The psychological, psychosocial and emotional wellbeing of children and young people with leukaemia

The World Health Organization (WHO) (2021) has estimated that 400 000 children and adolescents develop cancer each year. Leukaemia is one of the most common forms of cancer within the paediatric population (WHO, 2021). It is estimated that there are at least 35 000 people in the UK who are alive having been diagnosed with a childhood cancer and survived more than 5 years (Children's Cancer and Leukaemia Group, 2022). Around 2500 children and young people (CYP) are diagnosed every year (Cancer Research UK, 2017; Children with Cancer UK, 2022a), with leukaemia likely accounting for the largest percentage of this population.

Years of medical research and trials have contributed to the survival rates for leukaemia in the UK now being between 70% and 92% (Children with Cancer UK, 2022b), successfully changing what was once an acute, life-threatening disease into a curable illness (Roick et al, 2020). However, it is important to note that this often comes at the cost of long-term side effects, the full consequences of which are not yet completely understood (Roick et al, 2020).

Numerous studies worldwide have been conducted to establish the long-term side effects that treatment for leukaemia leaves behind, often with a focus on the physical symptoms, such as cardiac problems and the risk of secondary malignancies (Oeffinger et al, 2020). However, in addition to the physical ramifications of leukaemia treatment, it is vital that the impact on the individual's psychological, psychosocial and emotional wellbeing are also understood. Surprisingly, there is comparatively little research providing clarity on this subject, despite the fact that the National Institute for Health and Care Excellence (NICE) (2005) has identified this as a significant area of unmet need. Subsequently, there are very few guidelines available, despite the evolving numbers within this patient group.

The National Cancer Institute (2022) has defined a person's psychosocial health as their state of mental, emotional, social and spiritual wellbeing. It follows this statement by stressing the importance of positive psychosocial wellness, identifying its influence on how a patient thinks and feels, alongside its impact on their coping mechanisms and relationships. Studies in CYP have identified that the impact of cancer on their psychosocial health can include altered life perspectives, differing perceptions of self and impacts on relationships (Brown et al, 2016).

Method

The search aimed to identify literature that discussed the psychosocial, psychological and emotional wellbeing of CYP following the diagnosis and treatment of leukaemia. The literature search was performed from March to September 2020, with the target cohort comprising CYP who had completed treatment within 5 years of participating in the study. This diagnosis bracket was chosen as it was felt that any longer since treatment may have reduced the accuracy of the participants' recollection of that time, as highlighted by Schmier and Halpern (2004). The search parameters were then set to articles published within the past 20 years. The rationale for this broader time frame was to allow for the substantial medical advances that have occurred in recent times, while also taking into consideration the specialised nature of the subject area, which limited search results. The articles were restricted to being in English, with the addition of only free full text due to time restraints. Four databases were employed for the literature search, these were Medline, CINAHL, PsycInfo and Cochrane Library.

The search terms are set out in Table 1. Once combined, these three groups yielded 3239 articles. Box 1 lists the literature search's inclusion and exclusion criteria.

Box 1.Inclusion and exclusion criteria
CYP who had been diagnosed with leukaemia within 5 years at the time of participation

Additionally, to ensure this article provided a robust literature review, the authors hand searched through the journals and searched reference lists (including Cochrane Library).

In total, 3239 studies were considered during the selection process. Once duplicates were removed and the inclusion and exclusion criteria were applied, nine pieces of data remained for review. Most articles were excluded due to their inclusion of other cancers; however, another reason was articles that related the experiences of solely a parent or healthcare provider, removing the voice of CYP. The Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) chart in Figure 1 details the full search process executed (Moher et al, 2015). Table 2 briefly summarises the included literature.

A thematic analysis was applied to all studies rather than a direct comparison. To do this efficiently, a data extraction tool was used in order to provide focus and consistency when reviewing the studies, encouraging the same level of uniformity throughout each article (Bettany-Saltikov, 2012; Wakefield, 2015). The themes were identified by grouping together the results compiled from all nine studies and extracting the most prevalent.

Within the field of paediatric cancer, there is a wealth of published information pertaining to practice development and quality improvement. As a result, a critical appraisal tool, developed by Caldwell et al (2011), was included in the screening process of this review to appraise the quality of the evidence and to ensure only high-quality studies were included.

Within the final nine studies selected for this review, two adopted a qualitative methodology (McGrath et al 2004; Han et al, 2011). The remaining seven articles adopted a quantitative methodology.

Results

By reviewing the themes and results of the nine pieces of literature, new themes emerged. There were five overarching themes identified:

• Experienced emotions
• Relationships with peers
• Relationships with family
• Academic challenges
• Differentiating factors.

Experienced emotions

The initial theme to emerge from the literature was the different emotions experienced by CYP with leukaemia. The two qualitative studies emphasised the impact of the challenges and changes to lifestyle that the CYP face after a leukaemia diagnosis (McGrath et al, 2004; Han et al, 2011). Similar terminology could be seen within both articles, with participants using self-reported terms such as: ‘fear’, ‘boredom’, ‘anxiety’ and ‘uncertainty’ when talking about themselves. Each of the qualitative researchers also included parental-by-proxy experiences, which echoed the experiences of the CYP, with anxiety and fear frequently cited.

McGrath et al (2004) found that the lack of understanding at diagnosis significantly contributed to CYP fears and anxieties (participants from birth up to 16 years). They further reported that CYP indicated that they were too upset and fearful at the time of diagnosis to absorb information. Furthermore, Han et al (2011) observed that their participants reported negative experiences and feelings at the beginning of treatment, usually within the first 3 months. These participants used terms such as ‘unlucky’, ‘anxious’ and ‘numbness’ when describing their emotions. Comparable results were uncovered in the quantitative studies, with Çavusoglu et al (2015) recording anxiety as one of the top four psychological symptoms for their participants with leukaemia, alongside hostility, depression and somatisation. However, in contrast, Tremolada et al (2020) found their healthy peer control group reported a higher percentage of anxiety than that of the leukaemia group.

Interestingly, Han et al (2011) and McGrath et al (2004) both identified emotions commonly associated with grief such as shock, denial and numbness. Low self-esteem was another element that was a reported concern among CYP with leukaemia. Tremolada et al (2017) found that 75% of their participants suffered from low self-esteem, specifically in the aspects of interpersonal relationships and family life. This study employed a control group of healthy age-matched participants, discovering that two-thirds of their leukaemia cohort reported lower scores than that of their healthy peers. Tremolada et al (2020) reported similar findings. However, they additionally observed that, despite the healthy peer group reporting a better perception of current life, they also demonstrated lower scores of hope and a higher percentage of cognitive problems.

Relationships with peers

Relationships with peers was an overarching theme present in six of the articles included in this review (McGrath et al, 2004; Han et al, 2011; Çavusoglu et al, 2015; Arpaci and Kilicarslan Toruner, 2016; Tremolada et al, 2017; Tremolada et al, 2019). Both qualitative studies noted that CYP self-reported that prolonged hospitalisation caused them to miss out on spending time with peers (McGrath et al, 2004; Han et al, 2011). Han et al (2011) elaborated on this, stating that their participants felt a strong desire to return to their pre-leukaemia diagnosis lives and personal relationships. This disruption to peer relationships was likewise reiterated in the quantitative studies. Arpaci and Kilicarslan Toruner (2016) found that 12.1% of their 91 participants experienced a negative effect on their peer relationships. The CYP recruited in this study reported ‘failure of continuity of communication with their friends due to the long-term treatment’ (n=3), ‘exclusion by a friend’ (n=1) and ‘having difficulties getting in touch with friends and adaptation problems due to the long-term treatment’ (n=7). Tremolada et al's results (2019) support this finding, additionally acknowledging that social communication was problematic.

Han et al (2011) identified a ‘compulsion to adapt’ whereby CYP started to adapt to life with leukaemia until they had to return to hospital for a new course of treatment, at which point the impact on their school and home life would be felt again. In the same study, CYP who fully ‘adapted’ to their diagnosis, said that having playmates of a similar age was a supportive factor in helping them to cope (Han et al, 2011).

Çavusoglu et al (2015) examined the perceived social support and psychological symptoms of young people diagnosed with leukaemia. The authors identified a negative relationship to be found between social support from friends and the CYP's Brief Symptom Inventory scores. Their results demonstrated that CYP who perceived themselves as having reduced social support, also expressed a negative self-image, depression, anxiety and hostility scores. Interestingly, Çavusoglu et al (2015) also observed that their participants recognised they received greater social support from family members than from friends. This leads to the third theme identified: relationships with family.

Relationships with family

Some comparisons can be drawn between the way leukaemia impacts upon both peer and family relationships, as it was identified that CYP reported missing both their friends and family when subject to prolonged hospitalisation (McGrath et al, 2004; Han et al, 2011). However, some differences in the way these relationships are impacted can also be identified, as despite the earlier discussed finding, which demonstrated a greater perceived support from family members than from peers, Arpaci and Kilicarslan Toruner (2016) observed that leukaemia had a negative effect on family relationships in 12.1% of CYP.

Studies found that family relationships were occasionally strained, with parents reporting children to be aggressive and overwhelmed (McGrath et al, 2004; Han et al, 2011). Arpaci and Kilicarslan Toruner's (2016) parental participants reported their children as ‘being aggressive’ and ‘not listening to them due to the long treatment process’ (n=11) and found that ‘because of the family's extreme care and precision during the treatment period, children were overwhelmed by this interest and became estranged from their parents' (n=2). Similarly, Tremolada et al (2017) observed that interpersonal relationships and family life had the lowest reported score on a self-esteem test among their cohort of CYP. Tremolada et al (2019) and Arpaci and Kilicarslan Toruner (2016) included parental-by-proxy reports. Some parents described their CYP as demonstrating ‘irritability’ and ‘sensitivity’ (Arpaci and Kilicarslan Toruner, 2016), while Tremolada et al (2019) observed that the parents of children aged 2.5-4 years reported significantly lower interpersonal relationships than those in the healthy control group. This reveals the conflicting views on whether a leukaemia diagnosis can be an opportunity for increased family support (Çavusoglu et al (2015) or a strain on relationships.

Academic challenges

Academic challenges were reported in many of the studies included in this review, with some finding school-related issues and the lack of education to be the main challenge reported by CYP with leukaemia (Arpaci and Kilicarslan Toruner, 2016; Tremolada et al, 2017; Tremolada et al, 2019). To investigate this concern, some researchers recruited a control group to compare leukaemia participants with their healthy peers (Nazari et al, 2014; Tremolada et al, 2019; Tremolada et al, 2020). However, it should be noted that, despite CYP receiving lessons in hospital or via a home tutor, Tremolada et al (2017) found that, for 21.4% of their participants, the curriculum provided by tutors or in hospital was not always the same as that followed by their school peers.

Nazari et al (2014) concluded that their leukaemia cohort had significantly reduced scores in school performance compared with the healthy control group. Similarly, Tremolada et al (2017) found that 62.5% of their participants were concerned about their academic success, with parental reports revealing that 43.5% had difficulties at school, mainly in the areas of concentration or socialisation. Tremolada et al (2019) additionally recorded difficulties with articulation, use of plurals and verbs in different tenses, alongside their capacity to provide information. Moreover, they found reduced communication abilities among the leukaemia group of children aged 30–60 months, in particular in the subscales of receptive and expressive communication. Tremolada et al (2020) also stated that CYP displayed cognitive problems, difficulties with organisation, concentration and memory problems in their comparative study with peers.

Another study to implement a control group was Tremolada et al (2019). The researchers recruited 44 healthy peer-matched participants, observing the CYP with leukaemia to have significantly lower developmental scores than those of the control group. Despite not using a comparative approach, Arpaci and Kilicarslan Toruner (2016) recorded academic challenges with their group of CYP. They stated that school-related difficulties were accountable for a number (19.8%) of the children's psychosocial challenges, including ‘adaptation problems’ due to the break from school (n=14), ‘absence from school due to treatment’ (n=2) and ‘being educated at home due to the long-term treatment and side effects’ or children being unwilling to go to school (n=2).

Differentiating factors

Throughout the studies, numerous researchers tried to identify factors that may differentiate CYP with leukaemia from one another, to ascertain which CYP were most likely to experience which problems. Tremolada et al (2017) found there to be no significant relationship between their CYP's social demographics and their six self-esteem scales. This article used Spearman's correlation tests to investigate this relationship. These scales included: interpersonal relationships, environmental control competence, school achievement, family life, bodily experience and emotionality.

Çavusoglu et al (2015) sought to investigate whether the interruption period in education, stage of the illness, treatment methods, and time since diagnosis affected CYP's mean Brief Symptom Inventory scores, but observed no significant differences between their variables. The Brief Symptom Inventory tool measures to what extent the CYP has been troubled by various symptoms. Another variable considered was the difference between CYP with acute myeloid leukaemia (AML) and acute lymphoblastic leukaemia (ALL). McGrath et al (2004) identified that their AML group demonstrated a significantly stronger anxiety about death than that of other leukaemia groups. Additionally, Tremolada et al (2017) identified that their AML group had lower self-esteem scores, specifically in the areas of emotionality and bodily experience, than those of the ALL group members. More recently, Tremolada et al (2020) supported this finding, concurring their AML group declared lower life perception scores than that of their ALL participants. However, Çavusoglu et al (2015) found no significant differences between the two diagnoses.

The impact of age was discussed in several of the studies. Han et al (2011) found that children aged between 9 and 12 years struggled to contain their emotions, leading to them feeling impatient and irritable. However, they reported that 7-to-8 year olds, and 13-to-14 year olds adapted to their diagnosis better. These two groups were noted to accept hospitalisation as their ‘new normal’, reportedly creating ‘a new lifestyle’ and ‘felt more enriched’. Along similar lines, Choo et al (2019) found age to also have a significant influence, demonstrating a positive relationship between physical functioning, physical health and the participant's overall quality of life score in older CYP (CYP up to the age of 21 were included in this study).

Tremolada et al (2020) noted that a patient's age could influence their psychological wellbeing and reported intensity of problems. Their results demonstrated that their younger patients (8-13 years) had difficulties within the following domains: cognitive problems, disorganisation, concentration and memory problems, when compared with the older patient group (13-18 years). Han et al (2011) identified that children in the 7-8-year-old and 13-14-year-old age brackets who were extroverted, adapted well to their diagnosis.

Although Çavusoglu et al (2015) found no statistical significance in relation to sex, sex was observed to have an influence in Tremolada et al's (2020) study; they reportedly discovered a significant sex difference in their participants' current life perception, with females reporting lower scores than males. The impact of sex was not discussed in any of the other studies. However, Tremolada et al (2020) observed ethnicity as another variable of significant influence, particularly on the CYP's social functioning. Choo et al (2019) also found that ethnicity has a significant effect on social functioning and found this to a specific influence within the Chinese population.

Discussion

NICE (2014) has defined education, socialisation and relationships as major attributes of the psychosocial needs of CYP with cancer. The research articles in this review identified many negative emotions for CYP diagnosed with leukaemia. Researchers have uncovered similar responses among CYP with other cancers, with additional terms being disclosed such as ‘isolation’ and ‘abandonment’ (Duffey-Lind et al, 2006). However, when compared with the data emerging from generic childhood cancer studies, Reiter-Purtill et al (2003) described a more optimistic outcome. They reported that the children in their study exhibited ‘hardiness’ and claimed childhood cancer patients were minimally impacted when it came to social acceptance and social functioning.

Other studies suggest that many of the lower scores of quality of life and life perception had in fact been recorded via parent proxy and were not provided by the patients themselves (Stam et al, 2006; Eiser et al, 2017). This suggests that parents perceived the treatment journey as a negative experience on behalf of the CYP (Garas et al, 2019). Anclair (2017) suggested that this could be because of fatigue, distress and the fears that parents report themselves following their child's treatment.

The Royal Marsden NHS Foundation Trust (2022) noted that many family members ask for formal psychological support throughout their child's treatment. Formal psychological support begins with a referral from a practitioner (Royal Marsden NHS Foundation Trust, 2022) and, as a result, psychological support is not standard care, and therefore most CYP do not receive this support unless they are displaying problems.

One of the most frequently cited impacts of their cancer among the studies selected for this review was academic challenge (Nazari et al, 2014; Arpaci and Kilicarslan Toruner, 2016; Tremolada et al, 2017; Tremolada et al, 2019). This is consistent with findings in other cancer-related literature, because a decline in school attendance, attention problems and poor academic performance are frequently reported concerns among CYP diagnosed with cancer (French et al, 2013; Li et al, 2013).

The United Nations Convention on the Rights of the Child (UNCRC) (1990) states that all CYP have the right to an education. In a study by CLIC Sargent (2010), children reported that the disruption to their education was one of the most devastating impacts of cancer. It is therefore paramount that treating hospitals support CYP and ensure that home tuition is set up until they are fit to return to school.

Consistent with the results from this study, previous researchers also observed that CYP experienced problems within their relationships. For example, Stam et al (2006) and Schultz et al (2007) suggested that these issues may be triggered as a result of long-term treatment, leading to isolation from their social environments. In a qualitative study examining off-treatment leukaemia patients, An and Lee (2019) found CYP to have difficulties in creating or reviving friendships at school. The participants reportedly felt ‘alienated’ from their classmates and hurt by the perceptions that their physical appearance had changed because of treatment and suggested that more needs to be done to help children engage with their peers.

Due to treatment-related immunosuppression, CYP diagnosed with cancer adhere to strict hospital policies and infection control recommendations. This subsequently reduces their ability to socialise with others at school, home and/or in hospital. As a result, these individuals spend most of their time with parents, carers and health professionals. This may explain, at least in part, Çavusoglu et al's (2015) findings, as their participants were reported to have greater perceived social support from family members than from friends. Nevertheless, this study recognised that CYP value the support of their friends when coping with their diagnosis (Çavusoglu et al, 2015). It had been suggested that the provision of emotional support can help to lessen the psychological stress that accompanies a cancer diagnosis (Enskär et al, 2015). As a result, maintaining these relationships, in additional to social interactions throughout the trajectory of their treatment, is important for overall wellbeing (Corey et al, 2008; Beentjes and Cockett, 2019). Cassano et al (2008) also suggested that friendship networks are crucial at this time in CYP's development, consequently recommending the use of peer support groups to overcome the disruption to social life.

Other research has demonstrated that a parent/carer's way of disciplining their children can significantly impact upon the emotional, cognitive and social maturation of their children (Landry et al, 2000; 2001; Williams et al, 2013). Parents of children with an illness, such as cancer, often deploy overprotective parenting strategies as they perceive their child to be psychologically vulnerable (Thomasgard and Metz, 1993).

One of the research articles in this review revealed that there was no significant association between the patient's age at diagnosis and their self-esteem scores (Tremolada et al, 2017). Tremolada et al's (2017) study included participants aged 10-19 years. This finding is surprising, as in the authors' experience, older CYP usually have a more negative relationship with their body image and self-esteem than younger children. A survey by the Mental Health Association (2019) supports this hypothesis; the results demonstrated that 37% of young people reported being upset about their body image. The charity Macmillan Cancer Support (2022) noted that, in some cases, changes in appearance, such as hair loss, can be one of the hardest side effects of cancer treatment for young people. Another article by Fardell et al (2017) examined the health-related quality of life of children on treatment for ALL, and reported the increasing age of the child was associated with worse overall quality-of-life scores, specifically in the self-esteem and psychosocial domains.

Sex was another variable suggested to have an influence on the CYP's current life perception (Tremolada et al, 2020). It was not detailed within this article whether the females who were reporting lower scores were of pre- or post-puberty as this could affect the findings. Other existing literature on this area of study suggested that puberty can lead to females developing a negative self-image, in addition to behavioural and psychological problems (Copeland et al, 2010).

Strengths and limitations

The strength of this review lies in its choice to address CYP diagnosed specifically with leukaemia. This population of vulnerable individuals has not been independently considered in previous research on the psychological, psychosocial and emotional wellbeing of CYP diagnosed with cancer.

Unavoidably, there are limitations to this review: primarily that the research was restricted to being published in English and used the UK spelling of leukaemia, which inadvertently limited the data from other countries. Despite this, the authors included studies from a variety of geographical locations and although numbers were modest, it demonstrated an international perspective. Additionally, this review could be seen to have some interpretational bias, as the data were synthesised from previous research papers.

Conclusion and recommendations

This review clearly demonstrated the need for early intervention when supporting CYP emotionally, which has been reiterated in previous cancer studies (CLIC Sargent, 2012; Enskär et al, 2015). As a result, it is this article's first recommendation that all CYP diagnosed with leukaemia are offered a referral to the hospital's clinical psychology team from diagnosis.

NICE (2014) has advocated that all CYP diagnosed with cancer should be able to avail of the same standard of service provisions. This leads to the authors' second recommendation, the implementation of a standardised holistic nursing assessment (HNA) tool. Best practice would involve a standardised tool used across all CYP's cancer centres in the UK to ensure alignment with NICE guidelines (2014). Additionally, once the HNA tool has been chosen, staff training would be required to promote appropriate use of the tool.

Despite some of the studies reporting positive experiences within the hospital school, unfortunately this is not mirrored within the public education setting (McGrath et al, 2004; CLIC Sargent, 2012). This leads the authors to a third recommendation for better educational support. All CYP should have a referral for home tuition as soon as they are fit to participate, in concordance with international law (UNCRC, 1990; CLIC Sargent, 2012). All education boards have a legal obligation to ensure this home curriculum is up to the appropriate standard (UNCRC, 1990). Further research on this topic would be beneficial, to establish what is required to better support these CYP academically, and to standardise the approach to their education nationally.

Negative effects on normal parenting and discipline have been cited as an impact of leukaemia treatment, with parents articulating that their children were ‘not listening to them due to the long treatment process’ (Arpaci and Kilicarslan Toruner, 2016). Other articles have found that parents of CYP diagnosed with cancer use overprotective parenting strategies and struggle to enforce discipline (Thomasgard and Metz, 1993). As a result, the authors propose the development of an education resource for parents, as the fourth recommendation of this review. In conjunction, staff education on how to adequately support parents with this challenge would be beneficial.

This literature review identified a lack of peer support for younger children, suggesting this is an area requiring further study. Regardless of the conflicting results regarding the strain versus the support benefit of interpersonal relationships, it is an area frequently cited (McGrath et al, 2004; Han et al, 2011; Çavusoglu et al, 2015; Arpaci and Kilicarslan Toruner, 2016; Tremolada et al, 2017; Tremolada et al, 2019). The impact upon CYP's personal interactions is evident with both peers and family, with other studies suggesting that better peer relationships were associated with better family relationships (Noll et al, 1991; Erker et al, 2018). This finding highlights the need for more robust peer support in CYP with leukaemia, which is our fifth recommendation.

Although this article has identified several recommendations, in the opinion of the authors, the most important of these is that health professionals have a strong awareness of the psychological, psychosocial and emotional wellbeing impact this condition has on CYP.

KEY POINTS

• A cancer diagnosis instils many mixed emotions in children and young people (CYP), although it is frequently linked to many negative emotions such as fear and anxiety. Formal psychological support is recommended for CYP undergoing leukaemia treatment to support this group with their emotional wellbeing
• CYP with leukaemia identified that they benefit from social support from peers. Reduced social support is linked to negative self-image, depression and anxiety
• CYP with leukaemia occasionally reported strained familial relationships but said that they received greater social support from family than friends
• A lack of education was reported by many CYP with leukaemia. Improved educational support is recommended to better support these CYP

CPD reflective questions

• Thinking critically about the care provided in your area of work, are the psychological, psychosocial or emotional wellbeing impacts of a leukaemia diagnosis given equal consideration to the long-term physical impacts?
• What psychological support do you currently offer children and young people undergoing leukaemia treatment and how could this be improved?
• Considering the five themes discussed in this article, which can you identify as being one on which you could further reflect with your wider team?