Carrier screening for cystic fibrosis: costs and clinical outcomes.

OBJECTIVES: To evaluate the costs and clinical effects of 16 alternative strategies for cystic fibrosis (CF) carrier screening in the reproductive setting; and to test the sensitivity of the results to assumptions about cost and detection rate, stakeholder perspective, DNA test specificity, chance of nonpaternity, and couples' reproductive plans. METHOD: Cost-effectiveness analysis. RESULTS: A sequential screening strategy had the lowest cost per CF birth avoided. In this strategy, the first partner was screened with a standard test that identifies 85% of carriers. The second partner was screened with an expanded test if the first partner's screen was positive. This strategy identified 75% of anticipated CF births at a cost of $367,000 each. This figure does not include the lifetime medical costs of caring for a patient with CF, and it assumes that couples who identify a pregnancy at risk will choose to have prenatal diagnosis and termination of affected pregnancies. The cost per CF birth identified is approximately half this figure when couples plan two children. CONCLUSIONS: The cost-effectiveness of CF carrier screening depends greatly on couples' reproductive plans. CF carrier screening is most cost-effective when it is performed sequentially, when the information is used for more than one pregnancy, and when the intention of the couple is to identify and terminate affected pregnancies. These conclusions are important for policy considerations regarding population-based screening for CF, and may also have important implications for screening for less common diseases.

Response rates to mail surveys published in medical journals.

OBJECTIVE: The purpose of this study was to characterize response rates for mail surveys published in medical journals; to determine how the response rate among subjects who are typical targets of mail surveys varies; and to evaluate the contribution of several techniques used by investigators to enhance response rates. METHODS: One hundred seventy-eight manuscripts published in 1991, representing 321 distinct mail surveys, were abstracted to determine response rates and survey techniques. In a follow-up mail survey, 113 authors of these manuscripts provided supplementary information. RESULTS: The mean response rate among mail surveys published in medical journals is approximately 60%. However, response rates vary according to subject studied and techniques used. Published surveys of physicians have a mean response rate of only 54%, and those of non-physicians have a mean response rate of 68%. In addition, multivariable models suggest that written reminders provided with a copy of the instrument and telephone reminders are each associated with response rates about 13% higher than surveys that do not use these techniques. Other techniques, such as anonymity and financial incentives, are not associated with higher response rates. CONCLUSIONS: Although several mail survey techniques are associated with higher response rates, response rates to published mail surveys tend to be moderate. However, a survey's response rate is at best an indirect indication of the extent of non-respondent bias. Investigators, journal editors, and readers should devote more attention to assessments of bias, and less to specific response rate thresholds.

The limits of suffering: critical care nurses' views of hospital care at the end of life.

This study explores critical care nurses' beliefs, motivations, and experiences regarding end of life care. We performed a content analysis of the text comments provided by 468 U.S. critical care nurses in response to an anonymous mail survey about euthanasia. These comments reveal that these U.S. critical care nurses' feelings about issues surrounding the end of life are extremely complex. Eight themes emerged from 37 coded concepts, and the median number of themes volunteered per nurse was three. Among the most prevalent themes were patient concerns (53%), family concerns (33%), clinical circumstances (42%), the nurses' personal concerns (38%), and external or structural issues (68%). A profound sense of compassion and often conflicting forces were noted within and across themes. The nurses' comments offer a unique perspective on the care of critically ill patients and reveal much about that care that should be improved. In particular, (1) some nurses are frustrated about their limited role in the management of patients at the end of life, given their special understanding of these patients' experiences and wishes; (2) considerable confusion remains about the most appropriate way to care for these patients; and (3) the environment of some critical care setting may be unable to foster the compassion that many patients need.

Genetic screening for reproductive planning: methodological and conceptual issues in policy analysis.

OBJECTIVES: This paper explores several critical assumptions and methodological issues arising in cost-effectiveness analyses of genetic screening strategies in the reproductive setting. METHODS: Seven issues that arose in the development of a decision analysis of alternative strategies for cystic fibrosis carrier screening are discussed. Each of these issues required a choice in technique. RESULTS: The presentations of these analyses frequently mask underlying assumptions and methodological choices. Often there is no best choice. In the case of genetic screening in the reproductive setting, these underlying issues often touch on deeply felt human values. CONCLUSIONS: Space limitations for published papers often preclude explaining such choices in detail; yet these decisions determine the way the results should be interpreted. Those who develop these analyses need to make sure that the implications of important assumptions are understood by the clinicians who will use them. At the same time, clinicians need to enhance their understanding of what these models truly mean and how they address underlying clinical, ethical, and economic issues.

Methicillin-resistant Staphylococcus aureus colonization in a new nursing home.

Methicillin-resistant Staphylococcus aureus (MRSA) has been detected in nursing homes and long-term care facilities. Studies disagree about the risk of infection with MRSA in colonized patients. MRSA colonization and infection were tracked for one year in all admissions to a 60-bed ward at the Philadelphia VA Nursing Home Care Unit (NHCU) from the time of its opening in June, 1990. Patients and staff were blinded to culture results, and the NHCU followed universal precautions for all patients. Of the first 72 patients, 7 were found to be colonized with MRSA; only one of them was known to have had MRSA prior to NHCU transfer. Three patients died (2 had negative cultures prior to death), and 1 was discharged home. Three patients spontaneously cleared MRSA colonization and lived to the end of the study. Three patients appeared to be colonized by MRSA after admission; subsequent cultures were negative. No patients were infected by MRSA in the NHCU. At the close of the study, one year after the nursing home opened, no patient in the nursing home had a culture positive for MRSA. In conclusion, colonization with MRSA at the time of admission to the nursing home is not uncommon, but patients can spontaneously clear it. Besides, nursing homes that pre-screen only those patients with classic risk factors may be admitting many MRSA-colonized patients. Nonetheless, universal precautions appear to be effective in limiting transmission of MRSA in the nursing home; in this study, MRSA acquisition was sporadic and brief.