ArticlePDF AvailableLiterature Review

Abstract and Figures

Aphasia is a significant cause of disability and reduced quality of life. Two speech pathology treatment approaches appear efficacious: multimodal and constraint-induced aphasia therapies. In constraint-induced therapies, non-verbal actions (e.g., gesture, drawing) are believed to interfere with treatment and patients are therefore constrained to speech. In contrast, multimodal therapies employ non-verbal modalities to cue word retrieval. Given the clinical and theoretical implications, a comparison of these two divergent treatments was pursued. This systematic review investigated both approaches in chronic aphasia at the levels of impairment, participation and quality of life. After a systematic search, the level of evidence and methodological quality were rated. Meta-analysis was conducted on 14 single case experimental designs using Tau-U, while heterogeneity in the four group designs precluded meta-analysis. Results showed that high-quality research was limited; however, findings were broadly positive for both approaches with neither being judged as clearly superior. Most studies examined impairment-based outcomes without considering participation or quality of life. The application and definition of constraint varied significantly between studies. Both constraint and multimodal therapies are promising for chronic post-stroke aphasia, but there is a need for larger, more rigorously conducted studies. The interpretation of “constraint” also requires clearer reporting.
Content may be subject to copyright.
Constraint and Multimodal approaches to therapy for chronic aphasia: A
systematic review and meta-analysis
John E. Piercea,b, Maya Menahemi-Falkova, Robyn O’Hallorana, Leanne
Togherc and Miranda L. Rosea*
aSchool of Allied Health, La Trobe University, Melbourne, Australia; bSpeech Pathology,
Cabrini Health, Melbourne, Australia; cSpeech Pathology, Faculty of Health Sciences, The
University of Sydney, Lidcombe, Australia
John E Pierce. 494 Glen Huntly Road, Elsternwick 3185, Australia. +61430 130 893. ORCID
identifier: 0000-0001-5164-5106 Twitter:@johnpierce85 pierce.john.e@gmail.com
Maya Menahemi-Falkov. Health Sciences 1, La Trobe University, Plenty Road, Bundoora
3083, Australia. mayam10@gmail.com
Robyn O’Halloran. Health Sciences 1, La Trobe University, Plenty Road, Bundoora 3083,
Australia. ORCID identifier: 0000-0002-2772-2164 R.OHalloran@latrobe.edu.au
Leanne Togher. Faculty of Health Sciences, The University of Sydney, 75 East St, Lidcombe
2141, Australia. ORCID identifier: 0000-0002-4518-6748 Leanne.togher@sydney.edu.au
*Miranda L. Rose. Health Sciences 1, La Trobe University, Plenty Road, Bundoora 3083,
Australia. ORCID identifier: 0000-0002-8892-0965 M.rose@latrobe.edu.au
Disclosure of interest: The authors report no conflicts of interest
Constraint and Multimodal approaches to therapy for chronic aphasia: A
systematic review and meta-analysis
Aphasia is a significant cause of disability and reduced quality of life. Two speech
pathology treatment approaches appear efficacious; Multimodal and Constraint Induced
aphasia therapies. In Constraint Induced therapies, nonverbal actions (e.g. gesture,
drawing) are believed to interfere with treatment and patients are therefore constrained
to speech. In contrast, Multimodal therapies employ non-verbal modalities to cue word
retrieval. Given the clinical and theoretical implications, a comparison of these two
divergent treatments was sought. This systematic review investigated both approaches
in chronic aphasia at the levels of impairment, participation and quality of life. After a
systematic search, the level of evidence and methodological quality were rated. Meta-
analysis was conducted on 14 single case experimental designs using Tau-U, while
heterogeneity in the four group designs precluded meta-analysis. Results showed that
high-quality research was limited; however, findings were broadly positive for both
approaches with neither being judged as clearly superior. Most studies examined
impairment-based outcomes without considering participation or quality of life. The
application and definition of constraint varied significantly between studies. Both
constraint and multimodal therapies are promising for chronic post-stroke aphasia, but
there is a need for larger, more rigorously conducted studies. The interpretation of
“constraint” also requires clearer reporting.
Keywords: systematic review; aphasia; constraint; multimodal; therapy
Word count: 8050 word (inc. citations)
Introduction
The presence of aphasia after stroke results in significantly poorer quality of life than stroke
alone (Hilari, 2011). Aphasia is perceived as more detrimental for quality of life than any
other illness including cancer, Alzheimer’s Disease and quadriplegia (Lam & Wodchis,
2010). There are also significant financial and carer burdens (Flowers, Silver, Fang, Rochon,
& Martino, 2013; Patrício, Jesus, & Cruice, 2013) and thus, effective treatments for aphasia
are highly sought after.
Constraint and multimodal are two treatment approaches in aphasia. Multimodal
treatments have a long history of use in aphasia research and treatment, while constraint
principles were first introduced in 2001. However, these two approaches have theoretically
distinct rationales. Authors of constraint therapies such as Constraint Induced Aphasia
Therapy (CIAT) posit that the use of other communication modalities distract from, and
therefore weaken, recovery of verbal output, while authors of multimodal therapies suggest
that these additional modalities can facilitate word retrieval and learning. This results in a key
difference between treatments regarding the cueing of patient responses. These two therapy
approaches and their rationales will be described before their evidence is compared.
Multimodal
The concept of multimodal cueing in aphasia rehabilitation dates back at least to the 1940s,
when Luria put forward the principle of Intersystemic Reorganisation (Luria, 1970). This
principle proposes that a defective system can be supported and supplemented by another,
less damaged, system. Luria gave the example of recruiting the visual system to compensate
for impaired proprioception or balance during walking. In language, the impaired system
(spoken word retrieval) might be assisted by gesture production, for example. It is important
to note that in multimodality therapies, other modalities are used to facilitate spoken output
and not to replace it, though compensation may be a secondary goal if improvement of
spoken output is not successful. The most common communicative modalities used to
promote word retrieval and their proposed mechanisms are described below.
Reading.
Orthographic cues, typically manifested as first letter cues, are widely used in aphasia speech
pathology practice (Lorenz & Nickels, 2007). Research has found orthographic cues to be
generally successful for spoken naming (Nickels, 2002). The orthographic cue is thought to
provide complete or partial activation of the phonological form of the target word using
grapheme-phoneme conversion (Lorenz & Nickels, 2007); in other words, the letter/s provide
an internal phonemic cue to facilitate word production.
Writing.
As with reading, writing takes advantage of the deep links between graphemes and
phonemes. When a patient writes all or part of a target word, this might provide an alternative
route to the phonological output lexicon (a store of all phonological forms of known words)
via the reading route(s) described above (Nickels, 1992). Alternatively, the connection
between the orthographic and phonological output lexicons may be bidirectional, meaning
that phonological information is active whenever written word forms are accessed, as well as
vice versa (Kiran, 2005). This connection is proposed based on the fact that written naming
alone can improve spoken naming (DeDe, Parris, & Waters, 2003; Wright, Marshall, Wilson,
& Page, 2008).
Gesture.
The lexical retrieval hypothesis holds that, even in non-aphasic speakers, gestures used in
conversation are more for the speaker’s benefit than the listener’s in that they aid in word
retrieval (Beattie & Shovelton, 2006). Evidence for this hypothesis comes from findings that
a) more gestures are produced during word finding difficulties in the speech of both normal
and aphasic individuals, and b) restricting gestures during speech increases the frequency of
dysfluencies (Rose, 2006). There is “intense theorising” about exactly how gestures assist
with word finding with language (Hadar & Rumiati, 2006, p. 141), but two possible
mechanisms are described here.
Language is traditionally viewed as a discrete system in the brain. Embodied
language, a subset of embodied cognition, is the theory that language is connected to action
and sensory systems and there is experimental support for this theory (Fischer & Zwaan,
2008). For example, one study showed that reading of “action” words was immediately
followed by activation of a relevant motor cortex area, such as the word “kick” activating the
leg motor areas (Pulvermüller & Berthier, 2008). It follows from such close motor-language
connections that where a word cannot be produced, its corresponding gesture might aid
retrieval. The other explanation is that gesture aids the speaker in preverbal message
planning. That is, gesture helps the speaker mentally arrange the spatial and visual thoughts
behind a message, and this stimulates processing of semantic features which assist word
retrieval (Feyereisen, 2006).
Drawing.
In aphasia, drawing has typically been used to augment or compensate for speech loss
(Sacchett, 2002). However, there are case reports of individuals using drawing to self-cue
verbal word retrieval, or individuals whose naming has improved after treatment targeting
drawing accuracy (Cubelli, 1995). Farias, Davis and Harrington (2006) explored drawing as a
facilitator of word finding in 22 people with aphasia. While participants were drawing the
target, confrontation naming improved compared to both baseline and attempts at written
naming. Interestingly, this effect was not influenced by drawing quality (as measured by
recognisability ratings), suggesting that it could be a suitable treatment even for those with
limb apraxia or significant hemiplegia.
How would drawings assist word retrieval? Drawings, like gesture, have the
advantage of being free from linguistic symbolism. Drawing quality in aphasia correlates
with the integrity of the semantic system (Farias et al., 2006), which suggests that the process
of drawing requires access to semantic features. For naming, drawing is thought to stimulate
the semantic aspects of objects by placing attention on visual features (Farias et al., 2006) and
may do so for longer than naming attempts alone (Makuuchi, Kaminaga, & Sugishita, 2003).
For example, in drawing a truck, a person needs to focus more deeply on features such as its
large size, square shape and additional wheels, and perhaps its function, than when merely
naming it from sight. This process might suppress competing concepts whose features do not
match the target while activating sufficient semantic features to assist word retrieval.
Music.
Melodic Intonation Therapy (MIT; Sparks, Helm, & Albert, 1974) is probably the most
widely used and recognised music-related therapy for aphasia. MIT utilises “intoned speech”,
a song-like prosody, for phrases and utterances. Notes can be either high or low depending on
syllable stress, and syllables are rhythmicised. Simultaneous left hand tapping is also
employed during word or phrase production (Zumbansen, Peretz, & Hébert, 2014a). There
are other variations of music and rhythm treatments, including Modified MIT, Singen
Intonation Prosodie Atmung Rhytmusübungen Improvationen (SIPARI) and Speech-Music
Therapy in Aphasia (Hurkmans et al., 2012). As with other multimodal treatments, all these
treatments aim to encourage verbal output. Generalisation to untreated phrases in
conversational speech is the ultimate goal and patients are not expected to sing in everyday
life (Zumbansen, Peretz, & Hébert, 2014a).
There are several suggested mechanisms for melodic therapies. Sparks et al. (1974)
were initially uncertain as to how to explain the positive results of MIT, but did not believe
that the right hemisphere was learning to take over language production. Instead, they
proposed that the right hemisphere was assisting the left hemisphere. Tapping in MIT is left
handed for this reason — to encourage activation of the right hemisphere. Neuroimaging
evidence is mixed for the theory of increased right hemisphere activation in MIT, with some
showing increased perilesional left hemisphere activation and others, increased right
hemisphere activation (Zumbansen, Peretz, & Hébert, 2014a). Even if present, right
hemisphere activity might only increase due to the high intensity of “singing” that occurs
during such treatments, with the improvements in speech due to the repetition of phrases.
That is, right hemisphere activity and verbal improvement could be independent events
(Stahl, Kotz, Henseler, Turner, & Geyer, 2011).
A more recent hypothesis is that the rhythmic component of these treatments,
particularly the tapping of the left hand, is the true underlying mechanism (Stahl et al 2011).
This has some experimental support (Zumbansen, Peretz, & Hébert, 2014a), while other
studies found superiority of combined rhythm and pitch (Zumbansen, Peretz, & Hébert,
2014b).
Combined Multimodal
Multiple non-verbal modalities may be combined within a treatment to maximise cueing of
verbal output. M-MAT (Multi-modality Aphasia Therapy) is a high intensity combined
multimodal treatment (Attard, Rose, & Lanyon, 2013) which utilises a structured cueing
hierarchy of gesture, drawing and writing to cue word retrieval. A structured and detailed
protocol delineates that each time a patient is unable to produce a target, they are asked to
gesture, draw and write or copy the word while repeating it verbally. M-MAT has shown
improvements across receptive and expressive language measures, at both impairment and
activity/participation levels (Rose, Attard, Mok, Lanyon, & Foster, 2013).
Constraint
At the heart of constraint therapy lies the concept of learned nonuse. Taub and colleagues
developed the term based on observation of monkeys with deafferented upper limbs (Taub,
1976). They proposed that the deficit from an injury is not wholly due to the physiological
impairment, but also a subconscious preference not to use the affected body part (Taub,
Uswatte, Mark, & Morris, 2006). They hypothesised that nonuse is learned through (a)
punishment when trying to use the affected limb (e.g. issues with incoordination or
dropping), and (b) positive reinforcement when using the alternative limb. A “vicious spiral”
(Taub et al., 2006, p. 245) of nonuse then commences as reduced use leads to shrinkage of
the relevant cortical area, which results in less use of the limb, and so on. However, by
constraining function to the damaged limb, through either restraining the unaffected limb or
presenting tasks which necessitate the use of both limbs, improvements in the function of the
deafferented upper limbs were seen.
Constraint Induced Movement Therapy (CIMT) was subsequently developed to
address learned nonuse in adult stroke patients. The key elements of CIMT are intensive
training, use of functional transfer tasks, and constraint applied to the affected limb (Taub et
al., 2006). The efficacy of CIMT has been demonstrated extensively in upper and lower limbs
in stroke as well as other conditions (Smania, 2006; Taub et al., 2006).
CIAT was developed through combining the principles of CIMT with an existing
aphasia treatment, Communicative Aphasia Therapy (Pulvermüller & Berthier, 2008), which
employs language games that rely on the correct verbal response of patients. First reported in
2001 (Pulvermüller et al., 2001), CIAT has three primary principles: 1. Massed practice over
a short period, 2. Action-embedded, relevant language, and 3. Constraint to possible, but
avoided, verbal output. It is the third principle that addresses learned nonuse. In the context of
language, learned nonuse is proposed to occur where patients avoid problematic words or
phrases, reduce their attempts at verbal communication overall, or use alternative modalities
to compensate, such as gesture or writing. In CIAT, constraint is applied through progressive
difficulty of stimuli, gradual shaping of responses into more complex utterances, and
prohibiting nonverbal modes of communication.
More recently, the potentially detrimental effects of inhibiting multimodal self-cueing
have been recognised in CIAT and its use has now been permitted (Difrancesco,
Pulvermüller, & Mohr, 2012). Even so, any multimodal cues used within CIAT remain
incidental and patient-generated, in contrast to the systematic, clinician-prompted cueing of
multimodal treatments. A number of variations on the original CIAT protocol have been
described, including CIAT II (Johnson et al., 2014) and CIAT Plus (Attard et al., 2013;
Meinzer, Djundja, Barthel, Elbert, & Rockstroh, 2005). CIAT Plus builds upon CIAT by
assigning home tasks in order to improve carryover of language skills into real life and using
written stimuli as well as photographs (Meinzer et al., 2005). CIAT II uses a wider variety of
language activities than CIAT, including a role-playing task, picture description and
repetition drills (Johnson et al., 2014).
Summary
As outlined, constraint aphasia therapies seek to avoid learned nonuse of language
partly through constraining participants to the verbal modality, whereas multimodal
treatments seek to leverage intact modalities to aid verbal output.
The most recent Cochrane review of aphasia therapy called for further data comparing
different therapies in order to identify the most effective treatments (Brady, Kelly, Godwin,
Enderby, & Campbell, 2016). A comparison of constraint and multimodal approaches will
improve outcomes for people with chronic aphasia if one is found to be more effective (Rose
et al., 2013). If patients clearly respond better to being constrained to the verbal modality,
then use of multimodal cues should be re-examined. Conversely, if patients improve more
with multimodal cues, the contribution of verbal constraint in the CIAT protocol may be
questioned.
CIAT and M-MAT are two treatments that are operationally similar despite being
constraint and multimodal treatments, respectively. Both use group language games which
provide a social imperative for successful communication, have a high intensity of treatment
(30 hours over two weeks), and use shaping of responses to gradually increase the complexity
of utterances. To date, CIAT and M-MAT have been directly compared in two studies. In a
pilot study (Attard et al., 2013), a single case crossover design was used to provide CIAT
Plus and M-MAT to two participants. Confrontation naming of treated items was marginally
superior for M-MAT compared to CIAT Plus. In a phase 1 trial (Rose et al., 2013), 11
participants underwent both treatments in a multiple baseline crossover design. Both
produced strong positive effects for the primary outcome measure (confrontation naming),
with comparable mean effect sizes (M-MAT = 8.00, CIAT Plus = 8.58) according to Busk
and Serlin’s d (Busk & Serlin, 1992). These similar results in direct comparisons are
puzzling given the contrasting nature of cues provided.
In a narrative review of constraint and multimodal treatments, Rose (2013) examined
the theoretical explanations for constraint and multimodal treatments in detail as well as
critically examining the literature to date. The review concluded that there was limited
theoretical support for constraining people with aphasia to the verbal modality and that doing
so was potentially counterproductive for word retrieval. The research examined within the
review did not favour either approach. However, as a narrative review, the literature reviewed
was not exhaustive and was not subjected to meta-analysis.Therefore, the purpose of this
review was to systematically examine and compare the efficacy of constraint and multimodal
therapies more broadly than CIAT Plus and M-MAT. There were three key questions. Two
examined outcomes based on the World Health Organisation’s International Classification of
Functioning, Disability and Health (ICF). Quality of life was also considered, frequently
described as a missing component of the ICF and under consideration for inclusion in future
versions of the ICF (Ravenek, Skarakis-Doyle, Spaulding, Jenkins, & Doyle, 2015). Finally,
the outcome of carer burden was included due to the significant negative impacts of aphasia
on carers (Patrício et al., 2013).
For stroke-induced chronic aphasia, what is the influence of constraint and
multimodal treatments on measures of (1) language impairment, (2) communication
activity/participation, and (3) quality of life and carer burden?
Definitions
Multimodal therapy or multimodal training has many variations in speech pathology and
even within the field of aphasiology, so it is important to describe our definition for this
systematic review. In this paper we use multimodal therapy to mean treatments which expect
the patient to produce output in different modalities alongside speech production in order to
cue speech. Our own operationalised definition of multimodal therapy also formed the basis
for our inclusion criteria:
(1) The patient carries out a communication task (particularly writing, gesture, singing, or
drawing) at the same time as or immediately before an attempt at speech.
(2) Speech targets are practised repeatedly in a therapy task with the intention of
improving spontaneous speech in the long term rather than as a short-term facilitation
effect.
Based on this definition, studies looking at a simultaneous action that is not
communicative are not eligible. There is a series of research looking at the effects of
“intention gesture”, for example, where participants make a nonsymbolic, circular gesture
with their left hand while repeating the target word. These are not eligible as the nonsymbolic
gesture is designed to activate the right hemisphere and is not related to the target word in a
linguistic sense. Outside of this restriction we included any gesture, as classified in McNeill’s
(1992) model which differentiates gesticulation, pantomime, emblems and sign languages.
MIT and other melodic treatments were included as per the above definition as they
combine the modalities of singing and speech in a song-like prosody. Studies investigating
the use of rhythm on language without a melodic component do not meet our definition as
rhythm is not a communicative act in isolation.
Studies including orthographic cueing (i.e., reading the target word or part of it) as
their only multimodal cue were not included as they are so commonly used as to include the
majority of the aphasia literature. We also limited the writing studies to those focussing on
writing or typing the whole target word and not those that taught phoneme-grapheme
correspondences as an explicit strategy.
Table 1. Search strategy example – Medline (OVID, 1946-present)
Note. All subject headings were exploded, and all subheadings were
included.Method
Search terms relating to aphasia, constraint and multimodal therapies (including drawing,
writing, gesture and music) were used. An example of the search strategy employed is
displayed in Table 1. Three major databases were searched in September 2015: Medline
(OVID, 1946-present), CINAHL (Ebscohost) and Psycinfo (OVID, 1987-present). No limits
for date of publication were applied as we wanted to find as many relevant articles as
possible.
Keywor
ds:
aphasia, dysphasia,
anomia,
NOT "primary progressiv
e"
AN
D
Therapy,
Intervent
ion,
Treatme
nt
AN
D
CIAT*, CILT*,
"Constraint in
duced
language",
“constraint
induced
aphasia",
“Constraint
language",
“Constraint
aphasia",
“Forced
language
treatment",
ILAT,
“intensive lan
guage action"
O
R
M-MAT, MMAT,
"Multi-
modality aphasia th
erapy",
multimodal*, cross
modal*, Draw*,
Amerind, Amer-
ind, Pantomim*,
Sign, signs, signing,
Makaton, Gestur*,
gestic*, iconic,
Writ*, graphem*,
orthograph*,
Read*, music*,
melod*, sing,
singing, rhythm
OR
Subjec
t
headin
gs:
Aphasia, Anomia,
NOT Aphasia, primary pr
ogressive
Duplicate results were excluded using citation software and manual checking.
Resulting titles and abstracts were then screened by each of the first two authors as per the
following inclusion criteria:
Original data
Experimental design
Article from peer reviewed journal, text in English
Adults (18 years +) with stroke-induced, chronic aphasia (≥6 months)
Uses constraint to verbal output or uses multimodal therapy (see earlier definition)
Purposefully investigates effects on verbal output
If a study included participants who did not meet the demographic criteria, it was
excluded unless results for eligible participants could be separated.
After the screening process, the first two authors met to achieve consensus on
discrepancies regarding inclusion of studies. Full texts of the included articles were obtained
and checked again according to the inclusion criteria. Any discrepancies were discussed until
consensus was reached. The screening process is visualised in Figure 1. This selection
process resulted in 60 papers.
The final articles were
categorised according to
study type (Oxford Centre for
Evidence Based Medicine -
Levels of Evidence; 2011),
treatment type (constraint or
modality type) and outcome
(Impairment,
Activity/Participation,
Quality of life, Carer burden).
Author ROH resolved
discrepancies during this
process. The OCEBM levels
do not have a manual that
clearly describes categories
and the Level “Case series”
presumably includes pre/post
group studies, case reports and Single Case Experimental Designs (SCEDs) that are not the
traditional, medical N-of-1 design. However, there is a vast difference in rigour between
SCEDs, which can provide evidence of cause and effect of a treatment, and other single
participant studies such as case reports (Tate et al., 2013) or pre/post group designs.
Therefore, we have included an additional level of evidence for SCEDs, below randomised
trials and above non-randomised trials and have used this method in our categorisation. This
more accurately represents the levels of rigour of study designs. Using the guidelines in Tate
Figure 1. Flow diagram of search results
et al. (2013), this category included any multiple baseline, alternating treatments,
withdrawal/reversal or changing criterion designs. It also included quasi-experimental bi-
phasic (AB) designs.
Outcomes were classified as Activity/Participation where they rated or measured
performance in either real or simulated everyday life activities. Quality of life outcomes were
those using subjective ratings of life satisfaction, while carer burden outcomes were any that
proposed to measure the carer’s wellbeing or distress of any sort. An extraction template was
created and included data fields for sample size, treatment, details of cueing and constraint
used, and outcome measures. The completed data extraction for all 60 articles is included in
the appendix.
Randomised controlled trials (RCTs) and nonrandomised controlled trials (nonRCTs)
were assessed for methodological quality using the PEDro-P, an adapted version of the
PEDro scale (Sherrington, Herbert, Maher, & Moseley, 2000) used by the PsycBITE and
speechBITE teams (see Fitzpatrick, 2008). Ratings by the first author were compared against
those found on SpeechBITE and PsycBITE and any discrepancies were resolved with an
independent rater from the SpeechBITE team.
The RoBiNT scale (Tate et al., 2015) was used to assess methodological quality for
SCEDs. Non-experimental single case designs, including one phase designs, pre/post designs
and case descriptions, were not rated. Pre/post group designs were also not rated as these
have no experimental control and form a low level of evidence. Each study was rated by two
authors and discrepancies were discussed until agreement was reached.
For RCTs and nonRCTs, scores five and above on PEDro-P were considered
moderate to high quality based on commonly accepted consensus (Centre for Evidence-Based
Physiotherapy, 2016). For the RoBiNT, benchmarks have not yet been formally established,
but in a paper examining the reliability of the scale on a small number of papers (Tate et al.,
2013), the mean score was 12 with the highest score being 18 (Tate et al., 2015). In lieu of
existing benchmarks, SCEDs that scored 12 or higher were therefore considered moderate to
high quality. Studies that did not reach these quality cutoff scores (≥5 and ≥12) are included
in the Appendix but they were not considered further in the results.
Effect size calculation
Group Designs
We planned to calculate effect sizes for relevant group studies of Level 2 or 3 evidence
(RCTs and nonRCTs); however, studies were too heterogeneous in terms of outcome
measures and treatments (see results).
Single Case Experimental Designs
Calculation of effect sizes for SCEDs is a domain that continues to develop but it has a
number of promising methods (Parker, Vannest, Davis, & Sauber, 2011). Tau-U is a recent
effect size measure which is resistant to effects of autocorrelation, considers baseline trend,
deals well with only few data points and has the highest power of the non-overlap indices
(Brossart, Vannest, Davis, & Patience, 2014). Tau-U can also produce confidence intervals.
In this review, to be eligible for SCED effect size calculation, studies needed to be an
experimental design (viz., we excluded one phase designs, pre/post designs and case
descriptions), have raw data presented on case charts and have at least 2 data points in each of
the baseline and intervention phases. Only the outcome measure confrontation naming of
treated items was used for effect sizes, which included 94% of SCEDs with a RoBiNT score
of 12 or greater. Data was extracted by the first author and calculated using the online Tau-U
calculator at www.singlecaseresearch.org. A weighted average Tau-U was calculated across
all relevant participants and word sets for each paper. Positive baseline trend was corrected
where baselines had a Tau score of greater than 0.4 (Parker et al., 2011) and an increasing
trend apparent on visual inspection. Negative baseline was not corrected, nor was trend in
treatment phases, as this would have boosted effect sizes. These steps are consistent with
recommendations to correct for trend conservatively and to check statistical results against
visual analysis (Brossart et al., 2014; Parker et al., 2011). Maintenance and follow up phases
were not included in calculations.
Results
As outlined in Figure 1, of 1680 original results (1251 non-duplicates), 60 papers met the
criteria for this systematic review. Figure 2 summarises the number of studies for each
treatment approach.
Figure 2. Number of results by treatment approach
Descriptive characteristics of data
Overall, there were more multimodal treatment studies than constraint (36 vs. 24) but more
constraint studies than studies on any single modality. The first constraint paper was
published in 2001 (Pulvermüller et al.), while the earliest multimodal paper was published in
1975 (Sparks et al.), demonstrating the rapid growth in constraint therapy research.
No studies were identified that used drawing alone as a means of cueing verbal
output. Six studies utilised neurological stimulation (e.g. rTMS) or pharmacological
treatments (e.g., memantine) in combination with constraint or multimodal therapies. Two of
these were RCTs (Barbancho et al., 2015; Berthier et al., 2009), one a SCED (Al-Janabi et
al., 2014) and three were pre/post designs (Abo et al., 2012; Martin et al., 2014; Vines,
Norton, & Schlaug, 2011). While results of these mixed treatments were broadly positive, the
contribution of the constraint and multimodal therapies could not be differentiated from the
pharmacological and stimulation treatment aspects. These studies were therefore not
considered further in this review.
The majority of the twenty-four constraint studies reported use of the original CIAT
approach. Other variations included CIAT Plus (Attard et al., 2013; Meinzer et al., 2005),
CIAT II (Johnson et al., 2014), lower intensity CIAT (Goral & Kempler, 2009; Kempler &
Goral, 2011; Maul, Conner, Kempler, Radvanski, & Goral, 2014), or modifications such as
inclusion of grammatical shaping (Faroqi-Shah & Virion, 2009) or drill tasks (Kempler &
Goral, 2011).
The way “constraint” was applied varied considerably and was not well described in
many studies. While all studies constrained communication between participants to the verbal
modality only, three studies explicitly prevented participants from using gestures to self-cue
(Breier, Maher, Novak, & Papanicolaou, 2006; L. M. Maher et al., 2006; Martin et al., 2014)
whereas five studies allowed such gesture (Attard et al., 2013; MacGregor, Difrancesco,
Pulvermüller, Shtyrov, & Mohr, 2015; Meinzer, Streiftau, & Rockstroh, 2007; Mohr,
Difrancesco, Harrington, Evans, & Pulvermüller, 2014). The remaining 19 did not specify
whether they allowed self-cueing or not, including the original CIAT paper (Pulvermüller et
al., 2001). Similarly, while the majority of studies (14) allowed therapists to provide cueing
when necessary, one reported not providing cues (Maul et al., 2014) and the remaining 11
provided no description of whether therapists provided cues.
Figure 3. Number of papers per level (OCEBM levels of evidence)
Note.
Level 4 studies (Case Series) were not eligible for quality evaluation and meta-analysis. In addition to N-
of-1 designs, Level 1 ordinarily includes Systematic Reviews but these were not eligible as they are not original
data.
Figure 3 shows the levels of evidence found for each therapy type according to
OCEBM levels. There were a very limited number of RCTs and nonRCTS – five for
constraint and three for music – however, there were a number of SCEDs (19), particularly in
gesture studies. The remaining papers were low quality designs.
Figure 4 presents the methodological scores for papers for controlled trials (PEDro-P,
4b) and SCEDs (RoBiNT, 4b) and the red line shows the cutoff scores for inclusion in further
analysis. As visualised in Figure 4a, the methodological quality scores ranged from 3-7 on
PEDro-P (/10), with half the eight controlled trials (Level 2-3) below the cutoff score of 5.
Quality scores on the RoBiNT ranged from 9-21 of a possible 30, with three of the 18 SCED
studies (Level 2a) below the cutoff score of 12 (Figure 4b).
Figure 4. Pedro-P and RoBiNT scores
Note. Red line indicates cutoff score for methodological quality.
The Tau-U effect sizes for confrontation naming of treated items across 14 eligible
studies are shown in Figure 5. Many lacked regular intervals between probes and/or had
significant gaps in probing, and these are noted. Interpretation of Tau-U effect sizes is
difficult without benchmarks, which are not available for Tau-U at present. However, Tau-U
scores are an indication of the percentage of data in the treatment phase that has improved
over time compared to baseline (Parker et al., 2011). The limits are -1.0 and 1.0, which would
indicate 100% reduction and improvement in scores respectively, while 0.0 would indicate no
change in scores. A total effect size weighted by study was calculated for each modality.
Studies with more data points, whether due to more participants or more probes during
phases, have a greater weighting (Parker et al., 2011).
Results are described below in relation to the key questions of this review: by
outcome level (Impairment, Activity/Participation, Quality of life, Carer burden) and within
outcome level by treatment type (constraint, multimodal). Only studies Level 3 or higher
which met minimum quality cutoff scores are discussed.
Figure 5. Tau-U meta-analysis
Note. Solid lines -1.0 and 1.0 indicate the possible limits of Tau-U scoresnegative values demonstrate reduced naming performance and positive clues demonstrate improvement. Dotted
lines with diamonds indicate the weighted average for each area or modality. Larger squares indicate larger relative weighting.
CIAT = Constraint Induced Aphasia Therapy, M-MMAT = Multi-Modal Aphasia Therapy, Gest = Gesture, Sem = Semantic
Impairment Outcomes
Constraint Treatment
Impairment-based results are detailed in Table 2. Constraint had the highest number of top
tier studies according to the OCEBM levels; however, only three met quality criteria on the
PEDro-P and all had small sample sizes (range 9 - 27). In addition, two of the three did not
compare constraint to an equivalent non-constraint control. Meinzer, Streiftau and Rockstroh
(2007) compared CIAT to CIAT run by family, and Pulvermüller et al. (2001) compared
CIAT to conventional therapy of equal total hours but lower intensity.Wilssens et al. (2015)
did compare CIAT to equal intensity conventional therapy and included impairment based
assessments as secondary outcome measures.
Of the three constraint SCEDs that met quality criteria, two compared CIAT Plus to
M-MAT (Attard et al., 2013; Rose et al., 2013) while the other (Kurland, Pulvermüller, Silva,
Burke, & Andrianopoulos, 2012) compared CIAT to PACE (Promoting Aphasic
Communicative Effectiveness, Davis, 2005). Figure 5 shows Tau-U effect sizes with
constraint studies at the top followed by multimodal studies. In Attard et al. (2013), the Tau-
U effect size for CIAT Plus (1.0) was greater than M-MAT (0.81), though M-MAT was
within the 95% confidence interval of CIAT Plus. In Rose et al. (2013) the CIAT Plus effect
size was markedly lower than M-MAT (0.24 and 0.68) and the upper and lower confidence
interval bounds just overlapped.
The weighted Tau-U effect size for all three high-quality constraint SCEDs was
0.374, which means that approximately 37% of treatment phase naming scores were higher
than baseline scores. This was the lowest effect size relative to other modalities and was
reduced by Rose et al. (2013) which, due to its larger number of participants and probes, held
a greater weighting. However, Rose et al. (2013) was an outlier and the other three constraint
SCEDs were more in line with the effect sizes of other modalities (see Figure 5), though they
had lower weighting and RoBiNT scores.
As displayed in Table 2, group studies showed improvements on secondary, pre/post
impairment measures such as the Aachen Aphasia Test (AAT; Huber, Poeck, & Willmes,
1984). AAT results in Wilssens et al. (2015) appeared to favour CIAT over traditional
therapy but subtest results were reported without any overall profile scores. There were
indications of positive effects on blinded clinician ratings on the Communicative Activity
Log (CAL; Pulvermüller et al., 2001), while Boston Naming Test changes were variable
between studies (BNT; Kaplan, Goodglass, & Weintraub, 1983). Changes in pre/post
measures within SCEDs were obscured due to the crossover designs.
Multimodal Treatment
Combined Multimodal. There were two studies on combined multimodal treatment that
investigated M-MAT. Both used multiple impairment-based outcomes. As reported above,
the Tau-U effect size for M-MAT in Attard et al. (2013) was 0.81, lower than CIAT Plus but
suggesting a strong effect of the treatment compared to baseline nonetheless. The Tau-U
effect size was 0.68 for Rose et al. (2013), notably higher than CIAT Plus. The combined,
weighted Tau-U for combined multimodal was 0.70, the second highest of all total effect
sizes calculated; however, an effect size based on only two studies is far from conclusive.
Aside from confrontation naming of treated items, other impairment-based measures
were obscured by the difficulty of calculating pre/post changes in crossover designs.
Gesture. All studies using gesture to facilitate verbal output that met the quality and inclusion
criteria for gesture were SCEDs. Most compared the efficacy of gesture to another treatment
(see appendix), including repetition, semantic treatment and intention gestures. All used
impairment outcomes, with most investigating confrontation naming and/or the Western
Aphasia Battery (WAB, Kertesz, 2007).
As all eight studies used confrontation naming of treated items for probes, Tau-U was
calculated for each one. The narrower confidence intervals in some of the Tau-U effect sizes
and high relative weightings reflect the high number of data points, due to more participants
or more probes in each phase (Figure 5). The overall weighted effect size for the eight SCED
studies on gesture was 0.62, indicating that approximately 62% of treatment phase naming
scores were higher than baseline scores. This is promising for the effects of gesture on
confrontation naming of verbs and nouns. However, this included one negative (Ferguson,
Evans, & Raymer, 2012), low (Raymer et al., 2012) and moderate effect size (Rodriguez,
Raymer, & Gonzalez Rothi, 2006). Gesture therefore had the widest range of Tau-U effect
sizes. These lower effect sizes are not attributable to poor quality methodology, as Raymer et
al. (2012) scored 19 on the RoBiNT.
The small number of secondary, impairment-based outcome measures taken before
and after gesture treatment alone showed variable changes. Mean changes were small for
naming batteries. The mean change in those using the WAB Aphasia Quotient (WAB AQ)
reached the critical difference of five for one study (Raymer et al., 2012) but not the other
(Raymer et al., 2006).
Music. Despite 13 articles for music meeting the initial inclusion criteria — the second
highest yielding category — nine were pre/post designs, along with two low quality
nonRCTs. Only two studies with impairment-based outcomes reached adequate
methodological quality (Hough, 2010; Stahl, Henseler, Turner, Geyer, & Kotz, 2013).
While Stahl et al. (2013) aimed to disentangle the contributions of rhythm and singing
in melodic therapy, the results of the rhythm arm are not reported for this review. The singing
group rehearsed common phrases whilst the conventional therapy group received treatment
on other stimuli. After treatment, patients were assessed in their ability to sing and speak in
unison with recordings of both trained phrases and untrained phrases. Written prompts were
also provided. Unsurprisingly, the music group who had practised the trained phrases showed
greater improvement than the conventional therapy group. The conventional therapy group
demonstrated gains on the untrained phrases, however, while the singing group did not
improve.
Hough (2010) used successful repetition of phrases as the primary outcome for their
single participant. Thus, while the data presented in their case chart appears to show
improvement, it is not comparable to confrontation naming tasks, and a Tau-U effect size was
not calculated. Regardless, improvement of repetition performance was highly significant for
both common and personalised phrases (p < 0.0001), and the WAB AQ and CQ (cortical
quotient) showed large improvements (13 and 13.6 respectively).
Neither repetition (Hough, 2010) nor speaking in unison and with prompts (Stahl et
al., 2013) is a test of generative verbal output. These two papers show only that singing
specific phrases improves participants’ ability to produce those phrases with maximum
modelling and prompting.
Writing. There were no group studies utilising writing as a prompt for verbal output. All three
high quality SCEDs used the same primary outcome measure of confrontation naming of
treated items. These SCEDs had a high overall Tau-U effect size of 0.94, greater than all
other modalities and constraint. This is close to the ceiling of 1.0 and suggests that the
majority of treatment probes were improved over baseline.
In regards to pre/post impairment-based outcomes, the two participants in the multiple
baseline design of Wright et al. (2008) demonstrated improvement in pre/post WAB AQ (3.1
and 9.7) and contradictory improvement in pre/post BNT (-3 and 5).
Activity/Participation Outcomes
Constraint Treatment
Four high quality constraint studies included activity/participation outcomes: two RCTs
(Pulvermüller et al., 2001; Wilssens et al., 2015) and two SCEDs (Attard et al., 2013; Rose et
al., 2013). As displayed in Table 3, outcomes included the Communication Effectiveness
Index (CETI; Lomas, Pickard, Bester, & Elbard, 1989), the Amsterdam-Nijmegen Everyday
Language Test (ANELT, Blomert, Kean, Koster, & Schokker, 1994), the Scenario Test, and
one used a customised measure, the CAL.
The improvements on the CAL self-ratings in Pulvermüller et al. (2001) were
significant for constraint and not the control group, but no between-group comparisons were
made and the control group received lower intensity therapy.
Wilssens et al. (2015) was the only controlled trial to compare constraint to a non-
constraint treatment of the same intensity, a Dutch drill-based lexical-semantic therapy
program, BOX . They did not find a statistically significant between group change on the
CETI (p=.332). However, within group changes were significant for the BOX group and not
the CIAT group. The mean CETI change for the BOX group was also above the clinically
significant improvement level of 12 while it was less for the CIAT group (Lomas et al.,
1989). There were no significant between group differences on the ANELT.
There was no clinically significant improvement in CETI scores for CIAT in Attard,
Rose & Lanyon (2013) or in mean changes in Rose et al. (2013). Scenario Test scores
improved in both participants for CIAT in Attard, Rose & Lanyon but the mean change was
negligible for Rose et al.
Multimodal Treatment
Combined Multimodal. The CETI score changes post M-MAT in Attard, Rose and Lanyon
(2013) did not reach the minimum clinically significant change of 12. In Rose et al. (2013)
the mean change in CETI scores was 8.5 (range -2 to 33), again lower than the clinically
significant change. Scenario Test changes in both studies were minimal.
Gesture. There was limited use of activity/participation measures in gesture studies, but the
three studies that did use these were moderate to high quality SCEDs (RoBiNT 14-21).
However, two of these were comparisons of multiple treatments (Boo & Rose, 2011; Rose &
Sussmilch, 2008), and therefore pre/post measures of activity/participation cannot be
considered as they represent changes from all treatments. The third, Raymer et al. (2012),
used family member ratings on two measures — the CETI and the Functional Outcomes
Questionnaire for Aphasia (FOQ-A; Glueckauf et al., 2003). Changes on the CETI were
inconsistent. Two participants had a negative change — one clinically significant — and two
had a positive change — one clinically significant. The mean change was 5.20 (range -15 to
33). On the FOQ-A changes were positive but small, with only one score changing greater
than one standard deviation from the original FOQ-A paper (Glueckauf et al., 2003). The
mean change was 0.44 (-0.19 to 1.21).
Music. A single high quality study investigated activity/participation outcomes in a multiple
baseline design (Hough, 2010). The CETI was completed by the participant’s caregiver and
improved by 28.2, which is well above the clinically significant change of 12. The ASHA
FACS (American Speech-Language Hearing Association Functional Assessment of
Communication Skills; Frattali, Thompson, Holland, & Wohl, 1995) improvement was
2.05/7 (to our knowledge there are no established benchmarks for clinically significant
change for the ASHA FACS).
Writing. No high quality studies on writing used activity/participation outcomes.
Quality of Life/Carer Burden Outcomes
Carer Burden
No studies were retrieved that used assessments of carer burden.
Constraint Treatment
In two constraint studies (Attard et al., 2013; Rose et al., 2013), quality of life was measured
using the Stroke and Aphasia Quality of Life Scale-39 (SAQOL-39; Hilari, Byng, Lamping,
& Smith, 2003), but no others investigated this domain. Both administered SAQOL-39 before
and after their crossover design comparing CIAT Plus and M-MAT. As they were not
measured between crossover of treatments, it is not possible to attribute changes to one of M-
MAT or CIAT Plus.
Multimodal Treatment
Combined Multimodal. As reported above, Rose et al. (2013) and Attard, Rose and Lanyon
(2013) did not measure the SAQOL-39 between CIAT and M-MAT, therefore individual
contributions of the treatments cannot be determined.
Gesture. No gesture studies were found that investigated quality of life.
Music. One study meeting quality criteria investigated quality of life (Hough, 2010). The
single participant demonstrated a 25 point increase on the ASHA QCL (Paul et al., 2003) — a
64% improvement from baseline score.
Writing. No writing studies investigated quality of life.
Discussion
The aim of this systematic review was to examine and compare evidence for
constraint and multimodal therapies in chronic aphasia. Such a review is important for the
treatment of people living with aphasia long term and for the evidence-based application of
learned nonuse and multimodal cueing. The effectiveness of these approaches also inform our
theoretical understanding of language processing. This review shows that there is limited
high quality evidence to support the use of either constraint or multimodal approaches for
impairment, activity/participation and quality of life outcomes. The amount and strength of
evidence varied between communication modalities.
Impairment outcomes
There were few high-quality studies comparing constraint therapies to equivalent
intensity controls, and for those that did, results did not favour constraint. Indeed, Tau-U
scores for one study favoured multimodal over constraint. The existing research therefore
fails to demonstrate superiority of CIAT over any non-constraint treatment in chronic
aphasia. Comparisons aside, overall evidence does suggest positive effects for impairment
outcomes in constraint, but data is far from conclusive and further research is required.
There is scant evidence for combined multimodal treatment with only two SCED
studies. While this preliminary evidence has positive results in impairment measures with an
effect size of 0.702 for naming, the effect size could change with further research. Changes in
noun production during semi-structured conversation also hinted at positive outcomes, but
more research is indicated before any conclusions can be drawn.
Gesture evidence came from high quality SCED papers, yet effect sizes varied widely
for impairment outcomes. Further investigation is needed to explain this variability. In
addition to improvements in confrontation naming, there were signs of positive effects on
other impairment-based assessments. However, these data are only preliminary as results
were inconsistent and obscured at times by crossover designs. Group designs with control
groups would address the limitations of crossover designs and allow direct comparison of
changes on multiple outcome measures.
It is also worth noting that all eight SCEDs for gesture were conducted by Rose or
Raymer. Though a variety of participants and study designs were used, the evidence would be
enhanced with research from other authors. Group designs such as RCTs are also needed to
confirm current findings.
Despite MIT being a well-known treatment for aphasia, in chronic aphasia the
evidence is of low quality in terms of both study design and methodology. Future studies into
music-based treatment of verbal output in aphasia need to employ more rigorous research
designs. Rather than investigating speech production in unison or repetition, research should
investigate the presumed end goal of such treatments — the independent production of
trained words or phrases — and probe for generalisation to discourse and conversation.
Results for impairment outcomes from writing are very positive thus far but
inconclusive due to low replication. It is possible that research specifically examining the
effects of writing on verbal output are limited because pairing writing with speech is already
widely accepted and frequently embedded in cueing hierarchies. Nonetheless, further
research is necessary to confirm the impact of writing on verbal output, especially group
studies.
No studies were found investigating drawing. Research on this promising modality is
needed.
In summary, there is encouraging but insufficient evidence for constraint or
multimodal treatments on impairment-based measures, in terms of both quality and quantity.
Activity & Participation
In group studies on constraint therapy, there was insufficient evidence for effects on
Activity/Participation following treatment, both in terms of pre/post improvement and
between-group comparisons. CIAT Plus and M-MAT changes in the CETI, though not
clearly attributable to either treatment due to the crossover designs, suggested positive
outcomes, again with high variability amongst participants. In gesture studies, the limited
results for Activity/Participation measures were contradictory and inconclusive. Improvement
and deterioration of scores effectively cancelled each other out, with disparity even between
self and carer improvement ratings at times. In music studies, there was only quality data for
a single participant, though this was clinically and statistically significant.
Quality of Life and Carer Burden
Quality of life outcomes were rarely investigated. A subjective improvement in a person’s
life experience should be a goal of aphasia therapy, yet in this review, only five studies
employed quality of life outcomes, and only three were high quality.
Similarly, the effect of treatment on carer burden remains unexplored. Carers of
people with aphasia experience changes such as loneliness, anxiety, increased responsibilities
and need for support (Patrício et al., 2013), and supporting them should also be a crucial goal
of rehabilitation (Rombough, Howse, & Bartfay, 2006). Yet the impact of treatment on carers
and family was not investigated in any study.
Future research should include both quality of life and carer burden as outcome
measures.
Systematic review design
In this systematic review, we chose to include high quality SCEDs. The inclusion of SCEDs
allowed closer inspection of research areas that would have returned no results if a traditional
evidence hierarchy was used. We made this decision based on increasing recognition that
high quality SCEDs can have equal or even superior rigour to RCTs (e.g. Medical N-of-1
designs) (Tate et al., 2016).
Use of the RoBiNT scale allowed us to rank SCEDs according to specific features that
contribute to internal and external validity. This approach is more fine-grained than simply
classifying SCEDs by design subtype or as experimental/non-experimental. As RoBiNT is a
relatively new scale there is not yet an established cutoff for what constitutes a quality study.
Our use of 12 as the cutoff was based on early uses of this new scale. While this formed a
limited empirical basis, the widely accepted cutoff of 5 for the PEDro/PEDro-P is also based
on common scores (Teasell et al., 2007). Neither approach considers relative weighting of
individual items. Nevertheless, higher scoring studies will have stronger methodological
quality and internal validity and in our results there were no borderline papers on the RoBiNT
that were excluded.
As far as we are aware, this is the first time that the Tau-U effect size calculations
have been applied within a systematic review of aphasia treatments. The Tau-U effect size
provided ranking of SCEDs based on improved data points in the treatment phases, after
correction for baseline trend. These effect sizes allowed comparisons between papers in this
study but we know of no external benchmarks for Tau-U effect sizes. In addition, many
SCEDs analysed with Tau-U had gaps in treatment probe intervals (e.g. washout periods
between crossover trials) or irregular probing, which may have influenced the validity of
resulting effect sizes. Another challenge in utilising Tau-U is that there is no agreed protocol
yet; for example, the threshold for applying baseline trend correction varies between studies.
The first author therefore combined visual inspection with the baseline Tau to eliminate
overcorrection (see methods). While this did introduce a subjective element to calculation,
the overall process remains a defensible calculation of effect sizes in a family of designs
previously resistive to meta-analysis.
Finally, the exclusion of non-English articles is an unfortunate, but in our case,
unavoidable, criterion. It is possible that relevant research published in languages other than
English was missed by this systematic review.
Treatment reporting
Interventions should be reported thoroughly to allow future replication and synthesis of
results (Hoffmann, Glasziou, Boutron, & Milne, 2014). A problem noted across retrieved
constraint studies, regardless of quality or design, was the disparity in what was considered to
constitute constraint. While the original authors have recently clarified that self-cueing with
actions (e.g., gesture) is permitted as long as it is not communicative (Difrancesco et al.,
2012), prior to this publication some protocols banned gesture and the majority made no
comment on this aspect, including the original CIAT article (Pulvermüller et al., 2001).
Likewise, whether or not therapists provided cues to participants was often not described in
methods and is not addressed in Difrancesco, Pulvermüller and Mohr (2012). The term
“constraint” therefore currently represents therapies with significant procedural differences.
Without clearer reporting of methods, there is a risk of continued bleeding of the term
“constraint” to an increasingly diverse range of game-based language treatments. All future
constraint-based research should provide comprehensive description of methods and state
explicitly in which ways they depart from the outline in Difrancesco, Pulvermüller and Mohr
(2012). A template such as TIDieR (Hoffmann et al., 2014) also provides a framework for
thorough reporting. Without this detail, it will be difficult to determine the aspects of CIAT
that contribute to effectiveness.
Outcome measures
The majority of studies in this review focused on improvements at the Impairment level.
However, outcomes in aphasia research should be those that are important to people with
aphasia and their families (Wallace, 2016). Recent work has shown that these include a range
of outcomes across the ICF as well as quality of life and patient satisfaction with treatment
(Wallace et al., 2016). While these constructs can be difficult to measure directly, especially
in people with aphasia (e.g.; Szaflarski et al., 2015), without the inclusion of such outcomes
in future, the benefit of these treatments to people living with aphasia will remain unknown.
There was also a high number of different outcomes within this review. Heterogeneity
of outcome measures reduces research efficiency by limiting synthesis and meta-analysis of
results, which is an important way to overcome the small sample sizes that are common in
aphasia research (Brady et al., 2016). The development of a core outcome for aphasia
research set is currently underway which will recommend preferred outcomes measures for
the constructs identified as important to those with aphasia and other stakeholders (Wallace,
Worrall, Rose, & Le Dorze, n.d.). The core outcome set should be adhered to in future
research wherever possible.
Conclusion
Overall, this review has found a limited evidence base for constraint therapy in chronic
aphasia, especially in proportion to its prominence in research and clinical practice. While
studies indicated positive outcomes, there is a need for rigorous high level studies comparing
CIAT and its derivatives to non-constraint therapies or controls. We also found a very limited
evidence base for multimodal therapies. Studies on some modalities had limited research of
any quality (drawing, writing, combined multimodal) while others had more research but
little of adequate quality (music, gesture).
Accordingly, there is insufficient data to suggest superiority of either constraint or
multimodal approaches in chronic aphasia. There were not enough comparable high-quality
group studies to perform meta-analysis. Meta-analysis of SCEDs favoured multimodal
treatments but this is not yet conclusive.
In addition, there was insufficient examination of “real world” endpoints. Aphasia
research needs to expand beyond the use of basic impairment outcomes such as confrontation
naming and toward consistent outcome measures based on the wishes of people with aphasia
and their families.
Examination of constraint and multimodalities against control treatments of equal
intensity and duration are needed, as well as direct, rigorous comparison between constraint
and multimodal treatments. It is further recommended that future research provides a
comprehensive description of treatment methods and readily accessible treatment materials
arising from clinical trials to enable translation into practice.
Clinicians should not adopt either treatment approach exclusively until further
research is published demonstrating the superiority of one treatment, or, more likely, the
suitability of each to particular patient characteristics.
Acknowledgements
This work was supported by an Australian Government Research Training Program
Scholarship.
References
Abo, M., Kakuda, W., Watanabe, M., Morooka, A., Kawakami, K., & Senoo, A. (2012).
Effectiveness of low-frequency rTMS and intensive speech therapy in poststroke patients
with aphasia: A pilot study based on evaluation by fMRI in relation to type of aphasia.
European Neurology, 68(4), 199–208. https://doi.org/10.1159/000338773
Al-Janabi, S., Nickels, L. A., Sowman, P. F., Burianová, H., Merrett, D. L., & Thompson, W.
F. (2014). Augmenting melodic intonation therapy with non-invasive brain stimulation to
treat impaired left-hemisphere function: Two case studies. Frontiers in Psychology,
5(37), 1–12. https://doi.org/10.3389/fpsyg.2014.00037
Attard, M. C., Rose, M. L., & Lanyon, L. E. (2013). The comparative effects of Multi-
Modality Aphasia Therapy and Constraint-Induced Aphasia Therapy-Plus for severe
chronic Broca's aphasia: An in-depth pilot study. Aphasiology, 27(1), 80–111.
https://doi.org/10.1080/02687038.2012.725242
Ball, A. L., de Riesthal, M., Breeding, V. E., & Mendoza, D. E. (2011). Modified ACT and
CART in severe aphasia. Aphasiology, 25(6-7), 836–848.
https://doi.org/10.1080/02687038.2010.544320
Barbancho, M. A., Berthier, M. L., Navas-Sanchez, P., Dàvila, G., Green-Heredia, C.,
Garcia-Alberca, J. M., et al. (2015). Bilateral brain reorganization with memantine and
constraint-induced aphasia therapy in chronic post-stroke aphasia: An ERP study. Brain
and Language, 145-146, 1–10. https://doi.org/10.1016/j.bandl.2015.04.003
Beattie, G., & Shovelton, H. (2006). A critical appraisal of the relationship between speech
and gesture and its implications for the treatment of aphasia. International Journal of
Speech-Language Pathology, 8(2), 134–139.
https://doi.org/10.1080/14417040600667392
Beeson, P. M., & Egnor, H. (2006). Combining treatment for written and spoken naming.
Journal of the International …, 12, 816–827.
https://doi.org/10.10170S1355617706061005
Berthier, M. L., Green, C., Lara, J. P., Higueras, C., Barbancho, M. A., Dàvila, G., &
Pulvermüller, F. (2009). Memantine and constraint-induced aphasia therapy in chronic
poststroke aphasia. Annals of Neurology, 65(5), 577–585.
https://doi.org/10.1002/ana.21597
Blomert, L., Kean, M. L., Koster, C., & Schokker, J. (1994). Amsterdam—Nijmegen
everyday language test: construction, reliability and validity. Aphasiology, 8(4), 381–407.
https://doi.org/10.1080/02687039408248666
Bonakdarpour, B., Eftekharzadeh, A., & Ashayeri, H. (2003). Melodic Intonation Therapy in
Persian aphasic patients. Aphasiology, 17(1), 75–95.
https://doi.org/10.1080/02687030244000464
Boo, M., & Rose, M. L. (2011). The efficacy of repetition, semantic, and gesture treatments
for verb retrieval and use in Broca's aphasia. Aphasiology, 25(2), 154–175.
https://doi.org/10.1080/02687031003743789
Brady, M. C., Kelly, H., Godwin, J., Enderby, P., & Campbell, P. (2016). Speech and
language therapy for aphasia following stroke. The Cochrane Database of Systematic
Reviews, 6(6), CD000425–CD000425.
https://doi.org/10.1002/14651858.CD000425.pub4
Breier, J. I., Juranek, J., & Papanicolaou, A. C. (2011). Changes in maps of language function
and the integrity of the arcuate fasciculus after therapy for chronic aphasia. Neurocase,
17(6), 506–517. https://doi.org/10.1080/13554794.2010.547505
Breier, J. I., Juranek, J., Maher, L. M., Schmadeke, S., Men, D., & Papanicolaou, A. C.
(2009). Behavioral and neurophysiologic response to therapy for chronic aphasia.
Archives of Physical Medicine and Rehabilitation, 90(12), 2026–2033.
https://doi.org/10.1016/j.apmr.2009.08.144
Breier, J. I., Maher, L. M., Novak, B., & Papanicolaou, A. C. (2006). Functional imaging
before and after constraint-induced language therapy for aphasia using
magnetoencephalography. Neurocase, 12(6), 322–331.
https://doi.org/10.1080/13554790601126054
Breier, J. I., Maher, L. M., Schmadeke, S., Hasan, K. M., & Papanicolaou, A. C. (2007).
Changes in language-specific brain activation after therapy for aphasia using
magnetoencephalography: A case study. Neurocase, 13(3), 169–177.
https://doi.org/10.1080/13554790701448200
Breier, J. I., Randle, S., Maher, L. M., & Papanicolaou, A. C. (2010). Changes in maps of
language activity activation following melodic intonation therapy using
magnetoencephalography: two case studies. Journal of Clinical and Experimental
Neuropsychology, 32(3), 309–314. https://doi.org/10.1080/13803390903029293
Brossart, D. F., Vannest, K. J., Davis, J. L., & Patience, M. A. (2014). Incorporating
nonoverlap indices with visual analysis for quantifying intervention effectiveness in
single-case experimental designs. Neuropsychological Rehabilitation, 24(3-4), 464–491.
https://doi.org/10.1080/09602011.2013.868361
Busk, P. L., & Serlin, R. C. (1992). Meta-analysis for single case research. In T. R.
Kratochwill & J. R. Levin (Eds.), Single-Case Research Design and Analysis: New
Directions for Psychology and Education (pp. 187–212). Hillsdale, NJ: Lawrence
Erlbaum Associates.
Carragher, M., Sage, K., & Conroy, P. (2013). The effects of verb retrieval therapy for people
with non-fluent aphasia: Evidence from assessment tasks and conversation.
Neuropsychological Rehabilitation, 23(6), 846–887.
https://doi.org/10.1080/09602011.2013.832335
Centre for Evidence-Based Physiotherapy. (2016). PEDro Statistics. Retrieved June 14, 2016,
from http://www.pedro.org.au/english/downloads/pedro-statistics/
Cubelli, R. (1995). More on drawing in aphasia therapy. Aphasiology, 9(1), 78–83.
https://doi.org/10.1080/02687039508248693
Davis, G. A. (2005). PACE revisited. Aphasiology, 19(1), 21–38.
https://doi.org/10.1080/02687030444000598
DeDe, G., Parris, D., & Waters, G. (2003). Teaching self-cues: A treatment approach for
verbal naming. Aphasiology, 17(5), 465–480.
https://doi.org/10.1080/02687030344000094
Difrancesco, S., Pulvermüller, F., & Mohr, B. (2012). Intensive language-action therapy
(ILAT): The methods. Aphasiology, 26(11), 1317–1351.
https://doi.org/10.1080/02687038.2012.705815
Farias, D., Davis, C., & Harrington, G. (2006). Drawing: Its contribution to naming in
aphasia. Brain and Language, 97(1), 53–63. https://doi.org/10.1016/j.bandl.2005.07.074
Faroqi-Shah, Y., & Virion, C. R. (2009). Constraint-induced language therapy for
agrammatism: Role of grammaticality constraints. Aphasiology, 23(7-8), 977–988.
https://doi.org/10.1080/02687030802642036
Ferguson, N. F., Evans, K., & Raymer, A. M. (2012). A comparison of intention and
pantomime gesture treatment for noun retrieval in people with aphasia. American Journal
of Speech-Language Pathology, 21(2), s126–s139.
Feyereisen, P. (2006). How could gesture facilitate lexical access? International Journal of
Speech-Language Pathology, 8(2), 128–133.
https://doi.org/10.1080/14417040600667293
Fischer, M. H., & Zwaan, R. A. (2008). Embodied language: A review of the role of the
motor system in language comprehension. The Quarterly Journal of Experimental
Psychology, 61(6), 825–850. https://doi.org/10.1080/17470210701623605
Fitzpatrick, R. B. (2008). PsycBITE™: Psychological Database for Brain Impairment
Treatment Efficacy. Journal of Electronic Resources in Medical Libraries, 5(2), 171–
178. https://doi.org/10.1080/15424060802064428
Flowers, H. L., Silver, F. L., Fang, J., Rochon, E., & Martino, R. (2013). The incidence, co-
occurrence, and predictors of dysphagia, dysarthria, and aphasia after first-ever acute
ischemic stroke. Journal of Communication Disorders, 46(3), 238–248.
https://doi.org/10.1016/j.jcomdis.2013.04.001
Frattali, C. M., Thompson, C. M., Holland, A. L., & Wohl, C. B. (1995). ASHA functional
assessment of communication skills (FACS). Rockville, MD: American Speech-
Language Hearing Association.
Glueckauf, R. L., Blonder, L. X., Ecklund-Johnson, E., Maher, L. M., Crosson, B., &
Gonzalez-Rothi, L. (2003). Functional Outcome Questionnaire for Aphasia: overview
and preliminary psychometric evaluation. NeuroRehabilitation, 18(4), 281–290.
Goldfarb, R., & Bader, E. (1979). Espousing melodic intonation therapy in aphasia
rehabilitation: A case study. International Journal of Rehabilitation Research, 2(3), 333–
342.
Goral, M., & Kempler, D. (2009). Training verb production in communicative context:
Evidence from a person with chronic non-fluent aphasia. Aphasiology, 23(12), 1383–
1397. https://doi.org/10.1080/02687030802235203
Hadar, U., & Rumiati, R. I. (2006). I gesture therefore I speak: Cartesian cures. International
Journal of Speech-Language Pathology, 8(2), 140–142.
https://doi.org/10.1080/14417040600667301
Hilari, K. (2011). The impact of stroke: are people with aphasia different to those without?
Disability and Rehabilitation, 33(3), 211–218.
https://doi.org/10.3109/09638288.2010.508829
Hilari, K., Byng, S., Lamping, D. L., & Smith, S. C. (2003). Stroke and Aphasia Quality of
Life Scale-39 (SAQOL-39): Evaluation of Acceptability, Reliability, and Validity.
Stroke, 34(8), 1944–1950. https://doi.org/10.1161/01.STR.0000081987.46660.ED
Hillis, A. E. (1989). Efficacy and generalization of treatment for aphasic naming errors.
Archives of Physical Medicine & Rehabilitation, 70(8), 632–636.
Hoffmann, T. C., Glasziou, P. P., Boutron, I., & Milne, R. (2014). Better reporting of
interventions: template for intervention description and replication (TIDieR) checklist
and guide. Bmj.
Hough, M. S. (2010). Melodic Intonation Therapy and aphasia: Another variation on a theme.
Aphasiology, 24(6-8), 775–786. https://doi.org/10.1080/02687030903501941
Huber, W., Poeck, K., & Willmes, K. (1984). The Aachen Aphasia Test. Advances in
Neurology, 42, 291–303.
Hurkmans, J., de Bruijn, M., Boonstra, A. M., Jonkers, R., Bastiaanse, R., Arendzen, H., &
Reinders-Messelink, H. A. (2012). Music in the treatment of neurological language and
speech disorders: A systematic review. Aphasiology, 26(1), 1–19.
https://doi.org/10.1080/02687038.2011.602514
Johnson, M. L., Taub, E., Harper, L. H., Wade, J. T., Bowman, M. H., Bishop-McKay, S., et
al. (2014). An enhanced protocol for constraint-induced aphasia therapy II: A case series.
American Journal of Speech-Language Pathology, 23(1), 60–72.
https://doi.org/10.1044/1058-0360(2013/12-0168)
Kaplan, E. F., Goodglass, H., & Weintraub, S. (1983). The Boston naming test. Philadelphia,
PA: Lea & Febiger.
Kavian, S., Khatoonabadi, A. R., Ansari, N. N., Saadati, M., & Shaygannejad, V. (2014). A
single-subject study to examine the effects of constrained-induced aphasia therapy on
naming deficit. International Journal of Preventive Medicine, 5(6), 782–786.
Kempler, D., & Goral, M. (2011). A comparison of drill- and communication-based treatment
for aphasia. Aphasiology, 25(11), 1327–1346.
https://doi.org/10.1080/02687038.2011.599364
Kertesz, A. (2007). Western Aphasia Battery (Revised). San Antonio, TX: PsychCorp.
Kiran, S. (2005). Training phoneme to grapheme conversion for patients with written and oral
production deficits: A model-based approach. Aphasiology, 19(1), 53–76.
https://doi.org/10.1080/02687030444000633
Kurland, J., Baldwin, K., & Tauer, C. (2010). Treatment-induced neuroplasticity following
intensive naming therapy in a case of chronic Wernicke's aphasia. Aphasiology, 24(6-8),
737–751. https://doi.org/10.1080/02687030903524711
Kurland, J., Pulvermüller, F., Silva, N., Burke, K., & Andrianopoulos, M. (2012).
Constrained versus unconstrained intensive language therapy in two individuals with
chronic, moderate-to-severe aphasia and apraxia of speech: Behavioral and fMRI
outcomes. American Journal of Speech-Language Pathology, 21(2), S65–S87.
https://doi.org/10.1044/1058-0360(2012/11-0113)
Lam, J. M. C., & Wodchis, W. P. (2010). The Relationship of 60 Disease Diagnoses and 15
Conditions to Preference-Based Health-Related Quality of Life in Ontario Hospital-
Based Long-Term Care Residents. Medical Care, 48(4), 380–387.
https://doi.org/10.1097/MLR.0b013e3181ca2647
Lim, K.-B., Kim, Y.-K., Lee, H.-J., Yoo, J., Hwang, J. Y., Kim, J.-A., & Kim, S.-K. (2013).
The therapeutic effect of neurologic music therapy and speech language therapy in post-
stroke aphasic patients. Annals of Rehabilitation Medicine, 37(4), 556–562.
https://doi.org/10.5535/arm.2013.37.4.556
Lomas, J., Pickard, L., Bester, S., & Elbard, H. (1989). The Communicative Effectiveness
Index: Development and Psychometric Evaluation of a Functional Communication
Measure for Adult Aphasia. Journal of Speech and …, 54, 113–124.
Lorenz, A., & Nickels, L. A. (2007). Orthographic cueing in anomic aphasia: How does it
work? Aphasiology, 21(6-8), 670–686. https://doi.org/10.1080/02687030701192182
Luria, A. R. (1970). Traumatic aphasia: Its syndromes, psychology and treatment. (D.
Bowden, Trans.). The Hague, Netherlands: Mouton & Co.
MacGregor, L. J., Difrancesco, S., Pulvermüller, F., Shtyrov, Y., & Mohr, B. (2015). Ultra-
rapid access to words in chronic aphasia: The effects of intensive language action therapy
(ILAT). Brain Topography, 28(2), 279–291. https://doi.org/10.1007/s10548-014-0398-y
Maher, L. M., Kendall, D. L., Swearengin, J. A., Rodriguez, A., Leon, S. A., Pingel, K., et al.
(2006). A pilot study of use-dependent learning in the context of Constraint Induced
Language Therapy., 12(6), 843–852. https://doi.org/10.1017/S1355617706061029
Makuuchi, M., Kaminaga, T., & Sugishita, M. (2003). Both parietal lobes are involved in
drawing: a functional MRI study and implications for constructional apraxia. Cognitive
Brain Research, 16(3), 338–347. https://doi.org/10.1016/S0926-6410(02)00302-6
Marangolo, P., Bonifazi, S., Tomaiuolo, F., Craighero, L., Coccia, M., Altoè, G., et al.
(2010). Improving language without words: First evidence from aphasia.
Neuropsychologia, 48(13), 3824–3833.
https://doi.org/10.1016/j.neuropsychologia.2010.09.025
Martin, P. I., Treglia, E., Naeser, M. A., Ho, M. D., Baker, E. H., Martin, E. G., et al. (2014).
Language improvements after TMS plus modified CILT: Pilot, open-protocol study with
two, chronic nonfluent aphasia cases. Restorative Neurology and Neuroscience, 32(4),
483–505. https://doi.org/10.3233/RNN-130365
Maul, K. K., Conner, P. S., Kempler, D., Radvanski, C., & Goral, M. (2014). Using
informative verbal exchanges to promote verb retrieval in nonfluent aphasia. American
Journal of Speech-Language Pathology, 23(3), 407–420.
https://doi.org/10.1044/2014_AJSLP-13-0004
McNeill, D. (1992). Hand and Mind. Chicago, IL: University of Chicago Press.
Meinzer, M., Djundja, D., Barthel, G., Elbert, T., & Rockstroh, B. (2005). Long-term stability
of improved language functions in chronic aphasia after constraint-induced aphasia
therapy. Stroke, 36(7), 1462–1466.
https://doi.org/10.1161/01.STR.0000169941.29831.2a
Meinzer, M., Streiftau, S., & Rockstroh, B. (2007). Intensive language training in the
rehabilitation of chronic aphasia: Efficient training by laypersons., 13(5), 846–853.
https://doi.org/10.1017/S1355617707071111
Mohr, B., Difrancesco, S., Harrington, K., Evans, S., & Pulvermüller, F. (2014). Changes of
right-hemispheric activation after constraint-induced, intensive language action therapy
in chronic aphasia: fMRI evidence from auditory semantic processing. Frontiers in
Human Neuroscience, 8(309), 919. https://doi.org/10.3389/fnhum.2014.00919
Morrow-Odom, K. L., & Swann, A. B. (2013). Effectiveness of melodic intonation therapy in
a case of aphasia following right hemisphere stroke. Aphasiology, 27(11), 1322–1338.
https://doi.org/10.1080/02687038.2013.817522
Nickels, L. A. (1992). The autocue? self-generated phonemic cues in the treatment of a
disorder of reading and naming. Cognitive Neuropsychology, 9(2), 155–182.
https://doi.org/10.1080/02643299208252057
Nickels, L. A. (2002). Therapy for naming disorders: Revisiting, revising, and reviewing.
Aphasiology, 16(10-11), 935–979. https://doi.org/10.1080/02687030244000563
OCEBM Levels of Evidence Working Group. (2011). The Oxford Levels of Evidence 2.
Oxford Centre for Evidence-Based Medicine. Retrieved from
http://www.cebm.net/index.aspx?o=5653
Parker, R. I., Vannest, K. J., Davis, J. L., & Sauber, S. B. (2011). Combining Nonoverlap and
Trend for Single-Case Research: Tau-U. Behavior Therapy, 42(2), 284–299.
https://doi.org/10.1016/j.beth.2010.08.006
Patrício, B., Jesus, L., & Cruice, M. (2013). Quality of life of the caregivers of people with
aphasia. A systematic review.
Paul, D. R., Frattali, C. M., Holland, A. L., Thompson, C. K., Caperton, C. J., & Slater, S. C.
(2003). ASHA Quality of Communication Life Scale (QCL). Rockville, MD: American
Speech-Language-Hearing Association.
Pulvermüller, F., & Berthier, M. L. (2008). Aphasia therapy on a neuroscience basis.
Aphasiology, 22(6), 563–599. https://doi.org/10.1080/02687030701612213
Pulvermüller, F., Hauk, O., Zohsel, K., Neininger, B., & Mohr, B. (2005). Therapy-related
reorganization of language in both hemispheres of patients with chronic aphasia.
NeuroImage, 28(2), 481–489. https://doi.org/10.1016/j.neuroimage.2005.06.038
Pulvermüller, F., Neininger, B., Elbert, T., Mohr, B., Rockstroh, B., Koebbel, P., & Taub, E.
(2001). Constraint-induced therapy of chronic aphasia after stroke. Stroke, 32(7), 1621–
1626. https://doi.org/10.1161/01.STR.32.7.1621
Ravenek, M. J., Skarakis-Doyle, E., Spaulding, S. J., Jenkins, M. E., & Doyle, P. C. (2015).
Enhancing the conceptual clarity and utility of the international classification of
functioning, disability & health: the potential of a new graphic representation. Disability
and Rehabilitation, 35(12), 1015–1025. https://doi.org/10.3109/09638288.2012.717582
Raymer, A. M., McHose, B., Smith, K. G., Iman, L., Ambrose, A., & Casselton, C. (2012).
Contrasting effects of errorless naming treatment and gestural facilitation for word
retrieval in aphasia. Neuropsychological Rehabilitation, 22(2), 235–266.
https://doi.org/10.1080/09602011.2011.618306
Raymer, A. M., Singletary, F., Rodriguez, A., Ciampitti, M., Heilman, K. M., & Gonzalez
Rothi, L. J. (2006). Effects of gesture+verbal treatment for noun and verb retrieval in
aphasia. Journal of the International Neuropsychological Society, 12(6), 867–882.
https://doi.org/10.10170S1355617706061042
Richter, M., Miltner, W. H. R., & Straube, T. (2008). Association between therapy outcome
and right-hemispheric activation in chronic aphasia. Brain, 131(Pt 5), 1391–1401.
https://doi.org/10.1093/brain/awn043
Rodriguez, A. D., Raymer, A. M., & Gonzalez Rothi, L. J. (2006). Effects of gesture+verbal
and semantic-phonologic treatments for verb retrieval in aphasia. Aphasiology, 20(2-4),
286–297. https://doi.org/10.1080/02687030500474898
Rombough, R. E., Howse, E. L., & Bartfay, W. J. (2006). Caregiver strain and caregiver
burden of primary caregivers of stroke survivors with and without aphasia. Rehabilitation
Nursing : the Official Journal of the Association of Rehabilitation Nurses, 31(5), 199–
209.
Rose, M. L. (2006). The utility of arm and hand gestures in the treatment of aphasia.
Advances in Speech Language Pathology, 8(2), 92–109.
https://doi.org/10.1080/14417040600657948
Rose, M. L. (2013). Releasing the constraints on aphasia therapy: the positive impact of
gesture and multimodality treatments. American Journal of Speech-Language Pathology,
22(2), S227–39. https://doi.org/10.1044/1058-0360(2012/12-0091)
Rose, M. L., & Douglas, J. (2008). Treating a semantic word production deficit in aphasia
with verbal and gesture methods. Aphasiology, 22(1), 20–41.
https://doi.org/10.1080/02687030600742020
Rose, M. L., & Sussmilch, G. (2008). The effects of semantic and gesture treatments on verb
retrieval and verb use in aphasia. Aphasiology, 22(7-8), 691–706.
https://doi.org/10.1080/02687030701800800
Rose, M. L., Attard, M. C., Mok, Z., Lanyon, L. E., & Foster, A. M. (2013). Multi-modality
aphasia therapy is as efficacious as a constraint-induced aphasia therapy for chronic
aphasia: A phase 1 study. Aphasiology, 27(8), 938–971.
https://doi.org/10.1080/02687038.2013.810329
Rose, M. L., Douglas, J., & Matyas, T. (2002). The comparative effectiveness of gesture and
verbal treatments for a specific phonologic naming impairment. Aphasiology, 16(10-11),
1001–1030. https://doi.org/10.1080/02687030143000825
Sacchett, C. (2002). Drawing in aphasia: moving towards the interactive. International
Journal of Human-Computer Studies, 57(4), 263–277.
https://doi.org/10.1006/ijhc.2002.1018
Schlaug, G., Marchina, S., & Norton, A. (2008). From singing to speaking: why singing may
lead to recovery of expressive language function in patients with broca's aphasia. Music
Perception, 25(4), 315–323. https://doi.org/10.1525/MP.2008.25.4.315
Sherrington, C., Herbert, R. D., Maher, C. G., & Moseley, A. M. (2000). PEDro. A database
of randomized trials and systematic reviews in physiotherapy. Manual Therapy, 5(4),
223–226. https://doi.org/10.1054/math.2000.0372
Smania, N. (2006). Constraint-induced movement therapy: an original concept in
rehabilitation.
Sparks, R., Helm, N., & Albert, M. (1974). Aphasia rehabilitation resulting from melodic
intonation therapy. Cortex, 10(4), 303–316.
Stahl, B., Henseler, I., Turner, R., Geyer, S., & Kotz, S. A. (2013). How to engage the right
brain hemisphere in aphasics without even singing: Evidence for two paths of speech
recovery. Frontiers in Human Neuroscience, 7(Article 35), 1–12.
https://doi.org/10.3389/fnhum.2013.00035
Stahl, B., Kotz, S. A., Henseler, I., Turner, R., & Geyer, S. (2011). Rhythm in disguise: why
singing may not hold the key to recovery from aphasia. Brain, 134(Pt 10), 3083–3093.
https://doi.org/10.1093/brain/awr240
Sugishita, M., Seki, K., Kabe, S., & Yunoki, K. (1993). A material-control single-case study
of the efficacy of treatment for written and oral naming difficulties. Neuropsychologia,
31(6), 559–569.
Szaflarski, J. P., Ball, A. L., Vannest, J., Dietz, A. R., Allendorfer, J. B., Martin, A. N., et al.
(2015). Constraint-Induced Aphasia Therapy for Treatment of Chronic Post-Stroke
Aphasia: A Randomized, Blinded, Controlled Pilot Trial. Medical Science Monitor, 21,
2861–2869. https://doi.org/10.12659/MSM.894291
Szaflarski, J. P., Ball, A., Grether, S., Al-fwaress, F., Griffith, N. M., Neils-Strunjas, J., et al.
(2008). Constraint-induced aphasia therapy stimulates language recovery in patients with
chronic aphasia after ischemic stroke. Medical Science Monitor, 14(5), CR243–CR250.
Tate, R. L., Perdices, M., Rosenkoetter, U., Shadish, W., Vohra, S., Barlow, D. H., et al.
(2016). The Single-Case Reporting Guideline In BEhavioural Interventions (SCRIBE)
2016 Statement. Aphasiology, 30(7), 862–876.
https://doi.org/10.1080/02687038.2016.1178022
Tate, R. L., Perdices, M., Rosenkoetter, U., Wakim, D., Godbee, K., Togher, L., &
McDonald, S. (2013). Revision of a method quality rating scale for single-case
experimental designs and n-of-1 trials: The 15-item Risk of Bias in N-of-1 Trials
(RoBiNT) Scale. Neuropsychological Rehabilitation, 23(5), 619–638.
https://doi.org/10.1080/09602011.2013.824383
Tate, R. L., Rosenkoetter, U., Wakim, D., Sigmundsdottir, L., Doubleday, J., Togher, L., et
al. (2015). The Risk of Bias in N-of-1 Trials (RoBiNT) Scale: An expanded manual for
the critical appraisal of single-case reports. Sydney, Australia: Author.
Taub, E. (1976). Movement in nonhuman primates deprived of somatosensory feedback.
Exercise and Sport Sciences Reviews, 4, 335–374.
Taub, E., Uswatte, G., Mark, V. W., & Morris, D. M. (2006). The learned nonuse
phenomenon: implications for rehabilitation. Europa Medicophysica, 42(3), 241–255.
Teasell, R., Bayona, N., Marshall, S., Cullen, N., Bayley, M., Chundamala, J., et al. (2007). A
systematic review of the rehabilitation of moderate to severe acquired brain injuries.
Brain Injury, 21(2), 107–112. https://doi.org/10.1080/02699050701201524
van der Meulen, I., van de Sandt-Koenderman, M. E., & Ribbers, G. M. (2012). Melodic
Intonation Therapy: Present controversies and future opportunities. Archives of Physical
Medicine and Rehabilitation, 93(S1), S46–S52.
https://doi.org/10.1016/j.apmr.2011.05.029
Vines, B. W., Norton, A. C., & Schlaug, G. (2011). Non-invasive brain stimulation enhances
the effects of melodic intonation therapy. Frontiers in Psychology, 2(Article 230), 230.
https://doi.org/10.3389/fpsyg.2011.00230
Wallace, S. (2016, December 18). Improving Research Outcome Measurement in Aphasia
(ROMA): Development of a Core Outcome Set. Retrieved from
http://espace.library.uq.edu.au/view/UQ:415571
Wallace, S. J., Worrall, L. E., Rose, T., Le Dorze, G., Cruice, M., Isaksen, J., et al. (2016).
Which outcomes are most important to people with aphasia and their families? an
international nominal group technique study framed within the ICF. Disability and
Rehabilitation, 91, 1–16. https://doi.org/10.1080/09638288.2016.1194899
Wallace, S., Worrall, L. E., Rose, T., & Le Dorze, G. (n.d.). Improving Research Outcome
Measurement in Aphasia (ROMA): Development of a Core Outcome Set. Retrieved
March 13, 2017, from http://www.cometinitiative.org/studies/details/287
Wan, C. Y., Zheng, X., Marchina, S., Norton, A., & Schlaug, G. (2014). Intensive therapy
induces contralateral white matter changes in chronic stroke patients with Broca's
aphasia. Brain and Language, 136, 1–7. https://doi.org/10.1016/j.bandl.2014.03.011
Weill-Chounlamountry, A., Capelle, N., Tessier, C., & Pradat-Diehl, P. (2013). Multimodal
therapy of word retrieval disorder due to phonological encoding dysfunction. Brain
Injury, 27(5), 620–631. https://doi.org/10.3109/02699052.2013.767936
Wilson, S. J., Parsons, K., & Reutens, D. C. (2006). Preserved singing in aphasia: A case
study of the efficacy of Melodic Intonation Therapy. Music Perception, 24(1), 23–36.
Wilssens, I., Vandenborre, D., van Dun, K., Verhoeven, J., Visch-Brink, E., & Marien, P.
(2015). Constraint-induced aphasia therapy versus intensive semantic treatment in fluent
aphasia. American Journal of Speech-Language Pathology, 24(2), 281–294.
https://doi.org/10.1044/2015_AJSLP-14-0018
Wright, H. H., Marshall, R. C., Wilson, K. B., & Page, J. L. (2008). Using a written cueing
hierarchy to improve verbal naming in aphasia. Aphasiology, 22(5), 522–536.
https://doi.org/10.1080/02687030701487905
Zumbansen, A., Peretz, I., & Hébert, S. (2014a). Melodic intonation therapy: back to basics
for future research. Frontiers in Neurology, 5(7), 1–11.
https://doi.org/10.3389/fneur.2014.00007/abstract
Zumbansen, A., Peretz, I., & Hébert, S. (2014b). The combination of rhythm and pitch can
account for the beneficial effect of melodic intonation therapy on connected speech
improvements in Broca's aphasia. Frontiers in Human Neuroscience, 8(Article 592).
https://doi.org/10.3389/fnhum.2014.00592/abstract
Appendix
Summary table of all eligible papers
Treatment
Oxford level
Publication
Title
Treatment(s)
n Outcome type
Constraint
Level 2 - Randomised
trial
(Pulvermüller et al.,
2001)
Constraint-Induced Therapy of Chronic Aphasia After Stroke
CIAT vs. Standard therapy, less intensive
17 (10 constraint) Impairment
Activity/Participation
Constraint
Level 2 - Randomised
trial
(Meinzer et al., 2007)
Intensive language training in the rehabilitation of chronic aphasia: efficient
training by laypersons.
CIAT by therapists vs. CIAT by laypersons
20 Impairment
Constraint
Level 2 - Randomised
trial
(Wilssens et al., 2015)
Constraint-Induced Aphasia Therapy Versus Intensive Semantic Treatment
in Fluent Aphasia
CIAT vs. Standard Therapy
9 (5 constraint) Impairment
Activity/Participation
Constraint
Level 3 - Non-
randomised, controlled
cohort/followup study
(L. M. Maher et al.,
2006)
A pilot study of use-dependent learning in the context of Constraint
Induced Language Therapy.
CIAT vs. PACE
9 (4 constraint) Impairment
Constraint
Level 3 - Non-
randomised, controlled
cohort/followup study
(Meinzer et al., 2005)
Long-term stability of improved language functions in chronic aphasia after
constraint-induced aphasia therapy
CIAT vs. CIAT Plus
27 Impairment
Activity/Participation
Constraint
Level 4 - Case series
(Johnson et al., 2014)
An enhanced protocol for constraint-induced aphasia therapy II: a case
series.
CIAT II
4 Impairment
Activity/Participation
Constraint
Level 4 - Case series
(Faroqi-Shah & Virion,
2009)
Constraint-induced language therapy for agrammatism: Role of
grammaticality constraints
CIAT vs. CIAT-G
4 Impairment
Constraint
Level 4 - Case series
(Kempler & Goral,
2011)
A comparison of drill- and communication-based treatment for aphasia
Generative CIAT vs. Drill CIAT
2 Impairment
Constraint
Level 4 - Case series
(Goral & Kempler,
2009)
Training verb production in communicative context: evidence from a
person with chronic non-fluent aphasia
Modified CIAT (reduced intensity)
1 Impairment
Constraint
Level 4 - Case series
(Maul et al., 2014)
Using informative verbal exchanges to promote verb retrieval in nonfluent
aphasia
Modified CIAT (reduced intensity, modelling of
target sentences)
4 Impairment
Constraint
Level 4 - Case series
(Breier et al., 2009)
Behavioral and neurophysiologic response to therapy for chronic aphasia
CIAT
23 Impairment
Constraint
Level 4 - Case series
(Breier, Juranek, &
Papanicolaou, 2011)
Changes in maps of language function and the integrity of the arcuate
fasciculus after therapy for chronic aphasia
CIAT
1 Impairment
Constraint
Level 4 - Case series
(Breier et al., 2006)
Functional imaging before and after constraint-induced language therapy
for aphasia using magnetoencephalography.
CIAT
6 Impairment
Constraint
Level 4 - Case series
(Breier, Maher,
Schmadeke, Hasan, &
Papanicolaou, 2007)
Changes in language-specific brain activation after therapy for aphasia
using magnetoencephalography: A case study
CIAT
1 Impairment
Constraint
Level 4 - Case series
(MacGregor et al.,
2015)
Ultra-rapid access to words in chronic aphasia: The effects of intensive
language action therapy (ILAT)
CIAT
12 Impairment
Treatment
Oxford level
Publication
Title
Treatment(s)
n Outcome type
Constraint
Level 4 - Case series
(Mohr et al., 2014)
Changes of right-hemispheric activation after constraint-induced, intensive
language action therapy in chronic aphasia: fMRI evidence from auditory
semantic processing.
CIAT
12 Impairment
Constraint
Level 4 - Case series
(Pulvermüller, Hauk,
Zohsel, Neininger, &
Mohr, 2005)
Therapy-related reorganization of language in both hemispheres of
patients with chronic aphasia
CIAT
10 Impairment
Constraint
Level 4 - Case series
(Richter, Miltner, &
Straube, 2008)
Association between therapy outcome and right-hemispheric activation in
chronic aphasia
CIAT
24 (16 constraint) Impairment
Constraint
Level 4 - Case series
(Szaflarski et al., 2008)
Constraint-induced aphasia therapy stimulates language recovery in
patients with chronic aphasia after ischemic stroke
CIAT (individualised goals and stimuli)
3 Impairment
Activity/Participation
Constraint
Level 4 - Case series
(Kurland, Baldwin, &
Tauer, 2010)
Treatment-induced neuroplasticity following intensive naming therapy in a
case of chronic wernicke's aphasia
CIAT vs. PACE
1 Impairment
Constraint
Level 4 - Single case
experimental design
(Kavian, Khatoonabadi,
Ansari, Saadati, &
Shaygannejad, 2014)
A Single-subject Study to Examine the Effects of Constrained-induced
Aphasia Therapy on Naming Deficit.
CIAT
2 Impairment
Constraint
Level 4 - Single case
experimental design
(Kurland et al., 2012)
Constrained Versus Unconstrained Intensive Language Therapy in Two
Individuals With Chronic, Moderate-to-Severe Aphasia and Apraxia of
Speech: Behavioral and fMRI Outcomes.
CIAT vs. PACE
2 Impairment
Combined
Multimodal &
Constraint
Level 4 - Single case
experimental design
(Attard et al., 2013)
The comparative effects of Multi-Modality Aphasia Therapy and Constraint-
Induced Aphasia Therapy-Plus for severe chronic Broca's aphasia: An in-
depth pilot study.
CIAT Plus vs. MMAT
2 Impairment,
Activity/Participation
Quality of Life
Combined
Multimodal &
Constraint
Level 4 - Single case
experimental design
(Rose et al., 2013)
Multi-modality aphasia therapy is as efficacious as a constraint-induced
aphasia therapy for chronic aphasia: A phase 1 study
CIAT Plus vs. MMAT
11 Impairment,
Activity/Participation
Quality of Life
Gesture
Level 4 - Case series
(Carragher, Sage, &
Conroy, 2013)
The effects of verb retrieval therapy for people with non-fluent aphasia:
Evidence from assessment tasks and conversation
Semantic Feature Analysis + Gesture + phonemic
cueing
9 Impairment
Gesture
Level 4 - Case series
(Marangolo et al.,
2010)
Improving language without words: first evidence from aphasia
Action observation vs. Action observation and
execution vs. Action observation and
meaningless movement
6 (5 with stroke) Impairment
Gesture
Level 4 - Single case
experimental design
(Boo & Rose, 2011)
The efficacy of repetition, semantic, and gesture treatments for verb
retrieval and use in Broca's aphasia
Repetition vs. semantic vs. gesture vs. semantic +
gesture
2 Impairment
Activity/Participation
Gesture
Level 4 - Single case
experimental design
(Ferguson et al., 2012)
A comparison of intention and pantomime gesture treatment for noun
retrieval in people with aphasia
Intention gesture vs. pantomime gesture
4 Impairment
Gesture
Level 4 - Single case
experimental design
(Raymer et al., 2012)
Contrasting effects of errorless naming treatment and gestural facilitation
for word retrieval in aphasia
Errorless naming vs. gesture
8 Impairment
Activity/Participation
Gesture
Level 4 - Single case
experimental design
(Raymer et al., 2006)
Effects of gesture+verbal treatment for noun and verb retrieval in aphasia
Gesture + verbal
9 Impairment
Gesture
Level 4 - Single case
experimental design
(Rodriguez et al., 2006)
Effects of gesture+verbal and semantic-phonologic treatments for verb
retrieval in aphasia
Gesture + verbal vs. Semantic + phonologic
4 Impairment
Treatment
Oxford level
Publication
Title
Treatment(s)
n Outcome type
Gesture
Level 4 - Single case
experimental design
(Rose & Douglas, 2008)
Treating a semantic word production deficit in aphasia with verbal and
gesture methods
Verbal vs. Gesture vs. Verbal + gesture
1 Impairment
Gesture
Level 4 - Single case
experimental design
(Rose, Douglas, &
Matyas, 2002)
The comparative effectiveness of gesture and verbal treatments for a
specific phonologic naming impairment
Verbal vs. Gesture vs. Verbal + gesture
1 Impairment
Gesture
Level 4 - Single case
experimental design
(Rose & Sussmilch,
2008)
The effects of semantic and gesture treatments on verb retrieval and verb
use in aphasia
Semantic vs. Gesture vs. Semantic + Gesture vs.
Repetition
3 Impairment
Activity/Participation
Music
Level 3 - Non-
randomised, controlled
cohort/followup study
(Wan, Zheng,
Marchina, Norton, &
Schlaug, 2014)
Intensive therapy induces contralateral white matter changes in chronic
stroke patients with Broca's aphasia
MIT vs. no therapy
20 (11 MIT) Impairment
Music
Level 3 - Non-
randomised, controlled
cohort/followup study
(Lim et al., 2013)
The therapeutic effect of neurologic music therapy and speech language
therapy in post-stroke aphasic patients
MIT vs. Standard Therapy
21 (11 chronic, 6 chronic
and music)
Impairment
Music
Level 4 - Case series
(Bonakdarpour,
Eftekharzadeh, &
Ashayeri, 2003)
Melodic intonation therapy in Persian aphasic patients
MIT (adapted to Farsi)
7 Impairment
Music
Level 4 - Case series
(Breier, Randle, Maher,
& Papanicolaou, 2010)
Changes in maps of language activity activation following melodic
intonation therapy using magnetoencephalography: two case studies.
MIT
2 Impairment
Music
Level 4 - Case series
(Goldfarb & Bader,
1979)
Espousing melodic intonation therapy in aphasia rehabilitation: a case
study
MIT
1 Impairment
Music
Level 4 - Case series
(Morrow-Odom &
Swann, 2013)
Effectiveness of melodic intonation therapy in a case of aphasia following
right hemisphere stroke
MIT
1 Impairment
Activity/Participation
Quality of Life
Music
Level 4 - Case series
(Schlaug, Marchina, &
Norton, 2008)
From singing to speaking: Why singing may lead to recovery of expressive
language function in patients with Broca's aphasia
MIT vs. Speech Repetition Therapy
2 Impairment
Music
Level 4 - Case series
(Sparks et al., 1974)
Aphasia rehabilitation resulting from melodic intonation therapy
MIT
9 Impairment
Music
Level 4 - Case series
(van der Meulen, van
de Sandt-Koenderman,
& Ribbers, 2012)
Melodic Intonation Therapy: Present Controversies and Future
Opportunities
MIT
2 Impairment
Music
Level 4 - Case series
(Wilson, Parsons, &
Reutens, 2006)
Preserved Singing in Aphasia: A Case Study of the Efficacy of Melodic
Intonation Therapy
MIT vs. Rhythmic therapy
1 Impairment
Music
Level 4 - Single case
experimental design
(Hough, 2010)
Melodic intonation therapy and aphasia: Another variation on a theme
Modified MIT (no tapping)
1 Impairment
Activity/Participation
Quality of Life
Music
Level 3 - Non-
randomised, controlled
cohort/followup study
(Stahl et al., 2013)
How to engage the right brain hemisphere in aphasics without even
singing: Evidence for two paths of speech recovery
Therapy vs. Repetition Therapy vs. Rhythmic
Therapy
15 (5 music, 5 rhythm) Impairment
Activity/Participation
Music
Level 4 - Case series
(Zumbansen, Peretz, &
Hébert, 2014b)
The combination of rhythm and pitch can account for the beneficial effect
of melodic intonation therapy on connected speech improvements in
Broca's aphasia
Melodic therapy vs. Rhythmic therapy vs.
Standard Therapy
3 Impairment
Quality of Life
Treatment
Oxford level
Publication
Title
Treatment(s)
n Outcome type
Pharma/stimula
tion
Level 2 - Randomised
trial & Level 4 - Case
series
(Barbancho et al.,
2015)
Bilateral brain reorganization with memantine and constraint-induced
aphasia therapy in chronic post-stroke aphasia: An ERP study.
CIAT + Memantine
28 Impairment
Pharma/stimula
tion
Level 2 - Randomised
trial & Level 4 - Case
series
(Berthier et al., 2009)
Memantine and Constraint-Induced Aphasia Therapy in Chronic Poststroke
Aphasia
CIAT + Memantine
27 Impairment
Activity/Participation
Pharma/stimula
tion
Level 4 - Case series
(Abo et al., 2012)
Effectiveness of Low-Frequency rTMS and Intensive Speech Therapy in
Poststroke Patients with Aphasia: A Pilot Study Based on Evaluation by fMRI
in Relation to Type of Aphasia
Constraint therapy + rTMS
24 Impairment
Pharma/stimula
tion
Level 4 - Case series
(Martin et al., 2014)
Language improvements after TMS plus modified CILT: Pilot, open-protocol
study with two, chronic nonfluent aphasia cases.
Modified CILT + TMS
2 Impairment
Pharma/stimula
tion
Level 4 - Case series
(Vines et al., 2011)
Non-invasive brain stimulation enhances the effects of melodic intonation
therapy
MIT + tDCS
6 Impairment
Pharma/stimula
tion
Level 4 - Single case
experimental design
(Al-Janabi et al., 2014)
Augmenting melodic intonation therapy with non-invasive brain
stimulation to treat impaired left-hemisphere function: two case studies
MIT + rTMS
2 Impairment
Writing
Level 4 - Case series
(Sugishita, Seki, Kabe,
& Yunoki, 1993)
A material-control single-case study of the efficacy of treatment for written
and oral naming difficulties
Naming with written cueing hierarchy
22 (3 chronic) Impairment
Writing
Level 4 - Case series
(Weill-
Chounlamountry,
Capelle, Tessier, &
Pradat-Diehl, 2013)
Multimodal therapy of word retrieval disorder due to phonological
encoding dysfunction
Therapy software Au fil des mots(anagrams,
copying, writing)
1 Impairment
Activity/Participation
Writing
Level 4 - Single case
experimental design
(Ball, de Riesthal,
Breeding, & Mendoza,
2011)
Modified ACT and CART in severe aphasia
ACT + CART
3 Impairment
Writing
Level 4 - Single case
experimental design
(Beeson & Egnor,
2006)
Combining treatment for written and spoken naming
CART + repetition
2 Impairment
Writing
Level 4 - Single case
experimental design
(DeDe et al., 2003)
Teaching self-cues: A treatment approach for verbal naming
Written naming + tactile cueing + verbal naming
1 Impairment
Writing
Level 4 - Single case
experimental design
(Hillis, 1989)
Efficacy and generalization of treatment for aphasic naming errors
Written naming + verbal naming
2 (1 within chronic criteria) Impairment
Writing
Level 4 - Single case
experimental design
(Wright et al., 2008)
Using a written cueing hierarchy to improve verbal naming in aphasia
Written cueing hierarchy based on CART
2 Impairment
Table 2 – Impairment-based outcomes of high-quality studies
Treatment
Authors
Outcomes
Results
Constraint
(Pulvermüller et al., 2001)
AAT (comprehension, repetition,
naming, token test)
CAL (blinded clinician ratings)
Group x Time effect in favour of CIAT (F[1,15]=5.0; P<0.04)
7/10 CIAT participants improved. Significant but statistical testing only conducted on
seven (F[1,6] 10.5, P<0.01). Control group CAL scores not reported.
Constraint
(Meinzer et al., 2007)
AAT
Time effect significant for both groups
Clinicians: t(9) = 7.05, p<.0001
Laypersons: t(9) = 5.65, p<.002
Group x Time effect non-significant (F [1,18] = 1.26; p>.2)
19/20 improved as per critical difference in manual
Constraint
(Wilssens et al., 2015)
AAT
BNT (/60)
SAT
PALPA 51, 49, 8, 5, 6)
All participants improved on at least one subtest, no overall profile scores reported.
CIAT group: Statistically significant improvement on 4/5 subtests
BOX group: Statistically significant improvement on 1/5 subtests
Pre/post CIAT group (p = .004), Pre/post BOX group (p = .094), no between group
testing.
No significant change in 8/9, no between group testing.
Pre/post semantic scores favoured BOX, Pre/post phonological scores favoured CIAT
Constraint
(Attard et al., 2013)
Confrontation naming treated
items
WAB AQ (0-100)
BNT (/60)
Cinderella Narrative Retell
Semi-structured conversation
Tau-U 1.00
No change pre/post CIAT Plus
Non-significant changes pre/post CIAT Plus
No statistically sig. improvement
No statistically sig. improvement
Constraint
(Rose et al., 2013)
Confrontation naming treated
items
Tau-U 0.24
Treatment
Authors
Outcomes
Results
WAB-R AQ (0-100) a
BNT (/60) a
Semi-structured conversation a
Mean change 2.47 (range -3.00 to 7.70)
Mean change 1.9 (range -6.0 to 15.0)
Substantive nouns mean change 4.67 (range -64.0 to 76.0), substantive verbs
unchanged overall
Constraint
(Kurland et al., 2012)
Confrontation naming treated
items
BNT (/60)
BDAE-3
Tau-U 0.69
P1: 25 to 35 (though pre-treatment was 32); P2: 23 to 33
P1: Unchanged; P2: Responsive naming 6 to 11, others largely unchanged
Combined
Multimodal
(Attard et al., 2013)
Confrontation naming treated
items
WAB AQ (0-100)
BNT (/60)
Cinderella Narrative Retell
Semi-structured conversation
Tau-U 0.81
Non-significant changes pre/post M-MAT
Non-significant changes pre/post M-MAT
No statistically sig. improvement
No statistically sig. improvement
Combined
Multimodal
(Rose et al., 2013)
Confrontation naming treated
items
WAB-R AQ (0-100) a
BNT (/60)a
Semi-structured conversation a
Tau-U 0.68
Mean change 1.40 (range -4.2 to 9)
Mean change 2.2 (range -9.0 to 15.0).
Substantive nouns mean change 20.4 (range -24.0 to 87.0), substantive verbs largely
unchanged
Gesture
(Boo & Rose, 2011) b
Confrontation naming treated
items
Tau-U 0.85 (all gesture sets)
Gesture
(Ferguson et al., 2012)
Confrontation naming treated
items
Tau-U -0.04 (pantomime)
Treatment
Authors
Outcomes
Results
Gesture
(Rodriguez et al., 2006) b
Confrontation naming treated
items
Tau-U 0.46 (all gesture sets)
Gesture
(Raymer et al., 2006)
Confrontation naming treated
items
WAB AQ (0-100)
BNT (/60)
ANT (/60)
Tau-U 0.67, one participant’s results not published due to poor response
Mean change 4.8 (p = 0.15)
Mean change 1.2 (range -3 to 9)
Mean change -1.4 (range -11.6 to 7.4)
Gesture
(Rose et al., 2002)
Confrontation naming treated
items
Tau-U 0.74
Gesture
(Raymer et al., 2012)
Confrontation naming treated
items
WAB AQ (0-100) (pre/post
gesture)
BNT (/60) (pre/post gesture)
Tau-U 0.25
Mean change 5.5 (range 0.2 to 16.1)
Mean change 3 (range -15 to 13)
Gesture
(Rose & Sussmilch, 2008) b
Confrontation naming treated
items
Tau-U 0.73
Gesture
(Rose & Douglas, 2008) b
Confrontation naming treated
items
Tau-U 0.91
Music
(Stahl et al., 2013)
All in unison with recordings &
written prompts:
Singing & speaking trained
phrases (% correct)
Singing & speaking untrained
phrases (% correct)
Singing therapy: Mean change = 36.47, 95% CI [28.24, 44.70]; Standard therapy: Mean
change = 4.98, 95% CI [−3.25, 13.21]
Singing therapy: Mean change = −0.36, 95% CI [−2.62, 1.90]; Standard therapy: Mean
change = 6.21, 95% CI [3.96, 8.47]
Treatment
Authors
Outcomes
Results
Music
(Hough, 2010)
Repetition of treated phrases
WAB AQ (0-100)
WAB CQ (0-100)
Pre/post change (p < 0.0001)
Improved 13.0
Improved 13.6
Writing
(Wright et al., 2008)
Confrontation naming treated
items
WAB AQ (0-100)
BNT (/60)
Tau-U 0.90
Pre/post improvement P1: 3.1, P2: 9.7
Pre/post improvement P1: -3; P2: 5
Writing
(Hillis, 1989)
Confrontation naming treated
items
Tau-U 0.99
Writing
(Beeson & Egnor, 2006)
Confrontation naming treated
items
PALPA 53 (/40)
Tau-U 0.86
Unchanged
Note. AAT = Aachen Aphasia Test (Profile score), CAL = Communicative Activity Log, BNT = Boston Naming Test, SAT = Verbal Semantic
Association Test, PALPA = Psycholinguistic Assessment of Language Processing in Aphasia, WAB-R AQ/CQ = Western Aphasia Battery
[Revised] (Aphasia Quotient/Cortical Quotient), BDAE = Boston Diagnostic Aphasia Examination, ANT = Action Naming Test, VAST = Verb
And Sentence Test, OANB = Object Action Naming Battery.
aThe crossover design makes it difficult to isolate the contributions of pre/post outcome measures, but taking a conservative approach, where the
treatment of interest was administered second, the post treatment measure of the first treatment was taken as the baseline.
bPre/post measures represent changes from multiple treatments and so are not listed.
Table 3 – Activity/Participation outcomes of high-quality studies
Treatment
Authors
Outcomes
Results
Constraint
(Pulvermüller et al.,
2001)
CAL (self ratings)
Pre/post CIAT group (F[1,7]=25.0, P<0.001); Pre/post control group (F<1); no between
group comparison.
Constraint
(Wilssens et al., 2015)
CETI (/100) (self and family ratings)
ANELT
No between-group difference in improvements (t(6) = 1.01, p = .332); however:
Pre/post CIAT group (t(4) = 1.47, p = .216), < clinically significant difference
Pre/post BOX group (t(2) = 7.40, p = .019), > clinically significant difference
Statistically significant improvement for both groups; no significant difference
between groups t(7) = 0.85, p = .426
Constraint
(Attard et al., 2013)
CETI (/100)
Scenario test (/54)
Both less than clinically significant difference (12). P1: 3 point increase; P2: 3 point
increase
Improved by 3 and 8 points
Constraint
(Rose et al., 2013)
CETI (/100) a
Scenario test (/54) a
Mean change 4 points (range -3 to 13). 2/11 participants > clinically significant
difference
Mean change -0.1/54 (range -10.0 to 7.70)
Combined
Multimodal
(Attard et al., 2013)
CETI (/100) a
Scenario test (/54)
Both less than clinically significant difference (12). P1: 8 point increase; P2: 9 point
increase
P1: +1 point; P2: -3 points
Combined
Multimodal
(Rose et al., 2013)
CETI (/100) a
Scenario test (/54) a
Mean change 8.5 points (range -2 to 33). 3/11 participants > clinically significant
difference
Mean change 0.80/54 (range -3.0 to 9.0)
Gesture
(Raymer et al., 2012)
CETI (pre/post gesture) (/100) a
FOQ-A (pre/post gesture) (/5) a
Mean change 5.20 points (range -15 to 33). One participant > clinically significant
difference, one participant negative change > clinically significant difference.
Mean change 0.44 (-0.19 to 1.21)
Treatment
Authors
Outcomes
Results
Gesture
(Rose & Sussmilch, 2008)
LCQ (/90)b
n/a
Gesture
(Boo & Rose, 2011)
LCQ (/90)b
n/a
Music
(Hough, 2010)
CETI (caregiver rating) (/100)
ASHA FACS (/7)
28.2 increase (> clinically significant difference)
2.05 increase
Note. ANELT = Amsterdam-Nijmegen Everyday Language Test, CAL = Communicative Activity Log, CETI = Communication Effectiveness
Index, LCQ = La Trobe Communication Questionnaire, FOQ-A = Functional Outcomes Questionnaire for Aphasia, ASHA FACS = American
Speech-Language Hearing Association Functional Assessment of Communication Skills.
aThe crossover design makes it difficult to isolate the contributions of pre/post outcome measures, but taking a conservative approach, where the
treatment of interest was administered second, the post treatment measure of the first treatment was taken as the baseline.
bPre/post measures represent changes from multiple treatments and so are not listed.
Table 4 – Quality of life outcomes of high-quality studies
Treatment
Authors
Outcomes
Results
Constraint
(Attard et al., 2013)
SAQOL-39a
n/a
Constraint
(Rose et al., 2013)
SAQOL-39 a
n/a
Combined
Multimodal
(Attard et al., 2013)
SAQOL-39 a
n/a
Combined
Multimodal
(Rose et al., 2013)
SAQOL-39 a
n/a
Music
(Hough, 2010)
ASHA-QCL (/90)
25 point increase
Note. SAQOL-39 = Stroke and Aphasia Quality of Life Scale-39, ASHA-QCL = American Speech-Language Hearing Association Quality of
Communication Life Scale.
aPre/post measures represent changes from multiple treatments and so are not listed
... Kvalitní výsledky jsou závislé na kontinuitě vhodně zvoleného individualizovaného léčebného postupu a konečně i na synergii interdisciplinárního týmu (1). Nicméně životní spokojenost, motivace, emocionální a sociální podpora (17), aktivní participace samotných pacientů a pečujících rodin patří k faktorům, které významným způsobem ovlivňují samotný léčebný proces (4). ...
... Jen pět studií z celkového počtu 60 zahrnutých studií se věnovalo hodnocení kvality života a pouze v jedné z nich bylo zaznamenáno zvýšení jejího celkového skóre. Dle závěrů autorů Pierce et al. (17) je v této oblasti nutné vytvořit větší a kvalitnější studie. ...
... Zdá se také, že slibnou strategií pro logopedickou rehabilitaci afatiků po CMP je multimodální (M-MAT) a omezením indukovaná afázická terapie (CIAT), i když jistota důkazů je nedostatečná. Tyto přístupy dokážou zlepšit kvalitu života, ale není jasné, který typ poskytované intervence je v rámci léčby výhodnější, protože žádná strategie není v účinnosti nadřazenější té druhé (17). I když programy self-managementu nejsou podpořeny silnými důkazy (nízká až střední kvalita důkazů), je nutno zdůraznit, že představují další možný potenciál studiemi prověřených intervencí k úspěšnému zotavení takto postižených osob žijících v komunitním prostředí (4,27). ...
... CIAT-Plus and M-MAT are hypothesised to rely on different underlying neural recovery mechanisms and may be differentially effective based on aphasia severity. 9 10 Systematic reviews of trials of CIAT-Plus and M-MAT reveal moderate-high effect sizes [11][12][13] but studies are limited by small sample sizes (n<15), inadequate comparator groups, and recruitment and detection bias. Determining the most effective intervention for severitybased and other sub-groups of people with aphasia may lead to improved patient outcomes and reduced healthcare costs. ...
... 21 The primary outcome measure was change in aphasia severity from baseline to immediately post-intervention as assessed by the WAB-R-AQ. Secondary outcomes included change in word retrieval (COMPARE naming battery: treated 80-item set; untreated 100-item set 13 (online supplemental material p.1)), functional communication (Communication Effectiveness Index (CETI) 22 ), multimodal communication (Scenario Test, 23 quality of life (Stroke and Aphasia Quality of Life Scale-39g (SAQOL-39g), 24 and efficiency of connected speech (correct information units (CIUs)/ min. 25 Additional secondary outcomes included maintenance of treatment effects as measured by repeating all outcome measures at a 12-week follow-up visit. ...
Article
Full-text available
Background While meta-analyses confirm treatment for chronic post-stroke aphasia is effective, a lack of comparative evidence for different interventions limits prescription accuracy. We investigated whether Constraint-Induced Aphasia Therapy Plus (CIAT-plus) and/or Multimodality Aphasia Therapy (M-MAT) provided greater therapeutic benefit compared with usual community care and were differentially effective according to baseline aphasia severity. Methods We conducted a three-arm, multicentre, parallel group, open-label, blinded endpoint, phase III, randomised-controlled trial. We stratified eligible participants by baseline aphasia on the Western Aphasia Battery-Revised Aphasia Quotient (WAB-R-AQ). Groups of three participants were randomly assigned (1:1:1) to 30 hours of CIAT-Plus or M-MAT or to usual care (UC). Primary outcome was change in aphasia severity (WAB-R-AQ) from baseline to therapy completion analysed in the intention-to-treat population. Secondary outcomes included word retrieval, connected speech, functional communication, multimodal communication, quality of life and costs. Results We analysed 201 participants (70 in CIAT-Plus, 70 in M-MAT and 61 in UC). Aphasia severity was not significantly different between groups at postintervention: 1.05 points (95% CI −0.78 to 2.88; p=0.36) UC group vs CIAT-Plus; 1.06 points (95% CI −0.78 to 2.89; p=0.36) UC group vs M-MAT; 0.004 points (95% CI −1.76 to 1.77; p=1.00) CIAT-Plus vs M-MAT. Word retrieval, functional communication and communication-related quality of life were significantly improved following CIAT-Plus and M-MAT. Word retrieval benefits were maintained at 12-week follow-up. Conclusions CIAT-Plus and M-MAT were effective for word retrieval, functional communication, and quality of life, while UC was not. Future studies should explore predictive characteristics of responders and impacts of maintenance doses. Trial registration number ACTRN 2615000618550.
... Our focus will be on aphasia therapy studies. It is still unclear which exact forms of therapy and neural activations would produce optimal recovery for any given PWA (Cocquyt et al., 2017;Hartwigsen & Saur, 2019;Kiran, 2012;Pierce et al., 2017). Individual differences in the demographic and lesion factors, as well as variations in specific deficits, mean that definitive prognostic predictions remain very challenging and unlikely to be accurate (Plowman et al., 2012;Tippett, 2015). ...
... In this work, quality of life outcomes, impairment, and activity were assessed. A meta-analysis was impossible due to the methodological discrepancy and too few high-quality studies [41]. ...
Article
Full-text available
Aphasia is one of the most common clinical features of functional impairment after a stroke. Approximately 21–40% of stroke patients sustain permanent aphasia, which progressively worsens one’s quality of life and rehabilitation outcomes. Post-stroke aphasia treatment strategies include speech language therapies, cognitive neurorehabilitation, telerehabilitation, computer-based management, experimental pharmacotherapy, and physical medicine. This review focuses on current evidence of the effectiveness of impairment-based aphasia therapies and communication-based therapies (as well as the timing and optimal treatment intensities for these interventions). Moreover, we present specific interventions, such as constraint-induced aphasia therapy (CIAT) and melodic intonation therapy (MIT). Accumulated data suggest that using transcranial magnetic stimulation (TMS) and transcranial direct current stimulation (tDCS) is safe and can be used to modulate cortical excitability. Therefore, we review clinical studies that present TMS and tDCS as (possible) promising therapies in speech and language recovery, stimulating neuroplasticity. Several drugs have been used in aphasia pharmacotherapy, but evidence from clinical studies suggest that only nootropic agents, donepezil and memantine, may improve the prognosis of aphasia. This article is an overview on the current state of knowledge related to post-stroke aphasia pharmacology, rehabilitation, and future trends.
... Future research using case series should focus on maximizing their robustness and intervention reporting quality. Systematic reviews and metasyntheses of good quality single case studies and case series would also strengthen the field and increase our knowledge concerning the effectiveness of specific interventions for specific aphasias (e.g., Pierce, Menahemi-Falkov, O'Halloran, Togher, & Rose, 2019). Large, well-powered RCTs with good assessment of participants, replicable description of intervention should crucially include post-hoc regression to look at efficacy of different interventions for different aphasia profiles. ...
Article
Background: Recent reviews conclude that aphasia intervention is effective. However, replication and implementation require detailed reporting of intervention is and a specification of participant profiles. To date, reviews concentrate more on efficacy than on intervention reporting quality. Aims : The aim of this project is to review the descriptions of aphasia interventions and participants appearing in recent systematic reviews of aphasia intervention effectiveness. The relationship between the quality of these descriptions and the robustness of research design is explored, and the replicability of aphasia interventions is evaluated. Methods and Procedures : The scope of our search was an analysis of the aphasia intervention studies included in the and EBRSR 2018 systematic reviews, and in the RCSLT 2014 literature synthesis. Intervention descriptions published separately from the intervention study (i.e. published online, in clinical tools, or a separate trial protocols) were not included. The criteria for inclusion were that participants had aphasia, the intervention involved language and/or communication, and included the following research designs: Randomised Controlled Trial (RCT), comparison or control, crossover design, case series. Exclusion criteria included non-SLT interventions, studies involving fewer than four participants, conference abstracts, studies not available in English. Studies were evaluated for completeness of intervention description using the TIDieR Checklist. Additionally, we rated the quality of patient and intervention description, with particular reference to replicability. Outcomes and Results: Ninety-three studies were included. Only 14 studies (15%) had >50 participants. Fifty-six studies (60%) did not select participants with a specific aphasia profile, and a further 10 studies only described participants as non-fluent. Across the studies, an average of eight (of 12) TIDieR checklist items were given but information on where, tailoring, modification and fidelity items was rarely available. Studies that evaluated general aphasia intervention approaches tended to use RCT designs, whereas more specific intervention studies were more likely to use case series designs. Conclusions: Group studies were generally under-powered and there was a paucity of research looking at specific aphasia interventions for specific aphasia profiles. There was a trade-off between the robustness of the design and the level of specificity of the intervention described. While the TIDieR framework is a useful guide to information which should be included in an intervention study, it is insufficiently sensitive for assessing replicability. We consider possible solutions to the challenges of making large-scale trials more useful for determining effective aphasia intervention.
... However, in a paper examining the reliability of the RoBiNT scale [38] the mean score of included studies was 12 points. This score has been used in lieu of formalised benchmarks in a previous systematic review in aphasia [54] and was adopted for this review. ...
Article
Purpose Aphasia is a debilitating chronic acquired language disorder that impacts heavily on a person’s life. Behavioural treatments aim to remediate language processing skills or to enhance communication between the person with aphasia and others, and a number of different treatments are efficacious. However, it is unclear how much of a particular treatment a person needs in order to optimise recovery of language and communication skills following stroke. Materials and methods Systematic search for and meta-analysis of experimental studies that directly compared different amounts of the same behavioural aphasia treatment, following PRISMA guidelines. Results Treatment dose research in aphasia is an emerging area. Just six studies comparing different doses of the same intervention met all criteria for inclusion. Evidence from these studies was synthesised and meta-analysed, where possible. Meta-analyses were inconclusive due to limited data; however, there are indications that suggest increased dose may confer greater improvement on language and communication measures, but with diminishing returns over time. Aphasia severity and chronicity may affect dose–response relationships. Conclusions There is currently insufficient evidence to determine the effect of dose on treatment response. A dedicated and coordinated research agenda is required to systematically explore dose–response relationships in post-stroke aphasia interventions. A video abstract is available in the Supplementary Material. • Implications for rehabilitation • The investigation of the effect of dose on treatment outcomes in post-stroke aphasia is an emerging research area with few studies reporting comparison of different amounts of the same intervention. • In the acute phase of recovery following stroke, higher doses of treatment provided over short periods may not be preferable, tolerable, or superior to lower doses of the same treatment. • In the chronic phase, providing additional blocks of treatment may confer additional benefit for some people with aphasia but with diminishing returns. • People with chronic aphasia can achieve and maintain significant gains in picture naming after a relatively brief period of high-dose treatment.
Article
Purpose The purpose of this review was to examine the effects of aphasia therapy on language and/or communication outcome measures when treatment was initiated within four months post-onset. The review evaluated the methodological quality of relevant studies and summarised the findings of the high-quality studies according to three clinical questions about the provision of aphasia treatment in the early period of recovery: (1) Is treatment better than no treatment? (2) Is one type of treatment more effective than another? (3) Do different treatment intensities result in different outcomes? Methods A literature search was performed for articles in which aphasia treatments were initiated fewer than four months post-aphasia onset and evaluated with a control or comparison group. Two authors rated the studies on defined methodological quality criteria and extracted data for addressing the clinical questions. Results A total of 23 studies met the inclusion criteria. Sixteen of the studies received high-quality ratings. Nine studies provided data addressing clinical question 1; however, only four of them were considered as high-quality studies. Results from the high-quality studies were mixed: two studies demonstrated treatment efficacy for early aphasia therapy, and two studies found no differences in outcome measures between participants who received treatment and a no-treatment control. Eleven studies provided data addressing clinical question 2; six of them were considered as high-quality. None of the eleven treatment-comparison studies found that one type of treatment resulted in greater gains compared to another type of treatment on primary outcome measures. Finally, six studies contributed data for addressing clinical question 3; all of them were considered as high-quality studies. Five studies found no significant difference in outcomes between participants assigned to lower- and higher-intensive weekly treatment schedules, and one study reported superior findings in outcomes when participants received less intensive treatment. Conclusion This review found mixed results across studies that examined whether early aphasia treatment improved language/communication outcomes more than no treatment. The review also found that when different types of aphasia treatments were compared, no treatment was more efficacious than another treatment and that increasing the weekly intensity of treatment beyond 2–5 hours did not improve outcomes on language/communication measures. The review highlights the need for additional research on the effects of early aphasia therapy. We suggest that future research considers participant characteristics that might influence how a person will respond to a specific therapeutic approach and intensity.
Book
Praxiswissen Logopädie Das erfolgreiche Lehrbuch in der 7. Auflage vermittelt Lernenden und Lehrenden in der Logopädie Grundlagen und evidenzbasiertes Wissen für eine Therapie von Aphasie nach ICF. Die detaillierte Beschreibung spezifischer Beeinträchtigungen, z.B. von Alexien, Agraphien und Akalkulien, ermöglicht eine umfassende Diagnostik und Therapie kommunikativer Störungen nach Schlaganfall. Hinweise zur Phase der Rehabilitation und zu Erholungsprozessen des Gehirns helfen gezielt bei der Planung und Durchführung der Aphasietherapie. Zu den einzelnen Störungsbereichen wie beim Sprachverständnis, Wortfindung oder Wortabruf oder der Satzbildung finden Therapeuten bewährte Übungen, Hilfen und ausgewähltes Übungsmaterial. Auch auf Krankheitsbewältigung, soziale Integration und berufliche Reintegration wird eingegangen. Damit können Sie ein störungsspezifisches und individuell angepasstes Vorgehen für Ihre Patienten entwickeln. Der Inhalt • Grundlagen der Aphasiologie • Sprachverarbeitungsmodelle und ihre Anwendung in der Diagnostik und Therapie • Anamnese, Diagnostik, Planung und Durchführung der Behandlung • Beratung von Patienten und Angehörigen • Qualitätssicherung und evidenzbasierte Praxis Die 7. Auflage wurde umfassend überarbeitet und um zahlreiche Themen ergänzt, w.z.B. Bildgebung und Neuromodulation, partizipationsorientierte Diagnostik, Methoden der Unterstützten Kommunikation und technologiegestützten Aphasietherapie. Plus: Exkurse ermöglichen gezielt eine thematische Vertiefung. Ein Standard für die Behandlung von Patienten mit Aphasie für Auszubildende und Studierende der Logopädie sowie bereits erfahrene Sprachtherapeutinnen und Sprachtherapeuten zum Nachschlagen und Vertiefen.
Chapter
Dieses Kapitel gilt der Qualitätssicherung in der Aphasie-Therapie. Hierbei werden Maßnahmen und Prinzipien evidenzbasierter Praxis vorgestellt sowie Möglichkeiten beschrieben, wie einfache Therapie-Effekte in der Arbeit mit dem einzelnen Patienten überprüft werden können. Weiterhin werden Informationen zu medizinischen Leitlinien gegeben und der Leser erhält eine Einführung in das Clinical Reasoning. Entstehung, Ziel sowie die unterschiedlichen Formen des Clinical Reasonings werden kurz dargestellt und anhand von Patientenbeispiele erläutert. Informationen zu Weiterbildungsmöglichkeiten und Informationsquellen runden das Kapitel ab.
Article
Purpose To investigate the effectiveness of non-drug interventions for people with aphasia in the prevention and/or treatment of anxiety post-stroke as either a primary or secondary outcome. Materials and Methods A systematic search of Medline, CINAHL, PsycINFO and Cochrane Library up to March 2021 was carried out. Studies of stroke populations were included if people with aphasia represented 25% or more of the enrolled participants. Quality of the evidence was assessed. A narrative synthesis of results is presented. The PROSPERO record ID for this study is 106451. Results Ten studies were included: five randomised controlled trials (RCTs), a single case experimental design, and four pre-post studies. The quality of the RCT trials was at least adequate but none demonstrated a benefit to anxiety outcomes. Those studies that reported benefit were of lower-level evidence with respect to National Health and Medical Research Classifications. No studies were found that evaluated the prevention of anxiety after stroke for people with aphasia. Conclusion Definitive conclusions about the effectiveness of non-drug interventions for the prevention and/or treatment of anxiety in people with aphasia post-stroke cannot be made. Interventions that may show promise for those with aphasia and symptoms of anxiety include mindfulness meditation, modified cognitive behaviour therapy, unilateral nostril breathing, and the “Enhance Psychological Coping after Stroke” programme. Further high-quality research with better reporting of the inclusion of participants with aphasia and their specific sub-group results are required. • Implications for Rehabilitation • It is important for rehabilitation professionals to consider prevention of anxiety post-stroke as well as treatment. • Mindfulness meditation, modified cognitive behavioural therapy, unilateral nostril breathing, and the and the “Enhance Psychological Coping after Stroke” programme may be of benefit to people with aphasia post-stroke. • Adapting intervention protocols to be more communicatively accessible and training health professionals in supported communication may help people with aphasia engage in psychological therapies.
Book
Full-text available
Single-case designs have a long history in psychological research. They have also gained in popularity in medical research in recent years, where they are known as N-of-1 (or N=1) trials. They can provide high-level evidence if conducted well, and at the same level as systematic reviews (OCEBM, 2011). But these designs can be complex and difficult to conduct well. This manual describes the basic structure of single-case experimental designs and N-of-1 trials. It then presents a set of criteria that are important for the scientific value of a single-case study: the Risk-of-Bias in N-of-1 Trials (RoBiNT) Scale. This scale is a critical appraisal tool for published single-case experimental research and N-of-1 trials. The manual is also directed at applied researchers as a helpful resource for planning and conducting single-case research projects. In Chapter 1, the basic components and logic of single-case design methodology are introduced. In Chapter 2, background information on the RoBiNT Scale explains the history of its development, as well as investigations into its psychometric properties. Guidelines and suggestions on how to apply the scale to research output are provided. Chapter 3 addresses the questions: What are the crucial aspects of internal and external validity in single-case research? How can risk-of-bias be minimised to increase the scientific quality of single-case research? Detailed rating criteria are stipulated for each of the 15 items, which represent a comprehensive, yet succinct selection of the pertinent topics concerning validity in single-case research. Several examples of how to score each item are described and explained, providing users with more than 100 'real life' and fictitious examples of how the rating criteria are applied.
Article
Full-text available
We developed a reporting guideline to provide authors with guidance about what should be reported when writing a paper for publication in a scientific journal using a particular type of research design: the single-case experimental design. This report describes the methods used to develop the Single-Case Reporting guideline In BEhavioural interventions (SCRIBE) 2016. As a result of 2 online surveys and a 2-day meeting of experts, the SCRIBE 2016 checklist was developed, which is a set of 26 items that authors need to address when writing about single-case research. This article complements the more detailed SCRIBE 2016 Explanation and Elaboration article (Tate et al., 2016) that provides a rationale for each of the items and examples of adequate reporting from the literature. Both these resources will assist authors to prepare reports of single-case research with clarity, completeness, accuracy, and transparency. They will also provide journal reviewers and editors with a practical checklist against which such reports may be critically evaluated. We recommend that the SCRIBE 2016 is used by authors preparing manuscripts describing single-case research for publication, as well as journal reviewers and editors who are evaluating such manuscripts.
Article
Full-text available
Background: To provide a preliminary estimate of efficacy of constraint-induced aphasia therapy (CIAT) when compared to no-intervention in patients with chronic (>1 year) post-stroke aphasia in order to plan an appropriately powered randomized controlled trial (RCT). Material and methods: We conducted a pilot single-blinded RCT. 24 patients were randomized: 14 to CIAT and 10 to no-intervention. CIAT groups received up to 4 hours/day of intervention for 10 consecutive business days (40 hours or therapy). Outcomes were assessed within 1 week of intervention and at 1 and 12 weeks after intervention and included several linguistic measures and a measure of overall subjective communication abilities (mini-Communicative Abilities Log (mini-CAL)). Clinicians treating patients (CIAT group) did not communicate with other team members to maintain blinding and the testing team members were blinded to treatment group assignment. Results: Overall, the results of this pilot RCT support the results of previous observational studies that CIAT may lead to improvements in linguistic abilities. At 12 weeks, the treatment group reported better subjective communication abilities (mini-CAL) than the no-intervention group (p=0.019). Other measures trended towards better performance in the CIAT group. Conclusions: In this pilot RCT intensive language therapy led to an improvement in subjective language abilities. The effects demonstrated allow the design of a definitive trial of CIAT in patients with a variety of post-stroke aphasia types. In addition, our experiences have identified important considerations for designing subsequent trial(s) of CIAT or other interventions for post-stroke aphasia.
Article
Without a complete published description of interventions, clinicians and patients cannot reliably implement interventions that are shown to be useful, and other researchers cannot replicate or build on research findings. The quality of description of interventions in publications, however, is remarkably poor. To improve the completeness of reporting, and ultimately the replicability, of interventions, an international group of experts and stakeholders developed the Template for Intervention Description and Replication (TIDieR) checklist and guide. The process involved a literature review for relevant checklists and research, a Delphi survey of an international panel of experts to guide item selection, and a face-to-face panel meeting. The resultant 12-item TIDieR checklist (brief name, why, what (materials), what (procedure), who intervened, how, where, when and how much, tailoring, modifications, how well (planned), how well (actually carried out)) is an extension of the CONSORT 2010 statement (item 5) and the SPIRIT 2013 statement (item 11). While the emphasis of the checklist is on trials, the guidance is intended to apply across all evaluative study designs. This paper presents the TIDieR checklist and guide, with a detailed explanation of each item, and examples of good reporting. The TIDieR checklist and guide should improve the reporting of interventions and make it easier for authors to structure the accounts of their interventions, reviewers and editors to assess the descriptions, and readers to use the information.
Article
Purpose: To identify important treatment outcomes from the perspective of people with aphasia and their families using the ICF as a frame of reference. Methods: The nominal group technique was used with people with aphasia and their family members in seven countries to identify and rank important treatment outcomes from aphasia rehabilitation. People with aphasia identified outcomes for themselves; and family members identified outcomes for themselves and for the person with aphasia. Outcomes were analysed using qualitative content analysis and ICF linking. Results: A total of 39 people with aphasia and 29 family members participated in one of 16 nominal groups. Inductive qualitative content analysis revealed the following six themes: (1) Improved communication; (2) Increased life participation; (3) Changed attitudes through increased awareness and education about aphasia; (4) Recovered normality; (5) Improved physical and emotional well-being; and (6) Improved health (and support) services. Prioritized outcomes for both participant groups linked to all ICF components; primary activity/participation (39%) and body functions (36%) for people with aphasia, and activity/participation (49%) and environmental factors (28%) for family members. Outcomes prioritized by family members relating to the person with aphasia, primarily linked to body functions (60%). Conclusions: People with aphasia and their families identified treatment outcomes which span all components of the ICF. This has implications for research outcome measurement and clinical service provision which currently focuses on the measurement of body function outcomes. The wide range of desired outcomes generated by both people with aphasia and their family members, highlights the importance of collaborative goal setting within a family-centred approach to rehabilitation. These results will be combined with other stakeholder perspectives to establish a core outcome set for aphasia treatment research. Implications for Rehabilitation Important outcomes for people with aphasia and their families span all components of the ICF. The relevancy and translation of research findings may be increased by measuring and reporting research outcomes which are important to people living with aphasia. The results of this study indicate that important treatment outcomes for people living with aphasia most frequently link to the activity/participation and body function components of the ICF. The outcomes identified in this study suggest a broad role for clinicians working in aphasia rehabilitation. The categories of identified outcomes may be used clinically as a starting point in goal-setting discussions with clients and their families.
Chapter
Background: Aphasia is an acquired language impairment following brain damage that affects some or all language modalities: expression and understanding of speech, reading, and writing. Approximately one third of people who have a stroke experience aphasia. Objectives: To assess the effects of speech and language therapy (SLT) for aphasia following stroke. Search methods: We searched the Cochrane Stroke Group Trials Register (last searched 9 September 2015), CENTRAL (2015, Issue 5) and other Cochrane Library Databases (CDSR, DARE, HTA, to 22 September 2015), MEDLINE (1946 to September 2015), EMBASE (1980 to September 2015), CINAHL (1982 to September 2015), AMED (1985 to September 2015), LLBA (1973 to September 2015), and SpeechBITE (2008 to September 2015). We also searched major trials registers for ongoing trials including ClinicalTrials.gov (to 21 September 2015), the Stroke Trials Registry (to 21 September 2015), Current Controlled Trials (to 22 September 2015), and WHO ICTRP (to 22 September 2015). In an effort to identify further published, unpublished, and ongoing trials we also handsearched the International Journal of Language and Communication Disorders (1969 to 2005) and reference lists of relevant articles, and we contacted academic institutions and other researchers. There were no language restrictions. Selection criteria: Randomised controlled trials (RCTs) comparing SLT (a formal intervention that aims to improve language and communication abilities, activity and participation) versus no SLT; social support or stimulation (an intervention that provides social support and communication stimulation but does not include targeted therapeutic interventions); or another SLT intervention (differing in duration, intensity, frequency, intervention methodology or theoretical approach). Data collection and analysis: We independently extracted the data and assessed the quality of included trials. We sought missing data from investigators. Main results: We included 57 RCTs (74 randomised comparisons) involving 3002 participants in this review (some appearing in more than one comparison). Twenty-seven randomised comparisons (1620 participants) assessed SLT versus no SLT; SLT resulted in clinically and statistically significant benefits to patients' functional communication (standardised mean difference (SMD) 0.28, 95% confidence interval (CI) 0.06 to 0.49, P = 0.01), reading, writing, and expressive language, but (based on smaller numbers) benefits were not evident at follow-up. Nine randomised comparisons (447 participants) assessed SLT with social support and stimulation; meta-analyses found no evidence of a difference in functional communication, but more participants withdrew from social support interventions than SLT. Thirty-eight randomised comparisons (1242 participants) assessed two approaches to SLT. Functional communication was significantly better in people with aphasia that received therapy at a high intensity, high dose, or over a long duration compared to those that received therapy at a lower intensity, lower dose, or over a shorter period of time. The benefits of a high intensity or a high dose of SLT were confounded by a significantly higher dropout rate in these intervention groups. Generally, trials randomised small numbers of participants across a range of characteristics (age, time since stroke, and severity profiles), interventions, and outcomes. Authors' conclusions: Our review provides evidence of the effectiveness of SLT for people with aphasia following stroke in terms of improved functional communication, reading, writing, and expressive language compared with no therapy. There is some indication that therapy at high intensity, high dose or over a longer period may be beneficial. HIgh-intensity and high dose interventions may not be acceptable to all.
Article
Background: Few studies have documented the possibility of treatment-induced improvements in language functions 12 months or longer after stroke. The purpose of the current study was to provide a preliminary estimate of efficacy of constraint-induced aphasia therapy (CIAT) when compared to no-intervention in patients with chronic (> 1 year) post-stroke aphasia in order to provide the data needed to design an appropriately powered trial. Material/Methods: This was a randomized, controlled, single-blinded, pilot trial. We identified 32 patients with chronic post-stroke aphasia. Of these, 27 were offered participation, and 24 were randomized (CONSORT diagram): 14 to CIAT and to 10 to no-intervention. CIAT groups received up to 4 hours/day of intervention for 10 consecutive business days (40 hours of therapy). Outcomes were assessed within 1 week of intervention and at 1 and 12 weeks after intervention and included several linguistic measures and a measure of overall subjective communication abilities (mini-Communicative Abilities Log (mini-CAL)). To maintain blinding, clinicians treating patients (CIAT group) did not communicate with other team members and the testing team members were blinded to treatment group assignment. Results: Overall, the results of this pilot trial support the results of previous observational studies that CIAT may lead to improvements in linguistic abilities. At 12 weeks, the treatment group reported better subjective communication abilities (mini-CAL) than the no-intervention group (p=0.019). Other measures trended towards better performance in the CIAT group. Conclusions: In this randomized, controlled, and blinded pilot study, intensive language therapy (CIAT) led to an improvement in subjective language abilities. The effects demonstrated allow the design of a definitive trial of CIAT in patients with a variety of post-stroke aphasia types. In addition, our experiences have identified important considerations for designing subsequent trial(s) of CIAT or other interventions for post-stroke aphasia.