Original Article
Cardiac Autonomic Dysfunction in Patients With Infantile Spasm and the Effect of Adrenocorticotropic Hormone Treatment
Journal of Child Neurology 1-4 ª The Author(s) 2015 Reprints and permission: sagepub.com/journalsPermissions.nav DOI: 10.1177/0883073815583998 jcn.sagepub.com
¨ zgu¨r Duman, MD1, Pinar Gencpinar, MD1, Abdullah Kocabas, MD2, O 3 1 Nihal Olgac¸ Du¨ndar, MD , Senay Haspolat, MD , and Fırat Kardelen, MD4
Abstract Infantile spasm is an age-dependent epileptic-encephalopathy syndrome. Cardiac autonomic function is frequently altered in epilepsy. In this study, we examined heart rate variability in patients with infantile spasm before and after treatment. Nineteen patients with infantile spasm and 13 healthy comparisons were enrolled in the study. Cardiac rhythm was recorded with a Holter device for 24 hours before adrenocorticotropic hormone (ACTH) (Synacthen depot) and B6 vitamin administration and 1 month after treatment. Heart rate variability analysis found lower heart rate variability parameters in patients with infantile spasm at the onset of the syndrome, prior to treatment with ACTH. The time domain parameters of heart rate variability values showed a statistically significant increase following ACTH treatment. Our data suggest that patients with infantile spasm exhibit lower heart rate variability parameters, and the treatment of spasms with ACTH and B6 together diminished the autonomic dysfunction in our cohort. Keywords infantile spasm, autonomic dysfunction, heart rate variability, heart rate variability, ACTH treatment Received May 31, 2014. Received revised March 23, 2015. Accepted for publication April 01, 2015.
Infantile spasm is an age-dependent epileptic encephalopathy that is characterized by tonic spasms and hypsarrhythmia on electroencephalography (EEG). Prognosis is poor, and the mortality rate is higher than that seen in patients with epilepsy. In clinical follow-up, death occurs in 5% to 31% of patients with infantile spasm, secondary to underlying diseases, epileptic encephalopathy, or treatment.1,2 Cardiac autonomic dysfunction has been reported in patients with epilepsy.3,4 Jansen et al5 reported autonomic dysfunction and chronic autonomic changes in patients with West syndrome. Hattori et al6 found altered autonomic function and bradycardia as a result of adrenocorticotropic hormone (ACTH) treatment in patients with West syndrome. The autonomic system can be measured by evaluating heart rate variability, that is, the fluctuation in the duration of consecutive R-R intervals.6 In this study, we investigate autonomic function using heart rate variability analysis in patients with infantile spasm to demonstrate the probable existence of autonomic dysfunction and the effect of ACTH treatment.
were not receiving any antiepileptic drugs. Before diagnosis, at least one 16-channel EEG recording was performed on all patients during stage 2 sleep states. Electrodes were placed using the 10-20 International System with bipolar and referential montages (including the Oz electrode). Interelectrode resistance was below 3 kΩ and filters were set at 0.3 to 70 Hz. All patients had hypsarrhythmia on EEG according to criteria defined by Gibbs and Gibbs.7 Cranial magnetic resonance imaging (MRI), metabolic and intrauterine infection examinations, and chromosomal analysis were performed on all patients. None of the patients had hypothalamic or brain stem lesions. All participants were free of any cardiac abnormalities. Cardiac rhythm was recorded with a Holter device for 24 hours twice, once before
Materials and Methods
Corresponding Author: Senay Haspolat, Department of Child Neurology, Akdeniz University Hospital, 07070 Konyaaltı, Antalya, Turkey. Email:
[email protected]
Nineteen newly diagnosed patients with infantile spasm participated in the study. There was no previous history of seizures and patients
1
Department of Pediatric Neurology, Akdeniz University Hospital, Antalya, Turkey 2 Department of Pediatric Cardiology, Ankara Children’s Hematology and Oncology Research and Training Hospital, Ankara, Turkey 3 Department of Pediatric Neurology, Katip C ¸ elebi University Hospital, _Izmir, Turkey 4 Department of Pediatric Cardiology, Akdeniz University Hospital, Antalya, Turkey
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Journal of Child Neurology
Table 1. The Heart Rate Variability Parameters of the Patient and Control Groups. Parameters SDNN SDANN RMSSD NN50 PNN50 TAI TINN DIFFIDIX LOGIDX
Patients (n ¼ 13) 67.2 + 58 + 25.9 + 5951 + 3.8 + 18.7 + 147 + 63 + 66.8 +
4.8 4.4 4.2 1522 0.98 1.1 11.7 6.1 11.6
Controls (n ¼ 13)
P
Before ACTH treatment
84.8 + 4.6 74.8 + 4.3 30.4 + 4.2 8546 + 1900 5.7 + 1.4 22.3 + 1 162 + 18.6 77.7 + 5 53.3 + 6.4
