ObjectiveLovastatin has been shown to reverse learning deficits in a mouse model of Neurofibromatosis Type 1 (NF1), a common monogenic disorder caused by a mutation in the Ras‐MAPK pathway and associated with learning disabilities. We conducted a randomized double‐blind placebo‐controlled trial to assess lovastatin's effects on cognition and behavior in patients with NF1.MethodForty‐four NF1 patients (mean age 25.7+/−11.6 years; 64% female) were randomly assigned to 14 weeks of lovastatin (N = 23; maximum dose of 80 mg/day for adult participants and 40 mg/day for children) or placebo (N = 21). Based on findings in the mouse model, primary outcome measures were nonverbal learning and working memory. Secondary outcome measures included verbal memory, attention, and self/parent‐reported behavioral problems, as well as tolerability of medication. Participants also underwent neuroimaging assessments at baseline and 14 weeks, to determine whether neural biomarkers were associated with treatment response. Linear mixed models assessed for differential treatment effects on outcome measures.ResultsTwelve participants dropped from the study prior to completion (8 placebo, 4 lovastatin), resulting in 32 completers (15 placebo, 17 lovastatin). Lovastatin was well‐tolerated, with no serious adverse events. Differential improvement favoring lovastatin treatment was observed for one primary (working memory; effect size f
2 = 0.70, P < 0.01) and two secondary outcome measures (verbal memory, f
2 = 0.19, P = 0.02, and adult self‐reported internalizing problems, f
2 = 0.26, P = 0.03). Exploratory moderator analyses revealed that higher baseline neural activity in frontal regions was associated with larger treatment effects.InterpretationThese preliminary results suggest beneficial effects of lovastatin on some learning and memory functions, as well as internalizing symptoms in patients with NF1.
Children of parents with cognitive impairment are overrepresented in the child protection system (CPS). The aim of this study was to examine the relationship between primary caregiver cognitive impairment (CCI) and CPS investigation outcomes using the Canadian Incidence Study of Reported Child Abuse and Neglect (CIS-2008). The CIS-2008 includes process and outcomes data on a national sample of CPS investigations involving a total of 15,980 children under the age of 16 years. This secondary data analysis found that primary CCI was noted in 6.3% of all investigations. When primary CCI was noted, investigations were 3–4 times more likely to result in child removal and court action. The results further suggest that some case characteristics, including case history variables, are weighted differently in decision making depending on whether or not primary CCI is indicated. A systematic, national strategy is needed to build capacity in government and community sectors for the provision of research-informed child and youth protection and family support services for these parents and their children.
The purpose of this study was to pilot a flexible, group-based program designed to strengthen the social connections and enhance the psychological well-being of mothers with intellectual impairment. A multi-site, mixed-method, pretest-posttest design was employed. To obtain rich process and outcome data, the evaluation incorporated measures of psychosocial well-being, goal achievement scaling, and interviews with group facilitators and participants. A total of 18 mothers completed the program. Participation was associated with a meaningful reduction in psychological distress with effect sizes ranging from .57 for depression to .71 for anxiety. Participating in the group program gave the mothers something to look forward to each week, opportunities to learn from and support others, and the feeling of "being a part of society". Further research is needed to determine whether these promising results can be replicated, and to evaluate the long-term impact of the program on mothers and their children.
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