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Syst Rev. 2017 Sep 15;6(1):189. doi: 10.1186/s13643-017-0581-9.

Risk of mortality among children, adolescents, and adults with autism spectrum disorder or attention deficit hyperactivity disorder and their first-degree relatives: a protocol for a systematic review and meta-analysis of observational studies.

Author information

1
Department of Medicine, University of Valencia/INCLIVA Health Research Institute and CIBERSAM, Valencia, Spain. ferran_catala@hotmail.com.
2
Fundación Instituto de Investigación en Servicios de Salud, Valencia, Spain. ferran_catala@hotmail.com.
3
Clinical Epidemiology Program, Ottawa Hospital Research Institute, Ottawa, ON, Canada. ferran_catala@hotmail.com.
4
Clinical Epidemiology Program, Ottawa Hospital Research Institute, Ottawa, ON, Canada.
5
School of Epidemiology, Public Health and Preventative Medicine, University of Ottawa, Ottawa, ON, Canada.
6
School of Public Health and Preventive Medicine, Monash University, Melbourne, VIC, Australia.
7
Instituto Aragonés de Ciencias de la Salud, Red de Investigación en Servicios de Salud en Enfermedades Crónicas (REDISSEC), Zaragoza, Spain.
8
Fundación para el Fomento de la Investigación Sanitaria y Biomédica de la Comunitat Valenciana (FISABIO-Salud Pública), Valencia, Spain.
9
Geriatric Research Education and Clinical Center, VA Boston Healthcare System, Boston, MA, USA.
10
Division of Aging, Department of Medicine, Brigham and Women's Hospital, Harvard Medical School, Boston, MA, USA.
11
Department of History of Science and Documentation, University of Valencia, Valencia, Spain.
12
Unidad de Información e Investigación Social y Sanitaria-UISYS, University of Valencia-Spanish National Research Council (CSIC), Valencia, Spain.
13
Department of Medicine, University of Valencia/INCLIVA Health Research Institute and CIBERSAM, Valencia, Spain.
14
Division of Pharmacoepidemiology and Pharmacovigilance, Spanish Medicines and Healthcare Products Agency, Madrid, Spain.
15
Department of Health Systems and Services, Pan American Health Organization, Unit of Medicines and Health Technologies, Washington, DC, USA.
16
Hospital Clínic, Universitat de Barcelona, Institut d'Investigacions Biomèdiques August Pi i Sunyer (IDIBAPS) and CIBERSAM, Barcelona, Spain.
17
Life Sciences Department, Barcelona Supercomputing Center, Barcelona, Spain.
18
Structural Biology and Biocomputing Programme, Spanish National Cancer Research Centre (CNIO), Madrid, Spain.

Abstract

BACKGROUND:

Autism spectrum disorder (ASD) and attention deficit hyperactivity disorder (ADHD) are childhood onset neurodevelopmental disorders that may persist into adulthood. ASD and ADHD tend to run in families and may have a significant negative impact on the health and longevity of those with the disorder and their relatives. The aim of this study will be to analyze the risk of mortality among children, adolescents, and adults with ASD or ADHD and their first-degree relatives.

METHODS/DESIGN:

We will conduct a systematic review and meta-analysis of observational studies. Searches of PubMed/MEDLINE, EMBASE, PsycINFO, SCOPUS, and ISI Web of Science will be used to identify epidemiological studies. Eligible studies will be observational studies reporting study-specific data for all-cause mortality or cause-specific mortality in children, adolescents, or adults with ASD or ADHD and/or their first-degree relatives. Cohort studies and case-control studies will be included. The primary outcome will be all-cause mortality. The secondary outcome will be cause-specific mortality. Two reviewers will independently screen references identified by the literature search, as well as potentially relevant full-text articles. Data will be abstracted, and study risk of bias/methodological quality will be appraised by two reviewers independently. The methodological quality of epidemiological studies will be appraised using the Newcastle-Ottawa Scale (NOS). Conflicts at all levels of screening and abstraction will be resolved through discussion. Random-effects meta-analyses of primary studies will be conducted where appropriate. Subgroup analyses for exploring statistical heterogeneity, if feasible, will include gender, age group, ethnicity, comorbidities, classification of cause of death, and relevant study characteristics.

DISCUSSION:

Our study will establish the extent of the epidemiological evidence underlying the risk of mortality among children, adolescents, and adults with ASD or ADHD and their first-degree relatives. We anticipate that our findings will be of interest to patients, their families, caregivers, healthcare professionals, scientists, and policy makers. Implications for future epidemiological research will be discussed.

SYSTEMATIC REVIEW REGISTRATION:

PROSPERO CRD42017059955 .

KEYWORDS:

Adolescent; Adult; Attention deficit hyperactivity disorder; Autism spectrum disorders; Child; Child development; Epidemiology; Meta-analysis; Mortality; Neurodevelopmental disorders; Systematic review

PMID:
28915839
PMCID:
PMC5603059
DOI:
10.1186/s13643-017-0581-9
[Indexed for MEDLINE]
Free PMC Article

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