Children with Autism Spectrum Disorder in Times of COVID-19: Examining Emotional and Behavioral Problems, Parental Well-Being, and Resilience.

This longitudinal study assessed the impact of the COVID-19 pandemic on children with autism spectrum disorder (ASD; n = 62; Mage = 13 years) by measuring emotional and behavioral problems before and during the pandemic, and by comparing this change to a matched sample of children without ASD (n = 213; Mage = 16 years). Moreover, we examined whether indicators of parental well-being promoted resilience of children with ASD. Results showed that the mean change in problems did not differ between children with and without ASD. Importantly, some children showed an increase in problems, while others showed resilience. Parental well-being indicators were not related to resilience among children with ASD. The interindividual variability in responses, particularly among children with ASD, highlights the need for personalized support.

Autism Symptoms in Children and Young Adults With Fragile X Syndrome, Angelman Syndrome, Tuberous Sclerosis Complex, and Neurofibromatosis Type 1: A Cross-Syndrome Comparison.

Objective: The etiology of autism spectrum disorder (ASD) remains unclear, due to genetic heterogeneity and heterogeneity in symptoms across individuals. This study compares ASD symptomatology between monogenetic syndromes with a high ASD prevalence, in order to reveal syndrome specific vulnerabilities and to clarify how genetic variations affect ASD symptom presentation. Methods: We assessed ASD symptom severity in children and young adults (aged 0-28 years) with Fragile X Syndrome (FXS, n = 60), Angelman Syndrome (AS, n = 91), Neurofibromatosis Type 1 (NF1, n = 279) and Tuberous Sclerosis Complex (TSC, n = 110), using the Autism Diagnostic Observation Schedule and Social Responsiveness Scale. Assessments were part of routine clinical care at the ENCORE expertise center in Rotterdam, the Netherlands. First, we compared the syndrome groups on the ASD classification prevalence and ASD severity scores. Then, we compared individuals in our syndrome groups with an ASD classification to a non-syndromic ASD group (nsASD, n = 335), on both ASD severity scores and ASD symptom profiles. Severity scores were compared using MANCOVAs with IQ and gender as covariates. Results: Overall, ASD severity scores were highest for the FXS group and lowest for the NF1 group. Compared to nsASD, individuals with an ASD classification in our syndrome groups showed less problems on the instruments' social domains. We found a relative strength in the AS group on the social cognition, communication and motivation domains and a relative challenge in creativity; a relative strength of the NF1 group on the restricted interests and repetitive behavior scale; and a relative challenge in the FXS and TSC groups on the restricted interests and repetitive behavior domain. Conclusion: The syndrome-specific strengths and challenges we found provide a frame of reference to evaluate an individual's symptoms relative to the larger syndromic population and to guide treatment decisions. Our findings support the need for personalized care and a dimensional, symptom-based diagnostic approach, in contrast to a dichotomous ASD diagnosis used as a prerequisite for access to healthcare services. Similarities in ASD symptom profiles between AS and FXS, and between NF1 and TSC may reflect similarities in their neurobiology. Deep phenotyping studies are required to link neurobiological markers to ASD symptomatology.

Caring for Children with an Autism Spectrum Disorder: Factors Associating with Health- and Care-Related Quality of Life of the Caregivers.

This study investigated the association of child, caregiver, and caregiving measurements with the quality of life (QoL) in 81 caregivers (mostly parents) of clinically referred children with autism spectrum disorder (ASD). We used the EuroQol five-dimensional (EQ-5D) questionnaire and the care-related QoL questionnaire (CarerQol) to respectively assess health-related QoL and care-related QoL. Health-related QoL was associated with the caregiver's internalizing problems and adaptive coping, explaining 38% of the variance. Parenting stress and adaptive coping were associated with the care-related QoL and explained 60% of the variance. Child variables were not associated with the caregiver's health- and care-related QoL if caregiver and caregiving variables were taken into account. Findings indicate the importance of the caregiver's mental health, coping, and parenting stress in caring for children with ASD.

Children with an Autism Spectrum Disorder and Their Caregivers: Capturing Health-Related and Care-Related Quality of Life.

This study investigated health-related QoL (HRQoL) and care-related quality of life (CarerQol) in clinically referred children with an autism spectrum disorder (ASD), and their primary and secondary caregivers. The EuroQol five-dimensional (EQ-5D) and the CarerQol questionnaires were used to respectively measure health-related QoL and care-related QoL. Primary caregivers reported pain/discomfort (42%) and anxiety/depression (40%). In caring, they mostly experienced problems in the relationship with the child (84%), and in combining care with daily activities (51%). Children with ASD had a relevantly lower QoL. Despite negative effects, almost all caregivers (96%) derived fulfillment from caring for their affected children. HRQoL and CarerQol reports of primary caregivers and children were correlated, both providing useful information to ASD measurement and treatment.

Systematic Review and Meta-analysis of Virtual Reality in Pediatrics: Effects on Pain and Anxiety.

BACKGROUND: Medical procedures often evoke pain and anxiety in pediatric patients. Virtual reality (VR) is a relatively new intervention that can be used to provide distraction during, or to prepare patients for, medical procedures. This meta-analysis is the first to collate evidence on the effectiveness of VR on reducing pain and anxiety in pediatric patients undergoing medical procedures. METHODS: On April 25, 2018, we searched EMBASE, MEDLINE, CENTRAL, PubMed, Web of Science, and PsycINFO with the keywords "VR," "children," and "adolescents." Studies that applied VR in a somatic setting with participants ≤21 years of age were included. VR was defined as a fully immersive 3-dimensional environment displayed in surround stereoscopic vision on a head-mounted display (HMD). We evaluated pain and anxiety outcomes during medical procedures in VR and standard care conditions. RESULTS: We identified 2889 citations, of which 17 met our inclusion criteria. VR was applied as distraction (n = 16) during venous access, dental, burn, or oncological care or as exposure (n = 1) before elective surgery under general anesthesia. The effect of VR was mostly studied in patients receiving burn care (n = 6). The overall weighted standardized mean difference (SMD) for VR was 1.30 (95% CI, 0.68-1.91) on patient-reported pain (based on 14 studies) and 1.32 (95% CI, 0.21-2.44) on patient-reported anxiety (based on 7 studies). The effect of VR on pediatric pain was also significant when observed by caregivers (SMD = 2.08; 95% CI, 0.55-3.61) or professionals (SMD = 3.02; 95% CI, 0.79-2.25). For anxiety, limited observer data were available. CONCLUSIONS: VR research in pediatrics has mainly focused on distraction. Large effect sizes indicate that VR is an effective distraction intervention to reduce pain and anxiety in pediatric patients undergoing a wide variety of medical procedures. However, further research on the effect of VR exposure as a preparation tool for medical procedures is needed because of the paucity of research into this field.

Temperament and character in men with autism spectrum disorder: A reanalysis of scores on the Temperament and Character Inventory by individual case matching.

BACKGROUND: Interest in autism spectrum disorders (ASD) in adulthood is increasing. Although a person may be diagnosed with ASD, the diagnosis reveals little about the individual's temperament, character, and personality. Also, relatively little is known about the personality of adults with ASD. METHOD: A reanalysis of scores on the Temperament and Character Inventory (TCI) administered to a group of 66 normally intelligent men aged 18-63 years, diagnosed with ASD, by individual case matching to a comparison group of 66 men from the general population drawn from the TCI manual. RESULTS: Compared to the comparison group, men with ASD scored significantly higher on the scale for Harm Avoidance, and lower on Novelty Seeking, Reward Dependence, Self-Directedness, and Cooperativeness. CONCLUSIONS: In this study the score pattern for temperament and character found in men with ASD by individual case matching confirms and strengthens earlier general group matching findings emerging from our 2012 study and from studies from Sweden and the Netherlands.

Development of emotional and behavioral problems in neurofibromatosis type 1 during young childhood.

This retrospective longitudinal study in young children with neurofibromatosis type 1 (NF1) aimed to identify if, and how early problems in behavior, intelligence, and language development are associated with later behavioral problems. At the first assessment at preschool age, we evaluated language skills, intelligence, and emotional and behavioral problems as reported by parents. The second assessment at school-age we evaluated intelligence, and emotional and behavioral problems as reported by parents and teachers. Association of baseline assessments with secondary assessment was evaluated using multivariable linear regression analysis. Of the 61 patients (25 males, 36 females; mean age 4;5 years [SD 1;1 years]) with NF1 who had a first assessment, 38 children (21 males, 17 females; mean age 7;11 years [SD 2;1 years]) had a second assessment after a mean period of 3;5 years. Longitudinal data on behavioral problems were collected for 23 of these children. Intelligence and language development were not associated with internalizing problems. Parent-rated internalizing behavioral problems significantly increased with age in this subgroup. Baseline internalizing problems predicted later internalizing problems (adjusted R2 = 0.33, p = 0.003). The presence of these problems at pre-school age may be predictive of internalizing problems at a later age.

Motor problems in children with neurofibromatosis type 1.

BACKGROUND: Children with the neurogenetic disorder neurofibromatosis type 1 (NF1) often have problems with learning and behaviour. In both parent reports and neuropsychological assessment, motor problems are reported in approximately one third to one half of the children with NF1. Studies using broad motor performance test batteries with relatively large groups of children with NF1 are limited. The aim of this cross-sectional observational study was to describe the severity of motor problems in children with NF1 and to explore the predictive value of demographics, intelligence, and behavioural problems. METHODS: From 2002 to 2014, 69 children with NF1, aged 4 to 16 years (age = 9.5 ± 2.8 years; 29 girls) had a motor, psychological, and neurological evaluation in an NF1 expertise centre. Data were collected about (1) motor performance (M-ABC: Movement Assessment Battery for Children), (2) intelligence, and (3) emotional and behavioural problems as rated by parents. RESULTS: Sixty-one percent of these children scored within the clinical range of the M-ABC. In ordinal logistic regression analyses, motor problems were associated with symptoms of attention-deficit/hyperactivity disorder (ADHD), symptoms of autism spectrum disorder (ASD), and externalising behavioural problems. Motor outcome was not predicted by age, intelligence, scoliosis, hypotonia, nor hypermobility. CONCLUSIONS: Motor problems are among the most common comorbid developmental problems in children with NF1, and these problems do not diminish with age. Because of their impact on daily functioning, motor problems need to be specifically addressed in diagnosis, follow-up, and treatment of NF1.

Empirically based phenotypic profiles of children with pervasive developmental disorders: interpretation in the light of the DSM-5.

This study aimed to contribute to the Diagnostic and Statistical Manual (DSM) debates on the conceptualization of autism by investigating (1) whether empirically based distinct phenotypic profiles could be distinguished within a sample of mainly cognitively able children with pervasive developmental disorder (PDD), and (2) how profiles related to diagnoses and co-occurring behavioral and emotional problems. Six classes with distinct profiles were discerned. Three classes showed profiles not completely in line with the proposed DSM-5 conceptualization of autism. These classes included relatively many cognitively able individuals with PDD-not otherwise specified. However, profiles seemed to suit other diagnostic categories, such as social communication disorder. These alternative diagnoses could retain eligibility for services, and might adequately fit more specifically targeted interventions.

The association of quality of social relations, symptom severity and intelligence with anxiety in children with autism spectrum disorders.

Limited quality of social relations, milder symptom severity and higher intelligence were shown to account for higher anxiety levels in autism spectrum disorders. The current study replicated and extended earlier findings by combining these three determinants of anxiety in autism spectrum disorders in one study. The sample consisted of 134 school-aged children with autism spectrum disorders, of whom 58 (43%) had a co-morbid anxiety disorder according to the Diagnostic Interview Schedule for Children-Parent version. In this sample, we tested associations between these determinants and anxiety univariately and multivariately to clarify the unique contribution of all determinants. Since we hypothesized that the association between limited quality of social relations and anxiety would be amplified by low symptom severity and/or high intelligence, we additionally tested for moderating effects. We found that higher anxiety levels were associated with a lower quality of social relations and lower symptom severity. In this mainly high-functioning sample, intelligence was not related to anxiety levels. No moderation effects were found. Since lower quality of social relations and lower symptom severity are associated with higher anxiety levels in children with autism spectrum disorders, therapeutic interventions aimed at reducing anxiety in autism spectrum disorders should pay attention to improving social relations, and presumably children with a lower symptom severity could benefit most from such interventions.

The association between formal thought disorder and finger print asymmetry in children with a psychiatric disorder: an exploratory study.

Dermatoglyphics are the ridge constellations found on the hand palms and foot soles that are permanently formed by the 24th week of pregnancy. Associations have been found between adult schizophrenia and irregularities and asymmetries in dermatoglyphics. Children have not been studied before. The aim of this study was to assess the association between formal thought disorder (FTD), as a possible forerunner of schizophrenia, in children and asymmetry or discordance (DISC) of the finger prints. 222 children, aged 6-14, from an outpatient department of child psychiatry participated. Finger prints were rated with the three-pattern system (whorls, loops or arches). FTD criteria were illogical thinking (ILL), loose associations (LA), incoherence (INC), and poverty of content of speech (POC), as rated by the clinician. When boys with and without DISC were compared, no differences in FTD were found. In contrast, however, girls with DISC showed significantly more FTD than girls without DISC, t (72) = -2.39, p = 0.02. Further, for boys, only total FTD was positively correlated with DISC of the middle finger, r = 0.20, p = 0.02. For girls, total FTD was positively correlated with DISC of the index finger: r = 0.30, p = 0.02; DISC of the middle finger: r = 0.27, p = 0.03; and with total DISC dichotomous: r = 0.27, p = 0.02. In addition, total DISC correlated positively with ILL: r = 0.31, p = 0.01; LA: r = 0.23, p = 0.05; INC: r = 0.30, p = 0.01; and total FTD: r = 0.31, p = 0.01. Overall, the existence of finger print discordance, as a possible marker of prenatal instability, was associated with the occurrence of FTD in girls.

Autistic features in girls from a psychiatric sample are strongly associated with a low 2D:4D ratio.

Autistic features such as deficits in social interactions and communication have been associated with a low 2D:4D ratio in normal children.This study assessed this association in a large sample of children with a variety of psychiatric disorders (n = 35 girls and n = 147 boys). Autistic features were assessed with a highly valid and reliable measure (Autism Diagnostic Observation Schedule-Generic). Correlations between the 2D:4D ratio and autistic features were computed separately for boys and girls. Some small negative correlations (r = -0.17 and r = -0.19) were found in the right hand for boys; however, particularly in girls, large negative correlations (r = -0.51 to r = -0.64) were found in the left hand. A low 2D:4D ratio in girls was highly predictive of the presence of autistic features. Thus, a low ratio could possibly be used as a diagnostic predictor in clinical practice.

Classes of disruptive behavior problems in referred adolescents.

BACKGROUND: Previous studies have found considerable overlap between attention/hyperactivity problems, aggressive/oppositional problems and delinquent/conduct problems in adolescents. SAMPLING AND METHODS: Mothers of 1,965 11- to 18-year-olds (1,116 boys, 849 girls), referred to mental health agencies, completed the Child Behavior Checklist (CBCL). Latent class analysis was conducted on the Attention Problems scale (representing problems with attention, impulsivity and hyperactivity), Aggressive Behavior and Rule-Breaking Behavior scales of the CBCL. RESULTS: Six latent classes were found. One of these classes contained individuals who suffered predominantly from attention problems and to a far lesser degree from aggressive or rule-breaking behaviors. The other 5 classes represented individuals with varying degrees of attention problems, aggressive behaviors and rule-breaking behaviors. CONCLUSIONS: Contrary to previous studies, the present study indicated that, in a large referred sample, problems with attention, impulsivity and hyperactivity can be considered as a diagnostic construct that should be distinguished from aggressive or rule-breaking behaviors. However, the present study did not support the existence of diagnostic classes constituted by individuals who primarily suffer from aggressive behaviors or rule-breaking behaviors, and not from attention problems or hyperactivity. Implications of these findings for future research and clinical practice are discussed. The value of the study was limited by the use of parent reports only.

No hyperactive-impulsive subtype in teacher-rated attention-deficit/hyperactivity problems.

The aim of this study was to investigate which homogeneous groups, according to teacher reports of attention-deficit/hyperactivity (ADH) Problems on the Teacher's Report Form (TRF), can be identified in a referred sample (n = 4,422; age = 6-18 years; mean age = 9.9 years; 66% boys, 34% girls). Latent class analysis (LCA) was conducted on ADH Problems. In addition, co-morbidity levels in the different ADH Problems groups were compared. LCA yielded three different groups of children and adolescents with both Inattention and hyperactivity-impulsivity, and one group with high scores on Inattention but low scores on hyperactivity-impulsivity. A group of patients with predominantly hyperactivity and impulsivity was not found. Individuals in groups with higher levels of ADH Problems had significantly higher levels of oppositional defiant (OD) and conduct problems, and, although to a lesser extent, significantly higher levels of affective and anxiety problems than individuals in groups with lower levels of ADH Problems. It may not be useful to discern the hyperactive-impulsive type of ADHD.

High rates of psychiatric co-morbidity in PDD-NOS.

Rates of co-morbid psychiatric conditions in children with Pervasive Developmental Disorder-Not Otherwise Specified (PDD-NOS) are hardly available, although these conditions are often considered as more responsive to treatment than the core symptoms of PDD-NOS. Ninety-four children with PDD-NOS, aged 6-12 years were included. The DISC-IV-P was administered. At least one co-morbid psychiatric disorder was present in 80.9% of the children; 61.7% had a co-morbid disruptive behavior disorder, and 55.3% fulfilled criteria of an anxiety disorder. Compared to those without co-morbid psychiatric disorders, children with a co-morbid disorder had more deficits in social communication. Co-morbid disorders occur very frequently in children with PDD-NOS, and therefore clinical assessment in those children should include assessment of co-morbid DSM-IV disorders.

Multiple complex developmental disorder delineated from PDD-NOS.

The objective of this study was to identify behavioral differences between children with multiple complex developmental disorder (MCDD) and those with pervasive developmental disorder-not otherwise specified (PDD-NOS). Twenty-five children (6-12 years) with MCDD and 86 children with PDD-NOS were compared with respect to psychiatric co-morbidity, psychotic thought problems and social contact problems using the child behavior checklist/4-18, the Dutch version of the diagnostic interview schedule for children-Version IV, the child and adolescent functional assessment scale, and the autism diagnostic observation schedule-generic. MCDD was associated with anxiety disorders, disruptive behavior, and psychotic thought problems. PDD-NOS was associated with deficits in social contact. MCDD differs from autistic disorder, and can also be delineated from PDD-NOS.

Latent class analysis of anxiety and depressive symptoms in referred adolescents.

BACKGROUND: Comorbidity between anxiety disorders and depressive disorders in adolescents occurs very frequently. The aim of this study was to investigate if anxiety and depression represent distinct disease entities. METHODS: Two thousand thirty two 11- to 18-year-olds, referred to mental health services, were investigated. Self-reported symptoms of anxiety and depression were analyzed with latent class analysis. RESULTS: The numbers of adolescents with severe depression symptoms, but with less severe, or absent, anxiety symptoms, were negligible. Further, two subgroups could be discerned in those with severe anxiety symptoms: one with severe depressive symptoms, and one with mild depressive symptoms. LIMITATIONS: A self-report questionnaire was used, whereas diagnostic interview information regarding DSM-IV disorders was not available. DISCUSSION: It seems less useful to develop treatments, or to conduct studies aimed at etiological mechanisms, that are specifically aimed at depression and not at anxiety. Further, it may be relevant to discern specific subgroups of anxious adolescents, with severe versus mild comorbid depression symptoms.

Attention-deficit/hyperactivity disorder (ADHD): parents' judgment about school, teachers' judgment about home.

OBJECTIVE: The aim of this study was to separate sources of observer and situational variance in reporting attention-deficit/hyperactivity disorder (ADHD) symptomatology. METHOD: In a sample of 30 children diagnosed with ADHD, ADHD symptomatology was assessed with the Diagnostic Interview Schedule for Children-Parent Version (DISC-P), with parents and teachers as informants. Both parents and teachers reported about the child's ADHD symptomatology at home as well as at school. RESULTS: Parents and teachers showed high within-observer cross-situational presence of ADHD symptoms. However, the between-observer agreement on the presence of ADHD symptoms within the same situation (home or school) was low. This pattern held equally true for attention/concentration and hyperactivity/impulsivity symptom scores. CONCLUSIONS: In evaluating ADHD symptomatology, it is important to obtain independent reports about the child's behaviour at school from the teacher and about the child's behaviour at home from the parents.