Health care utilization and outcomes in older adults after Traumatic Brain Injury: A CENTER-TBI study.

INTRODUCTION: The incidence of Traumatic Brain Injury (TBI) is increasingly common in older adults aged ≥65 years, forming a growing public health problem. However, older adults are underrepresented in TBI research. Therefore, we aimed to provide an overview of health-care utilization, and of six-month outcomes after TBI and their determinants in older adults who sustained a TBI. METHODS: We used data from the prospective multi-center Collaborative European NeuroTrauma Effectiveness Research in Traumatic Brain Injury (CENTER-TBI) study. In-hospital and post-hospital health care utilization and outcomes were described for patients aged ≥65 years. Ordinal and linear regression analyses were performed to identify determinants of the Glasgow Outcome Scale Extended (GOSE), health-related quality of life (HRQoL), and mental health symptoms six-months post-injury. RESULTS: Of 1254 older patients, 45% were admitted to an ICU with a mean length of stay of 9 days. Nearly 30% of the patients received inpatient rehabilitation. In total, 554/1254 older patients completed the six-month follow-up questionnaires. The mortality rate was 9% after mild and 60% after moderate/severe TBI, and full recovery based on GOSE was reported for 44% of patients after mild and 6% after moderate/severe TBI. Higher age and increased injury severity were primarily associated with functional impairment, while pre-injury systemic disease, psychiatric conditions and lower educational level were associated with functional impairment, lower generic and disease-specific HRQoL and mental health symptoms. CONCLUSION: The rate of impairment and disability following TBI in older adults is substantial, and poorer outcomes across domains are associated with worse preinjury health. Nonetheless, a considerable number of patients fully or partially returns to their preinjury functioning. There should not be pessimism about outcomes in older adults who survive.

Effectiveness of a minimal virtual motivational interviewing training for first years medical students: differentiating between pre-test and then-test.

BACKGROUND: Shifting towards patient-centeredness, medical doctors need patient-centered communication skills. Motivational Interviewing (MI) is an evidence-based, collaborative, goal-oriented communication technique to strengthen a person's own motivation and commitment to change. The purpose of this study is to evaluate the effectiveness of a brief virtual role-play MI-training program on MI-knowledge and skills in first-year undergraduate medical students, making use of both a pre-test and a then-test (retrospective pre-test) to check for response shift in evaluating the educational intervention. METHODS: Four 10-15 min MI-game-based training conversations embedded in the Kognito Conversation Platform™ were offered to the students using a single-group Interrupted Time Series design. RESULTS: Participants included 339 undergraduate medical students (RR= 83.1%). The one-hour MI virtual training proved effective in two ways: participants gained knowledge and skills, and increased awareness of the existing intrinsic knowledge and skill they already possess to communicate with future patients in a patient-centered way. CONCLUSION: A brief one-hour MI-training simulation can be effective even if offered at an early stage during medical education. Furthermore, response shift varied and was not present in all students. PRACTICE IMPLICATION: The addition of a then-test to the study design reveals results that otherwise would not have been found.

Reference Values and Psychometric Properties of the Quality of Life After Traumatic Brain Injury-Overall Scale in Italy, The Netherlands, and the United Kingdom.

OBJECTIVES: The Quality of Life after Brain Injury-Overall Scale (QOLIBRI-OS) is a short screening instrument for assessing disease-specific health-related quality of life (HRQoL) after traumatic brain injury. To date, no reference values are available for the QOLIBRI-OS in general populations. Thus, this study aimed to establish reference values for the QOLIBRI-OS in general population samples from Italy, The Netherlands, and the United Kingdom. METHODS: Data were collected using an online survey. The total sample comprised 11759 participants, consisting of 3549 Italian, 3564 Dutch, and 4646 British subjects. In this sample, 49% of the total sample did not report any health complaints, whereas 51% had at least 1 chronic health condition. Reference values were deduced for the QOLIBRI-OS for health-condition-related samples and total general population samples per country. To ensure the comparability of these values, measurement invariance was assessed using a multigroup confirmatory factor analysis. Covariates characterizing the reference values were selected with the help of regression analyses. RESULTS: The confirmatory factor analysis confirmed that the QOLIBRI-OS scores measured the same traumatic brain injury-specific HRQoL construct across the 3 countries. Healthy individuals reported significantly higher HRQoL than individuals with at least 1 chronic health condition. Older age and higher education levels were significantly associated with higher HRQoL. CONCLUSIONS: Because the reference values displayed differences in terms of age and education level across the 3 countries, we recommend using country-specific reference values stratified by sociodemographic and health status in research and clinical practice.

Translation and Linguistic Validation of Outcome Instruments for Traumatic Brain Injury Research and Clinical Practice: A Step-by-Step Approach within the Observational CENTER-TBI Study.

Assessing outcomes in multinational studies on traumatic brain injury (TBI) poses major challenges and requires relevant instruments in languages other than English. Of the 19 outcome instruments selected for use in the observational Collaborative European NeuroTrauma Effectiveness Research in TBI (CENTER-TBI) study, 17 measures lacked translations in at least one target language. To fill this gap, we aimed to develop well-translated linguistically and psychometrically validated instruments. We performed translations and linguistic validations of patient-reported measures (PROMs), clinician-reported (ClinRO), and performance-based (PerfO) outcome instruments, using forward and backward translations, reconciliations, cognitive debriefings with up to 10 participants, iterative revisions, and international harmonization with input from over 150 international collaborators. In total, 237 translations and 211 linguistic validations were carried out in up to 20 languages. Translations were evaluated at the linguistic and cultural level by coding changes when the original versions are compared with subsequent translation steps, using the output of cognitive debriefings, and using comprehension rates. The average comprehension rate per instrument varied from 88% to 98%, indicating a good quality of the translations. These outcome instruments provide a solid basis for future TBI research and clinical practice and allow the aggregation and analysis of data across different countries and languages.

The Association of Post-Concussion and Post-Traumatic Stress Disorder Symptoms with Health-Related Quality of Life, Health Care Use and Return-to-Work after Mild Traumatic Brain Injury.

Patients with mild traumatic brain injury (mTBI) are at risk for post-concussion (PC) symptoms and post-traumatic stress disorder (PTSD). The co-occurrence of PC and PTSD symptoms after mTBI in relation to health-related quality of life (HRQoL), health care utilization, and return to work has not yet been investigated. PC and PTSD symptoms were measured six months post-TBI by respectively the Rivermead Post-Concussion Symptoms Questionnaire (RPQ) and the Post-Traumatic Stress Disorder Checklist for DSM-5 (PCL-5). Of the 1566 individuals after mTBI who met the inclusion criteria, 26.1% experienced PC symptoms (RPQ ≥16). Additionally, 9.8% experienced PTSD symptoms (PCL-5 ≥ 33), of which the vast majority (81%) also reported experiencing PC symptoms. Differences between patients with no/mild symptoms, with only PC, only PTSD, and both PC and PTSD symptoms in HRQoL, return to work, and rehabilitation were analyzed using logistic and linear regression analyses. Patients with PC and/or PTSD symptoms reported lower HRQoL, higher rates of rehabilitation, and lower return to work rates compared to patients with no/mild symptoms. Patients with both PC and PTSD symptoms reported significantly lower HRQoL (B = -2.73, CI = -4.65; -0.83, p < 0.001) compared to those with only PC symptoms, while there were no significant differences in their ongoing rehabilitation care (OR = 1.39, CI = 0.77-2.49, p = 0.272) and return to work rates (OR = 0.49, CI = 0.15-1.63, p = 0.246) at six months. These results underline the importance of the diagnosis and appropriate treatment of patients with mTBI, experiencing PC and/or PTSD symptoms.

Psychometric Characteristics of the Patient-Reported Outcome Measures Applied in the CENTER-TBI Study.

Traumatic brain injury (TBI) may lead to impairments in various outcome domains. Since most instruments assessing these are only available in a limited number of languages, psychometrically validated translations are important for research and clinical practice. Thus, our aim was to investigate the psychometric properties of the patient-reported outcome measures (PROM) applied in the CENTER-TBI study. The study sample comprised individuals who filled in the six-months assessments (GAD-7, PHQ-9, PCL-5, RPQ, QOLIBRI/-OS, SF-36v2/-12v2). Classical psychometric characteristics were investigated and compared with those of the original English versions. The reliability was satisfactory to excellent; the instruments were comparable to each other and to the original versions. Validity analyses demonstrated medium to high correlations with well-established measures. The original factor structure was replicated by all the translations, except for the RPQ, SF-36v2/-12v2 and some language samples for the PCL-5, most probably due to the factor structure of the original instruments. The translation of one to two items of the PHQ-9, RPQ, PCL-5, and QOLIBRI in three languages could be improved in the future to enhance scoring and application at the individual level. Researchers and clinicians now have access to reliable and valid instruments to improve outcome assessment after TBI in national and international health care.

Influence of Sociodemographic, Premorbid, and Injury-Related Factors on Post-Concussion Symptoms after Traumatic Brain Injury.

BACKGROUND: Post-concussion symptoms (PCS) are often reported as consequences of mild and moderate traumatic brain injury (TBI), but these symptoms are not well documented in severe TBI. There is a lack of agreement as to which factors and covariates affect the occurrence, frequency, and intensity of PCS among TBI severity groups. The present study therefore aims to examine the association between sociodemographic, premorbid, and injury-related factors and PCS. METHODS: A total of 1391 individuals (65% male) from the CENTER-TBI study were included in the analyses. The occurrence, frequency (number of PCS), and intensity (severity) of PCS were assessed using the Rivermead Post-concussion Symptoms Questionnaire (RPQ) at six months after TBI. To examine the association between selected factors (age, sex, living situation, employment status, educational background, injury and TBI severity, and premorbid problems) and PCS, a zero-inflated negative binomial model (ZINB) for occurrence and frequency of PCS and a standard negative binomial regression (NB) for intensity were applied. RESULTS: Of the total sample, 72% of individuals after TBI reported suffering from some form of PCS, with fatigue being the most frequent among all TBI severity groups, followed by forgetfulness, and poor concentration. Different factors contributed to the probability of occurrence, frequency, and intensity of PCS. While the occurrence of PCS seemed to be independent of the age and sex of the individuals, both the frequency and intensity of PCS are associated with them. Both injury and TBI severity influence the occurrence and frequency of PCS, but are associated less with its intensity (except "acute" symptoms such as nausea, vomiting, and headaches). Analyses focusing on the mTBI subgroup only yielded results comparable to those of the total sample. DISCUSSION: In line with previous studies, the results support a multifactorial etiology of PCS and show the importance of differentiating between their occurrence, frequency, and intensity to better provide appropriate treatment for individual subgroups with different symptoms (e.g., multiple PCS or more intense PCS). Although PCS often occur in mild to moderate TBI, individuals after severe TBI also suffer from PCS or post-concussion-like symptoms that require appropriate treatment. The chosen statistical approaches (i.e., ZINB and NB models) permit an ameliorated differentiation between outcomes (occurrence, frequency, and intensity of PCS) and should be used more widely in TBI research.

Outcomes after Complicated and Uncomplicated Mild Traumatic Brain Injury at Three-and Six-Months Post-Injury: Results from the CENTER-TBI Study.

The objective of this study was to provide a comprehensive examination of the relation of complicated and uncomplicated mild traumatic brain injury (mTBI) with multidimensional outcomes at three- and six-months after TBI. We analyzed data from the Collaborative European NeuroTrauma Effectiveness Research (CENTER-TBI) research project. Patients after mTBI (Glasgow Coma scale (GCS) score of 13-15) enrolled in the study were differentiated into two groups based on computed tomography (CT) findings: complicated mTBI (presence of any traumatic intracranial injury on first CT) and uncomplicated mTBI (absence of any traumatic intracranial injury on first CT). Multidimensional outcomes were assessed using seven instruments measuring generic and disease-specific health-related quality of life (HRQoL) (SF-36 and QOLIBRI), functional outcome (GOSE), and psycho-social domains including symptoms of post-traumatic stress disorder (PTSD) (PCL-5), depression (PHQ-9), and anxiety (GAD-7). Data were analyzed using a multivariate repeated measures approach (MANOVA-RM), which inspected mTBI groups at three- and six-months post injury. Patients after complicated mTBI had significantly lower GOSE scores, reported lower physical and mental component summary scores based on the SF-36 version 2, and showed significantly lower HRQoL measured by QOLIBRI compared to those after uncomplicated mTBI. There was no difference between mTBI groups when looking at psychological outcomes, however, a slight improvement in PTSD symptoms and depression was observed for the entire sample from three to six months. Patients after complicated mTBI reported lower generic and disease specific HRQoL and worse functional outcome compared to individuals after uncomplicated mTBI at three and six months. Both groups showed a tendency to improve from three to six months after TBI. The complicated mTBI group included more patients with an impaired long-term outcome than the uncomplicated group. Nevertheless, patients, clinicians, researchers, and decisions-makers in health care should take account of the short and long-term impact on outcome for patients after both uncomplicated and complicated mTBI.

Post-Concussion Symptoms in Complicated vs. Uncomplicated Mild Traumatic Brain Injury Patients at Three and Six Months Post-Injury: Results from the CENTER-TBI Study.

The aim of this study was to assess the occurrence of post-concussion symptoms and post-concussion syndrome (PCS) in a large cohort of patients after complicated and uncomplicated mild traumatic brain injury (mTBI) at three and six months post-injury. Patients were included through the prospective cohort study: Collaborative European NeuroTrauma Effectiveness Research (CENTER-TBI). Patients enrolled with mTBI (Glasgow Coma Scale 13-15) were further differentiated into complicated and uncomplicated mTBI based on the presence or absence of computed tomography abnormalities, respectively. The Rivermead Post-Concussion Symptoms Questionnaire (RPQ) assessed post-concussion symptoms and PCS according to the mapped ICD-10 classification method. The occurrence of post-concussion symptoms and syndrome at both time points was calculated. Chi square tests were used to test for differences between and within groups. Logistic regression was performed to analyse the association between complicated versus uncomplicated mTBI and the prevalence of PCS. Patients after complicated mTBI reported slightly more post-concussion symptoms compared to those after uncomplicated mTBI. A higher percentage of patients after complicated mTBI were classified as having PCS at three (complicated: 46% vs. uncomplicated: 35%) and six months (complicated: 43% vs. uncomplicated 34%). After adjusting for baseline covariates, the effect of complicated versus uncomplicated mTBI at three months appeared minimal: odds ratio 1.25 (95% confidence interval: 0.95-1.66). Although patients after complicated mTBI report slightly more post-concussion symptoms and show higher PCS rates compared to those after uncomplicated mTBI at three and six months, complicated mTBI was only found a weak indicator for these problems.

The psychometric validation of the Dutch version of the Rivermead Post-Concussion Symptoms Questionnaire (RPQ) after traumatic brain injury (TBI).

BACKGROUND: Traumatic brain injury (TBI) is one of the most common neurological conditions. It can have wide-ranging physical, cognitive and psychosocial effects. Most people recover within weeks to months after the injury, but a substantial proportion are at risk of developing lasting post-concussion symptoms. The Rivermead Post-Concussion Syndrome Questionnaire (RPQ) is a short validated 16-items self-report instrument to evaluate post-concussive symptoms. The aim of this study was to test psychometrics characteristics of the current Dutch translation of the RPQ. METHODS: To determine the psychometric characteristics of the Dutch RPQ, 472 consecutive patients six months after they presented with a traumatic brain injury in seven medical centers in the Netherlands (N = 397), and in two in Belgium (Flanders) (N = 75) took part in the study which is part of the large prospective longitudinal observational CENTER-TBI-EU-study. Psychometric properties at six months post TBI, were assessed using exploratory and confirmatory factor analyses. Sensitivity was analyzed by comparing RPQ scores and self-reported recovery status of patients with mild vs. moderate and severe TBI. FINDINGS: The Dutch version of RPQ proved good, showing excellent psychometric characteristics: high internal consistency (Cronbach's α .93), and good construct validity, being sensitive to self-reported recovery status at six months post TBI. Moreover, data showed a good fit to the three dimensional structure of separate cognitive, emotional and somatic factors (Chi2 = 119; df = 117; p = .4; CFI = .99; RMSEA = .006), reported earlier in the literature. DISCUSSION: Psychometric characteristics of the Dutch version of RPQ proved excellent to good, and can the instrument therefore be applied for research purposes and in daily clinical practice.

Measuring patient outcomes in chronic heart failure: psychometric properties of the Care-Related Quality of Life survey for Chronic Heart Failure (CaReQoL CHF).

BACKGROUND: The Care-Related Quality of Life survey for Chronic Heart Failure (CaReQoL CHF) is a newly developed patient-reported outcome measure (PROM) that measures care-related quality of life in patients diagnosed with chronic heart failure. This study describes the psychometric properties of the questionnaire and its relationship with disease severity and global rating of quality of care. METHOD: Insurance companies selected patients with a recorded diagnosis of chronic heart failure and for whom the hospital submitted a billing statement in the last year. Exploratory factor analysis, Cronbach's alpha and item-rest correlation were used to construct the CaReQoL CHF. Construct validity was assessed by examining the mean values of the CaReQoL CHF scales for the categories of the widely-used New York Heart Association (NYHA) functional classification and by correlating the global rating of quality of care with the CaReQoL CHF scales. RESULTS: One thousand eighteen patients with chronic heart failure filled out the CaReQoL CHF (RR: 35.7%). The CaReQoL CHF consists of 20 items and three scales: social and emotional problems, physical limitations, and being in safe hands. The mean scores of the three scales differed significantly for the NYHA categories, particularly for the social-emotional problems and physical limitation scales. The 'being in safe hands' scale showed a moderate positive correlation with the global rating of quality of care. CONCLUSIONS: The CaReQoL CHF is a concise and valid PROM that matches patients' priorities in healthcare. It adds a new element to existing quality of life questionnaires for patients with chronic heart failure, that is 'being in safe hands' scale. This scale is relevant for patients because they experience anxiety and tension about their condition. Future research should determine whether the CaReQoL CHF can help healthcare providers in daily practice to focus treatment on outcomes of care that are relevant to individual patients.

Clients' perspective on quality of audiology care: Development of the Consumer Quality Index (CQI) 'Audiology Care' for measuring client experiences.

OBJECTIVE: Clients' perspective on the quality of audiology care has not been investigated thoroughly. Research has focused primarily on satisfaction with, and limitations of hearing aids. We developed a Consumer Quality Index (CQI) questionnaire 'Audiology Care' to systematically assess client experiences with audiology care. DESIGN: The CQI Audiology Care was developed in three steps: (1) posing open-ended questions through e-mail (n = 14), (2) two small-scale surveys assessing psychometric properties of the questionnaire (n = 188) and importance of quality aspects (n = 118), and (3) a large-scale survey (n = 1793) assessing psychometric properties and discriminatory power of the questionnaire. STUDY SAMPLE: People with complex hearing impairments and/or balance and communicative disorders who visited an audiology care centre during the past year. RESULTS: Important quality aspects were translated into seven reliable scales: accommodation and facilities, employees' conduct and expertise, arrangement of appointments, waiting times, client participation and effectiveness of treatment. Client experiences differed among the participating centres concerning accommodation and facilities, arrangement of appointments, waiting times and client participation. CONCLUSION: The CQI Audiology Care is a valid and reliable instrument to assess clients' experiences with audiology care. Future implementation will reveal whether results can be used to monitor and improve the quality of audiology care.

Implementation intention and planning interventions in Health Psychology: Recommendations from the Synergy Expert Group for research and practice.

The current article details a position statement and recommendations for future research and practice on planning and implementation intentions in health contexts endorsed by the Synergy Expert Group. The group comprised world-leading researchers in health and social psychology and behavioural medicine who convened to discuss priority issues in planning interventions in health contexts and develop a set of recommendations for future research and practice. The expert group adopted a nominal groups approach and voting system to elicit and structure priority issues in planning interventions and implementation intentions research. Forty-two priority issues identified in initial discussions were further condensed to 18 key issues, including definitions of planning and implementation intentions and 17 priority research areas. Each issue was subjected to voting for consensus among group members and formed the basis of the position statement and recommendations. Specifically, the expert group endorsed statements and recommendations in the following areas: generic definition of planning and specific definition of implementation intentions, recommendations for better testing of mechanisms, guidance on testing the effects of moderators of planning interventions, recommendations on the social aspects of planning interventions, identification of the preconditions that moderate effectiveness of planning interventions and recommendations for research on how people use plans.

A case study of haemoglobinopathy screening in the Netherlands: witnessing the past, lessons for the future.

OBJECTIVES: In 2007 neonatal screening (NNS) was expanded to include screening for sickle cell disease (SCD) and beta-thalassaemia. Up until that year no formal recommendations for haemoglobinopathy (carrier) screening existed in the Netherlands. Although it has been subject to debate in the past, preconceptional and prenatal haemoglobinopathy carrier screening are not part of routine healthcare in the Netherlands. This study aimed to explore the decision-making process of the past: why was the introduction of a screening programme for haemoglobinopathy considered to be untimely, and did ethnicity play a role given the history in other countries surrounding the introduction of haemoglobinopathy screening? DESIGN: A witness seminar was organised, inviting key figures to discuss the decision-making process concerning haemoglobinopathy screening in the Netherlands, thereby adding new perspectives on past events. The transcript was content-analysed. RESULTS: The subject of haemoglobinopathy screening first appeared in the 1970s. As opposed to a long history of neglect of African-American health in the United States, the heritage of the Second World War influenced the decision-making process in the Netherlands. As a consequence, registration of ethnicity surfaced as an impeding factor. However, overall, official Dutch screening policy was restrained regarding reproductive issues caused by fear of eugenics. In the 1990s haemoglobinopathy screening was found to be 'not opportune' due to low prevalence, lack of knowledge and fear of stigmatisation. Currently the registration of ethnicity remains on the political agenda, but still proves to be a sensitive subject. DISCUSSION: Carrier screening in general never appeared high on the policy agenda. Registration of ethnicity remains sensitive caused by the current political climate. Complexities related to carrier screening are a challenge in Dutch healthcare. Whether carrier screening will be considered a valuable complementary strategy in the Netherlands, depends partly on participation of representatives of high-risk groups in policy making.

Feasibility of preconception screening for thalassaemia in Indonesia: exploring the opinion of Javanese mothers.

BACKGROUND: Thalassaemia has become a major public health issue in Indonesia. It has been estimated that up to 10% of the population carries a gene associated with beta-thalassaemia. Currently, there is no formal recommendation for thalassaemia screening. This study aimed to explore awareness of thalassaemia, and to explore attitudes regarding carrier testing among Javanese mothers. METHODS: A quantitative questionnaire, designed using constructs of the Theory of Planned Behaviour, was applied cross-sectionally. RESULTS: Out of 191 mothers who were invited, 180 agreed to participate (RR = 94%), of whom 74 had a child affected with thalassaemia. Both attitudes towards receiving information about thalassaemia, and attitudes towards carrier testing were very positive. Awareness of thalassaemia was poor. Mothers, both those with and without an affected child, had barely heard of thalassaemia, nor of carrier testing. However, all mothers, including those with an affected child expressed high levels of interest in carrier testing. Respondents did not perceive that they had any control over carrier testing, and feared stigmatization and being discriminated against if their carrier status was identified. Attitudes towards carrier testing explained 23% of future reproductive intentions, in addition to perceived stigmatization, education level and 'mother's age' (R (2)=0.44; p=0.001). CONCLUSION: Responding mothers expressed high levels of interest in receiving information on both thalassaemia and carrier testing. The less educated and the more deprived they were, the keener they were to receive this information. Overall, awareness of thalassaemia was low. Even mothers with affected children seemed unaware of the inheritance pattern and the recurrent risk of having an affected child in a subsequent pregnancy, showing the need for genetic counselling in Indonesia. It is therefore recommended not only to raise awareness about thalassaemia, but to improve the education of healthcare professionals as well.

General practitioner management of genetic aspects of a cardiac disease: a scenario-based study to anticipate providers' practices.

It is increasingly recognised that genetics will have to be integrated into all parts of primary health care. Previous research has demonstrated that involvement and confidence in genetics varies amongst primary care providers. We aimed to analyse perceptions of primary care providers regarding responsibility for genetic tasks and factors affecting those perceptions. Postal questionnaire including a hypothetical case management scenario of a cardiac condition with a genetic component was sent to random samples of medically qualified general practitioners in France, Germany, Netherlands, Sweden and UK (n = 1,168). Logistic regression analysis of factors affecting primary care practitioners' willingness to carry out genetic tasks themselves was conducted; 61% would take a family history themselves but only 38% would explain an inheritance pattern and 16% would order a genetic test. In multivariate analysis, only the country of practice was consistently predictive of willingness to carry out genetic tasks, although male gender predicted willingness to carry out the majority of tasks studied. The stage of career at which education in genetics had been provided was not predictive of willingness to carry out any of the tasks analysed. Country of practice is significantly predictive of attitudes towards genetics in primary care practice and therefore genetic education structure and content in Europe will need to be significantly tailored towards country-specific approaches.

Neonatal screening for treatable and untreatable disorders: prospective parents' opinions.

OBJECTIVE: In the Netherlands, in 2007, the national newborn screening program was expanded from 3 to 17 disorders that met the World Health Organization's Wilson and Jungner screening criteria, especially regarding treatability. The decision of whether to add diseases to the program is generally based on experts' advice, whereas the opinion of those whom it concerns--prospective parents--remains unknown. In this study, we investigated the opinion of prospective parents concerning newborn screening for disorders that are incurable yet treatable to some extent or even untreatable. METHODS: A structured questionnaire that consisted of 3 parts in which similar questions were posed about treatable, less treatable, and untreatable childhood-onset disorders was posted on the Web site of a national pregnancy fair. RESULTS: A total of 1631 prospective parents filled out the questionnaire, 259 of whom were excluded. In contrast to current policy, respondents showed a positive attitude toward inclusion of less treatable (88%) or untreatable childhood-onset disorders (73%) within the national newborn screening program. Respondents who already had children at the time of completing the questionnaire were even more in favor of screening for especially untreatable disorders. The most important reason mentioned was to prevent a long diagnostic quest. Obtaining information to enable reproductive choices in future pregnancies was hardly mentioned. CONCLUSIONS: Prospective parents in the Dutch population seem interested in newborn screening for untreatable childhood-onset disorders; therefore, we argue that additional debate of pros and cons is needed among policy makers, health care professionals, and consumers.

Raising awareness of carrier testing for hereditary haemoglobinopathies in high-risk ethnic groups in the Netherlands: a pilot study among the general public and primary care providers.

BACKGROUND: In the Netherlands no formal recommendations exist concerning preconceptional or antenatal testing for carriership of hereditary haemoglobinopathies. Those at highest risk may be unaware of the possibility of carrier screening. While universal newborn screening has recently been introduced, neither preconceptional nor antenatal carrier testing is routinely offered by health care services to the general public. A municipal health service and a foundation for public information on medical genetics undertook a pilot project with the aim of increasing knowledge and encouraging informed choice. Two groups were targeted: members of the public from ethnic groups at increased risk, and primary health care providers. This study examines the effectiveness of culturally specific 'infotainment' to inform high-risk ethnic groups about their increased risk for haemoglobinopathies. In addition, the study explores attitudes and intentions of primary care providers towards haemoglobinopathy carrier testing of their patients from high-risk ethnic groups. METHODS: Informational sessions tailored to the public or professionals were organised in Amsterdam, and evaluated for their effect. Psychological parameters were measured using structured questionnaires based on the Theory of Planned Behaviour. RESULTS: The pre-test/post-test questionnaire showed that members of the public gained understanding of inheritance and carriership of haemoglobinopathies from the "infotainment" session (p < 0.01). Perceived behavioural control, i.e. the feeling that they could actually get tested if they wanted to, increased in the targeted age group of 18-45 years (N = 41; p < 0.05). 191 surveys were collected from general practitioners or midwives. Their attitude towards the education programme for high-risk ethnic groups was positive, yet they did not show strong intention to effectuate carrier testing of their patients on the basis of ethnicity. The main factor which explained their (lack of) intention was social norm, i.e. their perception of negative peer opinion (41% variance explained). The majority of primary health care providers felt that policy change was unnecessary. CONCLUSION: The "infotainment" programme may have a positive effect on people from high-risk groups, but informed general practitioners and midwives were reluctant to facilitate their patients' getting tested. Additional initiatives are needed to motivate primary care providers to facilitate haemoglobinopathy carrier testing for their patients from high-risk backgrounds.

Facilitating the use of COBRA combination therapy in early rheumatoid arthritis: a pilot implementation study.

OBJECTIVE: COBRA combination therapy is well known and has uncontested efficacy in the treatment of rheumatoid arthritis (RA). However, it is infrequently applied in Dutch clinical practice. Based on qualitative research on opinions of physicians and patients towards COBRA therapy, our study describes the development and pilot testing of an implementation package to facilitate prescription and use of COBRA therapy in early RA. METHODS: The implementation package was developed to address specific barriers towards prescription of COBRA therapy and comprised informational handouts (an information booklet and leaflet for patients), preprinted prescription orders, and background information on COBRA therapy for the rheumatologists. Twenty-two rheumatologists agreed to participate, including the arthritis nurse where available. Rheumatologists, nurses, and patients were asked to record their experience. All Dutch arthritis nurses were invited to an educational session on COBRA therapy. RESULTS: Sixteen rheumatologists accompanied by 10 arthritis nurses used the material to prescribe COBRA therapy to a total of 27 patients. Rheumatologists and nurses both gave high marks to the supplied materials. Eighty-eight percent of rheumatologists reported that the material sped up the prescription process, and 65% indicated they would prescribe COBRA therapy more frequently if these materials were available routinely. Patients expressed great satisfaction with the information handouts, rating it 2.8 (standard deviation 0.5) on a scale of -3 (very negative) to +3 (very positive). Most patients (89%) planned to keep the information booklet as a reference and 70% used it as a tool to remember the correct intake of medication. The attitude and perceived capability of nurses towards the guidance of patients with RA receiving COBRA therapy was improved through a brief educational intervention. CONCLUSION: Rheumatologists, patients, and arthritis nurses all highly appreciated the implementation package and indicated that its availability would increase uptake of COBRA therapy.

Testing the children: do non-genetic health-care providers differ in their decision to advise genetic presymptomatic testing on minors? A cross-sectional study in five countries in the European Union.

BACKGROUND: Within Europe many guidelines exist regarding the genetic testing of minors. Predictive and presymptomatic genetic testing of minors is recommended for disorders for which medical intervention/preventive measures exist, and for which early detection improves future medical health. AIM: This study, which is part of the larger 5th EU-framework "genetic education" (GenEd) study, aimed to evaluate the self-reported responses of nongenetic health-care providers in five different EU countries (Germany, France, Sweden, the United Kingdom, and the Netherlands) when confronted with a parent requesting presymptomatic testing on a minor child for a treatable disease. METHODS: A cross-sectional study design using postal, structured scenario-based questionnaires that were sent to 8129 general practitioners (GPs) and pediatricians, between July 2004 and October 2004, addressing self-reported management of a genetic case for which early medical intervention during childhood is beneficial, involving a minor. RESULTS: Most practitioners agreed on testing the oldest child, aged 12 years (81.5% for GPs and 87.2% for pediatricians), and not testing the youngest child, aged 6 months (72.6% for GPs and 61.3% for pediatricians). After multivariate adjustment there were statistical differences between countries in recommending a genetic test for the child at the age of 8 years. Pediatricians in France (50%) and Germany (58%) would recommend a test, whereas in the United Kingdom (22%), Sweden (30%), and the Netherlands (32%) they would not. CONCLUSION: Even though presymptomatic genetic testing in minors is recommended for disorders for which medical intervention exists, EU physicians are uncertain at what age starting to do so in young children.