Is It Safe to Operate without Frozen Section Biopsies in Short-Segment Hirschsprung’s Disease? An Overview of 60 Cases

Round 1

Reviewer 1 Report

Comments and Suggestions for Authors

Well writen study, material and methods section is confusing, results are interesting, language is acceptable

Material and Methods are confusing, do not correlate with title and do not clarify primary and secondary results.

Authors do not describe the surgical technique(s) they used during these 20 years

Lines 93-95. sentence does not make sense. How can you have a microscopic evaluation during macroscopic assessment without sample under microscope. If you used a microscope please refrase and describe findings.

LInes 97-99. "resection above TZ" means from the beginning of TZ or from the end of TZ?

Lines 99-100. It seems that there was an intraoperative microscopic evaluation of healthy gagglionated bowel. Thus material and methods do not correlate with the title

Conclusion. Needs refrasal. Single macroscopic evaluation of the resection limit during definitve operation cannot be accepted and proposed. And is not supported by the study too, since authors used intraoperative H&E staining.

Author Response

Thank you very much for taking the time to review this manuscript. Please find the detailed responses below and the corresponding revisions/corrections in the re-submitted files.

Comment 1: [Authors do not describe the surgical technique(s) they used during these 20 years.]

Response 1: [Thank you for pointing this out. The operative technique used in most of the cases was trans anal pull-through but we didn’t mention theme since we didn’t find it affecting the resection level in none of the techniques used in the operation].

Comment 2: [Lines 93-95. sentence does not make sense. How can you have a microscopic evaluation during macroscopic assessment without sample under microscope. If you used a microscope please refrase and describe findings.]

Response 2: Agree. We have made corrections; it was mistake in writing. Instead of macroscopic we had written microscopic. It changes the context we agree, accordingly, we have corrected it in the revised manuscript this change can be found in the same line as commented from reviewer 1]

“[updated text in the manuscript if necessary]”

Comment 3: [LInes 97-99. "resection above TZ" means from the beginning of TZ or from the end of TZ?.]

Response 3: Resection was made proximally from the defined transition zone in the normally ganglionated segment. Change can be found in the same line as commented from reviewer 1]

 

Comment 4: [Lines 99-100. It seems that there was an intraoperative microscopic evaluation of healthy gagglionated bowel. Thus material and methods do not correlate with the title]

Response 4: There was no intra operative microscopic assessment. The samples taken from resected bowel after the operation were examined with routine examination (with HE). [The clarifications are in the line 99-100].

 

5. Additional clarifications for coment in the “conclusion”

In the lines 330-335 the conclusions are clarified and there is clear that no intraoperative H&E staining was used.

Reviewer 2 Report

Comments and Suggestions for Authors

Summary:

The manuscript pediatrrep-2992365 describes a study analyzing the impact of intraoperative macroscopic assessment only on outcomes and accuracy of diagnosing normally ganglionated colon in the surgical treatment of Hirschsprung's disease in a single center in Kosovo. The authors show that their macroscopic assessment has a 100% accuracy in their selected cohort of patients with short segment Hirschsprung's disease operated at an average of 9 months of age. 

Comments:
Study design: the study is a retrospective single center cohort analysis looking at accuracy of macroscopic evaluation of the transition zone and proximal ganglionated colon intraoperatively to post-op histology without using intraoperative frozen section to confirm the site of resection and pull-through level. The authors do not describe how these patients were managed to the time of surgery, and how many admissions these patients had for enterocolitis (if any) or other complications? Did the parents irrigate and decompress the patients at home? Any morbidity with this approach? Do the authors perform laparoscopy to evaluate the colon intraoperatively, or only resect the bowel transanally and evaluate the caliber at that time? Given that their approach differs somewhat from standard of care that is probably possible in Kosovo (pathology is available), did the surgeons and care team inform parents of that? Please comment. 

Given that a majority of resections went significantly more proximal than the usually described 5 cm, do the authors feel that they had to resect excessive amounts of colon to be in a "safe" zone? How does that affect their patients, especially if the TZ is in the descending colon and they may need to mobilize the splenic flexure? 

Did the authors perform a bowel prep or any preoperative decompression as this may affect bowel caliber as well? Please comment. 

A table with age at operation, distance from TZ, and potentially an analysis of the level of ganglionated bowel in the 10 and 15 cm resected specimen could be helpful. 

The manuscript addresses a very interesting question about the need for frozen section in surgical treatment of Hirschsprung's disease. The discussion is well-written, however should also raise concerns about overall safety and mention their selected cohort and impact of repeat imaging and difficulty to apply this approach at a younger / neonatal age. More granular long term outcomes data should be included in results, including possible reoperations, length of follow up and other complications to state that macroscopic determination is safe (not only comparing to permanent histological data, but also function).

Comments on the Quality of English Language

The manuscript is well-written and only very few spelling errors and minimal grammatical errors should be corrected.

Author Response

Thank you very much for taking the time to review this manuscript. Please find the detailed responses below and the corresponding revisions/corrections in the re-submitted files.

Comment: The authors do not describe how these patients were managed to the time of surgery, and how many admissions these patients had for enterocolitis (if any) or other complications?

Response: Most of these patients were treated from Pediatric gastroenterologists until were diagnosed for Hirschsprung disease. There was no clear evidence for the conservative treatment, but some of theme also had colostomas due to enterocolitis.

Comment: Did the parents irrigate and decompress the patients at home? Any morbidity with this approach? Do the authors perform laparoscopy to evaluate the colon intraoperatively, or only resect the bowel transanally and evaluate the caliber at that time? Given that their approach differs somewhat from standard of care that is probably possible in Kosovo (pathology is available), did the surgeons and care team inform parents of that? Please comment.

Response: In the last 8 years I personally do not operate Hirschsprung’s patients if the parents don’t learn irrigation procedure. Preoperatively most of my patients are well prepared with washouts at home but yet the transition zone is well defined intraoperatively. I also operate with assisted laparoscopic pull-through but intra operatively we don’t do frozen section biopsy and the level of resection is decided in the time of operation. 

Comment: Given that a majority of resections went significantly more proximal than the usually described 5 cm, do the authors feel that they had to resect excessive amounts of colon to be in a "safe" zone? How does that affect their patients, especially if the TZ is in the descending colon and they may need to mobilize the splenic flexure? 

Response: The dilated bowel that we resect we feel comfortable since it is well known that it usually is associated with dysmotility. Yes, we agree, in some cases we did splenic flexure mobilisation to make anastomosis without tension.

Comment: Did the authors perform a bowel prep or any preoperative decompression as this may affect bowel calibre as well? Please comment.

Response: yes, in every patient is performed bowel prep but yet the dilated bowel was clearly visible. It was somewhat difficult in those under age of 1 year which were managed with irigations at home but still the morphological changes were clearly differentiated.  

Comment: A table with age at operation, distance from TZ, and potentially an analysis of the level of ganglionated bowel in the 10 and 15 cm resected specimen could be helpful. 

Response: agree.

Comment: The manuscript addresses a very interesting question about the need for frozen section in surgical treatment of Hirschsprung's disease. The discussion is well-written, however should also raise concerns about overall safety and mention their selected cohort and impact of repeat imaging and difficulty to apply this approach at a younger / neonatal age. More granular long term outcomes data should be included in results, including possible reoperations, length of follow up and other complications to state that macroscopic determination is safe (not only comparing to permanent histological data, but also function).

Response: even in the patients that had many Hirschsprung associated enterocolitis the re biopsy and the biopsies from the margins of resections showed that there was no TZ left.

Reviewer 3 Report

Comments and Suggestions for Authors

Nice study. However, I would change the conclusion to be softer (i.e. cannot say its highly accurate). The standard of practice is still to perform frozen section. I suggest changing the conclusion to "May be accurate".

Authors clearly stated the limitations of their study.

Author Response

Thank you very much for taking the time to review this manuscript and thank you for suggesting the conclusion that was appropriate to add to it.

Round 2

Reviewer 1 Report

Comments and Suggestions for Authors

Thank you for the revisions

Author Response

Thank you very much for taking the time to review this manuscript. Please find the detailed responses below and the corresponding revisions/corrections in the re-submitted files.