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Candidate diagnostic biomarkers for neurodevelopmental disorders in children and adolescents: a systematic review

Candidate diagnostic biomarkers for neurodevelopmental disorders in children and adolescents: a systematic review
Candidate diagnostic biomarkers for neurodevelopmental disorders in children and adolescents: a systematic review
Neurodevelopmental disorders – including attention-deficit/hyperactivity disorder (ADHD), autism spectrum disorder, communication disorders, intellectual disability, motor disorders, specific learning disorders, and tic disorders – manifest themselves early in development. Valid, reliable and broadly usable biomarkers supporting a timely diagnosis of these disorders would be highly relevant from a clinical and public health standpoint. We conducted the first systematic review of studies on candidate diagnostic biomarkers for these disorders in children and adolescents. We searched Medline and Embase + Embase Classic with terms relating to biomarkers until April 6, 2022, and conducted additional targeted searches for genome-wide association studies (GWAS) and neuroimaging or neurophysiological studies carried out by international consortia. We considered a candidate biomarker as promising if it was reported in at least two independent studies providing evidence of sensitivity and specificity of at least 80%. After screening 10,625 references, we retained 780 studies (374 biochemical, 203 neuroimaging, 133 neurophysiological and 65 neuropsychological studies, and five GWAS), including a total of approximately 120,000 cases and 176,000 controls. While the majority of the studies focused simply on associations, we could not find any biomarker for which there was evidence – from two or more studies from independent research groups, with results going into the same direction – of specificity and sensitivity of at least 80%. Other important metrics to assess the validity of a candidate biomarker, such as positive predictive value and negative predictive value, were infrequently reported. Limitations of the currently available studies include mostly small sample size, heterogeneous approaches and candidate biomarker targets, undue focus on single instead of joint biomarker signatures, and incomplete accounting for potential confounding factors. Future multivariable and multi-level approaches may be best suited to find valid candidate biomarkers, which will then need to be validated in external, independent samples and then, importantly, tested in terms of feasibility and cost-effectiveness, before they can be implemented in daily clinical practice
ADHD, Biological markers, autism spectrum disorder, communication disorders, genome-wide association studies, intellectual disabil­ity, motor disorders, neurodevelopmental disorders, neuroimaging, neurophysiology, specific learning disorders, tic disorders
2051-5545
129-149
Cortese, Samuele
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Solmi, Marco
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Michelini, Giorgia
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Bellato, Alessio
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Blanner, Christina
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Canozzi, Andrea
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Eudave, Luis
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Farhat, Luis C.
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Højlund, Mikkel
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Köhler-Forsberg, Ole
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Leffa, Douglas Teixeira
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Rohde, Christopher
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de Pablo, Gonzalo Salazar
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Vita, Giovanni
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Wesselhoeft, Rikke
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Martin, Joanna
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Baumeister, Sarah
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Bozhilova, Natali S.
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Carlisi, Christina O.
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Leno, Virginia Carter
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Floris, Dorothea L.
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Holz, Nathalie E.
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Kraaijenvanger, Eline J.
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Sacu, Seda
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Vainieri, Isabella
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Ostuzzi, Giovanni
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Barbui, Corrado
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Correll, Christoph U.
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Cortese, Samuele
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Solmi, Marco
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Michelini, Giorgia
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Bellato, Alessio
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Blanner, Christina
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Canozzi, Andrea
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Eudave, Luis
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Farhat, Luis C.
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Højlund, Mikkel
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Köhler-Forsberg, Ole
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Leffa, Douglas Teixeira
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Rohde, Christopher
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de Pablo, Gonzalo Salazar
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Vita, Giovanni
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Wesselhoeft, Rikke
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Martin, Joanna
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Baumeister, Sarah
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Bozhilova, Natali S.
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Carlisi, Christina O.
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Leno, Virginia Carter
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Floris, Dorothea L.
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Holz, Nathalie E.
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Kraaijenvanger, Eline J.
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Sacu, Seda
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Vainieri, Isabella
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Ostuzzi, Giovanni
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Barbui, Corrado
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Correll, Christoph U.
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Cortese, Samuele, Solmi, Marco, Michelini, Giorgia, Bellato, Alessio, Blanner, Christina, Canozzi, Andrea, Eudave, Luis, Farhat, Luis C., Højlund, Mikkel, Köhler-Forsberg, Ole, Leffa, Douglas Teixeira, Rohde, Christopher, de Pablo, Gonzalo Salazar, Vita, Giovanni, Wesselhoeft, Rikke, Martin, Joanna, Baumeister, Sarah, Bozhilova, Natali S., Carlisi, Christina O., Leno, Virginia Carter, Floris, Dorothea L., Holz, Nathalie E., Kraaijenvanger, Eline J., Sacu, Seda, Vainieri, Isabella, Ostuzzi, Giovanni, Barbui, Corrado and Correll, Christoph U. (2023) Candidate diagnostic biomarkers for neurodevelopmental disorders in children and adolescents: a systematic review. World Psychiatry, 22 (1), 129-149. (doi:10.1002/wps.21037).

Record type: Article

Abstract

Neurodevelopmental disorders – including attention-deficit/hyperactivity disorder (ADHD), autism spectrum disorder, communication disorders, intellectual disability, motor disorders, specific learning disorders, and tic disorders – manifest themselves early in development. Valid, reliable and broadly usable biomarkers supporting a timely diagnosis of these disorders would be highly relevant from a clinical and public health standpoint. We conducted the first systematic review of studies on candidate diagnostic biomarkers for these disorders in children and adolescents. We searched Medline and Embase + Embase Classic with terms relating to biomarkers until April 6, 2022, and conducted additional targeted searches for genome-wide association studies (GWAS) and neuroimaging or neurophysiological studies carried out by international consortia. We considered a candidate biomarker as promising if it was reported in at least two independent studies providing evidence of sensitivity and specificity of at least 80%. After screening 10,625 references, we retained 780 studies (374 biochemical, 203 neuroimaging, 133 neurophysiological and 65 neuropsychological studies, and five GWAS), including a total of approximately 120,000 cases and 176,000 controls. While the majority of the studies focused simply on associations, we could not find any biomarker for which there was evidence – from two or more studies from independent research groups, with results going into the same direction – of specificity and sensitivity of at least 80%. Other important metrics to assess the validity of a candidate biomarker, such as positive predictive value and negative predictive value, were infrequently reported. Limitations of the currently available studies include mostly small sample size, heterogeneous approaches and candidate biomarker targets, undue focus on single instead of joint biomarker signatures, and incomplete accounting for potential confounding factors. Future multivariable and multi-level approaches may be best suited to find valid candidate biomarkers, which will then need to be validated in external, independent samples and then, importantly, tested in terms of feasibility and cost-effectiveness, before they can be implemented in daily clinical practice

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Candidate diagnostic biomarkers for neurodevelopmental disorders in children and adolescents: a systematic review - Accepted Manuscript
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Accepted/In Press date: 7 October 2022
e-pub ahead of print date: 14 January 2023
Published date: 14 January 2023
Additional Information: Funding Information: S. Cortese is supported by the UK National Institute for Health and Care Research, NIHR (grant nos. NIHR203035, RP-PG-0618-20003, NIHR203684 and NIHR130077). G. Michelini is supported by a Klingenstein Third Generation Foundation fellowship; L.C. Farhat by the São Paulo Research Foundation; D. Teixeira Leffa by a NARSAD Young Investigator Grant from the Brain & Behavior Research Foundation; G. Salazar de Pablo by the Alicia Koplowitz Foundation: C. Carlisi and V. Carter Leno by a Wellcome Trust Postdoctoral Fellowship; D. Floris by the European Union's Horizon 2020 research and innovation programme (Marie Skłodowska-Curie grant agreement no. 101025785); N. Holz by the German Research Foundation and the Radboud Excellence Fellowship. S. Cortese and M. Solmi have contributed equally to this work. Supplementary information on the study is available at https://osf.io/wp4je/?view_only=8c349f45a9ac441490981acf946c8d9a.
Keywords: ADHD, Biological markers, autism spectrum disorder, communication disorders, genome-wide association studies, intellectual disabil­ity, motor disorders, neurodevelopmental disorders, neuroimaging, neurophysiology, specific learning disorders, tic disorders

Identifiers

Local EPrints ID: 473991
URI: http://eprints.soton.ac.uk/id/eprint/473991
ISSN: 2051-5545
PURE UUID: 7ab4cb98-9a51-4ab2-b860-6c6b8eb6ee17
ORCID for Samuele Cortese: ORCID iD orcid.org/0000-0001-5877-8075
ORCID for Alessio Bellato: ORCID iD orcid.org/0000-0001-5330-6773

Catalogue record

Date deposited: 07 Feb 2023 18:04
Last modified: 17 Mar 2024 07:40

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Contributors

Author: Samuele Cortese ORCID iD
Author: Marco Solmi
Author: Giorgia Michelini
Author: Alessio Bellato ORCID iD
Author: Christina Blanner
Author: Andrea Canozzi
Author: Luis Eudave
Author: Luis C. Farhat
Author: Mikkel Højlund
Author: Ole Köhler-Forsberg
Author: Douglas Teixeira Leffa
Author: Christopher Rohde
Author: Gonzalo Salazar de Pablo
Author: Giovanni Vita
Author: Rikke Wesselhoeft
Author: Joanna Martin
Author: Sarah Baumeister
Author: Natali S. Bozhilova
Author: Christina O. Carlisi
Author: Virginia Carter Leno
Author: Dorothea L. Floris
Author: Nathalie E. Holz
Author: Eline J. Kraaijenvanger
Author: Seda Sacu
Author: Isabella Vainieri
Author: Giovanni Ostuzzi
Author: Corrado Barbui
Author: Christoph U. Correll

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