Haighton C

Powell S

Watson RM

Wilson JA

English

Newcastle University E-Prints

C L I N I C A L E X P E R I E N C ECaregiver acceptability of a UK trial for paediatric sleepdisordered breathing: A qualitative interview study1 | INTRODUCTIONPaediatric sleep disordered breathing, a general term for breathingdifficulties during sleep, can range from frequent loud snoring toobstructive sleep apnea (OSA), a condition where part, or all, of the air-way is blocked repeatedly during sleep.1 Paediatric sleep disorderedbreathing is usually caused by large adenoids and tonsils, and the com-monest treatment in the UK is adenotonsillectomy.2 While seriouscomplications from adenotonsillectomy are low, bleeding and return totheatre are relatively common.3 Mortality is very rare, but can occur inup to 1:26683 children.4 Twelve percent of 4–5 year olds snore and upto 2% have OSA in the UK.1 Cross sectional and longitudinal data high-light significant quality of life issues for both children and their fami-lies.5 Remarkably, given the impact of the paediatric sleep disorderedbreathing spectrum on quality of life, and the need to balance benefitsof this treatment with the risks of intervention, worldwide there havebeen only two large scale randomised controlled trials.6,7 However,these were skewed samples confined to polysomnography provenOSA.6,7 There is little evidence about the most effective and safest wayto treat children with sleep disordered breathing8 or whether treatmentis always necessary. We aimed to investigate the acceptability of per-forming a trial for paediatric sleep disordered breathing in the UK interms of whether caregivers would hypothetically participate.2 | MATERIALS AND METHODSApproval was obtained from: South Central—Hampshire B ResearchEthics Committee (ref number 18/SC/0378); Health Research Author-ity (IRAS ID: 239892); Newcastle Upon Tyne Hospitals NHS Founda-tion Trust (R&D number: 8841). This study is reported according tothe consolidated criteria for reporting qualitative research.A clinical secretary screened ENT appointments each month toidentify eligible patients aged 2–9 years with sleep disordered breath-ing symptoms referred to the clinic. Appointment letters included astudy information sheet. At appointment parents of patients con-sented to the collection of a small amount of clinical data about theirchild and to complete validated questionnaires consisting of: T14, a14-item parent-reported outcome questionnaire for paediatric throatdisorders; OSA-18, an 18-item parent-reported evaluation of sleepdisordered breathing; PedsQL, a 28-item assessment of a child's qual-ity of life. Patients with serious comorbidities or existing health condi-tions were excluded. With consent, parent contact details werepassed to the study team who invited them to participate in an in-depth qualitative interview.Parents were purposively sampled to ensure variation in regard togender and age of child as presentation of sleep disordered breathing dif-fers by these characteristics. Gender also influences decisions to partici-pate in trials. GPs who might refer children to ENT and hospital doctorsinvolved in treating children with sleep disordered breathing wereapproached by the study team through known contacts as a conveniencesample. Judgements about sample size and data saturation in thematicanalysis are subjective, and therefore could not be determined (wholly) inadvance of analysis but based on previous research experience it wasestimated that around 20 interviews would provide sufficient data.All participants were sent a letter confirming their interview withan information sheet and a consent form signed prior to the interview.A study identification number was allocated to each participant.Parent participants received a £10 high street shopping voucher tocompensate for expenses. Interviews were carried out either face-to-faceor over the telephone at the convenience of the participant and werenot repeated. Interviews lasted up to 1 h and were semi-structuredbased on flexible topic guides (see Supporting Information). Pilotedtopic guides, based on literature and discussion among the studyteam, explored symptoms, effects, management and treatment ofpaediatric sleep disordered breathing (reported elsewhere9) plusviews about data collection tools and willingness to be randomisedinto a surgical trial (reported here). Interviews, conducted betweenSeptember 2018 and April 2019 by a trained and experienced femalesocial scientist and qualitative researcher unknown to the participants,were audio recorded, transcribed verbatim, anonymised and coded(managed using NVivo). Field notes were not made. Coding was thendiscussed as a team to develop themes. Participants did not providefeedback on transcripts or findings.3 | RESULTSEleven parents (out of 23 approached) participated (three declined, sixunable to be contacted, three lost contact). Five parents wereReceived: 13 March 2023 Accepted: 27 October 2023DOI: 10.1111/coa.14125This is an open access article under the terms of the Creative Commons Attribution License, which permits use, distribution and reproduction in any medium,provided the original work is properly cited.© 2023 The Authors. Clinical Otolaryngology published by John Wiley & Sons Ltd.Clinical Otolaryngology. 2023;1–4. wileyonlinelibrary.com/journal/coa 1interviewed face-to-face and six over the telephone (see Table 1). FiveGPs and six hospital doctors also participated, most were interviewedover the telephone, with two face-to-face interviews (see Table 2).Themes are presented below with illustrative quotations.3.1 | Acceptability of a trial for paediatric sleepdisordered breathingDespite not seeing many children with sleep disordered breathing, allGPs reported willingness to refer eligible children into a surgical trialof sleep disordered breathing:“…if there are trials going on that can improve thehealth and wellbeing of the population, and contributeto new research, I don't have a problem with referringunless I have significant ethical concerns about thestudy, or I think the science is dubious.” (GP1)All hospital doctors were supportive of a trial although they acknowl-edged the inherent difficulties such as cost, parental preference,eligibility criteria, length of follow up, objective outcome measures,seasonable effects and generalisability. However not all hospitaldoctors were in clinical equipoise:“I appreciate that there's no evidence, but we all believe itworks, or we wouldn't be referring children for it.” (HD3)While most parents were willing for their child to be randomised intoa trial there were four parents who would be reluctant due to therisk that it would either delay surgery or that it would force them intosurgery that they did not want:“…just for the sake of getting them [tonsils] out andseeing if it works, I wouldn't do it. I would ratherwait.” (P11)3.2 | Acceptability of T14, OSA-18 and PedsQLAlmost all parents reported the T14, OSA-18 and PedsQL as accept-able. Most parents reported the questions easy to understand,straightforward and brief to complete while they waited for theirappointment:“The questions were all-I liked the way they were allworded and set out. Very clear to understand them. Ithink there is nothing worse than having a questionand reading it and thinking, “Well I'm not sure. Whatdoes this mean, or do you mean this,” but no I found itall very clear. Very understandable” (P1)In contrast, one parent did report that the questionnaires were drawnout and difficult to complete but this may have been because theycompleted the questionnaires after their appointment and reportedfinding it difficult to keep their children entertained while they filledthem in. Some parents commented on how relevant the questionswere “because I was going through it and it was so raw when doing theTABLE 1 Parent participants.Number ID code Parent Child gender Child age (years)1 P1 Mother Male 92 P3 Father Male 53 P4 Mother Male 34 P6 Mother Female 45 P8 Mother Female 76 P9 Mother Male 97 P10 Mother Female 48 P11 Mother Female 99 P12 Mother Female 410 P13 Mother Male 811 P15 Mother Female 6Key points1. There is no conclusive evidence about the safest andmost effective way to treat children with sleep disor-dered breathing.2. Some parents are reluctant for their child to be random-ised into a trial where surgery could be delayed.3. General Practitioners are willing to refer children to atrial for paediatric sleep disordered breathing.4. Hospital doctors may not be in equipoise regarding themost effective way to treat children with sleep disor-dered breathing.5. A partially randomised patient preference trial may bethe best way to establish the most effective way to treatchildren with sleep disordered breathing.TABLE 2 Health professional participants.Number ID code Role Gender1 GP1 GP, ad-hoc primary care GP sessions Male2 GP2 Senior partner in GP practice Female3 GP3 Locum GP Female4 GP4 Practice GP Female5 GP5 Practice GP Male1 HD1 General and respiratory paediatrician Female2 HD2 Consultant ENT surgeon Male3 HD3 Respiratory paediatrician Female4 HD4 Consultant ENT surgeon Male5 HD5 Consultant in respiratory paediatrics Male6 HD6 Consultant ENT surgeon Male2 HAIGHTON ET AL. 17494486, 0, Downloaded from https://onlinelibrary.wiley.com/doi/10.1111/coa.14125 by Newcastle University, Wiley Online Library on [17/01/2024]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons Licensequestionnaire” (P8) while others found the questionnaires highlighteda much wider spectrum of symptoms than some parents had initiallyassociated with sleep disordered breathing:“Really, really useful, to be honest…because I had noidea all of those things were linked to just havingmassive tonsils…lack of sleep, lack of concentration,mood swings, poor appetite.” (P12)One parent reported confusion over how certain questions wererelevant to sleep disordered breathing in particular questions aboutfeeling afraid and those relating to social functioning.3.3 | Suggested outcomes measure for a trial forpaediatric sleep disordered breathingGPs found it difficult to suggest what outcomes measures might bemost appropriate for a surgical trial for sleep disordered breathing butsuggested a variety including sleep scores, academic performance,cardiac pressures, qualitative parental views of child's sleep, snoringvolume, parental stress, and child daytime tiredness. GPs wereconcerned with identifying an objective outcome measure as parentswould not be blind to their child's intervention:“But that could be skewed…by bias in terms of theparents obviously would know if their child had anoperation or not.” (GP4)Hospital doctors suggested sleep studies as the primary objectiveoutcome measure which could be potentially used in a trial but alsomentioned academic performance, growth, or a snoring score. Theyalso suggested other more subjective outcome measures such asquality of sleep, quality of life, parental views/satisfaction, self-reportedchest infections/cough. Parents reported preferring qualitative methodsof data collection to quantitative:“Yes. I mean, you can fill in a form, that's fine, butsometimes…it's not yes or no. It's this grey area in themiddle that might not suit everybody. So, it is nice tobe able to sit and talk to somebody face-to-face” (P1)4 | DISCUSSIONWhile most parents were willing for their child to be randomised, someparents would be reluctant for their child to be randomised into a trialof sleep disordered breathing. GPs were willing to refer children intosuch a trial however hospital doctors may not be in equipoise. Random-ised controlled trials are the gold standard to provide unbiased datahowever when patients have a treatment preference, randomisationmay influence participation and outcomes. A recent meta-analysisassessed the influence of patients' preference in randomised controlledtrials by analysing partially randomised patient preference trials; a ran-domised controlled trial and preference cohort combined.10 Findingsrevealed that patients' preference led to a substantial proportion of aspecific patient group refusing randomisation, while it did not influencethe primary outcome within a partially randomised patient preferencetrial. Therefore, partially randomised patient preference trials couldincrease external validity without compromising the internal validitycompared with randomised controlled trials.10 While there is still a needto establish the most effective way to treat paediatric sleep disorderedbreathing8 a partially randomised patient preference trial may be themost suitable way forward. However, this study was carried out in onlyone geographical location and the sample of parents included only onefather. Further research is needed within other regions and with agreater variation in sample.AUTHOR CONTRIBUTIONSCatherine Haighton made substantial contributions to the design ofthe study, analysis and interpretation of the data. Rose Mary Watsonmade substantial contributions to the acquisition, analysis and inter-pretation of the data. Janet A. Wilson made substantial contributionsto the design of the study and interpretation of the data. StevenPowell made substantial contributions to the conception and designof the study and interpretation of the data. All authors contributedto drafting the work, revising it critically for important intellectualcontent and approved the final version. All authors agree to beaccountable for all aspects of the work in ensuring that questionsrelated to the accuracy or integrity of any part of the work areappropriately investigated and resolved.ACKNOWLEDGEMENTSWe thank all the parents, hospital doctors and general practitionerswho participated in this research.FUNDING INFORMATIONThis work was supported by the Newcastle upon Tyne Hospitals NHSCharity grant number (NFH/3078).CONFLICT OF INTEREST STATEMENTJanet A. Wilson was previously, and Steven Powell is currentlyemployed by Newcastle upon Tyne Hospitals NHS Foundation Trust.PEER REVIEWThe peer review history for this article is available at https://www.webofscience.com/api/gateway/wos/peer-review/10.1111/coa.14125.DATA AVAILABILITY STATEMENTThe data that support the findings of this study are available onrequest from the corresponding author. The data are not publiclyavailable due to privacy or ethical restrictions.ETHICS STATEMENTParticipants provided written informed consent to participate in thisstudy. In line with the terms of consent to which participants agreed,HAIGHTON ET AL. 3 17494486, 0, Downloaded from https://onlinelibrary.wiley.com/doi/10.1111/coa.14125 by Newcastle University, Wiley Online Library on [17/01/2024]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons Licensethe data are not publicly available. There are ethical restrictions onsharing the de-identified data set. The data contain potentially identi-fying and sensitive participant information and we do not have partici-pant consent to share this dataset. Data requests may be sent toSouth Central—Hampshire B Research Ethics Committee (ref number18/SC/0378).Catherine Haighton1Rose Mary Watson1Janet A. Wilson2Steven Powell2,31Faculty of Health and Life Sciences, Northumbria University, Newcastleupon Tyne, UK2Population Health Sciences Institute, Newcastle University, Newcastleupon Tyne, UK3The Department of Otolaryngology (Ear, Nose and Throat) Newcastleupon Tyne Hospitals NHS Foundation Trust, Newcastle upon Tyne, UKCorrespondenceCatherine Haighton, Northumbria University, B125, Coach LaneCampus West, Newcastle upon Tyne NE7 7XA, UK.Email: katie.haighton@northumbria.ac.ukORCIDCatherine Haighton https://orcid.org/0000-0002-8061-0428Rose Mary Watson https://orcid.org/0000-0003-4549-3462Janet A. Wilson https://orcid.org/0000-0002-6416-5870Steven Powell https://orcid.org/0000-0001-7181-4608REFERENCES1. Bonuck KA, Chervin RD, Cole TJ, Emond A, Henderson J, Xu L, et al.Prevalence and persistence of sleep disordered breathing symptomsin young children: a 6-year population-based cohort study. Sleep.2011;34:875–84.2. NHS Digital. Hospital episode statistics. Leeds, UK: NHS Digital; 2016.3. Connolly H, Tomaselli L, McKenna BM. Adenotonsillectomy for pedi-atric obstructive sleep apnea: how to predict those at risk for postop-erative complications. J Clin Sleep Med. 2020;16:3–4.4. Keltie K, Donne A, Daniel M, Stephenson K, Wyatt M, Kuo M, et al.Paediatric tonsillectomy in England: a cohort study of clinical practiceand outcomes using Hospital Episode Statistics data (2008-2019).Clin Otolaryngol. 2021;46:552–61.5. Torretta S, Rosazza C, Pace ME, Iofrida E, Marchisio P. Impact of ade-notonsillectomy on pediatric quality of life: review of the literature.Ital J Pediatr. 2017;43:1–8.6. Marcus CL, Moore RH, Rosen CL, Giordani B, Garetz SL, Taylor HG,et al. A randomized trial of adenotonsillectomy for childhood sleepapnea. N Engl J Med. 2013;368:2366–76.7. Waters K, Chawla J, Harris MA, Heussler H, Cheng A, Black R. ThePOSTA study: 24-month follow-up of IQ after randomised adenoton-sillectomy for mild OSA in preschoolers. J Sleep Res. 2019;28:35.8. Venekamp RP, Hearne BJ, Chandrasekharan D, Blackshaw H, Lim J,Schilder AGM, et al. Tonsillectomy or adenotonsillectomy versus non-surgical management for obstructive sleep-disordered breathing inchildren. Cochrane Database Syst Rev. 2015;10:1–44.9. Haighton C, Watson RM, Wilson JA, Powell S. Perspectives on paedi-atric sleep disordered breathing in the UK: a qualitative study.J Laryngol Otol. 2022;136:520–6.10. Wasmann K, Wijsman P, van Dieren S, Bemelman W, Buskens C. Par-tially randomised patient preference trials as an alternative design torandomised controlled trials: systematic review and meta-analyses.BMJ Open. 2019;9:1–12.SUPPORTING INFORMATIONAdditional supporting information can be found online in the Support-ing Information section at the end of this article.How to cite this article: Haighton C, Watson RM, Wilson JA,Powell S. Caregiver acceptability of a UK trial for paediatricsleep disordered breathing: A qualitative interview study.Clinical Otolaryngology. 2023. https://doi.org/10.1111/coa.141254 HAIGHTON ET AL. 17494486, 0, Downloaded from https://onlinelibrary.wiley.com/doi/10.1111/coa.14125 by Newcastle University, Wiley Online Library on [17/01/2024]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License

Caregiver acceptability of a UK trial for paediatric sleep disordered breathing: A qualitative interview study

https://eprints.ncl.ac.uk/file_store/production/295858/AF43D6A3-ECCB-4BA3-B137-10F3D15AF5DA.pdf

Caregiver acceptability of a UK trial for paediatric sleep disordered breathing: A qualitative interview study

Abstract

Similar works

Full text

Available Versions

Newcastle University E-Prints