Maksym Situkho1,2*
Tetsuo Ikezono3
Marte Myhrum1,2
Arne Kirkhorn Rødvik1
Yukihide Maeda3
Han Matsuda3
Masafumi Sawada3
Greg Eigner Jablonski1,2- 1Department of Otorhinolaryngology and Head and Neck Surgery, Oslo University Hospital, Oslo, Norway
- 2Institute for Clinical Medicine, Faculty of Medicine, University of Oslo, Oslo, Norway
- 3Department of Otorhinolaryngology, Faculty of Medicine, Saitama Medical University, Saitama, Japan
Objective: Perilymphatic fistula (PLF) is a challenging inner ear condition, characterized by the abnormal connection between inner and middle ear and subsequent leakage of inner ear liquid (perilymph) into the middle ear. Early surgical intervention for PLF might lead to favorable audiological and vestibular outcomes. The conventional reference standard of PLF detection is the intraoperative visualization of perilymph leakage during explorative tympanotomy or endoscopic inspection; the specificity and sensitivity of this method is unknown. Systematic reviews assessing the accuracy of diagnostic tests for PLF are not available.
To systematically review the literature for qualitative evidence exploring the diagnostic tests for the PLF diagnosis. The proposed systematic review will answer the following question: What is the accuracy of diagnostic tests in detecting perilymphatic fistula in humans?
Methods: Eligibility criteria: original peer-reviewed articles regarding studies on humans of any age containing data with diagnostic test accuracy estimation (sensitivity and specificity) for PLF diagnosis or in which diagnostic test accuracy could be calculated based on data provided, without language, study design or study date limits. MEDLINE, EMBASE, the Cochrane Central Register of Controlled Trials, Scopus, and Web of Science will be searched for eligible articles. Additional manual searches will be performed. Covidence software will be used for title and abstract screening, full text review and data extraction. The risk of bias assessment will be conducted using the Quality Assessment of Diagnostic Test Accuracy 2 (QUADAS-2) tool. If two or more high-quality articles evaluating the same diagnostic test will be identified, their findings will be quantitatively synthesized; if a quantitative synthesis is not feasible, the results will be described in a narrative summary. Grading of Recommendations, Assessment, Development, and Evaluations (GRADE) framework will be used to evaluate evidence strength. Study funded by Oslo University Hospital.
1 Introduction
1.1 Background
Perilymphatic fistula (PLF) is a difficult to diagnose inner ear condition, which is characterized by the abnormal connection between inner and middle ear and subsequent leakage of inner ear liquid (perilymph) into the middle ear (1, 2). The primary manifestations of the PLF are sudden or progressive, fluctuating sensorineural hearing loss or deafness and/or vestibular symptoms ranging from dizziness to rotatory vertigo (3).
Several diagnostic criteria were published previously (4–6); the recent one is published from a Japanese PLF research group (7). However, due to the lack of internationally accepted diagnostic criteria and the difficulty of making a definitive diagnosis, the epidemiology of PLF needs to be clarified. The heterogeneity of the causing factors adds to the difficulty of disease prevalence and incidence measures (2, 8). It has been reported that PLF was intraoperatively diagnosed in 24% of patients with severe to profound sudden sensorineural hearing loss, not responding to high doses of steroids (9).
PLF can be caused by a wide range of factors, such as head trauma (10–12), changes in air pressure (e.g., during air travel) (13–16), barotrauma (5), chronic ear infections (17, 18), congenital abnormalities (19), or as a result of an iatrogenic injury during cochlear implantation or stapes surgery (20, 21). Individuals with occupations or activities involving exposure to sudden pressure changes or repeated physical strain may be at higher risk, especially divers and deployed military personnel (22–24).
Early diagnosis and surgical intervention for PLF can lead to favorable audiological and vestibular outcomes (1, 25). However, the prognosis varies among individuals, and not all patients experience complete resolution of symptoms. Improvement in vestibular symptoms tends to be more significant than auditory symptoms (3).
The diagnosis of perilymphatic fistula (PLF) is challenging and depends on the integration of clinical evaluations and functional tests. Investigations include comprehensive audio-vestibular testing, such as pure tone, speech, and positional audiometry (7, 26), electro- and videonystagmography (27), vestibular evoked myogenic potentials (28), electrocochleography (29), video Head Impulse Testing (30), posturography (31), MRI and high-resolution computed tomography (1, 5, 32). Biomarkers, such as beta-2 transferrin, beta-trace protein, and Cochlin-tomoprotein, are also being used in diagnosis (33–37).
The conventional reference standard of PLF detection is the intraoperative visualization of perilymph leakage during explorative tympanotomy or endoscopic inspection (9, 38, 39). The reference standard is far from being perfect; the specificity and sensitivity of this method is unknown (22). Direct visualization of the round and oval windows is not always possible, and the diagnosis cannot be excluded in case of the absence of perilymph leakage. The presence of transparent fluid collection in the round and oval window niches may not necessarily represent perilymph, as tissue fluid, local anesthetic, and irrigation fluid can all be mistaken for perilymph (1, 40, 41).
1.2 Relevance
The accuracy of diagnostic tests for PLF significantly affects individual health outcomes. Early and precise diagnosis facilitates appropriate treatment, potentially improving auditory and vestibular functions, and enhancing the quality of life of affected individuals.
Healthcare professionals could be more certain in selecting appropriate diagnostic methods for suspected PLF cases, thereby providing better patient management.
Accurate diagnostic tests for PLF are important for public health, as they enable awareness and early symptom recognition. This can prevent treatment delays, reduce PLF burden, and enhance public health outcomes.
This review’s evidence-based recommendations on PLF diagnostic tests can guide healthcare and reimbursement policies, guidelines, and resource allocation.
The review will also have strong implications for future PLF research. Identifying promising diagnostic tests can guide new studies, including the development of new diagnostic criteria, validation of existing tests in different cohorts, and biomarker exploration. This can advance diagnostic accuracy, deepen the understanding of PLF pathophysiology, and promote targeted treatment strategies. Moreover, comprehensive analysis of bias sources in previous studies, as provided by systematic reviews, can pave the way for new high-quality research.
1.3 Rationale
There are two existing systematic reviews related to PLF, but they do not specifically address the accuracy of diagnostic tests (42, 43). The first systematic review focuses on the presentation, management, and hearing outcomes of labyrinthine fistula secondary to cholesteatoma, while the second review explores the association between sneezing and perilymphatic fistula of the round window. Thus, existing systematic reviews on PLF offer valuable insights but do not address diagnostic test accuracy, leaving the current evidence insufficient to answer this question.
1.4 Objective
Our study’s objective is to systematically review the literature for qualitative evidence exploring the diagnostic tests for the PLF diagnosis. PIT (population, index tests, and target condition) methodology was used to define the research question (44).
The proposed systematic review will answer the following question:
What is the accuracy of diagnostic tests in detecting perilymphatic fistula in humans?
What are the PIT components of the review question/objective?
P (Population) – humans.
I (Index tests) – all available diagnostic tests.
T (target condition) – perilymphatic fistula.
2 Methods
2.1 Protocol development
The protocol has been developed using the Cochrane handbook for reviews of diagnostic test accuracy and the Preferred Reporting Items for a Systematic Review and Meta-analysis of Diagnostic Test Accuracy Studies (PRISMA-DTA) statement (45, 46). This protocol is reported in line with the PRISMA statement for review protocols (PRISMA-P), which is attached in Supplementary Table S1 (47).
2.2 Eligibility criteria
Studies on humans of any age containing data with diagnostic test accuracy estimation (sensitivity and specificity) for PLF diagnosis.
Studies on humans of any age on PLF, in which diagnostic test accuracy could be calculated based on data provided in the article.
Original peer-reviewed articles will be included.
No language limits will be imposed.
No study design restriction will be imposed.
2.3 Information sources
For the identification of relevant studies, we will use following electronic databases: MEDLINE (Ovid interface, 1948 onwards), EMBASE (Ovid interface, 1980 onwards), the Cochrane Central Register of Controlled Trials (CENTRAL, Wiley interface, latest issue), Scopus, and Web of Science. In addition, the reference lists of the included studies and relevant reviews will be manually examined.
After the completion of the search, a bibliography of the identified studies will be distributed among the research team and top experts in the field of perilymphatic fistula for review.
2.4 Search strategy
Two factors are important to consider in the context of our review search strategy. First, diagnostic test accuracy studies are often poorly reported in the titles and abstracts. Use of search filters to identify diagnostic accuracy studies might lead to missing relevant studies (48). Second, PLF is a rare condition and the amount of publication regarding it is limited. Therefore, our search strategy was fitted to identify as many studies considering the target condition as possible.
The strategy (Supplementary Table S2) was built with an expert in systematic searches from The Library of Medicine and Science, University of Oslo library.
Search results will be deduplicated using EndNote reference management software (Clarivate, London, United Kingdom) and then uploaded to the specialized online review tool, Covidence (Veritas Health Innovation, Melbourne, Australia). Two investigators will screen the titles and abstracts. Any disagreements will be resolved by consensus decision. Priority screening with machine learning to focus early screening effort on most relevant records will be utilized. If no consensus is achieved, a third investigator will be invited to resolve the disagreement. Two authors will independently conduct full text screening on the chosen articles. Subsequently, the selected articles will proceed to data extraction.
2.5 Data extraction
A structured data extraction form will be developed from scratch in the Covidence software, containing all relevant data fields based on the key data elements identified. The form will be designed to capture study characteristics, participant characteristics, details of the index test and reference standards, diagnostic accuracy measures, and other relevant outcomes.
Study characteristics:
• Author(s) and year of publication
• Dates of data collection of the study (if available)
• Study design (e.g., prospective cohort, case–control, diagnostic accuracy study)
• Country or setting where the study was conducted
• Duration of the study, if applicable
Participant characteristics:
• Total number of participants included in the study
• Characteristics of the study population (e.g., age, gender, and clinical presentation)
• Inclusion and exclusion criteria for participant selection
• Reason for the referral for the index test (symptoms, other disease, etc.)
Index test information:
• Description of the index test(s) used to diagnose PLF
• Type of index test (e.g., clinical, instrumental, biomarker, imaging)
• Details on the procedure and protocol of the index test (time between index test and reference standard)
• Threshold(s) used for dichotomous interpretation of the index test results
Reference standard information:
• Description of the reference standard(s) used to confirm or exclude PLF
• Details on the procedure and protocol of the reference standard
• Threshold(s) used for dichotomous interpretation of the reference standard results
Diagnostic accuracy measures:
• Sensitivity: The proportion of true positive results among individuals with PLF
• Specificity: The proportion of true negative results among individuals without PLF
• 95% confidence intervals or standard errors for the above measures (if available)
• If sensitivity/specificity is not calculated, True Positive/True Negative/False Positive/False Negative cases data will be retrieved (using 2*2 table)
The data extraction form will be pilot-tested on a subset of included studies to ensure that it captures all necessary information and is easy to use. Necessary revisions will be made based on feedback from the pilot testing.
A narrative summary of the key findings extracted from the included studies will be presented, highlighting important trends, variations, or inconsistencies in the data.
2.6 Assessment of the risk of bias
The risk of bias assessment for individual studies will be conducted using the Quality Assessment of Diagnostic Test Accuracy 2 (QUADAS-2) tool, which is designed to evaluate the quality of primary diagnostic accuracy studies, and consists of four key domains - patient selection, index test, reference standard, and flow and timing (49). QUADAS-2 checklist is provided in Supplementary Table S3. The assessment will be carried out independently by two reviewers. Any disagreements between the two reviewers will be resolved through discussion and consensus. If consensus cannot be reached, a third reviewer will be consulted. The information obtained from the assessment will be provided in a narrative summary and displayed in a table.
Articles with high risk of bias may be excluded from the data analysis if studies with less risk of bias provide similar information or when the extent of the bias could significantly distort the overall results.
2.7 Data synthesis
If two or more articles of appropriate quality evaluating the same diagnostic test are identified, their findings will be quantitatively synthesized into an analysis of sensitivity and specificity (45). If quantitative synthesis is not appropriate, data will be organized into tabular forms and explained in a detailed descriptive summary.
Based on the analysis of potentially relevant articles, the review will investigate two distinct study categories in perilymphatic fistula research.
1. Diagnostic studies in which explorative tympanotomy and perilymph leakage visualization were used as reference standard and performed after the index test in patients with suspected PLF. Data synthesis will consider the limitations of the reference standard. To address the imperfect reference standard and heterogeneity of index tests, a Bayesian methodology can be employed (50).
2. Diagnostic studies with perilymph leakage occurring during surgery, such as cochlear implantation or stapedotomy. The index test, usually biomarker detection, followed the reference standard. Assuming the definite presence of a connection between inner and middle ear during the surgery, separate data synthesis will be performed.
No assessment of meta-bias is planned
2.8 Assessment of confidence in cumulative evidence
Strength of evidence and clinical practice recommendations will be evaluated and presented using the GRADE (Grading of Recommendations, Assessment, Development, and Evaluations) framework (51). Evidence strength will be assessed as high (“We are very confident that the true effect lies close to that of the estimate of the effect”), moderate (“We are moderately confident in the effect estimate: The true effect is likely to be close to the estimate of the effect, but there is a possibility that it is substantially different”), low (“Our confidence in the effect estimate is limited: The true effect may be substantially different from the estimate of the effect”), or very low (“We have very little confidence in the effect estimate: The true effect is likely to be substantially different from the estimate of the effect”).
3 Amendments
If we need to amend this protocol, we will give the date of each amendment, describe the change and give the rationale in this section. Changes will not be incorporated into the protocol.
4 Reporting the review
The systematic review will be reported in accordance with Preferred Reporting Items for a Systematic Review and Meta-analysis of Diagnostic Test Accuracy Studies: The PRISMA-DTA Statement (46). A flowchart will be used to present the flow of study selection, screening, and inclusion, as well as any relevant findings related to the diagnostic accuracy of tests for PLF.
Ethics statement
The underlying study is based on a systematic assessment of publicly accessible documents. No personal, sensitive or confidential information will be collected. Therefore, the systematic review does not require ethical approval (52). The results of the systematic review will be published in a peer-reviewed journal.
Author contributions
MakS: Conceptualization, Investigation, Methodology, Software, Writing – original draft, Writing – review & editing. TI: Conceptualization, Supervision, Writing – original draft, Writing – review & editing. MM: Conceptualization, Funding acquisition, Project administration, Supervision, Writing – original draft, Writing – review & editing. AR: Methodology, Writing – original draft, Writing – review & editing, Conceptualization. YM: Writing – original draft, Writing – review & editing. HM: Writing – original draft, Writing – review & editing. MasS: Writing – original draft, Writing – review & editing. GJ: Writing – original draft, Writing – review & editing, Conceptualization, Funding acquisition, Methodology, Resources, Supervision, Project administration.
Funding
The author(s) declare that financial support was received for the research, authorship, and/or publication of this article. This research is funded by the Oslo University Hospital, Department of Otorhinolaryngology and Head and Neck Surgery. Non-financial sources of support. The University of Oslo provided access to Covidence software for systematic reviews. Oslo University Hospital, University of Oslo and Saitama Medical University granted access to scientific databases.
Acknowledgments
We wish to thank Tarjei Fiskegård Werner from the Library of Medicine and Science, University of Oslo Library, for providing expertise in search strategy development. We also extend our gratitude to Corina Silvia Rüegg and Hilde Kjelgaard Brustad from the Oslo Center for Biostatistics and Epidemiology (OCBE) for their guidance in methods development. Specially developed by University of Oslo version of Chat GPT (GPT UiO) was used for the grammar check according to the University’s privacy requirements.
Conflict of interest
TI was developing the Cochlin-tomoprotein detection test for diagnosing perilymphatic fistula, the basic patent associated with this test expired in June 2023. TI has not received any financial benefit from the patent since its expiration. Furthermore, no royalties are currently being paid for the divisional patent. As a result, neither TI nor Saitama Medical University receive any financial benefit from the technology. HM has co-authored articles focused on Cochlin-tomoprotein detection in PLF diagnosis. GJ has co-authored articles related to Beta-trace protein detection for diagnosing PLF. However, it is important to note that TI, HM, and GJ will not be involved in the data extraction or risk of bias assessment for this study. The review will follow rigorous methodology and ensure transparency in data collection, analysis, and interpretation.
Publisher’s note
All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.
Supplementary material
The Supplementary material for this article can be found online at: https://www.frontiersin.org/articles/10.3389/fneur.2024.1500780/full#supplementary-material
References
1. Deveze, A, Matsuda, H, Elziere, M, and Ikezono, T. Diagnosis and treatment of Perilymphatic Fistula. Adv Otorhinolaryngol. (2018) 81:133–45. doi: 10.1159/000485579
2. Furhad, S, and Bokhari, AA. Perilymphatic Fistula. Treasure Island, FL: StatPearls Publishing LLC (2023).
3. Sarna, B, Abouzari, M, Merna, C, Jamshidi, S, Saber, T, and Djalilian, HR. Perilymphatic Fistula: a review of classification, etiology, diagnosis, and treatment. Front Neurol. (2020) 11:1046. doi: 10.3389/fneur.2020.01046
4. Kohut, RI . Perilymph fistulas: clinical criteria. Arch Otolaryngol Head Neck Surg. (1992) 118:687–92. doi: 10.1001/archotol.1992.01880070017003
5. Choi, JE, Moon, IJ, Kim, H, Lee, K, Cho, Y-S, and Chung, W-H. Diagnostic criteria of barotraumatic perilymph fistula based on clinical manifestations. Acta Otolaryngol. (2017) 137:16–22. doi: 10.1080/00016489.2016.1213419
6. Ikezono, T . The perilymphatic fistula: new diagnostic criteria and clinical characteristics. Equilib Res. (2013) 72:215–21. doi: 10.3757/jser.72.215
7. Matsuda, H, Sakamoto, K, Matsumura, T, Saito, S, Shindo, S, Fukushima, K, et al. A nationwide multicenter study of the Cochlin Tomo-protein detection test: clinical characteristics of perilymphatic fistula cases. Acta Otolaryngol. (2017) 137:S53–9. doi: 10.1080/00016489.2017.1300940
8. Møller, P, Molvaer, OI, and Lind, O. Perilymphatic fistula. Tidsskr Nor Laegeforen. (2001) 121:162–5.
9. Heilen, S, Lang, CP, Warnecke, A, Lenarz, T, and Durisin, M. Exploratory tympanotomy in sudden sensorineural hearing loss for the identification of a perilymphatic fistula - retrospective analysis and review of the literature. J Laryngol Otol. (2020) 134:501–8. doi: 10.1017/S0022215120000948
10. Goda, M, Murata, K, Tsuda, M, and Fujiki, M. A case of perilymphatic fistula accompanied with traumatic brain injury. Neurotraumatology. (2022) 45:12–6. doi: 10.32187/neurotraumatology.45.1_12
11. Nakajima, T, Motegi, M, and Yamamoto, Y. Penetrating trauma-induced Perilymphatic Fistula: a case report and literature review. Cureus. (2023) 15:e36106. doi: 10.7759/cureus.36106
12. Maillot, O, Attyé, A, Boyer, E, Heck, O, Kastler, A, Grand, S, et al. Post traumatic deafness: a pictorial review of CT and MRI findings. Insights Imaging. (2016) 7:341–50. doi: 10.1007/s13244-016-0490-9
13. Mirza, S, and Richardson, H. Otic barotrauma from air travel. J Laryngol Otol. (2005) 119:366–70. doi: 10.1258/0022215053945723
14. Klokker, M, and Vesterhauge, S. Perilymphatic fistula in cabin attendants: an incapacitating consequence of flying with common cold. Aviat Space Environ Med. (2005) 76:66–8.
15. Mallen, JR, and Roberts, DS. SCUBA medicine for otolaryngologists: part I. Diving into SCUBA physiology and injury prevention. Laryngoscope. (2020) 130:52–8. doi: 10.1002/lary.27867
16. Goplen, FK, Grønning, M, Aasen, T, and Nordahl, SHG. Vestibular effects of diving — a 6-year prospective study. Occup Med (Lond). (2010) 60:43–8. doi: 10.1093/occmed/kqp148
17. Kobayashi, T, Sakurai, T, Okitsu, T, Yuasa, R, Kawase, T, Kusakari, J, et al. Labyrinthine fistulae caused by cholesteatoma. Improved bone conduction by treatment. Am J Otol. (1989) 10:5–10.
18. Herzog, JA, Smith, PG, Kletzker, GR, and Maxwell, KS. Management of labyrinthine fistulae secondary to cholesteatoma. Am J Otol. (1996) 17:410–5.
19. Lee, K, Ochi, N, Yamahara, K, Makino, K, and Ikezono, T. A case of Perilymphatic Fistula with inner ear anomaly diagnosed preoperatively by the Cochlin-Tomoprotein detection test. Case Rep Otolaryngol. (2020) 2020:1–4. doi: 10.1155/2020/9476915
20. Özüer, MZ, Olgun, L, and Gültekin, G. Revision stapes surgery. Otolaryngol Head Neck Surg. (2012) 146:109–13. doi: 10.1177/0194599811423523
21. Todt, I, and Ikezono, T. Objective assessment of Perilymphatic Fistula in cases of postoperative Vertigo after Cochlear implantation by Cochlin Tomoprotein (CTP). Brain Sci. (2023) 13:1525. doi: 10.3390/brainsci13111525
23. Sherbul, OV, Srebniak, IA, and Kuzmuk, IO. Fluctuating hearing loss as a symptom of acquired perilymphatic fistula (PLF) under external injuries factor. Wiad Lek. (2023) 76:1246–51. doi: 10.36740/WLek202305217
24. Science and Technology Efforts and Programs Relating to the Prevention, Mitigation, and Treatment of Blast Injuries . FY14 report to the executive agent: US Department of Defence, blast injury research coordinating office (2014).
25. Ahn, J, Son, SE, Choi, JE, Cho, YS, and Chung, WH. Surgical outcomes on hearing and vestibular symptoms in Barotraumatic Perilymphatic Fistula. Otol Neurotol. (2019) 40:e356–63. doi: 10.1097/MAO.0000000000002160
26. McNeill, C, Tonkin, J, and Newall, P. Testing for perilymphatic fistula based on Hennebert's sign and Tullio phenomenon. Aust J Otolaryngol. (1994) 1:422–5.
27. Elliott, EJ, and Smart, DR. The assessment and management of inner ear barotrauma in divers and recommendations for returning to diving. Diving Hyperb Med. (2014) 44:208–22.
28. Hermann, M, and Coelho, DH. Perilymph fistula presenting with contralateral symptoms. Otol Neurotol. (2014) 35:301–4. doi: 10.1097/MAO.0b013e3182a43639
29. Gibson, WP . Electrocochleography in the diagnosis of perilymphatic fistula: intraoperative observations and assessment of a new diagnostic office procedure. Am J Otol. (1992) 13:146–51.
30. Gutkovich, YE, Shlizerman, L, Paker, M, Mazzawi, S, Siag, K, and Shupak, A. Barotrauma-induced perilymph Fistula: video head impulse test and high-resolution temporal bones computed tomography role in evaluation and FollowUp. J Int Adv Otol. (2023) 19:350–4. doi: 10.5152/iao.2023.22771
31. Shepard, NT, Telian, SA, Niparko, JK, Kemink, JL, and Fujita, S. Platform pressure test in identification of perilymphatic fistula. Otol Neurotol. (1992) 13:49–54. doi: 10.1097/00129492-199201000-00012
32. Kurihara, YY, Fujikawa, A, Tachizawa, N, Takaya, M, Ikeda, H, and Starkey, J. Temporal bone trauma: typical CT and MRI appearances and important points for evaluation. Radiographics. (2020) 40:1148–62. doi: 10.1148/rg.2020190023
33. Bachmann-Harildstad, G, Stenklev, NC, Myrvoll, E, Jablonski, G, and Klingenberg, O. β-Trace protein as a diagnostic marker for perilymphatic fluid fistula: a prospective controlled pilot study to test a sample collection technique. Otol Neurotol. (2011) 32:7–10. doi: 10.1097/MAO.0b013e3181fc74d0
34. Levenson, MJ, Desloge, RB, and Parisier, SC. Beta-2 transferrin: limitations of use as a clinical marker for perilymph. Laryngoscope. (1996) 106:159–61. doi: 10.1097/00005537-199602000-00010
35. Bachmann-Harildstad, G, Müller, R, and Michel, O. Beta-trace protein in pediatric otitis media with effusion. Int J Pediatr Otorhinolaryngol. (2014) 78:659–62. doi: 10.1016/j.ijporl.2014.01.029
36. Ikezono, T, Shindo, S, Sekiguchi, S, Morizane, T, Pawankar, R, Watanabe, A, et al. The performance of Cochlin-tomoprotein detection test in the diagnosis of perilymphatic fistula. Audiol Neurootol. (2010) 15:168–74. doi: 10.1159/000241097
37. Ikezono, T, Matsumura, T, Matsuda, H, Shikaze, S, Saitoh, S, Shindo, S, et al. The diagnostic performance of a novel ELISA for human CTP (Cochlin-tomoprotein) to detect perilymph leakage. PLoS One. (2018) 13:1498. doi: 10.1371/journal.pone.0191498
38. Lou, Z, and Lou, Z. Surgical indications or inclusion/exclusion criteria of explorative tympanotomy on sudden sensorineural hearing. Am J Otolaryngol. (2018) 39:365–6. doi: 10.1016/j.amjoto.2018.01.015
39. Prenzler, NK, Schwab, B, Kaplan, DM, and El-Saied, S. The role of explorative tympanotomy in patients with sudden sensorineural hearing loss with and without perilymphatic fistula. Am J Otolaryngol. (2018) 39:46–9. doi: 10.1016/j.amjoto.2017.10.006
40. Nomura, Y . Perilymph Fistula: concept, diagnosis and management. Acta Otolaryngol Suppl. (1994) 114:52–4. doi: 10.3109/00016489409127559
41. Lee, JJ, Ryu, G, Moon, IJ, and Chung, WH. Two cases of Barotraumatic perilymph Fistula mimicking atypical benign paroxysmal positional Vertigo with sudden hearing loss. Res Vestib Sci. (2015) 14:26–31.
42. Tomasoni, M, Arcuri, M, Dohin, I, Zorzi, S, Borsetto, D, Piazza, C, et al. Presentation, management, and hearing outcomes of labyrinthine Fistula secondary to cholesteatoma: a systematic review and Meta-analysis. Otol Neurotol. (2022) 43:e1058–68. doi: 10.1097/MAO.0000000000003716
43. Comacchio, F, and Mion, M. Sneezing and Perilymphatic Fistula of the round window: case report and systematic review of the literature. J Int Adv Otol. (2018) 14:106–11. doi: 10.5152/iao.2018.4336
44. Leeflang, MM, and Bossuyt, PM. Defining the review question. In: Cochrane handbook for systematic reviews of diagnostic test accuracy version 20 (updated July 2023). Cochrane. (2023). Available at: https://training.cochrane.org/handbook-diagnostic-test-accuracy/current.
45. Deeks, JJ, Bossuyt, P, Leeflang, MM, and Takwoingi, Y. Cochrane handbook for systematic reviews of diagnostic test accuracy. (2010).
46. McInnes, MDF, Moher, D, Thombs, BD, McGrath, TA, Bossuyt, PM, Clifford, T, et al. Preferred reporting items for a systematic review and Meta-analysis of diagnostic test accuracy studies: the PRISMA-DTA statement. JAMA. (2018) 319:388–96. doi: 10.1001/jama.2017.19163
47. Moher, D, Shamseer, L, Clarke, M, Ghersi, D, Liberati, A, Petticrew, M, et al. Preferred reporting items for systematic review and meta-analysis protocols (PRISMA-P) 2015 statement. Syst Rev. (2015) 4:1. doi: 10.1186/2046-4053-4-1
48. Beynon, R, Leeflang, MM, McDonald, S, Eisinga, A, Mitchell, RL, Whiting, P, et al. Search strategies to identify diagnostic accuracy studies in MEDLINE and EMBASE. Cochrane Database Syst Rev. (2013) 2013:22. doi: 10.1002/14651858.MR000022.pub3
49. Whiting, PF, Rutjes, AW, Westwood, ME, Mallett, S, Deeks, JJ, Reitsma, JB, et al. QUADAS-2: a revised tool for the quality assessment of diagnostic accuracy studies. Ann Intern Med. (2011) 155:529–36. doi: 10.7326/0003-4819-155-8-201110180-00009
50. Cheung, A, Dufour, S, Jones, G, Kostoulas, P, Stevenson, MA, Singanallur, NB, et al. Bayesian latent class analysis when the reference test is imperfect. Rev Sci Tech. (2021) 40:271–86. doi: 10.20506/rst.40.1.3224
51. Balshem, H, Helfand, M, Schünemann, HJ, Oxman, AD, Kunz, R, Brozek, J, et al. GRADE guidelines: 3. Rating the quality of evidence. J Clin Epidemiol. (2011) 64:401–6. doi: 10.1016/j.jclinepi.2010.07.015
52. Suri, H . Ethical considerations of conducting systematic reviews in educational research In: O Zawacki-Richter, M Kerres, S Bedenlier, M Bond, and K Buntins, editors. Systematic reviews in educational research: Methodology, perspectives and application. Wiesbaden: Springer Fachmedien Wiesbaden (2020). 41–54.
Keywords: perilymphatic fistula, diagnostic test accuracy, systematic review, biomarkers, vestibular disorders
Citation: Situkho M, Ikezono T, Myhrum M, Rødvik AK, Maeda Y, Matsuda H, Sawada M and Jablonski GE (2024) Accuracy of diagnostic tests for perilymphatic fistula: protocol for a systematic review. Front. Neurol. 15:1500780. doi: 10.3389/fneur.2024.1500780
Edited by:
Andrea Castellucci, Azienda USL – IRCCS di Reggio Emilia, ItalyReviewed by:
Eduardo Martin-Sanz, Getafe University Hospital, SpainGerard Joseph Gianoli, The Ear and Balance Institute, United States
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*Correspondence: Maksym Situkho, maksym.situkho@medisin.uio.no