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A number of studies have reported a decrement in fine motor skills following acute and chronic sleep deprivation in typical populations [1-3]. For example, Ayalon and Friedman [1] suggested that acute sleep deprivation among medical residents led to excessive daytime sleepiness and further reduced fine manual dexterity, as assessed by the Purdue pegboard test, . In addition, patients with chronic obstructive sleep apnea performed poorly in the dominant hand and assembly subtests of the Purdue pegboard test [4]. Furthermore, a high prevalence of sleep disorders has also been reported in individuals with Down syndrome (DS). For example, obstructive sleep apnea (OSA), bedtime resistance, sleep anxiety, night waking, parasomnias and daytime sleepiness were commonly reported sleep disorders and behaviors amongst children and adolescents with DS [5, 6]. Moreover, individuals with DS already have challenges in motor skills compared with their typical peers and other individuals with intellectual disabilities (ID) without DS [7-9]. Thus, it seems logical that their poor fine motor skills may be exacerbated by sleep disorders. However, to my knowledge, there is no research exploring the relationship between sleep disorders and fine motor skills in this population. Therefore, research is needed to determine whether or not sleep disorders and fine motor deficits may be linked in individuals with DS.
In the past years, most studies interested in motor performance in individuals with DS focused on gross motor skills, such as postural control [10-11] and locomotion [12]. To date, only a few studies investigated the importance of fine motor skills in this population. For example, manual dexterity is vitally important for self-care domains of daily life for individuals with DS. Dolva, Coster, and Lilja [13] reported that 5-year-old children with DS have low capabilities to perform fine motor skills, such as tooth brushing, tying shoelaces and toileting tasks. Only 11% and 0 % of participants were able to perform tooth brushing and tying shoelaces respectively. de campos, Rocha, and Savelsbergh [8] indicated that there appeared to be a relationship between chronological age (CA) and fine motor skills (i.e., reaching and grasping). Moreover, Sourtiji, Hosseini, Soleimani, and Hosseini [14] indicated a close relation between mental age (MA) and fine motor skill level, as measured by Peabody Developmental Motor Scales. Therefore, while studying the effect of sleep disorders on fine motor skills in individuals with DS, the effects of age (i.e., CA and MA) should also be taken into consideration.
Taken together, the objective of the current study was to examine the association between different factors of sleep disorders and fine motor skills, as measured by Purdue pegboard test, in young adults with DS. The Sleep Questionnaire by Simonds and Parraga [15] which was specifically designed and validated, for individuals with ID, including DS was utilized. In line with past literature, we expected that age would be a factor that attributed to fine motor deficits in individuals with DS. Furthermore, we expected that the increased caregiver ratings in sleep disorders, particularly ratings of OSA, would be related to fine motor deficits in persons with DS.

2. Materials and Methods
2.1 Participants
Participants in this study included 30 community-dwelling young adults with DS, aged 14-31 years. Sixty-seven percent of whom were males and 33% of whom were females. Participants were recruited from a variety of local DS organizations (e.g., Sharing Down syndrome Arizona, Down syndrome network, Special Olympics, and Raising Special Kids newsletters, meetings and emails lists, etc.). All participants resided at home with their parents. 
Participants� mental age was tested using the Peabody Picture Vocabulary test (3rd Ed.; PPVT-III). Then, vision and hearing assessments were conducted to ensure they had the capabilities to perform the tests. Participants with normal or at least 20/100 vision, normal or corrected-to-normal hearing, and no history of sensory impairment, physical disabilities, and neurological disorders were included. No participants were excluded based on pre-screening assessments.

2.2. Procedure
The parent or guardian signed a consent form and answered the Sleep Questionnaire. Participants read (or were read) and signed the informed consent or assent forms before test started. First, PPVT-III was conducted.  A stimulus word was orally presented to the participant with a set of pictures and then the participant was requested to select the picture that represented the meaning of the stimulus word. In addition, a vision test was conducted using a standard eye chart (i.e., Snellen) and a modified version which consists of E�s pointing in different directions for participants who could not recognize letters. In this test, they were instructed to say or point which direction the E�s were pointing. Further, hearing was tested using an audiometer (the Maico Ma 25). Moreover, handedness was also tested by using a seven-item handedness inventory [16]. Participants physically performed writing with a pen, drawing a circle with a pen, using scissors, using a hammer, throwing a ball, pretending to brush their teeth, and pretending to eat with a spoon. If they performed four out of seven items with their right/left hand, this right/left hand were deemed as their dominant hands and the other hands as non-dominant hands. All procedures were approved by the human ethics committee at our university. The total experiment lasted about an hour.
 	2.2.1. Sleep Questionnaire by Simonds and Parraga [15]
	The Sleep Questionnaire was a 7-point Likert-type scale, developed by Simonds and Parraga (SQ�SP; 1982) and modified by Stores et al. [17] to explore sleep problems in children with DS and other ID. Based on these studies, the test�retest reliability for the total SQ�SP score ranged from, r = .83 to 1.00. Four types of sleep factors were derived from SQ-SP, including 1) Disorders of initiating and maintaining sleep, 2) Features associated with OSA, 3) Disorders occurring during sleep, and 4) Sleep disorders occurring during the day. The first factor, consisted of eight items, rated bedtime resistance, the second factor, consisted of six items, rated snoring or gasping for breath, the third factor, consisted of six items, rated nightmares or sleep walking, and the fourth factor, consisted of six items, rated daytime sleepiness, naps, or daytime overactivity. Higher scores represented poorer sleep behavior.
	2.2.2. Purdue Pegboard Test
For evaluating fine manual dexterity of the hand, the Purdue Pegboard test was used (Lafayette Instrument Model #32020). It consists of a board with four cups across the top and two vertical rows of 25 small holes down the center. The cups from right to left side each contained 25 pegs, 20 collars, 40 washers and 25 pegs. Four subtests comprised the test: dominant hand, non-dominant hand, bimanual, and assembly. Each subtest was administered three times in a row. The predictive validity of employability in adults with intellectual disabilities is .70 [18] and the internal reliability of this methodology is reported to be .86 [19].
The dominant hand and non-dominant hand subtests of the Purdue Pegboard consisted of placing as many pegs as possible in the column corresponding to the hand being tested. The dominant hand is tested first followed by the non-dominant hand. The scores on both subtests were the number of pegs placed in a column within 30 sec. In the bimanual subtest, the dominant and non-dominant hands simultaneously placed pegs in both columns. The scores on this subtest were the number of pairs of pegs placed within 30 sec. The assembly subtest required picking up and placing pegs, washers, and collars using alternating hands. The assembly subtest was limited to 60 sec. This score represents the number of pieces assembled (i.e., pin, washer, collar, second washer).

2.3. Data Analysis
Statistical analysis were performed SPSS 20.0. For all analyses, the significance level was set at p <0.05. The correlations between the performance of the Purdue Pegboard test and age (CA, MA) were investigated with the Pearson product-moment correlation coefficient. In addition, a partial correlation analysis was conducted to examine the correlations between sleep problems and the fine manual dexterity, controlling for age. 

3. Results
The means and standard divisions for participants� background information (i.e., chronological age (CA), mental age (MA), sleep problems and performance in each Purdue pegboard subtest) are presented in Table 1.

Insert Table 1 about here

3.1 Relationship between age and fine manual dexterity
Table 2 shows the correlation between CA, MA and the performance of the Purdue pegboard subtests. The Non-dominant subtest was negatively correlated with MA (r=-0.46, p=0.01). The Bimanual subtest was also significantly negatively correlated with MA (r=0.39, p=0.03). Therefore, participants with high MA successfully completed more work on the Non-dominant and Bimanual subtests.

Insert Table 2 about here

3.2 Relationship between sleep problems and fine manual dexterity 
Table 3 shows the correlation between each sleep factor and the performance of Purdue pegboard subtests, controlling for MA. The �sleep related disorders during the day� factor was negatively correlated with Non-dominant hand (r=-0.47, p=0.01), Bimanual (r=-0.49, p=0.01), and Assembly subtests (r=-0.44, p=0.02). Hence, high ratings of sleep disorders on this scale related to poorer scores on the Purdue pegboard test. 

Insert Table 3 about here

4. Discussion
	This study first examined the relationship between age and fine manual dexterity in 30 young adults with DS. Inconsistent with a previous study [8], our results show that, only MA was related to the performance of fine motor skill in individuals with DS. The possible interaction with reduced language development may lead to the association between MA and fine manual dexterity in non-dominant and bimanual subtests. For example, Groen, Yasin, Laws, Barry, and Bishop [20] showed the association between poor language and poor motor development in children with DS. They found an association between hand preference and language skills in individuals with DS. Interestingly our results were only evident in Non-dominant and Bimanual subtests.  This may be because, compared to dominant hand, more motor unit recruitment, initial firing rate, and high discharge variability were needed to control the non-dominant hand [21]. Other researchers have also found that individuals with DS showed more deficits in bimanual coordination compared to their typical developing peers [22]. Therefore, high mental age may play a more important role than chronological age for individuals with DS in understanding instructions and monitoring their movements during non-dominant and bimanual subtests of the Purdue Pegboard test.   
Furthermore, inconsistent with previous studies [4, 23], patients with OSA had severe impairments in manual dexterity as assessed by the Purdue Pegboard test. Our preliminary findings indicated that a negative, but not statistically significant, association between the features of OSA and fine motor impairment in individuals with DS. The Purdue Pegboard is a normative data test of motor speed and coordination. Beebe, Groesz, Wells, Nicols and McGee [24] even conducted a meta-analysis to understand the neuropsychological effects of OSA in norm-referenced and case-controlled data and suggested the effect size was smaller and non-significant in the norm-reference data, consistent with the current results. In addition, they also suggested that OSA had less effect on motor speed. To my knowledge, motor functions in patients with OSA have been investigated by other means of the grooved pegboard [25] and finger tapping tests [26]. Thus, a direction of future studies may consider other fine motor tests to explore the effects of OSA on the development of control and coordination of fine motor skills in individuals with DS. 
Moreover, an interesting  results the significant correlation between sleep related disorders during the day (e.g., excessive daytime sleepiness, nap, overactivity) and impaired fine manual dexterity in individuals with DS,. The Controlled Attention Model may be the approach that can be applied to explain this relationship [27]. While performing the Purdue Pegboard test, participants were required to pay attention to the task and it seems likely that the individuals with DS were less capable of maintaining attention. In particular, sleep related disorders during the day have also been shown to affect cognitive performance in individuals with DS [28]. Therefore, it is possible that attentional and cognitive deficits that result from sleep disorders during the day prevent individuals with DS from processing and keeping information long enough and which cause the poor fine manual dexterity in Purdue Pegboard test. 
As for disorders of initiating and maintain sleep (e.g., insomnia), there were weak and non-significant associations with the performance of Purdue pegboard subtests. These results may be consistent with previous studies because researchers found that the performance on the pegboard test did not differ between patients with insomnia and healthy participants [29-30]. Other disorders during sleep (e.g., nightmares, sleep walking) also showed weak and non-significant associations with the performance of Purdue pegboard subtests. However, to my knowledge, there is no research indicating its relationship with fine motor deficits in both typical and atypical populations.
Hence, mental age, sleep disorders, as well as cognitive function [31] have been related to the performance of the Purdue pegboard test in individuals with DS. Thus, the development of fine motor skills in individual with DS seems to be a complex process that involves in physiological as well as psychological factors. Future studies should consider the extent to which factor related to their fine motor skills. In addition, the current results may provide coaches, therapists and parents the information about how to improve fine manual dexterity in individuals with DS. 
For the present study, although the parent-report questionnaire had high validity and reliability to individuals with intellectual disabilities including DS, sleepiness in individuals with DS is difficult to measure because parents may not interpret or be aware of sleep issues with their child. For example, the participant may awaken without the parent�s knowledge. Therefore, differences in parental reporting highlight the need for more objective instruments or direct measures of sleep in individuals with DS.
Conclusions:
In summary, the mediating role of sleep disorders in the development of fine motor skills in individuals with DS remains unclear. However, this is the first study provides some support for the association between sleep disorders and impaired fine motor skills in individuals with DS. The results show sleep disorders during the day may indicate attentional and cognitive deficits that in turn have an effect on the development of fine manual dexterity in individuals with DS. 




















Acknowledgement
We appreciate the City of Tempe Special Olympics for helping us with participant recruitment.  Thank you to Michelle Snow who was the student who helped with data collection. This research was supported by The Graduate Research Support Program at Arizona State University and Health Professionals Student Grant Program from Special Olympics. Jamie Edgin�s involvement was funded through grants from the Thrasher Research Fund, the Down syndrome Research and Treatment Foundation, and Research Down Syndrome. 















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