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J Dev Phys Disabil (2008) 20:209–216

DOI 10.1007/s10882-008-9105-9
O R I G I N A L A RT I C L E

Learning in Post-coma Persons with Profound Multiple


Disabilities: Two Case Evaluations

Giulio E. Lancioni & Marta Olivetti Belardinelli &


Claudia Chiapparino & Maria Teresa Angelillo &
Fabrizio Stasolla & Nirbhay N. Singh &
Mark F. O’Reilly & Jeff Sigafoos & Doretta Oliva

Published online: 15 April 2008


# Springer Science + Business Media, LLC 2008

Abstract Finding signs of learning in post-coma persons with profound multiple


disabilities (i.e., in a vegetative state or minimally responsive) would underline (a)
an awareness/consciousness of their responding and its links with environmental
stimuli and (b) a positive development in their immediate situation with the
possibility of treatment (rehabilitation) advances. This study was aimed at assessing
signs of learning in two of these persons (i.e., two adults). The learning setup
involved eye blinking as the persons’ responses and microswitch technology to
detect such responses and to present stimuli. The technology consisted of an
electronically regulated optic sensor mounted on an eyeglasses’ frame that the
persons wore during the sessions and a control system connected to stimulus
sources. The study involved an ABABCB sequence, in which A represented baseline
periods, B intervention periods with stimuli contingent on the responses, and C a
control condition with stimuli presented non-contingently. Data showed that the
level of responding during the B phases was significantly higher than the levels

G. E. Lancioni (*) : C. Chiapparino : M. T. Angelillo : F. Stasolla


Department of Psychology, University of Bari, Via Quintino Sella 268, 70100 Bari, Italy
e-mail: g.lancioni@psico.uniba.it

M. O. Belardinelli
University “La Sapienza” of Rome, Rome, Italy

N. N. Singh
ONE Research Institute, Midlothian, VA, USA

M. F. O’Reilly
University of Texas at Austin, Austin, TX, USA

J. Sigafoos
Victoria University of Wellington, Wellington, New Zealand

D. Oliva
Lega F. D’Oro Research Center, Osimo, Italy
210 J Dev Phys Disabil (2008) 20:209–216

observed during the A phases as well as the C phase (i.e., indicating clear signs of
learning by both persons). The implications of these data for assessment and
rehabilitation of post-coma persons with profound multiple disabilities are discussed.

Keywords Microswitch technology . Eye-blinking responses . Multiple disabilities .


Vegetative state . Minimal responsiveness . Minimally conscious state

Treatment strategies frequently employed for improving the condition of post-coma


persons with profound multiple disabilities (i.e., in a vegetative state or minimally
responsive) include systematic sensory stimulation and music therapy (Crews et al.
1997; Gerber 2005; Giacino 1996; Lombardi et al. 2002; Magee 2005; Oh and Seo
2003; Wales and Waite 2005; Wilson et al. 1991, 2002). In spite of the large
emphasis on these forms of intervention, the literature available presents relatively
limited amounts of usable (reliable) data and fairly heterogeneous outcomes
(Aldridge et al. 1990; Avesani et al. 2006; Giacino and Trott 2004; Lombardi et
al. 2002; Wales and Waite 2005). The heterogeneous outcomes seem to underline,
among others, differences in the persons’ neurological conditions as well as
difficulties in drawing diagnostic statements with predictive implications for
intervention effects and rehabilitation results (Jennett 2002; Lomber and Eggermont
2006).
At least two hypotheses may be formulated with regard to persons with profound
multiple disabilities who seem to remain in a vegetative state or are minimally
responsive (e.g., showing some smiles to specific environmental stimuli) after long
periods of systematic stimulation and music therapy. First, one may hypothesize that
their level of functioning is so low that it precludes any strong increase in
responsiveness (Jennett 2002). Second, one might also hypothesize (a) that they
remain passive because the stimulation presented in those programs is not related to
any specific response that could then become actively linked to it and/or (b) that the
motor repertoire is so limited that no obvious responsiveness can easily emerge
(Avesani et al. 2006; Haggard 2005; Lancioni et al. 2005c; O’Brien et al. 1994;
Whitnall et al. 2006).
Assuming that the second hypothesis is correct, a learning setup involving a most
basic response and the use of stimuli contingent on it through the help of
microswitch technology might be reliably adopted as an assessment strategy as well
as a treatment procedure to help the person progress in his or her condition (cf.
Boyle and Greer 1983; Lancioni et al. 2005b, c; Naude and Hughes 2005; Ptak et al.
1998). If the response increases when contingent stimuli are used and declines in the
absence of stimuli or when the stimuli are unrelated to the response, one may
suggest that the person is showing an awareness of the response–stimuli link (i.e.,
signs of learning) (Lancioni et al. 2003, 2005c; O’Brien et al. 1994; Saunders et al.
2003). Observing signs of learning might help replace a rating of vegetative state
with a rating of minimally conscious state (cf. Giacino 2004; Morin 2006) or may
provide explicit support and justification for the use of the latter rating if this was
already being adopted (cf. Giacino and Trott 2004; Haggard 2005). In both cases, the
learning signs would assert the person’s new knowledge and call for an extension of
the intervention process in use with the establishment of new responses (Giacino
J Dev Phys Disabil (2008) 20:209–216 211

2004; Watson et al. 1999). This in turn would increase the person’s overall level of
activity and provide basic choice opportunities with the possibility of further
progress (Davis and Gimenez 2003; Giacino 2004; Giacino and Kalmar 2005;
Lancioni et al. 2005c).
This study was aimed at applying the learning setup outlined above with two
post-coma adults who had profound multiple disabilities. The response adopted was
eye blinking (i.e., a single blink or a sequence of two blinks within a 2-s interval,
based on the characteristics of the participants). The microswitch technology
consisted of an electronically regulated optic sensor mounted on an eyeglasses’
frame that the participants wore during the sessions and a control system linked to
stimulus sources (cf. Lancioni et al. 2005a, 2006).

Method

Participants

The participants (Luke and Hilda) were 29 and 49 years old, respectively. Both
participants had a normal life until they were involved (about 5 and 4 years prior to
the beginning of this study, respectively) in road accidents with severe brain injury.
Following the injury, they spent 2 and 7 weeks, respectively, in a coma from which
they emerged in a condition that was rated as vegetative state combined with
pervasive motor disabilities (spastic tetraplegia with minimal motor behavior) and
lack of speech. Hilda also presented with general drowsiness and epilepsy. Both
participants were fitted with a gastrostomy tube for nutrition, although Luke could
be partially fed via the mouth as well. Their condition required them to spend their
time in a wheelchair or in bed but they could orient their eyes to various stimuli.
Luke also showed smiles in relation to some environmental stimuli (e.g., joyful
laughing). This behavior had led to re-labeling his condition as minimal
responsiveness (or minimally conscious state; see Giacino 2004; Giacino and Trott
2004). They lived at home with both parents and a sister (Luke) or the mother and a
sister (Hilda) and received rehabilitation programs involving essentially physiother-
apy and stimulation sessions. Their families had provided informed consent for their
participation in this study.

Position, Response, Microswitch Technology, and Stimuli

During the study, both participants sat in their wheelchair. The response recorded for
Luke (who tended to have low rates of eye blinking) consisted of a single eye blink
(Tota et al. 2006). The response recorded for Hilda (who tended to have relatively
high rates of eye blinking) consisted of a sequence of two blinks occurring within a
2-s interval (Lancioni et al. 2005a). The technology consisted of an electronically
regulated optic sensor mounted on an eyeglasses’ frame that the participants wore
during the sessions and a control system linked to stimulus sources (see Lancioni et
al. 2005a; Tota et al. 2006). The optic sensor involved an infrared light-emitting
diode and a mini infrared light-detection unit (positioned in front and to the external
side of the participants’ left or right eye) (see Tota et al. 2006). When a response was
212 J Dev Phys Disabil (2008) 20:209–216

detected, a signal was transmitted to the control system. This system in turn activated
one or more stimuli (see below) for 4 or 6 s for the two participants, respectively,
during the intervention phases.
The stimuli used for the two participants were selected following interviews with
their families and brief stimulus preference screening (Crawford and Schuster 1993).
Screening involved four to ten nonconsecutive presentations of each of the stimuli
suggested by the families as presumably pleasant. A stimulus was selected for the
study if the participant seemed to alert to it during more than half of the
presentations. The stimuli selected included familiar voices and stories, songs and
musical items.

Experimental Conditions

The study involved an ABABCB sequence in which A represented baseline, B


intervention phases with stimuli contingent on the response, and C a non-contingent
stimulation phase (Barlow et al. 2006). Sessions lasted 10 min for Luke and 5 min
for Hilda (on medical and caregivers’ advice) and typically occurred three to six
times a day, based on participants’ availability. During the first baseline and first
intervention phase, the participants were prompted to respond (i.e., a light air-puff at
the corner of the eye) at the start of the sessions and after 30 s of nonresponding
during the sessions. Prompting during the first intervention phase was to increase the
frequency of response–stimulation pairings and possibly speed up response
strengthening. Prompting during baseline served as a basic control for its use/impact
during the first intervention phase. Responses were recorded automatically through a
counter connected to the control system. A response would be ignored by the system
if it occurred during a stimulus presentation interval (i.e., within the B and C phases)
or an equivalent time interval (i.e., within the A phases) (Lancioni et al. 2005a).

Baseline (A) Phases The two baseline (A) phases included six and 19 sessions for
Luke and six and eight sessions for Hilda. The microswitch technology was
available, but responses did not produce any stimuli.

Intervention (B) Phases The three intervention (B) phases included 57, 87 and 28
sessions for Luke and 62, 33 and 21 sessions for Hilda. Procedural conditions were
as during baseline except that responses produced the occurrence of stimuli.

Non-contingent Stimulation (C) Phase This phase included 14 and 22 sessions for
Luke and Hilda, respectively. Stimuli such as those used in the B phases were
presented independent of responses, at a mean frequency matching that of the last 20
sessions of the second B phase.

Results

The participants’ data are summarized in the two graphs of Fig. 1. Luke’s mean
frequency of responses was about 20 per session during the first A phase, increased
to 43 per session during the first B phase, and declined and increased again (to levels
J Dev Phys Disabil (2008) 20:209–216 213

Fig. 1 The upper graph shows Luke’s data, the lower graph Hilda’s data. Each data point represents the
mean frequency of responses over a block of two sessions. Data points with a single session can occur at
the end of the single phases of the study

similar to those just reported) during the next A and B phases, respectively. A new
decline in response levels (with a mean frequency of 22 responses per session) was
observed during the C phase. This decline was followed by a new response increase
(with a mean frequency of about 50 responses per session) during the final B phase.
Hilda’s data differed from those of Luke in terms of overall frequencies (partially
due to shorter sessions and a double blinking response), but presented a similar
trend. That is, the mean frequencies were 24 to 27 for the B phases, 14 and 13 for
the A phases, and 15 for the C phase. The differences between the A and B phases as
well as between the B phases and the C condition were statistically significant (p<
0.01) for both participants on the Kolmogorov–Smirnov test (Siegel and Castellan
1988).

Discussion

These data indicate that the learning setup using eye-blinking responses and
microswitch technology with positive stimuli produced a fairly clear outcome with
both participants. They showed response increases during the intervention periods
when stimuli were contingent on their responding and response declines during non-
contingent stimulation (i.e., indicating an awareness of the link between responding
and consequences). These findings, which are noteworthy for both participants,
appear even more remarkable for Hilda given her more severe condition including a
tendency to drowsiness (cf. Lippert-Gruner et al. 2003).
214 J Dev Phys Disabil (2008) 20:209–216

The implications of these findings are multiple. For example, the learning setup
seems to add to the diagnostic strategies available for these persons and, specifically,
to the neurological evidence of learning. This basically relies on some form of
perceptual adjustment (relating to stimulus habituation and mismatch) rather than on
active association (Kotchoubey et al. 2006; Morin 2006). The appearance of the
aforementioned awareness (i.e., learning) could prompt a change of diagnostic label
from vegetative state to minimally conscious state for Hilda and add strong,
clarifying support to the rating of minimal responsiveness (minimally conscious
state) available for Luke (Davis and Gimenez 2003; Giacino 2004; Giacino and
Kalmar 2005). The learning evidence could raise expectations of cognitive and
behavioral progress and encourage an extension of the intervention with the
inclusion of additional responses (Avesani et al. 2006; Bekinschtein et al. 2005;
Boyle and Greer 1983; Giacino and Trott 2004; Lomber and Eggermont 2006; Pape
et al. 2005a, b). The responses could be used with microswitches allowing direct
access to stimuli (as in the present study) or with Voice Output Communication Aids
(VOCAs), that is, devices that the participant can activate to ask for contact with or
specific help by the caregiver (cf. Schlosser 2003; Schlosser and Sigafoos 2006).
The latter form of devices could extend the participant’s activation opportunities and
also allow the assessment of his or her communication abilities, interests, and social–
emotional conditions. Finally, extending the range of responses would provide the
participant personal enrichment, in terms of output as well as environmental input,
with positive consequences for his or her overall status, dignity, and quality of life
(Schalock et al. 2005).
New research could (a) assess the possibilities of a program extension (as
discussed above) with both participants of this study and (b) pursue the replication of
the present study with new persons with profound multiple disabilities (i.e., in a
vegetative state or minimally responsive) to ascertain the generality of the reported
findings and the strength and practicality of the intervention setup used here (Barlow
et al. 2006; Kennedy 2005). New research could also examine possible correlations
of the data obtained with a learning setup, such as that used in this study, with other
types of data obtained from different behavioral and neurological assessments
(Bekinschtein et al. 2005; Haggard and Eimer 1999; Kotchoubey et al. 2006; Pape et
al. 2005a, b).
In conclusion, the present study has suggested that a learning setup with suitable
technology and apparently positive stimuli may be profitably used to enhance the
assessment and treatment of post-coma persons with pervasive disabilities. Definite
statements about such an approach would need to wait for additional evidence, given
the limited amount of data available to date. The acquisition of new data in line with
those obtained in this study could be highly encouraging as to the practically
valuable function of the approach and provide fresh impetus for testing it further.

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