Core Outcome Measurement Set for Research and Clinical Practice in [Long COVID] in Children and Young People: “PC-COS Children”, 2024, Seylanova et al

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Core Outcome Measurement Set for Research and Clinical Practice in Post COVID-19 Condition Long COVID in Children and Young People: An International Delphi Consensus Study “PC-COS Children”
Nina Seylanova; Anastasia Chernyavskaya; Natalia Degtyareva; Aigun Mursalova; Ali Ajam; Lin Xiao; Khazhar Aktulaeva; Philipp Roshchin; Polina Bobkova; Olalekan Lee Aiyegbusi; Anbarasu Theodore Anbu; Christian Apfelbacher; Ali Akbar Asadi-Pooya; Liat Ashkenazi-Hoffnung; Caroline Brackel; Danilo Buonsenso; Wouter de Groote; Janet V. Diaz; Daniele Dona; Audrey Dunn Galvin; Jon Genuneit; Helen Goss; Sarah E. Hughes; Christina J. Jones; Krutika Kuppalli; Laura A. Malone; Sammie McFarland; Dale M. Needham; Nikita Nekliudov; Timothy R Nicholson; Carlos R. Oliveira; Nicoline Schiess; Terry Y Segal; Louise Sigfrid; Claire Thorne; Susanne Vijverberg; John O. Warner; Wilson Milton Were; Paula R. Williamson; Daniel Munblit; the PC-COS Children Study Group

The COVID-19 pandemic substantially impacted different age groups, with children and young people (CYP) not exempted. Many have experienced enduring health consequences. Presently, there is no consensus on the health outcomes to assess in CYP with post COVID-19 condition. Furthermore, it is unclear which measurement instruments are appropriate for use in research and clinical management of CYP with post-COVID-19.

To address these unmet needs, we conducted a consensus study, aiming to develop a core outcome set (COS) and an associated core outcome measurement set (COMS) for evaluating post-COVID-19 condition in CYP. Our methodology comprised of two phases. In phase 1 (to create a COS), we performed an extensive literature review and categorisation of outcomes, and prioritised those outcomes in a two-round online modified Delphi process followed by a consensus meeting. In phase 2 (to create the COMS), we performed another modified Delphi consensus process to evaluate measurement instruments for previously defined “core outcomes” from phase 1, followed by an online consensus workshop to finalise recommendations regarding the most appropriate instruments for each core outcome. In phase 1, 214 participants from 37 countries participated, with 154 (72%) contributing to both Delphi rounds.

The subsequent online consensus meeting resulted in a final COS which encompassed seven critical outcomes: fatigue; post-exertion symptoms; work/occupational and study changes; as well as functional changes, symptoms, and conditions relating to cardiovascular, neuro-cognitive, gastrointestinal, and physical outcomes. In phase 2, 11 international experts were involved in a modified Delphi process, selecting measurement instruments for a subsequent online consensus workshop where 30 voting participants discussed and independently scored the selected instruments.

As a result of this consensus process, four instruments met a priori consensus criteria for inclusion: “PedsQL multidimensional Fatigue scale” for “fatigue”; “PedsQL Gastrointestinal symptom scales” for “gastrointestinal”; “PedsQL Cognitive Functioning Scale” for “Neuro-cognitive” and “EQ5D family” for “physical functioning”. Despite proposing outcome measurement instruments for the remaining three core outcomes (“cardiovascular”, “post-exertional malaise”, “work/occupational and study changes”), a consensus was not achieved.

Our international, consensus-based initiative presents a robust framework for evaluating post-COVID-19 condition in CYP in research and clinical practice via a rigorously defined COS and associated COMS. It will aid in the uniform measurement and reporting of relevant health outcomes worldwide.


Link | PDF (European Respiratory Journal)

 

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Summary of additional comments from the experts in rounds 1/2: Chalder fatigue questionnaire —

The Chalder Fatigue Questionnaire has elicited varied opinions among experts. Some highlight the presence of unrelated questi ons, notably on memory, while others question its applicability in children, given its predominant use in adult populations. Additionally, it appears to elicit cognitive issues, with responses ranging from appreciation for its succinctness to critique for its potential controversy. The scale's validation in paediatric populations also raises questions

Further, some experts cast doubt on the scale due to controversy surrounding Chalder's work, particularly in relation to cognitive behavioural therapy for Myalgic Encephalomyelitis/Chronic Fatigue Syndrome, which may not adequately capture long COVID population. However, others appreciate its simplicity, shortness, and its established use in conditions like Myalgic Encephalomyelitis/Chronic Fatigue Syndrome. They argue that controversies related to the authors should not discount its use. It is also noted that the scale's age range is 18-65, questioning its content validity and highlighting the lack of validation in paediatric populations.

 

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3.3 Fatigue or exhaustion outcome measures —

The group highlighted the value of questionnaires which integrate self-reports, with the consensus being that children are quite adept at articulating their fatigue symptoms. An essential aspect identified was the inclusion of cognitive components in these measures. Some participants raised concerns about the lack of such components in certain questionnaires, such as PROMIS. Another area of contention, similarly to earlier discussions, was the timeframe with the general feeling that a '7-day' window was not sufficiently representative of the nature of Long COVID fatigue.

The Chalder Fatigue Scale gained some praise for its straightforwardness, but there were reservations regarding its validation. One of the health professionals/researchers expressed a preference for tests validated in multiple languages, emphasising the im portance of accessibility to a wider audience.

An underlying theme was the desire to encompass basic functioning in the measures. Carers voiced their wish for questions that reflect everyday tasks like dressing and showering. Unfortunately, they noted that none of the current questionnaires delve into these nuances.

Concluding the discussions, there seemed to be a collective nod towards the PedsQL Multidimensional Fatigue Scale. Participants appreciated its extensive coverage, the inclusion of self-reports, and its timeframe, which captures a month, thus allowing for the consideration of symptom fluctuation. The overall sentiment was that the PedsQL offered a comprehensive insight into the fatigue experienced by children and young people with post-COVID-19 conditions.

 

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3.4 Post-exertion symptoms outcome measures —

Researchers identified an overlap between post-exertion malaise (PEM) and fatigue, acknowledging the challenge in distinguishing the two due to the limited number of tools that specifically measure PEM. Health professionals expressed concerns about the wording in the CDC's set of questions, deeming it too intricate, which might lead to misinterpretations. Carers voiced their belief in the necessity to adapt and modernise specific questions in the DePaul questionnaire to better represent the current realities, such as the shift to remote learning brought about by the pandemic. They suggested revising the phrase ‘attending school’ to a broader term like ‘Participating in any education.’

An interesting dynamic emerged wherein health professionals initially expressed a positive perspective on PEM items from DePaul Symptom Questionnaire as a measure. However, after hearing some carers articulate their reservations, their stance evolved, leading to a change in opinion.

A common thread of concern from the researchers was the belief that the available questions and their framing may not accurately encapsulate the unique manifestation of PEM in Long COVID and the exacerbation of symptoms that accompany it. In echoing this sentiment, carers resonated with the feeling that the existing instruments fall short of truly portraying their children's experiences and the impact of the condition.

 

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Further, some experts cast doubt on the scale due to controversy surrounding Chalder's work, particularly in relation to cognitive behavioural therapy for Myalgic Encephalomyelitis/Chronic Fatigue Syndrome, which may not adequately capture long COVID population. However, others appreciate its simplicity, shortness, and its established use in conditions like Myalgic Encephalomyelitis/Chronic Fatigue Syndrome

Ugh. Talk about missing the point. The CFQ is her work, and it's terrible, that's why it's controversial. It's not about her, it's that her work is bad, the CFQ is bad and is not applicable here. And it's not established for ME/CFS, but the idea that a terrible questionnaire about fatigue doesn't adequately capture the Long Covid population is the exact same controversy: her stuff is terrible and misleading. If she did one competent thing it would be accepted without reservation, but nothing she ever worked isn't garbage of no interest.

At least some of the later discussion expands on this, but this is really basically a discussion on ME/CFS in all but name. The exact same issues, including the terrible quality of some of the work that has distorted understanding of the illness. And although it's mostly about pediatric patients, the exact same issues remain, maybe with substituting with education vs work, but then again pediatrics is about children so there's still a large % of patients with these issues, so that makes it a really tiny difference of little importance.

Carers voiced their wish for questions that reflect everyday tasks like dressing and showering. Unfortunately, they noted that none of the current questionnaires delve into these nuances.

There is a recent questionnaire built around this. I think about Long Covid. Published not too long ago, no more than a year.

We've been saying this stuff for decades, FFS.