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Neonatal Total Gastric Rupture with Bilateral Pneumoscotum in a Preterm Infant

Hemant Kumar, Ramnik Patel, Bharat More, Bala Eradi, Haitham Dagash, Ashok Rajimwale

Department of Paediatric Surgery, Leicester Royal Infirmary, University Hospitals of Leicester NHS Trust, Infirmary Square, Leicester, United Kingdom

 

Correspondence:

Hemant Kumar

Department of Paediatric Surgery

Leicester Royal Infirmary

University Hospitals of Leicester

NHS Trust Infirmary Square Leicester LE1 5WW

United Kingdom

Phone: 00447956896641, Fax: 00441162586089

E-mail: This email address is being protected from spambots. You need JavaScript enabled to view it.

 

Abstract

A preterm infant and twin 1 of the dichorionic diamniotic twins born at 27+3/40 weeks gestation weighting 790 grams who had on-going respiratory distress developed total gastric rupture following difficult intubation. Plain chest, abdominal and right lateral decubitus radiographs helped in diagnosis. Infant had resuscitation and underwent exploratory laparotomy, closure of massive gastric rupture and temporary gastrostomy for decompression initially and feeding later uneventfully. Neonatal gastric perforation is rare, serious and potentially lethal if not detected early and treated effectively. It could be traumatic, spontaneous or ischaemic. Our case had acute barotrauma following accidental oesophageal intubation and closed loop obstruction due to cardiac and pyloric sphincter at both ends and relatively high velocity air entering under pressure leading to anterior gastric wall blast. Primary repair of the stomach rupture with temporary gastrostomy is very useful and effective therapeutic strategy in these very sick preterm infants.

Keywords: preterm, gastric perforation, pneumoperitoneum, acute pneumoscotum, peritonitis, neonatal, gastrostomy, traumatic, iatrogenic

 

Introduction

Neonatal gastric perforation if rare but acutely catastrophic event and if not recognized and treated in time could be fatal [1]. Massive pneumoperitoneum with a rare air leak in an infant with bronchiolitis and high-frequency oscillatory ventilation without any gastrointestinal perforation have been reported on the other extreme requiring no treatment [2]. Massive neonatal pneumovesical distention may present as an acute abdomen in an intensive care unit environment [3]. We wish to report an extreme preterm and very low birth weight twin1 infant who had near total gastric rupture following difficult and traumatic intubation who recovered well with prompt and appropriate treatment.

Case report

Twin 1 male infant was born at 27+3 weeks weighing 790grams by vaginal delivery with vertex presentation. Dating scan at 14+1 revealed spontaneous dichorionic diamniotic (DCDA) twin pregnancy. Anomaly scan at 20+2 showed no obvious foetal defects for both twins. Growth scan at 24+3 weeks revealed normal growth and amniotic fluid indices for both twins. Emergency admission at 27+2 weeks gestation with irregular tightenings rapidly progressed to labour and delivery. Mother has used cannabis during pregnancy but had not taken any prescribed medications. One dose of steroids was given 3 hours prior to delivery.

Twin 1 was born in good condition, however, his respiratory effort quickly deteriorated and he was intubated and ventilated from birth. He received two doses of surfactant. A brief trial of continuous positive airway pressure (CPAP) had failed and hence he continued to be ventilated. He had respiratory distress leading to chronic lung disease, ventricular septal defect, patent ductus arteriosus, neonatal cardiac failure, pulmonary oedema, bilateral inguinal hernias, retinopathy of prematurity grade 2 and later on developed urea plasma positive nasopharyngeal aspiration (NPA) which was treated with 14 days of clarithromycin.

Despite ventilation with high pressures, his gases continued to show worsening respiratory acidosis and he was changed to high frequency oscillation on day three. After 24 hours on high frequency oscillation, infant was weaned back to conventional ventilation. He was again extubated to CPAP on day six but required re-intubation secondary to deterioration. The re-intubation was difficult and failed twice. Soon after the intubation, infant was noted to have deterioration of general condition and a tense distended abdomen and scrotum with blood stained nasogastric aspirates. Transillumination of the abdominal and scrotum wall was brilliant and suggested pneumoperitoneum.

The haematological and biochemical investigations were all within normal limits. Chest radiograph showed picture of advanced bilateral respiratory distress and massive free gas under both domes of diaphragm and saddle bag sign and nasogastric tube was going down into the pelvis (Fig. 1). An abdominal radiograph revealed free air in the abdomen tracking into scrotum with massive bilateral pneumo-scrotum (Fig. 2). Right lateral decubitus radiograph confirmed massive pneumoperitoneum and scrotum (Fig. 3).

Figure 1: Chest radiograph showing features of respiratory distress and gross pneumoperitoneum- note saddle bag and American football signs. Note nasogastric tube going straight down towards pelvis without following the gastric curvature.

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Figure 2: Abdominal radiograph showing massive pneumoperitoneum and bilateral pneumo-scrotum

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Figure 3: Right decubitus lateral view showing massive pneumo peritoneum and scrotum- note umbilical venous (UVC) and arterial catheters (UAC) in situ.

10.4 6 3

The diagnosis of gastrointestinal perforation was evident and in view of sudden deterioration following difficult intubation and massive pneumoperitoneum and scrotum; a diagnosis of traumatic perforation of stomach was made and necrotizing enterocolitis was unlikely clinically, pathologically and radilologically.

He was resuscitated with intravenous fluid boluses and started on triple antibiotics. Once stabilised, he was taken to theatre for exploratory laparotomy. At exploration, there was massive pneumoperitoneum and large amount of free milk feed in the peritoneal cavity. He was found to have a large gastric perforation extending from the fundus above to the antrum and pylorus below parallel to greater curvature of the stomach leaving the whole stomach open like a book and the anterior wall of the stomach was necrotic. Large stomach rent was defined and trimmed and closure was carried out in two layers with a size 10 French Malecot gastrostomy insertion for initial decompression and then feeding uneventfully. He remained nil by mouth post operatively with total parentral nutrition (TPN). Feeds were started from day 15 of life via gastrostomy and increased gradually as tolerated with weaning TPN. The infant underwent bilateral inguinal herniotomy on day 51 following bilateral obstructed inguinal hernia, his gastrostomy tube was taken out at the same time.

Following removal of gastrostomy tube he started leaking feeds through gastrostomy site which was treated with cauterisation of skin edges and application of glue. He had a symptomatic PDA which was ligated on his 56th day of life, and a VSD. He was fed via continuous nasojejunal (NJ) feeds. Once NJ feeds were well tolerated he was changed to NG feeds. Bottles were introduced and currently he is on demand bottle feeds with a good weight gain. He needed 58 days of ventilation via endotracheal tube/tracheostomy, 50 days of CPAP and 21 days of oxygen therapy. Overall he had 61 days of Intensive (level 1) care, 48 days of high dependency (level 2) care and 23 days of special (level 3) care. At follow up, he was thriving well and asymptomatic.

Discussion

Neonatal gastric perforation is an unusual, rare and life threatening emergency associated with high morbidity and mortality [4]. Exact causation is unknown but three possible mechanisms responsible for its pathogenesis have been postulated namely traumatic, ischaemic and spontaneous or idiopathic.

Most cases are secondary to traumatic oro-gastric or nasogastric tube insertions in a large series [5]. These perforations are generally a minute puncture like or a small laceration usually along the greater curvature side. Neonatal gastric perforation during the course of positive pressure ventilation and treated successfully have been reported [6].

Search of literature could not find near total gastric rupture in these cases as seen in our case and being extremely premature and extremely low birth weight with bilateral inguinal hernias immediate development of massive abdominal distention and pneumoscotum suggested accidental oesophageal intubation and positive pressure ventilation associated with close loop obstruction at cardiac and pyloric sphincters leading to anterior gastric wall near total rupture with necrotic gastric walls secondary to acute massive gastric pneumo distention in our case.

Perforation being very proximal with air swallow or leak may lead to rapid massive pneumoperitoneum and plain radiography generally helpful in early diagnosis as happened in our case and presence of bilateral inguinal hernia made pneumoscrotum quite easy to see. These infants can have massive tension pneumoperitoneum and if they need to travel long geographical distances for access to definitive treatment, decompression using cannula similar to tension pneumothorax has been recommended to reduce cardiorespiratory compromise during transfer [1].

Laparoscopic repair of neonatal gastric perforation has been reported which may be feasible in full term infants with good weight and small perforation [7]. In our case it was not feasible due to small size of the preterm twin 1 and the gastric rupture was extensive. The gold standard treatment of gastric perforation is exploratory laparotomy and primary suture repair of the perforation. In a puncture or small laceration primary closure with peritoneal toilet is recommended. In our case with acute stomach blast and near total rupture of anterior gastric wall parallel to greater curvature addition of temporary gastrostomy was helpful. The spontaneous or idiopathic variety has recently questioned as the gastric wall had microscopic defects and gaps due to developmental problems [8]. Non-steroidal anti-inflammatory drugs and steroids may be responsible for perforation in minority of case.

Neonatal gastric perforation has a very high morbidity and mortality associated with it which has added factors of extreme prematurity and extreme low birth weight. High index of suspicion, transillumination and radiological confirmation followed by immediate surgical repair helps reduce complications and allows rapid recovery.

Conclusion

Neonatal gastric perforation is rare but serious and potentially lethal condition. Traumatic, ischaemic and spontaneous neonatal gastric perforations can occur. High index of suspicion, clinical and plain radiological features helps early diagnosis. Prompt recognition, early resuscitation and surgical repair allows reduction in morbidity and mortality. Laparoscopic or open approach, primary closure/repair with a temporary gastrostomy in cases of near total rupture helps initial decompression followed by feeding later in these extremely vulnerable infants.

 

 

References

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