Inguinoscrotal Hernia, a Possible Cause of Rapidly Developing Fetal Scrotal Mass: Case Report and Literature Update

Round 1

Reviewer 1 Report

I would like to thank for the possibility to review the manuscript, and also acknowledge the Authors for their effort. Here are some of my comments to improve the text:

1.       “The inguinoscrotal hernia is common during infancy, especially in the first year of 18 life when the peritoneal-vaginal duct can still be patent. However, during fetal life, it is a 19 very rare condition.” – please add some reference to evidence the statement

2.       „The pregnancy was complicated by HIV infection and treated with antiretroviral therapy (ART) and antidepressant drugs.” – Do You have the information for how long and which drugs were taken? Were they taker also during 1^st trimester?

3.       “amniotic fluid at the upper limits”- What does it mean? AFI 20 or 22 or 25 cm?Or Do You use MPV?

4.       “The contralateral scrotum was normal, with the presence of mild hydrocele”- I would rather recommend not to write “normal” as the hydrocele could be an effect of the contrlateral hernia, so that is a pathological observation.

5.       Figure 1 presents perfect ultrasound photos of the hernia, but actually Fig 1. A and B and C presents the same thing. It would be better to change Fig 1B to henia with Power Doppler or Color Doppler with slower flow and Fig 1C to fetal bowels (maybe they were normal, but this is also important observation).

Author Response

I would like to thank for the possibility to review the manuscript, and also acknowledge the Authors for their effort. Here are some of my comments to improve the text:

COMMENT

“The inguinoscrotal hernia is relatively common during infancy, especially in the first year of 18 life when the peritoneal-vaginal duct can still be patent. However, during fetal life, it is a 19 very rare condition.” – please add some reference to evidence the statement

RESPONSE

As suggested by the Reviewer, we have modified the introduction as follows:

Inguinal hernia is common among neonates and infants as it occurs in up to 4% of full-term babies and up to 9-11% of those born prematurely due to the persistence of a patent processus vaginalis (PPV) [1,2]. It occurs more frequently in males (90%) and represents one of the main indications for pediatric surgery since a timely correction is recommended to reduce the risk of bowel incarceration, strangulation and overall morbidity and mortality [1].

COMMENT

 The pregnancy was complicated by HIV infection and treated with antiretroviral therapy (ART) and antidepressant drugs.” – Do You have the information for how long and which drugs were taken? Were they taker also during 1^st trimester?

RESPONSE

The patient took Emtricitabina, tenofovir, raltegravir and citalopram from the first trimester of pregnancy. Line 41

COMMENT

“amniotic fluid at the upper limits”- What does it mean? AFI 20 or 22 or 25 cm?Or Do You use MPV?

RESPONSE

We mean a deepest pocket of 7.5 cm. Line 45

COMMENT

“The contralateral scrotum was normal, with the presence of mild hydrocele”- I would rather recommend not to write “normal” as the hydrocele could be an effect of the contrlateral hernia, so that is a pathological observation

RESPONSE

“The contralateral scrotum showed the presence of mild hydrocele”. Line 53

COMMENT

Figure 1 presents perfect ultrasound photos of the hernia, but actually Fig 1. A and B and C presents the same thing. It would be better to change Fig 1B to henia with Power Doppler or Color Doppler with slower flow and Fig 1C to fetal bowels (maybe they were normal, but this is also important observation).

RESPONSE

Figure 1 A, B and C present the photos of the inguinoscrotal hernia from different points of view in order to provide to the reader the best ultrasound representation of the hernia. The signs of the presence of fetal bowel inside the hernia (through few small echo-free cystic areas) are visible only in FIG C while the box of color Doppler is present in each of the three images, indicating the absence of blood flow signal in Fig a, B and C.

So we can modify as follows: Line 82

“Figure 1. Case of fetal inguinoscrotal hernia ( Fetal sonographic findings at 38+6 weeks of gestation: a-b) a 4.0 × 3.5 cm right scrotal mass mainly solid, with regular wall, complex echotexture, no blood flow signal and c) few small echo-free cystic areas in its inside; d-e. Postnatal examination).”

Reviewer 2 Report

The authors presented a case of newborn with inguinoscrotal hernia. In addition the conducted a review of literature.

I read the study with great interest. Unfortunately, the review revealed significant methodological flaws. My concerns are as follows:

1. Presentation of abstract is poor, below any standard. The authors should add more details in regards to specific case.

2. Introduction is poor and not informative. More informations regarding differential diagnosis, diagnostic procedures and treatment modalities should be presented.

3. Presentation of a case is also insuficient. More information regarding surgical approach and intraoperative finding, as well as preoperative diagnostics (as child underwent surgery due to intestinal obstruction) should be presented.

4. This is not the optimal search performed by the authors. An optimal search in a systematic review requires at least 4 databases to be searched. (The authors only searched only one database, which is under the standard). Please see the link below:

 https://systematicreviewsjournal.biomedcentral.com/articles/10.1186/s13643-017-0644-y

5. Flow chart of the study should be in accordance to PRISMA guidelines.

6. Authors should indicate the exact date on which they conducted the literature search and the initials of the authors involved in the process.

7. Redolution of Table 1 is below any standard, very hard to follow and read.

8. The discussion is very extensive, consists largely of a repetition of known facts from the literature and needs to be significantly improved. Do not give an overview of the literature in this section. Do not discuss your results piece by piece. Focus on the results of the main objectives of the study. Write in four consecutive paragraphs (without headings): (i) summary (no data) of the findings of this study; (ii) logical and coherent comparison with the existing literature focusing on the main aim(s); (iii) limitations of the study; and (iv) implications for practise/policy/research with a concluding statement.

9. A similar review was recently published (doi: 10.1016/j.jpedsurg.2022.07.001.). I wonder what use this report in the same journal really has.

10. Conclusions are general and does not follow from present case.

The quality of the English language should be improved. The manuscript should be edited by a native English speaker or a professional language editor to improve grammar and readability.

Author Response

The authors presented a case of newborn with inguinoscrotal hernia. In addition the conducted a review of literature. I read the study with great interest. Unfortunately, the review revealed significant methodological flaws. My concerns are as follows:

COMMENT

Presentation of abstract is poor, below any standard. The authors should add more details in regards to specific case.

RESPONSE

Modified as follows:

Inguinoscrotal hernia is a common pediatric disease, but a rare condition in the fetus. We present a case from our institution of fetal inguinoscrotal hernia with a possible rapid development. In addition to our case, we present a literature update on fetal inguinoscrotal hernia to enhance the ability to recognize it from the other scrotal masses on ultrasound. Antenatal management, differential diagnosis and postnatal management are also discussed.

COMMENT

Introduction is poor and not informative. More informations regarding differential diagnosis, diagnostic procedures and treatment modalities should be presented.

RESPONSE

In the introduction, we cannot give more information about the topic of the paper (FETAL INGUINOSCROTAL HERNIA) because is a very rare condition in utero and the literature is poor. To provide information regarding prenatal pathogenesis, differential diagnosis, diagnostic procedures and management is the aim of the paper. Anyway, we can give some information about the post-natal hernia in general and modify as follows:

Inguinal hernia is common among neonates and infants as it occurs in up to 4% of full-term babies and up to 9-11% of those born prematurely due to the persistence of a patent processus vaginalis (PPV) [1,2]. It occurs more frequently in males (90%) and represents one of the main indications for pediatric surgery since a timely correction is recommended to reduce the risk of bowel incarceration, strangulation and overall morbidity and mortality [1]. Surgery, called herniorrhaphy, consists of the incision of the groin and the repositioning of the hernia sac in the abdomen.

COMMENT

Presentation of a case is also insufficient. More information regarding surgical approach and intraoperative finding, as well as preoperative diagnostics (as child underwent surgery due to intestinal obstruction) should be presented.

RESPONSE

The objective of the presented case is mainly to characterize the antenatal sonographic features of the inguinoscrotal hernia.  The diagnostic exams performed before surgery were only the ultrasound as mentioned in the text.  Suspicion of bowel obstruction is a clinical diagnosis.

We can detail the surgical approach and the intraoperative findings as follows:

Surgical correction of the inguinal-scrotal hernia was performed at the age of 15 days be-cause of intestinal obstruction, since the hernia was no longer reducible. Surgery consisted of a transverse incision of the groin, the individuation of the spermatic cord, and the repositioning of the hernia sac in the abdomen after its incision and verification of the intestinal content. There were no signs of bowel strangulation but incarceration was confirmed.

COMMENT

This is not the optimal search performed by the authors. An optimal search in a systematic review requires at least 4 databases to be searched. (The authors only searched only one database, which is under the standard). Please see the link below:

 https://systematicreviewsjournal.biomedcentral.com/articles/10.1186/s13643-017-0644-y

RESPONSE

As reported in the title and in the text, we have done a review, not a systematic review of the literature. We can modify the title and the text with a ʺliterature updateʺ.

COMMENT

Flow chart of the study should be in accordance to PRISMA guidelines.

RESPONSE

See reply to previous comment.

COMMENT

Authors should indicate the exact date on which they conducted the literature search and the initials of the authors involved in the process.

RESPONSE

The search terms used were “fetal scrotal hernia” from inception until the 31 of September 2023. Inclusion criteria were all human cases of fetal inguinoscrotal hernia with prenatal US assessment and confirmed by postnatal examination and surgery.  Exclusion criteria included all animal cases of fetal inguinoscrotal hernia, cases with no full-text available, cases without prenatal US assessment, cases without postnatal confirmation and cases of postnatal inguinoscrotal hernia. A total of 43 articles matched the keywords “fetal scrotal hernia”. Titles and abstracts of these articles were screened by authors (R.M.  and I.C.) to determine which articles could undergo full-text review.

COMMENT

Resolution of Table 1 is below any standard, very hard to follow and read.

RESPONSE

Table 1 has been fully revised to improve reading.

COMMENT

The discussion is very extensive, consists largely of a repetition of known facts from the literature and needs to be significantly improved. Do not give an overview of the literature in this section. Do not discuss your results piece by piece. Focus on the results of the main objectives of the study. Write in four consecutive paragraphs (without headings): (i) summary (no data) of the findings of this study; (ii) logical and coherent comparison with the existing literature focusing on the main aim(s); (iii) limitations of the study; and (iv) implications for practise/policy/research with a concluding statement.

RESPONSE

In line with the proposed objectives, the discussion aim to enhance the knowledge of the prenatal diagnosis and management of the inguinoscrotal hernia in the light of the data reported, with references to both the case report and the literature. These are not known facts since the incidence during prenatal period account only 18 cases so far.

That is why we discuss piece by piece about:

• Prental pathogenetic hypotheses
• The typical antenatal presentation (side, gestational age presentation of presentation, association with structural anomalies/genetic disorders, ultrasonographic features of the hernia and the contralateral scrotum )
• Differential diagnosis ( we resume the main ultrasonographics findings of the other scrotal masses to provide the reader a better understanding of differential diagnosis)
• Ultrasonographic signs predictive of intestinal obstruction/ strangulation
• Antenatal management ( how to follow up the patient, what to look at during each visit, when decide for urgent delivery)

We have removed the Initial general information about the hernia.

Limitations of the study are added at line 260.

COMMENT

A similar review was recently published (

RESPONSE

The review found at doi: 10.1016/j.jpedsurg.2022.07.001 has a different objective: to define the optimal timing to perform surgical repair of inguinal hernia (postnatal period).  Our paper deals exclusively with the inguinoscrotal hernia of the fetus. This is a very rare condition during the prenatal period, which is still poorly considered in the differential diagnosis of fetal scrotal masses. It is necessary firstly to improve the sonographer's ability to recognize this lesion during fetal life and secondly to improve their capability to manage this lesion before birth.

We think that our paper provides the more detailed ultrasound images of fetal inguinoscrotal hernia that we have seen in literature so far. Moreover, adding our case to the only 18 cases present in the current literature  will be useful to enhance the knowledge of this rare condition during fetal life, adding for the first time the hypothesis that it is a rapidly developing condition.

COMMENT

Conclusions are general and does not follow from present case.

RESPONSE

The conclusions aim to summarize the evidence obtained from our case and the review of the literature to provide greater tools to the gynaecologist in the diagnosis and prenatal management of the rare inguinoscrotal hernia.

They report:

• typical prenatal ultrasound presentation, (follow our case)
• the pathognomonic sign at ultrasound (follow our case)
• what to do in case of doubtful diagnosis : MRI/longitudinal scan / the development time of the lesion. (the attention to the time of development of the lesion is reported for the first time in our paper)
• the prenatal follow-up strategy
• when to suspect an intestinal obstruction and decide for urgent delivery ( since the prediction of bowel strangulation seems to be poor if polyhydramnios and abdominal bowel dilatation do not appear together and the blood flow signal is present---> the poor prognosis of intestinal obstruction in the inguinoscrotal hernia is first mentioned in our paper).

COMMENT

The quality of the English language should be improved. The manuscript should be edited by a native English speaker or a professional language editor to improve grammar and readability.

RESPONSE

We have already done the grammar check. We have made other corrections to improve the quality of the English language.

Reviewer 3 Report

This is a well-written case study presentation. The details are appropriate to the content, and the flow of the case is well done. Table 1 is quite difficult to read; it needed to be magnified to 175%, in order to see the words. And even then, it was still hard to read. Suggest to divide this into two tables, as currently the amount of columns are too many. This is the focus of the literature review, thus it needs to be clear to the reader. Otherwise, this is very well reported!

Author Response

COMMENT

This is a well-written case study presentation. The details are appropriate to the content, and the flow of the case is well done. Table 1 is quite difficult to read; it needed to be magnified to 175%, in order to see the words. And even then, it was still hard to read. Suggest to divide this into two tables, as currently the amount of columns are too many. This is the focus of the literature review, thus it needs to be clear to the reader. Otherwise, this is very well reported!

RESPONSE

Table 1 has been fully revised to improve reading.

Round 2

Reviewer 2 Report

The authors made minimal improvements to the paper. Narative review is of lowest scientific quality. In line with that I cannot recommend this work for publication due to the poor quality of the evidence. I see no benefit to readers from this  review compared to previously published data. In addition, the quality of the study is questionable due to several significant methodological problems. Finally, this is a simple case report of a well known condition.

Moderate editing of English language required

Author Response

We thank Reviewer for his attention to our Manuscript. We would like to clarify that our study, after invitation from the Journal, we submitted it as a case report and an update of the literature on a particular topic of interest to those involved in ultrasound, fetal malformations, and neonatal surgery, however, we respect the judgment.