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We highly appreciate that Hall and colleagues have critically reviewed our protocol and acknowledge their concerns regarding the lack patient and public involvement (PPI). We are aware of the COS-STAD recommendations and have carefully considered PPI. Nevertheless, we ultimately decided not to include parental input in the Delphi process. The most important consideration was that parents of asymptomatic patients may have difficulty providing input on desired outcome measures, since most of them are not confronted with symptoms or complications. Furthermore, to the best of our knowledge, patient societies or support groups for CPAM patients are scarce and widespread. This makes it difficult to establish an unbiased general opinion on the most important outcome parameters for this patient-group. Moreover, gathering a well-balanced and unbiased patient panel could prove to take a considerable amount of time. We would rather put this time to use in the development of clinical trials, since currently no prospective outcome studies have been published yet. Having said this, we intend to incorporate PPI in the development of clinical studies, thus including their essential input in the study design. We believe we have adequately addressed the concerns of our colleagues, and trust we can count on them for future collaboration in the field of congenital lung disease.
We read with interest your protocol to develop a core outcome set (COS) for congenital pulmonary airway malformations and applaud your intention to develop a COS for this population of children. We wish to highlight one area about which we have significant concerns, which is your decision to specifically exclude parents' and patients' views from your Delphi process.
There are strong arguments for involving patients' and parents' views in COS development. Only by specifically including them can researchers be certain that outcomes of particular importance to patients and parents have at least an opportunity of being represented in the COS. The specific exclusion of these important stakeholders risks not only excluding outcomes that are most important to this vital stakeholder group but also risks sending completely the wrong message to patients and parents. By deliberately excluding this group the researchers risk sending the message to patients and parents that their views simply don’t matter.
To ensure that important considerations such as this are not overlooked during COS development, the COS-STAD set of standards has been developed. A specific recommendation is that ‘COS developers should involve those who have experienced or who are affected by the condition (e.g., patients, family members, and carers)’. The authors of this protocol claim to have developed it in accordance with the COS-STAD guidance yet do not appear to incorporated...
To ensure that important considerations such as this are not overlooked during COS development, the COS-STAD set of standards has been developed. A specific recommendation is that ‘COS developers should involve those who have experienced or who are affected by the condition (e.g., patients, family members, and carers)’. The authors of this protocol claim to have developed it in accordance with the COS-STAD guidance yet do not appear to incorporated this particular recommendation within their plans. We note their justification that patients and parents were not included since ‘the final COS is intended specifically intended for patients with asymptomatic CPAM’ yet we wish to register our disagreement with this justification. Arguably patients who are currently asymptomatic are those affected most greatly by outcomes and therefore have the most to offer.
Consequently we urge these researchers to take stock and pause before proceeding with their Delphi process and specifically to amend their proposed protocol to facilitate the inclusion of patients and parents. Failure to do so risks their positive endeavour being futile and misinterpreted.