Case Report: Congenital diaphragmatic hernia in one twin followed by ex-utero intrapartum treatment (EXIT) procedure: Pearls & pitfalls

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This study aimed to report a rare case while also analyzing the pearls and pitfalls in diagnosing and managing this case. A 32-year-old multiparous woman referred to a tertiary hospital with multiple pregnancies and suspected CDH on the 1st baby. The patient underwent further investigation and the first fetus had a severe CDH on the left side. The second fetus was in a transverse lie and no other abnormalities were found. The babies were delivered through caesarean section and the first baby underwent the ex-utero intrapartum treatment (EXIT) procedure without prior antepartum fetal therapy. A large defect measuring 7.5 x 6 centimeters on the left side (Bochdalek type) was found in the first baby during surgery to make repairs. The baby died the next day due to some complications from the previous repair. As a pearl of this case, prenatal diagnosis and multidisciplinary approach hold pivotal roles. In prenatal imaging, CDH is distinguished by the visualization of thorax filled with abdominal contents and the mediastinal shifting from the defect to the contralateral side. EXIT procedures could be performed after the baby with CDH is delivered and the infant will still require post-natal surgical repair and its success rate depends on many factors. In conclusion, proper prenatal diagnosis with ultrasound evaluation from the second trimester, close and regular maternal-fetal monitoring, prenatal intervention, intrapartum ultrasound guidance, and post-natal procedures are all critical." } { "@context": "http://schema.org", "@type": "BreadcrumbList", "itemListElement": [ { "@type": "ListItem", "position": "1", "item": { "@id": "https://f1000research.com/", "name": "Home" } }, { "@type": "ListItem", "position": "2", "item": { "@id": "https://f1000research.com/browse/articles", "name": "Browse" } }, { "@type": "ListItem", "position": "3", "item": { "@id": "https://f1000research.com/articles/13-44", "name": "Case Report: Congenital diaphragmatic hernia in one twin followed..." } } ] } Home Browse Case Report: Congenital diaphragmatic hernia in one twin followed... ALL Metrics - Views Downloads Get PDF Get XML Cite How to cite this article Pribadi A, Santoso DPJ, Kharismawati N et al. Case Report: Congenital diaphragmatic hernia in one twin followed by ex-utero intrapartum treatment (EXIT) procedure: Pearls & pitfalls [version 1; peer review: 2 not approved] . F1000Research 2024, 13 :44 ( https://doi.org/10.12688/f1000research.138290.1 ) NOTE: If applicable, it is important to ensure the information in square brackets after the title is included in all citations of this article. Close Copy Citation Details Export Export Citation Sciwheel EndNote Ref. Manager Bibtex ProCite Sente EXPORT Select a format first Track Share ▬ ✚ Case Report Case Report: Congenital diaphragmatic hernia in one twin followed by ex-utero intrapartum treatment (EXIT) procedure: Pearls & pitfalls [version 1; peer review: 2 not approved] Adhi Pribadi 1 , Dhanny Primantara Johari Santoso 1 , Nuniek Kharismawati 1 , Fatima Zahra 1 , Cut Razianti ZB 1 , Annisa Dewi Nugrahani https://orcid.org/0000-0003-4734-1753 1 Adhi Pribadi 1 , Dhanny Primantara Johari Santoso 1 , [...] Nuniek Kharismawati 1 , Fatima Zahra 1 , Cut Razianti ZB 1 , Annisa Dewi Nugrahani https://orcid.org/0000-0003-4734-1753 1 PUBLISHED 08 Jan 2024 Author details Author details 1 Maternal Fetal Medicine Division, Department of Obstetrics and Gynecology, Faculty of Medicine, University of Padjadjaran, Padjadjaran University, Bandung, West Java, 40161, Indonesia Adhi Pribadi Roles: Conceptualization, Data Curation, Formal Analysis, Funding Acquisition, Investigation, Methodology, Project Administration, Resources, Validation, Writing – Original Draft Preparation, Writing – Review & Editing Dhanny Primantara Johari Santoso Roles: Conceptualization, Data Curation, Formal Analysis, Investigation, Methodology, Resources, Validation, Writing – Original Draft Preparation Nuniek Kharismawati Roles: Data Curation, Formal Analysis, Investigation, Methodology, Validation Fatima Zahra Roles: Data Curation, Formal Analysis, Investigation, Methodology, Validation Cut Razianti ZB Roles: Data Curation, Formal Analysis, Investigation, Methodology, Validation Annisa Dewi Nugrahani Roles: Conceptualization, Visualization, Writing – Original Draft Preparation, Writing – Review & Editing OPEN PEER REVIEW DETAILS REVIEWER STATUS Abstract Cases of congenital diaphragmatic hernia (CDH) in one twin have been reported rarely. This study aimed to report a rare case while also analyzing the pearls and pitfalls in diagnosing and managing this case. A 32-year-old multiparous woman referred to a tertiary hospital with multiple pregnancies and suspected CDH on the 1st baby. The patient underwent further investigation and the first fetus had a severe CDH on the left side. The second fetus was in a transverse lie and no other abnormalities were found. The babies were delivered through caesarean section and the first baby underwent the ex-utero intrapartum treatment (EXIT) procedure without prior antepartum fetal therapy. A large defect measuring 7.5 x 6 centimeters on the left side (Bochdalek type) was found in the first baby during surgery to make repairs. The baby died the next day due to some complications from the previous repair. As a pearl of this case, prenatal diagnosis and multidisciplinary approach hold pivotal roles. In prenatal imaging, CDH is distinguished by the visualization of thorax filled with abdominal contents and the mediastinal shifting from the defect to the contralateral side. EXIT procedures could be performed after the baby with CDH is delivered and the infant will still require post-natal surgical repair and its success rate depends on many factors. In conclusion, proper prenatal diagnosis with ultrasound evaluation from the second trimester, close and regular maternal-fetal monitoring, prenatal intervention, intrapartum ultrasound guidance, and post-natal procedures are all critical. READ ALL READ LESS Keywords Case Report, Congenital Diaphragmatic Hernia, Ex utero Intrapartum Technique Procedures, Hernia Repair, Twins Corresponding Author(s) Adhi Pribadi ( [email protected] ) Close Corresponding author: Adhi Pribadi Competing interests: No competing interests were disclosed. Grant information: This research is conducted by financial support from the Ministry of Research, Technology and Higher Education and University of Padjadjaran with contract numbers 4414/HK.03.01/J.6/2018, 591/UN6.L/PKS/2018 and 1827/UN6.3.1/LT/2020. The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript. Copyright: © 2024 Pribadi A et al . This is an open access article distributed under the terms of the Creative Commons Attribution License , which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. The author(s) is/are employees of the US Government and therefore domestic copyright protection in USA does not apply to this work. The work may be protected under the copyright laws of other jurisdictions when used in those jurisdictions. How to cite: Pribadi A, Santoso DPJ, Kharismawati N et al. Case Report: Congenital diaphragmatic hernia in one twin followed by ex-utero intrapartum treatment (EXIT) procedure: Pearls & pitfalls [version 1; peer review: 2 not approved] . F1000Research 2024, 13 :44 ( https://doi.org/10.12688/f1000research.138290.1 ) First published: 08 Jan 2024, 13 :44 ( https://doi.org/10.12688/f1000research.138290.1 ) Latest published: 08 Jan 2024, 13 :44 ( https://doi.org/10.12688/f1000research.138290.1 ) Introduction Congenital diaphragmatic hernia occurs when abdominal contents herniate into the thoracic cavity, causing lung hypoplasia and pulmonary vascular alteration. Hernias are classified into different types based on the defects’ location in the diaphragm which are: (a) The most frequent type, Bochdalek hernias (occur in the posterolateral part of the diaphragm) which mostly occurs on the left side rather than the right side, (b) Morgagni hernias (20% to 25%), and (c) central hernias, accounting for 2% to 5% of all cases. 1 , 2 Cases of CDH in one twin have rarely been reported. Studies reporting CDH in twin pregnancies are still rare and reported as cases of CDH in general. A retrospective study from 2010 to 2016 reported only 11 twin pregnancies with CDH in one of the twins. 3 Infants with CDH have a higher mortality rate without prompt management, and they also have a wide range of long-term issues that last beyond infancy, such as complications of the respiratory system, gastrointestinal system, neurobehavioral disorders, and orthopaedic deformities. Hernia recurrence is another risk that can develop later, especially in newborns with significant defects. It has been established that each of these clinical issues has an impact on the quality of life of affected children. Thus, prenatal diagnosis especially with ultrasound holds a pivotal role to prevent further fetal morbidity and mortality. 4 , 5 The majority of CDH cases are identified prenatally between 18–24 weeks of pregnancy during a standard abnormality scan. CDH is distinguished by the visualization of thorax filled with abdominal contents and the mediastinal shifting from the defect to the contralateral side in prenatal imaging. Milder cases, on the other hand, might not be discovered until later due to minor defects that lead on to cause many complications. 6 , 7 The prognosis of CDH is influenced by the extent of lung hypoplasia, the location of the liver, and co-existing abnormalities. Fetal lung volume is measured by the lung-to-head ratio (LHR) which is obtained from the contralateral lung area divided by the fetal head circumference. Fetal LHR measurements have been used to predict neonatal pulmonary hypoplasia. Between 12 and 32 weeks of gestation, there is an 18-fold increase in lung area and a four-fold increase in head circumference. Thus, LHR on the left or right-side increases exponentially with gestational age because the measurements to determine LHR differ depending on gestational age. The value of the ratio is very important, in the study by Metkus, all neonates died if the ratio reached 0.6, on the contrary, they would survive better with a survival rate of 100% if the ratio reached 1.35, while the ratio between 0.6 to 1.35 had a survival rate of 61%. 8 Advances in prenatal diagnosis have made it possible to identify a number of birth anomalies that may obstruct the fetus’s airway or impede ventilation at birth. Following the confirmation of CDH diagnosis, close monitoring to check fetal well-being is strongly advised. 6 , 8 , 9 Selective centers in developed countries are providing fetal therapy Tracheal Occlusion To Accelerate Lung Growth Trial (TOTAL) and Fetoscopic Endoluminal Tracheal Occlusion or FETO as management for qualified patients in moderate and severe cases of CDH. 10 The delivery of infants with CDH is recommended until term gestation. It is also strongly recommended that the infants are delivered at a tertiary center with expertise in CDH management and have that access to extracorporeal membrane oxygenation (ECMO) therapy. 11 , 12 In cases of prenatally diagnosed CDH, it is advised to place a nasogastric tube shortly after delivery for stomach and intestines decompressions. Thus, in twin pregnancy, ultrasound guidance plays a critical role in determining which twin suffered from CDH. Ex utero intrapartum treatment procedures could be performed on the neonates after the baby with CDH was delivered. A variety of fetal airway obstructions have been successfully treated with EXIT procedures, enabling control of the airway at birth. The EXIT procedure can be performed on babies with CDH at birth, however, babies with CDH still need postnatal surgical repair. 11 – 13 Here, we present a case report of prenatally diagnosed CDH in one of the twins who underwent EXIT procedure without previous antepartum fetal therapy. This case is very rare since diagnosing one of the twins with CDH during the intrapartum period to determine which one will undergo EXIT procedure is quite challenging. There have been no case reports published regarding prenatally diagnosed CDH in one of the twins followed by EXIT procedure, especially in developing countries. Therefore, the aim of this study was to report a rare case as well as to analyze the pearls and difficulties in diagnosing and managing these patients. Case report This following case is described according to the CARE checklist. 14 A 32-year-old multiparous Indonesian woman with no occupation only a housewife, came to the Maternal Fetal Clinic, in a tertiary hospital in Indonesia, as referred by an obstetrician with multiple pregnancy and suspected CDH on the 1 st baby. The mother had a history of twin babies in her family. She had no comorbidities and no previous history of surgery. The patient was compos mentis. Her vital signs, physical examination results, and laboratory profiles were within normal limits. The patient underwent further investigation with ultrasound and it revealed multiple pregnancies with placenta inserted at the posterior, positive septum confirmed monochorionic-diamniotic multiple pregnancies with discordance of 18%. The first fetus was alive with a cephalic lie equal to 32–33 weeks of pregnancy, and an estimated fetal weight of 1,504 grams. In the thoracal region, the four-chamber view diagnostic planes showed a dextroposition with narrow left ventricle; the measured cardiothoracic area (CTAR) was 20%, and the axis was 29.13 degree. A mass (stomach) was found in this thoracal region, which was 1.98 cm × 1.20 cm in size, located as high as the four-chamber view, and the right ventricle was pushed forward anteriorly as shown in Figure 1 and Figure 2 (Video of Babies’ Ultrasound). LHR was 0.14 and observed per expected LHR (o/e LHR) was 4.79%. Ultrasound assessment showed urinary bladder filled with fluid which shows normal kidney function, shape and appearance of the fetal kidney within normal limits, also umbilical artery within normal limits. The single deepest pocket (SDP) was 5.44 cm. Further examination with Doppler velocimetry revealed middle cerebral artery pulsatile index (MCA-PI) and umbilical pulsatile index (umbilical PI) were within normal limit with normal flow of ductus venosus. No abnormality was found in other regions. These findings suggested that the first fetus had a severe left-side CDH with no lung hypoplasia, polyhydramnios, hyperexpanded lungs, and anatomic compressions were found. Figure 1. The four-chamber view diagnostic planes showed a mass (stomach) (yellow arrow) was found in this thoracal region, was 1. The four-chamber view diagnostic planes showed a mass (stomach) (yellow arrow) was found in this thoracal region, was 1.98 cm × 1.20 cm in size. The right ventricle is pushed forward anteriorly (red arrow). LHR was 0.14 and o/e LHR was 4.79%. Lung hypoplasia prediction was also within normal limit. Figure 2. Video of Babies' Ultrasound. This is the video recording of the four-chamber view diagnostic planes which showed a mass (stomach) that was found in this thoracal region. It was 1.98 cm × 1.20 cm in size. The right ventricle is pushed forward anteriorly. LHR was 0.14 and o/e LHR was 4.79%. Lung hypoplasia prediction was also within normal limit. https://doi.org/10.6084/m9.figshare.24632139 The second fetus was alive with transverse lie equal to 31–32 weeks of pregnancy and an estimated fetal weight of 1,824 grams. No abnormality was found in the thoracal and abdominal regions. Further examination with Doppler velocimetry revealed MCA-PI and umbilical pulsatile index (umbilical PI) were within normal limits with normal flow of ductus venosus. The single deepest pocket (SDP) was 5.44 cm. Three weeks later, she was admitted to the obstetric ward and had undergone expectant management until term pregnancies. Caesarean delivery was planned for the patients in our case due to an obstetric indication (transverse lie of the second fetus), which was also accompanied by CDH of the first fetus. Following the confirmation of the CDH and the completion of the imaging workup, a multidisciplinary team of obstetricians, anesthesiologists, neonatologists, and pediatric surgeons was assembled. An intervention aimed at establishing and maintaining airway patency immediately after cesarean delivery was considered mandatory, thus direct laryngoscopy intubation as the first approach in EXIT-to-airway procedure was planned. The first baby was a male born weighing 1,600 grams with a 1 st minute APGAR score of 3 and the 5 th minute APGAR score was 5, while the second baby was also a male; born weighing 2,000 grams, with a 1 st minute APGAR score of 6, while the 5 th minute APGAR score was 8. Following informed consent, the group in charge carried out the EXIT-to-airway procedure planned previously. The babies were admitted to the neonatal intensive care unit (NICU) for general condition improvement and adaptation of pulmonary vasculature before definite surgical repair for CDH. The mother was given supportive treatments in obstetric wards. Four days later after stabilization, the first baby underwent primary repair surgery for his CDH. Intraoperative findings demonstrated left-sided Bochdalek type of CDH with a large defect size of 7.5 cm × 6 cm in size. Unfortunately, the baby died the next day due to some complications from previous CDH primary repair. Discussion CDH in one twin is rarely reported. As the pearl of this case, advances in prenatal diagnosis played an important role because this method allows the detection of a number of birth anomalies that can obstruct the fetal airway or obstruct ventilation at birth. Therefore, this condition should be referred to a tertiary center with standard neonatal management in the NICU and optimal pediatric surgery services which will improve outcomes. 15 More than half of all CDH cases are discovered prenatally in the second trimester. However, in this case, the patient was diagnosed in the third trimester in tertiary care, because it was not diagnosed earlier by a specialist or perhaps because it had an initial minor defect that was not identified until the third trimester. 6 , 7 , 16 Thus, accurate prediction of outcome is essential because prenatal interventions, which currently consist of FETO, may be beneficial for the fetus with the worst prognosis. The location of the defect, the estimated lung size, and the presence of liver herniation, predicted or measured by ultrasound and MRI, are used to make predictions and case-specific prognoses. In prenatal imaging, CDH is distinguished by the visualization of thorax filled with abdominal contents and the mediastinal shifting from the defect to the contralateral side. 6 The size of the heart is tiny (CTAR 20%), especially the left ventricle due to mass pressure from the left. In this case, the stomach pressure had a 100% impact on the small size of the heart. Pressure by other organs on the intrathoracic side affect the effectiveness of the heart’s workload, causing an urgent effect on the heart when it beats. The heart does not have enough space during diastole because of pressure from that organ. In the end the amount of blood pumped by the heart is limited because of the limitations of the heart expanding during diastole. 7 Metkus et al. 8 first described the lung-to-head ratio (LHR). LHR provides an indirect estimation of a lung size contralateral to the hernia. It is commonly measured using two-dimensional ultrasound at the four-chamber view by dividing the contralateral lung area divided by the fetal head circumference. In this case the LHR ratio was only 0.14. When referring to the Metkus study, postpartum babies have a poor prognosis and are unlikely to survive because the LHR ratio is below 0.6. 8 The most precise techniques for measuring the lung include tracing its contours or applying the long axis method. Severe lung hypoplasia is considered when the o/e LHR in left-sided CDH (LCDH) is <25%. However, if LHR 25% with o/e LHR 35% regardless of liver position, or LHR 35% with o/e LHR 45% with “up” type of liver position, this case is considered a “moderate” level even though the survival rate is limited to only 50–60% and survivors have significant morbidity. An o/e LHR emerges as a better predictor of fetal survival. 6 – 8 The patient in this case was diagnosed prenatally with CDH on the first fetus with an o/e LHR of 4.79%, thus this condition was considered a severe CDH. It is highly recommended to closely monitor and check the well-being of the fetus after confirming the diagnosis of CDH. 6 , 8 , 9 Eligible patients can be offered intrauterine management such as TOTAL and FETO especially in cases of moderate or severe CDH. However, because both of these invasive procedures were performed intrauterine, the potential for preterm labor due to rupture of the membranes or the emergence of early labor must also be considered. However, studies have shown that the group of fetuses that underwent this procedure had a higher survival rate than the control group. 16 One advantage of the EXIT procedure is that it allows intubation and ventilation during labor. However, the patient and family refused prenatal intervention in this case after informed consent was obtained, with the consideration that the family refused because it could worsen the prognosis of the first fetus if there was an earlier delivery. The delivery of infants with CDH is recommended until term gestation. Therefore, patients underwent expectative management until term pregnancy in this case. 17 As another pearl, diagnosing one of the twins with CDH during intrapartum period to determine which one will undergo EXIT procedure is quite challenging. Therefore, many aspects are required to be considered in evaluating the diagnosis and therapy. Following patient consent after delivery, the assembled group carried out the EXIT-to-airway procedure planned. With an overall survival rate of 83%, the EXIT-to-airway procedure reduced mortality of neonates. Nonetheless, maternal and fetal complications persist. The fetus is at risk of barotrauma and inadequate ventilation during airway securing, hence appropriate tube placement must be confirmed before cord clamping. 18 All infants undergoing intrauterine fetal surgery still require postnatal CDH repair and the success rate depends on many factors. 11 – 13 , 15 So far, CDH has been managed as a disorder requiring emergency surgery for many years, but even so, if surgical therapy is successful, there are still other pathological conditions, namely a relatively frequent threat of death, often from pulmonary hypertension (PH). Previous studies have challenged the practice of sending newborns directly from the delivery room to the operating room and have found no appreciable therapeutic benefit in the presence of pulmonary hypertension. There is currently advice for surgical correction to be delayed for at least 48 to 72 hours after delivery to allow time for the pulmonary vessels to adapt. Surgery is delayed longer in children with severe PH until the condition is effectively controlled. If ECMO is required, the survival rate of newborns with CDH drops to approximately 50%. 18 Historically, CDH was treated surgically through a subcostal abdominal incision. Nowadays, thoracoscopic surgery is used to conduct many CDH repairs on stable children since minimally invasive surgery has become increasingly popular over time. 19 The use of minimally invasive procedures may reduce postoperative discomfort and prevent thoracotomy and laparotomy-related complications. When the diaphragmatic defect is significant, a primary repair may be impractical or impossible. Based on the defect size in this patient, the patient underwent a primary repair. To fill the defect, one may employ an abdominal or thoracic muscle flap, a biologic patch, or a prosthetic patch. 20 However, all of these treatments carry the risk of future surgical failure and CDH recurrence. In this case, the baby died the next day due to some complications from previous CDH primary repair using synthetic patches. Synthetic patches were chosen as widely used patches of defect closure, but have been implicated as a significant contributor to morbidity and further complications in recent studies. 20 , 21 This condition might be also affected by technical rather than intrinsic to the patch. Thus, considering the risk and benefit of surgical method based on the defect size and patches or mesh used become important to determine patient’s survival expectations. Conclusion Although CDH in one of the twins is rarely reported, it has a high morbidity and mortality rate. The diagnosis and management are quite challenging since they involve several specialists as well as the adoption of particular uncommon procedures in clinical practices. In consequences, proper prenatal diagnosis using ultrasound since the second trimester, close and regular maternal-fetal monitoring, prenatal intervention, intrapartum ultrasound guidance, and post-natal procedures hold a pivotal role. Accurate preoperative planning with a multidisciplinary approach, followed by all necessary precautions and procedures are crucial for a successful EXIT-to-airway procedure. The pearls and pitfalls previously described become considerations for further provision and guidelines for CDH. Patient perspective The patient would be happy to see if all physician all over the world will learn from her baby’s case. She did not mind if her baby condition is discussed in this report in order to prevent another same case occur in the future. Case report consent Written informed consent for publication of their clinical details and clinical images was obtained from the patient. Data availability All data underlying the results are available as part of the article and no additional source data are required. Reporting guidelines Open Science Framework: CARE checklist for ‘Case Report: Congenital diaphragmatic hernia in one twin followed by ex-utero intrapartum treatment (EXIT) procedure: Pearls & pitfalls’. https://doi.org/10.17605/OSF.IO/W2TX8 . 14 Data are available under the terms of the Creative Commons Attribution 4.0 International license (CC-BY 4.0). Acknowledgment The great gratitude to Department Obstetrics & Gynecology, Faculty of Medicine, University of Padjadjaran/Dr. Hasan Sadikin General Hospital. References 1. Kosiński P, Wielgoś M: Congenital diaphragmatic hernia: pathogenesis, prenatal diagnosis and management - literature review. Ginekol. Pol. 2017; 88 (1): 24–30. PubMed Abstract | Publisher Full Text 2. Montedonico S, Nakazawa N, Puri P: Congenital diaphragmatic hernia and retinoids: searching for an etiology. Pediatr. Surg. Int. 2008; 24 (7): 755–761. PubMed Abstract | Publisher Full Text | Free Full Text 3. Wang W, Pan W, Chen J, et al. : Outcomes of Congenital Diaphragmatic Hernia in One of the Twins. Am. J. Perinatol. 2019; 36 (12): 1304–1309. PubMed Abstract | Publisher Full Text 4. Cordier AG, Russo FM, Deprest J, et al. : Prenatal diagnosis, imaging, and prognosis in Congenital Diaphragmatic Hernia. Semin. Perinatol. 2020; 44 (1): 51163. Epub 2019 Jul 30. PubMed Abstract | Publisher Full Text 5. Jani J, Nicolaides KH, Keller RL, et al. : Observed to expected lung area to head circumference ratio in the prediction of survival in fetuses with isolated diaphragmatic hernia. Ultrasound Obstet. Gynecol. 2007; 30 (1): 67–71. PubMed Abstract | Publisher Full Text 6. Shue EH, Miniati D, Lee H: Advances in prenatal diagnosis and treatment of congenital diaphragmatic hernia. Clin. Perinatol. 2012; 39 (2): 289–300. PubMed Abstract | Publisher Full Text 7. Pribadi A, et al. : Adverse effect left congenital diaphragmatic hernia to position and function of the fetal heart. Int. J. Reprod. Contracept. Obstet. Gynecol. 2015; 4 (6): 1863–1867. Publisher Full Text 8. Metkus AP, Filly RA, Stringer MD, et al. : Sonographic predictors of survival in fetal diaphragmatic hernia. J. Pediatr. Surg. 1996; 31 (1): 148–152. discussion 151-2. PubMed Abstract | Publisher Full Text 9. Belfort MA, Olutoye OO, Cass DL, et al. : Feasibility and Outcomes of Fetoscopic Tracheal Occlusion for Severe Left Diaphragmatic Hernia. Obstet. Gynecol. 2017; 129 (1): 20–29. PubMed Abstract | Publisher Full Text 10. Van der Veeken L, Russo FM, De Catte L, et al. : Fetoscopic endoluminal tracheal occlusion and reestablishment of fetal airways for congenital diaphragmatic hernia. Gynecol. Surg. 2018; 15 (1): 9. PubMed Abstract | Publisher Full Text | Free Full Text 11. Al-Maary J, Eastwood MP, Russo FM, et al. : Fetal Tracheal Occlusion for Severe Pulmonary Hypoplasia in Isolated Congenital Diaphragmatic Hernia: A Systematic Review and Meta-analysis of Survival. Ann. Surg. 2016; 264 (6): 929–933. PubMed Abstract | Publisher Full Text 12. Foglia EE, Ades A, Hedrick HL, et al. : Initiating resuscitation before umbilical cord clamping in infants with congenital diaphragmatic hernia: a pilot feasibility trial. Arch. Dis. Child Fetal Neonatal. 2020; 105 (3): 322–326. PubMed Abstract | Publisher Full Text | Free Full Text 13. Kays DW: ECMO in CDH: Is there a role? Semin. Pediatr. Surg. 2017; 26 (3): 166–170. PubMed Abstract | Publisher Full Text 14. Pribadi A: Case Report: Congenital diaphragmatic hernia in one of twins followed by ex-utero intrapartum treatment (EXIT) procedure: Pearls & pitfalls. OSF 2023. Publisher Full Text 15. Canadian Congenital Diaphragmatic Hernia CollaborativePuligandla PS, Skarsgard ED, et al. : Diagnosis and management of congenital diaphragmatic hernia: a clinical practice guideline. CMAJ. 2018; 29; 190 (4): E103–E112. PubMed Abstract | Publisher Full Text | Free Full Text 16. Hutcheon JA, Butler B, Lisonkova S, et al. : Timing of delivery for pregnancies with congenital diaphragmatic hernia. BJOG. 2010; 117 (13): 1658–1662. Publisher Full Text 17. Sengupta S, Carrion V, Shelton J, et al. : Adverse neonatal outcomes associated with early-term birth. JAMA Pediatr. 2013; 167 (11): 1053–1059. Erratum in: JAMA Pediatr. 2014;168(1):53. Publisher Full Text 18. Lefebvre C, Rakza T, Weslinck N: et al; French CDH Study Group. Feasibility and safety of intact cord resuscitation in newborn infants with congenital diaphragmatic hernia (CDH). Resuscitation. 2017; 120 : 20–25. PubMed Abstract | Publisher Full Text 19. Snoek KG, Reiss IK, Greenough A, et al. : Standardized Postnatal Management of Infants with Congenital Diaphragmatic Hernia in Europe: The CDH EURO Consortium Consensus - 2015 Update. Neonatology. 2016; 110 (1): 66–74. PubMed Abstract | Publisher Full Text 20. Tsao K, Lally PA, Lally KP, et al. : Minimally invasive repair of congenital diaphragmatic hernia. J. Pediatr. Surg. 2011; 46 (6): 1158–1164. PubMed Abstract | Publisher Full Text | Free Full Text 21. Kamal TR, Tyraskis A, Ghattaura H, et al. : Synthetic versus Biological Patches for CDH: A Comparison of Recurrence Rates and Adverse Events, Systematic Review, and Meta-Analysis. Eur. J. Pediatr. Surg. 2022. Publisher Full Text Comments on this article Comments (1) Version 1 VERSION 1 PUBLISHED 08 Jan 2024 Reader Comment 24 Jun 2025 Wiku Andonotopo , Obstetrics & Gynecology, Women Health Center, Fetomaternal Clinic, Eka Hospital BSD, Department of Obstetrics & Gynecology, Women Health Center, Fetomaternal Clinic, Eka Hospital BSD, Serpong, Tangerang, Banten, INDONESIA., Tangerang, Indonesia 24 Jun 2025 Reader Comment Dear Dr. Pribadi and Co-authors, Thank you for submitting your case report on a rare and complex presentation of congenital diaphragmatic hernia (CDH) in one twin with EXIT procedure . The ... Continue reading Dear Dr. Pribadi and Co-authors, Thank you for submitting your case report on a rare and complex presentation of congenital diaphragmatic hernia (CDH) in one twin with EXIT procedure . The topic is clinically important, particularly in showcasing the challenges of fetal diagnosis and surgical coordination in a developing country context. However, to make your manuscript worthy of publication in a peer-reviewed medical journal , several major areas must be addressed to meet professional and academic standards. �� MAJOR ISSUES TO FIX 1. ENGLISH LANGUAGE & GRAMMAR The manuscript requires substantial English editing to correct grammar, improve sentence structure, and reduce awkward phrasing. Many sentences are repetitive, overly long, or unclear. This affects reader comprehension and the scientific impact of your findings. �� Recommendation : Have the entire manuscript professionally proofread by a native English speaker or editing service. 2. STRUCTURE AND STYLE The abstract lacks clear structure (background, case description, conclusion) and includes unnecessary narrative elements. It should concisely reflect the content and key findings. Figures and videos are poorly integrated , with captions repeated or mismatched. There are redundant explanations (e.g., LHR and o/e LHR described multiple times across sections with the same phrasing). The main discussion lacks focus at times and wanders into generic textbook material rather than emphasizing what was unique about your case . �� Recommendation : Streamline the content. Focus more on critical insights from your case and how it advances understanding of CDH in twins and EXIT procedures in low-resource settings. 3. REFERENCES The citation format is inconsistent . Several citations are incomplete or out of place, and many PubMed links are pasted without being properly referenced. References need to follow a uniform style with journal abbreviation, year, volume, and page numbers. �� Recommendation : Reformat all references according to the target journal’s style guide (e.g., Vancouver style or AMA). 4. ETHICS AND REPORTING GUIDELINES There is no explicit statement of IRB approval or confirmation that the case followed the Declaration of Helsinki or institutional ethical standards. While CARE checklist use is mentioned, this section is underemphasized. �� Recommendation : Add a formal Ethical Approval section in the methods or end of the manuscript, even if local guidelines do not require approval for single case reports. 5. CLINICAL DEPTH AND FOLLOW-UP The report would be strengthened by: Postmortem insights (if available) explaining the complications leading to death. A clearer justification for choosing EXIT without prenatal intervention, considering an o/e LHR of only 4.79% (extremely poor prognosis). Discussion of the technical aspects of EXIT and surgery—what could have been done differently? �� Recommendation : Provide deeper critical analysis of the decision-making process and outcomes. Expand on limitations and what clinicians in similar settings should take away. ✅ STRONG POINTS The case is original and rare. Prenatal imaging is well-documented. Ethical patient consent is addressed. It gives valuable insight into fetal surgery in developing countries. �� BOTTOM LINE Your case is important, but it is not yet ready for indexing in its current form. With substantial improvements in language, structure, reference formatting, ethical transparency, and focused clinical insight, it could become a valuable contribution to fetal medicine literature. Please consider revising carefully with these points in mind. I would encourage resubmission once these issues are addressed. Respectfully, Wiku Andonotopo, MD, PhD, HDGO, FICS. Department of Obstetrics & Gynecology, Women Health Center, Fetomaternal Clinic, Eka Hospital Serpong, Tangerang, Banten, INDONESIA Dear Dr. Pribadi and Co-authors, Thank you for submitting your case report on a rare and complex presentation of congenital diaphragmatic hernia (CDH) in one twin with EXIT procedure . The topic is clinically important, particularly in showcasing the challenges of fetal diagnosis and surgical coordination in a developing country context. However, to make your manuscript worthy of publication in a peer-reviewed medical journal , several major areas must be addressed to meet professional and academic standards. �� MAJOR ISSUES TO FIX 1. ENGLISH LANGUAGE & GRAMMAR The manuscript requires substantial English editing to correct grammar, improve sentence structure, and reduce awkward phrasing. Many sentences are repetitive, overly long, or unclear. This affects reader comprehension and the scientific impact of your findings. �� Recommendation : Have the entire manuscript professionally proofread by a native English speaker or editing service. 2. STRUCTURE AND STYLE The abstract lacks clear structure (background, case description, conclusion) and includes unnecessary narrative elements. It should concisely reflect the content and key findings. Figures and videos are poorly integrated , with captions repeated or mismatched. There are redundant explanations (e.g., LHR and o/e LHR described multiple times across sections with the same phrasing). The main discussion lacks focus at times and wanders into generic textbook material rather than emphasizing what was unique about your case . �� Recommendation : Streamline the content. Focus more on critical insights from your case and how it advances understanding of CDH in twins and EXIT procedures in low-resource settings. 3. REFERENCES The citation format is inconsistent . Several citations are incomplete or out of place, and many PubMed links are pasted without being properly referenced. References need to follow a uniform style with journal abbreviation, year, volume, and page numbers. �� Recommendation : Reformat all references according to the target journal’s style guide (e.g., Vancouver style or AMA). 4. ETHICS AND REPORTING GUIDELINES There is no explicit statement of IRB approval or confirmation that the case followed the Declaration of Helsinki or institutional ethical standards. While CARE checklist use is mentioned, this section is underemphasized. �� Recommendation : Add a formal Ethical Approval section in the methods or end of the manuscript, even if local guidelines do not require approval for single case reports. 5. CLINICAL DEPTH AND FOLLOW-UP The report would be strengthened by: Postmortem insights (if available) explaining the complications leading to death. A clearer justification for choosing EXIT without prenatal intervention, considering an o/e LHR of only 4.79% (extremely poor prognosis). Discussion of the technical aspects of EXIT and surgery—what could have been done differently? �� Recommendation : Provide deeper critical analysis of the decision-making process and outcomes. Expand on limitations and what clinicians in similar settings should take away. ✅ STRONG POINTS The case is original and rare. Prenatal imaging is well-documented. Ethical patient consent is addressed. It gives valuable insight into fetal surgery in developing countries. �� BOTTOM LINE Your case is important, but it is not yet ready for indexing in its current form. With substantial improvements in language, structure, reference formatting, ethical transparency, and focused clinical insight, it could become a valuable contribution to fetal medicine literature. Please consider revising carefully with these points in mind. I would encourage resubmission once these issues are addressed. Respectfully, Wiku Andonotopo, MD, PhD, HDGO, FICS. Department of Obstetrics & Gynecology, Women Health Center, Fetomaternal Clinic, Eka Hospital Serpong, Tangerang, Banten, INDONESIA Competing Interests: Competing Interests: I have no competing interests to declare. Close Report a concern Comment ADD YOUR COMMENT Author details Author details 1 Maternal Fetal Medicine Division, Department of Obstetrics and Gynecology, Faculty of Medicine, University of Padjadjaran, Padjadjaran University, Bandung, West Java, 40161, Indonesia Adhi Pribadi Roles: Conceptualization, Data Curation, Formal Analysis, Funding Acquisition, Investigation, Methodology, Project Administration, Resources, Validation, Writing – Original Draft Preparation, Writing – Review & Editing Dhanny Primantara Johari Santoso Roles: Conceptualization, Data Curation, Formal Analysis, Investigation, Methodology, Resources, Validation, Writing – Original Draft Preparation Nuniek Kharismawati Roles: Data Curation, Formal Analysis, Investigation, Methodology, Validation Fatima Zahra Roles: Data Curation, Formal Analysis, Investigation, Methodology, Validation Cut Razianti ZB Roles: Data Curation, Formal Analysis, Investigation, Methodology, Validation Annisa Dewi Nugrahani Roles: Conceptualization, Visualization, Writing – Original Draft Preparation, Writing – Review & Editing Competing interests No competing interests were disclosed. Grant information This research is conducted by financial support from the Ministry of Research, Technology and Higher Education and University of Padjadjaran with contract numbers 4414/HK.03.01/J.6/2018, 591/UN6.L/PKS/2018 and 1827/UN6.3.1/LT/2020. The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript. Article Versions (1) version 1 Published: 08 Jan 2024, 13:44 https://doi.org/10.12688/f1000research.138290.1 Copyright © 2024 Pribadi A et al . This is an open access article distributed under the terms of the Creative Commons Attribution License , which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. The author(s) is/are employees of the US Government and therefore domestic copyright protection in USA does not apply to this work. The work may be protected under the copyright laws of other jurisdictions when used in those jurisdictions. Download Export To Sciwheel Bibtex EndNote ProCite Ref. Manager (RIS) Sente metrics Views Downloads F1000Research - - PubMed Central info_outline Data from PMC are received and updated monthly. - - Citations open_in_new 0 open_in_new 0 open_in_new SEE MORE DETAILS CITE how to cite this article Pribadi A, Santoso DPJ, Kharismawati N et al. Case Report: Congenital diaphragmatic hernia in one twin followed by ex-utero intrapartum treatment (EXIT) procedure: Pearls & pitfalls [version 1; peer review: 2 not approved] . F1000Research 2024, 13 :44 ( https://doi.org/10.12688/f1000research.138290.1 ) NOTE: If applicable, it is important to ensure the information in square brackets after the title is included in all citations of this article. COPY CITATION DETAILS track receive updates on this article Track an article to receive email alerts on any updates to this article. TRACK THIS ARTICLE Share Open Peer Review Current Reviewer Status: ? Key to Reviewer Statuses VIEW HIDE Approved The paper is scientifically sound in its current form and only minor, if any, improvements are suggested Approved with reservations A number of small changes, sometimes more significant revisions are required to address specific details and improve the papers academic merit. Not approved Fundamental flaws in the paper seriously undermine the findings and conclusions Version 1 VERSION 1 PUBLISHED 08 Jan 2024 Views 0 Cite How to cite this report: Ma L and Liu C. Reviewer Report For: Case Report: Congenital diaphragmatic hernia in one twin followed by ex-utero intrapartum treatment (EXIT) procedure: Pearls & pitfalls [version 1; peer review: 2 not approved] . F1000Research 2024, 13 :44 ( https://doi.org/10.5256/f1000research.151480.r254457 ) The direct URL for this report is: https://f1000research.com/articles/13-44/v1#referee-response-254457 NOTE: it is important to ensure the information in square brackets after the title is included in this citation. Close Copy Citation Details Reviewer Report 30 May 2024 Lishuang Ma , Department of Pediatric and Neonatal Surgery, Capital Institute of Pediatrics, Beijing, China Chao Liu , Department of Pediatric and Neonatal Surgery, Capital Institute of Pediatrics, Beijing, China Not Approved VIEWS 0 https://doi.org/10.5256/f1000research.151480.r254457 This study aimed to report a rare case while also analyzing the pearls and pitfalls in diagnosing and managing this case. However, throughout the full text, the article did not describe the highlights of the experience of the diagnosis and ... Continue reading READ ALL This study aimed to report a rare case while also analyzing the pearls and pitfalls in diagnosing and managing this case. However, throughout the full text, the article did not describe the highlights of the experience of the diagnosis and treatment of this patient, nor extracted the lessons that should be learned and the hidden dangers that need to be avoided, but generally made a literature review, and the cited literature was older. It is not unusual for one twin to have CDH. Therefore, this article does not reflect the scientific value and guiding significance for clinical practice. 1. "Milder cases, on the other hand, might not be discovered until later due to minor defects that lead on to cause many complications." Smaller and less severe defects may be detected later, and they may present clinically slightly after birth, some even without respiratory or circulatory disturbances, but only incidentally when chest radiographs are examined for other reasons. However, incarceration of herniated thoracic organs may also occur with critical illness and be first diagnosed. This is inconsistent with the authors' view. 2. "This case is very rare since diagnosing one of the twins with CDH during the intrapartum period to determine which one will undergo EXIT procedure is quite challenging." We all know that it is a challenge for twin children to distinguish diaphragmatic hernia, but it is not difficult to judge by prenatal and intrapartum ultrasonography, or what are the key points and difficulties in this process, please elaborate. 3. "The first baby was a male born weighing 1,600 grams with a 1st minute APGAR score of 3 and the 5th minute APGAR score was 5." Describe in detail how to judge a fetus with diaphragmatic hernia during EXIT. Why is this process challenging? 4. "Unfortunately, the baby died the next day due to some complications from previous CDH primary repair." What complication? What is it related to? Please specify. 5. "As the pearl of this case, advances in prenatal diagnosis played an important role because this method allows the detection of a number of birth anomalies that can obstruct the fetal airway or obstruct ventilation at birth." There was no particular point in this case. The birth process for one twin to have CDH is indeed crucial, but not specific for the EXIT process. Please specify. 6. "The patient in this case was diagnosed prenatally with CDH on the first fetus with an o/e LHR of 4.79%, thus this condition was considered a severe CDH." Lung head ratio and o/e lung head ratio are indeed very important for the assessment of CDH, but the accuracy of detection is not optimistic in clinical tests. However, for the children in this treatise, please give necessary explanation on what is the special significance and how it is different from the evaluation of singleton CDH children. 7. "As another pearl, diagnosing one of the twins with CDH during intrapartum period to determine which one will undergo EXIT procedure is quite challenging." We all know that it is a challenge to distinguish diaphragmatic hernia of one twin, but it is not difficult to judge by prenatal and intrapartum ultrasonography, or what are the key points and difficulties in this process, please elaborate. 8. "In this case, the baby died the next day due to some complications from previous CDH primary repair using synthetic patches." What mesh related complications can lead to the child 's death the day after surgery? This should be highlighted. Is the background of the case’s history and progression described in sufficient detail? Partly Are enough details provided of any physical examination and diagnostic tests, treatment given and outcomes? Partly Is sufficient discussion included of the importance of the findings and their relevance to future understanding of disease processes, diagnosis or treatment? No Is the case presented with sufficient detail to be useful for other practitioners? No Competing Interests: No competing interests were disclosed. We confirm that we have read this submission and believe that we have an appropriate level of expertise to state that we do not consider it to be of an acceptable scientific standard, for reasons outlined above. Close READ LESS CITE CITE HOW TO CITE THIS REPORT Ma L and Liu C. Reviewer Report For: Case Report: Congenital diaphragmatic hernia in one twin followed by ex-utero intrapartum treatment (EXIT) procedure: Pearls & pitfalls [version 1; peer review: 2 not approved] . F1000Research 2024, 13 :44 ( https://doi.org/10.5256/f1000research.151480.r254457 ) The direct URL for this report is: https://f1000research.com/articles/13-44/v1#referee-response-254457 NOTE: it is important to ensure the information in square brackets after the title is included in all citations of this article. COPY CITATION DETAILS Report a concern Respond or Comment COMMENT ON THIS REPORT Views 0 Cite How to cite this report: Masahata K. Reviewer Report For: Case Report: Congenital diaphragmatic hernia in one twin followed by ex-utero intrapartum treatment (EXIT) procedure: Pearls & pitfalls [version 1; peer review: 2 not approved] . F1000Research 2024, 13 :44 ( https://doi.org/10.5256/f1000research.151480.r259539 ) The direct URL for this report is: https://f1000research.com/articles/13-44/v1#referee-response-259539 NOTE: it is important to ensure the information in square brackets after the title is included in this citation. Close Copy Citation Details Reviewer Report 03 Apr 2024 Kazunori Masahata , Pediatric surgery, Aizenbashi Hospital (Ringgold ID: 97811), Osaka, Osaka Prefecture, Japan Not Approved VIEWS 0 https://doi.org/10.5256/f1000research.151480.r259539 The paper by Pribadi et al. entitled “Congenital diaphragmatic hernia in one twin followed by ex-utero intrapartum treatment (EXIT) procedure: Pearls & pitfalls” is an interesting case report. The authors presented a rare case report of prenatally diagnosed CDH ... Continue reading READ ALL The paper by Pribadi et al. entitled “Congenital diaphragmatic hernia in one twin followed by ex-utero intrapartum treatment (EXIT) procedure: Pearls & pitfalls” is an interesting case report. The authors presented a rare case report of prenatally diagnosed CDH in one of the twins who underwent EXIT procedure without previous antepartum fetal therapy, especially in developing countries. They concluded that proper prenatal diagnosis with ultrasound evaluation from the second trimester and accurate preoperative planning with a multidisciplinary approach, followed by all necessary precautions and procedures are crucial for a successful EXIT-to-airway procedure. In general, this is a well-written paper that presents important findings. However, this paper needs major revisions to be accepted. I have the following concerns. ・Regarding CDH, several prenatal prognostic parameters of postnatal survival in fetuses with CDH such as lung-to-thorax transverse area ratio (L/T raio), observed-to-expected total fetal lung volume (o/e-TFLV), liver and stomach herniated into the thorax, have been reported. In the Introduction section, these prenatal predictors of CDH should be described. ・In the Introduction section, please describe the respiratory management of gentile ventilation in CDH and the defect size of the diaphragm, which was classified into the four scales from A to D as described by the CDHSG. The size of the diaphragmatic defect is associated with mortality rate and appears to be an important factor determining outcomes in patients with CDH. ・Regarding the presentation of the case, did the fetal echocardiographic findings show herniation of the liver into the chest? ・The figure legend describes that “lung hypoplasia prediction was also within normal limits,” but the LHR was 0.14 and the o/e LHR was 4.79%, which is severe CDH. Please correct this sentence. ・Please present the demographic data such as gestational age at birth, the use of ventilator settings and modes, pulmonary vasodilators, use of inhaled nitric oxide (NO), and ECMO support. In addition, postnatal echocardiographic findings including patent ductus arteriosus shunt flow should be presented. We believe it would help the reader to better understand the severity of pulmonary hypertension after birth. ・Please describe in detail the intraoperative findings and procedure for CDH repair. What defect size of the diaphragm is this case classified as a CDHSG system (A-D)? Did the surgical repair of the diaphragmatic defect perform by either closing the defect primarily or by means of a patch to reconstruct the diaphragm? ・Please provide intraoperative photographs if available. In addition, please describe any organs that are herniated into the thorax during surgery, as very little information is available at the surgery. ・Please discuss and describe in detail the postoperative course and cause of death. Regarding EXIT procedure, ・Please describe the surgical procedure of EXIT in detail: anesthesia method and fetal anesthesia for EXIT, method of airway management, time from the fetal head exposure to endotracheal intubation, time on placental support, total anesthesia or operative time, and amount of intraoperative blood loss. ・What medications were used to maintain to uterine relaxation during the EXIT procedure? Please describe in detail. ・In general, during the EXIT procedure, the mother requires general anesthesia and is in danger of increased blood loss due to the uterine relaxation, which is required to preserve the uteroplacental exchange. Did this mother need a blood transfusion? ・Although the EXIT procedure has been reported to be safe and effective, please discuss whether the procedure was an appropriate treatment for the case. I speculate that even if the EXIT procedure is given to extremely severe patient with CDH, it is too difficult to save this case. ・I would like to ask the authors the following comments: If a patient with the same severity of illness came to you, would you perform the EXIT procedure in the same way as you did this time? Is the background of the case’s history and progression described in sufficient detail? Partly Are enough details provided of any physical examination and diagnostic tests, treatment given and outcomes? Partly Is sufficient discussion included of the importance of the findings and their relevance to future understanding of disease processes, diagnosis or treatment? Partly Is the case presented with sufficient detail to be useful for other practitioners? Partly Competing Interests: No competing interests were disclosed. Reviewer Expertise: Pediatric surgery I confirm that I have read this submission and believe that I have an appropriate level of expertise to state that I do not consider it to be of an acceptable scientific standard, for reasons outlined above. Close READ LESS CITE CITE HOW TO CITE THIS REPORT Masahata K. Reviewer Report For: Case Report: Congenital diaphragmatic hernia in one twin followed by ex-utero intrapartum treatment (EXIT) procedure: Pearls & pitfalls [version 1; peer review: 2 not approved] . F1000Research 2024, 13 :44 ( https://doi.org/10.5256/f1000research.151480.r259539 ) The direct URL for this report is: https://f1000research.com/articles/13-44/v1#referee-response-259539 NOTE: it is important to ensure the information in square brackets after the title is included in all citations of this article. COPY CITATION DETAILS Report a concern Respond or Comment COMMENT ON THIS REPORT Comments on this article Comments (1) Version 1 VERSION 1 PUBLISHED 08 Jan 2024 Reader Comment 24 Jun 2025 Wiku Andonotopo , Obstetrics & Gynecology, Women Health Center, Fetomaternal Clinic, Eka Hospital BSD, Department of Obstetrics & Gynecology, Women Health Center, Fetomaternal Clinic, Eka Hospital BSD, Serpong, Tangerang, Banten, INDONESIA., Tangerang, Indonesia 24 Jun 2025 Reader Comment Dear Dr. Pribadi and Co-authors, Thank you for submitting your case report on a rare and complex presentation of congenital diaphragmatic hernia (CDH) in one twin with EXIT procedure . The ... Continue reading Dear Dr. Pribadi and Co-authors, Thank you for submitting your case report on a rare and complex presentation of congenital diaphragmatic hernia (CDH) in one twin with EXIT procedure . The topic is clinically important, particularly in showcasing the challenges of fetal diagnosis and surgical coordination in a developing country context. However, to make your manuscript worthy of publication in a peer-reviewed medical journal , several major areas must be addressed to meet professional and academic standards. �� MAJOR ISSUES TO FIX 1. ENGLISH LANGUAGE & GRAMMAR The manuscript requires substantial English editing to correct grammar, improve sentence structure, and reduce awkward phrasing. Many sentences are repetitive, overly long, or unclear. This affects reader comprehension and the scientific impact of your findings. �� Recommendation : Have the entire manuscript professionally proofread by a native English speaker or editing service. 2. STRUCTURE AND STYLE The abstract lacks clear structure (background, case description, conclusion) and includes unnecessary narrative elements. It should concisely reflect the content and key findings. Figures and videos are poorly integrated , with captions repeated or mismatched. There are redundant explanations (e.g., LHR and o/e LHR described multiple times across sections with the same phrasing). The main discussion lacks focus at times and wanders into generic textbook material rather than emphasizing what was unique about your case . �� Recommendation : Streamline the content. Focus more on critical insights from your case and how it advances understanding of CDH in twins and EXIT procedures in low-resource settings. 3. REFERENCES The citation format is inconsistent . Several citations are incomplete or out of place, and many PubMed links are pasted without being properly referenced. References need to follow a uniform style with journal abbreviation, year, volume, and page numbers. �� Recommendation : Reformat all references according to the target journal’s style guide (e.g., Vancouver style or AMA). 4. ETHICS AND REPORTING GUIDELINES There is no explicit statement of IRB approval or confirmation that the case followed the Declaration of Helsinki or institutional ethical standards. While CARE checklist use is mentioned, this section is underemphasized. �� Recommendation : Add a formal Ethical Approval section in the methods or end of the manuscript, even if local guidelines do not require approval for single case reports. 5. CLINICAL DEPTH AND FOLLOW-UP The report would be strengthened by: Postmortem insights (if available) explaining the complications leading to death. A clearer justification for choosing EXIT without prenatal intervention, considering an o/e LHR of only 4.79% (extremely poor prognosis). Discussion of the technical aspects of EXIT and surgery—what could have been done differently? �� Recommendation : Provide deeper critical analysis of the decision-making process and outcomes. Expand on limitations and what clinicians in similar settings should take away. ✅ STRONG POINTS The case is original and rare. Prenatal imaging is well-documented. Ethical patient consent is addressed. It gives valuable insight into fetal surgery in developing countries. �� BOTTOM LINE Your case is important, but it is not yet ready for indexing in its current form. With substantial improvements in language, structure, reference formatting, ethical transparency, and focused clinical insight, it could become a valuable contribution to fetal medicine literature. Please consider revising carefully with these points in mind. I would encourage resubmission once these issues are addressed. Respectfully, Wiku Andonotopo, MD, PhD, HDGO, FICS. Department of Obstetrics & Gynecology, Women Health Center, Fetomaternal Clinic, Eka Hospital Serpong, Tangerang, Banten, INDONESIA Dear Dr. Pribadi and Co-authors, Thank you for submitting your case report on a rare and complex presentation of congenital diaphragmatic hernia (CDH) in one twin with EXIT procedure . The topic is clinically important, particularly in showcasing the challenges of fetal diagnosis and surgical coordination in a developing country context. However, to make your manuscript worthy of publication in a peer-reviewed medical journal , several major areas must be addressed to meet professional and academic standards. �� MAJOR ISSUES TO FIX 1. ENGLISH LANGUAGE & GRAMMAR The manuscript requires substantial English editing to correct grammar, improve sentence structure, and reduce awkward phrasing. Many sentences are repetitive, overly long, or unclear. This affects reader comprehension and the scientific impact of your findings. �� Recommendation : Have the entire manuscript professionally proofread by a native English speaker or editing service. 2. STRUCTURE AND STYLE The abstract lacks clear structure (background, case description, conclusion) and includes unnecessary narrative elements. It should concisely reflect the content and key findings. Figures and videos are poorly integrated , with captions repeated or mismatched. There are redundant explanations (e.g., LHR and o/e LHR described multiple times across sections with the same phrasing). The main discussion lacks focus at times and wanders into generic textbook material rather than emphasizing what was unique about your case . �� Recommendation : Streamline the content. Focus more on critical insights from your case and how it advances understanding of CDH in twins and EXIT procedures in low-resource settings. 3. REFERENCES The citation format is inconsistent . Several citations are incomplete or out of place, and many PubMed links are pasted without being properly referenced. References need to follow a uniform style with journal abbreviation, year, volume, and page numbers. �� Recommendation : Reformat all references according to the target journal’s style guide (e.g., Vancouver style or AMA). 4. ETHICS AND REPORTING GUIDELINES There is no explicit statement of IRB approval or confirmation that the case followed the Declaration of Helsinki or institutional ethical standards. While CARE checklist use is mentioned, this section is underemphasized. �� Recommendation : Add a formal Ethical Approval section in the methods or end of the manuscript, even if local guidelines do not require approval for single case reports. 5. CLINICAL DEPTH AND FOLLOW-UP The report would be strengthened by: Postmortem insights (if available) explaining the complications leading to death. A clearer justification for choosing EXIT without prenatal intervention, considering an o/e LHR of only 4.79% (extremely poor prognosis). Discussion of the technical aspects of EXIT and surgery—what could have been done differently? �� Recommendation : Provide deeper critical analysis of the decision-making process and outcomes. Expand on limitations and what clinicians in similar settings should take away. ✅ STRONG POINTS The case is original and rare. Prenatal imaging is well-documented. Ethical patient consent is addressed. It gives valuable insight into fetal surgery in developing countries. �� BOTTOM LINE Your case is important, but it is not yet ready for indexing in its current form. With substantial improvements in language, structure, reference formatting, ethical transparency, and focused clinical insight, it could become a valuable contribution to fetal medicine literature. Please consider revising carefully with these points in mind. I would encourage resubmission once these issues are addressed. Respectfully, Wiku Andonotopo, MD, PhD, HDGO, FICS. Department of Obstetrics & Gynecology, Women Health Center, Fetomaternal Clinic, Eka Hospital Serpong, Tangerang, Banten, INDONESIA Competing Interests: Competing Interests: I have no competing interests to declare. Close Report a concern Comment ADD YOUR COMMENT keyboard_arrow_left keyboard_arrow_right Open Peer Review Reviewer Status info_outline Alongside their report, reviewers assign a status to the article: Approved The paper is scientifically sound in its current form and only minor, if any, improvements are suggested Approved with reservations A number of small changes, sometimes more significant revisions are required to address specific details and improve the papers academic merit. Not approved Fundamental flaws in the paper seriously undermine the findings and conclusions Reviewer Reports Invited Reviewers 1 2 Version 1 08 Jan 24 read read Kazunori Masahata , Aizenbashi Hospital (Ringgold ID: 97811), Osaka, Japan Lishuang Ma , Capital Institute of Pediatrics, Beijing, China Chao Liu , Capital Institute of Pediatrics, Beijing, China Comments on this article All Comments (1) Add a comment Sign up for content alerts Sign Up You are now signed up to receive this alert Browse by related subjects keyboard_arrow_left Back to all reports Reviewer Report 0 Views copyright © 2024 Ma L et al. This is an open access peer review report distributed under the terms of the Creative Commons Attribution License , which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. 30 May 2024 | for Version 1 Lishuang Ma , Department of Pediatric and Neonatal Surgery, Capital Institute of Pediatrics, Beijing, China Chao Liu , Department of Pediatric and Neonatal Surgery, Capital Institute of Pediatrics, Beijing, China 0 Views copyright © 2024 Ma L et al. This is an open access peer review report distributed under the terms of the Creative Commons Attribution License , which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. format_quote Cite this report speaker_notes Responses (0) Not Approved info_outline Alongside their report, reviewers assign a status to the article: Approved The paper is scientifically sound in its current form and only minor, if any, improvements are suggested Approved with reservations A number of small changes, sometimes more significant revisions are required to address specific details and improve the papers academic merit. Not approved Fundamental flaws in the paper seriously undermine the findings and conclusions This study aimed to report a rare case while also analyzing the pearls and pitfalls in diagnosing and managing this case. However, throughout the full text, the article did not describe the highlights of the experience of the diagnosis and treatment of this patient, nor extracted the lessons that should be learned and the hidden dangers that need to be avoided, but generally made a literature review, and the cited literature was older. It is not unusual for one twin to have CDH. Therefore, this article does not reflect the scientific value and guiding significance for clinical practice. 1. "Milder cases, on the other hand, might not be discovered until later due to minor defects that lead on to cause many complications." Smaller and less severe defects may be detected later, and they may present clinically slightly after birth, some even without respiratory or circulatory disturbances, but only incidentally when chest radiographs are examined for other reasons. However, incarceration of herniated thoracic organs may also occur with critical illness and be first diagnosed. This is inconsistent with the authors' view. 2. "This case is very rare since diagnosing one of the twins with CDH during the intrapartum period to determine which one will undergo EXIT procedure is quite challenging." We all know that it is a challenge for twin children to distinguish diaphragmatic hernia, but it is not difficult to judge by prenatal and intrapartum ultrasonography, or what are the key points and difficulties in this process, please elaborate. 3. "The first baby was a male born weighing 1,600 grams with a 1st minute APGAR score of 3 and the 5th minute APGAR score was 5." Describe in detail how to judge a fetus with diaphragmatic hernia during EXIT. Why is this process challenging? 4. "Unfortunately, the baby died the next day due to some complications from previous CDH primary repair." What complication? What is it related to? Please specify. 5. "As the pearl of this case, advances in prenatal diagnosis played an important role because this method allows the detection of a number of birth anomalies that can obstruct the fetal airway or obstruct ventilation at birth." There was no particular point in this case. The birth process for one twin to have CDH is indeed crucial, but not specific for the EXIT process. Please specify. 6. "The patient in this case was diagnosed prenatally with CDH on the first fetus with an o/e LHR of 4.79%, thus this condition was considered a severe CDH." Lung head ratio and o/e lung head ratio are indeed very important for the assessment of CDH, but the accuracy of detection is not optimistic in clinical tests. However, for the children in this treatise, please give necessary explanation on what is the special significance and how it is different from the evaluation of singleton CDH children. 7. "As another pearl, diagnosing one of the twins with CDH during intrapartum period to determine which one will undergo EXIT procedure is quite challenging." We all know that it is a challenge to distinguish diaphragmatic hernia of one twin, but it is not difficult to judge by prenatal and intrapartum ultrasonography, or what are the key points and difficulties in this process, please elaborate. 8. "In this case, the baby died the next day due to some complications from previous CDH primary repair using synthetic patches." What mesh related complications can lead to the child 's death the day after surgery? This should be highlighted. Is the background of the case’s history and progression described in sufficient detail? Partly Are enough details provided of any physical examination and diagnostic tests, treatment given and outcomes? Partly Is sufficient discussion included of the importance of the findings and their relevance to future understanding of disease processes, diagnosis or treatment? No Is the case presented with sufficient detail to be useful for other practitioners? No Competing Interests No competing interests were disclosed. We confirm that we have read this submission and believe that we have an appropriate level of expertise to state that we do not consider it to be of an acceptable scientific standard, for reasons outlined above. reply Respond to this report Responses (0) Ma L and Liu C. Peer Review Report For: Case Report: Congenital diaphragmatic hernia in one twin followed by ex-utero intrapartum treatment (EXIT) procedure: Pearls & pitfalls [version 1; peer review: 2 not approved] . F1000Research 2024, 13 :44 ( https://doi.org/10.5256/f1000research.151480.r254457) NOTE: it is important to ensure the information in square brackets after the title is included in this citation. The direct URL for this report is: https://f1000research.com/articles/13-44/v1#referee-response-254457 keyboard_arrow_left Back to all reports Reviewer Report 0 Views copyright © 2024 Masahata K. This is an open access peer review report distributed under the terms of the Creative Commons Attribution License , which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. 03 Apr 2024 | for Version 1 Kazunori Masahata , Pediatric surgery, Aizenbashi Hospital (Ringgold ID: 97811), Osaka, Osaka Prefecture, Japan 0 Views copyright © 2024 Masahata K. This is an open access peer review report distributed under the terms of the Creative Commons Attribution License , which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. format_quote Cite this report speaker_notes Responses (0) Not Approved info_outline Alongside their report, reviewers assign a status to the article: Approved The paper is scientifically sound in its current form and only minor, if any, improvements are suggested Approved with reservations A number of small changes, sometimes more significant revisions are required to address specific details and improve the papers academic merit. Not approved Fundamental flaws in the paper seriously undermine the findings and conclusions The paper by Pribadi et al. entitled “Congenital diaphragmatic hernia in one twin followed by ex-utero intrapartum treatment (EXIT) procedure: Pearls & pitfalls” is an interesting case report. The authors presented a rare case report of prenatally diagnosed CDH in one of the twins who underwent EXIT procedure without previous antepartum fetal therapy, especially in developing countries. They concluded that proper prenatal diagnosis with ultrasound evaluation from the second trimester and accurate preoperative planning with a multidisciplinary approach, followed by all necessary precautions and procedures are crucial for a successful EXIT-to-airway procedure. In general, this is a well-written paper that presents important findings. However, this paper needs major revisions to be accepted. I have the following concerns. ・Regarding CDH, several prenatal prognostic parameters of postnatal survival in fetuses with CDH such as lung-to-thorax transverse area ratio (L/T raio), observed-to-expected total fetal lung volume (o/e-TFLV), liver and stomach herniated into the thorax, have been reported. In the Introduction section, these prenatal predictors of CDH should be described. ・In the Introduction section, please describe the respiratory management of gentile ventilation in CDH and the defect size of the diaphragm, which was classified into the four scales from A to D as described by the CDHSG. The size of the diaphragmatic defect is associated with mortality rate and appears to be an important factor determining outcomes in patients with CDH. ・Regarding the presentation of the case, did the fetal echocardiographic findings show herniation of the liver into the chest? ・The figure legend describes that “lung hypoplasia prediction was also within normal limits,” but the LHR was 0.14 and the o/e LHR was 4.79%, which is severe CDH. Please correct this sentence. ・Please present the demographic data such as gestational age at birth, the use of ventilator settings and modes, pulmonary vasodilators, use of inhaled nitric oxide (NO), and ECMO support. In addition, postnatal echocardiographic findings including patent ductus arteriosus shunt flow should be presented. We believe it would help the reader to better understand the severity of pulmonary hypertension after birth. ・Please describe in detail the intraoperative findings and procedure for CDH repair. What defect size of the diaphragm is this case classified as a CDHSG system (A-D)? Did the surgical repair of the diaphragmatic defect perform by either closing the defect primarily or by means of a patch to reconstruct the diaphragm? ・Please provide intraoperative photographs if available. In addition, please describe any organs that are herniated into the thorax during surgery, as very little information is available at the surgery. ・Please discuss and describe in detail the postoperative course and cause of death. Regarding EXIT procedure, ・Please describe the surgical procedure of EXIT in detail: anesthesia method and fetal anesthesia for EXIT, method of airway management, time from the fetal head exposure to endotracheal intubation, time on placental support, total anesthesia or operative time, and amount of intraoperative blood loss. ・What medications were used to maintain to uterine relaxation during the EXIT procedure? Please describe in detail. ・In general, during the EXIT procedure, the mother requires general anesthesia and is in danger of increased blood loss due to the uterine relaxation, which is required to preserve the uteroplacental exchange. Did this mother need a blood transfusion? ・Although the EXIT procedure has been reported to be safe and effective, please discuss whether the procedure was an appropriate treatment for the case. I speculate that even if the EXIT procedure is given to extremely severe patient with CDH, it is too difficult to save this case. ・I would like to ask the authors the following comments: If a patient with the same severity of illness came to you, would you perform the EXIT procedure in the same way as you did this time? Is the background of the case’s history and progression described in sufficient detail? Partly Are enough details provided of any physical examination and diagnostic tests, treatment given and outcomes? Partly Is sufficient discussion included of the importance of the findings and their relevance to future understanding of disease processes, diagnosis or treatment? Partly Is the case presented with sufficient detail to be useful for other practitioners? Partly Competing Interests No competing interests were disclosed. Reviewer Expertise Pediatric surgery I confirm that I have read this submission and believe that I have an appropriate level of expertise to state that I do not consider it to be of an acceptable scientific standard, for reasons outlined above. reply Respond to this report Responses (0) Masahata K. Peer Review Report For: Case Report: Congenital diaphragmatic hernia in one twin followed by ex-utero intrapartum treatment (EXIT) procedure: Pearls & pitfalls [version 1; peer review: 2 not approved] . F1000Research 2024, 13 :44 ( https://doi.org/10.5256/f1000research.151480.r259539) NOTE: it is important to ensure the information in square brackets after the title is included in this citation. The direct URL for this report is: https://f1000research.com/articles/13-44/v1#referee-response-259539 Alongside their report, reviewers assign a status to the article: Approved - the paper is scientifically sound in its current form and only minor, if any, improvements are suggested Approved with reservations - A number of small changes, sometimes more significant revisions are required to address specific details and improve the papers academic merit. Not approved - fundamental flaws in the paper seriously undermine the findings and conclusions Adjust parameters to alter display View on desktop for interactive features Includes Interactive Elements View on desktop for interactive features Competing Interests Policy Provide sufficient details of any financial or non-financial competing interests to enable users to assess whether your comments might lead a reasonable person to question your impartiality. 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