Parent versus Child Donor Perceptions of the Bone Marrow Donation Experience

Although the use of minors as HSC donors is considered medically safe⁴ and legally acceptable given that no alternative approach of comparable effectiveness exists, several published literature reviews and national organizations like the American Academy of Pediatrics have noted a lack of understanding of the physical/psychosocial effects of pediatric HSC donation and have called for investigations of such effects.^5,6,7,8,9

Prior to our recently completed investigation of health-related quality-of-life (HRQoL) in sibling pediatric HSC donation (RDSafe: A Multi-Institutional Study of HSC Donor Safety and Quality Life), the few reviews and other published investigations of pediatric HSC donors' experiences indicated that pediatric donors might experience a variety of psychosocial issues around donation – but limitations in prior investigations prevented definitive conclusions.^{6,7,8,9,10,11,12}

Key findings from our own RDSafe study – which interviewed donors and their parents/guardians at pre-donation and at 4-weeks and 1-year post-donation – included:(1) ∼20% of donors at each time point had very poor HRQoL, (2) child self-reported HRQoL was significantly lower than parent proxy-reported HRQoL and significantly lower than that of health normative comparison children, and (3) younger children (ages 5-7) were at particular risk of poor HRQoL.¹⁸

The goals of this follow-up analysis were to (1) examine whether parents of donor children believed that their child's age made a difference in donation-related experiences, (2) determine whether donor, recipient, or family characteristics differed by whether or not parents believed that their child's age made a difference, (3) qualitatively examine how parents explained the effects of their child's age, and (4) determine whether parents characterized their child's donation experience differently depending on whether their child self-reported very poor HRQoL.

This study was approved by IRBs at the University of Pittsburgh, the National Marrow Donor Program (NMDP), and participating transplant centers. Parents signed informed consent and children gave assent. The study included randomly selected pediatric sibling HSC donors aged 5-18 who were first-time bone marrow or PBSC donors in the US between April, 2010 and May, 2013 and their parent/guardian.

Pediatric donors and parents completed telephone interviews in English four weeks pre-donation and 4-weeks and 1-year post-donation. See Switzer et al., for additional methodological details.¹⁸ Measures included: (1) socio-demographics, (2) donor HRQoL assessed with the PedsQL™ 4.0 Generic Core Scales with both child and parent independent reports on child donor HRQoL,^19-21 and (3) parents' beliefs about the effects of their child's age assessed with a question asking whether they believed that their child's age made a difference in his/her donation experience (yes, no), and, if yes, how they believed that age affected the child's experience of the process (open-ended). The PedsQL™ is scored 0-100 with higher scores indicating better HRQoL. An “at risk” cutoff score of ≤ 69.71 is defined by the PedsQL™ developers.²¹ Children scoring at or below this cutoff have HRQoL similar to that of chronically ill children.^19-21

See Switzer et al. for descriptive statistics on socio-demographics and HRQoL for the entire sample.¹⁸ Socio-demographic comparisons of parents who reported that their child's age made a difference versus not and comparisons of children with HRQoL scores above versus below the at-risk cutoff were performed using t-tests and chi-square tests as appropriate. Qualitative data from parent responses to the open-ended question were reviewed for themes by three co-authors (GES, JB, and GP) and independently coded by two coders (GES and GP). Any coding disagreements were coded by a third co-author (JB) and adjudicated during group discussion.

This analysis includes 102 donor/parent pairs who completed both the pre-donation and 4-weeks post-donation interviews and 94 pairs who completed pre- and 1-year post-donation interviews. The majority of donors (93%) donated bone marrow and 7% donated PBSC.

At 4-weeks post-donation, 95/102 parents answered the question about their donor child's age making a difference (yes/no) – of those, 80% indicated that they believed their child's age made a difference in the donation experience. At 1-year post-donation, 89/94 parents answered the question – of those, 81% indicated that they believed their child's age made a difference in the donation experience.

Table 1 compares the two groups of parents (yes/no) by donor, recipient, and parent/family characteristics. At 4-weeks post-donation, parents of younger, male, and white donors; parents of younger recipients; and families with higher incomes were significantly more likely to indicate that their child's age made a difference. At 1-year post-donation, the pattern of results was similar but not statistically significant.

Table 1.

Participant characteristics by parents' beliefs about the effects of donor age at 4-weeks and 1–year post-donation.

Characteristic	4-weeks Post-donation				1-year Post-donation
	Yes n=76	No n=19	X2 or t	p	Yes n=72	No n=17	X2 or t	p
Demographic
Donor
Age group (%)			5.90	0.05			2.71	0.26
5-7	100	0			94	6
8-12	80	20			82	18
13-17	72	28			74	26
Sex (%)			6.18	0.01			0.38	0.54
Male	89	11			83	17
Female	68	32			78	22
Ethnicity (%)			8.85	0.03			6.15	0.09
White	90	10			89	11
Black	65	35			63	37
Hispanic	75	25			75	25
Other	57	43			86	14
Relationship to recipient (%)			4.55	0.47			6.93	0.23
Recipient is older brother	84	16			71	29
Recipientis younger brother	80	20			91	9
Recipient is older sister	84	16			72	23
Recipientis younger sister	78	22			82	18
Recipient is twin	75	25			50	50
Recipient
Age, mean (sd)	10.3 (5.5)	14.3 (7.6)	-2.16	0.04	10.9 (3.5)	12.2 (3.4)	-1.43	0.16
Sex (%)			0.04	0.83			0.53	0.47
Male	79	21			83	17
Female	81	19			77	23
Parent/Guardian
Respondent relationship to donor (%)			4.10	0.12			1.01	0.61
Mother	81	19			83	17
Father	79	21			74	26
Other	0	100			100	0
Age, mean (sd)	40.3 (6.9)	43.4 (9.4)	-1.60	0.11	40.0 (7.5)	41.4 (6.0)	-0.67	0.50
Education (%)			0.81	0.37			0.31	0.58
< bachelor's degree	77	23			79	21
≥ bachelor's degree	85	15			84	16
Relationship status (%)			0.39	0.54			0.51	0.48
Married	81	19			82	18
Not married	73	27			73	27
Number of children (mean, sd)	3.5 (1.5)	4.0 (1.8)	-1.05	0.30	3.6 (1.5)	3.6 (1.6)	0.02	0.98
Income (%)			6.72	0.04			0.29	0.87
<$35,000/year	67	33			80	20
$35,000-$75,000/year	85	15			78	22
> $75,000/year	91	9			83	17

Three themes emerged in the open-ended responses: (1) whether or not the parent believed that the child's age was a positive or negative influence on the donor's experience, (2) whether the comments indicated he effects of age were physical or emotional, and (3) the parent's beliefs about whether the effects of age had something to do with the donor child either understanding or not understanding the donation process. The following statement is a hybrid of two actual parent quotes and represents all three themes.

I guess in a way it helped her manage her feelings because she didn't fully understand or comprehend what all was going to be involved.

This quote would have been coded as (1) age effect positive, (2) effects were emotional, and (3) parent believed that the child didn't fully understand the donation process. Total percent agreement across the three coding categories was 83%-96% -- Kappa 0.69-0.84.²²

We examined qualitative data by donor age group (5-7, 8-12, 13-18). See Table 2 for sample quotes for each theme by age-group. Across all age groups, parents reported that their child's age had a positive effect on donation experiences. Only 11/73 parents at 4-weeks and 6/72 at 1-year post-donation reported negative effects of age. Across all age categories, parents were more than twice as likely to mention emotional rather than physical consequences. When parents discussed emotional consequences, they often described the donation as being like an adventure for the donor, how the child's age helped him/her avoid potential fear/anxiety/stress, and the positive feelings of being able to help their ill sibling. When parents mentioned physical consequences, they often linked their child's age to being more resilient than adults, having the ability to bounce back, and to recover more quickly.

Table 2.

Examples of comments about the positive effects of age

Ages 5-7 “I think being young helped him to take it all in stride more. To him it was just an exciting adventure. Being young helped.” (age 6) “I guess with him being younger he was more resilient, and it didn't bother him as much as maybe somebody older. He handled everything appropriately.” (age 7)

Ages 8-12 “He recovered real quick, he was up and around quick, someone older may have felt the aches and pains more.” (age 9) “As far as I think she is a little more mature than if she was younger. Handled the stress well. Even though she was worried about her brother, I think being older she got over it pretty quick.” (age 12)

Ages 13-18 “Well because she's younger I think she bounced back pretty fast. Because she's younger- like a person my age would have aches and pains but because she's young and healthy she healed better.” (age 14) “Being 15 at the time, she was fully aware of what she was doing and contributing. I think emotionally she was much more prepared than anyone who was a lot younger. For her, it was very rewarding. There was no fear. She was wanting to do it full-heartedly (age 15)

Examples of comments about physical and emotional effects

Ages 5-7 Physical: “Because she didn't really know what to expect, she didn't know it was gonna hurt so it didn't hurt.” (age 7) Emotional: “He was very young, only 7 so he didn't really have that 'scared' capacity, he just knew he was helping his sister” (age 7)

Ages 8-12 Emotional: “Emotionally at his age he just thinks he's just helping his brother. He wasn't thinking of the medical aspects, like being put to sleep, etc. He just thought the whole thing was cool, being in the hospital, getting special treatment was cool.” (age 8) Emotional: “Well because he was old enough to rationalize it and not be overly scared but was young enough to not overly think it.” (age 10)

Ages 13-18 Emotional: “Just cause we could explain it to him. I think he was emotionally capable of knowing what he was doing.” (age 13) Physical: “In a positive way. I think he was able to recover quickly because of his age. He was just, he was happy to do it. I think mostly because he was able to recover very quickly from it.” (age 14)

Examples of comments about donor's understanding of the process

Ages 5-7 “I don't think she fully understood the importance or the gravity of the situation, exactly what she was doing. You know she doesn't know the difference between bone marrow and blood. I think she was, I don't know if willing would be the right word, but maybe more willing to do it because she didn't understand everything that was going to happen.” (age 6) “Probably because she doesn't know much about what was going on. For that reason, I think it went smoother. If she were older, it would've been difficult.” (age 6)

Ages 8-12 “Because she was old enough to understand why it was worth going through that and the impact she will have from it” (age 9) “If she was older, it might have been worse because she would have known more, and there would have been more fear there” (age 10)

Ages 13-18 “Just because at 13, they know a little more than younger children do. Like, he knew what he was doing and why he was donating. He was mentally capable of understanding that.” (age 13) I think that he is old enough to understand and be able to ask the questions that he wanted to ask in order for it to be less to him. I also think that he understood it more than if he would have been a lot younger.” (age 14)

Approximately 70% of parent respondents across both post-donation time points indicated that the donors' age was linked to their ability to understand the donation process – which, in turn affected (generally positively) their donation experience. Large majorities of parents of the youngest age group (65% and >80% at the two time points) reported that their child was too young to understand the process. For the middle age group, parental respondents were almost equally split between those who thought their child understood the process (and benefitted) versus those who did not (and benefitted), and for the oldest age group, 60% and ∼90% at the two time points reported that their child understood the process.

Finally, we dichotomized PedsQL scores as above or below the at-risk cutoff and compared the groups by parents' reports of whether the child's age made a difference (yes/no) and their responses to the open-ended follow-up question. There were no significant differences in the percentage of parents who indicated that their child's age made a difference by cutoff score nor were there any obvious patterns of difference in their responses to the open-ended questions.

Our previous work indicated that ∼20% of sibling pediatric HSC donors experience poor HRQoL during and following donation, that the youngest donors appear to be most at-risk for poor HRQoL, and that parental proxy reports overestimate donor HRQoL.¹⁸ Although the question of whether poor pediatric donor HRQoL is a function of donation itself or of being part of a family with a chronically and potentially terminally ill child remains unanswered, these analyses provide additional insights about parents' beliefs about their donor child's donation experiences.

A first key conclusion is that the majority of parents (75%-80%) believed that their child's age made a difference in how they experienced the donation process. Second, parents were overwhelmingly likely to report that their child's age had a positive effect on the donation experience (85%- 90% of parents reported positive effects of age) and positive emotional effects were more often reported. Finally, 70% of parents linked their child's experience (positive or negative) to his/her ability to understand the donation process. Parents of the youngest group of donors, the group with the highest risk of poor HRQoL in our previous work, most often believed that the donation experience was positive because their child was too young to understand the process. Parents of the oldest group attributed the positive experience to their child's ability to understand. We found no quantitative or qualitative differences in how parents responded based on whether their child self-reported very poor HRQoL versus not.

Findings from this investigation are encouraging, in the sense that parents view the donation experience as generally positive for their donor children. However, they are also cautionary because they point out that some parents are unable to accurately recognize HRQoL deficits that some child donors experience around the donation/transplantation process. Ultimately, this investigation highlights the urgent need for additional investigations focused on the sources of poor HRQoL among some pediatric donors and the apparent discrepancies between parents' reports about their child's donation experiences and the child's self-reported HRQoL scores.

Abbreviations

HSC

Hematopoietic Stem Cell

HRQoL

Health-related Quality of Life

RDSafe

RDSafe: A Multi-Institutional Study of HSC Related Donor Safety and Quality of Life