Abstract
Objective
We have analysed incidence and survival trends of children and adolescents with leukaemia registered in Spanish population-based cancer registries during the period 1983–2007.
Methods
Childhood and adolescent leukaemia cases were drawn from the 11 Spanish population-based cancer registries. For survival, registries with data for the period 1991–2005 and follow-up until 31-12-2010 were included. Overall incidence trends were evaluated using joinpoint analysis. Observed survival rates were estimated using Kaplan–Meier, and trends were tested using the log-rank test.
Results
Based on 2606 cases (2274 children and 332 adolescents), the overall age-adjusted incidence rate (ASRw) of leukaemia was 47.9 cases per million child-years in children and 23.8 in adolescents. The ASRw of leukaemia increased with an annual percentage change of 9.6 % (95 % CI: 2.2–17.6) until 1990 followed by a stabilisation of rates. In adolescents, incidence did not increase. Five-year survival increased from 66 % in 1991–1995 to 76 % in 2001–2005. By age, survival was dramatically lower in infants (0) and adolescents (15−19) than in the other age groups and no improvement was observed. In both children and adolescents, differences in 5-year survival rates among major subgroups of leukaemias were significant.
Conclusions
The increasing incidence trends observed in childhood leukaemias during the study period were confined to the beginning of the period. Remarkable improvements in survival have been observed in Spanish children with leukaemias. However, this improvement was not observed in infants and adolescents.
Similar content being viewed by others
References
Coebergh JW, Reedijk AM, de Vries E, Martos C, Jakab Z, Steliarova-Foucher E, et al. Leukaemia incidence and survival in children and adolescents in Europe during 1978–1997. Report from the Automated Childhood Cancer Information System project. Eur J Cancer. 2006;42(13):2019–36.
Linet MS, Ries LA, Smith MA, Tarone RE, Devesa SS. Cancer surveillance series: recent trends in childhood cancer incidence and mortality in the United States. J Natl Cancer Inst. 1999;91(12):1051–8.
Marcos-Gragera R, Cervantes-Amat M, Vicente ML, de Sanjose S, Guallar E, Godoy C, et al. Population-based incidence of childhood leukaemias and lymphomas in Spain (1993–2002). Eur J Cancer Prev. 2010;19(4):247–55.
Maule MM, Zuccolo L, Magnani C, Pastore G, Dalmasso P, Pearce N, et al. Bayesian methods for early detection of changes in childhood cancer incidence: trends for acute lymphoblastic leukaemia are consistent with an infectious aetiology. Eur J Cancer. 2006;42(1):78–83.
Pollan M, Lopez-Abente G, Ardanaz E, Moreo P, Moreno C, Vergara A, et al. Childhood cancer incidence in Zaragoza and Navarre (Spain): 1973–1987. Eur J Cancer. 1997;33(4):616–23.
Hjalgrim LL, Rostgaard K, Schmiegelow K, Soderhall S, Kolmannskog S, Vettenranta K, et al. Age- and sex-specific incidence of childhood leukaemia by immunophenotype in the Nordic countries. J Natl Cancer Inst. 2003;95(20):1539–44.
Dreifaldt AC, Carlberg M, Hardell L. Increasing incidence rates of childhood malignant diseases in Sweden during the period 1960–1998. Eur J Cancer. 2004;40(9):1351–60.
McNally RJ, Birch JM, Taylor GM, Eden OB. Incidence of childhood precursor B-cell acute lymphoblastic leukaemia in north-west England. Lancet. 2000;356(9228):485–6.
McNally RJ, Alexander FE, Birch JM. Space-time clustering analyses of childhood acute lymphoblastic leukaemia by immunophenotype. Br J Cancer. 2002;87(5):513–5.
van Duijn CM, van Steensel-Moll HA, Coebergh JW, van Zanen GE. Risk factors for childhood acute non-lymphocytic leukaemia: an association with maternal alcohol consumption during pregnancy? Cancer Epidemiol Biomarkers Prev. 1994;3(6):457–60.
Ma X, Metayer C, Does MB, Buffler PA. Maternal pregnancy loss, birth characteristics, and childhood leukaemia (United States). Cancer Causes Control. 2005;16(9):1075–83.
Inaba H, Greaves M, Mullighan CG. Acute lymphoblastic leukaemia. Lancet. 2013;381(9881):1943–55.
Brenner H, Coebergh J, Parkin D, Izarzugaza I, Clavel J, Arndt V, et al. Up-to-date monitoring of childhood cancer long-term survival in Europe: leukaemias and lymphomas. Ann Oncol 2007;18(9):1561–68.
Peris-Bonet R, Salmeron D, Martinez-Beneito MA, Galceran J, Marcos-Gragera R, Felipe S, et al. Childhood cancer incidence and survival in Spain. Ann Oncol. 2010;21(Suppl 3):iii103–10.
Navarro C, Martos C, Ardanaz E, Galceran J, Izarzugaza I, Peris-Bonet R, et al. Population-based cancer registries in Spain and their role in cancer control. Ann Oncol. 2010;21(Suppl 3):iii3–13.
Frit A, Jack A, Percy C, Shanmugaratnam K, Sobin L, Parkin M, et al. International classification of diseases for oncology. 3rd ed. Geneva: World Health Organisation; 2000.
Steliarova-Foucher E, Stiller C, Lacour B, Kaatsch P. International classification of childhood cancer, third edition. Cancer. 2005;103(7):1457–67.
Ferlay J, Burkharc C, Whelan S, Parkin DM. Check and conversion programs for cancer registries (IARC/IACR Tools for Cancer Registries). IARC Technical Report No 42 Lyon, 2005. Available from: URL: http://www.iacr.com.fr/iacr_iarccrgtools.htm. Accessed 18 Jul 2016.
Instituto Nacional de Estadística. http://www.ine.es/dynt3/inebase/index.htm?padre=1949&capsel=1951. Accessed 18 Jul 2016.
Stiller CA, Marcos-Gragera R, Ardanaz E, Pannelli F, Almar ME, Canada MA, et al. Geographical patterns of childhood cancer incidence in Europe, 1988-1997. Report from the Automated Childhood Cancer Information System project. Eur J Cancer. 2006;42(13):1952–60.
Kim HJ, Fay MP, Feuer EJ, Midthune DN. Permutation tests for joinpoint regression with applications to cancer rates. Stat Med. 2000;19(3):335–51.
Joinpoint Regression Program [computer program]. Version 4.1.1. 2014. http://surveillance.cancer.gov/joinpoint/revisions.html. Accessed 18 Jul 2016.
Esteve J, Benhamou E, Croasdale M, Raymond L. Relative survival and the estimation of net survival: elements for further discussion. Stat Med. 1990;9(5):529–38.
Gatta G, Botta L, Rossi S, Aareleid T, Bielska-Lasota M, Clavel J, et al. Childhood cancer survival in Europe 1999–2007: results of EUROCARE-5–a population-based study. Lancet Oncol. 2014;15(1):35–47.
Peto R, Pike MC, Armitage P, Breslow NE, Cox DR, Howard SV, et al. Design and analysis of randomized clinical trials requiring prolonged observation of each patient. II. analysis and examples. Br J Cancer. 1977;35(1):1–39.
Stiller CA. Childhood cancer in Britain. New York: Oxford University Press; 2007.
Coebergh JW, der Does-van Van, den Berg, van Wering ER, van Steensel-Moll HA, Valkenburg HA, van’t Veer MB, et al. Childhood leukaemia in The Netherlands, 1973–1986: temporary variation of the incidence of acute lymphocytic leukaemia in young children. Br J Cancer. 1989;59(1):100–5.
Dalmasso P, Pastore G, Zuccolo L, Maule MM, Pearce N, Merletti F, et al. Temporal trends in the incidence of childhood leukaemia, lymphomas and solid tumors in north-west Italy, 1967–2001. A report of the Childhood Cancer Registry of Piedmont. Haematologica. 2005;90(9):1197–204.
Magnani C, Dalmasso P, Pastore G, Terracini B, Martuzzi M, Mosso ML, et al. Increasing incidence of childhood leukaemia in Northwest Italy, 1975–98. Int J Cancer. 2003;105(4):552–7.
Shah A, Coleman MP. Increasing incidence of childhood leukaemia: a controversy re-examined. Br J Cancer. 2007;97(7):1009–12.
McWhirter WR, Dobson C, Ring I. Childhood cancer incidence in Australia, 1982–1991. Int J Cancer. 1996;65(1):34–8.
Xie Y, Davies SM, Xiang Y, Robison LL, Ross JA. Trends in leukaemia incidence and survival in the United States (1973–1998). Cancer. 2003;97(9):2229–35.
Desandes E, Clavel J, Berger C, Bernard JL, Blouin P, De LL, et al. Cancer incidence among children in France, 1990–1999. Pediatr Blood Cancer. 2004;43(7):749–57.
Steliarova-Foucher E, Stiller C, Kaatsch P, Berrino F, Coebergh JW. Trends in childhood cancer incidence in Europe, 1970–99. Lancet. 2005;365(9477):2088.
Freedman MH, Estrov Z, Chan HS. Juvenile chronic myelogenous leukaemia. Am J Pediatr Hematol Oncol. 1988;10(3):261–7.
Niemeyer CM, Kratz CP. Paediatric myelodysplastic syndromes and juvenile myelomonocytic leukaemia: molecular classification and treatment options. Br J Haematol. 2008;140(6):610–24.
Niemeyer CM, Baumann I. Myelodysplastic syndrome in children and adolescents. Semin Hematol. 2008;45(1):60–70.
Niemeyer CM, Kang MW, Shin DH, Furlan I, Erlacher M, Bunin NJ, et al. Germline CBL mutations cause developmental abnormalities and predispose to juvenile myelomonocytic leukaemia. Nat Genet. 2010;42(9):794–800.
Kramarova E, Stiller CA. The international classification of childhood cancer. Int J Cancer. 1996;68(6):759–65.
Li J, Thompson TD, Miller JW, Pollack LA, Stewart SL. Cancer incidence among children and adolescents in the United States, 2001–2003. Pediatrics. 2008;121(6):e1470–7.
Lacour B, Guyot-Goubin A, Guissou S, Bellec S, Desandes E, Clavel J. Incidence of childhood cancer in France: National Children Cancer Registries, 2000–2004. Eur J Cancer Prev. 2010;19(3):173–81.
Kaatsch P, Spix C. German Childhood Cancer Registry-annual report 2011 (1980–2010). Institute for Medical Biostatistics, Epidemiology and Informatics (IMBEI), University Medical Center of the Johannes Gutenberg University Mainz. 2012. http://www.kinderkrebsregister.de.
AIRTUM Working Group. Italian cancer figures, report 2012: Cancer in children and adolescents 21. Epidemiol Prev 2013;37(1)Suppl 1:1–296.
UK: Childhood Cancer Research Group. Survival from Childhood Cancer, Great Britain, 1971–2005. 2015. http://www.ccrg.ox.ac.uk/datasets/survivalrates.shtml. Accessed 18 July 2016.
Gatta G, Zigon G, Capocaccia R, Coebergh JW, Desandes E, Kaatsch P, et al. Survival of European children and young adults with cancer diagnosed 1995–2002. Eur J Cancer. 2009;45(6):992–1005.
Gatta G, Luksch R, Coleman MP, Corazziari I. Survival from acute non-lymphocytic leukaemia (ANLL) and chronic myeloid leukaemia (CML) in European children since 1978: a population-based study. Eur J Cancer. 2001;37(6):695–702.
Coebergh JW, Pastore G, Gatta G, Corazziari I, Kamps W. Variation in survival of European children with acute lymphoblastic leukaemia, diagnosed in 1978–1992: the EUROCARE study. Eur J Cancer. 2001;37(6):687–94.
Pastore G, Viscomi S, Gerov GL, Terracini B, Madon E, Magnani C. Population-based survival after childhood lymphoblastic leukaemia in time periods corresponding to specific clinical trials from 1979 to 1998–a report from the Childhood Cancer Registry of Piedmont (Italy). Eur J Cancer. 2003;39(7):952–60.
Stiller CA, Eatock EM. Patterns of care and survival for children with acute lymphoblastic leukaemia diagnosed between 1980 and 1994. Arch Dis Child. 1999;81(3):202–8.
Dores GM, Devesa SS, Curtis RE, Linet MS, Morton LM. Acute leukaemia incidence and patient survival among children and adults in the United States, 2001–2007. Blood. 2012;119(1):34–43.
Pieters R, Schrappe M, De LP, Hann I, De RG, Felice M, et al. A treatment protocol for infants younger than 1 year with acute lymphoblastic leukaemia (Interfant-99): an observational study and a multicentre randomised trial. Lancet. 2007;370(9583):240–50.
Hunger SP, Lu X, Devidas M, Camitta BM, Gaynon PS, Winick NJ, et al. Improved survival for children and adolescents with acute lymphoblastic leukaemia between 1990 and 2005: a report from the children’s oncology group. J Clin Oncol. 2012;30(14):1663–9.
Gatta G, Rossi S, Foschi R, Trama A, Marcos-Gragera R, Pastore G, et al. Survival and cure trends for European children, adolescents and young adults diagnosed with acute lymphoblastic leukaemia from 1982 to 2002. Haematologica. 2013;98(5):744–52.
Ribera JM, Ribera J, Genesca E. Treatment of adolescent and young adults with acute lymphoblastic leukaemia. Mediterr J Hematol Infect Dis. 2014;6(1):e2014052.
Patrick K, Wade R, Goulden N, Mitchell C, Moorman AV, Rowntree C, et al. Outcome for children and young people with Early T-cell precursor acute lymphoblastic leukaemia treated on a contemporary protocol, UKALL 2003. Br J Haematol. 2014;166(3):421–4.
Pui CH, Schrappe M, Ribeiro RC, Niemeyer CM. Childhood and adolescent lymphoid and myeloid leukaemia. Hematology Am Soc Hematol Educ Progr. 2004;1:118–45.
Falini B, Mecucci C, Tiacci E, Alcalay M, Rosati R, Pasqualucci L, et al. Cytoplasmic nucleophosmin in acute myelogenous leukaemia with a normal karyotype. N Engl J Med. 2005;352(3):254–66.
Gale RE, Hills R, Kottaridis PD, Srirangan S, Wheatley K, Burnett AK, et al. No evidence that FLT3 status should be considered as an indicator for transplantation in acute myeloid leukaemia (AML): an analysis of 1135 patients, excluding acute promyelocytic leukaemia, from the UK MRC AML10 and 12 trials. Blood. 2005;106(10):3658–65.
Burnett AK, Wheatley K, Goldstone AH, Stevens RF, Hann IM, Rees JH, et al. The value of allogeneic bone marrow transplant in patients with acute myeloid leukaemia at differing risk of relapse: results of the UK MRC AML 10 trial. Br J Haematol. 2002;118(2):385–400.
Ofran Y, Rowe JM. Acute myeloid leukaemia in adolescents and young adults: challenging aspects. Acta Haematol. 2014;132(3–4):292–7.
Hijiya N, Schultz KR, Metzler M, Millot F, Suttorp M. Pediatric chronic myeloid leukaemia is a unique disease that requires a different approach. Blood. 2016;127(4):748–57.
Kantarjian H, O’Brien S, Jabbour E, Garcia-Manero G, Quintas-Cardama A, Shan J, et al. Improved survival in chronic myeloid leukaemia since the introduction of imatinib therapy: a single-institution historical experience. Blood. 2012;119(9):1981–7.
de la Fuente J, Baruchel A, Biondi A, de Bont E, Dresse MF, Suttorp M, et al. Managing children with chronic myeloid leukaemia (CML): recommendations for the management of CML in children and young people up to the age of 18 years. Br J Haematol. 2014;167(1):33–47.
Curado MP, Edwards B, Shin HR, Storm H, Ferlay J, Heanue M, et al. Cancer incidence in five continents, vol. IX. Lyon: IARC Scientific Publications No. 160; 2007.
Jaffe ES, Harris NL, Stein H, Vardiman J. World Health Organisation classification of tumours. Pathology and genetics of tumours of haematopoietic and lymphoid tissues. Lyon: IARC Press; 2001.
Swerdlow SH, Campo E, Harris NL, Jaffe ES, Pileri SA, Stein H, et al. World Health Organisation classification of tumours of haematopoietic and lymphoid tissues. 4th ed. Lyon: WHO; 2008.
Rossig C, Juergens H, Schrappe M, Moericke A, Henze G, von Stackelberg A, et al. Effective childhood cancer treatment: the impact of large scale clinical trials in Germany and Austria. Pediatr Blood Cancer. 2013;60(10):1574–81.
Vassal G, Fitzgerald E, Schrappe M, Arnold F, Kowalczyk J, Walker D, et al. Challenges for children and adolescents with cancer in Europe: the SIOP-Europe agenda. Pediatr Blood Cancer. 2014;61(9):1551–7.
Warsaw: SIOP-Europe 2. European standards of care for children with cancer. https://www.siope.eu/european-research-and-standards/standards-of-care-in-paediatric-oncology/2016. Accessed 18 Jul 2016.
The SIOPE Strategic Plan. SIOP-Europe, 2015. https://www.siope.eu/SIOPE_StrategicPlan2015/. Accessed 01 Jul 2016.
Acknowledgments
The authors thank G. Gatta, L. Botta and R. Capocaccia of EUROCARE for their help with the standardisation of survival in Spain and all the staffs of the cancer registries participating in the study and Elena Pardo for her support with statistical data.
Author information
Authors and Affiliations
Corresponding author
Ethics declarations
Conflict of interest
The authors declare that they have no conflict of interest.
Funding
This work was supported by FIS (PI 061742) and (PI 10/01617); Enriqueta Villavecchia Foundation; Red de Investigación temática en cancer RD12/0036/0056 (Rafael Marcos-Gragera) and RD12/0036/0053 (R Peris-Bonet and Jaume Galceran).
Additional information
R. Marcos-Gragera and J. Galceran contributed equally to this study.
Rights and permissions
About this article
Cite this article
Marcos-Gragera, R., Galceran, J., Martos, C. et al. Incidence and survival time trends for Spanish children and adolescents with leukaemia from 1983 to 2007. Clin Transl Oncol 19, 301–316 (2017). https://doi.org/10.1007/s12094-016-1531-z
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s12094-016-1531-z