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Psychosocial support interventions for children and adolescents experiencing gender dysphoria or incongruence: a systematic review
  1. Claire Heathcote,
  2. Jo Taylor,
  3. Ruth Hall,
  4. Stuart William Jarvis,
  5. Trilby Langton,
  6. Catherine Elizabeth Hewitt,
  7. Lorna Fraser
  1. Department of Health Sciences, University of York, York, UK
  1. Correspondence to Dr Jo Taylor, Department of Health Sciences, University of York, York, UK; dohs-gender-research{at}york.ac.uk

Abstract

Background National and international guidelines recommend that psychosocial support should be a key component of the care offered to children and adolescents experiencing gender dysphoria/incongruence. However, specific approaches or interventions are not recommended.

Aim To identify and summarise evidence on the outcomes of psychosocial support interventions for children and adolescents (age 0-18) experiencing gender dysphoria/incongruence.

Methods Systematic review and narrative synthesis. Database searches (MEDLINE; EMBASE; CINAHL; PsycINFO; Web of Science) were performed in April 2022, with results assessed independently by two reviewers. Peer-reviewed articles reporting the results of studies measuring outcomes of psychosocial support interventions were included. Quality was assessed using the Mixed Methods Appraisal Tool.

Results Ten studies were included. Half were conducted in the US, with others from Australia, Canada, New Zealand and the UK. Six were pre–post analyses or cohort studies, three were mixed methods, and one was a secondary analysis of intervention data from four trials. Most studies were of low quality. Most analyses of mental health and psychosocial outcomes showed either benefit or no change, with none indicating negative or adverse effects.

Conclusions The small number of low-quality studies limits conclusions about the effectiveness of psychosocial interventions for children/adolescents experiencing gender dysphoria/incongruence. Clarity on the intervention approach as well as the core outcomes would support the future aggregation of evidence. More robust methodology and reporting is required.

PROSPERO registration number CRD42021289659.

Data availability statement

Data sharing is not applicable as no datasets were generated and/or analysed for this study.

http://creativecommons.org/licenses/by-nc/4.0/

This is an open access article distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited, appropriate credit is given, any changes made indicated, and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/.

https://doi.org/10.1136/archdischild-2023-326347

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    WHAT IS ALREADY KNOWN ON THIS TOPIC

    • The number of children and adolescents identifying as a gender different from the sex they were registered at birth has increased markedly over the last 10-15 years.

    • Children and adolescents experiencing gender dypshoria/incongruence have higher rates of mental health needs compared with their peers.

    • National and international guidelines recommend that psychosocial interventions should be a key component of the care offered to children and adolescents experiencing gender dysphoria/incongruence.

    WHAT THIS STUDY ADDS

    • There is limited evidence on the outcomes of psychosocial interventions for children and adolescents experiencing gender dysphoria/incongruence.

    • The evidence base for outcomes of psychosocial interventions for children and adolescents experiencing gender dysphoria/incongruence is of low quality.

    • Most analyses of mental health, psychological and/or psychosocial outcomes showed either benefit or no change, with none indicating negative or adverse effects.

    HOW THIS STUDY MIGHT AFFECT RESEARCH, PRACTICE OR POLICY

    • Identification of the core approach and outcomes for psychosocial interventions would ensure they are addressing key clinical goals, attending to the needs of children/adolescents and families as well as supporting future aggregation of evidence. More robust methodology and reporting is required.

    Introduction

    The number of children and adolescents who are experiencing gender dysphoria/incongruence has risen markedly over the last 10-15 years.1 A considerably higher proportion of children and adolescents who present to specialised gender services experience mental health difficulties compared with their peers in the general population, with evidence showing higher recorded rates of psychiatric diagnoses,2–4 including depression,3–6 anxiety3 4 7 and eating disorders,4 ,8 and higher recorded rates of suicidality9 10 and self-harm.10 11

    The causes of mental health concerns and the ways in which well-being issues might contribute to the emergence of distress in this population are not fully understood. Some authors argue that children and adolescents experiencing gender dysphoria/incongruence present with complex and diverse needs so a developmental approach is necessary to understand the multiple factors that might be contributing to social and emotional distress.12–14 There is also evidence that the experience of being in a minority group can have a negative impact on development and well-being.15 16 Meyer’s minority stress model proposes that protective or coping factors can help to interrupt or moderate the impact of minority stress.17 18 Identifying, strengthening or developing coping and psychosocial support factors is reported to help build well-being and improve mental health.19 20

    National and international guidelines recommend that psychosocial interventions should be a key component of the care offered to children and adolescents experiencing gender dysphoria/incongruence. It is recommended that support should be offered to enhance psychological functioning, improve mood and well-being, and support coping factors, in addition to managing any co-occurring or contributory mental health difficulties.21–24

    This systematic review aims to identify and summarise the current evidence on the effectiveness of psychosocial support interventions for children and adolescents experiencing gender dysphoria/incongruence.

    Methods

    The review forms part of a linked series of systematic reviews examining the epidemiology, care pathways, outcomes and experiences of children and adolescents experiencing gender dysphoria/incongruence and is reported according to Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines.25 The systematic review protocol was registered on PROSPERO (CRD42021289659).26

    Search strategy

    A single search strategy was used to identify studies comprising two combined concepts: ‘children’, which included all terms for children and adolescents; and ‘gender dysphoria’, which included associated terms such as gender-related distress and gender incongruence, and gender identity terms including transgender, gender diverse and non-binary. MEDLINE (see online supplemental table 1), Embase and PsycINFO through OVID, CINAHL Complete through EBSCO and Web of Science (Social Science Citation Index) were searched (13–23 May 2021; updated 27 April 2022). The reference lists of eligible studies and any relevant identified systematic reviews were also checked.27 28

    Inclusion criteria

    Studies were included in relation to the following criteria:

    Population: children and adolescents up to 18 years with gender incongruence, gender dysphoria/gender-related distress or referral to a paediatric or adolescent gender service. Studies with a mixed population of adolescents/young adults (up to 25) were included, as were studies of other mixed populations if separate analyses of the groups were undertaken or if most participants (80% or more) were from the population of interest.

    Intervention: any psychological or psychosocial intervention provided to children and/or adolescents. Unstructured peer support groups and interventions designed to modify gender behaviours or identity were excluded. Interventions only provided to parents were excluded.

    Comparator: any.

    Outcomes: any child/adolescent outcome related to the intervention.

    Study design: studies published in English in a peer-reviewed journal of any design apart from case series or case reports.

    Study selection

    The results of the database and other searches were uploaded to Covidence and screened independently by two reviewers.29 Full texts for potentially relevant articles were retrieved and reviewed against the inclusion criteria by two reviewers independently. Disagreements were resolved through discussion and the inclusion of a third reviewer.

    Data extraction

    Data was extracted by one reviewer and checked by another using a piloted extraction sheet.

    Quality assessment

    Quality was assessed using the Mixed Methods Appraisal Tool (MMAT),30 a tool designed to appraise methodological quality in systematic reviews that include a combination of quantitative, qualitative and/or mixed methods research. Critical appraisal was undertaken by two reviewers independently, with consensus reached through discussion and involvement of a third reviewer where necessary. A score of 0–2 was deemed low quality, 3 medium, and 4–5 high (max score 5).

    Synthesis methods

    Due to extensive heterogeneity, a narrative approach to synthesising outcome data was adopted. This included any qualitative data pertaining to perceived or experienced effects as well as quantitatively measured outcomes. The Template for Intervention Description and Replication (TIDieR) Checklist31 was used to describe intervention reporting. For TIDieR items 1–10 (which cover intervention aims, theory, content and delivery), studies were rated as providing ‘full’, ‘partial’ or no (‘none’) information.

    Results

    Overall, searches yielded 28 147 records, of which 3181 were identified as potentially relevant for the linked series of systematic reviews (see figure 1). From these, nine studies met the inclusion criteria for this review of psychosocial intervention studies. One additional study meeting the inclusion criteria was identified from citation searches.32 Therefore, 10 studies were included in this review.32–41

    Figure 1
    Figure 1

    Study flow diagram.

    Study characteristics

    There were four cohort studies,36 39–41 two pre–post studies32 34 (one of which included analysis of open-ended survey responses in addition to outcome data34), three mixed methods,33 35 37 and one was a secondary analysis of intervention group data from four randomised controlled trials (RCTs)38 (the individual RCTs were excluded because they presented combined results for those experiencing and not experiencing gender dysphoria/incongruence). Five studies included a comparator group of children and/or adolescents not experiencing gender dysphoria/incongruence36 38–41 and one included a historical cohort of adolescents with gender dysphoria/incongruence.33 Half of the studies were conducted in the US (n=5),32 35 38 40 41 two in the UK36 37 and single studies from Australia,33 Canada34 and New Zealand.39 Table 1 provides a summary of study characteristics.

    View this table:
    Table 1

    Study characteristics

    Studies were published between 2015 and 2021 with data from 2012 to 2021. In total, studies included analyses of 10 583 individuals (854 who were transgender and gender diverse or experiencing gender dysphoria/incongruence, 2 sexual minority youth from a mixed population study32 and 9727 who did not identify as transgender or experience gender dysphoria/incongruence). Study sample sizes ranged from 8 to 9079. The age of participants ranged from 7 to 25 years with average age ranging from 10 to 19 years. Three studies included a mixed sample of adolescents and young adults32 39 41 and only two studies included children under the age of 12 years.33 38

    The gender identity of participants experiencing gender dysphoria/incongruence and how this was reported varied across the studies. Birth-registered sex was reported in four studies,33 36–38 with more birth-registered females in three of the four studies.33 36 37 Four studies reliably reported the ethnicity of participants experiencing gender dysphoria/incongruence, with the percentage from a minority ethnic group ranging from 19.5% to 50%.32 35 38 39

    Outcomes

    Studies employed a range of measures across different mental health and well-being outcomes with little congruence between studies (see table 1). All studies used validated instruments to measure outcomes. One study also devised a measure to determine the level of social support, degree of social transition, medication use and healthcare usage.33 Most relied on self-report measures and the timing of outcomes varied, with four studies measuring outcomes post-intervention only32 37 39 41 and others including a later follow-up ranging from 1 to 18 months.

    Interventions

    Various interventions were used: cognitive behavioural therapy (CBT) (n=2),34 39 mindfulness and self-compassion (n=1),35 attachment-based family therapy (n=1),32 ‘a community system of care approach’ (n=1),41 a gender service triage model including psychosocial support (n=1)33 and interventions using multiple different therapeutic approaches/techniques (n=4).36–38 40 All studies stated the goal of the intervention, and nine described the modality or theoretical approach.32 34–41 Table 2 provides a summary of intervention characteristics.

    View this table:
    Table 2

    Intervention characteristics

    Three interventions were developed for children and/or adolescents with mental health difficulties or diagnoses,38–40 one for sexual and gender minority adolescents,34 and two for sexual or gender minority adolescents and young adults with specific needs (clinical levels of suicidal ideation or moderate depression32; housing, mental health or substance misuse problems41). Three interventions were developed for children and/or adolescents referred to a specialist gender service,33 36 37 and one was a self-compassion intervention partially modified for adolescents experiencing gender dysphoria/incongruence.35

    The majority of studies gave an indication of intervention content, activities and/or processes used in the intervention,33–35 37–39 although this varied from a brief explanation33 38–40 to more detailed content.34 35 Eight studies provided information about who delivered the intervention, which included staff (professionals/roles not reported),36 psychologists37 and a specialist nurse33 working in gender services, trained facilitators/instructors34 35 or therapists,32 38 and case managers.41

    Most interventions were delivered face-to-face (n=8)32–34 36–38 40 41 with two online.35 39 Interventions were delivered individually (n=3),33 38 39 in groups (n=3),34 35 37 in families (n=1)32 or in combination (n=3).36 40 41 The setting for face-to-face interventions varied including specialist gender services (n=3),33 36 37 community settings for sexual and gender minorities (n=2),32 41 community mental health clinics (n=1),38 a weekend retreat format34 and an acute residential treatment programme (n=1)40 (the specific setting for these last two was not reported).

    Intervention duration ranged from a single session33 to having no fixed duration or number of sessions,36 38 with some participants receiving psychosocial care for around 18 months from these. The number and/or frequency of sessions varied from a single 90 minute session33 to three times weekly for 3–6 months.41 Three interventions had 8–9 sessions34 35 37 and one had 16,32 with frequency ranging from daily to weekly. Three studies reported session duration, all were 90 minutes.33 35 37 The computerised CBT programme had seven modules, each taking around 30 minutes to complete.39

    Intervention adherence was reported for the three group-based and family therapy interventions (100% completion,32 34 79% full attendance,37 73% retention35) and for the computerised CBT programme (53.6% completed module 1 but only 2.4% completed all seven).39

    When studies were assessed against the TIDieR framework, the most comprehensive information was provided on intervention name/label, rationale/aims and mode of delivery, with varying detail on intervention procedures, setting and tailoring. The least detail was provided on intervention materials, facilitator expertise and training and whether modifications were made during study delivery (see table 3 for summary of TIDieR assessment).

    View this table:
    Table 3

    TIDieR (Template for Intervention Description and Replication) assessment

    Study quality

    Nine studies were rated as low quality and one as moderate quality32 (see table 4 for critical appraisals). Limited reporting of recruitment methods meant it was difficult to determine how representative samples were of the target population. Three studies had no control or comparator group,32 35 37 and for others, the comparator was not always appropriate. Across the studies, significant confounding factors were not accounted for within the analysis. Other methodological issues included a small sample used for analysis,32 34 35 37 39–41 and reliance on self-report or a proxy indicator of gender dysphoria/incongruence or gender identity, rather than a validated and widely used assessment tool.38–41 There was also a lack of information about what happened to participants who dropped out,36 39 and several interventions were single site, limiting potential generalisability.33 34 36 37 40

    View this table:
    Table 4

    MMAT assessment

    Synthesis of results

    Eight studies reported outcomes related to mental health,32–35 38–41 five to psychological changes33–35 37 41 and five to psychosocial or healthcare changes.33 35–37 41

    Mental health outcomes

    Measures of depression were reported in six studies.32–35 39 40 All six studies focused on within-group changes either at a single33 39 or multiple32 34 35 40 post-intervention timepoints. Four studies found significant reductions in depression levels post-intervention33–35 40 and/or at a later timepoint (3 months34 35; 1 month40). Two studies found non-significant differences in depression levels during mid-treatment32 and/or post-intervention.32 39 Two studies provided comparisons with control groups (historical controls who did not receive the intervention33; adolescents not experiencing gender dysphoria/incongruence who received the same intervention40). One had a single timepoint post-intervention33 and the other had multiple post-intervention timepoints.40 One study compared the proportion of participants reaching borderline or clinical depression scores following an intervention, comparing children/adolescents experiencing gender dysphoria/incongruence with historical controls. The study reported significant reductions for those receiving the intervention in self-report but not caregiver-report measures.33 The study comparing adolescents experiencing gender dysphoria/incongruence with those not experiencing gender dysphoria/incongruence found no significant difference between groups over time in self-reported and caregiver-reported depression levels.40

    Measures of anxiety were reported in three studies.33 35 40 All three reported within-group changes either at a single33 or multiple35 40 post-intervention timepoints. All three studies found significant reductions in anxiety levels post-intervention.33 35 40 One study found significant reductions at a later timepoint (1 month40) and one did not (3 months35). The same two studies that provided comparisons with control groups for depression also did this for symptoms of anxiety (historical controls33; adolescents not experiencing gender dysphoria/incongruence40). The study including a historical control group found significantly greater reductions in self-reported but not carer-reported anxiety among those receiving the intervention.33 The final study found no significant difference between groups (those experiencing and not experiencing gender dysphoria/incongruence) over time in anxiety levels.40

    Two studies provided a combined mental health outcome.38 41 One study looked at within-group changes in mental health issues (anxiety or depression in past 30 days) and found no significant, although marginal, difference from baseline to 6 months.41 The second study measured internalising and externalising symptoms, comparing children and adolescents experiencing and not experiencing gender dysphoria/incongruence. The study found similar improvements between groups over time in self-report and caregiver report for internalising symptoms and in self-report for externalising symptoms. The caregiver report for externalising symptoms showed slower improvement among those experiencing gender dysphoria/incongruence compared with the control group. A significantly greater proportion of the participants experiencing gender dysphoria/incongruence had clinically elevated internalising problems post-intervention compared with those not experiencing gender dysphoria/incongruence.38

    One study looked at emotional dysregulation. This found significant reductions post-intervention and at a later timepoint (1 month), but no significant differences between adolescents experiencing and not experiencing gender dysphoria/incongruence over time.40

    Three studies evaluated the impact of the psychosocial intervention on suicidality and related outcomes.32 33 35 Two studies found significant decreases in suicidality scores,32 thwarted belongingness35 or perceived burdensomeness35 from baseline to mid-treatment32 and/or post-intervention32 35 and/or at a later timepoint (3 months35). One study found no difference in the proportion of patients at high suicide risk before and after the intervention (nurse triage; median time pre–post was 259 days).33

    Psychological changes

    Five studies focused on psychological changes33–35 37 41: three explored relationships quantitatively34 35 41 and four reported qualitative findings.33–35 37 For resilience,35 self-compassion35 and self-acceptance,41 there were significant increases in scores between baseline and post-intervention,35 and between baseline and 3 months for self-compassion35 and 6 months for self-acceptance,41 but the difference was no longer observed for resilience at 3-month follow-up (although it was of marginal significance).35 No significant differences were found in coping scores between baseline and post-intervention or between post-intervention and a later timepoint (3 months34).

    In the studies that reported qualitative data about perceived effects, participants reported more positive coping and problem-solving34 35 37 and increased confidence.33 37 Participants also talked about being more comfortable and accepting of their identity,33 35 37 having a better sense of self and self-worth,33 35 and reduced feelings of isolation and distrust towards others.35 37 These changes were reported to contribute to the adolescents having more agency over their lives in one study,33 increased life satisfaction in another35 and a different outlook in three studies,33 35 37 including more positivity and certainty about the future. In the group-based interventions, being able to talk openly with and learn from others about gender issues in a safe and supportive environment was reported to contribute to these positive outcomes34 35 37; whereas in the triage intervention, receiving information about what is available to them in terms of transition-related interventions was reported as a key mechanism leading to change.33

    Psychosocial and other changes

    Five studies focused on psychosocial changes33 35–37 41: all explored relationships quantitatively and three also reported qualitative findings.33 35 37

    Three studies found significant improvements in quality of life/global functioning/well-being post-intervention33 35 36 and/or at later timepoints36; however, for well-being the significant difference was no longer observed at 3 months.35 One study found no significant differences in well-being scores (physical and psychological) before and after receiving the intervention.37

    One study evaluated the impact of a single assessment and triage appointment in which information and signposting support was given to young people and families on a waiting list for a paediatric gender service. Clinical measures and qualitative interviews looked at rates of depression, anxiety, quality of life and access to health professional input and medication use, as well as knowledge relating to social transition, perceived support from social networks and family functioning.33 In the group who had received the single session intervention, there were improvements in mental health and well-being measures, family functioning was found to be significantly improved, and there were higher rates of social transition and knowledge about social transition. There was no significant change to participants’ reports of social support, no changes seen in access to broader healthcare input and overall safety had not increased. Significantly more birth-registered females had commenced medication to suppress menstruation having been signposted to information relating to this.33

    Significant improvements were also recorded, 6 months post-intake, for measures of employment and housing stability, in participants of a community system of care intervention.41

    In the studies that reported qualitative data pertaining to perceived effects, participants reported having better peer relationships and increased acceptance and/or support from peers (from meeting and forging relationships with others in the same situation).35 37 Some participants reported increased parental support or acceptance, while others did not.33

    Discussion

    There is limited research evaluating the outcomes of psychosocial interventions for children and adolescents experiencing gender dysphoria/incongruence. Low study quality and inadequate reporting in the 10 studies included limit any firm conclusions about their effects. Selection criteria for participation were not clearly defined, a range of different interventions were used, studies included heterogeneous measurements of different treatment outcomes and lacked appropriate comparators. This review highlights that to date there has been a lack of robust research and inadequate methodologies have been used to assess the effects of psychosocial interventions for children and adolescents experiencing gender dysphoria/incongruence.

    Assessed interventions included those developed specifically for children and/or adolescents experiencing gender dysphoria/incongruence,33 36 37 interventions developed or adapted for gender and sexual minority youth,32 34 41 and broader psychological interventions for adolescents with mental health difficulties either partially modified for those experiencing gender dysphoria/incongruence35 or with no tailoring.38–40 Only three were designed for children and/or adolescents referred to a specialist gender service and these varied considerably: one being a structured CBT and systemic theory-informed peer support group delivered over 8 weeks,37 one a specialist single session nurse-led triage clinic33 and one comprising the range of psychosocial support provided by a paediatric gender service.36

    Low study quality, inadequate reporting of intervention details and heterogeneity of interventions and their aims and outcome measurement prevented comparison of the different types of interventions identified or of different psychological approaches.

    Similar results were reported across studies, which showed either benefit or no change with no indication of adverse or negative effects. This result suggests that existing evidence-based interventions tailored for children and/or adolescents with gender dysphoria/incongruence as well as those developed explicitly for this population have the potential to result in positive outcomes. However, there remain unanswered questions about which specific approach works best, for whom and in what circumstances, and the acceptability and feasibility of these different approaches. There is also limited understanding of how psychosocial interventions might support a reduction of gender-related distress and the sorts of interventions that might be most suitable for prepubertal children and for adolescents with more complex needs.

    Strengths and limitations

    Strengths include a published protocol with robust search strategies and comprehensive synthesis. As searches were conducted to April 2022 this review does not include more recently published studies; as this is a rapidly evolving area this is a limitation.

    Conclusions

    There is limited research evaluating outcomes of psychosocial interventions for children and adolescents experiencing gender dysphoria/incongruence, and low quality and inadequate reporting of the studies identified. Therefore, firm conclusions about their effects cannot be made. Most analyses of mental health, psychological and/or psychosocial outcomes showed either benefit or no change, with none indicating any negative/adverse effects of the interventions offered. Identification of the core approach and outcomes for these interventions would ensure they are addressing key clinical goals, attending to the needs of children and families as well as supporting future aggregation of evidence. More robust methodology and reporting is required.

    Data availability statement

    Data sharing is not applicable as no datasets were generated and/or analysed for this study.

    Ethics statements

    Patient consent for publication

    Ethics approval

    Not applicable.

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    Footnotes

    • Contributors LF, CEH and TL contributed to the conception of this study. LF, CH, CEH, JT, and TL designed the study. Data collection was led by CH, JT and RH. Analyses was undertaken by CEH, RH, SWJ, JT and LF. CH drafted the first version of the manuscript. All authors reviewed the manuscript prior to submission. CEH accepts full responsibility for the finished work and/or the conduct of the study, had access to the data, and controlled the decision to publish.

    • Funding This work was funded by NHS England to inform the Cass Review (Independent review of gender identity services for children and young people). The funder and Cass Review team had a role in commissioning the research programme but no role in the study conduct, interpretation or conclusion.

    • Competing interests None declared.

    • Provenance and peer review Commissioned; externally peer reviewed.

    • Supplemental material This content has been supplied by the author(s). It has not been vetted by BMJ Publishing Group Limited (BMJ) and may not have been peer-reviewed. Any opinions or recommendations discussed are solely those of the author(s) and are not endorsed by BMJ. BMJ disclaims all liability and responsibility arising from any reliance placed on the content. Where the content includes any translated material, BMJ does not warrant the accuracy and reliability of the translations (including but not limited to local regulations, clinical guidelines, terminology, drug names and drug dosages), and is not responsible for any error and/or omissions arising from translation and adaptation or otherwise.

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