588215 research-article2015
HPQ0010.1177/1359105315588215Journal of Health PsychologyBrown et al.
Article
Family caregiving for adults with sickle cell disease and extremely high hospital use
Journal of Health Psychology 1–10 © The Author(s) 2015 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav DOI: 10.1177/1359105315588215 hpq.sagepub.com
Shan-Estelle Brown1, Daniel F Weisberg2 and William H Sledge1
Abstract This study investigated coping with chronic illness in the adult patient–caregiver relationship for sickle cell disease, marked by debilitating acute and chronic pain. One-on-one interviews (N = 16) were conducted with eight primary caregivers of eight adults with extremely high hospital use, severe sickle cell disease with hospital admissions several times monthly over successive years. Caregivers were predominantly parents; two were romantic partners. Caregivers attributed disruptions to the disease’s variability, tensions in how much support to give, and adults’ inability to fulfill parental obligations. Both groups expressed fears of patients’ increasing age, declining health, and early death. Targeted counseling and resilience training is recommended.
Keywords adults, chronic illness, coping, pain, social support
Introduction The act of caregiving can be a source of joy and bring families together, provided that caregivers have adequate information, social and financial support, and support from medical staff (Ohaeri and Shokunbi, 2002). Caring for a person affected by sickle cell disease (SCD), however, can precipitate “chronic sorrow” (Northington, 2000), the emotional distress that caregivers of people with SCD may experience. SCD is complex due to its clinical variability (Ballas et al., 2012), treatment as a racialized disease (Bediako and Moffitt, 2011; Rouse, 2009), as well as financial stress and possibly daily disruptions in family interactions (Brown et al., 2010). SCD is an inherited hemoglobinopathy known for its acute painful vaso-occlusive crises and chronic pain
(Ballas et al., 2012; Brousseau et al., 2010). Although most individuals manage crises at home (Smith et al., 2008), a small minority of patients are admitted as often as several times per month, over successive years (Carroll et al., 2009; Shankar et al., 2005), a phenomenon called extremely high hospital use (EHHU).
1Yale
School of Medicine, USA and Women’s Hospital, USA
2Brigham
Corresponding author: Shan-Estelle Brown, Department of Internal Medicine, Yale School of Medicine, 135 College Street, Suite 323, New Haven, CT 06510-2283, USA. Email: [email protected]
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The purpose of this study is to understand the interaction between adult patients with EHHU and their family caregivers. Previous studies on EHHU found that SCD patients with EHHU demonstrated increased hospitalization and opioid use from a young age, which perpetuated missed developmental milestones and a cycle of increasing hospitalization (Weisberg et al., 2013) and difficulty negotiating pain management with medical providers (Brown et al., 2014). Although most literature presents such difficulties for pediatric and adolescent patients (Tsao et al., 2013), generations of patients with SCD are living into adulthood, which present different challenges related to stress (Porter et al., 2000) and stigma (Bediako et al., 2014), different actors (e.g. spouses), and added complexity in the patient–caregiver relationship. Caregivers are problem-solvers (Barakat et al., 2013) who are frequently responsible for the education and home care of children with SCD, but this role often comes with a cost (Beyer and Simmons, 2004; Hoyt Drazen et al., 2014; Wonkam et al., 2014). Caregivers of patients with SCD had a significantly lower quality of life on all subscales of depressive moods, daily activities, and vitality compared to non-caregivers (Van Den Tweel et al., 2008). Compared with other caregivers to individuals with chronic illnesses, caregivers of children with SCD experience higher depressive mood scores even if the caregiving burden is similar (Moskowitz et al., 2007). Caregiving affects the entire family, as grandparents may additionally act as caregivers and siblings of affected children also encounter stress and possible negative psychosocial outcomes (Dilworth-Anderson, 1994; Gold et al., 2011; Graff et al., 2012). As caregivers are predominantly mothers (Hill, 1994; Sharpe et al., 1994), fear of their children’s deaths, separation anxiety, and helplessness blunt their ability to care (Burnes et al., 2008). This study attempts to fill several gaps in the literature of SCD, caregiving, and chronic illness. First, literature on SCD and caregivers depicts caregivers primarily as parents caring
for sick children. This study intends to expand the picture of caregiving to include nonparental roles, such as spouses, when the affected person is an adult. Second, the majority of articles on adult sickle cell care focus on the clinical transition from pediatric to adult SCD care, not what happens afterward. When an adolescent patient transitions from pediatric to adult care, the number of emergency department visits increases (Hemker et al., 2011). At this time, family caregivers are especially concerned about medication adherence and adult providers’ lack of familiarity with their child’s needs (Porter et al., 2014; Telfair et al., 1994). As recent research suggests that younger and older adults have different coping strategies (Sanders et al., 2010), the experiences of adults with SCD who have completed this transition need further investigation. Third, SCD is frequently described as an acute disease and the chronic illness component is less understood. This study uses a framework of illness uncertainty for chronic disease, which affects illness experiences and communication between family members to understand the unpredictability of SCD pain (Checton et al., 2012; Johnson Wright et al., 2009). This study attempts to fill these gaps by characterizing illness experiences for individuals with severe chronic illness from SCD and the relationships to their caregivers who are often their parents and grandparents to the individuals’ children.
Method Studies of caregivers have often involved the development of coping models (Brown et al., 2000), quantitative survey instruments to assess caregiver quality of life and stressors (Karlson et al., 2012; Schlenz et al., 2012), and focus group studies (Neely-Barnes et al., 2010; Porter et al., 2014). This study employed a qualitative study design of one-on-one, face-to-face interviews with patients with EHHU and their caregivers. EHHU was defined as at least 100 hospital days/year over each index 12 months. Patients were first identified as having EHHU and SCD through the Yale-New Haven Hospital
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Brown et al. Resource Information Management System for the period 1 January 2008 to 31 December 2010. Chart review further compared patients’ clinical histories for similarities in disease profile, complications, and medication regimens. Participants consisted of 16 individuals—8 SCD patients with EHHU (“high hospital-utilizing,” “EHHU”) and their primary caregivers, whom patients identified as the family member, significant other, spouse, or friend, most important to their care. Individuals identified were approached by telephone or in person; two individuals refused to participate. Table 1 presents the patients’ age, gender, and hemoglobinopathy type, as well as relationship to the caregiver. All the affected adults were unemployed. Five of the adults were parents. Demographic information was not collected on the caregivers.
Interviews An interview guide was created with openended questions to address the following themes: (1) disease, pain, and medication; (2) hospitalization; (3) support structures; (4) daily life; and (5) personal relationships. Family caregivers were asked to describe their role in caring for the patient, improvements, and setbacks as the disease has progressed, the evolution of their relationship, and helpful and harmful ways that the patients cared for themselves. Affected adults were interviewed by DW at the hospital. Family caregivers were interviewed by DW and a second interviewer; four interviews were conducted in research buildings near the hospital and four were conducted at the caregivers’ homes. Interviews lasted approximately 1 hour and were conducted one-on-one. Interviews were audio-recorded in private settings and transcribed by a professional transcriptionist. Participants were compensated US$25 for a completed interview.
Analysis Analysis began with pilot testing of the interview guide and creation of narrative summaries of the interviews (Weisberg et al., 2013).
Interviews were also coded by content analysis using Atlas.Ti software (Saldaña, 2009). Similarities in words and phrases were grouped together as codes, and the codes representing themes were linked to make broader inferences about participants’ experiences. We generated themes from the interview guide, and two coders (SB, DW) identified the themes in the transcripts. The entire research team met regularly to discuss themes and saturation. An audit trail to track emergence of new codes and progress of coding was maintained throughout. The research protocol was approved by the Human Investigation Committee at Yale School of Medicine, New Haven, CT.
Results The analysis identified differences in caregivers’ and patients’ descriptions of the care that caregivers provide; caregivers described difficulty balancing patients’ independence with limitations due to SCD and how they also care for the affected persons’ children; both groups expressed fear of the patients’ death.
Care that family caregivers provide Interviews with the caregivers reflected empathy about the person they cared for, but they described different kinds of support. Forms of support included rides to appointments, household routines, strength and encouragement, bringing food to the hospital, cooking, shopping, hospital visits, and errands. Patients described several people in their support network, not only the caregiver we interviewed. Patients broadly described the support they received: My aunts, they support me sometimes, but not all the time. Most of my support comes from my grandparents and my daughter. In each and every way they care for me. They come and visit me. If I don’t have the money to get what I want, they give it to me. They take care of me. (S1)
Only one sickle cell patient, S4, considered the support reciprocal:
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34 27 26 34 37 25 24 32
F F M M F F F M
Yes No No No Yes Yes Yes Yes
HbSS HbSS HbS-B thalassemia HbSC HbSS HbS-B thalassemia HbSS HbSC
SA + LA SA SA + LA SA + LA SA + LA SA + LA SA SA + LA
Yes No No Yes No Yes Yes No
PH AChS AVN CKD AChS AVN PH AChS PH AVN AChS None identified AVN PH AChS AVN PH AChS IO
171 263 151 111* 202 104 123 94
Stepdaughter Younger sister Romantic partner Son Daughter Daughter Grandniece Romantic partner
Average Relationship to hospital days caregiver 2008–2010
F1 F2 F3 F4 F5 F6 F7 F8
Caregiver ID
SA: short acting; LA: long acting; EHHU: extremely high hospital use. HbSS: homozygosity for the sickle cell gene (HBB glu6val), sickle cell anemia; HbSC: heterozygosity for the sickle cell gene (HBB glu6val) and the hemoglobin C gene (HBB glu6lys), sickle-hemoglobin C disease; HbS-B thalassemia: heterozygosity for the sickle cell gene (HBB glu6val) and one of the B-thalassemia gene mutations. AVN: joint avascular necrosis; PH: pulmonary hypertension; CKD: chronic kidney disease; AChS: acute chest syndrome; IO: iron overload. *Pt spent 1 year incarcerated—not included in calculation of average hospital days.
EHHU S1 Patients S2 S3 S4 S5 S6 S7 S8
Patient Age Sex Is a Hemoglobinopathy Outpatient opioid Hydroxyurea Major SCDID parent? regimen (short related acting/long acting) complications
Table 1. Patients’ clinical and demographic characteristics.
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Brown et al. If I need something, or if [my mother] can help me with something, she can help me. And it goes both ways. Like I help her, right now, she going through her sickness. I’ll help her out, like she do dialysis at home, I help her out. Just stuff like, carry the boxes upstairs, carry the bags upstairs, or whatever. Go over there and clean her house up and stuff. Take the trash out. And if she could help me with something, she helps me. (S4)
Caregivers used different kinds of knowledge to provide care. One caregiver searched the internet and “read everything she could get her hands on” (F2) while others relied on their often life-long experience with caring for a child with SCD, emphasizing “because I’m her mother!” (F5). One caregiver felt that the care she provided to her sister was limited, compared to the hospital’s services: I think it’s actually to the point where she might feel she has to be here [at the hospital]. [My sister thinks,] “This is the only place that’s gonna take care of me. They’re gonna take care of me better than my family can.” (F2)
As part of their care, two caregivers advised their patients on managing hospital staff: I don’t think he says anything. Especially as far as his care is concerned. Let them know how he feels, or what’s going on. Sometime you gotta be aggressive. He’s pretty much laid back. He won’t say anything. (F4) Although right or bad, if they don’t do the right thing, she gets on them. I told her to be nice to the doctors and nurses, and they’d be nice to her. I can’t tell them what to do. All I know is I told her to be nice to people. (F7)
wife, she would have to clean up. You know what I mean? Because I was telling my wife, make her do something. Don’t let her just sit on the couch. When she’s at grandma’s she doesn’t have to do anything. (F1)
Work was a recurring theme in the interviews with caregivers, as the majority of the EHHU patients interviewed are in the hospital too many consecutive days to work regularly. Despite the fact that caring for a person with SCD interrupted caregivers’ own employment, caregivers still wanted adult patients to work: I wish he could have a job. Give him something to do. (F4) He’s like, a different type of person. He can’t do what the average guy does. He can’t work. And I don’t—I just, I don’t know. I feel like I’m so used to it now that it doesn’t really bother me. Because he helps out as much as he can? He gives everything and we just work on it from there. But, intimately, it sometimes messes up our relationship intimately, but like I said, I’ve learned to live with so much that it doesn’t really bother me anymore. (F3) There will be no job for her. She says she wanted to work. But I know she, that she go to work, that she got to stay on the job. She can’t be running back and forth from the job, to the hospital. (F7)
Care of the patients’ children—three generations of caring Five of the eight adult individuals with SCD were parents. An important tension that arose between caregivers and patients was the caretaking of the adults’ children:
Balancing independence and dependence
She loves her daughter. And she … she take care of her when she can with her. (F7)
Caregivers wished that the person would be more of an active contributor to the household, despite having an unpredictable illness,
I have three kids, plus him, you know, my oldest is eighteen; and, like I said, I been with him for eleven years. So you can just imagine, I had babies then. Having to pack the kids up, four o’clock in the morning to take him to the hospital, you know? And then, sometimes I can’t stay with
That’s why she stays in the hospital a lot of times too, because when she was living with me and my
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Journal of Health Psychology him, “cause I gotta get the babies home. So it makes me feel bad that I can’t be there for him all the time. Because I also have to take care of the babies. So, you know, it hurts a lot.” (F8)
Affected individuals expressed frustration and stress about not being able to be fully invested in raising their children: It’s tough on them. Because they don’t like to see me like that. Especially my son, even though he’s the youngest, me and him have the best bond and it’s really rough on him. It’s worse on him, but it’s real bad on everyone. (S8) Sometimes I feel like I’m neglecting my son, being here [in the hospital]. Not a good mom, sometimes, that’s how I feel. People tell me, you know, I can’t think like that “cause I have this issue. You know? It’s not like you’re just doing it to do it. You have to take care of yourself in order for you to be there for him. But it just stresses me out.” (S6) Look where I’m at. I’m in the hospital. I’ve been in the hospital two years in a row for my daughter’s birthday, July 17th, 2010 I’m in the hospital, my daughter’s birthday was July 17th. July 17th, 2009, I was in the hospital. You know, it’s like, and nothing ever gives—Christmas, you know, Thanksgiving, New Years, it seems like I’m always in the hospital for the holidays. It’s like nothing gives, you know? I’m like, God, why, why me? (S1)
Fear of the patients’ death Caregivers and patients alike worried about worsening physical limitations, organ degeneration, and death, especially as caregivers and patients believed that the patients were approaching the average life expectancy for people with SCD. These thoughts provoked anxiety in the patients and caregivers; members of both groups stated that they were unable to broach these subjects with each other and found it difficult to plan for the future or be optimistic about it. The following two quotations come from affected adult S8 and his primary caregiver F8,
who were both concerned about death due to SCD complications. Sometimes I just get into a rut where I start feeling sorry for myself and, you know, stuff like that. Asking stuff like “why does it have to be me.” I get really down sometimes … I keep everything bottled up. It’s like, I don’t know what it is, I just don’t really like talking about that stuff. My family gets mad at me because I don’t talk to them about it, but, you know, every now and then I meet someone at the hospital and I end up talking to them. Being able to get it off my chest, or whatever. (S8) He told me how he wasn’t supposed to live past eighteen. The doctors had told him he wasn’t supposed to live past eighteen. And that really hit me hard. So, that’s when I was, like, you know, I need to really study this and see what’s going on. Because he didn’t, himself, know that much. (F8)
Life expectancy of adults with SCD weighed heavily on other caregivers and affected individuals: To my knowledge, sickle cell life expectancy is only 25. I know a lot of people that lived well past it, but you know, that’s the life expectancy. I try not to think about that. I mean, it comes at me sometimes, but, you know, what can you do? What can you do? I’m not one to, you know, sit there and just, “oh my God, I can’t live, I can’t go on.” You know? Life goes on. But, you know, it definitely has an effect on me. I can’t even say how it would affect me if it happens, because, you know, I’m not, I haven’t experienced those shoes. All I could do is think positive right now. (F6) I was born in the 80’s. When I was born, women with sickle cell were not living past the age of 16. Men were, but women weren’t. They told my mother I would die before I was 12 years old. Do you know how that feels to have an expiration date put on your life? And, you know, for me to be young; I was six years old when I heard the doctors tell my mom that. And she never knew that I knew, but I knew. I would be scared to go to sleep, because I would think I was gonna die in my sleep. I was like that for a long time, a long time. I was depressed for a long time and I thought
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Brown et al. I was gonna die. I could not figure out why I was given something and now I’m gonna be taken away from my family. And you know, I got over it. (S2) I hear a lot of stories about people that has sickle cell. Like, people live to like, 35, with sickle cell. It’s more help now so that people can live, like, longer lives. But it’s still to me, it’s still not right or normal. Because, you know, people—like, they say now, 50. You know, that’s good for a person with sickle cell. And to me it’s not. I want to live till I’m 90, just like other people. That bothers me. Not knowing when you come [to the hospital], it’s always something new. Like, when I came to the hospital, after I had my son, my heart enlarged. So I have an enlarged heart now. And they tell me I can’t have any more kids, which hurt me, because I wanted more kids. So that bugs me a lot, too. (S6)
Discussion SCD repeatedly interferes in daily events for patients and family caregivers. A study of parent caregivers of children with autism, cerebral palsy, Down syndrome, and SCD revealed that parents viewed their roles in their communities as advocating, educating, informing, ignoring, and hiding (Neely-Barnes et al., 2010). The caregivers interviewed in this study described exhibiting many of these behaviors. For the caregivers, taking care of adults with SCD and EHHU is difficult because of the disease’s variability and the tensions embedded in caring for adults. These tensions included caring for the affected person’s children and encouraging the patients’ independence while acknowledging their limitations. As aging adults themselves, the caregivers’ ability to care for the patient was limited for several reasons: they have their own lives, they have their own chronic illnesses, or they are too elderly to meet the caregiving burden for a person with SCD and EHHU. Despite these limitations, the caregivers seemed everpresent advocates for the affected individuals. Patients and family caregivers believed that death was close at hand because of the patients’ advanced age for individuals with SCD. Adults
with SCD represent an understudied population that needs updated metrics for measuring life expectancy, more research on the effects of pregnancy on crises and interventions to improve caregivers’ support systems and quality of life. Only half of the patients interviewed were taking hydroxyurea, a medication known to be efficacious for SCD but is underprescribed and may also have patient-level barriers to uptake (Wong et al., 2014). A possible clinical recommendation for adolescents and young adults with SCD would be a hydroxyurea regimen, because caregivers who cared for individuals not taking hydroxyurea experience an increased burden for providing care compared with those who do take hydroxyurea (Da Silva et al., 2012). Caregivers struggled with establishing how much support to give, but the patients’ inability to have regular employment, regularly care for children, or act as a contributing partner to a household meant that patients needed more support than they might have anticipated. In turn, caregivers, especially those who are parents, might overcompensate for feelings of sympathy and guilt and act in ways that undermine patients’ independence, thereby reinforcing the sick role (Parsons, 1978). Caregivers were close relatives of the patients, which further complicated communication about sensitive topics such as death and depression. Since caregiving for people with SCD is sometimes a life-long commitment, and given that caregivers for people with SCD experience lower quality of life scores than other caregivers and non-carers, there should be resilience training for any caregivers, but especially for the parents of infants with SCD and that should continue throughout the life cycle. Periodic measurement of quality of life for affected individuals and caregivers can provide insight into tensions happening outside the hospital. Consistent mental health services for caregivers and patients provided at low cost would also improve communication between these groups as well as would formal resilience training. A better alliance and improved communication is needed between caregivers and medical staff who understand the patient, not just the disease.
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More research is also needed on how being a spouse or romantic partner complicates the caregiver role. Two of the caregivers were patients’ partners, which caused stress in the relationship regarding how the household should run, how the family can subsist on one person’s income, and how all the responsibilities are shifted to the caregiver. Policy implications of the Family Medical Leave Act, which was mentioned by one partner, should be further investigated to determine its effectiveness for a disease like SCD.
Study limitations Specific characteristics of our study may limit the generalizability of our findings. The sample sizes of caregiver and patient interviews are small, but for qualitative studies with a purposive sampling, the reasons for choosing these patients was to understand the experiences of people with a very high average number of hospital days over successive years and the individuals who care for them. Patients were asked to identify who is the family member, significant other, spouse, or friend, most important to their care. Patients described their supporters in generalities as being synonymous with their family members, without being able to describe explicitly the activities that the caregivers did for them. We surmise that this is because patients thought of support as primarily emotional support rather than the time-consuming but necessary smaller tasks that they are usually too ill to complete.
Suggestions from caregivers An interview question asked caregivers to suggest improvements for patient care and describe what support they would like to see for caregivers of someone with SCD. Caregivers made the following concrete suggestions •• Medical staff needs to understand the patient: “You understand a person better, you’ll know how to take care of them better.”
•• Establish a local support group for caregivers. •• Give the caregiver more information. •• Raise awareness for sickle cell (e.g. a walk—The Sickle Cell Disease Association of America held its first annual national fundraising walk on 6 September 2014). SCD is an unpredictable disease that touches not only the affected individuals but also those who are related to them and who care for them. Adults with SCD represent an understudied population, and caregivers of adults deserve their own support system. Developing a set of “best practices” with caregivers addressed to caregivers could help them create networks. The caregivers interviewed did not know many more people with SCD but perhaps each caregiver might know another person who could benefit from sharing advice. Acknowledgements This work was supported by the George D. and Esther S. Gross Professor of Psychiatry endowment to Dr. Sledge [no grant number]; and the Office of Medical Student Research, Yale School of Medicine for Dr. Weisberg [no grant number]. When the research was conducted, Daniel F. Weisberg, MD was affiliated with Department of Psychiatry, Yale School of Medicine, New Haven, Connecticut, USA.
Funding This research received no specific grant from any funding agency in the public, commercial, or not-forprofit sectors.
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HPQ0010.1177/1359105315588215Journal of Health PsychologyBrown et al.
Article
Family caregiving for adults with sickle cell disease and extremely high hospital use
Journal of Health Psychology 1–10 © The Author(s) 2015 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav DOI: 10.1177/1359105315588215 hpq.sagepub.com
Shan-Estelle Brown1, Daniel F Weisberg2 and William H Sledge1
Abstract This study investigated coping with chronic illness in the adult patient–caregiver relationship for sickle cell disease, marked by debilitating acute and chronic pain. One-on-one interviews (N = 16) were conducted with eight primary caregivers of eight adults with extremely high hospital use, severe sickle cell disease with hospital admissions several times monthly over successive years. Caregivers were predominantly parents; two were romantic partners. Caregivers attributed disruptions to the disease’s variability, tensions in how much support to give, and adults’ inability to fulfill parental obligations. Both groups expressed fears of patients’ increasing age, declining health, and early death. Targeted counseling and resilience training is recommended.
Keywords adults, chronic illness, coping, pain, social support
Introduction The act of caregiving can be a source of joy and bring families together, provided that caregivers have adequate information, social and financial support, and support from medical staff (Ohaeri and Shokunbi, 2002). Caring for a person affected by sickle cell disease (SCD), however, can precipitate “chronic sorrow” (Northington, 2000), the emotional distress that caregivers of people with SCD may experience. SCD is complex due to its clinical variability (Ballas et al., 2012), treatment as a racialized disease (Bediako and Moffitt, 2011; Rouse, 2009), as well as financial stress and possibly daily disruptions in family interactions (Brown et al., 2010). SCD is an inherited hemoglobinopathy known for its acute painful vaso-occlusive crises and chronic pain
(Ballas et al., 2012; Brousseau et al., 2010). Although most individuals manage crises at home (Smith et al., 2008), a small minority of patients are admitted as often as several times per month, over successive years (Carroll et al., 2009; Shankar et al., 2005), a phenomenon called extremely high hospital use (EHHU).
1Yale
School of Medicine, USA and Women’s Hospital, USA
2Brigham
Corresponding author: Shan-Estelle Brown, Department of Internal Medicine, Yale School of Medicine, 135 College Street, Suite 323, New Haven, CT 06510-2283, USA. Email: [email protected]
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The purpose of this study is to understand the interaction between adult patients with EHHU and their family caregivers. Previous studies on EHHU found that SCD patients with EHHU demonstrated increased hospitalization and opioid use from a young age, which perpetuated missed developmental milestones and a cycle of increasing hospitalization (Weisberg et al., 2013) and difficulty negotiating pain management with medical providers (Brown et al., 2014). Although most literature presents such difficulties for pediatric and adolescent patients (Tsao et al., 2013), generations of patients with SCD are living into adulthood, which present different challenges related to stress (Porter et al., 2000) and stigma (Bediako et al., 2014), different actors (e.g. spouses), and added complexity in the patient–caregiver relationship. Caregivers are problem-solvers (Barakat et al., 2013) who are frequently responsible for the education and home care of children with SCD, but this role often comes with a cost (Beyer and Simmons, 2004; Hoyt Drazen et al., 2014; Wonkam et al., 2014). Caregivers of patients with SCD had a significantly lower quality of life on all subscales of depressive moods, daily activities, and vitality compared to non-caregivers (Van Den Tweel et al., 2008). Compared with other caregivers to individuals with chronic illnesses, caregivers of children with SCD experience higher depressive mood scores even if the caregiving burden is similar (Moskowitz et al., 2007). Caregiving affects the entire family, as grandparents may additionally act as caregivers and siblings of affected children also encounter stress and possible negative psychosocial outcomes (Dilworth-Anderson, 1994; Gold et al., 2011; Graff et al., 2012). As caregivers are predominantly mothers (Hill, 1994; Sharpe et al., 1994), fear of their children’s deaths, separation anxiety, and helplessness blunt their ability to care (Burnes et al., 2008). This study attempts to fill several gaps in the literature of SCD, caregiving, and chronic illness. First, literature on SCD and caregivers depicts caregivers primarily as parents caring
for sick children. This study intends to expand the picture of caregiving to include nonparental roles, such as spouses, when the affected person is an adult. Second, the majority of articles on adult sickle cell care focus on the clinical transition from pediatric to adult SCD care, not what happens afterward. When an adolescent patient transitions from pediatric to adult care, the number of emergency department visits increases (Hemker et al., 2011). At this time, family caregivers are especially concerned about medication adherence and adult providers’ lack of familiarity with their child’s needs (Porter et al., 2014; Telfair et al., 1994). As recent research suggests that younger and older adults have different coping strategies (Sanders et al., 2010), the experiences of adults with SCD who have completed this transition need further investigation. Third, SCD is frequently described as an acute disease and the chronic illness component is less understood. This study uses a framework of illness uncertainty for chronic disease, which affects illness experiences and communication between family members to understand the unpredictability of SCD pain (Checton et al., 2012; Johnson Wright et al., 2009). This study attempts to fill these gaps by characterizing illness experiences for individuals with severe chronic illness from SCD and the relationships to their caregivers who are often their parents and grandparents to the individuals’ children.
Method Studies of caregivers have often involved the development of coping models (Brown et al., 2000), quantitative survey instruments to assess caregiver quality of life and stressors (Karlson et al., 2012; Schlenz et al., 2012), and focus group studies (Neely-Barnes et al., 2010; Porter et al., 2014). This study employed a qualitative study design of one-on-one, face-to-face interviews with patients with EHHU and their caregivers. EHHU was defined as at least 100 hospital days/year over each index 12 months. Patients were first identified as having EHHU and SCD through the Yale-New Haven Hospital
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Brown et al. Resource Information Management System for the period 1 January 2008 to 31 December 2010. Chart review further compared patients’ clinical histories for similarities in disease profile, complications, and medication regimens. Participants consisted of 16 individuals—8 SCD patients with EHHU (“high hospital-utilizing,” “EHHU”) and their primary caregivers, whom patients identified as the family member, significant other, spouse, or friend, most important to their care. Individuals identified were approached by telephone or in person; two individuals refused to participate. Table 1 presents the patients’ age, gender, and hemoglobinopathy type, as well as relationship to the caregiver. All the affected adults were unemployed. Five of the adults were parents. Demographic information was not collected on the caregivers.
Interviews An interview guide was created with openended questions to address the following themes: (1) disease, pain, and medication; (2) hospitalization; (3) support structures; (4) daily life; and (5) personal relationships. Family caregivers were asked to describe their role in caring for the patient, improvements, and setbacks as the disease has progressed, the evolution of their relationship, and helpful and harmful ways that the patients cared for themselves. Affected adults were interviewed by DW at the hospital. Family caregivers were interviewed by DW and a second interviewer; four interviews were conducted in research buildings near the hospital and four were conducted at the caregivers’ homes. Interviews lasted approximately 1 hour and were conducted one-on-one. Interviews were audio-recorded in private settings and transcribed by a professional transcriptionist. Participants were compensated US$25 for a completed interview.
Analysis Analysis began with pilot testing of the interview guide and creation of narrative summaries of the interviews (Weisberg et al., 2013).
Interviews were also coded by content analysis using Atlas.Ti software (Saldaña, 2009). Similarities in words and phrases were grouped together as codes, and the codes representing themes were linked to make broader inferences about participants’ experiences. We generated themes from the interview guide, and two coders (SB, DW) identified the themes in the transcripts. The entire research team met regularly to discuss themes and saturation. An audit trail to track emergence of new codes and progress of coding was maintained throughout. The research protocol was approved by the Human Investigation Committee at Yale School of Medicine, New Haven, CT.
Results The analysis identified differences in caregivers’ and patients’ descriptions of the care that caregivers provide; caregivers described difficulty balancing patients’ independence with limitations due to SCD and how they also care for the affected persons’ children; both groups expressed fear of the patients’ death.
Care that family caregivers provide Interviews with the caregivers reflected empathy about the person they cared for, but they described different kinds of support. Forms of support included rides to appointments, household routines, strength and encouragement, bringing food to the hospital, cooking, shopping, hospital visits, and errands. Patients described several people in their support network, not only the caregiver we interviewed. Patients broadly described the support they received: My aunts, they support me sometimes, but not all the time. Most of my support comes from my grandparents and my daughter. In each and every way they care for me. They come and visit me. If I don’t have the money to get what I want, they give it to me. They take care of me. (S1)
Only one sickle cell patient, S4, considered the support reciprocal:
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34 27 26 34 37 25 24 32
F F M M F F F M
Yes No No No Yes Yes Yes Yes
HbSS HbSS HbS-B thalassemia HbSC HbSS HbS-B thalassemia HbSS HbSC
SA + LA SA SA + LA SA + LA SA + LA SA + LA SA SA + LA
Yes No No Yes No Yes Yes No
PH AChS AVN CKD AChS AVN PH AChS PH AVN AChS None identified AVN PH AChS AVN PH AChS IO
171 263 151 111* 202 104 123 94
Stepdaughter Younger sister Romantic partner Son Daughter Daughter Grandniece Romantic partner
Average Relationship to hospital days caregiver 2008–2010
F1 F2 F3 F4 F5 F6 F7 F8
Caregiver ID
SA: short acting; LA: long acting; EHHU: extremely high hospital use. HbSS: homozygosity for the sickle cell gene (HBB glu6val), sickle cell anemia; HbSC: heterozygosity for the sickle cell gene (HBB glu6val) and the hemoglobin C gene (HBB glu6lys), sickle-hemoglobin C disease; HbS-B thalassemia: heterozygosity for the sickle cell gene (HBB glu6val) and one of the B-thalassemia gene mutations. AVN: joint avascular necrosis; PH: pulmonary hypertension; CKD: chronic kidney disease; AChS: acute chest syndrome; IO: iron overload. *Pt spent 1 year incarcerated—not included in calculation of average hospital days.
EHHU S1 Patients S2 S3 S4 S5 S6 S7 S8
Patient Age Sex Is a Hemoglobinopathy Outpatient opioid Hydroxyurea Major SCDID parent? regimen (short related acting/long acting) complications
Table 1. Patients’ clinical and demographic characteristics.
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Brown et al. If I need something, or if [my mother] can help me with something, she can help me. And it goes both ways. Like I help her, right now, she going through her sickness. I’ll help her out, like she do dialysis at home, I help her out. Just stuff like, carry the boxes upstairs, carry the bags upstairs, or whatever. Go over there and clean her house up and stuff. Take the trash out. And if she could help me with something, she helps me. (S4)
Caregivers used different kinds of knowledge to provide care. One caregiver searched the internet and “read everything she could get her hands on” (F2) while others relied on their often life-long experience with caring for a child with SCD, emphasizing “because I’m her mother!” (F5). One caregiver felt that the care she provided to her sister was limited, compared to the hospital’s services: I think it’s actually to the point where she might feel she has to be here [at the hospital]. [My sister thinks,] “This is the only place that’s gonna take care of me. They’re gonna take care of me better than my family can.” (F2)
As part of their care, two caregivers advised their patients on managing hospital staff: I don’t think he says anything. Especially as far as his care is concerned. Let them know how he feels, or what’s going on. Sometime you gotta be aggressive. He’s pretty much laid back. He won’t say anything. (F4) Although right or bad, if they don’t do the right thing, she gets on them. I told her to be nice to the doctors and nurses, and they’d be nice to her. I can’t tell them what to do. All I know is I told her to be nice to people. (F7)
wife, she would have to clean up. You know what I mean? Because I was telling my wife, make her do something. Don’t let her just sit on the couch. When she’s at grandma’s she doesn’t have to do anything. (F1)
Work was a recurring theme in the interviews with caregivers, as the majority of the EHHU patients interviewed are in the hospital too many consecutive days to work regularly. Despite the fact that caring for a person with SCD interrupted caregivers’ own employment, caregivers still wanted adult patients to work: I wish he could have a job. Give him something to do. (F4) He’s like, a different type of person. He can’t do what the average guy does. He can’t work. And I don’t—I just, I don’t know. I feel like I’m so used to it now that it doesn’t really bother me. Because he helps out as much as he can? He gives everything and we just work on it from there. But, intimately, it sometimes messes up our relationship intimately, but like I said, I’ve learned to live with so much that it doesn’t really bother me anymore. (F3) There will be no job for her. She says she wanted to work. But I know she, that she go to work, that she got to stay on the job. She can’t be running back and forth from the job, to the hospital. (F7)
Care of the patients’ children—three generations of caring Five of the eight adult individuals with SCD were parents. An important tension that arose between caregivers and patients was the caretaking of the adults’ children:
Balancing independence and dependence
She loves her daughter. And she … she take care of her when she can with her. (F7)
Caregivers wished that the person would be more of an active contributor to the household, despite having an unpredictable illness,
I have three kids, plus him, you know, my oldest is eighteen; and, like I said, I been with him for eleven years. So you can just imagine, I had babies then. Having to pack the kids up, four o’clock in the morning to take him to the hospital, you know? And then, sometimes I can’t stay with
That’s why she stays in the hospital a lot of times too, because when she was living with me and my
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Journal of Health Psychology him, “cause I gotta get the babies home. So it makes me feel bad that I can’t be there for him all the time. Because I also have to take care of the babies. So, you know, it hurts a lot.” (F8)
Affected individuals expressed frustration and stress about not being able to be fully invested in raising their children: It’s tough on them. Because they don’t like to see me like that. Especially my son, even though he’s the youngest, me and him have the best bond and it’s really rough on him. It’s worse on him, but it’s real bad on everyone. (S8) Sometimes I feel like I’m neglecting my son, being here [in the hospital]. Not a good mom, sometimes, that’s how I feel. People tell me, you know, I can’t think like that “cause I have this issue. You know? It’s not like you’re just doing it to do it. You have to take care of yourself in order for you to be there for him. But it just stresses me out.” (S6) Look where I’m at. I’m in the hospital. I’ve been in the hospital two years in a row for my daughter’s birthday, July 17th, 2010 I’m in the hospital, my daughter’s birthday was July 17th. July 17th, 2009, I was in the hospital. You know, it’s like, and nothing ever gives—Christmas, you know, Thanksgiving, New Years, it seems like I’m always in the hospital for the holidays. It’s like nothing gives, you know? I’m like, God, why, why me? (S1)
Fear of the patients’ death Caregivers and patients alike worried about worsening physical limitations, organ degeneration, and death, especially as caregivers and patients believed that the patients were approaching the average life expectancy for people with SCD. These thoughts provoked anxiety in the patients and caregivers; members of both groups stated that they were unable to broach these subjects with each other and found it difficult to plan for the future or be optimistic about it. The following two quotations come from affected adult S8 and his primary caregiver F8,
who were both concerned about death due to SCD complications. Sometimes I just get into a rut where I start feeling sorry for myself and, you know, stuff like that. Asking stuff like “why does it have to be me.” I get really down sometimes … I keep everything bottled up. It’s like, I don’t know what it is, I just don’t really like talking about that stuff. My family gets mad at me because I don’t talk to them about it, but, you know, every now and then I meet someone at the hospital and I end up talking to them. Being able to get it off my chest, or whatever. (S8) He told me how he wasn’t supposed to live past eighteen. The doctors had told him he wasn’t supposed to live past eighteen. And that really hit me hard. So, that’s when I was, like, you know, I need to really study this and see what’s going on. Because he didn’t, himself, know that much. (F8)
Life expectancy of adults with SCD weighed heavily on other caregivers and affected individuals: To my knowledge, sickle cell life expectancy is only 25. I know a lot of people that lived well past it, but you know, that’s the life expectancy. I try not to think about that. I mean, it comes at me sometimes, but, you know, what can you do? What can you do? I’m not one to, you know, sit there and just, “oh my God, I can’t live, I can’t go on.” You know? Life goes on. But, you know, it definitely has an effect on me. I can’t even say how it would affect me if it happens, because, you know, I’m not, I haven’t experienced those shoes. All I could do is think positive right now. (F6) I was born in the 80’s. When I was born, women with sickle cell were not living past the age of 16. Men were, but women weren’t. They told my mother I would die before I was 12 years old. Do you know how that feels to have an expiration date put on your life? And, you know, for me to be young; I was six years old when I heard the doctors tell my mom that. And she never knew that I knew, but I knew. I would be scared to go to sleep, because I would think I was gonna die in my sleep. I was like that for a long time, a long time. I was depressed for a long time and I thought
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Brown et al. I was gonna die. I could not figure out why I was given something and now I’m gonna be taken away from my family. And you know, I got over it. (S2) I hear a lot of stories about people that has sickle cell. Like, people live to like, 35, with sickle cell. It’s more help now so that people can live, like, longer lives. But it’s still to me, it’s still not right or normal. Because, you know, people—like, they say now, 50. You know, that’s good for a person with sickle cell. And to me it’s not. I want to live till I’m 90, just like other people. That bothers me. Not knowing when you come [to the hospital], it’s always something new. Like, when I came to the hospital, after I had my son, my heart enlarged. So I have an enlarged heart now. And they tell me I can’t have any more kids, which hurt me, because I wanted more kids. So that bugs me a lot, too. (S6)
Discussion SCD repeatedly interferes in daily events for patients and family caregivers. A study of parent caregivers of children with autism, cerebral palsy, Down syndrome, and SCD revealed that parents viewed their roles in their communities as advocating, educating, informing, ignoring, and hiding (Neely-Barnes et al., 2010). The caregivers interviewed in this study described exhibiting many of these behaviors. For the caregivers, taking care of adults with SCD and EHHU is difficult because of the disease’s variability and the tensions embedded in caring for adults. These tensions included caring for the affected person’s children and encouraging the patients’ independence while acknowledging their limitations. As aging adults themselves, the caregivers’ ability to care for the patient was limited for several reasons: they have their own lives, they have their own chronic illnesses, or they are too elderly to meet the caregiving burden for a person with SCD and EHHU. Despite these limitations, the caregivers seemed everpresent advocates for the affected individuals. Patients and family caregivers believed that death was close at hand because of the patients’ advanced age for individuals with SCD. Adults
with SCD represent an understudied population that needs updated metrics for measuring life expectancy, more research on the effects of pregnancy on crises and interventions to improve caregivers’ support systems and quality of life. Only half of the patients interviewed were taking hydroxyurea, a medication known to be efficacious for SCD but is underprescribed and may also have patient-level barriers to uptake (Wong et al., 2014). A possible clinical recommendation for adolescents and young adults with SCD would be a hydroxyurea regimen, because caregivers who cared for individuals not taking hydroxyurea experience an increased burden for providing care compared with those who do take hydroxyurea (Da Silva et al., 2012). Caregivers struggled with establishing how much support to give, but the patients’ inability to have regular employment, regularly care for children, or act as a contributing partner to a household meant that patients needed more support than they might have anticipated. In turn, caregivers, especially those who are parents, might overcompensate for feelings of sympathy and guilt and act in ways that undermine patients’ independence, thereby reinforcing the sick role (Parsons, 1978). Caregivers were close relatives of the patients, which further complicated communication about sensitive topics such as death and depression. Since caregiving for people with SCD is sometimes a life-long commitment, and given that caregivers for people with SCD experience lower quality of life scores than other caregivers and non-carers, there should be resilience training for any caregivers, but especially for the parents of infants with SCD and that should continue throughout the life cycle. Periodic measurement of quality of life for affected individuals and caregivers can provide insight into tensions happening outside the hospital. Consistent mental health services for caregivers and patients provided at low cost would also improve communication between these groups as well as would formal resilience training. A better alliance and improved communication is needed between caregivers and medical staff who understand the patient, not just the disease.
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More research is also needed on how being a spouse or romantic partner complicates the caregiver role. Two of the caregivers were patients’ partners, which caused stress in the relationship regarding how the household should run, how the family can subsist on one person’s income, and how all the responsibilities are shifted to the caregiver. Policy implications of the Family Medical Leave Act, which was mentioned by one partner, should be further investigated to determine its effectiveness for a disease like SCD.
Study limitations Specific characteristics of our study may limit the generalizability of our findings. The sample sizes of caregiver and patient interviews are small, but for qualitative studies with a purposive sampling, the reasons for choosing these patients was to understand the experiences of people with a very high average number of hospital days over successive years and the individuals who care for them. Patients were asked to identify who is the family member, significant other, spouse, or friend, most important to their care. Patients described their supporters in generalities as being synonymous with their family members, without being able to describe explicitly the activities that the caregivers did for them. We surmise that this is because patients thought of support as primarily emotional support rather than the time-consuming but necessary smaller tasks that they are usually too ill to complete.
Suggestions from caregivers An interview question asked caregivers to suggest improvements for patient care and describe what support they would like to see for caregivers of someone with SCD. Caregivers made the following concrete suggestions •• Medical staff needs to understand the patient: “You understand a person better, you’ll know how to take care of them better.”
•• Establish a local support group for caregivers. •• Give the caregiver more information. •• Raise awareness for sickle cell (e.g. a walk—The Sickle Cell Disease Association of America held its first annual national fundraising walk on 6 September 2014). SCD is an unpredictable disease that touches not only the affected individuals but also those who are related to them and who care for them. Adults with SCD represent an understudied population, and caregivers of adults deserve their own support system. Developing a set of “best practices” with caregivers addressed to caregivers could help them create networks. The caregivers interviewed did not know many more people with SCD but perhaps each caregiver might know another person who could benefit from sharing advice. Acknowledgements This work was supported by the George D. and Esther S. Gross Professor of Psychiatry endowment to Dr. Sledge [no grant number]; and the Office of Medical Student Research, Yale School of Medicine for Dr. Weisberg [no grant number]. When the research was conducted, Daniel F. Weisberg, MD was affiliated with Department of Psychiatry, Yale School of Medicine, New Haven, Connecticut, USA.
Funding This research received no specific grant from any funding agency in the public, commercial, or not-forprofit sectors.
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