Background
Local Aboriginal leaders in the remote Fitzroy Valley region of Western Australia introduced alcohol restrictions in 2007 because they were concerned about the social and health effects of chronic alcohol misuse. These concerns included the potential harm caused by alcohol consumption during pregnancy, which can cause Fetal Alcohol Spectrum Disorder (FASD). In 2009 local leaders initiated ‘The Lililwan Project’ (‘Lililwan’ is Kimberley Kriol for ‘all the little ones’) to determine the prevalence of FASD [
1]. Diagnoses on the FASD spectrum include Fetal Alcohol Syndrome (FAS) and partial Fetal Alcohol Syndrome (pFAS), both with characteristic facial anomalies and impaired growth; and Alcohol-Related Neurodevelopmental Disorder (ARND) or Neurodevelopmental Disorder – Prenatal/Alcohol Exposed (ND-PAE/ND-AE) with neurodevelopmental impairment in the absence of physical features [
2,
3].
PAE can affect the development and function of the corpus callosum [
4], cerebellum [
5], basal ganglia [
6], and motor cortex [
7], and children with FASD may have skeletal malformations [
8], abnormal muscle development [
9], tremor [
10], and impaired nerve conductivity [
11]. All these factors may impair fine motor performance. Fine motor skills include basic skills such as grip strength, and more complex skills including visual (or fine) motor integration, manual dexterity, and upper-limb coordination. These skills underpin many self-care, academic, and recreational activities, including handwriting, dressing, and ball sports. Fine motor skills are particularly important in primary school aged children, who can spend more than half of their day completing tasks which require fine motor skills [
12]. Handwriting quality can be affected by poor fine motor skills, and students with poor handwriting often receive poorer grades [
13]. Teacher reports indicate that 20.6% of first year students at Fitzroy Fitzroy Crossing are below the Australian population 10th percentile for fine and gross motor skills [
14]. Many Australian Aboriginal students perform below-average on the National Assessment Program – Literacy and Numeracy (NAPLAN), which is conducted annually with students in Years 3, 5, 7, and 9 [
15].
Few studies of children with PAE or FASD have reported whether they have a motor impairment, and of those that do, many report a motor score that is a combination of fine motor and gross motor skills [
16‐
18], or a score based on subtests of generalised developmental assessment tools [
19], such as the Eye and Hand Coordination subscale from the Griffith’s Mental Development Scales [
20]. Individuals with FASD can have subtle neurological impairment, and researchers have highlighted the importance of assessing a range of specific areas of function rather than reporting amalgamated scores [
18,
19]. Motor scores that are an average of fine and gross motor skills provide little insight into deficits, which is essential for understanding the child’s neurological profile and developing appropriate therapy goals.
Several studies have assessed a range of fine motor skills in children with PAE or FASD [
21‐
24], but each has used varying assessment tools and none report data from an entire population age-cohort. Motor skills in children with PAE or FASD are summarised in three systematic reviews. In one review, ‘visual and motor’ skills were not associated with mild, moderate, or binge PAE, however, none of the included studies assessed children older than 5 years [
25]. Another review found an association between motor impairment and levels of PAE, but did not differentiate between fine and gross motor skills [
26]. We reviewed fine motor skills in primary school aged children with PAE or FASD [
27], and found that complex fine motor skills, such as visual-motor integration, were more likely to be impaired than basic skills, such as grip strength. We identified a range of assessment tools used to assess fine motor skills in children with PAE or FASD, but few that comprehensively assessed a range of different skills.
Study hypotheses
Fine motor proficiency and prevalence of impairment amongst children in the remote Fitzroy Valley, Western Australia were evaluated. We hypothesised that rates of fine motor impairment would be high due suspected high rates of neurodevelopmental and socioeconomic risk factors, including PAE. We also hypothesised that children with PAE, particularly those with FASD, would have the most impairment due to the teratogenic effect of alcohol on the central and peripheral nervous systems involved in performance of fine motor skills.
Study aims
1. Assess and evaluate fine manual control (fine motor precision and fine motor integration) and manual coordination (manual dexterity and upper-limb coordination) in a cohort of children in the Fitzroy Valley.
2. Compare fine motor skills of children (i) without PAE; (ii) with PAE but not FASD; and (iii) with FASD.
3. Determine the prevalence of moderate (≤ 16th percentile) and significant (≤ 2nd percentile) fine motor impairments in the cohort.
Discussion
This is the first study to comprehensively assess fine motor skills in a population-based cohort of predominantly Aboriginal children in Australia. Many children in our study had high levels of PAE and were diagnosed with FASD. The cohort’s mean BOT-2 Fine Motor Composite scores were in the ‘average’ range, an unexpected finding given the high levels of PAE and other neurodevelopmental risk factors in our cohort. However, in keeping with our hypothesis, children with FASD had poorer fine motor skills than children without PAE. Manual coordination skills, including fine motor speed, manual precision, and coordinated arm and hand movement were specific areas of difficulty for children with FASD. Few children had severe impairment (below the 2nd percentile), but rates of moderate impairment (below the 16th percentile) were very high.
Other studies of fine motor impairment in children with PAE or FASD have also reported a mixed profile of strengths and difficulties. A range of assessment tools have been used to evaluate fine motor skills in children with PAE or FASD, including the Visuomotor Precision subtest from the Developmental Neuropsychological Evaluation (NEPSY) [
40], the Movement Assessment Battery for Children (M-ABC) [
41], and The Beery Buktenica Developmental Test of Visual-Motor Integration (Beery VMI) [
42]. Other studies have reported mixed findings for fine motor precision [
24,
43] and manual dexterity [
44,
45] skills, which weren’t impaired in children with PAE or FASD in our study. Ball skills were also not impaired, which is consistent with other reported findings [
44‐
46]. We found that visual-motor integration (termed ‘fine motor integration’ in the BOT-2) wasn’t impaired, but this contradicts other studies which commonly report visual-motor integration impairment in children with FASD [
47‐
49]. This may be due to the limited number of tasks used to evaluate this skill in the BOT-2 (
n = 8), compared to the more commonly used Beery VMI (
n = 30). The Beery VMI formed part of the neurodevelopmental assessment battery in the Lililwan Project, and we reported that the Fine Motor Coordination subtest of the Beery VMI was significantly lower in children with FASD [
50].
Only one other study group [
17] has published motor outcomes in children with FASD using the BOT. These authors used an earlier version of the BOT (1st edition), which does not include a Fine Motor Composite score. The authors reported that the motor score (an amalgamation of fine and gross motor skills) was not significantly different in children with FASD (M = 49.1) compared to ‘typically developing’ (M = 57.7,
p = 0.36) children. These non-significant findings may result from areas of stronger skills masking fine motor impairments, in much the same way that children in our cohort with FASD had an ‘average’ Fine Motor Composite score (M = 45.2), which was derived from relatively stronger Manual Coordination (M = 51.8) and weaker Fine Manual Control scores (M = 41.1).
Implications of prevalence rates
The very low prevalence of severe fine motor impairment in our cohort has implications for FASD diagnosis. The University of Washington 4-digit Diagnostic Code [
51] and the Canadian FASD Diagnostic Guidelines [
3] each advise that scores 2
SD below the mean (≤ 2nd percentile) indicate impairment when diagnosing FASD. In contrast, 1
SD below the mean (≤ 16th percentile) indicates impairment according to the Centers for Disease Control (CDC) [
2]. Other authors have also proposed a 1
SD cut-off for identifying impairment for ND-PAE [
52]. Only one child in our cohort (who had FASD) had fine motor scores below the 2nd percentile, which seems conservative given the high levels of PAE and other neurodevelopmental risk factors in our cohort. This issue warrants further consideration and investigation.
Strengths
This study is the first comprehensive, population-based study of fine motor skills in Aboriginal children in Australia. It is also the first to use a standardised fine motor assessment to develop a comprehensive profile of fine motor skills in children with PAE and/or FASD.
Limitations
Most children in our study identified as Australian Aboriginal and all were living in remote communities, and so the results should not be generalised. Nevertheless, outcomes may be relevant to other populations with similar demographics. Although the study involved almost two entire age cohorts and had a high participation rate (%), the sample size was too small to statistically control for potentially confounding factors. However, many risk factors, such as early life trauma and low socioeconomic status, were common to almost all children in our study. Many children without PAE also had a moderate level of fine motor impairment, and thus impairments cannot be solely attributed to PAE. However, the high proportion of children in our cohort with “risky” or “high risk” levels of PAE make it likely that PAE contributed, at least in part, to the identified fine motor impairment.
Recommendations and future directions
This study highlights the importance of comprehensively assessing a range of fine motor skills in children with PAE or suspected FASD. Other researchers have expressed concerns that composite scores may not be sensitive enough to detect subtle neurological impairment in children with FASD [
18,
19]. Our findings support these concerns. We recommend that a range of fine motor skills be assessed in children with PAE, and outcomes not be amalgamated with other fine or gross motor scores, because an averaged ‘motor’ score could mask specific difficulties, resulting in inaccurate diagnoses and missed opportunities for therapeutic support.
Conclusions
Children in our cohort had Fine Motor Composite scores in the ‘average’ range. Upper-limb coordination (ball skills) was a strength, while fine motor integration skills (copying complex shapes) were an area of weakness. Children with FASD had significantly lower Fine Motor Composite and Manual Coordination scores than children without PAE. These outcomes highlight the importance of reporting specific types of fine motor skills, rather than an amalgamated ‘motor’ or even ‘fine motor’ score. The very high levels of impaired fine motor precision and fine motor integration skills highlight the need for therapeutic intervention for many children in the Fitzroy Valley, regardless of PAE, to encourage successful participation in self-care, academic, and recreational activities.
Acknowledgements
Thanks to the people and the children of the Fitzroy Valley who have participated in the Lililwan Project. The people of the Fitzroy Valley have bravely acknowledged the issues caused by alcohol in their communities, and have taken positive steps to support the needs of their children.
Members of the Lililwan Project team who contributed clinical, cultural, and administrative support: Fabrice Bardy, Dr. Joshua Bowyer, Dr. Robyn Bradbury, Dr. Heather Olson, Vanessa Carson, Emily Carter, Natalie Davey, Dr. Harvey Dillon, Sharon Eadie, Dr. Emily Fitzpatrick, Marmingee Hand, Carolyn Hartness, Genevieve Hawkes, Lorian Hayes, Dr. Samantha Kaiser, Meredith Kefford, Annette Kogolo, Aimee Leong, Denise Macoun, Dr. Raewyn Mutch, Juliette O’Brien, Marilyn Oscar, Trine Pedersen, Claire Salter, Charlie Schmidt, Rhonda Shandley, Stanley Shaw, Dr. Gemma Sinclair, Julianne Try, Dr. Angus Turner, Dr. Amanda Wilkins, and Harry Yungabun.