Psychological interventions for parents of children and adolescents with chronic illness.

This is an update of a previously published review published in 2012 investigating the efficacy of psychological therapies for parents of children with a longstanding or life-threatening physical illness. This review updates includes studies that have been conducted in the previous two years to give an up-to-date review of the evidence. Parenting a child with a longstanding or life-threatening illness is very difficult, and can have a negative impact on many aspects of the parents’ life. Parents of these children often have difficulty balancing caring for their child with other responsibilities and demands. As a result, parents may experience more stress, worries, mood disturbance, family arguments, and their children may show troubling or problematic behaviour. Parents also have a major influence on their child’s well-being and adjustment, and play an important role in how their child adapts to living with an illness. Treatments for parents of children with a longstanding illness aim to improve parent distress, parenting behaviours, family conflict, child distress, child disability and the child’s medical symptoms. To evaluate the effectiveness of psychological therapies for parents of children and adolescents with chronic illnesses including painful conditions, cancer, diabetes mellitus, asthma, traumatic brain injury (TBI), inflammatory bowel diseases (IBD), skin diseases, or gynaecological disorders. Psychological therapies will be compared to active, treatment as usual, or wait-list controls. There were two primary outcomes of interest: parent mental health and parenting behaviour. We included five secondary outcomes; child behaviour/disability, child mental health, child symptoms, family functioning, and adverse events. The search was completed in July 2014. Forty-seven studies were found in the search including 3778 participants. The average age of the children was 14.6 years. We found studies that focused on six chronic illnesses (painful conditions, cancer, diabetes, asthma, traumatic brain injury and eczema) and evaluated four types of psychological therapies (cognitive behavioural therapy, family therapy, problem solving therapy, multisystemic therapy). Outcomes were extracted from the time point immediately after the treatment and at the first available follow-up. We analysed the data in two ways: first we grouped the studies by each individual illness (across all therapies) and then we grouped the studies by each individual psychological therapy (across all chronic illnesses). Psychological therapies improved parenting behaviour of parents of children with cancer immediately following treatment. Parent distress also improved for parents of children with cancer. Children with painful conditions and those with symptoms of diabetes showed benefit immediately following treatment, and for diabetes the reduction in symptoms was maintained at follow-up. When analysing different psychological therapies, we found cognitive behavioural therapy can improve the child’s medical symptoms. Problem-solving therapy can improve a parent’s distress and their ability to solve problems, with the reduction in parental distress continuing long-term. Five studies reported that there were no adverse events during the study period. The remaining studies failed to report or discuss adverse events. Risk of bias assessments of included studies were predominantly unclear due to poor reporting. There is evidence that psychological therapies including parent interventions can benefit parents of children with a chronic illness, particularly for parents of children with cancer. However, due to the small number of studies in this review, future studies are likely to change the findings in this review.

We searched for randomised controlled trials (RCTs) of psychological interventions that include a parent component, compared with attention control, other active treatment, or waiting-list control groups. The parent intervention had to be primarily psychological in nature. Studies that met the inclusion criteria consisted of the following: RCT, published in full in a peer-reviewed journal; Primary aim of the trial was an evaluation of a psychological intervention; Involved parents of children who have an illness for three months or more ( Van der Lee 2007 ); Involved parents of children adjusting to a diagnosis of cancer; Had a participant n of 10 or more in both the treatment and control arm at end of treatment or at follow-up. RCT, published in full in a peer-reviewed journal; Primary aim of the trial was an evaluation of a psychological intervention; Involved parents of children who have an illness for three months or more ( Van der Lee 2007 ); Involved parents of children adjusting to a diagnosis of cancer; Had a participant n of 10 or more in both the treatment and control arm at end of treatment or at follow-up. Parents of a child who has endured a chronic illness for three months or more, or who was recently diagnosed with a condition (e.g., cancer) that is expected to last more than three months. We regard parents as the primary caregiver of a child or adolescent under the age of 19 years. We define parents for the purposes of this review as any adult who adopts the responsibility for the role of parenting the child (this could include biological parent, guardian, or other adult family member). There was no lower age limit for the children; however, by the definition of ’chronic illness’, the child must be three months or older. Physical illnesses that were considered for inclusion were: Asthma; Cancer; Diabetes mellitus; Gynaecological disorders (e.g. chronic dysmenorrhoea and endometriosis); Inflammatory bowel diseases (IBD); Painful condition (including but not exclusively limited to arthritis, back pain, complex regional pain syndrome (CRPS), fibromyalgia, headache, idiopathic pain conditions, irritable bowel syndrome (IBS), recurrent abdominal pain); Skin diseases (e.g. eczema); Traumatic brain injury (TBI). Asthma; Cancer; Diabetes mellitus; Gynaecological disorders (e.g. chronic dysmenorrhoea and endometriosis); Inflammatory bowel diseases (IBD); Painful condition (including but not exclusively limited to arthritis, back pain, complex regional pain syndrome (CRPS), fibromyalgia, headache, idiopathic pain conditions, irritable bowel syndrome (IBS), recurrent abdominal pain); Skin diseases (e.g. eczema); Traumatic brain injury (TBI). Chronic illnesses were selected from the National Survey of Children with Special Health Care Needs 2009 to 2010 ( Data Resource Center 2010 ). It was impractical to include all chronic illnesses on this list therefore we selected the most common. However, three illnesses (cancer, inflammatory bowel diseases and gynaecological disorders) were not included in the list of ‘Current Health Conditions and Functional Difficulties’ but were added for the purposes of this review. Cancer has a high incidence level, and in the UK alone 1600 0–14 year-old children are diagnosed with cancer each year ( Cancer Research UK 2014 ). In the USA, it is estimated that 15,780 0–19 year-olds are diagnosed with cancer ( National Cancer Institute 2014 ). Studies that delivered interventions to parents of children who have ’survived’ an illness but still experienced distress, such as childhood survivors of cancer, were also eligible for inclusion. Inflammatory bowel diseases and gynaecological disorders are also common conditions in childhood and adolescence, and are included because they are thought to be prevalent but under-represented in the academic literature. Studies were included if the interventions were primarily psychological, and had credible, recognisable psychological/psychotherapeutic content, and were specifically developed for, or included parents. Psychological interventions were defined as any psychotherapeutic treatment specifically designed to change parent cognition or behaviour, or both, and had the intention of improving parent or child outcomes. However, studies that included parents as ’coaches’ to support exclusively child-focused interventions were excluded from this review. The intervention had to aim to provide treatment to the parent rather than teach them to deliver an intervention to their child. Similarly, we also excluded health promotion therapies such as intervening with the parent to cease smoking to improve their child’s asthma. We have excluded studies that combine psychological interventions with pharmacological interventions or are qualitative in nature, because it is difficult to combine qualitative and quantitative data. Parent outcomes were the primary target of our review. However, if the study also reported child outcomes as stated below, we also analysed and reported these data as secondary outcomes. We analysed data at post-treatment and the first available follow-up period, where reported. Primary outcomes for the purposes of this review include parenting behaviour and parent mental health. Secondary outcomes include child behaviour/disability, child mental health, child illness-related symptoms, family function and adverse events. We made a judgement when studies reported multiple measures within one of the six outcome domains and did not define their primary or secondary outcome measure. The rules of this judgement were to select the most generic, reliable, and most frequently used measure within the field, and most appropriate for the given outcome category. When both parents and children reported on a measure, we extracted the self-reported item unless the non-self-reported measure was a more generic measure. For family functioning measures, we preferentially extracted parent data over child data, as the review is focused on whether interventions can help parents of children with a chronic illness. Two searches have been conducted. The first, from inception to March 2012, and the second from March 2012 to July 2014. We searched four databases for studies for this update. The dates listed below state the most recent date of our search. Cochrane Central Register of Controlled Trials (CENTRAL), the Cochrane Library, Issue 6, 2014; MEDLINE via Ovid, 1946 to 30/6014; EMBASE via Ovid, 1974 to 30/6/14; PsycINFO via Ovid, 1806 to June week 4 2014. Cochrane Central Register of Controlled Trials (CENTRAL), the Cochrane Library, Issue 6, 2014; MEDLINE via Ovid, 1946 to 30/6014; EMBASE via Ovid, 1974 to 30/6/14; PsycINFO via Ovid, 1806 to June week 4 2014. We adapted the search strategies from the MEDLINE search (for all search strategies see Appendix 1 ). There was no language restriction imposed and no unpublished literature or grey material was included, so only the highest quality trials were included. The search strategy included four categories of words: psychological interventions, parents, children/adolescents and chronic illnesses (as stated above), and was refined by a methodological filter used to identify RCTs according to Cochrane guidance ( Higgins 2011 ). We performed a reference list and citation search of all included studies. Relevant meta-analyses and systematic reviews were searched for additional studies. We also contacted authors of selected studies and experts in the field for further studies that had not already been identified from the search. In addition, we searched online trial repositories for additional studies including metaRegister of controlled trials (mRCT) ( www.controlledtrials.com/mrct/ ), ClinicalTrials.gov ( clinicaltrials.gov ), and the World Health Organization International Clinical Trials Registry Platform (ICTRP) ( www.who.int/ictrp/en/ ). Three review authors (EF, EL, JB) sifted through potential studies and identified those eligible to be included, with CE acting as arbiter. No blinding of study authors’ names, institutions or journals occurred during this process. We resolved any disagreements by discussion between all review authors. We made selection of abstracts using the following criteria. Participants ◦ Parents had to be referred to in the title or abstract of each study; ◦ The parent had to be the primary caregiver of the child; ◦ Children had to have one or more of the chronic illnesses listed above; ◦ Children had to be in the age range three months to 19 years; ◦ There had to be 10 or more participants in each condition at the end of the treatment assessment. Intervention ◦ The intervention had to be primarily psychological in at least one treatment arm; ◦ Design was an RCT; ◦ One or more parents had to be treated by the intervention; ◦ The parents or child or both had to complete assessments at baseline and at a point in time during or after the intervention. Comparison groups ◦ Active treatment group; ◦ Treatment-as-usual group (e.g. usual doctors’ appointments and treatment without added psychological therapy); ◦ Waiting-list control. Quantitative outcomes had to be presented We then obtained the selected studies meeting the criteria in full and EF, EL and JB read and assessed them independently. Participants ◦ Parents had to be referred to in the title or abstract of each study; ◦ The parent had to be the primary caregiver of the child; ◦ Children had to have one or more of the chronic illnesses listed above; ◦ Children had to be in the age range three months to 19 years; ◦ There had to be 10 or more participants in each condition at the end of the treatment assessment. Parents had to be referred to in the title or abstract of each study; The parent had to be the primary caregiver of the child; Children had to have one or more of the chronic illnesses listed above; Children had to be in the age range three months to 19 years; There had to be 10 or more participants in each condition at the end of the treatment assessment. Intervention ◦ The intervention had to be primarily psychological in at least one treatment arm; ◦ Design was an RCT; ◦ One or more parents had to be treated by the intervention; ◦ The parents or child or both had to complete assessments at baseline and at a point in time during or after the intervention. The intervention had to be primarily psychological in at least one treatment arm; Design was an RCT; One or more parents had to be treated by the intervention; The parents or child or both had to complete assessments at baseline and at a point in time during or after the intervention. Comparison groups ◦ Active treatment group; ◦ Treatment-as-usual group (e.g. usual doctors’ appointments and treatment without added psychological therapy); ◦ Waiting-list control. Active treatment group; Treatment-as-usual group (e.g. usual doctors’ appointments and treatment without added psychological therapy); Waiting-list control. Quantitative outcomes had to be presented We then obtained the selected studies meeting the criteria in full and EF, EL and JB read and assessed them independently. Three review authors (EF, EL and JB) carried out data extraction from studies that were identified by all review authors as appropriate for inclusion. We extracted demographics of parents and children (e.g. age, sex), characteristics of the child’s illness (e.g. diagnosis, length of illness), and characteristics of therapy (e.g. setting, components, treatment team, therapy type). Finally, we extracted relevant outcomes for analysis. We assessed risk of bias using the recommended Cochrane guidance ( Higgins 2011 ). Of the six suggested ’Risk of bias’ categories, we judged studies on random sequence generation (selection bias), allocation concealment (selection bias), blinding of outcome assessment (detection bias), incomplete outcome data (attrition bias) and selective reporting (reporting bias). We excluded the option of ’blinding participants and personnel’ because we deemed it redundant because personnel cannot be blinded to whether they deliver a treatment. Decisions about random sequence generation were based on whether authors gave a convincing method of randomisation. Participants being stratified by age or sex did not count as biased. Allocation concealment judgements were based on whether sufficient methods were employed for random allocation to take place. We judged risk of blinding of outcome assessment on whether the measures were administered and collected by an assessor who was blind to the treatment allocation. For attrition bias, we assigned a low risk of bias when authors gave both a description of attrition and stated that there were no significant differences between completers and non-completers. We judged there to be unclear risk of bias when there was an adequate description of attrition but authors did not report whether there were significant differences between completers and non-completers. We judged there to be a high risk of attrition bias when no description of attrition was reported. We judged selective reporting bias on whether data were fully reported in the study or if authors later responded to data requests. We assigned a low risk of bias when all the data were reported in the paper, an unclear risk of bias when authors responded to data requests, and a high risk of bias when the data were not reported in the paper and authors did not respond to data requests. Previously we had rated the concordance between the aims, measures, and results of studies; however, we decided not to conduct this assessment for this update and deleted previous ratings. The original version of this review included quality assessments advocated by Yates 2005 . However, for this update we used the GRADE assessment of quality in accordance with Cochrane guidance ( Higgins 2011 ). Quality of evidence using GRADE was assessed in order to determine the quality of evidence and to enable a summary of the level of confidence in the estimate of effect. First, GRADE assessments for parent outcomes combining all psychological therapies are presented. Second, GRADE assessments for all outcomes for each medical condition are presented. There were five categories that were assessed to obtain a GRADE rating for an outcome: limitations in the design, indirectness of evidence, unexplained heterogeneity, imprecision of results, and probability of publication bias. Quality ratings could be downgraded from high to either moderate, low, or very low quality evidence. For limitations in the design, the category was downgraded once if the majority of risk of bias ratings from included studies in an outcome were rated as ’unsure’ or ’high’ risk of bias. It was downgraded twice if there were a high proportion of high risk ratings. For indirectness of evidence, if 50% or more of studies had a waiting-list control the outcome was downgraded once, however if 75% or more had a waiting-list control, the outcome was downgraded twice. The inconsistency of results was downgraded once when the heterogeneity of the analysis was more than 45% and downgraded twice when the heterogeneity was 75% or more. For imprecision of results, we downgraded the outcome if the included studies had fewer than 500 participants. Outcomes were downgraded twice if there were fewer than 150 participants contributing to an outcome. Last, for publication bias, we downgraded outcomes where 50% or more of the contributing studies had a high risk of bias rating for publication bias. We investigated four classes of psychological therapies: cognitive behavioural therapy (CBT), family therapy (FT), problem-solving therapy (PST) and multisystemic therapy (MST). CBT is based on theories of behavioural analysis (Bergin1975), cognitive theory ( Beck 1979 ) and social learning theory ( Bandura 1977 ). CBT therefore includes a range of strategies with the goals of modifying social/environmental and behavioural factors that may exacerbate or cause symptoms, and modifying maladaptive thoughts, feelings and behaviours to reduce symptoms and prevent relapse. FT is based on family systems theory ( Haley 1976 ; Minuchin 1974 ), which emphasises the role of the family context in an individual’s emotional functioning. FT interventions typically focus on altering patterns of interactions between family members, and include structural family therapy ( Minuchin 1974 ), strategic family therapy ( Haley 1976 ) and behavioural systems family therapy ( Robin 1989 ). PST is based on the D’Zurilla 1982 social problem-solving model, which defines problem solving in terms of an individual’s ability to recognise problems and use a positive orientation and problem-solving skills to solve them. PST includes didactic instruction in problem-solving skills, followed by in-session modelling, behavioural rehearsal and performance feedback, as well as homework assignments ( D’Zurilla 2007 ). Finally, MST is an intensive family- and community-based intervention based on the Bronfenbrenner 1979 social ecological model and family systems theory ( Haley 1976 ; Minuchin 1974 ). MST therefore targets the child, their family and broader systems such as the child’s school, work or medical team as needed. MST incorporates a wide range of evidence-based intervention techniques based on the individual needs of the child and family ( Henggeler 2003 ), including cognitive-behaviour approaches, parent training and family therapies. We extracted data immediately post-treatment (i.e. immediately after the treatment programme had finished). Where data were available, we also analysed studies at follow-up, which is classed as the first available time point after post-treatment. We categorised outcomes into one of six outcome domains: parenting behaviour, parent mental health, child behaviour/disability, child mental health, child symptoms and family functioning. Where studies had more than one comparator group, we chose the ‘active control group’ over ‘standard treatment’ or ‘wait-list control’ groups. There are four therapies (CBT, FT, PST and MST), eight conditions (asthma, cancer, diabetes mellitus, gynaecological disorders, inflammatory bowel diseases, painful conditions, skin diseases, and traumatic brain injury), two time points (post-treatment and follow-up) and six possible outcomes (parenting behaviour, parent mental health, child behaviour/disability, child mental health, child symptoms and family functioning). There are six categories by which we analysed data: For each condition, across all types of psychological therapy, what is the efficacy for the six outcomes immediately post-treatment? For each condition, across all types of psychological therapy, what is the efficacy for the six outcomes at follow-up? For each psychological therapy, across all conditions, what is the efficacy for the six outcomes immediately post-treatment? For each psychological therapy, across all conditions, what is the efficacy for the six outcomes at follow-up? The interaction between the condition and the psychological therapy efficacy. Investigation of characteristics of particularly effective treatments. For each condition, across all types of psychological therapy, what is the efficacy for the six outcomes immediately post-treatment? For each condition, across all types of psychological therapy, what is the efficacy for the six outcomes at follow-up? For each psychological therapy, across all conditions, what is the efficacy for the six outcomes immediately post-treatment? For each psychological therapy, across all conditions, what is the efficacy for the six outcomes at follow-up? The interaction between the condition and the psychological therapy efficacy. Investigation of characteristics of particularly effective treatments. Analyses are presented for each of the six outcomes, however, due to the heterogeneous nature of the conditions and studies, this was not always possible. We pooled data using the standardised mean difference (SMD) and a random-effect model, as studies did not consistently use the same scales when measuring the same outcomes. Cohen’s d effect sizes can be interpreted as follows: 0.2 = small, 0.5 = medium, 0.8 = large ( Cohen 1992 ). We contacted authors of studies when data were not reported fully in publications. However, when authors could not send data to the review authors or were non-responsive to emails, we excluded data. When there were multi-arm trials or trials that compared more than one active treatment, we used the primary active treatment and compared with the least biased comparator (active control). Analyses of the following subgroups are presented where data permitted: Parent-only interventions versus family-based interventions; Intervention effects within specific illnesses; Intervention effects across specific types of psychological interventions. Parent-only interventions versus family-based interventions; Intervention effects within specific illnesses; Intervention effects across specific types of psychological interventions. We also explored heterogeneity through subgroup analysis.

See: Characteristics of included studies and Characteristics of excluded studies for a detailed description of included and excluded studies. The first search which was conducted from inception to March 2012 identified 35 studies for inclusion. For results of the initial search see Appendix 2 . The updated search identified studies from March 2012 to July 2014. Four hundred and eighteen abstracts were identified in the database search and read for inclusion, of these 376 were excluded. See Figure 1 for a flowchart of studies. Sixteen papers were identified in the updated search which met the inclusion criteria, three of which ( Levy 2010 ; Stark 2005 ; Wade 2011 ) were identified as follow-up papers of already included trials. Therefore, 13 new trials are included in this update, adding to the 35 previously included studies. One previously included study was excluded for the purposes of this review ( Grey 2011 ), as it combined data with another study already included in this review and would inflate the results if included. Therefore, in total there are 60 included papers and 47 included studies ( Allen 1998 ; Ambrosino 2008 ; Antonini 2014 ; Askins 2009 ; Barakat 2010 ; Barry 1997 ; Celano 2012 ; Connelly 2006 ; Duarte 2006 ; Ellis 2004 ; Ellis 2005 ; Ellis 2012 ; Gulewitsch 2013 ; Hicks 2006 ; Hoekstra-Weebers 1998 ; Kashikar-Zuck 2005 ; Kashikar-Zuck 2012 ; Kazak 2004 ; Laffel 2003 ; Lask 1979 ; Lehmkuhl 2010 ; Levy 2010 ; Marsland 2013 ; Mullins 2012 ; Naar-King 2014 ; Nansel 2009 ; Nansel 2012 ; Ng 2008 ; Niebel 2000 ; Olivares 1997 ; Palermo 2009 ; Robins 2005 ; Sahler 2002 ; Sahler 2005 ; Sahler 2013 ; Sanders 1994 ; Saßman 2012 ; Seid 2010 ; Shekarabi-Ahari 2012 ; Stark 2005 ; Stehl 2009 ; Tsiouli 2014 ; Wade 2006a ; Wade 2006b ; Wade 2011 ; Wysocki 1999 ; Wysocki 2006 ). There were previously 35 studies (45 papers) included in this review. Grey 2011 was previously included in the review but is now excluded for the purposes of this update as the manuscript replicates data already included from another included trial. The updated search identified an additional 13 studies ( Antonini 2014 ; Ellis 2012 ; Gulewitsch 2013 ; Marsland 2013 ; Mullins 2012 ; Naar-King 2014 ; Nansel 2009 ; Nansel 2012 ; Sahler 2013 ; Saßman 2012 ; Shekarabi-Ahari 2012 ; Stark 2005 ; Tsiouli 2014 ) and three follow-up papers of studies already included ( Levy 2010 ; Stark 2005 ; Wade 2011 ), resulting in a total of 47 studies (60 papers). Of the 47 studies included in this review, 43 had two comparator arms and four studies had three comparator arms ( Niebel 2000 ; Seid 2010 ; Wysocki 1999 ; Wysocki 2006 ). Of the 43 studies that had two arms, 16 studies used active controls (e.g. education), 17 studies used ’treatment-as-usual controls’ and 10 studies used wait-list controls. The total number of participants at the end of treatment was 3778 (mean = 80 per study). The total number of participants entering treatment was 4607 (mean = 98 per study). Therefore, the completion rate for all studies was 82%, making the attrition percentage 18%. The proportion of completers across studies ranged from 55% to 100%. The age of children receiving treatment could be extracted from 30 studies (mean = 14.6, standard deviation (SD) = 2.71). We categorised the studies by the primary illness of the children. There were 14 studies of children with painful conditions ( Allen 1998 ; Barakat 2010 ; Barry 1997 ; Connelly 2006 ; Duarte 2006 ; Gulewitsch 2013 ; Hicks 2006 ; Kashikar-Zuck 2005 ; Kashikar-Zuck 2012 ; Levy 2010 ; Palermo 2009 ; Robins 2005 ; Sanders 1994 ; Stark 2005 ). Ten studies with the primary illness of cancer met the inclusion criteria ( Askins 2009 ; Hoekstra-Weebers 1998 ; Kazak 2004 ; Marsland 2013 ; Mullins 2012 ; Sahler 2002 ; Sahler 2005 ; Sahler 2013 ; Shekarabi-Ahari 2012 ; Stehl 2009 ), 13 studies investigated children with diabetes ( Ambrosino 2008 ; Ellis 2004 ; Ellis 2005 ; Ellis 2012 ; Laffel 2003 ; Lehmkuhl 2010 ; Nansel 2009 ; Nansel 2012 ; Olivares 1997 ; Saßman 2012 ; Tsiouli 2014 ; Wysocki 1999 ; Wysocki 2006 ), five investigated asthma ( Celano 2012 ; Lask 1979 ; Naar-King 2014 ; Ng 2008 ; Seid 2010 ), four studies treated children with traumatic brain injury ( Antonini 2014 ; Wade 2006a ; Wade 2006b ; Wade 2011 ) and one study included children with atopic eczema ( Niebel 2000 ). No studies met the inclusion criteria for gynaecological disorders or inflammatory bowel diseases. Similarly, we also categorised studies by the type of psychological therapy delivered. There were 22 studies that delivered cognitive behavioural therapy (CBT) ( Allen 1998 ; Ambrosino 2008 ; Barakat 2010 ; Barry 1997 ; Connelly 2006 ; Duarte 2006 ; Gulewitsch 2013 ; Hicks 2006 ; Hoekstra-Weebers 1998 ; Kashikar-Zuck 2005 ; Kashikar-Zuck 2012 ; Laffel 2003 ; Levy 2010 ; Marsland 2013 ; Niebel 2000 ; Olivares 1997 ; Palermo 2009 ; Robins 2005 ; Sanders 1994 ; Shekarabi-Ahari 2012 ; Stehl 2009 ; Tsiouli 2014 ), eight studies delivered family therapy (FT) ( Celano 2012 ; Kazak 2004 ; Lask 1979 ; Lehmkuhl 2010 ; Ng 2008 ; Saßman 2012 ; Wysocki 1999 ; Wysocki 2006 ), 12 studies delivered problem-solving therapy (PST) ( Antonini 2014 , Askins 2009 ; Mullins 2012 ; Nansel 2009 ; Nansel 2012 ; Sahler 2002 ; Sahler 2005 ; Sahler 2013 ; Seid 2010 ; Wade 2006a ; Wade 2006b ; Wade 2011 ), four studies delivered multisystemic therapy (MST) ( Ellis 2004 ; Ellis 2005 ; Ellis 2012 ; Naar-King 2014 ) and one study delivered an exclusively behavioural intervention ( Stark 2005 ). The proportion of therapy received by parent and child varied between studies. The majority of studies gave equal attention to both parent and child (29 studies). In 12 studies only the parent received therapy, eight of which were delivered treatment to parents whose children had been diagnosed with cancer. Four further studies spent the majority of treatment time with the child. The final two studies did not specify how much therapy the parent and child respectively received (see Table 1 ). Forty studies treated participants in-person with the therapist, six studies used online programmes to deliver part or all of the therapy to participants and one study used telephone calls. Of the 47 studies, 36 studies carried out therapy with individuals or with individual families, whilst 10 studies carried out therapy in groups, and one study did not specify how treatment was carried out. A summary of the characteristics of therapy and a narrative summary of treatment content are presented in Table 1 and Table 2 respectively. We were unable to extract quantitative data from 10 of the 47 studies ( Barry 1997 ; Celano 2012 ; Duarte 2006 ; Kazak 2004 ; Lask 1979 ; Lehmkuhl 2010 ; Nansel 2012 ; Olivares 1997 ; Robins 2005 ; Stark 2005 ). These studies did not present means or standard deviations. Stark 2005 delivered behavioural therapy for children with juvenile rheumatoid arthritis and provided outcome data on calcium intake. This was heterogeneous with other outcomes we extracted for this condition and therapy and therefore was not appropriate to include in the meta-analysis. Therefore 37 studies (2984 participants post-treatment) presented data that were included in at least one analysis. Eleven studies that were excluded in the update of this review ( Bellin 2013 ; Borhani 2011 ; Chen 2013 ; Fedele 2013 ; Groß 2013 ; Gulewitsch 2012 ; Katz 2014 ; Kurowski 2013 ; Lyon 2013 ; Murphy 2012 ; Van der Veek 2013 ). One previously included study Grey 2011 was excluded in this update as it combined two sets of trial data, one of which is already included independently ( Grey 2011 ). Therefore a total of 73 studies were excluded, as they did not meet the inclusion criteria for this review. Thirty-six studies had insufficient psychotherapeutic content, such as instruction, education, parents trained as ’coaches’ for their children or health prevention interventions ( Aleman 1992 ; Anderson 1999 ; Braga 2005 ; Bellin 2013 ; Burke 1997 ; Burke 2001 ; Chen 2013 ; Chernoff 2002 ; Chiang 2009 ; Evans 1999 ; Field 1998 ; Garbutt 2010 ; Giallo 2008 ; Glang 2007 ; Groß 2013 , Hovell 1994 ; Humphreys 2000 ; Ireys 1996 ; Ireys 2001 ; Johnson 1987 ; Kaslow 2000 ; Katz 2014 ; Kazak 1996 ; Ketchen 2006 ; Klinnert 2005 ; Klinnert 2007 ; Kupfer 2010 ; Logan 1997 ; Mendez 1997 ; Murphy 2012 ; Nelson 2011 ; Pérez 1999 ; Staab 2002 ; Sullivan-Bolyai 2010 ; Szczepanski 2010 ; Walders 2006 ). Twenty-two studies had an aim that was irrelevant to the objectives of this review, such as fidelity studies, mixed illnesses or the intervention focusing on the parents’ communication with professionals ( Borhani 2011 ; Bruzzese 2008 ; Cakan 2007 ; Canino 2008 ; Carey 2008 ; Ellis 2007 ; Ellis 2008 ; Fedele 2013 ; Forsander 1995 ; Gerber 2010 ; Gulewitsch 2012 ; Harris 2001 ; Hommel 2012 ; Jay 1990 ; Kurowski 2013 ; Lyon 2013 ; Rasoli 2008 ; Scholten 2011 ; Van der Veek 2013 ; Wade 2010 ; Walker 1996 ; Wysocki 1997 ). Thirteen studies had an insufficient number of participants (n < 10) post-treatment in one or more arms of treatment ( Forsander 2003 ; Gustafsson 1986 ; Haus 1976 ; Hernandez 1998 ; Kamps 2008 ; Kazak 2005 ; Kroner-Herwig 1998 ; Lasecki 2008 ; Sanders 1989 ; Sanders 1996 ; Satin 1989 ; Sieberg 2011 ; Warner 2011 ), one paper recruited participants prospectively ( Betancourt 2004 ), and as mentioned above, one combined two sets of trial data ( Grey 2011 ). These judgements were often difficult to make and led to extended discussion between review authors. We used five ’Risk of bias’ categories: random sequence generation (selection bias), allocation concealment (selection bias), blinding of outcome assessment (detection bias), incomplete outcome data (attrition bias) and selective reporting (reporting bias) ( Figure 2 ; Figure 3 ). Twenty-three studies described a convincing method of randomisation and were judged as low risk of bias, a further 24 studies did not provide an adequate description and were judged as unclear. No studies were rated as high risk of bias for randomisation. There were 15 studies that described a convincing method of allocation and we judged them to have a low risk of allocation bias, a further 32 studies did not provide an adequate description and were judged as unclear. No studies were rated as high risk of allocation bias. For detection bias, 20 studies reported outcome assessors that were blinded to treatment allocation and were judged to have a low risk of bias, a further 27 studies did not provide an adequate description and were judged as unclear. No studies were assessed as having a high risk of outcome bias. For attrition bias, 15 studies reported attrition and found no significant differences between completers and non-completers, so we judged them to have a low risk of bias. Twenty-one studies reported attrition but did not report differences between completers and non-completers and so they were judged as unclear and 11 studies did not give an adequate description of attrition and so we judged them to be of high risk. For selective reporting bias, data could be fully extracted from the published paper in 18 studies and were judged to have a low risk of selective reporting bias. A further 14 studies were unclear, meaning data could not be extracted from the published manuscript but authors responded to data requests. We found 15 studies to have a high risk of selective reporting bias because data could not be extracted and we received no response from our requests for data. See: Summary of findings for the main comparison Psychological therapies for parent behaviour post-treatment; Summary of findings 2 Psychological therapies for parent mental health post-treatment. We analysed data in two categories. In the first, outcomes for each individual condition across all psychological therapies are analysed post-treatment and follow-up. For the second, outcomes for each psychological therapy across all conditions post-treatment and at follow-up are presented. No analyses could be conducted for gynaecological disorders due to lack of studies meeting the inclusion criteria, and no adverse events were reported in any study reviewed. Two studies of children with chronic pain (n = 92) were entered into an analysis of parent adaptive behaviour post-treatment. The analysis revealed that when all psychological therapies were combined, there was no effect for parent adaptive behaviour (Z = 0.80, p = 0.43; Analysis 1.1 ). Only one study could be included at follow-up, therefore no conclusions can be drawn. Seven studies of children with chronic pain (n = 457) were entered into an analysis investigating the effect of psychological therapies for reducing child behaviour/disability post-treatment, and three studies could be included at follow-up (n = 289). However, analyses revealed that therapies were not beneficial at reducing disability in children with chronic pain post-treatment (Z = 1.85, p = 0.06; Analysis 1.2 ), or at follow-up (Z = 0.95, p = 0.34; Analysis 2.1 ). We entered four studies of children with chronic pain post-treatment (n = 356) and two studies at follow-up (n = 255) into an analysis of child mental health. Combined psychological therapies did not have an effect on child mental health post-treatment (Z = 0.14, p = 0.89; Analysis 1.3 ) or at follow-up (Z = 0.14, p = 0.89; Analysis 2.2 ). Nine studies of children with chronic pain post-treatment (n = 540), and six studies at follow-up (n = 391) were entered into an analysis investigating the effects of psychological interventions at reducing child pain-related symptoms. A small beneficial effect of psychological treatments was found post-treatment (SMD = −0.39, 95% confidence interval (CI) −0.67 to −0.11; Z = 2.69, p < .01; Analysis 1.4 ; Figure 4 ). However, at follow-up no effect was found suggesting that a reduction in pain was not maintained (Z = 1.57, p = 0.12; Analysis 2.3 ). There was only one study of children with chronic pain that could be entered into an analysis of family functioning post-treatment and at follow-up, therefore no conclusion could be drawn. No studies presented extractable data on parent mental health post-treatment or at follow-up. Five studies of parents of children with cancer were entered into an analysis of parent behaviour post-treatment (n = 836) and five studies at follow-up (n = 789) to investigate the efficacy of psychological therapies. Psychological therapies had a small beneficial effect for parenting behaviour post-treatment (SMD = −0.20, 95% CI −0.36 to −0.04, Z = 2.44, p = 0.01; Analysis 3.1 ; Figure 5 ). However, this effect was not maintained at follow-up (Z = 1.39, p = 0.16; Analysis 4.1 ). Nine studies of parents of children with cancer were entered into an analysis of parent mental health post-treatment (n = 1010) and six studies (n = 819) were included at follow-up. There was no effect of psychological therapies on parent mental health post-treatment (Z = 1.86, p = 0.06; Analysis 3.2 ). However, at follow-up psychological therapies had a small beneficial effect for improving parent mental health (SMD = −0.18, 95% CI −0.32 to −0.04, Z = 2.58, p = 0.01; Analysis 4.2 ; Figure 6 ). There was only one study of children with cancer that we could be entered into an analysis of child symptoms post-treatment, and no studies were available at follow-up, therefore no conclusions can be drawn. No studies presented extractable data on child behaviour/disability, child mental health and family functioning post-treatment or at follow-up. There was only one study of children with diabetes that could be entered into an analysis of parenting behaviour post-treatment, therefore no conclusions could be drawn. Three studies of children with diabetes (n = 175) were entered into an analysis investigating the effect of psychological interventions for reducing parent mental health post-treatment. There was no beneficial effect found for psychological therapies post-treatment (Z = 1.43, p = 0.15; Analysis 5.1 ). Only one study could be entered at follow-up, therefore no conclusion could be drawn. Three studies of children with diabetes (n = 329) were entered into an analysis of child behaviour post-treatment, however, no effect was found (Z = 0.58, p = 0.56; Analysis 5.2 ). Only one study was available at followup therefore, no conclusions can be drawn. Two studies of children with diabetes (n = 198) were entered into an analysis of child mental health post-treatment, however, no effect of psychological therapies was found (Z = 0.28, p = 0.78; Analysis 5.3 ). Nine studies of children with diabetes post-treatment (n = 774), and four studies at follow-up (n = 385) were entered into an analysis of child diabetes-related symptoms. Psychological therapies were found to have a small beneficial effect at reducing child symptoms post-treatment (SMD = −0.19, 95% CI −0.37 to −0.01, Z = 2.12, p = 0.03; Analysis 5.4 ) and this effect was maintained at followup (SMD = −0.25, 95% CI to −0.45 to −0.05, Z = 2.41, p = 0.02, Analysis 6.1 ). We entered five studies of children with diabetes into an analysis of family functioning post-treatment (n = 422) however, no effect was found (Z = 0.12, p = 0.90; Analysis 5.5 ). No studies presented extractable data on parenting behaviour, child mental health and family functioning at follow-up. There was only one study of children with asthma that could be entered into analyses on parenting behaviour post-treatment, therefore no conclusions could be drawn. Two studies of children with asthma could be entered into an analysis of parent mental health post-treatment (n = 74). Psychological therapies were not beneficial for parent mental health (Z = 0.86, p = 0.39; Analysis 7.1 ). Similarly, two studies of children with asthma could be entered into an analysis of child behaviour/disability post-treatment (n = 200), however no effect was found (Z = 1.48, p = 0.14; Analysis 7.2 ). Four studies of children with asthma were entered into an analysis of child asthma-related symptoms post-treatment (n = 337) and two studies were entered at follow-up (n = 132). The overall effect of all psychological therapies on child symptoms was not beneficial post-treatment (Z = 1.53, p = 0.13; Analysis 7.3 ) or at follow-up (Z = 0.55, p = 0.58; Analysis 8.1 ). No studies presented extractable data on child mental health and family functioning post-treatment or at follow-up, or parenting behaviour, parent mental health and child behaviour/disability at follow-up. There was only one study of children with traumatic brain injury that could be entered into analyses on parenting behaviour post-treatment and none at follow-up, therefore no conclusions could be drawn. We entered two studies of children with traumatic brain injury into an analysis of parent mental health post-treatment (n = 72) but no effect of psychological therapies was found (Z = 1.49, p = 0.14; Analysis 9.1 ). Four studies of children with traumatic brain injury were entered into an analysis of child behaviour/disability post-treatment (n = 144), however, psychological therapies were not found to have an effect (Z = 1.40, p = 0.16; Analysis 9.2 ). Two studies of children with traumatic brain injury were entered into an analysis of family functioning post-treatment (n = 67), however, no effect was determined (Z = 0.33, p = 0.74; Analysis 9.3 ). No studies presented extractable data on child mental health or child symptoms post-treatment and no data were available for any outcomes at follow-up. There was only one study of children with skin diseases that could be entered into an analysis of parenting behaviour, parent mental health, child behaviour and child symptoms post-treatment, therefore no conclusions could be drawn. No studies presented extractable data on child mental health or family functioning post-treatment or any outcomes at follow-up. Four studies post-treatment (n = 166) and two studies at followup (n = 85) were entered into an analysis to investigate the effects of cognitive behavioural therapy (CBT) across all conditions on parenting behaviour. The overall effect of CBT on parenting behaviour was not beneficial post-treatment (Z = 0.08, p = 0.94; Analysis 10.1 ) or at follow-up (Z = 0.56, p = 0.58; Analysis 11.1 ). Seven studies post treatment (n = 325) and two studies at followup (n = 115) presented data on parent mental health, however, no effect of CBT for parent mental health could be determined post-treatment (Z = 0.66, p = 0.51; Analysis 10.2 ) or at follow-up (Z = 1.26, p = 0.21; Analysis 11.2 ). Eight studies post-treatment (n= 487) and three studies at follow-up (n=289) were entered into an analysis to investigate the effects of CBT on child behaviour/disability. No effect could be determined post-treatment (Z = 1.34, p = 0.18; Analysis 10.3 ) or at follow-up (Z = 0.95, p = 0.34; Analysis 11.3 ). Five studies post-treatment (n = 439) and two studies at follow-up (n = 257) were entered into an analysis to investigate the effects of CBT on child mental health, however, there was no effect of CBT post-treatment (Z = 0.21, p = 0.83; Analysis 10.4 ) or at follow-up (Z = 0.27, p = 0.78; Analysis 11.4 ). We entered 12 studies post-treatment (n = 754) and seven studies at follow-up (n = 472) into an analysis on child symptoms. The overall effect of CBT on child symptoms post-treatment was beneficial (SMD= −0.32, 95% CI −0.53 to −0.11, Z = 2.98, p < .01; Analysis 10.5 ). However this effect was not maintained at follow-up (Z = 1.70, p = 0.09; Analysis 11.5 ). We entered three studies post-treatment (n = 211) and two studies at follow-up (n = 107) into an analysis of the effects on family functioning. No beneficial effect of CBT for family functioning was found post-treatment (Z = 0.40, p = 0.69; Analysis 10.6 ) or at follow-up (Z = 0.61, p = 0.54; Analysis 11.6 ). Only one study could be entered into an analysis on the efficacy of family therapy (FT) across all conditions on parenting behaviour post treatment, therefore no analysis was conducted. Three studies (n = 131) were entered into an analysis of the effects of FT on parent mental health post-treatment, however, no effect was found (Z = 0.16, p = 0.88; Analysis 12.1 ). Only one study could be entered at follow-up, therefore no conclusions could be drawn. Two studies post-treatment (n = 107) were entered into an analysis on child behaviour/disability. The overall effect of FT was not beneficial for children with a chronic condition (Z = 1.44, p = 0.15; Analysis 12.2 ). We entered five studies post-treatment (n = 259) and two studies at follow-up (n = 96) into an analysis investigating the effects of FT on child symptoms. No beneficial effect was found for FT post-treatment (Z = 0.35, p = 0.73; ( Analysis 12.3 ) or at follow-up (Z = 0.12, p = 0.91; Analysis 13.1 ). We entered two studies (n = 132) into an analysis on family functioning, however, no effect was found (Z = 0.45, p = 0.65; Analysis 12.4 ). No studies presented extractable data on child mental health post-treatment or at follow-up. Furthermore, no data were available for parenting behaviour, child behaviour/disability and family functioning at follow-up. We entered five studies post-treatment (n= 832) and four studies at follow-up (n = 748) into an analysis investigating the effects of problem solving therapy (PST) across all conditions on parenting behaviour. PST had a small beneficial effect on parenting behaviour post-treatment (SMD = −0.25, 95% CI −0.39 to −0.11, Z = 3.59, p < .01; Analysis 14.1 ), however this was not maintained at follow-up (Z = 1.75, p = 0.08; Analysis 15.1 ). Seven studies post-treatment (n = 907) and five studies at follow-up (n = 778) were entered into an analysis on parent mental health. There was a small beneficial effect of PST on parent mental health post-treatment (SMD = −0.24, 95% CI −0.42 to −0.05, Z= 2.50, p = 0.01; Analysis 14.2 ). This beneficial effect was maintained at follow-up (SMD = −0.19, 95% CI −0.34 to −0.04, Z = 2.55, p = 0.01; Analysis 15.2 ). We entered five studies post-treatment (n=260) into an analysis of the effects of PST on child behaviour/disability, however no effect was found (Z = 1.21, p = 0.22; Analysis 14.3 ). There was only one study that could be entered at follow-up, therefore no conclusions could be drawn. We entered two studies post-treatment (n = 216) into an analysis on child symptoms. There was no beneficial effect of PST post-treatment (Z = 1.41, p = 0.16; Analysis 14.4 ). There was only one study that could be entered at follow-up, therefore no conclusions could be drawn. We entered three studies post-treatment (n = 183) into an analysis of the effects of PST on family functioning, however no effect was found (Z = 0.54, p = 0.59; Analysis 14.5 ). No studies presented extractable data on family functioning at follow-up, or child mental health post-treatment or at follow-up. We entered two studies post-treatment (n = 313) into an analysis investigating the effects of multisystemic therapy (MST) on child behaviour/disability. No effect was found at reducing child behaviour/disability (Z = 0.99, p = 0.32; Analysis 16.1 ). There was only one study at follow-up that could be entered into an analysis, therefore no conclusions could be drawn. Only one study post-treatment that could be entered into an analysis on the effects of MST across all conditions on child mental health; therefore no conclusions could be drawn. We entered four studies post-treatment (n = 455) and two studies at follow-up (n = 247) into an analysis of the effects of MST on child symptoms. There was no beneficial effect of MST on child symptoms post-treatment (Z = 1.52, p = 0.13; Analysis 16.2 ) or at follow-up (Z = 1.47, p = 0.14, Analysis 17.1 ). No studies presented extractable data on parenting behaviour, parent mental health and family functioning post-treatment and at follow-up. Furthermore, no studies presented extractable data on child mental health at follow-up.

There is little evidence available to guide parents as to the most effective intervention expected to produce changes in their own mental health or behavioural functioning. Limited data are available. Problem-solving therapy (PST) produced change in parenting behaviour and mental health, in part by reducing parenting stress. However, there are positive findings to suggest that cognitive behavioural therapy (CBT) that include parents are beneficial for reducing children’s primary symptoms such as pain. The only therapy that was expressly developed to reduce parent distress and was delivered to parents of children with a chronic illness was PST, which we found led to improvements in parent mental health and parenting behaviour. These trials were predominantly delivered to parents of children with cancer, but have also been trialled in parents of children with diabetes. Although CBT, family therapy (FT), and multisystemic therapy (MST) also treated parents, no beneficial effect of therapy was found, although CBT did reduce child symptoms post-treatment. These therapies include parents but remain focused on the child as the agent of behaviour change, and do not purposefully target strategies to improve parent mental health or behaviour. It is surprising how few trials have targeted parent behaviour or mental health, given the longstanding interest of psychologists in understanding the relationship between child and parent adjustment to chronic illness. When combining all therapies for parenting outcomes, we concluded that the quality of evidence was low to very low, meaning further research is very likely to change the estimates of effects. This is primarily due to the low number of studies that reported parenting outcomes. Thus, little guidance is currently available to understand the most effective interventions to implement with parents of youth with chronic health conditions. Since the first version of this review, there have been 13 new studies. However, many of the suggestions we made in 2012 still stand ( Eccleston 2012b ). There are relatively few studies of psychological interventions that target parents of children with a chronic illness and further work is needed to develop and evaluate additional parent-focused interventions that aim to alter parent mental health or parenting behaviour. For example, there were no studies of children with gynaecological disorders or inflammatory bowel diseases that met the inclusion criteria, and only one with children with skin diseases, meaning we were not able to conduct any meta-analyses for these conditions. The next generation of trials should improve by taking account of the limitations identified in this review, including: Larger sample sizes; Following CONSORT guidelines ( Schulz 2010 ); The clearer identification of primary outcomes; Designing treatment content to specifically target change in the primary outcomes; More consistency of measurement and greater consensus within the field around appropriate measures to use within and across illness groups; Placing treatment manuals and data in a shared database to facilitate replication of intervention trials and re-analysis of results; Measuring and reporting parent-specific outcomes; Inclusion of fathers as trial and intervention participants; Reporting of adverse events. Larger sample sizes; Following CONSORT guidelines ( Schulz 2010 ); The clearer identification of primary outcomes; Designing treatment content to specifically target change in the primary outcomes; More consistency of measurement and greater consensus within the field around appropriate measures to use within and across illness groups; Placing treatment manuals and data in a shared database to facilitate replication of intervention trials and re-analysis of results; Measuring and reporting parent-specific outcomes; Inclusion of fathers as trial and intervention participants; Reporting of adverse events. This review has also highlighted several future directions for research that examines interventions targeting parents of children with chronic illness. Problem-solving therapy (PST) improved parent mental health and parenting behaviour. Previously, we suggested that studies were needed to evaluate PST in populations other than cancer and traumatic brain injury. In this update, two studies delivered PST to parents of children with diabetes, however more PST studies on a broader range of chronic illnesses are still required (e.g. parents of children with pain). Replication studies are also needed for interventions that have been evaluated by only one research team, such as multisystemic therapy for families of children with diabetes, and PST for families of children with traumatic brain injury. We recognise that this goal may be difficult to achieve given the high degree of competition for funding and lack of interest among funding agencies for replication studies. Research is also needed to evaluate interaction effects such as the impact of changes in parent outcomes on child outcomes, as well as evaluation of specific treatment characteristics such as the intensity of intervention delivered to children versus parents. Fathers have also not been included in most trials and we do not know anything about the effects of the psychological interventions included in this review if delivered to fathers, which is a common critique of the field of paediatric psychology. Finally, in regards to duplication and piecemeal publication. Editorial policies are needed to inform authors regarding reporting standards for multiple publications from the same trial. Editors play a crucial role in creating and enforcing these policies, and need to take a pro-active approach to identifying such papers during the review process ( Committee on Publication Ethics 2011 ; World Association of Medical Editors 2012 ).

This is an updated version of the original Cochrane review published in Issue 8, 2012 ( Eccleston 2012b ). Chronic illness affects the lives of many children and their families. The prevalence of illness and disability differs by geographical and economic context. In the USA, Canada, Northern Europe, UK and Australia chronic activity-limiting conditions are reported to be frequent, with painful illness, allergy, asthma and obesity being common ( McDougall 2004 ). The changing demographic of childhood illness in economically wealthy countries has prompted a re-analysis of the role of paediatric medicine, as chronic illness becomes more prevalent than acute (e.g. Halfon 2010 ; Van Cleave 2010 ). Other parts of the world present different clinical challenges. In Africa, for example, life expectancy is 54 years and shorter in sub-Saharan Africa where almost half the population are children and the most prevalent chronic conditions are related to communicable diseases, in particular HIV-related disease, malaria and tuberculosis ( WHO 2011 ). The existing published literature shows a bias towards the medical management of chronic illness related to environment or lifestyle. Chronic pain in childhood is known to have widespread negative outcomes for children and parents ( Palermo 2000 ). Psychological intervention reviews have also been undertaken on the impact of sickle cell disease ( Anie 2012 ), recurrent abdominal pain/irritable bowel syndrome ( Huertas-Ceballos 2008 ), type 1 diabetes ( McBroom 2009 ), traumatic brain injury in children ( Soo 2007 ) and asthma ( Yorke 2005 ). The impact of childhood chronic illness on other family members, including parents, has been of growing interest for two reasons. First, it is now recognised that parents who have significant emotional distress of their own, and poor family functioning, can either directly or indirectly affect child outcomes by engaging in problematic responses to children’s pain behaviours ( Logan 2005 ; Palermo 2007 ). Second, it is now recognised that adaptive strategies used by parents can have a positive effect on child adjustment to chronic illness ( Logan 2005 ). Addressing the high level of parenting stress and mental health problems of parents, while enabling parents to be agents of change in the management of their child’s chronic illness, have recently been promoted as viable components of intervention in paediatric chronic conditions ( Jordan 2007 ; Palermo 2009b ). Studies have focused on the education of parents about the specific condition or treatment (e.g. cystic fibrosis; Savage 2014 ), whilst others evaluate the benefit of lay- or nurse-mediated social support (e.g. Lewin 2010 ). In psychological science, specific treatment approaches have been developed that focus on reducing the emotional distress expressed by parents, or on altering parenting behaviours to promote better child outcomes, whether this be decreasing emotional distress, or improving physical symptoms or behaviour. Psychological interventions of interest are defined as any psychotherapeutic treatment specifically designed to change parent cognition or behaviour, or both, with the intention of improving child outcomes. Psychological interventions are varied in their approaches and there is still debate surrounding which treatment is most effective for improving mental health and behaviour in parents and children with chronic illnesses. Such interventions include cognitive behavioural therapy (CBT), which has been found to be effective for modifying parent behaviour in children with a painful condition (e.g. Palermo 2009a ; Williams 2012 ). Problem-solving therapy (PST) has also been used to reduce distress in parents of children with various chronic illnesses ( D’Zurilla 1971 ; Sahler 2002 ). Other treatments have emerged from a family-systems approach that focuses explicitly on the family as a unit of intervention ( Ellis 2005 ; Wysocki 2000 ) such as multisystemic therapy (MST) or family therapy (FT). There are a variety of interventions described as psychological. Cognitive and cognitive behavioural therapies dominate, but therapies with a psychodynamic or systemic tradition are also represented. Family and couple therapies have also been developed. All psychological interventions include a rationale for therapy and specific goals for therapy. Education around illness and behaviour is common. Establishing the therapy and the therapist as credible is an important general stage ( Nock 2001 ). Next, a therapeutic relationship is established that will enable a confidential, non-blaming investigation of behaviour. Then, depending on the illness and behavioural presentation, specific components may include anxiety management, problem-solving skills, cognitive therapy for depression, and relationship management. Finally, most treatments will include a maintenance component that focuses on robust behavioural change within a normal home environment outside the clinic, over time. Such components have been used in parent interventions using different therapies to improve parental functioning, child behaviour and mental health. Cognitive behavioural interventions specifically are based on a number of foundational assumptions ( Beck 2011 ). First, behaviour is socially and historically contingent ( Skinner 1953 ). Second, cognition is an emergent property of behavioural context ( James 1980 ). Third, behaviour is regulated by cognitive goals ( Bandura 1989 ). Fourth, emotions influence both behaviour and cognition ( Ashby 1999 ; Gilliom 2002 ). Fifth, most behaviour is deployed outside of conscious awareness or control ( Bargh 2008 ). Finally, some attempts to control cognition and behaviour can have paradoxical negative effects on desired outcomes ( Beck 2011 ; Wegner 1994 ). Other interventions such as PST ( D’Zurilla 1971 ) are based on enhancing social competence through constructive problem-solving attitudes and skills. PST is based on a model of social problem-solving ( D’Zurilla 1999 ). Specific problem-solving skills are taught in sequential steps that typically include defining the problem, generating alternative solutions, decision making, and solution verification and implementation. PST has previously been effective with depression, anxiety and stress-related syndromes ( D’Zurilla 1999 ) and has been implemented with caregivers in a number of contexts. Family and systemic therapies specifically focus on a contextual and relational view of the aetiology and maintenance of behaviour. In particular, the target of health behaviour change is typically related to family functioning, or in the cognitive representation of the family, rather than on individual attitudes, beliefs or behaviour. Typically, family or systems therapy approaches will include multiple family members, and outcomes are often expressed on behalf of the family or dyad (two individuals regarded as a pair). Chronic illness is experienced by children, but within the context of the family. Parents are often detrimentally affected by their child’s illness, which adds a burden of adult distress to the child’s distress and disability. Further, parent distress can impair their performance in supporting their child in adapting to a chronic illness. Psychological interventions are available which focus on helping parents to help both themselves and their children. Establishing the evidence at this stage of development can guide best practice and further treatment development.

There were two objectives of this review. First, we aimed to evaluate psychological therapies delivered to parents of children with a chronic condition. Second, we sought to evaluate the risk of bias and quality of evidence for the included studies. Evidence was evaluated in two ways. First, we chose to determine the efficacy across psychological therapies for individual child health conditions. Second, we chose to determine the efficacy of individual psychological therapies across condition type. Data were analysed post-treatment and at follow-up. We did not identify any studies for gynaecological disorders or inflammatory bowel diseases. The majority of studies did not report whether they encountered any adverse events due to treatment, however five studies reported that there were no adverse events ( Gulewitsch 2013 ; Kashikar-Zuck 2012 ; Nansel 2009 ; Nansel 2012 ; Stark 2005 ). Kazak 2004 did not report any adverse events, but reported that participants reporting higher levels of distress were more likely to drop out of the treatment compared to less distressed participants, suggesting that the treatment may not be suitable for those who perhaps need it most. Because few studies reported whether or not they encountered adverse events, we are unable to comment further on the relevance of any adverse events on treatment safety. There were a number of analyses that could not be run due to missing data. This reflects the status of this developing field that has not yet produced a consensus of common core outcomes. First, we analysed data by each medical condition across all treatment classes, giving 72 possible analyses post-treatment and at follow-up ( Table 3 ). We were unable to conduct 48 analyses due to lack of data, 19 analyses were conducted but no effect was found, and five analyses found a beneficial effect of psychological therapies. First, psychological therapies were found to be beneficial at improving parenting behaviour for parents of children with cancer conditions post-treatment and for improving parent mental health at follow-up. Second, for children with diabetes and painful conditions, psychological therapies were beneficial at reducing medical symptoms (e.g., improving glycemic control, reducing pain intensity) for these conditions post-treatment, and these effects were maintained at follow-up for children with diabetes. We found no effect for 19 outcomes for four chronic conditions. There were insufficient data for 48 outcomes for all six conditions. Second, we analysed data by each treatment class across all medical conditions, giving 60 possible analyses post-treatment and at follow-up ( Table 4 ). We were unable to conduct 21 analyses due to lack of data, 23 analyses were conducted but no effect was found, and four analyses showed a beneficial effect across conditions. Cognitive behavioural therapy (CBT) was found to be beneficial at improving the primary disease-related symptoms of children with chronic illness conditions post-treatment. Problem-solving therapy (PST) improved parenting behaviour and mental health post-treatment and the effects for mental health were maintained at follow-up. It was not possible to conduct planned subgroup analyses for comparisons of the interaction between condition and psychological therapy effectiveness or the characteristics of particular treatments. We were unable to identify any trials for children with gynaecological disorders or inflammatory bowel diseases therefore studies investigating these disorders are still needed. One study including children with functional abdominal pain (FAP) and irritable bowel syndrome (IBS) was included in this update, where previously there was no evidence for parents of children with IBS ( Gulewitsch 2013 ). Despite this, IBS is a prevalent condition during adolescence ( Karabulut 2013 ). There were 21 studies that provided extractable data for parent outcomes. Parent outcomes were not included in the majority of trials. Only studies investigating children and adolescents with cancer had sufficient data for parent outcomes post-treatment and at follow-up. The remaining chronic conditions reported only one parent outcome post-treatment and no analyses could be conducted for parent outcomes at follow-up. Future randomised controlled trials (RCTs) of parent interventions should include relevant parent mental health and behavioural outcomes using standardised measures. When evaluating individual psychological therapies, we were able to evaluate parent outcomes for those who had received CBT or PST. However, follow-up evaluations must be conducted, particularly for parents receiving CBT. In addition, most studies evaluating PST investigated parents of children with cancer, although two new trials for this update evaluated PST for parents of children with diabetes. It is noticeable that MST is the only therapy type that does not have data for any parent outcomes post-treatment and at follow-up. Further, at follow-up no analyses could be conducted for parent outcomes when parents were treated with family therapy (FT), due to lack of data. Therefore, we are unable to comment on the maintenance of psychological therapies for parents receiving these treatments. The number of participants included in some studies is still small, with an average number of 80 participants entered into analyses. Although more trials have been included in this update, there are still insufficient data for many outcomes. Five ’Summary of findings’ tables are presented in this updated review. First, parent outcomes across all conditions post-treatment are presented ( Summary of findings for the main comparison ; Summary of findings 2 ). Due to a lack of evidence for parent outcomes at follow-up we were unable to assess these outcomes. Second, ’Summary of findings’ tables are presented for pain, cancer, and diabetes conditions ( Table 5 ; Table 6 ; Table 7 ). Due to a lack of data we did not produce ’Summary of findings’ tables for the remaining conditions. All outcomes in these tables were judged to be of low or very low quality. Contributing reasons for this are small sample sizes that were included in analyses, unclear or high risk of bias across studies, high heterogeneity within analyses, and suspected publication bias when authors are unable or unwilling to share data. Risk of bias was also assessed to determine the quality of individual studies. The majority of assessments were unclear or high risk across studies, highlighting issues with conducting and reporting of trials. However, assessments for the updated studies were mostly judged to be low or unclear risk of bias, indicating that risk of bias is improving in more recent trials. Random sequence generation, blinding of outcome assessors, and selective reporting bias had almost 50% of ratings judged as low risk. Incomplete outcome data and selective reporting were the only categories to contain any high risk of bias ratings. The following points contribute to lower quality of individual studies and evaluation of outcomes, and remain prominent issues within this field. First, multiple measurement tools within a given domain are often employed in individual studies and there is little agreement as to the preferred measurement tool across studies. This increases the heterogeneity of analyses. In some cases measurement is relatively homogeneous (e.g. pain intensity), whereas in others there is greater variety (e.g. family functioning scales in diabetes). These trials do not routinely identify a-priori the primary outcome, and there is unusual variety of outcome reporting. For example, one study discussed parent judgement of child outcome when the more valid measure, but non-significant finding, of child report was available ( Levy 2010 ). A posteriori selection of outcome measures is a significant problem in this field. As per our protocol, we were uninfluenced by the primary reporting of measures and focused on the best measure available in each domain. This field needs to take account of reporting biases and establish standards to improve the reporting of a-priori decisions regarding measurement. Second, we attempted to review trials with a parent intervention component. Therefore, trials inevitably included varied amounts of parent-directed content. Although we planned subgroup analyses, the data were not of sufficient quantity and quality to enable such an investigation. For some analyses we combined studies that were designed specifically with parents as the sole focus, and in others they were part of a combined treatment. Further, the philosophy of some treatments (e.g. MST) was antithetical to our strategy of determining an individual as a treatment target, however, we have included them in this review. It should be noted that significant findings in this review emerged when there was homogeneity of approach, homogeneity of outcome measurements, and a larger number of participants. Third, it should be noted that we had some difficulties in data retrieval due to incomplete and partial data reporting. Data were sometimes reported graphically, and participant means or standard deviations or both were often missing. As a result, complete or partial data were available to extract from 37 (n = 2985) of the 47 studies (n = 3778) included in this review. Twenty-eight trials produced complete outcome data in their published paper ( Allen 1998 ; Barakat 2010 ; Connelly 2006 ; Ellis 2004 ; Ellis 2005 ; Gulewitsch 2013 ; Hicks 2006 ; Hoekstra-Weebers 1998 ; Kashikar-Zuck 2005 ; Kashikar-Zuck 2012 ; Laffel 2003 ; Levy 2010 ; Marsland 2013 ; Nansel 2009 ; Ng 2008 ; Palermo 2009 ; Sahler 2013 ; Sanders 1994 ; Saßman 2012 ; Seid 2010 ; Shekarabi-Ahari 2012 ; Stehl 2009 ; Tsiouli 2014 ; Wade 2006a ; Wade 2006b ; Wade 2011 ; Wysocki 1997 ; Wysocki 2006 ) and nine authors responded to our requests for data ( Ambrosino 2008 ; Antonini 2014 ; Askins 2009 ; Ellis 2012 ; Mullins 2012 ; Naar-King 2014 ; Niebel 2000 ; Sahler 2002 ; Sahler 2005 ). Other authors were unable or unwilling to provide additional data or did not respond. The non-production of data is a problem in science ( Nature 2009 ), and has been particularly discussed in psychology ( Wicherts 2006 ; Wicherts 2011 ). We support the general move toward central registries for all trial data. Fourth, piecemeal and repeat publication was found in eight cases where multiple manuscripts were published from the same trial. In particular, one study ( Ellis 2005 ) was reported six times in five different journals while another trial ( Wysocki 1999 ) was reported five times in four different journals, with variable citation of previous publications in later publications. Such practices are unhelpful, create confusion and increase unnecessary labour ( American Psychological Association 2011 ). Many journals now have policies regarding publication of multiple manuscripts from the same trial, including a detailed description of previous publications from that trial and a statement regarding the unique contribution of the present manuscript (e.g. Drotar 2010 ). Finally, replication by other research teams independent to the therapy progenitors is uncommon. For example, Ellis and colleagues are the only group who have evaluated MST in young people with diabetes ( Ellis 2004 ; Ellis 2005 ; Ellis 2012 ). Similarly, PST for children and adolescents with traumatic brain injury (TBI) has not been evaluated by any research team outside of Wade and colleagues ( Antonini 2014 ; Wade 2006a ; Wade 2006b ; Wade 2011 ). Only a handful of reviews have also investigated psychological interventions for parents of children with a chronic illness. The results are consistent with a previous meta-analysis regarding the effectiveness of CBT in reducing child symptoms in young people with chronic pain ( Eccleston 2014 ), and are in agreement with the finding that children with painful conditions have reduced symptoms after psychological interventions ( Fisher 2014 ). The results are somewhat consistent with a meta-analysis of psychological paediatric oncology interventions, which showed no effects on child behaviour or child mental health, but positive effects for parent mental health and parenting behaviour ( Pai 2006 ). The results are consistent with previous reviews of psychological interventions that included parents of children with diabetes, which reported positive effects on child symptoms ( Armour 2005 ); however, they are not consistent with a review on families of children with diabetes, which found that psychological interventions improved family functioning ( McBroom 2009 ). Previous reviews of psychological interventions that included parents of children with asthma or skin diseases were inconclusive due to a lack of trials that met the inclusion criteria ( Ersser 2014 ; Yorke 2005 ). Notably, disagreements between the present meta-analysis and previous reviews may be attributable to differences in inclusion criteria, selection of outcome measures and/or selection of comparator group. The results are consistent with a systematic review and meta-analysis which found that PST has a beneficial effect on parent mental health and parenting behaviour post-treatment and at followup ( Law 2014 ), and consistent with the Law 2014 findings for other therapy types. One prior review indicated that psychological interventions which include coping skills training for adolescents and young adults with chronic illness (cancer, diabetes, juvenile idiopathic arthritis, sickle cell disease and asthma) and their parents/families had mixed effects on child psychosocial functioning and family functioning ( Sansom-Daly 2012 ). We were unable to find any previous reviews that compared results from individual psychological therapies across chronic illness conditions for parent outcomes or child symptoms. Therefore, we cannot draw any conclusions regarding the consistency of our results with previous reviews by treatment type for parent outcomes or child symptoms.

To evaluate the efficacy of psychological therapies that include parents of children and adolescents with chronic illnesses including painful conditions, cancer, diabetes mellitus, asthma, traumatic brain injury (TBI), inflammatory bowel diseases (IBD), skin diseases, or gynaecological disorders. We also aimed to evaluate the adverse events related to implementation of psychological therapies for this population. To evaluate the risk of bias of included studies and the quality of outcomes using the GRADE assessment.