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ORIGINAL ARTICLE
Year : 2019  |  Volume : 8  |  Issue : 4  |  Page : 94-98

Paraclinical cardiac findings of children with breath-holding Spells in Taleghani Hospital of Gorgan, Iran, during 2014–2016


1 Department of Pediatrics, Taleghani Medical and Educational Center, Golestan University of Medical Sciences, Gorgan, Iran
2 Pediatrics Center of Excellence, Childrens Medical Center, Tehran University of Medical Sciences, Tehran, Iran
3 Student Research Committee, Golestan University of Medical Sciences, Gorgan, Iran
4 General Practitioner, Golestan University of Medical Sciences, Gorgan, Iran

Date of Submission07-May-2019
Date of Decision26-Jun-2019
Date of Acceptance23-Nov-2019
Date of Web Publication30-Jan-2020

Correspondence Address:
Dr. Zohre Rahatab
General Practitioner, Golestan University of Medical Sciences, Gorgan
Iran
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/rcm.rcm_12_19

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  Abstract 


Context: Breath-holding spells (BHSs) are episodes of brief, involuntary cessation of breathing that occur in children in response to stimuli such as anger, frustration, fear, or injury. It occurs in about 0.1%–4.6% of healthy children. Although the etiology is not known, autonomic dysfunction and increased vagal tonus leading to cardiac arrest and cerebral anoxia are considered to play a role. Increase of QT dispersion increases the risk of sudden death. Because of this, children are always referred to cardiac clinics. Aims: Our aim was to evaluate According to these problems, our aim was the evaluation of ECG and Echocardiography findings in children diagnosed with BHSs in Taleghani Hospital in Gorgan. Settings and Design/ Methods and Material: All patients diagnosed with BHSs were enrolled in this study. In the first step, demographic characteristics and echocardiography and electrocardiogram (ECG) findings were obtained from the patients. Statistical Analysis Used: For statistical analysis, the demographic variables and ECG and echocardiography information of the patients were analyzed using frequency, percentage, and statistical tests such as Chi-square. Results: Sixty-three (9%) children were male and 36 (1%) children were female. Thirty-eight (9%) children were aged between 1 and 2 years, and this group was the most common age group. The most common type of attack was cyanotic (68/5%); 24 (1%) children had a positive family history, 78 (8%) had incomplete or absent iron supplementation, and 68% of children had anemia. Five (6%) children had long QT in ECG and five (6%) had abnormal echocardiography. There was a significant correlation between echocardiography findings and age, whereas there was no significant relationship between ECG findings with age, echocardiography findings with sex, and type of attack with age and sex. Conclusions: Although BHS among children with LQTS are relatively rare and occur at similar frequency as the general population, they can be the presenting symptom for a heart rhythm disorder.

Keywords: Breath-holding attacks, breath-holding spells, cardiac findings, paraclinical findings


How to cite this article:
Esmaeili H, Hosseini SA, Montazeri M, Rahatab Z. Paraclinical cardiac findings of children with breath-holding Spells in Taleghani Hospital of Gorgan, Iran, during 2014–2016. Res Cardiovasc Med 2019;8:94-8

How to cite this URL:
Esmaeili H, Hosseini SA, Montazeri M, Rahatab Z. Paraclinical cardiac findings of children with breath-holding Spells in Taleghani Hospital of Gorgan, Iran, during 2014–2016. Res Cardiovasc Med [serial online] 2019 [cited 2020 Feb 24];8:94-8. Available from: http://www.rcvmonline.com/text.asp?2019/8/4/94/277272




  Introduction Top


Breath-holding spells (BHSs) are a short and involuntary experience of respiratory arrest that occurs in children in response to stimuli such as anger, disappointment, fear, and physical damages.[1] The occurrence of this attack can be a terrifying experience for parents because the children seems dead and unresponsive due to cerebral anoxia during the attack. There are two major types of BHS, including cyanotic BHS, which are more common, are usually predictable, and are caused by an emotional upset of the infant. The incidence of this attack is associated with the rapid onset of generalized cyanosis and a reduction in consciousness, which could result in repetitive generalized clonic seizures, opisthotonos, and bradycardia. The other BHS type is pallid, which is less common compared to the cyanotic type. However, these BHS types have many similar characteristics.[2] BHS has been reported to occur in 0.1%–4.6% of children worldwide.[3]

BHS attacks typically start between 6 and 18 months of age and will stop by the age of 4–5 years. In rare cases, BHS might continue to the age of 7 years, and more than 4%–5% of children above the age of 8 experience this attack.[4] Some of these children may experience one attack annually and some might experience attacks several times a day.[5] About 10% of these children experience BHS once a month.[6] Even though the pathogenesis of BHS is not well understood, some studies suggest that imbalance between sympathetic and parasympathetic activities can be responsible for the manifestations.[7] Moreover, a genetic causative factor may be responsible for the disease due to detecting a positive family history in 20%–30% of patients with autosomal dominant disorder.[8] Some studies have reported iron deficiency and anemia (caused by decreased production of red blood cells) in these patients.[9] Others believe that these attacks occur due to psychological problems, depression, anxiety in mothers, and stressful accidents in the parents of infants.[10]

While the etiology of BHS is still unknown, autonomic dysfunction and increased vagal tone, which lead to cardiac arrest and anoxia, can be considered as major factors in this regard.[4] This cardiac arrest and anoxia may ultimately lead to the emergence of bradycardia and asystole that last longer than 2 s, leading to cerebral hypoperfusion. Studies have shown that children with pallid BHS are susceptible to vasovagal syncope. In addition, delay in demyelination of nerve cells is observed in these patients.[11] In general, BHSs resolve spontaneously and have a good prognosis, with the exception of increased risk of syncope that occurs in subsequent years.[12]

The possibility of an increase in QT is another dangerous complication of the disease that can lead to early neonatal mortality.[5],[13] However, due to contradictions between limited studies and evaluation of the possibility of the long QT, detection of QT syndrome and its risks should be considered in these children.[4] This is mainly because long QT can increase the risk of arrhythmias and endanger the life of the individual.[14] Some studies have indicated that implantation of a pacemaker can effectively reduce or improve the severity of symptoms in BHS.[15] As such, children with this condition are referred to cardiology clinics for examination and diagnosis of heart diseases.[16] Therefore, with regard to the cardiac complications and problems of these infants, this study aimed to evaluate Long QT and echocardiography finding of Children with Breath-holding Spells in Taleghani Hospital of Gorgan, Iran.


  Materials and Methods Top


This descriptive cross-sectional study was performed on neonates with BHS (selected by census sampling) referred to Taleghani Hospital during April 2014–2016. After obtaining the necessary approvals from the university and applying the inclusion criteria (no specific organic diseases, age below 6 years, diagnosis of BHS, normal growth and development, no history of epilepsy in first-degree relatives, no systemic diseases, and normal electroencephalogram) and exclusion criteria (unwillingness to participate in the study, the diagnosis of organic diseases or those affecting the electrocardiogram [ECG] findings, the history of using drugs that affect QT [erythromycin, clarithromycin, and furosemide], more than 0.48-0.49 seconds, sodium below 130 and above 150 mg/L, calcium below 8 mg/ml, and fasting blood sugar below 50 mg/ml). In total, 108 children with possible BHS were selected after the examination of clinical symptoms by a pediatric cardiologist. The criterion for diagnosing BHS was the patient's medical history. After obtaining informed consent from the parents, echocardiography and ECG recordings were taken from all children between BHSs and in the first stage of the study.

Electrocardiography was performed with a specific 12-lead ECG for all children. Cardiac examination and ECG interpretation were performed by the pediatric cardiologist. Demographic characteristics (age, gender, birth weight, family history of BHS in first-degree relatives, type of attack, frequency of attack, medication use during pregnancy, and method of giving iron to the child) and information related to ECG and echocardiography are presented in a previously prepared checklist.

In addition, laboratory tests including complete blood count (CBC) were requested for children suspected of anemia. After encoding the data collected, data analysis was performed in SPSS version 18 using descriptive indicators, including mean and standard deviation. In addition, qualitative data were described applying frequency and percentage.


  Results Top


In this study, 108 children referred to Taleghani Hospital of Gorgan during 2014–2016 were enrolled to evaluate paraclinical cardiac results. In terms of gender, 63.9% of the children were male and 36.1% were female. In addition, 8.3% of the participants were aged below 6 months, whereas 19.4%, 38.9%, and 33.3% of the neonates were aged 6–12 months, 1–2 years, and above 2 years, respectively [Table 1] and [Table 2].
Table 1: Frequency of gender of childre

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Table 2: Frequency of age of children

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The frequency of birth weight (<2500 g) and normal weight (2500–4000 g) is summarized in [Table 3].
Table 3: Frequency of birth weight

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In this research, the most common type of BHS was cyanotic (68.5%), and the highest incidence rate of attacks was one–three times (47.2%). In addition, 24.1% of the neonates had a positive family history and 21.3% of the neonates had complete iron supplementation [Table 4].
Table 4: Frequency of type of attack, repeat attack, and how to use iron

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In the present study, 5.6% of the children had long QT based on EKG results and 1.9% of the neonates had patent ductus arteriosus (PDA) according to echocardiography [Table 5].
Table 5: Frequency of echocardiography finding

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Moreover, the CBC test was requested for fifty patients due to an anemia-related clinical suspicion. Among the evaluated participants, 26% had leukocytosis, whereas 68% and 18% had anemia and thrombocytosis, respectively [Table 6].
Table 6: Frequency of laboratory test finding

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Based on Chi-square results, the frequency of ECG results showed no statistically significant relationship between BHS and the variables of gender and age (P = 0.058 and P= 0.579, respectively) [Table 7].
Table 7: Frequency of electrocardiogram finding according to age

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The frequency of echocardiographic findings in terms of gender and age in children with BHS is presented in [Table 8]. According to Chisquare test, while echocardiography results had no statistically significant correlation with gender (P = 0.753), there was a statistically significant relationship between these results and age (P = 0.017) [Table 8].
Table 8: Frequency of echocardiography finding according to age and gender

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The frequency of the type of attack in terms of gender and age in neonates with BHS is shown in [Table 9]. Furthermore, no statistically significant correlation was found between the type of BHS and the variables of gender and age (P = 0.736 and P= 0.538, respectively).
Table 9: Frequency of the type of attack in terms of gender and age in neonates with BHS

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  Discussion Top


BHS is a sudden benign event associated with apnea and changes in muscle tone conditions of infants. While the attacks are sometimes similar to seizures, they are not epileptic. Despite its generally good prognosis, BHS is often associated with complications such as decreased consciousness, tonic-clonic movements, and epilepsy, or cerebral hypoxemia in severe cases.[17] Some studies have shown the association between iron deficiency and anemia with BHS. In addition, neonates with iron deficiency are more prone to these attacks.[18]

There are two types of BHS, including cyanotic that begins with fear, anger, and despair, and pallid that starts with pain.[19] In general, BHSs occur between the ages of 6 months and6 years and vary in frequency from several times a day to three–four times a month. Furthermore, the number of attacks decreases by aging until the attacks completely stop. This condition might occur in infants with a positive family history, and several children in a family could have attacks. Therefore, studies have considered the role of genetics to be effective in the occurrence of these attacks.[20]

A family history of about 23%–38% can contribute to the disease, and some studies have reported autosomal dominant disorder to be the causative factor for this condition. Moreover, the condition has a male-to-female ratio of 3:1. Less than 10% of the disease continues until after the age of 2. The relationship between BHS and asystole has been reported in some studies as well; approximately 70% of patients with pallid BHS had asystole related to increased vagal tone. In addition, some studies have reported long QT syndromes in ECG. As such, it is recommended that ECG be carried out for patients with BHS.

The present study aimed to evaluate the cardiac findings of neonates with BHS at the Taleghani Hospital of Gorgan. A total of 108 children with BHS referred to Taleghani Hospital in Gorgan were diagnosed based on clinical suspicion and history taken by a pediatrician during 2014–2016. In the current research, 63.9% of the neonates were male and 36.1% were female. Regarding the variable of age, 8.3% of the participants were aged below 6 months, whereas 19.4%, 38.9%, and 33.3% of the neonates were aged 6–12 months, 1–2 years, and above 2 years, respectively. In 2014, Yilmaz et al. evaluated the neurological and cardiac status of children with BHS. In total, 165 children were enrolled in the study, 54.5% of whom were male and the rest were female. In addition, while 38.8% of the neonates were aged below 6 months, 30.9%, 23.6%, and 6.7% of the participants were aged 7–12 months, 12–24 months, and above 24 months, respectively.[21]

In another study by Mirzayiseyfabad et al. (2001) conducted to evaluate the therapeutic effects of iron on BHS, 65.6% of the neonates were male and 34.4% were female. Moreover, 3.1% of the participants were aged below 6 months, whereas 78.1%, 12.5%, and 6.3% of the neonates were aged 6 months–2 years, 2–4 years, and above 4 years, respectively.[22]

In the current research, the most common type of attack was cyanotic (68.5%), whereas 24.1% and 7.4% of the cases had pallid and mixed BHSs, respectively. In a study by Movahedian et al. (2016) on the QT status of children with BHS, 56 children were enrolled in the study, 83.9%, 12.5%, and 3.5% of whom had cyanotic, pallid, and mixed BHSs, respectively.[23]

In the mentioned study, family history was evaluated, reporting that 24.1% and 75.9% of the children had a positive and negative family history, respectively. In a study conducted by Azab et al. (2015), 76 children with BHS were enrolled, 31.3% of whom had a positive family history in this regard.[24]

In the present study, the highest incidence rate of BHS was one–three times per month (47.2%), followed by more than three times per month (45.4%) and without recurrence (7.4%). In 2017, Jain et al. performed a research on 100 children to evaluate the effect of iron supplementation on BHS. According to the results, 34% of the participants experienced attacks less than ten times per months, whereas 36% and 30% had attacks more than ten times per month and no attacks, respectively.[18]

In the present study, ECG showed long QT in 5.6% of children. In addition, echocardiography indicated PDA, PFO, ASD, and tricuspid regurgitation (TR) in 1.9%, 0.9%, 2.8%, and 1.9% of the participants, respectively. Moreover, there was no statistically significant correlation between ECG results (long QT) and the variables of age and gender based on Chi-square test (P = 0.579 and P= 0.058, respectively).

Moreover, no statistically significant correlation was found between echocardiography findings and gender (P = 0.753). In a study by Tomoum et al. (2018) on the association between autonomic impairment and iron deficiency with BHS, sixty people were enrolled, forty of whom had BHS. According to the results of the aforementioned study, there was no significant relationship between ECG findings and the variables of age and gender (P = 0.40 and P= 0.27, respectively).[1] In another study by Yilmaz et al. (2014), there was no echocardiographic impairment finding among patients with BHS.[21] However, there was a statistically significant relationship between age and echocardiography results in the present study (P = 0.017). According to our findings, there was no statistically significant relationship between the type of attack and the variables of gender and age (P = 0.736 and P= 0.538, respectively).

In 2016, a study was conducted by Movahedian et al. on QT changes in children with BHS, and the results were indicative of no relationship between the type of attack and gender (P > 0.05).[23] Furthermore, in a research by Azab et al. (2010), there was no statistically significant relationship between the type of attack and age (P > 0.05), which is consistent with our findings. In the current research, CBC was requested for fifty patients with regard to the clinical suspicion of anemia. Among these participants, 68% had hemoglobin (Hb) below 12. In the study by Jain et al. on the effect of iron supplementation on BHS, 73% of the patients had anemia and Hb <12. In general, children with anemia have lower oxygen-carrying capacity and are at increased risk for hypoxia, which could decrease by 52% with iron supplementation.[18]


  Conclusion Top


According to the results of the present research, there was no relationship between ECG findings and type of attack with age and gender and between echocardiography with gender and age. In addition, the most common type of attack was reported to be cyanotic. According to ECG and echocardiography findings, long QT and PDA, patent foramen ovale, TR, and atrial septal defect were observed in neonates with BHS.

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Conflicts of interest.

There are no conflicts of interest.



 
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    Tables

  [Table 1], [Table 2], [Table 3], [Table 4], [Table 5], [Table 6], [Table 7], [Table 8], [Table 9]



 

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