Keywords
Gastric pneumatosis - preterm - neonate - synchronized nasal intermittent positive
pressure ventilation
Gastric pneumatosis, defined as gas within the wall of the stomach, is an extremely
rare condition. It was first reported in a pediatric patient by the Children's Hospital
of Philadelphia in 1952.[1] Here, we describe the first case report of gastric pneumatosis associated with the
use of indomethacin in a preterm neonate with synchronized nasal intermittent positive
pressure ventilation (SNIPPV).
Case Report
A female neonate was born at 29 weeks' gestation with a birth weight of 1310 g. The
mother was Nepalese and suffered from severe gestational proteinuric hypertension.
An emergency lower-segment cesarean section was performed because of the maternal
condition. One dose of antenatal steroid was given 2 hours prior to delivery. The
neonate was delivered without difficulty with Apgar scores of 8 (1 minute) and 9 (5
minutes). She developed respiratory distress syndrome soon after birth and was treated
with surfactant replacement. Empirical penicillin and cefuroxime were also administered.
The surface swabs, gastric aspirate, and blood cultures taken at birth subsequently
yielded no bacterial growth. Both umbilical artery and venous catheters were inserted.
In view of her improved respiratory condition, she was extubated on day 3 of life.
The SNIPPV was used for respiratory support to improve the chance of successful extubation.
The Infant Star ventilator with the StarSync module (Infrasonics, San Diego, CA) was
used to deliver SNIPPV via the short binasal prongs, which provides thoracoabdominal
synchronization via the Graseby capsule placed on the abdomen. An orogastric tube
was placed. It was closed after each feed (at 2-hour intervals) and aspirated for
air and gastric residuals before the next feed. Trophic feeding, mainly in the form
of expressed breast milk, was commenced on day 2 of life and was increased gradually
by not more than 20 mL/kg/d. Feeding was well tolerated. The meconium was passed at
42 hours of life.
She developed increasing dyspnea and tachypnea on day 5, such that the ventilator
rate had to be increased from 30/min to 35/min. Peak inspiratory pressure was increased
from 17 to 21 cm H2O, and positive end-expiratory pressure was set at 5 cm H2O. Maximum peak inspiratory pressure arrived, increased from 15 to 16 cm H2O. The flow rate was kept at 8 to 10 L/min. A patent ductus arteriosus was subsequently
confirmed by echocardiogram. In view of its significant hemodynamic impact, a course
of indomethacin (0.2 mg/kg/dose for three doses) was started on day 7. A central venous
catheter was inserted percutaneously to replace the umbilical line for parenteral
nutrition. On the same day, she developed feeding intolerance and had gastric retention
of 16 mL milk mixed with fresh blood. An urgent abdominal X-ray revealed gastric pneumatosis
and portovenous gas. No pneumatosis was noted in the rest of the bowel or esophagus
([Fig. 1]). Using the necrotizing enterocolitis (NEC) classification by Bell et al, the presence
of bloody gastric aspirates, gastric pneumatosis, and portal venous gas should constitute
the diagnosis of definite necrotizing enterocolitis (modified Bell's staging II).[2]
[3]
[4] The neonate was reintubated for repeated apnea. Empirical cloxacillin, ceftazidime,
metronidazole, and an H2-blocker were given to the neonate. A repeat septic workup showed sterile blood cultures
and normal white cell counts. No fresh blood was yielded from the orogastric tube
by the next day. Serial abdominal X-rays revealed spontaneous resolution of the mural
gas in the stomach and the portal venous gas. She was extubated to nasal continuous
positive airway pressure 4 days later and received nothing by mouth for 1 week. After
the resumption of feeding, there was no feeding intolerance or recurrence of gastric
pneumatosis.
Figure 1 Abdominal X-ray of our patient showing gastric pneumatosis.
Subsequently, the neonate underwent a smooth neonatal course, without intraventricular
hemorrhage, retinopathy of prematurity, or chronic lung disease. She was ultimately
discharged on day 53. We last saw her at 3 years of age at our outpatient clinic,
and she was thriving. Apart from a mild language delay, her neurodevelopment was unremarkable.
Discussion
Gastric pneumatosis is a rarely reported condition and is primarily a radiological
diagnosis.[5] It has been found to be associated with necrotizing enterocolitis, concomitant with
pneumatosis intestinalis and hepatobiliary gas as detected by abdominal X-ray.[6]
[7] The other reported associations include pyloric stenosis,[1]
[8]
[9]
[10] duodenal obstruction,[11]
[12]
[13] and annular pancreas.[14] It has also been reported in a premature infant developing gastric pneumatosis secondary
to the placement of an intramural feeding catheter.[15] Krueger et al reported gastric pneumatosis in a neonate with hypoplastic left heart
syndrome on day 6 of life before cardiac operation.[16] Taylor et al[17] reported a neonate having this rare condition following a modified Norwood stage
I operation for complex cyanotic heart disease.
Nelson[18] summarized the possible mechanisms of the extraluminal gas collections in the gastrointestinal
tract, including those of the stomach. These gas collections could either be related
to infection of the visceral wall by gas-forming organisms or secondary to gastrointestinal
tract obstructions. In the former case, the extraluminal gas collections were related
to the direct inflammation and damage of the stomach wall. This inflammation is complicated
by the diffusion of gas through breaks in the necrotic mucosa, resulting in gastric
pneumatosis detected by X-ray. These observations of “emphysematous gastritis” were
primarily reported in adults, especially in alcohol abusers. The more commonly involved
bacterial pathogens include Escherichia coli and Streptococcus and Enterobacter species.[19] The patients were usually critically ill, and the condition was associated with
both high mortality and morbidity with gastric contractures.[19] In the latter group, the mechanical obstruction of the proximal gastrointestinal
tract resulted in elevated intragastric luminal pressure. These obstructions could
result in an increase in diffusion through the grossly intact mucosa or the escape
of the intraluminal gas through minor tears in an otherwise normal mucosa.[18] Unlike patients who develop gastric pneumatosis as a result of infection of the
visceral wall, this latter group of patients generally enjoyed good prognosis after
the prompt correction of the underlying obstruction.[1]
SNIPPV use in infants has been associated with reduced incidences of extubation failure,
bronchopulmonary dysplasia, neurodevelopmental impairment, and death, as compared
with infants managed with nasal continuous positive airway pressure.[20]
[21]
[22] No recent studies have reported any association of nasal intermittent positive pressure
ventilation use with necrotizing enterocolitis or gastric or other intestinal perforations.[20] Our center initiated the use of SNIPPV among very low-birth-weight infants in 2003.
In the case described in this report, the cause of the formation of gastric pneumatosis
remains unclear. By definition, she had stage II NEC. There was no evidence of sepsis
or an upper gastrointestinal tract obstruction in the neonate. We postulated that
there could have been mild damage in the mucosa, either related to the placement of
the feeding tube or secondary to the use of indomethacin or both. Indomethacin has
been well known to exacerbate gastric mucosal injury. The condition was further aggravated
by noninvasive ventilation, which introduced air into the stomach. An increase in
intragastric pressure resulted in the submucosal dissection of air followed by the
development of gastric pneumatosis detected by X-ray. This theory was also supported
by the prompt resolution of the pneumatosis after conservative management.
Conclusion
Gastric pneumatosis is a rarely reported condition. In our patient, it was associated
with the use of indomethacin and feeding tube and exacerbated with the use of noninvasive
ventilation. Prompt recognition and evaluation of this condition were essential for
making the diagnosis. The infant was also treated for NEC. Conservative management
strategies, including the use of a nasogastric tube for decompression and the withholding
of feeding, successfully managed the condition in our patient.
Acknowledgment
We would like to thank Dr. Y.Y. Chee for providing editorial assistance.
