Application of Low-Intensity Modified Constraint-Induced Movement Therapy to Improve the Affected Upper Limb Functionality in Infantile Hemiplegia with Moderate Manual Ability: Case Series

children Article Application of Low-Intensity Modified Constraint-Induced Movement Therapy to Improve the Affected Upper Limb Functionality in Infantile Hemiplegia with Moderate Manual Ability: Case Series Rocío Palomo-Carrión1, Rita-Pilar Romero-Galisteo 2,* , Elena Pinero-Pinto 3,* , Purificación López-Muñoz 1, Helena Romay-Barrero 1and Francisco García-Muro San José4 1Department of Nursery, Physiotherapy and Occupational Therapy, Faculty of Physiotherapy, University of Castilla-La Mancha, 13001 Ciudad Real, Spain; [email protected] (R.P.-C.); [email protected] (P.L.-M.); [email protected] (H.R.-B.) 2Department of Physiotherapy, Faculty of Science Health, University of Málaga, 29016 Málaga, Spain 3Department of Physical Therapy, Faculty of Nursery, Physiotherapy and Podiatry, University of Seville, 41004 Sevilla, Spain 4Department of Physiotherapy, Faculty of Medicine, University San Pablo-CEU, 28003 Madrid, Spain; fgarciamur[email protected] *Correspondence: rpromer[email protected] (R.-P.R.-G.); epiner[email protected] (E.P.-P.); Tel.: +34-95-1952862 (R.-P.R.-G.) Received: 15 July 2020; Accepted: 2 September 2020; Published: 4 September 2020   Abstract: Objective: To assess the functionality of the affected upper limb in children diagnosed with hemiplegia aged between 4 and 8 years after applying low-intensity modified Constraint-Induced Movement Therapy (mCIMT). Methods: Prospective case series study. A mCIMT protocol was applied for five weeks, with two hours of containment per day. The study variables were quality of movement of the upper limb, spontaneous use, participation of the affected upper limb in activities of daily living, dynamic joint position, grasp–release action, grasp strength, supination and extension elbow movements. Four measurements were performed, using the quality of upper extremity test (QUEST) scale, the Shriners Hospital for Children Upper Extremity Evaluation (SHUEE) Evaluation, a hand dynamometer and a goniometer. Results: The sample was composed of eight children with moderate manual ability. Statistically significant differences were detected in all the studied variables (p<0.05) between the pre-treatment and post–treatment results (Week 0–Week 5), except for upper limb dressing, putting on splints and buttoning up. In the first week, the changes were statistically significant, except for protective extension, grasp strength, grasp–release and all functional variables (level of functionality and participation of the patient’s upper limbs) in the SHUEE Evaluation (p>0.05). The greatest increase occurred in spontaneous use from Assessment 1 to Assessment 4 (p=0.01), reaching 88.87% active participation in bimanual tasks. The quality of movement of the upper limb exhibited a significant value due to the increase in dissociated movements and grasp (p=0.01). Conclusion: A low dose (50 h) of mCIMT increased the functionality of children diagnosed with congenital hemiplegia between 4 and 8 years of age with moderate manual ability. Keywords: family;infantilehemiplegia; modifiedConstraint-InducedMovementTherapy; physicaltherapy modalities; upper extremity 1. Introduction Infantile hemiplegia is a subtype of infantile cerebral palsy, characterized by the affectation of one of the hemibodies as a consequence of brain injury. Its prevalence is 1 case per 1300 live births [ 1 ]. Children 2020,7, 127; doi:10.3390/children7090127 www.mdpi.com/journal/children Children 2020,7, 127 2 of 20 There is more affectation of the upper limb than the lower limb due to the alteration of the corticospinal tract. The affected hand has a deficit in proprioception and tactile perception, which hinders fine motor skills, generally those of the fingers and the strength exerted by them [ 2 ]. Sensory abnormalities, weak grasp and loss of manual ability (fine movements) may appear, specifically in the fingers, with slower movements, poorer coordination and longer phases associated with mirror movements. This leads to a decrease in the use of the affected hand and often interferes with the manual ability of the healthy upper limb [3]. From early childhood, children with hemiplegia, even the least affected, use their healthy hand as the dominant hand in all tasks. Therefore, they learn “not to use” their affected upper limb, which is known as developmental disregard [ 4 ]. This “non-use” of the affected upper limb produces an increase in muscle tone in the affected segment, poor motor control, decreased active and passive range of motion, generalized weakness and delayed musculoskeletal maturation. The non-use affectation is caused by neural dysfunction as a result of brain injury. This neuronal alteration [ 5 ] can be improved through the activation of certain brain areas that remained inactive after the brain lesion and also through experience and learning (trial–error). Thus, in order to improve the affected upper limb “non-use”, Constraint-Induced Movement Therapy (CIMT) is used [ 6 , 7 ], which consists of constraining the healthy upper limb with a whole or partial containment (glove), thus promoting the use of the affected upper limb in activities of daily living. The programmed tasks integrate the repetition of the motor action with a variety of exercises. The use of CIMT has spread in recent years among physiotherapists and occupational therapists, due to the large number of studies that support the effectiveness of this intervention compared to traditional interventions that do not restrict the use of the healthy side [ 8 ]. However, in its original conception, two premises had to be met: the restriction of the less-affected upper limb and the application of an intensive treatment applied in a structured way to the upper limbs [ 9 ]. Different variants of CIMT have emerged over the years, under the term “modified Constraint-Induced Movement Therapy” (mCIMT). Likewise, there are a variety of protocols used for CIMT, in which different containment systems are proposed [ 10 – 15 ] in which it is the dose that varies [ 6 , 12 , 14 , 16 – 27 ]. In this line, one of the modifications widely used in pediatrics is the one based on mCIMT, which constrains the healthy upper limb for less than 3 h [ 23 , 28 – 30 ]. McConnell et al. [ 31 ] found that a less intensive treatment (63 h of treatment over 21 days) produced similar benefits compared to a more intensive approach (126 h of treatment over 21 days). Functional gains may be feasible for some children with a less intense program adjusted to 20 h of therapy in more than two consecutive weeks [ 31 ]. According to Schweighofer et al. [ 32 ], the existence of a “functional threshold” would be necessary for the maintenance of functionality after therapy, below which the use of the upper limb decreases while the benefits to the individual remain above such threshold. It would be useful to determine the specific doses of therapy in each patient. McConnell et al. [ 31 ] applied mCIMT for 2 weeks with two hours of dose per day in a clinical setting, designed for children aged 8–15 years, and the therapist increased the dose to continue with the use of the affected upper limb for 30 min at home. The children executed 20 h of total dose with functional changes. We proposed to apply 50 h of total dose, with the same distribution per day according to this study, although increasing the dose by 30 h, since the participants in our study were younger than those in McConnell et al. [ 31 ] and the therapy was performed at home, thus the children and their families needed more time to obtain significant results. Thus, we decided to assess the children at Week 2 of treatment (20 h) in order to verify whether the changes would be the same at Week 5 (after treatment with a total dose of 50 h). The systematic review [ 33 ] included 31 papers, each of which applied different doses per day, total doses, measurement tools, etc. All 31 studies were compared, with the main difficulty being that the children had different manual ability levels, and it would be ideal to know the correct dose for each level. In this systematic review, the manual ability levels were assessed with the Manual Classification System, MACS; some studies show Levels I–III, I–IV or I–V [ 34 ]. We could consider a moderate hand ability level for children classified as Level I–II in MACS. These levels reflect that the children are independent in the execution of activities using one or Children 2020,7, 127 3 of 20 two hands or including compensation strategies (neck, trunk, mouth, etc.) to complete the bimanual task; they also show that the movement restrictions do not impede the complete use of the affected upper limb. However, most of the published studies include functional activities in their treatment proposals. To our knowledge, a few of them contemplate an ecological vision of human development as initially proposed by Bronfenbrenner [ 35 ]. According to this perspective, it is crucial to incorporate the principles of therapy to the environment in which the child develops, which is essential to ensure the long-term persistence of the achieved results [ 36 ]. From the ecological point of view, the evolution of the child is understood as a process of progressive differentiation of the activities that he/she carries out, his/her role and his/her interactions with the environment. The interactions and transactions established between the child and the elements of his/her environment are very important, especially with his/her parents. One of the latest reviews on the use of CIMT [ 8 ] showed that the risk of bias of the analyzed clinical trials was between moderate and high; therefore, a new randomized controlled trial is proposed in this work, whose mainobjectivewastoanalyzetheeffectivenessofthe use of CIMT by modifying theapplied dose. Moreover, the ecological perspective of development was considered, introducing functional tasks that children usually carry out in their usual environment. To our knowledge, no study has been published in Spain that combines a low dose of treatment with the performance of functional activities at home with the parents. For this reason, we consider assessing the functionality of the affected upper limb in children diagnosed with congenital hemiplegia with moderate manual ability between 4 and 8 years of age after applying low-intensity modified constraint-induced movement therapy (50 h) at home 2. Materials and Methods This is a case series, prospective and longitudinal study with non-probability sampling (clinical.gov NCT02178371). The study was approved (060-13) by the ethics committee of the CEU-San Pablo University of Madrid in accordance with the Declaration of Helsinki of the World Medical Association. Before initiating the study, an informed consent form was given to the children’s families to participate, which guaranteed the right to withdraw from the study at any time, if required by the participants. The inclusion criteria were a medical diagnosis of left/right congenital infantile hemiplegia, age between 4 and 8 years, lack of activity of the affected upper limb, ability to exceed 10 ◦ extension in the metacarpophalangeal and interphalangeal joint, ability to complete a 20 ◦ extension of the wrist of the affected upper limb, adequate cognitive development to understand the verbal orders given for the execution of tasks and cooperation in their execution. In the same way, the exclusion criteria were visual problems that prevented the individual from carrying out the intervention, suffering from significant balance disturbances that put the child at risk of falling as a consequence of having the healthy upper limb contained, presenting uncontrolled epilepsy and having received botulinum toxin within 6 months prior to the intervention. All the children were selected according to the inclusion criteria by their rehabilitation doctor of the “Virgen de la Salud” Hospital in Toledo for the execution of the therapy. 2.1. Intervention Method The study was carried out over a period of 5 weeks of treatment, containing the healthy upper limb for 2 h per day [ 11 , 13 ] (not continuously) from Monday to Friday. McConnell et al. [ 31 ] and Al-Oraibi 2011 [ 21 ] used mCIMT for 2 h per day, obtaining positive results, and the children wore the hand containment for 96% of the total dose [ 13 ]. Thus, in our study, the children were requested to perform the structured activities for two non-consecutive hours, with the aim of increasing the adherence and avoiding great physical effort. The tasks were separated by at least 30 min of rest to allow the children to concentrate on the next activities, perform properly and stay motivated. The families were advised to set aside one hour in the early afternoon and another hour in the late afternoon to ensure that the child was attentive, frustration-free and effort-tolerant. The families were also instructed to run a Children 2020,7, 127 4 of 20 full hour, repeating the activity and designing a story, in which the child was the protagonist and the activity was an enjoyable game to complete. Each of the proposed hours could be divided into periods of 30 min to improve the child’s attention and motivation in case the child was tired during the treatment. We chose this minimum period of time, since it is the smallest dose of mCIMT used to treat babies aged 3 to 8 months with babyCIMT, in order to ensure sustained attention to activity [ 37 ]. This minimum dose of 30 min was used also in a systematic review [ 11 ]. When dealing with children from 4 to 8 years old, attention is greater and maintained for a longer time, which allows establishing a continuity of 1 h of intervention. The intervention was carried out by the family at home. Previously, an informative meeting was held with the parents, in which all the details of the study were explained to them and a program of unimanual activities was given to them to be executed with the affected upper limb every treatment week (Table 1). The activities were programmed to work different movements that were limited in the affected upper limb: shoulder flexion, elbow extension, forearm supination, wrist extension and grasp. Each activity was repeated for around 15 min to obtain a lesson about a functional strategy to use in their usual activities. Each intervention hour was completed with four activities (Table 1), which were repeated in the second hour to increase the movement learning and the possibility to modify and obtain new functional strategies to perform the activity. The same activities were performed every week (first and second hours from Monday to Friday) to improve the movement (shoulder flexion, forearm supination, elbow extension, wrist extension and grasp–release). Table 1. Example of “modified Constraint-Induced Movement Therapy” (mCIMT) designed tasks. Movements to Work in the Affected Upper Limb Examples of mCIMT Designed Tasks Shoulder flexion and elbow extension •Put stickers at different heights on the wall and try to cover them with the affected hand. •Take a small and light ball and try to throw it higher towards a target. •The parents will put a cardboard or continuous paper stuck on the wall; using finger paint, the child will try to draw a picture or put his/her hand with paint on the paper. •The parents will throw balloons or bubbles, which the child will try to hit with his/her hand. •The parents will give the child a small and light ball, which he/she will try to throw higher and higher or towards a target. Forearm supination •Put stickers on the palm of the affected hand or forearm. •Fishing game. •The parents will place a light object on the palm of the affected hand (for example, a colored pompom) and the child will keep it for a while. •The child will use the affected hand to remove objects stuck on his/her t-shirt (at the level of the abdomen). •Playing the trumpet or other instruments . . . Wrist extension •The parents will push cardboard boxes or other elements and the child will try to throw them offthe table. •The child will smash packing paper, balls and/or soft objects with the palm of his/her hand. •The child must roll a ball, bottle. •The child must hit a piano or a drum placed vertically. •The child will remove pieces fallen from the wall. Children 2020,7, 127 5 of 20 Table 1. Cont. Movements to Work in the Affected Upper Limb Examples of mCIMT Designed Tasks Grasp–release •Grasp, hold and transfer light and long objects. •Grasp, hold and transfer heavy, long and rough objects. •Grasp, hold and transfer rough, light and spherical objects. •Grasp, hold and transfer rough, heavy and spherical objects. •Grasp, hold and transfer different objects with a variety of the previous characteristics. The parents were instructed to correctly carry out the intervention avoiding possible errors during the treatment protocol. The weekly exercises, the containment technique and its correct use were also taught to them. The treatment was only initiated when the families and children were confident about it. The family and therapist met every week to assess the activities and make adjustments if necessary. Thecontainmentappliedwaspartial,suchasagloveorabandage[ 21 ]. Inthisway,manipulationwith the healthy hand was prevented and the wrist and elbow joints were also free to allow the child to react effectively to an external disturbance (Figure 1). Children 2020, 7, x FOR PEER REVIEW 5 of 20 • Grasp, hold and transfer rough, light and spherical objects. • Grasp, hold and transfer rough, heavy and spherical objects. • Grasp, hold and transfer different objects with a variety of the previous characteristics. The parents were instructed to correctly carry out the intervention avoiding possible errors during the treatment protocol. The weekly exercises, the containment technique and its correct use were also taught to them. The treatment was only initiated when the families and children were confident about it. The family and therapist met every week to assess the activities and make adjustments if necessary. The containment applied was partial, such as a glove or a bandage [21]. In this way, manipulation with the healthy hand was prevented and the wrist and elbow joints were also free to allow the child to react effectively to an external disturbance (Figure 1). Figure 1. Child with left hemiplegia wearing a bandage as a partial containment in the right hand (dominant hand). In this task, the child is working the grasp–release action. Each week, the therapist and the parents of the participants had a meeting to follow-up the tasks and solve problems. The follow-up with the families was conducted online, reviewing all the activities and modifying those that were too difficult for the child, maintaining great therapist– family–child feedback. The activities were designed according to the limitations of the children and with simplicity in order for the latter to be able to perform them. In the follow-up, the activities that were not adequate were valued and changed for others considering the interest of the child. The parents were asked to fill out a table with the performed tasks, completing these with photographs and videos of the tasks carried out daily and the execution time to complete 96% of total dose [13], in this case, 48 h (total dose: 50 h) (Figure 2). Figure 1. Child with left hemiplegia wearing a bandage as a partial containment in the right hand (dominant hand). In this task, the child is working the grasp–release action. Each week, the therapist and the parents of the participants had a meeting to follow-up the tasks and solve problems. The follow-up with the families was conducted online, reviewing all the activities and modifying those that were too difficult for the child, maintaining great therapist–family–child feedback. The activities were designed according to the limitations of the children and with simplicity in order for the latter to be able to perform them. In the follow-up, the activities that were not adequate were valued and changed for others considering the interest of the child. The parents were asked to fill out a table with the performed tasks, completing these with photographs and videos of the tasks carried out daily and the execution time to complete 96% of total dose [13], in this case, 48 h (total dose: 50 h) (Figure 2). Children 2020,7, 127 6 of 20 Children 2020, 7, x FOR PEER REVIEW 6 of 20 Figure 2. Weekly follow-up for the families. 2.2. Data Collection Four assessments (Basal (T0, before therapy), T1 (Assessment 2, after Week 1 of therapy), T2 (Assessment 3, after Week 2 of therapy) and T3 (Assessment 4, after Week 5 of therapy)) were performed to measure the study variables and to compare the results before, during and after the intervention (Figure 3). Figure 3. Representation of the assessment number in the 5 weeks of treatment (mCIMT). 2.2.1. Studied Variables and Measuring Instruments Quality of Movement and Bimanual Dexterity of Both Upper Extremities The quality of upper extremity test (QUEST) [38–40] was used, validated for children with neuromotor dysfunction with spasticity from 18 months to 8 years of age. It provides a numerical value that is obtained from the mean of the percentages in 36 items distributed in four categories: dissociated movements, grasp, weight bearing and protective extension of both extremities. It takes a value from 0 to 100 and can be expressed in percentages (%). Active Extension of the Wrist and Active Supination of the Forearm in the Affected Upper Limb Both variables were measured with an arm goniometer [41], making three measurements for each variable and selecting the best result. The measurements were made with the child sitting. Wrist extension was measured with elbow flexion, with the child leaning on a table to decrease muscle tension and associated reactions. Supination was measured with the forearm close to the body, avoiding trunk compensations to gain greater joint width. Grasp Strength in the Affected Hand This was measured using a hand dynamometer [18] with a scale between 0 and 150 that expressed grasp strength in PSI (pound per square inch, 1 psi = 0.0703 kg/cm2). The test was performed with the child sitting on a chair in front of a table with the healthy hand under the table and the affected forearm resting on the table to give stability to the upper limb (Figure 4). The child Assessment 2 Basal mCIM T WEEK 1 Assessment 4 Assessment 3 mCIM T WEEK 2 mCIM T WEEK 3-5 Figure 2. Weekly follow-up for the families. 2.2. Data Collection Four assessments (Basal (T0, before therapy), T1 (Assessment 2, after Week 1 of therapy), T2 (Assessment 3, after Week 2 of therapy) and T3 (Assessment 4, after Week 5 of therapy)) were performed to measure the study variables and to compare the results before, during and after the intervention (Figure 3). Children 2020, 7, x FOR PEER REVIEW 6 of 20 Figure 2. Weekly follow-up for the families. 2.2. Data Collection Four assessments (Basal (T0, before therapy), T1 (Assessment 2, after Week 1 of therapy), T2 (Assessment 3, after Week 2 of therapy) and T3 (Assessment 4, after Week 5 of therapy)) were performed to measure the study variables and to compare the results before, during and after the intervention (Figure 3). Figure 3. Representation of the assessment number in the 5 weeks of treatment (mCIMT). 2.2.1. Studied Variables and Measuring Instruments Quality of Movement and Bimanual Dexterity of Both Upper Extremities The quality of upper extremity test (QUEST) [38–40] was used, validated for children with neuromotor dysfunction with spasticity from 18 months to 8 years of age. It provides a numerical value that is obtained from the mean of the percentages in 36 items distributed in four categories: dissociated movements, grasp, weight bearing and protective extension of both extremities. It takes a value from 0 to 100 and can be expressed in percentages (%). Active Extension of the Wrist and Active Supination of the Forearm in the Affected Upper Limb Both variables were measured with an arm goniometer [41], making three measurements for each variable and selecting the best result. The measurements were made with the child sitting. Wrist extension was measured with elbow flexion, with the child leaning on a table to decrease muscle tension and associated reactions. Supination was measured with the forearm close to the body, avoiding trunk compensations to gain greater joint width. Grasp Strength in the Affected Hand This was measured using a hand dynamometer [18] with a scale between 0 and 150 that expressed grasp strength in PSI (pound per square inch, 1 psi = 0.0703 kg/cm2). The test was performed with the child sitting on a chair in front of a table with the healthy hand under the table and the affected forearm resting on the table to give stability to the upper limb (Figure 4). The child Assessment 2 Basal mCIM T WEEK 1 Assessment 4 Assessment 3 mCIM T WEEK 2 mCIM T WEEK 3-5 Figure 3. Representation of the assessment number in the 5 weeks of treatment (mCIMT). 2.2.1. Studied Variables and Measuring Instruments Quality of Movement and Bimanual Dexterity of Both Upper Extremities The quality of upper extremity test (QUEST) [ 38 – 40 ] was used, validated for children with neuromotor dysfunction with spasticity from 18 months to 8 years of age. It provides a numerical value that is obtained from the mean of the percentages in 36 items distributed in four categories: dissociated movements, grasp, weight bearing and protective extension of both extremities. It takes a value from 0 to 100 and can be expressed in percentages (%). Active Extension of the Wrist and Active Supination of the Forearm in the Affected Upper Limb Both variables were measured with an arm goniometer [ 41 ], making three measurements for each variable and selecting the best result. The measurements were made with the child sitting. Wrist extension was measured with elbow flexion, with the child leaning on a table to decrease muscle tension and associated reactions. Supination was measured with the forearm close to the body, avoiding trunk compensations to gain greater joint width. Children 2020,7, 127 7 of 20 Grasp Strength in the Affected Hand This was measured using a hand dynamometer [ 18 ] with a scale between 0 and 150 that expressed grasp strength in PSI (pound per square inch, 1 psi =0.0703 kg/cm 2 ). The test was performed with the child sitting on a chair in front of a table with the healthy hand under the table and the affected forearm resting on the table to give stability to the upper limb (Figure 4). The child was asked to press the dynamometer with the affected hand (global grasp) as hard as possible to obtain the best measurement. Children 2020, 7, x FOR PEER REVIEW 7 of 20 was asked to press the dynamometer with the affected hand (global grasp) as hard as possible to obtain the best measurement. Figure 4. Grasp strength measurement with a hand dynamometer. Spontaneous Use, Dynamic Positioning of the Affected Upper Limb, Grasping and Releasing Action (Wrist Position in Neutral Flexion–Extension), and Level of Functionality and Integration of the Affected Upper Limb in Various Activities of Daily Living The Shriners Hospital for Children Upper Extremity Evaluation (SHUEE) [40,42] was used to obtain the values in the four measurements. This evaluation consists of videotaping the children while they execute a series of tasks to observe the functionality and the joint alignment of the affected upper limb, and it has been validated for use in children with hemiplegia aged between 3 and 18 years. The results are expressed in percentages, with 100% being the best result. The level of functionality and participation of the patient’s upper limbs was determined through the SHUEE assessment as dependent, assisted or independent. Activities of daily living, such as dressing upper limbs, dressing lower limbs, buttoning, putting on socks, putting on shoes, putting on splints and personal hygiene were assessed. 2.3. Statistical Analysis Statistical analyses were performed using [43]. Given the small sample size, we used nonparametric tests. First, a Friedman’s test was used to evaluate the existence of statistical significance for the assessments performed at different times in each variable. Subsequently, a Wilcoxon test (paired samples) was performed on those variables that presented statistical significance, in order to observe statistically significant differences between paired assessments. Significance was set at a pvalue of 0.05. The results are shown as medians and interquartile ranges (IQRs) with 95% confidence intervals. Those with a p-value < 0.05 were considered as significant values. The qualitative variable of “functionality” was turned into a quantitative variable, graduating it in 5 levels, from 0 = worst functionality to 4 = maximum functionality (Table 2). Table 2. Description of the functionality levels. Dependent Value 0. Needs help from an adult (does not perform the action). Assisted Value 1. Needs help from an adult (partially performs the action). Independent Value 2. Uses the healthy upper limb exclusively. Value 3. Uses the upper limb to provide stability. Value 4. Uses both upper limbs to execute the action. 3. Results Twenty-four children were recruited; 14 of them did not meet the inclusion criteria and two of them declined to participate. The final sample consisted of eight children (Figure 5), with 50% males and 50% females, diagnosed with congenital hemiplegia. Figure 4. Grasp strength measurement with a hand dynamometer. Spontaneous Use, Dynamic Positioning of the Affected Upper Limb, Grasping and Releasing Action (Wrist Position in Neutral Flexion–Extension), and Level of Functionality and Integration of the Affected Upper Limb in Various Activities of Daily Living The Shriners Hospital for Children Upper Extremity Evaluation (SHUEE) [ 40 , 42 ] was used to obtain the values in the four measurements. This evaluation consists of videotaping the children while they execute a series of tasks to observe the functionality and the joint alignment of the affected upper limb, and it has been validated for use in children with hemiplegia aged between 3 and 18 years. The results are expressed in percentages, with 100% being the best result. The level of functionality and participation of the patient’s upper limbs was determined through the SHUEE assessment as dependent, assisted or independent. Activities of daily living, such as dressing upper limbs, dressing lower limbs, buttoning, putting on socks, putting on shoes, putting on splints and personal hygiene were assessed. 2.3. Statistical Analysis Statistical analyses were performed using [ 43 ]. Given the small sample size, we used non-parametric tests. First, a Friedman’s test was used to evaluate the existence of statistical significance for the assessments performed at different times in each variable. Subsequently, a Wilcoxon test (paired samples) was performed on those variables that presented statistical significance, in order to observe statistically significant differences between paired assessments. Significance was set at a p-value of 0.05. The results are shown as medians and interquartile ranges (IQRs) with 95% confidence intervals. Those with a p-value <0.05 were considered as significant values. The qualitative variable of “functionality” was turned into a quantitative variable, graduating it in 5 levels, from 0 =worst functionality to 4 =maximum functionality (Table 2). Table 2. Description of the functionality levels. Dependent Value 0. Needs help from an adult (does not perform the action). Assisted Value 1. Needs help from an adult (partially performs the action). Independent Value 2. Uses the healthy upper limb exclusively. Value 3. Uses the upper limb to provide stability. Value 4. Uses both upper limbs to execute the action. Children 2020,7, 127 8 of 20 3. Results Twenty-four children were recruited; 14 of them did not meet the inclusion criteria and two of them declined to participate. The final sample consisted of eight children (Figure 5), with 50% males and 50% females, diagnosed with congenital hemiplegia. Children 2020, 7, x FOR PEER REVIEW 8 of 20 Figure 5. Flowchart of the sample selection. Of the entire sample, 62.5% had affectation of the left upper limb. The average age was 6 years, with a standard deviation of 1.77 years. After assessing the motor ability of the children, they were classified as Level I in the Gross Motor Function Classification System (GMFCS) [44] and Level II in the manual ability classification system (MACS) [34]. (Table 3) Table 3. Baseline characteristics of the participants. Sample Age Sex Gest. Week Lesion MACS Level GMFCS Level Hemiplegia Side Add. Impair Child 1 4 F Term Perinatal stroke II I Left No Child 2 5 M Term Perinatal stroke II I Right No Child 3 5 M Preterm Perinatal stroke II I Left No Child 4 5 M Preterm Perinatal stroke II I Left No Child 5 7 F Term Perinatal stroke II I Right Speech Child 6 6 F Term Perinatal stroke II I Right Speech Child 7 8 F Preterm Perinatal stroke II I Left Speech Child 8 8 M Term Perinatal stroke II I Left No Sex: F: Female; M: Male. Gest. Week: Gestational Week: preterm (>32 weeks and <36 weeks); term: >36 weeks; MACS: Manual Ability Classification System; GMFCS: Gross Motor Function Classification System. Add. Impair: Added impairment. Figure 5. Flowchart of the sample selection. Of the entire sample, 62.5% had affectation of the left upper limb. The average age was 6 years, with a standard deviation of 1.77 years. After assessing the motor ability of the children, they were classified as Level I in the Gross Motor Function Classification System (GMFCS) [ 44 ] and Level II in the manual ability classification system (MACS) [34]. (Table 3) Table 3. Baseline characteristics of the participants. Sample Age Sex Gest. Week Lesion MACS Level GMFCS Level Hemiplegia Side Add. Impair Child 1 4 F Term Perinatal stroke II I Left No Child 2 5 M Term Perinatal stroke II I Right No Child 3 5 M Preterm Perinatal stroke II I Left No Child 4 5 M Preterm Perinatal stroke II I Left No Child 5 7 F Term Perinatal stroke II I Right Speech Child 6 6 F Term Perinatal stroke II I Right Speech Child 7 8 F Preterm Perinatal stroke II I Left Speech Child 8 8 M Term Perinatal stroke II I Left No Sex: F: Female; M: Male. Gest. Week: Gestational Week: preterm (>32 weeks and <36 weeks); term: >36 weeks; MACS: Manual Ability Classification System; GMFCS: Gross Motor Function Classification System. Add. Impair: Added impairment. Children 2020,7, 127 9 of 20 3.1. Quality of Movement of the Upper Limb The total score for the quality of movement in the upper limbs exhibited an increase of 94.07%. All the variables that compose it, i.e., dissociated movements, grasp, weight bearing and protective extension, showed statistical significance (p ≤ 0.001) in the Friedman test. When the Wilcoxon test was applied, dissociated movements and grasp obtained significant changes for all pairwise comparisons with a p-value <0.03. However, no statistically significant differences were found for weight bearing between the second and third assessments (p=0.14), second and fourth assessments (p=0.10) or third and fourth assessments (p=0.11), nor for protective extension between the baseline and second assessments (p=0.07) or second and third assessments (p=0.07). The results are shown in Table 4. Table 4. Results and pairwise comparisons for the quality of movement in the upper limbs. Variables Results Friedman’s Test Wilcoxon’s Test Median (IQR) Statistical Significance pValue Pairwise Comparisons Statistical Significance pValue Quality of Movement in the Upper Limbs (Total Score) Baseline 74.16 (63.55, 83.00) 24.000 0.000 * Baseline–2nd assessment −2.521 0.012 * 2nd assessment 83.32 (77.18, 88.49) Baseline–3rd assessment −2.521 0.012 * 3rd assessment 88.70 (84.42, 91.42) Baseline–4th assessment −2.521 0.012 * 4th assessment 94.07 (90.32, 94.92) 2nd assessment– 3rd assessment −2.521 0.012 * 2nd assessment– 4th assessment −2.521 0.012 * 3rd assessment– 4th assessment −2.521 0.012 * Dissociated Movements Baseline 59.38 (53.90, 80.48) 23.538 0.000 * Baseline–2nd assessment −2.524 0.012 * 2nd assessment 75.00 (73.83, 82.82) Baseline–3rd assessment −2.521 0.012 * 3rd assessment 83.60 (81.24, 87.11) Baseline–4th assessment −2.521 0.012 * 4th assessment 89.84 (84.77, 91.79) 2nd assessment– 3rd assessment −2.201 0.028 * 2nd assessment– 4th assessment −2.524 0.012 * 3rd assessment– 4th assessment −2.527 0.012 * Grasp Baseline 62.97 (46.30, 76.86) 21.808 0.000 * Baseline–2nd assessment −2.533 0.011 * 2nd assessment 79.60 (59.26, 87.96) Baseline–3rd assessment −2.527 0.012 * 3rd assessment 87.03 (63.89, 91.67) Baseline–4th assessment −2.527 0.012 * 4th assessment 96.30 (75.00, 96.30) 2nd assessment– 3rd assessment −2.200 0.028 * 2nd assessment– 4th assessment −2.384 0.017 * 3rd assessment– 4th assessment −2.384 0.017 * Children 2020,7, 127 16 of 20 obtained between the baseline and fourth measurements of the variables of dissociated movements and grasp. Appreciable benefits were obtained in each measurement produced by the acquisition of a more corrected posture of the trunk, head and shoulders in the execution of the grasp activities, present from the second measurement (after a week of treatment with mCIMT). A dynamic joint position occurred in wrist and elbow extension, increasing the median value for wrist extension by 21 ◦ from the baseline to the fourth measurement. In addition, the value reached in the fourth measurement for the median was 81.50 ◦ in the active supination of the forearm, allowing for greater control and support of the body structures for the execution of dissociated movements, grasp, weight bearing and protective extension due to the improvement of both active movements. There was a favorable evolution in the dynamic joint position of the affected upper limb due to the gain of active degrees for movement restriction in extension and supination, with an increase in this variable of 10.42% at the end of the treatment. In this way, the activities proposed during the evaluation were performed with greater ease of movement, such as eating a cookie, touching the opposite ear, picking up coins, opening a bottle or throwing a large ball, which require the selective motor control of certain muscles. In comparison with the results obtained for the quality of movement of the affected upper limb in the present study, we highlight an investigation [ 52 ] on mCIMT, which showed positive results for the assessment of the quality of movement of motor skills (measured through the QUEST scale) using an intervention protocol of 3 weeks of treatment with an intensity of 6 h per day of restriction and repetitive work. This study demonstrated the effectiveness of the intervention, as it had a larger sample and a control group (18 children with hemiplegia, nine children in the experimental group and nine children in the control group). In 2011, a different study [ 53 ], conducted exclusively with a girl with hemiplegia, used mCIMT for one hour per day for two weeks. No significant results were obtained at the end of the treatment, which was thereby prolonged for one more week of intervention. This last assessment showed an increase in the overall percentage of total quality of movement (also measured by the QUEST scale), appreciable in activities that involve the affected upper limb. Thus, the protocol chosen for the intervention and the initial functionality of the patient are two important factors to take into account in increasing the results obtained in the measurements. When comparing the results obtained in the second assessment after 20 h of treatment with those obtained after the treatment (50 h), improvements were detected in the former (20 h), although the greatest improvements and relevant changes were identified in the latter (50 h). At the end of the treatment, the families observed these functional changes (daily activities assessed) at different natural moments. Thus, it would be necessary to apply 50 h of dose instead of 20 h in children aged 4–8 years to increase the affected upper limb functionality, since the manual ability and age would be factors to consider in the dose to apply. Limitations and Future Lines of Research As this is an uncontrolled trial [ 54 ] due to the absence of a control group, it cannot be guaranteed that the observed response (changes produced throughout the intervention with respect to the baseline situation) is exclusively due to the mCIMT protocol used, since other uncontrolled factors may also have had an influence on it. Therefore, the effectiveness of the mCIMT in increasing the functionality of the affected upper limb seen in the present study cannot be generalized to the population of children with hemiplegia, and thus, the fundamental utility of this study is descriptive. It is important to highlight the statistically significant results obtained in the different studied variables throughout the 5 weeks of the intervention (It can be seen at Figures S1–S11. Progression of different variables in the four assessments over 5 weeks of mCIMT). This suggests that, although there was no control group, it could be inferred that the changes obtained in the improvement of the functionality were due to the efficacy of the treatment, since it was a short period of time, where important differences were observed in the affected upper limb, which is unlikely to occur due to the maturation effect (learning and natural development of the child over time). In Spain, this is the first study to provide preliminary data on the use of mCIMT. Although the results should be interpreted with caution due to the small sample size, the development of this pilot Children 2020,7, 127 17 of 20 scheme is the first step for this type of therapy, which has already demonstrated its effectiveness in other contexts; therefore, it should be taken into account by therapist and researchers who develop their work with this type of children in Spain. This study leads us to open different lines of research, such as including a control group to assess the effectiveness of the treatment in the functionality of the affected upper limb, to verify whether the gains obtained after the intervention are maintained over time (6 months or one year after therapy), and to study the influence of age on the obtained results due to neuronal plasticity and active participation of the subject. Likewise, it paves the road for clinicians and researchers to develop new treatment proposals in Spain. 5. Conclusions Alow dose(50 h)ofmodified Constraint-InducedMovementTherapy canincreasethe functionality of children diagnosed with congenital hemiplegia between 4 and 8 years of age with moderate manual ability. Supplementary Materials: The following are available online at http://www.mdpi.com/2227-9067/7/9/127/s1. Supplementary figures: Figures S1–S11. Progression of different variables in the four assessments over 5 weeks of mCIMT. Author Contributions: Conceptualization, R.P.-C. and R.-P.R.-G.; methodology, E.P.-P.; software, F.G.-M.S.J.; validation, R.P.-C., P.L.-M. and H.R.-B.; formal analysis, E.P.-P.; investigation, R.P.-C.; data curation, P.L.-M.; writing—original draft preparation, R.-P.R.-G. and R.P.-C.; writing—review and editing, R.-P.R.-G.; visualization, F.G.-M.S.J.; supervision, F.-G.-M.S.J. All authors have read and agreed to the published version of the manuscript. Funding: This research received no external funding. Acknowledgments: The authors thank the collaboration of Carmen L ó pez Zarzuela, a rehabilitation doctor of the “Virgen de la Salud” Hospital in Toledo, the parents who agreed to participate and allow the intervention to be carried out successfully due to their great dedication, and especially the children who participated in this study for their continuity and daily improvement. Conflicts of Interest: The authors declare no conflict of interest. References 1. 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