Background

Schizophrenia spectrum psychoses rank among the most disabling conditions of modern times (World Health Organisation, 2001). The personal and financial costs are considerable and well documented (Knapp, Mangalore & Simon, Reference Knapp, Mangalore and Simon2004). The first episode of psychosis frequently occurs during adolescence and early adulthood with an average delay of 22 weeks to 3 years before individuals begin formal treatment (Norman & Malla, Reference Norman and Malla2001; Stefan, Travis & Murray, Reference Stefan, Travis and Murray2002; Norman et al. Reference Norman, Malla, Verdi, Hassall and Fazekas2004). The initial episode, including the period of untreated illness, often coincides with a time when individuals are already encountering changes in their role and peer relationships, and are negotiating transitions to independence and responsibility (Mackrell & Lavender, Reference Mackrell and Lavender2004).

A critical period – the first 5 years

For service users, the onset of psychosis is commonly associated with a broad range of feelings, including shock, confusion, fear, distress and despair. The literature suggests that the first 5 years constitute a critical period for treatment and outcomes (Birchwood, Todd & Jackson, Reference Birchwood, Todd and Jackson1998; Lieberman & Fenton, Reference Lieberman and Fenton2000). We know, for example, there are high levels of trauma, depression and anxiety in people presenting to early psychosis services (Koreen et al. Reference Koreen, Siris, Chakos, Alvir, Mayerhoff and Lieberman1993; Birchwood, Spencer & McGovern, Reference Birchwood, Spencer and McGovern2000; Birchwood, Reference Birchwood2003; Mueser & Rosenberg, Reference Mueser and Rosenberg2003; Jackson et al. Reference Jackson, Knolt, Skeate and Birchwood2004). Suicide levels are elevated in psychosis, but particularly so in the early illness years (Caldwell & Gottesman, Reference Caldwell and Gottesman1990; Robinson et al. Reference Robinson, Harris, Harrigan, Henry, Farrelly, Prosser, Schwartz, Jackson and McGorry2010). Aggressive behaviour is common, with recent figures suggesting that about 40% of service users engage in aggressive acts at first episode (Dean et al. 2006). Rates of co-morbid substance abuse disorders and problematic drug misuse are also high in first-episode groups (Green et al. Reference Green, Tohen, Hamer, Strakowski, Lieberman, Glick and Clark2004; Van Mastright et al. Reference Van Mastrigt, Addington and Addington2004; Lambert et al. Reference Lambert, Conus, Lubman, Wade, Yuen, Moritz, Naber, McGorry and Schimmelmann2005). In contrast, adherence to prescribed pharmacological treatments is poor (Coldham, Addington & Addington, Reference Coldham, Addington and Addington2002). While remission from positive symptoms is often rapidly achieved following the first episode (Lieberman et al. Reference Lieberman, Tollefson, Tohen, Green, Gur, Kahn, McEvoy, Perkins, Sharma, Zipursky, Wei and Hamer2003; Schooler et al. Reference Schooler, Rabinowitz, Davidson, Emsley, Harvey, Kopala, McGorry, Van Hove, Eerdekens, Swyzen and De Smedt2005), social dysfunction is a significant issue from the very early phases (Addington et al. Reference Addington, Penn, Woods, Addington and Perkins2008). Service users tend to become isolated very quickly after onset and their social contacts may become restricted to paid mental health professionals and a few close family members (Macdonald, Hayes & Baglioni, Reference Macdonald, Hayes and Baglioni2000; Macdonald, Sauer, Howie & Albiston Reference MacDonald, Sauer, Howie and Albiston2005; Horan et al. Reference Horan, Subotnik, Snyder and Nuechterlein2006; Berry, Wearden & Barrowclough, Reference Berry, Wearden and Barrowclough2007; Stanghellini & Ballerini, Reference Stanghellini and Ballerini2007).

Effects on families

A large body of literature confirms that the negative impact of psychosis extends beyond the service user; families are also adversely affected. In general, many service users maintain regular contact with their families. This is the case particularly during the early phase, when they are more likely to be living with their families of origin (Fisher et al. Reference Fisher, Theodore, Power, Chisholm, Fuller, Marlowe, Aitchison, Tanna, Joyce, Sacks, Craig and Johnson2008). During this period, families play an active and key role in ensuring the care, well-being and recovery of service users. At the onset, they are often instrumental in initiating treatments and in helping the service user to engage with services and interventions (de Haan et al. Reference de Haan, Peters, Dingemans, Wouters and Linszen2002). However, the early illness phase can be a particularly challenging period for families as they struggle to make sense of the problems faced by their relative, including odd, unusual and embarrassing behaviours that may not always seem to be illness related. Carers spend a lot of time worrying about their relatives’ well-being and future care (Tennakoon et al. Reference Tennakoon, Fannon, Doku, O'Ceallaigh, Soni, Santamaria, Kuipers and Sharma2000; Barker, Lavender & Morant, Reference Barker, Lavender and Morant2001). They often face uncertainty about diagnosis, prognosis and the effectiveness of specific treatments. In addition, families must also adjust quickly to their new and unexpected role as ‘caregiver’, for which they often feel ill equipped and unprepared (Addington & Burnett, Reference Addington, Burnett, Gleeson and McGorry2004; Bergner et al. Reference Bergner, Leiner, Carter, Franz, Thompson and Compton2008). Carers rarely ‘choose’ their new role (Kuipers, Reference Kuipers and Thornicroft2001; Sawatzky & Fowler-Kerry, Reference Sawatzky and Fowler-Kerry2003). Psychosis often has a deleterious effect on the social networks of carers, rendering them isolated and lonely, with feelings of stigmatization, particularly if their relative has been involved in antisocial behaviour (MacInnes, Reference MacInnes, Chaloner and Coffey2000; Ferriter & Huband, Reference Ferriter and Huband2003; Magligano et al. Reference Magliano, Fiorillo, De Rosa, Malangone and Maj2005). It is not surprising, therefore, that research over a 50-year period has shown that carers are negatively affected by their role, with as many as 80% reporting a sense of burden and distress (Magliano et al. Reference Magliano, Marasco, Fiorillo, Malangone, Guarneri and Maj2002; Kuipers & Bebbington, Reference Kuipers, Bebbington, Sartorious, Leff and Lopez2005; Parabiaghi et al. Reference Parabiaghi, Lasalvia, Bonetto, Cristofalo, Marrella, Tansella and Ruggeri2007). Indeed, at least one-third of carers are clinically depressed (Dyck, Short & Vitaliano, Reference Dyck, Short and Vitaliano1999; Kuipers & Raune, Reference Kuipers, Raune, Birchwood, Fowler and Jackson2000; Lowenstein, Butler & Ashcroft, Reference Lowenstein, Butler and Ashcroft2010). Compared with those faced with the effects of more established illnesses, the carers of people attending early psychosis services tend to report equivalent or higher levels of distress and lower levels of positive caregiving experiences (Addington et al. Reference Addington, Coldham, Jones, Ko and Addington2003; Onwumere et al. Reference Onwumere, Kuipers, Bebbington, Dunn, Fowler, Freeman, Watson and Garety2008). Such carers often report levels of loss and grief similar to those seen in physical bereavement (Patterson, Birchwood & Cochrane, Reference Patterson, Birchwood and Cochrane2005). Many mourn the loss of their relationship with the service user and of their plans for the future. They report feelings of guilt and blame for various reasons, for example, feeling that they failed to help their relative soon enough, that they missed key signs and symptoms indicative of illness, and even that they played a role in causing the illness (McCann et al. 2009). Sadly, as part of their role, many carers experience verbal and physical aggression (Loughland et al. Reference Loughland, Lawrence, Allen, Hunter, Lewin, Oud and Carr2009). Almost 25% of homicides occur during the first episode, with close family members, particularly females, most likely to be victims (Nordstrom & Kullgren, Reference Nordstrom and Kullgren2003a, Reference Nordstrom and Kullgrenb; Nielssen & Large, Reference Nielssen and Large2010). Approximately one-third of carers report reactions indicative of post traumatic stress disorder (PTSD) (Barton & Jackson, Reference Barton and Jackson2008; Loughland et al. Reference Loughland, Lawrence, Allen, Hunter, Lewin, Oud and Carr2009). We know that early psychosis carers are likely to have limited previous experience of mental health. One consequence is that carers often engage in less adaptive (avoidant) styles of coping, in themselves, associated with impact of care and poorer outcomes (Raune, Kuipers & Bebbington. Reference Raune, Kuipers and Bebbington2004; Onwumere et al. in press). Moreover, indices of strained family relations, identified in terms of high-expressed emotion, are already present in at least 50% of first episode carers (Bachmann et al. Reference Bachmann, Bottmer, Jacob, Klaua-Thomas, Backentsrass, Mund, Renneberg, Fieldler and Schroder2002; Raune et al. Reference Raune, Kuipers and Bebbington2004). In summary, service users and their families have considerable and varied needs during the early phases.

Family interventions

Given the ubiquitous negative impact of psychosis on service users and their families, and the evidence for a critical early period, international clinical guidelines recommend family-focused interventions during the early illness phases (International Clinical Practice Guidelines for Early Psychosis, 2005), as delivered by specialist early intervention teams such as the flagship services in Melbourne, Birmingham, London and Calgary. The exact format and approach varies, but evidence-based family interventions include psychoeducation, stress reduction, emotional processing, cognitive reappraisal and structured problem solving. These are designed to support family carers in their role, improve adaptive coping and minimize the risk of relapse and readmission (Falloon, Boyd & McGill, Reference Falloon, Boyd and McGill1984; Barrowclough & Tarrier, Reference Barrowclough and Tarrier1992; Kuipers, Leff & Lam, Reference Kuipers, Leff and Lam2002; Addington et al. Reference Addington, Collins, McCleery and Addington2005; Bertolote & McGorry, Reference Bertolote and McGorry2005).

Overall, the evidence for the efficacy of family interventions is strong, with more than 50 controlled trials that have assessed service user outcomes (Cuijpers, Reference Cuijpers1999; Pilling et al. Reference Pilling, Bebbington, Garety, Kuipers, Geddes and Martindale2002; NICE Schizophrenia Update, 2009; Pharoah et al. Reference Pharoah, Mari, Rathbone and Wong2010, 2009). Family interventions can be cost effective (Mihalopoulos et al. Reference Mihalopoulos, Magnus, Carter and Vos2004). They significantly reduce rates of readmission and relapse in service users, and improve their social functioning (Pharoah et al. Reference Pharoah, Mari, Rathbone and Wong2010). The interventions have been linked to improved adherence with pharmacological interventions (Mari & Streiner, Reference Mari and Striener1994; Pilling et al. Reference Pilling, Bebbington, Garety, Kuipers, Geddes and Martindale2002). In terms of carer outcomes, we also know that family interventions improve the experience of care and the commitment to the caregiving role (Berglund, Vahlne & Edman, Reference Berglund, Vahlne and Edman2003; Giron et al. Reference Giron, Fernandez-Yanez, Maná-Alvarenga, Molina-Habas, Nolasco and Gomez-Beneyto2010).

It is clear that family intervention is effective. However, very few of the controlled trials have involved patients in the early stages of psychosis, and results have so far been equivocal. Moreover, where studies have been undertaken, family interventions have not been assessed independently. Thus, they have tended to be evaluated as part of an integrated approach to early intervention, making it difficult to assess the specific contribution of family intervention to service user and carer outcomes (e.g. Grawe et al. Reference Grawe, Falloon, Widen and Skogvoll2006). In a widely cited study, Linszen et al. (Reference Linszen, Dingemans, VanderDoes, Nugter, Scholte, Lenior and Goldstein1996) examined 76 first episode families in receipt of individual psychosocial interventions. They recorded no benefits on relapse rates in families who received an additional 18 sessions of a year-long behavioural family intervention. Disappointingly, families with low expressed emotion (EE) actually became worse following the intervention. However, 5-year follow-up data indicated that family interventions reduced the length of time service users spent in hospital when compared with the standard individual interventions (Lenior, Dingemans & Linszen, Reference Lenior, Dingemans and Linszen2001). In 106 families, Leavey et al. (Reference Leavey, Gulamhussein, Papadopoulos, Johnson-Sabine, Blizard and King2004) compared a brief, seven-session family intervention with standard care. The authors observed no differences between the two groups in readmission rates and carer satisfaction. In a study covering only male service users, Zhang et al. (Reference Zhang, Wang, Li and Phillips1994) recorded positive effects of family interventions on relapse rates and social functioning over an 18-month follow-up, when compared with standard outpatient provision. Interestingly, a recent meta-analysis and systematic review demonstrated that family interventions in early psychosis significantly reduce relapse and readmission rates (Bird et al. Reference Bird, Premkumar, Kendall, Whittington, Mitchell and Kuipers2010).

As there are still relatively few well-controlled randomized trials of family intervention in early psychosis, there is a clear need for additional studies (Askey et al. Reference Askey, Gamble and Gray2007, Reference Askey, Holmshaw, Gamble and Gray2009; Alvarez-Jimenez et al. Reference Alvarez-Jimenez, Parker, Hetrick, McGorry and Gleeson2009). Further work is also required to identify the key mediators of change, and to determine the most effective format and timing of interventions. It is unclear as to whether concepts relevant to longer-term carers have similar weight in early psychosis. High EE, for example, has proved a useful framework for understanding relationships and is a robust predictor of poor functioning and relapse in longer-term groups (Bebbington & Kuipers, Reference Bebbington and Kuipers1994). It has therefore been an important target for family interventions (Kuipers et al. Reference Kuipers, Leff and Lam2002). However, the role and predictive qualities of EE have been less clearly established in early psychosis (Butzlaff & Hooley Reference Butzlaff and Hooley1998). Recent evidence from Gleeson and colleagues suggest that following positive symptom remission, effective interventions offer a combination of individual cognitive behaviour therapy (CBT) for service users and family work for carers, and specifically target relapse prevention (Gleeson et al. 2009). However, there is a paucity of literature on whether interventions are more effective when offered to single families, to multiple families or to a combination.

A cognitive model of caregiving in psychosis

In our recent cognitive model of caregiving, we described the main types of caregiving relationships in psychosis and the proposed family interventions (Kuipers, Onwumere & Bebbington, Reference Kuipers, Onwumere and Bebbington2010). In a climate of limited resources and of training and implementation difficulties with clinicians (Fadden, Reference Fadden2006, Kuipers, Reference Kuipers2007), it is important that services have clear methods for identifying those in need of family interventions in early psychosis. However, evidence on how families successfully adapt following the first episode remains limited (Gleeson et al. Reference Gleeson, Cotton, Alvarez-Jimenez, Wade, Gee, Crisp, Pearce, Newman, Spiliotacopoulos, Castle and McGorry2010).

Triaged interventions?

Our clinical observations would suggest that, similar to longer-term groups, not all early psychosis families need full family intervention, particularly as we know that offering intensive interventions when they are not indicated may be harmful (Linszen et al. Reference Linszen, Dingemans, VanderDoes, Nugter, Scholte, Lenior and Goldstein1996). Given the range of issues they report, it seems likely that some early psychosis families might require only low-intensity interventions. These might offer clear and flexible opportunities for psychoeducation and social support. In the future, they might also use internet facilities to enhance the information provided (e.g. mentalhealthcare.org.uk – a website for carers and friends of those with psychosis; Rotondi et al. Reference Rotondi, Anderson, Haas, Eack, Spring, Ganguli, Newhill and Rosenstock2010). In contrast, intensive interventions are probably most suitable for families where there were previous as well as ongoing difficult relationships or when service users have persisting symptoms or frequent relapses or when carers have complicated grief reactions (Kuipers et al. Reference Kuipers, Onwumere and Bebbington2010). We have argued that it may well be helpful to offer carers an individualized support service in their own right, as they are a seriously neglected group with needs of their own (Kuipers Reference Kuipers2010).