Modified Children's sleep habits questionnaire for behavioral sleep problems: A validation study.

OBJECTIVES: Behavioral sleep problems (BSPs) are prevalent and consequential in young children. There is a need for screening tools that identify BSPs-which are often rooted in the parent-young child relationship-and typically respond to behavior management. Such a tool would increase capacity to identify and treat BSPs. We sought to validate a short-form version of the widely used Children's Sleep Habits Questionnaire (SF-CSHQ) that omitted items that would not be responsive to behavioral strategies. METHODS: The original 33-item CSHQ elicits parent report of "behaviorally-based" and "medically-based" sleep items (eg, parasomnias and sleep disordered breathing). We conducted analyses to develop a SF-CSHQ that excludes its "medically-based" items, to determine (a) the SF-CSHQ threshold score corresponding to the full CSHQ clinical cut-off score (≥41), and (b) preliminary validity of this SF-CSHQ. Data were re-analyzed from the original data that established the CSHQ's psychometric properties in 4-10 year olds, and a second dataset that established its validity in 24-66 month olds. RESULTS: In both datasets, a threshold score of 30 had correlations of 0.90-0.94 with the original cut-off. This 23-item SF-CSHQ cut-off functioned as well as the full CSHQ cut-off in discriminating between children with vs without a parent-reported behavioral sleep problem, and with vs without prolonged sleep latency (per actigraphy). CONCLUSION: We established preliminary validity of modified version of the widely-used CSHQ. This SF-CSHQ may be useful for widening screening and first-line guidance for behavioral sleep problems in young children, among professionals who are not sleep medicine specialists.

Methods for acquiring MRI data in children with autism spectrum disorder and intellectual impairment without the use of sedation.

BACKGROUND: Magnetic resonance imaging (MRI) has been widely used in studies evaluating the neuropathology of autism spectrum disorder (ASD). Studies are often limited, however, to higher functioning individuals with ASD. MRI studies of individuals with ASD and comorbid intellectual disability (ID) are lacking, due in part to the challenges of acquiring images without the use of sedation. METHODS: Utilizing principles of applied behavior analysis (ABA), we developed a protocol for acquiring structural MRI scans in school-aged children with ASD and intellectual impairment. Board certified behavior analysts worked closely with each child and their parent(s), utilizing behavior change techniques such as pairing, shaping, desensitization, and positive reinforcement, through a series of mock scanner visits to prepare the child for the MRI scan. An objective, quantitative assessment of motion artifact in T1- and diffusion-weighted scans was implemented to ensure that high-quality images were acquired. RESULTS: The sample consisted of 17 children with ASD who are participants in the UC Davis Autism Phenome Project, a longitudinal MRI study aimed at evaluating brain developmental trajectories from early to middle childhood. At the time of their initial scan (2-3.5 years), all 17 children had a diagnosis of ASD and development quotient (DQ) <70. At the time of the current scan (9-13 years), 13 participants continued to have IQs in the range of ID (mean IQ = 54.1, sd = 12.1), and four participants had IQs in the normal range (mean = 102.2, sd = 7.5). The success rate in acquiring T1-weighted images that met quality assurance for acceptable motion artifact was 100 %. The success rate for acquiring high-quality diffusion-weighted images was 94 %. CONCLUSIONS: By using principles of ABA in a research MRI setting, it is feasible to acquire high-quality images in school-aged children with ASD and intellectual impairment without the use of sedation. This is especially critical to ensure that ongoing longitudinal studies of brain development can extend from infancy and early childhood into middle childhood in children with ASD at all levels of functioning, including those with comorbid ID.

Social impairments in chromosome 22q11.2 deletion syndrome (22q11.2DS): autism spectrum disorder or a different endophenotype?

High prevalence of autism spectrum disorders (ASD) has been reported in 22q11.2DS, although this has been based solely on parent report measures. This study describes the presence of ASD using a procedure more similar to that used in clinical practice by incorporating history (Social Communication Questionnaire) AND a standardized observation measure (Autism Diagnostic Observation Schedule) and suggests that ASD is not as common as previously reported in 22q11.2DS. Differences in methodology, along with comorbid conditions such as anxiety, likely contribute to false elevations in ASD prevalence and information from multiple sources should be included in the evaluation of ASD.

Sleep and daytime functioning: a short-term longitudinal study of three preschool-age comparison groups.

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.

Six-month sleep-wake organization and stability in preschool-age children with autism, developmental delay, and typical development.

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.

Daytime sleep patterns in preschool children with autism, developmental delay, and typical development.

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.

Causes and correlates of frequent night awakenings in early childhood.

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.

Inconsolable night-time awakening: beyond night terrors.

CASE: Sophia is a 3-year-old girl who was brought to her pediatrician by her parents who were concerned about inconsolable night-time awakening. Her mother indicated that she has frequent (>6), early nocturnal awakenings accompanied by screaming and crying lasting up to 1 hour since her birth. These episodes increased in intensity and frequency in the past year since the birth of her brother. With a bedtime routine (a cup of water by bedside with a washcloth and touching mother's nose, chin, and cheeks), Sophia falls asleep easily; however, within 1 hour she awakes screaming and flailing unaware of her surroundings and unable to be comforted. There are no tonic-clonic movements. Prior interventions, including a sleep coach and "letting Sophia cry it out," did not change her sleep pattern. Sophia's mother reports that she needs to be on a specific daily routine including set times for awakening, activity, snacks, naps, and meals. Diversion from the routine and separation from her mother results in a tantrum (kicking, hitting, screaming, and inconsolability) often lasting more than 30 minutes. Sophia was born after an uncomplicated 37-week gestation. Neonatal hyperbilirubinemia required readmission for 24 hours of phototherapy; serum bilirubin levels were performed daily for 3 weeks after discharge. At 6 weeks, daily episodes of screaming, inconsolability, forceful vomiting, and inability to sleep led to a diagnosis of gastroesophageal reflux. Medication trials were not successful, but the symptoms resolved by 5 months. Formula intolerance and difficulty swallowing and chewing different textures of solid food occurred in the first year. Occupational therapy was of "no benefit"; Sophia was overwhelmed by the activity and took a long time to warm up to the therapist. Her texture aversion resolved by 2 years of age. She prefers one-on-one play and has minimal interactions with other children. She has met all her developmental milestones appropriately and has no other health issues. Sophia lives with her parents and infant brother. There is a maternal family history of insomnia and sleep walking and a paternal history of sleeping walking. Her mother adheres to a strict daily schedule. Sleep deprivation, differences parent child-rearing practices, social isolation, and lack of quality parent time were all identified by the mother as significant marital stressors. During the office visit, Sophia required 30 minutes to warm up and smile, and over 60 minutes before she spoke her first word. Physical examination was normal (including growth measurements) and the developmental examination was age-appropriate. Upon completion of the assessment, she was engaging, playful, and cooperative with the pediatrician.

The efficacy of melatonin for sleep problems in children with autism, fragile X syndrome, or autism and fragile X syndrome.

STUDY OBJECTIVE: To determine the efficacy of melatonin on sleep problems in children with autistic spectrum disorder (ASD) and fragile X syndrome (FXS). METHODS: 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. RESULTS: 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). CONCLUSION: The results of this study support the efficacy and tolerability of melatonin treatment for sleep problems in children with ASD and FXS.

A comprehensive volumetric analysis of the cerebellum in children and adolescents with autism spectrum disorder.

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.

Six-month persistence of sleep problems in young children with autism, developmental delay, and typical development.

OBJECTIVE: 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. METHOD: 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. RESULTS: 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. CONCLUSIONS: 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.

Sleep problems, sleepiness and daytime behavior in preschool-age children.

BACKGROUND: 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. METHODS: Actigraphy, structured questionnaires, laboratory assessments, and parent reports were obtained in 194 children. RESULTS: 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. CONCLUSIONS: 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.

Objective sleep measurement in typically and atypically developing preschool children with ADHD-like profiles.

OBJECTIVE: 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. METHODS: 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). RESULTS: 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. CONCLUSIONS: 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.

Inconsolable night-time awakening: beyond night terrors.

Sophia is a 3-year-old girl who was brought to her pediatrician by her parents who were concerned about inconsolable night-time awakening. Her mother indicated that she has frequent (>6), early nocturnal awakenings accompanied by screaming and crying lasting up to 1 hour since her birth. These episodes increased in intensity and frequency in the past year since the birth of her brother. With a bedtime routine (a cup of water by bedside with a washcloth and touching mother's nose, chin, and cheeks), Sophia falls asleep easily; however, within 1 hour she awakes screaming and flailing unaware of her surroundings and unable to be comforted. There are no tonic-clonic movements. Prior interventions, including a sleep coach and "letting Sophia cry it out," did not change her sleep pattern. Sophia's mother reports that she needs to be on a specific daily routine including set times for awakening, activity, snacks, naps, and meals. Diversion from the routine and separation from her mother results in a tantrum (kicking, hitting, screaming, and inconsolability) often lasting more than 30 minutes. Sophia was born after an uncomplicated 37-week gestation. Neonatal hyperbilirubinemia required readmission for 24 hours of phototherapy; serum bilirubin levels were performed daily for 3 weeks after discharge. At 6 weeks, daily episodes of screaming, inconsolability, forceful vomiting, and inability to sleep led to a diagnosis of gastroesophageal reflux. Medication trials were not successful, but the symptoms resolved by 5 months. Formula intolerance and difficulty swallowing and chewing different textures of solid food occurred in the first year. Occupational therapy was of "no benefit"; Sophia was overwhelmed by the activity and took a long time to warm up to the therapist. Her texture aversion resolved by 2 years of age. She prefers one-on-one play and has minimal interactions with other children. She has met all her developmental milestones appropriately and has no other health issues. Sophia lives with her parents and infant brother. There is a maternal family history of insomnia and sleep walking and a paternal history of sleep walking. Her mother adheres to a strict daily schedule. Sleep deprivation, different parental child-rearing practices, social isolation, and lack of quality parent time were all identified by the mother as significant marital stressors. During the office visit, Sophia required 30 minutes to warm up and smile, and over 60 minutes before she spoke her first word. Physical examination was normal (including growth measurements) and the developmental examination was age-appropriate. Upon completion of the assessment, she was engaging, playful, and cooperative with the pediatrician.

Sleep patterns in preschool-age children with autism, developmental delay, and typical development.

OBJECTIVE: A prominent noncore symptom of autistic disorder is disturbed sleep, but relatively few studies have investigated this symptom. METHOD: 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. RESULTS: 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. CONCLUSIONS: Our study provides objective evidence that sleep patterns are different in preschool children across the categories of AUT, DD, or typical development.

The Children's Sleep Habits Questionnaire in toddlers and preschool children.

OBJECTIVE: 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. METHODS: 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. RESULTS: 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. CONCLUSIONS: The somewhat higher subscale scores than previously reported for older children appear to be consistent with more sleep problems in younger children.

Sleep problems in children with autism spectrum disorders, developmental delays, and typical development: a population-based study.

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.

The effectiveness of parent-child interaction therapy for families of children on the autism spectrum.

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.

The use of actigraphy to study sleep disorders in preschoolers: some concerns about detection of nighttime awakenings.

STUDY OBJECTIVES: This study compared actigraphy with videosomnography in preschool-aged children, with special emphasis on the accuracy of detection of nighttime awakenings. DESIGN: Fifty-eight participants wore an actigraph for 1 week and were videotaped for 2 nights while wearing the actigraph. SETTING: Participants were solitary sleepers, studied in their homes. PARTICIPANTS: 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. INTERVENTIONS: N/A. MEASUREMENTS AND RESULTS: 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. CONCLUSIONS: Actigraphy has poor agreement for detecting nocturnal awakenings, compared with video observations, in preschool-aged children.

Autism profiles of males with fragile X syndrome.

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.