269) following TBI and 0.33 (95% CI, 0.25 to 0.40) and 0.09 (95% CI, 0.05 to 0.14) following chemoconditioning, respectively. CONCLUSION Improved OS and lower relapse risk were observed following TBI plus etoposide compared with chemoconditioning. We therefore recommend TBI plus etoposide for patients. 4 years old with high

Published
2021

19. Gynecologic oncology

Author

Oskav-Özcelik G., Hindenburg H. J., Klare P., Könsgen D., Mustea A., Heinrich G., Camara O., Lichtenegger W., Sehouli J., Tutuncu L., Ergur A. R., Gul I., Ertekin A., Yergok Y. Z., Ornek T., Tulunay G., Fetiel A., Tan O., Kose F., Haberal A., Noftolin F., Yermez E., Ata N., Sanci M., Sekü I., Karanfil C., Ispahi C., Akar M. E., Simsek T., Tamburaci E., Erdogan G., Pestereli E., Ingec M., Kadanali S., Erdogan F., Naki M. M., Tekcan C., Ergüler Y. S., Uysal A., Songülalp S., Kanadikirik F., Gezginc K., Görkemli H., Celik C., Acar A., Colakoglu M. C., Capar M., Akyürek C., Özbay K., Yardim T., Kurt S., Pilanci B., Tinar S., Camuzcuoglu H., Dicle N., Hanhan M., Inal M., Öztekin D., Dicle N., Özsaran Z., Tinar S., Demir B., Demir S., Gul T., Erden A. C., Bozaci E. A., Atabekoglu C., Taskin S., Sertcelik A., Ünlü C., Ortac F., Taskin S., Cengiz B., Bozaci E. A., Seval M., Ortac F., Taskin S., Yarci A., Kahraman K., Özmen B., Güngör M., Taskin S., Kahraman K., Özmen B., Yarci A., Güngör M., Taskin S., Bozaci E. A., Yarci A., Atabekoglu C., Ortac F., Hascalik S., Celik O., Ustun Y., Erdem G., Karadag N., Alkan A., Karakas H. M., Usta U., Mizrak B., Güzin K., Tekcan C., Naki M. M., Kayatas Eser S., Zemheri E., Kanadikirik F., Kurt S., Öztekin D., Karalti O., Inal M., Özsaran Z., Dicle N., Gunaydin G., Onan A., Taskiran C., Turp A., Yilmaz E., Kurdoglu M., Bozdayi G., Himmetoglu O., Kurdoglu Z., Gultekin M., Dursun P., Celik N. Y., Boynukalin K., Yuce K., Ayhan A., Dursun P., Gultekin M., Celik N. Y., Velipasaoglu M., Yuce K., Ayhan A., Gultekin M., Dursun P., Bozdag G., Celik N. Y., Guler Z., Yuce K., Ayhan A., Erkan L., Soylu F., Oztekin O., Tatli O., Eraslan T., Uysal D., Yavuzcan A., Yensel U., Baloglu A., Yildiz A., Köksal A., Tatli Ö., Tatli O., Ivit H., Yetimalar H., Cukurova K., Simsek E., Haydardedeoglu B., Asian E., Bulgan Kilicdag E., Erkanli S., Ozyurtseven Tarim E., Güzin K., Kayatas S., Tekcan C., Zemheri E., Kayabasoglu F., Kanadikirik F., Özmen B., Taskin S., Ünlü C., Ortac F., Uysal D., Aydin C., Yavuzcan A., Baloglu A., Salman M. C., Otegen U., Ozyuncu O., Bozdag G., Ayhan A., Salman M. C., Guven S., Ozyuncu O., Bozdag G., Usubutun A., Ayhan A., Oztekin D., Kurt S., Tinar S., Mit T., Balsak D., Hanhan M., Kurt S., Oztekin D., Tinar S., Karalti O., Inal M., Seyhan S., Turan T., Altinbas S., Boran N., Ozgul N., Ozer S., Ozfuttu A., Kose M. F., Boran N., Hizli D., Turan T., Halici F., Koc S., Bulbul D., Köse M. F., Vural M., Barut A., Tanriverdi H. A., Tutuncu L., Ergur A. R., Sancaktar M., Ertekin A., Yergok Y. Z., Iyibozkurt A. C., Topuz S., Bengisu E., Ilhan R., Berkman S., Boran N., Sarici S., Kose M. F., Tulunay G., Koc S., Ocalan R., Cavusoglu D., Haberal A., Boran N., Karacay Ö., Öztürkoglu E., Turan T., Cil A., Otken O. F., Köse M. F., Turan T., Öztürk F., Karacay Ö., Boran N., Özgül N., Tulunay G., Erdogan Z., Köse M. F., Koc S., Otken H., Yüksel K., Özdal B., Güngör T., Taner D., Tarhan I., Reyhan H., Aydogdu T., Mollamahmutoglu L., Daylan B. H., Zergeroglu S., Tunc I., Kahraman N., Gungor T., Bilge U., Güngör T., Yüksel K., Reyhan H., Aytan H., Tug M. T., Aydogdu T., Özdal B., Güngör T., Tug M., Cavkaytar S., Güngör T., Aydogdu T., Özdal B., Reyhan H., Daylan H. B., Tune I., Koc Ö., Gözübüyük S., Seckin S., Özdemir T., Abali R., Bozkurt S., Arikan I., Arikan D., Sahin A., Erdener O., Tülay Ö., Ergin S., Midilli K., Tan O., Kose F., Fatial A., Ornek T., Luk J., Tulunay G., Haberal A., Neftolin F., Aslan E., Kilicdag E., Bolat F., Erkanli S., Bal N., Kuscu E., Simsek E., Ayar A., Güzel Y., Vural M., Yetimalar M. H., Zeteroglu U., Köksal A., Soylu F., and Zeteroglu S.

Published
2005
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20. Menopause

Author

Tutuncu L., Arslanhan N., Ergur A. R., Mungen E., Yergok Y. Z., An E. S., Uner M., Eryilmaz M., Akar M. E., Inan I., Kelekci S., Yilmaz B., Kart C., Günaydin A. S., Cakir A., Yilmaz B., Oguz S., Gunyeli I., Kelekci S., Kalyoncu S., Ertas I. E., Kahyaoglu S., Gokturk U., Mollamahmutoglu L., Elter K., Yildizhan B., Basgul A., Pekin T., Gokaslan H., Kavak Z. N., Karas C., Gol M., Guclu S., Dogan E., Saygili U., Onvural A., Gol M., Akan P., Dogan E., Karas C., Saygili U., Posaci C., Biri A., Yurtcu E., Ciftci B., Ergun M. A., Gursoy R., Biberoglu K., Ozcagli E., Sardas S., Erkan A., Ergun M. A., Yilmaz A., Tiras B., Guner H., Yalcin R., Bozkurt N., Gursoy R., Yildirim M., Karabacak O., Himmetoglu O., Gulbahar O., Gursoy R., Nas T., Eskioglu A., Kumru S., Yildiz M. F., Godekmerdan A., Gürates B., Kiran H., Kiran G., Ekerbicer H. C., Guven A. M., Ürünsak I. F., Güzel A. B., Demir S. C., Kadayifci O., Dursun P., Gultekin M., Bozdag G., Aksan G., Aksu T., Bayrak A., Esinler D., Oguz S., Tapisiz O. L., Aytan H., Gunyeli I., Erdem S., Tuncay G., Mollamahmutoglu L., Aksakal O., Aytan H., Cavkaytar S., Tapisiz O. L., Gungor T., Ozdal B., Akhan S. E., Hanli U., Kalayci R., Kaya M., Ahisali B., Turfanda A., Hassa H., Tanir H. M., Tekin B., Oge T., Kahraman S., Yildirim A., Ürünasak I. F., Güzel A. B., Demir S. C., Özbilen N., Kadayifci O., Dane C., Cetin A., Dane B., Kiray M., Erginbas M., Döventas Y., Karabeyoglu N., Dursun P., Gultekin M., Aksu T., Kalli E., Kiran H., Kiran G., Guven A. M., Karakus S., Sapmaz K., Cetin T. M., Canda M. T., Bagriyanik H. A., Kaplan P. B., Yilmaz O., Gucer F., Yuce M. A., Tugyan K., Yoldemir T., Davas I., Tanrikulu A., Yazgan A., and Varolan A.

Published
2005
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21. Urogynecology

Author

Petri E., Niemeyer R., Petri E., Niemeyer R., Sivaslioglu A. A., Haberal A., Dölen I., Dede H., Akkök E., Deveci S., Demir B., Aksakal O., Ugur M., Yilmaz B., Yesilyurt H., Mollamahmutoglu L., Sivaslioglu A. A., Elhan A., Sakul U., Dölen I., Tunc E., Ercan F., Haberal A., Aksakal O., Tuncay G., Aytan H., Tapisiz O. L., Bilge U., Mollamahmutoglu L., Aksakal O., Tuncay G., Bal S., Bilge U., Tapisiz O. L., Mollamahmutoglu L., Unlu S., Aksakal O., Tapisiz O. L., Tuncay G., Aytan H., Ugur M., Bilge U., Mollamahmutoglu L., Yasar L., Yazicioglu F., Efe C., Sönmez S., Zebitay A. G., Süt N., Sensoy Y., Cebi Z., Bayrak O., Cimentepe E., Gümüs I. I., Dede H., Sivaslioglu A., Dolen I., Dede F. S., Seckin L., Haberal A., Sivaslioglu A. A., Dolen I., Dede H., Dilbaz S., Demir B., Sümer C., Gelisen O., Unlubilgin E., Deveci S., Dede S., Seckin L., Haberal A., Dönmez M. D., Atis A., Aydin Y., Tandogan T., Ozpak D., Oruc O., Aksakal O. S., Doganay M., Aytan H., Gungor T., Bal S., Bilge U., Mollamahmutoglu L., Ozdegirmenci O., Dede F. S., Haberal A., Karslioglu Y., Karadeniz S., and Gunhan O.

Published
2005
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23. The Menderes Massif of western Turkey and the Cycladic Massif in the Aegean - do they really correlate?

Author

Ring, U., Gessner, K., Gungor, T., and Passchier, C.W.

Subjects
Mediterranean Sea -- Natural history, Geology, Structural -- Research, Paleogeography -- Mediterranean Sea, Earth sciences
Abstract

Based on lithostratigraphic comparisons the Menderes Massif has been correlated with the Cycladic Massif, thereby implying that the eastern Mediterranean consists of a narrow pre-Alpine basement belt which is laterally continuous over a long distance and which experienced a similar Alpine orogenic history. Our work indicates that the architecture, the age of basement and the pre-Alpine and Alpine tectonometamorphic history of both massifs differ fundamentally from each other. The Menderes Massif consists of remnants of the Cycladic Massif which overly an exotic unit, the Menderes nappes. Both massifs do not represent lateral continuations which has implications for palaeogeographic reconstructions. Keywords: Eastern Mediterranean, Menderes Massif, Cycladic Massif, tectonics, palaeogeography.

Published
1999

25. Long-term outcome after allogeneic hematopoietic stem cell transplantation for Shwachman–Diamond syndrome: a retrospective analysis and a review of the literature by the Severe Aplastic Anemia Working Party of the European Society for Blood and Marrow Transplantation (SAAWP-EBMT)

Author

Cesaro, S., Pillon, M., Sauer, M., Smiers, F., Faraci, M., de Heredia, C. D., Wynn, R., Greil, J., Locatelli, Franco, Veys, P., Uyttebroeck, A., Ljungman, P., Chevalier, P., Ansari, M., Badell, I., Gungor, T., Salim, R., Tischer, J., Tecchio, C., Russell, N., Chybicka, A., Styczynski, J., Krivan, G., Smith, O., Stein, J., Afanasyev, B., Pochon, C., Menconi, M. C., Bosman, P., Mauro, M., Tridello, G., de Latour, R. P., Dufour, C., Locatelli F. (ORCID:0000-0002-7976-3654), Cesaro, S., Pillon, M., Sauer, M., Smiers, F., Faraci, M., de Heredia, C. D., Wynn, R., Greil, J., Locatelli, Franco, Veys, P., Uyttebroeck, A., Ljungman, P., Chevalier, P., Ansari, M., Badell, I., Gungor, T., Salim, R., Tischer, J., Tecchio, C., Russell, N., Chybicka, A., Styczynski, J., Krivan, G., Smith, O., Stein, J., Afanasyev, B., Pochon, C., Menconi, M. C., Bosman, P., Mauro, M., Tridello, G., de Latour, R. P., Dufour, C., and Locatelli F. (ORCID:0000-0002-7976-3654)

Abstract

Allogeneic hematopoietic stem cell transplantation (HSCT) is a curative procedure in patients with Shwachman–Diamond syndrome (SDS) with bone marrow abnormalities. The results of 74 patients with SDS (6 acute myeloid leukemia, 7 myelodysplastic syndrome, and 61 bone marrow failure) treated with HSCT between 1988 and 2016 are reported. The donor source was: 24% sibling, 8% parent, and 68% unrelated donor. The stem cell source was: 70% bone marrow, 19% peripheral blood stem cells, and 11% cord blood. The conditioning regimen was myeloablative in 54% and reduced intensity in 46%. Neutrophil engraftment was achieved in 84% of patients after a median time of 17.5 days. Graft failure occurred in 15% of HSCTs. Grades I–IV acute and chronic GVHD were observed in 55% and 20% of patients, respectively. After a median follow-up of 7.3 years (95% CI 4.8–10.2), 28 patients died for progression/relapse (7) or toxicity (21). The 5-year overall survival and nonrelapse mortality were 63.3% (95% CI 50.8–73.4) and 19.8% (95% CI 10.8–30.8), respectively. In conclusion, this is the largest series so far reported and confirms that HSCT is a suitable option for patients with SDS. Further efforts are needed to lower transplant-related toxicity and reduce graft failure.

Published
2020

26. Correction: Long-term outcome after allogeneic hematopoietic stem cell transplantation for Shwachman–Diamond syndrome: a retrospective analysis and a review of the literature by the Severe Aplastic Anemia Working Party of the European Society for Blood and Marrow Transplantation (SAAWP-EBMT) (Bone Marrow Transplantation, (2020), 55, 9, (1796-1809), 10.1038/s41409-020-0863-z)

Author

Cesaro, S., Pillon, M., Sauer, M., Smiers, F., Faraci, M., de Heredia, C. D., Wynn, R., Greil, J., Locatelli, Franco, Veys, P., Uyttebroeck, A., Ljungman, P., Chevalier, P., Ansari, M., Badell, I., Gungor, T., Salim, R., Tischer, J., Tecchio, C., Russell, N., Chybicka, A., Styczynski, J., Krivan, G., Smith, O., Stein, J., Afanasyev, B., Pochon, C., Menconi, M. C., Bosman, P., Mauro, M., Tridello, G., de Latour, R. P., Dufour, C., Locatelli F. (ORCID:0000-0002-7976-3654), Cesaro, S., Pillon, M., Sauer, M., Smiers, F., Faraci, M., de Heredia, C. D., Wynn, R., Greil, J., Locatelli, Franco, Veys, P., Uyttebroeck, A., Ljungman, P., Chevalier, P., Ansari, M., Badell, I., Gungor, T., Salim, R., Tischer, J., Tecchio, C., Russell, N., Chybicka, A., Styczynski, J., Krivan, G., Smith, O., Stein, J., Afanasyev, B., Pochon, C., Menconi, M. C., Bosman, P., Mauro, M., Tridello, G., de Latour, R. P., Dufour, C., and Locatelli F. (ORCID:0000-0002-7976-3654)

Abstract

An amendment to this paper has been published and can be accessed via a link at the top of the paper.

Published
2020

27. Hematopoietic cell transplantation in chronic granulomatous disease: a study of 712 children and adults

Author

Chiesa, R., Wang, J., Blok, H. -J., Hazelaar, S., Neven, B., Moshous, D., Schulz, A., Hoenig, M., Hauck, F., Seraihy, A. A., Gozdzik, J., Ljungman, P., Lindemans, C. A., Fernandes, J. F., Kalwak, K., Strahm, B., Schanz, U., Sedlacek, P., Sykora, K. -W., Aksoylar, S., Locatelli, Franco, Stepensky, P., Wynn, R., Lum, S. H., Zecca, M., Porta, F., Taskinen, M., Gibson, B., Matthes, S., Karakukcu, M., Hauri-Hohl, M., Veys, P., Gennery, A. R., Lucchini, G., Felber, M., Albert, M. H., Balashov, D., Lankester, A., Gungor, T., Slatter, M. A., Locatelli F. (ORCID:0000-0002-7976-3654), Chiesa, R., Wang, J., Blok, H. -J., Hazelaar, S., Neven, B., Moshous, D., Schulz, A., Hoenig, M., Hauck, F., Seraihy, A. A., Gozdzik, J., Ljungman, P., Lindemans, C. A., Fernandes, J. F., Kalwak, K., Strahm, B., Schanz, U., Sedlacek, P., Sykora, K. -W., Aksoylar, S., Locatelli, Franco, Stepensky, P., Wynn, R., Lum, S. H., Zecca, M., Porta, F., Taskinen, M., Gibson, B., Matthes, S., Karakukcu, M., Hauri-Hohl, M., Veys, P., Gennery, A. R., Lucchini, G., Felber, M., Albert, M. H., Balashov, D., Lankester, A., Gungor, T., Slatter, M. A., and Locatelli F. (ORCID:0000-0002-7976-3654)

Abstract

Chronic granulomatous disease (CGD) is a primary immunodeficiency resulting in life-threatening infections and inflammatory complications. Allogeneic hematopoietic cell transplantation (allo-HCT) can cure the disease, but the indication to transplant remains controversial. We performed a retrospective multicenter study of 712 patients with CGD who underwent allo-HCT transplantation from March 1993 through December 2018. We studied 635 children (aged <18 years) and 77 adults. Median follow-up was 45 months. Median age at transplantation was 7 years (range, 0.1-48.6). Kaplan-Meier estimates of overall survival (OS) and event-free survival (EFS) at 3 years were 85.7% and 75.8%, respectively. In multivariate analysis, older age was associated with reduced survival and increased chronic graft-versus-host disease. Nevertheless, OS and EFS at 3 years for patients ‡18 years were 76% and 69%, respectively. Use of 1-antigen-mismatched donors was associated with reduced OS and EFS . No significant difference was found in OS, but a significantly reduced EFS was noted in the small group of patients who received a transplant from a donor with a >1 antigen mismatch. Choice of conditioning regimen did not influence OS or EFS. In summary, we report an excellent outcome after allo-HCT in CGD, with low incidence of graft failure and mortality in all ages. Older patients and recipients of 1-antigen-mismatched grafts had a less favorable outcome. Transplantation should be strongly considered at a younger age and particularly in the presence of a well-matched donor.

Published
2020

28. Myeloablative conditioning for allo-HSCT in pediatric ALL: FTBI or chemotherapy?—A multicenter EBMT-PDWP study

Author

Willasch, A. M., Peters, C., Sedlacek, P., Dalle, J. -H., Kitra-Roussou, V., Yesilipek, A., Wachowiak, J., Lankester, A., Prete, A., Hamidieh, A. A., Ifversen, M., Buechner, J., Krivan, G., Hamladji, R. -M., Diaz-de-Heredia, C., Skorobogatova, E., Michel, G., Locatelli, Franco, Bertaina, A., Veys, P., Dupont, S., Or, R., Gungor, T., Aleinikova, O., Sufliarska, S., Sundin, M., Rascon, J., Kaare, A., Nemet, D., Fagioli, F., Klingebiel, T. E., Styczynski, J., Bierings, M., Nagy, K., Abecasis, M., Afanasyev, B., Ansari, M., Vettenranta, K., Alseraihy, A., Chybicka, A., Robinson, S., Bertrand, Y., Kupesiz, A., Ghavamzadeh, A., Campos, A., Pichler, H., Dalissier, A., Labopin, M., Corbacioglu, S., Balduzzi, A., Galimard, J. -E., Bader, P., Locatelli F. (ORCID:0000-0002-7976-3654), Willasch, A. M., Peters, C., Sedlacek, P., Dalle, J. -H., Kitra-Roussou, V., Yesilipek, A., Wachowiak, J., Lankester, A., Prete, A., Hamidieh, A. A., Ifversen, M., Buechner, J., Krivan, G., Hamladji, R. -M., Diaz-de-Heredia, C., Skorobogatova, E., Michel, G., Locatelli, Franco, Bertaina, A., Veys, P., Dupont, S., Or, R., Gungor, T., Aleinikova, O., Sufliarska, S., Sundin, M., Rascon, J., Kaare, A., Nemet, D., Fagioli, F., Klingebiel, T. E., Styczynski, J., Bierings, M., Nagy, K., Abecasis, M., Afanasyev, B., Ansari, M., Vettenranta, K., Alseraihy, A., Chybicka, A., Robinson, S., Bertrand, Y., Kupesiz, A., Ghavamzadeh, A., Campos, A., Pichler, H., Dalissier, A., Labopin, M., Corbacioglu, S., Balduzzi, A., Galimard, J. -E., Bader, P., and Locatelli F. (ORCID:0000-0002-7976-3654)

Abstract

Although most children with acute lymphoblastic leukemia (ALL) receive fractionated total body irradiation (FTBI) as myeloablative conditioning (MAC) for allogeneic hematopoietic stem cell transplantation (allo-HSCT), it is an important matter of debate if chemotherapy can effectively replace FTBI. To compare outcomes after FTBI versus chemotherapy-based conditioning (CC), we performed a retrospective EBMT registry study. Children aged 2–18 years after MAC for first allo-HSCT of bone marrow (BM) or peripheral blood stem cells (PBSC) from matched-related (MRD) or unrelated donors (UD) in first (CR1) or second remission (CR2) between 2000 and 2012 were included. Propensity score weighting was used to control pretreatment imbalances of the observed variables. 3.054 patients were analyzed. CR1 (1.498): median follow-up (FU) after FTBI (1.285) and CC (213) was 6.8 and 6.1 years. Survivals were not significantly different. CR2 (1.556): median FU after FTBI (1.345) and CC (211) was 6.2 years. Outcomes after FTBI were superior as compared with CC with regard to overall survival (OS), leukemia-free survival (LFS), relapse incidence (RI), and nonrelapse mortality (NRM). However, we must emphasize the preliminary character of the results of this retrospective “real-world-practice” study. These findings will be prospectively assessed in the ALL SCTped 2012 FORUM trial.

Published
2020

30. Incidence of development of factor VIII and factor IX inhibitors in haemophiliacs

Author

Ehrenforth, S., Kreuz, W., Scharrer, I., Linde, R., Funk, M., Gungor, T., Krackhardt, B., and Kornhuber, B.

Subjects
Hemophilia -- Complications, Hemophilia B -- Complications, Hemophilia in children -- Research, Blood coagulation factor VIII -- Adverse and side effects, Prothrombin complex concentrate -- Adverse and side effects
Published
1992

33. Disseminated Fusarium oxysporum Infection in Hemophagocytic Lymphohistiocytosis

Author

Albisetti, M., Lauener, R. P., Gungor, T., Schar, G., Niggli, F. K., and Nadal, D.

Subjects
Fusarium -- Health aspects, Enteral feeding -- Risk factors, Tube feeding -- Risk factors, Infection -- Causes of, Infection -- Risk factors, Health
Abstract

Byline: M. Albisetti (1), R. P. Lauener (1), T. Gungor (1), G. Schar (2), F. K. Niggli (1), D. Nadal (1) Abstract: Abstract The portal of entry of disseminated Fusarium spp. infections is still not clearly defined. We report on a disseminated Fusarium oxysporum infection occurring during a long period of severe neutropenia in a child with hemophagocytic lymphohistiocytosis. A nasogastric feeding tube was the possible source of entry of the fungus. Author Affiliation: (1) Divisions of Immunology, Hematology, Infectious Diseases and Oncology, University Children's Hospital of Zurich, Steinwiesstrasse 75, CH-8032, Zurich, Switzerland (2) Institute of Medical Microbiology, University of Zurich, Switzerland Article History: Registration Date: 01/01/2004 Received Date: 09/10/2003 Accepted Date: 29/12/2003

Published
2004

35. Sickle cell disease: An international survey of results of HLA-identical sibling hematopoietic stem cell transplantation

Author

Gluckman, E., Cappelli, B., Bernaudin, F., Labopin, M., Volt, F., Carreras, J., Simoes, B. P., Ferster, A., Dupont, S., De La Fuente, J., Dalle, J. -H., Zecca, M., Walters, M. C., Krishnamurti, L., Bhatia, M., Leung, K., Yanik, G., Kurtzberg, J., Dhedin, N., Kuentz, M., Michel, G., Apperley, J., Lutz, P., Neven, B., Bertrand, Y., Vannier, J. P., Ayas, M., Cavazzana, M., Matthes-Martin, S., Rocha, V., Elayoubi, H., Kenzey, C., Bader, P., Locatelli, Franco, Ruggeri, A., Eapen, M., Bordon, V., Labarque, V., Pereira, M., Bittencourt, H., Petersen, H., Deconninck, E., Jubert, C., Perrin, J., Cahn, J. Y., Bruno, B., Bordigoni, P., Mechinaud, F., Vernant, J. P., Stephan, J. L., Suttorp, M., Strahm, B., Da Cunha, C. B., Garwer, B., Rothmayer, M., Wendelin, K., Graphakos, S., Tbakhi, A., Naeimi, N., Zuckerman, T., Sharon, P. B., Yaniv, I., Amos, T., Prete, A., Lo Nigro, L., Lanino, E., Faraci, M., Ciceri, F., Marktel, S., De Simone, G., Messina, C., Bartolomeo, P. D. I., Santarone, S., Vallisa, D., Bertaina, A., Arcese, W., Foa, R., Berger, M., Maximova, N., Wallet, S., Bazuaye, G. N., Maschan, A., De Heredia, C. D., Bieler, C. B., Pato, J. R., Heras, I., Trevor, R., Abayomi, K., Thomson, J., Fasth, A., Frodin, U., Ljugman, P., Ansari, M., Gungor, T., Unal, E., Pehlivan, M., Anak, S., Ozturk, G., Unal, A., Lawson, S., Keshani, J., Drake, A., Wynn, R., Williams, J., Jagsia, M., Leung, W., Abraham, A., Sahdey, I., Margolis, D., Eames, G., Horwitz, E., Cowan, M., Kapoor, N., Rowley, S., Megason, G., Rogers, Z., Bolanos-Meade, J., Hudspeth, M., Rosenthal, J., Olson, T., Kassow, K., Selby, G., Haines, H., Chaudhury, S., Gluckman, E., Cappelli, B., Bernaudin, F., Labopin, M., Volt, F., Carreras, J., Simoes, B. P., Ferster, A., Dupont, S., De La Fuente, J., Dalle, J. -H., Zecca, M., Walters, M. C., Krishnamurti, L., Bhatia, M., Leung, K., Yanik, G., Kurtzberg, J., Dhedin, N., Kuentz, M., Michel, G., Apperley, J., Lutz, P., Neven, B., Bertrand, Y., Vannier, J. P., Ayas, M., Cavazzana, M., Matthes-Martin, S., Rocha, V., Elayoubi, H., Kenzey, C., Bader, P., Locatelli, F., Ruggeri, A., Eapen, M., Bordon, V., Labarque, V., Pereira, M., Bittencourt, H., Petersen, H., Deconninck, E., Jubert, C., Perrin, J., Cahn, J. Y., Bruno, B., Bordigoni, P., Mechinaud, F., Vernant, J. P., Stephan, J. L., Suttorp, M., Strahm, B., Da Cunha, C. B., Garwer, B., Rothmayer, M., Wendelin, K., Graphakos, S., Tbakhi, A., Naeimi, N., Zuckerman, T., Sharon, P. B., Yaniv, I., Amos, T., Prete, A., Lo Nigro, L., Lanino, E., Faraci, M., Ciceri, F., Marktel, S., De Simone, G., Messina, C., Bartolomeo, P. D. I., Santarone, S., Vallisa, D., Bertaina, A., Arcese, W., Foa, R., Berger, M., Maximova, N., Wallet, S., Bazuaye, G. N., Maschan, A., De Heredia, C. D., Bieler, C. B., Pato, J. R., Heras, I., Trevor, R., Abayomi, K., Thomson, J., Fasth, A., Frodin, U., Ljugman, P., Ansari, M., Gungor, T., Unal, E., Pehlivan, M., Anak, S., Ozturk, G., Unal, A., Lawson, S., Keshani, J., Drake, A., Wynn, R., Williams, J., Jagsia, M., Leung, W., Abraham, A., Sahdey, I., Margolis, D., Eames, G., Horwitz, E., Cowan, M., Kapoor, N., Rowley, S., Megason, G., Rogers, Z., Bolanos-Meade, J., Hudspeth, M., Rosenthal, J., Olson, T., Kassow, K., Selby, G., Haines, H., Chaudhury, S., France Monacord, Centre Scientifique de Monaco (CSM), CHI Créteil, Centre de Recherche Saint-Antoine (CR Saint-Antoine), Institut National de la Santé et de la Recherche Médicale (INSERM)-Université Pierre et Marie Curie - Paris 6 (UPMC), Eurocord, Assistance publique - Hôpitaux de Paris (AP-HP) (APHP)-Institut Universitaire d'Hématologie (IUH), Université Paris Diderot - Paris 7 (UPD7)-Université Paris Diderot - Paris 7 (UPD7), Hôpital Universitaire des Enfants - Reine Fabiola, Université Libre de Bruxelles, Equipe NEMESIS - Centre de Recherches de l'Institut du Cerveau et de la Moelle épinière (NEMESIS-CRICM), Centre de Recherche de l'Institut du Cerveau et de la Moelle épinière (CRICM), Centre National de la Recherche Scientifique (CNRS)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Université Pierre et Marie Curie - Paris 6 (UPMC)-Centre National de la Recherche Scientifique (CNRS)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Université Pierre et Marie Curie - Paris 6 (UPMC), Unité d'Hémato-Immunologie pédiatrique [Hôpital Robert Debré, Paris], Service d'Immuno-hématologie pédiatrique [Hôpital Robert Debré, Paris], Hôpital Robert Debré-Hôpital Robert Debré, Duke University Medical Center, Service de Greffe de Moelle - Unité AJA, Assistance publique - Hôpitaux de Paris (AP-HP) (APHP)-Groupe Hospitalier Saint Louis - Lariboisière - Fernand Widal [Paris], Assistance publique - Hôpitaux de Paris (AP-HP) (APHP), Service d'hématologie clinique, Assistance publique - Hôpitaux de Paris (AP-HP) (APHP)-Hôpital Henri Mondor-Université Paris-Est Créteil Val-de-Marne - Paris 12 (UPEC UP12), Hôpital de la Timone [CHU - APHM] (TIMONE), Department of Haematology, Hôpital Civil, Hopital Civil, Département d'Immunologie, hématologie et rhumatologie pédiatriques [Hôpital Necker-Enfants malades - APHP], CHU Necker - Enfants Malades [AP-HP], Centre Hospitalier Lyon Sud [CHU - HCL] (CHLS), Hospices Civils de Lyon (HCL), Hemostase, Endothelium, Angiogenese (UMR_S_553), Institut National de la Santé et de la Recherche Médicale (INSERM)-Université Paris Diderot - Paris 7 (UPD7), Physiopathologie, Autoimmunité, maladies Neuromusculaires et THErapies Régénératrices (PANTHER), Université de Rouen Normandie (UNIROUEN), Normandie Université (NU)-Normandie Université (NU)-Institut National de la Santé et de la Recherche Médicale (INSERM), Imagine - Institut des maladies génétiques (IMAGINE - U1163), Centre National de la Recherche Scientifique (CNRS)-Université Paris Descartes - Paris 5 (UPD5)-Institut National de la Santé et de la Recherche Médicale (INSERM), Churchill Hospital [Breast Care Unit], Churchill Hospital Oxford Centre for Haematology, Santa Lucia Foundation, IRCSS, Rome, Medical College of Wisconsin, National Institute for Health Research, Centre de Recherche Saint-Antoine (UMRS893), Université Pierre et Marie Curie - Paris 6 (UPMC)-Institut National de la Santé et de la Recherche Médicale (INSERM), Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Institut Universitaire d'Hématologie (IUH), Hôpital Universitaire des Enfants Reine Fabiola [Bruxelles, Belgique] (HUDERF), Université Pierre et Marie Curie - Paris 6 (UPMC)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS)-Université Pierre et Marie Curie - Paris 6 (UPMC)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS), Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Groupe Hospitalier Saint Louis - Lariboisière - Fernand Widal [Paris], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP), Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Hôpital Henri Mondor-Université Paris-Est Créteil Val-de-Marne - Paris 12 (UPEC UP12), Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP), Université Paris Diderot - Paris 7 (UPD7)-Institut National de la Santé et de la Recherche Médicale (INSERM), Université Paris Descartes - Paris 5 (UPD5)-Institut National de la Santé et de la Recherche Médicale (INSERM), UCL - SSS/IREC/PEDI - Pôle de Pédiatrie, and UCL - (SLuc) Service d'hématologie et d'oncologie pédiatrique

Subjects
Male, HYDROXYUREA, Transplantation Conditioning, [SDV]Life Sciences [q-bio], medicine.medical_treatment, CHILDREN, Hematopoietic stem cell transplantation, Biochemistry, THALASSEMIA, 0302 clinical medicine, HLA Antigens, Surveys and Questionnaires, 1114 Paediatrics And Reproductive Medicine, Child, ComputingMilieux_MISCELLANEOUS, CÉLULAS-TRONCO, Hazard ratio, Graft Survival, BONE-MARROW TRANSPLANT, Hematopoietic Stem Cell Transplantation, Hematology, Sickle cell anemia, 3. Good health, Survival Rate, medicine.anatomical_structure, Treatment Outcome, Settore MED/38 - PEDIATRIA GENERALE E SPECIALISTICA, 030220 oncology & carcinogenesis, Child, Preschool, Histocompatibility, [SDV.IMM]Life Sciences [q-bio]/Immunology, Female, Life Sciences & Biomedicine, medicine.medical_specialty, Adolescent, Anemia, Immunology, Anemia, Sickle Cell, 1102 Cardiovascular Medicine And Haematology, Disease-Free Survival, 03 medical and health sciences, Internal medicine, MANAGEMENT, medicine, Humans, ANEMIA, Survival rate, Transplantation, Science & Technology, business.industry, Siblings, Sickle cell disease, Infant, 1103 Clinical Sciences, ADULTS, Cell Biology, medicine.disease, LIFE, EXPERT PANEL, Bone marrow, FOLLOW-UP, business, 030215 immunology
Abstract

Despite advances in supportive therapy to prevent complications of sickle cell disease (SCD), access to care is not universal. Hematopoietic cell transplantation is, to date, the only curative therapy for SCD, but its application is limited by availability of a suitable HLA-matched donor and lack of awareness of the benefits of transplant. Included in this study are 1000 recipients of HLA-identical sibling transplants performed between 1986 and 2013 and reported to the European Society for Blood and Marrow Transplantation, Eurocord, and the Center for International Blood and Marrow Transplant Research. The primary endpoint was event-free survival, defined as being alive without graft failure; risk factors were studied using a Cox regression models. The median age at transplantation was 9 years, and the median follow-up was longer than 5 years. Most patients received a myeloablative conditioning regimen (n = 873; 87%); the remainder received reduced-intensity conditioning regimens (n = 125; 13%). Bone marrow was the predominant stem cell source (n = 839; 84%); peripheral blood and cord blood progenitors were used in 73 (7%) and 88 (9%) patients, respectively. The 5-year event-free survival and overall survival were 91.4% (95% confidence interval, 89.6%-93.3%) and 92.9% (95% confidence interval, 91.1%-94.6%), respectively. Event-free survival was lower with increasing age at transplantation (hazard ratio [HR], 1.09; P < .001) and higher for transplantations performed after 2006 (HR, 0.95; P = .013). Twenty-three patients experienced graft failure, and 70 patients (7%) died, with the most common cause of death being infection. The excellent outcome of a cohort transplanted over the course of 3 decades confirms the role of HLA-identical sibling transplantation for children and adults with SCD.

Published
2017
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36. STEM CELL TRANSPLANTATION IN PYRUVATE KINASE DEFICIENCY

Author

Straaten, S. van, Bierings, M., Bianchi, P., Akiyoshi, K., Kanno, H., Serra, I.B., Chen, J., Huang, X., Beers, E. van, Ekwattanakit, S., Gungor, T., Kors, W.A., Smiers, F., Raymakers, R., Yanez, L., Sevilla, J., Solinge, W. van, Segovia, J.C., and Wijk, R. van

Published
2017

38. ALEXITHYMIA, DEPRESSION, ANXIETY LEVELS AND QUALITY OF LIFE IN PATIENTS

Author

Karahan, AY, Kucuk, A, Balkarli, A, Kayhan, F, Ozhan, N, Nas, O, Gungor, T, and Kucuksen, S

Subjects
Alexithymia, rheumatoid arthritis, depression, anxiety, quality of life
Abstract

Introduction: Rheumatoid arthritis (RA) is a chronic inflammatory disease that manifests itself with joint swelling and pain. Although alexithymia is more commonly seen in painful rheumatic conditions such as RA, there is limited clinical data about the relations with other psychiatric conditions such as depression and anxiety and their impact on quality of life. We aimed to assess the level of alexithymia, depression and anxiety and their effects on quality of life in patients with rheumatoid arthritis. Materials and methods: A hundred forty-eight patients with RA and 100 healthy subjects were included in the study. After physical examination of the patients, Toronto Alexithymia Scale-20 (TAS-20), Beck Depression Inventory (BDI) and Beck Anxiety Inventory (BAI) were performed to determine levels of alexithymia depression and anxiety, respectively. The World Health Organization Quality of Life Scale Abbreviated Version (WHOQoL-BREF) was used to assess the quality of life. Disease Activity Score Calculator for Rheumatoid Arthritis-28 (DAS-28) was performed to evaluate disease activity. Results: The prevalence of alexithymia was 31.1% in patients with RA. Alexithymia was statistically significantly higher in the RA than the control group (p< 0.05). 41.9% (n= 62) of the patients with RA were diagnosed with depression. 20.9% (n= 31) of the patients had severe anxiety symptoms. Although there was no statistically significant difference between alexithymia and depression scores, disease activity scores (p> 0.05) in patient with RA. The anxiety scores were significantly higher (p < 0.05) in patients with RA. RA patients manifested poorer scores in all domains of WHOQoL-BREF than the control subjects (p< 0.05). Conclusion: In this clinical trial it has been demonstrated that regardless of disease activity, alexithymia, symptoms of depression and anxiety are more commonly seen in RA patients that can negatively affect the quality of life. Further studies are needed to confirm this association.

Published
2016

39. Effect of nicotine exposure during gestation on neonatal rat ovaries

Author

Gorkem, U., Togrul, C., Kerem Doga Seckin, Karsli, M. F., Deveci, E., Gungor, T., and Hitit Üniversitesi, Tıp Fakültesi, Cerrahi Tıp Bilimleri Bölümü

Subjects
endocrine system, Nicotine, E-Cadherin, Vimentin, Neonatal Rat, VEGF
Abstract

The purpose of present study was to investigate effects of nicotine on follicular development in the neonatal rats. In this study, 14 adult Sprague-Dawley rats weighing 230-260 g (±10 g) were used as experimental animal. After detecting pregnancies with vaginal smear method, pregnant rats were two equal groups were separated. The pregnant females were treated with the nicotine for 21 days and litters were sacrificed at first day of birth. The ovaries of litters were processed through routine paraffin embedding method, serially sectioned by microtome, stained with hematoxylin-Eosin and immunohistochemical technique investigated by light microscope.In the ovary of group treated nicotine, Preventing follicular development was observed in granular degeneration and luteal cells. It was also observed in inflammation and bleeding vessels. Immunohistochemical localization of vimentin was restricted to the granulosa cells and stromal area. Nicotine treatment group in granular cells and luteal cells in E-cadherin expression was weak. We concluded that nicotine might affect the cellular junctions in the ovarian follicular development. The high intensity in the staining with vimentin observed in the granulosa cells of the follicles is probably due to functional changes that occur during the process of cystogenesis. © 2016, E-Century Publishing Corporation. All rights reserved.

Published
2016

40. Alexithymia, depression, anxiety levels and quality of life in patients with rheumatoid arthritis

Author

Karahan, A.Y., Kucuk, A., Balkarli, Ayşe., Kayhan, F., Özhan, Nail., Nas, O., Gungor, T., and Kucuksen, S.

Subjects
Alexithymia, Quality of life, rheumatoid arthritis, Depression, adult, Beck Depression Inventory, prevalence, Anxiety, physical examination, major clinical study, Article, Toronto Alexithymia scale, female, male, middle aged, Beck Anxiety Inventory, world health organization, controlled study, human, disease activity score
Abstract

Introduction: Rheumatoid arthritis (RA) is a chronic inflammatory disease that manifests itself with joint swelling and pain. Although alexithymia is more commonly seen in painful rheumatic conditions such as RA, there is limited clinical data about the relations with other psychiatric conditions such as depression and anxiety and their impact on quality of life. We aimed to assess the level of alexithymia, depression and anxiety and their effects on quality of life in patients with rheumatoid arthritis. Materials and methods: A hundred forty-eight patients with RA and 100 healthy subjects were included in the study. After physical examination of the patients, Toronto Alexithymia Scale-20 (TAS-20), Beck Depression Inventory (BDI) and Beck Anxiety Inventory (BAI) were performed to determine levels of alexithymia depression and anxiety, respectively. The World Health Organization Quality of Life Scale Abbreviated Version (WHOQoL-BREF) was used to assess the quality of life. Disease Activity Score Calculator for Rheumatoid Arthritis-28 (DAS-28) was performed to evaluate disease activity. Results: The prevalence of alexithymia was 31.1% in patients with RA. Alexithymia was statistically significantly higher in the RA than the control group (p < 0.05). 41.9% (n=62) of the patients with RA were diagnosed with depression. 20.9% (n=31) of the patients had severe anxiety symptoms. Although there was no statistically significant difference between alexithymia and depression scores, disease activity scores (p > 0.05) in patient with RA. The anxiety scores were significantly higher (p < 0.05) in patients with RA. RA patients manifested poorer scores in all domains of WHOQoL-BREF than the control subjects (p < 0.05). Conclusion: In this clinical trial it has been demonstrated that regardless of disease activity, alexithymia, symptoms of depression and anxiety are more commonly seen in RA patients that can negatively affect the quality of life. Further studies are needed to confirm this association.

Published
2016

47. Diagnostic and therapeutic impact of whole body positron emission tomography using fluorine-18-fluoro-2-deoxy-D-glucose in children with chronic granulomatous disease

Author

Gungor, T, Engel-Bicik, I, Eich, G, Willi, U V, Nadal, D, Hossle, J P, Seger, R A, and Steinert, H C

Subjects
PET imaging -- Health aspects, Chronic granulomatous disease -- Care and treatment
Abstract

Abstract Aims--To compare whole body positron emission tomography (PET) using fluorine-18-fluoro-2-deoxy-D-glucose (FDG) with computed tomography (CT) in detecting active infective foci in children with chronic granulomatous disease. Methods--We performed 22 […]

Published
2001

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