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Meta-Analysis
. 2021 Jul 23;13(1):28.
doi: 10.1186/s11689-021-09362-5.

Autism spectrum disorder in females with fragile X syndrome: a systematic review and meta-analysis of prevalence

M Marlborough  1 , A Welham  2 , C Jones  1 , S Reckless  1 , J Moss  1
Affiliations
Meta-Analysis

Autism spectrum disorder in females with fragile X syndrome: a systematic review and meta-analysis of prevalence

M Marlborough et al. J Neurodev Disord. .

Abstract

Background: Whilst up to 60% of males with fragile X syndrome (FXS) meet criteria for autism spectrum disorder (ASD), the prevalence and nature of ASD in females with FXS remains unclear.

Method: A systematic literature search identified papers reporting ASD prevalence and/or symptomatology in females with FXS.

Results and conclusion: Meta-analysis suggested that rates of ASD for females with FXS are reliably higher than for females in the general population (a random effects model estimated weighted average prevalence at 14%, 95% CI 13-18%). Whilst papers highlighted a number of social and repetitive difficulties for females with FXS, characteristic profiles of impairment are not clear. Possible associations between ASD traits and IQ, and between ASD and levels of fragile X mental retardation protein, are suggested, but data are equivocal.

Keywords: Autistic spectrum disorder; Female; Fragile X syndrome; Meta-analysis; Prevalence.

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Conflict of interest statement

The authors declare that there are no competing interests.

Figures

Fig. 1
Fig. 1
Flow diagrams of review process based on PRISMA group flow chart [22]. a Search in July 2018. b Search in Feb 2020
Fig. 2
Fig. 2
Forest plots of random effects models and Baujat plot of ASD in fragile X females. a Forest plot for random effects model for FXS following omission of Symons et al. [62]. b Forest plot for random effects model for FXS all papers. c Baujatplot for FXS
Fig. 3
Fig. 3
Forest plots of quality effects model of ASD in fragile X females. a Forest plot for quality effects model for FXS omitting Symons et al. [62]. b Forest plot for quality effects model FXS all papers
Fig. 4
Fig. 4
Funnel plot, in which studies' reported proportion of participants meeting criteria for ASD is plotted against the square root of the studies' sampling variance
See this image and copyright information in PMC

References

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