Ilona Autti-Rämö

Toward a neurobehavioral profile of fetal alcohol spectrum disorders

BACKGROUNDnA primary goal of recent research is the development of neurobehavioral profiles that specifically define fetal alcohol spectrum disorders (FASD), which may assist differential diagnosis or improve treatment. In the current study, we define a preliminary profile using neuropsychological data from a multisite study.nnnMETHODSnData were collected using a broad neurobehavioral protocol from 2 sites of a multisite study of FASD. Subjects were children with heavy prenatal alcohol exposure and unexposed controls. The alcohol-exposed group included children with and without fetal alcohol syndrome (FAS). From 547 neuropsychological variables, 22 variables were selected for analysis based on their ability to distinguish children with heavy prenatal alcohol exposure from nonexposed controls. These data were analyzed using latent profile analysis (LPA).nnnRESULTSnThe results indicated that a 2-class model best fit the data. The resulting profile was successful at distinguishing subjects with FAS from nonexposed controls without FAS with 92% overall accuracy; 87.8% of FAS cases and 95.7% of controls were correctly classified. The same analysis was repeated with children with heavy prenatal alcohol exposure but without FAS and nonexposed controls with similar results. The overall accuracy was 84.7%; 68.4% of alcohol-exposed cases and 95% of controls were correctly classified. In both analyses, the profile based on neuropsychological variables was more successful at distinguishing the groups than was IQ alone.nnnCONCLUSIONSnWe used data from 2 sites of a multisite study and a broad neuropsychological test battery to determine a profile that could be used to accurately identify children affected by prenatal alcohol exposure. Results indicated that measures of executive function and spatial processing are especially sensitive to prenatal alcohol exposure.

The HTA Core Model: A novel method for producing and reporting health technology assessments

OBJECTIVESnThe aim of this study was to develop and test a generic framework to enable international collaboration for producing and sharing results of health technology assessments (HTAs).nnnMETHODSnTen international teams constructed the HTA Core Model, dividing information contained in a comprehensive HTA into standardized pieces, the assessment elements. Each element contains a generic issue that is translated into practical research questions while performing an assessment. Elements were described in detail in element cards. Two pilot assessments, designated as Core HTAs were also produced. The Model and Core HTAs were both validated. Guidance on the use of the HTA Core Model was compiled into a Handbook.nnnRESULTSnThe HTA Core Model considers health technologies through nine domains. Two applications of the Model were developed, one for medical and surgical interventions and another for diagnostic technologies. Two Core HTAs were produced in parallel with developing the model, providing the first real-life testing of the Model and input for further development. The results of formal validation and public feedback were primarily positive. Development needs were also identified and considered. An online Handbook is available.nnnCONCLUSIONSnThe HTA Core Model is a novel approach to HTA. It enables effective international production and sharing of HTA results in a structured format. The face validity of the Model was confirmed during the project, but further testing and refining are needed to ensure optimal usefulness and user-friendliness. Core HTAs are intended to serve as a basis for local HTA reports. Core HTAs do not contain recommendations on technology use.

Prenatal alcohol exposure alters the patterns of facial asymmetry

Directional asymmetry, the systematic differences between the left and right body sides, is widespread in human populations. Changes in directional asymmetry are associated with various disorders that affect craniofacial development. Because facial dysmorphology is a key criterion for diagnosing fetal alcohol syndrome (FAS), the question arises whether in utero alcohol exposure alters directional asymmetry in the face. Data on the relative position of 17 morphologic landmarks were obtained from facial scans of children who were classified as either FAS or control. Shape data obtained from the landmarks were analyzed with the methods of geometric morphometrics. Our analyses showed significant directional asymmetry of facial shape, consisting primarily of a shift of midline landmarks to the right and a displacement of the landmarks around the eyes to the left. The asymmetry of FAS and control groups differed significantly and average directional asymmetry was increased in those individuals exposed to alcohol in utero. These results suggest that the developmental consequences of fetal alcohol exposure affect a wide range of craniofacial features in addition to those generally recognized and used for diagnosis of FAS.

Collaborative initiative on fetal alcohol spectrum disorders: methodology of clinical projects

The Collaborative Initiative on Fetal Alcohol Spectrum Disorders (CIFASD) was created in 2003 to further understanding of fetal alcohol spectrum disorders. Clinical and basic science projects collect data across multiple sites using standardized methodology. This article describes the methodology being used by the clinical projects that pertain to assessment of children and adolescents. Domains being addressed are dysmorphology, neurobehavior, 3-D facial imaging, and brain imaging.

Two sides of the mirror: parents' and service providers' view on the family-centredness of care for children with cerebral palsy.

BACKGROUNDnIn order to best meet the needs of both families and their children with cerebral palsy, many rehabilitation service providers have adopted a family-centred service (FCS) approach. In FCS parents are seen as experts on their childs needs, and the family and professionals collaborate in the rehabilitation process. However, parents and service providers might look at FCS from different points of view, i.e. look into the mirror from two different sides. The objective of this study was to explore the degree to which parents experience the service as being family-centred and to which extent the service providers experience their service provision as family-centred.nnnMETHODSnA translated version of The Measure of Processes of Care 20 (MPOC-20) questionnaire was used to evaluate parents experience of FCS, and a Measures of Processes of Care for Service Providers (MPOC-SP) questionnaire was used to evaluate the FCS provided by professionals. Parents visiting two university hospital neuropediatric wards (n= 67) during a 2-month period and who were willing to participate received the questionnaire. Also the service providers working on the same wards (n= 49) were invited to participate.nnnRESULTSnA total of 53 families and 29 service providers completed the questionnaires. Both parents and professionals generally rated the FCS positively. General information was rated lowest and respectful treatment the highest by both parents and professionals. The results revealed that written information about the childs condition, the possibility to choose when to receive information, and contact with other families in the same situation are areas in need of improvement.nnnCONCLUSIONSnThe possibility to regularly evaluate services both from the families and the professionals perspectives should be part of quality development. Providing general information is a challenge for all service providers. The MPOC questionnaires can be used to highlight important areas of improvement in FCS.

Evidence on physiotherapeutic interventions for adults with cerebral palsy is sparse. A systematic review

Objectives: To identify evidence evaluating the effectiveness of physiotherapy in adolescents (>16 years of age) and adults with cerebral palsy. Data sources: Systematic literature search from the earliest available time until March 2009. Additional studies were identified through reference and citation tracking. Review methods: Two reviewers independently agreed on eligibility, methodological quality and quality of evidence assessment. Standard methods were used for quality assessments. Results: Included were 13 studies, two of which were randomized controlled trials. No article met the criteria for high methodological quality. Evidence of moderate quality was found on gait after strength training. Evidence of low quality was found on balance after strength training and workstation interventions. Low-quality evidence was also found on functionality after strength training in four studies evaluating gross motor capacity. There was very low-quality evidence on increased muscle strength and in outcome measures used to evaluate range of motion. Conclusion: Evidence for the effect of physiotherapy on adolescents and adults with cerebral palsy is sparse, and therefore there is an urgent need for well-designed physiotherapeutic trials for these people.

Factors associated with the utilization and costs of health and social services in frail elderly patients

BackgroundUniversal access is one of the major aims in public health and social care. Services should be provided on the basis of individual needs. However, municipal autonomy and the fragmentation of services may jeopardize universal access and lead to variation between municipalities in the delivery of services. This paper aims to identify patient-level characteristics and municipality-level service patterns that may have an influence on the use and costs of health and social services of frail elderly patients.MethodsHierarchical analysis was applied to estimate the effects of patient and municipality-level variables on services utilization.ResultsThe variation in the use of health care services was entirely due to patient-related variables, whereas in the social services, 9% of the variation was explained by the municipality-level and 91% by the patient-level characteristics. Health-related quality of life explained a major part of variation in the costs of health care services. Those who had reported improvement in their health status during the preceding year were more frequent users of social care services. Low informal support, poor functional status and poor instrumental activities of daily living, living at a residential home, and living alone were associated with higher social services expenditure.ConclusionsThe results of this study showed municipality-level variation in the utilization of social services, whereas health care services provided for frail elderly people seem to be highly equitable across municipalities.Another important finding was that the utilization of social and health services were connected. Those who reported improvement in their health status during the preceding year were more frequently also using social services. This result suggests that if municipalities continue to limit the provision of support services only for those who are in the highest need, this saving in the social sector may, in the long run, result in increased costs of health care.

Smoking strongly predicts disability retirement due to COPD: the Finnish Twin Cohort Study

No previous studies on the association of smoking behaviour with disability retirement due to register verified chronic obstructive pulmonary disease (COPD) exist. This 30-yr follow-up study examined how strongly aspects of cigarette smoking predict disability retirement due to COPD. The study population consisted of 24,043 adult Finnish twins (49.7% females) followed from 1975 to 2004. At baseline the participants had responded to a questionnaire. Information on retirement was obtained from the Finnish pension registers. Smoking strongly predicted disability retirement due to COPD. In comparison to never-smokers, age adjusted hazard ratio (HR) for current smokers was 22.0 (95% CI 10.0–48.5) and for smokers with ≥12 pack-yrs was 27.3 (95% CI 12.6–59.5). Similar estimates of risk were observed in within-pair analyses of twin pairs discordant for disability retirement due to COPD. Among discordant monozygotic pairs those with disability pension due to COPD were more often current smokers. The effect of early smoking onset (<18 yrs) on the risk of disability retirement due to COPD remained after adjustment for the amount smoked (HR 1.70, 95% CI 1.08–2.68). Smoking strongly predicts disability retirement due to COPD. Preventive measures against disability retirement and other harmful consequences of tobacco smoking should receive greater emphasis.

Professional background and the comprehension of family-centredness of rehabilitation for children with cerebral palsy

BACKGROUNDnChildren with cerebral palsy have difficulties in several areas of functioning, and they need long-lasting rehabilitation with a clear focus on the individuals needs. Finnish guidelines emphasize family-centred service. The values of family-centred service are widely known, but how the principles of family-centred service are adopted in clinical practice is not well documented. The objective of this study was to analyse the family-centred behaviour of professionals working with children and adolescents with cerebral palsy.nnnMETHODSnA translated version of the Measure of Processes of Care for Service Providers (MPOC-SP) questionnaire was used to evaluate the family-centred service. The questionnaire was sent to all the professionals in the multidisciplinary rehabilitation teams at all the hospitals and governmental special schools treating children and adolescents with cerebral palsy in Finland (n= 327). Furthermore, 438 physiotherapy service providers working in the childrens home region were invited to participate.nnnRESULTSnA total of 201 multidisciplinary team members and 311 physiotherapy service providers completed the questionnaire. Both the team members and the service providers generally rated their family-centred behaviour positively. There was statistically significant difference in how the team members in the multidisciplinary teams self-assessed their family-centred service. Physiotherapists working in multidisciplinary teams rated their family-centred service higher than physiotherapy service providers. The professionals apprehension of family-centred service increased with work experience.nnnCONCLUSIONSnProfessional background and professional context seem to affect the apprehension of family-centred service. Also work experience and being part of a multidisciplinary team have an influence on how the professionals embrace the family-centred service delivered. The MPOC-SP can be used to identify areas for improvement.

Maternal welfare, morbidity and mortality 6-15 years after a pregnancy complicated by alcohol and substance abuse: a register-based case-control follow-up study of 524 women.

BACKGROUNDnA register-based retrospective case-control study to investigate the long-term morbidity, mortality, and welfare among women with alcohol and/or substance misuse identified during pregnancy.nnnMETHODSnCohort of 524 women followed-up ante- and perinatally 1992-2001 at special out-patient clinics of maternity hospitals in the capital area of Finland. The control group of 1792 women with no evidence of alcohol or substance misuse was matched for maternal age, parity, date of birth and hospital of index delivery. Both groups were followed-up until end of 2007.nnnRESULTSn7.9% (42/524) of the cases and 0.2% (4/1792) of the controls had died by the end of the median follow-up of 9 years (OR 38, 95% CI 14-108). The cases displayed significant morbidity requiring in-patient care in the areas of mental disorders (AOR 8.8, 95% CI 6.5-11.9), viral (AOR 23.5, 95% CI 8.8-62.7) and bacterial (AOR 6.1, 95% CI 3.5-10.4) infections, skin diseases (AOR 3.9, 95% CI 2.0-7.8) and injury and poisoning (AOR 4.2, 95% CI 3.1-5.6). The cases displayed more out-patient visits (OR 2.7, 95% CI 2.7-2.8). Their mean length of hospital stay was longer compared to controls (10.3 vs. 4.4 days, p<0.001). The risk of pension granted due to a disorder, disease or disability (OR 8.8, 95% CI 6.0-13.0) and the risk for minimum unemployment benefit were higher compared to controls (OR 2.1, 95% CI 1.8-2.5).nnnCONCLUSIONSnThese women display significant long-term morbidity, mortality and loss of productivity after delivery. The results emphasize the importance of adequate postnatal follow-up and treatment for misuse.