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      Stuttering in individuals with Down syndrome: a systematic review of earlier research

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          Abstract

          The main objective of this systematic review was to synthesize the evidence on the occurrence and characteristics of stuttering in individuals with Down syndrome and thus contribute knowledge about stuttering in this population. Group studies reporting outcome measures of stuttering were included. Studies with participants who were preselected based on their fluency status were excluded. We searched the Eric, PsychInfo, Medline, Scopus, and Web of Science Core Collection databases on 3rd January 2022 and conducted supplementary searches of the reference lists of previous reviews and the studies included in the current review, as well as relevant speech and language journals. The included studies were coded in terms of information concerning sample characteristics, measurement approaches, and stuttering-related outcomes. The appraisal tool for cross-sectional studies (AXIS) was used to evaluate study quality. We identified 14 eligible studies, with a total of 1,833 participants (mean = 131.29, standard deviation = 227.85, median = 45.5) between 3 and 58 years of age. The estimated occurrence of stuttering ranged from 2.38 to 56%, which is substantially higher than the estimated prevalence (1%) of stuttering in the general population. The results also showed that stuttering severity most often was judged to be mild-to-moderate and that individuals with Down syndrome displayed secondary behaviors when these were measured. However, little attention has been paid to investigating the potential adverse effects of stuttering for individuals with Down syndrome. We judged the quality of the evidence to be moderate-to-low. The negative evaluation was mostly due to sampling limitations that decreased the representability and generalizability of the results. Based on the high occurrence of stuttering and the potential negative effects of this condition, individuals with Down syndrome who show signs of stuttering should be referred to a speech and language pathologist for an evaluation of their need for stuttering treatment.

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          The PRISMA 2020 statement: an updated guideline for reporting systematic reviews

          The Preferred Reporting Items for Systematic reviews and Meta-Analyses (PRISMA) statement, published in 2009, was designed to help systematic reviewers transparently report why the review was done, what the authors did, and what they found. Over the past decade, advances in systematic review methodology and terminology have necessitated an update to the guideline. The PRISMA 2020 statement replaces the 2009 statement and includes new reporting guidance that reflects advances in methods to identify, select, appraise, and synthesise studies. The structure and presentation of the items have been modified to facilitate implementation. In this article, we present the PRISMA 2020 27-item checklist, an expanded checklist that details reporting recommendations for each item, the PRISMA 2020 abstract checklist, and the revised flow diagrams for original and updated reviews.
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            Development of a critical appraisal tool to assess the quality of cross-sectional studies (AXIS)

            Objectives The aim of this study was to develop a critical appraisal (CA) tool that addressed study design and reporting quality as well as the risk of bias in cross-sectional studies (CSSs). In addition, the aim was to produce a help document to guide the non-expert user through the tool. Design An initial scoping review of the published literature and key epidemiological texts was undertaken prior to the formation of a Delphi panel to establish key components for a CA tool for CSSs. A consensus of 80% was required from the Delphi panel for any component to be included in the final tool. Results An initial list of 39 components was identified through examination of existing resources. An international Delphi panel of 18 medical and veterinary experts was established. After 3 rounds of the Delphi process, the Appraisal tool for Cross-Sectional Studies (AXIS tool) was developed by consensus and consisted of 20 components. A detailed explanatory document was also developed with the tool, giving expanded explanation of each question and providing simple interpretations and examples of the epidemiological concepts being examined in each question to aid non-expert users. Conclusions CA of the literature is a vital step in evidence synthesis and therefore evidence-based decision-making in a number of different disciplines. The AXIS tool is therefore unique and was developed in a way that it can be used across disciplines to aid the inclusion of CSSs in systematic reviews, guidelines and clinical decision-making.
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              The development of a critical appraisal tool for use in systematic reviews addressing questions of prevalence.

              Recently there has been a significant increase in the number of systematic reviews addressing questions of prevalence. Key features of a systematic review include the creation of an a priori protocol, clear inclusion criteria, a structured and systematic search process, critical appraisal of studies, and a formal process of data extraction followed by methods to synthesize, or combine, this data. Currently there exists no standard method for conducting critical appraisal of studies in systematic reviews of prevalence data.
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                Author and article information

                Contributors
                Journal
                Front Psychol
                Front Psychol
                Front. Psychol.
                Frontiers in Psychology
                Frontiers Media S.A.
                1664-1078
                29 November 2023
                2023
                : 14
                : 1176743
                Affiliations
                [1] 1Department of Education, Lillehammer, Inland Norway University of Applied Sciences , Lillehammer, Norway
                [2] 2Department of Special Needs Education, University of Oslo , Oslo, Norway
                Author notes

                Edited by: Susan Loveall, University of Nebraska-Lincoln, United States

                Reviewed by: Ellen Rombouts, KU Leuven, Belgium; Harrison Jones, Duke University, United States

                *Correspondence: Silje Hokstad, silje.hokstad@ 123456inn.no
                Article
                10.3389/fpsyg.2023.1176743
                10716236
                38094702
                42e70db3-6d99-4181-bcb4-888508080b13
                Copyright © 2023 Hokstad and Næss.

                This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.

                History
                : 28 February 2023
                : 09 October 2023
                Page count
                Figures: 1, Tables: 10, Equations: 0, References: 86, Pages: 16, Words: 12302
                Categories
                Psychology
                Review
                Custom metadata
                Developmental Psychology

                Clinical Psychology & Psychiatry
                down syndrome,stuttering,speech,disfluency,stuttering assessment,systematic review

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