Low-intensity hematopoietic stem-cell transplantation across human leucocyte antigen barriers in dyskeratosis congenita
Since the results of conventional hematopoietic stem-cell transplantation (HSCT) for patients with dyskeratosis congenita (DC) are poor owing to the high incidence of transplant-related complications, we explored the use of a low-intensity HSCT regimen. We report two children with DC with severe cyt...
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| Veröffentlicht in: | Bone marrow transplantation (Basingstoke) 2003-05, Vol.31 (10), p.847-850 |
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| creator | DROR, Y FREEDMAN, M. H LEAKER, M VERBEEK, J ARMSTRONG, C. A SAUNDERS, F. E DOYLE, J. J |
| description | Since the results of conventional hematopoietic stem-cell transplantation (HSCT) for patients with dyskeratosis congenita (DC) are poor owing to the high incidence of transplant-related complications, we explored the use of a low-intensity HSCT regimen. We report two children with DC with severe cytopenia, who underwent successful HSCT from a matched unrelated donor after conditioning with fludarabine, cyclophosphamide, and antithymocyte globulin. Graft-versus-host-disease (GVHD) prophylaxis consisted of corticosteroids and cyclosporin A. The regimen was well tolerated, no significant transplant-related complications were observed, and engraftment was rapid and complete. At 15 and 16 months after HSCT, the children were fully engrafted, in excellent clinical condition, full-donor chimerism, and no signs of GVHD. We conclude that a low-intensity regimen is sufficient to induce durable engraftment using matched unrelated donor HSCT in DC patients, with minimal 1-year transplant-related toxicity. Longer follow-up will determine whether this regimen also reduces long-term toxicity. |
| doi_str_mv | 10.1038/sj.bmt.1703931 |
| format | Article |
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H ; LEAKER, M ; VERBEEK, J ; ARMSTRONG, C. A ; SAUNDERS, F. E ; DOYLE, J. J</creator><creatorcontrib>DROR, Y ; FREEDMAN, M. H ; LEAKER, M ; VERBEEK, J ; ARMSTRONG, C. A ; SAUNDERS, F. E ; DOYLE, J. J</creatorcontrib><description>Since the results of conventional hematopoietic stem-cell transplantation (HSCT) for patients with dyskeratosis congenita (DC) are poor owing to the high incidence of transplant-related complications, we explored the use of a low-intensity HSCT regimen. We report two children with DC with severe cytopenia, who underwent successful HSCT from a matched unrelated donor after conditioning with fludarabine, cyclophosphamide, and antithymocyte globulin. Graft-versus-host-disease (GVHD) prophylaxis consisted of corticosteroids and cyclosporin A. The regimen was well tolerated, no significant transplant-related complications were observed, and engraftment was rapid and complete. At 15 and 16 months after HSCT, the children were fully engrafted, in excellent clinical condition, full-donor chimerism, and no signs of GVHD. We conclude that a low-intensity regimen is sufficient to induce durable engraftment using matched unrelated donor HSCT in DC patients, with minimal 1-year transplant-related toxicity. Longer follow-up will determine whether this regimen also reduces long-term toxicity.</description><identifier>ISSN: 0268-3369</identifier><identifier>EISSN: 1476-5365</identifier><identifier>DOI: 10.1038/sj.bmt.1703931</identifier><identifier>PMID: 12748659</identifier><identifier>CODEN: BMTRE9</identifier><language>eng</language><publisher>Basingstoke: Nature Publishing Group</publisher><subject>Abnormalities ; Adrenal Cortex Hormones - therapeutic use ; Adult ; Anesthesia. Intensive care medicine. Transfusions. Cell therapy and gene therapy ; Antigens ; Antilymphocyte serum ; Biological and medical sciences ; Bone marrow, stem cells transplantation. Graft versus host reaction ; Case studies ; Child, Preschool ; Children ; Chimerism ; Complications ; Corticoids ; Corticosteroids ; Cyclophosphamide ; Cyclosporin A ; Cyclosporine - therapeutic use ; Dermatology ; Dosage and administration ; Dyskeratosis ; Dyskeratosis Congenita - therapy ; Epithelial cells ; Female ; Fludarabine ; Globulins ; Graft vs Host Disease - immunology ; Graft vs Host Disease - prevention & control ; Graft-versus-host reaction ; Hematopoietic stem cells ; Histocompatibility Testing ; HLA Antigens - immunology ; Humans ; Immunosuppressive Agents - therapeutic use ; Leukocytes ; Male ; Medical sciences ; Patients ; Pigmentary diseases of the skin ; Prophylaxis ; Stem cell transplantation ; Stem Cell Transplantation - methods ; Stem cells ; Thymocytes ; Toxicity ; Transfusions. Complications. Transfusion reactions. 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H</creatorcontrib><creatorcontrib>LEAKER, M</creatorcontrib><creatorcontrib>VERBEEK, J</creatorcontrib><creatorcontrib>ARMSTRONG, C. A</creatorcontrib><creatorcontrib>SAUNDERS, F. E</creatorcontrib><creatorcontrib>DOYLE, J. J</creatorcontrib><title>Low-intensity hematopoietic stem-cell transplantation across human leucocyte antigen barriers in dyskeratosis congenita</title><title>Bone marrow transplantation (Basingstoke)</title><addtitle>Bone Marrow Transplant</addtitle><description>Since the results of conventional hematopoietic stem-cell transplantation (HSCT) for patients with dyskeratosis congenita (DC) are poor owing to the high incidence of transplant-related complications, we explored the use of a low-intensity HSCT regimen. We report two children with DC with severe cytopenia, who underwent successful HSCT from a matched unrelated donor after conditioning with fludarabine, cyclophosphamide, and antithymocyte globulin. Graft-versus-host-disease (GVHD) prophylaxis consisted of corticosteroids and cyclosporin A. The regimen was well tolerated, no significant transplant-related complications were observed, and engraftment was rapid and complete. At 15 and 16 months after HSCT, the children were fully engrafted, in excellent clinical condition, full-donor chimerism, and no signs of GVHD. We conclude that a low-intensity regimen is sufficient to induce durable engraftment using matched unrelated donor HSCT in DC patients, with minimal 1-year transplant-related toxicity. Longer follow-up will determine whether this regimen also reduces long-term toxicity.</description><subject>Abnormalities</subject><subject>Adrenal Cortex Hormones - therapeutic use</subject><subject>Adult</subject><subject>Anesthesia. Intensive care medicine. Transfusions. Cell therapy and gene therapy</subject><subject>Antigens</subject><subject>Antilymphocyte serum</subject><subject>Biological and medical sciences</subject><subject>Bone marrow, stem cells transplantation. Graft versus host reaction</subject><subject>Case studies</subject><subject>Child, Preschool</subject><subject>Children</subject><subject>Chimerism</subject><subject>Complications</subject><subject>Corticoids</subject><subject>Corticosteroids</subject><subject>Cyclophosphamide</subject><subject>Cyclosporin A</subject><subject>Cyclosporine - therapeutic use</subject><subject>Dermatology</subject><subject>Dosage and administration</subject><subject>Dyskeratosis</subject><subject>Dyskeratosis Congenita - therapy</subject><subject>Epithelial cells</subject><subject>Female</subject><subject>Fludarabine</subject><subject>Globulins</subject><subject>Graft vs Host Disease - immunology</subject><subject>Graft vs Host Disease - prevention & control</subject><subject>Graft-versus-host reaction</subject><subject>Hematopoietic stem cells</subject><subject>Histocompatibility Testing</subject><subject>HLA Antigens - immunology</subject><subject>Humans</subject><subject>Immunosuppressive Agents - therapeutic use</subject><subject>Leukocytes</subject><subject>Male</subject><subject>Medical sciences</subject><subject>Patients</subject><subject>Pigmentary diseases of the skin</subject><subject>Prophylaxis</subject><subject>Stem cell transplantation</subject><subject>Stem Cell Transplantation - methods</subject><subject>Stem cells</subject><subject>Thymocytes</subject><subject>Toxicity</subject><subject>Transfusions. 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H</au><au>LEAKER, M</au><au>VERBEEK, J</au><au>ARMSTRONG, C. A</au><au>SAUNDERS, F. E</au><au>DOYLE, J. J</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Low-intensity hematopoietic stem-cell transplantation across human leucocyte antigen barriers in dyskeratosis congenita</atitle><jtitle>Bone marrow transplantation (Basingstoke)</jtitle><addtitle>Bone Marrow Transplant</addtitle><date>2003-05-01</date><risdate>2003</risdate><volume>31</volume><issue>10</issue><spage>847</spage><epage>850</epage><pages>847-850</pages><issn>0268-3369</issn><eissn>1476-5365</eissn><coden>BMTRE9</coden><abstract>Since the results of conventional hematopoietic stem-cell transplantation (HSCT) for patients with dyskeratosis congenita (DC) are poor owing to the high incidence of transplant-related complications, we explored the use of a low-intensity HSCT regimen. We report two children with DC with severe cytopenia, who underwent successful HSCT from a matched unrelated donor after conditioning with fludarabine, cyclophosphamide, and antithymocyte globulin. Graft-versus-host-disease (GVHD) prophylaxis consisted of corticosteroids and cyclosporin A. The regimen was well tolerated, no significant transplant-related complications were observed, and engraftment was rapid and complete. At 15 and 16 months after HSCT, the children were fully engrafted, in excellent clinical condition, full-donor chimerism, and no signs of GVHD. We conclude that a low-intensity regimen is sufficient to induce durable engraftment using matched unrelated donor HSCT in DC patients, with minimal 1-year transplant-related toxicity. Longer follow-up will determine whether this regimen also reduces long-term toxicity.</abstract><cop>Basingstoke</cop><pub>Nature Publishing Group</pub><pmid>12748659</pmid><doi>10.1038/sj.bmt.1703931</doi><tpages>4</tpages></addata></record> |
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| ispartof | Bone marrow transplantation (Basingstoke), 2003-05, Vol.31 (10), p.847-850 |
| issn | 0268-3369 1476-5365 |
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| source | MEDLINE; Nature Journals Online; Elektronische Zeitschriftenbibliothek - Frei zugängliche E-Journals; SpringerLink Journals - AutoHoldings |
| subjects | Abnormalities Adrenal Cortex Hormones - therapeutic use Adult Anesthesia. Intensive care medicine. Transfusions. Cell therapy and gene therapy Antigens Antilymphocyte serum Biological and medical sciences Bone marrow, stem cells transplantation. Graft versus host reaction Case studies Child, Preschool Children Chimerism Complications Corticoids Corticosteroids Cyclophosphamide Cyclosporin A Cyclosporine - therapeutic use Dermatology Dosage and administration Dyskeratosis Dyskeratosis Congenita - therapy Epithelial cells Female Fludarabine Globulins Graft vs Host Disease - immunology Graft vs Host Disease - prevention & control Graft-versus-host reaction Hematopoietic stem cells Histocompatibility Testing HLA Antigens - immunology Humans Immunosuppressive Agents - therapeutic use Leukocytes Male Medical sciences Patients Pigmentary diseases of the skin Prophylaxis Stem cell transplantation Stem Cell Transplantation - methods Stem cells Thymocytes Toxicity Transfusions. Complications. Transfusion reactions. Cell and gene therapy Transplantation Transplants & implants |
| title | Low-intensity hematopoietic stem-cell transplantation across human leucocyte antigen barriers in dyskeratosis congenita |
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