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The predictive validity of Expressed Emotions (EE) in schizophrenia. A 20-year prospective study
Journal of Psychiatric Research 47 (2013) 208e214
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The predictive validity of Expressed Emotions (EE) in schizophrenia. A 20-year prospective studyq ska b, Igor Hanuszkiewicz b, Artur Daren b Andrzej Cechnicki a, *, Anna Bielan a b
Community Psychiatry Unit, Chair of Psychiatry, Collegium Medicum, Jagiellonian University, 2/8 Sikorskiego sq., 31-115 Cracow, Poland Association for the Development of Psychiatry and Community Care, Cracow, Poland
a r t i c l e i n f o
a b s t r a c t
Article history: Received 20 July 2012 Received in revised form 10 October 2012 Accepted 12 October 2012
Background: The level of expressed emotions in the family (EE) and components thereof (emotional overinvolvement EOI, and critical comments CC) have been found to be related to treatment outcomes in psychotic patients. One point of interest is the dynamic of various outcomes in relation to initial EE, CC and EOI levels. Method: 43 individuals with diagnosis of schizophrenia were assessed at 1, 3, 7, 12 and 20-year followups. The EE indicator was evaluated during the index hospitalization. Outcome indicators included: number of relapses, number and duration of in-patient rehospitalizations, and severity of psychopathological symptoms. Results: 1) A high EE indicator was associated with a higher number of relapses in every follow-up and differences between the groups with low and high EE indicators proved significant after 3, 7, 12 and 20 years of the illness. 2) A high EE indicator was associated with a higher number of rehospitalizations after 3 and 7 years, and showed as a clear trend after 12 and 20 years. 3) A high level of CC was associated with a higher number of relapses. 4) The severity of positive symptoms increased only in the group with high CC. 5) The negative syndrome was not associated with EE, EOI or CC. Conclusions: The EE indicator may be considered a valid prolonged predictor of relapses and rehospitalizations. A high level of CC was associated with a higher number of relapses and intensification of the positive syndrome in both the short-term and long-term course of the illness. Ó 2012 Elsevier Ltd. All rights reserved.
Keywords: Schizophrenia Expressed Emotions Long-term follow-up
1. Introduction Over the course of schizophrenia complicated relationships are observed between the course of the illness and the patient’s family environment. Over 30 years ago Vaughn and Leff (1976), building on the original work by Brown and Rutter (1966), compiled a description of the character and intensity of emotions expressed toward the suffering family member by their immediate relatives. They developed an indicator of expressed emotion in the family (EE), put forward a means of measuring it, and demonstrated its link with the course of the illness. Since that time, numerous replication studies have confirmed a high level of EE in over half of key relationships between patients and relatives. This has been observed in a variety of cultural and social contexts (Bebbington and Kuipers, 1994; Leff, 1998; Butzlaff and Hooley, 1998; Rostworowska, 1999; Lopez, 2004; Nomura, q This paper originated with Cracow Schizophrenia Research Group. * Corresponding author. Tel.: þ48 124215117. E-mail address: [email protected] (A. Cechnicki). 0022-3956/$ e see front matter Ó 2012 Elsevier Ltd. All rights reserved. http://dx.doi.org/10.1016/j.jpsychires.2012.10.004
2005; Hopper et al., 2007) including psychiatric staff members (Moore et al., 1992; Willets and Leff, 1997; Barrowclough, 2001; Humbeeck and Audenhove, 2003; Dennis and Leach, 2007; Berry et al., 2011) and in relation to a range of mental and somatic disorders (Butzlaff and Hooley, 1998). One very large group of studies addresses the influence of therapeutic intervention to reduce EE, in particular CC, and the associated therapeutic benefits (e.g. Tarrier et al., 1994; Bäuml et al., 1996; Linszen et al., 1997; Leff, 1998, and others). One issue of significance for comparison of the results is training for researchers in the method and tools (CFI and FMSS) used; others are assessment of the EE components (CC and EOI), changes in their levels over time, the criteria for qualification into groups (incidence vs. prevalence), observation time, and the criteria of course and outcome adopted. The EE index is a complex construct. Various relationships have been investigated between the course and outcome of the illness on the one hand and EE and its two components, criticism (CC) and emotional over-involvement (EOI), on the other (e.g. Vaughn and Leff, 1976; Ivanovi c et al., 1994; Schulze-Mönking et al., 1997;
A. Cechnicki et al. / Journal of Psychiatric Research 47 (2013) 208e214
King, 2000; Raune et al., 2004; Marom et al., 2005; Docherty et al., 2011; Singh et al., 2011). Most of these studies treat EE as a single construct, but even the earliest of them, Vaughn and Leff (1976), pointed to a link between high EE and high levels of CC. Marom et al. (2005) used the Five Minute Speech Sample (FMSS) in a study of 151 relatives, 93 patients with a schizophrenia diagnosis and 15 with a diagnosis of schizoaffective disorders. After seven years high CC was associated with a higher rate of readmissions and longer hospital stays than low CC. Similar results over an eight-year period of observation were obtained in a previous study by Schulze-Mönking et al. (1997) using the CFI. Kuipers et al. (1992) in their Practical Guide noticed that carers fail to understand that the negative symptoms are part of the illness, and blame the patients for being lazy. Similarly, Alvarez-Jimenez et al. (2008) construed CC as a reaction by the family associated with attributing the cause of the disease to the patient. Docherty et al. (2011) found an association between CC and intensification of anxiety in patients. Raune et al. (2004) found an association between a high level of EOI and number of relapses, while an average level of EOI could have a protective effect. In the study by King (2000), EOI in patients’ mothers assessed at the start of treatment was associated with a lower level of hostility and better cooperation by patients 18 months after the onset of the illness. One important group of studies addresses assessment of the change in EE. Kavanagh (1992) observed that CC is less stable than EOI. McCreadie et al. (1993), using the CFI, assessed EE three times over a period of five years in 32 families with a prevalence of patients with a schizophrenia diagnosis. EE was high at all three assessments in 25% of relatives, low at all three in 38%, and fluctuated in 38%; that is, in the majority of relatives (63%) the level of EE was stable over time. Lenior et al. (2002) in their nine-year observation using the FMSS found that the level of EE changed over time. They also noted a rise in EOI in successive years. However, these results should be interpreted with caution, as the FMSS is not as reliable as CFI, especially regarding EOI. Most of the research into EE has been short-term studies lasting between 9 and 24 months, e.g. Ivanovi c et al. (1994), Linszen et al. (1997), Macmillan et al. (1987) e two years and Patterson et al. (2005). Among the longer-term studies of the impact of EE households on the course of schizophrenia over many years are McCreadie et al. (five years, 1993), Tarrier et al. (five and eight years, 1994), Schulze-Mönking et al. (eight years, 1997), Lenior et al. (nine years, 2002), Marom et al. (seven years, 2005), and Cechnicki et al. (12 years, 2010). A number of studies have demonstrated that high EE in the families of patients suffering from schizophrenia is associated with a worse course of the disease in terms of a higher number of relapses (Vaughn et al., 1984; Tanaka et al., 1995; Ronald et al., 1998), rehospitalization and time spent in hospital (Marom et al., 2005), and severity of symptoms (Schulze-Mönking et al., 1997; Cechnicki et al., 2010). McCreadie et al. (1993) demonstrated that patients living with low-EE families had fewer relapses than those with a high or fluctuating EE. A few patients in consistently high-EE homes had no relapses over a five-year period, but the small size of the group dictates caution in interpretation of the results. Ronald et al. (1998) observed a possible stronger association between EE and severity of relapses in patients with a more chronic course of the illness. Ivanovi c et al. (1994) find the number of relapses to be as much as 10 times higher in the high-EE group. The Cracow-based prospective study of EE assessed using the CFI showed an association between high EE and a greater number of relapses over a 12-year follow-up period from index hospitalization (Cechnicki et al., 2010). Only Schulze-Mönking et al. (1997) observed that patients with a high EE obtained higher scores in terms of intensification of
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symptoms measured on the GAS scale, while Cechnicki et al. (2010) found differences between the two groups only during the index hospitalization, and these were of borderline statistical significance. The meta-analysis by Pharoah et al. (2000) finds no association between EE and intensification of symptoms, while Klingberg (2002) concludes: “High-EE behavior does not seem to cause symptoms”. The majority of the works cited above, however, confirmed the predictive value of EE, as well as CC, for the course of schizophrenia and, in particular, relapses of psychosis. 2. Study objective This study is part of the Cracow-based prospective study into the course of schizophrenia, which aims to observe schizophrenia patients for many years, from their first psychotic episode through the years of their life with the illness, and assess the outcomes of their treatment and prognostic factors. The objectives of this paper are to assess whether the EE indicator as an overall construct and its two major components, CC and EOI, measured in an incidence group of patients during their index hospitalization, remain associated with the course of their schizophrenia over a period of 20 years, and to examine their prognostic value for selected treatment outcome factors. The treatment outcome criteria used were number of relapses, number and duration of in-patient rehospitalizations, and severity of psychopathological symptoms measured using the BPRS-E scale in direct clinical examinations at the successive 3-, 7-, 12- and 20-year catamnesis post index hospitalization. 3. Subjects The original study group comprised of 80 people diagnosed in accordance with DSM III (and later rediagnosed in accordance with DSM IV) criteria for schizophrenia, who were consecutively admitted to the Krakow Psychiatry Clinic for Adults of the University Hospital. The selection criteria were following: 18 or more years old, residential status in Kraków, living with family of orientation or/and procreation. We did not include youths as they are treated at a separate clinic. Of those 80 participants 8 were not assessed with CFI as they refused to participate in the recording of their CFI interview. 29 of the remaining 72 subjects were not included, as they did not attend one, or sometimes more, of the five follow-up assessments we included in the analysis because of refusals to participate, change of the place of residence, death by somatic causes or by suicide within the 20 years period (Cechnicki, 2011). As their data were not complete we had to drop them from the analysis, which left us with 43 complete assessments. Over the 20-year period about half of subjects, both in the high EE and the low EE group, have changed their co-habitants (6 out of 12 subjects with low EE and 15 out of 31 subjects with high EE). The sociodemographic data of this group is presented in Table 1. All subjects’ data were collected in ambulatory setting, with wholly voluntary participation. At each assessment an informed consent was sought from the patients and their family members. They were informed of the objective, methods and the course of the assessment. The study has been approved by the Bioethical Committee of the Collegium Medicum, Jagiellonian University, Cracow. 4. Method The level of EE indicator was measured using the Camberwell Family Interview (CFI) tool. The interviews were rated by a researcher certified in the method by C. Vaughn. The interviewees
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Table 1 Demographic factors (group n ¼ 43) divided according to EE level. EE level
Age at first symptoms (years) Age at index hospitalization (years)
Mean
Standard dev.
Min.
max.
High
Low
High
Low
High
Low
High
Low
26.6 27.5
27.3 28.3
6.50 5.98
5.26 6.22
13 19
20 20
39 39
37 43
High
Low
High
Low
18 13 10 21 8 4 10 7 2 16 12 3 11 20 14 17 22 9 20 11 8 1 22 0 0 0 13 2 9 6 0 1
7 5 5 7 4 0 7 1 0 10 1 1 12 n/a 12 n/a 8 4 7 5 3 1 8 0 0 0 7 1 3 1 0 0
58.1 41.9 32.3 67.7 25.8 12.9 32.3 22.6 6.5 51.6 38.7 9.7 35.5 64.5 45.2 54.8 71.0 29.0 64.5 35.5 25.8 3.2 71.0 0.0 0.0 0.0 41.9 6.5 29.0 19.4 0.0 3.2
58.3 41.7 41.7 58.3 33.3 0.0 58.3 8.3 0.0 83.3 8.3 8.3 100 n/a 100 n/a 66.7 33.3 58.3 41.7 25.0 8.33 66.7 0.0 0.0 0.0 58.3 8.3 25.0 8.3 0.0 0.0
EE level
Gender Marital status Education
Work
EE e critical comments (CC) EE e emotional over-involvement (EOI) Beginning of illness Duration of untreated psychosis (DUP) Co-habitants of the patients at the beginning of the study
Co-habitants of the patients at 20-year follow-up
Incidence
Women Men Marital relationship Single Higher (Master level) Higher (Bachelor level) Secondary Vocational Primary In full-time work or study On sick leave (from work or study) Not working Absent Present Absent Present Chronic Acute Under 6 months Over 6 months Family of procreation Family of procreation & orient Family of orientation Without relatives Sheltered housing Social care home Family of procreation Family of procreation & orient Family of orientation Without relatives Sheltered housing Social care home
were parents (one or both) or/and spouses of the patients. All relatives living in a household with a patient were interviewed, however, a single high EE relative was sufficient to consider the entire household as high EE. EE was rated as high following the standard criteria of the EE evaluation, that is, a “high” result in any of the three components: EOI, CC or hostility. EOI was assessed based on a global assessment of the whole interview, on a scale of 0e5 points. A score of 3 or more is considered to indicate high level of emotional over-involvement. CC was assessed based on the number of critical remarks by a relative, where 6 or more remarks are considered to indicate high level of criticism. Hostility was assessed on a scale of 0e3 points, with 1 or more points indicating high level of hostility. Other EE components (warmth and positive remarks) were of no significance in assessing the family as low or high EE. The severity of symptoms was measured using the UCLAmodified BPRS-E (Lukoff et al., 1986). A relapse was defined as such a patient’s condition which requires a BPRS-E rating of 6 or 7 (on a 1e7 scale) in at least one of the positive or negative symptoms. This relapse measure was adopted from Lukoff et al. (1986). Treatment outcome data were gathered at successive catamneses at 1, 3, 7, 12 and 20 years after index hospitalization using an adaptation of the Alanen and Rekkolainen Anamnestic and Catamnestic Interview (Cechnicki, 2011). In the first set of analyses EE served as the independent between-group variable with two levels: high (any one or combination of the following: hostility, CC, EOI) and low (none of the above). In successive analyses the independent between-group
Percentage
variables were the dichotomous EOI indicator and the dichotomous CC indicator. Time was an independent variable with repeated assessments at successive follow-ups. Five points of measurement were analyzed e at the 1-, 3-, 7-, 12- and 20-year follow-ups. The indicators of treatment outcome (dependent variables) selected were number of relapses, number of rehospitalizations, duration of rehospitalizations (weeks), and severity of symptoms as per BPRS e overall result, BPRS e positive syndrome, and BPRS e negative syndrome. The independent variables formed a two-factor research plan with one between-group factor (EE, EOI or CC) and one repeated measurement factor (time). Potential differences were explored using the repeated measures analysis of variance. 5. Study results A preliminary comparison of the mean number of relapses was made for high-EE and low-EE groups, separately for each follow-up. This enabled accounting for the different numbers of people attending each follow-up, and, in effect, show between-group comparisons for relatively more numerous, although somewhat differing, samples. Low-EE groups evince a lower average number of relapses in case of every follow-up except one year after index hospitalization (Fig. 1). These results indicate possible relationships over time and in order to investigate them our next step was to properly analyze the 43 people who attended all five follow-ups. The indicators
A. Cechnicki et al. / Journal of Psychiatric Research 47 (2013) 208e214
Fig. 1. Mean numbers of relapses between index hospitalization and assessment at the given follow-up points showing division into low-EE and high-EE groups (significance levels of the differences in the U ManneWhitney test at successive follow-up points: p ¼ 0.240, p ¼ 0.002, p ¼ 0.007, p ¼ 0.042, p ¼ 0.024).
examined were changes over time in number of relapses, number of rehospitalizations, duration of rehospitalizations, and severity of symptoms measured using the BPRS, as well as interaction of these changes with EE level. Across the study group, irrespective of EE level, numbers of relapses and rehospitalizations, and duration of rehospitalizations increased in successive years, which was expected, as these data are additive. Parallel to this, a stabilization of BPRS-measured psychopathological symptoms was observed in the early catamneses, followed by slight intensification at 12- and 20-year assessments. Of major interest to us were interaction effects of repeated measures and the level of expressed emotions, as well as its components, which now follow. A significant interaction was found between EE level and change in relapses (F(4, 164) ¼ 5.02; p < 0.001) and rehospitalizations (F(4, 164) ¼ 2.85; p ¼ 0.025). The graphs below show the detailed differences at successive followups (Figs. 2 and 3). The differences in number of relapses between the two groups at the various follow-up points proved significant for the measurements at 3 (p ¼ 0.010), 7 (p ¼ 0.009), 12 (p ¼ 0.025) and 20 (p ¼ 0.011) years. Rises in numbers of relapses were higher in successive years in the high-EE group (p < 0.001 for differences between all the successive follow-up points) than in the low-EE
Fig. 2. Relationship between changes in number of relapses over time and EE level.
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Fig. 3. Relationship between changes in number of rehospitalizations over time and EE level.
group, where differences were statistically insignificant except for that between the first and 12th year of illness (p ¼ 0.42) and the third and 12th year of illness (p ¼ 0.046). Changes in number of rehospitalizations demonstrated similar association with EE level. The differences between the groups at each catamnesis were significant at three (p ¼ 0.020) and seven (p ¼ 0.023) years, and showed as a clear trend at 12 (p ¼ 0.055) and 20 (p ¼ 0.056) years. In the high-EE group results were significantly different at each successive follow-up (p ¼ 0.002 or lower), while in the low-EE group all the differences between follow-ups were insignificant (Fig. 3). In the case of the duration of rehospitalizations and severity of symptoms measured using the BPRS, the interactions with EE level were not statistically significant. 6. EOI and CC in relation to treatment outcomes The next step in our study was to examine the changes in number of relapses, number of rehospitalizations, duration of rehospitalizations and severity of symptoms measured using the BPRS and the relationship between change over time (at follow-up points) and levels of the EE constituents: CC and EOI (Figs. 4 and 5). No significant interaction was observed between the time of measurement and the level of EOI for any of the six outcome indicators. In the case of interaction between the time of measurement and the level of CC, however, two significant interaction effects were noted. In the first of them, the number of relapses rose higher in the high-CC group (F(4, 164) ¼ 4.11; p ¼ 0.003) (Fig. 4). The differences between the groups at the individual points of measurement were not statistically significant, but a trend was noted after 12 (p ¼ 0.069) and 20 (p ¼ 0.051) years. Both in the group with a high level of CC (p 0.001) and that with a low level of this indicator (p 0.021) the differences between numbers of relapses at successive catamneses were statistically significant. The second interaction was observed between CC and changes in the severity of positive symptoms (F(4, 164) ¼ 3.32; p ¼ 0.012) (Fig. 5). In the group in which a low level of CC was observed, positive symptoms did not change significantly over time. In the high-CC group, however, positive symptoms intensified over time, and statistically significant differences were noted between the 20-year follow-up and all the previous follow-ups (p 0.014). No
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A. Cechnicki et al. / Journal of Psychiatric Research 47 (2013) 208e214
Fig. 4. Relationship between changes in number of relapses over time and CC level.
statistically significant differences in severity of positive symptoms occurred at the various follow-up points between the high-CC and low-CC groups, though a borderline trend-level difference was observed after 20 years (p ¼ 0.100). 7. Discussion The results obtained in the present prospective study permit to interpret EE, assessed in the second week of the index hospitalization, as a specific predictor of early and long-term treatment outcomes. The associations observed relate to several indicators of the course of schizophrenia: relapses, rehospitalization, and severity of positive symptoms. A particularly important role is played by the CC component of EE. The majority of studies of EE confirm its predictive value, and the largest number of reports in the literature pertains to the dependence between EE and relapses. The Cracow study confirmed the association between EE and relapses in the short-term course of schizophrenia, and this outcome is concordant with studies including those by Kavanagh (1992), Ivanovi c et al. (1994), Linszen et al. (1997), Ronald et al. (1998), Rostworowska (1999), King
Fig. 5. Relationship between changes in the severity of positive symptoms over time (measured with BPRS) and CC level.
(2000), Patterson et al. (2005). Moreover, in long-term catamneses this association was also confirmed, as in McCreadie et al. (1993), Tarrier et al. (1994), Schulze-Mönking et al. (1997), Lenior et al. (2002), Cechnicki et al. (2010). A major new finding of the present study was that the difference in numbers of relapses between the high-EE and low-EE groups was sustained over 20 years. Mean numbers of relapses reach significant levels of difference at 3, 7, 12 and 20 years from index hospitalization. The study by Raune et al. (2004) offered an analysis of the complexity of issues surrounding the components of EE. High EOI proved to be related to an adverse impact on numbers of relapses, while an average EOI could have a protective effect. This is because moderate levels of EOI are associated with high warmth, while warmth is lower at high levels of EOI. Similar results were obtained by Breitborde et al. (2007), who also found that high level of warmth “appear to exert a protective effect on the course of illness”. In the present study both the high-CC and the low-CC groups evinced a statistically significant increase in numbers of relapses at successive follow-ups. Differences between the groups at the various points were not statistically significant, but a rising trend was observed at 12 and 20 years. As in the studies by Schulze-Mönking et al. (1997) and Marom et al. (2005), in the Cracow study the high EE proved to have a significant association with rehospitalizations at three and seven years, which was still visible at the trend level at 12 and 20 years. However, in contrast to those studies, there was no visible association between EE and duration of rehospitalizations, probably because the patients in Cracow were taking part in a psychosocial treatment program specifically aimed at reducing time in hospital. Rehospitalization and its duration is more reliable to record as an indicator of the course of the illness, but is dependent on many local circumstances, so should be taken in the context of other dimensions of treatment. Neither was confirmation obtained of the outcome found by Marom et al. (2005) that a high level of CC as a component of EE is associated with more frequent and longer rehospitalizations. Relatively few scholars have focused on the relationships between EE and severity of symptoms. In the meta-analysis by Pharoah et al. (2000) no association was observed between EE and symptom severity. On the other hand, in their psychophysiological studies, Sturgeon et al. (1984) found that patients had a higher arousal level in the presence of a high EE relative than when they were with a low EE relative. Woo et al. (2004) describe an association between EE and the symptoms of hostility, anxiety and arousal. Docherty et al. (2011) explored the relationship between CC and anxiety. Patients with high CC and a higher degree of anxiety had more severe symptoms at nine months from index hospitalization than those with low CC. Schulze-Mönking et al. (1997) also found a connection between EE and symptoms measured using the GAS, both at the start of treatment and after eight years. In the Cracow study CC measured at the onset of the illness is associated with intensification of the positive syndrome over 20 years of the illness. A significant difference was noted between the measurement at 20 years and all the previous measurements in the high-CC group, in which a gradual increase in severity of symptoms was observed over time. The study does not answer the question of whether high CC in the family is a cause of or a response to the psychopathology of the sickness of one of its members. Lenior et al. (2002) found instability in both overall EE and CC and EOI over nine years of observation, but as McCreadie (1993) noted, EE during index hospitalization is a 60% predictor of future EE, which may partly explain why in the Cracow study EE and CC levels measured at index hospitalization are connected to treatment results after 20 years. As a complex construct (Ronald et al.,
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1998), EE comprises relatively stable variables related to both personality (Hooley and Hiller, 2000) and circumstances, i.e. both reactive and temporary (King, 2000). Owing to this complexity, the issue of the stability of the construct is one for which it is hard to offer verification and theoretical justification. King (2000) found a dependence suggesting that both EOI and CC may be effects rather than causes of intensification of symptoms. Alvarez-Jimenez et al. (2008) indicate that CC may be a reaction by the family linked with attributing to the patient causality of the disease and a lack of understanding of the problem. Woo et al. (2004) moot bidirectional dependence between EE, the family and the patient’s psychopathology. As has been confirmed in many studies, lowering EE definitely reduces the relapse rate so that there is a strong vector between relatives’ EE and the patient’s vulnerability to relapse (Tarrier et al., 1994; Bäuml et al., 1996; Linszen et al., 1997; Leff, 1998, and others). Intervention designed to reduce CC in the family is thus a vital standard in treatment of schizophrenia, in particular in the context of the conclusions of Cutting et al. (2006), who studied patients’ perception of family behaviors. Their observations improve understanding of the processes in the family as they demonstrate that CC is stressful for patients because they perceive it clearly. An analysis of our results paired with the knowledge gathered from clinical research to date seem inviting to formulate a hypothesis explaining long-term effect of EE on treatment outcomes, in particular relapses and positive symptoms. One possibility is to think of family relations as being internalized by the patient many years before the onset of the illness. This may result in e.g. developing an overly critical self-attitude. Combined with a critical and rejecting reaction of their relatives at the onset of the illness, this situation renders the patient continuously subjected to intensive negative emotions, which may result in an unfavorable course of the illness. Constructing research models that would help to differentiate between more and less variable aspects of EE, as well as between variables dependent on the family and those dependent on the illness, would help to gain better insight into the nature of this phenomenon, which presents as both a predictor and an effect of the course of the illness. Irrespective of the outcomes of future studies, there is a need for effective therapeutic interventions focused on improving communication and relationships within the families of schizophrenia sufferers, and in particular on reducing CC. 8. Conclusions 1. EE level as measured at index hospitalization may be seen as a specific predictor of relapse and rehospitalization in the group under study over 20 years of the course of schizophrenia. 2. High CC is associated with intensification of the positive syndrome in the course of the illness.
Role of the funding source The research presented in the attached manuscript was founded from the research grant no. K/ZDS 000652 e an internal statutory grant of the Chair of Psychiatry, Collegium Medicum, Jagiellonian University in Cracow, Poland. Contributions Andrzej Cechnicki (AC) Anna Bielanska (AB) Igor Hanuszkiewicz (IH) Artur Daren (AD)
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AC designed the study and coordinated subsequent assessments, AC, AB wrote the first draft, AC, AB, IH managed the literature searches, analyses and discussion, AD and IH undertook statistical analysis. All authors participated in preparation and have approved the final version of the paper. Declaration of interest All four authors declare that there are no conflicts of interest regarding the submitted paper. Acknowledgments We would like to thank Mr Konrad Wronski, who kindly assisted with preparation of the figures, and Miss Joanna Palka, who assisted with the preparation of literature and proof-reading of this paper. References Alvarez-Jimenez M, Gleeson JF, Cotton SM, Wade D, Crisp K, Yap MBH. Differential predictors of critical comments and emotional over-involvement in firstepisode psychosis. Psychological Medicine 2008;40:63e72. Barrowclough C. Staff expressed emotion and causal attributions for client problems on a low security unit: an exploratory study. Schizophrenia Bulletin 2001; 27:517e26. Bäuml J, Pitschel-Walz G, Kissling W. Psychoedukative Gruppen bei schizophrenen Psychosen für Patienten und Angehörige. In: Stark A, editor. Verhaltenstherapeutische und psychoedukative Ansätze im Umgang mit schizophren Erkrankten. Tübingen: dgvt-Verlag; 1996. p. 217e55. Bebbington P, Kuipers L. The predictive utility of expressed emotion in schizophrenia: an aggregate analysis. Psychological Medicine 1994;24:707e18. Berry K, Barrowclough C, Haddock G. The role of expressed emotion in relationships between psychiatric staff and people with a diagnosis of psychosis: a review of the literature. Schizophrenia Bulletin 2011;37:958e72. Breitborde N, Lopez S, Wickens D, Jenkins J, Karno M. Toward specifying the nature of the relationship between expressed emotion and schizophrenic relapse: the utility of curvilinear models. International Journal of Methods in Psychiatric Research 2007;16:1e10. Brown GW, Rutter M. The measurement of family activities and relationships: methodological study. Human Relations 1966;19:241e63. Butzlaff RL, Hooley JM. Expressed emotion and psychiatric relapse. A meta-analysis. Archives of General Psychiatry 1998;55:547e52. Cechnicki A, Hanuszkiewicz I, Polczyk R, Cichocki q, Kalisz A, Rostworowska M. Wska znik ekspresji emocji (EE) jako rodzinny predyktor przebiegu schizofrenii [Expressed Emotion indicator as a family predictor of the course of schizophrenia]. Psychiatria Polska 2010;2:173e84. Cechnicki A. Schizofrenia e proces wielowymiarowy. Krakowskie prospektywne badania przebiegu, prognozy i wyników leczenia schizofrenii [Schizophrenia e A multidimensional process. An evaluation of its course, prognosis and longterm treatment results]. Warszawa: Instytut Psychiatrii i Neurologii; 2011. Cutting LP, Aakre JM, Docherty NM. Schizophrenic patients’ perceptions of stress, expressed emotion, and sensitivity to criticism. Schizophrenia Bulletin 2006; 26:1e8. Dennis AM, Leach C. Expressed emotion and burnout: the experience of staff caring for men with learning disability and psychosis in a medium secure setting. Journal of Psychiatric and Mental Health Nursing 2007;14:267e76. Docherty NM, St-Hilaire A, Aakre JM, Seghers JP, McCleery A, Divilbiss M. Anxiety interacts with expressed emotion criticism in the prediction of psychotic symptom exacerbation. Schizophrenia Bulletin 2011;37:611e8. Hooley JM, Hiller JB. Personality and expressed emotion. Journal of Abnormal Psychology 2000;109:40e4. Hopper K, Harrison G, Janca A, Sartorius N. Recovery from schizophrenia: an international perspective. A report from the WHO Collaborative Project, the international study of schizophrenia. Oxford: Oxford University Press; 2007. p. 277e285. Humbeeck GV, Audenhove CV. Expressed emotion of professionals towards mental health patients. Epidemiologia e Psichiatria Sociale 2003;12:232e7. Ivanovi c M, Vuletib Z, Bebbington R. Expressed emotion in the families of patients with schizophrenia and its influence on the course of illness. Social Psychiatry and Psychiatric Epidemiology 1994;29:61e5. Kavanagh DJ. Recent developments in expressed emotion and schizophrenia. British Journal of Psychiatry 1992;160:601e20. King S. Is expressed emotion cause or effect in the mothers of schizophrenic young adults? Schizophrenia Research 2000;45:65e78. Klingberg S. Family interventions: empirical evidence of efficacy and open questions. In: Häfner H, editor. Risk and protective factors in schizophrenia. Heidelberg: Springer; 2002. p. 285e93. Kuipers L, Leff J, Lam D. Family work for schizophrenia: a practical guide. Gaskell, London: Royal College of Psychiatrists; 1992.
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Leff J. Needs of the families of people with schizophrenia. Advances in Psychiatric Treatment 1998;4:277e84. Lenior M, Dingemans P, Schene A, Hart A, Linszen D. The course of parental expressed emotion and psychotic episodes after family intervention in recentonset schizophrenia. A longitudinal study. Schizophrenia Research 2002;57: 183e90. Linszen DH, Dingemans PM, Nugter MA, Van der Does JA, Scholte WF, Lenior MA. Patient attributes and expressed emotion as risk factors for psychotic relapse. Schizophrenia Bulletin 1997;23:119e30. Lopez SR. Ethnicity, expressed emotion, attributions, and course of schizophrenia: family warmth matters. Journal of Abnormal Psychology 2004;113:428e39. Lukoff D, Nuechterlein KH, Ventura J. Manual for the expanded Brief Psychiatric Rating Scale. Schizophrenia Bulletin 1986;13:261e76. Macmillan JF, Crow TJ, Johnson AL, Johnstone EC. Expressed emotion and relapse in first episodes of schizophrenia. British Journal of Psychiatry 1987;151:320e3. Marom S, Munnitz H, Jones PB, Wezman A, Hermesh H. Expressed emotion: relevance to rehospitalization in schizophrenia over 7 years. Schizophrenia Bulletin 2005;31:751e8. McCreadie RG, Robertson LJ, Hall DJ, Berry I. The Nithsdale schizophrenia surveys. XI: relatives’ expressed emotion stability over five years and its relation to relapse. British Journal of Psychiatry 1993;162:393e7. Moore E, Kuipers L, Ball R. Staff-patient relationships in the care of the long-term adult mentally ill. A content analysis of Expressed Emotion interviews. Social Psychiatry and Psychiatric Epidemiology 1992;27:28e34. Nomura H. A cross-cultural study on expressed emotion in carers of people with dementia and schizophrenia: Japan and England. Social Psychiatry and Psychiatric Epidemiology 2005;40:564e70. Patterson P, Birchwood M, Cochrane R. Expressed emotion as an adaptation to loss: prospective study in first-episode psychosis. British Journal of Psychiatry 2005; 187:59e64. Pharoah FM, Mari JJ, Streiner D. Family intervention for schizophrenia (Cochrane review). The Cochrane Library. Oxford: Update Software; 2000 (2).
Raune D, Kuipers E, Bebbington P. Expressed emotion at first-episode psychosis: investigating a carer appraisal model. British Journal of Psychiatry 2004;184: 321e6. Ronald L, Butzlaff AM, Hooley JM. Expressed emotions and psychiatric relapse. A meta-analysis. Archives of General Psychiatry 1998;55:547e52. Rostworowska M. Koncepcja wska znika Ujawnianych Uczu c [The concept of Expressed Emotion indicator]. In: de Barbaro B, editor. Schizofrenia w rodzinie [Schizophrenia in family]. Kraków: Wydawnictwo Uniwersytetu Jagiellon skiego; 1999. p. 51e67. Schulze-Mönking H, Hornung WP, Stricker K, Buchkremer G. Expressed emotion in an 8 year follow-up. European Psychiatry 1997;12:105e10. Singh SP, Harley K, Suhail K. Cultural specificity of emotional overinvolvement: a systematic review. Schizophrenia Bulletin 2011. http://dx.doi.org/10.1093/ schbul/sbr170 (online publication: December 20, 2011). Sturgeon D, Turpin G, Kuipers L, Berkowitz R, Leff J. Psychophysiological responses of schizophrenic patients to high and low expressed emotion relatives: a follow-up study. British Journal of Psychiatry 1984;145:62e9. Tanaka S, Mino Y, Inoue S. Expressed emotion and the course of schizophrenia in Japan. British Journal of Psychiatry 1995;167:794e6. Tarrier N, Barrowclough C, Porceddu K, Fitzpatrick E. The Salford Family Intervention Project: relapse rates of schizophrenia at five and eight years. British Journal of Psychiatry 1994;165:829e32. Vaughn C, Leff J. The influence of family and social factors on the course of psychiatric illness. British Journal of Psychiatry 1976;129:125e37. Vaughn C, Synder K, Jones S, Freeman W, Falkon I. Family factors in schizophrenia relapse: replication in California of British research on expressed emotion. Archives of General Psychiatry 1984;41:1169e77. Willets LE, Leff J. Expressed emotion and schizophrenia: the efficacy of a staff training programme. Journal of Advanced Nursing 1997;26:1125e33. Woo S, Goldstein M, Nuechterlein K. Relatives’ affective style and the expression of subclinical psychopathology in patients with schizophrenia. Family Process 2004;43:233e47.
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The predictive validity of Expressed Emotions (EE) in schizophrenia. A 20-year prospective studyq ska b, Igor Hanuszkiewicz b, Artur Daren b Andrzej Cechnicki a, *, Anna Bielan a b
Community Psychiatry Unit, Chair of Psychiatry, Collegium Medicum, Jagiellonian University, 2/8 Sikorskiego sq., 31-115 Cracow, Poland Association for the Development of Psychiatry and Community Care, Cracow, Poland
a r t i c l e i n f o
a b s t r a c t
Article history: Received 20 July 2012 Received in revised form 10 October 2012 Accepted 12 October 2012
Background: The level of expressed emotions in the family (EE) and components thereof (emotional overinvolvement EOI, and critical comments CC) have been found to be related to treatment outcomes in psychotic patients. One point of interest is the dynamic of various outcomes in relation to initial EE, CC and EOI levels. Method: 43 individuals with diagnosis of schizophrenia were assessed at 1, 3, 7, 12 and 20-year followups. The EE indicator was evaluated during the index hospitalization. Outcome indicators included: number of relapses, number and duration of in-patient rehospitalizations, and severity of psychopathological symptoms. Results: 1) A high EE indicator was associated with a higher number of relapses in every follow-up and differences between the groups with low and high EE indicators proved significant after 3, 7, 12 and 20 years of the illness. 2) A high EE indicator was associated with a higher number of rehospitalizations after 3 and 7 years, and showed as a clear trend after 12 and 20 years. 3) A high level of CC was associated with a higher number of relapses. 4) The severity of positive symptoms increased only in the group with high CC. 5) The negative syndrome was not associated with EE, EOI or CC. Conclusions: The EE indicator may be considered a valid prolonged predictor of relapses and rehospitalizations. A high level of CC was associated with a higher number of relapses and intensification of the positive syndrome in both the short-term and long-term course of the illness. Ó 2012 Elsevier Ltd. All rights reserved.
Keywords: Schizophrenia Expressed Emotions Long-term follow-up
1. Introduction Over the course of schizophrenia complicated relationships are observed between the course of the illness and the patient’s family environment. Over 30 years ago Vaughn and Leff (1976), building on the original work by Brown and Rutter (1966), compiled a description of the character and intensity of emotions expressed toward the suffering family member by their immediate relatives. They developed an indicator of expressed emotion in the family (EE), put forward a means of measuring it, and demonstrated its link with the course of the illness. Since that time, numerous replication studies have confirmed a high level of EE in over half of key relationships between patients and relatives. This has been observed in a variety of cultural and social contexts (Bebbington and Kuipers, 1994; Leff, 1998; Butzlaff and Hooley, 1998; Rostworowska, 1999; Lopez, 2004; Nomura, q This paper originated with Cracow Schizophrenia Research Group. * Corresponding author. Tel.: þ48 124215117. E-mail address: [email protected] (A. Cechnicki). 0022-3956/$ e see front matter Ó 2012 Elsevier Ltd. All rights reserved. http://dx.doi.org/10.1016/j.jpsychires.2012.10.004
2005; Hopper et al., 2007) including psychiatric staff members (Moore et al., 1992; Willets and Leff, 1997; Barrowclough, 2001; Humbeeck and Audenhove, 2003; Dennis and Leach, 2007; Berry et al., 2011) and in relation to a range of mental and somatic disorders (Butzlaff and Hooley, 1998). One very large group of studies addresses the influence of therapeutic intervention to reduce EE, in particular CC, and the associated therapeutic benefits (e.g. Tarrier et al., 1994; Bäuml et al., 1996; Linszen et al., 1997; Leff, 1998, and others). One issue of significance for comparison of the results is training for researchers in the method and tools (CFI and FMSS) used; others are assessment of the EE components (CC and EOI), changes in their levels over time, the criteria for qualification into groups (incidence vs. prevalence), observation time, and the criteria of course and outcome adopted. The EE index is a complex construct. Various relationships have been investigated between the course and outcome of the illness on the one hand and EE and its two components, criticism (CC) and emotional over-involvement (EOI), on the other (e.g. Vaughn and Leff, 1976; Ivanovi c et al., 1994; Schulze-Mönking et al., 1997;
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King, 2000; Raune et al., 2004; Marom et al., 2005; Docherty et al., 2011; Singh et al., 2011). Most of these studies treat EE as a single construct, but even the earliest of them, Vaughn and Leff (1976), pointed to a link between high EE and high levels of CC. Marom et al. (2005) used the Five Minute Speech Sample (FMSS) in a study of 151 relatives, 93 patients with a schizophrenia diagnosis and 15 with a diagnosis of schizoaffective disorders. After seven years high CC was associated with a higher rate of readmissions and longer hospital stays than low CC. Similar results over an eight-year period of observation were obtained in a previous study by Schulze-Mönking et al. (1997) using the CFI. Kuipers et al. (1992) in their Practical Guide noticed that carers fail to understand that the negative symptoms are part of the illness, and blame the patients for being lazy. Similarly, Alvarez-Jimenez et al. (2008) construed CC as a reaction by the family associated with attributing the cause of the disease to the patient. Docherty et al. (2011) found an association between CC and intensification of anxiety in patients. Raune et al. (2004) found an association between a high level of EOI and number of relapses, while an average level of EOI could have a protective effect. In the study by King (2000), EOI in patients’ mothers assessed at the start of treatment was associated with a lower level of hostility and better cooperation by patients 18 months after the onset of the illness. One important group of studies addresses assessment of the change in EE. Kavanagh (1992) observed that CC is less stable than EOI. McCreadie et al. (1993), using the CFI, assessed EE three times over a period of five years in 32 families with a prevalence of patients with a schizophrenia diagnosis. EE was high at all three assessments in 25% of relatives, low at all three in 38%, and fluctuated in 38%; that is, in the majority of relatives (63%) the level of EE was stable over time. Lenior et al. (2002) in their nine-year observation using the FMSS found that the level of EE changed over time. They also noted a rise in EOI in successive years. However, these results should be interpreted with caution, as the FMSS is not as reliable as CFI, especially regarding EOI. Most of the research into EE has been short-term studies lasting between 9 and 24 months, e.g. Ivanovi c et al. (1994), Linszen et al. (1997), Macmillan et al. (1987) e two years and Patterson et al. (2005). Among the longer-term studies of the impact of EE households on the course of schizophrenia over many years are McCreadie et al. (five years, 1993), Tarrier et al. (five and eight years, 1994), Schulze-Mönking et al. (eight years, 1997), Lenior et al. (nine years, 2002), Marom et al. (seven years, 2005), and Cechnicki et al. (12 years, 2010). A number of studies have demonstrated that high EE in the families of patients suffering from schizophrenia is associated with a worse course of the disease in terms of a higher number of relapses (Vaughn et al., 1984; Tanaka et al., 1995; Ronald et al., 1998), rehospitalization and time spent in hospital (Marom et al., 2005), and severity of symptoms (Schulze-Mönking et al., 1997; Cechnicki et al., 2010). McCreadie et al. (1993) demonstrated that patients living with low-EE families had fewer relapses than those with a high or fluctuating EE. A few patients in consistently high-EE homes had no relapses over a five-year period, but the small size of the group dictates caution in interpretation of the results. Ronald et al. (1998) observed a possible stronger association between EE and severity of relapses in patients with a more chronic course of the illness. Ivanovi c et al. (1994) find the number of relapses to be as much as 10 times higher in the high-EE group. The Cracow-based prospective study of EE assessed using the CFI showed an association between high EE and a greater number of relapses over a 12-year follow-up period from index hospitalization (Cechnicki et al., 2010). Only Schulze-Mönking et al. (1997) observed that patients with a high EE obtained higher scores in terms of intensification of
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symptoms measured on the GAS scale, while Cechnicki et al. (2010) found differences between the two groups only during the index hospitalization, and these were of borderline statistical significance. The meta-analysis by Pharoah et al. (2000) finds no association between EE and intensification of symptoms, while Klingberg (2002) concludes: “High-EE behavior does not seem to cause symptoms”. The majority of the works cited above, however, confirmed the predictive value of EE, as well as CC, for the course of schizophrenia and, in particular, relapses of psychosis. 2. Study objective This study is part of the Cracow-based prospective study into the course of schizophrenia, which aims to observe schizophrenia patients for many years, from their first psychotic episode through the years of their life with the illness, and assess the outcomes of their treatment and prognostic factors. The objectives of this paper are to assess whether the EE indicator as an overall construct and its two major components, CC and EOI, measured in an incidence group of patients during their index hospitalization, remain associated with the course of their schizophrenia over a period of 20 years, and to examine their prognostic value for selected treatment outcome factors. The treatment outcome criteria used were number of relapses, number and duration of in-patient rehospitalizations, and severity of psychopathological symptoms measured using the BPRS-E scale in direct clinical examinations at the successive 3-, 7-, 12- and 20-year catamnesis post index hospitalization. 3. Subjects The original study group comprised of 80 people diagnosed in accordance with DSM III (and later rediagnosed in accordance with DSM IV) criteria for schizophrenia, who were consecutively admitted to the Krakow Psychiatry Clinic for Adults of the University Hospital. The selection criteria were following: 18 or more years old, residential status in Kraków, living with family of orientation or/and procreation. We did not include youths as they are treated at a separate clinic. Of those 80 participants 8 were not assessed with CFI as they refused to participate in the recording of their CFI interview. 29 of the remaining 72 subjects were not included, as they did not attend one, or sometimes more, of the five follow-up assessments we included in the analysis because of refusals to participate, change of the place of residence, death by somatic causes or by suicide within the 20 years period (Cechnicki, 2011). As their data were not complete we had to drop them from the analysis, which left us with 43 complete assessments. Over the 20-year period about half of subjects, both in the high EE and the low EE group, have changed their co-habitants (6 out of 12 subjects with low EE and 15 out of 31 subjects with high EE). The sociodemographic data of this group is presented in Table 1. All subjects’ data were collected in ambulatory setting, with wholly voluntary participation. At each assessment an informed consent was sought from the patients and their family members. They were informed of the objective, methods and the course of the assessment. The study has been approved by the Bioethical Committee of the Collegium Medicum, Jagiellonian University, Cracow. 4. Method The level of EE indicator was measured using the Camberwell Family Interview (CFI) tool. The interviews were rated by a researcher certified in the method by C. Vaughn. The interviewees
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Table 1 Demographic factors (group n ¼ 43) divided according to EE level. EE level
Age at first symptoms (years) Age at index hospitalization (years)
Mean
Standard dev.
Min.
max.
High
Low
High
Low
High
Low
High
Low
26.6 27.5
27.3 28.3
6.50 5.98
5.26 6.22
13 19
20 20
39 39
37 43
High
Low
High
Low
18 13 10 21 8 4 10 7 2 16 12 3 11 20 14 17 22 9 20 11 8 1 22 0 0 0 13 2 9 6 0 1
7 5 5 7 4 0 7 1 0 10 1 1 12 n/a 12 n/a 8 4 7 5 3 1 8 0 0 0 7 1 3 1 0 0
58.1 41.9 32.3 67.7 25.8 12.9 32.3 22.6 6.5 51.6 38.7 9.7 35.5 64.5 45.2 54.8 71.0 29.0 64.5 35.5 25.8 3.2 71.0 0.0 0.0 0.0 41.9 6.5 29.0 19.4 0.0 3.2
58.3 41.7 41.7 58.3 33.3 0.0 58.3 8.3 0.0 83.3 8.3 8.3 100 n/a 100 n/a 66.7 33.3 58.3 41.7 25.0 8.33 66.7 0.0 0.0 0.0 58.3 8.3 25.0 8.3 0.0 0.0
EE level
Gender Marital status Education
Work
EE e critical comments (CC) EE e emotional over-involvement (EOI) Beginning of illness Duration of untreated psychosis (DUP) Co-habitants of the patients at the beginning of the study
Co-habitants of the patients at 20-year follow-up
Incidence
Women Men Marital relationship Single Higher (Master level) Higher (Bachelor level) Secondary Vocational Primary In full-time work or study On sick leave (from work or study) Not working Absent Present Absent Present Chronic Acute Under 6 months Over 6 months Family of procreation Family of procreation & orient Family of orientation Without relatives Sheltered housing Social care home Family of procreation Family of procreation & orient Family of orientation Without relatives Sheltered housing Social care home
were parents (one or both) or/and spouses of the patients. All relatives living in a household with a patient were interviewed, however, a single high EE relative was sufficient to consider the entire household as high EE. EE was rated as high following the standard criteria of the EE evaluation, that is, a “high” result in any of the three components: EOI, CC or hostility. EOI was assessed based on a global assessment of the whole interview, on a scale of 0e5 points. A score of 3 or more is considered to indicate high level of emotional over-involvement. CC was assessed based on the number of critical remarks by a relative, where 6 or more remarks are considered to indicate high level of criticism. Hostility was assessed on a scale of 0e3 points, with 1 or more points indicating high level of hostility. Other EE components (warmth and positive remarks) were of no significance in assessing the family as low or high EE. The severity of symptoms was measured using the UCLAmodified BPRS-E (Lukoff et al., 1986). A relapse was defined as such a patient’s condition which requires a BPRS-E rating of 6 or 7 (on a 1e7 scale) in at least one of the positive or negative symptoms. This relapse measure was adopted from Lukoff et al. (1986). Treatment outcome data were gathered at successive catamneses at 1, 3, 7, 12 and 20 years after index hospitalization using an adaptation of the Alanen and Rekkolainen Anamnestic and Catamnestic Interview (Cechnicki, 2011). In the first set of analyses EE served as the independent between-group variable with two levels: high (any one or combination of the following: hostility, CC, EOI) and low (none of the above). In successive analyses the independent between-group
Percentage
variables were the dichotomous EOI indicator and the dichotomous CC indicator. Time was an independent variable with repeated assessments at successive follow-ups. Five points of measurement were analyzed e at the 1-, 3-, 7-, 12- and 20-year follow-ups. The indicators of treatment outcome (dependent variables) selected were number of relapses, number of rehospitalizations, duration of rehospitalizations (weeks), and severity of symptoms as per BPRS e overall result, BPRS e positive syndrome, and BPRS e negative syndrome. The independent variables formed a two-factor research plan with one between-group factor (EE, EOI or CC) and one repeated measurement factor (time). Potential differences were explored using the repeated measures analysis of variance. 5. Study results A preliminary comparison of the mean number of relapses was made for high-EE and low-EE groups, separately for each follow-up. This enabled accounting for the different numbers of people attending each follow-up, and, in effect, show between-group comparisons for relatively more numerous, although somewhat differing, samples. Low-EE groups evince a lower average number of relapses in case of every follow-up except one year after index hospitalization (Fig. 1). These results indicate possible relationships over time and in order to investigate them our next step was to properly analyze the 43 people who attended all five follow-ups. The indicators
A. Cechnicki et al. / Journal of Psychiatric Research 47 (2013) 208e214
Fig. 1. Mean numbers of relapses between index hospitalization and assessment at the given follow-up points showing division into low-EE and high-EE groups (significance levels of the differences in the U ManneWhitney test at successive follow-up points: p ¼ 0.240, p ¼ 0.002, p ¼ 0.007, p ¼ 0.042, p ¼ 0.024).
examined were changes over time in number of relapses, number of rehospitalizations, duration of rehospitalizations, and severity of symptoms measured using the BPRS, as well as interaction of these changes with EE level. Across the study group, irrespective of EE level, numbers of relapses and rehospitalizations, and duration of rehospitalizations increased in successive years, which was expected, as these data are additive. Parallel to this, a stabilization of BPRS-measured psychopathological symptoms was observed in the early catamneses, followed by slight intensification at 12- and 20-year assessments. Of major interest to us were interaction effects of repeated measures and the level of expressed emotions, as well as its components, which now follow. A significant interaction was found between EE level and change in relapses (F(4, 164) ¼ 5.02; p < 0.001) and rehospitalizations (F(4, 164) ¼ 2.85; p ¼ 0.025). The graphs below show the detailed differences at successive followups (Figs. 2 and 3). The differences in number of relapses between the two groups at the various follow-up points proved significant for the measurements at 3 (p ¼ 0.010), 7 (p ¼ 0.009), 12 (p ¼ 0.025) and 20 (p ¼ 0.011) years. Rises in numbers of relapses were higher in successive years in the high-EE group (p < 0.001 for differences between all the successive follow-up points) than in the low-EE
Fig. 2. Relationship between changes in number of relapses over time and EE level.
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Fig. 3. Relationship between changes in number of rehospitalizations over time and EE level.
group, where differences were statistically insignificant except for that between the first and 12th year of illness (p ¼ 0.42) and the third and 12th year of illness (p ¼ 0.046). Changes in number of rehospitalizations demonstrated similar association with EE level. The differences between the groups at each catamnesis were significant at three (p ¼ 0.020) and seven (p ¼ 0.023) years, and showed as a clear trend at 12 (p ¼ 0.055) and 20 (p ¼ 0.056) years. In the high-EE group results were significantly different at each successive follow-up (p ¼ 0.002 or lower), while in the low-EE group all the differences between follow-ups were insignificant (Fig. 3). In the case of the duration of rehospitalizations and severity of symptoms measured using the BPRS, the interactions with EE level were not statistically significant. 6. EOI and CC in relation to treatment outcomes The next step in our study was to examine the changes in number of relapses, number of rehospitalizations, duration of rehospitalizations and severity of symptoms measured using the BPRS and the relationship between change over time (at follow-up points) and levels of the EE constituents: CC and EOI (Figs. 4 and 5). No significant interaction was observed between the time of measurement and the level of EOI for any of the six outcome indicators. In the case of interaction between the time of measurement and the level of CC, however, two significant interaction effects were noted. In the first of them, the number of relapses rose higher in the high-CC group (F(4, 164) ¼ 4.11; p ¼ 0.003) (Fig. 4). The differences between the groups at the individual points of measurement were not statistically significant, but a trend was noted after 12 (p ¼ 0.069) and 20 (p ¼ 0.051) years. Both in the group with a high level of CC (p 0.001) and that with a low level of this indicator (p 0.021) the differences between numbers of relapses at successive catamneses were statistically significant. The second interaction was observed between CC and changes in the severity of positive symptoms (F(4, 164) ¼ 3.32; p ¼ 0.012) (Fig. 5). In the group in which a low level of CC was observed, positive symptoms did not change significantly over time. In the high-CC group, however, positive symptoms intensified over time, and statistically significant differences were noted between the 20-year follow-up and all the previous follow-ups (p 0.014). No
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Fig. 4. Relationship between changes in number of relapses over time and CC level.
statistically significant differences in severity of positive symptoms occurred at the various follow-up points between the high-CC and low-CC groups, though a borderline trend-level difference was observed after 20 years (p ¼ 0.100). 7. Discussion The results obtained in the present prospective study permit to interpret EE, assessed in the second week of the index hospitalization, as a specific predictor of early and long-term treatment outcomes. The associations observed relate to several indicators of the course of schizophrenia: relapses, rehospitalization, and severity of positive symptoms. A particularly important role is played by the CC component of EE. The majority of studies of EE confirm its predictive value, and the largest number of reports in the literature pertains to the dependence between EE and relapses. The Cracow study confirmed the association between EE and relapses in the short-term course of schizophrenia, and this outcome is concordant with studies including those by Kavanagh (1992), Ivanovi c et al. (1994), Linszen et al. (1997), Ronald et al. (1998), Rostworowska (1999), King
Fig. 5. Relationship between changes in the severity of positive symptoms over time (measured with BPRS) and CC level.
(2000), Patterson et al. (2005). Moreover, in long-term catamneses this association was also confirmed, as in McCreadie et al. (1993), Tarrier et al. (1994), Schulze-Mönking et al. (1997), Lenior et al. (2002), Cechnicki et al. (2010). A major new finding of the present study was that the difference in numbers of relapses between the high-EE and low-EE groups was sustained over 20 years. Mean numbers of relapses reach significant levels of difference at 3, 7, 12 and 20 years from index hospitalization. The study by Raune et al. (2004) offered an analysis of the complexity of issues surrounding the components of EE. High EOI proved to be related to an adverse impact on numbers of relapses, while an average EOI could have a protective effect. This is because moderate levels of EOI are associated with high warmth, while warmth is lower at high levels of EOI. Similar results were obtained by Breitborde et al. (2007), who also found that high level of warmth “appear to exert a protective effect on the course of illness”. In the present study both the high-CC and the low-CC groups evinced a statistically significant increase in numbers of relapses at successive follow-ups. Differences between the groups at the various points were not statistically significant, but a rising trend was observed at 12 and 20 years. As in the studies by Schulze-Mönking et al. (1997) and Marom et al. (2005), in the Cracow study the high EE proved to have a significant association with rehospitalizations at three and seven years, which was still visible at the trend level at 12 and 20 years. However, in contrast to those studies, there was no visible association between EE and duration of rehospitalizations, probably because the patients in Cracow were taking part in a psychosocial treatment program specifically aimed at reducing time in hospital. Rehospitalization and its duration is more reliable to record as an indicator of the course of the illness, but is dependent on many local circumstances, so should be taken in the context of other dimensions of treatment. Neither was confirmation obtained of the outcome found by Marom et al. (2005) that a high level of CC as a component of EE is associated with more frequent and longer rehospitalizations. Relatively few scholars have focused on the relationships between EE and severity of symptoms. In the meta-analysis by Pharoah et al. (2000) no association was observed between EE and symptom severity. On the other hand, in their psychophysiological studies, Sturgeon et al. (1984) found that patients had a higher arousal level in the presence of a high EE relative than when they were with a low EE relative. Woo et al. (2004) describe an association between EE and the symptoms of hostility, anxiety and arousal. Docherty et al. (2011) explored the relationship between CC and anxiety. Patients with high CC and a higher degree of anxiety had more severe symptoms at nine months from index hospitalization than those with low CC. Schulze-Mönking et al. (1997) also found a connection between EE and symptoms measured using the GAS, both at the start of treatment and after eight years. In the Cracow study CC measured at the onset of the illness is associated with intensification of the positive syndrome over 20 years of the illness. A significant difference was noted between the measurement at 20 years and all the previous measurements in the high-CC group, in which a gradual increase in severity of symptoms was observed over time. The study does not answer the question of whether high CC in the family is a cause of or a response to the psychopathology of the sickness of one of its members. Lenior et al. (2002) found instability in both overall EE and CC and EOI over nine years of observation, but as McCreadie (1993) noted, EE during index hospitalization is a 60% predictor of future EE, which may partly explain why in the Cracow study EE and CC levels measured at index hospitalization are connected to treatment results after 20 years. As a complex construct (Ronald et al.,
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1998), EE comprises relatively stable variables related to both personality (Hooley and Hiller, 2000) and circumstances, i.e. both reactive and temporary (King, 2000). Owing to this complexity, the issue of the stability of the construct is one for which it is hard to offer verification and theoretical justification. King (2000) found a dependence suggesting that both EOI and CC may be effects rather than causes of intensification of symptoms. Alvarez-Jimenez et al. (2008) indicate that CC may be a reaction by the family linked with attributing to the patient causality of the disease and a lack of understanding of the problem. Woo et al. (2004) moot bidirectional dependence between EE, the family and the patient’s psychopathology. As has been confirmed in many studies, lowering EE definitely reduces the relapse rate so that there is a strong vector between relatives’ EE and the patient’s vulnerability to relapse (Tarrier et al., 1994; Bäuml et al., 1996; Linszen et al., 1997; Leff, 1998, and others). Intervention designed to reduce CC in the family is thus a vital standard in treatment of schizophrenia, in particular in the context of the conclusions of Cutting et al. (2006), who studied patients’ perception of family behaviors. Their observations improve understanding of the processes in the family as they demonstrate that CC is stressful for patients because they perceive it clearly. An analysis of our results paired with the knowledge gathered from clinical research to date seem inviting to formulate a hypothesis explaining long-term effect of EE on treatment outcomes, in particular relapses and positive symptoms. One possibility is to think of family relations as being internalized by the patient many years before the onset of the illness. This may result in e.g. developing an overly critical self-attitude. Combined with a critical and rejecting reaction of their relatives at the onset of the illness, this situation renders the patient continuously subjected to intensive negative emotions, which may result in an unfavorable course of the illness. Constructing research models that would help to differentiate between more and less variable aspects of EE, as well as between variables dependent on the family and those dependent on the illness, would help to gain better insight into the nature of this phenomenon, which presents as both a predictor and an effect of the course of the illness. Irrespective of the outcomes of future studies, there is a need for effective therapeutic interventions focused on improving communication and relationships within the families of schizophrenia sufferers, and in particular on reducing CC. 8. Conclusions 1. EE level as measured at index hospitalization may be seen as a specific predictor of relapse and rehospitalization in the group under study over 20 years of the course of schizophrenia. 2. High CC is associated with intensification of the positive syndrome in the course of the illness.
Role of the funding source The research presented in the attached manuscript was founded from the research grant no. K/ZDS 000652 e an internal statutory grant of the Chair of Psychiatry, Collegium Medicum, Jagiellonian University in Cracow, Poland. Contributions Andrzej Cechnicki (AC) Anna Bielanska (AB) Igor Hanuszkiewicz (IH) Artur Daren (AD)
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AC designed the study and coordinated subsequent assessments, AC, AB wrote the first draft, AC, AB, IH managed the literature searches, analyses and discussion, AD and IH undertook statistical analysis. All authors participated in preparation and have approved the final version of the paper. Declaration of interest All four authors declare that there are no conflicts of interest regarding the submitted paper. Acknowledgments We would like to thank Mr Konrad Wronski, who kindly assisted with preparation of the figures, and Miss Joanna Palka, who assisted with the preparation of literature and proof-reading of this paper. References Alvarez-Jimenez M, Gleeson JF, Cotton SM, Wade D, Crisp K, Yap MBH. Differential predictors of critical comments and emotional over-involvement in firstepisode psychosis. Psychological Medicine 2008;40:63e72. Barrowclough C. Staff expressed emotion and causal attributions for client problems on a low security unit: an exploratory study. Schizophrenia Bulletin 2001; 27:517e26. Bäuml J, Pitschel-Walz G, Kissling W. Psychoedukative Gruppen bei schizophrenen Psychosen für Patienten und Angehörige. In: Stark A, editor. Verhaltenstherapeutische und psychoedukative Ansätze im Umgang mit schizophren Erkrankten. Tübingen: dgvt-Verlag; 1996. p. 217e55. Bebbington P, Kuipers L. The predictive utility of expressed emotion in schizophrenia: an aggregate analysis. Psychological Medicine 1994;24:707e18. Berry K, Barrowclough C, Haddock G. The role of expressed emotion in relationships between psychiatric staff and people with a diagnosis of psychosis: a review of the literature. Schizophrenia Bulletin 2011;37:958e72. Breitborde N, Lopez S, Wickens D, Jenkins J, Karno M. Toward specifying the nature of the relationship between expressed emotion and schizophrenic relapse: the utility of curvilinear models. International Journal of Methods in Psychiatric Research 2007;16:1e10. Brown GW, Rutter M. The measurement of family activities and relationships: methodological study. Human Relations 1966;19:241e63. Butzlaff RL, Hooley JM. Expressed emotion and psychiatric relapse. A meta-analysis. Archives of General Psychiatry 1998;55:547e52. Cechnicki A, Hanuszkiewicz I, Polczyk R, Cichocki q, Kalisz A, Rostworowska M. Wska znik ekspresji emocji (EE) jako rodzinny predyktor przebiegu schizofrenii [Expressed Emotion indicator as a family predictor of the course of schizophrenia]. Psychiatria Polska 2010;2:173e84. Cechnicki A. Schizofrenia e proces wielowymiarowy. Krakowskie prospektywne badania przebiegu, prognozy i wyników leczenia schizofrenii [Schizophrenia e A multidimensional process. An evaluation of its course, prognosis and longterm treatment results]. Warszawa: Instytut Psychiatrii i Neurologii; 2011. Cutting LP, Aakre JM, Docherty NM. Schizophrenic patients’ perceptions of stress, expressed emotion, and sensitivity to criticism. Schizophrenia Bulletin 2006; 26:1e8. Dennis AM, Leach C. Expressed emotion and burnout: the experience of staff caring for men with learning disability and psychosis in a medium secure setting. Journal of Psychiatric and Mental Health Nursing 2007;14:267e76. Docherty NM, St-Hilaire A, Aakre JM, Seghers JP, McCleery A, Divilbiss M. Anxiety interacts with expressed emotion criticism in the prediction of psychotic symptom exacerbation. Schizophrenia Bulletin 2011;37:611e8. Hooley JM, Hiller JB. Personality and expressed emotion. Journal of Abnormal Psychology 2000;109:40e4. Hopper K, Harrison G, Janca A, Sartorius N. Recovery from schizophrenia: an international perspective. A report from the WHO Collaborative Project, the international study of schizophrenia. Oxford: Oxford University Press; 2007. p. 277e285. Humbeeck GV, Audenhove CV. Expressed emotion of professionals towards mental health patients. Epidemiologia e Psichiatria Sociale 2003;12:232e7. Ivanovi c M, Vuletib Z, Bebbington R. Expressed emotion in the families of patients with schizophrenia and its influence on the course of illness. Social Psychiatry and Psychiatric Epidemiology 1994;29:61e5. Kavanagh DJ. Recent developments in expressed emotion and schizophrenia. British Journal of Psychiatry 1992;160:601e20. King S. Is expressed emotion cause or effect in the mothers of schizophrenic young adults? Schizophrenia Research 2000;45:65e78. Klingberg S. Family interventions: empirical evidence of efficacy and open questions. In: Häfner H, editor. Risk and protective factors in schizophrenia. Heidelberg: Springer; 2002. p. 285e93. Kuipers L, Leff J, Lam D. Family work for schizophrenia: a practical guide. Gaskell, London: Royal College of Psychiatrists; 1992.
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