Background. Severe breath-holding spells (BHS) in children consist of a stereotypical sequence of provocation to cry, noiseless expiration, color change, and loss of consciousness. Parenting a child who exhibits BHS is likely more stressful than parenting a healthy child, owing to this additional medical concern. It is also likely more stressful than parenting a child with a convulsive seizure disorder (SD), because it is often not recognized or misdiagnosed as behavioral rather than medical. Mothers of children with BHS were hypothesized to suffer significantly greater stress than mothers of children with SD or mothers of children without any significant medical conditions.
Objectives. To examine maternal stress in parenting a child who exhibits BHS, and to identify the specific areas of stress impact, compared with mothers of children with a convulsive SD and mothers of control children.
Methods. The Parenting Stress Index and questions regarding how mothers coped were individually administered to 34 mothers of children with BHS, 16 mothers of children with SD, and 16 mothers of children with no medical conditions (controls).
Results. Mothers of children with BHS or SD experience more overall stress and disruption in their attachment or understanding of their child, compared with control mothers. These groups of mothers (BHS and SD) also perceived their child as more distractible/hyperactive, less adaptive, and more demanding than did control mothers. However, mothers of the BHS group alone showed significant disruption in their sense of competence as a parent, maintaining self-identity, and receiving positive reinforcement from their child. Mothers of children with SD showed a similar trend, but it was not significant. These findings were not related to maternal health or feelings of depression/isolation, insufficient spousal support, child's mood, or other life stresses.
Conclusion. Parenting a child with BHS or SD impacts a greater degree of life stress on mothers of these children than does parenting control children, although the stress is greater for the BHS group than for the SD group.
- BHS =
- breath-holding spells •
- SD =
- seizure disorder •
- PSI =
- Parenting Stress Index •
- TSI =
- Total Stress Index •
- ANOVA =
- analysis of variance
Severe breath-holding spells (BHS) in children consist of a stereotypical sequence, beginning with provocation to cry and leading to noiseless expiration, color change, and loss of consciousness.1 The child usually appears pallid or cyanotic in color before losing consciousness. BHS are involuntary, reflexive, and caused by an underlying autonomic dysregulation.1 A genetic influence (autosomal dominant) predisposing to BHS has been recognized.2Parenting a child who exhibits BHS likely is more stressful than parenting a healthy child because of this additional medical concern. It also likely is more stressful than parenting a child with a convulsive seizure disorder (SD), because children with BHS are often mislabeled as having a behavioral rather than a medical problem. This may make BHS more stressful than SD for parents to manage. It is important to study the stress or anxiety related to parenting a child who has BHS to identify the specific areas of parental difficulty and to develop strategies for helping parents to manage these difficulties.
Previous research on the stress of mothers of children with medical conditions and/or special needs indicates that they experience more stress in parenting their children than do mothers of healthy children.3–8 These mothers typically perceive their children as being less positively reinforcing, more demanding, more moody, more distractible, and less adaptable than comparative control children. In addition, this stress negatively influences the way these mothers perceive themselves, their health, and their marriage.3–5 ,7 These mothers view themselves as less competent, more depressed, more socially isolated, and more restricted in their parental role, compared with mothers of healthy children.3–5 ,7 Moreover, they perceive more problems with their spousal relationship and maintaining their own health than do mothers of healthy children.5 ,7
The stress related to parenting a child with BHS has not been studied specifically and only 1 known study examined the behavioral profiles of children showing BHS. DiMario and Burleson,9 using the Achenbach Child Behavior Checklist and the Revised Child Behavior Profile, did not find differences in the behavioral profiles of children with BHS and control children. Thus, despite common beliefs that children with BHS display more extreme behaviors and temperaments than do children without BHS, no particular behavior problems have been documented in research.
We decided to compare the stress of parenting a child with BHS not only with a control group but also with a comparison group, including mothers of children who display some type of acute-onset medical event. Mothers of children with a convulsive SD were chosen, because similar to mothers of children with BHS, they watch their child experience an acute-onset, dramatic, medical event that often is perceived as intimidating and anxiety provoking. Children displaying absence seizures (seizures only at night during their sleep) were not included in the SD group, because their presentation is not as dramatic as BHS or convulsive seizures.
The primary purpose of this study was to examine stress in mothers who have a child with BHS to identify specific areas of stress impact. As part of this study, questions regarding how mothers cope with their child's episodes of BHS or SD and the Parenting Stress Index (PSI) were administered individually to 34 mothers of children with BHS, 16 mothers of children with SD, and 16 mothers of children with no medical conditions (controls). Mothers of children with BHS are hypothesized to suffer significantly greater stress than are mothers of children with SD or control mothers.
The subjects consisted of 66 mothers of children 1 to 8 years old (mean age = 4 years 8 months). Three groups were chosen for comparison: mothers of children with BHS, mothers of children treated for a concurrent convulsive SD, and a control group of mothers of children without any significant medical conditions. Children with BHS were age- and gender-matched with children in the SD and control groups, before mothers in these groups were recruited. Subjects in the BHS and SD groups were drawn from mothers of patients evaluated and diagnosed in the neurology division of Connecticut Children's Medical Center by 1 of the authors (F.J.D.). Mothers of children with any major medical, neurological, or psychiatric condition, besides BHS or SD, were excluded. Control mothers were recruited from family members and friends of the mothers in the BHS and SD groups. Control children had no concurrent significant medical, neurological, or psychiatric conditions, and they were not taking any prescription medication. Final data collections demonstrated that all but 1 (from the SD group) was the biological mother of the child. Table 1 presents other demographic information on the mothers in the study and their children. In matching the BHS with SD groups, it is noted that age of onset of the condition was earlier for the BHS group than for the SD group. Length of illness was longer for the BHS group (4 years) than for the SD group (2 years).
The subjects were first contacted by a letter to introduce the study and then by telephone to reiterate the purpose of the study and obtain consent. If the subject agreed to participate, a telephone interview was conducted. Subjects were interviewed regarding demographic information and relevant background information on their children (eg, developmental and medical history). Subjects in the experimental groups also were asked specific questions about their child's BHS or SD (eg, age of onset, number of episodes, etc) as well as how they coped with their children's condition. Regarding the latter, subjects in the experimental groups were asked how they responded and coped with their child's episodes of BHS or SD, what was helpful and not helpful in helping them cope, whether there was anything that could have made it easier for them and/or their family to cope, and whether there was anything that made it more difficult for them to cope. Then subjects were administered the PSI as part of the telephone interview.
The PSI is a 120-item questionnaire designed to assess possible sources of stress in the parent–child relationship of parents with children between 1 month and 12 years of age. The PSI assesses 3 domains: 1) child characteristics and the role of the child's temperament and behavior (Child Domain); 2) characteristics of the parent and the family environment (Parent Domain); and 3) stressful circumstances that the parent or family may have experienced, such as death of a loved one, loss of a job, or financial problems (Life Stress Domain). Scoring responses to the questionnaire results in raw scores that can be converted to a Total Stress Index (TSI) score; scores for the Parent, Child, and Life Stress Domains; and several subscale scores from within the Child and Parent Domains. Percentile scores also may be obtained for each of the respective scores. Mean percentile scores were used as the unit of analysis in this study. The normative sample included 2633 mothers, mostly from Virginia, but also from Massachusetts, New York City, North Carolina, Georgia, and Wisconsin. The mothers ranged from 16 to 61 years old (mean age = 30.9 years). They were mostly white (76%), but 11% were black, 10% were Hispanic, and 2% were Asian. The PSI has established psychometric properties, including content validity, internal consistency, and test–retest reliability.8
The responses of the subjects to open-ended questions, regarding coping with their child's BHS or SD, were compiled into several categories by M.M.-L. Percentages were calculated by comparing the number of subjects who provided a specific response to the total number of subjects in the group.
Mean percentile scores for the TSI and the subscales for the Child, Parent, and Life Stress Domains are presented in Table 2 by group. Comparison of scores based on socioeconomic status or race yielded no significant differences. These mean percentile scores do not indicate levels of stress that are borderline or clinically significant (percentile > 80). However, analysis of variance (ANOVA) of the TSI on the PSI indicates a significant difference across groups in terms of the amount of stress reported F (2,63) = 8.21. Posthoc comparisons used the Tukey-Kramer honestly significant difference test for multiple comparisons to guard against type I error (Table 3). Posthoc comparisons indicate that the BHS and SD groups had significantly higher TSI scores and more overall stress than do mothers of healthy children. There was no difference among groups for the Life Stress Score (F [2,63] = 1.03). This suggests that no mother faced high levels of environmental or situational stress outside the realm of the parent–child relationship
Considering perceived characteristics of the child as a source of stress, 1-way ANOVA of the Child Domain subscales indicated significant differences across groups for Distractibility/Hyperactivity, Adaptability, and Demandingness (F [2,63] = 6.44,F [2,63] = 10.1, and F [2,63] = 9.98, respectively; see Table 2). When compared on these subscales, the BHS and SD groups had significantly higher scores than did controls. In addition, ANOVA revealed a significant effect across groups on the Reinforces Parent subscale (F [2,63] = 3.10). Posthoc comparisons revealed a significant difference only between the BHS group and the control group. There were no significant differences among groups in the areas of Mood and Acceptability.
With reference to the Parent Domain, significant ANOVA scores were noted on 3 subscales. Posthoc analysis using the Tukey-Kramer honestly significant difference test for multiple comparisons revealed significant differences between mothers in the BHS and SD groups, compared with controls on the Attachment subscale only (F[2,63] = 3.41). The other 2 subscales revealed significant scores on posthoc analysis only between the BHS and control group. These subscales were Competence (F [2,63] = 3.81) and Role Restriction (F [2,63] = 3.83). The SD group mean percentile scores trended intermediate to the BHS and control groups. There were no significant differences among groups in the areas of Isolation, Health, Depression, and Spouse.
The responses of subjects in the BHS and SD groups to open-ended questions regarding coping with their child's condition underwent qualitative review. This review revealed that the majority of mothers indicated that having information on BHS/SD (74% and 75%, respectively) and talking with a knowledgeable professional (68% and 50%, respectively) helped them cope with their child's condition. A fair number (13% for both groups) also said that participating in a support group would have made it easier for them to cope. A fair number of mothers of children with BHS (21%) indicated that finding out that BHS was benign was helpful. In addition, many of these mothers said that having a packet of information about BHS early on (38%) and having information on it in child health/developmental magazines or books (17%) would have made it easier for them to cope with their child's BHS. Many mothers of children with SD (38%) indicated that medication was helpful in coping with their child's SD.
In this study, it was hypothesized that mothers of children with BHS suffer significantly greater stress than mothers of children with SD or control mothers. Mothers of children with BHS or SD experience more overall stress and disruption in their perception of their children, compared with control group mothers. They perceive their child as showing certain negative characteristics (ie, short attention span, distractibility, impulsivity, hyperactivity, being overly demanding, and being less adaptive) more so than do mothers of children without medical conditions. In addition, mothers in the BHS group show disruption in their perceptions of themselves, such that they question their competence as a parent. They also show disruption in their perceptions of their relationship with their child, experiencing their child as less of a source of positive reinforcement than do mothers of children without a medical condition. Similar results were found for the SD group, although scores tended to be lower than for BHS mothers. These findings were not related to maternal health or feelings of depression/isolation, insufficient spousal support, child's mood, or other life stresses. The mean percentile scores in the above areas also were not in the borderline or clinically significant range, compared with the norms of the PSI.
A possible weakness of this study is that the PSI was administered on the telephone, a change from the standard administration in which the person completes the questionnaire in pencil and paper form. Because the PSI was not administered in the way in which it was standardized, the validity of the results may be compromised. Nonetheless, reading the measure to the subjects as opposed to the subjects reading the questions themselves likely did not have a major impact on the results. A second possible weakness is the difference in age of onset and length of illness for the BHS and SD groups. It is possible that the significant differences in the BHS mothers responses compared with those of SD mothers in terms of parent reinforcement by the child, adaptability and demandingness of the child, as well as role restriction of the mother may be associated with the earlier onset of BHS and longer duration rather than the medical condition itself. Further research will be required to clarify this issue.
It is important to note that although this study did not look at the longevity of these negative perceptions, research suggests that these perceptions persist as the child becomes older even if BHS or SD remits.6 Forgays et al6 found that the stress and negative perceptions of children with special needs persist after the peak period of the illness. This suggests that intervention needs to take place early, because the negative perceptions and their effects likely will last if not interrupted.6
The findings of this study suggest that, because the parent–child relationship is under stress, mothers of children with BHS or SD are at risk for developing dysfunctional parenting behaviors and/or their children are at risk for developing behavior problems. This vulnerability exists, although a previous study evaluating behavior profiles in a cohort of children with BHS did not identify any specific behavior profile or any clinically significant out of normal behaviors.9 Although the current study did not indicate clinically significant levels of stress or behavioral problems, it did identify specific behavioral profiles for mothers of children with BHS or SD and their children.
Specifically, mothers of children with BHS or SD develop negative views of themselves and their children that make it harder for mothers to parent these children. They perceive little reward from their effort, seemingly believing they lack the skills to adequately manage their child. In other words, mothers of children with BHS or SD not only experience more overall stress and often have to work harder at parenting than do mothers of healthy children, but they also perceive fewer rewards in doing so. Such perceptions or dynamics make these mothers vulnerable to feeling defeated and depressed and make their children vulnerable to developing behavior problems.
Mothers of children with BHS or SD may seek to deal with these negative feelings by being very protective of their children, trying to prove they are caring, good mothers, and/or trying to prevent a spell or seizure. Ultimately, this protectiveness may not be favorable, because it decreases the child's opportunities to develop certain coping or adaptive skills. For example, a mother may continually give into a child's demands, rather than set limits or say “no” out of concern or fear of inducing a spell or seizure. Consequently, the child may become very demanding, expecting to get his or her own way and never having the opportunity to develop patience or coping skills to manage disappointment, frustration, upset, or anger. Thus, these children are vulnerable to significant emotional or behavioral problems (eg, depression, anxiety, poor impulse control, and/or oppositional/defiant behavior) in the future. The results of this study indicate that mothers of children with BHS or SD perceive their children to show poor attention, hyperactivity, impulsivity, and tend to be overly demanding and less adaptive in their coping than control children.
Professionals working with these mothers must let them know that setting firm limits not only is helpful but necessary to avoid the above predicament and to help their child become mature, responsible, and adaptive. These mothers need to be reassured that setting firm limits, ultimately, is what is best for their child. Such limits more closely imitate circumstances in the real world and encourage these children to learn coping skills to accept delayed gratification. If these mothers have trouble setting such limits, a referral to a parenting group focusing on behavior management or to a behavior specialist may be necessary.
The results of this study also suggest that mothers of children with BHS experience the parental role as restricting their freedom and frustrating them in their attempts to maintain their own identity. Mothers of children with SD showed a similar trend in feelings of role restriction. This finding may stem from mothers' difficulty doing activities separate from their children because of problems finding competent childcare. Many of the mothers of children with BHS or SD admitted that they were concerned about leaving their child with a babysitter or other individual, because the individual may not know how to cope with their child if he/she had a spell or seizure. They admitted that they had difficulty not only finding a person whom they considered responsible or trustworthy enough to manage their child's BHS or SD but finding someone who was willing to take on the responsibility of watching their child, including relatives. Consequently, mothers of children with BHS or SD seem to receive few to no breaks from their children and, in turn, struggle with chronic significant stress without interruption. This maintains a negative view of themselves, their child, and their circumstances.
Pediatricians, neurologists, and other professionals working with mothers of children with BHS or SD can help them in several ways. They can provide support and a positive and realistic perspective to these mothers in an attempt to improve their perception of themselves, their child, and their circumstances. It was important and reassuring to mothers of children with BHS or SD, when a professional approached them with support, empathy, and guidance rather than with criticism. Professionals also must provide a clear message to these mothers that they need to take time to care for themselves and relax to best care for their child. They should encourage these mothers to take part in activities that promote their identity separate from their children and even connect these mothers to resources in the community that may help them do so (eg, respite care, well-trained babysitters, or extracurricular programs for their children). Taking some time for themselves should help to improve their perception of themselves, their child, and their circumstances.
Qualitative review suggested additional ways to help mothers of children with BHS or SD, identifying factors that both help and do not help them (see Table 4). The majority of mothers of children with BHS or SD indicated that having information about BHS or SDs and how to handle them, as well as talking with a knowledgeable professional, were helpful. Mothers of children with BHS were especially grateful to have BHS diagnosed correctly and to have their experience validated as frightful events rather than as episodes that are “nothing to worry about” or “behavioral.” Mothers of children with BHS also found it helpful to discover or be reassured that the condition was benign with an expected natural history. Therefore, spreading knowledge to medical professionals about BHS and adequately training pediatricians, neurologists, and emergency department physicians (the doctors most likely to see children with BHS) to recognize and diagnose BHS correctly would be very helpful. Mothers of children with BHS or SD also acknowledged that support from others (eg, doctors, nurses, or family members), who understood BHS, was helpful. A majority of mothers of children with BHS indicated that learning about BHS in child health/developmental books or getting a packet of information about BHS from doctors made it easier for them too. Thus, creating an information packet about BHS to be distributed to parents of children with BHS and putting information about BHS in child development literature are other appropriate treatment interventions.
Although mothers of children with BHS or SD tended to show a similar pattern of increased stress in parenting, negative perceptions of themselves, their children, and the parent–child relationship, mothers of children with BHS experienced a greater degree of stress and more negativity in the above areas than the SD group. Thus, the results suggest that mothers of children with BHS are somewhat more at risk for developing dysfunctional parenting behaviors and/or their children are at greater risk for developing behavior problems than are those in the SD group. One reason for this difference between the BHS group and the SD group is that BHS is not as well-known or -recognized by doctors as SD. Many mothers of children with BHS struggled to get doctors to take them seriously about their child's spells. Many mothers of children with BHS felt significant relief and comfort, when they finally found a doctor that recognized or accurately diagnosed BHS. This diagnostic ambiguity and lack of recognition of significance are less frequent in SD.
Another reason for the difference between the BHS and SD groups may be the earlier onset of a spell in the BHS group in comparison with the onset of a seizure in the SD group. Fifty-six percent of the BHS group had their first spell before they were 1 year old and 97% had their first spell by 3 years old, whereas only 1 child had a seizure before they were 1 year old and only 50% of the SD group had their first seizure by the time they were 4 years old. Thus, many of the mothers of children with BHS were dealing with a child having spells who was nonverbal. Finally, many of SD children were regulated by medication, an option not readily available to the BHS group. At the time of data collection, 50% of the BHS group were still having spells, whereas only 19% of the SD group were still having seizures.
Future research in this area should focus on the perceptions of fathers of children with BHS to see whether their perceptions differ significantly from mothers and, if so, how to incorporate such findings to help families with a child who has BHS. Future research also can focus on the degree to which the interventions suggested above have a positive influence on mothers of children with BHS.
We thank Sharon Maycock, LPN, for her assistance in data retrieval.
- Received March 19, 1999.
- Accepted October 25, 1999.
- Address correspondence to Francis J. DiMario, Jr, MD, Division Neurology, Connecticut Children's Medical Center, 282 Washington St, Hartford, CT 06106. E-mail:
This work was presented in part at the Annual Child Neurology Society Meetings; October 22–25, 1998; Montreal Canada.
- DiMario FJ,
- Sarafarazi M
- Fitzgerald M,
- Butler B,
- Kinsella A
- ↵Forgays DK, Hasazi JE, Wasserman JC. Recurrent otitis media and parenting stress in mothers of two-year-old children. Dev Behav Pediatr. 1992;321–325
- ↵Abidin RR. Parenting Stress Index. Charlottesville, VA: Pedicatric Psychology Press; 1986
- Copyright © 2000 American Academy of Pediatrics