Background

People with intellectual disability (ID), or formerly known as mental retardation (MR), have stereotypically been viewed as diseased or mentally handicapped.1 In the past, many people with ID were institutionalized in the hope of being cured and treated for having a debilitating condition.1 During this time, much of the therapeutic procedures were strengthened by the medical model of disability.2 The medical model views any form of illness or impairment as a problem that can only be fixed pathologically.2 However, the rise of normalization, a principle firstly articulated by Bengt Nirje in the 1960s and extended by Wolf Wolfensberger in the 1970s, challenged the underpinnings of the medical model.2 The normalization principle aimed for everyday living conditions to be made available to people with ID.2 It initiated the cessation of the institutional era in the attempt to support community integration and eradicate dehumanizing practices.2 The facilitation of social inclusion was also beginning to emerge as a result of normalization.3 The normalization principle was closely allied with the social role valorization (SRV) framework.3

Social role valorization was founded by Wolf Wolfensberger in 1983 as an extension to the normalization principle to address psychological and social challenges centered around human services toward vulnerable individuals with learning disabilities.3 This equality initiative provided people with ID opportunities to participate in society more inclusively.3 This gave rise to the social model of disability founded by Mike Oliver in 1983 which further supported the underpinnings of the SRV framework.4 The social model indicated that disability is socially constructed.4 This model did not disregard a person's disability or condition; however, it believed necessary accommodations should be made available to promote diversity and enable full participation in society.4 While the social model provides reassurance and hope to people with ID by integrating people with disabilities as part of mainstream society, societal stigma in the form of personal and systematic barriers remains prevalent as many are still in favor of ableism.5

Within an ableist culture, people with ID have been attributed as eternal children and asexual.6 Many have been thought to be incompetent of sexual expression and functioning and one's right to sexuality is usually subdued or coercively taken away.7 Moreover, heteronormativity enforces stringent gender binary in society.8 Heterosexuality is seen as the default sexual orientation.8 Individuals with ID who pursue sexual relationships with members of the same sex or embraced a sexual orientation different to the norm were seen as unnatural and immoral.9 Jones and Magowan10 noted that members of the lesbian, gay, bisexual and transgender (LGBT) populace with ID have articulated having one's sexual orientation denied and in some circumstances receiving disciplinary action in the form of adverse punishments.

The universal declaration of human rights proclaimed in 1994 that "all human beings are born free and equal in dignity and rights".11^(p.4) Contrary to this assertion, Noonan and Taylor Gomez12 reported that sexual inequalities remain prevalent among adults with ID who identify as LGBT. People with ID who identify as LGBT are seen as a minority within a minority.13 Abbott and Howarth14 coined this term as double minority. McCann et al.1 elaborated on the term double minority, proposing that LGBT individuals with ID are faced with layered stigma. Not only are individuals faced with discrimination as a result of one's ID label but also stigmatized for their LGBT status in society.6,14 The detrimental effects of layered stigma can generate segregation, therefore limiting the facilitation of meaningful social and sexual relationships.6 Cochran et al.15 noted that issues relating to homophobic and transphobic hatred, and emotional and physical violence such as bullying and harassment have increased the vulnerability of LGBT individuals with ID toward drug and alcohol misuse. Furthermore, Baxter et al.16 proposed that acceptability of same-sex affiliations among people with ID can also be difficult due to austere cultural and religious beliefs. People with ID have been faced with opposition from family members, friends and professional staff when sexual identities of a LGBT nature are discussed.17

A study conducted by Davidson-Paine and Corbett,18 which explored the experiences of homosexual men with ID, suggested that a vast majority of men experienced psychological distress at some point in their lives. D'Augelli and Hershberger19 and Rotheram-Borus et al.20 also noted that, in dire situations, gay men with ID have exercised self-harm, either intentionally ending one's life or attempted suicide, to alleviate emotional distress and combat the stigma associated with internalized homophobia. Burns and Davies21 reported that lesbian women with ID were underrepresented in society when compared to gay men with ID. For example, male homosexuality has been clearly defined and overrepresented through the media and other platforms.21 Gratton22 and Herek23 noted that gay men in general have higher visibility within the community. Visibility surrounding lesbianism, bisexualism and transsexualism is far less.21,24 Attitudes toward lesbianism remains ambiguous and unclear, which can limit opportunities for lesbian women with ID to develop positive sexual identities and express their true self more liberally.21

An existing literature review conducted by McCann et al.6 explored the experiences and support needs of people with ID who identify as LGBT from a service user point of view. The term "service user" in the context of disability refers to an individual seen as a client accessing healthcare services or is a recipient of support from an approved disability service provider.25 The literature review explored the support needs of participants from an objective standpoint and suggested that future research and educational and policy initiatives should embrace person centeredness.6

An integral element of person centered planning is taking into account the subjective knowledge by drawing upon individual perceptions and expressed needs to formulate a personalized meaning.26 Person centered planning also strengthens a person's voice by identifying what's important to that person based on individual merits.26 Carson et al.27 postulated that, due to the unfixed and multi-dimensional nature of realism, knowledge acquired from realities could only be subjectively determined. Wilson et al.28 recently conducted a narrative review of the literature pertaining to people with ID who identified as lesbian, gay, bisexual, transgender, intersex or questioning (LGBTIQ). This review however only included published studies and used limited search terms. Furthermore, it did not convey information pertaining to "lived experiences" but focused solely on the key issues faced by people with ID who identify as LGBTIQ.

Despite lacking generalizability and reliability, subjective knowledge from an interpretive perspective is weighted highly in terms of validity, enabling the researcher to gain an emphatic and deeper understanding of the lives of people.29 Personal reflections can also reinforce one's right to sexuality as it not only provides people with ID opportunities to actively voice any concerns relating to identity formation but also create further LGBT awareness in society.30 Abbott30 noted that other areas of life such as employment and mental health often take more precedence over sexuality, let alone homosexuality which continues to be a stigmatized sexual identity. Carabine31 argued that the impact of personal biographies in the form of first-hand accounts can also bring about positive change, triggering policy makers to reassess existing attitudes and values relating to non-conformist sexuality. In the hope of reducing prejudice and granting an improved social position among people with disabilities, Abbott30 indicated that the art of personal reflections in the form of firsthand accounts and storytelling could promote further awareness and exposure of LGBT sexuality within society.

To date, there is no evidence of existing systematic reviews conducted on the proposed topic. Given the emerging concept and importance placed on sexuality, in particular, the wide spectrum of non-heterosexual sexual orientations, as well as existing studies having focused solely on heterosexuals with ID,30 this systematic review is needed to give equal precedence to these sexual minorities within the disability populace. This review will consider the lived experiences of adults with ID who identify as lesbian,gay, bisexual, transgender, queer or questioning, intersex or asexual (LBGTQIA) to not only provide an authentic reflection about one's life pertaining to sexuality but also attempt to "bring lost or silent voices to the fore".32^(p.102) The findings from this review may also highlight gaps in current research and provide new perspectives for future research in a timely fashion, leading to better personal and social outcomes for adults with ID who identify as LGBTQIA.

The findings from this review will offer deeper understanding of the lives of adults with ID who identify as LGBTQIA. The findings will be of benefit to existing qualitative and quantitative research by contextualizing current evidence to inform areas for future research centered on an idiographic focus. The findings will also initiate further exploration of under-researched matters surrounding LGBTQIA and ID sexuality, and inform disability and allied health practitioners on the importance of considering the individual's perspective during service delivery to achieve the best possible outcomes pertaining to sexual health and overall wellness.

The review will not consider cross-dressers or formerly known as transvestites. The identity of cross-dressers do not necessarily denote homosexuality but merely involves gaining some of form of pleasure in dressing up in clothes of the opposite gender.33 Witten33 however proposed that most practicing cross-dressers identify as heterosexuals with a small minority who might experience gender dysphoria or identify as homosexuals undergoing the transitioning phase.

The review will also not include studies related to children. Clavering and McLaughlin34 asserted that while there was a growing body of recognition to consider the subjective voices of children, conducting sexuality research among minors could prove difficult due to the sensitive and confrontational content to which participants were likely to be exposed. Parents of children have a tendency to disapprove sexuality research among minors as many are afraid of causing further harm to their child's wellbeing.34 Furthermore, children with various disabilities may have not developed sexual maturation or fully understand the concept of sexuality and may therefore convey inaccurate information or feel obliged to respond biasedly.35 This can significantly affect the credibility and transferability of the research findings.35

Inclusion criteria

Types of participants

The review will consider studies that:

* Include adults (aged 18 and over) who have a formal diagnosis of ID under the DSM-5 or MR under the DSM-4. (As noted above, children have not been included due to the nature of sexuality research.)

* Do not state the ID or MR diagnosis but have clearly specified the participant's disability within the title, abstract or content.

* Include LGBTQIA adults who have self-identified or have an unconfirmed diagnosis of ID or MR.

Due to the prevalence of comorbidities associated with ID or MR, participants with dual/multiple diagnoses whereby ID or MR is their primary or secondary diagnosis will be also considered. All participants will need to identify as having a LGBTQIA sexual orientation in conjunction with their ID or MR diagnosis/condition. Cross-dressers with ID or MR who are subject to gender dysphoria or have self-identified as embodying a T, Q or I identity will be considered. However, as noted before, cross-dressers mostly identify as heterosexuals, and if that is the case, they will not be included.

Phenomena of interest

The current review will explore the lived experiences in the form of personal reflections and first-hand accounts of adults with ID related to identifying as LGBTQIA.

Context

The review will explore lived experiences of adults with ID who identify as LGBTQIA within mainstream society. Mainstream society in this review is a term commonly used within the disability discourse referring to the inclusion of people with disabilities in the wider community, that is, alongside people without disabilities as a result of deinstitutionalization/community inclusion initiatives. Studies conducted across diverse countries, support and rehabilitation groups, community housing, residential settings, disability organizations and various cultural and social settings will be considered.

Types of studies

The current review will mainly consider interpretive studies that draw on the lived experiences of adults with ID who identify as LGBTQIA but not limited to designs such as phenomenology, grounded theory, ethnography, action research and feminist research. To capture the totality of data, other research designs determined qualitatively will be considered and for studies that contain both qualitative and quantitative components, only the qualitative components will be considered. For papers that do not directly specify first-hand accounts of the researched populace, the author's interpretation of the lived experiences of adults with ID who identify as LGBTQIA may also be considered, depending on the credibility of findings. Authors of primary studies will be contacted for clarification or missing information.

Search strategy

The search strategy will aim to find both published and unpublished studies. The search strategy will consist of three phases. In the first phase, an initial search of CINAHL will be undertaken followed by the analysis of text words contained in the title and abstract and the index terms (subject headings) used to capture the essence of an article. In the second phase, the identified key words and index terms will form the development of a search strategy which will then be tailored to suit the various search platforms. A full search (phase two) on CINAHL is detailed in the Appendix I. In the third phase, hand searching will be used to review the reference list of all retrieved papers to extract additional studies that have been missed or not properly indexed.

The search will include studies published in English between 1990 to the most recent as research surrounding LGBTQIA and ID has progressively gained more precedence and recognition. The start date was attained through a rigorous review of the preliminary literature, outlined in the Appendix I where no studies relating to the researched topic were found prior to 1990. Date limitation was not used when searching for relevant papers. Reference lists of relevant papers were also examined to identify additional papers in the attempt to locate the appropriate "start date".

The databases within the designated platforms to be searched include:

* CINAHL (via EBSCOhost)

* MEDLINE Complete (via Ovid)

* PsycINFO (via Ovid)

* ScienceDirect (via Elsevier)

* Eric (via ProQuest)

* Scopus (via Elsevier)

* PubMed

* REHABDATA Database (via NARIC)

* Wiley Online Library (via Wiley)

* Google Scholar.

Reference lists of articles will be checked and key articles will be cross-checked in citation indexes. The search for unpublished studies will include gray literature and unpublished materials. For example, government reports, conference papers and dissertations will be sourced. The sources searched to locate unpublished studies include:

* ProQuest Dissertations and Theses Global

* Websites of relevant associations

* Internet search engines (e.g. Google Advanced)

* Government reports

* Book chapters.

Study selection

Following the search, all identified citations will be collated and uploaded into Endnote (Clarivate Analytics, PA, USA)/Joanna Briggs Institute System for the Unified Management, Assessment and Review of Information (JBI-SUMARI) and duplicates removed. Titles and abstracts will then be screened by the primary author and research supervisors who will act as independent reviewers for assessment against the inclusion criteria for the review. Studies that meet the inclusion criteria will be retrieved in full and their details imported into JBI-SUMARI. The full text of selected studies will be retrieved and assessed in detail against the inclusion criteria. Full text studies that do not meet the inclusion criteria will be excluded and reasons for exclusion will be provided in an appendix in the final systematic review report. Included studies will undergo a process of critical appraisal. The results of the search will be reported in full in the final report and presented in a Preferred Reporting Items for Systematic Review (PRISMA) flow diagram. Any disagreements that arise between the reviewers will be resolved through discussion, or with a third reviewer.

Assessment of methodological quality

Selected studies will be critically appraised by the primary author and research supervisors at the study level for methodological quality in the review using the Joanna Briggs Institute Qualitative Assessment and Review Instrument.36 Any disagreements that arise between the reviewers will be resolved through discussions, or with a third reviewer. The results of critical appraisal will be reported in narrative form and in a table. All studies, regardless of the results of their methodological quality, will undergo data extraction and synthesis.

Data extraction

Qualitative data will be extracted from papers included in the review using the standardized data extraction tool from JBI-SUMARI. The data extracted will include specific details about the populations, context, culture, geographical location, study methods and the phenomena of interest relevant to the review question and specific objectives. Findings, and their illustrations, will be extracted and assigned a level of credibility.

Data synthesis

Qualitative research findings will, where possible, be pooled using JBI-SUMARI with the meta-aggregation approach.36 This will involve the aggregation or synthesis of findings to generate a set of statements that represent that aggregation, through assembling the findings and categorizing these findings on the basis of similarity in meaning. These categories are then subjected to a synthesis in order to produce a single comprehensive set of synthesized findings that can be used as a basis for evidence-based practice. Where textual pooling is not possible, the findings will be presented in narrative form.

Assessing confidence

The final synthesized findings will be graded according to the ConQual approach for establishing confidence in the output of qualitative research synthesis and presented in a Summary of Findings table.37 The Summary of Findings table includes the major elements of the review and details how the ConQual score is developed. Included in the table is the title, population, phenomena of interest and context for the specific review. Each synthesized finding from the review is then presented along with the type of research informing it, a score for dependability, credibility and the overall ConQual score.

Acknowledgements

I would like to thank the following people for their comments and contributions that have assisted in the development of the protocol: Ms Nikki May and Mr Peter Newman (Liaison Librarian from Flinders University) and Associate Professor Zachary Munn (Transfer Science, Joanna Briggs Institute).

Appendix I: Search strategy (CINAHL)

A logic grid was used to facilitate systematic search of the literature. The table below illustrates the identified subject headings along with the diverse range of synonyms in the form of text words based on phase two of the search strategy. Truncations (^*) were used to capture the ending variance in root words.

Subject headings for concept 1: intellectual disability

Intellectual Disability+

Developmental Disabilities

Mentally disabled persons

Text words: ("mental^* deficien^*" or "mental^* handicap^*" or "mental^* subnormal^*" or "developmental^* disab^*" or "learning disab^*" or "mental^* retard^*" or "intellectual^* impair^*" or "intellectual^* disab^*" or ID or MR or "learning disorder^*" or "intellectual developmental disorder^*" or LD)

Subject headings for concept 2: lgbtqia identity

Homosexuality

Gay Persons+

Gay Men

Men Who Have Sex With Men

Lesbians

Bisexuality

Transgender Persons+

GLBT Persons+

Transsexualism

Transsexuals

Text words: ("gay^*" or "lesbian^*" or "homosex^*" or "homoerotic^*" or "homophi^*" or "queer^*" or "same-sex attraction^*" or "sexual inversion^*" or "bisexual^*" or "bi-sexual" or "transgender^*" or "genderqueer^*" or "same gender lov^*" or "same gender attract^*" or LGBT or LGBTQIA or LGB or GLBT or GBL or "transsexual^*" or "transexual^*" or "gender dysphoria^*" or "nongender^*" or "genderless^*" or "asexual^*" or "questioning" or "gender variant^*" or "transman" or "transwoman" or "men who have sex with men" or MSM or "male-male sex" or "women who have sex with women" or WSW or "bi-gender^*" or "bigender^*" or "gender fluid^*" or "third gender^*" or "third sex^*" or "dual gender^*" or "intergender^*" or "intersex")

Search history

Systematic search of the literature was undertaken on Saturday, April 1, 2017 at 11.20 pm. The "Flinders Database Syntax guide for Systematic Reviewers" was used to implement database specific search strategies. TI stands for Tittle, AB for Abstract and MM for Major Concept. BOOLEAN operators were used as conjunctions to include, exclude or combine search terms. This is to ensure a balance between search specificity and sensitivity was established.

References