The Impact of the Preferred Reporting Items for Case Reports in Endodontics (PRICE) 2020 Guidelines on the Reporting of Endodontic Case Reports

Aim The aim of this study is to evaluate the impact of the Preferred Reporting Items for Case reports in Endodontics (PRICE) 2020 guideline on the reporting of published endodontic case reports (CRs). Methodology All case reports published in the International Endodontic Journal, European Endodontic Journal, Journal of Endodontics and Restorative Dentistry and Endodontics, in the year before and after the release of PRICE 2020, were included for analysis. Two panels comprising dentists scored case reports against a scoring system adapted from the guideline. Individual items were scored up to a maximum of 1; scores were then summated to provide an overall maximum of 47 for each CR. Each report provided an overall percentage adherence, and panel agreement was calculated using the intraclass correlation coefficient (ICC). Disagreement on scoring was discussed until a consensus was reached. Scores before and after PRICE guideline publication were compared using an unpaired two-tailed t test. Results A total 19 CRs were identified in both the pre- and post-PRICE guideline publication. Mean adherence to PRICE 2020 increased by 7.9% (p=0.003) from 70.0%±8.89 to 77.9%±6.23 following its publication. Agreement between panels was moderate (ICC pre-PRICE: 0.673 {p=0.011}; ICC post-PRICE: 0.742 {p=0.003}). Items 1a, 6c, 6e, 6f, 6g, 6j, 6q, 6s, 7a, 9a, 11a, 12c and 12d experienced a fall in compliance. Conclusion The PRICE 2020 guideline has resulted in a modest improvement in the reporting of endodontic case reports. Greater awareness and a wider acceptance and implementation of the guideline in endodontic journals are needed to improve adherence to the novel guideline.


Introduction
Case reports (CR) provide detailed accounts of signs, symptoms, diagnosis and management of rare presentations or novel management techniques in order to share different aspects of clinical practice of interest [1]. The current classification of CRs is level 4 (poorest level) as per the Oxford Centre for Evidence-Based Medicine: Levels of Evidence [2]. Although CRs are graded as one of the lowest levels of evidence, they provide value in the rapid communication of rare incidents, conditions and associations, which may inform further higher-quality research. The lack of consistency, transparency, information and coherency in the reporting of CRs can result in poor educational value and weaker evidence. As a result, case reporting recommendations were introduced in 2013 in the form of CAse REport (CARE) guidelines, providing a 13item (30 individual points) detailed checklist for authors to follow [3]. In recent years, specialties have adapted CARE guidelines using Delphi consensus methodology to provide tailored frameworks for respective practices [4,5]. Endodontic case reporting practice is currently lacking; unpublished data by Dummer, reported by Nagendrababu et al., elucidates that a significant portion of CRs is rejected due to 'incomplete' and 'inaccurate' reporting. The Preferred Reporting Items for Case reports in Endodontics (PRICE) 2020 were therefore created with the intention of improving writing practices and enhancing the educational value of CRs [6]. PRICE introduced a comprehensive 47 individual-item checklist divided into 12 sections that are far more detailed than most case reporting guidelines (CARE: 30 individual items [3]; SCARE: 38 individual items [4]). Items were first developed in draft format, by integrating and adapting fundamental points from CARE [3] and the Clinical and Laboratory Images in Publications (CLIP) principles [7]. CLIP was introduced in 2012 in a concerted effort to promote the publication of clearer and more informative images, such as histopathology and radiography. PRICE was introduced to provide guidance for reporting singular case reports with no mention of case series articles. Draft PRICE items were reviewed via an online Delphi survey, where a group, comprising academics, endodontists, general dentists and patient representatives, scored items for inclusion. The findings from the Delphi survey and the revised PRICE items were thereafter discussed at an international endodontic meeting (19th European Society of Endodontology Biennial Congress), where the guideline was finalised. The combined emphasis on the reporting of both cases (CARE) and images (CLIP) makes PRICE the first of its kind, as previous recommendations have placed little emphasis on the reporting standards of images. PRICE also introduces a comprehensive flowchart for authors, summarising the process of writing a thorough and detailed case report [6].
Contemporaneous evaluations of endodontic case reporting practice have exemplified the need for PRICE [8]. Reporting guidelines are particularly necessary for the purposes of promoting clearer, more informative and uniform writing practice. A significant limitation to achieving such outcomes remains the authors' awareness, understanding and interpretation. The evaluations of both SCARE, guideline for the reporting of surgical case reports, and Preferred Reporting Of CasE Series in Surgery (PROCESS), guideline for the reporting of surgical case series, elucidated improvements in reporting practices [4,5,9]. Given the index of potential guidelines that possess influencing positive change, consistent long-term auditing of reporting practice is warranted. The present study therefore aimed to evaluate the early impact of the PRICE guidelines on the reporting of endodontic case reports in four reputable endodontic scientific journals.

Study design
A before-and-after study was conducted, evaluating adherence to endodontic CRs published prior to and following the publication of PRICE guidelines (23 February 2020) [6]. Two panels, comprising three dentists with interests in endodontics, independently scored CRs against the PRICE scoring system (Table 1), based on the detailed exposition and elaboration of items present in PRICE [10]. Each individual item was allocated a maximum score of 1 (e.g. item 6a=maximum of one and item 6c=maximum of one). Two independent panels were used to strengthen the reliability of CR scores. Each panel of three scored all the CRs as a group; a disagreement on finalised item scoring between panels was settled by discussion between both panels until a consensus was reached. Ethical approval was not required for this before-and-after study of published case reports; individual patient consent was obtained by the authors of respective CRs prior to publication. The strengths of the case report and its importance must be discussed with reference to the relevant literature 1/2 for the strengths of the report discussed with reference to the literature; 1/2 for the importance of the report discussed with reference to the literature; 0 for not mentioned Item 7c

Items PRICE Guideline Consensus Criteria Review Scoring Criteria
The limitations of the case report must be discussed 1 for the limitations of the report discussed; 0 for not mentioned Item 7d The rationale for the conclusion(s) must be discussed 1 for the rationale of the conclusion discussed; 0 for Item 12a The details of the equipment, software and settings used to acquire the image(s) must be described in the text or legend Markers/labels must be used to identify the key information in the image(s) and be defined in the legend or as a footnote 1/2 for markers to identify key information in the image; 1/2 for the marker defined in the legend; 0 for no markers

Item 12i
The legend of each image must include an explanation whether it is pre-treatment, intra-treatment or post-treatment and, if relevant, how images over time were standardised 1/2 for a statement of whether the image was pretreatment, intra-treatment or post-treatment; 1/2 for a mention in the legend of how images were standardised over time; 0 for not mentioned

Data analysis
Statistical analyses were performed using the Statistical Package for Social Sciences (SPSS) version 27.0 (IBM SPSS Statistics, Armonk, NY). Individual-item scores were collated to form a final overall score out of 47 for each CR; scores were then converted to percentages, by dividing CR score over maximal applicable scoring (items deemed not applicable were subtracted from the denominator) [5]. Intraclass correlation coefficient (ICC) was used to measure the agreement between final percentage panel ratings. Data was analysed for normality using the Shapiro-Wilk test, and parametric variables from before and after periods were compared using an unpaired two-tailed t test. A p-value of <0.05 was considered significant.

Results
A total of 19 CRs were identified in both the pre-and post-PRICE periods. The mean score for adherence to the PRICE guideline increased by 7.9% (t=-3.141, degrees of freedom {df}=36 and p=0.003) from 70.0% (standard deviation: ±8.89) in the pre-PRICE period to 77.9% (standard deviation: ±6.23) in the post-PRICE period ( Table 2). ICC between panels for pre-PRICE was 0.673 (p=0.011) and post-PRICE was 0.742 (p=0.003), indicating moderate agreement of scoring.   Figure 1 depicts the percentage adherence of pre-PRICE and post-PRICE period endodontic case reports. The five items with the greatest percentage increase in guideline adherence are outlined in Table 3; the five items with the greatest percentage decrease in guideline adherence are outlined in Table 4.  Quality of images: the details of the equipment, software and settings used to acquire the image(s) must be described in the text or legend +160.0

Item 6r
Case report information: the assessment method(s) used to determine the clinicianassessed and patient-assessed treatment outcomes and their results must be provided +66.7

Item 4a
Introduction: a background summary of the case(s) with relevant information must be provided +58.3

Item 5a
Informed consent: a clear statement that informed, valid consent was obtained from the patient(s) must be provided +42.9

Discussion
Reporting criteria are an important intervention to improve consistency and clarity in the narration of case reports, as has previously been demonstrated with case reporting (CARE [3]) and case series reporting (SCARE [4]) guidelines. This investigation identified a 7.9% overall improvement in adherence to the PRICE guideline following their publication in 2020, elucidating a promising early impact of the guidelines.
Although there was an improvement in adherence for the majority of reporting items, few items experienced a fall in compliance (13 items: 1a, 6c, 6e, 6f, 6g, 6j, 6q, 6s, 7a, 9a, 11a, 12c and 12d; Table 1). Most notable of such items is 'case report information' (item 6); a thorough, detailed case presentation is an essential component of the case report, and current evidence suggests that the endodontic community is lacking in this domain. Berlin-Broner and Levin reported similar findings in a large five-year evaluation of case reporting adherence to PRICE, identifying lowest scoring items as 6c, 6g, 6h, 6i, 8a, 12c and 12d [8]. Such findings heed the endodontic community not only to read PRICE prior to and during the writing of manuscripts but also to use it as a final screening checklist prior to submission.
PRICE is a substantial and comprehensive guideline with 47 reporting items, far greater than any other case reporting guideline currently available (CARE: 30 individual items [3]; SCARE: 38 individual items [4]). An agreement between the two independent panels of dentists was moderate for both pre-PRICE (ICC: 0.673; p=0.011) and post-PRICE (ICC: 0.742; p=0.003) periods, indicating that there is a small inter-rater variation in the interpretation of the guideline, particularly in elucidating the non-applicability (NA) of reporting items. Further clarity may be required to settle the potential differing interpretations of this aspect of the guideline.
An international, inter-journal, collaborative effort is needed to enhance awareness and adherence to PRICE in order to improve the universal reporting of endodontic case reports. This could be achieved by implementing the guideline as a primary screening tool at journal submission for authors, reviewers and editors. Currently, the International Endodontic Journal is the only endodontic journal that has updated its author guidelines to incorporate PRICE as a submission guide. Greater exposure to PRICE is needed within the endodontic community to establish comprehensive, transparent and educative case reporting practices. Therefore, it is strongly encouraged for authors to read Nagendrababu et al.'s explanation of the PRICE case reporting items to improve writing practice [10]. PRICE checklists and flowcharts can be downloaded by authors from the 'The Preferred Reporting Items for study Designs in Endodontology' (PRIDE) website [49]. Further studies are required to assess adherence following a wider acceptance and implementation of the PRICE guideline by endodontic journals internationally.

Limitations
There are some limitations to this study that must be considered. A short sample period was used, comparing the adherence of CRs in the years directly before and after the guideline was released. As such, a significant portion of the endodontic community may still be unaware of PRICE. Furthermore, the panels were not blinded to whether the papers were published prior to or following PRICE. CRs can be defined as articles describing up to four cases [1], but the present study only included CRs describing a maximum of one case to provide greater consistency in adherence grading between panels. There is also some variation in the proportion of journals in which reviewed articles were published in the before-and-after samples; this may affect the validity of the results as journals may have different processing guidelines. There is potential that reporting practices of articles describing multiple cases differ to those only reporting one case; further research is warranted to discern the reporting practices of such CRs and by extension endodontic case series.

Conclusions
Early indications demonstrate that the PRICE 2020 guideline has resulted in a modest improvement in the reporting of endodontic case reports. Greater exposure and a wider acceptance and implementation of the guideline in endodontic journals are needed to improve adherence to the novel guideline. Further studies are required in the future to assess PRICE 2020 adherence.

Additional Information Disclosures
Human subjects: All authors have confirmed that this study did not involve human participants or tissue. Animal subjects: All authors have confirmed that this study did not involve animal subjects or tissue.

Conflicts of interest:
In compliance with the ICMJE uniform disclosure form, all authors declare the following: Payment/services info: All authors have declared that no financial support was received from any organization for the submitted work. Financial relationships: All authors have declared that they have no financial relationships at present or within the previous three years with any organizations that might have an interest in the submitted work. Other relationships: All authors have declared that there are no other relationships or activities that could appear to have influenced the submitted work.