[show abstract][hide abstract] ABSTRACT: This study examined sleep, sleepiness, and daytime performance in 68 children with autism, 57 children with intellectual disability (ID), and 69 typically developing preschool children. Children in the autism and ID groups had poorer daytime performance and behaviors than the typically developing children. Children in the ID group also were significantly sleepier than children in both the autism and typically developing groups. These significant differences persisted over 6 months. Actigraph-defined sleep behaviors and problems did not relate to daytime sleepiness or daytime performance and behaviors for the children with autism or the typically developing group. For the ID group, longer night awakenings and lower sleep efficiency predicted more daytime sleepiness. For each group, parent-report sleep problems were associated with more daytime sleepiness and more behavior problems.
American Journal on Intellectual and Developmental Disabilities 07/2012; 117(4):275-90. · 2.08 Impact Factor
[show abstract][hide abstract] ABSTRACT: This study examined sleep-wake patterns in 3 matched comparison groups of preschool-aged children: children with autism (AUT), children with developmental delay (DD) without AUT, and children who are developing typically (TYP). Sleep was assessed via actigraphy and parent-report diaries for 7 consecutive 24-hr periods across 3 time points: at enrollment (n = 194), 3 months later (n = 179), and 6 months after enrollment (n = 173). At each recording period, children in the AUT group slept less per 24-hr period, on average, and were less likely to awaken at night than children in the other two groups. In contrast, children in the DD group had more frequent and longer duration nighttime awakenings than children in the AUT group. Overall, children in the 2 neurodevelopmentally disordered groups demonstrated more night-to-night variability in their sleep-wake measures than children in the TYP group.
Behavioral Sleep Medicine 04/2011; 9(2):92-106. · 1.13 Impact Factor
[show abstract][hide abstract] ABSTRACT: The present study examined daytime sleep patterns in 3 groups of preschool-aged children: children with autism, children with developmental delay, and children who were developing typically. Sleep was assessed in 194 children via actigraphy and parent-report sleep diaries for 7 consecutive days on 3 separate occasions over 6 months. Children with autism napped less often and for shorter periods of time than children with developmental disability, with whom they were matched on chronologic age. Children with developmental disabilities napped more like children in the typically developing group, who were, on average, 6 months younger. Each group displayed an expected shift in daytime sleep as more children matured out of their naps.
American Journal on Intellectual and Developmental Disabilities 03/2011; 116(2):142-52. · 2.08 Impact Factor
[show abstract][hide abstract] ABSTRACT: Night awakenings are a normative part of early development. In the first year, night awakenings are associated with birth order, feeding route, sleep aid use, sleep location, infant temperament and development, infant-parent attachment, family socioeconomics, and cultural norms. In the second year, additional factors build on these foundational features, including parenting practices and object attachment. As children grow, contextual factors like preschool entry or changes in family member status may influence the continuation or exacerbation of awakenings. Future research should consider the multitude of factors that influence not only awakenings but also parental perceptions, family dynamics, and cultural norms.
International Review of Neurobiology 01/2010; 93:177-91. · 1.65 Impact Factor
[show abstract][hide abstract] ABSTRACT: Magnetic resonance imaging (MRI) and postmortem neuropathological studies have implicated the cerebellum in the pathophysiology of autism. Controversy remains, however, concerning the nature and the consistency of cerebellar alterations. MRI studies of the cross-sectional area of the vermis have found both decreases and no difference in autism groups. Volumetric analysis of the vermis, which is less prone to "plane of section artifacts" may provide a more reliable assessment of size differences but few such studies exist in the literature. Here we present the results of a volumetric analysis of the structure of the whole cerebellum and its components in children and adolescents with autism spectrum disorders. Structural MRI's were acquired from 62 male participants (7.5 to 18.5 years-old) who met criteria for the following age-matched diagnostic groups: low functioning autism, high functioning autism (HFA), Asperger syndrome, and typically developing children. When compared to controls, the midsagittal area of the vermis, or of subgroups of lobules, was not reduced in any of the autism groups. However, we did find that total vermis volume was decreased in the combined autism group. When examined separately, the vermis of only the HFA group was significantly reduced compared to typically developing controls. Neither IQ nor age predicted the size of the vermis within the autism groups. There were no differences in the volume of individual vermal lobules or cerebellar hemispheres. These findings are discussed in relation to the pathology of autism and to the fairly common alterations of vermal morphology in various neurodevelopmental disorders.
Autism Research 11/2009; 2(5):246-57. · 3.99 Impact Factor
[show abstract][hide abstract] ABSTRACT: Sleep problems are a common complaint of parents of preschool children. Children with neurodevelopmental disorders have even more disrupted sleep than typically developing children. Although disrupted nighttime sleep has been reported to affect daytime behavior, the pathway from sleep disruption to sleep problems, to impairments in daytime performance or behavior is not clear. This multi-method, preliminary study assessed this path in 68 children with autism, matched to 57 children with developmental delay without autism and 69 children developing typically.
Actigraphy, structured questionnaires, laboratory assessments, and parent reports were obtained in 194 children.
Controlling for diagnosis and developmental age of the child, nighttime sleep problems determined by parent reports were significantly associated with decrements in daytime behavior, also measured by parent report instruments. However, actigraph-defined sleep problems and objective measures of daytime sleepiness were not associated with decrements in daytime performance.
Parent report measures substantiate relationships between disrupted sleep patterns and waking behavior. Further understanding of the pathway from sleep disorders to daytime sleepiness and decrements in waking performance, however, may require more rigorous methods of assessment such as polysomnography and the multiple sleep latency test.
Journal of Child Psychology and Psychiatry 08/2009; 50(12):1532-40. · 5.42 Impact Factor
[show abstract][hide abstract] ABSTRACT: This study examined the persistence of sleep problems in preschool children with autism and two matched comparison groups: children with developmental delay without autism and typically developing children. Sleep problems were defined subjectively by parent report, by the Children's Sleep Habits Questionnaire (CSHQ), and objectively by quantitative Research Diagnostic Criteria (RDC) derived from actigraphic recordings.
Children were studied on three occasions, each separated by a 3-month interval. At each assessment, the children were recorded actigraphically for 1 week, and parents completed sleep-wake diaries and the CSHQ. Descriptive statistics and odds ratios were used to assess the occurrence and stability of sleep problems within children and across groups and to explore how actigraph- and CSHQ-defined sleep problems affect parental sleep problem reports.
Parent reports of a generic sleep problem were more prevalent than RDC- and CSHQ-defined sleep problems, especially for children with neurodevelopmental disorders. For all groups, objectively measured sleep problems were rarely persistent during the 6-month period. The children in both neurodevelopmental groups, however, had more sleep problems on one or two occasions, using actigraph and the CSHQ, than typically developing children.
Objective and subjective measures of sleep problems in preschool-aged children produce different results. In a community sample, the rate of actigraph- and CSHQ-defined sleep problems in children with autism did not differ from rates for typically developing children, although the parent report of a generic sleep problem was significantly greater.
Journal of the American Academy of Child and Adolescent Psychiatry 07/2009; 48(8):847-54. · 4.98 Impact Factor
[show abstract][hide abstract] ABSTRACT: To determine the efficacy of melatonin on sleep problems in children with autistic spectrum disorder (ASD) and fragile X syndrome (FXS).
A 4-week, randomized, double blind, placebo-controlled, crossover design was conducted following a 1-week baseline period. Either melatonin, 3 mg, or placebo was given to participants for 2 weeks and then alternated for another 2 weeks. Sleep variables, including sleep duration, sleep-onset time, sleep-onset latency time, and the number of night awakenings, were recorded using an Actiwatch and from sleep diaries completed by parents. All participants had been thoroughly assessed for ASD and also had DNA testing for the diagnosis of FXS.
Data were successfully obtained from the 12 of 18 subjects who completed the study (11 males, age range 2 to 15.25 years, mean 5.47, SD 3.6). Five participants met diagnostic criteria for ASD, 3 for FXS alone, 3 for FXS and ASD, and 1 for fragile X premutation. Eight out of 12 had melatonin first. The conclusions from a nonparametric repeated-measures technique indicate that mean night sleep duration was longer on melatonin than placebo by 21 minutes (p = .02), mean sleep-onset latency was shorter by 28 minutes (p = .0001), and mean sleep-onset time was earlier by 42 minutes (p = .02).
The results of this study support the efficacy and tolerability of melatonin treatment for sleep problems in children with ASD and FXS.
Journal of clinical sleep medicine: JCSM: official publication of the American Academy of Sleep Medicine 04/2009; 5(2):145-50. · 2.93 Impact Factor
[show abstract][hide abstract] ABSTRACT: This study investigated the association between preschool children's sleep patterns measured by actigraphy and parent-reported hyperactivity symptoms. Many previous studies have reported sleep problems in children with attention deficit hyperactivity disorder (ADHD)-like symptoms.
This study examined a cross-sectional sample of 186 preschoolers age 2-5 years in three groups: children with autism, children with developmental delay without autism, and typically developing children recruited from the general population. One week of actigraphic sleep data plus a parent report of the presence or absence of a current sleep problem were collected. Parents completed the child behavior checklist; a subset of children in preschool had teachers who completed the caregiver-teacher report form. Sleep behavior was compared for those children with and without clinical levels of attention-deficit/hyperactivity symptoms (T scores > or = 65).
The prevalence of a parent-defined sleep problem across the entire sample was 36.1%. Thirty-four percent of the sample had a parent-reported ADHD composite in the clinical range. Those children with a clinical ADHD profile were more likely to be described by parents as having a sleep problem. However, no significant differences in actigraphic sleep patterns or night-to-night sleep-wake variability were found for children with an ADHD profile in the clinical range.
In this non-clinical sample of preschool age children, parental reports of clinical ADHD profiles were significantly associated with parental reports of sleep problems but not with actigraphically recorded sleep-wake data.
Child Psychiatry and Human Development 01/2009; 40(2):257-68. · 1.93 Impact Factor
[show abstract][hide abstract] ABSTRACT: Autism, which is common in individuals with fragile X syndrome, is often difficult to diagnose. We compared the diagnostic classifications of two measures for autism diagnosis, the ADOS and the ADI-R, in addition to the DSM-IV-TR in 63 males with this syndrome. Overall, 30% of the subjects met criteria for autistic disorder and 30% met criteria for PDD-NOS. The classifications on the ADOS and DSM-IV-TR were most similar, whereas the ADI-R classified subjects as autistic much more frequently. We further investigated the relationship of both FMRP and FMRI mRNA to symptoms of autism in this cohort and found no significant relationship between the measures of autism and molecular features, including FMRP, FMRI mRNA, and CGG repeat number.
American journal of mental retardation: AJMR 12/2008; 113(6):427-38. · 2.51 Impact Factor
[show abstract][hide abstract] ABSTRACT: A prominent noncore symptom of autistic disorder is disturbed sleep, but relatively few studies have investigated this symptom.
A multimethod approach assessed the quantity and quality of sleep in 194 children (68 with autism [AUT], 57 with developmental delay without autism [DD], 69 with typical development) recorded over 1 week. Parent perceptions, structured questionnaires, and actigraphy were compared. In addition, problem sleep as defined by parents was compared with research diagnostic criteria for behavioral insomnia obtained from actigraph recordings.
On actigraphy, children in the DD group, after sleep onset, exhibited more and longer awakenings than the other two groups. In contrast, children in the AUT group exhibited less total sleep time in 24 hours than the other two groups. Parent reports of sleep problems were higher in the AUT and DD groups than the typical development group, but parent reports did not concur with more objective RDC for behavioral insomnia. Parent reports of sleep problems in all of the groups were significantly associated with increased self-reports of stress. Total 24-hour sleep durations for all of the groups were shorter than recommended for preschool-age children.
Our study provides objective evidence that sleep patterns are different in preschool children across the categories of AUT, DD, or typical development.
Journal of the American Academy of Child and Adolescent Psychiatry 07/2008; 47(8):930-8. · 4.98 Impact Factor
[show abstract][hide abstract] ABSTRACT: This study compared parent-reported sleep characteristics in 2- to 5-year-old children with autism spectrum disorders (ASD) to children with other developmental delays (DD) and typical development (TD). We included 529 children (303 ASD [167 males], 63 DD [46 males], and 163 TD [134 males]) enrolled in the CHARGE study, an ongoing population-based case-control study. The mean age of participants was 3.6 years (standard deviation, 0.8 years). ASD diagnosis was confirmed with Autism Diagnostic Interview-Revised (ADI-R) and Autism Diagnostic Observation Schedules (ADOS). Cognitive and adaptive functioning was assessed using Mullen Scales of Early Learning (MSEL) and Vineland Adaptive Behavior Scales (VABS), respectively. Demographic, medical and sleep history information were ascertained from California birth records, telephone interview, medical assessments at clinic visit, and parent-administered questionnaires. Fifty-three percent of children with ASD had at least one frequent sleep problem, followed by 46% of children with DD, and 32% of the TD group (P < 0.0001). Exploratory factor analyses of sleep history data yielded two factors: sleep onset problems and night waking. Children with ASD had marginally higher sleep onset factor scores and significantly higher night waking factor scores compared with the TD group. Factor scores for children with DD were intermediate between the ASD and TD groups. Cognitive or adaptive development did not predict severity of sleep problems in the ASD group.
Journal of Sleep Research 06/2008; 17(2):197-206. · 3.04 Impact Factor
[show abstract][hide abstract] ABSTRACT: Twenty to 40% of young children are reported to have behavioral insomnias of childhood. Concerns about sleep at these ages are the most common problem expressed to pediatricians at the time of well child visits. A screening questionnaire, the Children's Sleep Habits Questionnaire (CSHQ), has been used in clinical settings and in research studies to assess children ages 4 to 10 for the presence of sleep problems. A CSHQ total score has distinguished clinical populations from community samples.
The current study assesses the CSHQ in a younger age group than previously reported and in a diverse population. A total of 194 children, ages 2 to 51/2 years, were recruited into 3 diagnostic groups: 68 children with autism, 57 children with developmental delay without autism, and 69 typically developing children. All children's parents completed the CSHQ and a sleep log, and all children were studied for 7 days and nights with actigraphy. The children were divided into problem sleep and non-problem sleep groups on the basis of a parent report of a generic sleep problem at the time of entry into the study. The CSHQ responses for the problem and non-problem sleep groups were then compared.
The results suggest that the CSHQ is clinically useful for screening of sleep problems in typically developing children at these young ages as well as in children with diverse neurodevelopmental diagnoses.
The somewhat higher subscale scores than previously reported for older children appear to be consistent with more sleep problems in younger children.
[show abstract][hide abstract] ABSTRACT: We report the results of a pilot trial of an evidence-based treatment-Parent-Child Interaction Therapy (PCIT; Eyberg et al. Psychopharmacology Bulletin, 31(1), 83-91, 1995) for boys aged 5-12 with high functioning autism spectrum disorders and clinically significant behavioral problems. The study also included an investigation of the role of shared positive affect during the course of therapy on child and parent outcomes. The intervention group showed reductions in parent perceptions of child problem behaviors and child atypicality, as well as an increase in child adaptability. Shared positive affect in parent child dyads and parent positive affect increased between the initial and final phases of the therapy. Parent positive affect after the first phase was related to perceptions of improvement in problem behaviors and adaptive functioning.
Journal of Autism and Developmental Disorders 05/2008; 38(9):1767-76. · 3.34 Impact Factor
[show abstract][hide abstract] ABSTRACT: This study compared actigraphy with videosomnography in preschool-aged children, with special emphasis on the accuracy of detection of nighttime awakenings.
Fifty-eight participants wore an actigraph for 1 week and were videotaped for 2 nights while wearing the actigraph.
Participants were solitary sleepers, studied in their homes.
One group (n = 22) was diagnosed with autism, another group (n = 11) had developmental delays without autism, and a third group (n = 25) were typically developing children; age ranged from 28 to 73 months (mean age 47 months); 29 boys and 29 girls.
Nocturnal sleep and wakefulness were scored from simultaneously recorded videosomnography and actigraphy. The accuracy of actigraphy was examined in an epoch-by-epoch comparison with videosomnography. Findings were 94% overall agreement, 97% sensitivity, and 24% specificity. Statistical corrections for overall agreement and specificity resulted in an 89% weighted-agreement and 27% adjusted specificity.
Actigraphy has poor agreement for detecting nocturnal awakenings, compared with video observations, in preschool-aged children.
[show abstract][hide abstract] ABSTRACT: The Prader-Willi phenotype (PWP) of fragile X syndrome (FXS) is associated with obesity and hyperphagia similar to Prader-Willi syndrome (PWS), but without cytogenetic or methylation abnormalities at 15q11-13. Thirteen cases of PWP and FXS are reported here that were identified by obesity and hyperphagia. Delayed puberty was seen in 5 of 9 cases who had entered puberty, a small penis or testicles in seven of 13 cases, and infant hypotonia and/or a poor suck in seven of 13 cases. Autism spectrum disorder occurred in 10 of 13 cases, and autism was diagnosed in seven of 13 cases. We investigated cytoplasmic interacting FMR1 protein (CYFIP) expression, which is a protein that interacts with FMR1 protein (FMRP) because the gene for CYFIP is located at 15q11-13. CYFIP mRNA levels were significantly reduced in our patients with the PWP and FXS compared to individuals without FXS (p < .001) and also individuals with FXS without PWP (p = .03).
[show abstract][hide abstract] ABSTRACT: Historically, there have been numerous proposals that the size of the brain correlates with its capacity to process information. Little is known, however, about which specific brain regions contribute to this correlation in children and adolescents. This study evaluated the relationship between intelligence and the size of various brain structures in typically developing male children 8-18 yrs of age. Magnetic resonance imaging (MRI) scans were used to measure the volume of the cerebrum, cerebral gray and white matter, cerebellum, amygdala, and hippocampus. Gray matter and hippocampal volume significantly correlated with full scale and verbal IQ. Since the hippocampus strongly correlated with verbal but not performance IQ, our findings reinforce the hypothesis that the hippocampus is involved in declarative and semantic learning, which contributes more notably to verbal IQ, than to performance IQ. Given the substantial evidence for environmentally induced changes in hippocampal structure, an unresolved issue is whether this relationship reflects genetically determined individual variation or learning induced plasticity.
[show abstract][hide abstract] ABSTRACT: This longitudinal, year-long study compared sleep–wake state organization in two groups of infants–infants whose mothers abused substances during their pregnancies and nonexposed, typically developing, age-matched comparison infants–to determine whether differences in sleep–wake state organization existed between the two groups. Seventeen infants of mothers who were participating in a parent–infant residential treatment program for substance abuse were enrolled. Their sleep–wake state organization over the first year of life was compared to that of 17 age-matched comparison infants. The intent was to follow each infant on five occasions over the first year of life using established methods of time-lapse videosomnography to record sleep–wake state organization; however, attrition in the substance-abusing group was problematic. Some sleep–wake variables (i.e., Active Sleep%, Quiet Sleep%, Awake%, number of nighttime awakenings) were similar for both groups of infants at comparable ages across the first year. Total sleep time and the longest sustained sleep period (sleep continuity variables) differed significantly at some of the ages measured. Although overall sleep architecture appears highly resilient and well organized, some indications of sleep fragmentation and shortened nighttime sleep periods were observed in the substance-exposed infants. More research is needed to explain why sleep-continuity variables and not sleep-state proportion variables differed between the two groups.
[show abstract][hide abstract] ABSTRACT: The authors describe and quantify the neuropsychiatric symptoms present in a cohort of males with the fragile X mental retardation 1 (FMR1) premutation allele who have developed fragile X-associated tremor/ataxia syndrome (FXTAS).
Fourteen male carriers of the FMR1 premutation who had clinical manifestations of the FXTAS syndrome and 14 age- and education-matched controls were assessed with the Neuropsychiatric Inventory (NPI), formal cognitive testing, and genetic analysis.
Males with FXTAS had significantly higher total NPI scores (p < .004) and significantly higher scores on the agitation/aggression (p < .004), depression (p < .004), apathy (p < .004), disinhibition (p < .004), and irritability (p < .004) scales, compared with controls. Cognitive performances on the Mini-Mental State Examination did not correlate with severity of symptoms on the NPI.
The neuropsychiatric manifestations of FXTAS, based on this preliminary report, appear to cluster as a fronto-subcortical dementia. Clinicians encountering patients with clinical dementia with motor symptoms suggesting FXTAS should consider genetic testing to determine whether the patient's dementia syndrome is secondary to a fragile X premutation carrier status.
The Journal of Clinical Psychiatry 02/2006; 67(1):87-94. · 5.81 Impact Factor
[show abstract][hide abstract] ABSTRACT: This article reviews evidence-based criteria that can guide practitioners in the selection, use, and interpretation of assessment tools for autism spectrum disorders (ASD). As Mash and Hunsley (2005) discuss in this special section, evidence-based assessment tools not only demonstrate adequate psychometric qualities, but also have relevance to the delivery of services to individuals with the disorder (see also Hayes, Nelson, & Jarrett, 1987). Thus, we use what is known about the symptoms, etiologies, developmental course, and outcome of ASD to evaluate the utility of particular assessment strategies and instruments for diagnosis, treatment planning and monitoring, and evaluation of outcome. The article begins with a review of relevant research on ASD. Next we provide an overview of the assessment process and some important issues that must be considered. We then describe the components of a core (minimum) assessment battery, followed by additional domains that might be considered in a more comprehensive assessment. Domains covered include core autism symptomatology, intelligence, language, adaptive behavior, neuropsychological functions, comorbid psychiatric illnesses, and contextual factors (e.g., parent well-being, family functioning, quality of life). We end with a discussion of how well the extant literature meets criteria for evidence-based assessments.