What is the prevalence of gross motor impairment in children 18 years or younger with a diagnosis of Fetal Alcohol Spectrum Disorder or exposed to "moderate" to "heavy" levels of alcohol prenatally?
Which gross motor domains are impaired in children 18 years or younger with a diagnosis of Fetal Alcohol Spectrum Disorder or exposed to "moderate" to "heavy" levels of alcohol prenatally?
The search strategy will be designed to access published material and will compromise three stages:
(i) Sources of published data will include electronic databases (MEDLINE, EMBASE, CINAHL, The Cochrane Library, AMED, PEDro and Google Scholar).
(ii) Reference lists from comprehensive reviews and included studies will be searched for possible references not otherwise found.
(iii) Experts in the field will be approached for additional references meeting the inclusion criteria.
We will not apply language, year of publication or type of publication restrictions.
Types of study to be included
All human observational studies including cross sectional, cohort, case control studies, population based surveys or randomized controlled trials.
Single case studies, reviews or animal studies.
Condition or domain being studied
Alcohol is a teratogen and exposure "in utero" may cause a range of lifelong conditions termed "Fetal Alcohol Spectrum Disorders" (FASD). These may result in significant cognitive, behavioural, physical and motor deficits which can persist into adulthood. The diagnostic process for FASD is standardized and involves the application of recognised criteria such as those specified by the University of Washington, Institute of Medicine, Centres for Disease Control, or the Canadian Guidelines. Diagnostic assessment includes examination of dysmorphology, growth restriction, structural central nervous system (CNS) abnormality and CNS domains of impairment. As part of the examination of CNS domains, FASD diagnostic guidelines recommend the accurate assessment of gross motor (GM) and fine motor (FM) functioning with use of standardised cut- offs for impairment.
Motor impairment in children with FASD is well established particularly with respect to fine motor (FM) function. Impairment of gross motor skills is often reported but its prevalence and the specific domains affected are not clear. Gross motor skills require the use of large muscle groups for the purpose of physical activity. In children they play an important part of skill repertoire which enables exploration of the environment, social interaction and play.
Understanding the nature and prevalence of gross motor impairment consequential to prenatal alcohol exposure will assist in informing assessments, establishing the needs of children with impairment, resourcing these needs and developing strategies to ameliorate impairment. It is timely to provide parents, clinicians, teachers and public health funders with the best available evidence on this matter.
Children aged 0 - 18 years diagnosed with a FASD or exposed to alcohol "in utero" deemed to be "moderate" to "high" risk levels by the study author. Gross motor performance must be measured with a standardized assessment tool. Outcomes need to be reported related to alcohol exposure or using a defined threshold for impairment .
Children not diagnosed with a FASD, or with absent or "low" risk levels of exposure to alcohol "in utero" as deemed by the study author. Studies reporting children with a FASD diagnosis in conjunction with a genetic disorder or other motor disorder associated with motor dysfunction will also be excluded.
Exposure Inclusion Criteria:
This review will consider all studies involving children diagnosed with FASD according to the application of recognised criteria as specified by the University of Washington, Institute of Medicine, or Centres for Disease Control, Canadian Guidelines or other, or exposed to alcohol "in utero" deemed at "moderate" to "high" risk levels by the study author.
Exposure Exclusion Criteria:
Studies reporting prenatal alcohol exposure in conjunction with other substance use where adjustment has not been made for the other substance will be excluded.
The comparison will be made to children ages 0 - 18 years without prenatal alcohol exposure or exposed to alcohol "in utero" deemed at "low" risk levels by the study author.
1. Prevalence of gross motor impairment.
2. Domains of gross motor impairment.
Outcomes need to be reported:
(i) related to alcohol exposure OR
(ii) using a defined threshold for impairment (Study needs to provide quantitative measures of gross motor function. Simply reporting a p value is insufficient.)
Data extraction, (selection and coding)
(i) Selection of studies: Two reviewers will independently determine if each study meets the inclusion criteria for the review using a clearly defined inclusion criteria checklist. Any differences regarding inclusion will be resolved by discussion and consensus with a third reviewer.
(ii) Data Extraction: Two reviewers will independently extract data from each study using clearly defined data extraction forms and will be cross-checked. Discrepancies will be resolved by consensus. The extraction of data includes study location, study design, demographics, sample size, study group characteristics, reference group characteristics, standardized assessment tool used to measure gross motor performance, assessors profession, gross motor areas significantly affected compared with control, prevalence of gross motor impairment, statistical analysis for gross motor outcome measure, confounders controlled for in gross motor statistical analysis. Papers will be categorized according to the following age ranges: 0 to < 2 years, 2 to < 5 years, 5 to < 10 years and = 10 and <18 years.
Risk of bias (quality) assessment
The methodological quality of the included studies will be also assessed by two independent reviewers. As recommended by the STROBE guidelines and Sanderson et al (2007), the following 7 point Critical Appraisal Tool (CAT) will be used to assess the following domains:
Control for Bias
1. Representative sample
2. Defined sample
3. Outcome rater blinding
Appropriate measurement of variables
4.Methods of Assessment
(i) Face validity
(ii) Evidence of clinometrics
5. Outcome Data reported
6. Adequate power calculation
Control for Confounding
7. Statistical Adjustment: multivariable or regression analysis conducted, with adjustment for potentially confounding factors
Each of these criteria will be explicitly judged using: Yes = criterion clearly satisfied; No = criterion clearly not satisfied or unclear if criterion is satisfied. Any differences between the two reviewers will be resolved by discussion and consensus with a third reviewer.
Sanderson S, Tatt LD, Higgins, JP. Tools for assessing quality and susceptibility to bias in observational studies in epidemiology: a systematic review and annotated bibliography. International Journal of Epidemiology. 2007;36: 666 – 676.
Strategy for data synthesis
If appropriate with available data, results from comparable groups of studies will be pooled for statistical meta-analysis. Where statistical pooling is not possible the findings will be presented in narrative form and descriptive statistics only will be used.
Analysis of subgroups or subsets
If data are suitable, analysis of outcomes will be performed by age and severity of gross motor impairment.
The essential messages from the review will be communicated to appropriate audiences through publication, conference presentations and other pediatric and FASD networks.
Contact details for further information
Royal North Shore Hospital
St Leonards 2065
Organisational affiliation of the review
1. Discipline of Paediatrics and Child Health – the University of Sydney, The University of Sydney 2. The George Institute for Global Health, Physiotheraoy Department , Royal North Shore Hospital
Ms Barbara Lucas, 1. Discipline of Paediatrics and Child Health – the University of Sydney, 2. The George Institute for Global Health, Royal North Shore Hospital Physiotherapy Department Professor Elizabeth Elliott, 1. Discipline of Paediatrics and Child Health – the University of Sydney, 2.The George Institute for Global Health, 3. Sydney Medical School - University of Sydney, 4.The Sydney Children’s Hospital Networks (Westmead). Professor Jane Latimer, 1. The George Institute for Global Health, 2. Sydney Medical School – the University of Sydney Dr Manuela Ferreira, 1. The George Institute for Global Health, 2. Sydney Medical School – the University of Sydney
Ms Danielle Dries, Physiotherapy Department - Royal North Shore Hospital Ms Taryn Jones, Faculty of Human Sciences - Macquarie University, The George Institute for Global Health Mrs Mandy Lau, The George Institute for Global Health
Details of any existing review of the same topic by the same authors
Anticipated or actual start date
02 April 2012
Anticipated completion date
31 December 2012
Discipline of Paediatrics and Child Health – the University of Sydney
Formal screening of search results against eligibility criteria
Risk of bias (quality) assessment
PROSPERO This information has been provided by the named contact for this review. CRD has accepted this information in good faith and registered the review in PROSPERO. CRD bears no responsibility or liability for the content of this registration record, any associated files or external websites.