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Case Report
Peer-Review Record

Arteriovenous Fistula with Pseudoaneurysm and Facial Palsy Following Bilateral Sagittal Split Osteotomy: A Case Report

by Michala Ivanic-Sefcikova 1, Vasco Starke 1,*, Lukas Groessing 1, Michael Augustin 2, Michael Schwaiger 1 and Wolfgang Zemann 1
Reviewer 1: Anonymous
Reviewer 2: Anonymous
Reviewer 3: Anonymous
Reviewer 4: Anonymous
Submission received: 1 November 2024 / Revised: 22 January 2025 / Accepted: 29 January 2025 / Published: 8 February 2025

Round 1

Reviewer 1 Report

Comments and Suggestions for Authors

The article reports a rare clinical case of vascular complication (arteriovenous fistula with pseudoaneurysm) associated with facial paralysis, which occurred after bilateral sagittal osteotomy of the lower jaw (BSSO). The main objective is to raise awareness of these rare complications and provide recommendations on diagnostic and therapeutic management.

The case described is unique, being the first to combine arteriovenous fistula and pseudoaneurysm post-BSSO. This represents a significant contribution to the literature, as similar cases are extremely rare.

The scientific impact could be relevant for maxillofacial surgeons and interventional radiologists, as it provides useful information to identify and manage rare vascular complications.

The bibliography is up to date and relevant, with recent and topic-specific references. However, it would be useful to include additional systematic review articles to strengthen the discussion.

The radiological images are of good quality and well annotated.

Suggestions for revision:

• Improve the clarity of some verbose sentences.

• Add a direct pre- and post-intervention visual comparison in the images.

• Provide more details on the rationale for choosing endovascular treatment.

• Possibly expand the discussion with further comparisons with cases documented in the literature.

Author Response

Reviewer 1:

Comments:

  1. Improve the clarity of some verbose sentences.
  2. Add a direct pre- and post-intervention visual comparison in the images.
  3. Provide more details on the rationale for choosing endovascular treatment.
  4. Possibly expand the discussion with further comparisons with cases documented in the literature.

Responses:

Thank you for the positive feedback

  1. Professional English language editing was carried out once again.
  2. The caption of Figure 3 was adapted to better illustrate the comparison with the preoperative imaging (Figure 2).
  3. The rationale for choosing endovascular treatment has been explained in more detail. The corresponding paragraph has been adapted (L94-99).
  4. The discussion has been adapted and further comparisons with other cases have been added (L153-157).

Reviewer 2 Report

Comments and Suggestions for Authors

The manuscript “Arteriovenous Fistula with Pseudoaneurysm and Facial Palsy Following Bilateral Sagittal Split Osteotomy: A Case Report“ is a case report of an 48-year-old male with a serious vascular complication after BSSO.

 

Case reports on this rare condition are highly appreciated. The manuscript is generally well structured and interesting. It raises awareness for rare complications that may occur with orthognathic surgery.

 

Just some minor comments that help to improve the article:

 

1. I recommend that the authors follow the CARE guidelines for reporting case reports (https://www.equator-network.org/reporting-guidelines/care/)

2. Figure 1: The addition of a posterior-anterior radiograph would be very interesting.

3. Discussion: Please include this recent article as it is relevant: https://doi.org/10.1186/s13005-023-00396-9

 

I hope that these points will help to improve the article.

Author Response

Reviewer 2:

Comments:

  1. I recommend that the authors follow the CARE guidelines for reporting case reports (https://www.equator-network.org/reporting-guidelines/care/)
  2. Figure 1: The addition of a posterior-anterior radiograph would be very interesting.
  3. Discussion: Please include this recent article as it is relevant: https://doi.org/10.1186/s13005-023-00396-9

Responses:

Thank you for the positive feedback

  1. The CARE guidelines were fully incorporated. See CARE checklist (supplementary file).
  2. The frontal cephalometric radiograph has been included in Figure 1.
  3. The article by Al-Dawoody et al. provides interesting findings on the splitting pattern following BSSO to prevent bad splits. Vascular complications such as pseudoaneurysms or ateriovenous fistulas are not addressed in the article. However, as bad splitting can definitely be ruled out in our case report, the paper is not included in the discussion.

Reviewer 3 Report

Comments and Suggestions for Authors

Dear authors,


Thank you for submitting your manuscript titled „Arteriovenous Fistula with Pseudoaneurysm and Facial Palsy Following Bilateral Sagittal Split Osteotomy: A Case Report “.


It is necessary to point out some concerns in the presented manuscript:


• Introduction is to brief and does not provide sufficient information about concerns of the presented study. The absence of case reports dealing with AV fistula in the literature is not a limitation in providing more details and references about the vascular complications and false aneurysms following sagittal split osteotomies.


• Case report: please describe the surgical procedure in a more detailed manner. Methods
should be described with sufficient detail to allow others to replicate and build on published
results.


• Discussion: OK.


• Conclusions: OK.


• References: ref 15 is not adequately written.


I find this manuscript suitable for publication after major revision.


Kind regards!

Author Response

Reviewer 3:

Comments:

  1. Introduction is to brief and does not provide sufficient information about concerns of the presented study. The absence of case reports dealing with AV fistula in the literature is not a limitation in providing more details and references about the vascular complications and false aneurysms following sagittal split osteotomies.
  2. Case report: please describe the surgical procedure in a more detailed manner. Methods

should be described with sufficient detail to allow others to replicate and build on published

results.

  1. References: ref 15 is not adequately written.

Responses:

Thank you for the positive feedback.

  1. The introduction was kept brief and precise, as this study is intended as a case report. In the discussions section, the vascular complications are discussed in more detail and linked to the existing literature.
  2. The surgical procedure has been described in more detail (L54-55).
  3. The Reference has been verified and updated.

Reviewer 4 Report

Comments and Suggestions for Authors

This article presents an important case report. Although it is well-written, it has some linguistic mistakes and requires more information regarding the correction surgery and ethical issues

Title: OK.

Abstract: OK.

Introduction: line 35, “Kumar et al,”, there should be a full stop after “al.”.

Line 40, “occur, which is often difficult to control”, remove the coma after “occur”.

Case Report: line 70, “worse and worse”, can be rephrased as “worsen over time”.

Who did the correction surgery?

Ethical issue: did the authors introduce the case for the second surgery to the patient and discuss all possible treatment options? Did the patient sign a consent for awareness of the case and agreement to the chosen treatment?

Line 105-106, “Unfortunately, the facial palsy remained twelve months postoperatively still subtly visible, despite active physiotherapy and electrotherapy”. Did the patient consult a neurosurgeon, or did the orthodontist discuss the case with other neurologists?

Discussion: Line 120, “hidden blood loss is always to be expected intraoperatively,” can be corrected to “hidden blood loss is always expected intraoperatively.”

Conclusions: OK.

Author Response

Reviewer 4:

Comments:

  1. Introduction: line 35, “Kumar et al,”, there should be a full stop after “al.”.
  2. Line 40, “occur, which is often difficult to control”, remove the coma after “occur”.
  3. Case Report: line 70, “worse and worse”, can be rephrased as “worsen over time”.
  4. Who did the correction surgery?
  5. Ethical issue: did the authors introduce the case for the second surgery to the patient and discuss all possible treatment options? Did the patient sign a consent for awareness of the case and agreement to the chosen treatment?
  6. Line 105-106, “Unfortunately, the facial palsy remained twelve months postoperatively still subtly visible, despite active physiotherapy and electrotherapy”. Did the patient consult a neurosurgeon, or did the orthodontist discuss the case with other neurologists?
  7. Discussion: Line 120, “hidden blood loss is always to be expected intraoperatively,” can be corrected to “hidden blood loss is always expected intraoperatively.”

Responses:

Thank you for the positive feedback.

1.;2.;3.;7. The wording has been adjusted (L35; L39; L72; L138).

  1. The corrective procedure was performed by the Interventional Radiology Department.
  2. The patient was informed about the possible need of a second intervention prior to the BSSO. This is standard practice at our university hospital. All possible treatment options were then discussed with the patient, in particular the consideration of a conservative versus an interventional procedure. The patient gave his verbal and written consent after sufficient, detailed explanation and clarification of all open questions.
  3. The patient was not referred to neurosurgery as the association with the BSSO was obvious (nerve compression due to the developed pseudoaneurysm). The clinical presentation was a mild, isolated peripheral facial nerve palsy with no other neurological abnormalities. Following the radiological procedure, a conservative approach (physiotherapy and electrotherapy) was pursued on an interdisciplinary basis.

Round 2

Reviewer 3 Report

Comments and Suggestions for Authors

Dear authors,

The manuscript is suitable for publication in a presented form. 

The references should be written using the same style throughout the entire article. Please use either a reference manager software or write down references manually, but in a correct manner. 

Kind regards.

Author Response

Dear reviewer,

The references were written in the same style (Vancouver) throughout the article, according to the MDPI standard. The reference management software Endnote 20 (Clarivate Analytics) was applied for sorting.

Thank you for approving the article in its current form.

Best regards.

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