Little is known about the possible causes of bone tumours in children and young adults. A recent study of national (UK) data has found that there is geographical variation for osteosarcomas diagnosed in children aged 0-14 years. This lends support to the possibility that certain environmental factors may be involved, particularly those that show geographical variation. Another recent study has found that bone sarcoma patients who were treated in specialist hospitals had better survival rates than patients who were treated in non-specialist hospitals. It is not known whether survival is related to socio-economic factors.

The main aim of the proposed study is to identify focused scientific hypotheses for further investigation. These hypotheses will be related to factors that may contribute to the development of bone tumours and factors that may be important for improved chance of survival after diagnosis of a bone tumour.

There are two parts to this study. The first part proposes carrying out a thorough review of what is currently known about the incidence, survival and causes of bone tumours in young people (aged 0-49 years). The second part proposes a set of analyses of population-based data from the North of England and the West Midlands.

Such analyses of population-based data are a cost-effective way of generating new hypotheses. This approach has been successfully used in studying childhood leukaemia and led to the development of plausible scientific hypotheses. Additionally factors that may affect survival will be studied.

The data analysis study will concentrate on the three regions from the North of England (the Northern Region, Yorkshire, the North West) and also the West Midlands. Data will be obtained from five sources: the Northern Region Young Persons’ Malignant Disease Registry (NRYPMDR), the Yorkshire Specialist Registry of Cancer in Children and Young People (YSRCCYP), the Northern and Yorkshire Cancer Registry and Information Service (NYCRIS), the West Midlands Regional Children’s Tumour Registry (WMRCTR)and the Manchester Children’s Tumour Registry (MCTR). All registries are population-based and are periodically cross-checked with data from regional and national cancer registries to ensure information is as accurate and complete as possible. Data for the Yorkshire and Northern regions will be available for 0-24 year olds from the NRYPMDR (from 1968 onwards) and the YSRCCYP (0-29 year olds from 1990 onwards and 0-14 year olds from 1974 onwards) and for 25-49 year olds from the NYCRIS (from 1975 onwards). Data for North West England and the West Midlands will be available for 0-14 year olds from the MCTR (from 1954 onwards) and the WMRCTR (from 1957 onwards). This novel piece of work aimed at children and uniquely young adults covers the entire age-incidence peaks for bone tumours.

Incidence patterns, trends and survival will all be described. Statistical modelling will be used to investigate the potential effect of socio-economic deprivation. Data on 0-24 year olds from the NRYPMDR will allow both the place of residence at birth and at diagnosis to be analysed. This will allow a distinction to be made between the possible effect of earlier and later exposures to environmental agents. Survival patterns will be analysed to determine whether there are differences in the survival between patients of different ages, gender and social class.

Findings from this study will be presented at scientific meetings and published in peer-reviewed journals. It is anticipated that this study will provide clues for investigation in a much larger study.

Dr Richard McNally, Newcastle University