Improved outcome of childhood acute myeloid leukemia in an Eastern European country: Lithuanian experience

Kairienė, Ignė; Pasaulienė, Ramunė; Lipunova, N. A.; Vaitkevičienė, Goda; Ragelienė, Lina; Rascon, Jelena

doi:10.1007/s00431-017-2978-9

Cited by 14 publications

(20 citation statements)

References 30 publications

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“…During the 2008–2014 period, paediatric solid tumours became increasingly divided between two paediatric oncology centres. The beneficial effect of patient concentration in a small country was demonstrated in our studies with childhood acute lymphoblastic and myeloid leukaemia enabling to achieve survival rates comparable to the numbers reported by research-intensive institutions ( 14 , 15 ). Third, the small number of children corresponding to the decreasing population (the number of inhabitants in Lithuania in 2000–2014 decreased from 3.51 to 2.94 million, ( 24 )) did not allow us to reach any statistically significant conclusions.…”

Section: Discussionsupporting

confidence: 74%

“…None of the 40 included children died from treatment-associated toxicity, which reflects sufficient experience of medical staff despite the small number of patients. Our previous research on acute myeloid leukaemia, one of the most challenging paediatric malignancy with regard to treatment-related toxicity, also demonstrated sufficient knowledge and experience accumulated at our institution for successful management of highly aggressive paediatric tumours ( 15 ).…”

Section: Discussionmentioning

confidence: 86%

“…Initiation of a clinical trial NOPHO ALL-2008 has led to a significant improvement of event-free survival in patients treated for acute lymphoblastic leukaemia ( 14 ). In addition, patients’ concentration, gain of experience and adequate diagnostic and treatment facilities have dramatically improved survival of patients with acute myeloid leukaemia ( 15 ).…”

Section: Introductionmentioning

confidence: 99%

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Survival of children treated for Ewing sarcoma in Lithuania: a single centre experience

Jakutis

Ragelienė

Rascon

2018

AML

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Introduction.Ewing sarcoma (ES) is a rare and aggressive malignant neoplasm that mostly affects children and adolescents. Recent studies reported a gap of 20% in childhood cancer survival rates between the Northern/Western and the Eastern European countries. We aimed to analyse the survival of patients treated for ES at our institution, to evaluate its correspondence to current survival rates in the expert centres, and to assess changes in treatment outcomes over time.Materials and methods.A retrospective single-centre study was performed. Children under 18 years of age, diagnosed and treated for Ewing sarcoma/PNET at our institution from 2000 to 2014 were included. To assess the hypothesized improvement of treatment outcomes over time, a comparative analysis of two time periods – 2000–2007 and 2008–2014 – was carried out. Five-year overall survival (OS5y) and disease-free survival (DFS5y) were chosen as primary study end-points. Clinical and laboratory data were retrieved from patient records.Results.In total, 40 patients were included in the study: 24 (60%) males and 16 (40%) females. Twenty-eight children (70%) presented with local and 12 (30%) with primary metastatic disease. Over the analysed time frame, improvement in treatment outcomes was observed: DFS5y improved from 46% in 2000–2007 to 61% in 2008–2014 (p = 0.27), whereas OS5y changed minimally from 62% in 2000–2007 to 65% in 2008–2014. Increase in DFS5y was more prominent for localized disease –from 50% in 2000–2007 to 74% in 2008–2014 (p = 0.14). Prognosis of initial metastatic disease remained poor with DFS5y: 25% in 2000–2007 and 38% in 2008–2014. Patients’ median follow-up was 12.3 years (the range from 8.1 to 15.6) and 3.9 years (the range from 1.1 to 8.0) in the first and second study groups, respectively.Conclusions.OS5y of the entire patient cohort did not change considerably over time and remained slightly inferior compared to the best reported survival rates. There was an evident trend for improvement of DFS5y in localized disease. Survival of children with primary metastases remained poor despite slight increase in DFS5y. Implementation of international clinical trials, consolidation of multidisciplinary approach, patients’ concentration and widening of research activities could be beneficial for the treatment of children in the future.

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Section: Discussionsupporting

confidence: 74%

Section: Discussionmentioning

confidence: 86%

Section: Introductionmentioning

confidence: 99%

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Survival of children treated for Ewing sarcoma in Lithuania: a single centre experience

Jakutis

Ragelienė

Rascon

2018

AML

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“…A cornerstone change in care of solid paediatric tumours in Lithuania was the transition from adult services (where children with WT were treated up to 1998) to specialized paediatric oncology centres. Centralisation of patient care and accumulation of specific knowledge and expertise was crucial for the improvement of cure rates as demonstrated in our previous study in which we analysed the outcome of treatment of paediatric acute myeloid leukaemia (19).…”

Section: Discussionmentioning

confidence: 99%

Wilms tumour in children: 18 years of experience at Vilnius University Hospital Santaros Klinikos, Lithuania

Rančelytė¹,

Nemanienė²,

Ragelienė³

et al. 2019

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Introduction. Wilms tumour (WT) is the most common childhood abdominal malignancy, with an average annual incidence of 1 in 10,000 children. The study published in 2002 reported lower survival rates of WT in Lithuania in comparison to the data of SIOP-9 study and the European Organization for Research and Treatment of Cancer (EORTC). We aimed to assess current diagnostic approach and treatment results of patients with WT treated at our institution and to compare the results with the previously published study. Materials and methods. A retrospective single-centre study was performed. 48 patients with WT registered at the institutional database from 2000 to 2018 were enrolled. An estimated 5-year overall survival (OS5y) and 2-year event-free survival (EFS2y) by stage and risk groups was calculated using IBM SPSS. A comparative analysis of two time periods – 2000–2008 and 2009–2018 – was carried out. Results. Forty-two (87.5%) patients presented with localised disease and 6 (12.5%) with primary metastatic disease. The majority of cases were of the intermediate-risk group (77%). The OS5yof all analysed children was 86.4%. The EFS2y was 88.9% in stage I, 91.7% in stage II, 83.3% in stage III, and 50% in stage IV. The EFS2y was 100% in the low-risk group, 86.5% in the intermediate-risk group, and 25% in the high-risk group. Improvement of outcomes was observed over the analysed period: OS5y changed from 81.0% in 2000– 2008 to 92.6% in 2009–2018. Among 48 cases, ten patients showed recurrence: eight – early relapse and two – late relapse. Six patients died. Conclusions. WT was diagnosed at early stages in most cases. The survival was better among the patients diagnosed in earlier stages and with favourable risk group. Better survival rates were observed in patients treated in 2009–2018 compared to the 2000–2008 period.

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“…The lack of improvement in cure rate over time (the OS 5y was 61.1% in 2000-2007 vs 57.9% in 2008-2015) was rather unexpected. National population-based studies on leukemia (22,23) (27). Of note, in our study a cancer predisposition syndrome was documented in five patients (Table 1).…”

Section: Treatment Outcomementioning

confidence: 49%

The impact of incomplete registration on survival rate of children with very rare tumors

Rascon

Šalaševičius

Rutkauskienė

et al. 2020

Preprint

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Background. Pediatric very rare tumors (VRTs) represent a heterogeneous subset of childhood cancers, with reliable survival estimates depending dramatically on each (un)registered case. The current study aimed to evaluate the number of VRTs among Lithuanian children, to assess the impact of the registration status on survival rate and to track changes in treatment outcome over the 16-year study period. Methods. We performed a population-based retrospective study across children below 18 years old diagnosed with a VRT in Lithuania between the years 2000 and 2015. The identified cases were cross-checked with the Lithuanian Cancer Registry – a population-based epidemiology cancer registry – for the fact of registration and survival status. The overall survival was calculated according to the registration status and treatment period. Results. Thirty-seven children with VRTs were identified within the defined time frame. Six of them (16.2%) were not reported to the Lithuanian Cancer Registry at diagnosis. The probability of overall survival at 5 years (OS5y) differed significantly between the registered (n = 31) and unregistered (n = 6) cohorts: 51.6% vs 100%, respectively (p = 0.049). A 5-year survival estimate for children diagnosed with a VRT at the age of 0-14 years differed by 10% according to the registration completeness: 52.1% calculated for the entire cohort vs 42.1% for registered patients only. The OS5y did not improve over the analyzed period: 61.1% in 2000-2007 vs 57.9% in 2008-2015 (p = 0.805). The survival continued to decline beyond 5 years due to late cancer-related adverse events: 59.5% of patients were alive at 5 years as compared to 44.3% at 10 years. Conclusions. The OS5y of children affected by VRT was lower than in more common childhood cancers. The survival rate of the unregistered patients was superior that mislead interpretation of treatment outcome. Meticulous registration of VRTs is crucial for correct evaluation of treatment outcome, especially across small countries with few cases.

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Improved outcome of childhood acute myeloid leukemia in an Eastern European country: Lithuanian experience

Cited by 14 publications

References 30 publications

Survival of children treated for Ewing sarcoma in Lithuania: a single centre experience

Survival of children treated for Ewing sarcoma in Lithuania: a single centre experience

Wilms tumour in children: 18 years of experience at Vilnius University Hospital Santaros Klinikos, Lithuania

The impact of incomplete registration on survival rate of children with very rare tumors

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