Prevalence and characteristics of autism spectrum disorders in children with cerebral palsy

Delobel‐Ayoub, Malika; Klapouszczak, Dana; Bakel, Marit Maria Elisabeth van; Horridge, Karen; Sigurðardóttir, Sólveig; Himmelmann, Kate; Arnaud, Catherine

doi:10.1111/dmcn.13436

Cited by 47 publications

(36 citation statements)

References 22 publications

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“…Some more children had been investigated and diagnosed since that study. The 18% reported in the present study is higher than in previous population‐based studies . Impairments are easier to detect at older age, and there is an increasing awareness of these impairments among clinicians, which may explain the higher rates in the present study.…”

Section: Discussioncontrasting

confidence: 69%

“…More children were diagnosed with ASD and ADHD with age. ASD has previously been reported from partly the same population, but at a younger age, by Delobel‐Ayoub et al with a prevalence of 15% . Some more children had been investigated and diagnosed since that study.…”

Section: Discussionsupporting

confidence: 55%

“…The generalisability of the present results may be limited to other high‐income countries, which are more likely to have a similar panorama of CP. However, traditions in diagnosing neuropsychiatric impairments may differ between countries , which may affect the possibility to compare different populations.…”

Section: Discussionmentioning

confidence: 99%

“…Behaviour problems are also common in children with CP , including neuropsychiatric disorders such as autism spectrum disorder (ASD) and attention‐deficit/hyperactivity disorder (ADHD). During the last decade, there has been growing awareness particularly of ASD being overrepresented in children with CP, reported from 3% to 16% , depending on the group studied and the methods used. In a European multicentre CP study by Delobel‐Ayoub et al, male sex, epilepsy, ID and better walking ability were associated with ASD in children with CP .…”

Section: Introductionmentioning

confidence: 99%

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One‐third of school‐aged children with cerebral palsy have neuropsychiatric impairments in a population‐based study

2019

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Aim: To describe motor function and associated impairments, particularly autism spectrum disorder (ASD) and attention-deficit/hyperactivity disorder (ADHD), in school-aged children with cerebral palsy (CP).Methods: Population-based study of all children with CP born 1999-2006 from the county of V€ astra G€ otaland, Sweden; 264 children (141 males, 123 females). Information was obtained from the CP Register of western Sweden (data collected at 4-8 years of age) and all medical records at 10-17 years of age.Results: Cerebral palsy was spastic in 76%, dyskinetic in 17% and ataxic in 7% of all children. Sixty-three per cent were independent walkers. Associated impairments were present in 75%. Vision was impaired in 19%, hearing in 8% and speech in 54%. Intellectual disability (ID) was present in 53% and epilepsy in 41%. ID had increased from 42% to 53% since preschool-age. Neuropsychiatric impairments were present in 32% of the children; ASD in 18%; and ADHD in 21%. All impairments, except for ASD and ADHD, increased with more severe motor impairment.Conclusion: Three in four school-aged children with CP have associated impairments, underscoring the need to broadly assess every child. The high rate of ASD and ADHD points to the importance of in-depth studies of such impairments in CP. 2048

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Section: Discussioncontrasting

confidence: 69%

Section: Discussionsupporting

confidence: 55%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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One‐third of school‐aged children with cerebral palsy have neuropsychiatric impairments in a population‐based study

2019

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“…Detailed cognitive assessments evaluating domains such as visual and perceptual functioning are necessary to determine whether some behavioural symptoms are due to existing deficits of the CP or represent an additional neurodevelopmental disorder. For example, other neurodevelopmental disorders such as autism spectrum disorder and attention‐deficit–hyperactivity disorder can co‐occur with CP. The symptoms of each of these disorders can create complex behavioural challenges for both the affected child and their family.…”

Section: Discussionmentioning

confidence: 99%

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

Downs

Blackmore²,

Epstein

et al. 2017

Develop Med Child Neuro

102

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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. What this paper adds Children with cerebral palsy and intellectual disability have a higher risk of mental health symptoms. The prevalence of mental health symptoms for age and severity groups is unclear.

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Diagnostic Yield of Exome Sequencing in Cerebral Palsy and Implications for Genetic Testing Guidelines

Gonzalez-Mantilla

Myers

et al. 2023

JAMA Pediatr

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ImportanceExome sequencing is a first-tier diagnostic test for individuals with neurodevelopmental disorders, including intellectual disability/developmental delay and autism spectrum disorder; however, this recommendation does not include cerebral palsy.ObjectiveTo evaluate if the diagnostic yield of exome or genome sequencing in cerebral palsy is similar to that of other neurodevelopmental disorders.Data SourcesThe study team searched PubMed for studies published between 2013 and 2022 using cerebral palsy and genetic testing terms. Data were analyzed during March 2022.Study SelectionStudies performing exome or genome sequencing in at least 10 participants with cerebral palsy were included. Studies with fewer than 10 individuals and studies reporting variants detected by other genetic tests were excluded. Consensus review was performed. The initial search identified 148 studies, of which 13 met inclusion criteria.Data Extraction and SynthesisData were extracted by 2 investigators and pooled using a random-effects meta-analysis. Incidence rates with corresponding 95% CIs and prediction intervals were calculated. Publication bias was evaluated by the Egger test. Variability between included studies was assessed via heterogeneity tests using the I2 statistic.Main Outcomes and MeasuresThe primary outcome was the pooled diagnostic yield (rate of pathogenic/likely pathogenic variants) across studies. Subgroup analyses were performed based on population age and on the use of exclusion criteria for patient selection.ResultsThirteen studies were included consisting of 2612 individuals with cerebral palsy. The overall diagnostic yield was 31.1% (95% CI, 24.2%-38.6%; I2 = 91%). The yield was higher in pediatric populations (34.8%; 95% CI, 28.3%-41.5%) than adult populations (26.9%; 95% CI, 1.2%-68.8%) and higher among studies that used exclusion criteria for patient selection (42.1%; 95% CI, 36.0%-48.2%) than those that did not (20.7%; 95% CI, 12.3%-30.5%).Conclusions and RelevanceIn this systematic review and meta-analysis, the genetic diagnostic yield in cerebral palsy was similar to that of other neurodevelopmental disorders for which exome sequencing is recommended as standard of care. Data from this meta-analysis provide evidence to support the inclusion of cerebral palsy in the current recommendation of exome sequencing in the diagnostic evaluation of individuals with neurodevelopmental disorders.

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Prevalence and characteristics of autism spectrum disorders in children with cerebral palsy

Abstract: Our findings support the need for a multidisciplinary approach to management of children with CP to adequately identify and address all facets of presentation, including ASD.

Cited by 47 publications

References 22 publications

One‐third of school‐aged children with cerebral palsy have neuropsychiatric impairments in a population‐based study

One‐third of school‐aged children with cerebral palsy have neuropsychiatric impairments in a population‐based study

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

Diagnostic Yield of Exome Sequencing in Cerebral Palsy and Implications for Genetic Testing Guidelines

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