What challenges do siblings of children with chronic disorders express to their parents? A thematic analysis of 73 sibling-parent dialogues.

PURPOSE: The study explored challenges experienced by siblings of children with chronic disorders, as expressed by siblings in parent-child dialogues. DESIGN AND METHODS: Seventy-three parent-child dialogues (M duration = 28.6 min) were analyzed using qualitative thematic analysis. The dialogues took place within the SIBS group intervention for siblings and parents of children with chronic disorders. The siblings (aged 8 to 14 years) had brothers and sisters with autism spectrum disorders, ADHD, rare disorders, cerebral palsy, or severe mental health disorders. The data are from session 5 in the SIBS intervention, in which the siblings are to express their wishes about family-related challenges (e.g., desired changes) to their parents. The parents are encouraged to listen, explore, and validate the child's perspective before discussing solutions. RESULTS: Most of the family-oriented challenges the siblings expressed were related to the diagnosis of the brother or sister with a disorder. Four main themes were identified: (1) Family life (e.g., limitations in family activities); (2) The diagnosis (e.g., concerns about the future); (3) Violence; and (4) Important relationships. CONCLUSION: The siblings experienced challenges and difficult emotions in interactional processes in which the diagnosis affected family life and relationships. The study adds a new dimension to the field by identifying siblings' expressed challenges based on parent-child dialogues. PRACTICE IMPLICATIONS: Identified themes can guide how parents should meet and address siblings' needs, how health care providers inform and support parents in doing so, and emphasize the relevance of interventions targeting family-level risk and resilience factors.

Suicidal Risk Behaviors in Adolescents With Rare Neurodevelopmental Disorders: The Role of Sex, Autistic Traits, and Mental Health Difficulties.

OBJECTIVE: Autistic traits are associated with mental health difficulties and risk of suicidal risk behaviors among adolescents. Little is known about how autistic traits affect the mental health of adolescents with rare neurodevelopmental disorders (RNDs). The aim of this study was to investigate the relationship between autistic traits, mental health difficulties, and suicidal risk behaviors in adolescents with RNDs. METHODS: Parents (N = 93) completed the Child Behavior Checklist, Social Communication Questionnaire, and Social Responsiveness Scale about their adolescent (Mage = 13.1, SD = 2.3, 62.4% females) with an RND (e.g., sex chromosome aneuploidies, Fragile X syndrome, 22q11.2 deletion syndrome). The data were analyzed with hierarchical logistic regression analysis. RESULTS: The prevalence of suicidal risk behaviors (16.1%) was similar to that reported among autistic youth and was higher among boys than girls. More autistic traits were associated with suicidal risk behaviors in bivariate analysis. In multivariate analysis, more anxiety/depressive symptoms were associated with more suicidal risk behaviors and externalizing problems associated with suicidal risk behaviors beyond autistic traits and anxiety/depressive symptoms. CONCLUSION: Adolescents with RNDs are at risk of suicidal risk behaviors, especially those with higher levels of autistic traits, anxiety/depressive symptoms, and externalizing problems. Assessment of autistic traits, mental health difficulties, and suicide risk may be indicated for adolescents with RNDs to determine if corresponding intervention is needed.

The Therapy Process Observational Coding System: Group cohesion scale in youth anxiety treatment: Psychometric properties.

BACKGROUND: There are no well-established measures of group cohesion, defined as the collaborative bond between group members, in group cognitive behavioral therapy (GCBT) with youth. We therefore examined the Therapy Process Observational Coding System for Child Psychotherapy-Group Cohesion Scale (TPOCS-GC), which has previously only been used with adult samples, in a youth sample. METHODS: Observers coded 32 sessions from 16 groups with 83 youth aged 8 to 15 years (90.7% European White). Youth had anxiety disorders and received manualized GCBT in community clinics. We examined psychometric properties of the TPOCS-GC and its' construct validity in terms of relations with pretreatment variables, alliance and fidelity during treatment, and post-treatment variables. Group cohesion was measured twice during treatment (early and late). RESULTS: The TPOCS-GC was internally consistent (α = 0.72) and was reliably coded (M ICC = 0.61). Higher clinical severity at pretreatment predicted lower early group cohesion. Higher youth age, higher clinical severity at pretreatment, and higher youth-rated early alliance predicted lower late group cohesion. Higher therapist-rated early alliance predicted higher early group cohesion. Higher therapist-rated late alliance predicted higher late group cohesion. Higher late group cohesion predicted lower clinical severity and higher client treatment satisfaction at post-treatment. Early group cohesion did not predict any post-treatment variables. CONCLUSIONS: A four-item version of the TPOCS-GC can be reliably used in youth GCBT. The TPOCS-GC is distinct from, but associated with, multiple clinical variables.

Process and Implementation Elements of Measurement Feedback Systems: A Systematic Review.

Measurement feedback systems (MFS) can help guide treatment and improve clinical outcomes. Studies of MFS are heterogeneous both in execution and results, and the effects of MFS seem restricted by limited attention to process and implementation elements and by limited adoption by health professionals. The current systematic review mapped the use of process and implementation elements in MFS studies. An overview of therapists' use of and attitudes toward MFS is provided. Three-level meta-analyses were used to test theoretically informed process and implementation elements as moderators of the effects of MFS. Hypotheses and general propositions from Clinical Performance Feedback Intervention Theory (CP-FIT) were used to organize the elements of the studies and were used as moderator variables. Previous studies on MFS interventions have had a limited focus on implementation efforts and process elements that may increase the effects of MFS and their use among therapists. Efforts have sparsely been made to reduce barriers to MFS use, and several studies have reported limited engagement with MFS among therapists. Therapists' attitudes toward MFS, feedback, or standardized measures were heterogeneously reported, making data synthesis challenging. Identified process and implementation elements were not significantly associated with effect sizes in the studies and the results did not support the propositions of CP-FIT. The lack of statistically significant associations may be due to limited reporting of details about process and implementation aspects. More research designed to test hypotheses regarding process and implementation elements is needed to improve the use and effects of MFS. Future studies should aspire to report findings in a manner that allows for an understanding of the implementation process and therapists' adoption of these systems.

Measuring family communication in pediatric nursing: Psychometric properties of the Parent-Child Communication Scale - Child Report (PCCS-CR).

PURPOSE: Communication quality is an essential indicator of family functioning and represents an important outcome after pediatric nursing interventions. However, few well-documented child-report questionnaires for family communication exist. We aimed to document the psychometric properties of a previously developed child-rated family communication scale for use in pediatric nursing. DESIGN AND METHODS: We examined the Parent-Child Communication Scale - Child Report (PCCS-CR) in terms of factor structure, convergent validity against the Experiences in Close Relationships-Relationship Structures scale (ECR-RS), and known-groups validity between a sample of siblings of children with pediatric health conditions and controls. The sample comprised 101 siblings of children with a pediatric health condition and 44 controls (M age = 11.5 years, SD = 2.2). RESULTS: We confirmed a two-factor structure of the PCCS-CR. One factor is communication from the child to the parent, labelled child communication (e.g., "I discuss problems with my parents") and the other is communication from the parent to the child, labelled parent communication (e.g., "My parent is a good listener"). Convergent validity of the PCCS-CR was demonstrated through correlations with ECR-RS (r = -0.73 to -0.22, p ≤ .05). Further, construct validity through differences between families with and without a child with a pediatric health condition was demonstrated (g = 0.36-0.83, p ≤ .052). CONCLUSION: The PCCS-CR appears to be a psychometrically sound measure of parent-child communication from the child's point of view. PRACTICAL IMPLICATIONS: The PCCS-CR can be administered in pediatric nursing care and can be used to target and measure the outcomes of interventions aimed at enhancing family functioning.

Age and depressive symptoms change predict the 4-year self-concept trajectory for youth after anxiety treatment.

OBJECTIVES: To investigate the self-concept trajectory from before to 4 years after cognitive behavioral therapy (CBT) for youth with anxiety disorders, including predictors. METHODS: Youth with anxiety diagnoses (N = 179; M = 11.5 years, SD = 2.1; 53.6% girls; 46.4% boys) received CBT in community clinics. Self-concept, anxiety/depression symptoms, and diagnostic status were assessed at pre-, post, 1-year, and 4-year posttreatment. RESULTS: Growth curve analyses showed that the self-concept improved significantly over time (d = 0.07 to 0.34). Higher age and a decrease in the depressive symptom trajectory predicted increased self-concept trajectory from baseline to 4 years posttreatment. Not dropping out of treatment also contributed positively to the self-concept trajectory, but not above and beyond decreased depressive symptoms. The correlation between self-concept and depressive symptoms was r = 0.60, indicating these are related but distinct. CONCLUSION: Self-concept can improve after CBT, also long-term. This change appears to primarily be associated with decreased depressive symptoms over time.

Therapist Alliance-Building Behaviors, Alliance, and Outcomes in Cognitive Behavioral Treatment for Youth Anxiety Disorders.

OBJECTIVE: The alliance influences outcomes in CBT for youth anxiety disorders. Thus, knowledge about how therapists can enhance the alliance is needed. METHOD: Seventy-three youth with anxiety diagnoses (M age = 11.5 years, SD = 2.2; range 8 to 15 years; 47.9% boys; 90.4% white-European) participated in 10-session cognitive behavioral therapy in community clinics. Therapist alliance-building behaviors in session 2 was reliably coded with the observer-rated Adolescent Alliance-Building Behavior Scale (Revised) (AABS(R)). Alliance was measured as youth- and therapist-rated alliance, and youth-therapist alliance discrepancy in session 3. Outcomes were diagnostic recovery and anxiety symptom reduction at post-treatment and one-year follow-up, and treatment dropout. We examined the direct effects of alliance-building on alliance, alliance on outcomes, and alliance-building on outcomes in multilevel mediation models, and between- versus within-therapist variance across these effects. RESULTS: The alliance-building behaviors collaborate, present treatment model, and explore motivation positively predicted alliance, whereas actively structuring the session (i.e., dominating) negatively predicted alliance. The alliance-building behaviors attend to experience, collaborate, explore motivation, praise, and support positively predicted outcomes. The alliance-building behaviors present treatment model, express positive expectations, explore cognitions, and support negatively predicted outcomes. The effect of collaborate on symptom reduction was mediated by youth-therapist alliance discrepancy. There was almost zero between-therapist variance in alliance-building, and considerable within-therapist variance. CONCLUSION: Therapist alliance-building behaviors were directly (positively and negatively) associated with alliance and/or outcomes, with only one effect mediated by alliance. Alliance-building behaviors varied far more within therapists (i.e., across clients) than between therapists.

Psychosocial Functioning in Siblings of Children With Rare Disorders Compared to Controls.

Siblings of children with chronic disorders are at increased risk of psychosocial problems. The risk may be exacerbated when the chronic disorder is rare and limited medical knowledge is available, due to more uncertainty and feelings of isolation. We examined mental health, parent-child communication, child-parent relationship quality, and social support among 100 children aged 8 to 16 years (M age 11.5 years, SD = 2.2; 50.0% boys, 50.0% girls). Fifty-six were siblings of children with rare disorders, and 44 were controls. The siblings of children with rare disorders (herein, siblings) were recruited from a resource centre for rare disorders and comprised siblings of children with a range of rare disorders including neuromuscular disorders and rare chromosomal disorders with intellectual disability. Controls were recruited from schools. Self-reported child mental health was significantly poorer for siblings compared to controls (effect size difference d = 0.75). Parent-reported child mental health was not significantly different between the groups (d = -0.06 to 0.16). Most child-parent relationships (anxiety/avoidance; mothers/fathers) were significantly poorer for siblings compared to controls (d = 0.47 to 0.91). There was no difference between groups in anxious relation with mother. Parent-child communication was significantly poorer for siblings compared to controls (d = -0.87 to -0.75). Social support was significantly poorer for siblings compared to controls (d = 0.61). We conclude that siblings of children with rare disorders display more psychosocial problems than controls. Interventions are indicated to prevent further maladjustment for siblings.

Neuropsychological functions, sleep, and mental health in adults with Klinefelter syndrome.

A few studies have examined neuropsychological functions, sleep, and mental health combined in Klinefelter syndrome (KS; 47,XXY). We investigated neuropsychological functions with standard tests, sleep with actigraphy, and self-reported mental health in 30 men with KS (Mean age = 36.7 years) compared to 21 controls (Mean age = 36.8 years). Men with KS scored significantly lower on mental speed, attention span, working memory, inhibition, and set-shifting tests, as well as overall IQ (mean effect size difference Cohen's d = 0.79). Men with KS had significantly longer night wakes, with no differences in other sleep variables (mean d = 0.34). Men with KS reported poorer mental health than controls (mean d = 1.16). Regression analyses showed neuropsychological functions explained variance in some sleep domains for men with KS but not for controls. Neuropsychological functions explained variance in some mental health domains for controls. For men with KS, however, verbal IQ was the only significant predictor of mental health. Altogether, men with KS display problems in neuropsychological functions and mental health but do not appear different from controls on most sleep parameters. Our findings indicate that relations between neuropsychological functions, sleep, and mental health differ between men with KS and controls.

School-based targeted prevention compared to specialist mental health treatment for youth anxiety.

BACKGROUND: The 'FRIENDS for life' program (FRIENDS) is a 10-session cognitive behavioral therapy (CBT) program used for prevention and treatment of youth anxiety. There is discussion about whether FRIENDS is best applied as prevention or as treatment. METHODS: We compared FRIENDS delivered in schools as targeted prevention to a previous specialist mental health clinic trial. The targeted prevention sample (N = 82; Mage  = 11.6 years, SD = 2.1; 75.0% girls) was identified and recruited by school nurses in collaboration with a community psychologist. The clinical sample (N = 88, Mage  = 11.7 years, SD = 2.1; 54.5% girls) was recruited for a randomized controlled trial from community child- and adolescent psychiatric outpatient clinics and was diagnosed with anxiety disorders. RESULTS: Both samples showed significantly reduced anxiety symptoms from baseline to postintervention, with medium mean effect sizes across raters (youths and parents) and timepoints (post; 12-months follow-up). Baseline youth-reported anxiety symptom levels were similar between the samples, whereas parent-reported youth anxiety was higher in the clinical sample. CONCLUSIONS: The study suggests that self-reported anxiety levels may not differ between youth recruited in schools and in clinic settings. The results indicate promising results of the FRIENDS program when delivered in schools by less specialized health personnel from the school health services, as well as when delivered in clinics by trained mental health professionals.

Health complaints and work experiences among adults with neurofibromatosis 1.

BACKGROUND: Neurofibromatosis type 1 (NF1) is a rare genetic disorder with usually benign nerve tumours, skin problems, pain and cardiovascular problems among common complications. AIMS: To examine work participation and experiences in relation to health complaints among adults with NF1. METHODS: We conducted a cross-sectional self-reported survey among 142 persons with NF1 (mean age = 50.3 years, SD = 12.0; 62% females). We measured physical and social dimensions of work experiences with eight items from the third wave of the epidemiological survey 'Nord-Trøndelag Health Study' (HUNT3). We compared NF1 data to 27 036 HUNT3 controls. We measured health complaints with the 29-item Subjective Health Complaints Inventory (SHC; subscales musculoskeletal pain, pseudoneurology and gastrointestinal problems) and compared NF1 data to norms. RESULTS: Compared to controls, significantly more persons with NF1 reported workplace bullying (13% versus 5%) and work as physically exhausting (74% versus 44%), whereas fewer reported work autonomy (64% versus 80%). Women with NF1 reported significantly more SHC than female norms (99% versus 86%). Men with NF1 reported significantly more pseudoneurology and gastrointestinal problems compared to male norms (76% versus 58%). SHC explained 9% of the variance in physical work experiences (ß = -0.47; 95% CI -0.76 to -0.19) and 8% of the variance in social work experiences (ß = 0.48; 95% CI 0.18-0.78). CONCLUSION: Persons with NF1 experience more physical and social work problems, and both work domains were related to health complaints. Individualized assessment and information to co-workers and managers may represent ways to increase work participation among persons with NF1.

The impact of treatment delivery format on response to cognitive behaviour therapy for preadolescent children with anxiety disorders.

BACKGROUND: Several delivery formats of cognitive behaviour therapy (CBT) for child anxiety have been proposed, however, there is little consensus on the optimal delivery format. The primary goal of this study was to investigate the impact of the child's primary anxiety diagnosis on changes in clinical severity (of the primary problem) during individual CBT, group CBT and guided parent-led CBT. The secondary goal was to investigate the impact of the child's primary anxiety diagnosis on rates of remission for the three treatment formats. METHODS: A sample of 1,253 children (5-12 years; Mage = 9.3, SD = 1.7) was pooled from CBT trials carried out at 10 sites. Children had a primary diagnosis of generalised anxiety disorder (GAD), social anxiety disorder (SoAD), specific phobia (SP) or separation anxiety disorder (SAD). Children and parents completed a semistructured clinical interview to assess the presence and severity of DSM-IV psychiatric disorders at preintervention, postintervention and follow-up. Linear mixture modelling was used to evaluate the primary research question and logistic modelling was used to investigate the secondary research question. RESULTS: In children with primary GAD, SAD or SoAD, there were no significant differences between delivery formats. However, children with primary SP showed significantly larger reductions in clinical severity following individual CBT compared to group CBT and guided parent-led CBT. The results were mirrored in the analysis of remission responses with the exception that individual CBT was no longer superior to group CBT for children with a primary SP. The difference between individual and group was not significant when follow-up data were examined separately. CONCLUSIONS: Data show there may be greater clinical benefit by allocating children with a primary SP to individual CBT, although future research on cost-effectiveness is needed to determine whether the additional clinical benefits justify the additional resources required.

Health Survey of Adults with Neurofibromatosis 1 Compared to Population Study Controls.

Neurofibromatosis type 1 (NF1) is a genetic, autosomal dominant multi-organ disease characterized by susceptibility to tumor formation, changes in skin pigmentation, skeletal abnormalities, and neuropsychological deficits. Clinical studies have shown impaired health-related quality of life (HQoL) in adults with NF1. However, little is known about HQoL in non-clinical NF1 samples. We conducted a cross-sectional self-report survey of 142 persons with NF1 (M age = 50.3 years, SD = 12.0, range 32 to 80; 62.0% females) recruited from non-clinical settings. Several HQoL domains, including life satisfaction, mental health, sleep, pain, gastrointestinal problems, oral health, and social support, were compared between the NF1 sample and 46,293 controls from the HUNT3 population study. We also examined gender differences within the NF1 sample and predictors of HQoL. Compared to controls, the NF1 sample reported significantly poorer life satisfaction, mental health, sleep, and oral health, and more pain, gastrointestinal problems, comorbid diseases, and memory problems. Several HQoL domains were significantly correlated. Mental health was the only unique significant predictor of overall life satisfaction. Women with NF1 reported significantly more mental health, sleep, and pain problems than men with NF1. Mental health assessment and management should be integrated into clinical care of persons with NF1 to potentially improve their HQoL.

Change in post-traumatic cognitions mediates treatment effects for traumatized youth-A randomized controlled trial.

Posttraumatic stress symptoms (PTSS) are associated with serious impairments in psychological, social, and academic functioning in youth. The aim of this study was to investigate whether changes in posttraumatic cognitions mediate treatment effects. Participants were multitraumatized youth (N = 156, mean age = 15.1 years, range = 10-18; 79.5% girls) randomly assigned to receive trauma-focused cognitive-behavioral therapy (TF-CBT) or treatment-as-usual (TAU). Mixed-effects models were applied to investigate the impact of treatment conditions on posttraumatic cognitions. Mediation analyses were applied to examine whether changes in posttraumatic cognitions mediated the relationship between treatment conditions and outcome in posttraumatic stress symptoms, depressive symptoms, and general mental health. Participants receiving TF-CBT reported significantly lower levels of negative posttraumatic cognitions at the end of treatment compared to participants in TAU. Change in posttraumatic cognitions mediated the treatment effect difference found for PTSS. When the overall change in cognition was divided into early and late changes, it was only the late change that significantly mediated the PTSS treatment effect. A mediation effect of posttraumatic cognitions was also found for the treatment effect difference in depressive symptoms and in general mental health symptoms. Traumatized youth report having many negative posttraumatic cognitions and changes in negative cognitions plays a key role for treatment outcome. (PsycINFO Database Record

Sleep Problems and Life Satisfaction as Predictors of Health in Men with Sex Chromosome Aneuploidies.

More knowledge is needed about men with sex chromosome aneuploidies (SCA). We present self-reported data from 53 men with SCA (Mage = 36.8 years, SD = 12.3, range 19-67). The Health Survey-Short Form (SF-36) measured eight health domains (physical functioning, role-physical, role-emotional, vitality, emotional health, social functioning, pain, general health). The Pittsburgh Sleep Quality Index measured sleep problems. The Personal Wellbeing Index measured satisfaction with eight life domains. Compared to norms, SCA reported poorer health (mean d = -0.80) and more sleep problems (mean d = -0.85). Differences between SCA and norms on personal well-being were small, except lower health satisfaction in SCA (d = -1.06). Seven of eight regression models predicting the SF-36 domains from life satisfaction and sleep problems were significant (explained variance 12.2% to 46.2%), except physical functioning (ns). Clinical assessment/intervention for a broad range of health and sleep problems is indicated for men with SCA.

A 6-year Follow-up survey of health status in middle-aged women with Turner syndrome.

OBJECTIVE: Studies suggest younger women with Turner syndrome (TS) have good quality of life. Less is known about everyday functioning in adults with TS. In a 6-year follow-up study, multiple areas of functioning were compared between TS women and controls. DESIGN: Women with TS and controls were mailed a self-report survey 6 years after a baseline study. PATIENTS: Fifty-seven women with TS (M age 40·6 ± 11·1 years) and 101 controls (M age 38·8 ± 10·6 years, ns) responded. MEASUREMENTS: Measures of background information, experienced life strain and presence/impact of health conditions were developed for this study. The QPS Nordic measured perceived workload challenges. The LiSat-9 measured life satisfaction. The Rosenberg Self-Esteem Scale measured self-esteem. RESULTS: More TS women lived alone, fewer had biological children, and more had adoptive children. TS women reported fewer sex partners and less sexual confidence. Controls had higher education. There was no difference in employment status. More TS women received disability pensions. TS women reported their work as more physically challenging, less positively challenging and requiring less knowledge skills. TS women experienced more life strain in school, adolescence and late working life. Controls reported higher overall life satisfaction, with no difference between samples on specific domains. TS women reported lower self-esteem. For TS women only, physical health at baseline predicted length of education and mental health at baseline predicted self-esteem. CONCLUSIONS: Women with TS face more challenges than controls on several domains of functioning. Early physical and mental health may influence later educational achievement and self-esteem for women with TS.

Therapist-youth agreement on alliance change predicts long-term outcome in CBT for anxiety disorders.

BACKGROUND: In individual cognitive behavioral therapy (ICBT) for youth anxiety disorders, it is unclear whether, and from whose perspective, the alliance predicts outcome. We examined whether youth- and therapist-rated alliance, including level of youth-therapist alliance agreement, predicted outcome in a randomized controlled trial. METHODS: Youth (N = 91, M age = 11.4 years (SD = 2.1), 49.5% boys, 86.8% Caucasian) diagnosed with separation anxiety disorder, social phobia, or generalized anxiety disorder drawn from the ICBT condition of an effectiveness trial were treated with an ICBT program. Youth- and therapist-rated alliance ratings, assessed with the Therapeutic Alliance Scale for Children (TASC-C/T), were collected following session 3 (early) and 7 (late). Early alliance, change in alliance from early to late, and level of youth-therapist agreement on early alliance and alliance change were examined, in relation to outcomes collected at posttreatment and 1-year follow-up. Outcome was defined as primary diagnosis loss and reduction in clinicians' severity ratings (CSR; Anxiety Disorders Interview Schedule; ADIS-C/P) based on youth- and parent-report at posttreatment and follow-up, and youth treatment satisfaction collected at posttreatment (Client Satisfaction Scale; CSS). RESULTS: Early TASC-C scores positively predicted treatment satisfaction at posttreatment. Higher levels of agreement on change in TASC-C and TASC-T scores early to late in treatment predicted diagnosis loss and CSR reduction at follow-up. CONCLUSIONS: Only the level of agreement in alliance change predicted follow-up outcomes in ICBT for youth anxiety disorders. The findings support further examination of the role that youth-therapist alliance discrepancies may play in promoting positive outcomes in ICBT for youth anxiety disorders. Clinical trial number NCT00586586, clinicaltrials.gov.

HPA AXIS RELATED GENES AND RESPONSE TO PSYCHOLOGICAL THERAPIES: GENETICS AND EPIGENETICS.

BACKGROUND: Hypothalamic-pituitary-adrenal (HPA) axis functioning has been implicated in the development of stress-related psychiatric diagnoses and response to adverse life experiences. This study aimed to investigate the association between genetic and epigenetics in HPA axis and response to cognitive behavior therapy (CBT). METHODS: Children with anxiety disorders were recruited into the Genes for Treatment project (GxT, N = 1,152). Polymorphisms of FKBP5 and GR were analyzed for association with response to CBT. Percentage DNA methylation at the FKBP5 and GR promoter regions was measured before and after CBT in a subset (n = 98). Linear mixed effect models were used to investigate the relationship between genotype, DNA methylation, and change in primary anxiety disorder severity (treatment response). RESULTS: Treatment response was not associated with FKBP5 and GR polymorphisms, or pretreatment percentage DNA methylation. However, change in FKBP5 DNA methylation was nominally significantly associated with treatment response. Participants who demonstrated the greatest reduction in severity decreased in percentage DNA methylation during treatment, whereas those with little/no reduction in severity increased in percentage DNA methylation. This effect was driven by those with one or more FKBP5 risk alleles, with no association seen in those with no FKBP5 risk alleles. No significant association was found between GR methylation and response. CONCLUSIONS: Allele-specific change in FKBP5 methylation was associated with treatment response. This is the largest study to date investigating the role of HPA axis related genes in response to a psychological therapy. Furthermore, this is the first study to demonstrate that DNA methylation changes may be associated with response to psychological therapies in a genotype-dependent manner.

Emotional Experiences Among Siblings of Children With Rare Disorders.

OBJECTIVE: This research aimed to provide new knowledge about the emotional experiences of siblings of children with rare disorders. METHODS: Qualitative thematic analysis was conducted based on videotapes of 11 support groups for 58 siblings aged 7-17 years (Mage = 11.4 years, SD = 2.4) of children with rare disorders. RESULTS: Participants described contradictory emotions in three main contexts: (1) implications of the disorder (knowledge, medical issues, prognosis); (2) consequences for family life (practical implications, limitations of the child with disorder, perspectives on the sibling relationship); and (3) consequences for social life (reflections about (ab)normality, reactions from others). Passive coping strategies were frequently described relative to active coping strategies. An emerging dilemma for participants was hiding versus sharing emotional experiences. CONCLUSIONS: Siblings' emotional experiences are characterized by diverse and contradictory feelings. Interventions to promote emotional health in siblings of children with rare disorder should address this complexity.

Autism traits, social withdrawal, and behavioral and emotional problems in a Norwegian cohort of adolescents with rare genetic disorders.

BACKGROUND: Adolescents with rare genetic disorders represent a population that can be at risk in psychosocial terms. Despite its importance, the association of autism traits and social withdrawal in behavioral and emotional problems in adolescents with rare genetic disorders remains understudied. AIM: The study aimed to empirically examine the clinical characteristics of adolescents with rare genetic disorders with a behavior theory-driven approach. METHOD: We investigated the behavioral and emotional problems and current and lifetime autistic traits in a sample of 93 Norwegian adolescents (Mage = 13.2 years, SDage = 2.4, rangeage 10-17, 62.4% females, 37.6% males) with various rare genetic disorders. The adolescents were investigated cross-sectionally utilizing standardized psychometric questionnaires rated by their parents. RESULTS: More current and lifetime autistic traits and social withdrawal were all associated with more internalizing problems. Further analyses demonstrated that social withdrawal partially mediated the positive association between current autistic traits and internalizing problems. In contrast, social withdrawal fully mediated the positive association between lifetime autistic traits and internalizing problems. CONCLUSION AND IMPLICATIONS: Our results demonstrate important characteristics of adolescents with rare genetic disorders that may guide clinicians and future interventions. Social withdrawal may be prodromal to internalizing problems such as anxiety and depression. Thus, clinically addressing social withdrawal can represent a means to prevent internalizing problems in adolescents with rare genetic disorders and autistic traits.