Caregiver Experience for Children Living with Digestive Tract Congenital Anomalies in Uganda: A Qualitative Study

Congenital anomalies are a significant cause of neonatal morbidity and mortality. The incidence of congenital anomalies approaches 1 in 1,500 live births, with 8.3% of all congenital anomalies relating to the gastrointestinal tract.[1] These anomalies affect 3-6% of live births globally, but low- and middle-income countries (LMICs) shoulder a higher prevalence and account for 95% of disease-related deaths.[2] Physicians in LMICs who treat these anomalies face a lack of specialized care units, insufficiently trained personnel, limited financial resources of families and caregivers, and poor patient follow-up.[3] These challenges persist even in areas where pediatric surgical services are available.[2] Challenges in caring for children with digestive tract congenital anomalies (DTCAs) persist for families at home. Qualitative data from studies on caregiver experiences in LMICs found that caregiver uncertainty and worry contributed to financial and familial instability and significant burden associated with the complex care of children with congenital anomalies despite surgical correction.[4, 5]

Programs exist to help address the burden that caregivers in LMICs face. The World Health Organization (WHO) Training of Trainers program and companion congenital disability manual for Community Health Extension Workers (CHEWs) expedited the surveillance and treatment of congenital anomalies in 9 sub-Saharan African (SSA) countries.[6] The WHO and UNICEF recognize that facility-based services are often insufficient to provide prompt and equitable care and that CHEWs are a viable alternative.[7] CHEWs have been deployed in 16 SSA countries and have improved survival for most high-burden childhood conditions in LMICs.[8] The success of CHEW programs in managing childhood conditions and promoting community engagement and education could serve as a useful model to apply to other conditions that affect these communities. This study proposes applying the CHEW model to caring for children with DTCAs, which frequently lead to prolonged disability pre- or post-operatively, to help address gaps in care and community understanding of these conditions.

Congenital anomalies are a significant challenge in pediatric health, particularly in sub-Saharan African (SSA) countries. In fact, 30% of the estimated 8.5 million newly diagnosed congenital anomalies were in SSA, and congenital anomalies were the 5th leading cause of death worldwide for children under 5 years in 2019.[9] In Uganda, only a fraction of children with DTCAs undergo surgical repair at tertiary care centers, underscoring a substantial unmet need.[10] Surgical correction of DTCAs is often staged with ostomies for gastrointestinal diversion. Living with an ostomy can have significant psychological, physical, and socioeconomic implications for patients and their families due to the constant attention required for specialized ostomy care.[11]

This qualitative study investigated the lived experiences of caregivers of children with DTCAs with and without stomas who were treated at a tertiary care center in Uganda through focus groups. The study findings highlighted the utility of establishing a CHEW program to facilitate healthcare in a community-based setting to help address the unmet needs of children’s surgical services and follow-up care.

Study Aim and Design

This qualitative study utilized thematic analysis of focus group discussions (FGD). The overarching goals of this study were to 1) understand the chronic burdens and lived experiences of caregivers of children living with DTCAs in Uganda and 2) identify opportunities where healthcare workers can facilitate care in the community setting. The focus group format was chosen to encourage caregivers to share similar or disparate experiences and organically form a consensus on shared barriers that may be mitigated with a CHEW program through participant discourse. The study followed the Consolidated Criteria for Reporting Qualitative Research (COREQ) guidelines.[12]

Research Team

The research team comprised a male Ugandan pediatric surgeon (NK), a male Ugandan surgical resident (BK), a female Canadian surgical resident (AY), a male Ugandan medical student (DB), a female American medical student (ER), and two female Ugandan registered nurses who worked in the inpatient wards and perioperative space (AN, AJ). Team members were selected for their diversity in demographics, pediatric surgical expertise, and training. All team members completed the prerequisite qualitative methods training. In February 2023, AN and AJ conducted and moderated in-person focus groups at tertiary care centers. Notably, AN also had prior clinical relationships with many focus group participants, which benefited the flow of the FGD conversation. Since participants were frequently faced with stigma from their child’s diagnosis, it was common for them to be reticent to share their personal stories of their care experiences. Therefore, it was important to have a focus group facilitator with existing therapeutic relationships with the participants to create a safe space to share experiences.

Participant Selection and Recruitment

Participants were selected via purposive sampling. Eight DTCA diagnoses were included: anorectal malformation, Hirschsprung’s disease, cloaca, omphalocele, gastroschisis, tracheoesophageal fistula, intestinal atresia, and malrotation. All focus groups included caregivers of children with DTCAs with and without stomas, as patients with stomas garnered caregiver perspectives around stoma care, a challenge unique to this subgroup. The inclusion criteria were caregivers of patients with DTCAs admitted to the tertiary care center pediatric surgical unit at least once. The FGDs were conducted only in Luganda, so we excluded caregivers who did not speak Luganda to minimize language barriers in the group. Caregivers whose children had died or who were admitted less than six months after participant recruitment were also excluded, as they were either no longer actively providing care or had not yet been in the caregiving role long enough to provide insight into chronic caregiving burdens.

Caregivers were contacted via telephone, with contact numbers obtained through an inpatient ward database. This call involved a recruitment pitch, verbal informed consent, an intake phone survey, and an invitation to an in-person FGD. During the recruitment phase, participants were compensated for their time via 20,000 Ugandan Shillings (UGX) in mobile money.

At the beginning of each FGD, researchers AN and AJ summarized the focus group contents, risks, benefits, and alternatives when needed, and participants were allowed to ask questions or decline to participate. Those caregivers who agreed to participate in the FGD signed a written consent before each FGD commenced. After the FGD, participants were reimbursed with 50,000 UGX in cash to cover their time and travel.

Participant recruitment started at the time of the first survey on December 13, 2022, and ended on February 19, 2023. Protected health information (PHI) was stored in a secure, encrypted device and erased after recruitment was completed. The original health-related information, housed in a secure local hospital system, was accessed with authorization.

Ethical Consideration

Ethical approval and permission to conduct the study were obtained from the tertiary care center in Uganda (IRB number #2022-354) and the tertiary care center in California (IRB number 22-36957). All participants provided written consent before study enrollment.

Study Setting for Data Collection

FGDs were held in a conference room within the hospital’s pediatric surgical unit. Given the lack of alternative childcare, participants were often accompanied by their child(ren) in the FGDs. A facilitator guide (Supplementary S1) was used to generate discussion, focusing on caregiver burden, child quality of life, and suggestions for a potential CHEW or similar outreach program. The main moderator (AN) fielded responses, while the assistant moderator (AK) documented field notes. Focus groups were conducted in Luganda, the local language of central Uganda. Each focus group lasted approximately 60 to 90 minutes, and voice audio was recorded. Transcription from audio recording into written Luganda and translation from Luganda to English were performed externally by a local, experienced, and bilingual social scientist. The accuracy of the transcription was ensured by comparing the transcription to the audio recordings.

Data analysis

Using thematic analysis, AY reviewed the transcripts and used open coding to identify response patterns, then 4 team members (AY, BK, ER, BD) iteratively tailored the codebook to optimize coder concordance over two rounds of coding a sample transcript. Codes were identified through transcript review and refined with pre-existing studies and constructs investigated in the facilitator guide. To reach code saturation, there had to be no new codes found in the third FGD reviewed by the team. The structured codebook is showcased in Table S2 in the supplementary materials. The same 4 team members (AY, BK, ER, BD) then acted as primary and secondary coders for each FGD transcript, such that a Ugandan researcher could code all transcripts. Intercoder agreement was measured by comparing the codes of the same FGD transcript that was coded independently by different team members. Areas of coding discrepancy were addressed by team-wide discussion until agreement on a single code for the section in question. Code reports were generated using Dedoose software.[13] Emerging themes were derived from reviewing and establishing relationships across transcripts in the aggregated code reports.

Participant Characteristics

A total of 30 caregivers participated in 4 FGDs during February 2023. The average FGD lasted 1 hour and 10 minutes. Most caregivers were mothers, though two fathers and one grandmother also participated. The median caregiver age was 32 years, and the majority (29/30, 97%) came from the surrounding neighborhoods of Wakiso, Mukono, and Kampala. A summary of caregiver and child demographics is shown in Table 1. Table S3 in the supplemental materials showcases individual patient characteristics, including the patient’s age, diagnosed DTCA, the procedure they underwent, and their age at the time of their first surgery.
 
Overview of Findings

The caregiver burden of children with DTCAs centered around the substantial time, financial cost, and material resources required for specialized childcare. Exacerbating factors included social stigma and isolation from community and family members, which stemmed from unfamiliarity with the DTCA diagnosis, and concern for the DTCA’s effect on the child’s quality of life and developmental delays. Alleviating factors of caregiver burden were faith and spiritual strength, positive interactions with healthcare workers, and collaborative support from spouses, family, and community members. This network of factors involved in the caregiver experience is shown in a mind map (Figure 1). Themes identified were:

1. Delays in definitive care hindered child development and adversely impacted the child’s and caregiver’s quality of life.
2. The community’s support was proportional to its appreciation of the child’s DTCA condition; minimal understanding led to social stigma, while faith-based empathy led to caregiver assistance.
3. Childcare for a DTCA patient carried a “double tax” of heavy financial cost and inability to finance these costs.
4. Healthcare worker (HCW) communication and education are critical to instilling caregiver preparedness.
5. A CHEW program with home visits could support caregivers of a child with a DTCA.

Major Themes

Theme 1: Delays in definitive care hindered child development and adversely impacted quality of life
Delays in definitive surgical repair were common for children living with DTCAs, hindering their development and suppressing their quality of life. The three delays framework summarizes caregiver experiences into the following categories: 1) failure to recognize symptoms related to the DTCA outside or within the medical system, 2) failure to access care due to confusing referral and discharge systems, and 3) extended waiting periods for necessary surgeries after seeking appropriate medical attention.[14] These treatment delays only added to the caregiver’s burden and economic strife.

Difficulty in diagnosing a DTCA at birth stymied finding appropriate care within the medical system, as one participant’s daughter narrated her daughter’s first days of life: “We gave her some water, but she could not keep it down. [The hospital] discharged us but told me to go to a clinic… People in clinics try to manage conditions that they do not understand.” (FG1). Another participant said, “You bring a sick child, and you have to go back without help. You feel like you have done nothing, and yet you will have spent money on transport, and the clinic attendants back there send us away” (FG2). Unfamiliarity with DTCAs in peripheral health centers led to unclear follow-up and referral plans, wasting caregiver time and resources. Financially strained caregivers remained helpless and uncertain about where to seek treatment for their child’s ailments.

Even after an appropriate diagnosis, definitive management of DTCAs was frequently prolonged due to a lack of hospital resources and surgical capacity. The waiting times dragged on for months or even years without answers, wearing on caregivers, especially those with children who required multiple surgical revisions. “That prolonged waiting time actually can make one lose hope. You get fed up.” (FG1). During the long wait, children often developed health issues such as weakness, fever, irregular bowel movements, and delays in reaching developmental milestones. These difficulties significantly impacted their everyday routines, impeded progress in toilet training, and restricted their engagement in various activities and socialization.

Theme 2: The community’s support was proportional to its appreciation of the child’s DTCA condition; minimal understanding led to social stigma, while faith-based empathy led to caregiver assistance.
FGD respondents frequently commented on the community’s lack of familiarity with DTCAs, which either materialized as an unsubstantiated fear of contracting the DTCA or unsolicited stares, both of which led to the child’s discrimination and ostracization. One respondent shared, “They fear letting their children play with him, saying that they will contract his things, such as the [ostomy]… Even the neighbors fear him” (FG2). The uncertainty surrounding DTCA led community members to display superficial pity but withhold true solidarity. One caregiver reported: “They feel sorry for you, but when they leave you, they start backbiting you.” (FG4). In this way, DTCAs’ novelty sensationalized the child’s plight into gossip, exacerbating the stigma against the caregiver and child. Other community members erroneously ascribe blame to the caregiver inflicting the DTCA on their child: “We encounter that problem of people asking what happened to the child or what you did to them. You get fed up with responding to them.” (FG3).

Stigma and isolation also stemmed from the caregiver’s spouse, who is usually the child’s father. Primary caregivers were almost exclusively mothers and reported that their husbands often blamed the mother for the child’s DTCA, denied paternity of the child, or even deserted the family. One respondent shared: “When I had my child, the father was not around. He had left. I was in the house alone, yet I had older children at home. I had to support them, and I had no way of looking for money because I was still caring for the baby. I had no help. I went hungry” (FG 3). Another respondent shared, “The child’s father refused to tell the family that he had a child because of that issue, and it wasn’t until all the operations were done that he went home and told them that he had a child” (FG 4). Spousal isolation contributes to the primary caregivers facing the community and their families.

Conversely, some fathers steadfastly supported the mother and child and maintained their spousal relationship. One of the two father participants said, “I have never abandoned my wife. For all the needs we have been working together. I did not want to let her down because she did not choose this situation. It was God’s choice” (FG 2). In this case, the father believed that the child’s condition was not the mother’s fault or due to circumstances directly under their control, which motivated him to play an active caregiving role. Respondents often relied on their faith in challenging situations and ascribed unlikely recovery and survival after surgical correction to God’s actions or “miracles.” This helped caregivers cope with the insurmountable difficulties of caring for their child’s condition.

Theme 3: Childcare for a DTCA patient carried a “double tax” of heavy financial cost and inability to finance these costs.
Caring for a child with a DTCA was financially destabilizing due to their specialized healthcare needs. Every aspect of childcare requires substantial time and money, from traveling to and from the hospital for repeated clinic visits to buying medical supplies to feeding and clothing the child. At times, destitute families could not afford their child’s necessary medical care entirely. As one respondent recounted witnessing a fellow caregiver’s harrowing experience: “…she did not even have a cloth to cover the baby. Then they told her to go and buy medicine. She just walked off and left.” (FG3). Hospital visits incurred the highest expenses. Medical costs accumulated abruptly and were financially catastrophic during hospital visits, which could extend over many days without upfront notice. As such, childcare frequently required all the savings and earnings of both parents. Some families resorted to selling possessions or fell into debt to pay for medical care, particularly surgery. Others had to ration their food intake for the sake of their child: “It’s about being creative and sacrificing. This means if you have been eating this or that, then you have to forgo them so that the child can be well” (FG1).

Paradoxically, caregivers of a child with a DTCA were frequently unable to earn a living. Opportunity costs were steep as childcare was time-consuming. Often, caregivers could not maintain a stable job due to their caregiving responsibilities. Given the unique needs of a child with a DTCA, caregivers could not entrust external parties to watch their children. Community members echo similar sentiments on the caregiver’s inability to support their child financially: “What scares me the most is they talk about the money. ‘How will you manage the child? You do not work. You do not do anything.’” (FG1). Additionally, some employers refuse to hire given their negative preconceptions of these caregivers: “No one offers you a job when you have a child with an intestinal problem because people see you as unclean” (FG4). Thus, caregivers suffered from a “double tax” of unemployment risk when they had urgent financial needs to care for their child.

Economic support from spouse, family, and religious community was critical in this dependent state. Those who received this welfare support were extraordinarily grateful. One respondent shared:

“The financial situation was not good, but we got help from other people. Even spiritually, we continued to praise God because it was not easy. Even in the church that I go to or in my faith, we continue to confirm that God is mighty, and everything is possible because if it were not for God’s grace, this child would not have recovered because his condition was very difficult” (FG3).

In fact, several caregivers’ hope in coping with the financial burden rested in their faith in God and their conviction that things would improve with supernatural intervention.

Theme 4: Healthcare worker (HCW) communication and education are critical to instilling caregiver preparedness.
Caregivers often felt unprepared to care for their child with a DTCA because of the special accommodations required. “It is different because when [the child] falls sick, I lack knowledge on what to do.” (FG2). As a result, caregivers relied heavily on the guidance from HCWs to learn the best practices of care, as it could not be obtained elsewhere. One respondent shared: “Most of the time, I call a health care provider because I cannot ask outsiders because they just scare you” (FG 1). As such, gaps in knowledge signified inadequate caregiver education on postoperative care. The teaching responsibility rested on the HCW, especially on specialized topics such as ostomy care: “…[W]hat is missing is a proper explanation being provided to people because I was also not told anything. They do the operation and return your child… It was challenging” (FG1). Failure to transmit this knowledge left the caregivers unprepared after surgery and hospital discharge.

However, HCWs also exhibited ignorance in diagnosing and treating DTCAs, which prevented effective knowledge transfer to the caregiver. One respondent noted that their doctor not only failed to recognize the child’s anorectal malformation but also jeered and belittled the caregiver’s concerns about the congenital anomaly.

“[The doctor said] “Can’t you see that that child is normal?” But on observation, you would see that her vagina and anus had a very small space in between, and there needed to be a distance between the two. I would tell the doctor, and she would say, “The child is fine. You are behaving like you have never had a child and don’t know what a normal child looks like.” She discouraged me.” (FG4).

Outpatient HCWs were equally reluctant to help caregivers and their affected children out of fear and unfamiliarity with their DTCA diagnosis. One respondent shared: “…you go to the nearby clinic; they fear to treat him. They first ask how you gave birth to him. ‘What is his health condition?’” (FG2). Respondents reported that HCWs at satellite facilities often defer patient care to the national referral hospital, which is usually difficult to access. This delay added to the care incompetence felt by caregivers and their children.

In contrast, HCWs who provided meaningful support, kindness, and communication bolstered caregiver preparedness. HCWs who kept caregivers engaged improved the child’s condition and clinical course. One respondent shared: “You would see that [HCW] were keen to help and ensured that they called and informed you of the status of things and asked you to come… You feel like someone cares for you… The doctors were kind. They were caring” (FG4).

Empathetic communication and open knowledge transfer from HCWs to caregivers facilitated understanding of the child’s condition from a caregiver’s standpoint and enhanced caregiver confidence. Caregivers found these HCWs’ teaching treatment strategies effective in instilling care principles. As such, HCWs served as an essential knowledge base for caregivers to address their concerns.

Respondents therefore felt HCWS needed to communicate caregiving expectations and anticipate care needs upfront during the hospitalization, perioperative period, and beyond. Key information to convey in advance was the medical supplies caregivers needed to purchase, their respective costs, the approximate length of hospitalization, and potential care delays. This was especially important given that local hospitals had chronic supply shortages. Several participants also suggested a caregiver triage hotline, operated by an on-call healthcare provider who could advise on acute medical issues at home. Additionally, this hotline could help streamline and triage who should be brought back to the hospital to limit readmissions. One respondent suggested:

“Someone I can go to crying about my problems, and they listen quickly. Even if it is a problem, someone who can pick up my phone call quickly. Some time back, I once called, I asked the health worker for their phone number so that I could call if things failed, but she said no. “You just have to come [to the hospital].” (FG3).

With consistent communication, HCWs could alleviate caregivers’ worries, ease care burdens, and avoid unnecessary and costly trips to the hospital.

Theme 5: A CHEW program with home visits could support caregivers of a child with a DTCA
Respondents wholeheartedly welcomed a community outreach program for continuity of care, favoring CHEWs to come to their homes to guide their care practices and assess their living conditions for caregiving preparedness. Respondents were open to learn from CHEWs on how to feed, clothe, and clean their children with limited resources and space. The knowledge gained from CHEWs would empower caregivers to independently handle their child’s condition and its complications and remedy the helplessness that is otherwise common.

The CHEW would need to develop rapport with families to establish comfort and trust with the caregivers to be effective. In return, the caregivers would be more engaged and adherent to the doctor’s and CHEW’s recommendations, especially when summoned to the hospital. “Visiting the patients also encourages us, the caretakers. We feel like the health worker cares. Even when you encounter a problem, you think that by the time the health worker has spared time to come here, why don’t I rush to the hospital now that the health worker has called me?” (FG1). In this way, a CHEW program would likely boost the morale of the caregivers, providing the emotional and social support they lacked.

The effects of allyship from CHEWs could extend into the community, as others mention the possibility of raising awareness of DTCAs among their neighbors to diffuse the surrounding stigma. One participant noted, “Even in the communities we live in, they may start holding us in high regard because the health worker has come all the way to visit” (FG1). The CHEW could also prompt the husband to participate in this allyship within the family circle, given that most primary caregivers were mothers. One respondent said:

“Doctor, my request is that the biggest number of people here are women, so please come and talk to our husbands so that they can also get to know you. Sometimes, they don’t take things seriously, and they may not understand what we explain to them. If you come home, you can also explain to him, and he understands what is going on” (FG2).

In this way, the CHEW could educate the household and raise the family’s and community’s awareness of the child’s condition and caretaking situation, leading to more positive involvement in the child’s care.

This study assessed the caregiver experience of children living with surgically correctable or corrected DTCAs in Uganda. Through our thematic analysis, we identified themes centered around dimensions of the caregiver burden and proposed ideas to alleviate this burden. Limited resources and delays in the healthcare system hindered children’s quality of life, added to caregiver burden, and exacerbated socioeconomic challenges. Notably, the community played a central role in the caregiver’s ability to handle the child’s condition, such that stigma stemming from poor awareness of these conditions left caregivers and children isolated, even from their closest relatives. In this setting, HCWs were critical in transferring knowledge to ensure the caregiver’s confidence and competence in managing the child’s specialized healthcare needs. Participants emphasized the importance of a long-term relationship with the surgical team, open communication via a hotline with a surgical HCW, and the potential benefits of a community outreach program that involved home visits. Figure 1 outlines this complicated network of factors contributing to positive and negative caregiver experiences.

Health System Issues Contributing to Caregiver Burden

In Uganda, diagnosis of DTCAs in neonates is often delayed until a week after birth, putting the child at risk for malnutrition and contributing to increased mortality rates and other long-term repercussions for the child and caregiver [15]. In our study, delays in care persisted after hospital discharge. Children with DTCAs must navigate through referral systems for their ongoing post-operative issues, and peripheral health units are often ill-equipped to manage their specialized diseases. Figure 1 outlines the connection between difficulties with accessing care or medical supplies and delays in care and overall delays in childhood development. These delays were observed in studies conducted in similar settings in Congo, South Africa, and Ethiopia and led to preventable morbidity, developmental delays, and social isolation.[16-19]

Caretakers consistently also expressed feeling overwhelmed by caring for their child with a DTCA and how it depleted time and finances. The stigma associated with caring for a child with a DTCA stonewalled employment opportunities, and a lack of understanding from the community made it challenging for caregivers to secure childcare options while remaining financially stable. This economic strife was also noted in similar studies in Rwanda and other African countries [6, 20]. While Uganda’s health financing policy is working toward aligning with WHO guidelines to achieve Universal Health Coverage (UHC), current health financing options have insufficient financial risk protection.[21] Additionally, Uganda lacks an insurance system to support individuals facing high healthcare costs, such that a third of pediatric surgical patients face catastrophic healthcare expenditures.[22] Implementing a National Health Insurance scheme could help alleviate these financial burdens at the system level.

Caregivers reported positive experiences when they feel supported by their belief systems, communities, families, spouses, and healthcare workers. Figure 1 outlines the contributions of faith and community support on caregiver experiences and draws a comparison to cases where a caregiver does not have such support. Our respondents often turned to their faith in God as a coping mechanism, finding comfort and hope in unlikely recoveries and survival. Their faith also provided peace of mind to cope with difficult times and societal stigma. While some questioned their faith and saw their children’s conditions as afflictions, they still maintained gratitude and trust in God’s plans. Overall, religious faith and social support from various sources helped caretakers to find strength, joy, and provision for their children’s well-being.

Uganda has a limited number of hospitals capable of managing DTCAs. Consequently, caretakers frequently cited difficulties accessing healthcare professionals skilled in managing such anomalies as a barrier to care. However, fully addressing these barriers requires HCW education and expansion of the HCW workforce. Specifically, educating birth delivery teams and pediatric surgical HCWs to recognize DTCAs and their post-treatment complications could facilitate prompt management and follow-up.

The sample of caregivers in this study (97%) is from an urban setting. Caregivers residing in rural settings are underrepresented in this study, who face the caregiving burdens to a greater extent, given a lack of resource availability and far distances to hospital facilities to obtain care. Because of this, it is likely that our study underestimates caregiver burden in these communities. Future studies should incorporate community-based recruitment strategies to include caregivers in rural settings or who are outside of tertiary care systems to provide a more in-depth understanding of caregiver experiences in various community settings in Uganda.

Future Directions: A CHEW Program

WHO guidelines for CHEW support programs recommend adopting a whole-system approach, which considers health system capacities and population needs to integrate CHEW programs appropriately into the local health system to deliver essential health services and education to communities.[23] In this context, CHEWs are critical contributors to community-based healthcare as an extension of hospital networks.[24] Studies demonstrate that CHEWs positively affect community and individual health because they serve as bridges between communities and formal healthcare services, systems, and resources.[25]

The goals of our proposed CHEW Program for the following children with DTCAs are outlined in Table 2, which describes the responsibilities of a CHEW worker and the actions they would carry out to fulfill those responsibilities. One responsibility of the CHEW outlined is establishing a registry of children living with surgically correctable or corrected DTCAs. This would support long-term health outcome monitoring and streamline the scheduling of surgeries and staged procedures. CHEWs could also staff a caregiver hotline, liaising between healthcare and surgical teams and families to triage caregiver concerns about best care practices for their child. CHEWs could help caregivers navigate confusing healthcare systems and obtain appropriate, timely care. CHEWs could also provide counseling and psychosocial support to families and engage in community education to raise awareness of these congenital anomalies and reduce stigma. Regular contact and communication with an HCW would help families access resources like financial assistance, transportation to and from healthcare appointments, housing, and peer support groups. Advocating for families in this way is another responsibility outlined in Table 2 that our team envisions for the CHEW program.

 For such programs to be effective, understanding the sustainability of their components is key.[26] Program management, personnel training, supervision, and funding must be secured for effective implementation. Flexible integration of healthcare services for caregivers and their children with DTCAs can increase the program’s acceptability to the communities served.[27, 28]

Strengths and Limitations

This was the first study to investigate the caregiver burden of children living with DTCAs in Uganda. The broad inclusion of multiple diagnoses and patients and the extended follow-up are significant strengths of this study. Limitations of this study include the second-hand source of the caregivers’ accounts, who represent their child’s experience living with a congenital anomaly. Some of the caregivers had to care for their children during the focus group, which could have limited the openness and attention a caregiver could give to the discussion at hand, since they also had to care for their child. However, this underscores the burdens caregivers face in caring for their children. We also needed to translate the focus group discussions conducted initially in Luganda to English transcripts for analysis. This meant that some of the nuances from the local language might not be captured, even though a certified translator was used. Future studies should back-translate FGD transcripts from English back into Luganda to ensure that cultural nuances are not lost in the translation process. Participants were recruited via hospital databases, meaning that caregivers who did not have the opportunity to obtain care at a tertiary care center were missed in recruitment, which is another limitation of this study. Furthermore, we cannot mitigate the risk of recall bias in the responses.

Improving the quality of life and long-term care outcomes for children with surgically correctable congenital anomalies in Uganda starts with understanding the experience of caregivers. The burden imposed on caregivers due to financial strain, social stigma, and delays in care is substantial and demands urgent attention. However, amidst these challenges, our study highlights factors that can make a positive difference, including the caregiver’s religious faith, community, spousal support, and positive interactions with healthcare workers. A CHEW program, consistently endorsed by caregivers, emerged as a potential solution in our study. By providing support through home visits, education, counseling, and home treatment, this program could bring about positive change for families of children with DTCAs. Implementing policy measures to improve and expand the health infrastructure, strengthen the pediatric surgical workforce, and raise public awareness through community campaigns about congenital anomalies would further support these efforts.

We would like to thank Dr. Susan Naiga for her excellent transcription and translation services, We would like to thank Dr. Marie Armstrong-Huff and Dr. Sophia Zamudio-Haas for their advisory role in quality control of our study methods. We would like to thank all the parents and caretakers who participated in this study.