Cerebral Palsy (CP) has an estimated prevalence of moderate and severe cases of 1.5 to 2.5 children per 1000 live births [1
]. Additionally, an increasing rate of children with CP has been observed due to the growing number of premature infants’ survival [2
]. Considering these figures, CP can be considered as one of the most common physical childhood disorders with lifelong impact [3
CP comprises a group of neurodevelopmental disorders of movement and posture, holding four criteria: (i) presence of a disorder of movement or posture; (ii) consequence of a cerebral abnormality; (iii) early developmental disorder (prenatal, perinatal, or postnatal development of the brain); and (iv) movement impairment stable in time associated to a non-progressive cerebral abnormality [6
]. As a broad clinical entity, the motor disorder is likely to vary in its features and associated severity [8
]. For example, CP motor impairment could be classified according to the topography of the lesion (e.g., hemiplegia—impairment in the upper and lower extremity of one side of the body) and type of the disorder (e.g., spastic—muscle spasticity) [8
]. Furthermore, motor impairment has frequently been associated with secondary disturbances, such as those in sensation, perception, cognition, communication, and behavior, as well as the presence of epilepsy, disequilibrium [10
], and secondary musculoskeletal problems [11
]. Importantly, people with CP present a predisposition to show working memory (WM) and executive function (EF) deficits [12
] with a clear impact on activities of daily living (ADL) and on the learning process [14
]. Specifically, regarding the latter, the risk that children with CP have to present learning disabilities is beyond the presence of cognitive impairment and highly related with EF deficits, since children with a normative cognitive level can present specific learning difficulties (frequently in mathematics and reading) [12
EFs are conceptualized as a family of complex and high cognitive processes involved in human cognition [16
]. Despite the multiplicity of definitions, EFs are commonly defined as a set of attention-regulation skills, responsible for goal-directed behavior, influenced by previous learning, and with repercussions in future actions and plans [19
Unlike automatic processes, EFs require effort and the use of self-regulation (SR) competences [20
]. Specifically, following Diamond [20
] EFs show two forms as follows: Core EFs (i.e., inhibition interference control, working memory, and cognitive flexibility) and High Order EFs (i.e., reasoning, problem solving, and planning). Literature suggests that EF and SR overlap, which may be explained by the activation of common prefrontal neural networks [19
]. According to Diamond [20
], SR behavior is highly related to the action of inhibitory control, which contributes to the response inhibition, control of attention, and to a functional level of emotional, motivational, and cognitive arousal. Moreover, EFs and SR behaviors are only activated by will-driven processes to achieve goals [19
]. In fact, growing evidence suggests that EFs improve school achievement and vice-versa [19
]. The EF skills (e.g., planning, anticipatory behavior) play a relevant role in SR behavior and are, theoretically, associated to School Engagement (SE) (e.g., follow rules; focus on the task; sustain attention). Children’s school trajectories show that, among other things, SR is positively associated with social competence, motivation, and SE [19
]. Furthermore, Bridgman [26
] argued that children’s SR skills and SE hold a strong relationship with school readiness and should start being promoted in kindergarten.
SE has been largely studied in recent literature and is considered the lighthouse guiding efforts to address major educational challenges [27
]. For example, SE has been considered a relevant framework to understand school dropout, disaffection, and low levels of academic achievement [24
]. Fredricks and colleagues [24
] proposed a model of SE that provides a relevant theoretical framework to the present study. According to these authors [24
], SE is a multidimensional and multifaceted construct comprising three interrelated but distinct dimensions: (i) behavioral engagement (BE); (ii) emotional engagement (EE); and (iii) cognitive engagement (CE). Behavioral engagement reflects the student’s involvement in school and social activities (intra or extracurricular). This SE component entails school attendance, fulfilment of schoolwork, and participation in class, school, and extracurricular activities [30
]. Emotional engagement refers to students’ feelings and affective reactions towards school [31
]. Students’ interest, happiness, and valuing of school activities reflects a positive emotional engagement [32
]. Emotional engagement is also related to school identification [31
] and feelings of belonging to the school community. Lastly, cognitive engagement refers to students’ efforts, will, and goals directed towards learning [24
]. Positive levels of cognitive engagement are associated with the use of SR strategies [24
]. In fact, student’s cognitive engagement draws on metacognitive strategies to manage time and effort allocated to tasks, which requires cognitive flexibility, persistence, and self-efficacy to cope with problem solving [34
Children with CP are especially prone to show poor SE. Due to motor limitations, children are likely to struggle to follow school routines and show limited participation in school activities [26
]. However, CP has been regarded as a mutable disorder, despite being a permanent developmental condition with persistent neurological prejudice [36
]. This changeable nature of CP opens an opportunity for educational interventions, in which “the earlier the better” is the rule of the thumb. Extant literature has highlighted the need to run SE promotional programs including the training of EFs and SR competences focused on ADL or on the school context to foster SE and autonomy [19
]. Grounded on these reasons, the present study aims to examine the efficacy of an educational program focused on the promotion of SE in children with CP.
“How do you know I’m on an adventure?” I asked curiously (Anastácio).
“Well, well. We are all engaged on an adventure. Whenever we challenge our limits, we are living an adventure. We may not recognize it, but it would be a shame because all the adventures are … an adventure” (Cucu) [33
] (p. 22).
The rationale to run this study is grounded on the: (i) predisposition of children with CP to show poor SE; (ii) the challenge launched by Piovesana and colleagues [38
] indicating the need to include EF training in the intervention program’s design; and (iii) work by Zelazo and colleagues (2013) [57
] highlighting the relevance of investigating SR associated with EF skills and its impact in school. All things considered, The Incredible Adventures of Anastácio, the Explorer
story-tool was designed with the purpose of promoting SE in children with CP through the training of EF and SR.
Current findings indicate that participants enrolled in the program improved in BE, EE, and CE. Data also show that time has a significant main effect in each of the dimensions of SE, but the variable sex does not show differences in the three SE dimensions analyzed. These results, despite preliminary, are very encouraging and indicate that the Anastácio program could be considered a valuable tool to promote SE among children with CP. To the best of our knowledge there are no publications reporting educational interventions focused on promoting SR competences and SE in children with CP. Still, according to the systematic review of Novak and colleagues [4
], educative interventions with the purpose of improving behavior and social skills (e.g., conductive education) have shown low results with this specific population. Current data are consistent with extent research using story tools to promote SRL [48
]. Reasons for the findings of the current study are believed to be twofold: the use of the narrative as a tool to deliver SRL strategies and the intervention group dynamics. The story tells the adventure of a boy struggling to overcome obstacles and attain his goals. For example, setting personal goals, understanding the nature of fear when coping with challenges, the importance of asking for, and accepting, the help offered by others to overcome obstacles, and ways to approach and solve problems, were topics analyzed in the sessions. Participants were invited to discuss the story and reflect on their own behavior. In these sessions, children met and worked with peers with a similar clinical condition, but with distinct symptoms and functioning (e.g., children with motor and communication limitations). Understanding a wide variety of challenges faced by other children with CP, and the actions needed to address them, may have helped each child to cope with their own problems and improve their competences, for example, in ADL challenges. Children were invited to reflect on Anastácio’s responses to the challenges of the adventure compared to their own responses to their daily adventures (e.g., doing the therapy homework; studying at home; interact with their peers). While discussing the story and reflecting on their own behaviors in the sessions, children were encouraged to complete the tasks of the program and to help each other (e.g., participant A15 struggled to write the task assignments and, alternately, participants helped him finish the tasks during the session). Working with other children with CP and discussing their difficulties to cope with ADL challenges is likely to have helped them reflect metacognitively and improve their efforts to attain their own goals. Our findings are consistent with those by Kim and colleagues (2014) [60
] which investigated the effects of action observation of physical training on the actions of the observer (i.e., children with CP). Findings showed positive effects on the upper extremity functions in children with CP as a result of having observed physical training.
The communitarian approach developed in the sessions of the current program may have promoted the feelings of belonging and empathy within the group and, consequently, their SE. This result is consistent with the work by Lovitt and colleagues (1999) [61
], which highlighted the positive effects of integrating children with special educational needs in the school community. Participants’ high improvement in EE may suggest the need to build safe environments focused on enabling children with CP to develop their potentialities.
Acknowledging these recommendations and considering the changeable nature of CP [36
], the Anastácio intervention program intentionally promoted SRL competences and EF. The development of children’s EF is modulated by experience; in fact, the neural circuit is characterized by high plasticity and is likely to change throughout children’s development [19
]. Therefore, children with CP should be supported with interventions focused on the training of strategies related with EF processes [22
Literature has reported that the three dimensions of SE are interrelated and are likely to influence each other on positive loops [62
]. Our findings are consistent with prior research stating that the quality of relationships, the approval by peers and teachers, and students’ sense of belonging at school, may contribute to the development of CE, which may lead to an increase in BE [63
]. This is likely to be particularly important for students with special needs due to their vulnerability and low engagement [61
]. However, future research should consider further analyzing the role of these aspects in the SE of children with CP. In fact, the specificity and complexity of some cases require complementary measures and a deep analysis (e.g., case study) to understand which factors may influence the development of learning strategies knowledge and of its use.
These findings stress the need to tailor interventions addressing each child’s own needs. Moreover, the analysis focused on the individual trajectories helped understand the importance of using measures sensitive to CP’s particularities. In fact, each child faces diverse challenges that the self-reports may not be able to capture. Future investigations may consider the need to use complementary instruments sensitive to the particularities of the CP condition. For example, repeated observational measures could provide an accurate picture of each child’s path during the intervention.
Other limitations may have influenced the findings reported and should be acknowledged. For example, SE is a complex process and to ensure the trustworthiness of the data, researchers should consider using observational measures to help capture all the dimensions of SE. Additionally, the number of participants is limited. Notwithstanding, it was a challenge to gather this number in regular sessions. These children have several health problems associated with the CP clinical condition which limits their regular participation in the sessions; in addition, they have busy schedules full with school activities and therapies. Future research should consider delivering this program using an online format to best meet the needs of children with CP. Our findings, despite preliminary and in need of further research, show that the training of SR and EF offered participants various opportunities to reflect on their own behavior in different contexts (e.g., school, home, therapy), to make choices, to assess the outcomes and assume responsibility for them, and, ultimately, to grow. This was a very important experience for all children; parents reported several anecdotal episodes confirming this proposition. However, to become autonomous and responsible, children with CP need the help of the educators that accompany them on a daily basis. Parents, rehabilitation therapists, and teachers, all play an important role in the promotion of the autonomy and SR of children with CP; thus, researchers may consider enrolling these educators in future investigative designs. For example, schools could consider training teachers to identify and meet the educational needs of their students with CP. To achieve this goal, data collection could be extended to parents, therapists, and teachers to capture the transfer of the acquired competences to ADL (e.g., planning daily study, setting suitable goals), and train them on how to improve children’s SRL strategies and EF. Equipping educators with a set of SRL and EF strategies is expected to further extend the impact of interventions on SE, as the end of the Anastácio’s story suggests: “Mom do you think I can do it too [attain self-set goals]?”/“What part of the story do you want to read again, son?” [33