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Aladağ E. et al: Risk Scores in Stem Cell TransplantationTurk J Hematol 2021;38:138-144undergoing allogeneic hematopoietic stem cell transplantation. Biol BloodMarrow Transplant 2018;24:1947-1951.25. Sorror ML, Maris MB, Storb R, Baron F, Sandmaier BM, Maloney DG, StorerB. Hematopoietic cell transplantation (HCT)-specific-comorbidity index: Anew tool for risk assessment before allogeneic HCT. Blood 2005;106:2912-2919.26. Charlson ME, Pompei P, Ales KL, MacKenzie CR. A new method of classifyingprognostic comorbidity in longitudinal studies: development and validation.J Chronic Dis 1987;40:373-383.27. Armand P, Kim HT, Logan BR, Wang Z, Alyea EP, Kalaycio ME, MaziarzRT, Antin JH, Soiffer RJ, Weisdorf DJ, Rizzo JD, Horowitz MM, Saber W.Validation and refinement of the Disease Risk Index for allogeneic stem celltransplantation. Blood 2014;123:3664-3671.28. Xhaard A, Porcher R, Chien JW, de Latour RP, Robin M, Ribaud P, Rocha V,Devergie A, Ferry C, Martin PJ, Socié G. Impact of comorbidity indexes onnon-relapse mortality. Leukemia 2008;22:2062-2069.29. Versluis J, Labopin M, Niederwieser D, Socie G, Schlenk RF, Milpied N,Nagler A, Blaise D, Rocha V, Cornelissen JJ, Mohty M. Prediction of nonrelapsemortality in recipients of reduced intensity conditioning allogeneicstem cell transplantation with AML in first complete remission. Leukemia2015;29:51-57.30. Barba P, Piñana JL, Martino R, Valcárcel D, Amorós A, Sureda A, Briones J,Delgado J, Brunet S, Sierra J. Comparison of two pretransplant predictivemodels and a flexible HCT-CI using different cut off points to determinelow-, intermediate-, and high-risk groups: the flexible HCT-CI is the bestpredictor of NRM and OS in a population of patients undergoing allo-RIC.Biol Blood Marrow Transplant 2010;16:413-420.31. Chien JW, Sullivan KM. Carbon monoxide diffusion capacity: How low canyou go for hematopoietic cell transplantation eligibility? Biol Blood MarrowTransplant 2009;15:447-453.32. Ljungman P, Brand R, Hoek J, de la Camara R, Cordonnier C, EinseleH, Styczynski J, Ward KN, Cesaro S; Infectious Diseases Working Partyof the European Group for Blood and Marrow Transplantation. Donorcytomegalovirus status influences the outcome of allogeneic stemcell transplant: a study by the European group for blood and marrowtransplantation. Clin Infect Dis 2014;59:473-481.144
Kaya Z. et al: ALPS in Chronic Immune Cytopenia and LymphomaBRIEF REPORTDOI: 10.4274/tjh.galenos.2020.2020.0618Turk J Hematol 2021;38:145-150Autoimmune Lymphoproliferative Syndrome in Children withNonmalignant Organomegaly, Chronic Immune Cytopenia, andNewly Diagnosed LymphomaMalign Olmayan Organomegali, Kronik İmmün Sitopeni ve Yeni Tanı Lenfomalı ÇocuklardaOtoimmün Lenfoproliferatif SendromZühre Kaya 1 , Melek Işık 1 , Nihan Oruklu 2 , Serap Kirkiz 1 , Emin Ümit Bağrıaçık 2 , Luis M. Allende 3 ,María J. Díaz-Madroñero 3 , Raquel Ruiz-García 3 , Faruk Güçlü Pınarlı 4 , Pınar Göçün Uyar 5 , Ülker Koçak 11Gazi University Faculty of Medicine, Department of Pediatric Hematology, Ankara, Turkey2Gazi University Faculty of Medicine, Department of Immunology and Life Science Research Center, Ankara, Turkey3Immunology Department and Research Institute i+12, Hospital Universitario 12 de Octubre, Madrid, Spain4Gazi University Faculty of Medicine, Department of Pediatric Oncology, Ankara, Turkey5Gazi University Faculty of Medicine, Department of Pathology, Ankara, TurkeyAbstractThis study investigated the frequency of and predictive factorsfor autoimmune lymphoproliferative syndrome (ALPS) in childrenwith lymphoma, chronic immune cytopenia, and nonmalignantorganomegaly. Thirty-four children with suspected ALPS (n=13,lymphoma; n=12, immune cytopenia; n=9, nonmalignantorganomegaly) were included. Double-negative T-cells, lymphocyteapoptosis, and genetic findings were analyzed. Patients were stratifiedinto two groups as proven/probable ALPS and clinically suspectedpatients according to the ALPS diagnostic criteria. Of the 34 patients,18 (53%) were diagnosed with proven/probable ALPS. One patient hada mutation (c.652-2A>C) in the FAS gene. The remaining 16 (47%)patients were defined as clinically suspected patients. Predictivefactors for ALPS were anemia and thrombocytopenia in patients withlymphoma, splenomegaly and lymphadenopathy in patients withimmune cytopenia, and young age in patients with nonmalignantorganomegaly. ALPS may not be rare in certain risk groups. Our studyindicates that screening for ALPS may be useful in children havinglymphoma with cytopenia at diagnosis, in those having nonmalignantorganomegaly with immune cytopenia, and in those having chronicimmune thrombocytopenic purpura or autoimmune hemolytic anemiawith organomegaly developing during follow-up.Keywords: Autoimmune lymphoproliferative syndrome, Immunecytopenia, LymphomaÖzBu çalışmanın amacı malign olmayan organomegali, kronik immünsitopeni ve lenfomalı çocuklarda otoimmün lenfoproliferatif sendrom(OILS) sıklığını ve belirleyici faktörlerini araştırmaktır. Bu çalışmayaOILS şüpheli 34 hasta dahil edildi (13 hasta lenfoma, 5 hasta otoimmünhemolitik anemi, 7 hasta kronik immün trombositopenik purpurave 9 hasta malign olmayan organomegali). Çift negatif T-hücreler,lenfosit apoptozis ve genetik bulgular analiz edildi. Hastalar OILStanı kriterlerine göre kesin ve yüksek olasılıklı OILS’li hastalar veklinik şüpheli OILS’li hastalar olarak iki gruba ayrıldı. Çalışmaya dahiledilen 34 hastanın, 18’i (%53) kesin ve yüksek olasılıklı OILS’di. Malignolmayan organomegalisi olan bir çocukta FAS geninde mutasyon(c.652-2A>C) saptandı. Klinik şüpheli hasta 16 (%47) idi. OILS içinbelirleyici faktörler; lenfomalı hastalarda anemi ve trombositopeni,kronik immün sitopenili hastalarda splenomegali ve lenfadenopati;malign olmayan organomegalisi olan hastalarda genç yaş idi. OILSbelirli risk gruplarında nadir olmayabilir. Çalışmamız, ilk tanıdasitopenisi olan lenfomalı çocuklarda, immün sitopenisi olan nonmalignorganomegalili çocuklarda ve izlem sırasında organomegalisi gelişenkronik immün trombositopenik purpura ve otoimmün hemolitikanemili çocuklarda OILS taramasının yararlı olabileceğine işaretetmektedir.Anahtar Sözcükler: Otoimmün lenfoproliferatif sendrom, İmmünsitopeni, Lenfoma©Copyright 2021 by Turkish Society of HematologyTurkish Journal of Hematology, Published by Galenos Publishing HouseAddress for Correspondence/Yazışma Adresi: Zühre Kaya, M.D., Gazi University Faculty of Medicine,Department of Pediatric Hematology, Ankara, TurkeyE-mail : zuhrekaya@gazi.edu.tr ORCID: orcid.org/0000-0002-3798-7246Received/Geliş tarihi: October 16, 2020Accepted/Kabul tarihi: December 29, 2020145
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