Childhood predictors of written expression in late adolescents with 22q11.2 deletion syndrome: a longitudinal study

Abstract

Background 22q11.2 Deletion Syndrome (22q11DS) is the second most prevalent genetic syndrome and has a characteristic academic and behavioural phenotype. The primary objective of the current study was to examine the childhood predictors of written expression achievement in adolescents with 22q11DS. Written expression is an important skill that can impact an individual’s overall academic performance, potentially resulting in increased levels of stress and exacerbation of psychiatric symptoms. Methods A total of 119 participants were included in this study. Sixty-nine late adolescents with 22q11DS and 50 controls (consisting of a combined sample of 23 unaffected siblings of youth with 22q11DS and 27 community controls) participated in a 6-year longitudinal research project and received neuropsychological test batteries every 3 years. The Written Expression subtest of the Wechsler Individual Achievement Test–2nd edition (WIAT-II) was the primary outcome measure in the current project. Results Findings indicated differences in childhood predictors of adolescent written expression between participants in the 22q11DS group and participants in the control group. Whereas childhood verbal IQ scores predicted adolescent written expression for participants in the control group, childhood executive function and language skills were unique predictors of adolescent written expression in individuals with 22q11DS. Conclusions Childhood predictors of late adolescent written expression in 22q11DS differ in meaningful ways from predictors in the non-22q11DS population. These results offer some guidance on the underlying factors that may be useful to consider when developing written expression interventions for children with 22q11DS.

Longitudinal Psychiatric and Developmental Outcomes in 22q11.2 Deletion Syndrome: A Systematic Review

“…32 Improvement of inhibition and working memory over an approximately 4-year period was minimal in early adolescents with 22q11DS and negative symptoms compared to those with 22q11DS without negative symptoms. 30 Verbal learning abilities The following studies are based on the same samples: Danish Registry: Hoeffding et al 17 and Vangkilde et al 16 ; Cardiff ECHO: Chawner et al 36 and Chawner et al 20 ; Rome/Geneva: Schneider et al 25 and Schneider et al 26 ; Syracuse: Albert et al 31 ; Antshel et al 32 ; Hamsho et al 37 ; Kates et al 27 ; Kates et al 18 ; Radoeva et al 33 ; Taylor et al 39 ; and Wagner et al 38 moderated the association between childhood avoidantanxious behaviors (i.e., anxiety, withdrawal, and atypicality) and adolescent positive prodromal psychotic symptoms; stronger verbal learning and memory abilities (low average to average abilities) protected individuals from developing positive symptoms of psychosis even if the individuals demonstrated significant avoidantanxious behaviors during childhood. 27 Finally, in the academic domain, individuals with 22q11DS who developed psychosis had weaker reading decoding skills in childhood and, after an initial improvement in adolescence, experienced a decline in reading skills in late adolescence.…”

Deletion Syndrome 22q11.2: A Systematic Review

“…Written expression can also be impaired. Hamsho et al [ 17 ] investigated possible predictors that could be related to good or poor development of this ability in adolescents with DS 22q11.2. These were working memory and “ set shifting ” (unconsciously shifting attention from one task to another).…”

“…While it is true that the daily activities of these individuals is diminished, this decline can be slowed or improved by taking a series of steps to help solve the problems they face. According to Alqarni et al, in their study on dental management in this type of patient, palate surgeries or going to a dentist during childhood could offer many advantages in terms of verbal communication [ 16 ]; in addition, executive function and written expression could be improved with various assistance programs, as indicated in the study by Hamsho et al [ 17 ].…”

Abnormalities in gray matter microstructure in young adults with 22q11.2 deletion syndrome

“…DMRI data acquisitions of the 4th time point are of high quality to allow analysis of microstructure in the cortex. The following publications have also used data from this same time point in the study and focused on investigation of structural analysis of brain gray and white matter and connectivity using functional MRI: ( Olszewski et al, 2017 ; Berhanu et al, 2017 ; Schreiner et al, 2017 ; Thompson et al, 2017 ; Hamsho et al, 2017 ; Antshel et al, 2016 ; Mattiaccio et al, 2016 ; McCarthy et al, 2015 ; Tylee et al, 2017 ; Mattiaccio et al, 2018 ; Taylor et al, 2018 ).…”