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T and NK Cells in IL2RG-Deficient Patient 50 Years After Hematopoietic Stem Cell Transplantation.
Melsen, Janine E; van Ostaijen-Ten Dam, Monique M; van den Akker, Erik B; Welters, Marij J P; Heezen, Kim C; Pico-Knijnenburg, Ingrid; Kolijn, P Martijn; Bredius, Robbert G M; van Doorn, Remco; Langerak, Anton W; Schilham, Marco W; Lankester, Arjan C.
Afiliación
  • Melsen JE; Department of Pediatrics, Stem Cell Transplantation Program and Laboratory for Pediatric Immunology, Willem-Alexander Children's Hospital, Leiden University Medical Center, Albinusdreef 2, 2333ZA, Leiden, The Netherlands. j.e.melsen@lumc.nl.
  • van Ostaijen-Ten Dam MM; Department of Pediatrics, Stem Cell Transplantation Program and Laboratory for Pediatric Immunology, Willem-Alexander Children's Hospital, Leiden University Medical Center, Albinusdreef 2, 2333ZA, Leiden, The Netherlands.
  • van den Akker EB; Department of Biomedical Data Sciences, Leiden University Medical Center, Leiden, The Netherlands.
  • Welters MJP; Pattern Recognition & Bioinformatics, Delft University of Technology, Delft, The Netherlands.
  • Heezen KC; Department of Medical Oncology, Leiden University Medical Center, Leiden, The Netherlands.
  • Pico-Knijnenburg I; Department of Immunology, Erasmus Medical Center, Rotterdam, The Netherlands.
  • Kolijn PM; Department of Pediatrics, Stem Cell Transplantation Program and Laboratory for Pediatric Immunology, Willem-Alexander Children's Hospital, Leiden University Medical Center, Albinusdreef 2, 2333ZA, Leiden, The Netherlands.
  • Bredius RGM; Department of Immunology, Erasmus Medical Center, Rotterdam, The Netherlands.
  • van Doorn R; Department of Pediatrics, Stem Cell Transplantation Program and Laboratory for Pediatric Immunology, Willem-Alexander Children's Hospital, Leiden University Medical Center, Albinusdreef 2, 2333ZA, Leiden, The Netherlands.
  • Langerak AW; Department of Dermatology, Leiden University Medical Center, Leiden, The Netherlands.
  • Schilham MW; Department of Immunology, Erasmus Medical Center, Rotterdam, The Netherlands.
  • Lankester AC; Department of Pediatrics, Stem Cell Transplantation Program and Laboratory for Pediatric Immunology, Willem-Alexander Children's Hospital, Leiden University Medical Center, Albinusdreef 2, 2333ZA, Leiden, The Netherlands.
J Clin Immunol ; 42(6): 1205-1222, 2022 08.
Article en En | MEDLINE | ID: mdl-35527320
The first successful European hematopoietic stem cell transplantation (HSCT) was performed in 1968 as treatment in a newborn with IL2RG deficiency using an HLA-identical sibling donor. Because of declining naive T and natural killer (NK) cells, and persistent human papilloma virus (HPV)-induced warts, the patient received a peripheral stem cell boost at the age of 37 years. NK and T cells were assessed before and up to 14 years after the boost by flow cytometry. The boost induced renewed reconstitution of functional NK cells that were 14 years later enriched for CD56dimCD27+ NK cells. T-cell phenotype and T-cell receptor (TCR) repertoire were simultaneously analyzed by including TCR Vß antibodies in the cytometry panel. Naive T-cell numbers with a diverse TCR Vß repertoire were increased by the boost. Before and after the boost, clonal expansions with a homogeneous TIGIT and PD-1 phenotype were identified in the CD27- and/or CD28- memory population in the patient, but not in the donor. TRB sequencing was applied on sorted T-cell subsets from blood and on T cells from skin biopsies. Abundant circulating CD8 memory clonotypes with a chronic virus-associated CD57+KLRG1+CX3CR1+ phenotype were also present in warts, but not in healthy skin of the patient, suggesting a link with HPV. In conclusion, we demonstrate in this IL2RG-deficient patient functional NK cells, a diverse and lasting naive T-cell compartment, supported by a stem cell boost, and an oligoclonal memory compartment half a century after HSCT.
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Verrugas / Trasplante de Células Madre Hematopoyéticas / Infecciones por Papillomavirus Límite: Adult / Humans / Newborn Idioma: En Revista: J Clin Immunol Año: 2022 Tipo del documento: Article País de afiliación: Países Bajos Pais de publicación: Países Bajos
Texto completo
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Verrugas / Trasplante de Células Madre Hematopoyéticas / Infecciones por Papillomavirus Límite: Adult / Humans / Newborn Idioma: En Revista: J Clin Immunol Año: 2022 Tipo del documento: Article País de afiliación: Países Bajos Pais de publicación: Países Bajos