Neuroendocrine Carcinoma at the Sphenoid Sinus Misdiagnosed as an Olfactory Neuroblastoma and Resected Using High-Flow Bypass
Round 1
Reviewer 1 Report
Dear Authors,
you made a great work. However, some improvements are mandatory before acceptance.
The paper is a case report study on a neuroendocrine carcinoma at the sphenoid sinus misdiagnosed as an olfactory neuroblastoma and resected using a high-flow bypass.
The Authors made a great work in terms of methodology and the paper sounds scientific and well written.
However, some improvements are mandatory before acceptance.
The abstract is well written, complete and summary in its various aspects. The keywords are complete and appropriate.
In the Introduction:
· in the introduction I think it may be useful to include some more information about the diagnostic imaging of this district, to highlight the possibility that these pathologies have an early diagnosis.
In the Case Report description:
· “was observed two and a half years after surgery (Figure 7).3.” I think it is more correct to indicate the number of months. Also I suggest authors to check punctuation in the text.
Discussion: I think it can be improved by adding references about other similar previous works or discussing previous works according to the evaluations that emerged from this one. The overall is comprehensive, concise and complete in its various aspects.
Conclusions are concise and clear.
Bibliography should be formatted respecting the journal’s requirements and no improper citations are evidenced.
Figures and labels are clear and easy to comprehend.
English is clear and easy to understand, can be made more readable.
Author Response
Response to reviewer 1
Thank you for your careful review and valuable comments and suggestions. We have responded to each of your comments below. The text in red font represents the changes made in response to your comments.
In the Introduction:
in the introduction I think it may be useful to include some more information about the diagnostic imaging of this district, to highlight the possibility that these pathologies have an early diagnosis.
Response:
Thank you for your suggestion. We have now added the following text in the Introduction section (Lines 34-36): “The imaging features of ONB are nonspecific; thus, diagnosis using imaging modalities alone is difficult and warrants histopathology [1].”
In the Case Report description:
“was observed two and a half years after surgery (Figure 7).3.” I think it is more correct to indicate the number of months. Also I suggest authors to check punctuation in the text.
Response:
Thank you for your advice. We have revised the terminology from two and a half years to 30 months at all relevant instances in the manuscript, including the Case Report section (Lines 25-26, 117, 156).
Discussion: I think it can be improved by adding references about other similar previous works or discussing previous works according to the evaluations that emerged from this one. The overall is comprehensive, concise and complete in its various aspects.
Response:
Thank you for your advice and positive feedback. We additionally searched PubMed (search terms: neuroendcrine carcinoma, misdiagnosis, olfactory neuroblastoma; neuroendocrine carcinoma, olfactory neuroblastoma, S-100; neuroendocrine carcinoma, S-100 positive) for studies on NEC misdiagnosed as ONB and found no reports of NEC misdiagnosed as ONB because of the presence of S-100-positive cells in biopsy tissue. The following text has been added in the Discussion section (Lines 173-175) to emphasize that it is extremely rare but possible to misdiagnose S-100 protein in normal tissue as sustentacular cells: “Within the extent of our investigation, we found no reports of NEC misdiagnosed as ONB because of the presence of S-100-positive cells in biopsy tissue.”
Conclusions are concise and clear.
Response:
We appreciate your evaluation.
Bibliography should be formatted respecting the journal’s requirements and no improper citations are evidenced.
Response:
Thank you for pointing this out. We have formatted the bibliography per the journal’s requirements.
Lines 303-324: References
Cohen, Z.R.; Marmor, E.; Fuller, G.N.; DeMonte, F. Misdiagnosis of olfactory neuroblastoma. Neurosurg. Focus. 2002, 12, e3. doi: 10.3171/foc.2002.12.5.4.
Mahooti, S.; Wakely, P.E., Jr. Cytopathologic features of olfactory neuroblastoma. Cancer. 2006, 108, 86–92. doi: 10.1002/cncr.21718.
Zahedi, F.D.; Gendeh, B.S.; Husain, S.; Kumar, R.; Kew, T.Y. Ectopic esthesioneuroblastoma of the sphenoclivus: A rare entity. Indian J. Otolaryngol. Head Neck Surg. 2017, 69, 125–129. doi: 10.1007/s12070-016-0978-0.
Yang, T.; Tariq, F.; Chabot, J.; Madhok, R.; Sekhar, L.N. Cerebral revascularization for difficult skull base tumors: A contemporary series of 18 patients. World Neurosurg. 2014, 82, 660–671. doi: 10.1016/j.wneu.2013.02.028.
Esposito, G.; Sebök, M.; Amin-Hanjani, S.; Regli, L. Cerebral bypass surgery: Level of evidence and grade of recommendation. Acta Neurochir. Suppl. 2018, 129, 73–77. doi: 10.1007/978-3-319-73739-3_10.
Mura, J.; Rojas-Zalazar, D.; de Oliveira, E. Revascularization for complex skull base tumors. Skull Base. 2005, 15, 63–70. doi: 10.1055/s-2005-868163.
Liu, J.K.; Couldwell, W.T. Interpositional carotid artery bypass strategies in the surgical management of aneurysms and tumors of the skull base. Neurosurg. Focus. 2003, 14, e2. doi: 10.3171/foc.2003.14.3.3.
Mizunari, T.; Murai, Y.; Kim, K.; Kobayashi, S.; Kamiyama, H.; Teramoto, A. Posttraumatic carotid-cavernous fistulae treated by internal carotid artery trapping and high-flow bypass using a radial artery graft--two case reports. Neurol Med Chir (Tokyo). 2011, 51, 113-116. doi: 10.2176/nmc.51.113.
Wormald, R.; Lennon, P.; O’Dwyer, T.P. Ectopic olfactory neuroblastoma: Report of four cases and a review of the literature. Eur. Arch. Otorhinolaryngol. 2011, 268, 555–560. doi: 10.1007/s00405-010-1423-8.
Zhong, W.; Wang, C.; Ye, W.; Wu, D. Primary olfactory neuroblastoma of the nasopharynx: A case report. Ear Nose Throat J. 2021, 145561321989436. doi: 10.1177/0145561321989436.
Figures and labels are clear and easy to comprehend.
Response:
We appreciate your evaluation.
English is clear and easy to understand, can be made more readable.
Response:
Thank you for advice. We have proofread the text and made minor changes.
Reviewer 2 Report
Dear Authors,
Thank you for submitting your manuscript about a diagnostic challenge involving sellar region. Study is a case report of a patient initially misdiagnosed as olfactory neuroblastoma. The text also points out the importance of total resection of lesions regardless of histopathology. From the surgeon's perspective, ideal conditions are achieved with total resection of masses. From an oncologist's perspective, I would like to know how it looks. Olfactory neuroblastoma or not, why didn't you refer the patient for adjuvant therapy? I think bypass surgery for total removal of this mass is a highly invasive approach. Another transnasal biopsy or partial resection could be preferred with ultimate diagnosis leading to chemoradiotherapy. Study of Cohen et al which is the number 1 reference in the manuscript, gives us the ambiguity of the neuroblastoma diagnosis and definitive diagnosis should be made after repeated biopsy examinations. Was this patient discussed within a multidisciplinary council including oncologists?
Other issues are as follows:
1) Line 83 involves a sentence: "Postoperatively, the patient had mild muscle weakness in the right hemisphere, transient expressive aphasia, and oculomotor nerve palsy, possibly due to surgery." "Right-sided muscle weakness" term may be used for correcting language misusage.
2) Figure 1 caption begins with a misspelled "Preoperative".
I believe the scientific value and interest is high in the study. After a minor revision of the above 2 issues I think your manuscript would be eligible for publication. Minor revision is selected for curiosity and to discuss choosing the treatment strategy for the best possible outcome and I really wonder about your reply to my concerns.
Best regards.
Author Response
Response to reviewer 2
Thank you for your careful review and valuable comments and suggestions. We have responded to each of your comments below. The text in blue font represents the changes made in response to your comments.
Thank you for submitting your manuscript about a diagnostic challenge involving sellar region. Study is a case report of a patient initially misdiagnosed as olfactory neuroblastoma. The text also points out the importance of total resection of lesions regardless of histopathology. From the surgeon's perspective, ideal conditions are achieved with total resection of masses. From an oncologist's perspective, I would like to know how it looks. Olfactory neuroblastoma or not, why didn't you refer the patient for adjuvant therapy? I think bypass surgery for total removal of this mass is a highly invasive approach. Another transnasal biopsy or partial resection could be preferred with ultimate diagnosis leading to chemoradiotherapy. Study of Cohen et al which is the number 1 reference in the manuscript, gives us the ambiguity of the neuroblastoma diagnosis and definitive diagnosis should be made after repeated biopsy examinations. Was this patient discussed within a multidisciplinary council including oncologists?
Response:
Thank you for your questions and suggestions. We agree that we need to make a comprehensive treatment decision that also includes the opinion of an oncologist, because bypass surgery is highly invasive and there is only a limited number of facilities that can perform it. Unfortunately, our institution does not have an oncologist dedicated to chemotherapy for head and neck tumors. Because the preoperative diagnosis in this case was ONB, we opted for complete removal using bypass instead of chemoradiotherapy because of the curative potential of the former.
We have added the following text in the last paragraph of the Discussion section (
Lines 247-251): “On the other hand, our institution does not have a specialized oncologist to treat head and neck malignancies, and this may have biased our treatment strategy. Because only a limited number of institutions can perform revascularization using high-flow bypass, the best treatment option may vary depending on the characteristics of the institution.”
Line 83 involves a sentence: "Postoperatively, the patient had mild muscle weakness in the right hemisphere, transient expressive aphasia, and oculomotor nerve palsy, possibly due to surgery." "Right-sided muscle weakness" term may be used for correcting language misusage.
Response:
Thank you for your advice. We have revised the terminology from mild muscle weakness in the right hemisphere to mild right-sided muscle weakness.
Lines 99-100: “Postoperatively, the patient had mild right-sided muscle weakness, transient expressive aphasia, and oculomotor nerve palsy, possibly due to surgery.”
Figure 1 caption begins with a misspelled "Preoperative".
Response:
Thank you for pointing this out. We have corrected the typographical error.
Lines 121-122: “Preoperative contrast-enhanced computed tomography (CT) findings. Axial section (A) and coronal (B) images are shown.”
Reviewer 3 Report
A well-writen manuscript to describe a clinically sucessful case
there are several concerns
1. not 100% ONB is S-100 positive, which has been proved by previous study
2. S-100 positive or not will not change our clinical policy
3. good case presentation but not fit the scope of this Journal "Diagnostics"
4. high-flow bypass procedure is not correlated with the diagnosis of this disease
Author Response
Response to reviewer 3
Thank you for your careful review and valuable comments and suggestions. We have responded to each of your comments below. The text in green font represents the changes made in response to your comments.
not 100% ONB is S-100 positive, which has been proved by previous study
Response:
Thank you for pointing this out. ONB with negative S-100 protein and no sustentacular cells is considered atypical. In addition, other morphological findings such as Homer–Wright rosettes were not present, so we concluded that the lesion was not ONB. We have modified the text in the Case Report section as follows (
Lines 95-98): “Differentiating between high-grade ONB and neuroendocrine tumors is sometimes difficult; the tumor was eventually diagnosed as NEC, based on the absence of sustentacular cells and the absence of morphological findings such as Homer–Wright rosettes (Figure 6D).”
S-100 positive or not will not change our clinical policy
Response:
We agree with your comment. In this case, however, the patient was diagnosed with NEC, and chemoradiotherapy was planned to be added if there was no prolonged loss of consciousness due to acute subdural hematoma. As a result, the positive and negative S-100 results may not have changed the treatment strategy in this case, because surgery alone prevented recurrence.
good case presentation but not fit the scope of this Journal "Diagnostics"
Response:
In this case report, the misdiagnosis was due to not only the difficulty in pathological diagnosis of skull base malignancy but also the assumptions during clinical evaluation by the clinician and the pathologist. We believe that the case is thought-provoking and will be of interest to the readers of Diagnostics.
high-flow bypass procedure is not correlated with the diagnosis of this disease
Response:
Thank you for your comment. There is no direct correlation between the diagnosis and the high-flow bypass procedure. However, for better understanding of the clinical course in this case, we would like to describe the details of the treatment.
Round 2
Reviewer 3 Report
A successful clinical case completed by an experienced surgical team!
However, I still feel that the content of this manuscript does not meet that of the article required by this journal
