Variations in EEG discharges predict ADHD severity within individual Smith-Lemli-Opitz patients

John M. Schreiber, Diane C. Lanham, William Trescher, Susan E. Sparks, Christopher A. Wassif, Brian S. Caffo, Forbes D. Porter, Elaine Tierney, Andrea L. Gropman, Joshua B. Ewen

Research output: Contribution to journalArticle

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Abstract

Objective: We sought to examine the prevalence of EEG abnormalities in Smith-Lemli-Opitz syndrome (SLOS) as well as the relationship between interictal epileptiform discharges (IEDs) and within-subject variations in attentional symptom severity. Methods: In the context of a clinical trial for SLOS, we performed cross-sectional and repeatedmeasure observational studies of the relationship between EEG findings and cognitive/behavioral factors on 23 children (aged 4-17 years). EEGs were reviewed for clinical abnormalities, including IEDs, by readers blinded to participants' behavioral symptoms. Between-group differences in baseline characteristics of participants with and without IEDs were analyzed. Within-subject analyses examined the association between the presence of IEDs and changes in attentiondeficit/ hyperactivity disorder (ADHD) symptoms. Results: Of 85 EEGs, 43 (51%) were abnormal, predominantly because of IEDs. Only one subject had documented clinical seizures. IEDs clustered in 13 subjects (57%), whereas 9 subjects (39%) had EEGs consistently free of IEDs. While there were no significant group differences in sex, age, intellectual disability, language level, or baseline ADHD symptoms, autistic symptoms tended to be more prevalent in the "IED" group (according to Autism Diagnostic Observation Schedule-2 criteria). Within individuals, the presence of IEDs on a particular EEG predicted, on average, a 27% increase in ADHD symptom severity. Conclusions: Epileptiform discharges are common in SLOS, despite a relatively low prevalence of epilepsy. Fluctuations in the presence of epileptiform discharges within individual children with a developmental disability syndrome may be associated with fluctuations in ADHD symptomatology, even in the absence of clinical seizures.

Original languageEnglish (US)
Pages (from-to)151-159
Number of pages9
JournalNeurology
Volume83
Issue number2
DOIs
StatePublished - Jul 8 2014

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Electroencephalography
Smith-Lemli-Opitz Syndrome
Autistic Disorder
Absence Epilepsy
Behavioral Symptoms
Developmental Disabilities
Sex Characteristics
Intellectual Disability
Observational Studies
Epilepsy
Appointments and Schedules
Seizures
Language
Observation
Clinical Trials

All Science Journal Classification (ASJC) codes

  • Clinical Neurology

Cite this

Schreiber, J. M., Lanham, D. C., Trescher, W., Sparks, S. E., Wassif, C. A., Caffo, B. S., ... Ewen, J. B. (2014). Variations in EEG discharges predict ADHD severity within individual Smith-Lemli-Opitz patients. Neurology, 83(2), 151-159. https://doi.org/10.1212/WNL.0000000000000565
Schreiber, John M. ; Lanham, Diane C. ; Trescher, William ; Sparks, Susan E. ; Wassif, Christopher A. ; Caffo, Brian S. ; Porter, Forbes D. ; Tierney, Elaine ; Gropman, Andrea L. ; Ewen, Joshua B. / Variations in EEG discharges predict ADHD severity within individual Smith-Lemli-Opitz patients. In: Neurology. 2014 ; Vol. 83, No. 2. pp. 151-159.
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Schreiber, JM, Lanham, DC, Trescher, W, Sparks, SE, Wassif, CA, Caffo, BS, Porter, FD, Tierney, E, Gropman, AL & Ewen, JB 2014, 'Variations in EEG discharges predict ADHD severity within individual Smith-Lemli-Opitz patients', Neurology, vol. 83, no. 2, pp. 151-159. https://doi.org/10.1212/WNL.0000000000000565

Variations in EEG discharges predict ADHD severity within individual Smith-Lemli-Opitz patients. / Schreiber, John M.; Lanham, Diane C.; Trescher, William; Sparks, Susan E.; Wassif, Christopher A.; Caffo, Brian S.; Porter, Forbes D.; Tierney, Elaine; Gropman, Andrea L.; Ewen, Joshua B.

In: Neurology, Vol. 83, No. 2, 08.07.2014, p. 151-159.

Research output: Contribution to journalArticle

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T1 - Variations in EEG discharges predict ADHD severity within individual Smith-Lemli-Opitz patients

AU - Schreiber, John M.

AU - Lanham, Diane C.

AU - Trescher, William

AU - Sparks, Susan E.

AU - Wassif, Christopher A.

AU - Caffo, Brian S.

AU - Porter, Forbes D.

AU - Tierney, Elaine

AU - Gropman, Andrea L.

AU - Ewen, Joshua B.

PY - 2014/7/8

Y1 - 2014/7/8

N2 - Objective: We sought to examine the prevalence of EEG abnormalities in Smith-Lemli-Opitz syndrome (SLOS) as well as the relationship between interictal epileptiform discharges (IEDs) and within-subject variations in attentional symptom severity. Methods: In the context of a clinical trial for SLOS, we performed cross-sectional and repeatedmeasure observational studies of the relationship between EEG findings and cognitive/behavioral factors on 23 children (aged 4-17 years). EEGs were reviewed for clinical abnormalities, including IEDs, by readers blinded to participants' behavioral symptoms. Between-group differences in baseline characteristics of participants with and without IEDs were analyzed. Within-subject analyses examined the association between the presence of IEDs and changes in attentiondeficit/ hyperactivity disorder (ADHD) symptoms. Results: Of 85 EEGs, 43 (51%) were abnormal, predominantly because of IEDs. Only one subject had documented clinical seizures. IEDs clustered in 13 subjects (57%), whereas 9 subjects (39%) had EEGs consistently free of IEDs. While there were no significant group differences in sex, age, intellectual disability, language level, or baseline ADHD symptoms, autistic symptoms tended to be more prevalent in the "IED" group (according to Autism Diagnostic Observation Schedule-2 criteria). Within individuals, the presence of IEDs on a particular EEG predicted, on average, a 27% increase in ADHD symptom severity. Conclusions: Epileptiform discharges are common in SLOS, despite a relatively low prevalence of epilepsy. Fluctuations in the presence of epileptiform discharges within individual children with a developmental disability syndrome may be associated with fluctuations in ADHD symptomatology, even in the absence of clinical seizures.

AB - Objective: We sought to examine the prevalence of EEG abnormalities in Smith-Lemli-Opitz syndrome (SLOS) as well as the relationship between interictal epileptiform discharges (IEDs) and within-subject variations in attentional symptom severity. Methods: In the context of a clinical trial for SLOS, we performed cross-sectional and repeatedmeasure observational studies of the relationship between EEG findings and cognitive/behavioral factors on 23 children (aged 4-17 years). EEGs were reviewed for clinical abnormalities, including IEDs, by readers blinded to participants' behavioral symptoms. Between-group differences in baseline characteristics of participants with and without IEDs were analyzed. Within-subject analyses examined the association between the presence of IEDs and changes in attentiondeficit/ hyperactivity disorder (ADHD) symptoms. Results: Of 85 EEGs, 43 (51%) were abnormal, predominantly because of IEDs. Only one subject had documented clinical seizures. IEDs clustered in 13 subjects (57%), whereas 9 subjects (39%) had EEGs consistently free of IEDs. While there were no significant group differences in sex, age, intellectual disability, language level, or baseline ADHD symptoms, autistic symptoms tended to be more prevalent in the "IED" group (according to Autism Diagnostic Observation Schedule-2 criteria). Within individuals, the presence of IEDs on a particular EEG predicted, on average, a 27% increase in ADHD symptom severity. Conclusions: Epileptiform discharges are common in SLOS, despite a relatively low prevalence of epilepsy. Fluctuations in the presence of epileptiform discharges within individual children with a developmental disability syndrome may be associated with fluctuations in ADHD symptomatology, even in the absence of clinical seizures.

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