PONE-D-20-16637

Modifying and Validating the Social Responsiveness Scale Edition 2 for use with Deaf Children and Young People

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Comments to the Author

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Reviewer #1: Yes

Reviewer #2: Yes

Reviewer #3: Yes

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Reviewer #1: Yes

Reviewer #2: Yes

Reviewer #3: Yes

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Reviewer #1: Yes

Reviewer #2: No

Reviewer #3: Yes

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Reviewer #2: Yes

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Reviewer #1: Review of “Modifying and Validating the Social Responsiveness Scale Edition 2 for use with Deaf Children and Young People”

This paper describes a recent adaptation and validation of the Social Responsiveness Scale (SRS-2) for use with deaf children. There are currently no validated instruments available for the screening or diagnosis of autism spectrum disorder (ASD) in deaf children. As such, this endeavor represents an essential step forward in beginning to provide culturally and linguistically appropriate tools for deaf children. I applaud the researchers for taking on this work, as it is sorely needed.

Overall the paper is quite complete, well written, and the procedure is adequately described. It is easy to understand how the researchers went about the adaptation and validation procedure, and the procedures selected are in line with best practices. The authors also successfully show that the tool is able to discriminate between ASD and non-ASD children (as assigned by clinical criteria). The sensitivity of the instrument is very good, only missing 2 children with ASD. Specificity also appears to be acceptable, despite initially classifying many non-ASD children as ASD-positive.

My concerns about the paper are mostly relatively minor. However, the paper could be strengthened by addressing the following issues:

1) Throughout the paper there is confusion about whether or not the instrument used in the validation study was in English or in British Sign Language (BSL). I understand that the SRS-2 was adapted in written English and then translated and again back-translated into BSL. However it is unclear how many families who then completed the instrument did so in English or BSL, and if the sensitivity and specificity are comparable in each modality. There are a number of places in the manuscript where this should be clarified. For example, p. 7, does “this newly modified instrument” refer to the BSL instrument, or Deaf-adapted English instrument? Similarly, p. 27, Table 3, including information about the hearing status of the parents and modality that the SRS was completed in would help the reader to better understand how the SRS was completed and the nature of the sample. There is also no discussion of any translation issues encountered in this process, although the translation from English into BSL is no small undertaking. If possible, I would like to see more detail about this.

2) The way that the paper is structured, it appears that some of the results are hinted at in the method. For example, p. 9, “some questions were deemed unsuitable to assess ASD in deaf children and were discarded” makes the reader want to know which questions were discarded, but those are not reported until the Results. Authors should consider restructuring this paragraph so as to not beg these questions. A construction such as “If questions were deemed unsuitable to assess ASD in deaf children, they were discarded” may work better. This is true for other sections in the Method: e.g., when you describe the Independent Research Review Team on p. 11, I was left wondering how many participants from each group were included.

3) p. 25, text reads: “Many deaf children have poor access to language and education and consequently, may not have a good understanding of English metaphor. This is particularly prevalent [sic] if they are exposed to spoken and signed languages, each having very different use of metaphorical expressions.” I find this section a bit problematic. Having poor access to language leads to much bigger problems than merely not understanding metaphors – it can lead to not understanding basic linguistic structures! Additionally, being exposed to sign and speech does not seem to me to be the problem – if a child is exposed to both, then they would likely understand metaphor in both modalities. However if a child is only exposed to speech, and has poor access to spoken language, they are unlikely to understand metaphor in either modality. In other words, the effects of language deprivation are not clearly described here, and currently the text is a bit misleading. In addition, the item that this section introduces does not focus on metaphor, but rather on general pragmatic abilities (“takes things too literally and doesn’t get the real meaning of a conversation”). I suggest that this section be rewritten to be clarified.

Global terminology concerns:

a) I’m not sure that the term “subjects” is correct when referring to deaf children (e.g. on p. 3 in the introduction) since the children are not technically participants in research. Similarly, the use of the term “participants” to refer to the children (e.g. in Fig. 3) is confusing, since both parents and children are technically participants in the research. I would suggest clarifying by using terms such as “children” and “parents” to specify which part of the research you are referring to.

b) please use “ASD” rather than “autism” throughout the paper, line with current DSM terminology.

Other/minor comments:

p. 3: definition of the term “deaf” is circular – please define in terms of hearing loss

p. 4: please report the comparable statistics of age of diagnosis for hearing children so that the reader can see the discrepancy between deaf and hearing

Missing punctuation: p. 5, end of paragraph 1; also, p. 25, line 562: punctuation missing from end of sentence. Also please use the serial (Oxford) comma between elements in a series of three of more items, in line with APA style.

p. 6, lines 128-131: this sentence is a bit awkward and confusing. The way it is currently written it reads as though the SRS is a parent semi-structured interview rather than the ADI-R

p. 8, there are two places where “see below” appears in the text; it would be better to replace these with more concrete directions (i.e., “see Fig.1”, etc.).

The Delphi Consensus Method is referred to rather extensively before it is finally described on p. 8. In addition, the reliability and validity of this method is described on lines 194-199, but should be described earlier.

The International Expert Panel consists of experts from a variety of countries, including non-English speaking countries, which introduces a wrinkle into the methodology, though not a big one (nearly all of the respondents were from English speaking countries). I find the description of the Panel on p. 9 a bit lacking. Perhaps you could mention here that most of the respondents were from English speaking countries, not just in Table 1.

p. 15, use of the SCQ also warrants caution, for the same reasons that the SRS-2 is being adapted. It is not validated for deaf children and contains items that are inappropriate (e.g., does the child respond when name is called). This should perhaps be noted.

Table 2: Round 1 says that 47 questions remained, but in Round 2 the sum of accepted and remaining items is 48. Is the reason for this discrepancy the addition of the new item? Perhaps I would asterisk the accepted item number as well to make this clear, if this is the case.

Table 3, p. 27, the column headers (“Diagnostic Group” vs “NICE guideline standard clinical assessments”) are confusing. Is one group a subset of the other? If so, perhaps this should be reformatted. Currently it looks like they are two independent groups.

p. 29, please specify in the text which groups there are statistically significant differences between.

p. 36, line 759: “…required all experts to be able to take part using the English language.” How did the deaf experts take part? Using written English?

Will the Appendix be included in the publication?

Reviewer #2: This well-conceived and soundly-executed study describes the modification and validation of a screening instrument designed to identify deaf children who should receive a full diagnostic evaluation for autism spectrum disorder. An existing screener was adapted via the guidance of a panel of international (English-speaking) experts, and was validated with a sample of 198 deaf children who did or did not already have an ASD diagnosis: a very large sample for this field. The modified screener had excellent internal consistency and test-retest reliability. Sensitivity was good and specificity was adequate for a screener.

I have no major concerns about this study, and only a handful of minor questions.

1. I am curious as to whether the recruitment efforts for the validation study specifically included ASD communities who view their diagnosis as a dimension of human neurodiversity rather than as pathology. (I am not sufficiently familiar with the British organizations listed to determine this on my own.) If so, I would view that as a strength and recommend that the authors make that explicit. If not, I would view that as a limitation and recommend that the authors include that in the list of limitations provided in the discussion.

2. Given the staggering heterogeneity of DHH children’s backgrounds, I would appreciate more detail on what constituted “a comprehensive history of the child’s development, family, and medical history”, especially with regard to their language history. Perhaps this information (or, at the very least, the questionnaires used) could be included as supplemental material.

3. I find Table 2 difficult to interpret, and note that it is not discussed anywhere in the text.

4. p. 24, ln 538-544: I support the inclusion of a caveat message like this; however, the particular verbiage here puzzles me. For a child who mixes mostly with hearing children, the caveat is barely needed, since those children would presumably be the only context that the carers could consider. For a child who mixes with both deaf and hearing children, the direction to consider only interactions with deaf children strikes me as inappropriate. The assumption seems to be that socializing with deaf children with allow barrier-free communication, but that is simply not the case. A deaf child who is a fluent signer may be surrounded by deaf children whose language skills are far below their developmental age. A deaf child who has been raised with a focus on listening and spoken language may be placed in a signing environment for the first time and be surrounded by deaf children who are using a language that is entirely new to them. It would seem more appropriate to simply truncate the instruction after the second sentence.

p. 25 ln 572-273: I assumed that the example item given here belonged to the original set; I recommend simply removing the text of the item from this sentence.

Table 3: I would appreciate more comment about the extent to which the demographics of the sample do or do not reflect the demographics of the UK population, as well as some discussion about the potential for existing assessments to over-/under-identify ASD in various demographic groups. Is there reason to believe that the SRS-2 Deaf Adaption fares any better or worse on this front?

Reviewer #3: Thank you for the opportunity to review this interesting manuscript. The authors report on a modification process and validation study of the SRS-2 for use in screening children and youth who are deaf for ASD. This is an area in which improvements are definitely needed, due to the complexities of assessing children who are deaf given differences in language development, behaviors in context, etc. Following a Delphi method informing modification of items, several psychometric properties of the adapted tool were assessed with a sample of British children (N=198) and parents; approximately 38% of the children met diagnostic criteria for ASD. ROC analyses were used to identify an optimal cut-point on the adapted tool. Strengths include the use of the Delphi process to engage international experts to review the original SRS-2 items, as well as subsequently modified items, over 4 waves of deliberation to develop an adapted version of the instrument. Participants for the validation study appear to have been recruited from a broad array of settings, increasing generalizability. The psychometric assessments of the screening tool, according to the authors, suggested that the adapted tool performs with similar levels of sensitivity and specificity as other ASD screening tools with hearing populations. The focus on this understudied population is important and appreciated.

Some weaknesses of the manuscript in its current form were noted, and these are listed below.

1) Table 2 and the text immediately following are very difficult to follow in terms of the process of reviewing, accepting, modifying, and discarding items. Perhaps the authors could describe this process in a different way. Just one example of difficulties interpreting the table – at the end of round 4, there were 5 “remaining” items, and it is unclear what “remained” means – they ended up being included in the final screening tool, but why?

2) Rather than the piecemeal description of which items were modified and why, it would be very helpful if the authors could organize this information in a more meaningful way. For example – if there were a certain number of “common” rationales for modifying items, perhaps a table could be developed listing each item, whether it was changed or not, and the rationale for the change. The final tool is presented in the additional materials, and it could instead be a part of the manuscript reporting the modifications, deletions, and addition to the items of the original SRS-2.

3) An example of a rationale for modifying an item that was not very clear is re: the item that was replaced because of the phrase “too tense,” with a statement that it was not clear how being “too tense” could be evaluated consistently in children who are deaf. That example begs the question of how being “too tense” is evaluated in hearing children. The rationale is not terribly clear – it may just be a vague item that does not clearly measure what it is intended to measure, regardless of the population of children targeted.

4) Similarly, additional information and justification for the items pulled from the two age-based versions of the SRS-2 would be helpful (i.e., not just that the group decided to go with an item from one rather than the other, but why).

5) Information on which 12 items did not load on the primary factor is needed.

6) It is unclear why parents of hearing children were not included in the test-retest reliability assessment.

7) Table 3 – percentages are missing for the child age categories under the NICE column.

8) Line 665 says diagnostic group, should refer to the NICE assessment group

9) Line 737, please clarify that 73% refers to the t-score

10) Line 751 refers to increasing agreement among the expert panel, but the authors initially described lowering the bar for agreement from 80% to 75% percent for the fourth round – please clarify.

11) As addressed in the introduction section, 90% of parents of children who are deaf are hearing, themselves. Were there deaf parents/carers in the study? There is no mention of this in Table 3. If not, the sign language version of the screening tool was not actually assessed for reliability and validity (and even if there were a few deaf parents/carers, there were likely not enough to make conclusions re: the translated tool’s psychometrics – this would need to be addressed in future studies).

12) I would strongly recommend a very close copy-editing of the manuscript. There are many issues with punctuation and phrasing that detract from the importance of the work.

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Reviewer #1: No

Reviewer #2: No

Reviewer #3: No

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Modifying and Validating the Social Responsiveness Scale Edition 2 for use with Deaf Children and Young People

PONE-D-20-16637R1

Dear Dr. Wright,

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PLOS ONE