ECHO autism adult healthcare: Training community clinicians to provide quality care for autistic adults.

Autistic adults experience significant unmet healthcare needs, with opportunities for improvement in both the systems and the practitioners who serve this population. Primary care physicians/practitioners (PCPs) are a natural choice to provide comprehensive care to autistic adults but often lack experience in serving this population. This pilot study developed and tested an Extension for Community Healthcare Outcomes (ECHO) Autism model adapted from our previous work, focused specifically on training PCPs in best-practice care for autistic adults. The project was informed directly by the perspectives and preferences of autistic adults, caregivers, and PCPs. Two consecutive cohorts of PCPs participated in ECHO Autism Adult Healthcare sessions. Each cohort met 1 h twice a month for 6 months, with 37 PCPs (n = 20 in Cohort 1, and n = 17 in Cohort 2) participating. Based on findings from the first cohort, adjustments were made to refine the session preparation, curriculum, conduct of the ECHO, resources, and evaluation. After participation in the ECHO Autism program, PCP self-efficacy and satisfaction improved, while the number of perceived barriers did not change. Knowledge did not improve significantly in Cohort 1, but after adjustments to the training model, participants in Cohort 2 showed significant knowledge gains. While attention to systems of care is critical to addressing barriers in healthcare in the autistic population, the ECHO Autism Adult Healthcare model is feasible and holds promise for improving PCP satisfaction and self-efficacy in working with autistic adults.

Health conditions in autism: Defining the trajectory from adolescence to early adulthood.

Autistic adults, as compared to non-autistic adults, have increased rates of nearly all medical and psychiatric conditions. Many of these conditions begin in childhood, although few longitudinal studies have been conducted to examine prevalence rates of these conditions from adolescence into early adulthood. In this study, we analyze the longitudinal trajectory of health conditions in autistic youth, compared to age and sex-matched non-autistic youth, transitioning from adolescence into early adulthood in a large integrated health care delivery system. The percent and modeled prevalence of common medical and psychiatric conditions increased from age 14 to 22 years, with autistic youth having a higher prevalence of most conditions than non-autistic youth. The most prevalent conditions in autistic youth at all ages were obesity, neurological disorders, anxiety, and ADHD. The prevalence of obesity and dyslipidemia rose at a faster rate in autistic youth compared to non-autistic youth. By age 22, autistic females showed a higher prevalence of all medical and psychiatric conditions compared to autistic males. Our findings emphasize the importance of screening for medical and psychiatric conditions in autistic youth, coupled with health education targeted at this population, to mitigate the development of adverse health outcomes in autistic adults.

Randomized, Placebo-Controlled Trial of Ferrous Sulfate to Treat Insomnia in Children With Autism Spectrum Disorders.

BACKGROUND: Insomnia and low iron stores are common in children with autism spectrum disorders, and low iron stores have been associated with sleep disturbance. METHODS: We performed a randomized placebo-controlled trial of oral ferrous sulfate to treat insomnia in children with autism spectrum disorders and low normal ferritin levels. Twenty participants who met inclusion criteria and whose insomnia did not respond to sleep education were randomized to 3 mg/kg/day of ferrous sulfate (n = 9) or placebo (n = 11) for three months. RESULTS: Iron supplementation was well tolerated, and no serious adverse events were reported. Iron supplementation improved iron status (+18.4 ng/mL active versus -1.6 ng/mL placebo, P = 0.044) but did not significantly improve the primary outcome measures of sleep onset latency (-11.0 minutes versus placebo, 95% confidence interval -28.4 to 6.4 minutes, P = 0.22) and wake time after sleep onset (-7.7 minutes versus placebo, 95% confidence interval -22.1 to 6.6 min, P = 0.29) as measured by actigraphy. Iron supplementation was associated with improvement in the overall severity score from the Sleep Clinical Global Impression Scale (-1.5 points versus placebo, P = 0.047). Changes in measures of daytime behavior did not differ between groups. CONCLUSION: This trial demonstrated no improvement in primary outcome measures of insomnia in subjects treated with ferrous sulfate compared with placebo. Interpretation was limited by low enrollment.

The Pediatric Sleep Clinical Global Impressions Scale-A New Tool to Measure Pediatric Insomnia in Autism Spectrum Disorders.

OBJECTIVE: To pilot a clinician-based outcome measure that provides complementary information to objective measures and parent-based questionnaires for insomnia in children with autism spectrum disorders (ASD). METHOD: The authors developed a Pediatric Sleep Clinical Global Impressions Scale (CGI). Questions included (1) the child's ability to fall asleep and remain sleeping independently (i.e., apart from parents); (2) bedtime resistance; (3) sleep onset delay; (4) night awakening; (5) parental satisfaction with their child's current sleep patterns; (6) family functioning as affected by their child's current sleep patterns; and (7) clinician's overall concern with the child's sleep. After refining the instrument through the evaluation of vignettes by ASD and sleep experts, the authors piloted the Pediatric Sleep CGI in a 12-week randomized trial of iron supplementation in children with ASD. Clinicians completed Pediatric Sleep CGIs and structured sleep histories, parents completed the Children's Sleep Habits Questionnaire (CSHQ), and children wore actigraphy watches. RESULTS: In repeated measures models, the Pediatric Sleep CGI and CSHQ were correlated for sleep onset delay (r = .66, p < .001), night wakings (r = .40, p < .001), and total score (r = .29, p < .001). The CGI-S sleep onset delay and actigraphy sleep onset delay scores (r = .75, p = .0095) were also correlated. The overall CGI-S showed improvement with therapy (p = .047). CONCLUSION: The Pediatric Sleep CGI shows promise in measuring clinician-rated outcomes in pediatric insomnia in children with ASD. Larger samples will be necessary to examine reliability, validity, and measure to change, as well as applicability to other populations with pediatric insomnia.

Melatonin in children with autism spectrum disorders: endogenous and pharmacokinetic profiles in relation to sleep.

Supplemental melatonin has been used to treat sleep onset insomnia in children with autism spectrum disorders (ASD), although the mechanism of action is uncertain. We assessed endogenous and supplemental melatonin profiles in relation to sleep in nine children with ASD. In endogenous samples, maximal melatonin concentration (C(max)) and time to peak concentration (T(max)) were comparable to those previously published in the literature for typically developing children, and dim light melatonin onsets were captured in the majority of children. In treatment samples (supplemental melatonin), melatonin parameters were also comparable to those previously published for typically developing children. Our findings support that children with ASD and insomnia responsive to low dose melatonin treatment have relatively normal profiles of endogenous and supplemental melatonin.

Relation of melatonin to sleep architecture in children with autism.

Children with autism often suffer from sleep disturbances, and compared to age-matched controls, have decreased melatonin levels, as indicated by urine levels of the primary melatonin metabolite, 6-sulfatoxymelatonin (6-SM). We therefore investigated the relationship between 6-SM levels and sleep architecture in children with autism spectrum disorders (ASD). Twenty-three children, aged 4-10 years, completed two nights of polysomnography and one overnight urine collection for measurement of urinary 6-SM excretion rate. Parents completed the Children's Sleep Habits Questionnaire. We found that higher urinary 6-SM excretion rates were associated with increased N3 sleep, decreased N2 sleep, and decreased daytime sleepiness. The results warrant further examination to examine the effects of supplemental melatonin on sleep architecture and daytime sleepiness.

Long-term outcome of nonsurgical candidates with medically refractory localization-related epilepsy.

PURPOSE: Epilepsy surgery can result in complete seizure remission rates of upto 80% in patients with mesial temporal sclerosis and unilateral seizures. The seizure-free rate after surgery for patients with extratemporal nonlesional epilepsy has ranged between 30% and 40%. Some patients with medically refractory localization-related epilepsy cannot be offered surgical resection because of inadequate localization of the epileptogenic zone, documentation of bilateral ictal onsets, or functionally important areas of cortex that prohibit resection. The short-term rate of complete remission with medications in temporal lobe epilepsy is poor. Less is known about remission rates in patients who are not surgical candidates. In this study, we evaluated the outcome of medical treatment in patients with medically refractory partial epilepsy who were evaluated for possible epilepsy surgery but deemed to be inadequate surgical candidates. METHODS: A retrospective chart review and telephone survey with a self-rating questionnaire were completed for all patients who underwent epilepsy surgery evaluation but were not ultimately offered surgical treatment at the University of Michigan from 1990 through 1998. We assessed changes in seizure frequency and type, imaging characteristics, ictal recordings, interim medication history, and subjective changes in quality of life. RESULTS: Thirty-four subjects were available for follow-up study, at an average of >4 years after surgical evaluation. A significant reduction in seizure frequency was noted at the time of follow-up compared with that at the time of surgical evaluation. Of patients, 21% achieved seizure remission and remained seizure free for an average of 2.5 years. Four of the seven seizure-free patients attributed their remission to new antiepileptic drugs (AEDs). On a global self-rating item, 15 of 34, or 44%, felt more or much more satisfied with their lives, and 41% felt their quality of life was stable. CONCLUSIONS: A surprisingly large number of patients we surveyed, with refractory partial epilepsy not eligible for surgical management, reported reduced seizure frequency at follow-up, and 21% were seizure free. Our findings suggest that the long-term prognosis in patients with refractory partial epilepsy who are not surgical candidates may be more positive than might be generally expected.

Effects of vagus nerve stimulation on sleep-related breathing in epilepsy patients.

PURPOSE: To describe the effects of vagus nerve stimulation (VNS) on sleep-related breathing in a sample of 16 epilepsy patients. METHODS: Sixteen adults with medically refractory epilepsy (nine men, seven women, ages 21-58 years) underwent baseline polysomnograms (PSGs). Three months after VNS therapy was initiated, PSGs were repeated. In addition, patient 7 had a study with esophageal pressure monitoring, and patient 1 had a continuous positive airway pressure (CPAP) trial. RESULTS: Baseline PSGs: One of 16 patients had an apnea-hypopnea index (AHI) >5 (6.8). Treatment PSGs: Five of 16 patients had treatment AHIs >5. Respiratory events were more frequent during periods with VNS activation (on-time) than without VNS activation (off-time; p = 0.016). Follow-up studies: Esophageal pressure monitoring in patient 7 showed crescendos in esophageal pressure during VNS activation, supporting an obstructive pattern. The CPAP trial of patient 1 showed that all respiratory events were associated with VNS stimulation at low CPAP levels. They were resolved at higher CPAP levels. CONCLUSIONS: Treatment with VNS affects respiration during sleep and should be used with care, particularly in patients with preexisting obstructive sleep apnea. The AHI after VNS treatment remained <5 in the majority of patients and was only mildly elevated (<12) in five patients. In one patient, CPAP resolved VNS-related respiratory events.