Abstract

BackgroundTumours of the central nervous system are the second most common group of childhood cancers in 0–14 year olds (24% of total cancers) and represent a major diagnostic group in 15–24 year olds. The pilot case–control study aimed to establish methodologies for a future comprehensive aetiological investigation among children and young adults.MethodsEligible cases were newly diagnosed with an intracranial tumour of neuroepithelial tissue aged 0–24 years. The pilot recruited patients through Leeds and Manchester Principal Treatment Centres. Controls were drawn from general practice lists. Controls were frequency matched by age and gender.ResultsWe interviewed 49 cases and 78 controls comprising 85% of the target sample size. Response rates were 52% for cases and 32% for controls. Completion of the questionnaire was successful, with a very small proportion of missing data being reported (5-10%). The age distribution of cases and controls was similar with around three-quarters of interviewed subjects aged 0–14. Half of cases and almost two-thirds of controls reported using a mobile phone with the majority starting between 10–14 years of age. Prevalence of breastfeeding was lower in cases than controls (Odds Ratio 0.4; 95% CI 0.2-1.2), whilst cases were more likely to be delivered by caesarean section (OR 1.6; 95% CI 0.6-4.4). Cases were significantly more likely to have a birthweight > 3.5 kg compared to controls. Cases were also more likely to come from a family with 3 or more siblings than controls (OR 3.0; 95% CI 0.7-13.6). The majority of participants (>80%) were in favour of taking either blood or saliva to aid molecular epidemiological research.ConclusionsSuccessful methods were established for identifying and recruiting a high proportion of case subjects, exploiting strong links with the clinical teams at the treatment centres. Control procedures proved more difficult to implement. However, working closely with national clinical and professional research networks will enable improved control identification and recruitment, with good prospects for collecting biological samples in the future.

Highlights

  • Tumours of the central nervous system are the second most common group of childhood cancers in 0–14 year olds (24% of total cancers) and represent a major diagnostic group in 15–24 year olds

  • Tumours of the central nervous system (CNS) are the second most common group of childhood cancers comprising a quarter of all malignancies in patients aged 0–14 years with approximately 350 children diagnosed each year in the UK [1]

  • Of the 12 cases and 25 controls who responded about their willingness to provide a blood or saliva sample to carry out future biological research, all cases and controls said they would agree to provide saliva whilst 89% of cases and 81% of controls would agree to provide a blood sample. Through this pilot study, we have demonstrated that by working closely as a multidisciplinary team, recruitment of participants diagnosed with brain tumours is feasible as part of a ‘case-control’ design to address aetiological questions, despite the huge challenges facing these young people shortly after diagnosis

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Summary

Introduction

Tumours of the central nervous system are the second most common group of childhood cancers in 0–14 year olds (24% of total cancers) and represent a major diagnostic group in 15–24 year olds. Tumours of the central nervous system (CNS) are the second most common group of childhood cancers comprising a quarter of all malignancies in patients aged 0–14 years with approximately 350 children diagnosed each year in the UK [1]. CNS tumours represent a major diagnostic group in teenagers and young adults. Childhood CNS tumours have been linked to residential pesticide exposure, traffic pollution, and parental occupations [8,9,10,11]. There is accumulating evidence of links between infections and CNS tumours in the young. Supportive evidence for the involvement of infections comes from analyses of spacetime clustering of incident cases, geographical and demographic variations in incidence and population mixing [12,13,14,15,16]

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