Gastric Serosal Tear: A Rare Complication of Congenital Diaphragmatic Hernia

 

Article By:

S. Khope

N.S. Phaldesai

 

Abstract

Gastric serosal tear (GST) in neonates is a very rare complication of congenital diaphragmatic hernia (CDH), the only previous report in literature being associated with congenital pyloric atresia. We report a 5-day-old male neonate, admitted for an initial suspicion of an intestinal obstruction. He presented with retching and vomiting, respiratory distress, epigastric distension and lethargy. A detailed radiological imaging study could not be done due to sepsis and suspicion of perforation. Laparotomy revealed CDH with GST. The diaphragmatic defect was closed after reduction of the herniated contents and gastric serosal tear was repaired. At the 3 and 6-month follow-up examinations, the  infant  remained  asymptomatic.

 

Introduction

Gastric serosal tear (GST) in neonates is a very rare complication of congenital diaphragmatic hernia (CDH). Gunaydin M et al. reported the only previous case of GST associated with congenital pyloric atresia.[1] Malrotation of the midgut is one of the common gastrointestinal malformations associated with CDH. While majority of CDH with severe pulmonary hypoplasia presents at birth with respiratory distress, mild hypoplasia may be asymptomatic and  missed  at  birth  if thorough examination not carried out. Presence of stomach in the chest can lead to gastric volvulus, with or without perforation. GST is a hitherto unknown complication of  CDH.

 

Case Report

A five-day-old male neonate with 3.2 kg weight was referred for non-bilious vomiting and refusal of feeds. He was delivered by caesarean section  and   no  congenital  anomaly  had  been detected  antenatally  or  at  birth.

On examination, he was lethargic, pale, tachypneic, and dehydrated, with mild icterus. There was no cyanosis. He had passed normal meconium at birth. Further physical examination revealed epigastric fullness with tenderness. His investigations showed hemoglobin of 6 grams per decilitre (gm/dL), white blood cell count of 16,000 per cubic millimetre, low serum sodium and potassium, with C-reactive protein (CRP) level of 12 milligrams per decilitre (mg/dL).

The chest X ray showed a left sided diaphragmatic hernia (Figure 1).

TC-July2018-021 - Plain abdominal radiograph showing left sided posterolateral diaphragmatic hernia

 

The stomach was massively distended with small-bowel gas shadows, but no air fluid levels were noted. Blood gas showed mild respiratory acidosis. A nasogastric tube was inserted, and dehydration and electrolyte imbalances were corrected. Preoperative blood transfusion and antibiotics were also started. Echocardiography showed normal cardiac anatomy.

Due to a strong suspicion of gastric perforation, an emergency exploratory laparotomy was done after stabilization of vital parameters. There was a left posterolateral diaphragmatic hernia with stomach, small bowel, and left lobe of liver in the hernia sac. A surprise finding was that of a large serosal tear along the greater curvature of stomach starting from middle point of the lesser curvature (Figure 2), without mucosal perforation. There was also malrotation of the midgut. The hernia was reduced, and the defect was repaired using interrupted non absorbable sutures. The gastric serosal tear was repaired (Figure 3)  and  Ladd’s  procedure  was  done.

TC-July2018-022 - Serosal tear at the anterior wall of the stomach TC-July2018-023 - Gastric serosal tear with irregular and necrotic edges and mucosa bulging out

The child recovered uneventfully and was discharged ten days postoperatively. A follow-up done two weeks later showed that the neonate was doing well. Further follow-ups done 3 and 6-months later showed that the patient remained asymptomatic  and  was  thriving.

 

Discussion :

Congenital diaphragmatic hernia is a very serious congenital defect associated with a high mortality rate and affects 1 in every 2,000 to 4,000 live births. CDH is diagnosed antenatally through routine ultrasonography screening. Patients with CDH usually present in the immediate neonatal period, however in healthy infant without respiratory distress CDH may be missed at birth. Alternately patients with less severe pulmonary hypoplasia may not have significant respiratory distress.

Herniated stomach when distended with trapped air during resuscitation, may lead to acute gastric volvulus with or without perforation or tension gastrothorax. While gastric perforations in newborns occur by three mechanisms: trauma, ischaemia, or spontaneous, gastric serosal tear without perforation is rare in newborns and has been reported only once before in English publications.[1]

Iatrogenic perforations mostly related to nasogastric tubes are more common and mostly observed at the greater curvature. Mask-ventilation during resuscitation may result with over distension of the stomach and perforation. Spontaneous perforations are seen in the first week of life in an otherwise healthy newborn and extremely rare. They are related with the sudden increase in intragastric pressure. In the amniotic fluid-filled stomach, the serosal layer is torn first, and the mucosa bulges out like a diverticulum. Further increase in the intragastric pressure due to vomiting, results in a full thickness tear.[1] Spontaneous gastric perforations occur at the greater curvature or ventral wall of the stomach where the wall is relatively thin.

Serious malnutrition, dehydration, acid-base imbalance and sepsis may further complicate the management.

Serosal tear without perforation and mild pulmonary hypoplasia had positive impact on the treatment and survival in this case.

 

Conclusion :

GST is a rare complication of CDH. We postulate that GST probably occurs as result of massive gastric distension due to trapped air and distal outlet obstruction due to associated malrotation of the midgut with Ladd’s bands simulating a closed loop obstruction.

TC-July2018-024 - Authors Pg38

 

References:

  1. Gunaydin M, Rizalar R, Bozkurter AT, Tander B, et al.Gastric serosal tear due to congenital pyloric atresia: a rare anomaly, a rare complication. Afr J Paediatr Surg. 2011; 8(2):232-4.