Efficacy and cultural appropriateness of psychosocial interventions for Aboriginal and Torres Strait Islander paediatric burn patients and caregivers: a systematic review. CURRENT

Background: Paediatric burns are highly painful and traumatising injuries that are overrepresented among Aboriginal and Torres Strait Islander people. Paediatric burn patients’ pain remains poorly managed by pharmacological interventions, leading to increased anxiety, distress and trauma in patients and their caregivers. Nonpharmacological psychosocial interventions have been suggested as effective in reducing pain and psychological morbidities among paediatric burn patients and their caregivers; however, the degree of effectiveness and appropriateness for Aboriginal and Torres Strait Islander people is unclear. Methods: A non-date restricted systematic review was conducted through four databases. Studies published in English assessing psychosocial interventions on paediatric burn patients’ physical pain along with theirs and/or their caregiver’s anxiety, distress, or trauma symptoms were identified and included in this review. Included studies were assessed for their ability to reduce one of the outcomes of interests and for their reflection of Aboriginal and Torres Strait Islander peoples’ perspectives of health. Results: Of the 3,178 identified references, 17 were eligible. These include distraction based techniques (n=8), hypnosis/familiar imagery (n=2), therapeutic approaches (n=4), and patient preparation/procedural control (n=3). Distraction techniques incorporating procedural preparation reduced pain, while discharge preparation and increased ‘patient control’ reduced patient and caregiver anxiety; and internet based Cognitive Behaviour Therapy reduced short-term but not long-term post-traumatic stress symptoms. No interventions reflected Aboriginal and Torres Strait Islander peoples’ perspectives of health; and few targeted caregivers

and focused on reducing their symptoms.
Conclusions: The development and assessment of psychosocial interventions to appropriately meet the needs of Aboriginal and Torres Strait Islander paediatric burn patients is required.

Background
Burn injuries cause severe pain [1][2][3][4][5] and can result in psychological trauma [2][3][4], anxiety [3,5] and distress [1,3]. These uniquely challenging injuries affect Aboriginal and Torres Strait Islander people at higher rates than non-Indigenous Australian people. This is highlighted by the Burns Registry of Australia and New Zealand's most recent report that between 2017-2018 Aboriginal and Torres Strait Islander people were hospitalised for burn injuries three times more often than non-Indigenous people, and experienced significantly larger burns covering 10-49% of their Total Body Surface Area (TBSA) [6]. Paediatric specific data indicates similar discrepancies with Aboriginal and Torres Strait Islander children and adolescents experiencing 2.4 times higher rate of hospitalisation from burn injuries than non-Indigenous Australian children between 2011-2013 [7].
A unique challenge of these injuries lies within the persistent and debilitating base level of pain that is further intensified by regular procedures undergone for months to years following the initial injury [8,9]. The complex nature of burn related pain often results in poor management despite the administration of standard doses of analgesia [8][9][10][11][12][13] and is particularly difficult to monitor among paediatric burn patients who are less able to articulate the intensity of their own pain [14]. This is further complicated for Aboriginal and Torres Strait Islander people who may not report their pain at all [15,16] or verbally express their pain differently to non-Indigenous Australians [17]. This is particularly concerning as poorly managed pain during hospitalisation strongly predicts burn patients' psychosocial adjustment and overall wellbeing up to two years following treatment and hospital discharge [18].
More specifically, the pain and discomfort experienced by burn patients is associated with increased distress and anxiety [19]. This, in turn, increases the risk of developing other psychological morbidities such as acute stress and posttraumatic stress disorders (PTSD) [19]. The impact of burn related pain and distress is further exacerbated for paediatric burn patients who have a limited understanding of their injury and treatment [20], restricted agency in their care [21], and reduced ability to cope with the unpredictability of a hospital setting [22].
The struggles faced by paediatric burn patients is also greatly felt by their caregivers [20] who often experience overwhelming feelings of guilt, worry, panic, and anxiety whilst struggling with drastic shifts in their parenting role and ability to assist their child [23]. A review of empirical data highlights that 10-20% of paediatric burn patients and 4-42% of their caregivers reportedly experience symptoms of PTSD following the burn injury [24]. This highlights the need to effectively treat paediatric burn patients' and their caregivers' anxiety, distress, and psychological trauma. This is further emphasised by the finding that early onset of such psychological morbidities have a high rate of relapsing later in life [25].
The use of non-pharmacological, psychosocial interventions in conjunction with pharmacological analgesia have been suggested for reducing burn related pain and consequential psychological morbidities [26]. Interventions incorporating Gate Control Theory [21] techniques into change of dressing (COD) procedures are suggested as particularly effective in distracting the patient and reducing their ability to concentrate on painful stimuli [27]. Following this theory, virtual reality has shown particularly favourable results on pain management among paediatric burn patients alone [28], and combined with young adult burn patients [29,30]. Likewise, music therapy has shown promising effects in reducing anxiety and distress among paediatric burn patients alone [31][32][33], and combined with adults [34,35]. Other psychosocial interventions utilising cognitive approaches and behavioural strategies have demonstrated similar effects and suitability for use among a wide age range of children and adolescents. More specifically, cognitive approaches including imagery, preparation techniques, information sharing, and coping strategies are suggested as particularly suitable for older children and adolescents [14]. While behavioural strategies including breathing exercises, desensitisation, and positive reinforcement are suggested as particularly suitable for younger children [14].
Several studies have presented the usefulness of such psychosocial interventions, however, no comprehensive comparison or systematic review has been conducted to assess their rigour, effectiveness, or appropriateness in meeting the needs of Aboriginal and Torres Strait Islander paediatric burn patients and their caregivers.
This study assessed the effectiveness of any psychosocial intervention in reducing pain and psychological trauma, distress, and/or anxiety among paediatric burn patients and their caregivers generally. Alongside this assessment, we systematically evaluated the appropriateness and applicability of such interventions for use among Aboriginal and Torres Strait Islander families to inform the necessity and directions for future developments of culturally appropriate interventions.

Protocol and registration
Details of the protocol for this systematic review were registered on PROSPERO, the international prospective register of systematic reviews (CRD42018073451) [36].

Eligibility criteria
The below eligibility criteria were applied. Associated search terms were developed in consultation with experts from the University of Queensland library [see Additional file 1]. In accordance with the model, data was extracted on five core aspects of health: physical (4 items), psychological (3 items), social (4 items), spiritual (3 items), and cultural wellbeing (3 items). All items was graded zero (not present) or one (present) with relevant details extracted. The cultural components data form is included as supplementary materials.
Risk of bias was assessed against the Cochrane Collaboration's tool for assessing risk of bias [ 39] (nine items), and an additional three items from the Cochrane suggested risk of bias criteria for EPOC reviews [ 40] to assess potential intervention contamination, and baseline outcome and characteristic similarities. All risk of bias items were graded zero (high/unclear bias) or one (low risk of bias). Study quality was evaluated by the Downs & Black Checklist [ 41] (28 items) to assess reporting, external validity, internal validity, and study power. Power was graded based on the smallest sample group (0 = n < 1 , 1 = n 1 -n 2 , 2 = n 3 -n 4 , 3 = n 5 -n 6 , 4 = n 7 -n 8 , 5 = n > 8 ), and all other items graded zero (no) or one (yes); possible maximum score of 31.

Data synthesis
Meta-analysis was not appropriate due to high heterogeneity among studies.
Therefore, data was synthesised narratively, presented in text by outcome (i.e. pain, anxiety, distress, and trauma), and tabulated by intervention type.

Study selection
Database searches returned 3,638 abstracts published prior to 18th November 2019. Duplicates were removed and the remaining 1,937 abstracts underwent title and abstract screening, resulting in 1,821 exclusions and 116 inclusions for full text revision. Exhaustive attempts to obtain full manuscripts was successful for 100 abstracts. Of the remaining 16, five were not able to be acquired and 11 were unavailable in English. Full manuscripts were double screened by the lead author and two co-authors, resulting in a further 82 exclusions. References of the remaining 18 studies were hand-screened, identifying an additional ten abstracts of interest, two of which were included for data extraction [see Additional file 3]. Data extraction was not possible for three studies despite repeated attempts to contact study authors to obtain missing data and further information. Data was extracted from the final 17 eligible studies and narratively synthesised. Exclusion rates are outlined in the PRISMA flow diagram (Fig. 1.).

Main results
The study and participant characteristics of included interventions are presented in Tables 2 and 3 respectively. The key findings of included interventions are presented by intervention type in Table 4, and narratively synthesised by outcome below.      Pain & distress • FLACC scores lower in MMD than control at post-removal (p = < 0.001), pre-application (p = 0.021), post-application (p < 0.001), 50% reduction at removal.
• Intervention group did not affect STAI-CH scores for any patient's or caregiver of patients with < 30% TBSA burns at discharge or 1st follow-up visit.
APPT     [45][46][47], caregiver observations of pain [46,47], and nurse observations of pain and distress [46,47] when the procedural preparation story "Bobby get's a burn" and interactive distraction games were provided together [45,47], and separately [46]. The benefits of MMD distraction increased with repeated use and was borderline more effective in reducing nurse observations of pain and distress behaviours when used alone than in combination with procedural preparation [46,47]. However, less interactive video game distraction was found to reduce self-reported pain and increase caregiver observations of pain over time compared to Multi-Modal Device -Procedure Preparation (MMD-PP), and Multi-Modal Device -Distraction (MMD-D) [46]; or reduce nurse observations of pain and distress but not affect self-reported pain compared to standard distraction [48].
Three-dimensional virtual reality increased self-reported pre-procedural pain and reduced self-reported procedural pain more effectively than passive distractions [49]. While off-the-shelf virtual reality increased nursing staff's observations of pain and distress behaviours and had no effect on patients' or caregivers' reports of pain [50]. Music therapy reduced self-reported pain compared to standard care when provided immediately following COD [51, 52]; however, did not affect patients' selfreported pain when provided during COD [51,52]. Likewise, 'medical play' prior to COD commencement did not affect patients' self-reported pain; however, reduced nursing staff observations of pain and distress behaviour at insignificant levels [43].
In contrast, the use of Child Life Therapy (CLT) reduced patients' pain as observed by caregivers and an independent assessor, and increased nursing staff's observations of pre-procedural pain [53]. Similarly, hypnotherapy reduced pain levels at the third COD as self-reported by patients less than 8 years of age and caregivers [44]. Stress management during COD reduced self-reported pain and distress from baseline to post-intervention; however increased similar to the control group when the therapist was absent [54]. Further, patients that received increased 'patient control' and 'predictability' required less analgesic medication during the first two weeks of hospitalisations and more in between CODs [55].

Distress
The MMD and Ditto™ devices' procedural preparation and distraction also reduced patient self-reported distress [45,47], compared to standard care which increased self-reported distress [45]. Similarly, stress and pain management during COD reduced patient self-reported distress but only in the presence of a therapist [54].
In contrast, the use of familiar imagery did not reduce patients' self-reported distress or investigators' observations of distress behaviours [56]. Likewise, music therapy during and following COD did not reduce nurse observations of distress [51, 52] but rather increased observations of distress when performed during COD procedures [52].
Anxiety Preparation for dressing procedures or hospital discharge reduced patients' and caregivers' anxiety [43,55,57]. Similarly, increased 'predictability' and 'patient control' during CODs reduced patient's anxiety during the first two weeks of hospitalisation; however, not significantly [55]. Hospital discharge preparation reduced anxiety among caregivers of children with burns affecting ≥ 30% of their TBSA, but did not impact other caregivers' or patients' state anxiety during CODs [57]. Blakeney et al. found that providing psychoeducational programs had similar effects as standard care in reducing patient's self-reported anxiety/depression scores from pre-intervention to 1 year post-intervention [42]. Hypnotherapy lowered patients' pre-removal anxiety as reported by patients aged older than 8 years at second COD, and caregivers for patients aged less than 8 years at second and third COD [44]. Child Life Therapy (CLT) reduced caregivers' observations of patients' procedural anxiety; however increased both caregiver anxiety at pre and during procedures, and nurse observations of anxiety at pre-procedure [53]. The Ditto™ device lowered self-reported anxiety at pre-removal for patient's > 8 years [45].
Likewise, tablet based electronic game distraction reduced anxiety during and after COD procedures compared to standard distraction [48]. In contrast, virtual reality did not reduce patient anxiety during COD [45,49,52]; and music therapy during CODs increased self-reported anxiety before and during procedure [52].

Trauma
Few studies measured psychological trauma symptoms and those that did had mixed results [44,45,58]. Sveen et al.'s online self-help program reduced patient posttraumatic stress scores six weeks post-baseline and three months postintervention; however symptoms returned to baseline levels 12 months postintervention [58]. The online self-help program also did not reduce caregivers' actual or perceived stress at any time [58]. Hypnotherapy significantly lowered patient self-reported trauma impairment severity compared to standard care; however, also increased caregiver's observations of trauma symptoms in children aged < 7 years at three months post-injury [44]. The Ditto™ device did not reduce children's stress or trauma symptoms one week following injury or three months following wound healing [45].

Discussion
This systematic review highlights a gap in understanding the effectiveness of psychosocial interventions for Aboriginal and Torres Strait Islander paediatric burn patients' and their caregivers. Previous systematic reviews have not accounted for this specific group [59][60][61][62][63], assessed the effects of all psychosocial interventions available [59,61], or considered psychological trauma as a primary outcome [59][60][61][62]. To the authors' knowledge, this systematic review is the first to assess the effectiveness of psychosocial interventions in reducing pain and/or anxiety, distress, and trauma symptoms among paediatric burn patients and their caregivers as well as their relevance to First Nations peoples.

The appropriateness of the included interventions for Aboriginal and Torres Strait
Islander people could not be determined due to the lack of cultural components and First Nations participants. The limited representation of First Nations people is disconcerting given all but one study [58] were conducted in countries with strong First Nations presence; including the United States of America (n = 9, 53%), Australia (n = 6, 35%), and South Africa (n = 1, 7%). The circumstance surrounding the omittance of First Nations people from most studies is unclear; however, some potential reasons may include inaccurate ethnicity records as exemplified by the categorisation of 'lighter skin' vs 'darker skin' by Brown et al. [45], or potential bias in intervention design appeal or accessibility for First Nations people.
Similarly, only two studies provided interventions to caregivers and focused on their symptoms [57,58]. The limited focus on caregivers is also concerning given the strong evidence of psychological implications of paediatric burn injuries on caregivers [20,23,64]. Furthermore, sufficient psychosocial support, education and involvement of caregivers in CODs can build caregivers' competency and aid with their coping during their child's burn treatment [65,66].
The relatively small number of included studies (n = 17) demonstrates the limited scope of formally assessed psychosocial interventions with clear comparison groups.
We acknowledge the difficulty of applying rigour and standardisation to psychosocial interventions due to their need for flexibility and adaptability; attributes often key to their responsiveness to individual needs. Standardisation is further compounded by the multi-factorial nature of 'standard' burns care, resulting in varied COD approaches [45], length of time from pain medication to dressing removal [43], days of hospitalisation [57], informal procedural preparation [46], and 'standard' distraction during COD [44]. However, some studies lacked standardisation of elements outside of 'standard care' that were provided to controls including additional investigator verbal support [56] and active distraction during CODs [55].
This review was limited to studies available in English and with Randomised Control Trial (RCT) and NRCT designs, resulting in the exclusion of seven otherwise relevant studies. These excluded studies were generally reflective of this review's findings; however, three reported on interventions not captured here [67][68][69]. One study found art therapy effective in allowing paediatric burn patient's to express their traumas [69], another found art and play therapy combined reduced patient anxiety [67], and the other found group therapy reduced caregiver anxiety [68]. It [56], and its inability to capture pain and distress differences between groups [43]. Similarly, the Achenbach Child Behavioral Checklist [ 42], State-Trait Anxiety Inventory [ 57], and anxiety tool used by Kavanagh [55] were reportedly too general to capture paediatric burn patients' anxiety during COD.
Despite these challenges, the included interventions demonstrated that procedural distraction via hand-held devices are effective in reducing patient's pain [45][46][47][48]; however, less effective when only games were available [48] opposed to procedural preparation stories as offered by the Ditto™ [45] and MMD [46,47]. This indicates that procedural information provided at any time during procedures can reduce patient pain. This is supported by Damanhuri et al.'s finding that active distraction incorporating additional COD information and encouragement was far more effective than passive distraction such as music therapy or games [14]. However, this review also found that distraction techniques did not reduce paediatric patient's anxiety [45,49,52], or trauma symptoms [45] regardless of incorporation of procedural preparation. In contrast, therapeutic approaches were effective in reducing psychological morbidities among patients and caregivers. In particular, CLT reduced caregiver and independent assessor's observations of patient pain, and caregiver's observations of patient anxiety [53]. An online self-help program was the only intervention found to effectively reduce patient trauma symptoms [58]. Similarly, incorporating stress and pain management into CODs reduced patient distress but was not sustained in the therapist's absence [54]. The results of the included studies should be interpreted with consideration of potential bias due to difficulties in intervention allocation blinding [42][43][44][45][46][47][48][49][50][51][52][53][54][55][56][57][58].

Conclusion
This review returned a limited number of interventions that effectively reduced paediatric burn patient and caregiver psychological morbidities. The scarcity of work on reducing psychological trauma symptoms is particularly disconcerting given the volume of work emphasising the highly traumatising nature of burn injuries for both patients and families [2][3][4]. This highlights a need for additional work to better support and prepare caregivers for their vital role in providing security and comfort to their children during procedures. Of main concern to this review, the well-   Risk of Bias