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Microswitch-Cluster Technology for Promoting Occupation and Reducing

Hand Biting of Six Adolescents with Fragile X Syndrome: New Evidence and

Social Rating

Article  in  Journal of Developmental and Physical Disabilities · October 2018

DOI: 10.1007/s10882-018-9634-9

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ORIGINAL ARTICLE

Microswitch-Cluster Technology for Promoting Occupation and Reducing Hand Biting of Six Adolescents with Fragile X Syndrome: New Evidence and Social Rating

Viviana Perilli1 & Fabrizio Stasolla2 & Alessandro O. Caffò3 & Vincenza Albano3 & Fiora D’Amico4

# Springer Science+Business Media, LLC, part of Springer Nature 2018

Abstract We further extended the use of microswitch-cluster technology for promoting occupa- tional activities and reducing hand biting of six adolescents with fragile X syndrome and severe to profound developmental disabilities. The primary rehabilitative goal was to enhance the adaptive response (i.e., inserting three different objects in the three containers within a 4 s time interval). The secondary objective was to evaluate the effects of the intervention on indices of positive participation as outcome measure of the participants ‘quality of life. Finally, a social validation assessment involving sixty- six external raters was conducted. The study was carried out according to an ABB1AB1

experimental sequence for each participant. Thus, A indicated baselines, B indicated the intervention focused on promoting the adaptive response irrespective of the challenging behavior, and B1 indicated the cluster phases with the provision of positive stimulation only if the adaptive response was exhibited with the absence of the challenging behavior. A one-year follow-up was implemented. Results showed an improved performance for all the participants, which was maintained over the time. Indices of positive participation increased as well. Social raters favorably scored the use of the microswitch-cluster technology. Clinical, educational, psychological, and reha- bilitative implications of the findings were critically discussed.

Keywords FragileXsyndrome.Clustertechnology.Qualityoflife.Positiveparticipation.

Social validation

Journal of Developmental and Physical Disabilities https://doi.org/10.1007/s10882-018-9634-9

* Fabrizio Stasolla [email protected]

1 Villa Apelusion Medical Care Center, Bari, Italy 2 Università BGiustino Fortunato^ of Benevento, Benevento, Italy 3 Department of Educational Sciences, Psychology, Communication, University of Bari, Bari, Italy 4 Villa Argento Medical Care Center, Locorotondo, Alberobello, Italy

Introduction

Fragile X syndrome (FXS) is a rare genetic disease and represents one of the frequent cause of severe to profound developmental disabilities. It is due to an excessive length of a cytosine-guanine-guanine (CGG) triplet repeat in the fragile mental retardation 1 (FMR1) gene located on the X chromosome. Consequently, a wide range of intellectual disabilities, learning difficulties, anxiety, attention deficits, impulsivity, and autistic-like behaviors are commonly observed. Additionally, self-injury, aggression, and disruptive behavior usually emerge. Hand biting and flapping stereotypic behaviors are described as part of the phenotype (Cornish et al. 2004; Morel et al. 2018; Purugganan 2018). Accordingly, individuals with FXS may be passive and isolated, have poor indepen- dence and constantly rely on caregivers’ assistance, and may have negative outcomes on their quality of life. To tackle the latter issue, one may envisage microswitch-based programs (MBP) (Haessler et al. 2016; Hardiman and McGill 2018; Stasolla et al. 2014a, b, c; Stasolla et al. 2016a, b).

Microswitches represent basic forms of assistive technology (AT) enabling persons with severe to profound developmental and/or multiple disabilities to interact with external environment. For example, brief periods of positive stimulation (e.g., 7–10 s) may be automatically delivered by an electronic system contingently to small behavioral responses (e.g., eye blinking, hand closing) detected by specific tools (e.g., optic or pressure sensors), based upon learning principles (i.e., causal association between behavioral responses and environmental consequences) (Lancioni et al. 2014a, b; Stasolla and Perilli 2015). In other words, a participant with severe to profound intellectual disabilities will be capable of determining the provision of positive environmental events independently (Lancioni et al. 2012; Stasolla et al. 2016a, b). One may reasonably argue that the above described process should have beneficial effects on the participant’ s quality of life, given that it is aimed at improving independence, constructive engagement, positive participation, and self- determination (Alborz 2017; Wehmeyer and Abery 2013). Within this framework, microswitch-cluster technology (MCT) pursues the dual and simultaneous goal of promot- ing an adaptive response and reducing a challenging behavior (Lancioni et al. 2005a). For example, Lancioni et al. (2013a) exposed a 10-year old girl who suffered from a post-coma (i.e., minimally conscious state due to a stroke), presented left hemiparesis, confused speech, was non-ambulatory, and tended to be passive, usually sat with her head and trunk leaning on the table, to a MCT focused on moving different daily objects across the table (i.e., adaptive response), and simultaneously decreasing inappropriate posture (i.e., head and trunk forward leaning as challenging behavior). The MCT consisted of (a) a vibration microswitch for detecting the adaptive response, (b) an optic microswitch fixed on her wheelchair’s back activated once the participant’s trunk distance was less than 6 cm from it, (c) a tilt microswitch attached to a wire frame, fixed at her left ear, activated once her head deviated less than 35° from an ideal straight line, and (d) a computer control system, connected to the microswitches, which automatically delivered brief periods (i.e., 10 s) of positive stimulation during intervention phases. The study was carried out according to an ABB1AB1 experimental sequence, in which A indicated baselines, B indicated an intervention phase, in which the adaptive responses were always followed contingently by preferred stimulation, and B1 indicated the cluster, in which the adaptive responses were followed contingently by preferred stimulation only if the challenging behavior (i.e., inappropriate posture) was absent. A 2-week post-intervention check was conducted.

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Results showed large increasing of adaptive responses and drastic reduction of the inappro- priate posture, during both B1 and post-intervention phases.

The literature on the use of MCT for teaching adaptive responding and decreasing challenging behaviors among individuals with multiple disabilities is widely substantial (Lancioni et al. 2003; Lancioni et al. 2004a, b; Lancioni et al. 2005a, b, c; Lancioni et al. 2006; Lancioni et al. 2007a, b, c, d; Lancioni et al. 2008a, b, c; Lancioni et al. 2009; Lancioni et al. 2011; Lancioni et al. 2013b; Stasolla et al. 2014a, b, c). However, its use for occupational (i.e., functional) purposes is largely limited (Stasolla et al. 2017a, b) and no empirical evidences are available on the use of MCT for individuals with FXS (Stasolla et al. 2014a, b, c). Within this framework, Stasolla et al. (2014a, b, c) exposed two boys with FXS and developmental disabilities to a MBP for promoting choice opportunities among preferred stimuli with positive outcomes, which were consolidated during the maintenance phase. Recently, Stasolla et al. (2017a, b) posi- tively extended the implementation of a MBP to three new participants with FXS for (a) promoting a new adaptive response (i.e., insert two different objects in two containers available, within 3 s), (b) monitoring its effect on participants’ positive mood, (c) conducting a three-month follow-up, and (d) assessing a social validation procedure involving 30 parents of children with developmental disabilities as external raters. All the participants profitably learned the use of the technology for occupational purposes and consolidated it during the follow-up. The MBP had beneficial consequences on participants’ mood. Social raters favorably endorsed the use of a MBP. Despite the aforementioned encouraging and promising findings, new extensions were warranted.

In light of the above, the current investigation would represent the first attempt to adopting a MCT for occupational goals and reducing hand biting of six adolescents with FXS and severe to profound developmental disabilities. Additionally, indices of positive participation, which should be closely linked to the participants’ constructive engagement, independence, and improved quality of life (Lancioni et al. 2014a; Stasolla et al. in press) were recorded. A 1-year follow-up was assessed. A new social validation procedure, which recruited three different groups of raters (i.e., caregivers, practitioners and psychologists) was carried out (Stasolla et al. in press).

Method

Participants and Setting

The eligibility criteria assessed at the beginning of the study were (a) a diagnosis of FXS, (b) a chronological age comprised between 13 and 19 years old (i.e., the study was focused on adolescents with FXS), (c) severe to profound intellectual and devel- opmental disabilities, (d) hand biting stereotypic behavior reported by parents and caregivers, and (e) the capacity of managing familiar objects if adequately rewarded (i.e., the capacity of keeping up a familiar object and inserting it in a container independently if rewarded) (i.e., being highly motivated), without the caregivers’ assistance was considered. Accordingly, six adolescents (Alfred, David, Gerard, Lee, Mathew, and Stephan) who were aged of 13.2, 16.4, 15.7, 18.6, 14.5, and 17.8 respectively, were selected for the current investigation. They were diagnosed with the full mutation of FXS from DNA results. Although no formal IQ score was available

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since no specific test was feasible due to their clinical conditions, the standard scores of the Vineland Adaptive Behavior Scale (VABS) (Perry et al. 2009), assessed by their psychologist, revealed that their mental ages were 3.2, 2.8, 2.3, 2.9, 3.6, and 2.7, respectively. Consequently, they were considered between the severe and the profound ranges of developmental and intellectual disabilities (i.e., a significant impairment of adaptive and intellectual functions).

Both parents and caregivers (i.e., psychologists, physiotherapists, and teachers) reported them with lack of speech, unawareness of sphincter control, isolated and passive with frequent hand biting stereotypic behavior, which seriously hampered their desirability, social image, and status. In other words, their isolation and passivity were deleterious for their social image and status. They were unable to learn new adaptive skills independently due to their general conditions but they had the ability of manip- ulating and inserting familiar objects in adaptive containers whenever sufficiently motivated, and would greatly benefit of a technological-aided program, as indicated and suggested by parents, psychologists, and neurologists. That skill was considered the adaptive response for this study since it seemed to be the most suitable positive behavior, in accordance with their families and caregivers. All the participants attended regular classes with a special training and support teacher 18 h per week. Additionally, they received stimulation and speech sessions, three days per week, within a medical center for individuals with developmental delays attended during the afternoon.

The rehabilitative program was carried out individually at participants’ homes. During the sessions, the participants sat in the wheelchair, in their quiet room, in front of a 160 × 80 cm rectangular table. Their parents considered the MCT highly desirable and signed a formal consent for the participation to the study, which was approved by a local ethic and scientific committee, and conducted according to Helsinki Declaration and its later amendments.

Selection of Stimuli

A formal screening of preferences (Crawford and Schuster 1993) and an informal 25– 30 min interview with parents and caregivers were used for selecting the positive stimulation to be used during the sessions as reinforcements. During the formal screening, 5 10-min sessions were collected. Auditory, visual stimuli and tactile vibrations were assessed. Overall, 15–20 randomized 6 s of presentations (with a 20– 25 s of rest interval) were provided by two research assistants. According to specific satisfaction criteria (i.e. alerting, orientation, and smiling evoked), stimuli preferred at least for the 80% of the time were retained. Amusing songs, pleasant songs, and tactile vibrations were considered as positive reinforcements for all the participants involved.

Technology and Response

The technology consisted of 3 40 × 40 cm square wicker containers fixed on the table and distanced of 10 cm one to each other, equipped with a square sensitive 40 cm pressure microswitch, activated with at least 3 g of weight on it, an optic sensor fixed on the participant’s lip corner, a laptop available behind the containers, visible but inaccessible to the participants, and an interface connecting the microswitches to the laptop. The laptop was equipped with a 17 in. monitor, and a Clicker 5 software

Journal of Developmental and Physical Disabilities

package (Crick House, Moulton Park, Northampton, UK). Both pressure and optic microswitches constituted the cluster technology. Familiar objects (i.e., little cars and bottles) were randomly available on the table around the containers.

The adaptive response consisted of inserting three different objects in the three containers (i.e., one in each container) within a 4 s time interval. It seemed to be the most suitable response for all the participants, selected in accordance with parents and caregivers. The challenging behavior consisted of hand biting (i.e., physical contact between participants’ hands and teeth). The rationale for choosing the adaptive response was to (a) preventing passivity, (b) promoting occupational activities, (c) improving constructive engagement, and (d) teaching a functional task (Stasolla et al. 2017a, b).

Sessions and Data Collection

Sessions lasted 10 min and were video-recorded. Typically, 3 sessions were collected 4 days per week. The study was completed within 15 months (i.e., considering 1 year follow-up). Overall, 130 sessions were carried out for each participant. Given that the adaptive response included 4 s and the contingent positive reinforcement lasted 6 s, the participants could provide up to 60 responses within a 10 min session. The laptop had three basic functions, (a) automatically record an adaptive response and the challenging behavior, (b) ignore a new adaptive response occurred during the stimulation period, and (c) deliver positive stimulation during intervention, cluster, and follow-up phases (i.e., see below experimental conditions).

Data collection concerned (a) the adaptive responses, (b) the challenging behavior, (c) indices of positive participation (i.e., gaze oriented to the containers and/or the laptop for 10 s within the observed interval, smiles, laughs, energized body movements with or without vocalizations), and (d) social validation scoring. The indices of positive participation were recorded by two blind research assistants according to a 15 s partial interval recording system, which included 10 s of observation and 5 s of dichotomous report (i.e., absence or presence) of the participation in the previously observed interval (Stasolla et al. 2015; Lancioni et al. 2005a, b, c, d, e). Verbal and physical prompts were provided by the research assistants every 30 s of non-independent responding.

Inter-Observers Agreement

The fidelity of the dependent variables was computed on the simultaneous and independent coding of the sessions by two blind research assistants. Thus, two inde- pendent and blind research assistants coded independently and simultaneously the collected sessions. The percentages of agreement were 98% (95–100), 99% (96– 100), and 97% (94–100) for the adaptive responses, the challenging behavior, and the positive participation, respectively. Furthermore, Koehn values were .98, .99, and .97 for the adaptive responses, the challenging behavior, and the positive participation, respectively.

Procedural Integrity

Both blind independent research assistants who assessed the fidelity, systematically followed each step of the study. For each checklist, three basic steps occurred. First, a

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blind research assistant coded singly the sessions. Second, the other research assistant checked autonomously the sessions. Finally, they compared systematically their results simultaneously. In fact, they checked the preference screening with the positive stimuli selected for the intervention phases, the containers positions, the management of the prompts, and the provision of the environmental consequences to be delivered contin- gently to the adaptive responding (except for the baselines), through a checklist of the procedural steps. Furthermore, they supervised the follow-up and the social validation procedure. The results revealed 100% of correct implementation, on all the collected sessions.

Experimental Conditions

The study was carried out according to an ABB1AB1 experimental sequence for each participant (Barlow et al. 2009; Stasolla et al. 2014a, b, c), in which A represented baselines, B represented intervention phases, and B1 the cluster. A one-year follow-up was conducted with a new AB1AB1 experimental sequence.

First Baseline (a)

During the first baseline, 5 sessions were collected within two days for each participant. The technology was available but inactive. Even if the participants produced an adaptive response with or without the challenging behavior, no environmental events were provided.

First Intervention (B)

During the first intervention, 30 sessions were collected for each participant within three weeks. The technology was available and active. Contingently to an adaptive response, 6 s of positive stimulation were automatically delivered by the system, irrespective of the challenging behavior, which was continuously monitored.

First Cluster Phase (B1)

During the first cluster phase, 30 sessions were collected within 3 weeks for each participant. Six seconds of positive stimulation were automatically delivered by the system contingently to an adaptive response only if it occurred free of the challenging behavior (i.e., with its simultaneous absence). Whenever a challenging behavior was recorded during the stimulation provision (i.e., 6 s), the stimulation itself was automat- ically interrupted by the system.

Second Baseline (a)

Once the learning process was consolidated (i.e., at least 75% of an adaptive responding free of the challenging behavior for each participant), a new baseline occurred. Experimental conditions were identical to the first baseline. Five sessions were collected for each participant within two days.

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Second Cluster (B1)

A new cluster phase followed the second baseline. Thirty sessions were collected for each participant within three weeks. Experimental conditions were identical to the first cluster phase.

Follow-Up

A one-year follow-up was conducted. An AB1AB1 experimental sequence was carried out for each participant. Experimental conditions were identical to the baselines and cluster phases, respectively. Thirty sessions (i.e., ten for both baselines and twenty for both clusters) were collected within three weeks.

Social Validation

Three groups of external raters (i.e., twenty-two for each group) were recruited for a social validation assessment. Specifically, caregivers, practitioners and psychologists, who worked in a local rehabilitative center for individuals with developmental disabil- ities, were selected as a convenience sample (Pedhazur and Schmelkin 1991). The eligibility criteria were (a) at least 10 years of professional experience, and (b) the supervision in at least one cognitive-behavioral rehabilitative intervention. Mean ages, and standard deviation scores were 34.6 (7.32), 44.8 (8.54), and 39.9 (6.34) for caregivers, practitioners, and psychologists, respectively.

They were equally and randomly divided in 6 groups of 11 raters (i.e., one for each participant). They were requested watching and assessing a 6 min video with a standard and representative part of session of an intervention (B), and a cluster (B1) phases. They were told that they would be expected to watch an adolescent FXS exposed to a rehabilitative intervention, focused on promoting an adaptive responding and reducing a challenging behavior. Subsequently, they would be requested to evaluate the videos on few relevant questions. The order of videos were systematically and randomly alternated. That is, half of the raters watched a BB1 sequence and the remaining watched a B1B sequence. Finally, they were requested to fill a 6 items questionnaire recently validated (see Table 1), through a Likert-type scale (i.e., strongly disagree, disagree, undecided, agree, and strongly agree), with 1–5 points scale, which indicated the least and most positive ratings, respectively (Stasolla et al. in press).

Results

Data were summarized over blocks of sessions and plotted in Figs. 1 and 2, for practical reasons. Original data were deposited in a public data base (i.e., DOI https://doi. org/10.6084/m9.figshare.7120835). Mean scores and ranges of the participants’ performances (i.e., adaptive responses, challenging behavior and indices of positive participation) were included in Tables 2, 3, and 4. Because the prompted responses were up to 3, 2, 4, 1, 2, and 2 for Alfred, David, Gerard, Lee, Mathew, and Stephan, respectively, at the beginning of the study, they were subtracted from the total and not reported.

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For all the participants, data evinced an improved adaptive responding during intervention and cluster phases, if compared to the baselines. Challenging behavior was drastically reduced. Indices with positive participation relevantly increased. The learning process was consolidated during the follow-up. Differences between baselines on one hand, and intervention and cluster phases on the other, were statistically significant (p < .01) to the Kolmogorov-Smirnov test (Siegel and Castellan 1988).

Mean, standard scores, paired T test and their significance for the social validation assessment were reported in Table 5. All the raters favorably scored the cluster technology on all the questionnaire’s items.

Because no statistical differences emerged between the three groups of raters, data were included as an unique sample of 66 raters for practical reasons (see Table 5) (Hastie et al. 2009). All the raters favorably considered the cluster if compared to the intervention phases on all the questionnaire’s items.

Discussion

Data demonstrated that a MCT may be effective and suitable for pursuing the dual goal of promoting an adaptive response and reducing a challenging behavior of adolescents with FXS and severe to profound developmental disabilities. All the participants significantly improved their performances during intervention and cluster phases if compared to the baselines. The occupational activities were enhanced and hand biting decreased. They consolidated the learning process during the follow-up. Indices of participation, which were expected to be closely linked to the participants’ constructive engagement and positive occupation, increased, with beneficial effects on their quality of life (i.e., they could be intended as an outcome measure of the quality of life). Social raters favorably endorsed the MCT. These findings were consistent with the literature available (Budimirovic et al. 2017; Fisch et al. 2012; Hare et al. 2014; Mirrett et al. 2003; Stasolla et al. 2017a, b), and suggested the following considerations.

First, the current investigation may be considered a profitable extension of previous findings focused on enhancing occupational activities and reducing hand biting of adolescents with FXS. It may be viewed as effective for promoting self-determination and independence of the participants involved. MCT enabled the participants with the autonomous access to positive stimulation. In fact, the participants first learned to performing a new adaptive response for occupational purposes (i.e., during intervention phases), and subsequently decreased their challenging behavior during cluster phases. At the same time, they positively participated and increased their active role towards their environmental preventing their isolation and passivity, with beneficial outcomes

Table 1 Social validation questionnaire

Do you think that the child enjoys (is comfortable with) this condition? Do you think that this condition promotes self-determination? Do you think that this condition enhances positive participation? Do you think that this condition has beneficial/rehabilitative outcomes? Do you think that this condition is suitable for daily contexts? Do you support (agree with) this condition?

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for their social image, desirability, and status, since they were constructively engaged (Chiapparino et al. 2011; Lancioni et al. 2014b; Stasolla and De Pace 2014).

Second, MCT may represent a valid option with regard to conventional programs (e.g., differential reinforcement of other/alternative behaviors) for reducing caregivers’ burden. It may be a critical educational and rehabilitative resource for fostering the participants’ inclusion in daily settings. In fact, MCT can be considered a relative

Fig. 1 The graph summarizes the data about performance for all six participants. The black and the light bars indicate the mean frequencies of adaptive responses and challenging behaviour, respectively, over blocks of sessions for the two baseline phases, the three intervention phases and the follow-up phases. The number of sessions included in each block is indicated by the numeral above it

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simple and realistic or practical solution for complex clinical conditions. Thus, the management of two dependent variables (i.e., both adaptive response and challenging behavior), the provision of contingent stimulation (i.e., simultaneous presence of an adaptive response and absence of a challenging behavior), and the interruption of it whenever the challenging behavior was present, may be uneasy and/or onerous for parents and caregivers to implement. The MCT can supply all the aforementioned requirements. Its use may be considered as affordable by parents and caregivers (i.e., by

Fig. 2 The graph summarizes the data about the participation for all six participants. The black diamonds refer to the mean percentage of intervals with indices of positive participation over blocks of sessions for the two baseline phases, the three intervention phases and the follow-up phases. The number of sessions included in each block is indicated by the numeral above it

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Ta b le 2

M ea n va lu es

an d ra ng es

of ad ap ti ve

re sp on se s

P ar ti ci pa nt s

In te rv en ti on

ph as es

F ol lo w

up

A B

B ′

A B ′

A B ′

A B ′

A lf re d

5. 25

(2 – 7 )

4 1. 3 7 (2 4– 50 )

51 .8

(4 8– 55 )

9. 5 (6 – 1 2)

47 .9 7 (3 6 – 55 )

6 (5 – 8)

31 .2 3 (3 5– 4 2)

8 .4 2 (5 – 12 )

4 3. 9 (3 7 – 4 8)

D av id

6. 5 (4 – 8)

2 8. 1 7 (1 4– 36 )

46 .9

(3 6– 52 )

10 .5

(6 – 12 )

44 .5 7 (2 6 – 50 )

7. 92

(7 – 9)

29 .5 7 (2 9– 4 8)

8 .4 2 (7 – 10 )

3 9. 9 (2 7 – 4 8)

G er ar d

4. 92

(4 – 6 )

2 8. 5 3 (1 4– 35 )

47 .1

(3 6– 52 )

8. 58

(7 – 10 )

44 .4

(2 5– 50 )

10 .0 8 (8 – 12 )

27 .7

(2 8 – 44 )

9 .4 7 (7 – 11 )

3 2. 2 (2 6 – 3 8)

L ee

7. 42

(5 – 9 )

2 7. 1 2 (1 8– 32 )

42 .8 7 (3 2 – 50 )

10 .9 2 (1 0– 1 2)

43 .2

(2 8– 50 )

7. 58

(6 – 9)

22 .7

(9 – 38 )

7 .4 2 (5 – 9)

3 6. 9 (2 5 – 4 5)

M at th ew

9. 58

(8 – 11 )

3 3. 5 3 (2 6– 38 )

49 .7 3 (4 0 – 54 )

8. 83

(4 – 12 )

44 .5 6 (3 0 – 51 )

7. 33

(4 – 10 )

26 .1 3 (2 4– 4 0)

7 .1 7 (4 – 9)

3 8. 2 (2 9 – 4 8)

S te p ha n

8. 83

(6 – 1 0)

3 2. 1 7 (2 6– 36 )

44 .8 3 (3 2 – 51 )

8. 92

(6 – 11 )

47 .2 7 (4 2 – 52 )

9 (5 – 11 )

31 .9 3 (3 4– 5 1)

7 .8 3 (5 – 10 )

4 6. 4 (3 9 – 5 0)

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Ta b le 3

M ea n va lu es

an d ra ng es

of ch al le ng in g be ha vi o r

P ar ti ci pa nt s

In te rv en ti on

ph as es

F ol lo w

up

A B

B ′

A B ′

A B ′

A B ′

A lf re d

5. 2 5 (2 – 7 )

1 3. 5 (1 0– 2 2)

3. 9 7 (1 – 8 )

8. 42

(5 – 10 )

3. 7 7 (1 – 8 )

6 (5 – 8)

3. 6 (2 – 5 )

7 .5 8 (5 – 10 )

4 .6

(2 – 7 )

D av id

6 (4 – 7 )

9 .9 3 (8 – 12 )

2. 9 7 (1 – 6 )

9. 83

(6 – 11 )

2. 5 3 (1 – 6 )

7 .6 7 (7 – 8)

5. 1 (3 – 6 )

7 .8 3 (6 – 9)

3 .6

(1 – 6 )

G er ar d

4. 7 5 (4 – 6 )

11 .4 7 (9 – 19 )

3. 7 (1 – 8)

8. 17

(7 – 9)

2. 3 (1 – 5)

9 .8 3 (8 – 11 )

4. 5 (2 – 6 )

9 .1 7 (7 – 10 )

4 .3

(2 – 6 )

L ee

7. 4 2 (5 – 9 )

9 .6 3 (7 – 12 )

3. 7 7 (1 – 6 )

10 .4 2 (1 0– 11

2. 8 7 (1 – 6 )

7 .5 8 (6 – 9)

3. 2 (2 – 6 )

7 .1 7 (5 – 8)

2 .3

(1 – 4 )

M at th ew

9. 17

(7 – 1 0)

11 .9

(1 0 – 14 )

4. 8 7 (2 – 8 )

8. 67

(4 – 12 )

4. 6 7 (2 – 8 )

7 .1 7 (4 – 10 )

4. 9 (2 – 7 )

6 .9 7 (4 – 9)

4 .3

(2 – 6 )

S te p ha n

8. 67

(6 – 1 0)

11 .6 7 (1 0– 1 4)

6. 3 3 (3 – 9 )

8. 75

(6 – 11 )

4. 7 3 (3 – 7 )

8 .6 7 (5 – 10 )

5. 7 (4 – 7 )

7 .4 2 (5 – 10 )

6 .2

(5 – 8 )

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Ta b le 4

M ea n pe rc en ta ge s an d ra n ge s of

in te rv al s w it h in di ce s o f p os it iv e pa rt ic ip at io n

P ar ti ci pa nt s

in te rv en ti on

p ha se s

fo ll ow

u p

A B

B ′

A B ′

A B ′

A B ′

A lf re d

20 .8

(1 5– 2 5)

7 2. 2 (6 0 – 80 )

83 .9

(7 5 – 90 )

27 .1

(2 0 – 35 )

80 .5

(6 7– 90 )

23 .7

(2 0– 30 )

7 7 (6 7 – 85 )

1 6. 7 (1 0– 2 2)

7 2. 7 (6 2– 8 2)

D av id

2 9. 3 (2 5– 3 2)

6 9. 9 (5 5 – 80 )

86 .2

(7 7 – 95 )

24 .2

(1 7 – 35 )

80 .8

(6 7– 90 )

29 .2

(2 0– 35 )

7 4. 2 (6 7– 8 0)

2 1. 1 (1 2– 2 7)

6 9. 7 (6 2– 7 5)

G er ar d

8 .1

(1 5 – 20 )

6 6. 2 (4 0 – 75 )

87 (7 7– 9 5)

22 .9

(2 0 – 25 )

81 .2

(7 0– 90 )

26 .8

(2 5– 30 )

7 5. 5 (6 7– 8 5)

2 6. 5 (2 0– 3 5)

8 2. 2 (7 2– 9 0)

L ee

2 2 .5

(2 0– 2 5)

6 6. 5 (5 5 – 75 )

85 .5

(7 7 – 75 )

12 .7

(1 0 – 15 )

80 .8

(7 2– 90 )

20 (1 5 – 25 )

7 8 (6 7 – 87 )

1 7. 5 (1 2– 2 0)

7 9. 5 (7 5– 8 7)

M at th ew

27 .5

(2 5 – 3 0)

7 2 (6 7– 75 )

86 .1

(7 7 – 93 )

26 .1

(2 0 – 30 )

81 .4

(7 5– 90 )

22 .3

(2 0– 25 )

7 7 (7 0 – 82 )

1 7. 5 (1 5– 2 0)

7 8. 7 (7 5– 8 2)

S te p ha n

1 6. 5 (1 0 – 2 0)

6 6. 2 (3 5 – 75 )

83 .1

(7 7 – 90 )

26 .5

(2 0 – 32 )

82 .7

(7 5– 90 )

32 .9

(2 5– 40 )

8 5. 5 (8 0– 9 0)

2 8. 5 (2 5– 3 2)

8 2 (7 2– 90 )

Journal of Developmental and Physical Disabilities

anecdotal and informal interview) in daily context since its cost is approximately 900 US dollars (Nazareth et al. 2016; Reichle 2011).

Third, the success of a MCT widely relied on the motivating potential of the delivered positive stimulation, the simplicity (i.e., low cost of response because already available in the behavioral repertoire), the manageability of the challenging behavior, and the suitability of the devices used. With regard to the first point, strong (i.e., highly rewarding) environmental events may profitably compensate the response cost. With regard to the second point, an easily reproducible adaptive response may be considered as crucial for the learning process. With regard to the third point, the manageability of the challenging behavior largely depends on the rapidity with it may be corrected and/ or redirected by or into a new adaptive response. With regard to the fourth point, the suitability of the devices used can be assessed with the easy matching between the adopted microswitches and the targeted responses/behaviors (Lancioni et al. 2012; Lancioni and Singh 2014). The data of the current investigation suggested a favorable outcome with regard to each of the four above mentioned points.

Fourth, the positive achievement of the study was favorably continued over the time. That is, the one-year follow-up demonstrated that all the participants maintained their learned capacities for occupational purposes. Furthermore, they drastically reduced their hand biting. One may argue that their cognitive and behavioral skills were enhanced, even with a long term intervention suspension (i.e., one year), indicating the quality, the effectiveness, and the suitability of the MCT for rehabilitative goals (Catania 2012; Fabio and Caprì 2015; Fabio et al. 2018; Kazdin 2001).

Fifth, the social raters favorably endorsed the use of MCT for promoting indepen- dent occupational activities, reducing hand biting, and increasing positive participation. Irrespective of their group (i.e., caregivers, practitioners, or psychologists), they all considered the cluster solution suitable for the dual goal of learning and an adaptive response and simultaneously decreasing the challenging behavior. Essentially, the clinical validity of the intervention program was significantly corroborated (Caffò et al. 2014; Perilli et al. 2013a, b).

Limitations

Despite the above discussed positive outcomes, this study presented some limitations. For example, it was based on a single-subject experimental design, even if the small sample considered was representative. Caution was mandatory and its generalization to

Table 5 Social validation scores

Items Mean B St. dev. B Mean B1 St. dev. B1 T df p

Comfort 3.66 .54 4.25 .55 8.75 65 <.001

Self-determination 3.88 .52 4.33 .56 9.15 65 <.001

Participation 3.75 .53 4.19 .51 7.86 65 <.001

Rehabilitation 3.42 .58 4.67 .48 12.27 65 <.0001

Suitability 3.53 .55 4.28 .50 11.54 65 <.0001

Support 3.29 .56 4.44 .57 14.67 65 <.0001

Journal of Developmental and Physical Disabilities

new participants with FXS and/other rare genetic syndromes (e.g., Angelman, Cornelia de Lange, and Rett) was warranted. Moreover, since the adaptive response and the microswitches triggered automatically the positive stimulation, the participants were not instructed to self-manage the reinforcements. Furthermore, a functional analysis of the challenging behavior would be useful (Hardiman and McGill 2017; Machalicek et al. 2014). The current study was focused on basic and simple responses. Different and more sophisticated behavioral responses could be trained for the future. A system- atic comparison with other behavioral interventions (e.g., differential or non-contingent reinforcement) would be useful (i.e., one delivered via microswitch and one adminis- trated via caregiver).

Future Research Perspectives

In light of the above, new research perspectives within this framework should deal with the following topics (a) new extension of the MCT to further participants with FXS and/other developmental disorders (e.g., autism and cerebral palsy), (b) a generalization of its use in daily contexts (e.g., school and/or medical settings), (c) a systematic comparison with different AT-based programs (e.g., request and choice) or other cognitive-behavioral interventions (e.g., positive reinforcement and/or extinction), (d) an enlargement of the social validation assessment to new groups of raters (e.g., parents of children with developmental disabilities, and/or physiotherapists), and (e) the envisagement of preference checks for the participants involved between two different rehabilitative programs implemented.

Compliance with Ethical Standards

Funding The authors received no financial support for the research, authorship, and publication of the article.

Ethical Approval All performed procedures of the study have been carried out in accordance with Helsinki Declaration (1964) and its later amendments or comparable ethical standards.

Informed Consent Informed consent was obtained for all the recruited participants by their legal represen- tatives (i.e., their parents).

Conflict of Interest The authors declared no conflicts of interest with respect to the research, authorship, and/ or publication of the article. The authors alone are responsible for the content and the writing of the article.

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  • Microswitch-Cluster...
    • Abstract
    • Introduction
    • Method
      • Participants and Setting
      • Selection of Stimuli
      • Technology and Response
      • Sessions and Data Collection
      • Inter-Observers Agreement
      • Procedural Integrity
      • Experimental Conditions
      • First Baseline (a)
      • First Intervention (B)
      • First Cluster Phase (B1)
      • Second Baseline (a)
      • Second Cluster (B1)
      • Follow-Up
      • Social Validation
    • Results
    • Discussion
      • Limitations
      • Future Research Perspectives
    • References