Design of a Multisite Study Assessing the Impact of Tic Disorders on Individuals, Families, and Communities.

BACKGROUND: Tic disorders, including Tourette syndrome, are complex, multisymptom diseases, yet the impact of these disorders on affected children, families, and communities is not well understood. METHODS: To improve the understanding of the impacts of Tourette syndrome, two research groups conducted independent cross-sectional studies using qualitative and quantitative measures. They focused on similar themes, but distinct scientific objectives, and the sites collaborated to align methods of independent research proposals with the aim of increasing the analyzable sample size. RESULTS: Site 1 (University of Rochester) was a Pediatric Neurology referral center. Site 2 (University of South Florida) was a Child Psychiatry referral center. A total of 205 children with tic disorders were enrolled from both studies. The University of Rochester also enrolled 100 control children in order to clearly isolate impacts of Tourette syndrome distinct from those occurring in the general population. The majority of children with tic disorders (n = 191, 93.1%) had Tourette syndrome, the primary population targeted for these studies. Children with Tourette syndrome were similar across sites in terms of tic severity and the occurrence of comorbid conditions. The occurrence of psychiatric comorbidities in the control group was comparable with that in the general pediatric population of the United States, making this a well-justified comparison group. CONCLUSIONS: Through collaboration, two sites conducting independent research developed convergent research methods to enable pooling of data, and by extension increased power, for future analyses. This method of collaboration is a novel model for future epidemiological research of tic disorders.

Structure and clinical correlates of obsessive-compulsive symptoms in a large sample of children and adolescents: a factor analytic study across five nations.

The underlying structure of obsessive-compulsive disorder (OCD) remains to be confirmed in child and adolescent populations. In this paper we report the first factor analytic study of individual OCD items from Children's Yale-Brown Obsessive Compulsive Scale (CY-BOCS). OCD symptoms were assessed using the CY-BOCS symptom checklist in a sample of 854 patients with OCD (7-18 years of age) recruited from clinics in five countries. Pooled data were subjected to exploratory and confirmatory factor analysis (CFA) to identify the optimal factor structure. Various models were tested for age and gender subgroups. Also, the invariance of the solution across age and gender was tested and associations with demographic and clinical factors were explored. A three-factor model provided the best-fit solution. It consisted of the following factors: (1) harm/sexual, (2) symmetry/hoarding, (3) contamination/cleaning. The factor structure was invariant for age and gender across subgroups. Factor one was significantly correlated with anxiety, and factor two with depression and anxiety. Factor three was negatively correlated with tic disorder and attention-deficit/hyperactivity disorder (ADHD). Females had higher scores on factor two than males. The OCD symptom structure in children and adolescents is consistent across age and gender and similar to results from recent child and adolescents although hoarding may not be a separate factor. Our three-factor structure is almost identical to that seen in early studies on adults. Common mental disorders had specific patterns of associations with the different factors.

Maternal history of autoimmune disease in children presenting with tics and/or obsessive-compulsive disorder.

OBJECTIVES: A commonality across a number of pediatric neuropsychiatric disorders is a higher than typical rate of familial - and especially maternal - autoimmune disease. Of recent interest, a subtype of obsessive-compulsive disorder (OCD) and tic disorders known collectively as Pediatric Autoimmune Neuropsychiatric Disorders Associated with Streptococcus (PANDAS) is believed to be secondary to central nervous system (CNS) autoimmunity that occurs in relation to group A streptococcal infection. Thus, we hypothesized that a sample of children with OCD and/or tics would have an increased maternal risk for an autoimmune response relative to population norms. We also expected maternal prevalence of various autoimmune diseases to be higher among those participants that met the putative criteria for PANDAS. METHODS: We examined, via structured interview, the medical history of the biological mothers of 107 children with OCD and/or tics. RESULTS: Autoimmune disorders were reported in 17.8% of study mothers, which is significantly greater than the general prevalence among women in the United States (approximately 5%). Further, study mothers were more likely to report having an autoimmune disease if their children were considered "likely PANDAS" cases versus "unlikely PANDAS" cases. CONCLUSIONS: The results offer preliminary support for hypothesized links between maternal autoimmune disease and both OCD/tics and PANDAS in youth. Further research is necessary to clarify these general associations; links to specific autoimmune disease; and relevance of autoimmune disease in other family members (e.g., fathers).

Evaluating the predictive validity of a single stimulus engagement preference assessment.

Most preference assessments require individuals to choose among two or more stimuli. More recently, the duration of engagement with items has been used as an index of preference. In the current study, the predictive validity of a single stimulus engagement (SSE) preference assessment was evaluated with 4 individuals. Stimuli were presented singly for a brief period while engagement with that stimulus was recorded. Although SSE preference rankings closely matched paired stimulus preference assessment rankings for only 2 of the 4 participants, relative preference rankings based on duration of engagement predicted relative reinforcer effectiveness for all participants in a subsequent concurrent-schedule reinforcer assessment. The SSE procedure took less time to administer than the paired stimulus procedure but produced less stable preference rankings across administrations. The SSE procedure may be appropriate for individuals who have difficulty selecting one stimulus from among two or more stimuli, and it may be well suited for evaluating activities that are difficult to present in a paired stimulus format.

Working with families of suddenly and critically ill children: physician experiences.

OBJECTIVE: To describe physicians' experiences in attempting to provide optimal care for families of children who suffer from sudden, acute life-threatening conditions (SALTC). DESIGN: To generate descriptive data in this exploratory study, we used qualitative methods including focus groups and in-depth interviews. Transcripts of focus groups and interviews were analyzed for content using standard phenomenologic analysis methods, which resulted in a participant-generated conceptual model of optimal care for families of children with SALTC. SETTING: The intensive care unit of an urban pediatric teaching hospital. PARTICIPANTS: Twenty-two pediatric intensive care unit physicians, including residents, fellows, and attendings. INTERVENTION: None. MAIN OUTCOME MEASURES: Each participating physician provided qualitative descriptions of experiences caring for families of children with SALTC. RESULTS: Physicians identified 4 components of optimal care for families: (1) providing timely, accurate information about their child; (2) maintaining privacy for confidential discussions and personal grieving; (3) giving adequate emotional support; and (4) granting family members the right to hold and comfort their dying child. Physicians also described barriers to, and facilitators of this optimal care. CONCLUSIONS: Descriptive information provided in this exploratory study offers a complex model of optimal family care. Issues that affect the quality of care to families include those related to the context of providing care in a large teaching hospital, as well as subtleties of communication between parents and staff. Physicians' beliefs about optimal care of families in the pediatric intensive care unit revealed implications for both practice and training in pediatrics.

Characteristics and perceived needs of mothers with serious mental illness.

This cross-sectional survey determined the prevalence of motherhood among severely mentally ill women hospitalized in an inner-city facility and examined the demographic characteristics and perceived needs of the 32 mothers who were identified. Slightly less than one-quarter of these mothers had seen their children within the last week, and only about 20 percent maintained full custody of their children. However, none of the respondents reported feeling that it was unimportant whether they continued mothering their children. Approximately half described needing help in dealing with their sadness about their children. The pilot data presented in this paper suggest that motherhood is common among severely mentally ill and hospitalized women and that issues relevant to mothering remain important to them.