Study Protocol Systematic Review Medicine ®

OPEN

Effects of social stories intervention for children

and adolescents with autism spectrum disorders
A protocol for a systematic review and meta-analysis of
randomized controlled trials
∗
Tingting Chen, MDa,b, Wenxu Yang, MDc, Qiu Wang, MDd, Ying Zhang, MDe, Zhigui Ma, MD, PHDa,

Abstract
Background: Autism spectrum disorder (ASD) is a common neurodevelopmental disorder, which lacks specific medical
treatment. Intervention is the key point of rehabilitation training for ASD. Social stories (SS) are a commonly used intervention practice
in individuals with ASD. However, there is mixed evidence on the effectiveness of SS. Thus, the objective of this systematic review and
meta-analysis is to assess studies of the effects of SS for children and adolescents with ASD.
Methods: To identify relevant studies, we will search PubMed, EMBASE, Cochrane Library, Web of Science, Google Scholar and
Downloaded from http://journals.lww.com/md-journal by BhDMf5ePHKbH4TTImqenVBZZxeh5YHRLJRAZs7Ufzg7jQvcoZgGxPfqm9D9pChkVZKveTMVNphE= on 09/16/2020

trials registers (the World Health Organization International Clinical Trial Registration Platform, ClinicalTrials.gov, and Chinese Clinical
Trial Register) from inception to May 2020. In addition, we will also perform handsearching of grey literature, such as conference
proceedings and academic degree dissertations. Only the randomized control trials will be accepted, no matter what the languages
they were reported. We will first focus on the effectiveness of the intervention on the behavior of the targets. Then we will do further
analysis of the study design, including the length and intensity of intervention, the characteristics of participants and interveners, the
methods of assessment, the place, the medium, and the economic feasibility. Two independent reviewers will carry out literature
identification, data collection, and study quality assessment. Discrepancies will be resolved by a third reviewer. The Cochrane Risk of
Bias Tool will be used to evaluate the risk of bias of the randomized controlled trials. Data analysis will be calculated using the STATA
13.0 software.
Result: This study will offer new evidence whether the SS is an appropriate intervention of benefiting the children and adolescents
with ASD, and to determine which factors affect the effectiveness of SS.
Conclusion: The conclusion drawn from this systematic review will benefit the children and adolescents with ASD.
Abbreviations: ASD = autism spectrum disorder, RCT = randomized controlled trial, SS = social stories.
Keywords: adolescent, autism spectrum disorders, children, meta-analysis, social stories, systematic review

1. Introduction nication skills, motor abnormalities, intellectual impairments,

Autism spectrum disorder (ASD) refers to a heterogeneous and various comorbidities.[2–4] Social impairments are common
neurodevelopmental condition characterized by impairment in symptoms in ASD, which encompass diverse impaired social
reciprocal social interaction and communication, together with cognitive processes, such as dullness of orienting reflex,
repetitive and restrictive behaviors.[1] There are wide clinical incomprehension of others’ cognitive states or actions and
characteristics with ASD, including variable degrees of commu- inappropriate self-referential thought.[5] As the prevalence of

This study is funded by the Municipal Health Department Program of Chengdu (no. 2018097).
This manuscript has been read and approved by all authors.
The authors have no conflicts of interest to disclose.
The datasets generated during and/or analyzed during the current study are publicly available.
a
Key Laboratory of Birth Defects and Related Diseases of Women and Children (Sichuan University), Ministry of Education, West China Second University Hospital,
Sichuan University, Chengdu, Sichuan, b Department of Pediatric Cardiology, c Department of Child Health Care, Chengdu Women’s and Children’s Central Hospital,
School of Medicine, University of Electronic Science and Technology of China, d Department of Pediatric Rehabilitation, West China Second University Hospital, Sichuan
University, e Department of Pediatric Respirology, Chengdu Women’s and Children’s Central Hospital, School of Medicine, University of Electronic Science and
Technology of China, Chengdu, China.
∗
Correspondence: Zhigui Ma, West China Second University Hospital, Sichuan University, No.20 section3 South Renmin Road, Chengdu, Sichuan Province 610041,
China (e-mail: ma_zg@yahoo.com).
Copyright © 2020 the Author(s). Published by Wolters Kluwer Health, Inc.
This is an open access article distributed under the Creative Commons Attribution License 4.0 (CCBY), which permits unrestricted use, distribution, and reproduction in
any medium, provided the original work is properly cited.
How to cite this article: Chen T, Yang W, Wang Q, Zhang Y, Ma Z. Effects of social stories intervention for children and adolescents with autism spectrum disorders: A
protocol for a systematic review and meta-analysis of randomized controlled trials. Medicine 2020;99:37(e22018).
Received: 24 July 2020 / Accepted: 31 July 2020
http://dx.doi.org/10.1097/MD.0000000000022018

1
Chen et al. Medicine (2020) 99:37 Medicine

ASD continues to rise, [4,6] it reaches 1:54 children in the United Table 1
States. [7] The increased risk for somatic and psychiatric illness, Search strategy for PubMed.
reduced quality of life and premature mortality suggest that ASD
Number Search terms
affects many families and represents a serious public health
problem. 1 Thematic Apperception Test [MeSH]
The appropriate intensive behavioral therapies are effective in 2 (social stories OR social story): [tiab]
reducing disability in many children with ASD. Developed in 3 1 OR 2
4 Autism Spectrum Disorder [MeSH]
1993,[8] Social stories (SS) describes various social situation
5 (autism OR autism spectrum disorder OR autistic OR
consisting of individualized phrases or stories that specify how a autistic spectrum disorder OR ASD):[tiab]
person should act in certain contexts or situations, which is 6 4 OR 5
accurate, convincing and easy to understand by children with 7 Child [MeSH]
ASD. The objective is to assist children with ASD in teaching 8 (children OR child):[tiab]
socially appropriate behaviors, and reduce disruptive behav- 9 Adolescent [MeSH]
iors.[9,10] Due to flexibility and capacity for individualization, 10 (adolescent OR adolescence OR teen OR teenager OR youth): [tiab]
they tend to have high acceptance and applicability.[10] 11 7 OR 8 OR 9 OR 10
There is mixed evidence on the effectiveness of SS. Previous 12 (randomized controlled trial): [pt]
literatures suggested that SS can improve understanding and 13 (controlled clinical trial): [tiab]
14 (randomized OR randomly OR trial OR groups): [tiab]
performance in social situations.[11,12] Other researchers had
15 12 OR 13 OR 14
come to inconsistent conclusions, noting uncertainty about the 16 3 AND 6 AND 11 AND 15
efficacy of SS for children with ASD.[13,14] A number of
systematic reviews have been conducted on the effect of SS
intervention for children with ASD.[13–15] However, there is
currently no systematic review and meta-analysis of randomized addition to active treatment compared with the same active
controlled trials (RCTs). The primary aim of this study is to treatment.
undertake a comprehensive systematic review and meta-analysis
to evaluate SS in children with strong experimental designs, and 2.2.4. Types of outcome measures. The primary outcome will
tease out what factors influence their effectiveness. be the effect of improvements in target behaviors, which include 3
broad categories: restricted and repetitive behaviors, impairment
in social communication, and appropriate life skills.
2. Methods The secondary outcomes will consider study design, the length
and intensity of intervention, the characteristics of participants
2.1. Design and registration
and interveners, the use of assessment, place, medium and
This systematic review is registered on the international economic feasibility.
prospective register of systematic reviews (PROSPERO). Regis-
tration number is CRD42020189708. This protocol is conducted
2.3. Data sources and search strategy
according to the Preferred Reporting Items for Systematic
Reviews and Meta-Analysis Protocol (PRISMA-P) statement We will search electronic databases including PubMed,
guidelines[16] and the Cochrane Handbook for Systematic EMBASE, Cochrane Library, Web of Science, Google Scholar
Reviews of Interventions[17]. and trials registers (the World Health Organization International
Clinical Trial Registration Platform, ClinicalTrials.gov and
Chinese Clinical Trial Register) from inception to May 2020.
2.2. Criteria for including studies in this review
2.2.1. Types of studies. To evaluate the efficacy of SS in the Grey literature, such as Conference proceedings and academic
intervention of ASD, this paper only reviewed the RCT between degree dissertations will be manually searched. For a compre-
SS and the control group, without any restrictions on blinding, hensive search, a search strategy that combines MeSH terms and
language, date of transmission or type of publication. Non- free words will be adopted. Search strategy in PubMed is shown
RCTs, quasi-RCTs, retrospective studies, case reports, non- in Table 1.
controlled trials, and animal mechanism studies will be excluded.
2.4. Data collection and analysis
2.2.2. Types of participants. Diagnosis of ASD patients (<18 2.4.1. study selection. In the literature screening process, we
years of age) will be included in the analysis, regardless of their will use EndNote X7 software. Two reviewers will independently
gender, ethnicity, and background, according to the International assess all relevant studies and select eligible articles that meet
Classification of Diseases (ICD) 11,[18] or the Diagnostic and inclusion criteria based on the title and abstract. The full texts of
Statistical Manual of Mental Disorders (DSM)-V,[19] research articles which are potentially eligible will be examined for further
diagnostic criteria. evaluation. In case of a discrepancy between the 2 review authors,
it will be discussed and made an agreement with the third author.
2.2.3. Types of interventions. We define SS as the experimental A flow diagram for the selection process will be developed using
intervention. SS included in this study will follow the Gray’s the Preferred Reporting Items for Systematic Reviews and Meta-
guidelines[8,20] as the primary intervention. If the studies included Analysis guidelines. (Fig. 1).[21]
use more than one intervention, the data of SS should be
separated. 2.4.2. Data extraction. Two authors will extract the data
There will be no restrictions with respect to the type of needed and fill out the data extraction form independently. The
comparator. The comparisons are likely to include: no interven- data form will include first author, year of publication, region,
tion, sham intervention, other active procedures and SS in sex, age, number of cases, number of controls, intervention

2
Chen et al. Medicine (2020) 99:37 www.md-journal.com

Records identified through Additional records identified

database searching through other sources

,GHQWLILFDWLRQ
(n=) (n=)

Records after duplicates removed

(n=)
6FUHHQLQJ

Records filtered by title Records exclude

and abstract (n=) (n=)
(OLJLELOLW\

Full-text articles assessed Full-text articles excluded,

for eligibility with reasons
(n=) (n=)

Studies included in
qualitative synthesis
(n=)
,QFOXGHG

Studies included in
quantitative synthesis
(meta-analysis)
(n=)

Figure 1. Flow chart of study flow from systematic search to selection process.

details, completion of intervention, missing participants, inter- missing or incomplete data, we will contact the author to obtain
vention time, control, outcome, and conflicts of interest. When the missing data. Statistical analyses will be performed using
encountering a necessary outcome that is inaccessible for direct STATA 13.0 software. The risk ratio with 95% confidence
data extraction, we will attempt to obtain information from the interval will be used to assess dichotomous data. Continuous
corresponding authors by e-mail. Any disagreements will be outcomes will be expressed as standardized mean difference
discussed and resolved in discussion with a third author. along with its 95% confidence interval. We will synthetize
primary studies to explore heterogeneity descriptively rather than
2.4.3. Assessment of risk of bias. Following the guidance in the
statistically. The statistical heterogeneity among studies was
latest version of Cochrane Handbook for systematic reviews of
assessed with the Q-test and I2 statistics. If significant
interventions,[22] two reviewers will independently assess the risk
heterogeneity is found, the random-effects model will be used
of bias for each included study with version 2 of the Cochrane
to estimate the data, otherwise, the fixed-effects model will be
risk-of bias tool for randomized trials, ROB 2.[23] We will
used. Funnel plot and Egger test will be used to detect the
examine five domains, including bias from the randomization
potential reporting biases if at least 10 studies are included.
process, bias due to deviations from intended interventions, bias
due to missing outcome data, bias in measurement of the
2.4.5. Sensitivity analysis and subgroup analysis. To check
outcome, and bias in selection of the reported result. If there is
the robustness of pooled outcome results, sensitivity analyses will
insufficient detail to assess the risk of bias, we will contact study
be conducted to assess how including and excluding studies
authors by e-mail. The third reviewer will arbitrate in the case of
influences the results, when studies are adequate. We will repeat
any disagreement.
the analysis after excluding cross-over trials and trials with a high
2.4.4. Data synthesis and statistical analysis. In accordance risk of bias. If appropriate data are available, subgroup analyses
with Higgins et al,[24] only the first phase of the data will be will be exploratory based on geographical location, age, control
included in the random crossover trial. If the primary result has interventions and different outcomes.

3
Chen et al. Medicine (2020) 99:37 Medicine

3. Discussion disabilities monitoring network, 11 sites, United States. MMWR Surveill

Summ 2020;69:1–2.
ASD symptoms often accompany the whole life of individuals. [8] Gray C, Garand J. Social stories: improving responses of students with
Without special intervention and rehabilitation, most of them autism with accurate social information. Focus Autistic Behav 1993;
will have lifelong intellectual and mental disability, which will 8:3–10.
[9] Balakrishnan S, Alias A. Usage of social stories in encouraging social
have a great impact on their social function.[25,26] SS could be a interaction of children with Autism Spectrum Disorder. J ICSAR
promising intervention to improve children’s appropriate 2017;1:91–7.
behavior and social skills and reduce destructive behavior. [10] Wright LA, McCathren RB. Utilizing social stories to increase prosocial
However, there is an ongoing debate regarding the effectiveness behavior and reduce problem behavior in young children with autism.
Child Dev Res 2012;4:1–3.
of SS in children with ASD due to uncertainty of factors affecting
[11] Balakrishnan S, Alias A. Usage of social stories in encouraging social
benefits. As we know, the current study will be the first meta- interaction of children with autism spectrum disorder. J Int Conf Spec
analysis of RCT for efficacy of SS in children with ASD. We will Edu Southeast Asia Reg 2017;1:91–7.
comprehensively evaluate randomized data characteristics, [12] Marshall D, Wright B, Allgar V, et al. Social stories in mainstream
including detailed information of interventions and primary schools for children with autism spectrum disorder: a feasibility
randomized controlled trial. BMJ Open 2016;6:1–10.
and secondary outcomes. According to the Cochrane method, [13] Test DW, Richter S, Knight V, et al. A comprehensive review and meta-
this study is based on the analysis of RCT evidence, searching and analysis of the Social StoriesTM literature. Focus Autism Other Dev
screening the main electronic literature database, providing Disabil 2011;26:49–62.
therapists with more convincing evidence in decision-making, to [14] Kokina A, Kern L. Social StoryTM intervention for students with autism
spectrum disorders: a meta-analysis. J Autism Dev Disord 2010;40:
better guide intervention.
812–26.
[15] Garwood JD, Van Loan C. Using social stories with students with social,
emotional, and behavioral disabilities: the promise and the perils.
Author contributions
Exceptionality 2019;27:133–48.
Conceptualization: Tingting Chen, Zhigui Ma [16] Shamseer L, Moher D, Clarke M, et al. Preferred reporting items for
systematic review and meta-analysis protocols (PRISMA-P) 2015:
Data curation: Tingting Chen, Wenxu Yang, Zhigui Ma
elaboration and explanation. BMJ 2015;350:g7647.
Formal analysis: Qiu Wang, Ying Zhang [17] Deeks JJ, Higgins JPT, Altman DG, et al. Cochrane Handbook for
Funding acquisition: Tingting Chen Systematic Reviews of Interventions Version 5.1. 0 (Updated March
Methodology: Tingting Chen, Wenxu Yang,Qiu Wang 2011). The Cochrane Collaboration; 2011.
Software: Tingting Chen, Ying Zhang [18] World Health Organization. International Classification of Diseases for
Mortality and Morbidity Statistics, 11th edition (ICD-11). 2018. Retrieved
Supervision: Zhigui Ma from https://www.who.int/classifcations/icd/en/.222. Accessed 20 May 2020.
Writing – original draft: Tingting Chen, Wenxu Yang [19] American Psychiatric Association. Diagnostic and Statistical Manual of
Writing – review & editing: Tingting Chen, Zhigui Ma Mental Disorders, 5th ed. American Psychiatric Association: Wash-
ington DC; 2013.
[20] Gray C. Writing Social Stories With Carol Gray. Arlington TX: Future
Horizons; 2000.
References
[21] Moher D, Shamseer L, Clarke M, et al. Preferred reporting items for
[1] American Psychiatric Association [APA]Diagnostic and Statistical systematic review and meta-analysis protocols (PRISMA-P) 2015
Manual of Mental Disorders (DSM-5). 5th ednArlington, TX: American statement. Syst Rev 2016;20:148–60.
Psychiatric Association; 2013. [22] Higgins JPT, Savovic J, Page MJ, et al. JAC S: chapter 8: assessing risk of bias in
[2] Kerns CM, Kendall PC, Berry L, et al. Traditional and atypical a randomized trial. Cochrane Handbook for Systematic Reviews of
presentations of anxiety in youth with autism spectrum disorder. J Interventions version 6 0 (updated 2019). The Cochrane Collaboration; 2019.
Autism Dev Disord 2014;44:2851–61. [23] Sterne JAC, Savovic J, Page MJ, et al. RoB 2: a revised tool for
[3] El Achkar CM, Spence SJ. Clinical characteristics of children and young assessing risk of bias in randomised trials. BMJ (Clin Res Ed)
adults with co-occurring autism spectrum disorder and epilepsy. Epilepsy 2019;366:1–7.
Behav 2015;47:183–90. [24] Higgins JPT, Eldridge S, Li T. Higgins JPT, Thomas J, Chandler J, Cumpston
[4] Baio J, Wiggins L, Christensen DL, et al. Prevalence of autism spectrum M, Li T, Page MJ, Welch VA. Chapter 23: including variants on randomized
disorder among children aged 8 years – autism and developmental trials. Cochrane Handbook for Systematic Reviews of Interventions version
disabilities monitoring network, 11 sites, United States. MMWR Surveill 6. 0 (updated July 2019). The Cochrane Collaboration; 2019.
Summ 2018;67:1–23. [25] Barendse EM, Hendriks M, Thoonen G, et al. Social behaviour and social
[5] Chevallier C, Kohls G, Troiani V, et al. The social motivation theory of cognition in high- functioning adolescent with autism spectrum disorder
autism. Trends Cogn Sci 2012;16:231–9. (ASD): two sides of the same coin? Cogn Process 2018;19:545–55.
[6] Lyall K, Croen L, Daniels J, et al. The changing epidemiology of autism [26] Wolstencroft J, Ribinson L, Srinivasan R, et al. A systematic review of
spectrum disorders. Annu Rev Public Health 2017;38:81–102. group social skill interventions, and meta-analysis of outcomes, for
[7] Maenner MJ, Shaw KA, Bak J, et al. Prevalence of autism spectrum children with high functioning ASD. J Autism Dev Disord 2018;
disorder among children aged 8 years - autism and developmental 48:2293–307.