Novel Findings in Breath-Holding Spells

The mechanism of breath-holding spells (BHS) is not fully understood and most probably multifactorial; so, this study was designed to clarify the pathophysiology of BHS through assessing some laboratory parameters and electrocardiographic (ECG) changes which might be contributing to the occurrence of the attacks. Another aim of the study was to evaluate the differences in the pathophysiology between pallid and cyanotic types of BHS. This was a prospective study performed in Zagazig University Hospitals. Seventy-six children diagnosed with BHS were included as follows: 32 children with cyanotic BHS, 14 children with pallid BHS, and 30 healthy children as a control group. All children were subjected to the following: full history taking, clinical examination, and laboratory work up in the form of CBC, serum iron, ferritin, and zinc levels. Twenty-four hours ambulatory ECG (Holter) recording was also performed. No significant statistical difference was found between cyanotic and pallid groups regarding family history of BHS, severity, and precipitating factors of the attacks. Frequent runs of respiratory sinus arrhythmia (RSA) during 24 hours ECG were significantly higher in children with BHS; the frequency of RSA was significantly correlated with the frequency (severity) of the attacks. Low serum ferritin was significantly associated with BHS groups but not correlated with the severity of the attacks. Autonomic dysregulation evidenced by frequent RSA is considered to be an important cause of BHS in children and is correlated with the frequency of the attacks. Low serum ferritin is additional factor in the pathophysiology. Both pallid and cyanotic BHS are suggested to be types of the same disease sharing the same pathophysiology. (Medicine 94(28):e1150) man A. Bendary, M . Farag, MD, , MD, and Osama M.A. Elazouni, MD INTRODUCTION B reath holding spells (BHS) is a common problem in children, and particularly so in infants and is a frightening experience for the parents. BHS is apparently due to acute cerebral hypoxia, and the child recovers spontaneously after a period of unconsciousness and sometimes opisthotonic posturing. BHS may occur in children with normal neurological development, and usually have no impact on the subsequent development. The diagnosis is based on stereotyped sequence of clinical events that are triggered by mild trauma/emotional upset/frustration, which is followed by crying leading to noiseless state of expiration associated with color changes either cyanosis or pallor and ultimately loss of consciousness. The spells begin most commonly during the first 12 months of life and almost always by 2 years of age. Most children outgrow those spells approximately at the age of 6 years. Rarely BHS continue and are replaced by vasovagal attacks. Two types of BHS are present based on the color of the child during the apneic episode following the end of prolonged expiration either pale (pallid attacks) or blue (cyanotic attacks); rarely both types may occur in the same child (mixed type). The mechanism of BHS is not fully understood and most probably multifactorial. Iron deficiency anemia is claimed to be associated with the occurrence of BHS supported by the response of some cases to iron therapy; associated zinc deficiency may be an additional factor. Autonomic dysregulation that leads to alteration in cardiac function and simultaneous decrease in cerebral blood flow is an important risk factor, so electrocardiogram (ECG) should be strongly considered in any patient with BHS. In this study, we aimed to clarify the pathophysiology of breath-holding spells by assessing factors claimed to be cks including laboratory parameters and continuous ECG monitoring and to identify differences in the pathophysiology between pallid and cyanotic BHS. METHODS This was a prospective cross-sectional study performed in Zagazig University Hospitals from December 2012 to January 2014. A total of 76 children diagnosed with BHS at the child neurology out-patient clinics were included and assessed at the Pedia trics, Neurology and Cardiology Departments of the same Hospi tals. Their age ranged from 15 to 48 months (mean 31 month s). We divided our cases into 3 groups: Group I: 32 patients with cyanotic BHS. Group II: 14 patients with pallid BHS. Group III: 30 healthy children, of comparable age and sex; (2) ediatric Department for preoperative ctive surgery, were enrolled as a control www.md-journal.com | 1 test. Categorical data were evaluated by Pearson x test. StatExclusion Criteria Children with primary cardiac or central nervous system disease were not included as well as any child with uncertain history of the type of BHS or suspected mixed type. Any child with possibility of seizure was subjected to EEG assessment and children with abnormal EEG were also excluded. Initial basic Azab et al