Late effects in survivors of childhood acute lymphoblastic leukemia in the context of selected gene polymorphisms

Abstract Background It has been shown that approximately half of survivors of childhood acute lymphoblastic leukemia (ALL) have symptomatic late effects (LE) that may be severe or life-threatening. The aim of our study was to assess the health status of childhood ALL survivors after over 10 years of...

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Main Authors: Kinga Kwiecinska, Wojciech Strojny, Danuta Pietrys, Miroslaw Bik-Multanowski, Maciej Siedlar, Walentyna Balwierz, Szymon Skoczen
Format: Article
Language:English
Published: BMC 2018-08-01
Series:Italian Journal of Pediatrics
Subjects:
Online Access:http://link.springer.com/article/10.1186/s13052-018-0526-5
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author Kinga Kwiecinska
Wojciech Strojny
Danuta Pietrys
Miroslaw Bik-Multanowski
Maciej Siedlar
Walentyna Balwierz
Szymon Skoczen
author_facet Kinga Kwiecinska
Wojciech Strojny
Danuta Pietrys
Miroslaw Bik-Multanowski
Maciej Siedlar
Walentyna Balwierz
Szymon Skoczen
author_sort Kinga Kwiecinska
collection DOAJ
description Abstract Background It has been shown that approximately half of survivors of childhood acute lymphoblastic leukemia (ALL) have symptomatic late effects (LE) that may be severe or life-threatening. The aim of our study was to assess the health status of childhood ALL survivors after over 10 years of follow-up and to assess its relationships with gene polymorphisms, numbers and types of LEs, as well as with intensity of chemotherapy and cranial radiotherapy (CRT). Methods We conducted a telephone survey in 125 ALL survivors (median time from completion of treatment was 12 years) and compared the results with those obtained in our previous study. Most of the patients were followed-up by local providers. Results The prevalence of LEs of approximately 50% was similar in both study groups. More than one LE was found in almost 25% of patients. Endocrine LEs were less frequent than in our previous study (44% vs 22%), probably due to underdiagnosis. The prevalence of hepatitis B/C decreased from 30%/50 to 18% (counted together), and prevalence of neurologic LEs decreased from 18 to 6%. The increase in the rate of second malignancies was not significant (2% vs. 3%). Sixty four percent of patients continued their education at the time of the study. Approximately 51% of ALL survivors who have completed their education by the time of the study had no permanent employment, including 4 mothers of infants and 3 persons qualified for a disability living allowance. These employment problems may have been due to cognitive impairment. The offspring of the ALL survivors included 11 children, all of them healthy. Further analysis showed higher prevalence of hepatitis in patients treated with CRT (p = 0.0001). Genetic studies revealed higher prevalence of hepatitis in patients homozygous for the rs9939609A variant of the FTO gene compared with other patients (p = 0.03). Moreover, wild-type rs1137101 polymorphism (Q223R) of the and leptin receptor gene was more frequent in patients with psychological LEs (p = 0.03). Conclusions The prevalence of LEs in ALL survivors is of key importance. The transition of childhood ALL survivors from pediatric to adult care should be urgently improved to maintain continued follow-up provide high-quality care. Trial registration Bioethics Committee of the Jagiellonian University approved the study protocol. Registration number: KBET/113/B/2006.
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spelling doaj.art-fa511bd9e0a64e0aba344d33c97314332022-12-22T01:44:59ZengBMCItalian Journal of Pediatrics1824-72882018-08-014411810.1186/s13052-018-0526-5Late effects in survivors of childhood acute lymphoblastic leukemia in the context of selected gene polymorphismsKinga Kwiecinska0Wojciech Strojny1Danuta Pietrys2Miroslaw Bik-Multanowski3Maciej Siedlar4Walentyna Balwierz5Szymon Skoczen6Department of Oncology and Hematology, Institute of Pediatrics, Jagiellonian University Medical CollegeDepartment of Oncology and Hematology, Institute of Pediatrics, Jagiellonian University Medical CollegeDepartment of Oncology and Hematology, Institute of Pediatrics, Jagiellonian University Medical CollegeDepartment of Medical Genetics, Institute of Pediatrics, Jagiellonian University Medical CollegeDepartment of Clinical Immunology, Chair of Clinical Immunology and Transplantation, Institute of Pediatrics, Jagiellonian University Medical CollegeDepartment of Oncology and Hematology, Institute of Pediatrics, Jagiellonian University Medical CollegeDepartment of Oncology and Hematology, Institute of Pediatrics, Jagiellonian University Medical CollegeAbstract Background It has been shown that approximately half of survivors of childhood acute lymphoblastic leukemia (ALL) have symptomatic late effects (LE) that may be severe or life-threatening. The aim of our study was to assess the health status of childhood ALL survivors after over 10 years of follow-up and to assess its relationships with gene polymorphisms, numbers and types of LEs, as well as with intensity of chemotherapy and cranial radiotherapy (CRT). Methods We conducted a telephone survey in 125 ALL survivors (median time from completion of treatment was 12 years) and compared the results with those obtained in our previous study. Most of the patients were followed-up by local providers. Results The prevalence of LEs of approximately 50% was similar in both study groups. More than one LE was found in almost 25% of patients. Endocrine LEs were less frequent than in our previous study (44% vs 22%), probably due to underdiagnosis. The prevalence of hepatitis B/C decreased from 30%/50 to 18% (counted together), and prevalence of neurologic LEs decreased from 18 to 6%. The increase in the rate of second malignancies was not significant (2% vs. 3%). Sixty four percent of patients continued their education at the time of the study. Approximately 51% of ALL survivors who have completed their education by the time of the study had no permanent employment, including 4 mothers of infants and 3 persons qualified for a disability living allowance. These employment problems may have been due to cognitive impairment. The offspring of the ALL survivors included 11 children, all of them healthy. Further analysis showed higher prevalence of hepatitis in patients treated with CRT (p = 0.0001). Genetic studies revealed higher prevalence of hepatitis in patients homozygous for the rs9939609A variant of the FTO gene compared with other patients (p = 0.03). Moreover, wild-type rs1137101 polymorphism (Q223R) of the and leptin receptor gene was more frequent in patients with psychological LEs (p = 0.03). Conclusions The prevalence of LEs in ALL survivors is of key importance. The transition of childhood ALL survivors from pediatric to adult care should be urgently improved to maintain continued follow-up provide high-quality care. Trial registration Bioethics Committee of the Jagiellonian University approved the study protocol. Registration number: KBET/113/B/2006.http://link.springer.com/article/10.1186/s13052-018-0526-5Late effectsAcute lymphoblastic leukemiaGene polymorphisms
spellingShingle Kinga Kwiecinska
Wojciech Strojny
Danuta Pietrys
Miroslaw Bik-Multanowski
Maciej Siedlar
Walentyna Balwierz
Szymon Skoczen
Late effects in survivors of childhood acute lymphoblastic leukemia in the context of selected gene polymorphisms
Italian Journal of Pediatrics
Late effects
Acute lymphoblastic leukemia
Gene polymorphisms
title Late effects in survivors of childhood acute lymphoblastic leukemia in the context of selected gene polymorphisms
title_full Late effects in survivors of childhood acute lymphoblastic leukemia in the context of selected gene polymorphisms
title_fullStr Late effects in survivors of childhood acute lymphoblastic leukemia in the context of selected gene polymorphisms
title_full_unstemmed Late effects in survivors of childhood acute lymphoblastic leukemia in the context of selected gene polymorphisms
title_short Late effects in survivors of childhood acute lymphoblastic leukemia in the context of selected gene polymorphisms
title_sort late effects in survivors of childhood acute lymphoblastic leukemia in the context of selected gene polymorphisms
topic Late effects
Acute lymphoblastic leukemia
Gene polymorphisms
url http://link.springer.com/article/10.1186/s13052-018-0526-5
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