key: cord-1023539-8jwxzofr authors: Putri, Nandita Melati; Kreshanti, Prasetyanugraheni; Aulia, Indri; Syarif, Akhmad Noviandi; Tunjung, Narottama; Sukasah, Chaula Luthfia title: Use of Local Perforator Flaps for Closure of a Thoraco-Omphalopagus Conjoined Twin Defect after Separation during the COVID-19 Pandemic date: 2021-06-30 journal: Heliyon DOI: 10.1016/j.heliyon.2021.e07443 sha: 6b880866b923553aa6a5c6f97723263aa01c948e doc_id: 1023539 cord_uid: 8jwxzofr INTRODUCTION: Conjoined twins are a rare medical phenomenon that poses unique challenges for surgeons. Separation of conjoined twins involves multidisciplinary teamwork, complex medical management and surgical planning, and multi-stage operations and often still has a high mortality and morbidity rate. In the times of the COVID-19 pandemic, separation of conjoined twins pose even greater challenges. Aiming for the best outcome possible, while minimizing the risk of COVID transmission and ensuring the safety of the personnel, is paramount. This case report presents thoraco-omphalopagus twins who were successfully separated at 4 months of age. The preoperative planning, operative details, postoperative follow-ups, and outcomes are discussed. METHODS: The absence of a tissue expander and the inability to acquire it due to travel restrictions from COVID-19 further complicated the management on this patient. A Routine Polymerase Chain Reaction (PCR) swab test was performed on the patients and personnel. Standardized personnel protective equipment (PPE) was worn during ward and surgical care. After separation of the twins by cardiothoracic and pediatric surgeons, one twin underwent immediate skin closure using a double keystone perforator island flap and a lower abdominal perforator flap. Due to extensive defects, closure was delayed for the second twin. After a series of dressing changes, eventually local perforator flaps could be raised to close the defect using staged tension sutures and skin grafts for secondary defects. RESULTS: Both twins were discharged with no significant morbidity, and no personnel were exposed to COVID-19 infection during the management CONCLUSION: Preoperative coordination and planning, multidisciplinary effort, adherence to screening protocols for COVID, and strict use of standardized PPE all contributed to the successful separation of thoraco-omphalopagus conjoined twins during the COVID-19 pandemic. Conjoined twins are a rare medical phenomenon that occurs as a result of monoamniotic and monochorionic gestation, when two identical individuals are joined by part of their anatomy and share one or more organs [1] . The incidence of conjoined twins ranges from 1:50,000 to 1:100,000 live births [2, 3] . Based on the classification proposed by Spencer et al [4] , thoracoomphalopagus conjoined twins join from the thoracal region to the umbilical with varying degree of fusion of heart, liver, and gastrointestinal tract. Separation surgery of conjoined twins still results in high mortality and morbidity [5] . In the times of COVID-19 pandemic, especially in a developing country where the cases are still increasing and peak of the curve is not yet reached [6] , the risks of disease transmission both to the patient and to the surgical teams are multiplied [7, 8] . Furthermore, a shortage of personal protective equipment, the inability to acquire tissue expanders that are widely used for conjoined twin separation, and the limited capacity of intensive care units and hospital resources also pose even greater challenges in the management of conjoined twins. Most reports on conjoined twins have mainly focused on the multidisciplinary teamwork and outcome of the separation, [1, 3, 5, [9] [10] [11] [12] but only a few reports have described the design and use of flaps for defect closure after separation. [13, 14] In this report, we present a case of a set of conjoined twins undergoing separation surgery. Our focus in this study is the use of local perforator flaps of the thoracoabdominal as soft tissue coverage of the defect after separation. We also present mitigation strategies for performing conjoined twin surgeries to prevent the risk of COVID transmission among the surgical teams, while still aiming for the best and safest outcome possible for the patients. The separation was a success technically, although one of the twins only survived a few weeks after being discharged. A set of female twins, weighing 3.5 kg at birth, were born prematurely. The twins were joined from the chest to abdomen. The caesarean delivery was performed in the regional hospital, and the twins were directly referred to our tertiary hospital and admitted to the neonatal intensive care unit (NICU). A multidisciplinary team consisting of pediatricians, J o u r n a l P r e -p r o o f anesthesiologists, pediatric surgery team, cardio-thoracic surgery team, and plastic surgery team was assembled to plan the separation. The cost for the separation surgery and inpatient care were all covered by the hospital, considering this was a really rare case. Informed consent was obtained from the patients for publication of the case report details. The study was performed in accordance with the principles of the Declaration of Helsinki. The twins were joined from the lower anterior thorax to the level of the umbilicus. The fusion was at the midline, and the area of skin on the lateral parts of the twins were relatively symmetrical. The CT scan imaging revealed that they shared a rib cage with 2 pairs of lungs and 2 separate hearts, and a shared liver, but with separate GI tracts ( Figure 1 ). Other than the fused parts, both babies showed normal anatomical presentations and normal extremities. One of the twins had smaller body proportions and was named Baby 1. After admission to the NICU, Baby 1 developed respiratory distress and was put on a ventilator. Both babies were screened for COVID-19 and Baby 1 was found positive for COVID-19. After a series of hemodynamic surges and ventilator-associated pneumonia, Baby 1 was finally stable at the age of 5 months, and the Polymerase Chain Reaction (PCR) test for COVID-19 was negative. To avoid further instability and deterioration in the hemodynamics of Baby 1, the separation surgery was scheduled at this period. See Table 1 for the timeline of multiple surgery on both babies. Both babies showed positive results for a rapid COVID-19 test during preoperative screening, ten days after the separation surgery. This made further management of the patients more complicated because they had to be moved to the COVID ward before finally testing negative twice by PCR for COVID-19. They were then transferred back to the PICU in the non-COVID ward. Closure of the thoracal defect and partial abdominal defect using double keystone perforator island flaps, an abdominal flap, and NPWT Debridement and resuturing of the flap, and applying NPWT* July 28, 2020 Debridement and resuturing of the flap, and applying NPWT* August 4, 2020 Re-elevation of the flap, dynamic suturing, and NPWT August 12, 2020 Debridement and primary closure September 7, 2020 STSG for the secondary defects NPWT: Negative-pressure wound therapy, STSG: Split-thickness skin graft *Period of suspected COVID-19 re-infection Initial preoperative screening tests for COVID-19 using the PCR test twice for each baby from oropharyngeal swabs and endotracheal swabs resulted in all negative tests for the twins preoperatively, so the first surgery to separate the babies was performed. The incision of the skin was done in a straight-line manner. The separation surgery was performed by a pediatric surgery team and a cardio-thoracic surgery team to separate the liver and rib cage. The liver was separated using cavitron ultrasonic surgical aspirator, a branch of portal vein which cross over the separation line was divided and ligated, the parenchyma was then sutured with polypropylene. The surgery was then continued by the plastic surgery team. The defect on Baby 2 was 9 × 15 cm, with the chest and abdomen covered by mesh. Perforators from the lateral abdomen were identified using hand-held Doppler ultrasound. Bilateral keystone design perforator island flaps (KDPIF) were designed, with the area of the perforators included in the design. The flaps were incised and elevated subfascially. More than fifty percent of the keystone design perforator island flaps were undermined for mobility, and the flaps were mobilized to close the thoracal and upper abdominal defect with an omega design, with a third flap from the lower abdominal flap advanced cranially to close the remaining abdominal defect. All the defects from Baby 2 were able to be covered in the separation surgery ( Figure 2 ). After the separation, the abdomen of Baby 1 was covered with mesh. A part of the chest wall was reconstructed with miniplates and the heart was covered with Gore-tex®. The defect on Three days after the separation, Baby 1 was scheduled for surgery to close the defect. Perforators from lateral thoracic area were identified with handheld Doppler ultrasound, and then the KDPIF was elevated in an omega manner to close the defect mainly to cover the chest with the exposed Gore-tex® and miniplates. A third perforator flap from the lower abdomen was raised to cover the lower abdominal area. After insetting the flap, a defect remained on the abdomen with a size of 6 × 10 cm (Figure 3 ). The remaining defect was covered with negative pressure wound therapy (NPWT). remaining defects. We re-elevated the lower limb of the right keystone perforator island flap and the lower abdominal perforator flap to cover the remaining defects ( Figure 3 ). Finally, almost 2 months after the separation, Baby 1 underwent the last surgery with a split thickness skin graft (STSG) to cover the secondary defects on the flank. All the defects were epithelialized and the patient was also discharged 1 week later (Figure 4 ). All the surgeries were performed with as minimal personnel as possible to reduce the risk of COVID transmission. Every surgical workforce must wear standardized personnel protective equipment according to the level determined by the hospital. The use of N95 masks and eye protectors was mandatory for surgery in the non COVID OR, while additional hazmat and boots were used in the COVID OR. During the follow up period, the twins showed good healing of the skin on the chest and abdomen and significant gains in the body weight. Unfortunately, two weeks after being discharged, Baby 1 developed symptoms of pneumonia, with fever and shortness of breath. Before having the chance to be brought back to our unit, Baby 1 passed away. The death was suspected to be related to COVID-19 but no autopsy was performed due to the cultural beliefs of the parents. Currently, Baby 2 is healthy, has normal growth, and is still under routine follow-up. The risk for ventral hernia will be assessed during follow-up and abdominal wall reconstruction will be done accordingly one until three years after the separation surgery [15] . Conjoined twins are regarded as monozygotic, monochorionic twins of the same sex with an identical chromosomal pattern [2, 4] . One of the most common types of conjoined twins is the thoraco-omphalopagus type, which accounts for 75% of these cases [16] . Surgical separation of these twins has been always a challenge and may require novel techniques. One of the challenges of the separation is tissue coverage of the defect after separation. Most cases of thoraco-omphalopagus conjoined twins result in large defects of the anterior chest and abdomen [5, 9] . Numerous strategies for soft tissue coverage have been reported. A variety of skin grafts and skin flaps and the use of tissue expander procedures have been described. Skin grafts, however, require an intact body wall for use in closure [17] . Skin flaps have been successful and reliable for tissue coverage [13, 14] , as well as pedicled flaps such as latissimus dorsi flap and superficial circumflex iliac artery perforator flap [18, 19] Tissue expanders have arisen as an optimal means of achieving wound closure in conjoined twin separation, with the first reported success in 1986 [20] . However, the use of tissue expanders has its own disadvantages, namely the need for multiple procedures and the possibility of the expander creating pressure into the abdominal cavity and causing intraabdominal compartment syndrome [5] . In this study, we used local perforator flaps from the lateral chest and abdomen area to close the defects on the anterior chest and abdomen. In this case, we used perforators from the lateral thoracic arteries. We used the hand-held doppler ultrasound to estimate the location of the perforator. A color doppler ultrasound would be more precise but it is not a standard operational procedure in our hospital and we tried to minimize the number of person involved in the management of the patients. We postulate that perforators from this region are a reliable source of vessels for the flaps. Based on the studies by Hocaoglu and Aydin [21] , Offman et al. [22] , and Taylor et al. [23] , the perforators in the flank region arise from the intercostal, lumbar, lateral thoracic, and thoracodorsal arteries. We opted for a keystone therefore, the respiratory condition of Baby 1 had not been as good as that of Baby 2. Neither PCR swab test or antigen swab examination was done to baby 1 due to socioeconomic issues, but during the postoperative visits, baby 2 and the mother also had a positive antigen swab test of COVID-19 with no respiratory symptoms. 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We thank the 5 th ICE on IMERI committee, who had supported the peer review and manuscript preparation before submitting it to the journal.J o u r n a l P r e -p r o o f