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Telerehabilitation for People with Aphasia: A Systematic Review and

Meta-Analysis

Cacciante Luisa , Kiper Pawel , Garzon Martina ,

Baldan Francesca , Federico Sara , Turolla Andrea ,
Agostini Michela

PII: S0021-9924(21)00034-4
DOI: https://doi.org/10.1016/j.jcomdis.2021.106111
Reference: JCD 106111

To appear in: Journal of Communication Disorders

Received date: 6 April 2020

Revised date: 27 April 2021
Accepted date: 4 May 2021

Please cite this article as: Cacciante Luisa , Kiper Pawel , Garzon Martina , Baldan Francesca ,
Federico Sara , Turolla Andrea , Agostini Michela , Telerehabilitation for People with Aphasia:
A Systematic Review and Meta-Analysis, Journal of Communication Disorders (2021), doi:
https://doi.org/10.1016/j.jcomdis.2021.106111

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Title page

TELEREHABILITATION FOR PEOPLE WITH APHASIA: A SYSTEMATIC REVIEW AND

META-ANALYSIS

Authors

Cacciante Luisa¹, Kiper Pawel2,*, Garzon Martina¹, Baldan Francesca¹, Federico Sara¹, Turolla

Andrea¹, Agostini Michela¹

Affiliation
1
Laboratory of Rehabilitation Technologies, San Camillo IRCCS SRL, Venice, Italy
2
Rehabilitation Unit, Azienda ULSS 3 Serenissima, Venice, Italy

*Corresponding Author

Kiper Pawel, PhD

Email: pawelkiper@hotmail.com

Phone: +390415295485

Address: Piazzale Ravà 1, 30126, Venice, Italy

Key words

Telemedicine, Telehealth, Telerehabilitation, Aphasia, Meta-analysis, Stroke

Conflict of interest

Authors declare no conflict of interest.

Funding sources

No special funds have supported this project.

Protocol registration number

PROSPERO 2019 CRD42019136545

1
Abstract

Objectives: To evaluate effectiveness or non-inferiority of telerehabilitation for people with

aphasia when compared to conventional face-to-face speech and language therapy.

Materials and Methods: Five electronic databases (PUBMED, EMBASE, WEB OF SCIENCE,

SCOPUS and the Cochrane Library) were searched. We extrapolated data from the included studies

and evaluated the methodological quality using the Revised Cochrane risk-of-bias tool for

Randomized Trials (RoB 2) and the Risk Of Bias In Non-randomized Studies of Interventions

(ROBINS-I). A meta-analysis compared effects of intervention, and it was conducted using the

Review Manager 5.3 software. GRADE profile to assess overall quality of evidence was carried

out.

Results: Out of a total of 1157 records, five studies met the inclusion criteria and were eligible for

meta-analysis with a total of 132 participants with post-stroke aphasia.

Discussion: Results revealed that telerehabilitation and face-to-face speech and language treatment

are comparable with respect to the gains achieved in auditory comprehension (SMD = -0.02; 95%

CI -0.39, 0.35), naming accuracy (SMD = -0.09; 95% CI -0.44, 0.25), Aphasia Quotient (MD = -

2.18; 95% CI -16.00, 11.64), generalization (SMD = 0.77; 95% IC -0.95, 2.49) and functional

communication skills (SMD = -0.08; 95% IC -0.54, 0.38).

Conclusion: Although evidence is still insufficient to guide clinical decision making due to the

relatively low quality of the evidence identified, the analysis of the results suggest that

telerehabilitation training for aphasia seems to be as effective as the conventional face-to-face

treatment.

2
INTRODUCTION

Description of aphasia language impairment

Stroke is one of the most common causes of death and a major cause of disability worldwide

(Donnan, Fisher, Macleod, & Davis, 2008). People who suffered a stroke, commonly experience a

wide range of symptoms, including impairment of motor functions, speech disturbances,

swallowing difficulties, cognition deficits, impaired vision and sensation disorders (Langhorne,

Bernhardt, & Kwakkel, 2011). In particular, approximately one third of stroke survivors is affected

by aphasia (Engelter et al., 2006). Aphasia often leads to communication difficulties hindering

participation in social activities, both for patients and caregivers. People with aphasia (PWA) show

several impairments in some or all language modalities that can influence production and/or

comprehension of speech, reading and/or writing and gesture (Brady, Kelly, Godwin, Enderby, &

Campbell, 2016). Aphasic symptoms can be considered mild, when the subject shows word-finding

difficulties, or global, where all language modalities are severely impaired (Code & Herrmann,

2003). Evidence shows that patients who suffer from aphasia can benefit from rehabilitative

programs even after many years from its onset. However, only some receive self-tailored therapy,

and when it happens it is usually limited to the first few months post-stroke (Brady et al., 2016).

Almost half of stroke survivors complains about the lack of clinical or social support within the first

5 years after the event (McKevitt et al., 2011). Nevertheless, long-term therapy is hardly accessible

for stroke survivors, especially for those living in rural areas. Among the other reasons, the fact that

patients who suffered from brain damage often have several comorbidities that make it more

difficult to reach a rehabilitation center (Agostini et al., 2014). A study by Howe et al. explored the

environmental factors that hinder the participation of adults with aphasia in social activities. PWA

perceived the lack of services after hospital-based speech therapy and the lack of opportunities to

meet with other people with aphasia as barriers to social integration. Furthermore, participants

reported difficulties in getting to and from social environments (Howe, Worrall, & Hickson, 2008).

Considering all these limitations, it becomes crucial to provide treatments that ensure the
3
continuity of care for people with aphasia, in order to enhance participation in their social life.

Thus, the innovative telecommunication technologies seem to be the solution to address this issue.

Intervention description

Telerehabilitation (TR) is defined as the delivery of rehabilitation services via information

and communication technologies (Winters, 2002). It has been primarily developed to reduce

inpatient hospital stay and to increase community services, including rehabilitative treatment after

discharge from a hospital ward. TR can include the following specific services: assessment,

monitoring, prevention, intervention, supervision, education, consultation and counseling, and are

directed to support individuals with disabilities (D. Brennan et al., 2010).

There is a growing body of evidence that underlies the validity and reliability of speech and

language TR systems: some studies showed good validity and reliability of telepractice in the

assessment of acquired language disorders (Theodoros, Hill, Russell, Ward, & Wootton, 2008) and

in their treatment (Cherney & van Vuuren, 2012; Hall, Boisvert, & Steele, 2013), in particular on

speech and voice deficits in Parkinson’s disease (Constantinescu et al., 2010). Moreover, TR turned

out to be advantageous in the treatment of fluency disorders: a series of research trials provided

evidence that the use of TR is feasible and effective, leading to satisfactory clinical outcomes in the

treatment of stuttering in children (Wilson, Onslow, & Lincoln, 2004), adolescents (Sicotte,

Lehoux, Fortier-Blanc, & Leblanc, 2003) and adults (O'Brian, Packman, & Onslow, 2008). On the

other hand, there is limited evidence regarding the efficacy of TR application for aphasic

impairments (D. M. Brennan, Georgeadis, Baron, & Barker, 2004), including TR for anomia

(Dechêne et al., 2011) and assessment of apraxia of speech (Hill, Theodoros, Russell, & Ward,

2009). Agostini et al. demonstrated the feasibility and effectiveness of TR applied to lexical deficits

in chronic stroke patients, reporting comparable results between face-to-face and TR settings

(Agostini et al., 2014). Furthermore, Latimer et al. reported that computer therapy for people with

4
long-standing aphasia has the potential to reduce the social burden associated with health services

management (Latimer, Dixon, & Palmer, 2013).

Approaches to telerehabilitation intervention

The rationale for TR is that similar mechanisms can be triggered to those observed in

conventional treatments (Laver et al., 2013). Preliminary data from our clinical practice showed that

speech and language treatment fits well with the TR approach and could be used for lexical retrieval

training. For instance, the interaction with a touchscreen and writing on a paper share similar

characteristics. However, this study is ongoing, more participants will be enrolled and an in depth

analysis will be carried out.

Two technological approaches are promising in the delivery of remote speech and language

treatments. The first approach is referred to as "asynchronous" TR. It does not need the presence of

the speech and language therapist and allows the provision of self-administered computer-based

exercises, so that patients can complete extensive training. Therapists can then evaluate progress

and increase exercise difficulty according to patient's needs (Wade, Mortley, & Enderby, 2003).

Indeed, Kurland et al. demonstrated the effectiveness of unsupervised home practice, with weekly

video teleconferencing support for people with chronic aphasia (Kurland, Liu, & Stokes, 2018).

The second modality is known as "synchronous" TR and is based on two-way videoconferencing in

the presence of the speech and language therapist. This kind of TR allows therapists to provide real

time remote treatments, and to reduce both time and travel costs (Meltzer, Baird, Steele, & Harvey,

2017).

Why it is important to do this review

According to available evidence, there is a lack of systematic reviews and meta-analyses

addressing the problem of speech and language TR. It has been noted that TR may not be inferior to

in-person therapy and, therefore, it appears to be a reasonable model of service delivery for people
5
with motor disorders after stroke (Laver et al., 2020). However, the same findings have yet to be

confirmed for PWA. Thus, it is pivotal to incorporate new evidence regarding the use of TR in

aphasia treatment that could be relevant for further development of clinical practice.

OBJECTIVES

The main aim of this review was to assess the effectiveness or non-inferiority of speech and

language TR in people with post-stroke aphasia when compared to conventional face-to-face

treatment. The primary outcome was to evaluate improvements of linguistic abilities of patients

who were treated remotely. The secondary outcome was to evaluate cost-effectiveness and

feasibility of TR for PWA.

METHODS

Electronic searches

The protocol of this systematic review was registered in the PROSPERO database under the

following registration number: CRD42019136545. We searched articles written in English in

PUBMED, EMBASE, WEB OF SCIENCE, SCOPUS and the Cochrane Library (see

Supplementary Table 1). The last search was performed on 30th of March 2020.

Study selection

Selection criteria for inclusion comprised studies which included post-stroke PWA as study

participants, and remotely provided treatments of linguistic functions and communication abilities.

Randomized controlled trials, quasi-randomized controlled trials and case-control studies were

considered for qualitative and quantitative assessment.

We excluded from the meta-analysis studies involving patients with Primary Progressive

Aphasia; studies examining the computer based face-to-face delivery as experimental intervention;

studies designed as case series, case report or study protocol were excluded as well.
6
 Two reviewers, independently screened records identified through the search strategy

described in Appendix A. An inclusion/exclusion criteria template helped to form a decision on

whether to proceed or not with full text study evaluation. The same two reviewers independently

evaluated the studies and decided what to include or to exclude, according to the above described

criteria. Any disagreement was resolved after a discussion with a third reviewer. Included full texts

were analyzed for their quality.

Types of participants and interventions

We selected trials that included participants with post-stroke aphasia and mixed impairments

in linguistic functions (i.e. impairments in production and/or comprehension of speech, reading

and/or writing and gesture). We included studies comparing PWA who underwent TR therapy to

improve linguistic functions and/or communication abilities, with PWA who received conventional

face-to-face treatments. In particular, we considered all types of speech and language TR delivery

methods, e.g. clinic-home or two separate rooms in the same clinic, where the distance between

patient and therapist was kept and provided in different locations.

Outcome measures

The primary outcome of this systematic review was to analyze the effectiveness of TR and

the improvement in linguistic abilities. Depending on the outcome measure identified in each study,

this resulted in examining changes in: auditory comprehension (comprehension subtests of the

Comprehensive Aphasia Test [CAT], the Western Aphasia Battery [WAB] or the Norwegian Basic

Aphasia Assessment [NGA]), naming accuracy for expressive language (calculated as the mean of

corrected items named at pre- and post- treatment evaluations), overall severity of language

impairment (Aphasia Quotient arising from the Western Aphasia Battery [WAB-AQ]),

generalization of intervention (evaluated as picture-naming performance with the score taken in the

assessment of the untreated items after treatment) or functional communication skills (assessed with
7
the Communicative Effectiveness Index [CETI] or with the Communicative Abilities in Daily

Living [CADL]). The secondary outcomes included all financial reports that aimed to assess cost-

effectiveness of TR and all kinds of feasibility assessment methods, such as recruitment and

attrition rates, participant observations, questionnaires and interviews, and treatment fidelity

checking.

Grade and quality of evidence

The quality of the evidence is described in the GRADE assessment (Guyatt et al., 2008).

The GRADE approach involves separate grading of certainty of evidence for each patient-important

outcome followed by determining an overall certainty of evidence across outcomes. The certainty of

evidence reflects the extent to which we are confident that an estimate of the effect is correct.

Furthermore, the GRADE approach suggests selecting all patient-important outcomes and

rating the importance of each outcome. To facilitate ranking of outcomes according to their

importance (see “Importance”, in Table 1), outcomes were rated numerically on a 9-point scale,

from 1 to 9 (i.e. 7 to 9 – critical; 4 to 6 – important; 1 to 3 – of limited importance), to distinguish

between importance categories. Table 1 presents outcome-specific information concerning the

overall quality of evidence. We evaluated the evidence for study limitations (risk of bias),

inconsistency of the results, indirectness of evidence, imprecision of effects estimates or potential

publication bias (other consideration). We examined the following outcomes: auditory

comprehension, naming accuracy (both in RCT and non-RCT), WAB-AQ, generalization and

functional communication. To achieve transparency, the GRADE system classifies the certainty of

evidence in one of four grades. (1) High: further research is very unlikely to change our confidence

in the estimate of effect; (2) Moderate: further research is likely to have an important impact on our

confidence in the estimate of effect and may change the estimate; (3) Low: further research is very

likely to have an important impact on our confidence in the estimate of effect and is likely to change

the estimate; (4) Very low: any estimate of effect is very uncertain.
8
DATA COLLECTION AND ANALYSIS

Data extraction and management

We extracted study data and recorded information in standardized tables for data extraction,

i.e. : authors and year of publication, study design, methods of randomization, blinding, attrition

from intervention, co-interventions, confounder details, number of participants, age, time post

onset, inclusion and exclusion criteria, details of intervention in accordance with the Template for

Intervention Description and Replication (TIDieR) checklist (Hoffmann et al., 2014), outcome

measures and when they were administered.

Assessment of risk of bias in included studies

Methodological quality of trials was assessed using the Revised Cochrane risk-of-bias tool

for Randomized Trials (RoB 2) (Sterne et al., 2019) and the Risk Of Bias In Non-randomized

Studies of Interventions (ROBINS-I) tool for quasi-randomized control trials and case-control

studies (Sterne et al., 2016). Two review authors (LC and PK) independently assessed the risk of

bias for each included study and a third reviewer (MG) resolved any disagreement. For RCTs we

evaluated the following types of bias: bias arising from the randomization process (allocation

sequence generation), bias due to deviations from intended interventions (the intervention effect of

interest was the effect of assignment to the interventions at baseline), bias due to missing outcome

data, bias in measurement of the outcome and bias in selection of the reported results. We coded

risk of bias for each domain and overall risk of bias as "low risk", "some concerns" or "high risk",

according to the RoB 2 guidance. Regarding non-randomized studies, we assessed methodological

quality by analyzing seven bias domains: confounding bias, selection bias, measure intervention

bias, performance bias, attrition bias, detection bias, reporting bias. We coded risk of bias for each

domain and overall risk of bias as "low risk", "moderate risk", "serious risk", "critical risk" or "no

information", according to the ROBINS-I guidance.

9
 Measures of treatment effect

We used Review Manager 5.3 (RevMan 2014) to conduct review, to record descriptive

information for each trial in the characteristics of the included studies table, and for statistical

analysis. Methodological quality of studies was recorded in the risk of bias tables. We assigned

outcome measures to each assessed domain (auditory comprehension, naming accuracy, overall

severity impairment, generalization of intervention and functional communication skills). Treatment

effect was evaluated using Mean Difference (MD) in case of homogeneous outcome measures, or

Standardized Mean Difference (SMD) when the outcomes were assessed with different scales.

Confidence interval (CI) for continuous outcomes was identified at 95%, as appropriate.

Data synthesis

We conducted a meta-analysis based either on a random-effects model or fixed model,

depending on the heterogeneity of the results, with 95% CIs using RevMan 5.3. We contacted trial

authors to ask for missing data (i.e. methods of randomization and/or allocation, or additional

information related to results in order to carry out the meta-analysis).

Subgroup analysis and investigation of heterogeneity

We planned analysis of subgroups in relation to study design (i.e. RCT or non-RCT).

Heterogeneity was assessed considering intervention and outcome measures. Statistical

heterogeneity was assessed with the I² test, establishing the cut-off value at 50%.

RESULTS

Results of the search

Search strategy identified 1157 records from 5 electronic databases. After removing 265

duplicates and 885 studies with unrelated target topics, 7 studies remained for full-text review. At
10
the end of the process, 5 studies met the inclusion criteria and were eligible for quantitative

analysis. The flow of literature through the searching and screening process is shown in the

PRISMA flow diagram (Figure 1).

Included and excluded studies

Among the five included studies, there were three randomized controlled trials and two non-

randomized controlled trials. Both synchronous and asynchronous TR were considered in this

review. However, we found trials evaluating only synchronous TR. The overall number of analyzed

participants was 132. The intervention groups in included studies received TR therapies, with a total

of 67 patients who completed the training program. In the control groups, 65 participants received

conventional face-to-face treatments that aimed to improve linguistic and communicative functions.

The therapy length ranged from 2 weeks to 6 months, and a follow-up assessment was performed

by Agostini et al., Ora et al. and Woolf et al. within four months post-randomization. Meltzer et al.

and Zhou et al. did not provide information about follow-up administration. The sample size of the

included studies ranged from 5 to 62 patients with language impairments due to stroke. Only one

study recruited participants in the acute stage post-stroke (Zhou et al., 2018), and three trials

included participants in subacute and chronic stages (Agostini et al., 2014; Meltzer et al., 2017;

Woolf et al., 2016). One study (Ora et al., 2020) included participants with no limitations regarding

post-stroke time. Three studies excluded participants with cognitive or other neurological

impairments. Study by Meltzer et al. included participants with cognitive-linguistic communication

disorders and stratified them to an aphasic or a cognitive group. Treatments for both aphasic and

cognitive groups were administered both in-person or remotely (Meltzer et al., 2017). Study by

Zhou et al. included patients with language and cognitive impairments and delivered both language

and cognitive training to all participants (Zhou et al., 2018). All experimental interventions were

delivered at the patient's home via videoconferencing. Only Zhou et al. provided an in-person

computerized speech-language and cognitive treatment to one of the two intervention groups. The
11
primary aim of the intervention varied across studies. Two studies evaluated the feasibility of

treatment remotely delivered, (Agostini et al., 2014; Woolf et al., 2016). Meltzer et al. and Ora et. al

aimed to evaluate the effectiveness of TR, whereas Zhou et al. investigated the efficacy of a

computerized intervention for aphasia that combined speech-language and cognitive training

delivered on an inpatient unit (i.e. face-to-face) or via TR. All studies evaluated improvements in

linguistic and communicative abilities.

More specifically, Agostini et al. aimed to explore the feasibility of TR as compared with

conventional face-to-face treatment for anomia. All 5 participants included in the study carried out

two versions of the naming treatment: face-to-face treatment and TR. The presentation of the first

and second treatment was balanced so that participants started either with the face-to-face or with

TR treatments, and then proceeded with the other one. The outcome measure was naming accuracy

(percent correct) on the therapy set, measured at baseline, immediately after the therapy, and three

weeks later (follow-up). To examine generalization effects to untreated items, they compared

accuracy on treated items with non-treated control items. In the assessments administered

immediately after the treatments they observed a main effect of item type (p = 0.02), due to a better

performance on the treated compared to the control items. There was no main effect of treatment

type (p = 0.934). Authors indicated that the treatment of naming deficits provided remotely is not

inferior to the conventional face-to-face treatment (Agostini et al., 2014).

In the study by Meltzer et al. authors evaluated the effectiveness of TR by conducting a

randomized non-inferiority trial. They included 16 PWA in the in-patient group and 17 PWA in the

TR group. For PWA, they evaluated improvements in linguistic function through the Western

Aphasia Battery-Revised, Part 1 (WAB-R). A total of 30 participants completed the WAB before

and after treatment. Results showed a significant increase in WAB-AQ score after treatment (p <

0.001). Authors stated that the TR was highly effective, producing objective and subjective gains

that were comparable with in-person treatment (Meltzer et al., 2017).

12
 In the study by Ora et al., 62 participants were included and allocated to the intervention

group (TR in addition to usual care; N=32) and to the Control Group (usual care alone; N=30). As

primary outcome they assessed naming ability through the NGA. For the evaluation of language

functioning beyond naming, the NGA subtests auditory comprehension and repetition were also

included, as well as the CETI. Results showed no significant treatment effects for the percentile

score of the subtests naming (p = 0.489) and comprehension (p = 0.332) from the NGA. However,

the mixed models analysis revealed a significant larger improvement over time in the TR group

compared to the control group for the subtest sentence production of the Verb and Sentence Test (p

= 0.002) and on the repetition percentile score of the NGA (p = 0.026). For the CETI, no statistical

significance was seen between the groups. Authors concluded that TR may be a viable

rehabilitation model for PWA (Ora et al., 2020).

Woolf et al. aimed to test the feasibility of a quasi-randomized controlled trial comparing

face-to-face and TR for word-finding therapy for PWA. Twenty participants were allocated to four

groups, i.e. remote therapy from University (N=5), remote therapy from clinical site (N=5), face-to-

face therapy (N=5) and an attention control group (N=5). Feasibility measures consisted in

recruitment and attrition rates, as well as the results of fidelity checking. The primary outcome was

the naming ability, assessed with the test of spoken picture naming of the CAT. Authors reported

that compliance and satisfaction with the intervention were good. Treatment fidelity was high for

both remote and face to face delivery. In relation to the primary outcome, they demonstrated that

both remote and face-to-face therapy improved word retrieval (p <0.001). Authors stated that word

finding therapy delivered via TR was feasible and well accepted by participants. Moreover, results

showed that word production, at least in picture naming tasks, was improved by therapy more than

by the attention control condition, regardless of delivery (i.e. remote or face-to-face) (Woolf et al.,

2016).

13
 Zhou et al. investigated the efficacy of a computerized training for aphasia that combined

speech-language and cognitive training delivered on an inpatient unit or via TR to discharged

patients. They assessed language function with the WAB and practical communication skills with

the CADL at two time points (T1 and T2). Results demonstrated that the training group showed

statistically greater improvement than the control group from T1 to T2 on WAB-AQ (p < 0.001).

Results on CADL were similar to those on the WAB-AQ: within each site, there was a significant

main effect of time (p < 0.001) but not of group (p > 0.75). Authors concluded that this combined

form of computerized training promoted aphasia recovery more effectively than a traditional

training, for both hospitalized and discharged patients (Zhou et al., 2018). More detailed

information about characteristics of included studies is presented in Supplementary Table 2.

We deemed 2 studies to be ineligible for meta-analysis, as they did not have control groups

(Macoir, Sauvageau, Boissy, Tousignant, & Tousignant, 2017; Marshall et al., 2016).

Risk of bias in included studies

Supplementary Table 3 and Supplementary Table 4 describe the risk of bias of the included

studies according to ROB 2 and ROBINS-I standards, respectively.

The following risks of bias for the randomized trials were observed, according to ROB-2:

- Bias arising from the randomization process: two randomized controlled trials (Meltzer et

al., 2017; Zhou et al., 2018) were judged with “some concerns” in this domain, due to

missing information about allocation methods; study by Ora et al. was judged with a low

risk of bias, as allocation sequence was random and adequately concealed.

- Bias arising from deviations from the intended interventions: Study by Meltzer et al. and

Zhou et al. resulted in "some concerns", as analysis of the effect of assignment to

intervention were not reported. Only study by Ora et al. resulted with a low risk of bias in

this domain: due to the nature of speech and language intervention participants, caregivers

and therapists were aware of assigned intervention, but we deemed this domain related to
14
 the nature of intervention instead of to the study quality. In addition, intention-to-treat (ITT)

analysis was provided.

- Bias due to missing outcome data: all studies were at low risk of bias.

- Bias in measurement of the outcome: both Meltzer et al. and Zhou et al. trials were judged

with “some concerns”: even though there was no blinding of outcome assessments, outcome

tests used have high validity and reliability. Study by Ora et al. was at low risk of bias in this

domain, as assessors were blinded.

- Bias in selection of reported results: Studies by Meltzer and Zhou’s were judged with “some

concerns”, as no information about study protocols were provided. In Ora et al. study all

outcome measurement and analyses correspond to the trial protocol, resulting in a low risk

of bias.

The following risks of bias for the Non-randomized studies, according to ROBINS-I, were

observed:

- Confounding bias: the two non-RCT studies (Agostini et al., 2014; Woolf et al., 2016) did

not report confounding biases.

- Selection bias: both trials had a low risk of bias in the selection of participants into the

study.

- Measure intervention bias: in the classification of the intervention, a low risk of bias was

found in both studies.

- Performance bias: this domain was assessed with a low risk in both trials, despite the lack of

blindness. However, due to the nature of speech and language therapy it is difficult to blind

either patients or therapists to the intervention received.

- Attrition bias: the two trials reported drop-out rates that had a low risk of bias of missing

data.

- Detection bias: study of Woolf et al. (Woolf et al., 2016) reported that outcome assessors

were unaware of the intervention received by study participants, thus outcome assessments
15
 were not influenced by the knowledge of the participants and the risk of bias was low. In the

other study (Agostini et al., 2014) the assessor was not blind; however, the outcome tests

employed have high validity and reliability, and this domain was judged with a moderate

risk of bias.

- Reporting bias: for the selection of the reported results, Agostini’s trial was judged with a

moderate risk of bias. Conversely, in the trial by Woolf et al., all expected and pre-specified

outcomes were reported, and the risk of bias was low.

Effects of intervention

Comparison 1. Auditory comprehension. Telerehabilitation versus face-to-face treatment.

Regarding auditory comprehension three RCTs were analysed. Since outcome measures

differ in the trials, we examined Standardized Mean Difference (SMD) with fixed-effect model and

Confidence Interval (CI) of 95%. Results did not demonstrate a statistically significant difference

between conventional treatment and TR (SMD = -0.02; 95% CI -0.39, 0.35) and there was no

evidence of heterogeneity (I² = 0%). The results are shown in Figure 2.

Comparison 2. Naming Accuracy. Telerehabilitation versus face-to-face treatment.

For naming accuracy outcome, we separately analysed the three RCTs and the two non-

RCTs. In both cases the analyses were conducted using SMD with a fixed-effect model, since

outcome measures differed across groups. The subgroup analysis of post-stroke aphasia in RCTs

shows that the two modalities used to provide treatments did not differ between groups in RCTs

with PWA (SMD = -0.16; 95% CI -0.53, 0.21). In the subgroup analysis of post-stroke aphasia

treatments in non-RCT, a statistically significant difference was not demonstrated between

interventions (SMD = 0.30; 95% CI -0.59, 1.20). Overall results did not reveal a statistically

significant difference between conventional and TR treatment (SMD = -0.09; 95% CI -0.44, 0.25),

and there was no evidence of heterogeneity (I² = 0%). The analysis of naming accuracy is shown in

Figure 3.
16
 Comparison 3. Western Aphasia Battery Aphasia Quotient WAB-AQ. Telerehabilitation

versus face-to-face treatment.

For the Aphasia Quotient (AQ) arising from the WAB, analyses were performed with Mean

Difference (MD) with fixed-effect model, since the same battery was used to test overall language

impairments. The analysis did not evidence statistically significant differences between

interventions (MD = -2.18; 95% CI -16.00, 11.64). No heterogeneity was observed (I² = 0%). The

results of the comparison are shown in Figure 4.

Comparison 4. Generalization. Telerehabilitation versus face-to-face treatment.

In relation to generalization, analyses were based on SMD with random-effects model.

Generalization assessment is based on the evaluation of patients’ performance in naming of

untreated items. There were no statistically significant differences between the two modalities of

providing treatment (SMD = 0.77; 95% IC -0.95, 2.49). In this case we observed a critical value of

heterogeneity (I² = 66%). Figure 5 shows the results of the analysis of generalization.

Comparison 5. Functional communication. Telerehabilitation versus face-to-face treatment.

We analyzed functional communication skills in two RCTs that reported results of the

Communicative Effectiveness Index (CETI) and the Communicative Abilities in Daily Living

(CADL). Also in this case, we did not find a statistically significant difference between

interventions (SMD = -0.08; 95% IC -0.54, 0.38). No heterogeneity was found (I² = 0%). The

results are shown in Figure 6.

17
 GRADE assessment

The rating of the quality of the evidence is described in GRADE assessment (Table 1).

Table 1 Telerehabilitation compared to face-to-face SLT for people with aphasia.

Certainty assessment № of patients Effect

Certainty Importance
№ of Study Risk of Other face-to-face Relative Absolute
Inconsistency Indirectness Imprecision Telerehabilitation
studies design bias considerations SLT (95% CI) (95% CI)

Receptive language: Auditory comprehension (assessed with: WAB and NGA)

3
randomised not serious not serious serious a not serious none 57 55 - SMD 0.02 SD lower ⨁⨁⨁◯ CRITICAL
(0.39 lower to 0.35 MODERATE
trials
higher)

Naming accuracy - post-stroke aphasia in RCTs (assessed with: WAB and NGA)

3
randomised not serious not serious serious a not serious none 57 55 - SMD 0.16 SD lower ⨁⨁⨁◯ IMPORTANT
(0.53 lower to 0.21 MODERATE
trials
higher)

Naming accuracy - post-stroke aphasia in non-RCTs (assessed with: naming of treated items)

2 observational serious b not serious serious a not serious none 10 10 - SMD 0.3 SD higher ⨁⨁◯◯ IMPORTANT
(0.59 lower to 1.2 LOW
studies
higher)

Western Aphasia Battery - Aphasia Quotient for post-stroke aphasia (assessed with: WAB-AQ)

2 randomised serious b not serious serious not serious none 25 25 - MD 2.18 lower ⨁⨁⨁◯ IMPORTANT
(16 lower to 11.64 MODERATE
trials
higher)

18
Generalization in post-stroke aphasia (assessed with: naming of untreated items)

2 observational serious b serious c serious a not serious none 10 10 - SMD 0.77 SD higher ⨁◯◯◯ CRITICAL
(0.95 lower to 2.49 VERY LOW
studies
higher)

Functional communication (assessed with: CETI and CADL)

2 randomised not serious not serious serious a not serious none 38 35 - SMD 0.08 SD lower ⨁⨁⨁◯ CRITICAL
(0.54 lower to 0.38 MODERATE
trials
higher)

CI: Confidence interval; SMD: Standardised mean difference; MD: Mean difference

Explanations:

a. Downgraded 1 level from high to moderate as the test used to assess language abilities falls within a standardized setting of language evaluation but not within
natural speech context (i.e. communication used in everyday life).
b. Downgraded 1 level from moderate to low as there were serious limitations identified in the risk of bias (either unclear randomization sequence, unclear or high
risk of bias for allocation concealment, or both in 1 or more of the trials).
c. Downgraded 1 level from low to very low due to the presence of heterogeneity for which we cannot identify a plausible explanation.

19
DISCUSSION

Summary of main results

This systematic review included 5 studies with 132 participants (67 patients in

experimental groups and 65 patients in control groups, overall) analyzed for speech and language

disorders. We synthesized the data to compare TR with conventional face-to-face treatment. We

evaluated auditory comprehension and naming performances, the Aphasia Quotient (AQ), the

generalization of treatment by analyzing patient’s performance in the untreated items, and

functional communication skills. This review pointed out that results obtained from TR seem to

be non-inferior to conventional treatment. This suggests that speech and language treatment

provided via videoconference could bring similar benefits as those obtained from the

conventional face-to-face treatment.

Overall completeness and applicability of evidence

Despite our wide search strategy, only five trials on 1157 search results met the inclusion

criteria of this review. Furthermore, the examined studies involved small sample sizes and only

three were RCTs, while the other two were non-randomized studies. Although one of the aims

was to evaluate the feasibility of TR, only one study (Woolf et al., 2016) reported feasibility

assessed by recruitment and attrition rates, participant observations and interviews and treatment

fidelity checking; patients reported high levels of satisfaction and good compliance in system

usability. In addition, limited information was available about the cost-effectiveness of TR.

However, all the included studies were published over the last five years, demonstrating that

speech and language TR is a relatively new approach to aphasia rehabilitation and that new

developments are expected, therefore increased research in this area is needed.

20
 Quality of the evidence

All included studies provided results of small sample sizes, thus studies recruiting larger

samples are necessary to provide stronger evidence. In many cases, it was unclear whether

studies were at risk of bias because of poor reporting and lack of clarification from study

authors: for instance, Zhou et al. declared that patients were randomly assigned to the training

group or to the control group, but did not describe randomization procedure. Furthermore, very

low to moderate methodological quality, which emerged from the GRADE assessment, indicates

that we cannot draw firm conclusions about the effectiveness of TR when compared to

conventional face-to-face treatment. The domains of risk of bias and indirectness of evidence

had an important impact on the ratings in the certainty assessment. More specifically, the non-

randomization of participants across groups, the lack of information regarding the allocation

concealment and the methods used to randomize patients in RCTs, downgraded the quality of

evidence by one level. Moreover, ratings related to the indirectness of evidence were

downgraded by one level for all outcomes, except for the WAB, as recommended in the Core

Outcome Set for aphasia research provided by Wallace et al. (Wallace et al., 2019). All the other

tests used to assess language abilities fall within a standardized setting of language evaluation

but not within a natural communication context, resulting in the use of surrogate outcome

measures, which are identified as measures that are used in place of a clinical endpoint

(Johnston, 1999). Indeed, the ability to successfully communicate relates to language or

communicational skills sufficient to permit the transmission of the message via spoken, written,

or non-verbal modalities, or through a combination of these channels. Attempts to measure this

communication success in a formal way vary from analysis of discourse interaction in real life or

sampling of discourse during specific tasks (Brady et al., 2016). Thus, as Brady et al. already

stated, given the lack of comprehensive, reliable, valid and globally accepted communication

evaluation tools, surrogate outcome measures of communicative and linguistic abilities are used

21
as formal measures of receptive or expressive language, where receptive and expressive

language are measured using language tests in a standardized setting (Brady et al., 2016). The

certainty assessment for generalization outcome was also downgraded by one level for

inconsistency, as we found a substantial heterogeneity of results for which we cannot identify a

plausible explanation. These results confirm that further research is needed and is likely to have

an important impact on our confidence in estimating the treatment’s effect. Thus, it is necessary

to improve future research in speech and language TR by increasing the number of high-quality

RCTs and by incorporating information about cost-effectiveness of TR.

Potential biases in the review process

Although our search strategy included five databases and we considered studies

published in conference proceedings which were retrieved through the databases, we did not

search grey literature. Authors were contacted in the case of missed information. However, we

did not receive replies from any of them.

CONCLUSIONS

Implications for practice

Speech and language TR has the potential to facilitate access to services and to give

continuity to treatment, without decreasing treatment intensity and frequency after discharge

from the hospital. Analysis of the results shows that TR training for aphasia seems to be as

effective as the conventional face-to-face treatment and that similar linguistic goals can be

achieved whether treatment is provided via videoconferencing or in-person. However, since the

assessment of the quality of the evidence was relatively low, we cannot draw firm conclusions

about the effectiveness of TR, and evidence is still insufficient to be used to help clinical

decision making. Thus, the challenge for speech and language therapists and for researchers is to

22
design clinical trials with a larger sample size, in order to identify differences between the two

interventions.

Implications for research

There are numerous potential advantages for using TR, such as decreased travel time,

cost reductions and access to otherwise unavailable services (Pramuka & van Roosmalen, 2009).

Another strength of TR is that this type of intervention for people with long-standing aphasia is

likely to represent a cost-effective use of resources (Latimer et al., 2013). However, analyses of

cost-effectiveness are only exploratory because of the small sample size of the trials. Thus, it

becomes important for aphasia researchers, funders, reviewers and editors to publish all the new

evidence from completed trials and to adhere to the recommendations of the CONSORT

Statement, in order to better report on RCTs and to improve the quality of speech and language

TR trials conducted.

Acknowledgements

Authors would like to acknowledge Francesco Cacciante, PhD; Annamaria Cortese, MD and

Daniele Rimini, PhD for their help at the final stage of this work and for the final English

review.

23
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Figure 1. PRISMA Flow diagram.

27
Figure 2. Comparison 1. Auditory comprehension post-intervention. Telerehabilitation versus face-to-face treatment

28
Figure 3. Comparison 2. Naming accuracy post-intervention. Telerehabilitation versus face-to-face treatment

29
Figure 4. Comparison 3. Western Aphasia Battery Aphasia Quotient WAB AQ. Telerehabilitation versus face-to-face treatment.

30
Figure 5. Comparison 4. Generalization post-intervention. Telerehabilitation versus face-to-face treatment

31
Figure 6. Comparison 5. Functional communication post-intervention. Telerehabilitation versus face-to-face treatment.

32