A neonate with meconium peritonitis,first-trimester hepatitis A affected pregnancy: A case report

Case presentation

Case of a female infant born at 34 2/7 weeks of gestation via cesarean section to a 25-years-old mother’s G2P2A0 with adequate prenatal care. During the first trimester of pregnancy, the mother’s screening tests were negative for human immunodeficiency virus, syphilis, and hepatitis B. She was also rubella immune. The mother did not have a history of alcohol, smoking, or drug use. The routine third-trimester pelvic sonogram findings raised concerns for fetal bowel obstruction, or a pelvic mass accompanied by polyhydramnios (AFI 43). The mother went into preterm delivery with a persistent Category II tracing, and the baby was delivered by cesarean section due to malpresentation. Due to preterm birth, the pediatric surgery service had not yet been involved in the case. At birth, the newborn required neonatal resuscitation with positive pressure ventilation and admission to the neonatal intensive care unit. The neonate had respiratory depression at birth associated with significant abdominal distention.

Physical examination exhibited a distended abdomen, measuring 38 cm at the mid-abdominal circumference, with mottling and visible vein engorgement (Figure 1). The abdominal ultrasound revealed a large, well-defined, centrally located intra-abdominal cystic mass extending from the epigastric to the pelvic regions. In addition, the study described a significant amount of layering and floating internal debris. The radiologic findings on the infant suggested a complicated mesenteric cyst or a meconium pseudocyst. However, as no gross abnormal calcifications were recognized as expected on the mentioned diagnoses, differential diagnoses were broadened, including choledochal cyst, duplication cyst, or ovarian lesion (Figure 2).

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Figure 1.

Distended abdomen, measuring 38 cm at the mid-abdominal circumference, with mottling and visible vein engorgement.

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Figure 2.

Abdominal ultrasound. Left lower quadrant transverse view of patient abdomen demonstrating a large, well defined intra-abdominal cystic mass with a significant amount of layering and floating internal debris.

The infant was taken emergently to the operating room, where she underwent an exploratory celiotomy. The surgical team carefully drained the large cystic structure identified on imaging and dissected off the small bowel and surrounding structures. However, upon entry into the abdominal cavity, ileal atresia with associated proximal perforation was identified as remarkable for meconium peritonitis, requiring an enterorrhaphy, a segmental ileal resection, and lysis of the extensive adhesions with the creation of ileostomy and mucous fistula.

A month later, the infant underwent a second surgery for ileostomy takedown, but a slow recovery with minimal oral tolerance led to a third intervention, unveiling an anastomotic stricture. Strictureplasty with successful recovery ultimately resulted in an improved nutritional status and adequate weight gain.

Despite the association of ileal atresia and perforation with meconium peritonitis (MP), the extent of the case prompted an investigation into coexisting etiologies. Comprehensive workup included the newborn screening and cystic fibrosis gene sequence, which yielded normal results. Rectal biopsy excluded Hirschsprung’s disease as it showed the presence of ganglion cells and a regular pattern of calretinin immunoreactive mucosa innervation.

Further inquisition of prenatal history revealed a benign course of flu-like syndrome and diarrhea during the first trimester managed conservatively with complete resolution of symptoms. The newly obtained information raised suspicion of intrauterine Hepatitis A infection as a possible etiology of ileal atresia with meconium peritonitis. Hepatitis A titers for immunoglobulin M and total antibodies were positive in both the infant and her mother, with the mother also testing positive for hepatitis Anti-HAV ll reactive, indicative of a past infection.

One year after discharge, the infant continued to receive comprehensive care from her pediatrician, as well as the gastroenterology, genetics, and surgery teams. The postsurgical recovery was carefully monitored, with a focus on stool passage and diet tolerance. While the patient is gaining weight, she is still below the expected range for her age. A thorough evaluation of cystic fibrosis was conducted, yielding negative results from both the sweat and stool tests.

Discussion

MP results from intestinal perforation in utero. Leakage of meconium into the peritoneal cavity causes a sterile chemical inflammatory reaction that may progress to intestinal necrosis and lead to calcification and extensive scarring. ¹ Any cause of bowel obstruction may result in meconium peritonitis; these include atresia of the bowel, volvulus, intussusception, bands, and meconium plug syndrome in cystic fibrosis. ² The estimated prevalence is around 1 in 35,000. Antenatal ultrasound with visualization of calcifications in the fetus after 18 weeks of gestation is the standardized test for a prenatal diagnosis of MP. Intra-peritoneal meconium usually calcifies, sometimes within 24 h. ³ However, intra-abdominal calcifications are nonspecific as they may be seen in multiple etiologies, such as meconium pseudocyst, intraluminal meconium, congenital infections, mesenteric nodes, and hepatic granulomas. ⁴ Other etiologies include adrenal and liver calcifications.

Intestinal atresias have long been hypothesized to result from either failure of recanalization of the intestinal lumen or in utero vascular accidents. Based on data from mouse models, intestinal atresias begin forming very early in development between E10.5 and E12.5 (the equivalent of days 33–48 in humans). ⁵ The impact of maternal systemic viral infection, whose pathophysiology affects primarily the gastrointestinal tract, on early intestinal fetal development has not been evaluated.

Ileal atresia caused by an in-utero vascular accident during fetal development involving branches of mesenteric vessels in the midgut, resulting in ischemic necrosis, leaving a blind proximal loop with distal being atretic.

Hepatitis A virus (HAV), a Heptavirus of the family of Picornavirus, is transmitted by a fecal-oral route, replicates in the liver, and is shed in the stool for a prolonged period. ⁶ The infectious stage lasts from the incubation period, approximately 28 days until symptoms appear. ⁷ The most common presentation is the abrupt onset of nausea, vomiting, anorexia, fever, malaise, and abdominal pain that can be related to jaundice and, or elevated liver enzymes. ⁸ The presence of serum IgM anti-HAV antibodies confirms the diagnosis. This antibody can be detectable up to 6 months after infection. Anti-HAV IgG is the marker immunity and appears last. ⁹ Anti-HAV II reactive, an immunoassay for the in-vitro qualitative detection of total (IgM and IgG) antibodies to HAV in human serum and plasma, is used as an aid to detect a past or existing HAV infection or to determine the presence of antibody response to HAV in vaccine recipients. ¹⁰

It is well known that viruses like hepatitis B and C, as well as rubella, among others, can cause congenital infection and malformations in neonates.^11,12 Moreover, HAV infection typically has minimal impact on pregnancy; it is a self-limited disease, and treatment is supportive for mild cases. Rarely hospitalization may be indicated for severe hepatitis or in uncommon cases of fulminant hepatitis.^13,14 Despite being a well-described infection, little is understood about its implications in congenital infections or malformations. Nevertheless, acute HAV infection has been implicated in preterm labor and gestational complications. ¹⁵ Routine screening for HAV during pregnancy is currently not recommended. Hence, its contribution to congenital disease may be underestimated.

Conclusion

In this case report, we present an atypical presentation of meconium peritonitis secondary to ileal atresia associated with intrauterine hepatitis A infection. The absence of typical intra-abdominal calcifications, commonly seen in patients with MP in imaging studies, including fetal ultrasound, postnatal abdominal ultrasound, and plain abdominal X-ray, further adds to the uniqueness of this case.

The history of flu-like symptoms and diarrhea, in conjunction with serologic evidence of hepatitis A immunoglobulin M and hepatitis A total antibodies in the neonate and the mother, confirmed the diagnosis of intrauterine hepatitis A infection. In conclusion, although multiple causes may contribute to meconium peritonitis and bowel rupture, this case underscores the importance of a comprehensive prenatal history in identifying risk factors for specific conditions. In addition, it suggests that intrauterine exposure to HAV may contribute to fetal intra-abdominal vascular accidents, causing meconium peritonitis. Increased awareness and further research are warranted to understand better the implications of maternal hepatitis A infection on congenital diseases.