Abstract
AIM:
This study evaluated the inter-observer reliability and stability over time of the Eating and Drinking Ability Classification System (EDACS) for children and young people with cerebral palsy (CP).
METHOD:
Case records for 97 children with CP were examined to collect retrospective data about eating and drinking abilities at four time-points with a minimum of 2 years between each time-point. Sex, Gross Motor Function Classification System (GMFCS) level, presence of feeding tube and orthopaedic issues were recorded from case records. One speech and language therapist (SaLT1) classified eating and drinking ability using EDACS for all cases at all time-points; SaLT2 assigned EDACS levels for 50 cases at time-point 1; SaLT3 assigned EDACS levels for 24 cases at all time-points. Inter-observer reliability and stability over time were assessed using the Intraclass Correlation Coefficient (ICC). Associations between EDACS levels and functioning recorded with other Functional Classification Systems (FCSs) were calculated using Kendall’s tau (
RESULTS:
Out of 97 children, 48 were male, 48 had feeding tubes, and 83 had orthopaedic issues. ICC for EDACS levels recorded by SaLT1 across all time-points was 0.97 (95% CI 0.96–0.98); changes in EDACS levels occurred infrequently and never by more than one level. ICC between SaLT1 and SaLT2 at time-point 1 was 0.8 (95% CI 0.67–0.89); ICC between SaLT1 and SaLT3 across all time-points was 0.95 (95% CI 0.92–0.98). Association between GMFCS and EDACS was moderate (
INTERPRETATION:
Retrospective use of EDACS to classify children’s eating and drinking abilities appears reliable; EDACS appeared stable over 6 or more years in 86% of the cases.
Simplified summary descriptions of functional classification systems suitable for use with people with cerebral palsy
Simplified summary descriptions of functional classification systems suitable for use with people with cerebral palsy
In the field of developmental disability, it is well known that children and young people with the same condition such as cerebral palsy (CP), vary considerably in their functional abilities. In recent years, there has been a recognition of the utility of functional classification systems (FCSs) which provide far more detail than a diagnostic label alone [1]. The oldest and most widely used of these FCSs is the Gross Motor Function Classification System (GMFCS [2]); others have been developed to describe manual ability (Manual Ability Classification System MACS [3]), communication function (Communication Function Classification System CFCS [4]) and speech production (Viking Speech Scale VSS [5]). Each of these FCSs describe function using distinct levels which are meaningful in daily life, replacing poorly defined, value-laden terms such as mild, moderate and severe. The GMFCS, MACS and CFCS describe the full range of ability in 5 levels whereas the Viking Speech Scale uses 4 levels. See Table 1 for summary headings for each of the FCS.
FCS are considered to be useful in both clinical and research contexts because they can facilitate clear communication and planning at local and national levels. Different FCS levels can be used to consider different clinical management options and enable clear reporting of research findings contributing to the clinical evidence base. In some cases, FCS enable prediction of future outcomes through the stability of the assigned FCS level [1].
The impact of eating and drinking difficulties on poor respiratory health has been well documented [6, 7, 8, 9]. Eating and drinking difficulties have also been associated with limited growth and poor health because of compromised nutritional intake [10, 11], and in some instances can lead to premature death [12]. Prevalence figures for eating and drinking difficulties for children with CP vary widely depending upon definitions and measures used [13]. Prevalence rates include: 21% with “swallowing and chewing difficulties” [14]; 40% with “difficulties with eating” [15]; 55% with limitations to “chewing and swallowing” [16]; and 85% with “oro-pharyngeal dysphagia” assessed using two standardised measures [17]. A systematic review of ordinal scales used to measure the eating and drinking ability of people with CP [18] identified 15 different scales: 13/15 were for clinician or researcher use only; 8/15 used the terms mild, moderate and severe with varying definitions to describe different aspects of eating and drinking impairment; none met recommended psychometric quality standards [41]. The review clearly identified the need for a new classification system of eating and drinking ability [18].
The Eating and Drinking Ability Classification System (EDACS) has recently been added to this group of FCS [19, 1]. EDACS describes the full range of eating and drinking ability of children and young people with CP from age 3 years in five distinct levels, using the key features of safety and efficiency. EDACS focuses on a person’s usual performance of biting, chewing, drinking, and swallowing and the co-ordination of these with respiration. Descriptions of different levels of ability include details of food and fluid textures that can be managed without choking or aspiration (entry of food or fluid into the lungs). Descriptions also include the extent to which food and fluid are retained in the mouth and the speed and range of movement brought to the task. Like the other FCSs, EDACS has been shown to be valid and reliable for children and young people with CP [19]. Studies have demonstrated that EDACS meets quality standards for inter-observer reliability both between health professionals and between parents and health professionals [19, 20, 21, 22]. High intra-rater reliability [20] and strong construct validity [21, 22] have also been demonstrated.
Each FCS provides broad categorical descriptions of function such that a level assigned to a child with CP is unlikely to change over time; if change does happen, it is likely to be by just one level [1]. Öhrvall et al. [23] stated that it is necessary to consider the extent to which stability is influenced by potential changes in ability or whether it is due to inconsistency in the use of the tool by different or the same raters. Research evaluating the GMFCS and MACS has demonstrated the stability of function over time in retrospective [24] and prospective studies [23, 25, 26].
There is limited evidence from longitudinal observations of the eating and drinking abilities of people with CP, which is hampered by the lack of consensus concerning measurement tools [27]. There is no clear understanding of the natural history of eating and drinking development in CP and no context within which to assess the impact of interventions to improve function. Currently, parents and health professionals make significant and emotive clinical decisions such as use of tube feeding without evidence of the stability of children and young people’s eating and drinking ability. Conflict can arise between parents and health professionals when engaged in decision-making linked to children’s limitations in eating and drinking abilities [28, 29, 30]. Some parents resist proactive recommendations by the clinical team to use alternative and supplementary tube feeding for their child at a young age; this can result in limited growth and compromised health associated with chronic malnutrition for their child’s lifetime [10]. The EDACS is a measurement tool that parents are able to understand, recognising their own child’s eating and drinking abilities within the levels [19]. Discussions with parents could be enhanced with a clear statement about a child’s eating and drinking ability using EDACS along with research evidence about how likely it is that a particular level will change in the future.
The use of EDACS in clinical and research contexts will be supported by evidence concerning the stability of eating and drinking function measured by EDACS throughout childhood. The purpose of this study was to: 1) to measure the inter-observer reliability of EDACS applied retrospectively using case notes, 2) to assess the stability over time of a child’s EDACS level, and 3) to compare EDACS levels with other areas of function measured by other FCSs.
Method
This study was carried out as a retrospective case note review, following a similar study design employed in retrospective examination of GMFCS levels from case notes by Wood and Rosenbaum [24]. The study took place at a centre providing specialist healthcare and education to children and young people with complex neuro-disability, in the UK. The multi-professional nutrition team manages the nutritional and hydrational intake of children with complex neuro-disability; team members include dietitian, neuro-developmental paediatrician, speech and language therapist and specialist children’s nurse. Recommendations for safe and efficient mealtime support are provided for parents and care staff within a prescribed format in order to optimise nutrition and mealtime experiences. Electronic case records are available which detail the overall function and eating and drinking ability of children with CP dating from 2001.
NHS Health Research Authority approval was conferred by the London – Camden and Kings Cross Research Ethics Committee with REC reference: 16/LO/ 0344 IRAS Project ID: 197498.
Identification of cases
Children were included in the study if a diagnosis of CP was confirmed by a neuro-developmental paediatrician. Children had to have had at least 6 years contact with the specialist centre from age 3 years and above, between the years 2001 and 2016. Contact may not have been over consecutive years. The 15 year time frame was pragmatically determined because key documents from case records stored on computer databases could be routinely accessed from 2001. Data were collected for each child at four time-points (TP) with a minimum of 2 years between each TP. The selected TPs extend across the period of time that children accessed services, including TPs before and after adolescence. Children were excluded from the study if they had less than 6 years of contact or where there were insufficient data on their eating and drinking abilities.
Data extraction and coding
Case notes were used to record the following information: sex, CP type following the Surveillance of Cerebral Palsy in Europe classification tree [31], GMFCS level at each TP, presence of feeding tube and age at which tube insertion was carried out, presence of seizures, gastro-oesophageal reflux and orthopaedic issues. Case notes include annual medical reports which routinely summarise diagnosis, present and past problems, medication(s), investigations and interventions. Annual therapy reports are produced for each child which include descriptions of gross motor and communication function, manual and eating and drinking ability. The following FCSs were used to describe different aspects of children’s function: Manual Ability Classification System [3], Communication Function Classification System [4] and Viking Speech Scale [5] at TP1. See Table 1 for summary descriptions of levels for GMFCS [2], MACS [3], CFCS [4] and Viking Speech Scale [5].
The lead author (DS) extracted case note information. DS conferred with a neuro-developmental paediatrician (VC) who checked CP diagnosis and clinical summaries for each child.
Eating and Drinking Ability Classification System levels
The most detailed clinical records of eating and drinking function for each child were selected by the lead author (DS) across 4 Time Points. Clinical records were used by 3 specialist speech and language therapists (SaLTs) to identify the EDACS level which best described that child’s eating and drinking ability at the first time-point (TP1) and across all time-points (TP1, TP2, TP3, TP4). The summary descriptions for each EDACS level are given in Table 2.
Eating and drinking ability Classification System – summary description of levels
Eating and drinking ability Classification System – summary description of levels
EDACS was published in 2014 and not routinely included in case records at the specialist centre until 2015. Each of the 5 levels of EDACS systematically describes the safety and efficiency of someone’s eating and drinking ability using similar content to that contained in case records. Retrospective use of EDACS involved the conversion of qualitative clinical data into ordinal scale data. Clinical reports, case notes and annual mealtime guidance sheets produced following NHS National Patient Safety Agency [32] recommendations contain information about safety of swallowing, chewing ability, risk of aspiration or choking, recommended food textures and fluid consistencies, positioning, assistance required at mealtimes and required techniques for each child. The reliability of classifying function using EDACS using clinical data as source material was tested in two ways. The lead author and first SaLT1 (DS) assigned EDACS levels for all cases across all time points. The second SaLT2 assigned EDACS levels for 51% of randomly selected cases at TP1. The third SaLT3 assigned EDACS levels for 25% of randomly selected cases across each of the time points. Reliability testing followed guidance set out in international quality standards [41]. The reliability of the use of EDACS by pairs of SaLTs was examined using two way contingency tables to consider percentage absolute agreement and patterns of disagreement. The inter-rater reliability of EDACS levels assigned independently by pairs of SaLTs at TP1 and across all time points was examined using the Intra-Class Correlation Coefficient (ICC). The ICC (two-way random effects, single measure, absolute agreement) was calculated to examine the level of agreement between raters. ICC values of 0.9 or higher are required for the use of EDACS to be considered clinically reliable; ICC values of 0.7 or higher are acceptable for measures in groups [33].
The stability of EDACS over time was examined by comparing children’s EDACS levels recorded at each of the TPs by SaLT1. The ICC was calculated to examine the level of overall agreement in EDACS levels across all time points (two-way random effects, single measure, absolute agreement). ICC values higher than 0.9 indicate high levels of agreement and stability of EDACS levels over time [33]. Five case studies were selected and summarised to illustrate study findings.
The association between eating and drinking ability and other functional abilities measured using other Functional Classification Systems was examined using Kendall’s tau b (
A computer search of the clinical services caseload identified 97 eligible children with CP from 373 case records. 276 records were of children who did not have CP or where there was insufficient data to record EDACS levels over time. Information recorded from case notes is summarised in Table 3.
Summary of clinical information extracted from case notes including annual medical summaries, health reviews and therapy reports
Summary of clinical information extracted from case notes including annual medical summaries, health reviews and therapy reports
SaLT1 and SaLT2 used EDACS to independently rate the eating and drinking abilities of 50 children at TP1: absolute agreement between SaLT1 and SaLT2 was 62% (ICC
Reliability measures associated with use of EDACS at time point 1 (TP1) by SaLT1 vs SaLT2, for 51% of randomly selected cases
50
Reliability measures associated with use of EDACS at time point 1 (TP1) by SaLT1 vs SaLT2, for 51% of randomly selected cases
*Disagreement of 3 levels between raters linked to difference of interpretation of case notes for child with a gastrostomy because of restricted food intake linked to behavioural issues: SaLT2 understood presence of gastrostomy to indicate unsafe swallow.
Reliability measures associated with use of EDACS across all time points (TP1–TP4) for 25% of randomly selected cases by SaLT1 vs SaLT3
The ICC examining the level of overall agreement in EDACS levels across all time points was 0.97 (95% CI 0.96–0.98). The high ICC of 0.97 indicates that EDACS levels remained stable over time, with excellent agreement across time points [33].
The assigned EDACS level remained constant over time for 86% of children. The EDACS level assigned changed by one level for 14 children. 3/14 showed improvements to eating and drinking abilities from Level IV to Level III. 10/14 children had increased limitations to eating and drinking abilities which occurred between 12 and 19 yrs. Increasing limitation occurred at different EDACS levels: 6 children moved from Level IV to V; 4 children moved from Level III to IV and 1 child moved from Level II to III. 10/11 children who lost function had orthopaedic issues and/or seizures (7 children GMFCS V; 3 children GMFCS IV). See Table 6 for summary of changes to function over time.
Summary of changes to function over time by EDACS level for children with CP (
97)
Summary of changes to function over time by EDACS level for children with CP (
There was a statistically significant moderate positive correlation between EDACS levels and all FCSs (ranging from 0.53–0.75) [34]: the highest associations were between someone’s ability to use intelligible speech and their eating and drinking ability and their ability to use their hands and eating and drinking ability. See Table 7 for associations between EDACS levels and other areas of function.
Associations between children’s EDACS levels and levels of other Functional Classification Systems using Kendall’s tau b (
) [34]
Associations between children’s EDACS levels and levels of other Functional Classification Systems using Kendall’s tau b (
Children with the most limitations to eating and drinking were the most dependent upon enteral nutrition: all 16 children classified as EDACS V received enteral nutrition/hydration; 26/36 children classified as EDACS IV received some form of enteral nutrition; 6 children classified as EDACS I, II or III received some form of enteral nutrition/hydration. Enteral nutrition was used to address concerns linked to: aspiration; sub-optimal hydration and nutrition; and in some instances behavioural issues. The presence of a gastrostomy did not indicate unsafe swallow.
Case study 1: Female (GMFCS I, MACS I, VSS III, CFCS III, EDACS IV at TP1). EDACS level changed from Level IV to Level III between ages 3 and 6 years as she learnt skills to bite and chew soft lumps of food, and drink thin fluids.
Case study 2: Male (GMFCS V, MACS V, VSS IV, CFCS V, EDACS IV at TP1). EDACS level changed from Level IV to V between 17 and 19 years. He experienced progressive scoliosis in adolescence and other orthopaedic challenges. He also experienced a series of chest infections prompting a videofluoroscopic investigation of swallowing (VFSS) which revealed aspiration of food and fluids when eating and drinking. He needed to rely solely on enteral feeding for nutrition and hydration.
Case study 3: Male (GMFCS IV, MACS IV, VSS IV, CFCS IV, EDACS III at TP1). EDACS level changed from III to IV between 12 and 14 yrs. Silent aspiration was demonstrated on VFSS linked to strong dystonic spasms affecting posture and respiratory control. The risk of aspiration during eating and drinking was reduced by modification of food/fluid textures with increased opportunities to exercise and change position throughout the day.
Case study 4: Female (GMFCS III, MACS III, VSS III, CFCS III, EDACS III at TP1). EDACS III remained stable from age 5 yrs to 14 yrs. Concerns about weight were linked to her limited inefficient intake of food at age 5; at age 14 she managed larger volumes of food with no concerns about her weight gain, although EDACS level remained the same.
Case study 5: Male (GMFCS IV, MACS IV, VSS IV, CFCS III, EDACS IV at TP1). EDACS IV remained stable over time with concern about lack of weight gain linked to limited oral intake; introduction of gastrostomy age 16 yrs led to a gradual loss of interest in eating/drinking although he was always offered food and drink.
Discussion
EDACS is a member of the family of functional classification systems for people with CP, which includes the GMFCS, MACS and CFCS [1]. The application of the GMFCS and MACS in retrospective [24] and prospective [23, 25, 26] studies provides strong evidence for their discriminative and predictive validity. The discriminative and predictive validity of EDACS requires further investigation. This study is the first to investigate the stability of EDACS levels over time for a group of children and young people with CP, providing some preliminary findings to inform future research. Demonstrating stability of EDACS levels over time is the first step in the process of building the case for its use prognostically. EDACS has potential to provide a map for health professionals working with children with CP and their families to consider likely future outcomes and limits to change.
The different levels of EDACS make clinical sense as a way to describe the eating and drinking abilities of children and young people with CP in both clinical and research contexts. It has been highly recommended as a research tool to describe the characteristics of a study population [38, 39]. Important clinical information about children’s usual eating and drinking performance at mealtimes can be reliably captured and shared with other health professionals in order to improve treatment and management, including the prevention of respiratory harm [12, 39]. It can form the basis of conversations with parents about their children’s abilities and a context within which to identify risks associated with eating and drinking along with options to manage these in different settings. However, the full potential of EDACS to inform clinical practice has yet to be exploited.
This study demonstrates the reliability and stability of EDACS when applied retrospectively using case records supporting its use in clinical and research contexts. Speech and language therapists were able to consistently assign EDACS levels retrospectively from case records. The reliability of the conversion of qualitative clinical data into ordinal scale data by different raters was tested at the initial time point, and over all time points. This study supports the proposition that a child’s eating and drinking ability would remain at the same EDACS level over time. If change in eating and drinking ability occurs for some individuals at the margins between levels, this is likely to be by one level only.
The retrospective application of EDACS in this study reveals changes to eating and drinking abilities that sometimes occur in adolescence. Experienced clinicians anecdotally report changes to eating and drinking ability associated with ageing [37]; the lack of a measurement tool suitable for use in epidemiological studies has hampered the collection of such evidence. Each case study illustrates the stability of eating and drinking ability defined by EDACS, including limits to change by one level, where it occurs. Case 1 illustrates a change of EDACS level by one level with learning of new skills. Cases 2 and 3 show increasing limitations to eating and drinking ability associated with adolescence, orthopaedic and postural changes. Closer examination of cases where EDACS levels remain stable, reveal changes to the extent to which someone makes use of underlying eating and drinking abilities (Cases 4 and 5).
The use of EDACS in combination with other FCSs communicates a helpful summary about a child’s function to others including the wider health care team. The moderate association between EDACS and other FCSs is evidence of discriminative construct validity: it measures aspects of function which are connected to but distinct from other aspects of function. The GMFCS is used as a measure of severity of CP, and has been used to estimate life expectancy and risks to health associated with unsafe eating [35, 36]. However, the GMFCS does not discriminate between those children whose eating and drinking is safe and efficient and those at risk of choking or of aspiration. The ability to use speech (VSS) is most closely related to someone’s eating and drinking ability (EDACS). However, the relationship is not strong enough to use VSS to predict mealtime safety and efficiency. The relationship between MACS and CFCS and EDACS levels also show only a moderate positive correlation. Each of the FCSs used in this study measures distinct aspects of someone’s day-to-day function and none can be used as proxy measures of eating and drinking ability. Similarly, the presence of a feeding tube cannot be used as a proxy measure of unsafe swallowing.
The study population demonstrated the full range of eating and drinking ability captured by EDACS level I to V; in contrast, the sitting, standing and walking ability of the majority of children would be classified as GMFCS III-V. The population represents children who experience the greatest limitations to function as a result of CP. The clinical impetus to develop EDACS arose from the acknowledged need to consider eating and drinking ability as a separate aspect of functioning [19].
There are a number of limitations to this study because it is based on the retrospective examination of case records of a clinical population accessing multi-professional healthcare in a community setting.
The collection of retrospective data is limited by the quality of historical records. Some case records contained limited information about eating and drinking abilities. The gaps between time points were determined by availability of case record data rather than by pre-determined ages. Consequently, there is variation between time points for each case. The earlier case records lacked the consistent format of later records, reflected in the lower reliability value across Time Point 1 between SaLTs 1 and 2. The case note materials could only be accessed by SaLTs who were members of the clinical team. All three SaLTs had worked at the specialist centre for 10 or more years. They each knew some of the children included in the study and were sometimes familiar with the details of individual children’s eating and drinking; this may have had an impact on how they assigned EDACS levels from case records. All ratings by SaLTs were undertaken independently of one another. SaLT2 assigned a level to each child only once and was blind to EDACS levels assigned by SaLT1 and SaLT3. SaLT1 and SaLT3 assigned EDACS levels with knowledge of previous EDACS level they had each assigned to that individual.
The strength of the study is that it provides new insights into the eating and drinking abilities of children with CP over six or more years. It captures changes in eating and drinking ability associated with adolescence.
Like the other FCSs, EDACS provides ordinal descriptions of function that is not suitable for use as an outcome measure in the context of therapeutic intervention [1]. All children within the study received some input from therapists as part of ongoing health care and habilitation. Twenty-four hour postural management programmes [40] and multi-professional patient-centred healthcare typify the interventions received by each child. Therapy was targeted to support safe and efficient mealtime management and participation, and to optimise available movements associated with eating and drinking. In some cases, therapy was specifically targeted to improve eating and drinking function. This study does not identify the impact of therapeutic intervention on children’s eating and drinking abilities. Whilst an intervention would not be expected to change a classification level, at the outset of this study, it was not clear that EDACS would perform in the same way as the GMFCS and MACS over time.
The next step in assessing the stability of EDACS would be a prospective cohort study charting the eating and drinking ability of children with CP over time to evaluate the predictive validity of EDACS.
Footnotes
Acknowledgments
Funding for the study was awarded by the British Academy of Childhood Disability and Royal College of Paediatrics and Child Health Polani Prize, March 2015. Ethical approval for the research was conferred by NHS Health Research Authority REC reference 16/LO/0344. Diane Sellers has received a research grant and honorarium funding from Nutricia Advanced Medical Nutrition UK 2017–2019.
Conflict of interest
All other authors have no conflict of interest to report.
