DOI: https://doi.org/10.21203/rs.3.rs-1804500/v1
Many therapies for enhancing motor function in children with Down syndrome are regarded to be beneficial. Multiple systematic reviews have analyzed the available evidence to determine which physical therapy interventions are the most effective. However, readers are now confronted with a slew of systematic studies that produce contradictory results.
The goal of this study was to compile current evidence from systematic reviews on the effects of physical therapy interventions in children with Down syndrome, such as treadmill training, progressive resistive training, whole-body vibration training, virtual reality therapy, and neuromuscular training.
Google scholar, PEDro, Cochrane, PubMed, and Scopus were used to search for relevant health resources. Titles, abstracts (k = 0.78), and full-text articles (k = 1.0) were chosen by two reviewers separately. Systematic reviews were considered if they addressed a specific research issue, explicitly stated the search strategy criteria and study selection/inclusion criteria, and conducted a thorough literature search. The modified R-AMSTAR technique was used to assess the methodological quality of systematic reviews. The collected primary studies were subjected to a meta-analysis.
Each systematic review's findings were tabulated according to evidence levels, with outcomes classified using the International Classification of Functioning, Disability, and Health framework. Even though different reviews had different interpretations of the results, the conclusions were reached.
These physical therapy programs involving treadmill training, progressive resistive training, whole-body vibration training, virtual reality therapy, and neuromuscular training; improved muscular strength and balance provided moderate evidence, while other outcomes (such as muscle endurance, cardiovascular fitness, and body composition) provided less conclusive or limited evidence.
Systematic review registration PROSPERO 2021, CRD42021264910
Down syndrome (DS) is a chromosomal abnormality caused by one of three chromosomal abnormalities: trisomy 21 (the most well-known), translocation, and/or mosaicism. This chromosome change occurs during the creation of the fetus, more specifically during cell division, and will characterize the syndrome's indications and symptoms [1, 2]. In the United States, 1 in 732 live births is expected to be affected. It is the most common hereditary cause of intellectual impairment and developmental delay. Individuals with DS have had an average annual raise of 0.94life-years during the last 50 years, with an average life expectancy of 60 years, according to recent studies. Individuals with DS may live as long as the general population, according to life expectancy trends [3].
The basic characteristics of DS are delayed neuro-psychomotor development, global muscular hypotonia, and ligament laxity, which cause gait acquisition to take an average of two years and cognitive functions to be affected. Motor disorders frequently result in the development of aberrant postural control, resulting in instability and a disrupted gait pattern, as well as increased energy expenditure and diminished performance [4, 5].
As people with DS live longer, it's more vital than ever to assess the impact of a lifetime with a disability, as well as areas where improvements may be made to improve participation. It may be feasible to influence an individual's everyday activities and social participation. Exercise therapies and physical exercise have been thoroughly explored for individuals in a variety of populations, including those with obesity, cardiovascular disease, and cancer, with good evidence to back up the benefits [6, 7].
Exercise intervention has also been shown to improve cardiovascular and muscular endurance, and strength, and reduce total body fat mass percentage in people with DS, according to research. Recently, research has begun to focus on the psychological consequences of exercise intervention and physical activity, favorable results, looking at characteristics including self-efficacy, motivation, mood, satisfaction, and quality of life [8].
With the World Health Organization's (WHO) introduction of the International Classification of Functioning, Disability, and Health (ICF) model, a shift in rehabilitation has occurred that focuses on the full person and individual quality of life. Instead of focusing on impairment, this standard framework for classification focuses on an individual's health. The International Classification of Functioning, Structure, and Involvement (ICF) model explains health and health-related domains as they relate to an individual's body function and structure, activity, and participation [11].
The psychological and physiological functions of body systems are defined as body function; body structure refers to all physical aspects of the body. An individual's activity is defined as a task or action that they perform. The term "participation" means "to take part in anything." Involvement in a life/social context is defined as participation. Instead of focusing solely on impairment, the ICF model places a greater emphasis on an individual's overall health. The ICF model can be used to give a framework for systematically comparing research outcomes across investigations. As a result, the ICF model could be used to categorize research that focuses on the psychosocial, long-term effects of exercise intervention and physical activity. However, limited research has examined the impact of an exercise intervention on daily activities and motor performance in people with DS, and few systematic reviews have examined the impact of various interventions on physical engagement in people with DS across the lifespan [9, 10].
The goal of this meta-analysis, to the best of our knowledge, was to assess the effectiveness of various intervention regimens on motor competence and their impact on daily living activities and social interactions in individuals with DS using all systematic review studies in published literature. The methodological quality of prior reviews, as well as the outcome measures and participant selection of included main studies, were reviewed in the analysis to discover the cause for inconsistencies in the literature and directions for future study. The contradictory findings were thought to be explained by discrepancies in the methodological quality of the systematic reviews and the breadth of the research included.
Systematic reviews had to answer a focused research question, explicitly specify the search strategy criteria in addition to study selection/inclusion, and conduct a comprehensive literature search to be considered for this analysis.
Systematic reviews, whether they included or did not include meta-analysis, had to be published in a peer-reviewed publication. Cohort studies, case-control studies, and cross-sectional studies were eliminated because they used a non-systematic review process. Language imposed limitations on study selection. Studies that were not published in English were eliminated.
For this review, we looked at all systematic reviews and meta-analyses that looked at the impact of various physical therapy modalities on the Down Syndrome population.
Postural control dysfunctions, as well as issues in motor coordination, sensorimotor integration disorders, and the fact that patients take time to adapt to new situations, are all frequently reported in children with DS. Table 1 shows how each included systematic review described participants' characteristics. The homogeneity of the population among primarily included studies was assessed using inclusion and exclusion criteria.
| Criteria for Inclusion | (1) child participants (18 years or younger) diagnosed with DS (trisomy 21 (nondisjunction), translocation, or mosaicism), and results specific to participants with DS were presented separately if other diagnoses were included, and (2) a diagnosis consistent with having a motor impairment was presented separately if other diagnoses were included |
|---|---|
| Criteria for Exclusion | Subject with a history of (1) Previous surgeries to musculoskeletal structures involving bone, ligaments, and/or nerves, (2) musculoskeletal fracture in either lower limb requiring realignment. |
| 1 | ((MH ‘‘Review’’) OR (MH ‘‘Meta-Analysis’’) OR (MH ‘‘Meta-Analysis as Topic’’) OR systematic review OR meta-analysis OR meta-analysis) |
|---|---|
| 2 | ((MH ‘‘Down) OR (MH ‘‘Down syndrome’’) OR (MH ‘‘Trisomy 21’’) OR Genetic Disorder * OR Developmental delay) |
| 3 | ((MH ‘’Postural control ’’) OR (MH ‘‘Strength ’’) OR (MH ‘‘Endurance’’) |
| 4 | ((MH ‘’Physical modalities ’’) OR (MH ‘‘therapy approaches’’) OR (MH ‘‘physical activities’’) |
| 5 | 1 AND 2 AND 3 AND 4 |
The primary investigator (SR) did a complete and systematic search of Google Scholar, MEDLINE, PubMed, Scopus, and PEDro to June 2021, which was revised by the second author (FA). The search phrases included database-specific subject categories as well as free-text terms such as systematic review, meta-analysis, Down syndrome, and physical approaches. Table 2 shows the PubMed, Scopus, PEDro, and Google scholar search strategies.
All publications found through database searching were individually examined by two reviewers (SR and FA). The titles of the articles that were returned were evaluated for study eligibility. The same criteria were used to evaluate abstracts that were recognized as potentially relevant based on the title. After that, full texts were evaluated to see if they could be used in the review and meta-analysis. Additional relevant systematic reviews were found by hand-searching reference lists. Disagreements between the two reviewers were handled through dialogue to reach an agreement.
Each systematic review was data-extracted independently by two reviewers (SR and FA). When missing data was discovered, the associated author was contacted and data was sought. The aim, population age, R-AMSTAR total score (Table 3) [12], and rank, No. of studies/No. of children, Outcome variable, Standardized measurements Interventions, and Meta-analysis summary were among the data retrieved. In terms of participant selection/inclusion and procedures, the homogeneity of the included studies was assessed. Authors, year of publication, outcome variables and measurement method, participant inclusion criteria, number of included participants, and so on were retrieved from the individual main studies of each systematic review to be included in the meta-analysis.
| Items | Score |
|---|---|
| 1. Was an ‘a prior design provided? | 4 |
| 2. Was there duplicate study selection and data extraction? | 4 |
| 3. Was a comprehensive literature search performed? | 4 |
| 4. Was the status of publication (i.e. grey literature) used as an inclusion criterion? | 4 |
| 5. Was a list of studies (included and excluded) provided? | 4 |
| 6. Were the characteristics of the included studies provided? | 4 |
| 7. Was the scientific quality of the included studies assessed and documented? | 4 |
| 8. Was the scientific quality of the included studies used appropriately in formulating conclusions? | 1 |
| 9. Were the methods used to combine the findings of studies appropriate? | 4 |
| 10. Was the likelihood of publication bias assessed? | 4 |
| 11. Was the conflict of interest included? | 3 |
| Total score | 40 |
| R-AMSTAR Revised—A Measurement Tool to Assess systematic Reviews | |
Assessment Independent critical appraisal and data extraction were completed by two reviewers (SR and FA). Disagreement was resolved by discussion to reach a consensus. Systematic review quality and potential bias were assessed using the modified R-AMSTAR tool (Revised—A Measurement Tool to Assess Systematic Reviews) [13, 14]. A detailed version of the modified R-AMSTAR is available online: (https://static-content.springer.com/esm/art%3A10. and attached as an additional file of the protocol paper [52]. A summary of the R-AMSTAR tool is shown in Table 3. Using the modified R-AMSTAR, studies were given a score out of 40. A higher score indicated higher methodological quality, greater internal validity, and a lower risk of bias. The methodological quality of systematic reviews was ranked from highest to lowest based on the total score and percentile rank (A 90–100%, B 80–89%, C 70–79%, D 60–69%, E > 60%) [15]. Impacts of bias and methodological flaws on the internal validity of the review were considered in the synthesis of review findings. Reviews were not excluded based on quality.
The percent agreement between reviewers regarding eligibility screening and methodological quality of systematic reviews was calculated using kappa scores of agreement. To avoid confounding in the inclusion of the same individual primary studies by multiple systematic reviews, averages and standard deviations of individual studies were extracted instead of the total mean difference calculated by the reviews. Duplicate studies were then removed. Heterogeneity was assessed using the chi-square (I2) calculation and interpreted as 0–40% representing unimportant heterogeneity, 41–60% moderate heterogeneity, 61–90% substantial heterogeneity, city, and 91–100% considerable heterogeneity [47]. Sub-group analyses were performed concerning the International Classification of Functioning, Disability, and Health (ICF) model by the World Health Organization (WHO) [10, 11], the method used to measure outcomes and organized by the methodological quality of the systematic review. Meta-analyses were conducted in Rev- Manager version 5.0 (Copenhagen, Denmark: the Nordic Cochrane Centre, the Cochrane Collaboration, 2008). Standard mean difference (SMD) and 95% confidence intervals (CIs) were calculated. An SMD of 0.3, 0.5, and 0.8 indicated a weak, moderate, e, and strong effect size, respectively. Given the significant heterogeneity within the data, prediction intervals (PIs) were calculated to estimate the uncertainty around the effect estimate.
Database searching returned 425 potential systematic reviews. The numbers of systematic reviews screened, assessed for eligibility, and included in the review after taking into account The PRISMA extension statement for reporting of systematic reviews [37], are shown in Fig. 1. A total of 12 systematic reviews involving 117 studies included publications from 2007 to 2020. Inter-rater kappa scores of agreement were high for both screening of abstracts (k = 0.947, SE = 0.52) and full-text articles (k = 0.957, SE = 0.42). The percent agreement for admissibility of systematic reviews during full-text critical appraisal was 91% (10/11). The consensus during the inclusion of reviews was reached through discussion between the two primary reviewers (SR and FA) and did not require further deliberation from a third, independent reviewer. Fig. 1 Search strategy results.
Six systematic reviews examined muscular fitness (strength and endurance), six systematic reviews examined dynamic and/or static balance [16, 18, 21, 23, 24, 25, 26, 27], four systematic reviews examined cardiovascular fitness[16, 24, 25, 26], and three systematic reviews examined anthropometric measurements [16, 22, 26], and three systematic reviews examined locomotor and functional performance [17, 18, 20]. Tables 4 and 5 show the outcomes of each review, as well as the number of papers included, the meta-analysis summary, and the adjusted R-AMSTAR ratings. The following were the reasons for the exclusion of nine carefully examined reviews. three studies focused solely on children with cerebral palsy, two were not systematic reviews, two did not look at motor functions affecting performance, and two did not have full-text versions accessible.
| Systematic review | Modified R- AMSTAR items | |||||||||||||
|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|
| 1 | 2 | 3 | 4 | 5 | 6 | 7 | 8 | 9 | 10 | 11 | total | % | Rank | |
| Li et al, 2013 | 3 | 4 | 4 | 2 | 3 | 4 | 4 | 0 | 4 | 1 | 3 | 32 | 80% | B |
| Khondowe et al, 2007 | 3 | 4 | 4 | 3 | 2 | 4 | 2 | 1 | 4 | 1 | 2 | 30 | 75% | C |
| Sugimoto et al, 2016 | 3 | 1 | 3 | 2 | 1 | 4 | 4 | 1 | 4 | 1 | 3 | 27 | 67.5% | D |
| Damiano and Dejong 2009 | 3 | 4 | 4 | 4 | 3 | 4 | 4 | 0 | 1 | 2 | 2 | 31 | 77.5% | C |
| Gudiol et al, 2017 | 4 | 4 | 4 | 4 | 3 | 4 | 4 | 0 | 4 | 3 | 2 | 36 | 90% | A |
| Koopman, 2020 | 4 | 1 | 3 | 1 | 1 | 4 | 3 | 0 | 1 | 1 | 1 | 20 | 50% | E |
| Espinosa et al., 2020 | 4 | 2 | 3 | 4 | 3 | 4 | 4 | 0 | 3 | 1 | 2 | 30 | 75% | C |
| Zago et al., 2020 | 3 | 4 | 3 | 2 | 3 | 4 | 4 | 0 | 1 | 1 | 2 | 25 | 62.5% | D |
| Hardee and Fetters, 2017 | 4 | 4 | 4 | 3 | 3 | 4 | 4 | 1 | 1 | 1 | 3 | 32 | 80% | B |
| Gonzalez et al., 2019 | 4 | 4 | 4 | 4 | 3 | 4 | 3 | 1 | 4 | 3 | 3 | 37 | 92.5% | A |
| Paul et al.,2019 | 3 | 3 | 4 | 1 | 4 | 3 | 3 | 1 | 2 | 1 | 4 | 29 | 72.5% | C |
| Maiano et al., 2019 | 4 | 3 | 4 | 3 | 4 | 4 | 3 | 1 | 1 | 1 | 3 | 31 | 77.5% | C |
| Average | 3.5 | 3.2 | 3.6 | 2.7 | 2.7 | 3.9 | 3.5 | 0.5 | 2.5 | 1.4 | 2.5 | 30 | 75% | C |
| Systematic review | Aim | ICF level | Range of age (years) | Modified R-AMSTAR Total / Rank | No. of studies/No. of children | Outcome variable | Standardized measures | Interventions | Meta-analysis summary |
|---|---|---|---|---|---|---|---|---|---|
| 1. Lie et al.,2013[16] | To assess the benefits of physical fitness in D.S children | Body structure and function | 3–18 | 32 / B | 10/349 | Muscular fitness(strength &endurance) -balance, cardiovascular fitness (the peak vo2) and -body composition | -Isokinetic strength -Standing balance test -Treadmill test and rowing ergometer test -BMI and body fat | a treadmill program, a bicycle program, a rowing ergometer intervention, a progressive resistance training game-like balance exercise, cardiovascular and strength exercise, and a weight-bearing exercise program | No meta-analysis performed. |
| 2. Khondowe et al, 2007 [17] | To evaluate the effect of physical activity on motor development in children with D.S | Activity & participation | > 10 | 30/ C | 4/ 146 | Independent walking Locomotor skills (developmental quotient) | -Bayley scale of infant development -10 step forward walking and 10 step side walking | sensory integrative therapy ; vestibular stimulation, Neurodevelopmental therapy | Meta-analysis performed on 2 studies showed a significant effect for both measures. |
| 3. Sugimoto et al,2016 [18] | To examine the effects of neuromuscular training on general strength, maximal strength, and functional mobility tasks in children with D.S | Body structure and function & activity | > 20 | 27/ D | 7 /309 | -general strength studies) -Maximal strength-Functional mobility performance | -isometric, isokinetic strength -One repetition maximum(1 RM) of leg and chest press -grocery shelving and timed up- and- downstairs | weight machine exercises(resistance training), a 5-min treadmill exercise and a 20-min virtual-reality based activity, a whole-body vibration training | Meta-analysis performed on 5 studies showed a significant effect for general and maximum strength & non-significant for functional performance. |
| 4. Damiano and Dejong, 2009 [19] | to investigate the evidence supporting treadmill training with and without body weight support across broader diagnostic categories within pediatric neurorehabilitation. | Body structure and function | 13–15 | 31/ C | 6 out 29/ 186 | -Trunk & leg activity duration -cadence, velocity, step length, width, dynamic base, double support percentage, foot rotation & symmetry - the onset of independent walking | -Acti watch activity monitors for 24 hrs -Milestone achievement scale | treadmill training | Meta-analysis not performed |
| 5. Gudiol et al., 2017 [20] | To assess the effectiveness of treadmill interventions on locomotor development in children with delayed ambulation or in pre-ambulatory children | Activity & participation | > 6 | 36/ A | 7/ 175 | The onset of independent walking and locomotor skills | -GMFM, BSITD; PDMS – 2 -motion analysis systems | treadmill training | Meta-analysis on 2 studies showed no significant effect on independent walking and significant on locomotor skills. |
| 6.Koopman, 2020 [21] | To identify which interventions have been used between 2010 and 2020 for improving balance in individuals with Down Syndrome | Body structure and function | +_ 18 | 20/ E | 6/ 147 | Static & dynamic balance and LL strength | BOTMP, KTK, PBS & Handheld dynamometer for lower extremity strength testing | BOSU Ball training exercises, Six-week strength and balance training program, Dance based training program, Whole body vibration training, Virtual reality therapy, Wiihabilitation | Meta-analysis not performed |
| 7. Espinosa et al., 2020 [22] | to analyze the relationship between dietary intervention, physical exercise, and body composition, in DS with overweight and obesity | Body structure and function | > 19 | 30/ C | 6/ 163 | anthropometric measures (BMI & body fat) | Bod Pod® plethysmography equipment& a Holtain lipometer | a physical exercise program with different aerobic and resistance activities | Meta-analysis not performed |
| 8. Zago et al., 2020 [23] | To describe (1) the current knowledge on gait and postural control in individuals with DS, and (2) relevant rehabilitation strategies | Body structure and function & activity | > 18 | 25/D | 9 out of 37/ 324 out of 1299 | Postural control & spatiotemporal parameters | an unstable foam rubber mat over a pressure plate | treadmill training | Meta-analysis not performed |
| 9. Hardee and Fetters, 2017 [24] | To evaluate the effectiveness of exercise intervention on daily life activities and social participation in individuals with DS | Body structure and function & activity & participation | > 18 | 32/ B | 11 out of 19/ 289 out of 525 | Balance Cardiovascular , muscle strength and endurance | -BOTMP - maximal oxygen consumption VO2, heart rate (HR),respiratory rate (RR), height/weight/skinfolds handheld manual muscle tester& 10 repetition maximum (RM)1 RM seated chest press), (1 RM seated leg press) | progressive resistance training, aerobic and strength training, bike riding, dance | Meta-analysis not performed |
| 10. Gonzalez et al., 2019 [25] | To evaluate the effectiveness of PT on the physical outcomes (such as vestibular, cardiovascular and respiratory, weight maintenance and movement-related functions, motor skills, carrying out tasks, mobility and walking indexes) in people with DS. | Body structure and function & activity & participation | > 18 | 37/ A | 21out of 27/ 689 out of 842 | muscle strength balance Cardiovascular function Motor development - | - Dynamometer,-1RM repetition maximum tests. -Bruininks Oseretsky Test of Motor Proficiency, Balance platform, Biodex balance system. -Treadmill exercise test with gas consumption control and electrocardiogram. -Gross Motor Function Measure, Bayley Scales of Infant Development, Peabody Developmental Motor Scales. | treadmill training, Progressive resistance training, Full-body vibration, Neurodevelopment therapy and massage therapy, Supramalleolar orthosis | Meta-analysis of 4 studies on muscle strength & 2 studies on balance showed a significant effect. In Cardiovascular function, the meta-analysis of 2studies shows inconclusive results. |
| 11. Paul et al.,2019 [26] | To discusses the benefits of exercise therapy on body composition, aerobic capacity, muscle strength, proprioception and cardiometabolic profiles of PWDS. | Body structure and function | a mean age of 18.1 ± 6.8 years | 29/C | 19/1331 | Anthropometric measurements, muscle strength and endurance of DS | -BMI, WC, BW -Bruininks Oseretsky Test of Motor Proficiency, - accelerometer, a 6 -min walk test | Regular aerobic exercises, Regular muscle strengthening exercises | Meta-analysis not performed |
| 12. Maiano et al., 2019 [27] | To summarize the findings from studies examining the effects of exercise interventions designed to improve balance in youths with Down syndrome | Body structure and function | 5–22 | 31/C | 11/281 | Static balance Dynamic balance | -BOTMP (balance subtest), Pressure platform. -Biodex Balance System, Heel-to-Toe Dynamic Balance Test. | Strength and balance exercises, Whole body vibration, and Wii Fit balance game training | Meta-analysis not performed |
Diverse outcome measures were used corresponding to the different kinds of intervention programs. The majority of the studies assessed muscular fitness and balance, four studies evaluated cardiovascular fitness; three studies for body composition, and three studies tested locomotor skills which terms classified according to ICF (detailed information is presented in Table 5).
Six systematic reviews looked into the effects of an intervention program on muscle strength and endurance, with different training methods used in each study.
In the study conducted by Li et al. (2013), a treadmill program, a bicycle program, a rowing ergometer intervention, and progressive resistance training were employed; the meta-analytic data demonstrated that exercise programs can improve muscular strength. The conflicting results among research may be due to the effects of different exercise types [16].
In Sugimoto et al. (2016); weight machine exercises(resistance training), A 5-minute treadmill activity were employed as weight machine exercises (resistance training). Their findings suggested that neuromuscular training could be an effective strategy for improving overall and maximal muscle strength in Down syndrome children and young adults [18].
In Hardee and Fetters, (2017); A favorable impact of an exercise intervention on daily living activities and involvement for people with DS was demonstrated using progressive resistance training [24].
Treadmill training with progressive resistance training was employed by Gonzalez et al ( 2019); the meta-analytic data on strength levels emphasize the importance of resistance training programs in improving muscle strength in patients with DS [25].
Regular muscular strengthening exercises, such as circuit training, plyometric, and swimming, as well as regular resistance training, were used by Paul et al (2019). Regular exercise improves the health condition of PWDS by improving their body composition, aerobic capacity, and muscle strength, according to clinical research [26].
In Koopman et al., (2020), a Six Week Strength and Balance Training Program was used to examine lower limb strength. Resistance exercises with sandbags for hip and knee flexors, hip and knee extensors, hip abductors, and ankle plantar flexors made up the strength training element of the intervention program. Subjects did two sets of ten repetitions at 50% of their one-repetition maximum (1RM), and resistance was increased only after the participants could complete the sets with ease; the results showed that after a six-week exercise program, the participants' one repetition maximum (1RM) had decreased by 10%. The intervention group was stronger than the control group in all muscle groups whose strength was measured. These results were statistically significant (p < 0.05) [21].
Six systematic evaluations looked at how different exercise regimens affected the result of balance interventions. A treadmill program, particular balance exercise training, and a weight-bearing exercise program were used by Lie et al (2013). Exercise innovations increased balance, according to meta-analytic data [d = 1.10; 95 percent CI (0.55, 1.63); Q2 = 1.10; P = 0.58; I 2 = 0; fail-safe n = 14 [16].
Aerobic and strength training, bike riding, and dance were used by Hardee and Fetters(2017). Studies with statistically significant body structure and function measures were shown to have lesser validity than studies with Activity and Participation measures [24].
Gonzalez et al. (2019) employed full-body vibration. The intervention had a beneficial effect on mediolateral oscillations of the center of gravity controlling body posture, according to the meta-analysis [25].
Strength and balance workouts, whole-body vibration, and Wii Fit balance game training were used by Maiano et al, (2019). The findings revealed that the exercise programs studied are more effective than control conditions in improving static and static-dynamic balance in children with Down syndrome [27].
BOSU Ball training activities, a Dance-based training program, Whole body vibration training, Virtual reality therapy, and Wii rehabilitation were employed by Koopman et al (2020). Children with Down Syndrome achieved much-improved balance in both static and dynamic balance tasks when using the BOSU ball. Although the results of the dance program did not show any significant variations in the static balance of people with Down syndrome, the findings suggest that sensory integration training could help these people with their balance and coordination [21].
The results of the whole-body vibration training study reveal that it is only useful in people who have a problem with their balance. The virtual reality program's results imply that virtual reality therapy is an effective tool for improving balance and motor coordination in people with Down syndrome and that it might be suggested as a clinical treatment for this population. The results of the Wii-Fit Virtual Reality, or Wii-habilitation, study demonstrate that VR-based therapy in the form of Wii-habilitation, when used in conjunction with regular physical therapy, can help people with Down syndrome improve their balance [21].
Treadmill training was employed by Zago et al (2020). After a 6-month treadmill training program, improvements in dynamic balance function were reported in the elderly with DS [23].
As a result of four systematic reviews, cardiovascular fitness was assessed and the peak VO2 was measured. A treadmill test and a rowing ergometer test were employed by Lie et al., (2013). Through rowing ergometer training, there was no group difference in peak VO2, as determined by a treadmill test [d = 0.37; 95 percent CI (– 0.62, 1.36)] and a rowing ergometer test [d = 0.30; 95 percent CI (– 0.68, 1.29). Exercise therapies enhanced cardiovascular fitness [d = 0.60; 95 percent CI (0.07, 1.14); Q1 = 1.74; P = 0.19; I 2 = 42.41; fail-safe n = not applicable], according to the meta-analysis [d = 0.60; 95 percent CI (0.07, 1.14); Q1 = 1.74; P = 0.19; I 2 = 42.41; fail-safe n = not applicable [16].
Aerobic and weight training, bike riding, and dance were used to quantify peak aerobic capacity (maximal oxygen consumption, heart rate) in Hardee and Fetters(2017) [24].
The results of the meta-analysis revealed that the data presented by the studies were equivocal, according to Gonzalez et al.,(2019). VO2 max examined the maximal heart rate with an intervention based on aerobic exercise [25].
The study by Paul et al.,(2019) showed that regular aerobic exercise boosted the aerobic capacity of PWDS, which was the main contributor to maximal oxygen consumption, and thus needs further validation through more randomized controlled trials [26].
The effects of physical fitness training on body composition were studied in three systematic reviews. In Lie et al.,2013; cardiovascular, bicycle, and strength exercise training had no statistically significant impact on body weight [d = 0.05; 95 percent CI (– 0.50, 0.60)], BMI [d = 0.04; 95 percent CI (– 0.51, 0.59)], or skinfold score [d = – 0.37; 95 percent CI (– 0.93, 0.18)], but the combined exercise training programme (i.e. jumps, press ups, elastic bands) had statistical significant difference [16].
Espinosa et al., 2020; used a physical training program with diverse aerobic and resistance activities to measure anthropometric measures (BMI and body fat) utilizing Bod Pod® plethysmography equipment and a Holtain lipometer. The procedures that caused the largest variation in body composition in children and adolescents were those based on planned physical activity, considering intensity, duration, number of repetitions, days per week, and programming by macrocycles, according to this review [22].
Paul et al.,2019; Clinical research suggests that regular exercise improves the health status of PWDS by improving their body composition, aerobic capacity, muscle strength, proprioception, and postural stability. The benefits of improved aerobic work capacity and body composition contribute to a decreased cardiometabolic risk profile in people with Parkinson's disease [26].
The impact of physical therapy training techniques on locomotor skills was studied in three systematic reviews.
Independent walking and locomotor skills (developmental quotient) were measured using the Bayley scale of baby development and 10-step forward and 10-step side walking (Khondowe et al, 2007). The therapies used were sensory integrated treatment, vestibular stimulation, and neurodevelopmental therapy. The time it takes for Down Syndrome youngsters to walk independently is greatly reduced when they get intensive physical care. Furthermore, intense physical therapy considerably improves the total and locomotor development quotients [17].
The effect of neuromuscular training on functional mobility tasks was investigated by Sugimoto et al. in 2016. (grocery shelving and timed up-and-down stairs). The intervention had a tiny, statistically non-significant effect on grocery shelving and timed up-and-down stairs, according to the meta-analysis results (SE: 0.11, 95 percent CIs: 0.47, 0.69, p = 0.71) [18].
Gudiol et al., 2017; GMFM, BSITD, PDMS – 2, and motion analysis systems were used to examine the start of independent walking and locomotor skills using treadmill intervention. The meta-analysis found no difference between the two intervention groups in the age of onset of independent walking (MD 0.10, 95 percent CI-5.96 to 6.16). Other gait measures such as step width (MD -0.58, 95 percent CI -2.11 to 0.95) and step length (MD2.68, 95 percent CI-0.99 to 6.35) did not differ between the high-intensity and low-intensity treadmill intervention groups at the 12-month follow-up assessment. Gait ankle dorsiflexion (MD -2.80, 95 percent CI -5.96 to 0.36); and toe-off (MD -0.90, 95 percent CI-5.49 to 3.69) [20].
R-AMSTAR rank did not differ between meta-analyses reporting significant and non-significant findings. Thus, the methodological quality of each systematic review was not a strong predictor of overall effect or significance. A meta-analysis of included primary studies from systematic reviews of the same methodological quality was performed, if outcomes were comparable. Findings not suitable for meta-analyses were summarized qualitatively [13, 14]. Meta-analyses were conducted in Rev-Manager version 5.0 (Copenhagen, Denmark: the Nordic Cochrane Centre, the Cochrane Collaboration, 2008). Standard mean difference (SMD) and 95% confidence intervals (CIs) were calculated. A summary of total effect sizes for four primary included studies, in addition to respective 95% CIs and PIs, is provided for locomotor developmental skills outcome in Fig. 2.
This study reviewed the effects of physical therapy modalities on motor proficiency among children with DS. Twelve studies using varying interventions (i.e. progressive resistance training, aerobic training, balance training, and training that combined the elements above fulfilled the inclusion criteria and were reviewed to examine their effects on four major components of motor fitness outcomes. These fitness outcomes were, (a) muscle strength and endurance, (b) balance, (c)cardiovascular fitness, (d) body composition, and (e) locomotor skills. Specifically, the six systematic reviews showing significant impacts (d = 0.50–1.30) of exercise training on muscle strength used a combination of cardiovascular and strength exercises, a combination of resistance training and balance exercises, a combination of treadmill walking, and game-like exercises (virtual reality therapy) [16, 18, 21, 24, 25, 26].
The methodological quality of systematic reviews was ranked using the modified R-AMSTAR tool to assess the internal validity of included reviews showed that there were two studies [20, 25] ranked with higher scores (A), two studies ranked with (B) score [16, 24], five studies ranked as (C) score [17, 19, 22, 26, 27], two studies ranked as (D) score [18, 23], and one ranked as (E) score [21]. Overall results of methodological quality of included systematic reviews indicated moderate evidence [12].
The ICF model can be used to give a framework for systematically comparing research outcomes across investigations [9, 10]. As a result, the ICF model assessed body structure and function in nine reviews and tested activity and participation in five reviews.
Other findings found an improvement in upper-limb muscular endurance but no positive effects on the endurance of lower-limb muscle. The poor exercise economy observed in individuals with DS (e.g. disturbed gait kinetics and kinematics) may be attributed to the non- effectiveness of the intervention program for lower limb muscular endurance. In children and young people with Down syndrome, neuromuscular training may be a useful strategy for improving overall and maximum muscle strength and engaging in greater physical adaptation as their motor proficiency improves, leading to a more physically active lifestyle [28, 29, 30, 44].
Resistance training interventions are useful in improving the strength of the upper and lower limbs; Furthermore, vibration therapy interventions have a good effect on balance, particularly in the decrease of the mediolateral center of gravity displacements during independent walking [33, 34, 35, 42].
These data imply that physical therapy is beneficial for improving strength and balance. The meta-analytic findings showed that exercise programs can improve muscular strength. The inconsistent findings across individual studies could be because of the effects of different modes of exercise [36, 37, 40, 41].
Cardiovascular exercises are essential for maintaining optimal cardiovascular health [31, 32]. The effects of aerobic exercise on peak VO2 uptake in individuals with DS were inconsistent. The duration of aerobic training sessions from each study may contribute to this inconsistent finding. the finding from these study suggests that longer training duration should be used to improve aerobic fitness in individuals with DS [43, 34, 38, 39]. Aerobic workouts were largely chosen to alter the cardiometabolic profile of PWDS, according to this review.
Body composition as an outcome of interventions was reported. Although individuals with DS often have low to mild obesity, only three out of the 12 studies included assessed body composition, Thus, the frequency and duration of physical exercise intervention should be increased to improve their body composition compared with the general population [45]. Increased body composition diversity will assist to lessen the negative impacts of obesity and overweight, resulting in lower healthcare expenses [46].
As a result of a meta-analysis done for four involved studies for locomotor developmental skills [48, 49, 50, 51], there was no -significant effect of treadmill interventions on independent walking in these children Fig. 2. In pediatric rehabilitation, the state of the evidence for bodyweight-assisted treadmill exercise differs by demographic [53]. Treadmill intervention may be associated with an earlier onset of independent and supported walking in children with Down syndrome, according to the findings of this analysis (both primary outcomes; a high-intensity customized treadmill intervention and a low-intensity universal treadmill intervention) had the same effect on the initiation of independent walking[20].
Physical therapy modalities for individuals with DS had evidence on improving motor skills that get a great benefit from activities such as weight-bearing exercises, progressive resistance training, neurodevelopmental therapy, and treadmill training, that improved muscle strength outcome also interventions such as whole-body vibration and virtual reality therapy that improved balance measure providing moderate evidence, according to the findings of the current study; In contrast, other outcomes (such as muscle endurance, cardiovascular fitness, and body composition) were less conclusive or had minimal positive evidence. To reinforce the outcomes and promote motor development in children with Down Syndrome, experts recommend that physical exercise programs be intensive and that parents be included. This would allow for a more comprehensive study of the evidence on the influence of exercise intervention on daily living activities and social participation in people with DS, as well as broader inter-disciplinary application
The present study was limited by the exclusion of non-English journal articles, which may preclude potential studies fulfilling the inclusion criteria. In addition, there was a wide range of variability in outcome measures, specifically balance measurements. Also, participants’ ages in the reviewed studies were diverse, ranging from under 10 years to eighteens. Finally, other issues must be more thoroughly investigated such as the safety of using a treadmill over longer periods for those who are at risk for joint deformity in the short term such as fracture and hip dislocation, or at risk for osteoarthritis in young adulthood, must be more systematically and carefully evaluated.
Intervention programs are more successful than control circumstances in improving motor proficiency in children with Down syndrome, according to findings. However, results on cardiovascular fitness and body composition are contradictory affecting quality of life and social participation. Finally, our findings show that more specific interventions and reporting standards are needed to aid professionals in the selection, development, and implementation of preprograms to improve the physical health of Down syndrome children. The findings of this study suggest an evidence-based paradigm for clinical therapists to use when working with DS patients.
Conflict of interest: All authors have no conflict of interest to disclose.
Funding source: No honorarium, grant, or other form of payment was given to anyone to produce this manuscript.
Contributor’s statement
Shimaa Reffat: Designed the study, collected, analyzed the data, contributed to writing the initial draft*and revised the manuscript draft. Faten Abd Elaziem: Conceptualized the study, delivered directional guidance, and contributed to the writing and critical review of the manuscript. All of the named authors approved the final manuscript as submitted, and agree to be accountable for all aspects of the work.