Identifying the Resource Needs of Young People with Differences of Sex Development

REVIEWER

RESPONSE

CHANGE

1

You have mentioned male/female participation; what about those who felt indeterminate

The number of people identifying as other than male or female was low in this group. Only 2 participants identified as intersex (line 109, table 1), while none of those declining participation had a non-binary gender recorded on their file (although some had not been seen at the hospital for a long time so only information about their sex assigned at birth was available).

To make this clearer, we have added to line 83 (Age and gender/sex of non-participants was collected from their file) and line 106 (Numbers of people identifying with non-binary identities were too low to compare in this group.)

2

What were the opinion of participants on timing of sex assignment. Do they support early assignment or would have liked to have left it to later date when they had grown up

Unfortunately, this question was not covered by our survey as the focus of our survey was to explore their resource needs.

3

What is the incidence of gender dysphoria

No participant reported previous gender incongruity in response to “Some people's gender identity can change over time, has this ever been the case for you?”

However, one did not know and nine did not respond to this question.

We have now modified line 111 to read: “No participant reported previous gender incongruity, however one ‘did not know’ and nine did not respond.”

4

Was there any decisional support/ regret on early surgery

As the purpose of this project was to explore resource needs, this was not explored in detail. Overall participants were accepting of their medical intervention. For example, the timing of their intervention was not asked. We choose not to include the following information as it did not appear to add to the central question focused on their information and support needs.

Of respondents who had genital surgery(n=21), 17(81%) reported the decision was appropriate, 1(5%) said it wasn’t and 3(14%) didn’t know.

Of respondents who had gonadal surgery(n=13), 10(77%) reported that the decision was appropriate, 1(8%) said it wasn’t and 2(15%) didn’t know.

Of respondents who used self-dilatation(n=17), 15(88%) reported the decision was appropriate and 2(12%) didn’t know.

Of respondents who had hormone therapy(n=30), 29(97%) reported the decision was appropriate and 1(3%) did not know.

REVIEWER

RESPONSE

CHANGE

1

There are no descriptions of the measures used in the study. Were these validated measures or measures developed by the authors?

We agree this should be clarified:

Patients were surveyed using a specifically designed questionnaire delivered online. A link to the survey is in the references and supplementary material.

Added to line 87 (Patients were surveyed using a specifically designed questionnaire delivered online)

2

How is "current distress" operationally defined? Is this related to anxiety (worried/troubled/distressed) or depression or uncertainty or something else?

We agree this should be clarified: Current distress was defined as respondents strongly agree or agree to feeling worried/troubled/distressed on a 5-point Likert scale

To clarify this more clearly, we have now added a sentence to line 93: “Distress was assessed by responding ‘strongly agree’ or ‘agree’ to feeling worried/troubled/distressed on a 5-point Likert scale.”

3

Were any data collected from the parents of the participants? It would be interesting to know what their memories were about the time of disclosure, especially for children who were diagnosed at birth or a very young age.

A separate study of parents' support and information needs occurred at the same time. However, the respondents were separate families to this study population. This study involved only self-reports from the participants themselves.

4

While CAH and TS are included in the diagnoses mentioned in the 2016 Consensus Statement (Lee et al., 2016), patients with CAH strongly advocate that their diagnosis is not a DSD. Typically, patients with TS are also not treated under the "umbrella" of DSD. I think the authors need to acknowledge this information some where in the "Materials and Methods" section.

We agree, and acknowledge that there is disagreement about which diagnoses fit into the “umbrella” of DSD. After consideration, we thought it most consistent to follow the classification proposed by the consensus statement.

5

For figures 1 & 2, are these differences statistically significant?

Yes, a chi-squared test was performed for all figures. We agree this should be added.

We have amended Figures 1 and 2 to include the test performed and the corresponding p-value.

6

While I appreciate the authors providing quantitative data, I would encourage them to consider presenting this as more of a qualitative study. By definition, it is quantitative because Likert scales were used, but because of the unclear definition of "distress", and lack of statistical significance (I'm assuming data in Figures 1 & 2 are not significantly different) there is a very large subjective piece to the study.

We agree that there is a subjective component to this study however no qualitative methods were used. We have tried to adjust some of the wording to reflect the exploratory nature of the study. We have also defined our definition of distress and added the statistics to the figures.

To clarify this more clearly, we have now adjusted:

Line 94: “Distress was assessed by responding ‘strongly agree’ or ‘agree’ to feeling worried/troubled/distressed on a 5-point Likert scale.”

Line 159: “(X² = 61.6, p<0.0001).”

Line 165, 166: “(X² = 26.1, p<0.0001), (X² = 59.4, p<0.0001).”

Line 208: removed “provides quantitative evidence”