Parental Experiences with Newborn Screening and Gene Replacement Therapy for Spinal Muscular Atrophy.

BACKGROUND: Spinal muscular atrophy (SMA) is a genetic neurodegenerative disorder with onset predominantly in infants and children. In recent years, newborn screening and three treatments, including gene replacement therapy (Onasemnogene abeparvovec-xioi), have become available in the United States, aiding in the diagnosis and treatment of children with SMA. OBJECTIVE: To evaluate parents' experiences with newborn screening and gene replacement therapy and to explore best practices for positive newborn screen disclosure and counseling of families. METHODS: We conducted semi-structured interviews (n = 32) and online surveys (n = 79) of parents whose children were diagnosed with SMA (on newborn screening or symptomatically) and treated with gene replacement therapy. RESULTS: Gene replacement therapy was most parents' first treatment choice, although concerns regarding long term efficacy (65%) and safety (51%) were common. Information provided during the newborn screening disclosure was quite variable. Only 34% of parents reported the information provided was sufficient and expressed need for more information about treatment. Although many parents experienced denial of the diagnosis at initial disclosure, 94% were in favor of inclusion of SMA on newborn screening. Parents were almost universally anxious following diagnosis and over half remained anxious at the time of study participation with uncertainty of the future being a key concern. Many parents had difficulty processing information provided during their first clinic appointment due to its complexity and their emotional state at the time. CONCLUSIONS: Utilizing this data, we provide a recommendation for the information provided in newborn screening disclosure, propose adjustments to education and counseling during the first clinic visit, and bring awareness of parents' mental health difficulties.

Face-to-Face Versus Online Behavioral Parent Training for Young Children at Risk for ADHD: Treatment Engagement and Outcomes.

Attention deficit/hyperactivity disorder (ADHD) is associated with significant challenges in child functioning. Although behavioral parent training (BPT) can reduce ADHD symptoms, factors associated with traditional face-to-face (F2F) delivery results in only about half of families receiving BPT. The purpose of this pilot randomized controlled trial was to examine parent engagement and program acceptability of F2F and online BPT, as well as the efficacy of both formats relative to a waitlist control (WLC) group. Participants were 47 families with preschool children (30 boys, 17 girls) who were between the ages of 3 years 0 months (3;0) and 5 years 11 months (5;11) old who were identified at risk for ADHD. Children were randomly assigned to F2F or online BPT or to WLC; parents in the two treatment conditions received 10 sessions of BPT. Assessment data for all groups were collected at pre-, mid-, and posttreatment. Both intervention formats resulted in high attendance (M = 80%) and significantly improved parent knowledge of interventions, treatment implementation fidelity, and child behavior (reduced restlessness and impulsivity, improved self-control, affect, and mood) compared with WLC. However, parents in the F2F group reported significantly higher acceptability ratings than parents in the online group. Findings indicate a streamlined BPT delivered online or F2F results in high levels of engagement, acceptability, as well as parent treatment knowledge and fidelity. Online BPT is associated with similar efficacious outcomes with F2F BPT, suggesting the need for further research to determine variables that predict greater acceptability for and adoption of this format.

What a rude e-mail! Examining the differential effects of incivility versus support on mood, energy, engagement, and performance in an online context.

Existing research on workplace incivility has demonstrated an association with a host of negative outcomes, including increased burnout, turnover intentions, and physical symptoms. With the rise in Internet communication over the last decade, interpersonal mistreatment has spilled over to the Internet, but little is known about the impact of incivility communicated via e-mail on employee psychological and performance outcomes. The current study presents a within-subjects experiment wherein incivility and support were manipulated in a laboratory-based simulated workplace setting. Eighty-four participants completed a series of math tasks while interacting with either an uncivil or a supportive supervisor via e-mail. Data were collected on energy, cardiac activity, mood, task performance, and engagement. Findings indicate that participants reported higher levels of negative affect and lower levels of energy after working with the uncivil supervisor than with the supportive supervisor. Additionally, participants performed significantly worse on the math tasks and had lower engagement in the uncivil condition than the supportive condition, and these relationships were mediated by energy. No differences were found in cardiac activity between the two conditions. Results are discussed in terms of their implications for the 21st century world of work.

Cyber incivility @ work: the new age of interpersonal deviance.

The current study was designed to extend the interpersonal deviance literature into the online domain by examining the incidence and impact of supervisor cyber incivility and neuroticism on employee outcomes at work. Conservation of Resources (COR) theory was used as the guiding framework because cyber incivility is thought to deplete energetic resources in much the same way that other stressors do, ultimately leading to negative outcomes like burnout. Results indicate that supervisor cyber incivility is positively related to burnout, absenteeism, and turnover intentions. Support was also found for the role of neuroticism as a moderator of the relationship between supervisor cyber incivility and outcomes. In general, the relations between cyber incivility and outcomes were stronger for those individuals reporting higher levels of neuroticism. Results are discussed in terms of COR theory, and possible mechanisms for the role of neuroticism in the stressor-strain relationship are discussed. The current study highlights the importance of understanding workplace online behavior and its impact on employee health and organizational well-being. Future research directions examining online interpersonal deviance are suggested.