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探讨颅面短小畸形患者的言语表型:系统评价方案

Characterising the speech phenotype in individuals with craniofacial microsomia: a scoping review protocol.

机构信息

Craniofacial Center, Seattle Children's Hospital, Seattle, Washington, USA

Pediatrics, Division of Craniofacial Medicine, University of Washington, Seattle, Washington, USA.

出版信息

BMJ Open. 2023 Feb 28;13(2):e069233. doi: 10.1136/bmjopen-2022-069233.

Abstract

INTRODUCTION

Asymmetric mandibular hypoplasia, microtia, tongue and laryngeal anomalies, and soft palate and facial nerve dysfunction are clinical features observed in children with craniofacial microsomia (CFM). Despite involvement of all these structures in hearing and speech, there is limited evidence reporting speech outcomes in this population. Systematic reviews of clinical and surgical interventions related to CFM have been published, but no methodological review of speech outcomes exists. This scoping review will summarise what is known about speech production in individuals with CFM as well as illustrate gaps in the existing body of literature that will guide future research.

METHODS/ANALYSIS: This review will follow the methodological framework for scoping reviews first reported by Arksey & O'Malley and revised by Levac and others. Databases searched will include Ovid MEDLINE, EMBASE, CINAHL, PsycINFO and grey literature. Articles reporting any parameter of speech production in individuals with CFM will be considered for inclusion. Articles published in a language other than English will be excluded. Articles will be screened in three stages: (1) title review, (2) abstract review and (3) full text review. Ten per cent of articles will be rescreened by a second reviewer. Reference lists will be hand reviewed to identify additional relevant articles. Data charting will capture article metadata, study population and design, CFM diagnostic criteria, speech outcome measurement and key findings. The Preferred Reporting Systems for Systematic Reviews and Meta-Analyses Protocols-Extension for Scoping Reviews checklist will guide reporting of results. Descriptive analysis and data visualisation strategies will be used.

ETHICS AND DISSEMINATION

Institutional review board approval is not required for a scoping review, as it does not directly involve human subjects. Results will be disseminated through peer-reviewed publication as well as conference presentation.

摘要

简介

不对称性下颌发育不全、小耳畸形、舌和喉畸形以及软腭和面神经功能障碍是颅面畸形(CFM)患儿的临床特征。尽管所有这些结构都参与了听力和言语,但关于该人群言语结局的证据有限。已经发表了与 CFM 相关的临床和手术干预的系统评价,但不存在关于言语结局的方法学评价。本范围综述将总结已知的 CFM 患者的言语产生情况,并说明现有文献中的空白,这将指导未来的研究。

方法/分析:本综述将遵循 Arksey 和 O'Malley 首次报道的范围综述方法框架,并由 Levac 等人进行修订。搜索的数据库将包括 Ovid MEDLINE、EMBASE、CINAHL、PsycINFO 和灰色文献。将考虑纳入报告 CFM 患者任何言语产生参数的文章。将排除以英语以外语言发表的文章。文章将分三个阶段进行筛选:(1)标题审查,(2)摘要审查,(3)全文审查。将由第二名评审员重新筛选 10%的文章。将手动审查参考文献列表以确定其他相关文章。数据图表将捕获文章元数据、研究人群和设计、CFM 诊断标准、言语结局测量和主要发现。系统评价和荟萃分析报告的扩展范围综述的首选报告系统清单将指导结果报告。将使用描述性分析和数据可视化策略。

伦理与传播

范围综述不直接涉及人类受试者,因此不需要机构审查委员会批准。结果将通过同行评议的出版物以及会议报告进行传播。

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