Guanfacine Use in Children With Down Syndrome and Comorbid Attention-Deficit Hyperactivity Disorder (ADHD) With Disruptive Behaviors

Capone, George T.; Brecher, Liza; Bay, Mihee

doi:10.1177/0883073816634854

Cited by 24 publications

(8 citation statements)

References 32 publications

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“…In a study of 154 children and adolescents with intellectual disability (ID), a Conners Parent Rating Scale total score of 42 provided a sensitivity of 0.9 and a specificity of 0.67 with an area under the curve of 0.84, with strong discriminant validity between children with ID with and without ADHD 39 . The parent version of the Conners-3 also has been used previously in other studies targeting ADHD symptoms of individuals with DS 17 , 40 , with mean T-scores consistent with diagnostic features of ADHD 40 . For our primary analyses, we used T-scores for the inattentive and the hyperactive-impulsive subscales as the primary symptoms of ADHD, as well as the global index T-score.…”

Section: Methodsmentioning

confidence: 99%

Gestational age is related to symptoms of attention-deficit/hyperactivity disorder in late-preterm to full-term children and adolescents with down syndrome

Soriano

Rosser

Hamilton

et al. 2020

Sci Rep

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Attention-deficit/hyperactivity disorder is frequently reported in individuals with Down syndrome, with considerable variation in the expression and severity of the symptoms. Despite growing evidence that gestational age predicts later symptoms of attention-deficit/hyperactivity disorder in the euploid population, this has not been studied in down syndrome. The current study is designed to investigate the influence of gestational age in later symptoms of attention-deficit/hyperactivity disorder in 105 individuals (49 males and 56 females; aged 6–18 years) with Down syndrome who were born at or after 35 weeks gestation. Maternal age at birth, maternal level of education, household income, as well as sex, chronological age, and cognitive level of the participant with Down syndrome were considered in our analysis. Results from this study show that gestational age is related to inattentive and hyperactive/impulsive symptoms in children and adolescents with Down syndrome. Therefore, gestational age should be addressed when considering symptoms of attention-deficit/hyperactivity disorder, as it may have implications for early interventions. More attention is needed toward the advancement of care and follow-up for infants with down syndrome who are born even late preterm or early term.

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Section: Methodsmentioning

confidence: 99%

Gestational age is related to symptoms of attention-deficit/hyperactivity disorder in late-preterm to full-term children and adolescents with down syndrome

Soriano

Rosser

Hamilton

et al. 2020

Sci Rep

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“…Another reason is that scientific evidence available on the pharmacological treatment of ADHD in DS is limited to case reports and an open‐label trial of guanfacine (Capone et al . 2016; Palumbo & McDougle 2018). As the French Health Authority only recommends guanfacine as a drug of last resort because of its adverse effects (Haute Autorité de Santé n.d.…”

Section: Discussionmentioning

confidence: 99%

Efficacy and safety of methylphenidate on attention deficit hyperactivity disorder in children with Down syndrome

Roche

Mircher

Toulas

et al. 2021

J intellect Disabil Res

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Background Attention deficit hyperactivity disorder (ADHD) is a common co‐morbidity that affects up to 44% of children with Down syndrome (DS). There is a need for reliable, good quality research on the use of methylphenidate within this population. The objective of this study is to report our experience regarding the management of ADHD in these children using methylphenidate. Methods This study is a retrospective observation of 21 children with DS, followed at Jérôme Lejeune Institute between 2000 and 2018. The diagnosis of ADHD was made using the Diagnostic and Statistical Manual of Mental Disorders criteria. Efficacy was measured as response or non‐response on two main symptoms: attention/concentration and hyperactivity/impulsivity. Safety was evaluated by the presence or absence of side effects. Results Sixteen out of the 21 children (76%) showed improvement with methylphenidate. The average age of treatment onset in responding children was 8 years and 10 months versus 6 years and 3 months in non‐responders (P = 0.05). Average dose/weight was significantly different in responders and non‐responders (0.82 vs. 0.54 mg/kg/day, respectively; P = 0.03). Twelve children out of 21 (57%) experienced side effects; only three experienced side effects severe enough to require treatment interruption. Most common side effects were loss of appetite and difficulties in falling asleep. Conclusion Methylphenidate was effective and safe in treating ADHD in 76% of cases in children with DS, with few serious side effects to report. Early diagnosis of ADHD is important to improve the quality of life, learning, inclusion and socialisation of children with DS.

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“…DS occurs in approximately 1 in 700 live births, and its incidence increases with a high maternal age (Carothers et al, 1999). DS involves a remarkably broad spectrum of human disorders (Capone et al, 2016;Zhao Y. et al, 2017), including chronic systemic inflammation, lupus erythematosus, amyotrophic lateral sclerosis, various cancers, and autism spectrum disorder (Zis et al, 2017). The neuropathological changes noted in nearly all DS cases > 35 years closely resemble Alzheimer's disease (AD) (Tramutola et al, 2020), suggesting an overlap of the underlying molecular mechanisms between these two disorders.…”

Section: Introductionmentioning

confidence: 99%

Integrative Analysis Extracts a Core ceRNA Network of the Fetal Hippocampus With Down Syndrome

et al. 2020

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Accumulating evidence suggests that circular RNAs (circRNAs)—miRNA–mRNA ceRNA regulatory network—may play an important role in neurological disorders, such as Alzheimer’s disease (AD). Interestingly, neuropathological changes that closely resemble AD have been found in nearly all Down syndrome (DS) cases > 35 years. However, few studies have reported circRNA transcriptional profiling in DS cases, which is caused by a chromosomal aberration of trisomy 21. Here, we characterized the expression profiles of circRNAs in the fetal hippocampus of DS patients (n = 8) and controls (n = 6) by using microarray. MiRNA, mRNA expression profiling of DS from our previous study and scRNA-seq data describing normal fetal hippocampus development (GEO) were also integrated into the analysis. The similarity between circRNAs/genes with traits/cell-types was calculated by weighted correlation network analysis (WGCNA). miRanda and miRWalk2 were used to predict ceRNA network interactions. We identified a total of 7,078 significantly differentially expressed (DE) circRNAs, including 2,637 upregulated and 4,441 downregulated genes, respectively. WGCNA obtained 15 hub circRNAs and 6 modules with cell type–specific expression patterns among scRNA-seq data. Finally, a core ceRNA network was constructed by 14 hub circRNAs, 17 DE miRNA targets and 245 DE mRNA targets with a cell type–specific expression pattern annotation. Known functional molecules in DS or neurodegeneration (e.g., miR-138, OLIG1, and TPM2) were also included in this network. Our findings are the first to delineate the landscape of circRNAs in DS and the first to effectively integrate ceRNA regulation with scRNA-seq data. These data may provide a valuable resource for further research on the molecular mechanisms or therapeutic targets underlying DS neuropathy.

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Guanfacine Use in Children With Down Syndrome and Comorbid Attention-Deficit Hyperactivity Disorder (ADHD) With Disruptive Behaviors

Cited by 24 publications

References 32 publications

Gestational age is related to symptoms of attention-deficit/hyperactivity disorder in late-preterm to full-term children and adolescents with down syndrome

Gestational age is related to symptoms of attention-deficit/hyperactivity disorder in late-preterm to full-term children and adolescents with down syndrome

Efficacy and safety of methylphenidate on attention deficit hyperactivity disorder in children with Down syndrome

Integrative Analysis Extracts a Core ceRNA Network of the Fetal Hippocampus With Down Syndrome

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