Cost-effectiveness of screening with contrast enhanced magnetic resonance imaging vs X-ray mammography of women at a high familial risk of breast cancer.

Contrast enhanced magnetic resonance imaging (CE MRI) is the most sensitive tool for screening women who are at high familial risk of breast cancer. Our aim in this study was to assess the cost-effectiveness of X-ray mammography (XRM), CE MRI or both strategies combined. In total, 649 women were enrolled in the MARIBS study and screened with both CE MRI and mammography resulting in 1881 screens and 1-7 individual annual screening events. Women aged 35-49 years at high risk of breast cancer, either because they have a strong family history of breast cancer or are tested carriers of a BRCA1, BRCA2 or TP53 mutation or are at a 50% risk of having inherited such a mutation, were recruited from 22 centres and offered annual MRI and XRM for between 2 and 7 years. Information on the number and type of further investigations was collected and specifically calculated unit costs were used to calculate the incremental cost per cancer detected. The numbers of cancer detected was 13 for mammography, 27 for CE MRI and 33 for mammography and CE MRI combined. In the subgroup of BRCA1 (BRCA2) mutation carriers or of women having a first degree relative with a mutation in BRCA1 (BRCA2) corresponding numbers were 3 (6), 12 (7) and 12 (11), respectively. For all women, the incremental cost per cancer detected with CE MRI and mammography combined was pound28 284 compared to mammography. When only BRCA1 or the BRCA2 groups were considered, this cost would be reduced to pound11 731 (CE MRI vs mammography) and pound15 302 (CE MRI and mammography vs mammography). Results were most sensitive to the unit cost estimate for a CE MRI screening test. Contrast-enhanced MRI might be a cost-effective screening modality for women at high risk, particularly for the BRCA1 and BRCA2 subgroups. Further work is needed to assess the impact of screening on mortality and health-related quality of life.

Newborn screening for congenital heart defects: a systematic review and cost-effectiveness analysis.

OBJECTIVES: To provide evidence to inform policy decisions about the most appropriate newborn screening strategy for congenital heart defects, identifying priorities for future research that might reduce important uncertainties in the evidence base for such decisions. DATA SOURCES: Electronic databases. Groups of parents and health professionals. REVIEW METHODS: A systematic review of the published medical literature concerning outcomes for children with congenital heart defects was carried out. A decision analytic model was developed to assess the cost-effectiveness of alternative screening strategies for congenital heart defects relevant to the UK. A further study was then carried out using a self-administered anonymous questionnaire to explore the perspectives of parents and health professionals towards the quality of life of children with congenital heart defects. The findings from a structured review of the medical literature regarding parental experiences were linked with those from a focus group of parents of children with congenital heart defects. RESULTS: Current newborn screening policy comprises a clinical examination at birth and 6 weeks, with specific cardiac investigations for specified high-risk children. Routine data are lacking, but under half of affected babies, not previously identified antenatally or because of symptoms, are identified by current newborn screening. There is evidence that screen-positive infants do not receive timely management. Pulse oximetry and echocardiography, in addition to clinical examination, are alternative newborn screening strategies but their cost-effectiveness has not been adequately evaluated in a UK setting. In a population of 100,000 live-born infants, the model predicts 121 infants with life-threatening congenital heart defects undiagnosed at screening, of whom 82 (68%) and 83 (69%) are detected by pulse oximetry and screening echocardiography, respectively, but only 39 (32%) by clinical examination alone. Of these, 71, 71 and 34, respectively, receive a timely diagnosis. The model predicts 46 (0.5%) false-positive screening diagnoses per 100,000 infants with clinical examination, 1168 (1.3%) with pulse oximetry and 4857 (5.4%) with screening echocardiography. The latter includes infants with clinically non-significant defects. Total programme costs are predicted of pound 300,000 for clinical examination, pound 480,000 for pulse oximetry and pound 3.54 million for screening echocardiography. The additional cost per additional timely diagnosis of life-threatening congenital heart defects ranges from pound 4900 for pulse oximetry to pound 4.5 million for screening echocardiography. Including clinically significant congenital heart defects gives an additional cost per additional diagnosis of pound 1500 for pulse oximetry and pound 36,000 for screening echocardiography. Key determinants for cost-effectiveness are detection rates for pulse oximetry and screening echocardiography. Parents and health professionals place similar values on the quality of life outcomes of children with congenital heart defects and both are more averse to neurological than to cardiac disability. Adverse psychosocial effects for parents are focused around poor management and/or false test results. CONCLUSIONS: Early detection through newborn screening potentially can improve the outcome of congenital heart defects; however the current programme performs poorly, and lacks monitoring of quality assurance, performance management and longer term outcomes. Pulse oximetry is a promising alternative newborn screening strategy but further evaluation is needed to obtain more precise estimates of test performance and to inform optimal timing, diagnostic and management strategies. Although screening echocardiography is associated with the highest detection rate, it is the most costly strategy and has a 5% false-positive rate. Improving antenatal detection of congenital heart defects increases the cost per timely postnatal diagnosis afforded by any newborn screening strategy but does not alter the relative effects of the strategies. An improvement of timely management of screen positive infants is essential. Further research is required to refine the detection rate and other aspects of pulse oximetry, to evaluate antenatal screening strategies more directly, and to investigate the psychosocial effects of newborn screening for congenital heart defects.

Screening with magnetic resonance imaging and mammography of a UK population at high familial risk of breast cancer: a prospective multicentre cohort study (MARIBS).

BACKGROUND: Women genetically predisposed to breast cancer often develop the disease at a young age when dense breast tissue reduces the sensitivity of X-ray mammography. Our aim was, therefore, to compare contrast enhanced magnetic resonance imaging (CE MRI) with mammography for screening. METHODS: We did a prospective multicentre cohort study in 649 women aged 35-49 years with a strong family history of breast cancer or a high probability of a BRCA1, BRCA2, or TP53 mutation. We recruited participants from 22 centres in the UK, and offered the women annual screening with CE MRI and mammography for 2-7 years. FINDINGS: We diagnosed 35 cancers in the 649 women screened with both mammography and CE MRI (1881 screens): 19 by CE MRI only, six by mammography only, and eight by both, with two interval cases. Sensitivity was significantly higher for CE MRI (77%, 95% CI 60-90) than for mammography (40%, 24-58; p=0.01), and was 94% (81-99) when both methods were used. Specificity was 93% (92-95) for mammography, 81% (80-83) for CE MRI (p<0.0001), and 77% (75-79) with both methods. The difference between CE MRI and mammography sensitivities was particularly pronounced in BRCA1 carriers (13 cancers; 92%vs 23%, p=0.004). INTERPRETATION: Our findings indicate that CE MRI is more sensitive than mammography for cancer detection. Specificity for both procedures was acceptable. Despite a high proportion of grade 3 cancers, tumours were small and few women were node positive. Annual screening, combining CE MRI and mammography, would detect most tumours in this risk group.

Provision, uptake and cost of cardiac rehabilitation programmes: improving services to under-represented groups.

OBJECTIVES: To estimate UK need for outpatient cardiac rehabilitation, current provision and identification of patient groups not receiving services. To conduct a systematic review of literature on methods to improve uptake and adherence to cardiac rehabilitation. To estimate cost implications of increasing uptake of cardiac rehabilitation. DATA SOURCES: Hospital Episode Statistics (England). Hospital Inpatient Systems (Northern Ireland). Patients Episode Database for Wales. British Association for Cardiac Rehabilitation/British Heart Foundation surveys. Cardiac rehabilitation centres. Patients from general hospitals. Electronic databases. REVIEW METHODS: The study analysed hospital discharge statistics to ascertain the population need for outpatient cardiac rehabilitation in the UK. Surveys of cardiac rehabilitation programmes were conducted to determine UK provision, uptake and audit activity, and to identify local interventions to improve uptake. Data were also examined from a trial estimating eligibility for cardiac rehabilitation and non-attendance. A systematic review of interventions to improve patient uptake, adherence and professional compliance in cardiac rehabilitation was conducted. Estimated costs of improving uptake were identified from national survey, systematic review and sampled cardiac rehabilitation programmes. RESULTS: In England, Wales and Northern Ireland nearly 146,000 patients discharged from hospital with primary diagnosis of acute myocardial infarction, unstable angina or following revascularisation were potentially eligible for cardiac rehabilitation. In England in 2000, 45-67% of these patients were referred, with 27-41% attending outpatient cardiac rehabilitation. If all discharge diagnoses of ischaemic heart disease were considered, nearly 299,000 patients would be potentially eligible and in England rates of attendance and referral would be 22-33% and 13-20% respectively. Rates of referral and attendance were similar in Wales, but somewhat lower in Northern Ireland. It was found that referral and attendance of older people and women at cardiac rehabilitation tended to be low. It was also suggested that patients from ethnic minorities and those with angina or heart failure were less likely to be referred to or join programmes. A wide range of local interventions suggested awareness of the problem of uptake. In an NHS-funded randomised controlled trial, possibly representing more optimal protocol-led care, medical and nursing staff identified 73-81% of patients with acute myocardial infarction as eligible for cardiac rehabilitation. Excluded patients tended to be older with more severe presentation of cardiac disease. Experiences of patients suggested that uptake may be improved by addressing issues of motivation and relevance of rehabilitation to future well-being, co-morbidities, site and time of programme, transport and care for dependents. Systematic review of studies supported the use of letters, pamphlets or home visits to motivate patients and the use of trained lay visitors. Self-management techniques showed some value in promoting adherence to lifestyle changes. Studies examining professional compliance found that professional support for practice nurses may have value in the coordination of postdischarge care. Average costs in 2001 of cardiac rehabilitation to the health service per patient completing a cardiac rehabilitation programme were about GBP350 (staff only) and GBP490 (total). If services were modelled on an intermediate multidisciplinary configuration with three to five key staff, approximately 13% more patients could be treated with the same budget. Depending on staffing configuration an approximate 200-790% budget increase would be required to provide cardiac rehabilitation to all potentially eligible patients. CONCLUSIONS: Provision of outpatient cardiac rehabilitation in the UK is low and little is known about the capacity of cardiac rehabilitation centres to increase this provision. There is an uncoordinated approach to audit data collection and few interventions aimed at improving the situation have been formally evaluated. Motivational communications and trained lay volunteers may improve uptake of cardiac rehabilitation, as may self-management techniques. Experience of low-cost interventions and good practice exists within rehabilitation centres, although cost information frequently is not reported. Increased provision of outpatient cardiac rehabilitation will require extra resources. Further trials are required to compare the cost-effectiveness of comprehensive multidisciplinary rehabilitation with simpler outpatient programmes, also research is needed into economic and patient preference studies of the effects of different methods of using increased funding for cardiac rehabilitation. An evaluation of a range of interventions to promote attendance in all patients and under-represented groups would also be useful. The development of standards is suggested for audit methods and for eligibility criteria, as well as regular and comprehensive data collection to estimate the need for and provision of cardiac rehabilitation. Further areas for intervention could be identified through qualitative studies, and the extension of low-cost interventions and good practice within rehabilitation centres. Regularly updated systematic reviews of relevant literature would also be useful.