Abstract
Background:
Caregivers of children with intestinal failure (IF) face difficult decisions without a clear best alternative. Providers assist in decision-making but often lack knowledge of caregiver perspectives. using decision-making around anemia treatment as a focal point, we explored how caregivers of children with IF prefer to make decisions. Our goal was to offer insight to guide providers as they assist in decision-making.
Methods:
We conducted 12 half-hour semistructured interviews with parents of children with IF. interview questions addressed general decision-making and specifics of iron supplementation, including key factors and stakeholders in decision-making. interviews were transcribed verbatim. Two investigators coded the transcripts and inductively derived themes.
Results:
Four themes were identified regarding decision-making. They involved the search for reliable, accurate, and positive information; the role of caretakers on the medical team; the relationships between caretakers and the medical team; and effective communication. Themes around anemia treatment included: identification of anemia by bloodwork; proactive supplementation; individualized regimens; prioritizing safety and convenience.
Conclusions:
Understanding caregiver perspectives regarding anemia treatment in pediatric If identifies opportunities for systematic quality improvement.
Keywords: chronic disease, clinical guideline, parenteral nutrition, semistructured interviews, short bowel syndrome
Children with intestinal failure (IF) require parenteral nutrition (PN) for adequate nutrition (1). Conditions leading to IF can present within the first few days of life, resulting in a chronic illness that requires significant caregiver support. Medical providers must guide families through many difficult decisions through the disease course.
Iron supplementation presents a unique challenge in this population. Iron deficiency contributes to anemia, often necessitating blood transfusions (2–4). Iron supplementation can be given either enterally or parenterally to treat anemia, alleviate associated symptoms, and lessen the need for blood transfusions. Enteral iron can have limited absorption or poor palatability in a population with frequent gastrointestinal symptoms and oral aversion (5). Parenteral iron requires access of the central line and must be given separately from the PN mixture because of incompatibility with many lipid emulsions (4). As such, deciding an optimal iron supplementation strategy for children with IF represents a complex decision involving medical and personal factors. A survey of IF dietitians showed that iron supplementation is often only recommended in response to deficiency and that monitoring practices vary between centers in terms of specific bloodwork and frequency (6). At our own institution, we previously found that many children with IF were not receiving routine iron supplementation and that monitoring was not standard between providers (7).
Because of insufficient information about caretaker preferences, providers may struggle to effectively support this decision-making process. This may interfere with the caregiver’s ability to make the best decision for their child and their situation. Lack of information has been identified as a major barrier to utilizing shared decision-making in pediatrics (8). We aim to inform providers how to approach iron supplementation in children with IF. To develop such guidance, we studied the values (what a person feels is important), beliefs (how a person thinks things work), and preferences (wishes) of caregivers of children with IF focusing on their decision-making processes regarding anemia treatment.
METHODS
We used qualitative methods to elucidate the values, beliefs, and preferences of caregivers of children with IF regarding iron deficiency anemia treatment. A single primary caregiver for each patient participated in half-hour semistructured interviews via phone. Caregivers were the primary participants as many children were not developmentally able to participate in the study. Interviews were preferable to focus groups to allow researchers to discern individual viewpoints. We used purposive sampling by reviewing medical records of our institutional roster of children with IF to identify those with anemia. We aimed to recruit enough caregivers to achieve thematic saturation, which typically occurs between 12 and 20 interviews in a homogeneous population (9). Caregivers whose children were identified by medical record review were approached face-to-face at their routine intestinal rehabilitation clinic visits to obtain consent. This study was approved by the University of Pittsburgh Institutional Review Board (STUDY# 19050168).
The principal investigator (PI) developed the interview guide, which was reviewed, refined, and approved by all members of the research team before conducting the first interview (see Table, Supplemental Digital Content 1, http://links.lww.com/MPG/C24, which contains the final interview guide). The interviews focused both on questions specific to iron supplementation and general decision-making preferences.
The PI, who had experience working with this population and basic training in qualitative research, conducted each interview. Interviews were audiorecorded and transcribed verbatim. The PI and 1 additional investigator reviewed all transcripts and developed a codebook inductively. The final codebook was applied to all transcripts by these 2 investigators using NVivo (10) with adjudication of all coding for full agreement. A senior author was available to resolve disagreements during adjudication. The investigators performed thematic analysis using the methods described by Braun and Clarke (11) to inductively derive themes about anemia treatment and decision-making more broadly.
RESULTS
Fifteen caregivers, all biological parents, were recruited, and all consented to participate. Three interviews were not completed because of scheduling conflicts after consent. This analysis included transcripts from the 12 completed parent interviews. Thematic saturation was felt to be achieved by the 11th interview. Table 1 contains demographic information of the parents and children. In the following sections, we will discuss the themes that emerged regarding decision-making in general followed by themes regarding anemia treatment. Table 2 summarizes these results.
TABLE 1.
Caregiver and child characteristics
| Caregiver sex, female (%) | 11 (92%) |
| Child age, median (range) | 7 y (17 mo to 15 y) |
| Child sex, female (%) | 7 (58%) |
| Diagnosis, n (%) | |
| Hirschsprung disease | 4 (33%) |
| Abdominal wall defects | 3 (25%) |
| Megacystis-microcolon-intestinal Hypoperistalsis syndrome | 2 (17%) |
| Necrotizing enterocolitis | 1 (8%) |
| Midgut volvulus | 1 (8%) |
| Other | 1 (8%) |
| Child history of anemia, n (%) | 12 (100%) |
| Child anemia treatment, n (%) | |
| Enteral iron | 10 (83%) |
| Parenteral iron | 9 (75%) |
| Blood transfusion | 11 (92%) |
TABLE 2.
Summary of themes
General-decision making among caregivers
|
Anemia treatment
|
Decision-making
Four themes identified regarding decision-making are discussed below.
Caregivers Search for Reliable, Accurate, and Positive Information, Often from Other Families and Support Groups
Immediately after diagnosis, caregivers searched for information about their children’s condition, many turning first to the internet. What they found online was, however, usually not comforting. Much of the information was grim, leaving them feeling alarmed and worried. Several caregivers described searching for information online that provided an “inkling of hope” (P7) yet rarely finding it. It was not always immediately apparent to them that some of this information was outdated and unreliable. For instance, 1 mother described reading online that her child was unlikely to live beyond its first birthday, only to learn later from the medical team that the prognosis was far better.
Many caregivers joined Facebook groups to directly communicate with families with firsthand experience living with IF, often learning more current information than from older publications. One person said the Facebook group she belonged to provided “a good community for the parents, giving advice in a way [we] all kind of understand” (P13). For some, the value of that “wider perspective” led them to “turn [to social media] first and the doctors second” (P6). As 1 mom said, “Going to parents who have gone through similar issues […] often provides more benefit than trying to do research online through medical journals and things like that because they live the nitty gritty of it” (P4).
Caregivers Feel that They Are Members of the Medical Team, Which Comes With the Responsibility of Being Involved in Medical Decisions
Caregivers quickly assimilated to medical culture and viewed themselves as members of the medical team responsible for making medical decisions. As a caregiver became more comfortable, they assumed more responsibility, including checking labs and “look[ing] for trends” (P13). They would then call the outpatient nurse to discuss a treatment plan with some caregivers proposing the plan themselves. One caregiver felt that she has “made the best decisions […] often with very limited input [from providers]” (P4). Thus, despite that initial search for information and lack of medical training, caregivers assumed a medical provider role with the associated duties and responsibilities. Often, 1 caregiver had the “ultimate final say” (P6) with others deferring decision-making. Many shared a view that “at the end of the day, its [their] responsibility” (P8) as caregivers. Occasionally, this led to a feeling of remorse if a decision led to a poor outcome or, more commonly, if a test result was not immediately acted upon. For example, caregivers described regret that they did not proactively request iron supplementation if it had not been started by the medical team.
Caregivers Have Strong Often Personal Relationships With the Medical Team that Develop Over Time, Which Makes Interactions With Unfamiliar Providers Difficult
In addition to increasing medical comfort over time, caregivers felt strong attachments with their providers. For some, the “medical team is like [their] family” (P9) or they noted “a special connection” (P3) between their child and a particular provider. The connection was difficult to describe but associated with an expectation that providers would carry a complete knowledge of their children, as is often the case with intestinal rehabilitation teams and their patients.
This also led to negative interactions with newer or unfamiliar providers. For example, 1 caregiver was dismissive when describing a new provider, saying said that she “doesn’t value [that provider’s] opinion as much as somebody that [she] know[s]” (P3). Another caregiver expressed frustration with “people who don’t know [her] kid” (P4). Unfamiliar providers also seemed less willing to accept the medical expertise that experienced caregivers felt they had obtained, ranging from performing line care themselves to participating in medical decision-making. These interactions led caregivers to seek out familiar providers. For instance, some caregivers only felt comfortable with unfamiliar attending physicians when they were accompanied by a familiar nurse practitioner. This mistrust with new providers could have a profound impact on medical decision-making in the way families engage in shared decision-making.
Effective Communication Improves the Decision-making Process, Including Sitting Down, Educating the Caregiver, and Eliciting Their Preferences
Caregivers brought up 3 main components of effective communication. Effective communicators were “sitting down” and “face-to-face” (P14). They used “terms that you would use with a 12-year-old” and “anything that’s illustrative or like a picture” (P7) to explain medical concepts. Caregivers expected their “opinion is very much taken into every single decision” (P13). One simply said, “You’re missing the boat if you’re not involving the parents” (P5). Conversely, a few caregivers expressed sentiments, such as “I’d just rather have a doctor just let us know what to do” (P1), hoping the medical team could make decisions for them. The context needs to be carefully considered as making decisions for a family can also lead to ill effects. As 1 mother said about the stress of working with the medical team, “You feel like you have no control. I really don’t feel like he’s my child. I feel like he belongs to the medical team. I feel like they make the decisions” (P6). In the end, the majority of caregivers agreed that at least eliciting their preferences played an important role in decision-making.
Treatment of Anemia
The following 4 themes arose specific to anemia treatment.
Caregivers Could not Identify Anemia Reliably by Symptoms Alone, Relying on Blood Test Results
Caregivers described many ways of determining if their children were anemic. Some noticed behavioral changes and decreases in energy level. Others focused more on numbers, citing low hemoglobin, ferritin, or blood cell count. No matter whether they could identify symptoms or not, every caregiver made reference to their child’s anemia being diagnosed by bloodwork. In fact, caregivers commonly only found out their children were anemic after a blood test. One mom said that her daughter “can be all active and [mom] get[s] a call from the hospital that she needs blood or she needs iron” (P9). Caregivers could not identify consistent signs or symptoms of anemia outside of blood test results.
Caregivers Prefer Pre-emptive Iron Supplementation Over Reacting to Iron Deficiency
Iron supplementation often started after a diagnosis of anemia. In some cases, the anemia was severe enough to require a blood transfusion before starting iron. One caregiver commented that she “think[s] it’s fairly obvious in retrospect that a kid in intestinal failure who’s only getting [PN] is going to get iron-deficient anemia” (P5) as iron cannot easily be added to PN. Caregivers expressed negative feelings towards a system of reacting to anemia, comparing this to “flying by the seat of their pants” (P4). As such, they clearly preferred providing iron supplementation even before the development of anemia or iron deficiency.
Each Caregiver Wanted a Plan that Met the Specific Needs of Their Child
In treating anemia, caregivers felt that the unique needs of their children needed to be considered. One caregiver whose child receives home iron infusions felt that for them “that really made sense” but that she did not “think it’s the right choice for all families or for all kids” (P6).
For those with some enteral tolerance, caregivers preferred oral therapy. Some caregivers liked the idea of providing iron-containing foods but others quickly pointed out that their child “can only receive so much from his diet” (P2). In terms of oral supplements, families found taste and absorption to be limiting factors. One caregiver whose child receives enteral supplements said, “if the kid doesn’t have a G-tube, I would find it really hard to believe that they’re going to take the iron by mouth” (P12).
For some, oral iron is not even considered as the child “doesn’t absorb anything really” (P7). Intravenous iron provides a well tolerated, effective alternative. That made it “the easiest ‘yes’ [one caregiver] ever said because [they] can do them at home and it’s not another trip to the hospital” (P4). Several caregivers shared this sentiment although one cautioned that they faced a barrier because of lack of a home health nurse. Receiving therapy at home limits disruptions to a family’s normal schedule, as 1 caregiver reminded that she isn’t “just like living on vacation” (P3) and that these interruptions mean “a day off work, […] a day off school” (P3) to a family already dealing with many such days.
Thus, families had many individual patient-level factors that had a significant impact on their preferred iron therapy.
Caregivers Identified Safety and Convenience as Important Factors in Determining Iron Therapy
In each discussion, families held 2 values above all else, namely safety and convenience. From a safety standpoint, families found blood transfusions to be the least safe, and thus preferred some form of iron therapy. As 1 mother said, “it makes you a little bit nervous so you try to think about any alternative that you could do to try to avoid [a blood transfusion]” (P3). As such, parents supported iron therapy as a preventative approach. Parents also preferred to avoid additional infection risk by limiting central line access that would be required with parenteral iron. Generally, parents who had used enteral iron had no concerns about safety.
Families identified convenience as a key driver of choosing parenteral iron. For some, enteral iron could be conveniently given at home, potentially through a gastrostomy tube. For others, convenience meant a home nurse administering parenteral iron acknowledging the low but present additional safety risk. A key part of convenience was avoiding trips to the hospital, both for blood transfusions or parenteral iron for those who lacked access to a home nurse.
DISCUSSION
Children with IF require complex care coordinated between the medical team and their caregivers. These teams must collaboratively navigate challenging decisions throughout each child’s life. We provide guidance regarding values, beliefs, and preferences towards decision-making among the caregivers of these children with a focus on anemia treatment.
Caregivers invoked many aspects of the well-described Chronic Care Model (CCM) in children (12–14). The CCM has been successfully used as a framework for improvement in pediatric chronic illness (12). The CCM describes how a combination of community and healthcare resources can strengthen the collaborative efforts of a motivated patient and a dedicated provider to produce quality care. In pediatrics, the motivation includes both the patient and caregiver. Community resources provide Self-management Support for the caregiver, which aligns with our identified themes regarding the desire for support groups, the identification of the caregiver as a medical team member, and the desire for a safe, convenient treatment individualized to each particular child. Health systems resources are divided into Delivery System Design, Decision Support, and Clinical Information Systems. The Delivery System Design incorporates the direct interaction between patient, caregiver, and provider. This corresponds with our themes describing the personal relationship between providers and families and the importance of effective communication. Decision Support refers to the use of evidence-based practice, which would be incorporated in the theme regarding proactive iron supplementation. Finally, Clinical Information Systems could be designed to incorporate routine monitoring of anemia since caregivers relied on blood tests for the diagnosis. Thus, although we used an inductive strategy to code and arrive at themes, the results fit into a well-established paradigm of providing chronic illness care, strengthening these results.
Incorporating the CCM into our results, we can make several recommendations regarding anemia treatment. First, we should routinely check hemoglobin and iron indices such as ferritin and total iron binding capacity in all at-risk patients in a way that can be integrated into the existing infrastructure. While each of these tests has limitations, we believe that simply increasing awareness of iron deficiency will improve treatment. Second, we should proactively supplement iron for these children before anemia development. Although our qualitative study cannot define strict guidelines for supplementation, it would be reasonable to attempt enteral iron before proceeding with parenteral iron in any child who may tolerate and absorb it. Third, a personalized protocol should be developed for each patient through a discussion with the family. This can be considered similar to developing a unique PN prescription for each patient and should include the aforementioned monitoring pattern. Finally, and perhaps most importantly, we must consider the value of involving caregivers in decision-making, at least in those who want to participate. Currently, much of their information comes from outside the medical team, including through social media. As families appreciate support from those who have been through the experience, we will be most successful by partnering with these support groups to create a cohesive community. This may be achieved through provider activity on social media or hospital-led parent groups or patient/parent-mentoring programs as part of routine care.
Qualitative interviews are hypothesis-generating methods. The results from this small sample will need to be evaluated in a broader population. We, however, did reach thematic saturation with a reasonable sample size despite the heterogeneity in age, diagnosis, and enteral tolerance that may affect responses. The views expressed in these interviews may not be generalizable but can be used to inform further inquiry. In particular, we plan to use our results to implement a quality improvement initiative in improving iron supplementation in our local population. Recently, it has been suggested that leveraging clinical informatics to create a learning health system, as has been successful in inflammatory bowel disease, could be an attractive way to implement a broader quality initiative in intestinal rehabilitation, although such efforts have not been successfully implemented to date (15). This work provides preliminary evidence that such an effort would not only be an effective way to improve care but also it is what caregivers of these children want.
CONCLUSION
Caregivers of children with chronic IF prefer an individualized but protocolized approach to anemia treatment. The CCM covers many important values, beliefs, and preferences of these caregivers when they discuss decision-making. Quality improvement initiatives using this framework may generate significant enhancements in medical care and quality of life.
Supplementary Material
What Is Known
Children with intestinal failure face many difficult decisions during their life.
Despite little guidance, providers must aid families with these decisions.
Iron supplementation can be challenging in children with intestinal failure because of poor tolerability of enteral iron and administration barriers to parenteral iron.
What Is New
Despite a strong trust in the medical team, caregivers frequently seek input from social media.
Caregivers consider themselves a member of the medical team, sharing in the responsibility of decision-making.
Caregivers prefer a protocol for iron deficiency anemia treatment that includes routine monitoring and pre-emptive supplementation.
Acknowledgments
This study is supported by NIDDK 5T32DK063922-17.
Footnotes
The authors report no conflicts of interest.
Supplemental digital content is available for this article. Direct URL citations appear in the printed text, and links to the digital files are provided in the HTML text of this article on the journal’s Web site (www.jpgn.org).
REFERENCES
- 1.Squires RH, Duggan C, Teitelbaum DH, et al. , Pediatric Intestinal Failure Consortium. Natural history of pediatric intestinal failure: initial report from the Pediatric Intestinal Failure Consortium. J Pediatr 2012;161:723.e2–8.e2. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 2.Yang CF, Duro D, Zurakowski D, et al. High prevalence of multiple micronutrient deficiencies in children with intestinal failure: a longitudinal study. J Pediatr 2011;159:39.el–44.e1. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 3.Ubesie AC, Kocoshis SA, Mezoff AG, et al. Multiple micronutrient deficiencies among patients with intestinal failure during and after transition to enteral nutrition. J Pediatr 2013;163:1692–6. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 4.Hwa YL, Rashtak S, Kelly DG, et al. Iron deficiency in long-term parenteral nutrition therapy. JPEN J Parenter Enteral Nutr 2016;40:869–76. [DOI] [PubMed] [Google Scholar]
- 5.Powers JM, Nagel M, Raphael JL, et al. Barriers to and facilitators of iron therapy in children with iron deficiency anemia. JPediatr 2020;219:202–8. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 6.Nucci AM, Ellsworth K, Michalski A, et al. , ASPEN Pediatric Intestinal Failure Section. Survey of nutrition management practices in centers for pediatric intestinal rehabilitation. Nutr Clin Pract 2018;33:528–38. [DOI] [PubMed] [Google Scholar]
- 7.Raghu VK, Rudolph JA, Jalal HJ, et al. Microsimulation model to compare enteral and parenteral iron supplementation in children with intestinal failure. JPEN J Parenter Enteral Nutr 2020[online ahead of print]. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 8.Boland L, Graham ID, Legare F, et al. Barriers and facilitators of pediatric shared decision-making: a systematic review. Implement Sci 2019;14:7. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 9.Guest G, Bunce A, Johnson L. How many interviews are enough?:an experiment with data saturation and variability. Field Methods 2006;18:59–82. [Google Scholar]
- 10.NVivo qualitative data analysis software [Computer Program]. Version 12. QSR International Pty Ltd; 2018.
- 11.Braun V, Clarke V. Using thematic analysis in psychology. Qual Res Psychol 2006;3:77–101. [Google Scholar]
- 12.Adams JS, Woods ER. Redesign of chronic illness care in children and adolescents: evidence for the chronic care model. Curr Opin Pediatr 2016;28:428–33. [DOI] [PubMed] [Google Scholar]
- 13.Bodenheimer T, Wagner EH, Grumbach K. Improving primary care for patients with chronic illness: the chronic care model, part 2. JAMA 2002;288:1909–14. [DOI] [PubMed] [Google Scholar]
- 14.Bodenheimer T, Wagner EH, Grumbach K. Improving primary care for patients with chronic illness. JAMA 2002;288:1775–9. [DOI] [PubMed] [Google Scholar]
- 15.Mezoff EA, Minneci PC, Hoyt RR, et al. Toward an electronic health record leveraged to learn from every complex patient encounter: health informatics considerations with pediatric intestinal rehabilitation as a model. J Pediatr 2019;215:257–63. [DOI] [PubMed] [Google Scholar]
Associated Data
This section collects any data citations, data availability statements, or supplementary materials included in this article.