Psychological Adjustment of Children and Adolescents with 22q11.2 Deletion Syndrome and Their Mothers’ Stress and Coping—A Longitudinal Study

Abstract

At present, there is a lack of longitudinal studies on the psychological adjustment of both children and adolescents with 22q11.2 deletion syndrome (22q11.2DS) and their primary caregivers. To fill this gap, we performed a four-year follow-up study. Mothers filled out the Child Behavior Checklist 4–18, the Social Orientation of Parents with Handicapped Children questionnaire to assess maternal stress and coping strategies, and the Freiburger Personality Inventory-Revised—subscales strain and life satisfaction. Fifty-five subjects with 22q11.2DS (26 males and 29 females; age: M = 10.79 years, SD = 3.56 years) and their biological mothers (age: M = 40.84 years, SD = 4.68 years) were included in this study. Significantly higher levels of behavior problems than in the general population and an increase in these problems, especially internalizing ones, over time could be found. In contrast, maternal stress did not change significantly over time, but mothers demonstrated increased levels of strain and reduced life satisfaction at T2. Thus, careful monitoring as well as early and adequate interventions, if indicated, should be offered to families with a child with 22q11.2DS, not only for somatic complaints but also for problems with psychological adjustment.

Social behavior in 16p11.2 and 22q11.2 copy number variations: Insights from mice and humans

“…Even after excluding subjects with a diagnosis of ASD from the sample, 46% of the remaining 22q‐DEL without ASD still presented impairments at the ADI‐R Social Interaction 33 . Presence of Social Problems, assessed with the CBCL, were often reported in children with 22q‐DEL 38,48–50 ranging from 27% to 37% 51–53 . Clinical elevations in the CBCL Withdrawal subscale was reported as a feature in 12%–14% of cases of 22q‐DEL children as well 49,53 .…”

“… 33 Presence of Social Problems, assessed with the CBCL, were often reported in children with 22q‐DEL 38 , 48 , 49 , 50 ranging from 27% to 37%. 51 , 52 , 53 Clinical elevations in the CBCL Withdrawal subscale was reported as a feature in 12%–14% of cases of 22q‐DEL children as well. 49 , 53 However, despite the deficits in this sub‐domain, it was suggested that 22q‐DEL children may still understand emotions and thoughts from others and may also be interested in social interactions.…”

Implications in Educational, Linguistic, and Psycho-Emotional Development in People with 22q11 Syndrome

“…This also happens in terms of behavior problems. In longitudinal studies, individuals with 22q11 have been shown to score significantly higher in areas such as withdrawal, thinking problems, attention problems, aggressive behavior, internalizing problems, externalizing problems, and total problems (Briegel et al, 2008;Briegel & Andritschky, 2021;Wagner et al, 2017).…”

Environmental Influences on the Relation between the 22q11.2 Deletion Syndrome and Mental Health: A Literature Review

“…One group of studies has investigated environmental effects in cohorts of 22q11DS patients. Studies looking into the role of maternal stress on children’s behaviour problems [ 28 ], of substance use on psychosis [ 35 ], of the character of a close relationship, of stressful events and neighbourhood danger on anxiety and depression [ 33 ], and general history of trauma [ 39 ] have reported non-significant findings. Other studies have shown significant effects of the following environmental factors: (1) 22q11DS patients with higher levels of parental anxiety or depression had significant increases in psychopathology [ 27 ]; (2) more usage of physical punishment was linked to higher levels of problem behaviours [ 29 ]; (3) individuals with higher parental education or parent intelligence showed higher IQ scores [ 30 , 31 ]; (4) higher parental socio-economic status (SES) correlated with higher social competency of patients [ 32 ], less frequent behaviour problems [ 37 ] or patients’ better global functioning [ 39 ]; (5) higher stress load—the number of negative life events, was associated with psychotic symptoms, e.g., hallucinations and delusions, as well as with dysfunctional coping strategies [ 34 ]; and (6) higher scores on peer victimisation and hostile close relationships were related to reports of both higher levels of anxiety and depression as well as to impaired tolerance to normal stress in patients [ 33 ].…”