Parental Reports on Early Autism Behaviors in Their Children with Fragile X Syndrome as a Function of Infant Feeding.
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ABSTRACT:
SUBMITTER: Westmark CJ
PROVIDER: S-EPMC8401950 | biostudies-literature |
REPOSITORIES: biostudies-literature
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Project description:Cerebellar dysfunction has been linked to autism spectrum disorders (ASDs). Although cerebellar pathology has been observed in individuals with fragile X syndrome (FXS) and in mouse models of the disorder, a cerebellar functional contribution to ASD-relevant behaviors in FXS has yet to be fully characterized. In this study, we demonstrate a critical cerebellar role for Fmr1 (fragile X messenger ribonucleoprotein 1) in ASD-relevant behaviors. First, we identify reduced social behaviors, sensory hypersensitivity, and cerebellar dysfunction, with loss of cerebellar Fmr1. We then demonstrate that cerebellar-specific expression of Fmr1 is sufficient to impact social, sensory, cerebellar dysfunction, and cerebro-cortical hyperexcitability phenotypes observed in global Fmr1 mutants. Moreover, we demonstrate that targeting the ASD-implicated cerebellar region Crus1 ameliorates behaviors in both cerebellar-specific and global Fmr1 mutants. Together, these results demonstrate a critical role for the cerebellar contribution to FXS-related behaviors, with implications for future therapeutic strategies.
| S-EPMC10831814 | biostudies-literature
Project description:PURPOSE: The goals of this study were to 1) describe the feeding skills of young children with cerebral palsy (CP); and 2) elucidate the type and severity of feeding problems for children with and without oral-motor involvement. METHOD: Parents of 37 children (16 females, 21 males) with CP, who ranged in age from 11-58 months (mean age = 41 months), completed questionnaires regarding their child's past and current feeding abilities. Children were also clinically evaluated to determine whether each had evidence of oral-motor involvement. RESULTS: Children with CP and oral-motor involvement had significantly more difficulty with self-feeding, increased frequency of coughing and choking, increased prevalence of swallowing evaluation and feeding therapy, and were introduced to solid food at a later age relative to children with CP who did not have oral-motor involvement. Both groups of children were similar in their history of tube feeding, bottle feeding, difficulty with solid foods, use of adaptive equipment, duration of mealtimes, and presence of choking, coughing, and gagging. CONCLUSIONS: Children with and without oral-motor involvement initially presented with similar feeding difficulties. However, feeding problems appeared to resolve to a greater extent in children without oral-motor involvement. The difficulties identified early in life, for children with oral-motor involvement, appeared to persist with development.
| S-EPMC2688002 | biostudies-literature
Project description:Parental feeding practices have been associated with children's dietary intakes, yet the directionality of these associations remains unclear. Among 1172 mother-child pairs from Project Viva, we aimed to examine associations of parental concerns and feeding behaviors at 2 years (behaviors dichotomized as yes vs. no), with diet quality (Youth Healthy Eating Index; YHEI) in early (mean 3.2, SD 0.3 years; n = 1076) and mid-childhood (mean 7.8, SD 0.7 years; n = 993). We used multivariable linear regression models adjusted for sociodemographic characteristics, parental body mass index (BMI), maternal diet quality in pregnancy, and child's BMI z-score and diet quality at 2 years. Early parental concerns about their child becoming overweight (15%) was associated with lower YHEI (β -1.54 points; 95%CI -2.75, -0.33; fully adjusted model) in early childhood. Early parental concerns about their child becoming underweight (7%) was associated with lower YHEI (-2.19 points; -4.31, -0.07) in early childhood, but the association was attenuated after adjustment for child's BMI z-score and diet quality at 2 years. We did not find associations of parental restrictive feeding (8%) and parental pressure to eat (47%) with child's YHEI through mid-childhood. In conclusion, we found no evidence that early parental concerns and feeding behaviors independently contribute to child's diet quality through childhood.
| S-EPMC7690442 | biostudies-literature
Project description:ObjectiveWe examined associations between household food insecurity status and parental feeding behavior, weight perception, and child weight status in a diverse sample of young children.MethodsCross-sectional analysis of 2-year-old children in Greenlight, a cluster randomized trial to prevent childhood obesity. The exposure was food insecurity, defined as a positive response to a validated screen. Outcomes were parent feeding behaviors/beliefs measured by the Child Feeding Questionnaire and child weight status. t tests and linear regression were used to assess associations between food insecurity and each outcome. We adjusted for child sex, race/ethnicity, parent education, employment, site, number of children in the home, and Special Supplemental Nutrition Program for Women, Infants, and Children status.ResultsFive hundred three households (37%) were food insecure. After adjusting for covariates, parents from insecure households reported more pressuring feeding behaviors (mean factor score 3.2 compared to food secure parents mean factor score 2.9, P = .01) and were more worried about their child becoming overweight (mean factor score 2.3 vs 2.0; P = .02). No differences were observed in monitoring or restrictive feeding behaviors. After adjusting for covariates, there was no difference in weight status or prevalence of overweight/obesity of children or parents based on household food insecurity status.ConclusionsParents from food insecure households reported more pressuring feeding behaviors. This finding underscores the need to address food insecurity and potentially prevent harmful effects on child feeding. Parents in food insecure households might benefit from linkage with resources and education to develop healthier feeding behaviors.
| S-EPMC7655687 | biostudies-literature
Project description:Autism spectrum disorder (ASD) is an early onset, developmental disorder whose genetic cause is heterogeneous and complex. In total, 70% of ASD cases are due to an unknown etiology. Among the monogenic causes of ASD, fragile X syndrome (FXS) accounts for 2-4% of ASD cases, and 60% of individuals with FXS present with ASD. Epigenetic changes, specifically DNA methylation, which modulates gene expression levels, play a significant role in the pathogenesis of both disorders. Thus, in this study, using the Human Methylation EPIC Bead Chip, we examined the global DNA methylation profiles of biological samples derived from 57 age-matched male participants (2-6 years old), including 23 subjects with ASD, 23 subjects with FXS with ASD (FXSA) and 11 typical developing (TD) children. After controlling for technical variation and white blood cell composition, using the conservatory threshold of the false discovery rate (FDR ≤ 0.05), in the three comparison groups, TD vs. AD, TD vs. FXSA and ASD vs. FXSA, we identified 156, 79 and 3100 differentially methylated sites (DMS), and 14, 13 and 263 differential methylation regions (DMRs). Interestingly, several genes differentially methylated among the three groups were among those listed in the SFARI Gene database, including the PAK2, GTF2I and FOXP1 genes important for brain development. Further, enrichment analyses identified pathways involved in several functions, including synaptic plasticity. Our preliminary study identified a significant role of altered DNA methylation in the pathology of ASD and FXS, suggesting that the characterization of a DNA methylation signature may help to unravel the pathogenicity of FXS and ASD and may help the development of an improved diagnostic classification of children with ASD and FXSA. In addition, it may pave the way for developing therapeutic interventions that could reverse the altered methylome profile in children with neurodevelopmental disorders.
| S-EPMC9602177 | biostudies-literature
Project description:PurposeIn this study, we examine joint engagement (JE) in young children with fragile X syndrome (FXS) and its relationship to language abilities and autism spectrum disorder symptomatology at 24 to 36 months (toddler period) and 59 to 68 months (child period).MethodParticipants were 28 children with FXS (24 boys, four girls) and their mothers. Videotaped home observations were conducted during the toddler period and coded for JE. Language abilities were measured at both ages from a developmental assessment, a functional measure, and from a language sample. The Childhood Autism Rating Scale (Schopler, Reichler, & Renner, 1988) was completed at both ages.ResultsChildren with FXS spent more time in supported JE than in coordinated JE. Using a weighted JE variable, we found that children with FXS who had higher weighted JE scores also had more advanced expressive language skills at both the toddler and child periods. Weighted JE was negatively related to autism symptomatology in the toddler period.ConclusionThis study provides evidence that children with FXS who use more JE also have more advanced expressive language skills in early development. Therefore, existing early interventions that target JE behaviors may be effective for promoting language, social communication, and social interaction in this population.
| S-EPMC5345555 | biostudies-literature
Project description:Fragile X syndrome is caused by the expansion of CGG triplets in the FMR1 gene, which generates epigenetic changes that silence its expression. The absence of the protein coded by this gene, FMRP, causes cellular dysfunction, leading to impaired brain development and functional abnormalities. The physical and neurologic manifestations of the disease appear early in life and may suggest the diagnosis. However, it must be confirmed by molecular tests. It affects multiple areas of daily living and greatly burdens the affected individuals and their families. Fragile X syndrome is the most common monogenic cause of intellectual disability and autism spectrum disorder; the diagnosis should be suspected in every patient with neurodevelopmental delay. Early interventions could improve the functional prognosis of patients with Fragile X syndrome, significantly impacting their quality of life and daily functioning. Therefore, healthcare for children with Fragile X syndrome should include a multidisciplinary approach.
| S-EPMC10469670 | biostudies-literature
Project description:BackgroundAlmost all children with autism spectrum disorder (ASD) have experienced challenging behavior, including disruptive and aggressive behavior symptoms to both themselves and others. In conjunction with appropriate strategic parenting, challenging behavior can be prevented by empowering children's sociality and optimizing their environment. However, a means of measuring such parenting has yet to appear. This study developed the Parental Self-Efficacy Scale for Preventing Challenging Behaviors in Children with Autism Spectrum Disorder (PASEC) and evaluated its reliability and validity.MethodSelf-administered questionnaires were distributed to 1,344 parents of children with ASD at all 521 child development support centers in Japan. Confirmed construct validity of the PASEC was determined using confirmatory factor analysis. Internal consistency of the PASEC was calculated using Cronbach's alpha. The self-efficacy subscale of the Parenting Sense of Competence (PSOC) was administered to assess criterion-related validity of the PASEC.ResultsIn total, 260 parents provided valid responses. Exploratory and confirmatory factor analyses identified six items from two factors: empowerment of children's sociality and optimization of children's environment. The final model showed goodness-of-fit index, 0.981; adjusted goodness-of-fit index, 0.944; comparative fit index, 0.999; and root mean square error of approximation, 0.019. Cronbach's alpha for the entire PASEC was 0.82; that for each factor was above 0.70. The correlation coefficient between the self-efficacy subscale of the PSOC and the entire PASEC was r = 0.52 (P <0.001).ConclusionsThe PASEC demonstrated adequate reliability and validity to assess parents' self-efficacy for preventing challenging behavior for children with ASD. That scale can help prevent challenging behavior; it can contribute to improving the mental health of parents and children with ASD as well as to primary prevention of child maltreatment and abuse.
| S-EPMC7470344 | biostudies-literature
Project description:Autism is a neurodevelopmental disorder consisting of a constellation of symptoms that sometimes occur as part of a complex disorder characterized by impairments in social interaction, communication and behavioral domains. It is a highly disabling disorder and there is a need for treatment targeting the core symptoms. Although autism is accepted as highly heritable, there is no genetic cure at this time. Autism is shown to be linked to several genes and is a feature of some complex genetic disorders, including fragile X syndrome (FXS), fragile X premutation involvement, tuberous sclerosis and Rett syndrome. The term autism spectrum disorders (ASDs) covers autism, Asperger syndrome and pervasive developmental disorders (PDD-NOS) and the etiologies are heterogeneous. In recent years, targeted treatments have been developed for several disorders that have a known specific genetic cause leading to autism. Since there are significant molecular and neurobiological overlaps among disorders, targeted treatments developed for a specific disorder may be helpful in ASD of unknown etiology. Examples of this are two drug classes developed to treat FXS, Arbaclofen, a GABA(B) agonist, and mGluR5 antagonists, and both may be helpful in autism without FXS. The mGluR5 antagonists are also likely to have a benefit in the aging problems of fragile X premutation carriers, the fragile X -associated tremor ataxia syndrome (FXTAS) and the Parkinsonism that can occur in aging patients with fragile X syndrome. Targeted treatments in FXS which has a well known genetic etiology may lead to new targeted treatments in autism.
| S-EPMC3498468 | biostudies-literature
Project description:Introduction Between the ages of 6 and 12 months is a crucial stage for children to develop appetitive self-regulation. Evidence suggests that a combination of parental responsive feeding and infant temperament (surgency, effortful control, negative affect) shapes infant appetitive traits (food approach, food avoidance). There is a need for research to explore these relationships, in order to provide guidance for the design of an effective intervention to improve appetitive traits. The objective of the current study was to explore the moderating role of infant temperament in the relationship between parental responsive feeding and infant appetitive traits. Methods A total of 616 questionnaires, measuring parental responsive feeding, infant appetitive traits, and infant temperament, were collected from parents with infants aged 6–12 months. Results Results revealed that responsive feeding was associated with both food approach and food avoidance. Furthermore, only lower levels of surgency significantly moderated the relationship between responsive feeding and food approach, while responsive feeding was associated with food avoidance irrespective of infant temperament. Discussion These findings suggest that a strategy embedding responsive feeding interventions should be adopted to reduce infant food avoidance and low-surgent infant food approach, and interventions that are tailored toward food approach for infants with effortful control, negative affect, or higher levels of surgency should be further sought.
| S-EPMC9939436 | biostudies-literature