Journal of Genetic Counselin~ VoL 4, No. 3, 1995
Resilience in Siblings of Children with SicMe Cell Disease Charmaine D. Royal, 1,2,5 Verle E. Headings, 1,3 Eva T. Molnar, 3 and Franklin R. Ampy 4
This pilot study was conducted to identify factors responsible for promoting resilience in siblings of children with sickle cell disease. Twenty siblings (10-17 years of age) of children (5-13 years) with sickle cell disease were selected from the Pediatric Clinic of Howard University Center for Sickle Cell Disease. The siblings responded to questionnaires, and the data obtained was analyzed by chi-square for association. The results indicated that age, birth order, and gender had no effect on resilience in the siblings. However, family size, number of parents in the home, sibling's knowledge of the illness, degree of morbidity of the illness, socioeconomic status of the family, and parents' attitudes and childrearing practices were all found to affect resilience. These findings provide additional insight into the psychosocial aspects of, and genetic counseling for sickle cell disease, as well as for other chronic genetic disorders. KEY WORDS: siblings; children; sickle cell disease and trait; coping; resilience.
INTRODUCTION The Sibling Relationship
Cicirelli (1982) described the relationship between biological siblings as being unique among human relationships for three main reasons. 1Department of Genetics and Human Genetics, Howard University, Washington, D.C. 2Center for Sickle Cell Disease, Howard University, Washington, D.C. 3Department of Pediatrics and Child Health, Howard University, Washington, D.C. 4Department of Biology, Howard University, Washington, D.C. 5Correspondence should be directed to Charmaine D. Royal, Center for Sickle Cell Disease, Howard University, 2121 Georgia Avenue, N.W., Washington, D.C. 20059. 199 1059-7700/95/0900-0199507.50/1 © 1995NationalSocietyof GeneticCounselors,Inc.
200
Royal, Headings, Molnar, and Ampy
Siblings share a common genetic heritage, common cultural environment, and common early experiences within the family. Ninety percent of children have at least one sibling (Cicirelli, 1982). Childhood is usually a time when siblings are the closest physically and emotionally, sharing much of their living space, and probably spending more time together than with their parents (Kahn and Lewis, 1988). In view of the unparalleled relationship shared by siblings, it is not surprising that life events or crises that affect one child in the family inevitably affect the others. Chronic illness in a sibling undoubtedly qualifies as one of those circumstances that have great impact on the healthy siblings.
Coping with Chronic Disease
Approximately five million families in this country live with a seriously disabled child (Featherstone, 1980). Battle (1976) noted that chronic illness stresses and drains the child and every family member over an indefinite period of time. Most research on coping has centered primarily on families of the mentally ill and physically handicapped (Darling and Darling, 1982; Dyson, 1989; Gath, 1989; Grossman, 1972; Seligman, 1988; Sensky, 1982; Vine, 1982), and although many cases of mental retardation and physical disability are genetic in origin, researchers rarely incorporate the genetic element into their investigations. The feelings and emotions experienced by parents and siblings of individuals with chronic illnesses have been extensively researched (Battle, 1976; Costello, 1988; Darling and Daring, 1982; Featherstone, 1980; Fields, 1982; Gath, 1989; Grossman, 1972; Jablow, 1982; Patterson, 1991; Seligman, 1988; Sensky, 1982; Sourkes, 1987; Tritt and Esses, 1988; Vine, 1982; Wahl and Harman, 1989; Wilson, 1983). However, most of these studies focused almost exclusively on the negative aspects of coping. They noted that healthy siblings often experience jealousy, anger, embarrassment, loneliness, guilt, fear (of getting the illness), and over-protectiveness. In addition, many of them are called upon to help care for their disabled sibling, a responsibility which some are not prepared for, and which may cloud their vision of parenthood, making them almost afraid to have children of their own. Life with a disabled or seriously ill sibling is not all negative, however, as some of these and other investigations have suggested. Featherstone (1980), observed that many siblings, in spite of experiencing some negative effects from the situation, reported an increased tolerance for human difference, a less casual acceptance of the blessing of good health, and a sense of specialness concerning family bonds. Similarly, Seligman (1988) observed
Resilience in Siblings of Children with Sickle Cell Disease
201
that some college students with retarded siblings appeared to be more tolerant, compassionate, and more knowledgeable about prejudice. Some also reported involvement in the growth and development of their retarded sibling and exhibited a sense of pride that they had been a part of it. Tritt and Esses (1988) found that some siblings felt that they had developed more patience, understanding, sensitivity, and awareness about how to deal with someone who is sick. In light of such observations, Featherstone (1980) asked, "Why does the relationship with a handicapped brother or sister enhance the lives of some siblings while diminishing those of others? What makes the difference?" In addition, Leonard (1991) subsequently stated, "The study of siblings of chronically ill brothers and sisters would benefit from a change in perspective. Research in the last decade still framed studies from the perspective of vulnerability rather than resilience. Having benefitted from these studies, the study questions need to be reframed. Because childhood chronicity is a given, identifying risk factors for both able-bodied brothers and sisters as well as disabled children can reduce negative outcomes for these children and their parents. Many children are living successfully with disabled siblings, and their success should be the focus of current research so that appropriate interventions can be initiated." These are the questions and statements to which this investigation responds. Upon review of the often contradictory findings by investigators seeking to identify factors affecting coping in siblings, we hypothesized in this pilot study that resilience in siblings of children with sickle cell disease may be related to the age of the sibling, birth order of the sibling and patient, gender of the sibling and patient, family size, number of parents in the home, sibling's knowledge of the illness, degree of morbidity of the illness, socioeconomic status of the family, and the parents' attitudes and childrearing practices.
Sickle Cell Disease
Persons with sickle cell disease most commonly experience pain caused by blockage of the blood vessels. Some patients may experience painful episodes (crises) once a year or less frequently, while some may have as many as 15-20 episodes annually, requiring hospitalizations for several days at a time. The blood clots made up of sickle cells could lead to strokes, or may damage such vital organs as the heart, kidney, lungs, liver, or eyes. Other possible complications of the disorder include skin ulcers, gallstones, jaundice, spleen enlargement, problems in bone growth, and a variety of infections (Gaston, 1990).
Royal, Headings, Molnar, and Ampy
202
The illness affects not only the physical, but also the psychosocial wellbeing of afflicted individuals. In outlining the social and emotional aspects of sickle cell disease, Molnar (1977) indicated that the patients' work and/or play activities may have to be scaled to the general level of disability that the illness imposes on them. In addition, their schooling, work, or social ties may be periodically interrupted by fluctuations of the illness. Due to the unpredictability of sickle cell disease, family members live with the constant foreboding of another crisis or other complications of the illness, and their financial security may be threatened by recurring medical expenses. She further pointed out that during the school years, patients sometimes develop at a slower rate than their peers, and that many are teased for their small stature and jaundiced appearance of their eyes.
METHODOLOGY
Subjects The subjects were healthy siblings of children with sickle cell disease from Afro-American families in the Washington, D.C. metropolitan area who attend the Pediatric Clinic of the Center for Sickle Cell Disease at Howard University. Selection was based primarily on their ages (10-17 years) and the ages of their affected brothers or sisters (5-18 years). Families with more than one child with sickle cell disease were excluded. Given that no two individuals respond identically to circumstances, each qualified healthy sibling in each family was treated as an independent subject. Upon identifying 26 families which met the requirements for participation in the study, a letter providing a brief description of the project and a telephone number for them to call for additional information or to express their willingness to participate, was mailed to each family. After a week of not receiving any response (except for two returned letters as a result of the families' relocation), the investigator telephoned all parents whose telephone numbers were available (there were no numbers for four families), and all but four of the 20 families contacted agreed to participate. The sample consisted of 20 siblings from 16 different families (there were two subjects from each of four of the families). There were ten male and ten female siblings of ten male and six female children with sickle cell disease. Eight (40%) of the siblings had sickle cell trait. The mean age of the siblings was 12.5 years, ranging from 10-17 years of age, while that of the affected children was 9.95 years, ranging from 5-13 years of age. Twelve of the families were from the District of Columbia, six from Maryland, and two from Virginia.
Resilience in Siblings of Children with Sickle Cell Disease
203
Procedure
A questionnaire, administered verbally by the investigator, was used to collect the data. It was designed to test all nine hypotheses, and therefore, was comprised of questions regarding demographic characteristics of the family, as well as the adaptive and functional patterns of its members, particularly the siblings. The investigator decided to develop her own instrument because she was unable to locate a standardized questionnaire which adequately addressed all of the factors being studied (see Appendix). Part I of the questionnaire was answered by the parents, and contained questions about age and gender of each child, family size, number of parents in the home, socioeconomic status of the family, and the severity of sickle cell disease in the affected child. Part II was addressed to the siblings, with questions regarding their knowledge of sickle cell disease, feelings and attitudes toward their affected brother or sister, benefit(s), if any, from the experience, advice to other healthy siblings, and parents' attitudes and child-rearing practices. The questions concerning parents' attitudes and child-rearing practices were also included in the parents' questionnaire. The seemingly" positive bias of the questions asked is due to the fact that the primary goal of this project is to isolate factors that promoted resilience in the siblings. At each interview, the parent (the mother, in all except one case) was first given a consent form to read, ask questions, and sign, and each sibling received a preamble (a shortened and simplified version of the parent's consent document) to read and ask any questions. In an effort to maintain confidentiality for each participant and to prevent possible conflict, parents and siblings were interviewed separately. Information provided by the parents was not shared with the siblings, and information provided by the siblings was not shared with the parents. The investigator recorded the responses to minimize the risks of misinterpretation of questions or uneven reading proficiency. The majority (16) of the interviews involved home visits by the investigator, because that was most convenient for most families. The others (four) were done at the Sickle Cell Center.
RESULTS Upon completion of all interviews, the information provided by the questionnaires was used to determine the classification for each sibling within the dependent and independent variables. The dependent variable, resilience, was defined as the ability of siblings to draw strength from their situations, creating positive outcomes
204
Royal, Headings, Molnar, and Ampy
and a variety of opportunities. It was measured in terms of the siblings' feelings; profit from their experience; advice to other siblings; complaints about the affected brother/sister or parent(s); and the investigator's clinical impression. Each of the five factors was rated as being "high" or "low" for each sibling. Siblings who had more positive than negative feelings (questions 8-25--Part II), stated some profit from their experience (question 32--Part II), had some advice for other siblings (question 33--Part II), and had minor or no complaints were rated as "high," while the others were "low." The investigator's clinical impression was one of the five quantified measures, and was not used to break an otherwise borderline result. In other words, it was determined immediately upon completion of each interview, and not after rating the other four factors. The investigator recorded her impression as "high" or "low," based on the sibling's responses and questions, mannerisms, interaction with the parents and affected child, and parents' reports about the sibling. Siblings receiving a rating of "high" for more than three factors were classified as very resilient, those with two or three "highs" were classified as fairly resilient, while those with less than two "highs" were classified as not resilient. However, only two of the three levels of resilience occurred in the study. There were no siblings who were not resilient--they were either fairly resilient or very resilient, and the frequencies were 6 and 14, respectively (Table I). The independent variables, displayed in Table II, were defined as follows: Age: low--siblings between 10 and 13 years of age; high--siblings between 14 and 17 years of age. Birth order: older--sibling older than patient; younger--sibling younger than patient. Gender: male~male--male sibling of male patient; male~female--male sibling of female patient; female~male--female sibling of male patient; female~female-female sibling of female patient. Family size: small--less than four children in the family; large--more than three children in the family. Number of parents: single--only one parent in the home; both--both parents in the home. Knowledge (questions 1-6--Part II): lack--siblings who answered the questions incorrectly or did not know basic information about the disease; adequate--ability to answer the questions and/or provide additional information about the disease.
Resilience in Siblings of Children with Sickle Cell Disease
205
Table I. Classification of Dependent Variable: Resilience Score* Degree of resilience
Subjectno.
High
Low
1 3
5 4
0 1
4
5
0
5 6 7 8 9 10 12 13 16 17 20
4 4 4 5 4 5 5 5 5 5 5
1 1 1 0 1 0 0 0 0 0 0
Very resilient
14 = 70% 2 11 14 15 18 19
Fairly resilient
Frequency
3 2 3 2 3 2
2 3 2 3 2 3 6 = 30%
*Determined by rating these factors: feelings, profit, advice, complaints, clinical impression.
Morbidity ( q u e s t i o n 9 - - P a r t I): mild--siblings w h o s e a f f e c t e d brother/sister had no serious complications and/or was hospitalized less than five times per year; severe--complications such as: chest syndrome, stroke, pneumonia, transfusions, and/or five or m o r e hospitalizations per year. S o c i o e c o n o m i c status: upper--families with private insurance or H M O ; lower--families with Medicaid or no insurance. P a r e n t s ' attitudes and child-rearing practices (questions 2 6 - 3 0 - - P a r t II): good--more than two positive responses to the related questions; poor--less than three positive responses to the questions. The distributions of both the dependent and independent variables were then combined and charted to reveal the frequency of subjects who were very resilient, and those who were fairly resilient within each subgroup (Table III). As indicated, 90% of the siblings in small families were very resilient. Similarly, 90% of those with adequate knowledge were very resilient. It is also interesting to note that of the six siblings in single-parent
206
Royal, Headings, Molnar, and Ampy Table II. Classification of Independent Variables
Variable
Absolute freq.
Relativefreq. (%)
Age low high
14 6
70 30
Birth order older younger
15 5
75 25
7 3 4 6
35 15 20 30
Family size small large
11 9
55 45
No. of parents single both
6 14
30 70
Knowledge lack adequate
9 11
45 55
Morbidity mild severe
16 4
80 20
S.E.S. upper lower
9 11
45 55
Parents' attitudes good poor
18 2
90 10
Gender male/male male/female female/male female/female
families, only one was very resilient, while of the 14 with both parents, only one was fairly resilient. Other observations are that all of the siblings of upper socioeconomic status were very resilient, whereas all of those who were fairly resilient were of lower socioeconomic status. Likewise, all of the siblings who were very resilient felt that their parents' attitudes and child-rearing practices were good, while all of those who felt that their parents' attitudes and childrearing practices were poor were fairly resilient. In order to determine the statistical correlations between resilience and the selected factors, chi-square for association was applied to the data in Table III. The results, presented in Table IV, indicate that age, birth
Fairly Resil.
Very Resil.
Degree of resil.
X
P12S15
Total
5
X
X
X
P12S14
P14S18 P15S19
X
1
X
3
X
X
X
12
9
5
x
x
x
x
x
x
x
x
x
x
x
x
x
x
x
x
x
x
x
x
x
x
x
x
x
3
X
X
X
2
x
x
Old Young
Low High
x
Birth order
Age
P10Sll
P2S2
Total
PISi P3S3 P4S4 P5S5 P5S6 P6S7 P7S8 P8S9 P9S10 PllS12 PllS13 P13S16 P13S17 P16S20
no.
Subject
2
X
X
5
x
x
x
x
x
1
2
X
×
x
1
X
3
x
x
x
1
X
5
x
x
x
x
x
MM MF FM FF
Gender
1
X
10
X
X
X
X
X
X
X
X
X
X
5
X
X
X
X
X
4
X
X
X
X
Small Large
Family size
5
X
X
X
X
X
1
X
1
X
13
X
X
X
X
X
X
X
X
X
X
X
X
X
Single Both
No. of parents
5
X
X
X
X
X
4
X
X
X
X
1
X
10
X
X
X
X
X
X
X
X
X
X
Lack Adeq.
Knowledge
Table 1II. Interaction Between Dependent and Independent Variables
3
x
x
x
13
x
x
x
x
x
x
x
x
x
x
x
x
x
1
3
x
x
x
x
Mild Severe
Morbidity
0
9
x
x
x
x
x
x
x
x
x
6
x
x x
x
x
x
5
x
x
x
x
x
Upp. Low
SES
4
x
x
x
x
14
x
x
x
x
x
x
x
x
x
x
x
x
x
x
2
x
x
0
Good Poor
Parent attitude
g
F
G~
¢3
=.
g
208
Royal, Headings, Molnar, and Ampy
Table IV. HypothesisTesting Using Correlations Between Dependent and Independent Variablesa Hypothesis 1 2 3 4 5 6 7 8 9
Correlation with dependent Independentvariable variable(chi-square) Age Birth order Gender Family size No. of parents Knowledge Morbidity S.E.S. Parents' attitudes
0.726 2.857 0.952 5.089* 11.610"** 5.089* 4.821" 7.013"* 5.185"
Confirmed Yes
No X X X
X X X X X X
aN = 20. *p < 0.05. **p < 0.01. ***p < 0.001.
order, and gender were not significantly related to resilience in the siblings. On the other hand, family size, number of parents, knowledge of the illness, degree of morbidity, socioeconomic status, and parents' attitudes and childrearing practices all influenced resilience. The number of parents in the home and the socioeconomic status of the family showed the strongest relationship with resilience in the siblings. The study provided other useful data which give rise to some meaningful implications. Forty percent of the siblings had sickle cell trait. However, they did not differ from the other siblings in their resilience indicators (chi-square = 0.586, p > 0.3). The questions regarding parents' attitudes and child-rearing practices were also asked of the parents in order to compare their responses with those of the children. Overall, there was no significant difference between the responses from each group (chi-square = 0.084, p > 0.3). In addition to determining the relationship between resilience and each independent variable, various pairs of independent variables were also tested for correlations between them. Upon visual observation of the data in Table III, it seemed apparent that both number of parents in the home and family size would be associated with socioeconomic status. However, the .,:suits indicate that family size was significantly related to socioeconomic status (chi-square = 5.130,p = 0.025), but the number of parents was not (chi-square = 2.780, p = 0.092). When the age of the siblings was tested for correlation with their knowledge of sickle cell disease, none was found (chi-square = 2.780, p = 0.092), i.e., the siblings in the high age range did not necessarily know more about sickle cell disease than those in the low age range.
Resilience in Siblings of Children with Sickle Cell Disease
209
DISCUSSION
The findings and implications of this study have generated a number of issues for discussion. Three of the nine hypotheses tested were not confirmed, i.e., age, birth order, and gender. These results contradict the findings of Leonard (1991) who suggested that the age of the sibling affected coping, and those of Lobato et al. (1988) and Breslau (1982) who reported that birth order affected coping. The findings regarding gender are contrary to those of Darling and Darling (1982); Leonard (1991); Lobato et al. (1988); and Seligman (1988), all of whom suggested that sisters of affected children have more difficulty in coping than brothers, primarily because they are more often called upon to assume care-giving roles and to assist with other household chores. The contradiction between the results of this study and those of other studies may be accounted for by cultural differences between the populations studied. As previously noted, the subjects in this study were all Afro-American. In general, siblings (older and younger, male and female) in Afro-American families are taught and encouraged by their parents, to take care of one another, and are given various responsibilities in the home. Therefore, whenever, one sibling is ill, it is natural for the others to become involved and assist as much as is necessary. The siblings in the study gladly participated in caring for their brother or sister with sickle cell disease. A 12-year-old sister, when asked about helping to care for her 8-year-old brother, responded, "I love my brother, and I love to take care of him, whether or not he is sick. He is my brother." Family size, number of parents, knowledge, morbidity, socioeconomic status, and parents' attitudes and child-rearing practices were all significantly related to coping in the siblings. Gath (1974) suggested that siblings cope better in small families, whereas Grossman (1972) found better coping in large families. Siblings coping better in small families may result from closer bonding and from parents being able to give more individual attention to each sibling. Lobato et aL (1988) suggested that the parent-parent relationship influences coping in siblings, and should also be included in investigations such as this. Their clinical impression was confirmed in this study, given that siblings raised by both parents were more resilient than siblings raised by a single parent. In fact, this variable proved to be the most significant in its relationship with resilience. A noteworthy observation is that of the six siblings who were fairly resilient, five were from large families, and four of those five had single parents. On the other hand, the four siblings from large families who were very resilient, all lived with both parents. It seems reasonable to presume
210
Royal, Headings, Molnar, and Ampy
that a large family has a greater need for both parents. Therefore, it was to be expected that the siblings in large families with single parents would be less resilient. Consistent with studies done by Tritt and Esses (1988), siblings who had adequate knowledge about sickle cell disease coped better than those who lacked knowledge, and the extent of knowledge and understanding was not significantly related to age. The siblings who had adequate knowledge seemed to be less anxious and worried, and their ages were varied. While Dyson (1989) found that siblings of children with highly morbid illnesses do not adapt as well as siblings of children with less morbid illnesses, Grossman (1972) found that the severity of the illness was not a significant factor in adaptation. The results of this study support Dyson's findings. Of the four siblings of children with severe sickle cell disease, only one was very resilient. This suggests that even though sickle cell disease is not a very morbid disease compared to mental retardation or physical disability, its effects on siblings are noticeable. Socioeconomic status was the second most significant variable in the study. Similar to the findings of Seligman (1988), the results suggested that siblings in families of lower socioeconomic status have more difficulty coping than those of higher socioeconomic status. In this study, all six of the siblings who were fairly resilient were from families of lower socioeconomic status, and although the statistical analysis showed a lack of correlation between number of parents and socioeconomic status, these siblings may have already been at risk for poor adaptation because most of them were from large families with single parents. Apparently, the more financially secure a family is, the better able are the parents to adequately provide for their children, both physically and emotionally. The attitudes and child-rearing practices of the parents had a definite impact on coping in the siblings, as was also observed by Trevino (1979) and Seligman (1988). All of the siblings who were very resilient believed that their parents had good attitudes and child-rearing skills. Only two siblings in the entire sample thought that they were poor. Overall, the parents of the siblings in this study seemed to be excellent with their children. This uniformity of parenting may reflect the quality of social service follow-up with these families at the Center for Sickle Cell Disease, and may not be representative of the universe of families coping with sickle cell disease. When asked about their attitudes and child-rearing practices, the general response from the parents was that they tried to treat the children equally, and endeavored not to dwell on the illness. This was evident in the siblings. In fact, one 12 year-old child stated, "My mother treats us the same, and I see my brother as an ordinary person with no disease."
Resilience in Siblings of Children with Sickle Cell Disease
211
As was previously shown, there was no significant difference between the responses of the siblings and those of the parents. Equally impressive, is the fact that the siblings in each of the four pairs from the four families in which two siblings were interviewed, coped alike. Considering that each interview was done separately, these findings certainly indicate congruence between parents and children, and also between the siblings themselves. This demonstration of cohesiveness, supportiveness, and closeness within the families is unparalleled, and deserves notice. Another very interesting finding was the lack of any significant difference between siblings with sickle cell trait and those without the trait. It seemed reasonable to expect that siblings with the trait would worry more about the illness, especially in terms of having their own children. However, their responses were quite contrary to the expectations of the investigator. This study did not gather data which could distinguish cultural variables that might account for this. A 14-year-old sister, when asked if she worries about having a child with sickle cell disease, said, "No, I don't worry about it, because even if it happens, I think I could handle it after watching my mother take care of my brother." Undoubtedly, that is a very positive and mature attitude to have, especially when the illness is genetic, and, as noted by Costello (1988), genetic conditions are permanent, chronic, complex, relate to family members, evoke labeling, and threaten the lifestyle and well-being of the entire family. However, it really is unproductive to worry and become anxious about the situation, because unless one chooses a mate without the trait, elects not to have children, or terminates a pregnancy involving an affected fetus, there is nothing that one can do, except utilize the resources available for care and management, in the best interest of the child and the family. Perhaps the most intriguing aspect of the interviews was listening to the siblings' responses to the two final questions on the questionnaire. When asked what they had gained from the experience of having a brother or sister with sickle cell disease, most siblings replied that they had become more patient, compassionate, understanding, helpful, responsible, and sensitive. These benefits are similar to those observed in other siblings by Seligman (1988) and Tritt and Esses (1988). The investigator was even more impressed, however, by those siblings who also reported that they had become more aware of, and knowledgeable about sickle cell disease. This benefit had not been identified by any of the studies previously reviewed. It certainly is commendable that acquiring knowledge about their brother's or sister's illness was important enough to them, for them to view it as being profitable. The question regarding advice to other siblings of children with sickle cell disease or other illnesses, produced far more varied responses. They
212
Royal, Headings, Molnar, and Ampy
encouraged other siblings not to worry, but to think positively, and become involved in other activities to keep their minds off the situation. They also urged them to be patient, understanding, and helpful, and not be jealous. Some of them gave such valuable advice, that they are worth quoting. Try to boost their image. Don't let them feel left out or different.--17-year-oId sister of a 13-year-oldboy It's O.K. to get sad, but don't blame yourself.--10-year-oldsister of a 13-year-old girl Your parents need to pay more attention to them, so don't get jealous. Pray for them.--12-year-old brother of a 10-year-oldboy Don't blame your parents because it is not their fault in most cases (except fetal alcohol, etc.).--17-year-old sister of a 12-year-oldgirl Put yourself in their place.--12-year-oldbrother of a 10-year-oldboy Be nice to them because they may feel that they are different because they have an illness or disease.--ll-year-old sister of a 12-year-oldgirl
LIMITATIONS OF STUDY There are some limitations which should be noted. First, due to the fact that this was a pilot study, the sample size was relatively small. This, together with the fact that these families are well-served by a social worker at the Center for Sickle Cell Disease, makes it unlikely that our sample was fully representative of the universe of families coping with sickle cell disease. With regard to the interviews, it is likely that some subjects refrained from talking about problems in the family, and gave expected answers to some of the questions asked. This could be because they did not want the investigator to know that there were family problems, and/or they could have felt that the investigator would share their responses with their parents. Perhaps the most evident limitation is the general lack of morbidity of sickle cell disease in the patients whose siblings were interviewed. In contrast to the previous studies on mental retardation and physical disability, the patients in this study had virtually no visible signs of illness. Therefore, there are some feelings and emotions (e.g., embarrassment) that the siblings were not likely to experience as a result of the illness in the family. This, as well as the previous limitation, may have been major contributors to the outcome of no subject being rated as not resilient.
Resilience in Siblings of Children with Sickle Cell Disease
213
RECOMMENDATIONS In spite of its limitations, this study has meaningful implications for the establishment of support groups and programs for siblings and parents of children with serious illnesses, and for future research. Interestingly, the three variables (age, birth order, and gender) which were not significantly related to resilience in the siblings, are all factors over which one has no control, whereas, all of the six which were significant, can, to some degree, be influenced by the individuals involved. Consideration should be given to the establishment of programs and support groups for siblings and their parents, with special emphasis on family size, number of parents, knowledge (including management) of the illness, socioeconomic status, and parents' attitudes and child-rearing skills. These programs should include counseling/educational sessions for the parents regarding family planning, parent-parent relations, and parental attitudes and child-rearing practices. In relation to socioeconomic status, information on adult education and employment opportunities should be provided, especially for young, single parents. These programs should also include sessions where the siblings can be informed about the particular illnesses in their families. Some parents may also need to attend these sessions, because in many instances, they lack knowledge about the illness, and are therefore unable to share the information with their children, or to answer their questions. Information concerning management of the illness should also be provided for the parents, because in some cases the morbidity of the illness could be decreased if parents did their part in adhering to the established and recommended protocol for care of their affected child. In children with sickle cell disease, for example, some parents fail to administer prophylactic penicillin as prescribed by physicians. Consequently, some of these children develop life-threatening infections. More support groups also need to be established, so that siblings of children with serious illnesses can interact with one another. This will enable them to talk about their thoughts and feelings with persons who have life experiences in common with them. The implication for future research is that more investigations need to focus on the resilience (instead of deficiencies) in siblings of children with serious illnesses and disabilities (genetic and otherwise), and to highlight characteristics and supportive programs that promote resiliency. These studies will benefit not only the siblings themselves, but the entire society. In fact, there is much that can be learned from these siblings.
214
Royal, Headings, Molnar, and Ampy APPENDIX
I. Q U E S T I O N N M R E
Part I 1. 2. 3. 4. 5.
Age of healthy sibling Age of child with sickle cell disease Gender of healthy sibling Gender of child with sickle cell disease How many children (of either parent or both parents) are there in the family (those that live in the home with the affected child)? 6. Do both parents raise the children together in the same home, or are they raised by a single parent? 7. Is either the sibling or the child with sickle cell disease in any type of special education program? 8. What grades are the children in, and how are they doing in school? (a) The child with sickle cell disease (b) The healthy sibling(s) 9. What complications of sickle cell disease has the affected child experienced? How frequently (times per year) has he/she been hospitalized?
10. What is your highest level of formal education? _ _ Less than high school _ _ Trade or vocational school _ _ Some high school _ _ College degree _ _ High school graduate _ _ Graduate/professional degree _ _ Some college _ _ Post-graduate work 11. What is your occupation? (a) Mother b) Father 12. What type of health insurance do you have? (a) Medicaid (b) HMO (c) Private insurance (d) None Do you (key: H--healthy child; S--sick child): 13. punish H more severely than S? (a) Never (b) Sometimes (c) Always 14. give H more chores and responsibilities than S? (a) Never (b) Sometimes (c) Always 15. listen to S more than you do to H? (a) Never (b) Sometimes (c) Always 16. give S more gifts, toys, etc. than H? (a) Never (b) Sometimes (c) Always 17. talk with H about S's illness? (a) Never (b) Sometimes (c) Always
Resilience in Siblings of Children with Sickle Cdl Disease
215
Part II 1. Do you sometimes worry about getting your brother's/sister's illness? (a) Yes
(b) No 2. Do (b) 3. Do 4. Do
you think that your brother/sister will be cured of sickle cell disease? (a) Yes No you ever worry about having a child with sickle cell disease? (a) Yes (b) No you ever wonder if you did something to make your brother/sister sick? (a) Yes
(b) No 5. Do you think that your parents did something that caused your brother/sister to become sick? (a) Yes (b) No 6. What do you know about sickle cell disease?
7. Who takes care of your brother/sister when he/she becomes ill? (a) The doctor (b) You (c) Your parent(s) (d) Other siblings or relatives
8. 9. 10. 11. 12. 13. 14. 15.
When your brother/sister is sick, how do you feel? Never Angry Frightened Sad Guilty Jealous Embarrassed Protective of him/her Neglected
16. 17. 18. 19. 20. 21. 22. 23. 24. 25.
How do you feel toward or about your brother/sister with sickle cell disease? Never Sometimes Always Compassionate Patient Distressed Resentful Sensitive Ashamed Understanding Tolerant Involved Proud
Sometimes
Always
Do your parents: 26. punish you more severely than your affected brother or sister? (a) Never (b) Sometimes (c) Always 27. give you more chores and responsibilities than your affected brother/sister? (a) Never (b) Sometimes (c) Always 28. listen to your affected brother/sister more than they do to you? (a) Never (b) Sometimes (c) Always 29. give him/her more gifts, toys, etc. than they give to you? (a) Never (b) Sometimes (c) Always 30. talk with you about your brother's/sister's illness? (a) Never (b) Sometimes (c) Always
216
Royal, Headings, Molnar, and Ampy 31.
Who in the family do you think is most unhappy because of your brother's/sister's illness? (a) Your affected sibling (b) Your parents (c) Your other brothers and sisters
(d) You 32.
What have you gained from the experience of living with a brother/sister with sickle cell disease?
33.
What advice would you give to other children who have a brother/sister with sickle cell disease or other chronic illness?
ACKNOWLEDGMENTS
The authors thank the staff of Howard University Center for Sickle Cell Disease for their invaluable support and assistance throughout the project. Special thanks also to the 16 families who so willingly participated.
REFERENCES Battle CU (1975) Chronic physical disease: Behavioral aspects. Pediat Ctin N Am 22(3):525531. Breslau N (1982) Siblings of disabled children: Birth order and age-spacing effects. JAbnorm Child Psychol 10:85-96. Cicirelli VG (1982) Sibling influence throughout the lifespan. In: Lamb ME, Sutton-Smith B (eds) Sibling Relationships: Their Nature and Significance Across the Lifespan. New Jersey: Lawrence Earlbaum Associates, pp 268-269. Costello AJ (1988) The psychosocial impact of genetic disease. In: Genetics Applications: A Health Perspective. Kansas: Learner Managed Designs, pp 149-166. Darling J, Darling RB (1982). Children Who are Different: Meeting the Challenges of Birth Defects in Society. St. Louis: The C.V. Mosby Company. Dyson LL (1989) Adjustment of siblings of handicapped children: A comparison. J Pediat Psychol 14(2):215-229. Featherstone H (1980) A Difference in the Family: Life with a Disabled Child. New York: Basic Books. Fields G (1982) Social implications of long-term illness in children. In Downey JA, Low NL (eds) The Child with Disabling Illness: Principles of Rehabilitation. New York: Raven Press, pp 595-610. Gaston M (1990) Sickle Cell Anemia. Medicine for the Public. U.S. Department of Health and Human Services. National Institutes of Health Publication No. 90-3058. Gath A (1974) Sibling reactions to mental handicap: A comparison of the brothers and sisters of mongol children. J Child Psychol Psychiat 15:187-198. Gath A (1989) Living with a mentally handicapped brother or sister. Arch Dis Childhood 64:513-516.
Resilience in Siblings of Children with Sickle Cell Disease
217
Grossman FK (1972) Brothers and Sisters' of Retarded Children: An Exploratory Study. New York: Syracuse University Press. Jablow MM (1982) Cara: Growing with a Retarded Child. Philadelphia: Temple University Press. Kahn MD, Lewis KG (1988) Siblings in Therapy. New York: W.W. Norton and Co. Leonard BJ (1991) Siblings of chronically ill children: A question of vulnerability versus resilience. Pediat Ann 20(9):501-506. Lobato D, Faust D, Spirito A (1988) Examining the effects of chronic disease and disability on children's sibling relationships. J Pediat Psychol 13(3):389-407. Molnar, ET (1977) Socio-economic factors in the lives of patients with sickle cell disease. In: Handbook for Genetic Counseling in Hemoglobinopathies. Washington, D.C.: Howard University Printing and Reproduction, pp 94-106. Patterson JM (1991) A family systems perspective for working with youth with disability. Pediatrician 18(2):129-141. Seligman M (1988) Psychotherapy with siblings of disabled children. In: Kahn MD, Lewis KG (eds) Siblings in Therapy. New York: W.W. Norton and Co., pp 167-189. Sensky T (1982) Family stigma in congenital physical handicap. Brit Med J 285:1033-1035. Sourkes BM (1987) Siblings of the child with cancer. Candlelighters Childhood Cancer Foundation 7(3/4):9-i2. Trevino F (1979) Siblings of handicapped children: Identifying those at risk. Soe Casework 60:488-493. Tritt SG, Esses LM (1988) Psychosocial adaptation of siblings of children with chronic medical illnesses. A m J Orthopsychiat 58(2):211-219. Vine P (1982) Families in Pain: Children, Siblings, Spouses, and Parents of the Mentally Ill Speak Out. New York: Pantheon Books. Wahl OF, Harman CR (1989) Family views of stigma. Schizo Bull 15(1):131-138. Wilson GN (1983) Counseling parents of children with genetic disorders. Ross Timesaver 25(4):13-16.