Investigation of Difference Between the Three Representatives of the Functional Status According to Executive Functions and Participation in Children With Hemiplegic Cerebral Palsy

doi:10.21203/rs.3.rs-475651/v1

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Investigation of Difference Between the Three Representatives of the Functional Status According to Executive Functions and Participation in Children With Hemiplegic Cerebral Palsy

https://doi.org/10.21203/rs.3.rs-475651/v1

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Aim: This study aimed to explore whether the three functional status representatives differentiate according to actual performance in everyday life.

Method: High to moderate functional motor and communication performance levels as described by Manual Ability Classification System (MACS), Gross Motor Function Classification System, and Communication Function Classification System (CFCS) were investigated in the context of the scaled scores of executive function tasks and participation in different life situations on ABILHAND-Kids and Child and Adolescent Scale of Participation (CASP) questionnaires. The data were collected from 98 children with hemiplegic cerebral palsy (CP) mainstreamed in regular schools (mean age 9,3 years [SD 3.0 years], [%95 CI:8.7-9.9]; 42 females, 56 males); 28,6% classified to level I, 29,6 % to level II, and 41,8 % to level III in MACS.

Results: The study findings demonstrated that scaled scores of the executive function tasks and participation in different life situations increased with the greater MACS, GMFCS, and CFCS levels (p<0,5).

Conclusion: Data from both MACS, GMFCS and CFCS might be useful to predict a child’s actual performance in daily life.

Pediatrics

cerebral palsy

hemiplegia

upper limb

functional classification systems

MACS

Assessment of the children with cerebral palsy (CP) has clinical importance in identifying the existing problems associated with the CP. Moreover, in either research or clinical settings, it is also beneficial to quick yet robust document the clinical characteristics of the disabled children[1]. Following that, classification systems were developed as practical tools to concisely provide knowledge to researchers or clinicians as a snapshot of a condition[2]. Consequently, classification systems might help both health professionals improve their communications with families/caregivers in clinical environments, and researchers constitute homogenous groups in research settings[3].

The Manual Ability Classification System (MACS), Gross Motor Function Classification System (GMFCS) and Communication Function Classification System (CFCS) are the most prevalent functional classification systems to report the functional motor and communication skills of children with CP into five-level, where level I represents the best and level V represents the worst performance in daily life[4; 5]. The Manual Ability Classification System was established to classify hand functions of children with CP based on the perceived hand use in daily living or structured settings[4; 6]. Although the MACS is commonly used to specify functional hand use both in clinical and in research settings [4; 7], there is still uncertainty related to the role of the affected hand when manipulating an object, e.g., uncertainty in the extent of involvement of the affected side while executing a meaningful activity. The Gross Motor Function Classification System was designed to discriminate the disabled children concerning gross motor functions[8]. Hence, using GMFCS and MACS together provides a broad range of information about functional motor profile, including gross and fine motor function, at a particular time point in children with CP[1], as well as enhances communication between families and health professionals. The CFCS instrument aims to grade communication performance concerning communication effectiveness with familiar or unfamiliar partners in daily life [9].

Consequently, the GMFCS, MACS, and CFCS are the evidence-based classification systems to describe both functional motor and communication levels of children with CP by sketching their functional motor and communication abilities in daily life[10]. A previous study has reported that these three classification systems complement each other to depict the child’s capacity associated with executive function, mobility, and communication performance [11] onto the World Health Organization's (WHO) international classification of functioning, disability, and health[12]. A potential inter-relationship among GMFCS, MACS, and CFCS was determined to strong or moderate in 222 children with CP aged 2-7 years. Besides, in the same study, it was found out that the GMFCS had a high correlation with MACS, however, less with CFCS [13]. Similarly, a relatively more recent study by Compagnone [11] reported a strong correlation between these three functional classification systems.

One aspect of the validation of the classification systems is their correspondence to the external references by using outcome measures [6]. Therefore, it is crucial to explore what means each of the classification system levels in the context of real-life utilizing the most appropriate outcome measures. In other words, validating the classification systems within the functional performance in daily life and understanding each level's potential difference in terms of an external reference is essential for clinicians, health professionals and caregivers/families. Following that, some studies have investigated a potential inter-relationship between these three prevailing functional classification systems in perspective of executive function tasks, demonstrating a meaningful correlation among classification systems in question and functional performance in daily living [3; 6; 14]. However, while their unique resultants, these studies included participants of different typologies of CP at all MACS and GMFCS levels, which strengthens better understanding of the differences or similarities between high to moderate levels (I-III) of classification systems. Furthermore, it would be crucial to differentiate between high to moderate levels of classification systems in the context of executive function and participation to enhance communication between families and health professionals in the clinic settings and as well as to construct homogenous groups in the research settings. An actual discrimination between classification systems’ levels based on the actual performance in basic daily living activities and involvement in different life situation is required to practically delineate the functional status of disabled children.

Based upon the premises mentioned above, the purpose of this study was to further investigate (1) actual differences between high to moderate levels of the prevailing functional classification systems in respect of executional functional tasks and participation in different life situations and (2) an inter-relationship between MACS, GMFCS, and CFCS. We hypothesized that there would be a potential difference between the high to moderate levels of the classification systems based on functional performance in real world.

2.1. Participant

Participants in this cross-sectional study were the children with hemiplegic CP between the ages of 7-14 years and were at the MACS, CFCS, and GMFCS levels I-III. Human Research Ethics Committee at the XXXX University approved the study protocols numbered 79236777-050.01.04. A convenience sample size of the 98 children diagnosed with congenital hemiplegia (56 males, 46 females) born in XXXX city was set from different rehabilitation centers. Informed consent was obtained and signed by the parents/guardian of the children. Participants’ functional motor and communication levels were evaluated by an experienced rehabilitation therapist who is expert in this field. Children with lower levels (IV-V) of functional motor and communication skills or diagnosed with acquired brain injury were excluded from the study.

2.3. Classification Tools of Functional Motor and Communications Performance

The participants' functional status in the study was documented using the GMFCS (interclass correlation coefficient [ICC]= 0,75)[15], MACS (ICC=0,7 to 0,9)[6], and CFCS (ICC= 0.71 to 0.89)[9] based on both clinical observation and the reporting of child's physiotherapist or families. These three instruments are universally accepted to describe the functional performance as a snapshot of functional motor and communication skills[1; 11] on a five-level scale from the score I to score V (lower level indicates the best capacity). Although some studies have attempted to explore the possible correlation between these instruments [9; 10; 14], the distinction among the classification systems’ levels, especially between high levels based on the activity and participation in natural settings, remains to be determined.

2.4.Outcome Measures of Executive Function and Participation

The ABILHAND-Kids (test retest reliability ICC=0,92) [16; 17] was developed as a Rasch-based measure to report perceived manual ability of children with CP aged 6 to 15 years by their caregiver/parents [18]. The ABILHAND-Kids questionnaire consists of 21 items that are the best representative of specific daily activities requiring one or both hand use [16]. The potential advantage of this questionnaire is its usefulness in clinical and research settings, as it can be quickly completed[18]. The total score can be calculated based on converting the raw score into a logit measure on the website of http://rssandbox.iescagilly.be[19] . Finally, ABILHAND-Kids has been validated in evaluating executive functions from the perspective of the manual ability of children with hemiplegic CP in clinical settings[16].

The Child and Adolescent Scale of Participation (CASP) (ICC=0.94)[20] was used to documenting the extent of child's involvement in different life situations, including participation in home, school, and community activities[21]. The CASP includes 20 ordinal-scaled items as to home participation (6 items), 2) community participation (4 items), 3) school participation (5 items), and 4) home and community living activities (5 items). Its assessment items are suitable for school-aged children (5 years or older) and coincide with many activities in different life situations. Psychometric properties of the CASP, such as internal consistency and test-retest reliability, were demonstrated to good [22] .

2.5. Statistical Analysis

Statistical analyses were performed using the SPSS software version 24. The variables were investigated using visual (histogram, probability plots) and analytical methods (Kolmogorov-Smirnov) to determine whether or not they are normally distributed. While investigating the associations between non-normally distributed and/or ordinal variables, the correlation coefficients and their significance were calculated using Spearman Test. Inter-relationship among MACS, GMFCS and CFCS was interpreted according to Spearman’s correlation coefficient strength: r<0.2 very weak relationship; 0.2-0.4 weak relationship; 0.4-0.6 moderate relationship; 0.6-0.8 strong relationship; r>0.8 very strong relationship [23]. Categorical data were presented with percentage or frequency as appropriate, while continuous data were given as mean ± standard deviation. One-way ANOVA and Tukey’s Test were performed to explore possible differences in mean executive functions and participation in different life situations scaled scores in three representatives of functional status on MACS, GMFCS, and CFCS. Standard error bars were used to demonstrate mean scores with %95 confidence interval (CI) on assessment tools by each classification system level. An overall p-value of less than 0.05 was considered to show a statistically significant result.

All study participants' demographic characteristics and prognostic variables, and mean ages of participants classified into different GMFCS, MACS, and CFCS levels were shown in Table I. Children at each of the three levels of the classification systems demonstrated similarity in the age (p>0,05). Among the 98 enrolled children, 57 (% 58,2) were in good functioning levels in MACS (level I-II), whereas 41 (% 41,8) were in moderate functioning level (MACS level III). On average, % 55,1 could walk independently in all environments (GMFCS I), % 38.1 could walk with some limitations (GMFCS II), and only % 6.1 required a hand-held device for mobility. Considering that the MACS is a classification system of disabled children's hand function in five levels (a lower value describes higher manual ability), most of the children in this study had effective hand use (MACS I-II). For communication skills, the predominant levels were level I (% 56.1) and level II (% 29.6) respectively, while fewer participants had a moderate score (level III; %14.3). That is, most of the study participants could independently and effectively communicate with people in most environments. Finally, except for hearing impairment, vision, speech, and cognition impairment was reported in 9 (% 9.2), 11 (%11,2), and 12 (% 12,2) of study participants, respectively.

The distribution of children at different MACS levels within GMFCS levels was demonstrated in Table II. As outlined, most children in MACS level I (89,3%) were qualified with GMFCS I; on the other hand, a very few percent (10,7%; 3 children) presented GMFCS II. Among children in MACS II, 55,2% was at GMFCS I; likewise, 44,8% was at GMFCS II. Finally, more than half children in GMFCS III (53,7%) featured level III of GMFCS.

Table III demonstrates comparison of participants’ executive function tasks (manual ability) and participation in different life situations scaled scores on ABILHAND-Kids and CASP questionnaires according to MACS levels. Analysis of one-way ANOVA showed that the executive function and participation in different life situations outcomes exhibited variability in participants at each of the three MACS levels (p= 0,000). In other words, children in high functioning levels in MACS gained greater both executive functions and participation scaled scores on ABILHAND-Kids and CASP questionnaires than children of moderate functioning level (Post-Hoc: a>b>c). Moreover, Figure 1 and 4 illustrated the range of scaled scores on ABILHAND-Kids and CASP questionnaires with % 95 CI. According to Figure 1, children in MACS I demonstrated greater variability as to manual performance than children in MACS II and III.

Children in level I, II, and III at GMFCS differed greatly for executive function tasks and participation in different life situations outcomes (p=0,000) (Table IV). Furthermore, results of pairwise post-hoc tests suggested that participants in a higher functioning GMFCS level had more significant outcomes in both executive function tasks and participation in different life situations than participants with a moderate functioning level (Post-Hoc: a>b>c). That is, the higher GMFCS levels were found to be associated with greater mean scores on both the ABILHAND-kids and CASP questionnaires. Children in GMFCS I presented greater variability for executive function tasks (ABILHAND-Kids) than those in GMFCS II and III. In contrast, children in GMFCS III demonstrated greater variability in scaled scores on CASP-Community Participation subdomains than those in GMFCS I and II (Figure 2 and 5)

Table V shows executive function tasks and participation in different life situation outcomes by communication skills on CFCS instrument. Results of one-way ANOVA displayed statistical meaningful differences among three levels related to both manual ability and participation in different life situation outcomes (p=0,000). However, isolated comparisons (pairwise post-hoc tests with Tukey) revealed a statistically significant difference only between high and moderate levels (CFCS I-III), whereas it was not found out statistically meaningful differences between level I and II, and between level II and III (Post-Hoc: a>c, a=b, b=c). In other words, only children at level I and level III in CFCS exhibited variability in executive function tasks, and participation scaled scores on ABILHAND-Kids and CASP questionnaires. In contrast, a significant difference could not be found between children classified into CFCS levels closer to each other for actual performance in daily life. Furthermore, as demonstrated in Figure 3 and 6, children qualified with CFCS III displayed greater variability in scaled scores on both executive function tasks and participation in different life situations than those in CFCS I and II.

As demonstrated in Table VI, MACS levels were found to be moderately correlated with GMFCS levels (r=0,491, p=0,000), whereas, weakly correlated with the CFCS levels (r=0.247, p=0,014). Finally, a moderate relationship was observed between GMFCS and CFCS levels (r=0,574, p=000).

This current study demonstrated significant differences among each of three levels of functional motor skills, as measured by the MACS and GMFCS, for executive function tasks and participation in different life situations, as measured by ABILHAND-kids and CASP-subdomains in children with hemiplegic CP. Conversely, for CFCS levels, a statistically meaningful difference was found only between high and moderate levels as to actual performance in daily life. In contrast, no significant difference was introduced between levels closer to each other. In other words, executive function tasks and participation in different life situations increased with greater functional motor and communication levels. Furthermore, this study has demonstrated weak to moderate inter-relationships among high to moderate functional ability representatives, as measured by the first three levels of MACS, GMFCS, and CFCS. Finally, our study demonstrated that most children with hemiplegic CP presented high functional skills on MACS, GMFCS, and CFCS (level I-II).

The gross motor function classification system, manual ability classification system, and communication function classification system objectively describe cerebral palsied children and adolescents’ functional profile through five levels to ensure a practical communication between health professionals and families [10; 24]. In brief, the GMFCS delineates motor disability or current motor function[25]; the MACS describes the manual ability of children with CP to manage daily activities;[26] and the CFCS measures communication performance regardless of the underlying functional or structural impairment [9], i.e., each level of these classification systems is expected to characterize disabled children capacity concerning functional motor and communication skills with the most appropriate level [11; 24]. Although some studies have reported a strong relationship among this classification systems[10; 13; 24], the difference between each functional classification system level was explored only by Öhrvall et al.’s study [3], indicating a strong correlation among functional motor levels (MACS,GMFCS) and self-care and mobility capability. Similarly, our study showed significant differences between three representatives of functional status for executive function tasks and participation in different life situations. Together with, our study is different from the Öhrvall et al.’s study for some aspects, by investigating a possible difference among three representatives of functional status as to executive function tasks (ability to manage daily activities) and participation in the different life situations. Then, our study was conducted on a more homogeneous group (involving only children with hemiplegic CP), whereas the study in question enrolled participants with different typology of CP. Consequently, our study findings suggested that MACS, GMFCS and CFCS tools may have complementary nature in predicting executive functions and the extent of participation in different life situations. As well, our study findings demonstrated that there was a meaningful difference only between high and moderate communication performance levels relating to executive function tasks and involvement in different life situations. In contrast, the CFCS levels closer to each other did not differentiate into functional performance in the real world. But nevertheless, these results displayed that high communication performance is necessary for greater participation in different life situations and executive function tasks. Therefore, both functional motor and communication performance classification system tools can be used as a concise and precise language by health professions, families, and researchers to describe a child's actual performance in daily life (participation and executive functions). In other words, both functional motor and communication performance instruments (GMFCS, MACS, and CFCS) can be used to predict the extent of participation in different life situations and performance in daily activities. These implications are in line with a previously published study' s results reporting a strong relationship between MACS and ABILHAND-Kids [6]

The actual performance of children with CP is intimately related to gross motor function, manual ability, and daily communication abilities [11; 27]. Besides, as reported previously, gross motor functions [28], communication skills [29], and ability to manage daily activities [6] are essence for executional autonomy in participation, especially of children with CP.

Unlike some studies demonstrating a strong or moderate correlation between classification instruments of gross motor function, communication performance, and manual ability [10; 11; 13; 14], we found out a weak to moderate among functional motor and communication classification systems’ levels. This variability may arise from difference in enrolled population. Eventually, the existing evidence confirms that manual ability, gross motor function, and communication performance, as measured by MACS, GMFCS, and CFCS, complement one another to constitute child’s functional performance in the real world. The significant difference among three levels of MACS, GMFCS, and CFCS for both the manual performance in daily living and participation in different life situations suggest that an appropriate functional motor and communication performance level might predict disabled children’s actual performance in daily life. In line with this purpose, this cross-sectional study sought to further validate GMFCS, MACS, and CFCS levels by using outcome measures as an external reference.

Although the homogeneous sample size was a strong aspect of our study, this may also have led to a limitation for the current study. Because our study participants’ functional profiles changed between level I and Level III on the MACS, GMFCS, and CFCS, our raw data could not meet assumptions for a linear regression model to separately investigate the influence of MACS GMFCS and CFCS on executive function tasks and participation in different life situations (lack of participants in level V on classification systems-lack of reference data). Therefore, future research is needed to investigate the possible impacts of functional motor and communication performance classification systems on the actual performance in daily life.

In conclusion, this study is novel in investigating possible differences among three representatives of functional status associated with executive function tasks and participation in different life situations by using ABILHAND-Kids and the CASP questionnaires as an external reference. As we hypothesized, our study findings revealed significant differences among each of the three levels of MACS and GMFCS for manual performance and involvement in different life situations. However, for the communication performance instrument, although it was demonstrated a significant difference between high and moderate levels (I-III), levels closer to each other (I-II or II-III) have not differed in actual performance in daily life. In conclusion, we assert that executive function tasks (manual performance in daily life) and participation in different life situations are more sensitive to levels of functional motor classification instruments than levels of communication performance instrument. In other words, our study's findings indicated that executive function tasks and participation scaled scores increase with greater GMFCS, MACS, and CFCS levels, except for CFCS levels closer to each other.

Acknowledgement:

The authors are very grateful to families, and schools’ teachers attended by the participants because, without their help, we could not have carried out the current study.

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Table I: Demographic Characteristics and Descriptive Information of the Participants

			(n=98) X ± SD, %95 CI		Age (y) X ± SD, %95 CI
Age (y)			9,3 ± 3,0 [%95 CI:8.7-9.9]
CASP	Home Participation		74,6 ± 17,3 [%95 CI:71.13-78.06]
	Community Participation		71,0 ± 18,1 [%95 CI:67.37-74.62]
	School Participation		68,9 ± 18,1 [%95 CI: 65.27-72.52]
	Home and Community Living Activities		59,4 ± 18,5 [%95 CI:55.69-63.1
ABILHAND-Kids (Logits)			2,2 ± 1,4 [%95 CI:1.91-2.48]
			n	(%)
Gender		Male	56	57,1
Gender		Female	42	42,9
Affected Side		Right	49	50,0
Affected Side		Left	49	50,0
Cognitive impairment		Yes	12	12,2
Cognitive impairment		No	86	87,8
Hearing impairment		Yes	0	0,0
Hearing impairment		No	98	100,0
Speech impairment		Yes	11	11,2
Speech impairment		No	87	88,8
Visual impairment		Yes	9	9,2
Visual impairment		No	89	90,8
					X	SD	%95CI	p
MACS		I	28	28,6	8,9	3,1	7.6-10.1	0,634
		II	29	29,6	9,7	2,9	8.5-10.8
		III	41	41,8	9,2	3,2	8.1-10.2
GMFCS		I	54	55,1	9,1	3,0	8.28-9.9	0,382
		II	38	38,8	9,7	3,1	8.6-10.7
		III	6	6,1	8,2	3,1	4.9-11.4
CFCS		I	55	56,1	9,0	3,0	8.1-9.8	0,454
CFCS		II	29	29,6	9,3	2,9	8.1-10.4
		III	14	14,3	10,1	3,4	8.1-12.0

X; mean, SD; Standard Deviation; CASP; Child and Adolescent Scale of Participation; MACS, Manual Ability Classification System; GMFCS, Gross Motor Function Classification System; CFCS, Communication Function Classification system

Table II: Distribution of Children in Different MACS Levels Within GMFCS Levels

		GMFCS
		I		II		III
		n	%	n	%	n	%
MACS	I	25	89,3%	3	10,7%	0	0,0%
	II	16	55,2%	13	44,8%	0	0,0%
	III	13	31,7%	22	53,7%	6	14,6%

MACS, Manual Ability Classification System; GMFCS, Gross Motor Function Classification System; n, no of participants

Table III: Performance of children on ABILHAND-kids and CASP by MACS Levels

		MACS
		I^a (n=28)			II^b (n=29)			III^c (n=41)				P
		X	SD	%95 CI	X	SD	%95 CI		X	SD	%95 CI
CASP	Home Participation	92,7	10,0	88.8-96.5	77,6	9,5	73.9-81.2		60,0	12,0	56.2-63.7		0,000
	Community Participation	87,9	13,6	82.6-93.1	72,6	11,1	68.3-76.8		58,2	14,7	53.5-62.8		0,000
	School Participation	87,3	13,2	82.1-92.4	71,2	8,9	67.8-74.5		54,8	13,2	50.6-58.9		0,000
	Home and Community Living Activities	75,9	14,5	70.2-81.5	63,4	13,3	58.3-68.4		45,2	12,5	41.2-49.1		0,000
ABILHAND-Kids (logits)		3,9	1,1	3.4-4.3	2,2	0,5	2.0-2.3		1,2	0,8	0.9-1.4		0,000

CASP, Child and Adolescent Scale of Participation; MACS, Manual Ability Classification System ; X; Mean, SS; Standard Deviation; Post-Hoc: a>b>c; n, no of participants

Table IV: Performance of children on ABILHAND-kids and CASP by GMFCS Levels

		GMFCS									P
		I^a (n=54)			II^b (n=38)			III^c (n=6)
		X	SD	%95 CI	X	SD	%95 CI	X	SD	%95 CI
CASP	Home Participation	82,6	15,7	78.3-86.8	67,6	12,2	63.5-71.6	45,8	7,5	37.9-53.6	0,000
	Community Participation	81,1	14,4	77.1-85.0	61,5	12,2	57.4-65.5	39,6	9,4	29.7-49.4	0,000
	School Participation	78,2	15,6	73.9-82.4	60,9	11,6	57.0-64.7	35,8	7,4	28.0-43.5	0,000
	Home and Community Living Activities	67,9	17,6	63.0-72.7	51,6	12,5	47.4-55.7	31,7	6,1	25.2-38.1	0,000
ABILHAND-Kids (logits)		2,8	1,5	2.3-3.2	1,8	0,8	1.5-2.0	0,3	0,2	0.0-0.5	0,000

CASP, Child and Adolescent Scale of Participation; GMFCS, Gross Motor Function Classification System X; Mean, SS; Standard Deviation, Post-Hoc: a>b>c; n, no of participants.

Table V. Performance of children on ABILHAND-kids and CASP by CFCS Levels

		CFCS									P
		I^a (n=55)			II^b (n=29)			III^c (n=14)
		SD	%95 CI	X	SD	%95 CI	X	SD	%95 CI
CASP	Home Participation	80,7	16,0	76.3-85.0	72,1	13,9	66.8-77.3	55,6	14,5	47.2-63.9	0,000
	Community Participation	79,4	14,6	75.4-83.3	65,6	15,2	59.8-71.3	49,0	13,6	41.14-56.8	0,000
	School Participation	76,1	15,8	71.8-80.3	65,5	15,4	59.6-71.3	47,9	13,4	40.1-55.6	0,000
	Home and Community Living Activities	65,8	18,2	60.8-70.7	56,4	14,6	50.8-61.9	40,4	11,8	33.5-47.2	0,000
ABILHAND-Kids (logits)		2,5	1,5	2.0-2.9	2,2	1,1	1.7-2.6	1,2	0,9	0.6-1.7	0,007

CASP, Child and Adolescent Scale of Participation; CFCS, Communication Function Classification system; X, Mean, SS; Standard Deviation; Post-Hoc: a>c, a=b, b=c; X, mean; SD, Standard Deviation; n, no of participant

Table VI: Inter-relationships between functional motor and communication systems

		MACS	GMFCS
GMFCS	r	0,491
GMFCS	p	0,000
CFCS	r	0,247	0,574
CFCS	p	0,014	0,000

MACS, Manual Ability Classification System; GMFCS, Gross Motor Function Classification System; CFCS, Communication Function Classification system; r, Spearman Correlation Coefficient; p, significance

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Investigation of Difference Between the Three Representatives of the Functional Status According to Executive Functions and Participation in Children With Hemiplegic Cerebral Palsy

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