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Management of the normal-appearing appendix during laparoscopy for clinically suspected acute appendicitis in the pediatric population
Journal Pre-proof Management of the Normal-Appearing Appendix during Laparoscopy for clinically suspected acute appendicitis in the pediatric population
Kathleen Logie, Tessa Robinson, Lisa VanHouwelingen PII:
S0022-3468(20)30078-6
DOI:
https://doi.org/10.1016/j.jpedsurg.2020.01.039
Reference:
YJPSU 59583
To appear in:
Journal of Pediatric Surgery
Received date:
21 January 2020
Accepted date:
25 January 2020
Please cite this article as: K. Logie, T. Robinson and L. VanHouwelingen, Management of the Normal-Appearing Appendix during Laparoscopy for clinically suspected acute appendicitis in the pediatric population, Journal of Pediatric Surgery(2020), https://doi.org/10.1016/j.jpedsurg.2020.01.039
This is a PDF file of an article that has undergone enhancements after acceptance, such as the addition of a cover page and metadata, and formatting for readability, but it is not yet the definitive version of record. This version will undergo additional copyediting, typesetting and review before it is published in its final form, but we are providing this version to give early visibility of the article. Please note that, during the production process, errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
© 2020 Published by Elsevier.
Journal Pre-proof Management of the Normal-Appearing Appendix During Laparoscopy for Clinically Suspected Acute Appendicitis in the Pediatric Population Kathleen Logiea, Tessa Robinsonb,c, Lisa VanHouwelingenb,c,d* a
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Division of General Surgery, Department of Surgery, McMaster University, Hamilton, Ontario, Canada b Division of Pediatric Surgery, Department of Surgery, McMaster University, Hamilton, Ontario, Canada c McMaster Pediatric Surgery Research Collaborative, McMaster University, Hamilton, Ontario, Canada d McMaster Children’s Hospital, Hamilton, Ontario, Canada
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Corresponding Author: Lisa VanHouwelingen McMaster Children’s Hospital Room 4E4 1200 Main Street West Hamilton, Ontario, Canada E-Mail: [email protected]
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COMPETING INTERESTS: The authors have no conflicts of interest to disclose.
Journal Pre-proof ABSTRACT Purpose: The widespread use of laparoscopy has brought forth the question of how to manage a macroscopically normal-appearing appendix in cases of clinically suspected appendicitis. This study aimed to determine the current practices of pediatric general surgeons in Canada regarding this matter. Methods: An online survey was created following the American Pediatric Surgical Association (APSA) guidelines and distributed via email to the Canadian Association of Pediatric Surgeons (CAPS) staff surgeons. The questions assessed clinician characteristics, standard practice, and rationale. Results were analyzed using descriptive statistics.
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Results: A total of 54/72 (75%) CAPS members practicing in Canada completed the survey. All (100%) agreed they would remove a normal-appearing appendix during laparoscopy for suspected acute appendicitis. The most common reasons were: possibility of microscopic appendicitis (39/54, 72.2%), avoiding future diagnostic confusion (28/54, 51.9%), and patient preference/consent discussion (21/54, 38.9%). Most (53/54, 98.1%) had performed a negative appendectomy and 49/54 (90.7%) agreed there were no sufficient guidelines.
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Conclusions: The majority of pediatric surgeons agree sufficient guidelines do not exist to support decision making when a normal-appearing appendix is found during laparoscopy for suspected acute appendicitis. This survey shows that removal of the appendix in this case would be supported by the majority of Canadian pediatric surgeons.
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KEYWORDS: Negative appendectomy; Appendicitis; Laparoscopic appendectomy; Pediatric
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TYPE OF STUDY: Survey
LEVEL OF EVIDENCE: VII (Expert Opinion)
ABBREVIATIONS: Negative Appendectomy (NA) Negative Appendectomy Rate (NAR) Canadian Association of Pediatric Surgeons (CAPS) American Pediatric Surgical Association (APSA) Diagnostic Laparoscopy (DL) Laparoscopic Appendectomy (LA)
Journal Pre-proof 1. INTRODUCTION Acute appendicitis represents the most common indication for abdominal surgery in the pediatric population [1]. Prior to the advent of laparoscopic surgery, most surgeons would remove the appendix regardless of intraoperative appearance due to the position of the scar raising concern for future diagnostic uncertainty. Thus, it was not uncommon for a histologically normal appendix to be removed, referred to as a negative appendectomy (NA). The NA rate (NAR) ranges in the literature from 1.8 up to 46% and trends higher in the pediatric population [2–12]. Although often considered relatively benign, NA has a 6% rate of morbidity [13].
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In the era of laparoscopy, the main argument supporting removal of a normal-appearing appendix is the high false negative rate of intraoperative diagnoses. Published data shows this to
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be as high as 76%, prompting the majority of authors to recommend appendectomy regardless of
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appearance [6,14–20]. However, when others have investigated the outcomes of leaving a normal-appearing appendix in situ, their rates of re-operation and were relatively low, ranging
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from 0-2.1% in available studies [7,21–28]. As a result of the confusion in the literature, current adult guidelines state that in the absence of other pathology seen during laparoscopy, the
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decision to remove the appendix should be based on the individual clinical scenario (level III, grade A) [31]. Despite this, two large surveys of adult surgeons have shown that 64-73% opt to
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remove all macroscopically normal-appearing appendixes as a practice. The most common reasons for removal were lack of clear guidelines and/or evidence on the topic, and the
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possibility of microscopic appendicitis [32,33].
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The pediatric population has a high incidence of appendicitis and negative appendectomy, with simultaneously low numbers of comorbidities to confound the data. Furthermore, to our knowledge, there remains no literature regarding surgeon decision making when faced with a macroscopically normal appendix during laparoscopy for clinically suspected acute appendicitis in pediatric centers. The purpose of this study was therefore to determine the current clinical practices and beliefs of pediatric general surgeons practicing in Canada regarding this matter. 2. METHODS A descriptive survey was created following the American Pediatric Surgical Association (APSA) guidelines for surveys [34]. Question formulation was performed by a specialty committee and informed by both a detailed literature search as well as feedback gained from the
Journal Pre-proof surgeons at the study center. The proposed survey was piloted to the same surgeons to test validity. The survey was then evaluated by the Canadian Association of Pediatric Surgeons (CAPS) research committee and further adjustments were made. Once finalized, the survey (Appendix 1) was distributed via email to all surgeon members of CAPS. The survey was distributed and all data collected in 2019. All Canadian surgeon members of CAPS were included; with all other non-staff level members or other personnel being excluded. Two additional reminders were sent at two-week intervals. No formal incentives were offered for completion.
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The data were collected using a Research Electronic Data Capture (REDCap) online
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database [35]. Data from staff surgeons practicing in Canada were analyzed. Participant demographics and survey responses were analyzed using descriptive statistics and basic thematic
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analysis. Counts and percentages were used for categorical variables.
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3. RESULTS 3.1 Participant Characteristics
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A total of 54/72 (75%) CAPS members practicing in Canada completed the survey. Of the 54 respondents, 49 (90.7%) trained in Canada with 1 (1.9%) training in the United States, and
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4 (7.4%) training elsewhere. The majority 92.6% (50/54) had practiced for > 5 years and 90.7% (49/54) performed on average > 25 appendectomies per year. All respondents completed the
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questionnaire in its entirety. A summary of participant characteristics can be found in Table 1.
3.2 Management of Appendicitis Operative management was the preferred method of treatment for acute uncomplicated appendicitis for all respondents (54/54), with one preferring the open approach and the rest preferring laparoscopic. Ultrasound was the preferred method of imaging by all (54/54) and 23/54 (42.5%) agreed they prefer to have imaging even when history and physical exam support a diagnosis of appendicitis.
3.3 The Macroscopically Normal-Appearing Appendix Almost all respondents (53/54, 98%) reported performing at least one negative appendectomy in their career. All 54 participants stated they would remove a normal-appearing
Journal Pre-proof appendix during laparoscopy for suspected acute appendicitis in the absence of other intraoperative pathology. The most common reasons cited for removal of a normal-appearing appendix were: the possibility of endo/microscopic appendicitis (39/54, 72.2%), avoiding future diagnostic confusion (28/54, 51.9%), and patient preference/terms of the consent discussion (21/54, 38.9%) (Figure 1). Additionally, 49/54 (90.7%) agreed sufficient guidelines do not exist to guide decision making in this clinical scenario.
3.4 Microscopic Appendicitis
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Although 52% (28/54) were unsure, 39% (21/54) of respondents believed that
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microscopic appendicitis is a cause of right lower quadrant pain. Only 9% (5/54) believed that it is not. Conversely, 50% (27/54) believe that microscopic appendicitis will not become acute
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appendicitis with 4% (2/54) believing that it will and 46% (25/54) being unsure.
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4. DISCUSSION Despite being so common, management of pediatric appendicitis continues to be quite
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varied. This is partly due to conflicting results in the literature, providing little definitive guidance. A recent review of twelve pediatric emergency departments across Canada highlights
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this. It showed statistically significant differences in almost all aspects of care, including imaging strategies, time to operation, operative approach, and use of antibiotics across centers [36]. This
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variability in practice is further supported by the data acquired in the American College of Surgeons National Surgical Quality Improvement Program (ACS-NSQIP) database. The ACS-
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NSQIP was initially developed in Veteran’s Affairs hospitals in the 1990s to guide quality improvement initiatives [37]. This database has since been expanded to its current form, containing prospective, peer-controlled, validated data from over 400 participating hospitals. The morbidity of negative appendectomy is not insignificant at 6% [13]. However, one common justification for performing negative appendectomies is the belief that it is a relatively benign procedure that carries no additional risks when compared to uncomplicated appendectomy. This school of thought is supported by two retrospective series, comparing groups of NA and true appendicitis. These studies found no statistically significant differences in readmission (14.8% vs 9.54%, p=1.0), length of stay during readmission (1.95 vs 3.2 days, p=0.271), or in complications (11.9% vs 16.6%, p=0.141) [15,37). These results however, did not differentiate between acute uncomplicated and complicated appendicitis in their comparison
Journal Pre-proof to NA. When this distinction is considered, NA is associated with a statistically higher complication rate than uncomplicated appendicitis and is comparable to that of complicated appendicitis [10]. Kotaluoto et al., reviewed 164,579 cases of clinical appendicitis and found the 30-day mortality rate of uncomplicated appendicitis to be 2.1/1000. There was 4-fold increase in mortality related to NA and a 3.2-fold increase related to complicated appendicitis [38]. Although the comorbidities were higher in the NA group, this likely does not explain the discrepancy entirely. An analysis of short- and long-term mortality after appendectomy in 223,543 patients, controlling for the differences in comorbidities, age, and sex, showed higher
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30-day and long-term mortality in NA as compared to the uncomplicated appendicitis group
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(Hazard Ratio=3.32 p<0.001; Hazard Ratio=1.76 p<0.001)[39]. Similarly, in the pediatric population, a large retrospective analysis of 250,783 patients comparing NA to perforated
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appendicitis demonstrated a longer length of stay (7days vs. 3days, p<0.05), higher cost, and increased overall mortality (4/1000 vs. 1/10 000, p<0.001) [11]. This literature showing
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increased morbidity, mortality, cost, and length of stay in those with NA, as compared to those
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with acute uncomplicated appendicitis, highlights the importance of continuing to minimize the negative appendectomy rate.
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According to the APSA guidelines, the average response rate for internet studies is 40%, with physician-targeted surveys being even lower at 23-36% [33,40,41]. Our survey had a much higher response rate at 75%. This likely reflects the ubiquity of this clinical scenario and the
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paucity of data, making it highly relevant to our sampled population. Overall there were high
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levels (over 90%) agreement amongst respondents on most questions. This could be indicative of bias created within the wording of the questions. However, it is more likely explained by the relatively small group of pediatric surgeons surveyed, 90.7% of whom trained in the same eight Canadian centers. Interestingly, despite the growing evidence that antibiotic management of acute uncomplicated appendicitis is safe and often effective, all 54 survey participants still prefer operative management [42]. Although we did not include questions on opinions regarding this, it is possible that the high reported recurrence rate coupled with the relatively low morbidity of appendectomy, deters people from the use of antibiotics alone. Our results also indicated that ultrasound is the preferred method of imaging, and slightly less than half of surgeons still prefer to have imaging when the clinical picture is in keeping with appendicitis, both being consistent
Journal Pre-proof with the literature. All but one of the survey participants had performed at least one negative appendectomy in their career, supporting the notion that intraoperative diagnoses are unreliable [14,15,19,43]. All 54 participants agreed they would remove a normal-appearing appendix during laparoscopy for clinically suspected acute appendicitis in the absence of other findings that could explain symptomatology. This is higher than the literature in the adult population. According to two separate surveys, 64-73% of adult surgeons opt to remove the appendix in this scenario [32,44]. Our results indicate that this decision is driven by a number of reasons. The most important
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well as the potential for future confusion for the patient.
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reasons cited in our population were the concern regarding possible microscopic appendicitis as
One of the most common reasons cited in the literature for removing a normal-appearing
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appendix during laparoscopy for clinically suspected acute appendicitis is the high rate of intraoperative misdiagnosis. The quoted rates are highly variable, up to 58% for false positive
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diagnosis and 76% for false negative diagnosis [14,15]. However, multiple authors have
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questioned the clinical relevance of this misdiagnosis by studying the effects of leaving a normal-appearing appendix in situ. In one study, five of 211 (2.4%) patients who underwent
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diagnostic laparoscopy (DL) had complications (none infectious) compared to 52/498 (10%) of laparoscopic appendectomy (LA) patients (19 infectious) [21]. The authors did not test for statistical significance, but concluded their results supported the practice of performing DL to
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avoid a more traumatic procedure if it is not necessary. Conversely, Bhangu et al.,
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retrospectively compared DL patients with LA patients and found a 44% greater likelihood of being readmitted at one year with acute abdominal pain in those undergoing DL, along with an emergency readmission rate of 18.6% vs. 10.7% (p<0.001). Interestingly, in their study population only 2.1% (213) underwent subsequent appendectomy with 1% (101) having positive appendicitis on histology. These competing results are potentially explained by the concept of microscopic appendicitis (endoappendicitis), which describes the case when inflammation is confined to the luminal and/or muscularis layers of the appendix [23]. Available data suggest that microscopic appendicitis accounts for the majority of false negative diagnoses, highlighting the question of its clinical significance [6,14,29,30]. Unfortunately, the proportions of endo-, acute, and complicated appendicitis were not reported in these studies, with the authors also failing to specify whether the abdominal pain resolved after subsequent LA. Overall, the results
Journal Pre-proof of these types of studies should be interpreted with caution. Owing to the retrospective nature, the group selected by clinicians to undergo DL alone likely had a lower pre-test probability for appendicitis. This selection bias would affect the false negative rate as well as make it difficult to directly compare the two cohorts. Much controversy surrounds microscopic appendicitis, with the natural history and clinical significance of this condition being increasingly questioned. Some authors postulate that simple instrumentation of the appendix can lead to inflammation and query whether this can skew pathologic reports [23,45,46]. A satisfying pathophysiologic explanation is lacking as to
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how mucosal inflammation that does not travel to the serosa can cause RLQ pain. From the small
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number of studies that report specific pathology results, we know that a high proportion of macroscopically-negative, microscopically-positive appendicitis is histologically classified as
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microscopic appendicitis [6,14,29,30]. This calls into question the clinical significance of this diagnosis as discussed above. Some consider microscopic appendicitis to represent its own entity
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that often self-resolves, and not as the precursor to acute suppurative appendicitis [28]. Although
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the majority (52%) of our survey population were unsure, 39% of respondents believed that microscopic appendicitis is a cause of right lower quadrant pain with only 9% (5) believing it is
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not. Conversely, 50% (27) agree with the sentiments above that microscopic appendicitis will not become acute appendicitis with only 4% (2) believing that it will. The remaining 46% were unsure.
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It is clear that little high-quality evidence exists to guide surgeons in their decision
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making when confronted by a macroscopically normal-appearing appendix during laparoscopy for acute right lower quadrant pain. There is an even larger paucity of data for the pediatric population, in which this is a particularly common problem. Most (90.7%) of our population agreed that sufficient guidelines do not exist. Our survey is the first to our knowledge to gain insight into the decision making of pediatric surgeons when faced with this clinical scenario. There are some limitations to the study. This survey was a descriptive survey aimed at getting the general opinions and ideas surrounding this topic to serve as a launch point. It was designed following rigorous guidelines but is not a validated tool and thus is vulnerable to certain sources of bias. We were however, able to limit many of the usual sources such as nonresponse, and response bias by using a highly relevant topic with clear rationale and keeping it short. This survey was restricted to surgeons practicing in Canada, meaning its applicability to
Journal Pre-proof other populations may be limited.
5. CONCLUSION: The majority of pediatric surgeons agree sufficient guidelines do not exist to support decision making when a macroscopically normal-appearing appendix is found during laparoscopy for suspected acute appendicitis. This survey shows that removal of the appendix in this case would be supported by the majority of Canadian pediatric surgeons. This combined with the potential for higher morbidity and cost associated with NA justifies the continued efforts
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to improve care of this common disease.
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Table and Figure Legends
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Table 1 Participant characteristics
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Figure 1 Percentage of respondents who cite these common reasons for removing a normal appearing appendix *Other reasons included: parasite infection 3.7%, other pathology identified 5.5%, fee for appendicitis 1.9%, experience has shown symptoms resolve 1.9%, eliminate confusion if pain persists 7.4%
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APPENDIX 1
What is your current position?
In which country did your complete (or are you currently completing) your pediatric surgery training?
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Please specify the other country in which you completed your pediatric surgery training. In which country are you currently practicing?
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What is your age?
Male Female Other 25-35 36-45 46-55 56-65 >65 Staff pediatric surgeon 1st year pediatric surgery fellow 2nd year pediatric surgery fellow Retired surgeon Canada United States Other
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What is your gender?
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Canada United States Other
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Please specific the other country in which you are currently practicing. How many years have you been practicing as an independent pediatric surgeon?
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What is your preferred method of treating acute, uncomplicated appendicitis? Please specify your preferred non-operative method for treating acute, uncomplicated appendicitis. Do you prefer to have imaging even when the physical and history support a diagnosis of appendicitis? What kind if imaging do you prefer?
Please specify the other type of imaging you obtain. In your individual practice, approximately how many appendectomies do you perform a year?
What is your preferred method of appendectomy? You are performing a laparoscopic appendectomy for suspected acute uncomplicated appendicitis and the appendix appears structurally normal. You do not find
Currently completing fellowship Less than 5 years 5-10 years 11-20 years More than 20 years Appendectomy Non-operative methods
Yes No Ultrasound CT Other None 0-25 26-50 51-75 76-100 >100 Open Laparoscopic Yes No
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1. The possibility of microscopic/endoappendicitis 2. To prevent future appendicitis 3. To avoid future confusion for the patient as to whether they have had their appendix removed or not 4. Experience has shown that it improves symptoms post-op 5. The consent dictated that you were going to remove the appendix 6. Patient/parent preference based on the consent discussion beforehand 7. Obligation to do something since the child is already in surgery 8. This is how I was trained 9. Other Check all that apply.
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any other obvious causes for the patient’s symptoms (i.e. ovarian torsion). Do you remove this appendix? Why would you chose to remove this appendix?
1. Based on this scenario there is no evidence that the appendix is causing the patient’s symptoms 2. Unnecessary risk of post-operative complications 3. Evidence for the use of the appendix in reconstructive procedures 4. Immunological benefit of the appendix 5. This is how I was trained 6. Other Check all that apply.
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Please specify the other reason that you WOULD remove this appendix. Why would you chose NOT to remove this appendix?
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Please specify the other reason that you would NOT remove the appendix. Do you feel that there are sufficient guidelines on the topic of whether or not to remove a normal-appearing appendix during laparoscopy for suspected acute appendicitis? What is the source of these guidelines? In your individual practice, have you ever removed an appendix that pathology later deemed to be normal (negative appendectomy)? Do you believe that microscopic appendicitis/endoappendicitis is a cause of right lower quadrant pain? Do you believe that all cases of microscopic/endoappendicitis will become acute appendicitis? Please share any additional comments you have.
Yes No
Yes No Unsure Yes No Unsure Yes No Unsure
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Author contributions
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Each author should have participated sufficiently in the work to take public responsibility for appropriate portions of the manuscript contents. Allowing one’s name to appear as an author without contributing significantly to the study or adding the name of an individual that has not contributed or has not agreed to the study in its current form is considered a breach of appropriate authorship and publication ethics. No individual other than the authors listed below should have contributed substantially to the preparation and revision of the manuscript. Ghost-writing is not acceptable.
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Individuals who do not meet the criteria for authorship can be recognized and listed in the acknowledgments. This includes individuals that allow their clinical experience to be included in the study, those that provide only technical assistance, copyediting, proofreading or translation assistance, or a departmental leader that provided only general support including a department Chair.
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For multi-institutional studies, groups of individuals and institutions that have materially contributed to the study but, whose contributions do not justify authorship may be listed in an appendix under a heading such as participating or clinical investigators. Their function should be described for example as a scientific advisor, critically reviewed the study proposal or participating institution. Type the name of each author next to the appropriate following categories:
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Study Conception and Design Kathleen Logie, Lisa VanHouwelingen Data Acquisition Kathleen Logie, Tessa Robinson Analysis and Data Interpretation Kathleen Logie, Tessa Robinson Drafting of the Manuscript Kathleen Logie Critical Revision Kathleen Logie, Tessa Robinson, Lisa VanHouwelingen
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Percentage of Respondents (%) 59.3 40.7
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5.6 29.6 25.9 35.2 3.7
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Table 1 Participant characteristics Number of Respondents (n=54) Gender Male 32 Female 22 Age 25-35 3 36-35 16 46-55 14 56-65 19 >65 2 Location of Training Canada 49 USA 1 Other 4 Years of Independent Practice <5 6 5-10 12 11-20 15 >20 21 Appendectomies per Year 0-25 8 26-50 22 51-75 20 76-100 2 >100 2
90.7 1.9 7.4 11.1 22.2 27.8 38.9 14.8 43.1 37.0 3.7 3.7
Figure 1
Kathleen Logie, Tessa Robinson, Lisa VanHouwelingen PII:
S0022-3468(20)30078-6
DOI:
https://doi.org/10.1016/j.jpedsurg.2020.01.039
Reference:
YJPSU 59583
To appear in:
Journal of Pediatric Surgery
Received date:
21 January 2020
Accepted date:
25 January 2020
Please cite this article as: K. Logie, T. Robinson and L. VanHouwelingen, Management of the Normal-Appearing Appendix during Laparoscopy for clinically suspected acute appendicitis in the pediatric population, Journal of Pediatric Surgery(2020), https://doi.org/10.1016/j.jpedsurg.2020.01.039
This is a PDF file of an article that has undergone enhancements after acceptance, such as the addition of a cover page and metadata, and formatting for readability, but it is not yet the definitive version of record. This version will undergo additional copyediting, typesetting and review before it is published in its final form, but we are providing this version to give early visibility of the article. Please note that, during the production process, errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
© 2020 Published by Elsevier.
Journal Pre-proof Management of the Normal-Appearing Appendix During Laparoscopy for Clinically Suspected Acute Appendicitis in the Pediatric Population Kathleen Logiea, Tessa Robinsonb,c, Lisa VanHouwelingenb,c,d* a
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Division of General Surgery, Department of Surgery, McMaster University, Hamilton, Ontario, Canada b Division of Pediatric Surgery, Department of Surgery, McMaster University, Hamilton, Ontario, Canada c McMaster Pediatric Surgery Research Collaborative, McMaster University, Hamilton, Ontario, Canada d McMaster Children’s Hospital, Hamilton, Ontario, Canada
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Corresponding Author: Lisa VanHouwelingen McMaster Children’s Hospital Room 4E4 1200 Main Street West Hamilton, Ontario, Canada E-Mail: [email protected]
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COMPETING INTERESTS: The authors have no conflicts of interest to disclose.
Journal Pre-proof ABSTRACT Purpose: The widespread use of laparoscopy has brought forth the question of how to manage a macroscopically normal-appearing appendix in cases of clinically suspected appendicitis. This study aimed to determine the current practices of pediatric general surgeons in Canada regarding this matter. Methods: An online survey was created following the American Pediatric Surgical Association (APSA) guidelines and distributed via email to the Canadian Association of Pediatric Surgeons (CAPS) staff surgeons. The questions assessed clinician characteristics, standard practice, and rationale. Results were analyzed using descriptive statistics.
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Results: A total of 54/72 (75%) CAPS members practicing in Canada completed the survey. All (100%) agreed they would remove a normal-appearing appendix during laparoscopy for suspected acute appendicitis. The most common reasons were: possibility of microscopic appendicitis (39/54, 72.2%), avoiding future diagnostic confusion (28/54, 51.9%), and patient preference/consent discussion (21/54, 38.9%). Most (53/54, 98.1%) had performed a negative appendectomy and 49/54 (90.7%) agreed there were no sufficient guidelines.
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Conclusions: The majority of pediatric surgeons agree sufficient guidelines do not exist to support decision making when a normal-appearing appendix is found during laparoscopy for suspected acute appendicitis. This survey shows that removal of the appendix in this case would be supported by the majority of Canadian pediatric surgeons.
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KEYWORDS: Negative appendectomy; Appendicitis; Laparoscopic appendectomy; Pediatric
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TYPE OF STUDY: Survey
LEVEL OF EVIDENCE: VII (Expert Opinion)
ABBREVIATIONS: Negative Appendectomy (NA) Negative Appendectomy Rate (NAR) Canadian Association of Pediatric Surgeons (CAPS) American Pediatric Surgical Association (APSA) Diagnostic Laparoscopy (DL) Laparoscopic Appendectomy (LA)
Journal Pre-proof 1. INTRODUCTION Acute appendicitis represents the most common indication for abdominal surgery in the pediatric population [1]. Prior to the advent of laparoscopic surgery, most surgeons would remove the appendix regardless of intraoperative appearance due to the position of the scar raising concern for future diagnostic uncertainty. Thus, it was not uncommon for a histologically normal appendix to be removed, referred to as a negative appendectomy (NA). The NA rate (NAR) ranges in the literature from 1.8 up to 46% and trends higher in the pediatric population [2–12]. Although often considered relatively benign, NA has a 6% rate of morbidity [13].
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In the era of laparoscopy, the main argument supporting removal of a normal-appearing appendix is the high false negative rate of intraoperative diagnoses. Published data shows this to
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be as high as 76%, prompting the majority of authors to recommend appendectomy regardless of
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appearance [6,14–20]. However, when others have investigated the outcomes of leaving a normal-appearing appendix in situ, their rates of re-operation and were relatively low, ranging
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from 0-2.1% in available studies [7,21–28]. As a result of the confusion in the literature, current adult guidelines state that in the absence of other pathology seen during laparoscopy, the
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decision to remove the appendix should be based on the individual clinical scenario (level III, grade A) [31]. Despite this, two large surveys of adult surgeons have shown that 64-73% opt to
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remove all macroscopically normal-appearing appendixes as a practice. The most common reasons for removal were lack of clear guidelines and/or evidence on the topic, and the
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possibility of microscopic appendicitis [32,33].
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The pediatric population has a high incidence of appendicitis and negative appendectomy, with simultaneously low numbers of comorbidities to confound the data. Furthermore, to our knowledge, there remains no literature regarding surgeon decision making when faced with a macroscopically normal appendix during laparoscopy for clinically suspected acute appendicitis in pediatric centers. The purpose of this study was therefore to determine the current clinical practices and beliefs of pediatric general surgeons practicing in Canada regarding this matter. 2. METHODS A descriptive survey was created following the American Pediatric Surgical Association (APSA) guidelines for surveys [34]. Question formulation was performed by a specialty committee and informed by both a detailed literature search as well as feedback gained from the
Journal Pre-proof surgeons at the study center. The proposed survey was piloted to the same surgeons to test validity. The survey was then evaluated by the Canadian Association of Pediatric Surgeons (CAPS) research committee and further adjustments were made. Once finalized, the survey (Appendix 1) was distributed via email to all surgeon members of CAPS. The survey was distributed and all data collected in 2019. All Canadian surgeon members of CAPS were included; with all other non-staff level members or other personnel being excluded. Two additional reminders were sent at two-week intervals. No formal incentives were offered for completion.
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The data were collected using a Research Electronic Data Capture (REDCap) online
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database [35]. Data from staff surgeons practicing in Canada were analyzed. Participant demographics and survey responses were analyzed using descriptive statistics and basic thematic
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analysis. Counts and percentages were used for categorical variables.
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3. RESULTS 3.1 Participant Characteristics
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A total of 54/72 (75%) CAPS members practicing in Canada completed the survey. Of the 54 respondents, 49 (90.7%) trained in Canada with 1 (1.9%) training in the United States, and
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4 (7.4%) training elsewhere. The majority 92.6% (50/54) had practiced for > 5 years and 90.7% (49/54) performed on average > 25 appendectomies per year. All respondents completed the
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questionnaire in its entirety. A summary of participant characteristics can be found in Table 1.
3.2 Management of Appendicitis Operative management was the preferred method of treatment for acute uncomplicated appendicitis for all respondents (54/54), with one preferring the open approach and the rest preferring laparoscopic. Ultrasound was the preferred method of imaging by all (54/54) and 23/54 (42.5%) agreed they prefer to have imaging even when history and physical exam support a diagnosis of appendicitis.
3.3 The Macroscopically Normal-Appearing Appendix Almost all respondents (53/54, 98%) reported performing at least one negative appendectomy in their career. All 54 participants stated they would remove a normal-appearing
Journal Pre-proof appendix during laparoscopy for suspected acute appendicitis in the absence of other intraoperative pathology. The most common reasons cited for removal of a normal-appearing appendix were: the possibility of endo/microscopic appendicitis (39/54, 72.2%), avoiding future diagnostic confusion (28/54, 51.9%), and patient preference/terms of the consent discussion (21/54, 38.9%) (Figure 1). Additionally, 49/54 (90.7%) agreed sufficient guidelines do not exist to guide decision making in this clinical scenario.
3.4 Microscopic Appendicitis
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Although 52% (28/54) were unsure, 39% (21/54) of respondents believed that
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microscopic appendicitis is a cause of right lower quadrant pain. Only 9% (5/54) believed that it is not. Conversely, 50% (27/54) believe that microscopic appendicitis will not become acute
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appendicitis with 4% (2/54) believing that it will and 46% (25/54) being unsure.
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4. DISCUSSION Despite being so common, management of pediatric appendicitis continues to be quite
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varied. This is partly due to conflicting results in the literature, providing little definitive guidance. A recent review of twelve pediatric emergency departments across Canada highlights
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this. It showed statistically significant differences in almost all aspects of care, including imaging strategies, time to operation, operative approach, and use of antibiotics across centers [36]. This
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variability in practice is further supported by the data acquired in the American College of Surgeons National Surgical Quality Improvement Program (ACS-NSQIP) database. The ACS-
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NSQIP was initially developed in Veteran’s Affairs hospitals in the 1990s to guide quality improvement initiatives [37]. This database has since been expanded to its current form, containing prospective, peer-controlled, validated data from over 400 participating hospitals. The morbidity of negative appendectomy is not insignificant at 6% [13]. However, one common justification for performing negative appendectomies is the belief that it is a relatively benign procedure that carries no additional risks when compared to uncomplicated appendectomy. This school of thought is supported by two retrospective series, comparing groups of NA and true appendicitis. These studies found no statistically significant differences in readmission (14.8% vs 9.54%, p=1.0), length of stay during readmission (1.95 vs 3.2 days, p=0.271), or in complications (11.9% vs 16.6%, p=0.141) [15,37). These results however, did not differentiate between acute uncomplicated and complicated appendicitis in their comparison
Journal Pre-proof to NA. When this distinction is considered, NA is associated with a statistically higher complication rate than uncomplicated appendicitis and is comparable to that of complicated appendicitis [10]. Kotaluoto et al., reviewed 164,579 cases of clinical appendicitis and found the 30-day mortality rate of uncomplicated appendicitis to be 2.1/1000. There was 4-fold increase in mortality related to NA and a 3.2-fold increase related to complicated appendicitis [38]. Although the comorbidities were higher in the NA group, this likely does not explain the discrepancy entirely. An analysis of short- and long-term mortality after appendectomy in 223,543 patients, controlling for the differences in comorbidities, age, and sex, showed higher
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30-day and long-term mortality in NA as compared to the uncomplicated appendicitis group
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(Hazard Ratio=3.32 p<0.001; Hazard Ratio=1.76 p<0.001)[39]. Similarly, in the pediatric population, a large retrospective analysis of 250,783 patients comparing NA to perforated
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appendicitis demonstrated a longer length of stay (7days vs. 3days, p<0.05), higher cost, and increased overall mortality (4/1000 vs. 1/10 000, p<0.001) [11]. This literature showing
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increased morbidity, mortality, cost, and length of stay in those with NA, as compared to those
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with acute uncomplicated appendicitis, highlights the importance of continuing to minimize the negative appendectomy rate.
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According to the APSA guidelines, the average response rate for internet studies is 40%, with physician-targeted surveys being even lower at 23-36% [33,40,41]. Our survey had a much higher response rate at 75%. This likely reflects the ubiquity of this clinical scenario and the
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paucity of data, making it highly relevant to our sampled population. Overall there were high
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levels (over 90%) agreement amongst respondents on most questions. This could be indicative of bias created within the wording of the questions. However, it is more likely explained by the relatively small group of pediatric surgeons surveyed, 90.7% of whom trained in the same eight Canadian centers. Interestingly, despite the growing evidence that antibiotic management of acute uncomplicated appendicitis is safe and often effective, all 54 survey participants still prefer operative management [42]. Although we did not include questions on opinions regarding this, it is possible that the high reported recurrence rate coupled with the relatively low morbidity of appendectomy, deters people from the use of antibiotics alone. Our results also indicated that ultrasound is the preferred method of imaging, and slightly less than half of surgeons still prefer to have imaging when the clinical picture is in keeping with appendicitis, both being consistent
Journal Pre-proof with the literature. All but one of the survey participants had performed at least one negative appendectomy in their career, supporting the notion that intraoperative diagnoses are unreliable [14,15,19,43]. All 54 participants agreed they would remove a normal-appearing appendix during laparoscopy for clinically suspected acute appendicitis in the absence of other findings that could explain symptomatology. This is higher than the literature in the adult population. According to two separate surveys, 64-73% of adult surgeons opt to remove the appendix in this scenario [32,44]. Our results indicate that this decision is driven by a number of reasons. The most important
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well as the potential for future confusion for the patient.
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reasons cited in our population were the concern regarding possible microscopic appendicitis as
One of the most common reasons cited in the literature for removing a normal-appearing
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appendix during laparoscopy for clinically suspected acute appendicitis is the high rate of intraoperative misdiagnosis. The quoted rates are highly variable, up to 58% for false positive
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diagnosis and 76% for false negative diagnosis [14,15]. However, multiple authors have
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questioned the clinical relevance of this misdiagnosis by studying the effects of leaving a normal-appearing appendix in situ. In one study, five of 211 (2.4%) patients who underwent
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diagnostic laparoscopy (DL) had complications (none infectious) compared to 52/498 (10%) of laparoscopic appendectomy (LA) patients (19 infectious) [21]. The authors did not test for statistical significance, but concluded their results supported the practice of performing DL to
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avoid a more traumatic procedure if it is not necessary. Conversely, Bhangu et al.,
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retrospectively compared DL patients with LA patients and found a 44% greater likelihood of being readmitted at one year with acute abdominal pain in those undergoing DL, along with an emergency readmission rate of 18.6% vs. 10.7% (p<0.001). Interestingly, in their study population only 2.1% (213) underwent subsequent appendectomy with 1% (101) having positive appendicitis on histology. These competing results are potentially explained by the concept of microscopic appendicitis (endoappendicitis), which describes the case when inflammation is confined to the luminal and/or muscularis layers of the appendix [23]. Available data suggest that microscopic appendicitis accounts for the majority of false negative diagnoses, highlighting the question of its clinical significance [6,14,29,30]. Unfortunately, the proportions of endo-, acute, and complicated appendicitis were not reported in these studies, with the authors also failing to specify whether the abdominal pain resolved after subsequent LA. Overall, the results
Journal Pre-proof of these types of studies should be interpreted with caution. Owing to the retrospective nature, the group selected by clinicians to undergo DL alone likely had a lower pre-test probability for appendicitis. This selection bias would affect the false negative rate as well as make it difficult to directly compare the two cohorts. Much controversy surrounds microscopic appendicitis, with the natural history and clinical significance of this condition being increasingly questioned. Some authors postulate that simple instrumentation of the appendix can lead to inflammation and query whether this can skew pathologic reports [23,45,46]. A satisfying pathophysiologic explanation is lacking as to
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how mucosal inflammation that does not travel to the serosa can cause RLQ pain. From the small
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number of studies that report specific pathology results, we know that a high proportion of macroscopically-negative, microscopically-positive appendicitis is histologically classified as
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microscopic appendicitis [6,14,29,30]. This calls into question the clinical significance of this diagnosis as discussed above. Some consider microscopic appendicitis to represent its own entity
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that often self-resolves, and not as the precursor to acute suppurative appendicitis [28]. Although
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the majority (52%) of our survey population were unsure, 39% of respondents believed that microscopic appendicitis is a cause of right lower quadrant pain with only 9% (5) believing it is
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not. Conversely, 50% (27) agree with the sentiments above that microscopic appendicitis will not become acute appendicitis with only 4% (2) believing that it will. The remaining 46% were unsure.
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It is clear that little high-quality evidence exists to guide surgeons in their decision
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making when confronted by a macroscopically normal-appearing appendix during laparoscopy for acute right lower quadrant pain. There is an even larger paucity of data for the pediatric population, in which this is a particularly common problem. Most (90.7%) of our population agreed that sufficient guidelines do not exist. Our survey is the first to our knowledge to gain insight into the decision making of pediatric surgeons when faced with this clinical scenario. There are some limitations to the study. This survey was a descriptive survey aimed at getting the general opinions and ideas surrounding this topic to serve as a launch point. It was designed following rigorous guidelines but is not a validated tool and thus is vulnerable to certain sources of bias. We were however, able to limit many of the usual sources such as nonresponse, and response bias by using a highly relevant topic with clear rationale and keeping it short. This survey was restricted to surgeons practicing in Canada, meaning its applicability to
Journal Pre-proof other populations may be limited.
5. CONCLUSION: The majority of pediatric surgeons agree sufficient guidelines do not exist to support decision making when a macroscopically normal-appearing appendix is found during laparoscopy for suspected acute appendicitis. This survey shows that removal of the appendix in this case would be supported by the majority of Canadian pediatric surgeons. This combined with the potential for higher morbidity and cost associated with NA justifies the continued efforts
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to improve care of this common disease.
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Data Capture (REDCap) - A metadata-driven methodology and workflow process for providing translational research informatics support. J Biomed Inform. 2009;42(2):377– 81. Thompson GC, Schuh S, Gravel J, Reid S, Fitzpatrick E, Turner T, et al. Variation in the Diagnosis and Management of Appendicitis at Canadian Pediatric Hospitals. Acad Emerg Med. 2015;22(7):811–22. Khuri SF, Daley J, Henderson W, Hur K, Demakis J, Aust JB, et al. The Department of Veterans Affairs’ NSQIP: the first national, validated, outcome-based, risk-adjusted, and peer-controlled program for the measurement and enhancement of the quality of surgical care. National VA Surgical Quality Improvement Program. Ann Surg [Internet]. 1998 Oct [cited 2019 Jul 25];228(4):491–507. Available from: http://www.ncbi.nlm.nih.gov/pubmed/9790339 Lee M, Paavana T, Mazari F, Wilson TR. The morbidity of negative appendicectomy. Ann R Coll Surg Engl. 2014;96(7):517–20. Kotaluoto S, Ukkonen M, Pauniaho SL, Helminen M, Sand J, Rantanen T. Mortality Related to Appendectomy; a Population Based Analysis over Two Decades in Finland. World J Surg. 2017;41(1):64–9. Andersson RE. Short and long-term mortality after appendectomy in Sweden 1987 to 2006. Influence of appendectomy diagnosis, sex, age, co-morbidity, surgical method, hospital volume, and time period. A national population-based cohort study. World J Surg. 2013;37(5):974–81. Kellerman SE, Herold J. Physician Response to Surveys A Review of the Literature. Am J Prev Med [Internet]. 2001 [cited 2019 Jun 26];20(1):61–7. Available from: www.elseveir.com/locate/ajpmonline. Cunningham CT, Quan H, Hemmelgarn B, Noseworthy T, Beck CA, Dixon E, et al. Exploring physician specialist response rates to web-based surveys. BMC Med Res Methodol [Internet]. 2015 Dec 9 [cited 2019 Jun 26];15(1):32. Available from: https://bmcmedresmethodol.biomedcentral.com/articles/10.1186/s12874-015-0016-z Xu J, Adams S, Liu YC, Karpelowsky J. Nonoperative management in children with early acute appendicitis: A systematic review. J Pediatr Surg [Internet]. 2017 Sep [cited 2019 Jun 26];52(9):1409–15. Available from: http://www.ncbi.nlm.nih.gov/pubmed/28535960 Jones MW, Paterson AG. The correlation between gross appearance of the appendix at aopendicectomy and histological examination. Ann R Coll Surg Engl. 1988;70(2):93–4. Turner E, Lightwood R. Management of the Normal Appendix during Laparoscopy for Right Iliac Fossa Pain. World J Laparosc Surg. 2009;2(2):15–7. Pieper R, Kager L, Nasman P. Clinical Significance of Mucosal Inflammation of the Vermiform Appendix. Ann Surg. 1983;197(3):368–74. Slotboom T, Hamminga JT, Hofker HS, Heineman E, Haveman JW, Evaluation ASGA and aparoscopic. Intraoperative motive for performing a laparoscopic appendectomy on a postoperative histological proven normal appendix. Scand J Surg. 2014;103(4):245–8.
Table and Figure Legends
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Table 1 Participant characteristics
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Figure 1 Percentage of respondents who cite these common reasons for removing a normal appearing appendix *Other reasons included: parasite infection 3.7%, other pathology identified 5.5%, fee for appendicitis 1.9%, experience has shown symptoms resolve 1.9%, eliminate confusion if pain persists 7.4%
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APPENDIX 1
What is your current position?
In which country did your complete (or are you currently completing) your pediatric surgery training?
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Please specify the other country in which you completed your pediatric surgery training. In which country are you currently practicing?
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What is your age?
Male Female Other 25-35 36-45 46-55 56-65 >65 Staff pediatric surgeon 1st year pediatric surgery fellow 2nd year pediatric surgery fellow Retired surgeon Canada United States Other
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What is your gender?
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Canada United States Other
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Please specific the other country in which you are currently practicing. How many years have you been practicing as an independent pediatric surgeon?
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What is your preferred method of treating acute, uncomplicated appendicitis? Please specify your preferred non-operative method for treating acute, uncomplicated appendicitis. Do you prefer to have imaging even when the physical and history support a diagnosis of appendicitis? What kind if imaging do you prefer?
Please specify the other type of imaging you obtain. In your individual practice, approximately how many appendectomies do you perform a year?
What is your preferred method of appendectomy? You are performing a laparoscopic appendectomy for suspected acute uncomplicated appendicitis and the appendix appears structurally normal. You do not find
Currently completing fellowship Less than 5 years 5-10 years 11-20 years More than 20 years Appendectomy Non-operative methods
Yes No Ultrasound CT Other None 0-25 26-50 51-75 76-100 >100 Open Laparoscopic Yes No
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1. The possibility of microscopic/endoappendicitis 2. To prevent future appendicitis 3. To avoid future confusion for the patient as to whether they have had their appendix removed or not 4. Experience has shown that it improves symptoms post-op 5. The consent dictated that you were going to remove the appendix 6. Patient/parent preference based on the consent discussion beforehand 7. Obligation to do something since the child is already in surgery 8. This is how I was trained 9. Other Check all that apply.
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any other obvious causes for the patient’s symptoms (i.e. ovarian torsion). Do you remove this appendix? Why would you chose to remove this appendix?
1. Based on this scenario there is no evidence that the appendix is causing the patient’s symptoms 2. Unnecessary risk of post-operative complications 3. Evidence for the use of the appendix in reconstructive procedures 4. Immunological benefit of the appendix 5. This is how I was trained 6. Other Check all that apply.
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Please specify the other reason that you WOULD remove this appendix. Why would you chose NOT to remove this appendix?
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Please specify the other reason that you would NOT remove the appendix. Do you feel that there are sufficient guidelines on the topic of whether or not to remove a normal-appearing appendix during laparoscopy for suspected acute appendicitis? What is the source of these guidelines? In your individual practice, have you ever removed an appendix that pathology later deemed to be normal (negative appendectomy)? Do you believe that microscopic appendicitis/endoappendicitis is a cause of right lower quadrant pain? Do you believe that all cases of microscopic/endoappendicitis will become acute appendicitis? Please share any additional comments you have.
Yes No
Yes No Unsure Yes No Unsure Yes No Unsure
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Author contributions
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Each author should have participated sufficiently in the work to take public responsibility for appropriate portions of the manuscript contents. Allowing one’s name to appear as an author without contributing significantly to the study or adding the name of an individual that has not contributed or has not agreed to the study in its current form is considered a breach of appropriate authorship and publication ethics. No individual other than the authors listed below should have contributed substantially to the preparation and revision of the manuscript. Ghost-writing is not acceptable.
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Individuals who do not meet the criteria for authorship can be recognized and listed in the acknowledgments. This includes individuals that allow their clinical experience to be included in the study, those that provide only technical assistance, copyediting, proofreading or translation assistance, or a departmental leader that provided only general support including a department Chair.
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For multi-institutional studies, groups of individuals and institutions that have materially contributed to the study but, whose contributions do not justify authorship may be listed in an appendix under a heading such as participating or clinical investigators. Their function should be described for example as a scientific advisor, critically reviewed the study proposal or participating institution. Type the name of each author next to the appropriate following categories:
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Study Conception and Design Kathleen Logie, Lisa VanHouwelingen Data Acquisition Kathleen Logie, Tessa Robinson Analysis and Data Interpretation Kathleen Logie, Tessa Robinson Drafting of the Manuscript Kathleen Logie Critical Revision Kathleen Logie, Tessa Robinson, Lisa VanHouwelingen
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Percentage of Respondents (%) 59.3 40.7
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5.6 29.6 25.9 35.2 3.7
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Table 1 Participant characteristics Number of Respondents (n=54) Gender Male 32 Female 22 Age 25-35 3 36-35 16 46-55 14 56-65 19 >65 2 Location of Training Canada 49 USA 1 Other 4 Years of Independent Practice <5 6 5-10 12 11-20 15 >20 21 Appendectomies per Year 0-25 8 26-50 22 51-75 20 76-100 2 >100 2
90.7 1.9 7.4 11.1 22.2 27.8 38.9 14.8 43.1 37.0 3.7 3.7
Figure 1