Study participants and recruitment

The study design was exploratory sequential mixed methods. A convenience sample of caregivers of children diagnosed with SCD through the pilot program at John F. Kennedy Medical Center (JFKMC), the teaching and referral hospital in Monrovia, Liberia was recruited. Caregivers who either regularly attended the SCD Clinic or those who were considered lost to follow-up and had either attended once or never attended were also included in the study. A caregiver was invited to participate in the study through contact by telephone, text message, in person at clinic visits, or during a home visit. A caregiver was eligible to participate if he/she self-identified as the primary caregiver, were aged 18 years or above, and had sufficient English language comprehension to participate in the interview and complete the questionnaire. There were no specific exclusion criteria. One caregiver per patient was recruited. Caregivers received remuneration for their time and travel.

Data collection

The research question guiding this investigation was whether factors that drove decision-making for families with children with SCD could be modified to improve retention in care and ultimately, to improve patient outcomes. A semi-structured interview guide was designed by the authors to elicit knowledge, perceptions and behaviors of caregivers related to their child’s SCD diagnosis and care. The guide was designed to assess five domains (intrapersonal, interpersonal, community, external, and organizational) of decision-making as part of a socio-ecologic model outlined in the pediatric HIV literature [11]. The socio-ecologic model takes into consideration the complex interplay of factors between individuals, families, communities, and society. Interviewers were healthcare workers trained in qualitative interview techniques. Interviews were conducted in private, between November 2016—April 2017. All interviews were conducted in English. Interviews were digitally recorded and transcribed verbatim. Examples of interview questions were: “Please describe your experience receiving the diagnosis of SCD?” and “What are some of the barriers preventing you from attending the clinic?”.

Prior to the interview, demographic information about the participant and patient were solicited with a 56-item questionnaire, along with information about family dynamics. The questionnaire was read aloud to participants. Visits to the JFKMC were validated by medical record review. Dates and ages were verified by comparing to records obtained in the newborn screening program [5].

Ethics

All study staff completed human subjects research training prior to the initiation of the study. Due to low literacy levels and concerns about comprehension the consent was read aloud prior to all participants providing consent. Written informed consent was obtained from all participants. The research was conducted in accordance with ethical research practices and reviewed by the Institutional Review Boards at University of Liberia—PIRE and Baylor College of Medicine.

Data analysis

Prior to analysis of qualitative data, transcripts were cross-checked for accuracy by one author who was also an interviewer (VNT). Final transcripts were uploaded into Dedoose Version 8.035. An initial codebook containing codes and corresponding definitions was developed by adapting a socioecological framework developed for HIV research for SCD [11]. Within each of five domains are subdomains that more specifically characterize drivers of decision-making.

Using the initial codebook as a guide, transcripts were coded by two independent coders (KU, HH) trained in qualitative research methodology using a hybrid thematic analytic approach [12]. This approach includes both structured and emergent codes. Structured codes were derived from the adapted socioecological model [11], supported by emergent codes which were generated during coding to capture additional features of the data in relation to the research question. Once finalized, each coder then used the codebook to independently apply codes to the transcripts, meeting periodically to compare. A third author (VNT) served as a tie-breaker for any disagreements. After coding was complete, codes were reviewed to identify, define, refine, and name themes and subthemes.

For quantitative data analysis, the International Wealth Index (IWI) calculator was used to determine the average household wealth of the study participants based on questionnaire responses [13]. The IWI is scored from 0–100, with 0 representing households with none of the assets in question and lowest quality housing and 100 representing households having all assets and highest quality housing. These assets include seven consumer durables (possession of a TV, fridge, phone, bike, car, a cheap utensil and an expensive utensil), access to two public services (water and electricity) and three housing characteristics (number of sleeping rooms, quality of floor material and of toilet facility). In 2020, the average national IWI for Liberia was 32.6, with a range across counties of 18.8 in Gbarpolu to 45.7 in Monteserado. Data from questionnaires were entered into RedCap [14] and descriptive statistics summarized using Microsoft Excel or GraphPad Prism. Data integration occurred by using the quantitative results to confirm and expand qualitative findings.

“I Do Not Believe It”: The stages of grieving inform caregivers’ health-seeking behavior

The emotions experienced by many caregivers between diagnosis and the interview aligned with stages of grieving and influenced retention in care. Grief manifested through intrapersonal, community factors, and interpersonal factors (Fig 1). Although there was wide variation among the caregivers in the description of the manifestations of grief, common threads surfaced among the interviews representing the stages of grief, including denial, anger, bargaining, depression, and acceptance.

Most caregivers demonstrated disbelief and shock upon disclosure of the SCD diagnosis (Table 2). In the questionnaire, 16 of 26 participants reported knowing another person with SCD, including 6 with an affected family member and 2 with a death in the family attributable to SCD (Table 1). Many refused to believe the diagnosis and reported that SCD was unfamiliar to them. Caregivers reported that their newborn had appeared healthy and thus, the diagnosis was inconsistent with that appearance. They reported that their extended family members were healthy, and thus they perceived their family to not be at risk for SCD. Caregivers expressed anger at family members and spouses who did not disclose that they were carriers of sickle cell trait, those who did not know their sickle cell carrier status, and those that had knowledge that the disease existed in the family. Caregivers described feelings of helplessness, often voiced in conjunction with fear of having a chronically ill child. Underlying the experiences relayed in the interviews were feelings of guilt for many caregivers. Most caregivers evolved toward acceptance of the diagnosis, generally in association with highlighting their role the importance of seeking care and medications. Where family support was available, the caregiver was able to more quickly move toward acceptance.

Several community-oriented factors highlighted grieving as both internally driven and externally amplified or mollified. Interwoven into the theme of grief, caregivers expressed concerns for how disease disclosure occurred or was delivered by a provider, as well as factors such as misinformation and myths about SCD that are prevalent in the community. Strong cultural and social links to religion enabled consultation with pastors and faith leaders to be a mechanism for accepting the diagnosis of chronic disease. All participants identified a religious affiliation (Table 1). Caregivers shared how faith communities provided a source of hope and space for non-judgmental disclosure of disease.

“The Child Will Die Soon”: Misconceptions drive stigma towards children, parents and families

While most study participants did not use the word stigma, they recounted negative experiences with community, interpersonal and structural/organizational factors which impacted healthcare decisions (Table 2). External factors such as the caregiver’s gender contributed to stigma. Many of the interviewed caregivers assigned blame to the mother for the diagnosis, misunderstanding the autosomal recessive nature of the disease. This was true for caregivers who were and those who were not the mother of the child.

Caregivers internalized stigma, which heightened social isolation, anxiety and despair. While 84.6% of participants reported disclosure to family, only 46.2% reported disclosing the diagnosis to friends (Table 1). Caregivers alluded to the similarity between SCD and other diseases associated with stigma and discrimination, such as HIV. A prevalent and damaging example is the myth of death around 18 years of age. This myth in particular led to feelings of shame and made caregivers reticent to disclose the child’s diagnosis for fear of stigmatization of themselves, the child, and their families. Caregivers suggested that their lack of knowledge and understanding of the disease made it difficult to disclose and discuss the diagnosis with family and community.

Some caregivers described receiving misinformation not just from the community but also from within the health system, including from within JFKMC and from other facilities. Perpetuation of misinformation within the health system itself served as a source of stigmatization and a deterrent to accessing the health system. Despite caregivers acknowledging the importance of seeking care and the severity of the disease, they reported being told not to accept the diagnosis for their child. Due to the fear of stigma by outsiders, a large number of caregivers saw continuity of care with known health providers to be safer and reduced fears of being stigmatized by the health system. This behavior led to non-disclosure of child’s SCD status to providers at other health facilities, increasing the likelihood for mismanagement of the condition.

“I Don’t Have Money”: The burdens of caring for a child with SCD affects access to healthcare

Caregivers described complicated financial, psychological and emotional burdens of caring for a child with a chronic disease which altered decisions around retention in care. The participants included 26.9% students, 23.1% working at least part-time, and 50% not working. Despite a recommended quarterly visit schedule, 23.1% of participants never returned to the SCD clinic after the initial visit, 42.3% had fewer than 5 visits over 4 years, and 19.2% had more than 10 visits in the observation period. This differs from frequent interactions with the healthcare system for routine childhood immunizations (Table 1). The burdens of the diagnosis manifest through intrapersonal, community, and external factors.

The economic status of the family contributed to healthcare decisions. The median IWI for study participants was 35.5 (Fig 2A). Monetary costs associated with the need to frequently access healthcare, such as costs of medications or visits and cost of transportation determined whether or not they were able to make a clinic appointment. There was no association between IWI and the number of return visits (Fig 2B). Of 26 participants, 73.1% relied on public transportation (Table 1). The logistical costs of accessing care included distance from place of residence to that of the health facility, the time spent in transit, and time spent away from work. For transportation to the hospital, 57.7% reported spending greater than 60 minutes in transit to the hospital for an appointment (Table 1).

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Fig 2. International Wealth Index by quartile and return visit.

A: Box and whisker plots representing the distributions of IWI scores for study participants. Dashed lines represent the mean IWI score for all of Liberia, along with means for selected regional, continental, and international low- and middle-income countries. B: Scattered plot depicting IWI scores for participants against number of return visits. Mean with standard deviation shown. Dashed line represent the mean IWI score for all of Liberia.

https://doi.org/10.1371/journal.pgph.0001705.g002

Caregivers expressed a feeling of being overwhelmed by the diagnosis, that the disease was “too big” or too serious. Caregivers reported being told not to “pronounce” such a diagnosis of their child. Sometimes, this advice came from community members, and sometimes it appeared as a representation of denial. Caregivers feared the voicing the diagnosis would lead to true manifestation of the disease. The psychological burden of the diagnosis was made greater by the perception that they were giving the diagnosis, rather than the diagnosis already existing. This emotional burden led to some avoidant behaviors. Some caregivers endorsed not wanting to be seen going to the hospital.

“Through God’s Will, the Child Will Survive”: Religious beliefs and cultural support networks serve as positive and negative drivers

Support networks for caregivers emerged as a key factor in decision-making. Systems within communities, religious organizations, and health healthcare organizations influenced health seeking behavior positively, by sustaining caregivers emotionally and financially, and influenced negatively, by perpetuating myths about SCD (Table 2).

Family and community networks negatively impacted caregiver decision making by perpetuating prevalent community misconceptions about SCD. Fear of stigma and discrimination led to failure to seek care or failure to disclose the diagnosis at outside facilities. Denial of the child’s diagnosis led to delays in care. Additionally, misinformation related to the survival of children with SCD enhanced a perception of futility of medical intervention.

Caregiver decisions were positively influenced by support networks to manage the diagnosis of SCD. The interviews captured how community and family support helped to reduce feelings of shock, grief, disappointment, and despair (Table 2). Strong religious beliefs and faith communities provided a supportive environment that both individuals and families relied on as they dealt with the chronic disease diagnosis. Caregivers were encouraged to seek care for their child and to continue to care for their child despite the diagnosis. Despite expressing fears of disclosing the disease to community members, most caregivers acknowledged the importance of confiding in faith leaders.

Caregivers revealed how family members and faith communities were critical in providing financial support to access healthcare. This includes gifts of transportation and payment for hospital services and medications. The enormous costs of caring for a chronically ill child in an LMIC were apparent in all interviews. Obvious costs such as transport, hospital fees, and medications were addressed. However, the costs of care for other children, missed wages, and time were also frequently mentioned.

Caregivers also highlighted organizational support such as personal connection with the healthcare team influencing their decision-making processes and contributing to continuity of care. Even years after an encounter, caregivers could recount an experience with a specific healthcare provider who gave knowledge or encouragement for an intervention. Caregivers perceived clinic attendance to be beneficial for medical assessment and advice. Intertwined with this was the perception that medications provided by the health system was beneficial.

“I will get better treatment for him to live longer” The perceived benefits of treatment versus the role of medications

Perceived benefits of healthcare emerged as a theme related to healthcare decision-making at intrapersonal, external, and organizational levels. Most caregivers saw the benefit of accessing free or subsidized medications and access to a physician who understood SCD. Caregivers perceived that the clinic visits, medications, and treatment could lead to a longer life for their children as a benefit. Caregivers expressed the perception that the benefits of treatment were enhanced when accompanied with prayer and their faith. For many caregivers, self-efficacy manifest as providing medications to their child: it was the only thing some parents felt they could do for their child.

Relationships developed between healthcare providers and caregivers seemed to influence the understanding of SCD and perceived benefits of medications and treatment at the clinic. Study participants recalled beneficial interactions reflecting a patient-centered care approach by healthcare workers (Table 2). A personal relationship with the healthcare provider was viewed as beneficial in obtaining prioritized treatment and as having an understanding of the child’s clinical diagnosis. Some caregivers expressed mixed feelings and confidence in the health system that was entwined with their economic status. Caregivers with more financial resources sought treatment outside of Liberia or at private health facilities in the hope that they would benefit from more definitive treatment outcomes. Those with fewer financial resources had lower expectations and less value placed of treatment provided by the facility.