Autor: Cheuk, Daniel - Prolib Integro

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Tytuł:: Survival outcomes of children with relapsed or refractory myeloid leukemia associated with Down syndrome
Autorzy:: Cellot, Sonia
Merli, Pietro
Hasegawa, Daisuke
Chopek, Ashley
Bartram, Jack
Locatelli, Franco
Ariffin, Hany
Antoniou, Evangelia
Klusmann, Jan-Henning
Hitzler, Johann K.
Raghuram, Nikhil
Skajaa, Torjus
Kolenova, Alexandra
Sung, Lillian
Cheuk, Daniel
Balwierz, Walentyna
Ab Rahman, Syaza
Norton, Alice
Rojas-Vasquez, Marta
Aftandilian, Catherine
Rabin, Karen R.
Goemans, Bianca F.
Kolb, E. Anders
McGuire, Bryan
Nakashima, Kentaro
O'Brien, Maureen
Verma, Anupam
Kotecha, Rishi Sury
Arad-Cohen, Nira
Hasle, Henrik
Jahnukainen, Kirsi
Opis:: Children with Down syndrome (DS) are at a significantly higher risk of developing acute myeloid leukemia, also termed myeloid leukemia associated with DS (ML-DS). In contrast to the highly favorable prognosis of primary ML-DS, the limited data that are available for children who relapse or who have refractory ML-DS (r/r ML-DS) suggest a dismal prognosis. There are few clinical trials and no standardized treatment approach for this population. We conducted a retrospective analysis of international study groups and pediatric oncology centers and identified 62 patients who received treatment with curative intent for r/r ML-DS between year 2000 to 2021. Median time from diagnosis to relapse was 6.8 (range, 1.1-45.5) months. Three-year event-free survival (EFS) and overall survival (OS) were 20.9 ± 5.3% and 22.1 ± 5.4%, respectively. Survival was associated with receipt of hematopoietic stem cell transplantation (HSCT) (hazard ratio [HR], 0.28), duration of first complete remission (CR1) (HR, 0.31 for > 12 months) and attainment of remission after relapse (HR, 4.03). Patients who achieved complete remission (CR) before HSCT, had an improved OS and EFS of 56.0 ± 11.8% and 50.5 ± 11.9%, respectively compared to those who underwent HSCT without CR (3-year OS and EFS of 10.0 ± 9.5%). Treatment failure after HSCT was predominantly because of disease recurrence (52%) followed by treatment-related mortality (10%). The prognosis of r/r ML-DS remains dismal even in the current treatment period and serve as a reference point for current prognostication and future interventional studies. Clinical trials aimed at improving the survival of patients with r/r ML-DS are needed.
Dostawca treści:: Repozytorium Uniwersytetu Jagiellońskiego

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