The Swiss Primary Hypersomnolence and Narcolepsy Cohort study (SPHYNCS): Study protocol for a prospective, multicentre cohort observational study.

Narcolepsy type 1 (NT1) is a disorder with well-established markers and a suspected autoimmune aetiology. Conversely, the narcoleptic borderland (NBL) disorders, including narcolepsy type 2, idiopathic hypersomnia, insufficient sleep syndrome and hypersomnia associated with a psychiatric disorder, lack well-defined markers and remain controversial in terms of aetiology, diagnosis and management. The Swiss Primary Hypersomnolence and Narcolepsy Cohort Study (SPHYNCS) is a comprehensive multicentre cohort study, which will investigate the clinical picture, pathophysiology and long-term course of NT1 and the NBL. The primary aim is to validate new and reappraise well-known markers for the characterization of the NBL, facilitating the diagnostic process. Seven Swiss sleep centres, belonging to the Swiss Narcolepsy Network (SNaNe), joined the study and will prospectively enrol over 500 patients with recent onset of excessive daytime sleepiness (EDS), hypersomnia or a suspected central disorder of hypersomnolence (CDH) during a 3-year recruitment phase. Healthy controls and patients with EDS due to severe sleep-disordered breathing, improving after therapy, will represent two control groups of over 50 patients each. Clinical and electrophysiological (polysomnography, multiple sleep latency test, maintenance of wakefulness test) information, and information on psychomotor vigilance and a sustained attention to response task, actigraphy and wearable devices (long-term monitoring), and responses to questionnaires will be collected at baseline and after 6, 12, 24 and 36 months. Potential disease markers will be searched for in blood, cerebrospinal fluid and stool. Analyses will include quantitative hypocretin measurements, proteomics/peptidomics, and immunological, genetic and microbiota studies. SPHYNCS will increase our understanding of CDH and the relationship between NT1 and the NBL. The identification of new disease markers is expected to lead to better and earlier diagnosis, better prognosis and personalized management of CDH.

Parent alcohol problems and peer bullying and victimization: child gender and toddler attachment security as moderators.

This study examined the association between parents' alcoholism and peer bullying and victimization in middle childhood in 162 community-recruited families (80 girls and 82 boys) with and without alcohol problems. Toddler-mother attachment was assessed at 18 months of child age, and child reports of peer bullying and victimization were obtained in 4th grade. There was a direct association between fathers' alcohol symptoms and bullying of peers, as well as indirect association via toddler-mother attachment security. Multiple group models indicated that the direct association between parents' alcohol symptoms and bullying was significant for boys but not girls. The association between maternal alcohol symptoms and bullying was significant for secure but not insecure boys or secure/insecure girls. The association between fathers' alcohol symptoms and bullying was significant for insecure boys but not secure boys or secure/insecure girls.

A longitudinal study of social competence among children of alcoholic and nonalcoholic parents: role of parental psychopathology, parental warmth, and self-regulation.

This study tested a conceptual model predicting children's social competence in a sample of children with alcoholic and non-alcoholic parents. The model examined the role of parents' alcohol diagnoses, depression, and antisocial behavior at 12-18 months of child age in predicting parental warmth/sensitivity at 2 years of child age. Parental warmth/sensitivity at 2 years was hypothesized to predict children's self-regulation and externalizing behavior problems at 3 years. Parenting, self-regulation, and behavior problems were expected to predict social competence in kindergarten. Structural equations modeling was supportive of this model. Fathers' alcohol diagnosis was associated with lower warmth/sensitivity. Lower maternal warmth/sensitivity at 2 years was predictive of lower child self-regulation at 3 years. Parenting, self-regulation, and externalizing behavior problems were predictive of social competence in kindergarten, although associations varied by reporter (parents or teacher). There was a direct association between fathers' alcohol diagnosis and father reports of social competence, and between fathers' depression and teacher reports of social competence. The study elucidates developmental processes in predicting social competence and the role of fathers' alcoholism and associated risk factors in this process. (PsycINFO Database Record (c) 2009 APA, all rights reserved).

A conceptual model for the development of externalizing behavior problems among kindergarten children of alcoholic families: role of parenting and children's self-regulation.

The purpose of this study was to test a conceptual model predicting children's externalizing behavior problems in kindergarten in a sample of children with alcoholic (n = 130) and nonalcoholic (n = 97) parents. The model examined the role of parents' alcohol diagnoses, depression, and antisocial behavior at 12-18 months of child age in predicting parental warmth/sensitivity at 2 years of child age. Parental warmth/sensitivity at 2 years was hypothesized to predict children's self-regulation at 3 years (effortful control and internalization of rules), which in turn was expected to predict externalizing behavior problems in kindergarten. Structural equation modeling was largely supportive of this conceptual model. Fathers' alcohol diagnosis at 12-18 months was associated with lower maternal and paternal warmth/sensitivity at 2 years. Lower maternal warmth/sensitivity was longitudinally predictive of lower child self-regulation at 3 years, which in turn was longitudinally predictive of higher externalizing behavior problems in kindergarten, after controlling for prior behavior problems. There was a direct association between parents' depression and children's externalizing behavior problems. Results indicate that one pathway to higher externalizing behavior problems among children of alcoholics may be via parenting and self-regulation in the toddler to preschool years.

Children's internalization of rules of conduct: role of parenting in alcoholic families.

This study examined the association between fathers' alcoholism and children's internalization of rules of conduct at 2 to 3 years of age. The sample consisted of 220 families (102 without alcoholism, 118 with alcoholism). Results indicated that there was no direct association between fathers' alcoholism and children's internalization measured with a behavioral paradigm at age 3 years. However, the indirect association between fathers' alcoholism and children's behavioral internalization was significant through fathers' sensitivity during play interactions at age 2 years. Children of fathers with alcoholism were rated by their mothers as having lower internalized conduct over the 2- to 3-year period. This direct association was not mediated by parental sensitivity.

The development of aggression in 18 to 48 month old children of alcoholic parents.

This study examined the development of aggressive and oppositional behavior among alcoholic and nonalcoholic families using latent growth modeling. The sample consisted of 226 families assessed at 18, 24, 36, and 48 months of child age. Results indicated that children in families with nonalcoholic parents had the lowest levels of aggressive behavior at all time points compared to children with one or more alcoholic parents. Children in families with two alcoholic parents did not exhibit normative decreases in aggressive behavior from 3 to 4 years of age compared to nonalcoholic families. However, this association was no longer significant once a cumulative family risk score was added to the model. Children in families with high cumulative risk scores, reflective of high parental depression, antisocial behavior, negative affect during play, difficult child temperament, marital conflict, fathers' education, and hours spent in child care, had higher levels of aggression at 18 months than children in low risk families. These associations were moderated by child gender. Boys had higher levels of aggressive behavior at all ages than girls, regardless of group status. Cumulative risk was predictive of higher levels of initial aggressive behavior in both girls and boys. However, boys with two alcoholic parents had significantly less of a decline in aggression from 36 to 48 months compared to boys in the nonalcoholic group.

Predictors of effortful control among children of alcoholic and nonalcoholic fathers.

OBJECTIVE: The purpose of this study was (1) to examine the association between fathers' alcoholism and children's effortful control and (2) to examine the role of parental warmth and toddler temperament as mediators or moderators of this relationship. METHOD: Families were recruited through New York State birth records when their infant was age 12 months. The final sample consisted of 226 families (116 boys) constituting two major groups: a nonalcoholic group consisting of parents with no or few current alcohol problems (n = 102) and a father alcoholic group (n = 124). Families were assessed when their child was ages 12, 18, 24 and 36 months. RESULTS: Results indicate that boys of alcoholic fathers exhibit lower overall levels of effortful control than boys of nonalcoholic fathers. For boys, fathers' warmth over the second year of life mediated the association between fathers' alcoholism and effortful control. Maternal warmth was a unique predictor of effortful control for boys. For girls, fathers' alcoholism was associated with lower paternal warmth, which was in turn a significant predictor of effortful control. Child activity level and negative affect were associated with effortful control for boys but did not account for significant variance when entered in regression models with fathers' alcoholism and parenting variables. CONCLUSIONS: Sons of alcoholic fathers are at an increased risk of problems in self-regulation when they are ages 2 to 3 years. Paternal warmth mediates the association between fathers' alcoholism and self-regulation for both boys and girls, although the nature of mediation may vary by child gender.

IMPACT OF FATHERS' ALCOHOLISM AND ASSOCIATED RISK FACTORS ON PARENT-INFANT ATTACHMENT STABILITY FROM 12 TO 18 MONTHS.

This study examined short-term attachment stability and sought to identify predictors of stability and change within a sample characterized by fathers' alcoholism. Results suggest moderate stability of attachment classifications (60% for mothers, 53% for fathers) from 12 to 18 months. Higher paternal and maternal alcohol symptoms, maternal depression, and maternal antisocial behavior were found in families with stable insecure mother-infant attachment compared to those who were stable secure. Mother-infant stable insecurity was associated with higher levels of maternal negative affect expression during play. Father-infant stable insecurity was associated with lower levels of paternal positive affect expression and decreased sensitivity during play. Stable insecure children also had higher levels of negative affect during parent-infant interactions and higher negative emotionality during other episodes compared to stable secure children. Results indicate that infants who were insecure at both time points had the highest constellation of family risk characteristics.

TEMPERAMENT AND BEHAVIORAL PROBLEMS AMONG INFANTS IN ALCOHOLIC FAMILIES.

This study examined the association between paternal alcoholism and 12-month infant temperament and 18-month behavior problems. The role of associated parental psychopathology and maternal drinking in exacerbating risk for maladaptive behavioral outcomes was also examined. Participants were 213 families (102 control families, 94 paternal alcoholic families, and 17 families with alcoholic fathers and heavy drinking mothers) who were assessed when their child was 12 months old and reassessed again when their child was 18 months old. Infants of alcoholics displayed marginally more stubborn/persistent temperaments at 12 months of age, but significantly more internalizing problems at 18 months. Analyses suggested that internalizing problems in the infants of alcoholics could be attributed to the paternal depression concomitant with paternal alcoholism. In addition, an interaction was observed, indicating that paternal alcohol problems predicted 18-month externalizing problems among families with low maternal depression, but not among families with high maternal depression. Children of depressed mothers exhibited uniformly higher externalizing scores, but were not further impacted by paternal alcohol problems. However, children of nondepressed mothers were adversely affected by fathers' drinking as reflected by higher externalizing behavior scores. The results highlight the necessity of addressing the overall contextual risks that occur with paternal alcoholism in studies of the development of children in alcoholic families.

Analyzing family data: A GEE approach for substance use researchers.

INTRODUCTION: Analyzing data that arises from correlated observations such as husband-wife pairs, siblings, or repeated assessments of the same individuals over time requires more specialized analytic tools. Additionally, outcomes that are not normally distributed such as count data, (e.g., number of symptoms or number of problems endorsed) also require specialized analytic tools. Generalized estimating equations (GEE) are a very flexible tool for dealing with correlated data (such as data derived from related individuals such as families). The objective of this report was to compare traditional ordinary least squares regression (OLS) to a GEE approach for analyzing family data. METHODS: Using data from an ongoing five-wave longitudinal study of newlywed couples, we examined a subset of 173 families with children between the ages of 4 and 11 at two data collection points. The relation between parental risk factors (e.g., heavy drinking, aggression, marital quality) and child internalizing symptoms was examined within the context of two regression-based models: traditional OLS regression and a GEE approach. RESULTS: Overall, the GEE approach allowed a more complete use of the available data, provided more robust findings, and produced more reliable parameter estimates. CONCLUSION: GEE models are a flexible regression-based approach for dealing with related data that arises from correlated data such as family data. Further, given the availability of the models in common statistical programs, family researchers should consider these models for their work.

Marital Aggression and Child Peer Competence: A Comparison of Three Conceptual Models.

This study examined longitudinal data linking marital aggression with child peer competence in kindergarten. The study compared three conceptual models for understanding the relation between marital aggression and child peer competence. Model 1 examines the direct effects of marital aggression, parental alcoholism, and parenting on child peer competence, model 2 posits that this relation is mediated by child social problem solving abilities (social information processing theory), while model 3 proposes that the relation is mediated by parental warmth/sensitivity (spillover theory). Structural Equation Modeling was most supportive of models 1 and 3 indicating that parenting behavior, but not social problem solving, partially mediates the relation between marital conflict and child peer competence.

Alcoholism, associated risk factors, and harsh parenting among fathers: Examining the role of marital aggression.

This study utilized a longitudinal design to examine relations between paternal alcoholism, paternal psychopathology, marital aggression and fathers' harsh parenting behavior in a sample of children with alcoholic (n = 89) and non-alcoholic (n = 94) fathers. Structural Equation Modeling revealed that paternal alcoholism, depression, and antisocial behavior at 12 months of child age each predicted higher levels of marital aggression at 36 months. Moreover, after controlling for prior parenting, marital aggression was predictive of harsher parenting at kindergarten. Alcoholism and psychopathology were not directly predictive of harsh parenting with marital aggression included in the model, thus indicating that marital aggression is mediating the relation between paternal risk factors and parenting outcome. Results of this study suggest that one pathway linking fathers' alcohol diagnosis to harsh parenting is via marital aggression.

A Longitudinal Examination of Physiological Regulation in Cocaine-Exposed Infants Across the First 7 Months of Life.

This study examined the association between prenatal exposure to cocaine and physiological regulation across the first 7 months of age. Measures of respiratory sinus arrhythmia (RSA) were obtained from 169 (82 cocaine-exposed and 87 nonexposed) infants during baseline periods at 1 month and 7 months of age and during tasks designed to elicit positive and negative affect at 7 months of age. After controlling for maternal age, gestational age, and obstetrical risk, structural equation modeling indicated that the association between prenatal exposure to cocaine and baseline RSA at 7 months of age was direct even in the presence of an indirect effect through baseline RSA at 1 month of age. There were no indirect effects through maternal affect during mother-infant interactions assessed at 1 month of age. Analyses also indicated a direct association between prenatal exposure to cocaine and RSA regulation to negative affect at 7 months of age.

Longitudinal prediction of early childhood discipline styles among heavy drinking parents.

This study sought to examine predictors of parenting trajectories in a sample of heavy drinking and abstaining/light drinking parents. Mixture modeling was used to estimate trajectories of parental discipline styles over time. Two dimensions of parenting were examined: laxness and overreactivity. Changes in these dimensions were examined for each parent. Trajectories for mothers and fathers were very similar and were generally stable from 18 months to 5-6 years child age. Fathers' binge drinking was associated with high levels of both paternal and maternal overreactivity. Mothers with depressed affect had the highest levels of overreactivity, whereas fathers reporting depressed affect were more likely to have moderate levels of laxness. Mothers with high levels of marital satisfaction were more likely to have partners in the stable low overreactivity group. Findings begin to elucidate the nature of early family processes that may contribute to maladaptive child outcomes in heavy drinking families.

A conceptual model predicting internalizing problems in middle childhood among children of alcoholic and nonalcoholic fathers: the role of marital aggression.

OBJECTIVE: The purpose of this study was to test a conceptual model predicting children's anxiety/depression in middle childhood in a community sample of children with parents who had alcohol problems (n = 112) and those without alcohol problems (n = 101). The conceptual model examined the role of parents' alcohol diagnoses, depression, and antisocial behavior among parents of children ages 12 months to kindergarten age in predicting marital aggression and parental aggravation. Higher levels of marital aggression and parental aggravation were hypothesized to predict children's depression/anxiety within time (18 months to kindergarten age and, prospectively, to age during fourth grade). METHOD: The sample was recruited from New York State birth records when the children were 12 months old. Assessments were conducted at 12, 18, 24, and 36 months; at kindergarten age; and during fourth grade. RESULTS: Children with alcoholic fathers had higher depression/anxiety scores according to parental reports but not self-reports. Structural equations modeling was largely supportive of the conceptual model. Fathers' alcoholism was associated with higher child anxiety via greater levels of marital aggression among families with alcohol problems. Results also indicated that there was a significant indirect association between parents' depression symptoms and child anxiety via marital aggression. CONCLUSIONS: The results highlight the nested nature of risk characteristics in alcoholic families and the important role of marital aggression in predicting children's anxiety/depression. Interventions targeting both parents' alcohol problems and associated marital aggression are likely to provide the dual benefits of improving family interactions and lowering risk of children's internalizing behavior problems.

Behaviour problems in 18- to 36-month old children of alcoholic fathers: secure mother-infant attachment as a protective factor.

This study examined the relationship between paternal alcoholism and toddler behavior problems from 18 to 36 months of age, as well as the potential moderating effects of 12-month infant-mother attachment security on this relationship. Children with alcoholic fathers had higher levels of internalizing and externalizing behavior than children of nonalcoholic fathers. Simple effects testing of an interaction effect of child age, group, and attachment security with mothers on externalizing behaviour suggested that at 24 and 36 months of age mother-infant attachment security moderated the relationship between alcohol group status and externalizing behaviour. Namely, within the alcohol group, those children with secure relationships with their mothers had significantly lower externalizing than insecure children of alcoholics. A similar pattern was noted for internalizing behavior at 36 months of age. Implications for intervention are discussed.

Mother-infant and father-infant attachment among alcoholic families.

This study examined the association between fathers' alcoholism and other risk factors such as parental depression, family conflict, infant temperament, and parent-infant attachment. The quality of parent-infant interactions was hypothesized to be a proximal mediator of the associations among alcoholism and other risk factors and attachment. The participants were 223 families (104 nonalcoholic families and 119 alcoholic families) with 12-month-old infants recruited through birth records. Infants in families with two parents with alcohol problem had significantly higher rates of insecure attachment with both parents. Structural Equations Modeling indicated that the fathers' alcohol problem was associated with lower paternal sensitivity (higher negative affect, lower positive engagement, and lower sensitive responding) during father-infant play interactions, and this in tum was associated with higher risk for infant attachment insecurity with fathers. The association between the fathers' alcohol problem and infant attachment security with the mother was mediated by matemal depression, and matemal alcohol problems and family conflict were associated with maternal sensitivity during play interactions. These results indicate that the fathers' alcoholism is associated with higher family risk including the quality of the parent-infant relationship; infant attachment develops in a family context; and this context has a significant association with attachment security.