Robin Michael Van Eck Transcending Illness Personal Recovery in Psychosis
Robin Michael Van Eck Transcending Illness Personal Recovery in Psychosis
Colophon All rights reserved. No parts of this publication may be reproduced or transmitted in any form or by any means without the written permission of the author. The author is grateful for the financial support to perform the research in this thesis provided by Amsterdam UMC, Mentrum/Arkin, Focus GGZ/Caleidozorg and Invivo Clinics. The infrastructure for the GROUP study, described in chapter 6, was funded through the Geestkracht programme of the Dutch Health Research Council (ZonMw, grant number 10-000-1001), and matching funds from participating pharmaceutical companies and universities and mental health care organizations (see details in chapter 6). The study described in chapter 7 was supported by a grant from ZonMw, as a part of the mental healthcare research programme (grant number 636330002). The printing of this thesis was financially supported by: Amsterdam UMC and Mentrum/Arkin. Author: Robin Michael Van Eck Design: Hans Schaapherder, Persoonlijk Proefschrift | www.persoonlijkproefschrift.nl All photos on the cover of and inside this thesis show Mount Etna Volcano, Sicily, Italy, in different stages of eruption. Photos chapters 1, 3 and dankwoord pages: Carlinde Broeks/Robin Van Eck; Photos chapters 2, 4-9 and appendix page: Shutterstock. Printing: Ridderprint | www.ridderprint.nl ISBN: 978-94-6522-460-2
“I have always experienced what happened to me as a volcano that erupts. It will never be like it was before, because of the explosion. Then it settles down and it becomes a new volcano.” (Quote from one of the participants of the qualitative research described in chapter 5)
TABLE OF CONTENTS Chapter 1 General introduction 9 Chapter 2 The relationship between clinical and personal recovery in patients with schizophrenia spectrum disorders: a systematic review and meta-analysis 19 Chapter 3 The impact of affective symptoms on personal recovery of patients with severe mental illness 49 Chapter 4 The association between change in symptom severity and personal recovery in patients with severe mental illness 65 Chapter 5 Clinical treatment interventions in personal recovery stories of patients with severe mental illness: a qualitative study 83 Chapter 6 Personal recovery suits us all: a study in patients with non-affective psychosis, unaffected siblings and healthy controls 103 Chapter 7 Perspective matters in recovery: the views of persons with severe mental illness, family and mental health professionals on collaboration during recovery, a qualitative study 123 Chapter 8 General discussion 157 Chapter 9 English summary 179 Appendix Nederlandse samenvatting Portfolio About the author Dankwoord 187 191 195 197
CHAPTER 1 General introduction Zicht op de Etna vanaf Tenuta San Michele, Santa Venerina, Sicilië, gemaakt door Carlinde Broeks, 4 juni 2025
10 Chapter 1 GENERAL INTRODUCTION Definition of recovery Recovery is a broad term with a wide range of meanings. The Oxford dictionary states: ‘senses relating to gaining or regaining possession’ and ‘senses related to the regaining of a state or position.’ Regarding the last category, one of the sub definitions is: ‘restoration or return to health from illness’ (1). In medicine, recovery generally means this ‘returning to a pre-existing state of health.’ In psychiatry and mental healthcare diverging descriptions of recovery have emerged, coming from different backgrounds. Usually, two types of recovery are defined: clinical and personal recovery (2). Clinical recovery Clinical recovery entails a medical point of view, in which symptoms of an illness need to be reduced by treatment, for instance by medication. This meaning corresponds with ‘cure’ of a disease and definitions come from professionals. According to Slade, clinical recovery usually has four key features: 1. it is an outcome, 2. it is objective, 3. it is rated by a clinician and 4. it is invariant across individuals (3). Usually, for research purposes, clinical remission is at the basis of the definition of recovery (4). The Remission in Schizophrenia Working Group (RSWG) defines remission as improvements in core signs and symptoms to the extent that they are of such low intensity that they no longer interfere significantly with behavior or cause suffering. Operational criteria include a score of mild or less on specific items of a symptom scale over a 6-month period, e.g. the Positive and Negative Syndrome Scale (PANSS) or the Brief Psychiatric Rating Scale (BPRS). The RSWG states that recovery is a more demanding and longer-term phenomenon than remission and that remission is a necessary but not sufficient step toward recovery. Recovery implies, besides being relatively free of disease-related psychopathology, the ability to function in the community, socially and vocationally (5). Some authors indeed include functional improvement in their definition of clinical recovery, e.g. having work, having friends, living independently (6), or a score of >65 on the Global Assessment of Functioning (GAF) (7). Others define these factors as an additional type of recovery: functional or social/societal recovery is about interpersonal relationships, work or study, daily living and self-care (8-11). Some limit functional recovery to regaining psychological functions, such as executive functions and self-regulation (12, 13). Usually, though, the abovementioned dichotomy of clinical and personal recovery is used in research. Personal recovery The definition of personal recovery has been developed by the patient movement and has been based on narratives of individuals who have experienced mental illness. The most cited definition is the one from William Anthony: “a deeply personal, unique
11 General introduction process of changing one’s attitudes, values, feelings, goals, skills, and/or roles. It is a way of living a satisfying, hopeful, and contributing life even with limitations caused by illness. Recovery involves the development of new meaning and purpose in one’s life as one grows beyond the catastrophic effects of mental illness (14).” Manuscripts with first person accounts on recovery have been published in scientific journals, which illustrate the individual character of recovery (15-18). Recent qualitative research shows that recovery narratives of people with different diagnoses show similar themes. This suggests that personal recovery is a transdiagnostic concept, i.e. not only applicable to patients with psychosis (19). In addition, Anthony mentions another interesting aspect: “Recovery transcends illness and the disability field itself. Recovery is a truly unifying human experience. Because all people (helpers included) experience the catastrophes of life (death of a loved one, divorce, the threat of severe physical illness, and disability), the challenge of recovery must be faced.” This implies that the concept of recovery is not limited to people with mental illness, but is universal. After that, Anthony writes: “Successful recovery from a catastrophe does not change the fact that the experience has occurred, that the effects are still present, and that one’s life has changed forever. Successful recovery does mean that the person has changed, and that the meaning of these facts to the person has therefore changed. They are no longer the primary focus of one’s life.” This suggests that recovery is not, as described in the classical dictionary definition above, a return to the same state as before, but an adaptation to disruptive circumstances. Like stated in the quote of a research participant at the beginning of this thesis, a volcano might be a good metaphor: after an eruption, the volcano will never be like it was before, and you don’t know if and when it will erupt again. This is in line with Deegan’s description of recovery as “a transformative process in which the old self is gradually let go of and a new sense of self emerges (15).” ‘To overcome’, rather than ‘to recover’, might have been a better word for this process. Efforts have been made to develop conceptual frameworks for personal recovery. A systematic review and narrative synthesis resulted in an, often cited, acronym CHIME, which stands for five core recovery processes: Connectedness; Hope and optimism about the future; Identity; Meaning in life; and Empowerment (20). Also, different phases of recovery have been defined: Overwhelmed by, struggling with, living with and living beyond the disability (21). There is a debate whether personal recovery is measurable in a quantitative manner, because it is highly individual (22). A ‘golden standard’ is difficult, if not impossible, to define. This also relates to the question whether personal recovery is an outcome or a process (23, 24). An outcome measure is a tool that can be used to assess the effectiveness of a treatment, usually by comparing scores before and after an intervention. Personal recovery as an outcome may be compared with other 1
12 Chapter 1 outcomes, like clinical scales. Many patients find the definition of recovery as an outcome to be unsatisfactory, because it suggests an evaluative component (25). Rudnick suggests, from a philosophical framework, to characterize recovery as “a process of adaptive or compensatory self-organization of the person as a whole and in relation to the environment (24).” The process of being ‘in recovery’ can be continuous, but can also be defined with stages. Several personal recovery measures have been developed (26). Some try to capture the process by identifying the stages of recovery, such as the Stages of Recovery Instrument (STORI) (27). Others define recovery as an outcome, for example the widely used Recovery Assessment Scale (RAS) (28, 29). The Individual Recovery Outcomes Counter (I.ROC) can be used to start a discussion with a patient about recovery and what is needed to support this process. The resulting spidergram visually shows individual areas of strength, and areas that are a priority to work on. As such, the I.ROC does not measure an outcome, but focuses on the individual evaluation of the patient (30). The relationship between clinical and personal recovery Traditionally, the primary goal of treatment of mental disorders is reduction/ remission of symptoms leading to clinical recovery (31). But if clinical and personal recovery do not overlap, are we helping the patient enough by focusing on symptom reduction? Does clinical improvement of specific symptom domains support personal recovery? Or do we need to extend our treatment strategies beyond clinical goals to promote personal recovery (32)? Earlier studies on the relationship between clinical and personal recovery were usually done in patients with schizophrenia spectrum disorders. They show substantial variation in direction and magnitude of the association (33-36). Evidence suggests that symptoms of psychosis, i.e. positive and negative symptoms, show a smaller association with personal recovery than affective symptoms (37, 38). It is important to know which symptom domains particularly influence personal recovery and vice versa, because this could also inform professionals about priorities in their treatment-focus. Anyway, the association between clinical and personal recovery varies. If both types of recovery are placed in a figure with clinical recovery on the x-axis and personal recovery on the y-axis, a model is established with four ‘profiles of recovery’ (see figure 1): A: personal recovery, but no clinical recovery B: clinical recovery and personal recovery C: no clinical recovery and no personal recovery D: clinical recovery, but no personal recovery
13 General introduction Profiles B and C seem logical from a medical perspective, because patients show less symptoms and experience recovery or still have symptoms and experience no recovery. Categories A and D are at first sight less obvious. Why do patients with ongoing symptoms do experience recovery (category A)? And why do patients with remission of symptoms do not feel recovered (category D)? To understand these patients better, qualitative research is needed to ask them if clinical treatment interventions were actually helpful or not to achieve personal recovery. Clinicians may use this knowledge to adapt their practices to stimulate patients’ process of recovery. Figure 1: Conceptual model of four profiles of recovery. Illustration adapted from a figure developed by Penumbra (http://www.penumbra.org.uk) Personal recovery suits us all? Starting from the idea, described above in the definition of Anthony, that personal recovery is universally human, the question arises whether (clinical) factors associated with personal recovery are similar in persons with and without a psychiatric diagnosis. If patients and non-patients share supportive factors of personal recovery, this underscores the hypothesis that recovery transcends illness and this may result in mutual understanding. No earlier research has been done into the possible predictors of personal recovery in the general population. Van der Krieke et al. already found that the usefulness of the Recovery Assessment Scale (RAS) in outcome assessments is questionable, because the differences detected in recovery between service users, siblings, and healthy controls had limited clinical relevance. This indicates that recovery suits us all. 1
14 Chapter 1 The recovery triad The journey of recovery is personal, but also undertaken with significant others: family and friends and professionals (39). It is important to know and understand the views of the members of this ‘triad’ on recovery, because they may influence the process in a positive or negative way (40). Having more knowledge about the collaboration between patients, family and mental health professionals during recovery may open opportunities to facilitate this partnership (41). Aim and research questions, and outline of this thesis The aim of the research in this thesis is to better understand the relationship between clinical and personal recovery in patients with psychosis and other severe mental illnesses (SMI). The questions that we want to answer are: • What is the strength of the relationship between clinical and personal recovery in patients with schizophrenia spectrum disorders (SSD)? In chapter 2 the relationship between clinical and personal recovery is explored by means of a systematic review and meta-analysis of existing literature in patients with SSD. • What is the correlation between the severity of clinical symptom domains and personal recovery in patients with SMI? In chapter 3 cross sectional data are presented of a study on the relationship between clinical symptom domains and personal recovery performed at Flexible Assertive Community Teams (42) of Mentrum, part of Arkin Institute for Mental Health, in Amsterdam, the Netherlands. The participants all have a severe mental illness (SMI), but vary in diagnosis (not only schizophrenia), which is common in mental healthcare. • What is the association between change in symptom severity and change in personal recovery in patients with SMI? In chapter 4 the longitudinal perspective of the study at Mentrum is described. After three years assessment was repeated to evaluate the association between change in symptom severity and change in personal recovery over time. • What is the subjective experience of clinical treatment interventions in personal recovery stories of patients with SMI? Chapter 5 shows the results of qualitative interviews with 26 participants from the quantitative study described in chapter 3 and 4. They were asked to reflect on the importance of clinical treatment interventions for their personal recovery process, both in a helping and a hindering sense.
15 General introduction • Which individual and environmental factors influence personal recovery in patients with non-affective psychosis, their unaffected siblings and healthy controls? Chapter 6 investigates the predictors of personal recovery in a large sample of not only patients, but also siblings and healthy controls. This approach is in line with the idea - as explained above - that recovery is a universal experience, rather than limited to persons with a diagnosed mental disorder. • What is the experience of the collaboration of persons with severe mental illness, family and mental health professionals during recovery? Chapter 7 explores different perspectives on the collaboration towards recovery in the triad of patient, family and professional. In chapter 8 the findings will be discussed and implications for clinical practice and directions for future research will be presented. Chapter 9 provides a summary of the thesis. 1
16 Chapter 1 REFERENCES 1. Oxford English Dictionary [Search ‘recovery’]. Available from: https://www.oed.com/. 2. Slade M, Amering M, Oades L. Recovery: an international perspective. Epidemiologia e psichiatria sociale. 2008;17(2):128-37. 3. Slade M. Personal Recovery and Mental Illness. A Guide for Mental Health Professionals. Cambridge, United Kingdom: Cambridge University Press; 2009. 4. Leucht S, Lasser R. The Concepts of Remission and Recovery in Schizophrenia. Pharmacopsychiatry. 2006;39(05):161-70. 5. Andreasen NC, Carpenter WT, Jr., Kane JM, Lasser RA, Marder SR, Weinberger DR. Remission in schizophrenia: proposed criteria and rationale for consensus. The American journal of psychiatry. 2005;162(3):441-9. 6. Liberman RP, Kopelowicz A, Ventura J, Gutkind D. Operational criteria and factors related to recovery from schizophrenia. International Review of Psychiatry. 2002;14(4):256-72. 7. Torgalsbøen A. What is recovery in schizophrenia? In: Provencher HL, Keyes CLM, editors. Severe Mental Illness, Research Evidence and Implications for Practice. Boston, US: Boston University; 2013. p. 302-15. 8. Castelein S, Timmerman ME, PHAMOUS investigators, van der Gaag M, Visser E. Clinical, societal and personal recovery in schizophrenia spectrum disorders across time: states and annual transitions. The British Journal of Psychiatry. 2021:1-8. 9. Crutzen S, Burger SR, Visser E, Ising HK, Arends J, Jörg F, et al. Societal recovery trajectories in people with a psychotic disorder in long term care: a latent class growth analysis. Social Psychiatry and Psychiatric Epidemiology. 2024. 10. Swildens WE, Visser E, Bähler M, Bruggeman R, Delespaul P, van der Gaag M, et al. Functional recovery of individuals with serious mental illnesses: Development and testing of a new short instrument for routine outcome monitoring. Psychiatric rehabilitation journal. 2018;41(4):341-50. 11. Castelein S, Visser E, Brilman MF, Wardenaar KJ, Bruins J. Identifying factors strongest associated with clinical, societal and personal recovery in people with psychosis with a long duration of illness. Comprehensive Psychiatry. 2025;136:152540. 12. van der Stel JC. [Functional recovery and self-regulation: assignments for both clients and psychiatrists]. Tijdschrift voor psychiatrie. 2015;57(11):815-22. 13. van Aken BC, Wierdsma AI, Voskes Y, Pijnenborg GHM, van Weeghel J, Mulder CL. The Association Between Executive Functioning and Personal Recovery in People With Psychotic Disorders. Schizophrenia Bulletin Open. 2022;3(1). 14. Anthony WA. Recovery from mental illness: the guiding vision of the mental health service system in the 1990s. Psychosocial Rehabil J. 1993;16(4):11-23. 15. Deegan PE. Recovery as a Self-Directed Process of Healing and Transformation. Occupational Therapy in Mental Health. 2002;17(3-4):5-21. 16. Deegan PE. I am a person, not an illness. Schizophrenia research. 2022;246:74. 17. Mead S, Copeland ME. What recovery means to us: consumers’ perspectives. Community mental health journal. 2000;36(3):315-28. 18. Boevink WA. From being a disorder to dealing with life: an experiential exploration of the association between trauma and psychosis. Schizophrenia bulletin. 2006;32(1):17-9. 19. Lases MN, Bruins J, Scheepers FE, van Sambeek N, Ng F, Rennick-Egglestone S, et al. Is personal recovery a transdiagnostic concept? Testing the fit of the CHIME framework using narrative experiences. Journal of Mental Health. 2024:1-9. 20. Leamy M, Bird V, Le Boutillier C, Williams J, Slade M. Conceptual framework for personal recovery in mental health: systematic review and narrative synthesis. The British journal of psychiatry : the journal of mental science. 2011;199(6):445-52. 21. Spaniol L, Wewiorski NJ, Gagne C, Anthony WA. The process of recovery from schizophrenia. International Review of Psychiatry. 2002;14:327–36. 22. Davidson L. Is “Personal Recovery” a Useful Measure of Clinical Outcome? Psychiatric services (Washington, DC). 2019;70(12):1079. 23. van Weeghel J, van Zelst C, Boertien D, Hasson-Ohayon I. Conceptualizations, assessments, and implications of personal recovery in mental illness: A scoping review of systematic reviews and meta-analyses. Psychiatric rehabilitation journal. 2019;42(2):169-81.
17 General introduction 24. Rudnick A. Recovery from Schizophrenia: A Philosophical Framework. American Journal of Psychiatric Rehabilitation. 2008;11(3):267-78. 25. Ralph RO, Corrigan PW. Recovery in mental illness: Broadening our understanding of wellness. Washington, DC: American Psychological Association; 2005. 26. Shanks V, Williams J, Leamy M, Bird VJ, Le Boutillier C, Slade M. Measures of personal recovery: a systematic review. Psychiatric services (Washington, DC). 2013;64(10):974-80. 27. Andresen R, Caputi P, Oades L. Stages of Recovery Instrument: Development of a Measure of Recovery from Serious Mental Illness. Australian & New Zealand Journal of Psychiatry. 2006;40(11-12):972-80. 28. Giffort D, Schmook A, Woody C, Vollendorf C, Gervain M. Recovery assessment scale. Chicago: Illinois: Department of Mental Health; 1995. 29. Law H, Morrison A, Byrne R, Hodson E. Recovery from psychosis: a user informed review of self-report instruments for measuring recovery. Journal of mental health (Abingdon, England). 2012;21(2):192-207. 30. Rudd B, Karatzias T, Bradley A, Fyvie C, Hardie S. Personally meaningful recovery in people with psychological trauma: Initial validity and reliability of the Individual Recovery Outcomes Counter (I.ROC). Int J Ment Health Nurs. 2020;29(3):387-98. 31. Bellack AS. Scientific and consumer models of recovery in schizophrenia: concordance, contrasts, and implications. Schizophrenia bulletin. 2006;3(32):432-42. 32. Slade M, Amering M, Farkas M, Hamilton B, O’Hagan M, Panther G, et al. Uses and abuses of recovery: implementing recovery-oriented practices in mental health systems. World Psychiatry. 2014;13(1):12-20. 33. Giusti L, Ussorio D, Tosone A, Di Venanzio C, Bianchini V, Necozione S, et al. Is personal recovery in schizophrenia predicted by low cognitive insight? Community mental health journal. 2015;51(1):30-7. 34. Roe D, Mashiach-Eizenberg M, Lysaker PH. The relation between objective and subjective domains of recovery among persons with schizophrenia-related disorders. Schizophrenia research. 2011;131(1-3):133-8. 35. Vass V, Morrison AP, Law H, Dudley J, Taylor P, Bennett KM, et al. How stigma impacts on people with psychosis: The mediating effect of self-esteem and hopelessness on subjective recovery and psychotic experiences. Psychiatry research. 2015;230(2):487-95. 36. Chan RCH, Mak WWS, Chio FHN, Tong ACY. Flourishing With Psychosis: A Prospective Examination on the Interactions Between Clinical, Functional, and Personal Recovery Processes on Well-being Among Individuals with Schizophrenia Spectrum Disorders. Schizophrenia bulletin. 2017;44(4):778-86. 37. Kukla M, Lysaker PH, Roe D. Strong subjective recovery as a protective factor against the effects of positive symptoms on quality of life outcomes in schizophrenia. Compr Psychiatry. 2014;55(6):1363-8. 38. Brown C, Rempfer M, Hamera E. Correlates of insider and outsider conceptualizations of recovery. Psychiatric rehabilitation journal. 2008;32(1):23-31. 39. Topor A, Borg M, Di Girolamo S, Davidson L. Not just an individual journey: social aspects of recovery. The International journal of social psychiatry. 2011;57(1):90-9. 40. Camacho-Gomez M, Castellvi P. Effectiveness of Family Intervention for Preventing Relapse in First-Episode Psychosis Until 24 Months of Follow-up: A Systematic Review With Meta-analysis of Randomized Controlled Trials. Schizophrenia bulletin. 2020;46(1):98-109. 41. Landeweer E, Molewijk B, Hem MH, Pedersen R. Worlds apart? A scoping review addressing different stakeholder perspectives on barriers to family involvement in the care for persons with severe mental illness. BMC Health Serv Res. 2017;17(1):349. 42. Remmers van Veldhuizen J. FACT: a Dutch version of ACT. Community mental health journal. 2007;43(4):421-33. 1
CHAPTER 2 The relationship between clinical and personal recovery in patients with schizophrenia spectrum disorders: a systematic review and meta-analysis Robin M. Van Eck, Thijs J. Burger, Astrid Vellinga, Frederike Schirmbeck, Lieuwe de Haan In: Schizophrenia Bulletin, 44 (2018), 631-642
20 Chapter 2 ABSTRACT Patients describe experiencing personal recovery despite ongoing symptoms of psychosis. The aim of the current research was to perform a meta-analysis investigating the relationship between clinical and personal recovery in patients with schizophrenia spectrum disorders. A comprehensive OvidSP database search was performed to identify relevant studies. Correlation coefficients of the relationship between clinical and personal recovery were retrieved from primary studies. Metaanalyses were performed, calculating mean weighted effect sizes for the association between clinical and personal recovery, hope, and empowerment. Additionally, associations between positive, negative, affective symptoms, general functioning, and personal recovery were investigated. The results show that heterogeneity across studies was substantial. Random effect meta-analysis of the relationship between symptom severity and personal recovery revealed a mean weighted correlation coefficient of r = −.21 (95% CI = −0.27 to −0.14, P < .001). We found the following mean weighted effect size for positive symptoms r = −.20 (95% CI = −0.27 to −0.12, P < .001), negative symptoms r = −.24 (95% CI = −0.33 to −0.15, P < .001), affective symptoms r = −.34 (95% CI = −0.44 to 0.24, P < .001) and functioning r = .21 (95% CI = −0.09 to 0.32, P < .001). The results indicate a significant small to medium association between clinical and personal recovery. Psychotic symptoms show a smaller correlation than affective symptoms with personal recovery. These findings suggest that clinical and personal recovery should both be considered in treatment and outcome monitoring of patients with schizophrenia spectrum disorders.
21 Clinical and personal recovery, a meta-analysis INTRODUCTION Recovery has become an increasingly important aspect of care in mental health services all over the world (1). Recovery-oriented practices have especially emerged for schizophrenia, which has traditionally been seen as a condition with an unfavorable course (2). Since the second half of the 20th-century patient organizations have challenged the assumption that people with schizophrenia cannot live a productive and satisfying life. Patients have emphasized that recovery can occur even when psychotic symptoms are persistent (3). Scientific- and patientbased influences have resulted in a clinical and a personal definition of recovery in schizophrenia (2). The clinical definition includes remission of symptoms and functional improvement. The Remission in Schizophrenia Working Group (RSWG) defines remission as improvements in core signs and symptoms to the extent that they are of such low intensity that they no longer interfere significantly with behavior (4). Operational criteria include a score of mild or less on specific items of a symptom scale over a 6-month period, eg, the Positive and Negative Syndrome Scale (PANSS) or the Brief Psychiatric Rating Scale (BPRS). The RSWG describes that recovery, besides being relatively free of disease-related psychopathology, implies the ability to function in the community, socially and vocationally. The RSWG states that recovery is a more demanding and longer-term phenomenon than remission and that remission is a necessary but not sufficient step toward recovery. Moreover, the RSWG writes that consensus regarding operational criteria for recovery, in particular, cognition or psychosocial functioning, was considered outside its scope, because more research is needed on this topic (4). Other authors have also included living independently, having friends (5) and scores of >65 on the Global Assessment of Functioning (GAF) to the criteria of clinical recovery (6). The patient-based definition of recovery has been developed based on narratives of individuals who have experienced mental illness (7, 8). Stories from the patient movement have shown that people with psychosis have the possibility of living a productive and satisfying life, despite ongoing symptoms (3). The most frequently cited patient-based definition is: “the development of new meaning and purpose in one’s life, as one grows beyond the catastrophic effects of mental illness (9).” Recovery from schizophrenia is not a uniform process but varies from person to person (10). The term personal recovery has been widely used in literature to describe the patientbased definition of recovery (11). Because personal recovery is different for every individual, it is hard to define common characteristics. Nonetheless, several authors have tried to capture important aspects of personal recovery in qualitative research. Andresen et al., by reviewing 2
22 Chapter 2 published experiential accounts of recovery by people with schizophrenia, have identified four key processes of personal recovery: finding hope; re-establishment of identity; finding meaning in life; and taking responsibility for recovery (12). Leamy et al. have identified similar categories of personal recovery processes, namely: connectedness, hope, identity, meaning, and empowerment (also known by the acronym “CHIME”) (13). In a more recent study, this framework has been validated as a defensible theoretical base for clinical and research purposes (14). Law and Morrison have found comparable elements in their Delphi study among individuals experiencing symptoms of psychosis (15). Several instruments have been developed, based on the experience of patients, to assess personal recovery (16-19), hope (20) and empowerment (21). In the treatment of patients with schizophrenia, the primary goal traditionally is clinical recovery. So far, inconsistent findings regarding the association between clinical and personal recovery have been published, from large (22) to very small correlations (23), usually negative, but also positive correlations (24). Insight into this association is important, because if clinical and personal domains do not overlap, this may inform mental health services to consider extending their treatment strategies beyond clinical goals to promote personal recovery. Moreover, it would support the implementation of instruments to measure personal recovery in outcome monitoring (25). In this meta-analysis, we aim to investigate the strength of the relationship between clinical and personal recovery in patients with schizophrenia spectrum disorders. Because connectedness, hope, and empowerment have most consistently been identified as relevant categories of personal recovery, we will also assess the relationship between these concepts and clinical measures (13). METHODS Literature search A comprehensive online OvidSP database search was performed, including Embase, MEDLINE, PsycINFO, EBM Reviews, and the Ovid Nursing database from the inception of the individual databases to February 2017, without limits concerning publication status or language domain. The following search terms were used: (“schizophrenia” OR “psychosis” OR “psychotic disorder”) AND ((“recovery” AND (“personal” OR “subjective”)) OR (“connectedness” OR “hope” OR “empowerment”)). In addition, the reference lists of articles that met inclusion criteria were used for further study identification. The title and abstract of each article were manually screened according to the following criteria: (1) the article possibly investigates the relationship between clinical and
23 Clinical and personal recovery, a meta-analysis personal recovery, or between aspects of both; (2) the article includes quantitative data. After the screening, the full-texts of the remaining studies were obtained. Study selection To be considered for inclusion in the meta-analysis, studies had to meet the following inclusion criteria: 1. DSM diagnosis of schizophrenia spectrum disorder in >90% of the study sample. The current DSM-5 definition of the schizophrenia spectrum was used, which includes the following diagnoses: schizophrenia, schizoaffective disorder, schizophreniform disorder, brief psychotic disorder and schizotypal (personality) disorder. The “other psychotic disorders”, including substance/medicationinduced psychotic disorder, psychotic disorder due to another medical condition, catatonia, and other specified and unspecified schizophrenia spectrum and psychotic disorders were excluded (26). 2. Clinical recovery was defined as severity of psychotic symptoms, measured with a valid clinical instrument, such as the Positive and Negative Syndrome Scale (PANSS); and/or as severity of emotional distress measured with a valid instrument such as the Calgary Depression Scale (CDS); and/or as functioning assessed with measures such as the GAF. 3. A valid measure was used to assess personal recovery (eg Recovery Assessment Scale [RAS]), hope (eg Beck Hopelessness Scale [BHS]), empowerment (eg Empowerment Scale [ES]) or connectedness (eg Social Connectedness Scale [SCS]). 4. Effect sizes or data that enabled effect size calculation or estimation were reported. We excluded studies without original data (ie articles which used the same patient sample), or not published in peer-reviewed journals (ie reviews, conference abstracts, book chapters). Data collection and items Data were collected directly from the text, correlation matrixes, or other statistical tables from the included studies. Sample size, mean age, gender, primary diagnosis, country of origin of the study, clinical setting, stage of the disorder (early psychosis or chronic), the mean duration of illness, clinical outcome measure used, personal recovery measure used, and effect sizes of the relationships between clinical and personal measures were extracted. Unfortunately, most studies did not report on the phase of the disorder (ie active or remission). Risk of bias in studies and study quality The methodological quality of individual studies was assessed by the Agency for Healthcare Research and Quality (AHRQ) assessment tool. This specific tool has been 2
24 Chapter 2 used in multiple reviews of observational studies (27-30). We used the version of the tool adapted by Taylor et al, which judges parameters such as sample selection, description of the cohort, and adequate reporting (31). We excluded 4 out of 10 items because these were only applicable for studies with comparison groups. We added 2 items, to assess whether the association between clinical and personal recovery was a primary or secondary outcome of the study, and to assess the method by which a diagnosis was made. This resulted in 8 items. For every study, we calculated a total quality score, see table 1. Data analysis Effect size calculation To assess the strength of the relationship between measures of clinical and personal recovery, Pearson’s correlation coefficient r was used as the measure of effect size. According to Cohen, r = .10 is considered a small effect, explaining 1% of the total variance); r = .30 a medium effect, accounting for 9% of the total variance) and r = .50 a large effect, accounting for 25% of the variance (32). When correlation coefficients were not given, they were either calculated from reported data according to Lipsey and Wilson, or estimated from reported standardized regression coefficients (33). Regarding the personal recovery category of hope, the direction of the reported correlation coefficient was reversed wherever necessary, such that all included effect sizes represented the association between clinical recovery and hope, instead of hopelessness. Integration of dependent effect sizes To ensure that every study only contributed one effect size to the analysis, we calculated average effect sizes within one study if multiple effect sizes were reported on similar outcomes (34-36). This was the case if 2 or more measures were used to assess overall symptom severity, or (an aspect of) personal recovery. When studies only reported the outcome of assessed subscales and not of the total score, we averaged effect sizes of subscales to estimate an overall effect size. For example, reported correlation coefficients between personal recovery and positive symptoms, negative symptoms and general psychopathology of the PANSS were averaged, to calculate an overall correlation between personal recovery and symptom severity. Meta-analysis procedure To account for expected heterogeneity between studies, random-effects models according to Hedges and Vevea (37) were calculated to obtain a combined effect weighted for sample size. The relationships between symptom severity on the one
25 Clinical and personal recovery, a meta-analysis hand and personal recovery, hope, and empowerment, on the other hand, were investigated separately. In addition to the analyses of overall symptom severity, separate analyses were conducted for positive, negative and affective symptoms and general functioning, if possible. The presence of heterogeneity was evaluated by calculating the I2 metric, i.e. the percentage of between-study variance due to systematic heterogeneity rather than chance (38). Data-analysis was performed with SPSS version 23 (39). Figures were plotted using Comprehensive Meta-analysis software (40). Heterogeneity was further explored by evaluating the effect of moderator variables. We were interested if estimated study quality as assessed with the AHRQ would affect reported effect sizes and/or cause heterogeneity. Furthermore, we planned to include age and illness duration or stage of illness as moderators. However, because the information was missing on these variables in a substantial part of the studies (table 1), only age could be added to the model. Meta-regression analyses were performed using SPSS 23 software, with macros provided by Lipsey and Wilson (33). Publication bias was examined by computing Rosenthal’s fail safe N and a funnel plot, which displays effect sizes plotted against the standard errors (35, 41). Data points on the funnel plot are ideally evenly distributed around the mean effect size, with asymmetry suggesting a publication bias. RESULTS Study selection Figure 1 shows the flow chart of the process of selecting relevant articles. The literature search resulted in 5191 publications, of which 3617 remained after removal of duplicates. A total of 178 publications were selected on the basis of title and abstract. Eventually, 35 studies from the literature search were included. By reviewing the references of the included publications, we found an additional 2 relevant articles (25, 42). Within the total of 37 included publications, 22 addressed overall personal recovery, 19 hope and 7 empowerment. No relevant articles were found concerning connectedness. See table 1 for study characteristics. Results of the meta-analysis Clinical and personal recovery The meta-analysis that investigated the relationship between overall symptom severity and overall personal recovery in patients with schizophrenia spectrum disorders included 20 studies, with a total of 3994 participants, most of which were male (70.4%) and outpatients. The mean age was 42.4, with an age range from 28.0 2
26 Chapter 2 to 54.3. One study did not report on gender and age (43). Individual sample sizes ranged from 25 to 825. Most studies reported on chronic patients, only one study specifically investigated early psychosis patients (44). The heterogeneity across studies was substantial (I2 = 75.8, 95% CI = 62.7-84.3) (45). Random effect meta-analysis of the relationship between overall symptom severity and personal recovery revealed a significant mean weighted correlation coefficient of r = -.21 (95% CI = -0.27 to -0.14, P < .001) (supplementary table 2). This implies that patients with higher severity of overall psychopathology reported slightly lower personal recovery. Figure 2 shows the forest plot of effect sizes and 95% confidence intervals, as well as z and P values, which provide an indication of the statistical significance of the association. Of the 20 studies, the majority reported on the association between personal recovery and different symptom domains: positive symptoms (k = 17, n = 3319), negative symptoms (k = 15, n = 2442), affective symptoms (k = 12, n = 2442). In addition to the association between personal recovery and overall psychopathology, we performed separate meta-analyses for all of these domains. The meta-analyses of positive and affective symptoms included one additional study, which only reported data on the association between these symptoms domains and personal recovery (46). One additional study only reported on the association between personal recovery and the GAF (47) resulting in 8 studies which reported on the association with general functioning (n = 1938). Again, heterogeneity was substantial across studies assessing positive symptoms (I2 = 74.8, 95% CI = 59.3-84.3), negative symptoms (I2 = 76.7, 95% CI = 61.8-85.8), and affective symptoms (I2 = 84.5, 95% CI = 74.4-90.6). Random effect meta-analyses revealed a significant negative mean weighted effect size for positive symptoms r = -.20 (95% CI = -0.27 to -0.12, P < .001), negative symptoms r = -.24 (95% CI = -0.33 to -0.15 P < .001) and affective symptoms r = -.34 (95% CI = -0.44 to -0.24, P < .001). A small significant positive effect size was found for the association with general functioning r = .21 (95% CI = -0.09 to 0.32, P < .001) (supplementary table 2). Meta-regression analysis We conducted a meta-regression analysis to evaluate if age or study quality potentially explained differences in reported effect sizes among studies and caused heterogeneity. A nonsignificant tendency for age to correlate with the effect size (B = .010, SE = .005, P = .056) was found and no effect for study quality. Together they only explained 14.5% of the variation between studies. Furthermore, no significant moderation effects were found in meta-regression analyses conducted for the different symptom domains. Differences in age and study quality explained 27.6% of the variation observed in the association with positive symptoms, 6.2% for negative symptoms, and 29.8% for affective symptoms.
27 Clinical and personal recovery, a meta-analysis Sensitivity analysis Rosenthal Fail-Safe N = 913 suggests that there would need to exist more than 900 negative unpublished studies to turn the estimated significant population effectsize into a nonsignificant result. The funnel plot does not indicate publication bias (supplementary material). Clinical recovery and hope Seventeen studies were included in the meta-analysis of the relationship between overall symptom severity and hope, with one publication included twice, because it reported on 2 studies in different populations (in Austria and Japan). Three thousand ninety-nine participants were analyzed, most were male (60.1%) and outpatients. The mean age was 37.2. One study did not report on gender and age (48). The majority of studies mainly included patients with longer duration of psychosis, only 2 specifically investigated patients with early psychosis (44, 49). Heterogeneity across studies was substantial (I2 = 64.9, 95% CI = 41.2-79.0). Results showed a significant mean weighted correlation coefficient of r = -.24 (95% CI = -0.30 to -0.187 P < .001), see supplementary table 2. Figure 3 shows the forest plot of effect sizes and 95% confidence intervals for the association between overall symptom severity and hope in the included individual studies. Subsequently, meta-analyses of the 3 separate symptom domains positive symptoms (k = 12, n = 2364), negative symptoms (k = 11, n = 1539) and affective symptoms (k = 14, n = 2669) were conducted. The meta-analyses of affective symptoms included 3 additional studies, which only reported data on the association between hope and affective symptoms (50-52). Again, heterogeneity between studies included in these analyses was substantially high for positive symptoms (I2 = 77.4, 95% CI = 60.7-87.0) and affective symptoms (I2 = 81.7, 95% CI = 70.4-88.7). Results showed significant negative mean weighted effect sizes for positive symptoms r = -.14 (95% CI = -0.23 to -0.05, P = .004), negative symptoms r = - .26 (95% CI = -0.32 to -0.19 , P < .001) and affective symptoms r = -.43 (95% CI = -0.51 to -0.35, P < .001). There were not enough studies to conduct a meta-analysis of general functioning. Meta-regression analysis Meta-regression analyses, which integrated age and study quality as potential predictors of the heterogeneity between studies, showed no significant effects. Together they only explained 4.7% of the variation observed regarding overall symptom severity, 25.4% regarding positive symptoms, 9.0% in the association with negative symptoms, and 17.4% in affective symptoms. 2
28 Chapter 2 Figure 1: Flow diagram study selection.
29 Clinical and personal recovery, a meta-analysis Sensitivity analysis Rosenthal Fail-Safe N = 869 suggests that there would need to be more than 800 negative unpublished studies included in the meta-analysis to result in a nonsignificant population effect size. The funnel plot does not suggest publication bias (supplementary material). Clinical recovery and empowerment Seven studies investigated the relationship between symptom severity and empowerment. Five of 7 studies (n = 1793) could be included in the meta-analysis, since in 2 studies it was not possible to calculate an effect size of the relationship between overall psychopathology and empowerment (53, 54). Of the 1793 participants, most were male (68.8%) and outpatients. The mean age was 40.6. All studies reported mainly on patients with longer duration of psychosis. Again, heterogeneity across studies was substantial (I2 = 86.7, 95% CI = 71.2-93.9, Q = 30.1, P > .001). The random-effect meta-analysis revealed a significant mean weighted correlation coefficient of r = -.23 (95% CI -0.36 to -0.09, P < .001). Figure 4 shows the forest plot of individual effect sizes and 95% confidence intervals for the association between overall symptom severity and empowerment. Due to the small number of included studies, no additional sub-sample analyses regarding different symptom domains and functioning, or meta-regression analyses were conducted. Sensitivity analysis Rosenthal Fail-Safe N = 118 suggests that there would need to be more than 100 negative unpublished studies included in the meta-analysis to result in a nonsignificant population effect size. The funnel plot does not suggest publication bias (supplementary material). 2
30 Chapter 2 Table 1: Studies on the Relationship Between Clinical Recovery and Personal Recovery, Hope and Empowerment in Schizophrenia Spectrum Disorders. Study N Age, Mean (SD) Gender, %M/%F Origin Setting Andresen et al(25) 110 39.5 (11.3) 54/46 Australia Outpatient Armstrong et al(55) 795 54.3 (9.4) 92/8 USA Outpatient Berry et al(56) 325 37.9 (9.7) 86.5/13.5 UK Outpatient Castelein et al(21) 50 31.4 (13) 72/28 Netherlands Mixed Cavelti et al(57) 156 44.5 (11.7) 66/34 Switzerland Outpatient Chou et al(58) 190 44.9 (11.6) 50/50 Taiwan Outpatient Díaz-Mandado et al(59) 43 36.7 (8.1) 74.4/25.6 Spain Outpatient Giusti et al(24) 76 45.8 (12.7) 57.9/42.1 Italy Inpatient Gottschalk et al(48) 23 ? ? USA ? Hofer et al(60) Japan: 60 Austria: 52 J: 45.9 (10.0) A: 44.4 (10.7) J: 37/63 A: 52/48 Japan and Austria Outpatient Jahn et al(61) 169 51.9 (8.5) 87.5/12.5 USA Outpatient Jørgensen et al(62) 101 37.5 (12.6) 47/53 Denmark Outpatient Kukla et al(63) 119 47.6 (8.8) 74/26 USA Outpatient Kukla et al(64) 68 50.2 (11.0) 94.1/5.9 USA Outpatient Landeen et al(65) 55 34.9 75/25 Canada Outpatient Landeen et al(66) 70 31.1 (6.3) 60/40 Canada Mixed Law et al(67) 335 36.0 (11.6) 66.3/33.7 UK Mixed Law et al(68) 110 37.3 (11.6) 69/31 UK Mixed Lecomte et al(49) 150 25.0 (6.4) 61/39 Canada Outpatient Lysaker et al(50) 52 44 100/0 USA Outpatient Lysaker et al(69) 25 50.7 (10.2) 96/4 USA Outpatient
31 Clinical and personal recovery, a meta-analysis Stage of disorder Duration of Illness in Years, Mean (SD) Clinical Recovery Instrument Personal Recovery Instrument Quality Score (0-12) Mixed <5: 14.2% ≥5: 71.8% Unknown: 14.1 % HoNOS K-10 GAF RAS MHRM 5 Chronic ? BPRS GAF MHRM 8 Chronic 12.1 (0.24) PANSS GAF MDES 9 Mixed 6.5 (6.3) CAPE ES PES MHCS 10 Chronic 17.9 (11.7) PANSS BDI CDS GAF RAS 10 Chronic 21.9 (10.4) PSS ES 9 Chronic 24.3 (6.6) PANSS RAS 9 Chronic 20.5 (10.9) PANSS BPRS RAS 11 ? ? BPRS HRSD GHS 7 Chronic J: 18.9 (10.6) A: 15.4 (10.5) PANSS BHS 11 Chronic Range: 3 or more PANSS BSI MARS 6 Chronic 10 PANSS RAS 9 ? ? PANSS RAS SHS 9 Chronic 20 PANSS RAS 10 Chronic 12.7 (range 2-26) PANSS MHS 10 Chronic 7.4 (4.7) PANSS BDI MHS 11 Mixed ? PANSS CDS QPR BHS 8 Mixed ? PANSS GAF QPR 6 Early psychosis Range: 0-2 BPRS LOT 10 Chronic 22 PANSS BHS (inverted) 10 Chronic 25 PANSS STAND 9 [continued on next page] 2
32 Chapter 2 Table 1: [continued] Study N Age, Mean (SD) Gender, %M/%F Origin Setting Lysaker et al(70) 143 46.8 (9.6) 92/8 USA Outpatient Macpherson et al(42) 403 44 (11) 64/36 UK Outpatient Morrison et al(71) 122 35.5 (11.5) 88/34 UK Mixed Norman et al(44) 84 28 (7.4) 69/31 Canada Outpatient Oliveira et al(47) 101 52.2 (13.8) 76.2/23.8 Portugal Outpatient Resnick et al(72) 825 44.6 (12) 71.6/28.4 USA Mixed Ringer et al(51) 52 46.6 (9.2) 100/0 USA Outpatient Roe et al(23) 159 43.2 (10.7) 66.7/33.3 Israel Outpatient Schrank et al(52) 200 40.3 (12.2) 62.5/37.5 Austria Mixed Schrank et al(73) 284 39.9 (12.6) 58.1/41.9 Austria Mixed Sibitz et al(53) 157 37.3 (11.9) 54.5/45.5 Austria Mixed Snyder et al(43) 104 ? ? USA Inpatient Vass et al(22) 80 39.2 (11.6) 61.3/38.7 UK Outpatient Wciórka et al(74) 110 38.3 (11.4) 39/61 Poland Mixed Wciórka et al(54) 110 38.3 (11.4) 39/61 Poland Mixed Wood and Irons(46) 52 37.0 (13.0) 59.6/40.4 UK Mixed Note: ADS = Allgemeine Depressionsskala: German version of the widely used CES-D (Center for Epidemiological Studies Depression Scale); BDI = Beck Depression Inventory; BHS = Beck Hopelessness Scale; BPRS = Brief Psychiatric Rating Scale, BSI: Brief Symptom Inventory; CAPE = Community Assessment of Psychic Experiences; CDS = Calgary Depression Scale; ES = Empowerment Scale; GAF = Global Assessment of Functioning; GHS = Gottschalk Hope Scale; HADS= Hospital Anxiety and Depression Scale; HHI = Herth Hope Index; HoNOS = Health of the Nation Outcome Scale; HRSD = Hamilton Rating Scale for Depression; IHS = Integrative Hope Scale; K-10 = Kessler Psychological Distress Scale; LOT = Life Orientation Test; MARS= Maryland Assessment of Recovery in People With Serious Mental Illness; MDES = Making Decisions and Empowerment Scale; MHCS = Mental Health Confidence Scale; MHRM = Mental Health Recovery Measure; MHS = Miller Hope Scale; PANSS = Positive and Negative Syndrome Scale; PES = Personal Empowerment Scale; PSS = Psychiatric Symptoms Scale;
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