Introduction
Methods
Diary development
Participant sample
Interview procedure
Concept elicitation
Cognitive debriefing
Data analysis
Results
Demographic Information | Round One Hybrid Interviews n = 6 | Round Two Hybrid Interviews n = 3 | Round Three Cognitive Debriefing Interviews n = 13 |
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Caregiver’s gender | |||
Male | 1 | 1 | 2 |
Female | 5 | 2 | 11 |
Caregiver’s education | |||
High school or equivalent | 0 | 1 | 4 |
Some college | 0 | 2 | 0 |
Associate’s degree | 1 | 0 | 2 |
Bachelor’s degree | 3 | 0 | 2 |
Post-graduate degree | 2 | 0 | 5 |
Caregiver’s relationship to the child | |||
Parent | 4 | 3 | 11 |
Grandparent | 1 | 0 | 1 |
Cousin | 1 | 0 | 0 |
Legal Guardian | 0 | 0 | 1 |
Region of residence | |||
Northeast | 3 | 0 | 3 |
Pacific | 0 | 0 | 2 |
Southeast | 3 | 0 | 6 |
Midwest | 0 | 3 | 2 |
Child’s gender | |||
Male | 2 | 2 | 4 |
Female | 4 | 1 | 9 |
Child’s age | |||
Mean (SD) | 8.3 years (2.8) | 8.7 years (1.5) | 5.1 years (3.1) |
Range | 3–11 years | 7–10 years | 11 months-11 years |
Child’s disease type | |||
HbSS disease | 2 | 0 | 8 |
HbSC disease | 2 | 3 | 3 |
Don’t know/unsure | 2 | 0 | 2 |
# of SCPCs in the past 12 months | |||
Mean (SD) | 5.8 (4.2) | 18.3 (2.9) | 6 (5.9) |
Range | 1–12 | 15–20 | 1–24 |
Frequency of healthcare use for SCPCs | |||
Every time or almost every time | 5 | 3 | 10 |
Sometimes but not all of the time | 1 | 0 | 3 |
Treat at home/see regular doctor | 0 | 0 | 0 |
Concept elicitation
Symptoms
Impacts and healthcare resource utilization
Area of Impact | n (%) n = 9 | Representative Caregiver Quote(s) |
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Emotional Health Anxiety Depression Anger Feeling judged Frustration | 7 (78) | And it was, 'Why do I have to do this? Why do I have to go through this?' You know, 'Why me?' So, it affects her because now she's getting older and she's realizing what she's missing. She’ll see the… kids that don't have sickle cell get to run, and play, and jump, and do –so it affects her. She doesn't like sitting on the sidelines sometimes. So, you'll catch her crying. You’ll catch her upset |
Social and Recreational Activities Modify or limit activities including: physical education, recess, field trips, or sports | 6 (67) | He would like to play football, and that’s just not going to happen for many reasons. Because, just getting bruised, that can be an issue… I might take him shopping or do something kind of fun. But not too fun… any little thing can trigger him going into a crisis or just a lot of pain |
Physical Functioning Limit physical activities including: running or playing | 5 (56) | Sometimes she likes to run, and she likes to jump, and she likes to play; she loves to dance. So, with her, she gets winded so quick that she has to stop and sit down, and then she has to get back up and so… with sickle cell, you have a lack of oxygen you can pull into all of your organs, so with her, she’s off top like, OK, I can't go as long or I can't, but it doesn't stop her, but she’s going to try it |
Daily Activities Interference in day-to-day activities General low activity | 5 (56) | [He’s] not as active as my other kids. Um mainly you got to, “Come on let’s go,” you got to prep him up to get him to go to school and stuff like that |
School Attendance School work Attentiveness during class | 4 (44) | He’s literally falling asleep in class. He’ll come home and fall asleep and won’t finish his studies or his homework |
Representative Caregiver Quotes on SCPCs | ||
---|---|---|
The pain crises. He would holler. Scream. Run through the house… Just more pain. The screaming, usually when he go through crises, everything starts to swell up. In his stomach, hands, lips, and ankles. And he don’t want to be bothered, so I know that’s when I really have to take him in When he’s in a crisis, it’s just intensifies about 10 times more. He’s screaming and in a lot of pain. And then sometimes—but the pain is kind of weird, because sometimes it’s like it’s certain body parts. It’s never in one area. Sometimes, 'My arm is so numb, and it hurts, and it feels like needles and pins sticking in.' And then there’s other times where he just, he has a really bad stomach ache, but he cannot hold anything down |
Impacts of SCPC | n (%) n = 9 | Representative Caregiver Quote |
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Emotional Health Sadness Agitation/Irritability Anger Frustration Worry Fear | 9 (100) | As bad as she want to [play with others], she’ll break down and cry… frustrated and angry. She gets so angry when you don’t let her, cause she be willing and wanting to |
Sleep At home or at hospital Trouble falling asleep Trouble staying asleep | 9 (100) | No one slept. Because when you're in the hospital, they're coming in and checking often, and it's just hard to get comfortable. Or we are waking him to give him the medicine…stay ahead of the pain, so then you have to try to get him to go back to sleep |
School Attendance School work Attentiveness during class | 8 (89) | She’s not able to take her test when she’s supposed to. She has to do make-up…and then sometimes she can’t catch—she’s not catching up because she has so much to do, and she’s a little bit behind |
Social and Recreational Activities Unable to participate or needing to stop participation | 6 (67) | Um, he likes to play basketball, and he’ll run, run, run, and then all of a sudden, he’ll stop, and I’m like, 'Why’d you stop? You know, finish it,' or whatever. He’s like, 'No… I think that’s enough. ‘I said… 'Why? ‘And he doesn’t really have a explanation for it. But I can look at him and tell it’s like, you know, 'I don’t want to, ' and I said, 'What is it? Are you afraid of being in pain or, you know, you think you might be in pain? ‘He’s like, 'Maybe.' |
Physical Functioning Unable to complete physical tasks Immobile | 4 (44) | I mean, he is not going to be climbing anything, that’s for sure, and he is not going to be jumping, because his bones are hurting. He is not going to be doing any of that, that’s why he just sits |
Daily Activities Difficulty getting ready in the morning General inactivity Unable to use bathroom | 3 (33) | No, he can’t move. No, I even have to help him to the bathroom. I even got him one of those urinals from the hospital. He has it in his room |
Caregiver burden during an SCPC
Caregiver burden | n (%) n = 9 | Representative caregiver quote(s) |
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Work | 7 (78) | I’m telling you, it’s hard. It’s really hard. My boyfriend is home with him now, and he plays a big role and everything. But if I have to leave work or I have to change something with my schedule, because he got ill at school or—anytime, it’s just anytime type thing |
I basically work my own schedule, but when I do get my schedule because we schedule a week ahead, I got to stick with that schedule. And if I don’t get them clients to—I end up paying [for] what day they miss, I don’t like it. If that fare costed $60, I have to pay that | ||
Emotional health | 6 (67) | Oh, so many times I have broke—I haven’t did it in front of her, but I have went in the bathroom, broke down because it’s like, this is my baby, and I can’t do nothing for to help her It’s very painful for the caregiver as well, it’s very painful for the caregiver, because sometimes when you know they are going through something, and you could see it on their face, and sometimes they just don’t want to be bothered, they are just sick of it themselves |
Saturation
Cognitive debriefing
Round one
Original Text (SCPD-C v.01) | Rationale for Modification | Caregiver Quote On Original Text | Final Modified Text (SCPD-CS v.01 and SCPD-CN v.01) | Caregiver Quotes On Final Modified Text |
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Item text: Did pain from your child's sickle cell pain crisis cause you to miss any work during the last 24 h? Response Options: Yes No | Caregivers suggested modifying response options to record how much of the work day was missed, and noted that sometimes they did not miss work because they were not scheduled to work that day, but otherwise would have missed work | If you wanted to streamline it, 'Did you miss a whole day? Did you miss a part of the day?' Because sometimes, if a sickle cell crisis starts, and your child is in school, and you have to go pick up your child, it might be halfway through the day | I missed all of my paid work I missed half or more (but not all) of my paid work I missed less than half of my paid work I did not miss any paid work I was not scheduled to go to work I do not currently work | I think they were clear. Because you can’t just have… three answers; you’re saying 'I missed all of my paid work' or 'I do not currently work' or 'I was not scheduled to work.' So the …answers… perfect |
I think those were fine. I think those made sense | ||||
Item text: During the last 24 h, how much, on average, did you observe signs of your child having a problem with pain not related to a pain crisis? | Caregivers found this item confusing and suggested that it be reworded for clarity | You don’t want to have to deal with worrying about reading something that’s long and drawn out or something that’s cumbersome, because it’s like why? You don’t want to deal with that. I just think it can be easier | Based on your observation, how much on average did your child display signs of pain during the past 24 h? | Yeah, I think it makes sense |
On a daily basis when he’s not in pain, that would make a whole lot of sense. It’s a relatively easy answer | ||||
Item text: During the last 24 h, did your child take any medication to control his or her pain? | Caregivers reported inconsistencies in how they interpreted “any medication” | I was thinking only medication I have at home and if the doctor gave me any. That’s medication period, it just says 'any medication' | Did your child take any medication to control his or her pain, including prescribed and over-the-counter medication, during the past 24 h? | It was very simple |
Yes, he did take medication, to control his pain. He took both over-the-counter medication, as well as, prescription pain medication | ||||
Item text: How long did the pain crisis episode last in total during the last 24 h? Select the time point that best represents the total duration (in hours and/or minutes) | Caregivers reported that it would be difficult to accurately provide the exact duration of time that a pain crisis lasts in hours and minutes | It can happen at any point in time. So, we could be anywhere, and I could look at her and be like, OK, something’s not right with her. I could be at church, I can be at school, I could be anywhere. Be in the mall, anywhere. And I can look at her and be like, 'What’s wrong with you?' And then, most of the time by the time I can do that she's already been in pain probably a good hour or two and just hasn’t said anything | Approximately how many hours did the pain crisis last during the past 24 h? | No, not too hard. It was just more so, just, yeah, it was just more so me just trying to figure out roughly about what time she woke up and what time she went to bed so I was just trying to get those answers, you know, get the number of hours |
That was easy | ||||
Yeah. I can estimate |
Rationale | Representative Caregiver Quote | Item Added to the SCPD-C v.02 |
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All 6 caregivers reported that SCPCs had an impact on their child’s emotional health | I would ask, ‘How does sickle cell affect the persons as a whole, emotionally? What are the thoughts behind a person with sickle cell, they are feeling?’ Things like that, because when you go into those—and you ask someone, I am sure if they were asked, because I ask them all the time, and he just wish it didn’t happen. Most time they wish it didn’t happen, and the main question that I get is, ‘Am I going to die?’ | Based on your observation, how much emotional difficulty did your child experience due to his or her sickle cell disease during the past 24 h (for example, being irritated or mad, worried or afraid, or feeling very sad)? |
Four of 6 caregivers reported an impact on their emotional health as a result of their child’s SCPC | I think sometimes it affects the parent, so I know you've given the gauge in how it affects the child… it affects the parent watching the child go through it. So that’s another part… I’m so sorry. I’m sniffling | How much emotional difficulty did you experience due to your child’s sickle cell pain crisis during the past 24 h (for example, stress, anxiety, sadness, or depression)? |
Caregivers discussed a tendency to go to the hospital when their child is experiencing a pain crisis. Two of 6 caregivers endorsed adding an item related to healthcare utilization | I can't count how many times we have gone to the emergency department. In the last year usually, it's just a blood test, they say it's a common cold or some little virus and they send us home, but for a while it meant an overnight stay for a couple days | Did the pain crisis lead to a visit with your child's primary care doctor, urgent care center, or emergency department during the past 24 h? |
Round two
Round three
Summary of final diary content
Item Content | SCPD-CS v1.0 Caregiver Report, School Age | SCPD-CN v1.0 Caregiver Report, Non-school Age |
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For Electronic Administration | ||
Age of child | 1 item | 1 item |
School enrollment | 1 item | 1 item |
During an SCPC: Presence, Duration, and Severity | ||
Observed signs of SCPC | 1 item | 1 item |
Duration of SCPC | 2 items | 2 items |
Healthcare utilization due to SCPC | 1 item | 1 item |
Medication use due to SCPC | 2 items | 2 items |
Observed Interference of SCPC-related Pain on Daily Activities | ||
Interference with… | ||
Child’s school attendance | 1 item | 1 modified item: Preschool or Daycare |
Caregiver’s paid work attendance | 1 item | 1 item |
Child’s schoolwork | 1 item | – |
Child’s activities of daily living | 1 item | 1 item: Only if the child is 2 years or older |
Child’s social and recreational activities | 1 item | 1 item: Only if the child is 2 years or older |
Child’s sleep | 1 item | 1 item |
Observed Impacts of SCPC-related Pain on Fatigue and Emotional Difficulty | ||
Fatigue | 1 item | 1 item |
Child’s emotional difficulty | 1 item | 1 item: Only if the child is 2 years or older |
Caregiver’s emotional difficulty | 1 item | 1 item |
Outside of an SCPC: Observed Pain, Fatigue, and Impacts of SCD | ||
Signs of pain outside of an SCPC | 1 item | 1 item |
Interference with sleep | 1 item | 1 item |
Fatigue | 1 item | 1 item |
Emotional difficulty | 1 item | 1 item: Only if the child is 2 years or older |