The outcomes of childhood acute lymphoblastic leukemia with hyperleukocytosis
Abstract
Background Hyperleukocytosis in childhood acute lymphoblastic leukemia (ALL) is an emergency in oncology. This condition showed high mortality and relapse rates, as well as low survival rate. The outcomes of this group of patients are not yet well studied.
Objective To evaluate the characteristics and outcomes of childhood acute lymphoblastic leukemia (ALL) with hyperleukocytosis.
Methods This was a retrospective cohort study. The patients were children less than 18 year of age who were diagnosed as ALL in Dr. Sardjito Hospital, Yogyakarta, from January 1st 2010 to November, 30th 2016. Event-free survival rate and overall survival rate were estimated for group of patients with the white blood cell (WBC) groups 50-200x109/L and >200x109/L using the Kaplan-Meier method.
Results There were 705 children diagnosed as ALL during the study period, 129 (18%) with hyperleukocytosis and 111 of them met the inclusion criteria, consisted of 76 children in a group of WBC 50-200 x 109/L and 35 children in a group of WBC >200 x 109/L. Presentation at diagnosis: median age were 7 years (range 1 month-18 years), male was 1.5 higher than female, 92% of cases with lymphoid infiltration, 5% with CNS involvement, 40% had bleeding tendency, and 10% had clinical tumor lysis syndrome (TLS). Median WBC was 122 (range 53.4-876) x 109/L; mean Hb was 8 (SD 3) g/dL; median platelet count was 30 (range 1-221) x 109/L. Immunophenotyping was done in 23 patients, 5/23 (8%) was T cell. The patients in lower WBC group showed lower death (26% vs. 34%, P=0,389), higher two-year event-free survival (EFS) 68% vs. 45%, P=0.003, and overall survival (77% vs. 68%, P= 0.16), compared to patients in higher WBC group. Univariate and multivariate Cox regression analyses revealed that none of the variables was a significant prognostic factor for 2 years EFS or overall survival.
Conclusion The group of children with ALL and hyperleukocytosis with lower WBC at diagnoses showed better outcomes than the higher WBC.
References
Campana D, Pui C. Childhood leukemia. In: Niederhuber JE, Armitage JO, Dorshow JH, Kastan MB, Tepper JE, eds. Abeloff’s clinical oncology. 5th ed. Philadelphia: Elsevier Inc; 2016. p.1849-71
Magrath I, Steliarova-Foucher E, Epelman S, Ribeiro RC, Harif M, Li CK, et al. Paediatric cancer in low-income and middle-income countries. Lancet Oncol. 2013;14:e104-16.
Wartenberg D, Groves FD, Adelman AS. Acute lymphoblastic leukemia: epidemiology and etiology. In: Estey EH, Faderl S, Kantarjian H, eds. Acute leukemia. Berlin: Springer; 2008. p. 77–93.
Yeoh, A.E., Tan, D., Li, C.K., Hori, H., Tse, E., Pui, C.H., 2013. Management of adult and paediatric acute lymphoblastic leukaemia in Asia: resoiurce-stratified guidlines from the Asian Oncology Summit 2013. Lancet Oncol 14, 1–32.
Pui CH, Yang JJ, Hunger SP, Pieters R, Schrappe M, Biondi A, et al. Childhood acute lymphoblastic leukemia: progress through collaboration. J Clin Oncol. 2015;33:2938-48.
Supriyadi E, Widjajanto PH, Purwanto I, Cloos J, Veerman AJ, Sutaryo S. Incidence of childhood leukemia in Yogyakarta, Indonesia, 1998–2009. Pediatr Blood Cancer. 2011;57:588–93.
Hussein UY, Yusof NM. Hyperleucocytosis in acute and chronic leukaemias (myeloid & lymphoid types ). Biohealth Sci Bull. 2011;3:20-8.
Oymak Y, Geter S, Aycicek A, Koc A, Soker M, Nice D. Clinical outcomes of childhood leukemia with hyperleukocytosis, without leukopheresis. Acta Medica Cordoba. 2014;3:89–94.
Vaitkevicience G, Heyman M, Jonsson OG, Lausen B, Harila-Saari A, Stenmarker M, et al. Early morbidity and mortality in childhood acute lymphoblastic leukemia with very high white blood cell count. 2013;27:2259–62.
Kong SG, Seo JH, Jun SE, Lee BK, Lim YT. Childhood acute lymphoblastic leukemia with hyperleukocytosis at presentation. Blood Res. 2014;49:29–35.
Lowe EJ, Pul CH, Hancock ML, Geiger TL, Khan RB, Sandlund JT. Early complications in children with acute lymphoblastic leukemia presenting with hyperleukocytosis. Pediatr Blood Cancer. 2005;45:10–5.
Eguiguren BJM, Schell MJ, Crist WM, Kunkel K, Rivera GK. Complications and outcome in childhood acute lymphoblastic leukemia with hyperleukocytosis. Blood. 1992;79:871-5.
Carroll WL, Bhatla T. Acute Lymphoblastic Leukemia. In: Lanzkowsky P, Lipton JM, Fish JD. Lanzkowsky's Manual of Pediatric Hematology and Oncology. 6th edition. New York: Academic Press; 2016. p. 367-89.
Yang X, Lu J, He H, Wang Y, Zhao W, Xiao P, et al. Complications and outcomes of pediatric patients with hyperleukocytic acute lymphoblastic leukemia with CCLG-2008 protocol. Int J Clin Exp Med. 2016;9:3324–33.
Porcu P, Cripe LD, Ng EW, Bhatia S, Danielson CM, Orazi A, et al. Hyperleukocytic leukemias and leukostasis: a review of pathophysiology, clinical presentation and management. Leuk Lymphoma. 2000;39:1–18.
Howard SC, Metzger ML, Wilimas JA, Quintana Y, Pui CH, Robison LL, et al. Childhood cancer epidemiology in low-income countries. Cancer; 2008;112:461-72.
Mostert S, Sitaresmi MN, Gundy CM, Sutaryo, Veerman AJ. Influence of socioeconomic status on childhood acute lymphoblastic leukemia treatment in Indonesia. Pediatrics. 2006;118:1600-6.
Arora RS, Eden T, Pizer B. The problem of treatment abandonment in children from developing countries with cancer. Pediatr Blood Cancer. 2007;49:941-6.
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Accepted 2018-08-10
Published 2018-08-15