Mental Health in Pre-Adolescents with Cerebral Palsy: Exploring the Strengths and Difficulties Questionnaire as a Screening Tool in a Follow-Up Study including Multi-Informants

There is a high prevalence of mental health problems in children with Cerebral Palsy (CP). Still, knowledge regarding the trajectory of mental health problems throughout childhood and differences according to informants is lacking. There is also a need for more knowledge regarding the validity of mental health screening tools. In the present study, we assessed changes in parent-rated mental health problems in a cohort of 36 children with CP from school-starting age to pre-adolescence and differences in mental health problems according to informants. Further, we assessed the validity of the Strengths and Difficulties Questionnaire (SDQ) for psychiatric disorders. The study cohort was assessed using the SDQ and a child psychiatric diagnostic instrument at school-starting age and at pre-adolescence. Mean parental SDQ scores increased significantly for emotional, hyperactivity and total problems. Self-reported impact of mental health problems was significantly lower than parent-reported impact, and parents and pre-adolescents reported significantly higher mean scores than teachers for emotional problems, conduct problems and total problem scores. Validated against psychiatric disorders, the SDQ was satisfactory for screening children with CP for risk of psychiatric disorders at pre-adolescence. We recommend that mental health screening be integrated into the regular follow-up for children with CP

Low frequency of parental mosaicism in de novo COL4A5

Abstract Background Alport syndrome is a progressive hereditary kidney disease clinically presenting with haematuria, proteinuria, and early onset end‐stage renal disease, and often accompanied by hearing loss and ocular abnormalities. The inheritance is X‐linked in the majority of families and caused by sequence variants in the COL4A5 gene encoding the α5‐chain of type‐IV collagen. The proportion of de novo COL4A5 sequence variants in X‐linked Alport syndrome has been reported between 12 and 15% in previous studies. Methods In the present study we have systematically investigated the mosaic status of asymptomatic parents of six patients with X‐linked Alport syndrome using next‐generation sequencing of DNA extracted from different tissues. The deleterious COL4A5 sequence variants in these patients were previously assumed to be de novo, based on Sanger sequencing of the parents. Results A low‐grade (1%) parental mosaicism was detected in only one out of six families (17%). In addition, in one out of six families (17%), we found that the mutational event probably occurred postzygotic. Conclusion These findings highlight the importance of testing for mosaicism in unaffected parents of patients with sequence variants considered to be de novo, as it may have implications for the recurrence risk and thereby for the genetic counseling of the family

The prevalence of mental health disorders and symptoms in children and adolescents with cerebral palsy: a systematic review and meta-analysis

Aim: Mental health conditions and problems are often reported in children and adolescents with cerebral palsy (CP). A systematic review was undertaken to describe their prevalence. Method: MEDLINE and PsycINFO databases from 1996 to 2016 were searched and reference lists of selected studies were reviewed. Studies were included if they reported point prevalence of mental health diagnoses or symptoms in a general population of children and/or adolescents with CP. Pooled prevalence for mental health symptoms was determined using a random effects meta-analysis. Results: Of the 3158 studies identified, eight met the inclusion criteria. Mental health disorders were diagnosed by psychiatric interview in one study, giving a prevalence of 57% (32 out of 56 children). The remaining seven studies (n=1715 children) used parent-report mental health screening tools. The pooled prevalence for mental health symptoms using the Strengths and Difficulties Questionnaire (n=5 studies) was 35% (95% confidence interval [CI] 20-61) and using the Child Behavior Checklist (n=2 studies) was 28% (95% CI 22-36). Evidence was characterized by a moderate level of bias. Interpretation: More studies are needed to ascertain the prevalence of mental health disorders. Mental health symptoms are common and mental health evaluations should be incorporated into multidisciplinary assessments for these children