Research methodology was approved by The Sydney Children’s Hospital’s Network Human Research Ethics Committee (LNR/15/SCHN/296) and Monash University Human Research Ethics Committee (30310) with no deviation to set protocols. This research comprised of two studies with different methodologies. Study 1 used an online modified Delphi technique to gain consensus and agreement on the core set of outcome measures. Study 2 was an online survey of families with lived experience of treatment from health professionals for their children’s toe walking gait. Both studies were performed in accordance with relevant guidelines and regulations. All participants provided written informed consent through either return of a paper consent form or through indication on an online check box.
Study 1 potential participants were identified through hospitals, health professional department networks, and recent publications found by the research team google scholar. Potential participants were sent individual email invitations. Following return of a signed consent form, participants were provided with a unique number to link responses, and a survey link to complete the online Round 1 of the Delphi survey. This purpose-built survey was trialed prior to use with two health professionals that were not invited to participate, and wording modified as suggested (Appendix 1). Participants were asked their qualifications, profession, gender, recency of practice, practice settings, asked to estimate the percentage of children they worked with clinically, or during research (compared to adults), and an estimated number of children treated annually who had ITW. Participants were continually reminded of the research aim relating to treatment outcome measurement rather than diagnosis, to minimise statements relating to investigations that aid condition specific diagnosis (e.g., medical imaging), or to focus on treatment outcomes relating to ITW, to minimise mention of assessments specific to other conditions known to cause toe walking such as spasticity.
Participants were guided through a series of multichoice and open-ended response boxes about the types of questions they asked parents during treatment. Survey logics were used when a participant indicated in a multichoice box response (e.g., when the participate identified pain as a question domain), they were then asked to specify how that data should be collected. Participants were also provided free text for additional questions that did not align to domains. Participants were then asked to describe physical assessments used to understand treatment success, with domains aligning to joint ranges of motion, balance, gait, strength. Again, participants were able to select none of those domains or provide further details if they collected different measures. Where a domain was chosen, they were asked to specify how that information was collected from the child.
Where 70% or greater participant responses in Round 1 were similar, these statements were accepted. Consensus responses were fed back to participants in Round 2 for confirmation on wording or display of any scale. Questions or assessments described by 50-69% of participants were refined and re-presented to participants in Round 2 for agreement rating. Where questions or assessments were described by less than 50% of participants, they were discarded.
In Round 2, participants were asked to provide agreement on the generated statements using a 5-point Likert scale (Strongly Disagree, Disagree, Neutral, Agree, Strongly Agree). Participants were also requested to comment about question phrasing. Similar to Round 1, any questions or assessments with 70% or greater agreement were included within final core set. Questions where 50-69% of participants agreed were re-phrased based on any feedback and re-presented to participants in Round 3. Any questions were less than 50% agreed were removed. This process was replicated in Round 3 and any outstanding questions meeting 50-69% agreement at the end of Round 3 were presented to parent participants in Study 2, and questions 50% or less were removed.
In Study 2, a purpose-built online survey (Appendix 2) was advertised through social media, online parent forums and through clinical networks of authors to pass on to potential participants. Parents of children who toe walked from any reason, were invited to provide online feedback on what outcome questions and assessments were important to them during toe walking treatment.
Parent participants were initially asked about which country they lived in, their relationship to the child who toe walks, how many children (<18 years) were in the family, how many children (<18 years) toe walked, reason given by health professional for toe walking gait, health professional/s accessed for treatment within past six months, overall satisfaction with questions and assessments about treatment and toe walking treatments offered, refused, or recently undertaken.
The online survey displayed the questions or assessments developed in Study 1 to parent participants. Survey logic supported parent participants to indicate importance of each question or assessment based on their treatment experiences. Where a parent provided a response of “not at all important” or “slightly important’, they were asked why, then presented with the question or assessment developed in Study 1 and asked to indicate agreement with it being used given the expert health professionals found this important.
Where parent participants responded “moderately important” or “very important’ to a question or assessment, they were presented with how it was described in Study 1 and asked the four questions of the validated Intervention Appropriateness Measure (IAM) . Parent participants were also asked to indicate their level of agreement in their own ability to answer the question without a health professional being present for coaching purposes or if they could be taught to take the measurement without the health professional being present. Parent participants were provided with open-ended questions to provide further information if there were additional questions or assessments, they considered important.