Early Introduction of Power Mobility Devices for Children with Fukuyama Congenital Muscular Dystrophy and Its Psychological Impact on Caregivers: A Case Report

Round 1

Reviewer 1 Report

Dear Authors,

This case study of a child with a rare diagnosis is interesting. The child is learning to use a Powered Mobility Device and the fathers changing perspectives over the course of the childs development of goal-directed driving is important to recognise. Understanding the parents perspective is necessary to support their persistance to use the device with the child, a period long enough to see achievements. The ALP can help the parents see the childs smaller steps forward, which may support their persistance. It is very important for professionals to build relationships with parents with children in very young ages, when introducing new interventions such as PMD.

However, there are needs of clarification and reorganisation of the text in this paper. Pleas see commentaries in attached file.

1) Please clearly state the aim (objective) of the study in the end of the Introduction. The aim has to be in one place and be in line with the reported Results and Discussion. Now different aims are stated at different places in the introduction which is very confusing.
At lines 44-46 is stated: In this study the authors hope to examine the reliability and validity of each (referring to the PMP and the ALP assessments). -Which is not examined as this is a case study and the PMP is not used in the data collection.
At lines 85-88 is stated: In this study, a case is reported to clarify clinical questions for future RCTs. -Which is rather a part of Future research in the Discussion.
Then at lines 92-99 two objectives are stated: first to clarify the childs developmental changes related to using a Powered Mobility Device, and second to understand how the fathers perspective shifted in alignment with the childs development of goal-directed driving with the device. -Which I find are the aims of this study.

2) I would suggest to start the Method by stating this is a case study.
And please see to that the ethics are described where the journal want them to be placed.
The method would also benefit from reorganising the information in the sections Procedure and Analysis.
Please add a section for Data collection, before Analysis. The Data collection could include descriptions of how and when the video footage was collected, of the Driving to Learn as a therapeutic intervention (Nilsson et al 2011); of the ALP assessment (Nilsson and Durkin, 2014) (also that it was the Japanese version of the ALP that was used - can be found at https://www.lisbethnilsson.se/en/alp-tool/alp-tool-in-japanese/), and of the interview guide and the interviews with the father (primary caregiver).

3) Please organise the Result so it clearly responds to the aims/objectives

4) Please reorganise the Discussion to begin with a short note on the most important results and reasoning on them. Please sort strengths and limitations of the study into Methodological considerations

5) References need to be corrected.

 

Comments for author File: Comments.pdf

There is a moderate need of improving the quality of English language.

This may be related to a different use of such as personal pronouns in Japanese, and shows especially in Table 3

Author Response

Please see the attachment.

Author Response File: Author Response.pdf

Reviewer 2 Report

Fukuyama congenital muscular dystrophy (FCMD) is almost exclusively found in Japan. This is the sole drawback of this present manuscript and based on this it is very difficult the results of this case report, confirmed by future large-scale, better-controlled studies (as author said in conclusions/limitations). Overall, it is a well written paper. Furthermore, the overall propose to manage children’s mobility disabilities in FCMD is well supported.  

Author Response

Please see the attachment.

Author Response File: Author Response.pdf

Reviewer 3 Report

The current study was a case report involving 1 participant who was a 1 year and 10 month old girl diagnosed with Fukuyama congenital muscular dystrophy. The purpose was to investigate the influence of a power mobility device (PMD) on the Assessment of Learning Powered mobility use tool (ALP). In addition, the father of the participant was interviewed and the responses accessed. The rationale for the study was that previous studies on PMDs in infants had mainly focused on cerebral palsy and had not investigated PMDs for Fukuyama congenital muscular dystrophy.

 

The study appeared to last a total of 20 months (Table 1) and the participant was given a PMD device which consisted of a motorized sitting chair with a joystick to control it. The ALP was measured every 3 months using video footage (6 total videos). At the 18 month time point, an interview with the father was conducted in which he was asked questions regarding what he had expected from the PMD, what communication changes occurred in the family, how were the child’s driving skills, and changes in the parents lives. Unfortunately, the child was hospitalized for weeks 12-15 and could not use the PMD during that part of the study. The primary results were that ALP levels increases from values of 1-2 to values of 8 for all 5 categories (attention, activity/movement, understanding of tool use, expressions/emotions, interactions/communication). The parent interview results were fairly complicated and difficult to interpret in detail from my reading of the paper. Overall, it appeared that the father thought the PMD was a net positive for the child.

 

 

This study is on an important topic and provides some useful information to clinicians who care for this type of patient and to readers of this journal. Being a case report, the study has all the normal and expected weaknesses of a case report, which is understandable. The study may ultimately be publishable and add to the literature, but needs sine major revisions due to the concerns below.

 The study is not that well controlled even for being a case study. There was no interview with the father before the intervention to compare the 18 month interview too. All of the measures used in the study were subjective and the evaluators were not blinded. All categories of the ALP increased but without a control or a comparison to previous data or even clinical expectations based on experience, it is difficult to understand how the author arrived at the conclusions presented in paragraph 2 of the Discussion. The author needs to explain how all of these concerns do not take away from the impact of the paper. In other words, the Discussion has to be much better and more precise.

 Overall, the English of the current manuscript is pretty good, but there were some grammatical and typographical errors. As just one example, the patient is referred to as “participants” in line 102. The interview text has numerous major grammatical mistakes, I am not sure if those occur because the parent talked that way or how the interview was transcribed. It makes it very difficult to understand exactly what he meant In summary, the paper unfortunately needs an English speaker to completely rewrite it before it can be evaluated fairly.

Some inconsistencies in the Methods. The abstract says the interview with the father was at 19 weeks, the methods say 18 weeks.

 Why were the physical therapy students that helped the single author, not included as authors if they did a lot of work on the evaluation of the videos etc?

 The Introduction could be shortened, made more concise, and the paragraphs that are only comprised of 1 or 2 sentences combined with other paragraphs.

 Line 32. Is “trigger” the best word to use in this sentence?

 Some mistakes in the Bibliography. For instance, if we only look at references 18, 21, and 23 as an example…. Some titles of articles have every word capitalized others don’t.

 

 

See main review.

Author Response

Please see the attachment.

Author Response File: Author Response.pdf

Round 2

Reviewer 1 Report

Dear Authors,

Thank you for the revised version of the manuscript.

I only have a few commentaries on the language that may cause misunderstandings, please see attached file.

It is an interesting paper highlighting parent perspective and need for therapists to work in partnership with parents

Comments for author File: Comments.pdf

I only have a few commentaries on the language that may cause misunderstandings, please see attached file.

Author Response

Thank you for your comment.
I have checked and revised the points you have suggested. (Lines 117, 119, 191-192, 214)

Reviewer 3 Report

The author has answered all my prior comments. Perhaps a little more English proofreading is needed, but other than that my concerns have been addressed. 

The author has answered all my prior comments. Perhaps a little more English proofreading is needed, but other than that my concerns have been addressed. 

Author Response

Thank you for your comment.
I have checked and revised the points you have suggested. (Lines 117, 119, 191-192, 214, Table3)