Corticosteroid therapy in regressive autism: a retrospective study of effects on the Frequency Modulated Auditory Evoked Response (FMAER), language, and behavior

• Type of Material: Article
• Authors: Frank H Duffy, Boston Children’s Hospital; Adit Shankardass, Harvard University; Gloria B McAnulty, Harvard University; Yaman Zorlu Eksioglu, Emory University; David Coulter, Boston Children’s Hospital; Alexander Rotenberg, Boston Children’s Hospital; Heidelise Als, Harvard University
• Language: English
• Date: 2014-05-15
• Publisher: BMC (part of Springer Nature)
• Publication Version: Final Published Version
• Copyright Statement: © 2014 Duffy et al.; licensee BioMed Central Ltd.
• License: Creative Commons Attribution 4.0 International
• Final Published Version (URL): http://dx.doi.org/10.1186/1471-2377-14-70
• Title of Journal or Parent Work: BMC Neurology
• ISSN: 1471-2377
• Volume: 14
• Issue: 1
• Start Page: 70
• End Page: 70
• Grant/Funding Information: This work was supported in part by National Institutes of Health/National Institute of Child Health and Human Development grant R01HD047730 to Heidelise Als, PhD, the Harris Foundation (to Dr Als), the Buehler Family Fund (to DrAls), and the Intellectual and Developmental Disabilities Research Center (IDDRC) grant P30HD018655 to Scott Pomeroy, MD, PhD, Boston Children’s Hospital and Harvard Medical School. The authors furthermore acknowledge support from the Tommy Fuss Fund to Joseph Gonzalez-Heydrich, MD, and thank the Chairmen of Neurology (Dr. Pomeroy) and Psychiatry (David R. DeMaso, MD) for their continuing support of these research efforts.; The funding bodies indicated above had no role in study design; in the collection, analysis, and interpretation of data; in the writing of the manuscript; and in the decision to submit the manuscript for publication.
• Keywords: EPILEPTIFORM ACTIVITY; Science & Technology; Neurosciences & Neurology; Distortion; Treatment; Frequency modulated auditory evoked response; INFANTILE SPASMS; ANOMALIES; Spectral; APHASIA; Autism; Electroencephalogram; Superior temporal gyrus; Steroids; LANDAU-KLEFFNER-SYNDROME; SPECTRUM DISORDERS; CHILDREN; Regression; Clinical Neurology; EEG; STG; EP; DEVELOPMENTAL-DISABILITIES; SLEEP; Evoked potential; Behavior; Life Sciences & Biomedicine; FMAER; CONTINUOUS SPIKE; Language; Corticosteroids
• Research Categories: Psychology, Behavioral; Psychology, General

Abstract

Background: Up to a third of children with Autism Spectrum Disorder (ASD) manifest regressive autism (R-ASD).They show normal early development followed by loss of language and social skills. Absent evidence-based therapies, anecdotal evidence suggests improvement following use of corticosteroids. This study examined the effects of corticosteroids for R-ASD children upon the 4 Hz frequency modulated evoked response (FMAER) arising from language cortex of the superior temporal gyrus (STG) and upon EEG background activity, language, and behavior. An untreated clinical convenience sample of ASD children served as control sample.Methods: Twenty steroid-treated R-ASD (STAR) and 24 not-treated ASD patients (NSA), aged 3 - 5 years, were retrospectively identified from a large database. All study participants had two sequential FMAER and EEG studies;Landau-Kleffner syndrome diagnosis was excluded. All subjects' records contained clinical receptive and expressive language ratings based upon a priori developed metrics. The STAR group additionally was scored behaviorally regarding symptom severity as based on the Diagnostic and Statistical Manual IV (DSM-IV) ASD criteria list. EEGs were visually scored for abnormalities. FMAER responses were assessed quantitatively by spectral analysis. Treated and untreated group means and standard deviations for the FMAER, EEG, language, and behavior, were compared by paired t-test and Fisher's exact tests.Results: The STAR group showed a significant increase in the 4 Hz FMAER spectral response and a significant reduction in response distortion compared to the NSA group. Star group subjects' language ratings were significantly improved and more STAR than NSA group subjects showed significant language improvement. Most STAR group children showed significant behavioral improvement after treatment. STAR group language and behavior improvement was retained one year after treatment. Groups did not differ in terms of minor EEG abnormalities. Steroid treatment produced no lasting morbidity.Conclusions: Steroid treatment was associated with a significantly increased FMAER response magnitude, reduction of FMAER response distortion, and improvement in language and behavior scores. This was not observed in the non-treated group. These pilot findings warrant a prospective randomized validation trial of steroid treatment for R-ASD utilizing FMAER, EEG, and standardized ASD, language and behavior measures, and a longer follow-up period.Please see related article http://www.biomedcentral.com/1741-7015/12/79.