Psychosocial adjustment of children who
have ended brain tumour treatment:
child and parent reports
A systematic literature review
Leire Cea
One year master thesis 15 credits Maria Björk
Interventions in Childhood
Examiner
SCHOOL OF EDUCATION AND COMMUNICATION (HLK) Jönköping University
Master Thesis 15 credits Interventions in Childhood Spring Semester 2020
ABSTRACT
Author: Leire Cea
Psychosocial adjustment of children who have ended brain tumour treatment: child and parent reports
A systematic literature review
Pages: 44
Paediatric brain tumour is the second most common childhood cancer type. As sur-vival of these children has increased, psychosocial aspects of after treatment quality of life has started to gain importance. They face several neurological deficits and late effects, which eventually cause problems with psychosocial adaptation. It is there-fore of great importance to understand what aspects affect psychosocial adjustment. This paper examines the psychosocial adjustment of children who have ended brain tumour treatment from the perspective of the child and the parents. A systematic lit-erature review was conducted and resulted in 10 suitable articles. The children were 6-18 years old and were brain tumour survivors. Findings showed that both children and parents reported brain tumour survivors to have several concerns affecting their daily life (e.g. low social support, low physical appearance rates, poor psychological functioning). The factors that had the most impact on the psychosocial adjustment were type and length of treatment. These results highlight the problems these chil-dren endure in their everyday lives. Therefore, it is of great importance to early iden-tify these problems in order to better ideniden-tify those factors that affect their psychoso-cial adjustment, plan early interventions and prevent those problems from increasing
Keywords: Systematic literature review, paediatric brain tumour survivors, psychosocial ad-justment, parent- reports, child- reports
Postal address Högskolan för lärande och kommunikation (HLK) Box 1026 551 11 JÖNKÖPING Street address Gjuterigatan 5 Telephone 036–101000 Fax 036162585
Table of Content
1 Introduction ... 1
1.1 Psychosocial adjustment …... 1
1.2 Brain tumours ... 1
1.2.1 Definition and classification ... 1
1.2.2 Incidence and survival ... 2
1.2..3 Diagnosis and treatment ... 2
1.3 Health in long- term survivors ... 3
1.4 Theoretical framework ... 5
1.5 Rationale, Aim and Research Questions ... 7
2 Methodology ... ... 8
2.1 Methods and Data-analysis ... 8
2.2. Study Design ... 8
2.3 Search Strategy ... 8
2.4 Selection Criteria ... 9
2.5 Selection Process ... 11
2.5.1 Protocol for study selection... 11
2.5.2 Study selection process ... 11
2.6 Ethical considerations ... 14
2.7 Quality assessment ... 14
3 Results ... 15
3.1 Study Characteristics ... 15
3.2 Participant Characteristics ... 20
3.3 Findings of the Articles ... 20
3.3.1 Problems that children who have ended brain tumour treatment report related to their psychosocial adjustment ... 20
3.3.2 Problems that parents of children who have ended brain tumour treatment report related to their psychosocial adjustment ... 21
4.3.3 Factors that influence how children adjust to the psychosocial aspects of everyday life after ending their cancer directed treatment toward brain tumour... 22
4 Discussion ... 27
4.1 Reflections on findings ... 27
4.1.1 Problems that parents of children who have ended brain tumour treatment report related to their psychosocial adjustment ... 28
4.1.2 Factors that influence how children adjust to the psychosocial aspects of everyday life after ending their cancer directed treatment toward brain tumour... 29
4.2 Methodological issued and limitations ... 30
4.3 Future Research ... 31
4.4 Clinical implications ... 31
5 Conclusion ... 32
6 References ... 33
1
1 Introduction
1.1.Psychosocial adjustment
The term “adjustment” refers to the changes in life that constantly occur when an individual suffers from a chronic illness. When it comes to the term “psychosocial”, it is a wider term that contemplates the social environment and the psychological condition (Larsen, 1990, p. 43). Thus, psychosocial adjustment is the psychosocial adaptation of a person to a life- modulating occurrence or change (Stubley & McCroy, 2014). In the field of psychology, research usually describes psychological adjustment, whereas psychosocial adjustment is more commonly en-countered in nursing (Larsen, 1990).
The journey through cancer and its psychosocial consequences are not very understood (Yallop, McDowell, Koziol-McLain & Reed., 2013). Several studies evoke that children and adolescents with a cancer diagnosis are at greater risk for diverse psychosocial adjustment prob-lems such as poor self- esteem, less ambitious ideals, poor self- satisfaction, depression, death anxiety, poor social skills, school reintegration problems and school phobia. Nevertheless, other studies reported healthy grades of psychosocial adjustment amongst long- term survivors (Bes-sell, 2001). A probable explanation for these opposing findings is the diverse heterogeneity of the participants regarding diagnosis, age at diagnosis, time since diagnosis and treatment- in-tensity (Bessell, 2001). For a paediatric brain tumour survivor, impairment in social capacity is a relevant psychosocial sequela. Children and adolescents who survive brain tumour treatment are less prone to have close friends and more socially isolated (Desjardins et al., 2019).
1.2. Brain tumours
1.2.1. Definition and classification
A brain tumour is a mass of abnormal cells that originate in the brain. Brain tumours cause neurologic symptoms that might differ depending on the location, dimension and invasiveness (Nejat, El Khashab & Rutka, 2008). The primary brain tumours can be benign, invasive or
2 malignant. The classification is based on the site of the tumour, the type of tissue that gets involved and whether it is benign or malignant (National Library of Medicine, 2019)
1.2.2. Incidence and survival
Paediatric brain tumours are the most usual type of solid childhood cancer, and they entail the second most common paediatric neoplasia (Bowers, Adhikari, El-Khashab, Gargan & Oeff-inger, 2009; Darcy et al, 2015; Dessens, Herwerden, Aarsen, Birnie & Catsman-Berrevoets, 2016; Fischer, Petriccione, Donzelli & Pottenger, 2016; Gupta & Jalali, 2017; Weinberger & Barakat, 2007; Soria et al, 2008). Amongst children, leukaemia (33%) is the most common type of cancer, followed by nervous system cancer (NSC) (20%), lymphomas (12%), neuroblasto-mas (8%), soft- tissue sarconeuroblasto-mas (7%), renal tumours (6%) and bone tumours (5%) (Ajovalasit et al, 2009).
Globally, the frequency of primary malignant brain and central nervous system (CNS) tu-mours is 3.7 per 100,000 people per year in males and 2.6 per 100,000 people per year in fe-males. In children, this incidence rate is 2.6 per 100,000 per year for 0 to 19 years old, and these rates are more elevated in developed countries than in developing ones (Ajovalasit et al, 2009). Close to 80% of children with brain tumour survive at least 5 years (Gunn et al, 2016). Accord-ing to the Childhood Survivor Study (CCSS), by 30 years after the diagnosis of the cancer, 73.4% of the children will have at least one chronic illness, with 42.4% of the survivors having a severe or life- threatening condition caused by a chronic disease (Bowers et al, 2009).
1.2.3. Diagnosis and treatment
The diagnosis of a brain tumour is frequently hard to establish in children since many of the symptoms and signs might mimic those of more usual paediatric pathologies (Nejat et al., 2008). Advances in neuroimaging have enabled professionals to perform earlier diagnosis and to be more precise about tumour disseminations and recurrence (Nejat et al., 2008). Survival rates are wide- varying depending on tumour stage and type (National Cancer Institute, 2019). Long- term outcomes associated with the initial appearance of the tumour and the treatment are usual. Debilitating effects on neurologic development and growth have commonly been ob-served after radiation therapy treatment, mainly in younger infants (National Cancer Institute, 2019). Secondary tumours have more frequently been diagnosed in cancer long- term survivors.
3 Due to this, the role of chemotherapy in enabling delay or decrease in administration of radia-tion therapy remains under study. Initial results reveal that chemotherapy can be used to limit, delay and sometimes obviate the need for radiation in children with benign and malignant le-sions (National Cancer Institute, 2019). This long- term management of the treatment and the subsequent consequences are challenging and complex and demand a multidisciplinary ap-proach (National Cancer Institute, 2019).
Cranial radiation therapy has been related to the highest risk of long- term cognitive morbidity, especially in younger infants, and there is an existing established dose- response relationship, being children who receive higher- dose of cranial radiation therapy suffering from more late effects (National Cancer Institute, 2004). The neurocognitive outcomes of CNS can-cers and treatment might have a reasonable impact on functional results for brain tumour sur-vivors. Neurocognitive deficits have been related to poor social adjustment, and these paediatric population are more likely to need special education support than survivors from other different malignancies (National Cancer Institute, 2004).
1.3.Theoretical framework
The WHO defines health as a “state of complete physical, mental and social well-being and not merely the absence of disease or infirmity”. The International Classification of Functioning, Disability and Health- Children and Youth version (ICF-CY) describes components of health and health-related components of well-being, and it offers a conceptual framework and a com-mon language and terminology for recording problems manifested in infancy, childhood and adolescence involving functions and structures of the body, activity limitations and participa-tion restricparticipa-tions, and environmental factors important for children and youth (World Health Organization, 2007). The ICF-CY defines participation as “involvement in a life situation (Imms, Granlund, Wilson, Steenbergen, Rosenbaum & Gordon, 2017), and a fundamental focus of this tool is to report children´s participation in everyday life (Granlund, 2013). It can as well help professionals working with children in need of special support to facilitate participation in their daily life activities (Granlund, 2013). The ICF-CY also offers a more integrative percep-tion of health creating a comprehensive profile of an individual instead of centring on one´s health status (Darcy et al., 2015).
4 As an individual, participation could be perceived as a universal outcome, which is sig-nificant for both learning and development as well as for health and well- being (Imms et al., 2017). The social and physical settings are important aspects of the child´s capacity to perceive health and be able to function in everyday life (Almqvist, Hellnäs, Stefansson & Granlund, 2006). Participation of children in distinct activities promotes both their physical and psycho-logical health, and they obtain skills and create social abilities that are important when becom-ing an adult (Gorter, Stewart & Woodbury‐Smith, 2011). As cancer survivors, these children tend to have lot of problems regarding socialization, and thus promoting and enhacing partici-pation could help them get involved in society and interact with peers. In addition, promoting participation could also improve psychosocial health and therefore meliorate their psychosocial adjustment.
The implications of utilizing the ICF-CY include accentuating the strengths of children and adolescents, supporting individuals in participating more substantially in society with in-terventions aimed at improving their capacities, and considering the environmental and per-sonal factors that might obstruct one´s participation (Granlund, 2013). Darcy et al. (2015) doc-umented the existing necessity to report health and functioning in the daily life of children and adolescents with cancer utilizing the multidimensional framework of the ICF- CY. They also expressed that this tool can be used to help and guide health care professionals´ clinical ap-praisal of the child´s health and everyday life performance. This idea could encourage future research to utilize the ICF-CY as an assessment instrument for analysing everyday life of chil-dren who have ended their cancer directed treatment and, this way, enabling a tool that could help professionals with improving the psychosocial adjustment of these children.
5 Figure 1. The framework of the International Classification of Functioning (ICF)
1.4.Health in long term survivors
As survival and life expectancy have ameliorated in the past two decades, rising focus has been given to long- term outcomes and adaptation of this paediatric population (Weinberger & Barakat, 2007). Thus, there is an existing and increasing need to focus on quality of life of these patients (Bhat et al, 2005). The cancer itself and the treatment might cause health- related prob-lems months or years after the successful treatment is over. These probprob-lems are considered late effects (National Cancer Institute, 2019). Late effects might affect organs, tissues, body func-tion, growth and development, mood, feelings, actions, thinking, learning, memory, social and psychological adjustment, and the risk of second cancers (National Cancer Institute, 2019). These late effects might depend on the degree of the harmfulness, age at event, location and treatment (Soria et al, 2008).
Children who survived brain cancer suffer from a considerably decreased health condi-tion compared with their peers or with other childhood cancer survivors (Soria et al, 2008). The urgent need to discover the actual importance and significance of health- related quality of life (HRQOL) for this paediatric population increases the need to ask children and adolescents di-rectly about their own experience and its effects on their thoughts, feelings, decisions and other health- related outcomes. Children´s own rating of their HRQOL ought to be considered as the primary rating rather than from a proxy such as parents or health professionals. Regularly used
6 and alternate labels for cancer- related HRQOL interview guidance include normal activities, symptoms and social/family interactions. Other identified but scarcely used classifications in-clude communication, disclosing behaviour and compliance. A small number of instruments for evaluating HRQOL focus on the perspective of the child or the teenager. Some self- report HRQOL instruments have been recounted as valid and reliable in infants as young as 5. How-ever, proper measures for children younger than 5 years have not been developed (King & Hinds, 2011).
Increasingly, there is an existing emphasis on considering HRQOL from a more holistic approach by acknowledging a link between physical and psychosocial domains. Interventions to improve this HRQOL would be different if the focus would be on helping children make meaning from experience rather than from improving functionality (King & Hinds, 2011). The main goal of care after cancer cure is to maximise the potential of paediatric patients affected by the emotional and physical burden of a brain tumour diagnosis and its treatment and acquire the best quality of life. This embraces the hope that teenagers and young adults will feel they have control of their destiny, and when possible, make paediatric patients feel “demedicalised” (Kelly & Gibson, 2008).
The need for long- term follow up for children who survived cancer remains supported by the American Society of Paediatric Haematology/Oncology, the International Society of Pae-diatric Oncology, the American Academy of PaePae-diatrics, the Children’s Oncology Group (COG), and the Institute of Medicine. A risk- based medical follow up is suggested, which includes a systematised plan for lifelong screening, surveillance and prevention that includes risk estimates based on cancer therapy, previous cancer, genetic predisposition, comorbid con-ditions, lifestyle behaviours and sex (National Cancer Institute, 2004).
1.5.Rationale, aim and research questions
Survival of paediatric oncologic patients has increased over time. Thus, the impact on survi-vors´ quality of life after treatment is starting to acquire importance. Medical and physical as-pects of HRQOL have gained prominence. Nonetheless, scant attention has been given to both social and psychological aspects. Brain Pediactric brain tumour survivors (PBTS) have to face several neurological deficits and late effects. In addition, their treatment result in social dys-function and psychological suffering that affect these children in different developmental
7 stages. It can therefore be appreciated that brain tumour survivors confront greater challenges of psychosocial adjustment. Bearing in mind the existing problems these children and adoles-cents suffer, it is of great importance to identify the factors that affect their psychosocial adjust-ment early in order to plan promotive interventions, as well as to understand how children themselves and parents perceive the psychosocial adaptation of these paediatric population after ending brain tumour treatment. Awareness and education of health professionals is also of great significance. Hence, this paper will try to systematically gather and review the current available literature about this paediatric population.
The aim of this systematic review is to report the psychosocial adjustment of children who have ended brain tumour treatment from the perspective of the child and the parents.
The following research questions will guide the research process:
(1) What problems do children who have ended brain tumour treatment report related to their psychosocial adjustment?
(2) What problems do parents of children who have ended brain tumour treatment report related to their psychosocial adjustment?
(3) Which factors influence how children adjust to the psychosocial aspects of everyday life after ending their cancer treatment?
8
2 Methodology
To identify research on psychosocial adjustment of children who have ended brain tumour treat-ment, a systematic literature review was conducted. A systematic review was used in order to synthesize the results of diverse primary investigations by utilizing strategies that limit bias. These strategies embrace an exhaustive search of all relevant articles and the use of a clear criteria when selecting the articles (Cook, Mulrow & Haynes, 1997). This section describes the study design, search strategies, selection criteria, selection process, peer review, ethics, quality assessment and data-analysis method utilized in this systematic review.
2.3. Search strategy
The literature search for this systematic review was performed between January and February 2020, using relevant electronic databases. The databases used for the search were Pubmed, Ci-nahl, Psycinfo and Sciencedirect. These databases were selected because they provide compre-hensive literature focusing on psychology and health,and include articles directed to children and their parents in paediatric oncology settings. Two search strategies were performed in order to broaden the information regarding the main aim. Initially, HRQOL was thought to be the main focus of this paper. Since the term was too broad to address, the central topic was changed but the search strategy remained included because it bestowed relevant information for the study purpose. The second strategy included search terms that complemented the main aim; the term “emotional adjustment” was added because it was found to be a Mesh synonym term. With both search strategies, information including psychosocial aspects was concluded. The first search strategy was Child* AND (Brain Tumor OR Brain Neoplasm OR Brain Cancer) AND (Life Quality OR HRQOL) AND Cancer survivor, and it was performed in the database Pubmed. The second search strategy was Child* AND (Brain Tumor OR Brain Neoplasm OR Brain Can-cer) AND Survivor AND (Psychological Adjustment OR Emotional Adjustment), and it was conducted in Pubmed, Psycinfo, Cinahl and Sciencedirect. Other databases like Scopus and Cochrane were also utilized, but due to the elevated amount of results, the author decided not to use them. All the searches were limited to scholarly articles published in English language. The search terms were developed in relation to the study aim and research questions.Several search strategies adopting different terms and strings were used beforehand, which were
even-9 tually dismissed. A truncation was used in order to broaden the search and include diverse end-ings. Different filters were used in different databases depending on the availability and main interests of the study.After applying the filters to the corresponding search strategy, the titles of the articles were examined. If they seemed to be relevant for the aim of this study, the corre-sponding articles were appraised after reading the abstract, and when necessary, the full text, aiming at ensuring that no relevant articles were missed.
2.4. Selection criteria
Selection criteria used for the screening were determined based on the research questions. The aim was to analyse the psychosocial adjustment of children who have ended brain tumour treat-ment, so research that was describing and evaluating such population was considered. The three steps in the data collection process (article election, data extraction, and quality assessment) were performed. The included articles had to be peer reviewed, describe empirical studies and be written in English. Grey literature and incomplete articles were excluded because they lack sufficient precision for the detailed comprehension and analysis of the articles. Systematic lit-erature reviews and litlit-erature studies were also excluded. No restrictions regarding publication date were applied. The participants of interest were children aged 6-18 years old who had ended brain tumour treatment, as well as parents or caregivers. In some cases, the age limit was ele-vated to 20 years old, as many paediatric care settings continue to treat children until this age. Children with health conditions other than brain tumour were excluded.
10 Table 1: Inclusion and Exclusion criteria.
Inclusion Exclusion
Population
Children aged 6-18/+2 years
Children who have ended brain tu-mour treatment
Ages >6 and <20
Children with other health conditions than brain tumour
Content
Children´s perceptions about their psychosocial adjustment
Parent´s or caregiver´s perceptions about their child´s psychosocial adjustment
Factors that affect children´s psy-chosocial adjustment
No mentioning of psychosocial aspect in the study
Design
Empirical studies Systematic literature reviews or literature studies
Publication type
Peer reviewed articles
Full text available
Language: English
Grey literature (e.g. books, conference ab-stracts)
11 2.5. Selection process
2.5.1. Protocol for study selection
A selection protocol aiming at screening and electing articles for the present study was devel-oped. Microsoft Excel was used in order to organize, assess and document the selection process. During the screening, the author categorized each article by choosing ’no’, ’maybe’ or ’yes’. In the title/abstract screening, ’yes’ meant including the article in the succeeding full-text screen-ing. In the full-text screening, that ’yes’meant including the article in the review, and in the last screening, inclusion and exclusion criteria were clarified and utilized to keep the selection process transparent.A detailed protocol (Appendix 1) was elaborated to examineand extract the data of the final articles. This protocol focused on the study’s research aims and questions (e.g. psychosocial adjustment and factors that did influence the psychosocial adjustment) and included a section about limitations and research implications. It was designed with the aim of providing a standardized content-analysis of the articles.
2.5.2. Study selection process
During the selection process, an evaluation of the articles was accomplished first on the title level, followed by the abstract and eventually the full- text level. They were all screened using the inclusion and exclusion criteria as a checklist.
The three steps in the process of data gathering were performed (selection, data extrac-tion and quality assessment). In the first search 206 articles were found in Pubmed. After ap-plying the database specific filters regarding age limit, 183 articles remained. Pursuant to title and abstract screening, and taking into consideration the inclusion criteria, 12 articles were en-compassed. In the end, 5 articles were chosen. In the second search, with the second search strategy, 37 articles were compiled in Pubmed, 28 in Psycinfo, 20 in Cinahl and 1885 in Sci-encedirect. Since too many articles were found, they were limited with database tools. Some filters were applied to the databases based on the established inclusion and exclusion criteria. In Pubmed age limit of 6-18 was stablished, in Pysicinfo same age limit and peer reviewed articles, in Cinahl peer reviewed articles were taken into consideration and in Sciencedirect the most relevant journals for the study aim were utilized. Pursuant to this, 32 articles remained in
12 Pubmed, 11 in Psyinfo, 20 in Cinahl and 142 in Sciencedirect. When titles did not fulfil the expectations for the aim of this study, they were removed. After reading the abstract of the remaining articles, 15 articles from Pubmed, 7 from Psycinfo, 11 from Cinahl and 12 from Sciencedirect were included because they met the inclusion criteria (table 1). In the end, 5 arti-cles were chosen. Taking into consideration both search strategies, 10articles remained for data extraction and quality assessment.
13 Figure 2. Search strategy
1st search strategy 2nd search strategy
Pumbed Cinahl Psycinfo ScienceDirect
2176
After filter use
Data analysis
Title and abstract screening
Full text screening
206 37
20
28
1885
388
57
17
10
Excluded: Duplicated Not related to aim Literature studies orsystematic reviews Not about PBTS
Excluded: Age (n=1) Not related to aim
(n=4)
Not sufficient refer-ence to PBTS (n= 1) Only parent- reports
14 2.6. Ethical considerations
There are several ethical issues that need to be taken into consideration when conducting re-searchwith children, especially with vulnerable groups of children such as pediatric brain tu-mour survivors (PBTS), meaning: obtaining informed consent, controlling the risk of potential or further harm to the child, and managing the information obtained from the participants (Chowdhury, 2014). The United Nations on the Rights of the Child is frequently used as a basis when accomplishing research with children, and the compliance of the Nuremberg Code and the Declaration of Helsinki should always be ensured (Chowdhury, 2014). Furthermore, a re-search ethics committee ought to ensure that rere-searches performed with children are reviewed and authorised before effectuating them (Royal College of Pediatrics and Child Health, 2000). Articles included in this study were peer- reviewed. Thus, they should be based on ethical prem-ises. Most of the included articles explicitly reported that they had received an ethical approval from the ethics committee (Barrera et al., 2017; Cheung et al., 2019; de Ruiter et al., 2016; Eilertsen et al., 2012; Emond et al., 2016). Two articles did not directly report an ethical ap-proval but mentioned being reviewed by a board (Meeske et al., 2007; Quast et al., 2018).
2.7. Quality assessment
A quality assessment tool was utilized in order to assess the level of evidence of the incorpo-rated articles. To suit this paper CASP checklists for quantitative (2017a) and qualitative (2017b) research were used, as well as the Critical review form of Law and colleagues (1998) for quantitative studies and the Critical review form (Letts et al., 2007) for qualitative studies. An assessment protocol for mixed- method studies was also created based on the Mixed Meth-ods Appraisal Tool (MMAT) for systematic mixed studies reviews of Pluye et al. (2009) and on the quantitative and qualitative assessment protocols. Questions from the quality assessment tools were merged into an adapted version for this study. The articles were later classified de-pending on the degree of quality. Each question was scored from 0 - 2 points, where 0 repre-sented” None or unknown”, 1 “Partial” and 2 “Adequate”. The score of each article was summed up and ranked as low (0-17 points for the quantitative studies and 0-13 for the mixed- method studies), medium (25-18 points for the quantitative studies and 19-14 for mixed- method studies) or high quality (26-34 points for the quantitative studies and 20-26 for mixed-
15 method studies). After the study selection process, solely quantitative and mixed- method stud-ies were selected.
2.8. Data analysis
Data analysis was performed both during and after data extraction. Even though three research questions were utilized for this study, the main purpose was to have an inductive approach towards the content of the included articles. In accordance with the aim of this paper, articles were analysed in order to gather relevant information during the analysis procedure. Only the parts that comprised information about the psychosocial adjustment of children who have ended their cancer directed treatment towards brain tumour were considered as relevant for the anal-ysis.
3 Results
3.1. Study characteristics
The 10 studies included in this systematic review were carried out in the following countries: Hong Kong (n= 1), Norway (n= 1), United Kingdom (n= 1), The Netherlands (n= 1), USA (n= 5) and Canada (n= 1). There was no restriction for publication dates, so articles that were rele-vant for the study aim were included regardless the date. Nine articles used merely quantitative design, whereas 1 article used both quantitative and qualitative methods (mixed- method). Most of the studies reported local hospitals (n= 4) and clinics (n= 2) to be the most common recruit-ment settings, being centres (n= 1) also utilized recruitrecruit-ment environrecruit-ments. Programs (n= 2) and databases (n= 1) were also ways of enlisting participants for the corresponding researches. All this information can be found in table 2, as well as utilized measurement- tools.
Some studies focused on quality of life (Barrera et al., 2017; Cheung et al., 2019; Ei-lertsen et al., 2012; Meeske et al., 2007; Quast et al., 2018;), whereas others focused on the social or the psychosocial adjustment (Carpeinteri et al., 2003; de Ruiter et al., 2016; Emond et al., 2016; Hardy et al., 2010; Radcliffe et al., 1996). It ought to be mentioned that those articles aimed at examining the health-related quality of life (HRQOL) included information about the psychosocial adjustment, which is why they were incorporated to this systematic review.
16 Regarding the quality of the included articles, since most of them were quantitative, the adapted quantitative assessment protocol was mainly utilized. The range for the implicated studies was from medium to high. Information concerning quality assessment requisites can be found in Appendix 2.
17
Table 2
Study characteristics
Article Location Year Type of article Recruitment setting Aim Measurement tool Quality range
Barrera et al., 2017
Canada 2017 Quantitative Four Canadian sites: (1) Hospital for Sick Children (Toronto, Canada) (2) London Health Sciences Centre (London, Ontario) (3) Alberta Chil-dren´s Hospital (Cal-gary, Alberta) (4) BC Children´s Hospital (Vancouver, British Columbia)
To describe the QOL of PBTS prospectively and to identify potential medical, personal and family contextual factors associated with QOL
(1) Outcome measures
- PedsQL self- and caregiver report (2) Neurocognitive battery
-WASI
- BRIEF parent version - GEC
- Behaviour Regulation Index (BR) - Metacognition Index (MI)
- Conner´s Continuous Performance Test- II (CPT- II)
- Children´s Auditory Verbal Learning Test- 2 (CAVLT- 2)
Medium
Carpeinteri et al., 2003
USA 2003 Quantitative Stop and Shop Fam-ily Pediatric Brain Tumor Clinic at the Dana- Farber Cancer Institute
To describe the psychosocial and behavioural functioning of 12-18 years old children who were evaluated between 1-5 years post- treat-ment for brain tumour
The Behavioural Assessment System for Chil-dren (BASC)
High
Cheung et al., 2019
Hong Kong 2019 Mixed Pediatric outpatient clinic
(1) Assess depressive symptoms, self- esteem, and QoL of Hong Kong Chinese paediatric brain tumour survivors
(2) Compare depressive symptoms, self- es-teem, and QoL between Hong Kong Chinese PBTS and survivors of other paediatric can-cers
(3) Understand how brain tumour and its treatment influence PBTS´ physical and psy-chosocial well- being and QoL
(1) Demographic sheet
- The Chinese version of the Center for
Epide-miological Studies Depression Scale for Chil-dren (CES-DC)
- The Chinese version of Rosenberg self- esteem scale (RSES)
- The Chinese version of the Pediatric QoL in-ventory 4.0 Generic Core Scale (PedsQL 4.0)
(2) Semi- structured interview
18 De Ruiter et
al., 2016
The Nether-lands
2015 Quantitative One of the participat-ing Dutch hospitals
To investigate various domains of psychoso-cial functioning of PBTS who suffer from par-ent- reported neurocognitive complaints: HRQOL, self- esteem, psychosocial adjust-ment, executive functioning, and fatigue
(1) Self- report
-HRQOL: The Kidscreen- 27 & Kidscreen- 10 -Self- esteem: Self- perception profile for
chil-dren (SPPC, 8-12) and adolescents (SPPA, 12-18)
-Psychosocial adjustment: SDQ
-Fatigue: Checklist individual strengths (CIS) (2) Parent report
-Psychosocial adjustment: SDQ
-Executive functioning: The Behaviour rating
inventory of executive function (BRIEF)
(3) Teacher report
-Psychosocial adjustment: SDQ
-Executive functioning: BRIEF teacher- report
version
High
Eilertsen et al., 2012
Norway 2012? Quantitative Paediatric Depart-ment, St Olav´s Uni-versity Hospital, Trondheim
To explore subjective and proxy reported QoL in children and adolescents surviving cancer three years after diagnosis compared with healthy controls
The Inventory of life quality in children and ad-olescents (ILC)
The Kinder Lebensqualität Fragebogen (KINDL) High Emond et al., 2016 United Kingdom 2016 Quantitative (cross- sec-tional) NHS site Pediatric Oncologic database
(1) Provide a multi- informant assessment of social competence in 8-16-year olds treated for a brain tumour
(2) To compare these assessment outcomes to peers
(1) Social competence measures
-The Social Skills Improvement System (SSIS)
rating scales
(2) Secondary measures
-The Wechsler Abbreviated Scale of
Intelli-gence (WASI)
-The Strengths and Difficulties Questionnaire (SDQ) High Hardy et al., 2010 USA 2010 Quantitative (cross- sec-tional) Division of Pediatric Neuro- Oncology at a large university- af-filiated medical cen-tre
(1) To evaluate academic and treatment- re-lated correlates of perceived social compe-tency of children on- and off- treatment for brain tumour
(2) To determine the medical, cognitive and psychosocial risk factors associated with per-ceived competency
(1) Cognitive and academic functioning - Wechsler Intelligence Scale for Children-
Third Edition (WISC-III)
- Woodcock- Johnson Tests of Achievement- Re-vised (WJ-R)
(2) Parent psychosocial functioning - Brief Symptom Inventory (BSI)
19 (3) Child Emotional, behavioural and psychoso-cial functioning
- Child Behaviour Checklist (CBCL)
- Harter´s Self- Perception Profile for Children (SPPC)
Meeske et al., 2007
USA 2007 Quantitative Children´s Hospital Los Angeles
To describe HRQOL in paediatric cancer sur-vivors attending a long- term follow- up clinic and to identify demographic and disease/treat-ment- related factors associated with poor QoL outcomes
LIFE Clinic Intake Questionnaire PedsQL 4.0 Generic Core Scale The Late Effect Severity Scale
High
Quast et al., 2018
USA 2018 Quantitative A Pediatric Neuro- Oncology program in the northeast United States
To examine prospectively the associations be-tween family functioning at the end of tu-mour- directed treatment and the HRQOL of PBTS approximately nine months later
PedsQL
McMaster Family Assessment Device (FAD) Family Management Measure (FaMM)
High
Radcliffe et al., 1996
USA 1996 Quantitative Regional Neuro- On-cology program
(1) To identify the psychosocial status of chil-dren and adolescents who are 2 to 5 years postdiagnosis of a brain tumour
(2) To identify the psychosocial impact of having a child with a brain tumour on mothers of former patients
(3) To identify factors associated with rela-tively good adjustment among a sample of children surviving brain tumours
(1) Child self- report
-The Children´s Depression Inventory (CDI)
- The Children´s Manifest Anxiety Scale- Re-vised (CMAS-R)
- The Self- Perception Profile for Children (SPP)
(2) Parent and teacher report on child
- CBCL
- Teacher Report Form (TRF)
(3) Mother report on child
- The Vineland Adaptive Behaviour Scale (VABS)
(4) Mother self- report
- The Beck Depression Inventory (BDI) - The State- Trait Anxiety Inventory (STAI) (5) Mother report on child and parent- child in-teraction
- The Parenting Stress Index- Short Form
(PSI-SF)
20 3.2. Participant characteristics
The number of children in the studies spanned from 30 until 170. Overall, there were more male participants than females. Some articles included control groups (Cheung et al., 2019; de Ruiter et al., 2016; Eilertsen et al., 2012; Emond et al., 2016), and one article compared on- treatment pediatric brain tumor survivors(PBTS) with off- treatment PBTS (Hardy et al., year). Most of the studies included both school- age children (6 to 12 years old) and adolescents. One article included cancer survivors until the age of 20 (Eilertsen et al., 2012), even though most of the participants in that study were within the established age range (6-18 years old). Time since treatment ended varied from one study to another (two- seven years). Three articles recounted that mean time since the end of treatment was four years (Barrera et al., 2017; Carpeinteri et al., 2003; Emond et al., 2016). Around 2.6 months (Quast et al., 2018), at least six months (Hardy et al., 2010), 6-24 months (Cheung et al., 2019), more than two years (de Ruiter et al., 2016), six years (Eilertsen et al., 2012) and around seven years (Meeske et al., 2007) off treat-ment were values that were also described. In eight articles parents were included in order to get reports about their children.
3.3. Findings
3.3.1. Problems that paediatric brain tumour survivors report related to their psychoso-cial adjustment
The most often reported concern was their physical well- being (n= 5), accompanied by low athletic competence (n=3) and low- rated physical appearance (n= 1), and followed by per-ceived low social support and integration (n= 5), scarce scores for peer- relations (n= 2), low psychological well- being (n= 4) and poor academic performance (n= 2). Moreover, there were some more manifested concerns, including fatigue (de Ruiter et al., 2016; Cheung et al., 2019; Meeske et al., 2007) headache (Cheung et al., 2019), stress (Cheung et al., 2019), lower self- esteem (Cheung et al., 2019; Eilertsen et al., 2012), more cognitive problems (Hardy et al., 2010), lower IQ (Hardy et al., 2010), concentration problems (de Ruiter et al., 2016), worse global self- worth (Hardy et al., 2010), poor family functioning (Eilertsen et al., 2012; Quast et al., 2018), pain (Meeske et al., 2007), severe late effects (Meeske et al., 2007) and worse HRQOL (de Ruiter et al., 2016; Meeske et al., 2007; Quast et al., 2018).
21 Some articles reported PBTS to be not worse than the normative population or their peers regarding both the social and psychosocial adjustment (Carpeinteri et al., 2013; Emond et al., 2016; Radcliffe et al., 1996). Eilertsen et al. (2012) reported no differences compared to peers for school performance, family functioning, activity and hobbies, physical health, self- esteem and total QOL. However, PBTS reported lower scores for family functioning, friends and social integration and physical health within the aforementioned aspects. Meeske et al. (2007) found that brain tumour survivors PBTS scored lower regarding physical, social and psychosocial functioning. With regard to the report- differences compared with parents, Quast et al. (2018) stressed that PBTS scored worse in family functioning in comparison with parent- reports. More detailed and specific information can be found in table 3.
3.3.2. Problems that parents of paediatric brain tumour survivors report related to their psychosocial adjustment
A total of 8eight articles had reports of parents regarding the psychosocial adjustment of their children after ending brain tumour treatment. The most often reported concern was social ad-justment or integration (n= 3), which included mainly concerns regarding communication skills, competence, interactions and information processing, followed by physical appearance and function (n=3), somatization (n= 3), family functioning (n= 2) and HRQOL (n= 2). Further-more, parents described a wide range of other worries such as aggressive behaviours (Hardy et al., 2010), mental problems (Eilertsen et al., 2012), poor emotional control (de Ruiter et al., 2016), attention problems (Carpeinteri et al., 2003) poor school performance (Eilertsen et al., 2012; Radcliffe et al., 1996), depression (Hardy et al., 2010), poor activity and hobby attend-ance (Eilertsen et al., 2012) and poor working memory (de Ruiter et al., 2016). Most of the included articles demonstrated that parents rated their children to be worse than what children reported themselves to be (n= 5). De Ruiter et al. (2016) recounted that children and parents reported similarly in diverse aspects with the exception of psychosocial adjustment, and Ei-lertsen et al. (2012) stated that parents and PBTS reported similarly on a general basis. More detailed and specific information can be found in table 3.
22 3.3.3. Factors that influence how paediatric brain tumour survivors adjust to the psycho-social aspects of everyday life
Type of treatment was the factor that appeared to have the most impact on the cancer survivor (Barrera et al., 2017; Cheung et al., 2019; Eilertsen et al., 2012; Quast et al., 2018; Radcliffe et al., 1996). Chemotherapy (Barrera et al., 2017; Quast et al., 2018) and radiation therapy (Rad-cliffe et al., 1996) were reported to be the most relevant ones. Furthermore, length of treatment also seemed to be relevant for the future psychosocial adjustment of the paediatric patients (Eilertsen et al., 2012). Different articles reported several other factors that did influence on how children who have ended brain tumour treatment adjust to the psychosocial aspects of everyday life, such as studying in a special education centre (de Ruiter et al., 2016), cultural and ethnical characteristics (Barrera et al., 2017; Cheung et al., 2019; Meeske et al., 2007), available support resources and strategies (Cheung et al., 2019), family income (Barrera et al., 2017), fear of cancer recurrence (Cheung et al., 2019; Eilertsen et al., 2012), personal charac-teristics (Barrera et al., 2017), family interactions (Carpeinteri et al., 2003; Quast et al., 2018), attitudes of denial and defensiveness (Carpeinteri et al., 2003; Eilertsen et al., 2012; Emond et al., 2016), age (Barrera et al., 2017; Emond et al., 2016), time since diagnosis (Hardy et al., 2010), gender (Barrera et al., 2016), how cancer survivors are treated (Eilertsen et al., 2012), lack of insight and willingness to acknowledge the difficulties (Carpeinteri et al., 2003) and the fact of suffering from neurocognitive complaints (de Ruiter et al., 2016; Eilertsen et al., 2012; Emond et al., 2016; Radcliffe et al., 1996).
23
Table 3
Findings of the articles
Study N Self- reported problems Parent- reported problems Factors that influence adjustment
Barrera et al., 2017
91 PBTS Physical scores
Higher scores at 8 months follow- up compared to 2 months follow- up
Emotional scores
Higher scores at 8 months follow- up compared to 2 months follow- up
Physical scores
Higher scores at 8 months follow- up compared to 2 months follow- up
Emotional scores
Higher scores at 8 months follow- up compared to 2 months follow- up
Cranial Irradiation Therapy (CIT)
- Child- report: Poorer QOL than children who didn´t receive this treatment
- Proxy- report: Poorer QOL than children who didn´t receive this treatment Gender
- Child- report: Boys better emotional QOL than girls
Age
- PBTS over 11 years of age better emotional and school QOL than younger PBTS Family income
- Child- report: PBTS form relatively high- in-come families better QOL
Ethnicity
-Child report: PBTS form white families bet-ter QOL
Personal characteristics Executive function Carpeinteri et
al., 2003
32 PBTS Not any greater risk for psychosocial problems than same age peers
Somatization
Excess of score elevation
Attention problems
Excess of score elevation
Leadership scales
Excess of score elevation
Mother- child interaction
Psychologically- based denial or defensive-ness
Lack of insight on the part of the adolescent Lack of willingness to acknowledge difficul-ties on paper- pencil type questionnaire Cheung et al., 2019 77 PBTS 80 survivors of other child-hood cancers Physical domain Fatigue Headache Psychological domain
Low or depressed mood
/ Cultural characteristics
Resources and supporting strategies Fear for cancer recurrence Type of treatment
24 Stress
Lower self- esteem
Social domain
Difficulties in making friends & maintaining relation-ships with others
School domain
Poor academic performance De Ruiter et al., 2016 82 PBTS participants 43 controls 45 PBTS non- participants HRQOL
Worse than normative sample for: - Physical and psychological well- being - Peers
- Social support
Self- esteem
8-11 years:
Higher behavioural conduct scores Lower scholastic and athletic competence 12-18 years:
Higher behavioural conduct scores Lower athletic competence
Psychosocial adjustment
Not worse than normative population
Fatigue
More concentration problems Decreased physical activity
Psychosocial adjustment
-Higher problem score
Executive functioning
- Significantly worse - Worse emotional control
- Worse initiate and working memory
Studying in special education Neurocognitive complaints Eilertsen et al., 2012 50 cancer sur-vivors 29 controls School performance No differences Family functioning No differences
For KINDL results, PBTS lower mean scores
Friends and social integration
Higher mean scores for PBTS
Activity and hobbies
No differences
Physical health
No differences
For KINDL results, PBTS lower mean scores
School performance
Lower scores for PBTS
Family functioning
Lower scores for PBTS
Friends and social integration
Lower scores for cancer survivors in general
Activity and hobbies
Lower scores for PBTS
Physical health
Lower scores for PBTS
Mental health
Lower scores for cancer survivors in general
Somatic and psychiatric side effects - Cancer diagnosis
- Type and length of cancer treatment - Cancer complications
Psychological strain of the cancer illness -Life- threatening disease
- Long term, severe and intensive treatment Long absences from normal social and school activities
How cancer survivors are treated
25
Mental health
Lower scores for PBTS
Emotional well- being Self- esteem
No differences
Psychological problems Total QOL
No differences
Emotional well- being Self- esteem
No differences
Somatic late- effects Psychological problems Total QOL
Lower scores for PBTS Emond et al.,
2016
33 PBTS 34 controls
No significant social adjustment and social interac-tion difficulties
Social adjustment
Risk of social competence difficulties
Poor social awareness and social cognitive skills
Social interaction
Less affiliative interactions
Social information processing
Poor functioning
Cognitive late effects
- Not understand basis for socially competent behaviour
Repressive coping style
- To minimise negative psychosocial effects of cancer experience
Age
-Become more aware of social differences as they become older and face more demanding and complex social situations
Age- appropriate social competence Peer relationships Hardy et al., 2010 15 on- treat-ment 15 off- treat-ment Cognitive scores Lower scores Difference in IQ
Behavioural and emotional functioning Social acceptance Lower scores Perceived competency Physical appearance - Lower scores Athletic competence - Lower scores Global self- worth - Lower scores
- As children´s worry increases, global self- worth decreases
Psychosocial functioning
More aggressive
Behavioural and emotional functioning
As children´s perceived social acceptance decreased - More aggressive
- More depressed - Somatic complaints As parental anxiety increased
- Children´s global self- worth scores and physical appearance increase
Time since diagnosis
- As it increases, children´s perceptions of ath-letic competence decreased
26 Meeske et al., 2007 86 cancer sur-vivors Physical functioning Lower for PBTS Psychosocial functioning Lower for PBTS HRQOL Fatigue Pain
Severe late effects
Social functioning Lower for PBTS / Ethnicity Quast et al., 2018 35 PBTS HRQOL
Worse than normative sample
Family function
Rated worse than mothers
HRQOL
Rated worse than children
Family function
Rated better than children
Chemotherapy treatment Family functioning - Single- parent status
- Lower caregiver educational attainment Radcliffe et al.,
1996
38 PBTS Within normal limits
Significantly less anxious and depressed than their normative counterparts
Less athletically competent
Overall social competence
Lower scores
Higher degree of social problems
Greater difficulties in communication skills More problematic mother- child interactions
School competence
Lower scores
27
4 Discussion
The aim of this systematic review was to report the psychosocial adjustment of children who have ended brain tumour treatmentfrom the perspective of the child and the parents. Findings of the articles in relation to the results and other literature are discussed and followed by clinical implications, methodological issues, limitations and recommenda-tions for future research.
4.1. Reflection on findings
4.1.1. Problems that brain tumour survivors report related to their psychosocial adjustment
Results show that children reported a wide number of concerns regarding their psychoso-cial adjustment, amongst many other worries. Pediatric brain tumour survivors (PBTS) most commonly reported concerns with their physical well- being, low athletic compe-tence, low rated physical appearance, perceived low social support and integration, mea-gre peer- relations, poor psychosocial well- being and poor academic performance. These children also recounted fatigue, headache, stress, lower self- esteem, more cognitive prob-lems, lower IQ, concentration probprob-lems, worse global self- worth, poor family function-ing, pain, severe late effects and worse health related quality of life.
On the whole, results expose that there were no differences in ratings between PBTS and other childhood cancer survivors or healthy population. In addition, it was comprehended that PBTS scored even higher for problems comparing with normative population, finding that was observed previously in existing literature (Blix & Birkeland, 2015). Nevertheless, results of this paper also showed that when comparing this paediatric population with peers and normative population, or even with other childhood cancer survivors, results showed worse ratings for PBTS. Other authors support these findings (Aarsen, Paquier, Reddingius, Streng, Arts, Evera‐Preesman & Catsman‐Berrevoets, 2006; Shah, Dellarole, Peterson, Bregy, Komotar, Harvey & Elhammady, 2015; Zebrack & Chesler, 2002), highlighting the added psychosocial and social difficulties with social functioning these children suffer. Long- term PBTS are at higher risk for poorer psycho-social adjustment taking into account their elevated risk for neurocognitive, psycho-social and
28 behavioural impairments, typically secondary to neurocognitive injuries, findings that Patenaude & Kupst (2005) also discovered.
4.1.2. Problems that that parents of brain tumour survivors report related to their psychosocial adjustment
Parents reported a wide range of concerns regarding the psychosocial adjustment of their children. The most commonly reported were social adjustment or integration, followed by physical appearance and function somatization family functioning and HRQOL. They also reported their child to suffer from aggressive behaviours, mental problems, poor emotional control, attention problems, poor school performance, depression, poor activity and hobby attendance and poor working memory.
When comparing parent and child reports, most of the articles suggest that parents reported their children to have more difficulties than what children reported themselves. In addition, parents recounted their children to be worse off than other childhood cancer survivors. Both healthy children and children with brain tumour described higher levels of psychosocial adjustment than their parents. However, some studies found the opposite (Theunissen, Vogels, Koopman, Verrips, Zwinderman, Verloove-Vanhorick & Wit, 1998). Dissimilarities could be due to influences of different factors (Sato et al., 2013). One of the articles included in this systematic review (Eilertsen et al., 2012) stated that parents and PBTS reported similarly on a general basis.
As parental anxiety increased, it was perceived that children´s global self- worth scores and physical appearance increased. Parents who are anxious about their own social performance tend to have more socially competent children than do parents with optimis-tic feelings about their own social abilities (Putallaz, Klein, Costanzo & Hedges, 1994). A possible explanation for this could be that parents who are more worried about their children´s functioning may also be those who make concrete efforts to meliorate their children´s experiences whenever it´s feasible, to the detriment of their own self- care. However, Mulhern, Fairclough, Smith & Douglas (1992) observed that elevations in ma-ternal depressive symptoms were related to elevated mama-ternal ratings of depressive symp-toms in the child. The illness of the child and the consequent problems might influence parent´s stress and therefore their perception of the child´s situation (Jozefiak, 2004)
29 4.1.3. Factors that influence how paediatric brain tumour survivors adjust to the
psychosocial aspects of everyday life
According to the results of this paper, type of treatment was the factor that appeared to have the most impact on the cancer survivor, being chemotherapy and radiation therapy the most relevant ones, as well as the length of the treatment. Other factors that appeared to have an impact on the psychosocial adjustment of PBTS were cultural and ethnical characteristics, available support resources and strategies, family income, fear of cancer recurrence, personal characteristics, family interactions, studying in special education centres, attitudes of denial and defensiveness, age, time since diagnosis, gender, how can-cer survivors are treated, lack of insight and willingness to acknowledge the difficulties and the fact of suffering from neurocognitive complaints. Time off treatment was also a factor that appeared to have an impact on the PBTS, mainly due to neurocognitive out-comes.
In the study performed by de Ruiter et al. (2016), a large proportion of the sam-ple studied in special education centres, where many of the peers suffered from diverse chronic conditions, which could have affected the psychosocial adjustment of PBTS. Turner, Rey-Casserly, Liptak & Chordas (2009) described that problems of PBTS in-crease once they leave the structured school environment, which signifies that his paedi-atric population may benefit from a structured environment. Contrary to this, another study reported that studying in special education placements was not related to worse adjustment (Radcliffe et al., 1996). It is likely that PBTS received suitable educational evaluations and placements and that they are in appropriate educational settings that promote well- being for both the child and their family.
30 4.2.Methodological issues and limitations
The present study shows several methodological issues and limitations that must be re-ported. Firstly, in order to conduct this systematic review, only four databases (Pubmed, Cinahl, Psycinfo and Sciencedirect) were utilized, which reduced the likelihood of find-ing articles that could have been relevant for the study aim. In addition, only studies in English language were included, there might have been other potential relevant studies published in other languages that were not incorporated in this review. These two meth-odological points might have hampered to obtain a broader range of articles, and thus a more comprehensive and exhaustive picture of the available literature; furthermore, the utilized search strategies may have removed relevant data for the definitive study aim
Secondly, this paper used adapted quality- assessment tools for all the study meth-ods. The adapted tools have never been verified, which gives uncertainty to the internal validity. Moreover, the author decided the content and the scores that determine the qual-ity ranges. These adjustments, as well as the qualqual-ity evaluation itself, were produced and performed by the same person and there is an existing risk for bias.
Peer reviewing would have improved the reliability of the study. The lack of sec-ond reviewer throughout the whole data collection and extraction process imperils the present study’s reliability. Another limitation that ought to be reported is the limited time for the performance of the present systematic review. This may have increased the likeli-hood for bias, some relevant information might be missing, and the study might lack proper revision.
Additionally, the author experienced difficulties defining what aspects psychoso-cial adjustment include. Existing literature examined different aspects of both psycholog-ical and social adjustment and psychosocial adjustment but did not clearly report and clarify these points. Due to this occurrence, the author decided to include diverse aspects that alter and have an impact on the psychosocial adjustment of this paediatric population. This event influenced the selection criteria for the articles, and thus there is an existing chance that useful articles may have been dismissed.
31 4.3. Future research
Bearing in mind the existing difficulties this paediatric population endure throughout the whole cancer experience, future research should provide more longitudinal studies that go from diagnosis through treatment and after treatment. When receiving more knowledge, this could promote early identification and thus early intervention. The im-portance of including caregivers in the whole assessment and intervention process ought to be highlighted, as well as utilizing both quantitative and qualitative methods when performing future research. In addition, larger samples and broader age ranges could make the results more representative.
Some authors reported the need of further research regarding several aspects. Car-peinteri et al., (2003) recounted the need for larger studies to verify if there is a concrete pattern of psychosocial vulnerability related to treatment or tumour variables. Cheung et al. (2019) suggest future studies that examine and develop suitable interventions in rein-forcing their sense of achievement, thereby making PBTS feel competent and able in ex-ecuting certain tasks and hence, helping improve their self- esteem. In addition, social competence screening tools could amend exactitude and reliability of evaluation methods and help recognise target areas of intervention (Emond et al., 2016).
4.4. Clinical implications
The results of this systematic review emphasise the problems PBTS endure in their eve-ryday lives after ending their cancer directed treatment towards brain tumour. There is an existing need for early identification in order to better and swiftly recognise those factors that affect their psychosocial adjustment, plan early interventions and prevent those prob-lems from increasing, and ensure that survivors are referred to the corresponding services. Furthermore, it might be beneficial to plan interventions aimed at meliorating and pro-moting social activities, such as keeping routines as normal as possible (attending school as much as possible, visiting friends) and conversing with peers routinely (Hardy et al., year), as well as interventions aimed at enhancing self- esteem (Cheung et al., 2019)
Some authors reported the importance of bearing in mind both children´s and parents´ perspectives when assessing in order to increase awareness of the psychosocial adjustment PBTS experience (Barrera et al., 2017; de Ruiter et al., 2016; Eilertsen et al.,
32 2012). Besides, the importance of the collaboration of a multidisciplinary team and ex-ternal assistance should also be highlighted. Cheung et al. (2019) suggested that health- care professionals ought to work together with non- governmental organizations, govern-mental policymakers and educational sectors in order to furnish support and assistance to PBTS and their caregivers to eliminate enduring challenges throughout their cancer jour-ney. In addition, PBTS should benefit from supportive interventions concerning rehabil-itation and long- term follow- up (Eilertsen et al., 2012).
5 Conclusion
Paediatric brain tumours are the most usual type of solid childhood cancer, and the second most common paediatric neoplasia after leukaemia. As survival and life expectancy of pediatric brain tumour survivors (PBTS) has improved, rising focus has been given to long- term outcomes and adaptation of this paediatric population. More attention has been given to the medical adjustment of these children, but little is known about their psycho-social adaptation. Even though this systematic literature review had certain limitations, the results showed that both PBTS and their parents reported concerns regarding psycho-social adjustment. There were differences in reports between PBTS and caregivers, show-ing that usually parents rate their child´s difficulties to be worse than as experienced by the children rate themselves. Type of treatment appeared to be the most influential factor that affected the psychosocial adjustment of these paediatric population, being chemo-therapy and radiation chemo-therapy the most relevant ones. In order to help these children im-prove their HRQOL quality of life and thus their psychosocial adjustment, early identifi-cation, early interventions and preventing those problems from increasing remain of great importance, as well as including parents in the whole assessment and intervention pro-cess. This paper identified the need of further research regarding larger studies that go from diagnosis through treatment and after treatment. The importance of providing a mul-tidisciplinary attendance, as well as adapting a holistic approach when assessing and treat-ing PBTS ought to be considered.
33
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