Despite decades and multiple techniques described, the management of GO is still debatable. Currently, two main strategies are available for GO repair, the staged surgical closure (SSC) and the nonoperative delayed closure (NDC), and there is literature background to support both [8, 9].
NDC is usually accomplished with the use dressings and topical agents on the omphalocele sac to stimulate epithelization of the membrane and are followed by an interval repair months later [9, 13]. Its main benefits include shorter time to enteral feeds and less morbidity.
For SSC, many techniques have been described such as placement of surgical silos [14], nonsurgical external silos [15], intraabdominal tissue expanders[16] and external compression methods[17–19] until complete reduction of the omphalocele content. All these are followed by another intervention for final closure. The main benefits of this approach include faster time to repair and discharge with already corrected defect.
Mostly all SSC techniques usually demand more than one trip to the OR to achieve abdominal wall closure, and these patients are usually required to be under mechanical ventilation, sedation and/or muscle paralysis throughout the process.
Furthermore, all these methods, even in the nonoperative approach, usually require additional techniques to be applied at the time of surgery such as component separation, skin or fascial flaps, negative pressure wound therapy and/or prosthesis implant [9, 15, 20–23]. Adding any of these techniques can significantly increase surgical morbidity and postoperative complications as well as recurrence rates [19, 24].
It has already been stated the benefits and the efficacy of the BTA application for the repair of large abdominal wall defects in adults [10] and there is evidence of its use for other purposes in children and neonates [12, 25].
A recent study in the adult population by Jacombs et al. [26] showed that 76.6% of patients with a defect from 5 to 11.9cm could be repaired with the use of BTA alone, and 20% of patients with a defect larger than 12cm. They also reported a mean unstretched length gain of 4.2cm per side.
Recently, we reported the first cases in the literature of BTA application in children with hernias secondary to GO. Both were performed in association with preoperative progressive pneumoperitoneum (PPP) [5, 11]. In the second and most complex case [5], the PPP was clearly not successful due to intraabdominal adhesions, but it was still possible to successfully achieve abdominal wall closure with no additional techniques. That made us wonder about the results of BTA alone in neonates with GO.
In both cases reported in this article, final surgical outcome was excellent. Midline approximation was easily performed, even in the subxiphoid region where usually the defect is larger, there is less aponeurosis, and the rigidity of the rib cage can hinder closure. In neither case an additional technique or a mesh reinforcement or patch were required for closure. Postoperative intraabdominal pressures were surprisingly low and neither had any ventilatory repercussions. Both could be extubated as soon as they were in condition to do so and had an uneventful recovery with a fast discharge.
During the process until surgery both patients had minimal manipulation with spaced dressing changes, only requiring sedation during BTA application, without need for mechanical ventilation, were both on full enteral feeds by the first week of life, were able to breast feed and were always in close contact with their parents.
Only minor skin complications were observed, that resolved with dressings and hygiene alone, without the need for antibiotics or suture removal. Despite this, aesthetic outcome was great in both cases. No skin necrosis was noted, no seroma or local infections. No complications regarding the use of BTA were seen, although they are rare [12]. After a follow-up of 8 and 24 months, no signs of recurrence, and both patients are developing well.
Regardless the choice of initial management, the final goal is always the same, to achieve fascial closure.
Our method reaches a middle ground between the staged repair in the neonatal period and the nonoperative delayed techniques. It does not require aggressive interventions early in life and discharges the patient with a repaired defect, apparently with a high chance of primary closure without additional techniques or the need for prosthesis use.
We understand the patients in our study did not have major malformations nor important pulmonary hypoplasia, which certainly contributed to the good outcome. Still, that does not nullify the fact that the process is more amenable by requiring less invasive procedures.
The application of BTA in the abdominal wall is a quite simple technique, that can be performed in any center, considering the availability of BTA and ultrasound equipment. It is mandatory the use of ultrasound for correct visualization of the needle and toxin infiltration, and to minimize possible complications. It is also important to infiltrate at least the two most superficial layers, the OE and IO [10]. A low volume injection per site seems to be better since the small space available in order to provide good ultrasound visualization.
It is also known that the synergy of stretching the pre-paralyzed lateral abdominal wall muscles can increase lateral gain [10]. This stretching can be either passive or active, which suggests that not just the compression itself, but possibly the awake and conscious neonate that is in constant motion, could alone, contribute to the stretching.
At the time of surgical correction, skin flaps should be raised until the aspect of “corset” is lost, which was usually at the level of the midaxillary lines. Excess of skin should only be removed after complete plicature of the flaps to the aponeurosis. Even though we could easily approximate the midline without additional techniques such as abdominal component separation or the use of prothesis, we still recommend having a mesh or patch available at the OR in the case of impossibility to close the midline or in the case of high intraabdominal pressures at the time of closure.
It is still early, but apparently this technique shows the following benefits: it is safe and is easily reproducible, does not demand intubation nor ventilation, does not need multiple trips to the operating room or submit the child to repeated sedation, enteral feeding is allowed and encouraged, maintains the mother-child bonding, apparently doesn’t require additional techniques or prosthesis for closure, and ultimately discharges the patient with a repaired defect. Nevertheless, it might increase costs due to time of hospitalization and the use of the toxin itself.
Another positive point in this management is the possibility to associate different methos for abdominal gain of domain, thus facilitating even more the final closure.
The literature confirming that BTA facilitates the repair of ventral hernias in the adult population has increased dramatically and it is just time until it has its proven benefits in children.
Due to giant omphalocele’s complexity, there is and there will always be room for developing new techniques or refining the previous ones, and the important is to keep advancing to further improve these patient’s care. Our objective in this study is to describe the technique of BTA application in neonates, and to provide another asset when managing this complicated condition. It is reasonable to say that this technique is in its early days, and we encourage further studying and present some of the questions that need to be assessed: (1) minimal BTA dosage needed for expected effect on the abdominal wall, (2) costs of the treatment, (3) is BTA required for all GO cases, (4) is BTA alone sufficient for all GO cases. We believe that this technique can be a great asset and we hope it will aid other surgeons and children worldwide.