{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,25]],"date-time":"2026-09-25T04:59:52Z","timestamp":1790312392579,"version":"4.1.0"},"reference-count":22,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2026,9,25]],"date-time":"2026-09-25T00:00:00Z","timestamp":1790294400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Immunol."],"abstract":"<jats:p>\n                    Experience in hematopoietic stem cell transplant (HSCT) as a long-term curative treatment for sickle-cell anemia (SCA) has been gathered over the last 30 years. Sustained engraftment to restore normal erythropoiesis remains the primary aim of HSCT in this disease. Yet, with the increasing use of reduced intensity conditioning as a toxicity-sparing alternative, a significant proportion of transplanted patients develop mixed chimerism. While a myeloid donor chimerism above 20% is deemed sufficient to prevent pain crisis, patients with donor chimerism below 50% can present recurrence of hemolytic features (\n                    <jats:xref>1<\/jats:xref>\n                    ). Although experimental studies have demonstrated the feasibility of chimerism analysis on CD71-positive erythroblasts as well as earlier erythroid precursors such as BFU-E and CFU-GM, their clinical benefit remains debated (\n                    <jats:xref>2<\/jats:xref>\n                    ,\n                    <jats:xref>3<\/jats:xref>\n                    ). Herein we report the unusual case of a 15 year-old patient transplanted for SCA who developed Pure Red Cell Aplasia (PRCA), followed by the recurrence of hemolytic anemia and rising HbS levels upon reticulocyte recovery. The patient failed to respond to standard treatment with Rituximab and Daratumumab for PRCA, which further raised concern for poor donor erythroid engraftment or secondary graft failure. In this setting, the information supplied by lineage-specific chimerism, particularly B-cell and erythroid progenitor chimerism, which were both predominantly of donor origin, helped distinguishing the two conditions and guide clinical decision-making. The hematological condition of the patient dramatically improved upon donor-derived B-cell recovery and initiation of plasmapheresis. This clinical report makes an argument for lineage-specific chimerism in patients with hemoglobinopathies experiencing recurrence of hemolytic anemia after PRCA.\n                  <\/jats:p>","DOI":"10.3389\/fimmu.2026.1932283","type":"journal-article","created":{"date-parts":[[2026,9,25]],"date-time":"2026-09-25T04:26:15Z","timestamp":1790310375000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":0,"title":["Case Report: Recurrence of recipient-derived erythropoiesis in hematopoietic stem cell transplant for sickle-cell anemia: multi-lineage chimerism analysis as a tool to distinguish atypical pure red cell aplasia from poor erythroid engraftment"],"prefix":"10.3389","volume":"17","author":[{"given":"Claire","family":"Geurten","sequence":"first","affiliation":[{"name":"Department of Paediatric Haemato-oncology & Stem Cell Transplantation (SCT), Universitaire Ziekenhuizen Leuven","place":["Leuven, Belgium"]},{"name":"Department of Paediatric Haemato-oncology & Stem Cell Transplantation (SCT), Centre Hospitalier Universitaire de Li\u00e8ge","place":["Li\u00e8ge, Belgium"]},{"name":"GIGA Immunobiology, University of Li\u00e8ge","place":["Li\u00e8ge, Belgium"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Evelyne","family":"Willems","sequence":"additional","affiliation":[{"name":"Department of Clinical Haematology, Centre Hospitalier Universitaire de Li\u00e8ge","place":["Li\u00e8ge, Belgium"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nathalie","family":"Dhedin","sequence":"additional","affiliation":[{"name":"Department of Haematology Adolescent & Young Adults, H\u00f4pital Saint-Louis","place":["Paris, France"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Corinne","family":"Pondarre","sequence":"additional","affiliation":[{"name":"Centre Hospitalier Intercommunal de Cr\u00e9teil, INSERM Unit\u00e9 955, Paris XII Universit\u00e9","place":["Cr\u00e9teil, France"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Annarita","family":"Miccio","sequence":"additional","affiliation":[{"name":"Laboratory of Chromatin and Gene Regulation During Development, Institut Imagine, Institut national de la sant\u00e9 et de la recherche m\u00e9dicale UMR 1163, Universit\u00e9 Paris Cit\u00e9","place":["Paris, France"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ivan","family":"Sloma","sequence":"additional","affiliation":[{"name":"Department of Haematology and Immunobiology, Institut Mondor de Recherche Biom\u00e9dicale, Institut national de la sant\u00e9 et de la recherche m\u00e9dicale UMR 955,Assistance Publique - H\u00f4pitaux de Paris (AP-HP) H\u00f4pitaux universitaires Henri Mondor","place":["Paris, France"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maria Bou","family":"Dames","sequence":"additional","affiliation":[{"name":"Laboratory of Chromatin and Gene Regulation During Development, Institut Imagine, Institut national de la sant\u00e9 et de la recherche m\u00e9dicale UMR 1163, Universit\u00e9 Paris Cit\u00e9","place":["Paris, France"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Louise","family":"Vrancken","sequence":"additional","affiliation":[{"name":"GIGA Immunobiology, University of Li\u00e8ge","place":["Li\u00e8ge, Belgium"]},{"name":"Department of Clinical Haematology, Centre Hospitalier Universitaire de Li\u00e8ge","place":["Li\u00e8ge, Belgium"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jaques","family":"Foguenne","sequence":"additional","affiliation":[{"name":"Department of Biological Haematology, Centre Hospitalier Universitaire de Li\u00e8ge","place":["Li\u00e8ge, Belgium"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"J\u00e9r\u00f4me","family":"Coupier","sequence":"additional","affiliation":[{"name":"Department of Genetics, Centre Hospitalier Universitaire de Li\u00e8ge","place":["Li\u00e8ge, Belgium"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Frederic","family":"Baron","sequence":"additional","affiliation":[{"name":"GIGA Immunobiology, University of Li\u00e8ge","place":["Li\u00e8ge, Belgium"]},{"name":"Department of Clinical Haematology, Centre Hospitalier Universitaire de Li\u00e8ge","place":["Li\u00e8ge, Belgium"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1965","published-online":{"date-parts":[[2026,9,25]]},"reference":[{"key":"B1","unstructured":"Sickle-Cell Disease\n          \n          \n          Geneva\n          World Health Organization"},{"key":"B2","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM198409203111207","article-title":"Bone-marrow transplantation in a patient with sickle-cell anemia","volume":"311","author":"Johnson","year":"1984","journal-title":"N Engl J 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