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PLOS One logoLink to PLOS One
. 2021 Feb 8;16(2):e0245796. doi: 10.1371/journal.pone.0245796

Traumatic dislocation of middle ear ossicles: A new computed tomography classification predicting hearing outcome

Georgios Mantokoudis 1,*, Njima Schläpfer 1, Manuel Kellinghaus 2, Arsany Hakim 2, Moritz von Werdt 1, Marco D Caversaccio 1, Franca Wagner 2
Editor: Rafael da Costa Monsanto3
PMCID: PMC7870152  PMID: 33556107

Abstract

Objectives

To assess the feasibility of radiologic measurements and find out whether hearing outcome could be predicted based on computer tomography (CT) scan evaluation in patients with temporal bone fractures and suspected ossicular joint dislocation.

Methods

We assessed 4002 temporal bone CT scans and identified 34 patients with reported ossicular joint dislocation due to trauma. We excluded those with no proven traumatic ossicular dislocation in CT scan and patients with bilateral temporal bone fractures. We measured four parameters such as malleus-incus axis distance, malleus-incus angle at midpoints, malleus- incus axis angle and ossicular joint space. The contralateral healthy side served as its own control. Hearing outcome 1–3 months after the index visit was analyzed. We assessed diagnostic accuracy and performed a logistic regression using radiologic measurement parameters for outcome prediction of conductive hearing loss (defined as >20dB air-bone gap).

Results

We found excellent inter-rater agreement on the measurement of axis deviation between incus and malleus in CT scans (interclass correlation coefficient 0.81). The larger the deviation of incus and malleus axis, the higher probability of poor hearing outcome (odds ratio (OR) 2.67 per 0.1mm, p = .006). A cut-off value for the axis deviation of 0.25mm showed a sensitivity of 0.778 and a specificity of 0.94 (p < .001) for discrimination between poor and good hearing outcome in terms of conductive hearing loss.

Conclusion

Adequate assessment of high resolution CT scans of temporal bone in which ossicular chain dislocation had occurred after trauma was feasible. Axis deviations of the incus and the malleus were strongly predictive for poor hearing outcome in terms of air conduction 1–3 months after trauma. We propose a 3-level classification system for hearing outcome prediction based on radiologic measures.

Introduction

Ossicular chain dislocation is often associated with a traumatic fracture of the temporal bone [13]. Often hearing dysfunction is overlooked in polytrauma patients because other trauma-related physical/brain injuries take medical priority. Immediate hearing assessment and outcome prediction in cases with suspected hearing loss is not possible because patients often have a traumatic head or brain injury and are not eligible for hearing tests in the acute stage. Even if they do not require bed rest, patients might still not be able to participate in audiometry since they might have cognitive impairment. Finally, temporal bone fractures are often associated with a hemotympanum, which makes an accurate early assessment of the middle ear impossible. Currently, patients with a suspected traumatic dislocation of the ossicular chain only undergo a comprehensive hearing test after some weeks or months, and the risk for loss to follow-up is high. Unilateral hearing loss (conductive or sensorineural) might therefore remain untreated in a large proportion of patients [4, 5]. Any technique to predict hearing outcome at the initial assessment might help to initiate early follow-up treatment with hearing aids, cochlear implants, or reconstructive middle ear surgery. Temporal bone computed tomography (CT) assessing the length of the fracture line sparing the otic capsule was reported to be useful for predicting sensorineural hearing loss; however, no prediction was made regarding conductive hearing loss due to ossicular chain disruption [6].

Transverse fractures often affect the labyrinth, the vestibular, and cochlear systems, as well as the facial nerve. Longitudinal fractures (sparing the otic capsule) commonly involve the external auditory canal, tympanic membrane, and the middle ear including the ossicular chain, which often results in conductive hearing loss [5, 7, 8]. Immediate surgery is only indicated in patients with primary facial nerve palsy due to traumatic neurotmesis or in patients with labyrinthine fistula (e.g. rupture of round window membrane) leading to perilymph loss and deafness [1, 9]. Cerebrospinal fluid leaks might also need early surgical intervention if conservative treatment fails. Spontaneous recovery of traumatic conductive hearing loss is reported in 77% with conservative treatment [4]. Reconstructive middle ear surgery is indicated as a second stage elective procedure in patients with persistent conductive hearing loss and/or traumatic rupture of the tympanic membrane [1, 2]. The prognosis for hearing after middle ear surgery is excellent [2, 1012].

We sought to perform a retrospective analysis of patients with a radiologically suspected ossicular chain dislocation and to assess the radiologic CT parameters and their association with conductive hearing outcome.

Material and methods

Patient population

This retrospective single-center cohort study included patients admitted to the emergency department (ED) with an ossicular injury caused by trauma and a fully documented audiometric examination at the time of the traumatic event in the period January 2010 to December 2017. All patients underwent a CT scan of the head as part of our standard emergency procedure for trauma patients.

In a primary screening of the radiological database, head trauma cases including petrous bone injury were identified using key words like “ossicular dislocation” “ossicular dehiscence” “petrous bone” by an experienced head and neck neuroradiologist (FW). In the second-stage screening conducted by a medical student (NS), only patients with a reported or suspected traumatic ossicular chain dislocation were included. All images were reviewed and assessed by 2 blinded neuroradiologists; one was a very experienced head and neck neuroradiologist (FW) and the other was a neuroradiology trainee (MK).

We excluded patients whose scans showed no proven dislocation of the ossicular chain in CT or had no head trauma. We further excluded patients with bilateral temporal bone fracture since the contralateral healthy side served as its own control. S1 Fig in S1 Appendix shows how patients were selected for the analysis.

Radiological assessment: Parameters

All CT examinations were performed with the patient in a supine position using a 128-slice CT scanner (SOMATOM® Definition Edge; Siemens Healthcare, Erlangen, Germany). A certified reporting workstation (Sectra IDS7, Linköping, Sweden) was used for evaluation by the 2 neuroradiologists, who were blinded to outcomes. Slight motion artifacts were considered acceptable. All the images collected were of sufficiently good quality to allow an accurate assessment of the middle ear.

Overall, 4002 CT scans of trauma patients were retrospectively reviewed and, of these, 34 patients with ossicular joint dislocation due to trauma were included in the study.

Image reconstruction according to our standard in-house trauma protocol included a soft-tissue window (kernel J45s) and a bone window (kernel J70h) of the acquired CT scan of the head; each in the axial, coronal, and sagittal plane. For our retrospective data analysis, we additionally reconstructed the CT scans in 3-D to provide a different view of the ossicular chain anomalies in our trauma cohort. Both, 2-D and 3-D views have been used for the measurements [13].

The acquisition parameters of our standard trauma CT scan of the head were: slice thickness 1.0 mm, matrix 512 × 512, field of view 200 mm, total acquisition time of 1 second by tube current-time product of 240 mA, and tube voltage 100 kV. This resulted on average in a computed tomography dose index of 35 mGy and a dose-length product of 650 mGycm.

Assessment of the presence of an ossicle fracture was followed by the evaluation of ossicle dislocation or luxation based on visual identification of a discernable gap between ossicles. The distance ‘D’ between malleus and incus was measured by drawing a virtual line between the long axis of the incus and the middle of the head of the malleus, and the offset of the incus in medial or lateral deviation measured in mm were reported (Fig 1). The presence or absence of luxation and/or dislocation of the ossicles was recorded as: none, incudostapedial, incudomalleolar, stapedo-vestibular, or complex if there was a luxation or dislocation in more than one direction (dislocation of several axis).

Fig 1. Radiologic measurement parameters.

Fig 1

Definition of all continuous variables and radiologically measured parameters.

The degree of the offset between malleus and incus dislocation was separately measured, and the malleus–incus axis angle ‘α’ measured at midpoints (degrees) and the malleus–incus axis angle ‘β’ (degrees) were documented (Fig 1). Furthermore, we measured the ossicular joint space ‘d’ in mm in the soft tissue and bone window to determine whether the appearance of the space was normal or was filled with hemorrhage or air, based on standard Hounsfield Units. Fig 1 shows the definitions of all continuous variables (measurement of distances and angles).

Audiometric testing

We assessed hearing outcome 1–3 months after the index ED visit and after resorption of any hemotympanon confirmed by tympanometry and otoscopy. Mixed hearing loss was not an exclusion criterion, however, sensorineural hearing loss due to labyrinthine concussion was not assessed since it was not considered a primary or secondary endpoint in this study. We used data from pure tone audiometry with air and bone conduction. The pure tone average including frequencies of 500Hz, 1000Hz, 2000Hz, and 3000Hz was calculated following the guidelines of the American Academy of Otolaryngology Committee on Hearing and Equilibrium [14]. An average air-bone gap <20 dB was considered a good hearing outcome.

Statistics

We used SPSS (IBM Version 25) for statistical analysis. We calculated the inter-rater agreement for each variable, Cohen’s kappa for categorical variables and the intraclass correlation coefficient for continuous data. A logistic regression was applied for the assessment of independent variables that might determine a poor hearing outcome with an air-bone gap of ≥20dB. Hearing outcome was coded as a binary, dependent variable.

A receiver operator characteristics (ROC) curve was constructed using the radiological variables. We assessed the diagnostic accuracy and calculated the optimal cut-point (Youden’s index) for the discrimination of poor hearing outcome. Normative data from the contralateral healthy ear (2 standard deviations from the mean) and data from the ROC curve served as a basis for the classification of traumatic ossicular chain luxations.

Ethics

The institutional review board and the local ethics committee (Kantonale Ethikkomission des Kantons Bern, Schweiz) gave approval for the access to and use of the data collected with the intention of using it for retrospective clinical research. Data have been fully anonymized. Informed consent was waived by the ethics committee.

Results

We included 34 patients (12 females) with a mean age of 44.7 years (SD 24.9), 20 with a dislocation of the incudomalleolar joint, 1 with involvement of the incudostapedial joint, 4 with a joint distension (malleus head still within the facet of the incus without axis deviation but expanded joint space), and 9 with a complex dislocation involving all 3 ossicles. All patients had a history of head trauma, 56% reported a fall from height, 27% were involved in a traffic accident, 11% had a blunt head trauma and 6% had an unknown trauma mechanism. Fifteen of the 34 patients had a severe brain injury with a Glasgow Coma Scale (GCS) score of 3–8, 5 patients had a moderate (GCS 9–12), and 6 patients a minor brain injury (GCS 13–15). The state of consciousness remained unknown for 8 patients. The main complaints at the index ED visit were dizziness (6 out of 34) and hearing loss (7) followed by nausea (3), vomiting (3), headache (3), facial palsy (2), otorrhea (2), and gait disturbance (1).

One-third of the patients had facial nerve palsy (12 out of 34). Twenty-two patients suffered from bloody ear discharge, 10 had a hemotympanon, and 1 patient a perforation of the tympanic membrane; however, only 7 patients complained of hearing loss. Nine of the 22 patients who underwent follow-up hearing tests 1–3 months after the initial trauma had a hearing loss ≥20dB (S1 Fig in S1 Appendix).

Radiological examination revealed that 25 patients had a longitudinal, 1 patient a transverse, 7 patients a mixed/complex temporal bone fracture. One trauma patient had no temporal bone fracture, another patient showed a fracture of the incus.

Inter-rater agreement on the radiological parameters for the assessment of ossicular chain luxations was excellent (see S1 Table in S1 Appendix), except regarding the categorical assessment of the incus axis deviation. Normative data for the incudomalleolar joint are shown in the Appendix (S2 Table in S1 Appendix).

The distance ‘D’ [mm] measured between the 2 axes from malleus and incus (Fig 1) was the most significant factor for predicting poor hearing outcome (air bone gap ≥20dB). The probability of poor hearing outcome increased if the measured deviation of incus and malleus axis increased (odds ratio (OR) 2.67 per 0.1mm, 95% CI, 1.32–5.41, p = .006, Table 1).

Table 1. Logistic regression for hearing outcome ≥20dB.

Parameter Odds Ratio Lower 95% CI Upper 95% CI P Value
Malleus-incus axis distance ‘D’ (per 1/10mm increase) 2.674 1.32 5.416 .006
Malleus-incus axis angle measured at midpoints ‘α’ (per degree increase) 1.781 1.056 3.003 .03
Malleus-incus axis angle ‘β’ (per degree increase) 1.033 0.979 1.09 .232
Ossicle joint space ‘d’ (per 1/10mm increase) 1.374 0.948 1.991 .094

The angle ‘α’ and the distance ‘D’ between the 2 measured axes also had the highest sensitivity and specificity for predicting a poor hearing outcome (Table 2). A cut-off axis distance of 0.25mm showed a sensitivity of 0.778 and a specificity of 0.94 (p < .001) for discrimination between poor and good hearing outcome in terms of conductive hearing loss. The axis angle measured at the midpoints ‘α’ was also a significant predictor (OR 1.78, 95% CI, 1.05–3.00, p = .03) with a high sensitivity (0.89, p = .002) but lower specificity (0.743).

Table 2. ROC (receiver operator characteristics) curves.

Parameter Cut-off Sensitivity Specificity AUC P Value
Malleus-incus axis distance ‘D’ (mm) 0.250 0.778 0.943 0.892 < .001
Malleus-incus angle measured at midpoints ‘α’ (deg) 0.950 0.889 0.743 0.840 .002
Malleus-incus axis angle ‘β’ (deg) 93.8 0.222 1.000 0.524 .827
Ossicle joint space ‘d’(mm) 0.850 0.500 0.829 0.664 .203

Abbreviations: AUC, area under the curve.

Fig 2 shows a receiver operator characteristics (ROC) curve with all the applied radiological parameters.

Fig 2. Receiver operator characteristics (ROC) curve.

Fig 2

ROC curve for all 4 radiological parameters. The best predictive parameter was the distance between the 2 axes [mm], which yielded a curve bending toward the left top corner. Curves along the diagonal line, such as the parameter “malleus-incus axis angle ‘β’” reflect a random guess and are not discriminative with respect to hearing outcome.

Table 3 shows our new 3-grade classification for the prediction of normal or poor hearing outcome based on the distance between incus and malleus axis. Grade I represents the normal, non-displaced axis distance of the incudomalleolar joint (Fig 3A, normative Data). Grade II includes cases with an anatomical axis configuration in the pathological range, but whose predicted hearing outcome is good (Fig 3B). Patients with grade III axis distance have a poorer prognosis for hearing outcome due to the abnormal incudomalleolar joint dislocation (Fig 3C). The discrimination cut-off between Grade II and Grade III was derived from the ROC analysis (Table 2).

Table 3. New classification system of ossicular chain dislocation.

Grade Malleus-incus axis distance ‘D’ (mm) Dislocation/subluxation Hearing outcomea Example
I 0–0.07 No Normal Fig 2A
II 0.08–0.25 Yes Normal Fig 2B
III >0.25 Yes Poorb Fig 2C

a1-3 months after trauma

bPTA air bone gap >20dB HL

Fig 3. Computed tomography examples of malleus-incus axis distance ‘D’.

Fig 3

Panel A, B and C shows a computed tomography (at index visit) of the incudomalleolar joint from 3 patients and the distances ‘D’ (mm) between the malleus axis and incus axis for grade I, II and III and the corresponding hearing outcome (pure tone average (PTA) of the air bone gap), dB HL) 1–3 months after trauma. The image in panel C was horizontally flipped for better visualization.

Discussion

Almost half of the patients suffered from conductive hearing loss 1–3 months after temporal bone fracture with associated luxation/dislocation of middle ear ossicles. High resolution temporal bone CTs with narrow slice widths allowed accurate radiologic measurements with high inter-rater agreement. The distance ‘D’ between the 2 axes through the short process of the incus and the midpoint of the malleus was an accurate predictor for hearing outcome. Each deviation difference of 1/10mm increased the odds for a poor predicted hearing outcome by 2.6. A cut-off value of 0.25mm had a statistically significant high sensitivity and specificity for discriminating poor from normal hearing outcome and served as the threshold for a new proposed classification.

The most frequent trauma mechanism was falls from height followed by traffic accidents. A rapid deceleration or high energetic trauma was the cause of temporal bone fractures with consecutive injury to the ossicular chain. This result is congruent with that of a recent study by Delrue et al. [11], but in most studies the main cause of injury was a traffic accident [2, 4, 7, 10].

There was a high proportion of longitudinal fractures in our cohort. The classical classification of temporal bone fractures into longitudinal, transverse, or mixed fractures, however, has a poor correlation with clinical symptoms [15, 16]. Therefore, new classifications have been proposed taking into account the involvement of the otic capsule [15], the petrous bone [17], or 4 parts of the temporal bone (squama, tympanic, mastoid, and petrous) [18]. These studies showed that only the categorization into petrous or non-petrous fractures was significantly associated with the clinical symptoms [1719]. Since we only included patients with petrous bone involvement, we decided to use the traditional classification, according to which the exact description of the fracture course and the affected regions is essential.

Ossicular chain dislocation or luxation is a frequent complication of temporal bone fracture. In this study, incudomalleolar dislocation was by far the most common type, as has been reported in other CT-based studies [20, 21]. However, surgical explorations or cadaveric dissections revealed frequent involvement of the incudostapedial joint [7, 8, 10, 22, 23]. This is plausible because dislocation of the incus, which is embedded between the malleus and stapes, should affect both joints. Another plausible explanation for the lower prevalence of the often subtle incudostapedial dislocation in our CT-based study might be the initial hemotympanum, which makes an accurate radiologic assessment difficult, especially in early trauma diagnostics and for slight dislocations [20, 24].

Our study data suggest that a latero-medial dislocation of the incus is more likely than a longitudinal traction. A lateral displacement changes the axis angle and axis distance, whereas traction leads to a widening of the joint space. The configuration of the ligamentous apparatus of the middle ear ossicles supports the observed mechanism of dislocation and the vulnerability of the incus. While the malleus is attached by 3 ligament folds (anterior, lateral, and posterior), the incus is only kept in place by a strong posterior incudal ligament fold, which is attached at the apex of the short incus process [25] (S2 Fig in S1 Appendix). The lateral incudomalleal fold, however, is purely membranous and is probably not sufficient to protect against shearing forces. In addition, the stapes and malleus are attached to the middle ear muscles, the tensor tympani muscle, and the stapedial muscle, resulting in stronger stabilization of these 2 ossicles. A dislocation of the malleus is less likely, since it is additionally attached to the fibrous layers of the tympanic membrane [7, 26, 27]. A large proportion of dislocated incudomalleolar joints showed a pattern of subluxation, but nevertheless led to a satisfactory hearing outcome in the long term. Reversible subluxations may only stretch the ligamentous apparatus. Possible spontaneous resolution of ossicular chain dislocations has been described by other authors [4, 28]. Such luxations were classified as grade II (Table 3).

Strengths and limitations

To our knowledge, this is the first study to propose a grading system for predicting conductive hearing outcome based on CT in patients with traumatic dislocations of the middle ear ossicles. This classification of hearing outcome prediction is limited to patients with conductive or mixed hearing loss after trauma. Sensorineural hearing loss due to labyrinthine concussion is not covered; however, sensation levels might be screened by the Weber tuning fork test at the bedside (lateralization toward the affected middle ear or the healthy inner ear) or assessed by bone conduction hearing tests. Patients classified as grade II had a pathological axis deviation in the index CT but still had normal hearing after 1–3 months; however, no follow-up imaging or surgical reports were available to confirm recovery in terms of anatomy.

CT spatial resolution was limited to 0.2–0.3mm, however, distance measurements close to the resolution limits were not performed between anatomical structures but were restricted to distances between two marked axis. Some calculated mean distances in our classification table were smaller than the spatial CT resolution which is the result from statistical analysis including values with zero millimeters (no dislocation). Further studies using the proposed classification system and its radiologic measuring techniques are necessary to detect possible technical limitations regarding the measurements. Overall, the inter-rater agreement was excellent indicating a reproducible and accurate radiologic measurement, which was also found by Maillot et al, provided that assessments were done by senior readers and taking into account 3D CT reconstructions [13].

Finally, luxations of the incudostapedial joint were not measured quantitatively due to the low frequency of occurrence in our cohort; however, qualitative radiological assessment was still possible. It is unclear whether such isolated joint luxations remain limited to the incudostapedial joint or—more likely—affect the entire ossicular chain.

Potential implications

This proposal for a grading system is a first attempt to classify patients according to whether a normal or poor hearing outcome is likely after temporal bone fractures. Future prospective studies are essential for its validation. Prospective studies with hearing assessments after resorption of the hemotympanon, long-term hearing results and intraoperative findings might give more insights about the mechanism and morphology of dislocations. Information about prognosis and encouragement might improve the follow-up of these patients.

Conclusions

Assessment of high-resolution CT scans of temporal bone with respect to ossicular chain dislocations after traumatic temporal bone fractures was feasible. Axis distance ‘D’ of the short incus process and the middle point of the malleus body were strongly predictive for hearing outcome in terms of air conduction 1–3 months after trauma. We propose a new 3-level classification system for the prediction of poor or normal hearing outcome in patients with ossicular chain dislocation caused by trauma based on radiologic measures.

Supporting information

S1 Appendix

(PDF)

Data Availability

The data underlying this study are available on the Bern Open Repository (https://boris.unibe.ch/147873/).

Funding Statement

G.M. received funding from the Swiss National Science Foundation (#320030_173081). All other authors received no external financial support for that study. The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.

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Decision Letter 0

Rafael da Costa Monsanto

13 Oct 2020

PONE-D-20-27974

Traumatic Dislocation of Middle Ear Ossicles: A New Computed Tomography Classification Predicting Hearing Outcome

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Although the reviewers considered the study well-written and scientifically sound, there are still some issues that need to be addressed before the manuscript is further considered for publications. The most critical concerns were regarding description of the methodology and results, especially concerning to patient selection and analysis of the results.

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Reviewers' comments:

Reviewer's Responses to Questions

Comments to the Author

1. Is the manuscript technically sound, and do the data support the conclusions?

The manuscript must describe a technically sound piece of scientific research with data that supports the conclusions. Experiments must have been conducted rigorously, with appropriate controls, replication, and sample sizes. The conclusions must be drawn appropriately based on the data presented.

Reviewer #1: Yes

Reviewer #2: Yes

Reviewer #3: Yes

**********

2. Has the statistical analysis been performed appropriately and rigorously?

Reviewer #1: Yes

Reviewer #2: Yes

Reviewer #3: Yes

**********

3. Have the authors made all data underlying the findings in their manuscript fully available?

The PLOS Data policy requires authors to make all data underlying the findings described in their manuscript fully available without restriction, with rare exception (please refer to the Data Availability Statement in the manuscript PDF file). The data should be provided as part of the manuscript or its supporting information, or deposited to a public repository. For example, in addition to summary statistics, the data points behind means, medians and variance measures should be available. If there are restrictions on publicly sharing data—e.g. participant privacy or use of data from a third party—those must be specified.

Reviewer #1: Yes

Reviewer #2: Yes

Reviewer #3: Yes

**********

4. Is the manuscript presented in an intelligible fashion and written in standard English?

PLOS ONE does not copyedit accepted manuscripts, so the language in submitted articles must be clear, correct, and unambiguous. Any typographical or grammatical errors should be corrected at revision, so please note any specific errors here.

Reviewer #1: Yes

Reviewer #2: Yes

Reviewer #3: Yes

**********

5. Review Comments to the Author

Please use the space provided to explain your answers to the questions above. You may also include additional comments for the author, including concerns about dual publication, research ethics, or publication ethics. (Please upload your review as an attachment if it exceeds 20,000 characters)

Reviewer #1: I congratulate the authors for this interesting paper.

How unfortunate that the study managed to analyze only 34 exams in a universe of more than 4000 CT scans. Anyway, the authors managed to adequately point out the limitations of the study and I believe that the idea should follow so that other studies use the same method to validate the tomographic correlation with the potential to predict the audiometric results.

Reviewer #2: This is a review of the temporal bone CT scans of patients with a radiologically suspectedossicular chain dislocation and their association with conductive hearing outcome. It states that Figure S1 in the Appendix shows how patients were included in the analysis, although I failed to find this figure in my copy of the manuscript. The severity of incudomalleal joint disruption was assessed with 1 mm slices of standard trauma CT scan of the head. Aside from the inter rater agreement, logistic regression was used to assess the association between 4 CT scan parameters with poor hearing outcome defined as conductive hearing loss of ≥20dB air-bone gap. The authors then found that the malleus-incus axis distance and malleus-incus angle measured at midpoints were statistically correlated with conductive hearing loss.

Here are some of my questions:

1. Was the patient selection process blinded to outcomes? Who did the selection, how was this done and how were disagreements to include patients addressed?

2. Were the 2 neuroradialogists blinded to outcomes?

3. I am concerned that the distance of 0.25 mm was found significant when the cuts were 1 mm in thickness. The “ice cream cone” constituting the incus and the malleal head appears discernible enough from the sample CT scan picture but how were differences in measurements settled? And how high were the inter rater agreements in each of the 4 radiologic parameters? (I did not find Appendix Table S1 in my copy).

4. I am not clear as to how the authors determined the ranges of the malleus-incus axis distances of 0-0.07, 0.08-0.25 and >0.25 for each category. I may have missed how they have correlated these ranges with normal or abnormal hearing.

I appreciate how the discussion attempts to explain the results in terms of the ligamentous attachments of the malleus and incus may explain the vulnerability of the incudomalleal joint to trauma. An illustration may more clearly drive home the point.

I also appreciate the limitations part and agree with all the points raised by the authors, particularly with their statement that “some calculated mean distances in our classification table were smaller than the spatial CT resolution which is the result from statistical analysis”. Given this potential technical difficulty in measurement, I think they should point out that their new classification system should be applied into another set of trauma patients to understand how well it performs.

Reviewer #3: The authors perform a study to determine the association between CT findings of post-traumatic ossicular injuries and conductive hearing outcome. They also propose a classification system to predict audiometric outcomes based on radiological measurements. Although this is an interesting area of research to explore, there are some major concerns that need to be addressed, as listed below:

1. The abstract should be revised to include more detail about methodology, such as summary of inclusion/exclusion criteria, measurements taken (e.g. incus-malleus axis distance, etc) and how good/poor hearing was defined by the authors. “CT” abbreviation should be provided in the Objectives.

2. Also, in the abstract — The authors write in the conclusion: “Adequate assessment of high resolution CT scans of temporal bone in which ossicular chain dislocation had occurred after traumatic fractures of temporal bone was feasible.” — did you mean your new radiological parameters (measurements) for assessment were feasible and reproducible? If the authors also wanted to assess their feasibility, it should be included in the aims (objectives).

3. Lines 80-83: Patients should also acknowledge the fact that hearing dysfunction is often overlooked in polytrauma patients because other trauma-related physical/brain injuries that take medical priority.

4. Lines 84: fractures are OFTEN associated with a hemotympanum.

5. Material and Methods: Inclusion criteria is confusing — were considered cases with ossicular chain injury following head trauma regardless of presence of TBF? Or was presence of TBF determinant for study inclusion? I also suggest providing # of excluded cases within each criterion in Figure 1S (no dislocation of the ossicular chain, no temporal fracture after second review, or who had a bilateral temporal bone fracture).

6. Line 152: What does “more than one direction” mean? More than one joint? Please clarify.

7. Did any patient have a history of asymmetric hearing loss or ear disease prior to the trauma?

8. Line 208, 217 — Keep consistency in terminology while reporting hearing loss (average air-bone gap vs. conductive hearing loss) throughout the Results.

9. Audiometric data timeline considered for inclusion in the study needs clarification. Did all patients have audiometric evaluation? Did the authors include audiometric data before 1 mo in addition to those performed 1-3 months after trauma? In the Results, Line 207-208: “Nine of the 22 patients who underwent follow-up hearing tests still had a hearing loss ≥20dB 1-3 months after the initial trauma” gives the impression that audiometric data before 1mo were also reviewed. Please clarify.

10. Line 109: Figure 1 does not relate to the sentence/paragraph.

11. Line 164-173: It is not clear if cases with mixed hearing loss were also included. Please specify.

12. Line 197: “4 with a joint distension” — Please clarify.

13. Line 210-211: The total accounts for 33 cases. However, authors report a total of 34 cases in this study. Similar to my question #5 — were cases without TBF included? Please clarify.

14. Keep consistency when reporting measurements in the main manuscript, table and figures (e.g. Incus-malleus or malleus-incus). Also, consider including symbols (D, d, α and β) in the tables.

15. Line 225: Table 2 indicates that D and α had the highest sensitivity and specificity for predicting a poor hearing outcome instead. Please review/clarify.

16. In Methods, authors mention to have performed 3D-CT reconstructions to provide different views of the ossicular chain anomalies — Were both 2D-CT axial and sagittal views and 3D-CT reconstructions used for measurements? It should be clarified in the Methods. Also, were there any ossicle fractures identified in your cohort?

17. I suggest including the recent publication from Maillot et al. (2020) — PMID: 28551022 — related to this topic to enrich the discussion.

**********

6. PLOS authors have the option to publish the peer review history of their article (what does this mean?). If published, this will include your full peer review and any attached files.

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Do you want your identity to be public for this peer review? For information about this choice, including consent withdrawal, please see our Privacy Policy.

Reviewer #1: No

Reviewer #2: Yes: Jose Acuin

Reviewer #3: No

[NOTE: If reviewer comments were submitted as an attachment file, they will be attached to this email and accessible via the submission site. Please log into your account, locate the manuscript record, and check for the action link "View Attachments". If this link does not appear, there are no attachment files.]

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PLoS One. 2021 Feb 8;16(2):e0245796. doi: 10.1371/journal.pone.0245796.r002

Author response to Decision Letter 0


26 Nov 2020

Reviewer #1:

I congratulate the authors for this interesting paper.

How unfortunate that the study managed to analyze only 34 exams in a universe of more than 4000 CT scans. Anyway, the authors managed to adequately point out the limitations of the study and I believe that the idea should follow so that other studies use the same method to validate the tomographic correlation with the potential to predict the audiometric results.

AUTHOR REPLY: We would like to thank the reviewer for the thorough review of the manuscript, the positive feedback and the appreciation of the work.

Reviewer #2:

This is a review of the temporal bone CT scans of patients with a radiologically suspected ossicular chain dislocation and their association with conductive hearing outcome. It states that Figure S1 in the Appendix shows how patients were included in the analysis, although I failed to find this figure in my copy of the manuscript. The severity of incudomalleal joint disruption was assessed with 1 mm slices of standard trauma CT scan of the head. Aside from the inter rater agreement, logistic regression was used to assess the association between 4 CT scan parameters with poor hearing outcome defined as conductive hearing loss of ≥20dB air-bone gap. The authors then found that the malleus-incus axis distance and malleus-incus angle measured at midpoints were statistically correlated with conductive hearing loss.

Here are some of my questions:

1. Was the patient selection process blinded to outcomes? Who did the selection, how was this done and how were disagreements to include patients addressed?

AUTHOR REPLY: Thank you for raising this important question. In a primary screening the radiological database has been searched for key words like “ossicular dislocation” “ossicular dehiscence” “petrous bone” by a experienced head and neck neuroradiologist (FW). In the second-stage screening, all reports of the 4002 CT scans were screened by a medical student (NS) for reported or suspected ossicular chain dislocation associated with trauma. All images were then again reviewed and assessed by 2 blinded neuroradiologists to determine whether a dislocation actually was present or not. Neurologist were blinded to outcomes. We have added this specification to “Material and Methods: Patient Population.

2. Were the 2 neuroradialogists blinded to outcomes?

AUTHOR REPLY: Both neuroradiologists were blinded to outcomes as stated in Material and Methods under the section “Patient population”.

REVISED: “A certified reporting workstation (Sectra IDS7, Linköping, Sweden) was used for evaluation by the 2 neuroradiologists, who were blinded to outcomes.”

3. I am concerned that the distance of 0.25 mm was found significant when the cuts were 1 mm in thickness. The “ice cream cone” constituting the incus and the malleal head appears discernible enough from the sample CT scan picture but how were differences in measurements settled? And how high were the inter rater agreements in each of the 4 radiologic parameters? (I did not find Appendix Table S1 in my copy).

AUTHOR REPLY: Please see appendix table 1S attached

REVISED: Limitations section: Some calculated mean distances in our classification table were smaller than the spatial CT resolution which is the result from statistical analysis including values with zero millimeters (no dislocation).

4. I am not clear as to how the authors determined the ranges of the malleus-incus axis distances of 0-0.07, 0.08-0.25 and >0.25 for each category. I may have missed how they have correlated these ranges with normal or abnormal hearing.¨

AUTHOR REPLY: Grade I was determined by the mean range of the contralesional, healthy side (normative data, mean + two standard deviations = 0.07). For the determination of the cut-off between grad 2 and 3, we used the ROC analysis and calculated the best discrimination cut-off (0.25). We added this information in the manuscript to make it more clear.

REVISED: “Grade I represents the normal, non-displaced axis distance of the incudomalleolar joint (Figure 3A, normative Data).”

“The discrimination cut-off between Grade II and Grade III was derived from the ROC analysis (Table 2).”

I appreciate how the discussion attempts to explain the results in terms of the ligamentous attachments of the malleus and incus may explain the vulnerability of the incudomalleal joint to trauma. An illustration may more clearly drive home the point.

AUTHOR REPLY: We added an additional figure in the appendix illustrating the incudal ligament folds (figure S2).

I also appreciate the limitations part and agree with all the points raised by the authors, particularly with their statement that “some calculated mean distances in our classification table were smaller than the spatial CT resolution which is the result from statistical analysis”. Given this potential technical difficulty in measurement, I think they should point out that their new classification system should be applied into another set of trauma patients to understand how well it performs.

AUTHOR REPLY: We agree with the reviewer that the new classification should be applied to other trauma patients.

REVISED: “Further studies using the proposed classification system and its radiologic measuring techniques are necessary to detect possible technical limitations regarding the measurements.”

Reviewer #3:

The authors perform a study to determine the association between CT findings of post-traumatic ossicular injuries and conductive hearing outcome. They also propose a classification system to predict audiometric outcomes based on radiological measurements. Although this is an interesting area of research to explore, there are some major concerns that need to be addressed, as listed below:

1. The abstract should be revised to include more detail about methodology, such as summary of inclusion/exclusion criteria, measurements taken (e.g. incus-malleus axis distance, etc) and how good/poor hearing was defined by the authors. “CT” abbreviation should be provided in the Objectives.

AUTHOR REPLY: Thank you for raising these important points. We revised the abstract and added inclusion/exclusion criteria. “CT” abbreviation provided. Radiologic measurements and hearing outcome defined.

2. Also, in the abstract — The authors write in the conclusion: “Adequate assessment of high resolution CT scans of temporal bone in which ossicular chain dislocation had occurred after traumatic fractures of temporal bone was feasible.” — did you mean your new radiological parameters (measurements) for assessment were feasible and reproducible? If the authors also wanted to assess their feasibility, it should be included in the aims (objectives).

AUTHOR REPLY: We assessed also the feasibility of radiological measurements and added this to the objectives in the abstract.

REVISED: “To assess the feasibility of radiologic measurements and find out whether hearing outcome could be predicted based on computer tomography (CT) scan evaluation in patients with temporal bone fractures and suspected ossicular joint dislocation.”

3. Lines 80-83: Patients should also acknowledge the fact that hearing dysfunction is often overlooked in polytrauma patients because other trauma-related physical/brain injuries that take medical priority.

AUTHOR REPLY: Corrected. We integrated this important fact in the introduction.

REVISED: “Often hearing dysfunction is overlooked in polytrauma patients because other trauma-related physical/brain injuries take medical priority.”

4. Lines 84: fractures are OFTEN associated with a hemotympanum.

AUTHOR REPLY: corrected

5. Material and Methods: Inclusion criteria is confusing — were considered cases with ossicular chain injury following head trauma regardless of presence of TBF? Or was presence of TBF determinant for study inclusion? I also suggest providing # of excluded cases within each criterion in Figure 1S (no dislocation of the ossicular chain, no temporal fracture after second review, or who had a bilateral temporal bone fracture).

AUTHOR REPLY: We agree that exclusion criteria were not clearly stated. We rephrased this sentence. Yes, we included cases with ossicular chain injury following any head trauma regardless of presence of TBF. However, we did not found any bilateral temporal bone fractures with associated bilateral ossicular dislocation (n=0). We corrected Figure S1.

REVISED: “We excluded patients whose scans showed no proven dislocation of the ossicular chain in CT or had no head trauma. We further excluded patients with bilateral temporal bone fracture since the contralateral healthy side served as its own control.”

6. Line 152: What does “more than one direction” mean? More than one joint? Please clarify.

AUTHOR REPLY: “more than one direction” means a dislocation in several planes (axes).

REVISED: or complex if there was a luxation or dislocation in more than one direction (dislocation of several axis).

7. Did any patient have a history of asymmetric hearing loss or ear disease prior to the trauma?

AUTHOR REPLY: These patients had no audiological assessment prior to the trauma, however, any preexisting asymmetric hearing cannot be excluded due to the retrospective nature of our study.

8. Line 208, 217 — Keep consistency in terminology while reporting hearing loss (average air-bone gap vs. conductive hearing loss) throughout the Results.

AUTHOR REPLY: corrected

9. Audiometric data timeline considered for inclusion in the study needs clarification. Did all patients have audiometric evaluation? Did the authors include audiometric data before 1 mo in addition to those performed 1-3 months after trauma? In the Results, Line 207-208: “Nine of the 22 patients who underwent follow-up hearing tests still had a hearing loss ≥20dB 1-3 months after the initial trauma” gives the impression that audiometric data before 1mo were also reviewed. Please clarify.

AUTHOR REPLY: We did not review audiologic data in the time period from trauma until 1 month after trauma. We rephrased this sentence.

10. Line 109: Figure 1 does not relate to the sentence/paragraph.

AUTHOR REPLY: We agree. This relates to figure S1 in the appendix.

11. Line 164-173: It is not clear if cases with mixed hearing loss were also included. Please specify.

AUTHOR REPLY: Thank you for pointing out this ambiguity. Mixed hearing loss was not an exclusion criterion. The study focuses on the ABG, whereas sensorineural hearing loss due to labyrinthine concussion was not assessed since it was not considered a primary or secondary endpoint in this study. We rephrased this in the methods section. We made it more clear in the methods section.

12. Line 197: “4 with a joint distension” — Please clarify.

AUTHOR REPLY: Joint distension means, that the malleus head was still within in the facet of the incus without axis deviation but the joint space was expanded. We revised accordingly.

13. Line 210-211: The total accounts for 33 cases. However, authors report a total of 34 cases in this study. Similar to my question #5 — were cases without TBF included? Please clarify.

AUTHOR REPLY: One trauma patient had no TBF. We added this in line 211 and clarified in the methods section.

14. Keep consistency when reporting measurements in the main manuscript, tables and figures (e.g. Incus-malleus or malleus-incus). Also, consider including symbols (D, d, α and β) in the tables.

AUTHOR REPLY: Thank you for pointing out these inconsistencies. In the revised manuscript the term “malleus-incus” is now used consistently and we included the symbols (D, d, α and β) consistently throughout the whole manuscript, including tables, figures and the appendix.

15. Line 225: Table 2 indicates that D and α had the highest sensitivity and specificity for predicting a poor hearing outcome instead. Please review/clarify.

AUTHOR REPLY: Corrected. It should be ‘D’ and ‘α’». We added labels ‘D’, ‘α’, ‘β’ and ‘d’ to all tables.

16. In Methods, authors mention to have performed 3D-CT reconstructions to provide different views of the ossicular chain anomalies — Were both 2D-CT axial and sagittal views and 3D-CT reconstructions used for measurements? It should be clarified in the Methods. Also, were there any ossicle fractures identified in your cohort?

AUTHOR REPLY: We used both, 2D and 3D views for the measurements. We clarified this in the methods. One patient had a fracture of the incus. This was added in the results section.

17. I suggest including the recent publication from Maillot et al. (2020) — PMID: 28551022 — related to this topic to enrich the discussion.

AUTHOR REPLY: We included the proposed publication in the methods and discussion.

Attachment

Submitted filename: Response to Reviewers PONE-D-20-27974.docx

Decision Letter 1

Rafael da Costa Monsanto

8 Jan 2021

Traumatic Dislocation of Middle Ear Ossicles: A New Computed Tomography Classification Predicting Hearing Outcome

PONE-D-20-27974R1

Dear Dr. Mantokoudis,

We’re pleased to inform you that your manuscript has been judged scientifically suitable for publication and will be formally accepted for publication once it meets all outstanding technical requirements.

Within one week, you’ll receive an e-mail detailing the required amendments. When these have been addressed, you’ll receive a formal acceptance letter and your manuscript will be scheduled for publication.

An invoice for payment will follow shortly after the formal acceptance. To ensure an efficient process, please log into Editorial Manager at http://www.editorialmanager.com/pone/, click the 'Update My Information' link at the top of the page, and double check that your user information is up-to-date. If you have any billing related questions, please contact our Author Billing department directly at authorbilling@plos.org.

If your institution or institutions have a press office, please notify them about your upcoming paper to help maximize its impact. If they’ll be preparing press materials, please inform our press team as soon as possible -- no later than 48 hours after receiving the formal acceptance. Your manuscript will remain under strict press embargo until 2 pm Eastern Time on the date of publication. For more information, please contact onepress@plos.org.

Kind regards,

Rafael da Costa Monsanto, M.D.

Academic Editor

PLOS ONE

Additional Editor Comments (optional):

Congratulations on the excellent piece of work.

Reviewers' comments:

Reviewer's Responses to Questions

Comments to the Author

1. If the authors have adequately addressed your comments raised in a previous round of review and you feel that this manuscript is now acceptable for publication, you may indicate that here to bypass the “Comments to the Author” section, enter your conflict of interest statement in the “Confidential to Editor” section, and submit your "Accept" recommendation.

Reviewer #1: All comments have been addressed

Reviewer #3: All comments have been addressed

**********

2. Is the manuscript technically sound, and do the data support the conclusions?

The manuscript must describe a technically sound piece of scientific research with data that supports the conclusions. Experiments must have been conducted rigorously, with appropriate controls, replication, and sample sizes. The conclusions must be drawn appropriately based on the data presented.

Reviewer #1: Yes

Reviewer #3: Yes

**********

3. Has the statistical analysis been performed appropriately and rigorously?

Reviewer #1: Yes

Reviewer #3: Yes

**********

4. Have the authors made all data underlying the findings in their manuscript fully available?

The PLOS Data policy requires authors to make all data underlying the findings described in their manuscript fully available without restriction, with rare exception (please refer to the Data Availability Statement in the manuscript PDF file). The data should be provided as part of the manuscript or its supporting information, or deposited to a public repository. For example, in addition to summary statistics, the data points behind means, medians and variance measures should be available. If there are restrictions on publicly sharing data—e.g. participant privacy or use of data from a third party—those must be specified.

Reviewer #1: Yes

Reviewer #3: Yes

**********

5. Is the manuscript presented in an intelligible fashion and written in standard English?

PLOS ONE does not copyedit accepted manuscripts, so the language in submitted articles must be clear, correct, and unambiguous. Any typographical or grammatical errors should be corrected at revision, so please note any specific errors here.

Reviewer #1: Yes

Reviewer #3: Yes

**********

6. Review Comments to the Author

Please use the space provided to explain your answers to the questions above. You may also include additional comments for the author, including concerns about dual publication, research ethics, or publication ethics. (Please upload your review as an attachment if it exceeds 20,000 characters)

Reviewer #1: I congratulate the authors to address a point-by-point revised document of their study. And I congratulate the authors for their very interesting work.

Reviewer #3: I would like to thank the authors for the revised manuscript. The paper is improved with the revisions made. I have no further comments or concerns.

**********

7. PLOS authors have the option to publish the peer review history of their article (what does this mean?). If published, this will include your full peer review and any attached files.

If you choose “no”, your identity will remain anonymous but your review may still be made public.

Do you want your identity to be public for this peer review? For information about this choice, including consent withdrawal, please see our Privacy Policy.

Reviewer #1: No

Reviewer #3: No

Acceptance letter

Rafael da Costa Monsanto

28 Jan 2021

PONE-D-20-27974R1

Traumatic Dislocation of Middle Ear Ossicles: A New Computed Tomography Classification Predicting Hearing Outcome

Dear Dr. Mantokoudis:

I'm pleased to inform you that your manuscript has been deemed suitable for publication in PLOS ONE. Congratulations! Your manuscript is now with our production department.

If your institution or institutions have a press office, please let them know about your upcoming paper now to help maximize its impact. If they'll be preparing press materials, please inform our press team within the next 48 hours. Your manuscript will remain under strict press embargo until 2 pm Eastern Time on the date of publication. For more information please contact onepress@plos.org.

If we can help with anything else, please email us at plosone@plos.org.

Thank you for submitting your work to PLOS ONE and supporting open access.

Kind regards,

PLOS ONE Editorial Office Staff

on behalf of

Dr. Rafael da Costa Monsanto

Academic Editor

PLOS ONE


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