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Experiences of stigma among family members of persons living with schizophrenia: a systematic review protocol

Maiocco, Susan1; Shelley, Ellen1; Salmond, Susan1,2; Jewell, Sarah T.3; Caldwell, Barbara1; Lieggi, Michelle4

JBI Database of Systematic Reviews and Implementation Reports: June 2017 - Volume 15 - Issue 6 - p 1575–1584
doi: 10.11124/JBISRIR-2016-003150
SYSTEMATIC REVIEW PROTOCOLS

Review question/objective: : The aim of this qualitative systematic review is to explicate the experiences of stigma among family members of persons living with schizophrenia in any setting where they receive care. More specifically, the review will: (i) describe the experiences of stigma among family members of persons living with schizophrenia, (ii) describe the factors that influence the experiences of stigma, and (iii) describe the strategies and approaches used to cope with the stigma.

1School of Nursing, Rutgers, The State University of New Jersey, Newark, USA

2The Northeast Institute for Evidence Synthesis and Translation (NEST): a Joanna Briggs Institute Center of Excellence

3George F. Smith Library of the Health Sciences, Rutgers, The State University of New Jersey, Newark, USA

4Fishbon Library, University of California San Francisco Medical Center, San Francisco, USA

Correspondence: Ellen Shelley, shelleee@sn.rutgers.edu

There is no conflict of interest in this project.

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Background

Schizophrenia is characterized by “disordered cognition, including a gain of function in psychotic symptoms and a loss of function in specific cognitive functions, such as working memory, but without the progressive dementia that characterizes classical neurodegenerative disorders”.1 (p.1432) It is a debilitating disease characterized by a diverse range of symptoms, none of which are unique to the disease itself.2 The Diagnostic and Statistical Manual of Mental Disorders-5 divides the clinical symptoms of schizophrenia into both positive and negative symptoms. Positive symptoms represent behaviors that are not generally seen in healthy people and include hallucinations, delusions, movement disorders, thought disorders as well as bizarre behaviors. The negative symptoms represent disruptions in or lack of normal feelings and include reductions in emotional expressions (flat affect and speech), social withdrawal, disinterest, reduced anticipatory pleasure and motivation, and feeling out of touch.3 Symptom patterns vary case by case, but manifestations typically impact emotion, affect, perception, thought, speech, behavior and motor activity.4 A consequence of these symptoms is a marked personality change, social isolation, occupational disability, cognitive impairment and poor health.5 Worldwide prevalence estimates range between 0.5% and 1.1%, regardless of racial, ethnic or economic background.6,7 Age at first episode is typically 21 years among men and 27 years among women.6 Approximately 3.5 million people in the United States are diagnosed with schizophrenia and over 21 million people worldwide.8 The rate ratio for schizophrenia for males to females is 1.4:1, hence it is more prevalent in men than women with overall estimates of 12 million males and nine million females.7,9

Significantly, schizophrenia is one of the leading causes of disability, ranking among the top 10 leading causes of disability-adjusted life years worldwide.7 It presents a devastating health, social and economic burden, not only for patients but families, other caregivers and the wider society.10 Treatment and other economic costs due to schizophrenia are estimated to be between $32.5 and $65 billion annually.7 People diagnosed with schizophrenia face one of the highest unemployment rates, among all occupational-deprived groups, and in part the low rates of employment are not the outcome of the condition itself but prejudices arising from the stigma accompanying the condition.11 The stigma associated with mental illness globally is the greatest impediment to improving the lives of people with schizophrenia and severe mental illness.

Goffman12 described stigma as an “attribute that is deeply discrediting” and makes the person carrying it “different from others and of a less desirable kind” with a quality of being “not quite human”.12 (p.3,5) The Center for Disease Control and Prevention similarly defines stigma as a discrediting attribute to the bearer, causing feelings of shame and isolation.13 The experience of stigma can impose a discrediting label and negative stereotype with consequences of social isolation, status loss and discrimination.14 As a result, prejudice, avoidance, rejection and discrimination are directed at the person and their family members/relatives, causing suffering, denial of symptoms, delay of treatment, alteration of daily activities, and withdrawal from social relationships and events.15

Stigma affects the experiences of not only individuals possessing the stigmatizing characteristics but also family members/relatives and close associates with whom the stigmatized person interacts – an experience referred to as courtesy stigma.16 Courtesy stigma is attached to those who merely associate with the stigmatized person. Associative stigma is an extension of psychiatric stigma to those who care for patients, like family members.17 The associates experience stigma, not because of any individual characteristic, but because of their affiliation with the stigmatized individual. Catthoor et al. 17 indicate that according to research, associative stigma appears to be culturally determined and is greater in family members who have mental health problems and who doubt if the diagnosis of the patient is of any significance. The results of a quantitative study conducted by Catthoor et al.,17 in Flanders assessed the presence and severity of associative stigma and factors for higher associative stigma in 150 family members of psychotic patients and showed that the prevalence of associative stigma in this sample was 86%. The prevalence was found to be higher than other previous international research. It was also found that disruptive behavior by the patient toward family members who resided with the patient was the most accurate predictor of higher associative stigma.17

Stigma, whether experienced by the patient or as a courtesy experience, is categorized as public stigma, perceived/anticipated stigma, experienced stigma and self-stigma.18 Public stigma is defined as “the general population endorsing prejudice and manifesting discrimination toward mental illness; perceived/anticipated stigma refers to people's beliefs about attitudes of the general population towards their condition and towards themselves as members of a potentially stigmatized group; experienced stigma refers to discrimination or restrictions met by the affected person; and self-stigma is defined as a type of identity transformation that might lead to the loss of previously held positive beliefs about the self which in turn yields negative consequences for the person such as diminished self-esteem and self-efficacy”.18 (p.155) Other literature describes public stigma as comprising negative attitudes (prejudice), beliefs (stereotypes) and behavior (discrimination) toward the stigmatized person, and self-stigma as internalization of these experiences by the stigmatized individual.19

Stigma is widespread and disrupts multiple life domains (e.g. resources, social relationships and coping behaviors) for both the individual with schizophrenia and their family members/relatives.14 In fact, the deleterious consequences of stigma may be even more problematic than the difficulties arising from the symptoms of the disease itself.20 Stigma or the reactions of the social environment associated with the illness become a dual burden as the family must deal with the disease and the stigma.21 It is said that “of all the mental illnesses, schizophrenia appears to be the most stigmatized disorder” and adds a “dimension of suffering to the illness experience” which contributes further to “social isolation, limited life chances and delayed help-seeking behavior”.18 (p.299) In focus group interviews with 122 members of relatives of family members/relatives with schizophrenia who were also members of advocacy groups, 65.3% experienced stigma in inter-personal interaction, 27.7% in structural discrimination, 4.5% in the public image of mental illness and 2.5% in access to social roles.16

The stigma of having a family member with schizophrenia, often characterized in the media as violent and threatening, can interfere with family members’/relatives’ social interactions, limit their personal activity, and reduce and strain friendships and relationships.22 The stigma of shame often keeps family members/relatives isolated and alienated with increased emotional distress.23 Family members/relatives report feelings of rejection and avoidance by others, resulting in feelings of hurt, shame and disappointment.24 Parents often feel isolated and historically have felt blame, which often leads to feelings of guilt and shame. A qualitative study examining how family members/relatives experiencing courtesy stigma associated with mental illness coped with these challenges found that families gave careful consideration to the major tasks of who to disclose to or conceal information from and whether or not to confront or ignore stigmatizing behavior.24 Reluctance to share information about the diagnosis, shame and feelings of guilt can lead to a delay or avoidance of treatment.25 Many report hiding the diagnosis for fear of the negative reactions from the community.26 In a study of family members/relatives in Taiwan, interviews revealed that the stigma of shame discouraged caregivers from looking for social support networks.23 In a cross-sectional study of Ethiopian family members/relatives, relatives of those living with schizophrenia experienced financial problems, social problems and difficulties with work and family burdens.27

Feelings of survivor guilt among siblings, the feeling of “being the normal one who has managed to escape”28 (p.20) and a sense of bereavement that they have lost the sibling they have once known have been documented. Siblings often experience resentment, feel sandwiched between the parent's loyalty to the sick sibling and the other family members/relatives, and are designated as the involuntary caregiver when something happens to the parent.29

The stigma of shame is an important factor in the isolation of the family or caregiver, and the sense of alienation from community and society at large, which have a significant impact on their quality of life.23 The widespread occurrence of stigma impacts on family members’ social relationships, personal relationships with the family member with schizophrenia and severe mental illness, personal relations with other family members/relatives, and their own health.26 International research reports consistently document that caregivers of persons with schizophrenia are critical of the negative attitudes toward them and describe experiences of being excluded from the decision making process of the person being cared for, being blamed for the affected person's illness, having their knowledge undervalued, and receiving insufficient medical information and support from the mental health clinician.30 Better understanding of public, perceived/anticipated, experienced and self-courtesy stigma will assist providers in being more attuned to and empathetic with families. Identifying the facilitators and barriers to the different courtesy stigma experiences and coping strategies can inform the development of evidence-based guidelines to pro-actively guide and support family members experiencing courtesy stigma and to help shape public policy in the prevention of stigma in patients and their family members. Understanding the strategies and coping mechanisms that family members utilize to deal with stigma can provide insight into practices that may help to alleviate the negative effects of stigma.

To date, no other systematic review of the experience of stigma among family members of persons with schizophrenia has been conducted. Learning about and understanding the experience of courtesy stigma among family members of persons with schizophrenia will contribute to the development of evidence-based practice guidelines for assisting family members in developing positive actions and behaviors to reduce and alleviate the psychological distress and isolation that is often experienced.

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Inclusion criteria

Definitions

For the purpose of this systematic review protocol, definitions of the participant are defined below:

Family member: “Any individual that may or may not be related to the participant by blood or affinity or in a commitment to a permanent relationship and a perceived reciprocal obligation to care for each other.”31 (p.117)

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Types of participants

The current review will consider individuals who have identified themselves as family members who have on-going contact, past or current, with individuals who have a self-reported diagnosis of schizophrenia, no matter what their age is.

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Phenomena of interest

The current review will consider studies that investigate the perceptions, experiences and meaning of courtesy stigma in family members of those persons living with schizophrenia and the strategies, approaches and factors that contribute to the stigma or that help to alleviate, cope and deal with stigma.

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Context

The current review will focus on studies that address courtesy stigma associated with family members of individuals with schizophrenia and severe mental illness, whether they are residing in the community, group home or outpatient residential facility or receiving intermittent hospital care.

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Types of studies

This review will consider studies that focus on qualitative data including, but not limited to, designs such as phenomenology, grounded theory, ethnography, action research and mixed methodology studies where the qualitative component yields in-depth descriptions, from 1963 to the present. In addition, case studies describing personal experiences of family members experiencing courtesy stigma will be included.

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Exclusion criteria

Those who have not had on-going contact with the individual with schizophrenia or have had rare contact will be excluded.

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Search strategy

The search strategy aims to locate both published and unpublished studies authored in English from 1963 through December 2016. Otey and Fenton32 state that Goffman first used the term stigma over 40 years ago in 1963 in connection with mental illness in his influential writing of Stigma: Notes on the Management of Spoiled Identity. 33 A three-step detailed search strategy will be used. An initial limited search of MEDLINE and Cumulative Index to Nursing and Allied Health Literature (CINAHL) will be undertaken followed by an analysis of the text words contained in the title and abstract, and of the index terms used to describe the article. A second search using all identified keywords and index terms will then be performed across all included databases. Appendix III provides the terms used for the database searches. The third search strategy step will be a search of the reference list of all identified reports and articles for additional studies. In addition, hand searching the table of contents of two key journals – Journal of Psychiatric Rehabilitation and Journal of the American Nurses Psychiatric Association – will also be performed for the most current three years. There are sufficient research-based data available to draw upon that are related to the aim of this systematic review protocol. All papers retrieved will be independently assessed, in duplicate, for methodological quality by two reviewers; for methodological validity prior to inclusion in the review using the standardized critical appraisal instruments from the Joann Briggs Institute Qualitative Assessment and Review Instrument (JBI-QARI) (Appendix I). Any disagreements that arise between the reviewers will be resolved through discussion or in consultation with a third reviewer.

The databases to be searched include:

  • Academic search Premier
  • CINAHL
  • HealthStar
  • JSTOR
  • MEDLINE (medical literature on-line)
  • PsycINFO
  • Psychology and Behavioral Sciences Collection
  • Sociology Abstracts
  • Web of Science.

The search for unpublished studies will include:

  • ProQuest Dissertations and Theses
  • Virginia Henderson Nursing Library
  • Robert Wood Johnson Foundation Research and Publications
  • Health Resources and Services Administration Special Projects of National Significance.

The search for gray literature will include Greynet at http://www.greynet.org

Initial keywords to be used will be: schizophrenia, prejudice, discrimination, public stigma, personal stigma, internalized stigma, courtesy stigma, qualitative research, phenomenology, grounded theory, ethnography, action research, family members, stigma and inherited stigma.

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Assessment of methodological quality

All papers retrieved will be assessed for methodological quality by two reviewers, independently and in duplicate, for methodological validity prior to inclusion in the review using the standardized critical appraisal instruments from the Joann Briggs Institute Qualitative Assessment and Review Instrument (JBI-QARI) (Appendix I). Any disagreements that arise between the reviewers will be resolved through discussion or in consultation with a third reviewer. Results of the search will be illustrated in a PRISMA diagram. To be eligible for inclusion, each study will be required to have a score of six out of the 10 criteria in the JBI-QARI instrument, in which ‘yes’ scores for a minimum of question numbers 2, 3, 4 and 8 are mandatory (Appendix I).

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Data extraction

Qualitative data will be extracted from papers included in the review by two reviewers, independently and in duplicate, using the standardized data extraction tool from JBI-QARI (Appendix II). Any disagreements that arise between the reviewers will be resolved through discussion or in consultation with a third reviewer. The data extracted will include specific details about the phenomena of interest, populations, study methods and outcomes of significance to the review question and specific objectives. The level of congruency between findings and supporting data will be graded for credibility based on three levels: unequivocal, credible or unsupported. Authors of relevant primary studies will be contacted for any required information.

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Data synthesis

Qualitative research findings will, where possible, be pooled using JBI-QARI. This will involve the aggregation or synthesis of findings to generate a set of statements that represent aggregations through assembling the findings rated according to their quality and categorizing these findings on the basis of similarity in meaning. These categories will then be subjected to a meta-synthesis to produce a single comprehensive asset of synthesized findings that can be used as a basis for evidence-based practice.

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Appendix I: Appraisal instruments

JBI critical appraisal checklist for qualitative research

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Appendix II: Appraisal instruments

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Appendix III: Search strategy

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Keywords:

Courtesy stigma; family members; qualitative research; schizophrenia; stigma

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