Measurement properties of outcome measures used in neurological telerehabilitation: A systematic review protocol.

Several measurement tools commonly used in face-to-face neurological rehabilitation have been used in telerehabilitation. However, it is not known whether these tools have adequate measurement properties and clinical utility. This systematic review aims to investigate the measurement properties and the clinical utility of measurement tools used in telerehabilitation in individuals with neurological diseases. A systematic review to investigate the measurement properties and clinical utility of measurement tools used in telerehabilitation in individuals with neurological conditions will be conducted. This systematic review will follow the Preferred Reporting Items for Systematic Review and Meta-Analyses (PRISMA) statement. this systematic review protocol was registered in the International Prospective Register of Systematic Reviews (PROSPERO) on 28 May 2021 (registration number: CRD42021257662). Electronic searches will be performed in following databases: Medical Literature Analysis and Retrieval System Online (MEDLINE Ovid), Excerpta Medica Database (Embase Classic + Embase Ovid), Physiotherapy Evidence Database (PEDro), Scientific Electronic Library Online (Scielo), and Literatura Latino-Americana e do Caribe em Ciências da Saúde (LILACS). Two trained independent reviewers will select the studies according to the inclusion criteria, and will also extract the data, evaluate the clinical utility and methodological quality. The relevant data such as design, participants, settings, and mode of administration, measurement properties, and clinical utility will be summarized. Disagreements between reviewers will be resolved by consensus or by the decision of a third independent reviewer. Hand searches of other relevant studies will be employed. The COnsensus-based Standards for the selection of health Measurement Instruments (COSMIN) checklist and the clinical utility scale will be used to assess the methodological quality and clinical utility of these tools, respectively. This systematic review will provide information regarding the measurement properties and the clinical utility of the measurement tools used in neurological telerehabilitation. This information will be useful to assist health professionals in choosing adequate measurement tools and planning new research studies.

Introduction

Neurological conditions have a high prevalence and incidence [1]. These disorders are the leading cause of disabilities and the second biggest cause of death worldwide [1]. From 1990 to 2016, the disabilities and deaths caused by neurological conditions increased approximately 39% and 15%, respectively [1]. It leads to impairments, activity limitations, and participation restrictions [2], as well as negative impacts on the quality of life [3-6]. This makes these individuals commonly in need of rehabilitation services. According to Cieza et al. 2021, neurological conditions were one of the largest determinants of the need for rehabilitation [7]. However, they usually face barriers to access the rehabilitation centers, such as transport problems, lack of a caregiver, high costs, among others [8-10]. In contexts as the COVID-19 pandemic, these barriers may be even greater, as social distancing is recommended to avoid the spread of the virus [11]. Therefore, telerehabilitation has been an innovative alternative to delivery rehabilitation services.

Telerehabilitation can be defined as the remote delivery of rehabilitation services using information and communication technologies, such as telephone, videoconferencing, and sensors [12]. Telerehabilitation sessions can be used to assess, goal‐setting, intervention, education, and monitoring [2]. The use of telerehabilitation has increased over time as technologies become increasingly prevalent and easily accessible [12].

Telerehabilitation has several benefits, such as easier access, can be carried out at the individual’s home, does not require transportation [13], and has a lower cost when compared to face-to-face rehabilitation [14-16]. In recent years, the use of telerehabilitation has increased, mainly due to the development of new computer technologies and more advanced devices that allow long-distance communication [12, 16]. Moreover, during the COVID-19 pandemic, this strategy was even more widespread.

Recent systematic reviews assessed the effectiveness of telerehabilitation in individuals with neurological conditions [17-19]. In these reviews, various outcomes were investigated, and many measurement tools commonly applied in the face-to-face evaluation were used, such as Barthel Index, Berg Balance Scale, Fugl-Meyer Upper Extremity, Action Research Arm Test, and Stroke Impact Scale [17-19]. However, assessing remotely an individual can be challenging [20].

It is important to consider that the results of these measurement tools may be different when comparing telerehabilitation and face-to-face rehabilitation. The measurement tools commonly used in face-to-face rehabilitation may require adaptations for use in telerehabilitation [20]. For example, the Fulg-Meyer Scale [21] commonly used in the face-to-face evaluation of individuals after stroke can be a challenge to remote use [20]. In addition, communication, comprehension, and interaction are different remotely when compared to face-to-face [22]. All these factors can interfere with the measurements provided by the tools. Hence, it is important to establish the measurement properties of the measurement tools, when used in telerehabilitation. The choice of tools with adequate and accurate measurement properties is important to guarantee the quality and reliability of the results, both in research and in clinical practice [23].

As previously discussed, in recent years a wide range of studies have investigated the use of telerehabilitation, consequently, the number of studies investigating the measurement properties of these measurement tools has also increased [17–19, 22]. Systematic reviews are considered the best way to synthesize existing information and provide a comprehensive analysis of the full range of literature on a particular topic [23]. To our knowledge, no systematic review gathered information on the measurement properties and clinical utility of the measurement tools used in telerehabilitation in individuals with neurological conditions. Given the growth of telerehabilitation in research and clinical practice [12, 16] and the challenges of using measurement tools in remote evaluation [20], a systematic review is necessary. Furthermore, this is an important source of information for researchers and professionals to choose measurement tools with adequate measurement properties and clinical utility. Therefore, this systematic review aims to investigate the measurement properties and the clinical utility of measurement tools used in telerehabilitation in individuals with neurological diseases.

Methods

This systematic review protocol was conducted following Preferred Reporting Items for Systematic Review and Meta-Analysis Protocols (PRISMA-P) [24, 25] and the systematic review results will be reported according to Preferred Reporting Items for Systematic Review and Meta-Analysis (PRISMA 2020 statement) [26].

Study registration

According to PRISMA-P guidelines [24, 25], this systematic review protocol was registered in the International Prospective Register of Systematic Reviews (PROSPERO) on 28 May 2021 (registration number: CRD42021257662).

Eligibility criteria

All full-text papers that aimed to investigate the measurement properties of measurement tools used in telerehabilitation in adults (age ≥18 years old), who had a neurological condition, for example, stroke, Parkinson’s disease, spinal cord injuries, multiple sclerosis, cerebellar ataxia, traumatic brain injuries, and peripheral nervous system diseases, will be included. The searches will not be limited by study design, language, or date of publication.

Research reports, working papers, conference proceedings, conference abstracts, commentaries, letters, dissertations, theses, and editorial papers will be excluded. Systematic reviews and qualitative studies will also be excluded, but their reference lists will be screened for relevant studies.

Search strategy for identification of relevant studies

Electronic search will be carried out for articles indexed on following databases: Medical Literature Analysis and Retrieval System Online (MEDLINE Ovid), Excerpta Medica Database (Embase Classic + Embase Ovid), Physiotherapy Evidence Database (PEDro), Scientific Electronic Library Online (Scielo), and Literatura Latino-Americana e do Caribe em Ciências da Saúde (LILACS). The search strategy was designed according to previous studies and with the assistance of an experienced researcher. The established search strategy for the MEDLINE database will be adapted to suit the other databases. The search strategy (S1 File) is composed of blocks of key terms related to the target population, telerehabilitation, as measurement properties, as follows:

Target population: Individuals with neurological conditions. The search was developed based on a systematic review by Marinho-Buzelli et al. 2015 [27].

Telerehabilitation: The search was developed based on a Cochrane systematic review by Laver et al. 2020 [28].

Measurement properties: The search was developed based on a systematic review by Silva et al. 2014 [29].

Selection of the studies

The selection of the studies will be carried out in three stages. In the first stage, searches will be carried out in the databases. Then, it will be saved and maintained in the Rayyan Systems Inc software [30]. In the second stage, the reviewers will screen the titles and abstracts of the records for eligibility, and the duplicates will be removed. In the third stage, selected full-text articles will be screened for eligibility. The excluded studies and the reason for the exclusion will be recorded. The selection will be performed by two reviewers (SAFB and PCP) independently. Disagreements between reviewers will be resolved by consensus or by the decision of a third independent reviewer (CDCMF). During the selection and screening of the studies, reviewers will be blinded to authors, journals, and outcomes. If additional information would be necessary, the authors of the paper will be contacted. According to the PRISMA 2020 statement [26], a study flow diagram will be created to depict the flow of information through the different stages of this systematic review.

Data extraction

Two reviewers (SAFB and PCP) will independently extract data from the articles using a pre-designed data extraction form. The relevant data extracted from all the included studies will be summarized in tables. Disagreements between reviewers will be resolved by consensus or by the decision of a third independent reviewer (CDCMF).

The tables will contain the following data: study authors; year and country of publication; participants and settings (including data on age, gender, type of neurological condition, and severity of the condition); name, outcomes, and characteristics of the measurement tools; settings and mode of administration (such as telephone, videoconferencing, and sensors); measurement properties (internal consistency, reliability, measurement error, content validity, face validity, construct validity, structural validity, cross-cultural validity, criterion validity, responsiveness); clinical utility; and methodological quality of the study.

Assessment of the methodological quality of the included studies

The methodological quality of the included studies will be assessed using the Consensus-based Standards for the selection of health Measurement Instruments (COSMIN) Risk of Bias checklist [31, 32]. In the COSMIN checklist three domains are distinguished (reliability, validity, and responsiveness), and each domain contains one or more measuring properties. The COSMIN checklist is composed of ten boxes (one box for instrument development and nine boxes for the measurement properties). The boxes contain various items, and each item can be scored on a 4‐point rating scale (i.e. very good, adequate, doubtful, inadequate). Standards that are considered not applicable can be skipped. An overall quality score can be obtained by taking the lowest rating for each item in one box (“worst score counts” method) for each measurement property [31-33]. Following the recommendation by Prinsen et al. 2018 [34], the criteria for good measurement properties will also be used [34, 35]. The criteria for good measurement properties present pooled or summarized results per measurement property per measurement tool. Each item can be scored as sufficient (+), insufficient (−), or indeterminate (?) [34, 35]. The assessment will also be carried out by two reviewers independently (SAFB and PCP). Disagreements will be discussed between the two reviewers and, if necessary, a third reviewer (CDCMF) will be consulted.

Assessment of the clinical utility of the identified tools

The clinical utility can be characterized by the ease with which an instrument is incorporated into clinical practice [36, 37]. This can be evaluated by the instrument’s ability to be brief and simple to apply, understand and score [23]. Parameters used to investigate clinical utility include the acceptability (individual friendliness of the instrument, often characterized by the total time to complete the tool and is influenced by the total number of items and the interpretability of the items) and the feasibility (ease of use, such as required specific training, costs, the need of supervision during the completion of the instrument, and time needed to score) [37]. In addition, the clinical utility can be assessed by criteria that may influence the clinicians in using a measurement tool in their practice, such as time to administer, analyze and interpret the measure, cost, need of specialized equipment/training, portability, and accessibility [36]. Studies that have investigated clinical utility using any of these definitions will be included.

Moreover, the clinical utility of all measurement tools will be collected and reported using the scale proposed by Tyson & Connell [36]. This is a 10-point scale that assessed the following criteria:

Time to administer, analyze and interpret the measure (<10 minutes = 3 points, 10–30 minutes = 2 points, 30–60 minutes = 1 point, >1 hour = 0 points)

Cost (<£100 = 3 points, £100–£500 = 2 points, £500–£1,000 = 1 point, >£1000 or unknown = 0 point)

Need of specialized equipment/training (‘No’ = 2 points, ‘Yes, but only simple, easy to use equipment which does not need specialist training’ = 1 point, ‘Yes’ or ‘Unknown’ = 0 points)

Portability, and accessibility (‘Yes, easily (can go in pocket)’ = 2 points, ‘Yes, in a briefcase or trolley’ = 1 point, ‘No or very difficult’ = 0 points) [36].

The assessment will also be carried out by two reviewers independently (SAFB and PCP).

Data synthesis

A narrative synthesis will be carried out, which will provide texts and tables to synthesize and discuss the data of the studies and methodological characteristics, as previously described in the data extraction section. In addition, texts and tables will be used to summarize the findings regarding the methodological quality [31, 32], quality of measurement properties [33-35], and clinical utility [36, 37].

Discussion

According to our knowledge, this systematic review will be the first one to assess the measurement properties and clinical utility of measurement tools used in telerehabilitation in individuals with neurological conditions. The purpose is to provide a discussion of the strengths and limitations of the different tools used in the evaluation performed during telerehabilitation of individuals with neurological conditions.

Remote delivery health services have been used for a few years. Recently, its use has increased in rehabilitation services, mainly in some specialties such as neurological and cardiac rehabilitation [16]. In the global crisis caused by the COVID-19 pandemic, the rehabilitation services had to adapt quickly to continue offering treatment to the patients [22]. One solution found was telerehabilitation since it allows the rehabilitation of individuals by substituting the traditional face-to-face approach, thus respecting safety rules preconized by health organizations, such as social distancing [38]. Therefore, telerehabilitation has increased exponentially in the last year, both in research and in clinical practice [22].

Investigating the measurement properties of the measurement tools is important to ensure an adequate evaluation of the outcomes [23]. The therapist-patient interaction, characteristics, and settings are different in telerehabilitation when compared to face-to-face rehabilitation [16]. This may reflect different measurement properties compared to those previously investigated in the face-to-face evaluation. Furthermore, functional evaluation has been identified as a challenge to telerehabilitation practice [20, 39]. The identification and use of measurement tools that have appropriate measurement properties and clinical utility may enhance the feasibility and credibility of the evaluation performed remotely, and the comparability of interventions carried out by telerehabilitation. The results of this systematic review will be useful to assist physiotherapists in choosing the measurement tools they will use in telerehabilitation practice. Moreover, it can direct the definition of future research goals and the planning of new research studies.

This review employs a systematic, clear, and replicable inclusion and exclusion criteria and search strategy, as well as the approach regarding the searching, screening, and extracting data. The methods and instruments used in this study are recommended and validated. The involvement of two reviewers in all stages from the selection to the data extraction phase, as well as the assessment of the methodological quality of the included studies, will enhance the methodological rigor and credibility of the results found. The results from this systematic review will be spread by scientific peer-review publications and presentations at conferences and scientific events.

PRISMA-P (Preferred Reporting Items for Systematic review and Meta-Analysis Protocols) 2015 checklist: Recommended items to address in a systematic review protocol*.

(DOC)

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Search strategy.

(DOCX)

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21 Dec 2021

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1. Does the manuscript provide a valid rationale for the proposed study, with clearly identified and justified research questions?

The research question outlined is expected to address a valid academic problem or topic and contribute to the base of knowledge in the field.

Reviewer #1: Partly

Reviewer #2: Yes

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2. Is the protocol technically sound and planned in a manner that will lead to a meaningful outcome and allow testing the stated hypotheses?

The manuscript should describe the methods in sufficient detail to prevent undisclosed flexibility in the experimental procedure or analysis pipeline, including sufficient outcome-neutral conditions (e.g. necessary controls, absence of floor or ceiling effects) to test the proposed hypotheses and a statistical power analysis where applicable. As there may be aspects of the methodology and analysis which can only be refined once the work is undertaken, authors should outline potential assumptions and explicitly describe what aspects of the proposed analyses, if any, are exploratory.

Reviewer #1: Partly

Reviewer #2: Yes

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3. Is the methodology feasible and described in sufficient detail to allow the work to be replicable?

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Reviewer #1: Yes

Reviewer #2: Yes

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Reviewer #2: Yes

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Reviewer #1: Yes

Reviewer #2: Yes

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6. Review Comments to the Author

Please use the space provided to explain your answers to the questions above and, if applicable, provide comments about issues authors must address before this protocol can be accepted for publication. You may also include additional comments for the author, including concerns about research or publication ethics.

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(Please upload your review as an attachment if it exceeds 20,000 characters)

Reviewer #1: In this article the authors investigate the measurement properties and the clinical utility of measurement used in telerehabilitation in individual with neurological disease.

Authors should better structure the manuscript and report the novel finding if any regarding this issue.

The used of the reviewed articles should be used to answer the main questions under study.

Although this is a well written paper with a quite extensive literature survey the major problem is it is not so “innovative”, and the lack of the results strongly limit the decision to proceed with publication.

Reviewer #2: Thank you for asking me to review this systematic review protocol. Given the increasing focus on telehealth and telerehabilitation, this review is timely as it aims to investigate the measurement properties and the clinical utility of measurement instruments used in telerehabilitation in individuals with neurological diseases.

I do have some suggestions and comments:

• How will “telerehabilitation” be defined? Is there a standard definition currently in use?

• Why were these neurological conditions such as stroke, Parkinson's disease, spinal cord injuries, multiple sclerosis, cerebellar ataxia, traumatic brain injuries, and peripheral nervous system diseases selected? What about other neurological conditions or co-morbidities? Will they be excluded?

• I assume you will be including only quantitative studies. If so, you will need to state that you will exclude qualitative studies.

• What will you do with articles which are in language other than English? How will you translate those?

• Will databases such as CINALH/Emcare (allied health specific databases), Cochrane, Scopus/Web of Science be searched?

• How will you address publication and location bias? Are you planning on searching grey literature?

• Will the search terms include both keywords and MeSH?

• Extraction of data is not part of study selection section. Please remove.

• How will discrepancies in data extraction be resolved?

• I am unclear what “systematic narrative synthesis” means. Could you provide more detailed overview of this process?

• Minor point – in some instances, the tense is future (which is correct as this is a protocol) and in other instances, it is in past tense (incorrect). Please amend.

**********

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Reviewer #1: No

Reviewer #2: No

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14 Jan 2022

Reviewer's Responses to Questions

Comments to the Author

1. Does the manuscript provide a valid rationale for the proposed study, with clearly identified and justified research questions?

The research question outlined is expected to address a valid academic problem or topic and contribute to the base of knowledge in the field.

Reviewer #1: Partly

Reviewer #2: Yes

As suggested, the text has been revised to meet this requirement, as follows:

Lines 79-84: “Telerehabilitation can be defined as the remote delivery of rehabilitation services using information and communication technologies, such as telephone, videoconferencing, and sensors [12]. Telerehabilitation sessions can be used to assess, goal‐setting, intervene, education, and monitoring [2]. The use of telerehabilitation has increased over time as technologies become increasingly prevalent and easily accessible [12].”

Lines 110-115: “As previously discussed, in recent years a wide range of studies have investigated the use of telerehabilitation, consequently, the number of studies investigating the measurement properties of these measurement tools has also increased [17-19, 22]. Systematic reviews are considered the best way to synthesize existing information and provide a comprehensive analysis of the full range of literature on a particular topic [23].”

Lines 117-122: “Given the growth of telerehabilitation in research and clinical practice [12,16] and the challenges of using measurement tools in remote evaluation,20 a systematic review is necessary. Furthermore, this is an important source of information for researchers and professionals to choose measurement tools with adequate measurement properties and clinical utility.”

2. Is the protocol technically sound and planned in a manner that will lead to a meaningful outcome and allow testing the stated hypotheses?

The manuscript should describe the methods in sufficient detail to prevent undisclosed flexibility in the experimental procedure or analysis pipeline, including sufficient outcome-neutral conditions (e.g. necessary controls, absence of floor or ceiling effects) to test the proposed hypotheses and a statistical power analysis where applicable. As there may be aspects of the methodology and analysis which can only be refined once the work is undertaken, authors should outline potential assumptions and explicitly describe what aspects of the proposed analyses, if any, are exploratory.

Reviewer #1: Partly

Reviewer #2: Yes

As suggested, the text has been revised to meet this requirement, as follows:

Lines 163-169: “The selection of the studies will be carried out in three stages. In the first stage, searches will be carried out in the databases. Then, it will be saved and maintained in the Rayyan Systems Inc software [30]. In the second stage, the reviewers will screen the titles and abstracts of the records for eligibility, and the duplicates will be removed. In the third stage, selected full-text articles will be screened for eligibility. The excluded studies and the reason for the exclusion will be recorded. The selection will be performed by two reviewers (SAFB and PCP) independently.”

Lines 169-171: “Disagreements between reviewers will be resolved by consensus or by the decision of a third independent reviewer (CDCMF).”

Lines 239-243: “A narrative synthesis will be carried out, which will provide texts and tables to synthesize and discuss the data of the studies and methodological characteristics, as previously described. In addition, texts and tables will be used to summarize the findings regarding the methodological quality [30, 31], quality of measurement properties [33; 34] and clinical utility [34-36].”

3. Is the methodology feasible and described in sufficient detail to allow the work to be replicable?

Descriptions of methods and materials in the protocol should be reported in sufficient detail for another researcher to reproduce all experiments and analyses. The protocol should describe the appropriate controls, sample size calculations, and replication needed to ensure that the data are robust and reproducible.

Reviewer #1: Yes

Reviewer #2: Yes

We would like to thank the reviewers for the positive comments.

4. Have the authors described where all data underlying the findings will be made available when the study is complete?

The PLOS Data policy requires authors to make all data underlying the findings described in their manuscript fully available without restriction, with rare exception, at the time of publication. The data should be provided as part of the manuscript or its supporting information, or deposited to a public repository. For example, in addition to summary statistics, the data points behind means, medians and variance measures should be available. If there are restrictions on publicly sharing data—e.g. participant privacy or use of data from a third party—those must be specified.

Reviewer #1: No

Reviewer #2: Yes

Due to the characteristics of the present study (protocol of a systematic review without meta-analysis), it does not involve statistics and/or data of participants. In the future, the results of the review will be published in full.

As suggested, this information was pointed out in the manuscript text, as follows:

Lines 30-31: “Data availability: All relevant data from this study will be made available upon study completion.”

Lines 153-154: “The search strategy (S1 File) is composed of blocks of key terms related to the target population, telerehabilitation…”

5. Is the manuscript presented in an intelligible fashion and written in standard English?

PLOS ONE does not copyedit accepted manuscripts, so the language in submitted articles must be clear, correct, and unambiguous. Any typographical or grammatical errors should be corrected at revision, so please note any specific errors here.

Reviewer #1: Yes

Reviewer #2: Yes

We would like to thank the reviewers for the positive comments.

6. Review Comments to the Author

Please use the space provided to explain your answers to the questions above and, if applicable, provide comments about issues authors must address before this protocol can be accepted for publication. You may also include additional comments for the author, including concerns about research or publication ethics.

You may also provide optional suggestions and comments to authors that they might find helpful in planning their study.

(Please upload your review as an attachment if it exceeds 20,000 characters)

Reviewer #1: In this article the authors investigate the measurement properties and the clinical utility of measurement used in telerehabilitation in individual with neurological disease.

Authors should better structure the manuscript and report the novel finding if any regarding this issue. The used of the reviewed articles should be used to answer the main questions under study. Although this is a well written paper with a quite extensive literature survey the major problem is it is not so “innovative”, and the lack of the results strongly limit the decision to proceed with publication.

Firstly, it is important to point out that the present manuscript is related to a systematic review protocol. Therefore, the results cannot be reported in this manuscript. The PLOS ONE Journal has published this type of manuscript, as “REGISTERED REPORT PROTOCOL”. An example of this type of publication at PLOS ONE is:

Tomacheusk RM et al. Measurement properties of pain scoring

instruments in farm animals: A systematic review protocol using the COSMIN checklist. PLOS ONE May 14, 2021 (https://doi.org/10.1371/journal.pone.0251435).

In the methods section, the first paragraph, we have pointed this out, as follows:

Lines 126-129: This systematic review protocol was conducted following Preferred Reporting Items for Systematic Review and Meta-Analysis Protocols (PRISMA-P) [24, 25] and the systematic review results will be reported according to Preferred Reporting Items for Systematic Review and Meta-Analysis (PRISMA 2020 statement) [26].

As pointed out by the second reviewer “given the increasing focus on telehealth and telerehabilitation, this review is timely as it aims to investigate the measurement properties and the clinical utility of measurement instruments used in telerehabilitation in individuals with neurological diseases.”

In the initial searches, it was possible to observe that the number of publications on telerehabilitation is growing, and in 2020 several systematic reviews were published on this topic. However, no systematic review has investigated the measurement properties nor the clinical utility of measurement tools used in telerehabilitation in individuals with neurological diseases. An initial electronic search was performed, resulting in more than 30 articles on measurement properties and the clinical utility of measurement tools used in telerehabilitation in individuals with neurological diseases. Therefore, the main question will be answered with the development of the systematic review. This highlights the importance of developing a systematic review on this topic.

This review will have important results for both research and clinical practice, including a significant number of articles. The identification and use of measurement tools that have appropriate measurement properties and clinical utility may enhance the feasibility and credibility of the evaluation performed remotely, and the comparability of interventions carried out by telerehabilitation. Furthermore, the results of this systematic review will be useful to assist professionals in choosing the measurement tools they will use in telerehabilitation practice.

Information regarding the “innovative” characteristics of the present study was included, as follows:

Lines 79-84: “Telerehabilitation can be defined as the remote delivery of rehabilitation services using information and communication technologies, such as telephone, videoconferencing, and sensors [12]. Telerehabilitation sessions can be used to assess, goal‐setting, intervene, education, and monitoring [2]. The use of telerehabilitation has increased over time as technologies become increasingly prevalent and easily accessible [12].”

Lines 110-115: “As previously discussed, in recent years a wide range of studies have investigated the use of telerehabilitation, consequently, the number of studies investigating the measurement properties of these measurement tools has also increased [17-19, 22]. Systematic reviews are considered the best way to synthesize existing information and provide a comprehensive analysis of the full range of literature on a particular topic [23].”

Lines 117-122: “Given the growth of telerehabilitation in research and clinical practice [12,16] and the challenges of using measurement tools in remote evaluation,20 a systematic review is necessary. Furthermore, this is an important source of information for researchers and professionals to choose measurement tools with adequate measurement properties and clinical utility.”

Reviewer #2: Thank you for asking me to review this systematic review protocol. Given the increasing focus on telehealth and telerehabilitation, this review is timely as it aims to investigate the measurement properties and the clinical utility of measurement instruments used in telerehabilitation in individuals with neurological diseases.

I do have some suggestions and comments:

• How will “telerehabilitation” be defined? Is there a standard definition currently in use?

As suggested, this information was included in the manuscript text, as follows:

Lines 79-84: “Telerehabilitation can be defined as the remote delivery of rehabilitation services using information and communication technologies, such as telephone, videoconferencing, and sensors [12]. Telerehabilitation sessions can be used to assess, goal‐setting, intervene, education, and monitoring [2]. The use of telerehabilitation has increased over time as technologies become increasingly prevalent and easily accessible [12].”

• Why were these neurological conditions such as stroke, Parkinson's disease, spinal cord injuries, multiple sclerosis, cerebellar ataxia, traumatic brain injuries, and peripheral nervous system diseases selected? What about other neurological conditions or co-morbidities? Will they be excluded?

These neurological conditions were used as examples only. Our search strategy was defined according to previous reviews and covered broader terms, such as Nervous System Diseases and Neuromuscular disease, as well as other neurological conditions such as Alzheimer’s Disease and Myelitis. Thus, studies identified through our search strategy that comprise individuals with any neurological condition will be included.

As suggested, the text was revised, as follows:

Lines 137-139: “…who had a neurological condition, for example, stroke, Parkinson's disease, spinal cord injuries, multiple sclerosis, cerebellar ataxia, traumatic brain injuries, and peripheral nervous system diseases, will be included.”

• I assume you will be including only quantitative studies. If so, you will need to state that you will exclude qualitative studies.

As suggested, this information was included in the manuscript text, as follows:

Lines 143-144: “Systematic reviews and qualitative studies will also be excluded, but their reference lists will be screened for relevant studies.”

• What will you do with articles which are in language other than English? How will you translate those?

The review authors are proficient in English, Portuguese, French, and Spanish. These languages comprise an extensive part of the scientific literature. If there are articles in other languages, the authors of the paper will be consulted about the existence of an English version of the material, and in its absence, a translator will be consulted.

• Will databases such as CINALH/Emcare (allied health specific databases), Cochrane, Scopus/Web of Science be searched?

The databases selected for conducting the searches were selected with the assistance of an experienced researcher, and according to previous studies. In addition, the indexing profile of each database was observed according to the revision area. Electronic searches will be performed in following databases: Medical Literature Analysis and Retrieval System Online (MEDLINE Ovid), Excerpta Medica Database (Embase Classic + Embase Ovid), Physiotherapy Evidence Database (PEDro), Scientific Electronic Library Online (Scielo), and Literatura Latino-Americana e do Caribe em Ciências da Saúde (LILACS).

The databases were selected because they cover international literature in the area of biomedicine and life science topics, covering topics from different areas, such as bioengineering, public health, clinical care, animal science, nursing, and dentistry.

Systematic reviews were not included in this review, so the Cochrane database was not used.

• How will you address publication and location bias? Are you planning on searching grey literature?

One way to avoid publication and location bias is to have no language restrictions.

“Because of the potential for bias when search strategies are limited to databases of peer-reviewed scholarly journals, the systematic review process should also include an examination of the "grey" literature.” (Portney & Watkins, 2009) Examples include research reports, working papers, conference proceedings, dissertations, and theses.

Only full-text articles published in indexed scientific journals will be included in this study. During the stage of selection of the studies, the “grey” literature will be excluded. In addition, conference abstracts, commentaries, letters, dissertations, and editorial papers will be excluded.

As suggested, this information was revised in the manuscript text, as follows:

Lines 135-136: “All full-text papers that aimed to investigate the measurement properties of measurement tools used in telerehabilitation in adults (age ≥18 years old) …”

Lines 141-142: “Research reports, working papers, conference proceedings, conference abstracts, commentaries, letters, dissertations, theses, and editorial papers will be excluded.”

• Will the search terms include both keywords and MeSH?

Our search strategy was designed according to previous studies and with the assistance of an experienced researcher, and used a combination of controlled vocabulary and text‐word terms. This is composed of blocks of key terms related to the target population, telerehabilitation, as measurement properties. Thus, the search terms include both keywords and MeSH.

• Extraction of data is not part of study selection section. Please remove.

As suggested, the text was revised, as follows:

Lines 163-169: “The selection of the studies will be carried out in three stages. In the first stage, searches will be carried out in the databases. Then, it will be saved and maintained in the Rayyan Systems Inc software [30]. In the second stage, the reviewers will screen the titles and abstracts of the records for eligibility, and the duplicates will be removed. In the third stage, selected full-text articles will be screened for eligibility. The excluded studies and the reason for the exclusion will be recorded.”

• How will discrepancies in data extraction be resolved?

Disagreements between reviewers will be resolved by consensus or by the decision of a third independent reviewer.

As suggested, this information was included in the manuscript text, as follows:

Lines 179-181: “Disagreements between reviewers will be resolved by consensus or by the decision of a third independent reviewer (CDCMF).”

• I am unclear what “systematic narrative synthesis” means. Could you provide more detailed overview of this process?

The expression “systematic narrative synthesis” was replaced by “narrative synthesis”. “Narrative’ synthesis’ refers to an approach to the systematic review and synthesis of findings from multiple studies that rely primarily on the use of words and text to summarise and explain the findings of the synthesis (Popay et al., 2006).”

According to PRISMA 2020, “many systematic review reports include narrative summaries of the characteristics and risk of bias across all included studies.” In addition, according to the PRISMA 2009, “following the presentation and description of each included study, as discussed above, reviewers usually provide a narrative summary of the studies. Such a summary provides readers with an overview of the included studies. It may for example address the languages of the published papers, years of publication, and geographic origins of the included studies”.

Lines 239-243: “A narrative synthesis will be carried out, which will provide texts and tables to synthesize and discuss the data of the studies and methodological characteristics, according to previously described. In addition, texts and tables will be used to summarize the findings regarding the methodological quality [30, 31], quality of measurement properties [33; 34] and clinical utility [34-36].”

• Minor point – in some instances, the tense is future (which is correct as this is a protocol) and in other instances, it is in past tense (incorrect). Please amend.

We are sorry for this mistake. As suggested, the errors have been fixed. The excerpt that remained in the past tense refers to the elaboration of the search strategy attached to the protocol. This has already been done.

Lines 151-153: “The established search strategy for the MEDLINE database will be adapted to suit the other databases.”

Lines 153-154: “The search strategy (S1 File) is composed of blocks of key terms related to the target population, telerehabilitation, as measurement properties, as follows:”

Submitted filename: Response to Reviewers.docx

Click here for additional data file.

9 Mar 2022

Measurement properties of outcome measures used in neurological telerehabilitation: a systematic review protocol

PONE-D-21-29425R1

Dear Dr. Faria,

Thank you for submitting your revised manuscript - the original reviewers have both kindly assessed the draft and we are all satisfied that the comments have been appropriately addressed. We are, therefore, pleased to inform you that your manuscript has been judged scientifically suitable for publication and will be formally accepted for publication once it meets all outstanding technical requirements.

Within one week, you’ll receive an e-mail detailing the required amendments. When these have been addressed, you’ll receive a formal acceptance letter and your manuscript will be scheduled for publication.

An invoice for payment will follow shortly after the formal acceptance. To ensure an efficient process, please log into Editorial Manager at http://www.editorialmanager.com/pone/, click the 'Update My Information' link at the top of the page, and double check that your user information is up-to-date. If you have any billing related questions, please contact our Author Billing department directly at authorbilling@plos.org.

If your institution or institutions have a press office, please notify them about your upcoming paper to help maximize its impact. If they’ll be preparing press materials, please inform our press team as soon as possible -- no later than 48 hours after receiving the formal acceptance. Your manuscript will remain under strict press embargo until 2 pm Eastern Time on the date of publication. For more information, please contact onepress@plos.org.

Kind regards,

Alastair D. Smith

Academic Editor

PLOS ONE

Additional Editor Comments (optional):

Reviewers' comments:

Reviewer's Responses to Questions

Comments to the Author

1. Does the manuscript provide a valid rationale for the proposed study, with clearly identified and justified research questions?

The research question outlined is expected to address a valid academic problem or topic and contribute to the base of knowledge in the field.

Reviewer #1: Yes

Reviewer #2: Yes

**********

2. Is the protocol technically sound and planned in a manner that will lead to a meaningful outcome and allow testing the stated hypotheses?

The manuscript should describe the methods in sufficient detail to prevent undisclosed flexibility in the experimental procedure or analysis pipeline, including sufficient outcome-neutral conditions (e.g. necessary controls, absence of floor or ceiling effects) to test the proposed hypotheses and a statistical power analysis where applicable. As there may be aspects of the methodology and analysis which can only be refined once the work is undertaken, authors should outline potential assumptions and explicitly describe what aspects of the proposed analyses, if any, are exploratory.

Reviewer #1: Yes

Reviewer #2: Yes

**********

3. Is the methodology feasible and described in sufficient detail to allow the work to be replicable?

Descriptions of methods and materials in the protocol should be reported in sufficient detail for another researcher to reproduce all experiments and analyses. The protocol should describe the appropriate controls, sample size calculations, and replication needed to ensure that the data are robust and reproducible.

Reviewer #1: Yes

Reviewer #2: Yes

**********

4. Have the authors described where all data underlying the findings will be made available when the study is complete?

The PLOS Data policy requires authors to make all data underlying the findings described in their manuscript fully available without restriction, with rare exception, at the time of publication. The data should be provided as part of the manuscript or its supporting information, or deposited to a public repository. For example, in addition to summary statistics, the data points behind means, medians and variance measures should be available. If there are restrictions on publicly sharing data—e.g. participant privacy or use of data from a third party—those must be specified.

Reviewer #1: Yes

Reviewer #2: Yes

**********

5. Is the manuscript presented in an intelligible fashion and written in standard English?

PLOS ONE does not copyedit accepted manuscripts, so the language in submitted articles must be clear, correct, and unambiguous. Any typographical or grammatical errors should be corrected at revision, so please note any specific errors here.

Reviewer #1: Yes

Reviewer #2: Yes

**********

6. Review Comments to the Author

Please use the space provided to explain your answers to the questions above and, if applicable, provide comments about issues authors must address before this protocol can be accepted for publication. You may also include additional comments for the author, including concerns about research or publication ethics.

You may also provide optional suggestions and comments to authors that they might find helpful in planning their study.

(Please upload your review as an attachment if it exceeds 20,000 characters)

Reviewer #1: No additional comments ,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,,

Reviewer #2: Thank you for addressing my comments and suggestions. I am happy with the revisions that have been undertaken.

**********

7. PLOS authors have the option to publish the peer review history of their article (what does this mean?). If published, this will include your full peer review and any attached files.

If you choose “no”, your identity will remain anonymous but your review may still be made public.

Do you want your identity to be public for this peer review? For information about this choice, including consent withdrawal, please see our Privacy Policy.

Reviewer #1: No

Reviewer #2: No

11 Mar 2022

PONE-D-21-29425R1

Measurement properties of outcome measures used in neurological telerehabilitation: a systematic review protocol

Dear Dr. Faria:

I'm pleased to inform you that your manuscript has been deemed suitable for publication in PLOS ONE. Congratulations! Your manuscript is now with our production department.

If your institution or institutions have a press office, please let them know about your upcoming paper now to help maximize its impact. If they'll be preparing press materials, please inform our press team within the next 48 hours. Your manuscript will remain under strict press embargo until 2 pm Eastern Time on the date of publication. For more information please contact onepress@plos.org.

If we can help with anything else, please email us at plosone@plos.org.

Thank you for submitting your work to PLOS ONE and supporting open access.

Kind regards,

PLOS ONE Editorial Office Staff

on behalf of

Dr Alastair Smith

Academic Editor

PLOS ONE