Development of a Core Outcome Set for Amblyopia
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Jabri et al. BMC Ophthalmology (2019) 19:47 https://doi.org/10.1186/s12886-019-1055-8 RESEARCHARTICLE Open Access Development of a core outcome set for amblyopia, strabismus and ocular motility disorders: a review to identify outcome measures Samia Al Jabri1, Jamie Kirkham2 and Fiona J. Rowe1* Abstract Background: Core Outcome Sets (COS) are defined as the minimum sets of outcomes that should be measured and reported in all randomised controlled trials to facilitate combination and comparability of research. The aim of this review is to produce an item bank of previously reported outcome measures from published studies in amblyopia, strabismus and ocular motility disorders to initiate the development of COS. Methods: A review was conducted to identify articles reporting outcome measures for amblyopia, strabismus and ocular motility disorders. Using systematic methods according to the COMET handbook we searched key electronic bibliographic databases from 1st January 2011 to 27th September 2016 using MESH terms and alternatives indicating the different subtypes of amblyopia, strabismus and ocular motility disorders in relation to treatment outcomes and all synonyms. We included Cochrane reviews, other systematic reviews, controlled trials, non- systematic reviews and retrospective studies. Data was extracted to tabulate demographics of included studies, primary and secondary outcomes, methods of measurement and their time points. Results: A total of 142 studies were included; 42 in amblyopia, 33 in strabismus, and 68 in ocular motility disorders (one study overlap between amblyopia and strabismus). We identified ten main outcome measure domains for amblyopia, 14 for strabismus, and ten common “visual or motility” outcome measure domains for ocular motility disorders. Within the domains, we found variable nomenclature being used and diversity in methods and timings of measurements. Conclusion: This review highlights discrepancies in outcome measure reporting within published literature for amblyopia, strabismus and ocular motility and it generated an item bank of the most commonly used and reported outcome measures for each of the three conditions from recent literature to start the process of COS development. Consensus among all stakeholders including patients and professionals is recommended to establish a useful COS. Keywords: Amblyopia, Strabismus, Ocular motility disorders, Outcome measures, Core outcome set * Correspondence: [email protected] 1Department of Health Services Research, University of Liverpool, Waterhouse Building Block B, 2nd Floor, 1-3 Brownlow Street, L69 3GL Liverpool, UK Full list of author information is available at the end of the article © The Author(s). 2019, corrected publication March/2019. Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. Jabri et al. BMC Ophthalmology (2019) 19:47 Page 2 of 22 Background Development of a COS involves a number of stages Amblyopia, strabismus and ocular motility disorders that commence with a systematic review of the literature occur in about 10% of the general population (amblyopia to identify existing knowledge about outcome measures 2–5%, strabismus 4%) [1]. They often present as child- [7]. This is then followed by qualitative studies, Delphi hood conditions and can constitute long-term problems surveys to consult widely on outcome measures and fi- for children and young adults. Strabismus and ocular nally, consensus meetings to discuss and agree on the motility disorders can also develop as acquired condi- COS [7]. This paper reports the first stage – the litera- tions due to neurological, endocrine and traumatic ture review to identify the reported range of outcome causes. There are several approaches to the management measures in the published literature for amblyopia, stra- of these conditions including occlusion, penalisation, bismus and ocular motility disorders. spectacles, prisms, drugs, surgery, botulinum toxin, exer- cises, watchful waiting, or a combination of two or more Objectives of the above [2]. The effects from these treatments such The primary aim of this review is to generate an item as improvements in symptoms or side effects are bank of relevant outcome measures previously reported assessed by outcome measures and are usually used to by researchers and clinicians in studies of treatment of formally evaluate management options in clinical stud- conditions under evaluation. The review aims also to de- ies. However varied outcome measures and several end- termine the variation in measuring methods used and – points are often used [3 5]. This lack of standardisation timings of assessments. makes it difficult to compare the conclusions of these The secondary objectives of this review are to investi- studies and, as a result, renders it challenging to discuss gate sources of variability of outcome measure defini- realistically the likely outcomes of treatment with pa- tions including different age groups, study designs, types tients in the clinic [6]. of amblyopia (e.g. refractive, strabismic, stimulus One strategy suggested to overcome the issues result- deprivation), types of strabismus (e.g. exotropia, esotro- ing from variable outcome measures is the development pia), and types of ocular motility disorder (e.g. accom- of Core Outcome Sets (COS). This is defined as the modation and convergence disorders, mechanical minimum set of outcomes that should be measured and restrictions, myogenic, neurogenic, nystagmus, patterns reported in all randomised controlled trials [7]. The deviation and gaze palsy). COS will make it easier for the results of trials to be compared, contrasted and combined, lead to research that is more likely to have measured relevant outcomes Methods due to involvement of relevant stakeholders, and en- A protocol for the development of this COS project was – hance the value of evidence synthesis by ensuring that written by a steering committee a team of stakeholders all trials contribute usable information [7]. Therefore, it including COS developers, ophthalmologists, orthoptists is postulated that the use of COS would increase the po- and journal editors. The review protocol was registered tential in carrying out future meta-analysis for target in the COMET initiative website (http://www.comet-ini- conditions. tiative.org/studies/details/900?result=true) and published The numerous and diverse outcome measures that as open access (http://pcwww.liv.ac.uk/~rowef/index_- may be used for amblyopia, strabismus and ocular motil- files/Page356.htm). The review, using systematic rigor- ity disorders include, amongst others, visual acuity, angle ous methods, was conducted in accordance with the of deviation, range of ocular movements, fixation stabil- guidelines from the COMET handbook [7]. A PRISMA ity and binocular vision measurements. There are a checklist [10] has been completed for the systematic re- number of Cochrane systematic reviews that consider a view and can be found in Additional file 1 : Table S1. range of treatment trials for amblyopia, strabismus and ocular motility disorders. Their recommendations call Eligibility criteria for clarification of dose/response effect and further in- Age vestigation of treatment regimens [2–4]. An attempt to Subjects of all ages with target conditions were included. utilise a COS is evident for the National Strabismus Data Target conditions: Set project [8]. A recent review recommended four out- comes for reporting results of surgery for intermittent 1. Amblyopia (unilateral, bilateral) of any type or exotropia [5] but was limited by the extent of literature severity (refractive, meridional, ametropic, review and lack of external consensus. A short narrative strabismic or stimulus deprivation). review of outcome measurements for size of deviation 2. Strabismus (latent, manifest, constant, intermittent, showed considerable variability across the tests available micro) of any type and severity (eso, exo, hyper, and the recommendations for their use [9]. hypo, cyclo deviation) Jabri et al. BMC Ophthalmology (2019) 19:47 Page 3 of 22 3. Ocular motility disorders (OMDs) of any type and September 2016. This period was selected given the con- severity (nystagmus, horizontal/vertical gaze palsy, siderable increase in studies, trials and reviews in recent cranial nerve palsy, convergence/divergence years and to extract treatment outcome measures that disorder, patterns of horizontal incomitance, are relevant to recent research and clinical practice. As mechanical restrictions, myogenic disorders like per COMET handbook guidance [7] we recognised that thyroid eye disease and myasthenia with ocular overly large reviews would be resource intensive and involvement). might not yield important additional outcomes. We did not search for unpublished studies or in clin- We included all three target conditions in recognition ical trials registries and we did not hand-search any add- of the considerable overlap between them, for example itional resources. We performed citation