Abstract
To understand the stressors experienced by parents of hemophiliac children in China and the coping behaviors utilized, semi-structured interviews were conducted with parents of 158 Chinese hemophiliac children. We administered the Coping Health Inventory for Parents to determine coping behaviors. Parents’ main stressors were in the domains of children’s illness and physical condition, economic problems, and so on. Parents engaged in various positive coping behaviors to manage these stressors. The helpfulness of these coping behaviors in maintaining a normal family life differed significantly by parents’ education and income (p < .01). Parents of hemophilic children in China face numerous stressors but engage in a number of coping behaviors to manage these stressors. While treating hemophilic children, Chinese medical workers should guide parents to adopt such positive coping behaviors.
Introduction
Hemophilia is a hereditary genetic defect of coagulation factors VIII and IX that causes insufficient or functional deficiencies in plasma factors VIII and IX, thereby leading to lifelong coagulation dysfunction. Hemophilia is characterized by a disease onset during childhood, unstoppable bleeding occurring spontaneously or after mild trauma, hematoma formation, and joint bleeding. Lifelong coagulation factor replacement therapy is usually required (Franchini and Mannucci, 2012). During childhood, this therapy not only affects children’s daily lives and physical and mental health but also greatly affects the lives of their parents, siblings, and family. For instance, home treatment for hemophilia enables flexibility and autonomy for patients and other family members, while mothers may experience this type of treatment as a burden (Dolatkhah et al., 2014; Von Der Lippe et al., 2017; Witt and DeLeire, 2009). Parents must invest substantive amounts of time, energy, emotion, and money to provide care and support to their hemophilic children. Furthermore, parents also must deal with various problems and difficulties that arise during therapy, such as complications of other diseases. Thus, these parents might exhibit worry, tension, anxiety, frustration, fear, and so on. These negative psychological conditions and behaviors can adversely affect the physical and mental health of both children and parents (Beeton et al., 2007; Tifferet et al., 2011). Conversely, parents who can maintain an optimistic attitude and cope positively with these stressors can better help children cope with similar problems (Dolatkhah et al., 2014; Morawska et al., 2015; Witt and DeLeire, 2009).
The current study aimed to understand the stressors and coping behaviors of parents of Chinese hemophiliac children and the effects of parents’ gender, education, and family income on their coping behaviors. The results of our study indicate that parents of Chinese hemophilic children mainly experienced stressors in the following seven domains: children’s illness and physical conditions, economic problems, children’s treatments, caring for children, genetic issues, role dysfunction, and communication problems. We intended to provide a basis for clinical workers to guide parents in engaging in positive and effective coping behaviors in order to better care for children, including maintaining family unity, coordination, and optimism; seeking social support; maintaining self-esteem and psychological stability; communicating actively with medical personnel; and obtaining greater access to disease knowledge.
Methods
Study participants
All children were registered before February 2015 at the Chinese Hemophilia Case Information Management Centre. Nonprobability purposive sampling was used to select the parents of children younger than 18 years old with various types of hemophilia. All participants were outpatients at the study hospital between February 2009 and May 2015. A total of 171 patients met these criteria and were contacted, and the investigation was explained to parents (via in-person discussion, telephone, mail, WeChat, etc.). A total of 158 parents agreed to participate. All hemophilic children were diagnosed according to the ‘Consensus of Chinese Experts on Diagnosis and Treatment of Haemophilia’ guidelines (Thrombosis and Hemostasis Group, Chinese Society of Hematology, Chinese Medical Association/Hemophilia Treatment Center Collaborative Network of China, 2013); the disease course was more than three months for all patients. The study was approved by the institutional review board of our university and conformed to the Declaration of Helsinki. All parents/legal guardians of the patients provided their written consent.
Measures
Stressors faced by parents of hemophilic children were assessed using semi-structured interviews. We created the interview questionnaire according to literature review on stressors faced by parents of children with chronic diseases (Cassis et al., 2012; Li et al., 2010a; Li et al., 2010b). The interview items were then reviewed and modified by experts at the Chinese Hemophilia Case Information Management Centre. The stressors assessed in the interview formed eight categories. Each stressor was rated in terms of severity of stress on a scale ranging from 0 to 3 (see Supplementary Material I). All participants were asked to write down and rate their stressors according to the eight categories mentioned above. Then, the experts at the center summarized the main stressors according participants’ responses, resulting in the most common 2–3 stressors in each categories being summarized. Other minor stressors were abandoned. Cronbach’s α was .89.
Parents’ coping behaviors were investigated using the Coping Health Inventory for Parents (CHIP) developed by McCubbin et al. (1983). The CHIP is reliable and valid (Allen and Marshall, 2010; Li and Wei, 2007; McCubbin et al., 1983). Li and Wei (2007) translated the CHIP into Chinese and then back translated it using the Brislin translation model (Jones et al., 2001) and tested its applicability to the Chinese culture. The Chinese version of the CHIP contains 45 items, each corresponding to a coping behavior in three subscales: ‘maintaining family solidarity, cooperation, and an optimistic attitude’ (19 items), ‘seeking social support and maintaining self-esteem and psychological stability’ (18 items), and ‘understanding the disease situation by consulting medical personnel and communicating with other parents’ (8 items). All items were rated on a five-point scale ranging from 1 (Never) to 5 (Always used). If a method was used (i.e. all responses other than ‘1’), participants indicated how helpful the method was in maintaining a normal family life on a four-point scale (0 = Not helpful, 1 = Minimally helpful, 2 = Moderately helpful, or 3 = Extremely helpful). The Chinese version of the CHIP had a Cronbach’s α of .91 and a content validity index of .82, thereby indicating that it was reliable, valid, and applicable to China (Li et al., 2010a; Li et al., 2010b; see Supplementary Material II in English and Chinese).
Procedure
Data were collected via face-to-face interviews in various settings, such as during children’s diagnostic visits, consultations with parents at a hemophilia center, or at hemophilia lectures. All interviews were conducted by Xiangya Hemophilia Care Center physicians.
Statistical analysis
EpiData 3.02 (The EpiData Association, http://www.epidata.dk) database management software was used. Additionally, statistical analyses were performed using SPSS 19.0 (IBM Corporation, Armonk, NY, USA). Mean ± standard deviation (SD;`x ± s) was calculated for measurement data. Frequency and percentage were calculated for count data. Comparison of measurement data was conducted with a single factor analysis of variance (ANOVA). Comparison of count data was conducted through χ 2 test. A p value <.05 was considered statistically significant.
The study was approved by Ethics Committee of Xiangya Hospital of Central South University (No.: 2012-230 (Date of approval: December 30, 2012)).
Results
Parent characteristics
One hundred and fifty-eight parents (60 (38.0%) fathers and 98 (62.0%) mothers) of hemophilic children were interviewed. Participants’ average age was 35.3 years (SD 5.0) and ranged from 26 to 53 years. Eighty-nine (56.3%) parents had completed junior high school or below and 69 (43.7%) had completed high school or above. Sixty-seven (42.4%) had a monthly family income of ≤1000 RMB (US$143.73), 51 (32.3%) had an income of 1001–2000 RMB (US$143.87–287.46), 22 (13.9%) had an income of 2001–5000 RMB (US$287.60–718.65), and 18 (11.3%) had an income of ≥5001 RMB (US$718.79; 2014 median national per capita disposable monthly income: 1464.17 RMB (US$210.44); US$ 1 = 6.96 RMB).
General conditions of children
Of the 158 hemophilic children whose parents were interviewed, the average age was 10.7 years (SD 7.1) and ranged from 3 months to 18 years, with 136 (86.1%) displaying hemophilia A and 22 (14.0%) displaying hemophilia B. Eighty-six (54.4%) had complications of osteoarthritis and 55 (34.8%) had complications of muscular dystrophy. The average time absent from school due to disease was 2.9 months per school year. Ninety-three (58.9%) patients were not covered by medical insurance (i.e. parents had to pay for treatment out-of-pocket).
Parent stressors
All parents were asked to write down and rate their stressors according to the eight categories. Experts at the center selected the most common 2–3 stressors in each category (summarized in Table 1). The categories were ranked in terms of mean stress score (from highest to lowest). Children’s illness and physical conditions, Economic problems, and Children’s treatments were the three main sources of stress. Almost all parents mentioned these three categories of stressors and perceived these stressors as very serious. The mean total number of stressors and mean total stress scores were used as dependent variables. One-factor ANOVAs revealed that the number of stressors and total stress scores differed significantly by family residence, having medical insurance, parents’ education level, and family monthly income (see Table 2). Specifically, fewer stressors and lower stress scores were found among parents who were from urban areas, more highly educated, had higher incomes, and had purchased medical insurance. Furthermore, the child’s diagnosis (i.e. hemophilia A or hemophilia B); whether the family had genetic history of hemophilia; whether the child had osteoarthritis, muscular dystrophy, or other disorders; whether the child was absent from school; or whether we investigated the father or the mother did not affect the number of stressors, but the total stress scores differed significantly.
Prevalence and severity of stressors faced by parents of Chinese hemophiliac children (N = 158).
Differences in number of stressors and stress scores by parent and child characteristics.
Coping behavior
ANOVA results on frequency of use of various coping behaviors and their level of helpfulness indicated that helpfulness scores differed significantly by parents’ educational levels (p < .01 for total score and all three subscale scores). Specifically, highly educated parents had higher CHIP scores. CHIP scores also differed according to family monthly income (p < .01 for total score and three subscale scores), whereby parents with higher monthly incomes tended to adopt more helpful coping behaviors for maintaining a normal family life. Additionally, we also found that coping behaviors and their level of helpfulness had no relationship with the child’s diagnosis (i.e. hemophilia A or hemophilia B); whether the family had genetic history of hemophilia; whether the child had osteoarthritis, muscular dystrophy, or other disorders; whether the child was absent from school, or whether we investigated the father or the mother. These results were similar to the results of stressors faced by parents and average total stress scores (Table 2). Additionally, it was found that children’s residence and children’s medical insurance demonstrated a relationship with the coping behaviors utilized and their perceived level of helpfulness, but these relationships were not statistically significant (Table 3).
Scores on the CHIP by parent and child characteristics (N = 158,
Note: CHIP: Coping Health Inventory for Parents.
aMaintaining family solidarity, cooperation, and an optimistic attitude.
bSeeking social support and maintaining self-esteem and psychological stability.
cUnderstanding the disease situation by consulting medical personnel and communicating with other parents.
Regarding which coping behaviors were more helpful, of the 45 described in the CHIP, ≥35 behaviors were exhibited by 77.8% of the parents and ≥40 behaviors were exhibited by 16.5%. The coping behaviors that parents considered the most and least helpful for maintaining a normal family life are listed in order in Table 4.
Behaviors that parents considered the most and least helpful in maintaining a normal family life.
Discussion
Stressors faced by parents of Chinese hemophilic children
A stressor is a stimulus causing physiological and psychological tension reactions and requires adaptive coping responses to alleviate (Figley and McCubbin, 1983). Despite the advancement of medical care standards, millions of children and adolescents with chronic illness experience stress-related emotional and behavioral problems during treatment that interfere with the appropriate implementation of treatment plans. Furthermore, parents’ coping behaviors change over the course of the disease (Compas et al., 2012; Hamner et al., 2015). Many medical centers worldwide have administered questionnaires about hemophilic families, such as disease management, family history, emotional stress, and financial burden, as these factors are common stressors faced by hemophilia families (DeKoven et al., 2014; Torres-Ortuño et al., 2014). The results of this study indicate that parents of Chinese hemophilic children mainly experienced stressors in the seven domains (e.g. children’s illness and physical conditions, economic problems, children’s treatments, etc.) mentioned above.
Most stressors related to children’s illness and physical conditions, and these stressors appeared to be the most severe. However, most (98.7%) parents reported stress related to financial problems because of long-term replacement therapy (for which costs range from 300 to 1200 RMB (US$43.56–174.24) per 200–250 IU of coagulation factor). This pressure is enormous for families with monthly income of ≤2000 RMB (US$287.46) or for children without medical insurance. Even in children who had medical insurance, coverage was basic, with many medicines, such as coagulation factor, considered self-funded medicine. Therefore, in these instances, these stressors appeared to be the most severe.
Regarding treatments, 94.3% of parents mentioned that it was difficult to obtain appropriate medical treatment for their children. It is often very difficult to obtain treatment in 3A (i.e. elite) hospitals in China, which can cause tremendous psychological and economic pressure among parents seeking medical treatment for their children (Li and Wei, 2007; Li et al., 2010a; Li et al., 2010b).
Regarding caring for children, the major problems faced by parents were ‘not having relevant knowledge of hemophilia’ and ‘not knowing how to care for children’. These results suggest that parents must be educated on how to care for their children with hemophilia. Some parents also felt pressure from society, because having children with genetic diseases is considered shameful in Chinese culture (Li and Wei, 2007; Li et al., 2010a; Li et al., 2010b).
Effects of education and family income on parents’ coping behaviors
Coping behaviors are methods, approaches, or strategies people use to cope with internal and external environmental demands that cause emotional distress (Compas et al., 2012). We showed that, in China, parents with higher education levels are more likely to engage in positive and effective coping behaviors, likely because highly educated parents are more knowledgeable or capable of obtaining knowledge about the disease and are therefore able to cooperate better with treatment. Less educated parents, however, mainly come from rural areas and, thus, would have limited access to relevant information and be less knowledgeable overall about the disease. Therefore, these parents would rarely engage in helpful coping behaviors, such as getting a second job, reading books to understand coping methods employed by people with similar problems, and attempting to understand the medical problems facing them in detail.
In China, parents with higher monthly incomes tended to engage in positive coping behaviors, which is similar to the trend with educational level. This is perhaps because socioeconomic status is closely related to educational level in China. Parents with higher incomes are generally better able to invest in their children’s health, thereby ensuring that their children will receive the necessary treatment in time to avoid future disabilities. However, parents with low income have difficulty in affording these long-term high medical expenses, which leads to an inability to provide the necessary treatment for hemophilic children. Having a low educational level also prevents these parents from improving their economic conditions.
Coping behaviors considered the most and least helpful in maintaining a normal family life
Among the five coping behaviors that parents considered the most helpful for maintaining a normal family life, four belonged to the subscale of ‘maintaining family solidarity, cooperation, and an optimistic attitude’. This indicates that the parents of Chinese hemophilic children generally attempted to enhance internal unity and stability among family members, which helped their families act as one entity in seeking and maintaining optimal treatment and care for their hemophilic children.
The five coping behaviors that the parents considered least helpful all belonged to the second subscale (‘seeking social support and maintaining self-esteem and psychological stability’). Parents of children suffering from a lifelong hereditary disease are commonly reluctant to inform people around them about their child’s disease, unless necessary. Thus, they rarely seek social support and help (Li et al., 2010a).
Conclusion
We investigated stressors and coping behaviors of parents with hemophilic children in China. Although possibly not true of all parents in China (we only surveyed approximately 160 parents), the results are likely representative of the current status of most parents with hemophilic children in China. The results can be of use to medical workers by providing them with greater understanding of parents’ coping behaviors and how to evaluate these behaviors. This would in turn allow medical workers to provide parents with greater support. Moreover, parents’ educational level and economic status will need to be considered by workers providing such support. Additionally, it should also be acknowledged that diverse problems will arise during each growth stage of hemophilic children; therefore, medical workers should evaluate those problems in time in order to guide parents in learning new coping behaviors and techniques before such problems occur (Bottos et al., 2007; Incledon et al., 2015). Finally, because the interviews were made by physicians of the Xiangya Hemophilia Care Center, the Hawthorne effect may have affected the results of the survey. Additionally, in the future, a larger sample survey will be conducted, increasing the generalizability of the results.
Footnotes
Declaration of conflicting interests
The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding
The author(s) disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: This study was funded by a grant from the Xiangya Hemophilia Care Center.
References
Supplementary Material
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