Generic Health-Related Quality of Life Utility Measure for Preschool Children (Health Utilities Preschool): Design, Development, and Properties.

OBJECTIVES: Health Utilities Preschool (HuPS) was developed to fill the need for a generic preference-based measure (GPM) applicable in early childhood. A GPM has all the properties for higher-order summary measures, such as quality-adjusted life-years, required to inform important policy decisions regarding health and healthcare services. METHODS: Development was in accordance with published standards for a GPM, statistical procedures, and modeling. HuPS incorporates key components of 2 existing measurement systems: Health Status Classification System for Preschool Children and Health Utilities Index Mark 3 (HUI3). The study included a series of 4 measurement surveys: definitional, adaptational, quantificational, and evaluational health-related quality of life (HRQL). HuPS measurements were evaluated for reliability, validity, interpretability, and acceptability. RESULTS: Definitional measurements identified 8 Health Status Classification System for Preschool Children attributes in common with HUI3 (vision, hearing, speech, ambulation, dexterity, emotion, cognition, and pain and discomfort), making the HUI3 scoring equation commensurate with HuPS health states. Adaptational measurements informed the content of attribute-level descriptions (n = 35). Quantificational measurements determined level scoring coefficients. HRQL scoring inter-rater reliability (intraclass correlation coefficient = 0.79) was excellent. Continuity of HRQL scoring with HUI3 was reliable (intraclass correlation coefficient = 0.80, P < .001) and valid (mean absolute difference = 0.016, P = .396). CONCLUSIONS: HuPS is an acceptable, reliable, and valid GPM. HRQL scoring is continuous with HUI3. Continuity expands the applicability of GPM (HUI3) scoring to include subjects as young as 2 years of age. Widespread applications of HuPS would inform important health policy and management decisions as HUI3 does for older subjects.

Commentary. In Praise of Studies That Use More Than One Generic Preference-Based Measure.

OBJECTIVES AND BACKGROUND: Generic preference-based (GPB) measures of health-related quality of life (HRQL) are widely used as outcome measures in cost-effectiveness and cost-utility analyses (CEA, CUA). Health technology assessment agencies favor GPB measures because they facilitate comparisons among conditions and because the scoring functions for these measures are based on community preferences. However, there is no gold standard HRQL measure, scores generated by GPB measures may differ importantly, and changes in scores may fail to detect important changes in HRQL. Therefore, to enhance the accumulation of empirical evidence on how well GPB measures perform, we advocate that investigators routinely use two (or more) GPB measures in each study. METHODS: We discuss key measurement properties and present examples to illustrate differences in responsiveness for several major GPB measures across a wide variety of health contexts. We highlight the contributions of longitudinal head-to-head studies. RESULTS: There is substantial evidence that the performance of GPB measures varies importantly among diseases and health conditions. Scores are often not interchangeable. There are numerous examples of studies in which one GPB measure was responsive while another was not. CONCLUSIONS: Investigators should use two (or more) GPB measures. Study protocols should designate one measure as the primary outcome measure; the other measure(s) would be used in secondary analyses. As evidence accumulates it will better inform the relative strengths and weaknesses of alternative GPB measures in various clinical conditions. This will facilitate the selection and interpretation of GPB measures in future studies.

What were they thinking when providing preference measurements for generic health states? The evidence for HUI3.

BACKGROUND: Multi-attribute generic preference-based measures of health-related quality of life are used as comprehensive outcome measures. Typically preferences for health states defined by these systems are elicited from a representative sample of the general population. An important element in that elicitation process is the information that respondents were instructed to consider in providing their responses. METHODS: A random sample of community-dwelling respondents in Canada was surveyed in face-to-face interviews. Respondents provided preference scores for selected Health Utilities Index Mark 3 (HUI3) health states. Respondents also answered questions about the most important attributes and the importance of various impacts of the health states in providing their preference scores. RESULTS: Fifty per cent of respondents reported that they focussed on two, and 21% on three, attributes of the eight HUI3 attributes. Each of the eight attributes was identified as important; pain (49%), vision (37%), cognition (34%), emotion (28%), and ambulation (28%) were the most important. The null hypothesis that all of the attributes were equally important was rejected (p < 0.001). With respect to the impacts, 89% of respondents indicated that the ability to take care of oneself was quite or very important; similarly 76% reported the same for impact on family life, 69% for impact on the happiness of others, 61% for the impact on their ability to work, and 42% for the impact on their leisure activities. The null hypothesis that all of the impacts were equally important was rejected (p < 0.001). CONCLUSIONS: In providing preference scores for HUI3 health states, respondents thoughtfully examined the implications of the health states for their ability to live, work, socialize, and function.

Health Status and Health-related Quality of Life Measurement in Pediatric Cancer Clinical Trials: An Examination of the DFCI 00-01 Acute Lymphoblastic Leukemia Protocol.

Health-related quality of life (HRQL) improved progressively during therapy and beyond in children treated for acute lymphoblastic leukemia on the Dana-Farber Cancer Institute (DFCI) 95-01 protocol. This study aimed to validate that trajectory in a successor study (DFCI 00-01) and to compare the HRQL of patients in the 2 studies. Children aged above 5 years were assessed during each phase of treatment (N=4) and 2 years after completion of therapy. Health status and HRQL were measured using Health Utilities Index (HUI) instruments, HUI2 and HUI3. Quality-adjusted life years (QALYs) were calculated and compared with the general population, and patients treated on DFCI 95-01. Over 5 intervals and 758 HUI assessments, mean HRQL increased progressively from remission induction to the time after treatment (P<0.001). During intensification, high-risk patients had lower HRQL than standard-risk patients (P<0.001). During remission induction, patients on DFCI 95-01 had lower HRQL than patients on DFCI 00-01. Patients on DFCI 00-01 had ~0.2 and 0.3 fewer QALYs than controls, measured by HUI2 and HUI3, respectively. QALYs for DFCI 00-01 patients during treatment were similar to those for DFCI 95-01 patients. The trajectory of improvement in HRQL during the treatment of acute lymphoblastic leukemia in children was confirmed.

Health-related Quality of Life in Long-term Survivors of Brain Tumors in Childhood and Adolescence: A Serial Study Spanning a Decade.

Survivors of brain tumors in childhood experience adverse sequelae that are greater in prevalence and severity than those encountered by survivors of all other forms of cancer in early life, reflected in a burden of morbidity by instruments measuring health-related quality of life (HRQL). However, there are few studies of the change in HRQL over time in such populations. Patients who were above 5 years of age, at least 2 years from completion of therapy, and able to communicate in English were eligible for study of HRQL by the Health Utilities Index HUI2 and HUI3 at study entry, and again 5 and 10 years later. An initial cohort of 40 patients was reduced to 37 and 25 at the second and third time points, respectively, although only 1 death occurred during the study. HRQL showed a progressive decline over the decade, reaching conventional levels of clinical significance for the sizes of the changes. Median scores for HUI2 were 0.93, 0.90, and 0.88; and for HUI3 were 0.88, 0.85, and 0.77 at baseline, 5, and 10 years, respectively. The serial decline in HRQL demands further examination and an exploration of potential targets for therapeutic intervention.

Surveillance and survival among adolescents and young adults with cancer in Ontario, Canada.

Gains in survival rates among adolescents and young adults (AYA) are reported from the USA to be lower than in both younger and older patients. Limiting factors include low accrual to clinical trials related to the type of institutional care. This study aimed to determine the incidence of cancer in the 15-29 age group in Ontario, and the 5-year survival of these cases by disease class, age at diagnosis group and highest level of institutional complexity of care. The primary data source was Cancer Care Ontario (CCO). Diseases were classified according to an AYA-specific system. Age at diagnosis was grouped as 15-19, 20-24 and 25-29 years; and institutional site of care was categorized as pediatric oncology group of Ontario (POGO) centers, regional cancer centers (RCC-tertiary care centers associated with CCO), RCC affiliate and satellite institutions and other institutions having no specialized cancer services. More than 10,000 incident cases were identified during 1990-2001. Carcinomas and lymphomas each accounted for > 20% of the total. Overall 5-year survival rate was 83%; significantly higher for lymphomas at POGO centers and RCC than elsewhere. About 40% of eligible AYA cases were treated at a POGO center and 25% of those were accrued to clinical trials. The low proportion of adolescents referred to pediatric cancer centers may result in a survival disadvantage for this group. All AYA, especially with lymphomas, should be referred to specialized centers. Accrual of AYA to clinical trials must be improved substantially.

Health-related quality of life among children with acute lymphoblastic leukemia.

BACKGROUND: The objective was to quantify the health-related quality of life (HRQL) of children treated for acute lymphoblastic leukemia (ALL) and identify specific disabilities for remediation. PROCEDURE: Two types of subjects were included: ALL patients 5 plus years old in a multi-center clinical trial and general population control groups. Patients were assessed during all four major phases of active treatment and approximately 2 years after treatment. Health status and HRQL were measured using HEALTH UTILITIES INDEX® (HUI®) Mark 2 (HUI2) and Mark 3 (HUI3). HRQL scores were used to calculate quality-adjusted life years (QALYs). Excess disability rates identified attributes for remediation. RESULTS: HUI assessments (n = 749) were collected during the five phases. Mean HRQL increased from induction through the post-treatment phase (P < 0.001). There were no significant demographic or treatment effects on HRQL, except for type of asparaginase during continuation therapy (P = 0.005 for HUI2 and P = 0.007 for HUI3). Differences in mean HRQL scores between patients and controls were important (P < 0.001) during the active treatment phases but not during the post-treatment phase. Relative to controls, patients lost approximately 0.2 QALYs during active treatment. Disability was evident in mobility/ambulation, emotion, self-care and pain, and declined over time. CONCLUSIONS: Patients with ALL experienced important but declining deficits in HRQL during active treatment phases: Equivalent to losing approximately 2 months of life in perfect health. HRQL within the 2-years post-treatment phase was similar to controls. The policy challenge is to develop new treatment protocols producing fewer disabilities in mobility/ambulation, emotion, self-care, and pain without compromising survival.

Translation and cultural adaptation of health utilities index with application to pediatric oncology patients during neutropenia and recovery in Turkey.

BACKGROUND: This is a report on the translation and cultural adaptation from English to Turkish languages of a standard HEALTH UTILITIES INDEX® (HUI®) questionnaire for interviewer-administration and proxy assessment of patients' health status, and its use for assessing 50 consecutive pediatric oncology patients during periods of and subsequent recovery from neutropenia. PROCEDURE: Preparation of the Turkish-language questionnaire was a collaborative effort by physician-researchers in Turkey, senior Health Utilities, Inc. staff with extensive experience in the development and translation of HUI questionnaires, and Turkish-born residents of Canada. Standard HUI questionnaires cover both HUI Mark 2 (HUI2) and HUI Mark 3 (HUI3). RESULTS: The mean improvement in overall health-related quality of life (HRQL) scores between neutropenic and non-neutropenic phases was clinically important according to both HUI2 and HUI3 instruments. Single-attribute utility scores showed clinically important size improvements between phases for HUI2 mobility, emotion, and HUI2 self-care. Significant reductions in disability rates between phases were also detected for pain (HUI2 and HUI3 rate decreases of 30%, P < 0.001) and HUI3 ambulation (rate decline of 14%, P = 0.020). CONCLUSIONS: The study results provide evidence that the Turkish-language questionnaire is acceptable, valid and useful for collecting parental assessments of health-status among young cancer patients in Turkey. Neutropenia was associated with important deficits in overall HRQL and disabilities in ambulation/mobility, emotion, self-care, and pain. Treatment plans should include strategies for addressing emotion and pain problems to improve the HRQL of neutropenic patients.

Canadian French translation and linguistic validation of the health-related quality of life utility measure for pre-school children.

BACKGROUND: There is a need to perform a Canadian French translation and linguistic validation of the health-related quality of life utility measure for pre-school children (HuPS) conceptually equivalent to the original Canadian English version. RESEARCH DESIGN AND METHODS: The translation process consisted of forward and back translations. The linguistic validation was performed with the parents of preschool children during face-to-face cognitive debriefing interviews. The whole process was done in accordance with academic standards and the guidance of the Food and Drug Administration (FDA) for patient-reported outcome instruments. RESULTS: The results of back translations indicated that 89% of the sentences were identical or almost identical to the original English-language wording. The review of the back translations led to a change in 13 sentences out of 91 from the reconciled forward translation, while the linguistic validation process with 13 parents led to 14 additional changes. Preliminary reliability validation results indicate a Cronbach's alpha of 0.73. CONCLUSION: The translation and linguistic testing processes were successful in creating a valid HuPS in Canadian French (HuPS-CF). This translation should be the subject of reliability and validity studies in a wide variety of clinical and general populations before to use in research projects.

Self and proxy-reported health status and health-related quality of life in survivors of childhood cancer in Uruguay.

BACKGROUND: The incidence of cancer in children in Uruguay is similar to that in industrialized societies but the survival rate is half as great. This study assesses another important measure of treatment effectiveness: the health-related quality of life (HRQL) of survivors. METHODS: All new patients diagnosed in a 3-year period were eligible if free of disease for at least 2 years after therapy and at least 7 years of age at the time of study. A sample of convenient subjects for comparison was obtained from schools and clinics for well children. During a 7-month period, Spanish language interviewer-administered questionnaires were used to collect Health Utilities Index data from survivors and the comparison group (self-reports), and from proxies (parents, physicians, and teachers). RESULTS: Of 113 eligible survivors, 95 (84%) participated together with 96 "control" subjects. Control subjects have a higher mean HRQL utility score than survivors (P < 0.001). The mean score for survivors of acute lymphoblastic leukemia (ALL): 0.72 (n = 49) is higher than the score for survivors of brain tumors: 0.60 (n = 20), as expected. Inter-rater agreement is highest between survivors and parents, and lowest between controls and physicians or teachers. CONCLUSIONS: The burden of morbidity in survivors of childhood cancer in Uruguay is considerable and greater than that in a comparative group of healthy children. Survivors of ALL have better HRQL than survivors of brain tumors, mirroring experience elsewhere. The level of inter-rater agreement is related to the degree of familiarity of the pair-members of respondents with each other.

Disability and health-related quality of life in long-term survivors of cancer in childhood in Brazil.

There is limited experience with patient-reported measurements of health status and health-related quality of life (HRQL) in survivors of cancer in childhood in low-income countries. The purposes of this study were to collect such measurements in Brazil, to test hypotheses about differences among diagnostic groups, and to compare results with those from other countries. Survivors were eligible if diagnosed with cancer in childhood, attending a long-term follow-up clinic, cancer free, literate, and at least 13 years of age. Health status measurements were collected using a Brazilian Portuguese Health Utilities Index questionnaire. Questionnaire responses were converted to scores for morbidity in individual health attributes and for overall HRQL. More than one-third of the 138 consecutive survivors who participated reported some cognitive disability or pain. Approximately one-quarter reported problems with vision, speech, or emotion. Mean HRQL was similar (P>0.05) among countries for survivors of acute lymphoblastic leukemia and Hodgkin disease. The results support the hypotheses that Brazilian survivors of cancer in childhood experience a wide range of disabilities and impaired HRQL, are similar to those in other countries, and should be assessed in long-term follow-up clinics.

Methodology and measurement properties of health-related quality of life instruments: a prospective study of patients undergoing breast reduction surgery.

BACKGROUND: Breast hypertrophy is associated with clinically important morbidity. A prospective study was conducted to assess the change in health-related quality of life (HRQL) following breast reduction mammoplasty. This paper describes the measurement properties of each of the HRQL questionnaires used. METHODS: The reliability, responsiveness, and the construct validity of each HRQL instrument (the Health Utilities Index Mark 2 (HUI2) and Mark 3 (HUI3) and the Breast Reduction Assessment Value and Outcomes (BRAVO) instruments) were assessed. The BRAVO instruments are a set of separate instruments including the Short Form-36 (SF-36), the Multidimensional Body Self Relations Questionnaire Appearance Assessment (MBSRQ-AS), and the Breast Related Symptoms Questionnaire (BRSQ). RESULTS: The HUI2, the HUI3, the MBSRQ-AS, and the breast severity symptom (BSS) score from the BRSQ all demonstrated good test-retest reliability. The SF-36 physical component summary, the MBSRQ-AS, and the BSS score demonstrated high responsiveness. The SF-36 mental component summary and the HUI3 had a moderate effect size and the HUI2 had a small effect size. All of the changes in scales are correlated in the same direction except for the SF-36 physical component summary and the SF-36 mental component summary. CONCLUSION: All four instruments were found to be reliable and responsive. These instruments can be used in similar clinical settings to evaluate the change in patients' HRQL.

Patient-focused measures of functional health status and health-related quality of life in pediatric orthopedics: a case study in measurement selection.

The objectives of this report are to review the assessment of patient-focused outcomes in pediatric orthopedic surgery, to describe a framework for identifying appropriate sets of measures, and to illustrate an application of the framework to a challenging orthopedic problem.A detailed framework of study design and measurement factors is described. The factors are important for selecting appropriate instruments to measure health status and health-related quality of life (HRQL) in a particular context. A study to evaluate treatment alternatives for patients with neurofibromatosis type 1 and congenital tibial dysplasia (NF1-CTD) provides a rich illustration of the application of the framework. The application involves great variability in the instrument selection factors. Furthermore, these patients and their supportive caregivers face numerous complex health challenges with long-term implications for HRQL. Detailed summaries of important generic preference-based multi-attribute measurement systems, pediatric health profile instruments, and pediatric orthopedic-specific instruments are presented. Age-appropriate generic and specific measures are identified for study of NF1-CTD patients. Selected measures include the Activities Scale for Children, Gillette Functional Assessment Questionnaire Walking Scale, Health Utilities Index, and Pediatric Inventory of Quality of Life. Reliable and valid measures for application to pediatric orthopedics are available. There are important differences among measures. The selected measures complement each other. The framework in this report provides a guide for selecting appropriate measures. Application of appropriate sets of measures will enhance the ability to describe the morbidity of pediatric orthopedic patients and to assess the effectiveness of alternative clinical interventions. The framework for measurement of health status and HRQL from a patient perspective has relevance to many other areas of orthopedic practice.

Exploration of Morbidity in a Serial Study of Long-Term Brain Tumor Survivors: A Focus on Pain.

PURPOSE: Children surviving brain tumors are frequently identified as having substantially decreased health-related quality of life (HRQL) in cross-sectional studies. This study explored the HRQL of a cohort of such survivors, who were recruited as adolescents and followed for a decade, in order to determine the trajectory of their morbidities. METHOD: Children diagnosed between January 1, 1985, and December 31, 1998, more than 2 years from diagnosis (N = 40), were recruited in 2000/2001 (T1) aged 16.74 ± 4.23 years. Health Utilities Index questionnaires (HUI2/3) were completed in 2000/2001 and again at 5 years (T2) and 10 years (T3), with 37 and 25 participants then aged 21.54 ± 4.29 and 27.97 ± 4.07 years, respectively. In addition to study subjects, parental proxies completed questionnaires at T1 and T2, while study subjects selected proxies at T3. Single attributes (domains/dimensions) of HRQL and details of pain were analyzed. RESULTS: Cognition was the attribute compromised most often (T1 = 66.7% of participants, T2 = 62.2%, T3 = 60.0%). Pain was also reported frequently (T1 = 35%, T2 = 25%, T3 = 52%), and at T3 correlated moderately with HUI2 sensation (0.77) and HUI3 vision (0.44), speech (0.51), and ambulation (0.50). The lower median utility score for pain at T3 than at T1/T2 was a clinically important difference. Severe pain was identified in the lower extremities, back, upper extremities, and abdomen. Morbidity was observed also in emotion (worry HUI2 and unhappiness HUI3), sensation, and vision. CONCLUSION: Decreased HRQL in survivors of brain tumors in childhood is multifaceted. Pain is a prominent burden, along with morbidity in cognition, emotion, sensation, and vision. Further studies should explore pain and neurologic deficits, and potential opportunities for therapeutic intervention.

Economic evaluation of treatments for cancer in childhood.

Treatment of cancer in childhood is an expensive undertaking for the health-care system and for the affected families. As there is a substantial burden of treatment-related morbidity, it is important to determine whether the effects of treatment are worth these monetary costs, especially from a societal perspective. Economic evaluation affords a comparison of the costs and consequences (effects) of relevant therapeutic alternatives. Preference-based measures of health-related quality of life are particularly useful for assessing the effects of treatment, for these tools integrate mortality and morbidity. These measures provide utility scores that can be used as weights on survival data to compute quality-adjusted life years (QALYs). Costs are incurred both within and outside of the health-care system. The former should include those in front-line patient care departments (e.g. nursing); the pro-rated share of the expenses of service departments (e.g. materials management) to those in the front line; and the fully allocated costs for capital invested in lands, building and equipment. The latter are costs borne by families that are both out-of-pocket (e.g. for over-the-counter drugs) as well as related to time spent in providing care, which may involve foregone income. Costs and consequences should be subject to discounting; a process for converting those items incurred in the future into contemporary equivalents. Economic evaluation provides estimates of incremental discounted costs per discounted QALY gained. By almost any interpretative standard this appears attractive with respect to cancer in childhood. Examples are provided with the encouragement that economic evaluation be undertaken in more clinical trials in paediatric oncology.

Comparing directly measured standard gamble scores to HUI2 and HUI3 utility scores: group- and individual-level comparisons.

Directly measured standard gamble (SG) utility scores reflect the respondent's assessment and valuation of their own health status. Scores from the health utilities index (HUI) are based on self-assessed health status but valued using community preferences obtained using the SG. Our objectives were to find if mean directly measured utility scores agree with mean HUI mark 2 (HUI2) and mean HUI mark 3 (HUI3) scores. Also, if individual directly measured utility scores agree with HUI2 and HUI3 scores, and whether HUI2 and HUI3 scores agree. Questionnaires based on the HUI2 and HUI3 health-status classification systems were administered by interviewers to 140 teenage survivors of extremely low birthweight (ELBW) and 124 control group teens. Respondents were asked to think about their own usual health states using six dimensions from HUI2 and value that state using the SG. Mean SG scores are compared with mean HUI2 and mean HUI3 scores using paired sample t-tests. Mean HUI2 scores are compared with mean HUI3 scores. Agreement among scores is assessed using intra-class correlation coefficient (ICC). The effect of severity of health-state morbidity on agreement was assessed using three approaches. ELBW cohort mean (standard deviation) SG, HUI2, and HUI3 scores were 0.90 (0.20), 0.89 (0.14), and 0.80 (0.22). Results for controls were 0.93 (0.11), 0.95 (0.09), and 0.89 (0.13). Mean SG and HUI2 scores did not differ; mean SG and HUI3 did differ; mean HUI2 and HUI3 also differed. At the individual level for ELBW, the ICCs between SG and HUI2, SG and HUI3, and HUI2 and HUI3 scores were 0.13, 0.28, and 0.64. For controls the ICCs were 0.14, 0.24, and 0.56. HUI2 scores appear to match directly measured utility scores reasonably well at the group level. HUI2 and HUI3 scores differ systematically. At the individual level, however, HUI2 and HUI3 scores are poor substitutes for directly measured scores.

The Health Utilities Index (HUI): concepts, measurement properties and applications.

This is a review of the Health Utilities Index (HUI) multi-attribute health-status classification systems, and single- and multi-attribute utility scoring systems. HUI refers to both HUI Mark 2 (HUI2) and HUI Mark 3 (HUI3) instruments. The classification systems provide compact but comprehensive frameworks within which to describe health status. The multi-attribute utility functions provide all the information required to calculate single-summary scores of health-related quality of life (HRQL) for each health state defined by the classification systems. The use of HUI in clinical studies for a wide variety of conditions in a large number of countries is illustrated. HUI provides comprehensive, reliable, responsive and valid measures of health status and HRQL for subjects in clinical studies. Utility scores of overall HRQL for patients are also used in cost-utility and cost-effectiveness analyses. Population norm data are available from numerous large general population surveys. The widespread use of HUI facilitates the interpretation of results and permits comparisons of disease and treatment outcomes, and comparisons of long-term sequelae at the local, national and international levels.

How robust is the Health Utilities Index Mark 2 utility function?

PURPOSE: The utility function for the Health Utilities Index Mark 2 (HUI2) system is based on preference measurements from a random sample of parents with exclusion of inconsistent respondents. Would results without exclusions or from a different group of parents have differed? METHODS: Scores were obtained from parents of patients (n = 59) undergoing treatment for cancer. Mean scores from the 2 sets of parents were compared:parents of patients and parents from the general population. Three multiattribute utility functions were estimated. Mean scores for HUI2 states using the functions were compared. RESULTS: Most differences in mean scores between different groups were not statistically significant (P < 0.05). Differences in parameter estimates among the 3 utility functions were 0.05 or less. The exponent on the power function for the parent-of-patient group was 2.16, within 6% of that for random sample parents. The intraclass correlation between scores for 144 health states derived from the random-sample-parents and parents-of-patients functions was 0.99; the mean difference per state in scores was 0.018. CONCLUSION: The HUI2 scoring function generalizes well in that different groups of parents give similar results. The HUI2 scoring function is robust in that the functions without and with exclusions generate scores that are very close in value.

Health-related quality-of-life assessment of prenatal diagnosis: chorionic villi sampling and amniocentesis.

This study assesses the health-related quality-of-life (HRQL) effects of chorionic villi sampling (CVS) and genetic amniocentesis (GA), including both process and outcomes of prenatal diagnosis. The HRQL of 126 women participating in a randomized controlled clinical trial of CVS versus GA in Toronto and Hamilton, Ontario, was assessed in four interviews at weeks 8, 13, 18, and 22 of pregnancy. Statistical analyses included analysis of variance, repeated measures analysis of covariance, chi-square, Fisher's exact test, Student's t-tests, and paired t-tests. Utility scores for patients undergoing CVS exceeded those for GA patients at week 18 (p = 0.04). Utility scores for hypothetical health states did not differ significantly by trial arm. CVS results in slightly improved HRQL during prenatal diagnosis. This advantage needs to be weighed against the high disutility patients attach to infrequent outcomes associated with pregnancy losses, equivocal diagnoses, and diagnostic inaccuracy.