Interventions for family members and carers of patients with borderline personality disorder: A systematic review

Carers of patients with borderline personality disorder (BPD) experience high levels of distress. Several studies have been carried out on interventions designed to decrease their burden. However, the evidence from these studies has not been summarized. The objective of this work is to explore the clinical utility of interventions developed for family members of patients with BPD. A systematic review was conducted following the PRISMA guidelines (registration number CRD42018107318), including psychological interventions focused on relatives of patients with BPD. The following databases were used: PsycINFO, PubMed, EBSCOhost, and Web of Science. Two independent researchers reviewed the studies to determine whether the eligibility criteria were met. A total of 2303 abstracts were identified. After duplicates had been removed, 1746 studies were screened. Finally, 433 full-text articles were reviewed, yielding 11 studies that satisfied the inclusion criteria. Results show that these interventions with different clinical formats and settings are effective. The quality of the included studies varies, and the empirical support for these programmes is still preliminary. The results help to establish a Page 1 of 42 Family Process

. In addition, participating in treatment enables them to know more about the disorder, set limits, validate their own experiences, and prioritize their own health in a supportive environment (Penney, 2008). Furthermore, the patients can also obtain benefits from the family's involvement in the treatment. Although it has not yet been demonstrated that the carer's burden can alter the outcome of a patient with BPD, some studies have found that interventions with family members can improve the effectiveness of treatments for people with BPD and their long-term prognosis, and reduce the interpersonal factors that can maintain BPD symptoms, taking into account family members' difficulties in a non-invalidating way (Fossati & Somma, 2018;Gunderson et al., 2006;Hooley & Hoffman, 1999). Furthermore, working with family members could have an influence on reducing patient relapse and rehospitalization and improving patient recovery (Dixon et al., 2001). Therefore, it is necessary to develop and test intervention programmes designed specifically for family members of patients suffering from BPD, and analyse existing studies. Thus, the objective of the present work, as described in detail below, is to explore the clinical utility of the programmes that have been developed so far in the field of interventions for family members of patients with BPD.
Moreover, some authors have focused on analysing the experiences of carers of people diagnosed with BPD, showing that carers feel discriminated against when they ask for help and support from health services (Miller & Skerven, 2017). They express a lack of recognition and support for the needs of the person with BPD and his/her relatives, and they state that professionals do not know how to adequately respond to their demands (Lawn & McMahon, 2015). Indeed, the National Institute for Health and Care Excellence (NICE) guidelines for BPD treatment have highlighted the need to provide interventions for family members who are living with and caring for a patient Some research has examined interventions that include family members of patients with BPD, and the studies differ in the focus or structure of the treatment. In general, there are programmes for patients where family members are included in a few sessions (Blum, Pfohl, John, Monahan, & Black, 2002;Rathus & Miller, 2002;Woodberry & Popenoe, 2008). There are also some programmes designed for patients and relatives that include different treatment components for each group. In these cases, the treatment is given jointly, but the main focus is on patients rather than on family members or carers (Santisteban, Muir, Mena, & Mitrani, 2003;Santisteban et al., 2015).
Thus far, small pilot studies have only provided feasibility and acceptability data for the intervention programme (Santisteban et al., 2003;Santisteban et al., 2015). In general, all these programmes recommend that family members be included in the treatment plan, but the fundamental focus is not on the family or carers. Finally, there are programmes specifically focused on relatives of patients with BPD, such as Family Connections (Flynn et al., 2017;Hoffman et al., 2005Hoffman et al., , 2007.

Search strategy
A systematic search of peer-reviewed literature was conducted using the following databases: PsycINFO, PubMed and Cochrane. The following search terms were used to represent the areas of: i) psychological interventions; ii) relatives, family members, carers; iii) borderline personality disorder (see Annex 1). The search was conducted until 26th June (2018). Articles from Google Scholar and references from relevant articles were also searched for additional studies. If the full text was not available or data were missing or unclear, we contacted the respective author. Only studies written in English or Spanish were included. We did not restrict the publication year.

Inclusion criteria
Page 7 of 42 Family Process systematic review. The study selection process is presented in the PRISMA flow chart ( Figure 1).
- ------------------------------------ Table S1 presents the fundamental results obtained in the studies taken into consideration in this review, as well as the description of a series of variables addressed in the process of extracting information from these studies: objectives, participants and type of relationship with the patient, age and sex of the participants, design followed in the study, description of the intervention used, content of the intervention, context in which it is applied and who applies the intervention, theoretical model on which the intervention is based, outcome measures used, effect sizes, limitations of the study, and summary of the results. As can be seen in Table S1, eleven treatment programmes that specifically focused on relatives of patients with BPD were found in this review work. Two of these programmes are psychoeducational, another study is based on mentalization, and the rest are DBT-based.
The objective of psychoeducational interventions is to provide family members of BPD patients with information about the disease and help them to understand some of their relative's behaviours, thus improving the relationship and family climate. The work by The most empirically supported study is Family Connections (FC) (Hoffman et al., 2005), one of the first interventions designed for relatives of patients with BPD, applied by either clinicians or trained relatives. To test the efficacy of FC, five uncontrolled clinical In terms of the people who implemented the intervention programme, in many studies they were psychiatrists, psychologists, or PhD-level clinicians trained in DBT, aided by a graduate student or support worker. Moreover, in several of the studies, (Bateman & Fonagy, 2018;Hoffman et al., 2005Hoffman et al., , 2007 applying FC or the mentalizationbased training, the programme was delivered by volunteer family members who had been previously trained. With regard to the outcome measures considered in these studies, as Table S1 shows, most of the studies evaluated the construct of burden (objective burden and subjective burden), emotional burnout, feelings of pain and grief, family climate, depressive-anxious symptoms, perceived level of coping and mastery, relationship skills, and family climate. Nevertheless, in several studies, other variables were also considered, such as: the number of conflicting or adverse incidents involving the patient with BPD, as reported by the carer, hopelessness, and other symptom patterns of psychological distress, quality of life, family empowerment, and mindfulness.

Discussion
The aim of this study was to carry out a systematic review of the intervention programmes developed and tested to date to help relatives of patients with BPD. This review has focused on interventions specifically designed for family members, relatives, or people living with these patients. The interventions focus on the relatives, unlike programmes for patients that merely include relatives in some sessions (Blum et al., 2002; both, but where the target is still the patient and no data are provided on the outcome of the intervention in the relatives (Santisteban et al., 2003(Santisteban et al., , 2015. The search identified a total of 11 studies that met the inclusion criteria. All these programmes are offered in group format, but they differ in the type of orientation and contents, as well as the structure of the intervention. As described in the results section, two studies present only psychoeducational contents (Grenyer et al., 2018;Pearce et al., 2017).
The rest of the programmes, despite containing some psychoeducation sessions, are skills training programmes, either based on mentalization (Bateman & Fonagy, 2018) or on DBT skills (Hoffman et al., 2005(Hoffman et al., , 2007. With regard to the DBT skills training studies, they have different structures and numbers of sessions. Regarding the results, as Table S1 shows, significant improvements were obtained in most of the outcome measures used in the different studies. Therefore, the first conclusion that can be drawn is that some programmes designed specifically to help family members of patients with BPD have obtained empirical support. In general, all these programmes have been shown to be useful for reducing emotional burnout, feelings of pain and guilt, overload, and depressive-anxious symptoms, and for improving relationship skills and the family climate. They provide family members with a series of strategies that help them to relate to the patient suffering from BPD and know how to act in a crisis situation. The FC programme deserves to be highlighted because it is the most advanced so far, both in terms of content specifically designed for families and in terms of strategies designed to improve its dissemination, such as training family members to hold the therapy groups themselves. Furthermore, conducting these kinds of programmes in different settings may be a time-and cost-efficient implementation option (Liljedahl et al., 2019).
Although these results are hopeful, our second conclusion is that the empirical support for these programmes is still preliminary. This line of research has not developed as much as it should, given the important implications for the whole family when a member has a problem as serious as BPD. In any case, these studies indicate that the development and implementation of intervention strategies for family members with BPD begins to gain relevance, in terms of the guidelines to follow to improve the family dynamics and, in turn, help to achieve patients' prompt recovery.
Nevertheless, these studies also have some limitations, given that so Taking into consideration what has been achieved so far, we consider it necessary to make progress in a number of research topics. First, the specific programme components responsible for improvements need to be identified, and the relative effectiveness of the components should be determined (Hoffman et al., 2007).
Second, there is a surprising lack of studies that explore the psychopathology or limited skills of family members or exclude them from the intervention if they have psychopathology (Grenyer et al., 2018). These studies could provide a more detailed analysis of the family members' different skills or gaps, in order to better orient and choose the intervention components. In this regard, the studies by the Fruzzetti group on validation skills in family members should be highlighted (Fruzzetti, Shenk, & Hoffman, 2005) because they showed that family invalidation can contribute to establishing and maintaining BPD.
Another question to examine in future research is whether it is more beneficial for interventions to be performed by volunteer family members who have already received them and been trained for this purpose, as in the FC programme (Hoffman et al., 2005(Hoffman et al., , 2007, or by expert clinicians (Flynn et al., 2017). From a cost-benefit perspective, and taking into account the possible benefits if the effectiveness of the intervention is similar when applied by expert clinical personnel or by family members, we understand that it is important to make an additional effort in this direction and compare these two formats (groups led by family members vs groups led by clinicians). This would represent a step in the direction defended by Kazdin (Kazdin & Blase, 2011) regarding the types of intervention needed to reduce the burden associated with suffering from mental disordersin this case, the possibility that the intervention could be applied by non-professionals.
In addition, with regard to relevant outcome measures, from our point of view, in order to state that an intervention for relatives is effective, it not only has to reduce the clinical symptomatology of the family members, but also the conflictive relationship between family members and patients, and achieve improvements in crisis management. In This work has some strengths. It is the first systematic review to analyse existing interventions specifically designed for relatives of patients with BPD that conforms to PRISMA guidelines and has a previous record in PROSPERO. It also has some limitations.
Although we tried to be comprehensive in our search strategy, it is possible that some studies were not located and have not been included in this review. Moreover, programmes that only included family members in one-off sessions were excluded, as well as general community interventions that were not specifically designed for family members of patients with BPD. Finally, we were not able to perform meta-analytic calculations because only two randomized controlled trials were identified that reported efficacy data (Bateman & Fonagy, 2018;Grenyer et al., 2018). Therefore, future meta-analyses are warranted when there is a minimum number of studies to conduct them.
helping family members and relatives of patients with BPD. Given the seriousness of the disorder and the significant burden for the family, it is necessary to think of helpful strategies for families and make it possible to achieve faster and more consistent patient recovery and better family dynamics. In conclusion, it is fundamental to focus attention, work, and resources on designing, developing, and testing specific interventions for family members of people with BPD. Currently, this line of research has only just begun, and the present study tries to make a modest contribution to its advancement. For more information, visit www.prisma-statement.org. "Family connections (FC)" (Hoffman et al., 2007) Replication and extension study of FC (Hoffman et al., 2005) To assess the impact of the FC program on the wellbeing of BPD carers as well as sex differences. The same description of the original FC study (Hoffman et al., 2005) The same content of the original FC study (Hoffman et al., 2005) The same as in the original FC study (Hoffman et al., 2005) The same as in the original FC study (Hoffman et al., 2005) -BAS -PFBS -CES-D -GAS -Master y Scale was adapted to be delivered over two intensive weekends split by 1 month (FC-R) compared to the usual approach, which was delivered weekly for 12 weeks (FC-S). The intervention is the same as described in the original FC study (Hoffman et al., 2005) FC: The same description as the original FC study (Hoffman et al., 2005) Given by therapists, all of whom were trained in DBT and FC. Clinical setting The same as the original FC study (Hoffman et al., 2005).  (Reinhard, Gubman, Horwitz, & Minksy, 1994); PFBS: Perceived Family Burden Scale (Struening et al., 1995); CES-D: Revised Centre for Epidemiologic Studies Depression Scale (Radloff, 1977); GAS: Grief Scale (Struening et al., 1995); Mastery Scale (Dixon et al., 2001); RCT: Randomized Controlled Trial; ZBI Zarit Burden Interview (Zarit, et al., 1980); SCL-90R: Symptom Checklist (Derogatis, 1994); HAD: Hospital Anxiety and Depression Scale (Bjelland, Dahl, Haug, & Neckelmann, 2002); OTAU: Optimized Treatment-as-usual; PMS: The Personal Mastery Scale (Pearlin et al., 1981); IIP-PD-25: Inventory of Interpersonal Problems-Personality Disorders (Kim & Pilkonis, 1999); FAS: The Family Attitude Scale (Kavanagh et al., 1997); CGSQ-SF7: Caregiver Strain