Alessandra Bernardi*, Pierpaolo Berti, Costantino Damico, Elisabetta Cicchetti, Fabiola Landi, Ottavia Porzio and Michele Vacca
Volume5-Issue7
Dates: Received: 2024-07-11 | Accepted: 2024-07-23 | Published: 2024-07-24
Pages: 834-837
Abstract
Hematopoietic Stem Cell Transplantation (HSCT) is a curative treatment for certain blood disorders, but can lead to complications such as Autoimmune Cytopenia (AIC), particularly Autoimmune Hemolytic Anemia (AIHA). AIHA after HSCT often involves donor-derived antibodies attacking donor red blood cells, but this case report describes a unique case in a 7-year-old male with beta-thalassemia major. Following HSCT, he developed AIHA nine months after transplantation from an unrelated donor. Despite initially successful engraftment, chimerism analysis revealed a decline in donor cells coincident with the onset of AIHA. The patient had severe hemolysis, a positive Direct Coombs Test (DAT) for IgG and C3d, and several antibodies including anti-e, anti- C, and anti-D. Treatment with cyclosporine and prednisone initially stabilized his condition, but discontinuation resulted in relapse, worsening chimerism, and increased antibody titers. Analysis confirmed the predominance of recipient immune cells, suggesting that AIHA was driven by the recipient's immune response against his own red blood cells. This case emphasizes the complexity of immune reconstitution after HSCT and highlights a recipient-versus-recipient mechanism in post-transplant AIHA.
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DOI: 10.37871/jbres1961
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Copyright
© 2024 Bernardi A, et al. Distributed under Creative Commons CC-BY 4.0 
How to cite this article
Bernardi A, Berti P, Damico C, Cicchetti E, Landi F, Porzio O, Vacca M. Donor vs. Donor or Recipient vs. Recipient? A Singular Case of Post Hematopoietic Stem Cell Transplantation Autoimmune Hemolytic Anemia in a Pediatric Patient. J Biomed Res Environ Sci. 2024 Jul 24; 5(7): 834-837. doi: 10.37871/jbres1961, Article ID: JBRES1961, Available at: https://www.jelsciences.com/ articles/jbres1961.pdf
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References
- Greenmyer JR, Ariagno S, Ali A, Pence L, O'Shea M, Greenmyer LA, Khan S, Kuhn A, Martin C, Ferdjallah A, Kohorst M. Autoimmune cytopenias following pediatric hematopoietic cell transplant. Bone Marrow Transplant. 2024 Jan;59(1):117-120. doi: 10.1038/s41409-023-02116-y. Epub 2023 Oct 4. PMID: 37794111.
- González-Vicent M, Sanz J, Fuster JL, Cid J, de Heredia CD, Morillo D, Fernández JM, Pascual A, Badell I, Serrano D, Fox L, de la Serna J, Benito A, Couselo JM, Molina B, Díaz MÁ, Sanz MÁ. Autoimmune hemolytic anemia (AIHA) following allogeneic hematopoietic stem cell transplantation (HSCT): A retrospective analysis and a proposal of treatment on behalf of the Grupo Español De Trasplante de Medula Osea en Niños (GETMON) and the Grupo Español de Trasplante Hematopoyetico (GETH). Transfus Med Rev. 2018 Mar 3:S0887-7963(17)30164-5. doi: 10.1016/j.tmrv.2018.02.005. Epub ahead of print. PMID: 29573905.
- Gabelli M, Ademokun C, Cooper N, Amrolia PI. Pathogenesis, risk factors and therapeutic options for autoimmune haemolytic anaemia in the post-transplant setting. Br J Haematol. 2022 Jan;196(1):45-62. doi: 10.1111/bjh.17648. Epub 2021 Jun 30. PMID: 34195990.
- Wang M, Wang W, Abeywardane A, Adikarama M, McLornan D, Raj K, de Lavallade H, Devereux S, Mufti GJ, Pagliuca A, Potter VT, Mijovic A. Autoimmune hemolytic anemia after allogeneic hematopoietic stem cell transplantation: analysis of 533 adult patients who underwent transplantation at King's College Hospital. Biol Blood Marrow Transplant. 2015 Jan;21(1):60-6. doi: 10.1016/j.bbmt.2014.09.009. Epub 2014 Sep 28. PMID: 25262883.
- Szanto CL, Langenhorst J, de Koning C, Nierkens S, Bierings M, Huitema ADR, Lindemans CA, Boelens JJ. Predictors for Autoimmune Cytopenias after Allogeneic Hematopoietic Cell Transplantation in Children. Biol Blood Marrow Transplant. 2020 Jan;26(1):114-122. doi: 10.1016/j.bbmt.2019.07.022. Epub 2019 Jul 22. PMID: 31344451.
- Koo J, Giller RH, Quinones R, McKinney CM, Verneris MR, Knight-Perry J. Autoimmune cytopenias following allogeneic hematopoietic stem cell transplant in pediatric patients: Response to therapy and late effects. Pediatr Blood Cancer. 2020 Sep;67(9):e28591. doi: 10.1002/pbc.28591. Epub 2020 Jul 13. PMID: 32658382.
- Ogonek J, Kralj Juric M, Ghimire S, Varanasi PR, Holler E, Greinix H, Weissinger E. Immune Reconstitution after Allogeneic Hematopoietic Stem Cell Transplantation. Front Immunol. 2016 Nov 17;7:507. doi: 10.3389/fimmu.2016.00507. PMID: 27909435; PMCID: PMC5112259.
- Li Z, Rubinstein SM, Thota R, Savani M, Brissot E, Shaw BE, Majhail NS, Mohty M, Savani BN. Immune-Mediated Complications after Hematopoietic Stem Cell Transplantation. Biol Blood Marrow Transplant. 2016 Aug;22(8):1368-1375. doi: 10.1016/j.bbmt.2016.04.005. Epub 2016 Apr 14. PMID: 27095688.
- Michniacki TF, Ebens CL, Choi SW. Immune-Mediated Cytopenias After Hematopoietic Cell Transplantation: Pathophysiology, Clinical Manifestations, Diagnosis, and Treatment Strategies. Curr Oncol Rep. 2019 Aug 15;21(10):87. doi: 10.1007/s11912-019-0838-7. PMID: 31414187.
- Michalak SS, Olewicz-Gawlik A, Rupa-Matysek J, Wolny-Rokicka E, Nowakowska E, Gil L. Autoimmune hemolytic anemia: current knowledge and perspectives. Immun Ageing. 2020 Nov 20;17(1):38. doi: 10.1186/s12979-020-00208-7. PMID: 33292368; PMCID: PMC7677104.
