Effectiveness of individualized,goal directed institutional based rehabilitation programme in children with developmental delay disorders,in a region with limited rehabilitation accessibility

Abstract

PURPOSE:

To evaluate effectiveness of individualized, goal directed rehabilitation protocol in improving functional outcomes in children with developmental delay disorders in a region with limited rehabilitation accessibility and to explore if goal setting influences changes observed in functional levels.

METHODS:

Children (6 months to 17 years) with developmental delay, who visited as out-patients to the Department of Physical Medicine and Rehabilitation and whose care givers were willing to participate in the study, were enrolled for a period of one year. Individualized rehabilitation protocol targeting the child’s specific problems with a simultaneous home programme was performed. A single group pre-post study design was used to assess the protocol’s effectiveness.

OUTCOME MEASURES:

Goal attainment scale (GAS), Gross Motor Function Classification System (GMFCS) Level, Gross Motor Functional Measure (GMFM) and Manual Ability Classification System (MACS) were administered pre and post-intervention monthly for 3 months. Statistical analysis was done using SPSS statistics version-22.

RESULTS:

Total of 32 children participated in the study. Significant differences between pre and post scores of GAS score (Wilcoxon’s signed rank test [ $W$ value] $=$ 4.937, $p$ -value $<$ 0.005), GMFCS ( $W$ value: $-$ 3.435, $p$ -value $<$ 0.005), GMFM ( $W$ value: 4.937, $p$ -value $<$ 0.005) and MAC score ( $W$ value: $-$ 4.714, $p$ -value $<$ 0.005) were noted at 3 months. No correlation was observed between the GAS pre-post score and GMFM, GMFCS and MAC pre-post scores.

CONCLUSION:

An effective rehabilitation programme in children with developmental delay should be individualized and should be goal directed to achieve maximum functional improvement. Improvement in the goals set were independent of the improvement in functional outcome levels. Care-givers play a pivotal role in both individualization and goal setting for rehabilitation, especially in a region with limited rehabilitation accessibility.

1. Introduction

Developmental Delay Disorders (DDD) involves a child’s inability to reach important developmental milestones within an expected time period, in one or more domains [1]. DDDs are very common. Nearly 200 million children worldwide do not attain all their developmental milestones in the first five years [2]. In India, developmental delay may affect nearly 10% of children [3]. Developmental delay disorders can affect motor control and posture. It may be associated with other co-morbidities like epilepsy, behavioural and cognitive deficits, communication issues and musculoskeletal deformities [4, 5]. Delay in a motor domain (gross motor and fine motor domain) may be the first or most obvious sign of a developmental disorder [1]. DDDs not only impacts the quality of life of the child, but also their family members [6]; making rehabilitation essential. Traditionally, rehabilitative interventions have focused on improving the dysfunctional control, namely posture and movement. However, in the recent times, there has been a paradigm shift towards adopting measures for maximizing the child’s environment, their independence in daily activities and their community participation i.e., from a child focussed to a context focussed approach [7]. This has been aided by goal based rehabilitation protocol, wherein goals, based on clinical assessment, are set and interventions targeting those goals are administered. In the past few years, studies have explored the benefit of developing a goal directed, individualized plan [7, 8].

However, the scenario for rehabilitative services differs between developed countries and developing countries. In a developing country, even today, the utilization of these available services are limited among these children due to low resources of family, poor accessibility and low awareness amidst care-givers of children with developmental delay. Hence, the current study was designed to evaluate if an individualized, goal directed rehabilitation protocol was effective in improving the functional outcomes in children with developmental delay disorders residing in a region with limited rehabilitation accessibility and further explore if goal setting had an impact on functional level changes in these children.

2. Methodology

2.1 Setting of study

The study was conducted at the out-patient unit of Department of Physical Medicine and Rehabilitation (PMR) in a tertiary care institution. The institute is a government hospital that caters to both rural and urban population of Jodhpur (a district in India). The institute has a separate Department of Physical Medicine and Rehabilitation, offering medical rehabilitation with multi-disciplinary approach to children and adults with impairments and disability. The department comprises of Physiatrists, Occupational Therapists and Physiotherapists. Speech therapy and psychological interventions are availed as referred services on an as-needed basis. It was noted that most of the children referred to the department had developmental delay disorders and care-givers of these children cited various reasons for their limited utilization of rehabilitative services on a regular basis. Loss of daily wages when care-givers brought their child to the department, resulting in financial constraints for family, low socioeconomic status, ignorance about condition, poor parental education, false family beliefs and unrealistic expectation of normalcy were a few reasons cited by the care-givers for the limited utilization. Hence, the study had to be formulated keeping these limiting factors into consideration.

2.2 Study design

A prospective study with single group pre and post-intervention design was opted for. The accessibility and utilization pattern of rehabilitation services was limited on a regular basis in the region where the study was being conducted for these children. Hence, due to ethical reasons, it was mandatory to provide the best possible services to those children, whose care-givers were at least willing to avail the facilities present. Therefore, the present study was designed such that each child served as their own control. One group pre-intervention and post intervention was chosen, wherein the pre-intervention period served as the control period itself and also, provided information about what the outcomes would have been if the intervention had not occurred.

The study was conducted at the out-patient unit of the Department of Physical Medicine and Rehabilitation (PMR) for a period of one year after approval from the Institutional Ethics Committee.

2.3 Participants

Children aged between 6 months to 17 years who had developmental delay with primary motor domain involvement were included in the study. These children were initially identified for developmental delay by Department of Paediatrics. Children with pure language and social domain involvement (Autism related disorders/intellectual disorders) and exclusive learning disabilities like dyslexia, dysgraphia were excluded. Of the selected children, care-givers were explained the following intervention. Those willing to participate and follow the instructions were enrolled in the study after written informed consent.

2.4 Intervention

Rehabilitative intervention was individualized for each child. Individualization was done with regards to the goals set as well as the rehabilitation protocol to be implemented. The goals were set using components of International Classification of Functioning, Disability and Health (ICF): body functions and structures, activity and participation and environmental factors influencing the child’s surroundings. Specific body function and structure problems of the child was assessed clinically by rehabilitation professionals in terms of motor function, muscle tone, gross and fine movement patterns, posture and balance. Activity limitation was observed by the physiatrist and therapists and also as reported by care-givers/parents. Inputs regarding the participation limitation and environmental/contextual factors influencing the child in community settings was provided by care-givers/parents of these children. Thus, during the initial visit, the child along with care-givers first visited the Physiatrist for consultation and then visited the occupational therapists and physiotherapists. A multidisciplinary team discussion was followed wherein the rehabilitation professionals discussed with care-givers the assessments in common language and treatment options available in the context that care-givers provided inputs for. Thereafter, the felt needs of care-givers/parents were asked for. This was done in order to address the low awareness levels of care-givers and to enhance their understanding of how rehabilitation works. Thus, goals set used both multidisciplinary approach and family centred approach. Goal setting added a specificity and direction to rehabilitation protocols. The protocol included interventions targeting specific body structures and functional impairments as assessed and also focussed on improving the functionality of these children in context to the environment where they resided. These individually defined programs used a combination of motor learning approaches as administered through physical therapy and occupational therapy along with age- appropriate training for activities of daily living.

Care-givers were advised to visit the hospital daily for 3 months initially, for the administration of the interventions so as to intensify practice and improve compliance with protocol [9, 10]. Home programme was also taught to augment practice of activities in natural environments. However, with limited resources of care-givers and accessibility issues, the protocol had to incorporate care-giver’s feasibility as well. In case, the care-giver was unable to visit daily, he/she was advised to visit the hospital at regular intervals either weekly, bi-weekly or monthly and follow the home programme on the intervening days, at least twice a day. Care-givers were also educated about various aspects of disorder of developmental delay, the interventions and ways to cope up with the chronic condition and various adaptations and environmental modifications in home settings. The consistency and compliance with home programme was maintained by the verbal/written feedback notebook brought by the care-givers on the day of visit. The parents were asked to demonstrate the therapy and were also asked about the child’s daily schedule. The goals set, the therapy administered and the home programme were titrated according to the clinical response noted and feedback of care-givers at regular intervals during the first three months. The following outcome parameters were chosen in order to assess the changes in the set goals and changes in the functionality of gross motor and fine motor levels of each child. The outcomes were measured pre-intervention and subsequently each month after initiation of intervention until completion of 3 months.

2.5 Outcome measures

2.5.1 Goal Attainment Scale (GAS)

It is a sensitive evaluation tool that demonstrates changes in the performance of a goal that is important for the child and the family. GAS is an individualized, criterion-referenced measure of change in goals set for the individual child, being a 5-point ordinal rating scale with score of $-$ 2 as ‘much less than expected outcome’ and score of $+$ 2 as ‘much greater than expected outcome’. Thus, it can capture subtle but important changes in activity skills and can be used to measure progress in the heterogeneous population with a variety of treatment goals [11].

2.5.2 Gross Motor Functional Measure (GMFM)

It is a standardised observational instrument developed to assess change in gross motor function in children with cerebral palsy aged 5 months to 16 years. Original GMFM version had 88 items with a 4-point ordinal scale scoring system of 0 to 3, where 0 indicated inability of child to initiate task and 3 indicated that child completes 100% task. Eighty- eight items were grouped into five dimensions of lying and rolling, sitting, crawling and kneeling, standing, and walking and running and jumping [12].

2.5.3 Gross Motor Function Classification System (GMFCS)

It is a 5-level ordinal classification system which helps in classifying and assessing the severity of child’s motor disability using one of the five levels, ordinal grading system, with Level 1 indicating the highest motor functional independence and Level 5 the lowest gross motor function [13].

2.5.4 Manual Ability Classification System (MACS)

It describes the ability of children with impairment to handle various objects manually in daily activities [14]. It includes 5-level ordinal classification system based on the child’s self-initiated ability to handle objects and their need for assistance or adaptation to perform those activities in everyday life. Level 1 includes children with minor limitations, while Level 4 and Level 5 are for children with severe functional limitations. This system was validated for children aged 4 to 18 years. Hence, Mini-MACS, an adaptation of MACS, was developed for children aged 1–4 years. The study used both these scales depending upon the age of the child to assess the manual handling of objects in them [14].

Table 1

Frequency distribution of demographic profile of participating children with developmental delay disorders

Variables	Demographic distribution ( $N=$ 32)	Percentage (%)
Gender
Male	20	63
Female	12	37
Age group (years)
$<1$	4	13
1–5	19	59
5–10	7	22
$>10$	2	6
Birth history (predisposing factor)
Preterm	10	31
Delay in birth cry	13	41
Post natal sepsis	2	6
Asphyxia while in labour	3	9
Others	4	13
Seizures
Yes	16	50
No	16	50
Type of tone
Hypertonia	21	66
Hypotonia	5	16
Dystonia	3	9
Dyskinesia	3	9

Table 2

Comparison of pre and post intervention scores of GAS, GMFM, GMFCS, MAC

Outcome measure	Pre-intervention P0	Post intervention P1	Post intervention P2	Post intervention P3	$W$	sig
GAS	25.44	34.57	41.53	48.38	4.937	0.000
GMFM	0.14	0.196	0.27	0.357	4.937	0.000
GMFCS	4.1	3.71	3.14	2.67	$-$ 3.435	0.001
MAC	4.59	4.39	3.5	2.68	$-$ 4.714	0.000

(GAS: Goal attainment scale, GMFCS: Gross Motor Function Classification System Level, GMFM: Gross Motor Functional Measure, MAC: Manual Ability Classification, P0: Pre-intervention mean score at day 1, P1: Post intervention mean score at first month P2: Post intervention mean score at second month P3: Post intervention mean score at third month, $W$ value: Wilcoxon’s signed rank test, of 32 participants, number of children assessed for GAS/GMFM: 32, for GMFCS/MAC: 28).

Table 3

Correlation of day 1 score and 3 ${}^{\mathrm{rd}}$ month score for GAS and GMFCS, GMFM, MAC respectively

For day 1
	GMFCS	GMFM	MAC
Goal attainment scale (GAS)	$p=-0.268$ , sig. $=0.137$	$p=0.333$ , sig. $=0.062$	$p=-0.259$ , sig. $=0.153$
For month 3
	GMFCS	GMFM	MAC
Goal attainment scale (GAS)	$p=-0.015$ , sig, $=$ 0.934	$p={0.377}^{*}$ , sig. $=$ 0.033 (*0.05 level of significance)	$p=-0.086$ , sig. $=$ 0.639

(GAS: Goal attainment scale, GMFCS: Gross Motor Function Classification System Level, GMFM: Gross Motor Functional Measure, MAC: Manual Ability Classification, $p$ : Spearman’s rank correlation coefficient or Spearman’s rho).

Table 4

Correlation of pre-post change in score for GAS and GMFCS, GMFM and MAC

	Pre-post Change of score
	GMFCS	GMFM	MAC
GAS	$p=-0.015$ , sig. $=0.934$	$p=0.155$ , sig. $=0.396$	$p=0.006$ , sig. $=0.973$

(GAS: Goal attainment scale, GMFCS: Gross Motor Function Classification System Level, GMFM: Gross Motor Functional Measure, MAC: Manual Ability Classification, $p$ : Spearman’s rank correlation coefficient or Spearman’s rho).

2.6 Statistical analysis

At the end of 1 year, the data was analysed using IBM SPSS statistics version-22. The demographic data was analysed for age and sex distribution and incidence of important findings in their clinical history. The children were also classified based on their findings in the clinical examination. The scores for each outcome measure was compared pre and post intervention taking day 1 (pre-intervention) and 3 ${}^{\mathrm{rd}}$ month score (post intervention) using Wilcoxon’s signed rank test, considering the data set to be ordinal type and the two sets of scores (pre and post) coming from the same participants. Also, the trend for change at day 1, 1 ${}^{\mathrm{st}}$ month, 2 ${}^{\mathrm{nd}}$ month and 3 ${}^{\mathrm{rd}}$ month was assessed for each outcome measure using Friedman’s one-way ANOVA test since each outcome measure was assessed at three different occasions and data was ordinal type. A correlation was done between GAS and the other three outcome measures of gross and fine motor level, in order to assess the association of goal setting on changes noted in functional outcome measures. The correlation was analysed using Spearman’s rank correlation coefficient, a non-parametric test used for ordinal data sets.

3. Results

A total of 32 children (Male $=$ 20, Female $=$ 12) participated in the study. The youngest participant was aged 6 months and oldest child participating in the study was 12 years old. Of them, 13 children had birth history of delayed cry and 10 were born pre-term with associated issues like low birth weight and hyper-bilirubinaemia. These children were identified and diagnosed by department of Paediatrics and referred to PMR department thereafter. Among the included children, 31 had been diagnosed as cerebral palsy and 1 child had Joubert Syndrome. The parents of 16 children reported history of seizure disorder (Table 1). Neuro-imaging (MRI scans) was available for 27 children, of which 23 children had features suggestive of hypoxic ischemic encephalopathy, one child each had features of encephalomalacia, atrophy of periventricular region, demyelination and one child had normal scan. Clinical examination showed that there were 21 children with hypertonia, 5 with hypotonia, 3 with dystonia and 3 with dyskinesia. Voluntary motor control (according to Daniels and Worthingham grading system) was poor for 19 children and 13 had fair control in affected limbs. Behavioural issues were noted in 6 children, out of which 2 children had overlapping features of autism and 4 had mild hyper activity. The development quotient (numerical expression of a child’s developmental level as measured by dividing the developmental age by the chronological age and multiplying by 100) was assessed by clinical psychologist and was available for 7 cases which ranged from 48 to 98. It could not be done for the remaining children because of the unwillingness of the care-givers. During pre-study period, gross motor functional level was tested for 28 children out of 32, since there were 4 children below 1 year of age. Of these, GMFCS was 5 for 16 children, level 4 for 5 children, level 3 for 3 children, level 2 for 2 children and level 1 for 2 children. Fine motor level as assessed through MAC was 5 for 18 children, level 4 for 7 children and level 3 for 3 children. During the study period, 15 children visited hospital daily for rehab, 12 children came fortnightly, 4 children visited bi-weekly and 1 child came at weekly interval. On the remaining days, home programme was followed by care-givers.

Comparison using Wilcoxon’s signed rank test for day 1 (pre-intervention) and post intervention 3rd month scores showed that there was significant difference in the scores of GMFM ( $W=$ 4.937, sig. $=$ 0.000), GMFCS ( $W=-3.435$ , sig. $=0.001$ ), MACS/mini MACS score ( $W=-4.714$ , sig $=0.000$ ) and GAS score ( $W=$ 4.937, sig. $=$ 0.000) (Table 2). Comparison on day 1, 1 ${}^{\mathrm{st}}$ month, 2 ${}^{\mathrm{nd}}$ month and 3 ${}^{\mathrm{rd}}$ month using Friedman’s 1 way ANOVA also showed that there was significant difference in scores at all the stages in GMFCS (F day 1 vs 1 ${}^{\mathrm{st}}$ month $=$ 38.151, F day 1 vs 2 ${}^{\mathrm{nd}}$ month $=$ 9.429, F day 1 vs 3 ${}^{\mathrm{rd}}$ month $=$ 9.880, $p$ -value $=$ 0.000) and MAC/mini MAC (F day 1 vs 1 ${}^{\mathrm{st}}$ month $=$ 33.781, F day 1 vs 2 ${}^{\mathrm{nd}}$ month $=$ 26.278, F day 1 vs 3 ${}^{\mathrm{rd}}$ month $=$ 16.326, $p$ -value $=$ 0.000). There was no significant difference between scores for GMFM and GAS when compared with day 1 and 1st month, 2nd month and 3rd months, respectively.

Correlation using Spearman’s rank correlation coefficient between day 1 pre-intervention score and 3 ${}^{\mathrm{rd}}$ month post intervention scores of GAS with day 1 and 3 ${}^{\mathrm{rd}}$ month scores of GMFCS, GMFM and MACS showed no correlation of GAS with GMFCS and MACS. However, there was a positive correlation between GAS and GMFM at 3 months (Table 3). Similarly, correlation in the change of scores of GAS with respect to change in scores for GMFCS, GMFM and MACS showed no correlation (Table 4).

4. Discussion

The study aimed to explore effectiveness of individualised, goal directed rehabilitation for children with developmental delay residing in a region with limited rehabilitation accessibility. A significant improvement in all outcome measures from pre intervention to post intervention state was noted. Not all goals set were achieved, but the functionality of children in terms of both gross and fine motor levels were improved.

In a region, where awareness regarding comprehensive rehabilitation was relatively new, understanding the very concept of abilities in a child with developmental delay, became itself a novel idea. As evident, out of 330 children visiting the department during the study period, only 32 (10%) were willing to participate in rehabilitation on a regular basis and therefore, could be enrolled into the study.

The children involved were a heterogeneous group with different types of topographical involvement and tone type. Smits et al. had reported that for children with bilateral spastic involvement, treatment has to focus on gross motor function enhancement and for unilateral spastic involvement, focus should be on continuous situation training stressing the need for individually defined protocols [15, 16]. Even in the present study, while individualizing the rehabilitation protocol for each child based on impairments of body structures and functions, a similar pattern was noted. Moreover, it was observed in the current study that individual protocols for children less than 5 years were mostly child focussed (at structural level) and for older children more than 5 years, it gradually shifted to a context focussed approach (environmental factors).

However, irrespective of approach used for an individual child, improvement was noted which was seen in a previous study by Kruijsen-Terpstra et al. [17] as well.

The variation in terms of community to which each child belonged to and the environmental factors influencing activity and participation of each child, led to usage of a combination of individual techniques for a specific problem faced by a child at a particular moment. This was based on clinical assessments by rehab professionals and inputs from child’s care-givers/parents. Thus, the individualized rehabilitation programs based on the individualized goals set in this study became more relevant in contextual settings of clinical practice. Literature has also showed that depending on the functional status and age of child, parents tend to prioritize different goals. In a study by Knox, where the majority of participants were below 6 years of age, the felt need of parents for children with GMFCS levels II to IV focussed on standing and walking [18]. In another study by Brandao et al. children aged over 7 years with GMFCS level 4, personal care activities were considered more important by parents and caregivers, followed by school activities and play. In their study, mobility demands was more frequent for the 3–6 year age group [19]. Similar findings were observed by Rosenbaum et al. [20]. Hence, the changing felt needs and priorities of care-givers made it essential to involve them in setting the goals, even in the current study where the patient group included a wide range of age group and functional level.

Further, in order to improve functionality in home setting and natural environment, it was essential to know the contextual factors in which these children resided and interacted. This was again contributed by the primary care-givers and family members. As suggested by Shumway et al., including contextual factors enhances transfer of new skills to the environments where they were meant to be used [21].

Moreover, the care-givers were responsible for carrying out the home programme, apart from the therapy session received at the Institute. It is well known that repetition and practice enhances motor learning [9]. Therefore, higher sessions of practise in a natural environment of child was more likely to improve a task performance and help in overall goal achievement. Previous studies have also emphasized about the positive effects of home programme with parents and care-givers as active participants in goal setting and training sessions [22, 23, 24].

Eliasson et al. reported that the use of a home program was less controllable and can lead to variations in its impact because of variable compliance [25]. Hence, in order to increase compliance, parental education is important. A recent systematic review by Myrhaug et al. showed that more than half of the effective interventions have reported parental education and their active involvement in training [9]. In the current study, it was noted that educating the parents/care-givers gave a better understanding of how rehabilitation would work, an objectivity in their expectation, defined what could be their role as care-givers, helped them in prioritizing their goals accordingly and actively participating in goal formulation. It may also have provided a vent out for the stress, they faced being a care-giver of the child with a chronic condition; although this wasn’t particularly explored.

Thus, as evident in the current study, individualisation of rehabilitation services in terms of goal set and protocol implemented in context to the place and environment, where the service is being provided, became important for effectiveness of rehabilitation programme provided for these children with developmental delay. The primary care-givers and family members play a significant role in formulation of such individualized programmes, apart from the essential contribution of rehabilitation professionals.

The study also showed that the change in score for goal attainment scale and the change in score for GMFCS and MACS did not have any significant correlation. This is in concordance with previous studies. A recent systematic review by Carlberg and Lowing did not provide support for influence of goal setting on functional treatment outcome. They did, however, acknowledge that it is difficult to separate the effect of goal setting from effect of activity focused intervention and that it should be incorporated in a family centred practice [7, 26]. As seen in the current study also, goal setting became an integral part of rehabilitation intervention, since it helped care-givers to understand in what direction rehabilitation was aimed and made the improvements visible in terms of outcome achieved. Thus, it improved specificity of rehabilitation and helped emphasizing activities which the family considered important for the child to learn. It also facilitated better coordination of service among health care professionals as well as between the family and professionals. Previous studies have also shown similar finding in terms of setting goals in activity focussed intervention of particular interest [26, 27].

The study had limitations of an overall small sample size and an even smaller sample for subgroups of care-givers’ frequency of hospital visit for rehab. The study could not formulate a uniform protocol in order to consider the social and environmental issues, as experienced by care-givers in availing the services. Though a rigid protocol would devoid even children with good rehab potential of the needful benefits of services, the study did bear this limitation. There was also no objective evaluation/tool used for investigating the role of the home programme administered by parents on effectiveness of rehab. Future studies are recommended with larger sample size with two group study, exploring the impact of frequency of hospital visit on effectiveness of rehab on functional outcomes, assessing factors limiting the accessibility and utilization of available services and objective assessment of role of home programme in improving effectiveness of rehab on functional measures.

To conclude, a successful and effective rehabilitation programme is multifactorial. It must include a goal-directed protocol, using a combination of interventions, individualized according to clinical assessment, age, functional level of child, contextual settings of the child and felt needs of the care –givers. It should include daily practice sessions both with the therapist and at home to enhance dosing of therapy and a regular weekly follow-up for better tailoring of the protocol, especially in a region with limited accessibility of services. It is essential to inculcate a multidisciplinary approach recognizing the role of different professionals as well as the important role of parents/care givers in the rehabilitative process.