Family caregivers of children with cystic fibrosis: supportive care needs – scoping review

Abstract

Background

Cystic fibrosis (CF) in children requires complex and time-consuming daily care, presenting significant challenges for families and caregivers. Family caregivers caring for children with CF report diverse and complex needs.

Objective

This review aimed to identify the supportive care needs of family caregivers of children with CF.

Review methods

This scoping review was conducted to identify the supportive care needs of family caregivers for children with CF using the methodology proposed by Arksey and O’Malley. The Preferred Reporting Items for Systematic review and Meta-Analysis extension for Scoping Reviews checklist was used as a guide. The inclusion criteria included full-text quantitative and qualitative English articles from January 2000 to Apr 2024.

Data sources

Structured searches were conducted using Magiran, MEDLINE (PubMed), Scopus, ScienceDirect, SID (Science Information Database), Web of Science and Google Scholar search engines.

Findings

A search across six databases, including Magiran (96), PubMed (680), Scopus (828), ScienceDirect (972), SID (Science Information Database) (47) and Web of Science (409), identified 3032 records. An additional 1185 related articles were found through a manual search of the reference lists and the Google Scholar search engine bringing the total to 4217. Out of 4217 initial articles, 21 eligible articles were reviewed. The findings from this study indicated that family caregivers of children with CF face multidimensional needs requiring comprehensive attention and support including educational/informational, psychological/emotional, spiritual, social, family-related, health and child development and growth needs.

Conclusion

Addressing the needs of family caregivers of children with CF requires a multidimensional approach including multidisciplinary team support across various domains.

WHAT IS ALREADY KNOWN ON THIS TOPIC

• The focus has been on patients and what caregivers can do to support their ill children rather than also supporting themselves.

• The lack of a clear framework for identifying the supportive care needs of family caregivers of children with cystic fibrosis (CF).

WHAT THIS STUDY ADDS

• Proposing a framework that focuses on the supportive care needs of family caregivers of children with CF.

HOW THIS STUDY MIGHT AFFECT RESEARCH, PRACTICE OR POLICY

• Designing appropriate programmes and policies that can meet the needs of caregivers of children with CF.

Introduction

The supportive care needs of family caregivers of children with cystic fibrosis (CF) may start during pregnancy with screening tests and the diagnosis of the child’s disease and intensify with the birth of the affected child. The shock of the newborn’s diagnosis may introduce a new crisis for the family members, an experience previously unknown.¹ In many developing countries, the absence of adequate prenatal screening facilities to test for genetic mutations often results in children being born with genetic diseases. The inability to manage and treat this disease after birth due to the unavailability of pancreatic enzyme supplements and inhaled antibiotics creates further challenges for families.^{2 3}

On receiving a CF diagnosis for their child, parents face an unforeseen future.^{1 4} Some parents may experience grief and mourning similar to the loss of a family member. These parents might grieve the loss of their ‘normal’ child or experience a vague sense of loss.⁵ Family caregivers of children with CF who are in this situation and who are committed to providing lifelong care face numerous supportive care needs.⁶ The child’s illness can also change the family’s lifestyle and parents of such children often feel responsible for their child’s condition, leading to feelings of anxiety, guilt, helplessness and incapacity.⁷ When parents are in this situation, they often face a lack of knowledge about the disease, the complexity of treatment, side effects and its management associated with high levels of emotional distress, reduced quality of life and poor psychosocial adaptation.^{1 8} Families experiencing these conditions may require changes in work patterns, income, household responsibilities, specialised health literacy, caregiving skills and resources beyond what is typically required by ordinary parents.⁶ Due to their dependence on families, children also need more care to manage their disease and families especially parents play a significant role in the treatment and care process. Therefore, the more parents participate in care programmes for children with CF, the more influential the disease control and the more significant the reduction in its severity and complications.⁹

Due to the involvement of different systems by CF disease, its different genetic nature from other chronic diseases and the involvement of affected children from birth, the parents of these children have different support care needs in their care compared with other chronic diseases. Unfortunately, due to the chronic nature of this disease and its scattered prevalence compared with common diseases, the parents of these children receive less attention from healthcare teams.^{10 11} Supportive care has been defined as a benefit to patients and their families by encouraging individuals to live as well as possible.¹² Almost all research has focused on identifying supportive care needs from the patient’s perspective, often neglecting the burden it places on parents and caregivers.^{13 14} Studies previously conducted in this field have generally focused on rare diseases¹⁵ or, specifically, chronic diseases other than CF in children.16,18

Given that the identification of the supportive care needs of family caregivers of children with CF has yet to be systematically attempted, initiatives to provide appropriate support have, until recently, been ad hoc.¹⁹ Since each disease has specific supportive needs that are constantly changing, the responsibility for caring for children falls on their family caregivers due to their age. Consequently, in line with the family-centred care model’s objective of promoting the overall health and well-being of patients and their families, identifying, comprehending and planning for the supportive needs of family caregivers is one of the health team’s top priorities for interventions. Achieving this goal requires a correct understanding of the supportive needs of family caregivers. Hence, we decided to conduct a study to identify the supportive needs of family caregivers of children with CF.

Study objective

This scoping review aimed to collect and evaluate all published texts regarding the supportive care needs of family caregivers for children with CF.

Study method

The Arksey and O’Malley methodological steps were used to perform a comprehensive and systematic literature search to identify the full range of supportive care needs of family caregivers of children with CF.²⁰ The five stages proposed in this framework are (1) identifying the research question, (2) identifying relevant studies, (3) selecting studies, (4) charting the data and (5) collating, summarising and reporting the results. The PAGER framework (Patterns, Advances, Gaps, Evidence for Practice and Research Recommendations) was used to improve reporting quality²¹ and the study was reported based on the Preferred Reporting Items for Systematic review and Meta-Analysis extension for Scoping Reviews (PRISMA-ScR) checklist.

A scoping review is a suitable method for identifying research gaps, providing recommendations for further research, determining the scope of existing evidence and ultimately packaging and conveying research findings.²² The Population, Concept, Context (PCC) criteria were established at the beginning of this study to ensure that only relevant articles were included and that irrelevant articles were eliminated (table 1).

Table 1. PCC framework.

PCC	Components of the research question in the scoping review
Population	Family, father, mother or primary caregiver of a child with CF.
Concept	Identifying the supportive care needs of family caregivers of children with CF.
Context	Any setting for support, whether clinical, clinic or in any geographical location.

CF, cystic fibrosis.

Step one: define the research question

The PCC framework which guides database searches and eligibility criteria addresses the following research question:

What are the supportive care needs of family caregivers for children with CF?

Step two: identifying relevant studies

To comprehensively search relevant texts, published articles from six electronic databases including Magiran, MEDLINE (PubMed), Scopus, ScienceDirect, SID (Science Information Database), Web of Science and Google Scholar search engines were searched between January 2000 and April 2024.

Key search phrases and word combinations include:

Family, parent, mother, father, caregiver, support, supportive, supportive care need, advocacy, cystic fibrosis, CF, child, children, pediatric, adolescence, adolescent.

We combined terms using Boolean operators such as AND, OR and NOT. Table 2 describes the search strategy used in PubMed which was adapted for the other databases. Additionally, manual searches of the reference lists of selected articles were performed and Google Scholar was used to access other primary sources and full-text articles. After completing the search, all identified relevant studies were entered into EndNote software.

Table 2. Search strategy used for the PubMed database.

Database	Keywords for database search
PubMed	Search terms Family, parent, mother, father, caregiver, support, supportive, ‘supportive care need’, advocacy, ‘Cystic fibrosis’, CF, Child, children, pediatric, paediatric, adolescence, adolescent
	Keywords combination (‘family’(Text Word) OR ‘parent’(Text Word) OR ‘father’(Text Word) OR ‘mother’(Text Word) OR ‘caregiver’(Text Word) OR (‘family’(MeSH Terms) OR ‘parents’(MeSH Terms) OR ‘mothers’(MeSH Terms) OR ‘fathers’(MeSH Terms) OR ‘caregivers’(MeSH Terms))) AND (‘support’(Text Word) OR ‘supportive’(Text Word) OR ‘supportive care’(Text Word) OR ‘supportive care need’(Text Word) OR ‘advocacy’(Text Word)) AND (‘cystic fibrosis’(Text Word) OR ‘CF’(Text Word) OR ‘cystic fibrosis’(MeSH Terms)) AND (‘child’(Text Word) OR ‘children’(Text Word) OR pediatric(Text Word) OR ‘paediatric’(Text Word) OR ‘adolescence’(Text Word) OR ‘adolescent’(Text Word) OR (‘adolescent’(MeSH Terms) OR ‘child’(MeSH Terms) OR ‘pediatrics’(MeSH Terms)))

Step three: selecting articles

The study’s inclusion criteria included quantitative and qualitative research, mixed-methods studies and full-text articles in English without restrictions on study type or methodology. Studies meeting the inclusion criteria involved families with children aged 18 and under with CF. The exclusion criteria included studies reporting supportive care needs of home caregivers for other types of chronic diseases, conference presentations, letters to the editor and studies published only as abstracts.

Reporting was based on PRISMA guidelines. Two independent researchers screened the titles and abstracts of the retrieved studies for eligibility. Studies that did not meet the inclusion criteria were fully retrieved and reviewed. Two researchers thoroughly reviewed all the full texts to decide which ones to include. In the case of any disagreements, a consensus was reached. The methodological quality of all the included studies was independently assessed by two researchers using the QualSyst tool, specifically designed to evaluate the quality of both quantitative and qualitative studies.²³ The quality scale of this tool consists of 10 items with a total of 20 statements. Each statement was scored based on adherence to the criteria (yes: 2, partial: 1, no: 0). A summary score was recorded for each study. Summary scores for quantitative studies were similarly calculated based on 14 items with a maximum possible total score of 28. Any scoring discrepancies between researchers were resolved through discussion to reach a consensus. A third researcher was used in case of disagreement between two assessors. No study was excluded based on methodological quality. Ultimately, 21 articles meeting the criteria were included in this review and approved for final consideration.

Step four: charting data

Step four involved creating a framework for charting the data. We used a modified version of the data charting form based on recommendations by Arksey and O’Malley. Based on this form, each primary article reviewed was summarised based on the author’s name, year, country of origin, type of source evidence, study objective, study design, sample characteristics, data collection and analysis methods, sample size and limitations. Two reviewers independently conducted the data charting and the results were subsequently discussed. The data charting was continuously updated in an iterative process. The extracted data were then analysed and interpreted.

Step five: summarising and reporting findings

This fifth and final review step involved collecting, summarising and reporting support care needs in texts. We used the PAGER framework to describe and critique the literature comprehensively (table 3). We reported the results in five aspects: (1) patterns, (2) advances, (3) gaps, (4) evidence for practice and (5) research recommendations.²¹ Key themes were structured as patterns according to the research question and background evidence for practice and research recommendations were also provided based on information related to patterns, advances and gaps.

Table 3. PAGER framework.

Research recommendations	Evidence for practice	Gaps	Advances	Pattern
Given the dynamic nature of supportive care needs which change over time and vary among individuals, the proposed supportive care needs must be updated and localised according to each country’s culture.	This supportive care needs to guide the development of a support programme for family caregivers of children with CF and provide a framework for their care.	A comprehensive study is necessary to determine the supportive care needs and their extent in family caregivers of children with CF.	There is no evidence of an international/global agreement on the supportive care needs and their scope for family caregivers of children with CF.	Supportive care is essential for family caregivers of children with CF.

CF, cystic fibrosis.

Selected study features

A search across seven databases, including Magiran (96), PubMed (680), Scopus (828), ScienceDirect (972), SID (Science Information Database) (47) and Web of Science (409), identified 3032 records. An additional 1185 related articles were found through a manual search of the reference lists and the Google Scholar search engine bringing the total to 4217. After the identified articles were imported into EndNote software, 708 duplicate records were removed and 3509 articles were screened, 3307 of which were excluded based on the exclusion criteria. Ultimately, 202 studies were retrieved for full-text review. Full-text screening for relevance and exclusion of articles that did not cover supportive care needs were related to other chronic diseases or were not relevant to family caregivers of children with CF reduced the number to 21 articles which were then entirely and textually evaluated (see figure 1). Two authors reviewed and approved the chosen studies. After reviewing and extracting the necessary information, the results were summarised in a table and manually analysed. These articles were published between 2002 and 2024 in countries including the USA (n=6), the UK (n=5), Australia (n=2), Ireland (n=2) and one study each in the Netherlands, Jordan, Germany, Ireland, Italy and Turkey. 14 studies used qualitative methods and 7 studies employed quantitative methods (online supplemental table 1).

Figure 1. Preferred Reporting Items for Systematic review and Meta-Analysis flow diagram. CF, cystic fibrosis.

Supportive care needs

After extracting and carefully reviewing the necessary information, the results were summarised in a table and analysed manually. The results of supportive care needs of family caregivers for children with CF were organised under seven main categories (educational/informational, psychological/emotional, spiritual, social, family-related, health and child development and growth needs) and 25 subcategories (table 4).

Table 4. Summary of supportive care needs.

Categories	Subcategories (references)
Informational/education needs	Medical information needs.8 24 25
	Nutritional information needs.26
	Care information needs.827,29
	Accuracy of information.8 30
	Perception of information.8
	Lack of organisation of information.8 24 30
	Lack of information from others.8 27 31
Psychosocial/emotional needs	Psychosocial/palliative care needs.2527 28 30 32,38
	Psychological and emotional problems.48 24,33 35 38
Spiritual needs	Contact with God.27 37
	Searching for meaning.37
Social needs	Interpersonal need.36 38
	Isolation.25 27 29 31 34 38
	School-related needs8 26 29 31
	Community support needs.27 28 30 31 35 38 39 41
Family-related needs	Intimacy needs.4 27 32 38
	Family function.427 28 30,32 34 38 40
	Work-related needs.4 27 31 35 41
	Economic needs.27 31 32 34 39
Health needs	Access to health facilities.827 34 38,40 42
	Home care skills.48 26,29 31 32 35 41 42
	Physical needs.4 27 39
	Health system/patient–clinician participating needs.8 24 27 36 42
Child growth and developmental needs	Child rearing.26 28 31 33 42
	Independence of the child.40 41

Category 1: informational/educational needs

In nine studies,824,31 the informational and educational needs of family caregivers of children with CF were identified. The needs were classified into seven subcategories: Medical information, nutritional information, care information, accuracy of information, perception of information, lack of information organisation and lack of information from others about CF. The most common information/educational needs of family caregivers of children with CF included medical information related to side effects, outcomes and risks associated with medications, distrust toward medications, concerns about antibiotic resistance, timing of medication doses,^{8 24 25} disease management knowledge and routine care prioritisation,27,30 unawareness of surroundings and even medical staff about CF^{8 27 31} and other essential needs including receiving information from a reliable and trustworthy source^{8 30} and knowledge about nutritional care.²⁶

Category 2: psychological/emotional needs

Psychological/emotional needs were mentioned in 18 studies48 24,41 including two subtopics: Psychological/palliative care needs and psychological/emotional problems of family caregivers of children with CF. Family caregivers of children with CF suffer from unpredictability,²⁸ uncertainty,²⁵ the unexpected nature of the disease, the potential for its exacerbation²⁵ and the lack of immediate tangible evidence of care benefits³² leading to emotional exhaustion,³⁸ constant worry,^{25 33} psychological devastation,²⁷ the feeling of being overwhelmed,^{28 30 34 35} inability,^{27 37} feelings of sorrow and distress all the time²⁷ and being trapped in a circle of suffering,²⁷ highlighting the importance and prominence of the need for extensive and continuous psychological palliative care. Family caregivers of children with CF often experience emotional and psychological issues such as anxiety,^{4 24 27 29 39 40} stress,^{4 8 26 28 30 31} fear,24,2631 32 feelings of depression426 35 38,40 and guilt.2730,32 These caregivers frequently battled intense emotions including shock,^{27 30} pressure,³² crying,²⁷ restlessness,⁴ pessimism,^{27 38} doubt,³² disappointment,^{27 41} sadness,^{27 39} distress,^{27 30} denial,²⁷ hopelessness, dilemmas,²⁹ inadequate feelings,³⁸ feelings of worthlessness³⁸ and frightened tension or wounding.⁴ These family caregivers often endure many mental and emotional burdens^{28 30 32} and must cope with being carriers.³¹ Family caregivers are usually concerned about their child’s future.^{4 33 38}

Category 3: spiritual needs

Two studies^{27 37} mentioned the need for spirituality: Contact with God and searching for meaning among family caregivers of children with CF. Family caregivers of children with CF sought spiritual support through praying, pleading and communicating with God to cope with the disease and achieve peace, expressing the need for spirituality.^{27 37} In some cases, family caregivers felt that their connection with God was disrupted believing that they were being punished by divine retribution and needed spiritual cleansing and reassessment of spirituality and religion as essential.

Category 4: social needs

Social needs were mentioned in 14 studies,825,31 34 including four subtopics: Interpersonal needs, isolation, school-related needs and community support needs among family caregivers of children with CF. Family caregivers of children with CF expressed their interpersonal needs as reduced time spent with others,³⁸ conflict within themselves and with others³⁸ and difficulty coexisting with their surroundings.³⁶ These families reported pressure from their surroundings and blame for their child’s disease,²⁷ feeling stigmatised and different from others^{27 29} and suffering from isolation.^{25 27 31 34 38} Due to the disease, family caregivers faced issues with their child’s education in school including uncertainty about school-based healthcare support,²⁹ concerns about adherence to treatment in school,⁸ adapting all treatments and educational obligations during the school day,²⁹ difficulty in managing the transition to school,²⁶ concerns about managing calorie intake throughout the school day,²⁶ ensuring academic welfare for the child³¹ and reduced available study time due to treatments.²⁹ Additionally, family caregivers experienced community issues such as a lack of social support,^{27 28 30 31 38 39 41} an absence of affordable insurance and assistance services³⁹ and a lack of informal assistance from relatives or the community.³⁵

Category 5: family-related needs

Family-related needs were mentioned in 10 studies,427 28 30,32 34 35 38 including four subtopics: Intimacy-related needs, family function, work-related needs and economic needs of family caregivers of children with CF. Caring for a child with CF led to conflicts in various parental roles within the family. One of these areas was intimacy-related needs. Parents of these children reported tension and conflict in marital relationships and dissatisfaction with their sexual relationships.^{4 27 32 38} Another family area affected by the disease was family functioning. Due to constant care for their sick child, families face numerous issues including a lack of opportunity for parenting and bonding with their child,³² challenges related to family commitments and responsibilities,^{30 34 38} family disunity,³¹ being not as good as other people,⁴ unequal attention distribution among siblings and other family members,^{27 31} lack of privacy,²⁷ having a busy family life,²⁸ less satisfaction with leisure activities,⁴ disruption in family plans,²⁸ the inevitable struggle for the child’s life and comfort,⁴⁰ less satisfaction with leisure activities and general life,⁴ affecting the dynamics and welfare of the entire family^{27 34} and leading to a lower quality of life for these families.^{27 40} One of the challenges these parents faced was work-related needs and economic concerns.²⁷ Due to constant daily caregiving, these parents faced work-related issues such as the inability to return to work, limited work hours, changed jobs or shifts, quitting and decreased job satisfaction.^{4 27 31 35 41} Additionally, due to high treatment and caregiving costs, these parents were concerned about earning income and reported financial pressure and economic challenges.^{27 31 32 34 39}

Category 6: health needs

Health needs were mentioned in 17 studies48 24 26,29 31 32 34 including four subtopics: Access to health facilities, home care skills and physical and health system/patient–clinician participation needs among family caregivers of children with CF. The results indicated that these family caregivers faced challenges accessing healthcare and treatment facilities including difficulties,³⁹ seeking resources,³⁴ seeking support from healthcare professionals,⁴⁰ practical problems with the supply of products or equipment,⁸ lack of time in busy clinics,⁸ frequent hospitalisations^{27 38} and long-term hospital stays.²⁷ These family caregivers also sought access to weekly home visits outside clinic appointments and clinic services to reduce the issues mentioned.⁴² Family caregivers of children face challenges in providing home care including the complexity of treatments,^{27 29} the relentless and repetitive cycle of the disease,³² constant adherence to respiratory therapy and chest physiotherapy,⁴² preparing separate meals,²⁶ the burden of responsibility and care,^{28 31 35 41} practical problems in disease treatment,⁸ the constant need for medication⁸ and difficulties in accommodating the regimen in everyday activities.⁸ Additionally, these parents reported physical issues such as headaches, pain, fatigue, lack of energy and lower satisfaction with their physical condition.^{4 27 39} Family caregivers of children with CF faced difficulties with the health system-patient‒physician relationship when visiting healthcare facilities including improved communications, limited opportunity to express any concerns or problems, non-cooperative hospital staff, inconsistencies in approaches, recommendations, practices and philosophies of different centres regarding their involvement in care, lack of a holistic approach, failure of staff to follow specialist recommendations and general practitioners’ uncertainty about medication treatments and diagnoses.^{8 24 27 36 42}

Category 7: child development and growth needs

Five studies^{26 28 31 33 42} mentioned child development and growth needs. Family caregivers of children with CF experience various needs related to their child’s development and growth. These family caregivers faced issues in raising their child including coping with social factors, general behavioural non-conformity, irregular eating habits, struggles with children, forced feeding, differing priorities between parents and children, challenges in transferring treatment responsibility from parents to children, considering the growing independence of the child and efforts to ensure social welfare for the child.^{26 28 31 33 42}

Discussion

This scoping review aimed to identify the supportive care needs of family caregivers for children with CF. Our findings indicate that these family caregivers face various needs across multiple dimensions, requiring comprehensive attention and support. These needs include educational–informational, psychological–emotional, spiritual, social, familial, health and child development and growth.

The results of our study align with Gill et al’s research which identifies the support needs of parents and caregivers of children with life-limiting diseases including informational and healthcare needs (such as emotional support and practical services).⁴³ Additionally, our study’s findings are in line with the research by Thomas et al which outlines the support needs of caregivers of children with cancer, asthma and congenital heart disease. These needs include communication, choice, information, education, practical, social, psychological, emotional, physical, home and sibling support.¹⁹ Previous studies^{19 43} in this field have mainly focused on chronic diseases other than CF in children. Therefore, the identified needs do not cover all the needs of caregivers of children with CF. The needs mentioned in these studies are part of the identified needs in our research. Additionally, these studies have encountered problems in classifying items.

One of the most essential conditions identified among the selected articles was educational–informational needs. Accurate and up-to-date information about CF care knowledge in all its aspects is crucial for family caregivers.³⁰ This educational–informational need is fundamental for informed decision-making and effective disease management.²⁹ Therefore, developing educational programmes tailored to these family caregivers’ needs is essential.

In addition to educational needs, significant psychological–emotional needs were observed. The chronic nature of this disease leads to emotional exhaustion,³⁸ constant psychological devastation,²⁷ feeling overwhelmed,^{28 30 34 35} inability to deal with the disease^{27 37} and being trapped in a circle of suffering²⁷ among family caregivers of children with CF as documented in various studies showing their lower quality of life^{27 40} highlighting the importance and prominence of the need for extensive and continuous palliative psychological care. The immense emotional and psychological burden of CF^{28 30 32} documented on family caregivers in this review indicates that this should be a priority for clinical practice and future research. Many family caregivers desire increased psychological care and access to support groups.²⁸ It appears that advances in supportive care have not kept pace with advancements in other treatment aspects. For instance, while anxiety and depression among family caregivers of children with CF have been thoroughly documented in recent years,48 24 26,31 39 40 specific therapeutic support for this group has not been considered; therefore, these findings emphasise the importance of access to counselling services and psychological support. Spiritual needs also constitute a significant part of family caregivers’ experiences, especially during challenging times.^{27 37} Spiritual support can be seen as a source of hope and resilience enhancement.

The study revealed that many caregivers expressed a desire to seek support from social networks or organisations by joining support groups and receiving increased psychosocial care. Patient advocacy organisations frequently play a critical role in providing supportive care services and are crucial in improving the quality of life for family caregivers of children. Creating spaces for sharing experiences and accessing support from one’s surroundings can reduce feelings of isolation and strengthen family and social relationships.⁴² These families, due to their child’s disease, frequently reported pressure from their surroundings, lack of societal understanding of CF and blame, adding to the distress of family caregivers of children with CF.^{25 27 31 34 38} The care burden of this disease affects the entire family’s dynamics and welfare leading to family disintegration and challenges in relationships between parents, other family members and effective family functioning contrary to the recommendations and goals of family-centred care.^{27 34 38} Given the chronic condition of the disease and the need for 24-hour care, these family caregivers have various needs for providing different facilities and equipment for home care services.^{8 42} Ensuring access to quality and specialised healthcare for family caregivers of affected children along with the participation of the health system patient‒doctor is one of the priorities of providing services. To improve services, specific areas of home care, the coordination and organisation of services, the interpersonal communication skills of professionals and the provision of support for the needs of each dimension should be considered. Families have reported issues of distrust toward physicians and medical staff. This is often due to ineffective communication, misinformation being provided to them and little opportunity for them to express concerns or problems. Furthermore, some families feel that there needs to be more cooperation from hospital medical staff.^{8 27} Therefore, improving care communication approaches and enhancing the skills of doctors and treatment staff to meet the needs of family caregivers of children with CF will be highly beneficial.

The care needs of those families will differ widely as will the remits of services and the capacity to provide for families. In connection with this are the geographical setting of these services, the areas where their families live and their different cultures. Supportive care provision varies internationally particularly regarding the level of specialty and advanced care delivery available to families. The findings discussed in this review may be limited to specific countries. Further exploration of supportive needs in particular locations to identify region-specific areas of improvement is necessary. According to the findings of Thomas et al,¹⁹ Gill et al and Pelentsov et al, the domains assessed in studies addressing support needs in families of children with chronic health conditions should likely be broader than those identified in the original patient-centred support care need framework.^{15 19 43} To date, no study has attempted to examine the entire constellation of supportive care needs of family caregivers of children with CF. Based on the findings of this scoping review, we suggest (see figure 2) that this is a preliminary suggestion for a supportive conceptual framework and there is a need for much more research in this area, mainly empirical justification for the proposed framework. We agree with Kerr et al¹⁶ who noted that if needs are not well defined, they cannot be satisfactorily met in providing care to their families.

Figure 2. Proposed caregivers supportive care needs framework.

Strengths and weaknesses

The strength of this review is the amalgamation of studies conducted across a wide range of settings to provide a comprehensive overview of the supportive care needs of family caregivers for children with CF. CF service providers can use these data to design care models that directly address the needs of caregivers. The limitations of this review were that we only focused on works published in English which may have limited the number and type of studies included in our review. This focus also means that the assessment of needs may not generalise to non-English speaking countries. Nevertheless, restricting the evidence syntheses to the English language is a viable method and has been found to have little impact on the conclusions of systematic reviews. Another limitation was our inability to access some full-text articles in the Embase and CINAHL databases. To address this, we consulted with an information specialist who recommended using the Scopus database due to overlap in clinical content. Additionally, they suggested using the Google Scholar search engine to find more related articles. We conducted a thorough and precise search of these databases to include all articles.

Conclusion

Ultimately, this study demonstrated that addressing the needs of family caregivers for children with CF requires a multidimensional approach including providing support across educational–informational, psychological–emotional, spiritual, social, familial, health and child development and growth domains. Attention to these needs and the design of appropriate programmes and policies can help improve the quality of life for these families. Future research should explore these needs more profoundly and evaluate the effectiveness of various solutions.

Data availability statement

All data relevant to the study are included in the article or uploaded as supplementary information.