Hydrops fetalis and neonatal abdominal compartment syndrome continuum from immature gastric teratoma: a case report

Abstract

Background: Hydrops fetalis as well as abdominal compartment syndrome (ACS) are conditions that are associated with high mortality rates. A rare case of immature gastric teratoma causing fetal hydrops and subsequent ACS is presented. The related pathophysiologic mechanisms are discussed, and the importance of timely recognition and appropriate interventions are highlighted. Case presentation: The male patient was born preterm, weighing 3.9 kg., by Cesarean section. Prior prenatal ultrasounds were normal, but a scan done just before delivery had findings indicating polyhydramnios, fetal ascites, and meconium peritonitis. Upon delivery, the patient had respiratory distress, anasarca and a massively distended abdomen. Resuscitation measures, including ventilatory support, were instituted. Imaging studies showed ascites as well as a large, complex intra-abdominal lesion with calcifications. In the succeeding hours, anuria persisted, anasarca worsened, the abdomen became more distended, and inotrope requirements increased. The occurrence of ACS, from what was presumed to be a retroperitoneal teratoma, was therefore considered. Laparotomy was done on the 28th hour of life, with en bloc excision of a massive tumor and attached section of the greater curvature of the stomach. Passage of urine occurred intra-operatively, and the patient was soon after weaned off inotropes and ventilator support. The histopathologic result was immature gastric teratoma. No chemotherapy was given, and the patient's serum AFP is at normal levels 15 months following surgery. Conclusion: The presence of a massive intra-abdominal lesion can result in the pathophysiologic continuum of hydrops fetalis and neonatal ACS. The early recognition of such an association can enable appropriate expectant management of similarly affected neonates, including emergent decompression laparotomy.

Enlarged Abdominal Lymph Node as a Cause of Polyhydramnios in the Course of Congenital Neonatal Leukaemia: A Case Report and Review of the Literature on Foetal Abdominal Tumours with Coexisting Polyhydramnios

“…Following abstract screening, 102 articles were determined to be outside the scope of the investigation and a further 14 full-text articles described cases of abdominal tumours without concomitant polyhydramnios. The remaining 27 publications describing 32 cases constituted the basis of this review ( Supplementary Table S1 ), [ 12 , 13 , 14 , 15 , 16 , 17 , 18 , 19 , 20 , 21 , 22 , 23 , 24 , 25 , 26 , 27 , 28 , 29 , 30 , 31 , 32 , 33 , 34 , 35 , 36 , 37 , 38 ].…”

“…It is a very rare tumour, accounting for less than 2% of abdominal tumours diagnosed in neonates. In three reported cases of immature gastric teratoma, ultrasound studies revealed not only polyhydramnios, but also placentomegaly, ascites and intraperitoneal calcifications [ 34 , 35 , 36 , 45 ]. Treatment of a tumour in the post-partum period involves removal of the lesion, sometimes combined with a partial gastric resection.…”

Immature gastric teratoma: A case report

“…Although the USG during pregnancy has become almost universal and more and more fetal MRIs are being carried out, GT was not diagnosed antenatally till now. In neonates with suspicious mass lesion with calcification suggestive of meconium peritonitis in fetal imaging[ 3 ] and nonimmune hydrops with suspicious meconium peritonitis in fetal imaging may be subjected to early postnatal CT/MRI/contrast upper gastrointestinal (GI) study[ 4 ] to confirm GT. This approach helps to plan emergency decompression of the abdomen to treat the abdominal compartment syndrome related to hydrops secondary to the mass and early and appropriate surgical procedure for complete excision.…”