Unresolved grief in parents of children with CF: A pilot randomised controlled trial on the use and delivery sequence of disease-related education and psychotherapeutic support


 Diagnosis of chronic disease in a child can result in unresolved grief (UG) in parents. This study aimed to evaluate the efficacy of psychological insight-oriented therapy (IOT) as a treatment for UG compared to disease related education in parents of children with cystic fibrosis (CF). Sequence of delivery, first IOT then disease related education (or vice versa ) was also examined, to let all participants experience both interventions.
Parents were screened for UG. Parents with UG were randomised to either five one-hour sessions of IOT or five one-hour sessions of education. Measures were assessed pre-intervention, after the first intervention period (primary efficacy assessment), and after the second intervention period (swapping intervention).
Forty-seven parents were screened of which 46.8% (22/47) had UG. Median duration of UG was 5 years (range: 6 months to 14 years). Anxiety (50% vs. 20%, p =0.03) and stress (59% vs. 28%, p =0.03) were significantly more prevalent in parents with UG. There was no difference between arms in the odds of UG resolving either following the first intervention period (OR 0.88; 95%CI 0.5, 1.5) or the second intervention period (OR 0.91; 95%CI 0.5, 1.6). While not statistically significant, adjusted mean values of all nine mental health measures were lower in the IOT (first) arm compared to the ED (first) arm, following the first intervention period.
Conclusions: UG is a significant burden for families affected by CF. Provision of disease related education and psychological support, regardless of sequence, can result in resolution of grief.

Parents report the period directly following diagnosis to be a time of shock, disbelief, grief 62 [5; 6], heightened emotional experiences and difficult thoughts [7]. This is also typically a 63 time of intense CF related education and engagement with a multidisciplinary team. For some 64 parents the initial disease related education has been found to be an overwhelming experience 65 in which coping capacities can vary greatly [7; 8]. other feelings that arise with these news, as well as the acceptance of the long-term aspect of 71 parenting a child with these challenges [9]. As the features of being unresolved are closely 72 associated and parallel with many features of grieving, for the purposes of this study, we will 73 refer to a lack of resolution as unresolved grief (UG) 74 75 Prolonged UG can have long-lasting effects on the quality of the parent-child relationship in 76 terms of the parent-child attachment, and consequently on the child's developing sense of self 77 [10; 11]. Specific and targeted interventions to address parental unresolved grief following a 78 diagnosis of CF have not been studied to-date.   In this pilot study, participants were screened for UG. Those with UG were then randomised 96 to a pre-post design to assess two interventions.
Setting, participants and screening 98 Parents of children with CF aged 6 months to 18 years and living within, or close to, the 99 Perth (Australia) metropolitan area were identified through the only paediatric CF service in 100 Western Australia and approached with or mailed information about the study. In total 105 101 families were approached and 7 opted out of being contacted further by the study team.

102
Parents who gave written informed-consent to participate in the study were screened for grief 103 in relation to their child's diagnosis with CF with the Reaction to Diagnosis Interview (RDI) 104 [12] administered during a one-hour interview with an RDI-trained psychologist. The    Participants were assessed again for UG and emotional wellbeing following each period of 120 intervention. Therefore, participants were assessed for UG and emotional wellbeing at three time-points pre (screening), post (after intervention one), and follow-up (after intervention 122 two).    The overall setting of education sessions could be described as optimistic and caring.

179
The CONSORT diagram is presented in Figure 1 207 Mean values for all eight mental health outcomes (scales and totals), were all lower post-208 invention compared to pre, in both arms (Table 2, Figure 2). This decrease was only 209 statistically significant for DASS-Stress in the IOT arm (-2.12; 95% CI -3.57, -0.68).   Parental unresolved grief (UG) related to their child being diagnosed with CF was highly 232 prevalent amongst parents, and for some this had been present over a decade post diagnosis.

233
Anxiety and stress were more frequently present in parents with UG. IOT and CF education 234 (in either delivery sequence) appeared to mitigate parental grief and resulted in resolution of grief. Whilst increased rates of depression and anxiety and higher levels of stress have been 236 well described in patients with CF and their caregivers, and UG related to other chronic and 237 permanent conditions has been described [16][17][18][19][20], to our knowledge this is the first study to 238 investigate UG related to a diagnosis of CF and also the first clinical trial to investigate 239 methods to facilitate resolution of such grief.    [16][17][18][19][20]. Therefore, improvements in both anxiety and stress levels 260 following resolution of grief was encouraging.

262
The study had a number of limitations. Firstly, the sample size was limited. However, results 263 clearly showed that grief that had been present for years resolved for multiple participants 264 following the relatively brief study period. For some participants the limited intervention may 265 not have been enough to fully explore the depth of their feelings regarding their child's 266 diagnosis and result in resolution of grief, hence the resolution of grief seen with the limited 267 intervention was encouraging. Secondly, study participants were mostly mothers. The 268 implications of our findings to fathers therefore require further study. Thirdly, only one 269 certified RDI coder was used to analyse the data to make the UG diagnosis. Despite this, the 270 reduction in 'coder assessed UG' coincided with a substantial reduction in mental wellbeing 271 indicators, supporting the study's ability to objectively assess change following intervention.

273
The present study also demonstrated that revisiting CF related education proved to assist 274 parents to resolve UG. Clearly, offering CF related education to parents shortly after 275 diagnosis (i.e. during a time of high stress, shock and disbelief) may not be conducive 276 towards the information being taken on board and processed, and might result in distortion of 277 information. Providing parents with an opportunity to revisit CF education after a period of 278 adjustment may enable parents to obtain clarity and reprocess information more accurately.

279
The measurement of UG using the RDI is both time consuming and costly, it requires