Intestinal microbiome changes in an infant with right atrial isomerism and recurrent necrotizing enterocolitis: A case report and review of literature

Aleksandra Kaplina; Ekaterina Zaikova; Artem Ivanov; Yulia Volkova; Tatiana Alkhova; Vladimir Nikiforov; Alexander Latypov; Marina Khavkina; Tatiana Fedoseeva; Tatiana Pervunina; Yulia Skorobogatova; Svetlana Volkova; Vladimir Ulyantsev; Olga Kalinina; Stanislav Sitkin; Natalia Petrova

doi:10.12998/wjcc.v10.i29.10583

Intestinal microbiome changes in an infant with right atrial isomerism and recurrent necrotizing enterocolitis: A case report and review of literature

World J Clin Cases. 2022 Oct 16;10(29):10583-10599. doi: 10.12998/wjcc.v10.i29.10583.

Authors

Aleksandra Kaplina¹, Ekaterina Zaikova², Artem Ivanov³, Yulia Volkova⁴, Tatiana Alkhova⁵, Vladimir Nikiforov⁶, Alexander Latypov⁴, Marina Khavkina⁷, Tatiana Fedoseeva¹, Tatiana Pervunina⁸, Yulia Skorobogatova⁹, Svetlana Volkova¹⁰, Vladimir Ulyantsev³, Olga Kalinina², Stanislav Sitkin^{11

12}, Natalia Petrova¹

Affiliations

¹ Research Laboratory of Physiology and Diseases of Newborns, Almazov National Medical Research Centre, St. Petersburg 197341, Russia.
² Research Laboratory of Autoimmune and Autoinflammatory Diseases, Almazov National Medical Research Centre, St. Petersburg 197341, Russia.
³ International Laboratory of Computer Technologies, ITMO University, St. Petersburg 197101, Russia.
⁴ Department of Cardiovascular Surgery for Children, Almazov National Medical Research Centre, St. Petersburg 197341, Russia.
⁵ Department of Neonatal Physiology with an ICU Ward, Almazov National Medical Research Centre, St. Petersburg 197341, Russia.
⁶ Pediatric Cardiac Intensive Care Unit, Almazov National Medical Research Centre, St. Petersburg 197341, Russia.
⁷ Neonatal and Preterm Special Care Unit (2Stage Care), Almazov National Medical Research Centre, St. Petersburg 197341, Russia.
⁸ Institute of Perinatology and Pediatrics, Almazov National Medical Research Centre, St. Petersburg 197341, Russia.
⁹ Express Laboratory of Perinatal Centre, Almazov National Medical Research Centre, St. Petersburg 197341, Russia.
¹⁰ Clinical Diagnostic Laboratory, Almazov National Medical Research Centre, St. Petersburg 197341, Russia.
¹¹ Epigenetics and Metagenomics Group, Institute of Perinatology and Pediatrics, Almazov National Medical Research Centre, St. Petersburg 197341, Russia.
¹² Department of Internal Diseases, Gastroenterology and Dietetics, North-Western State Medical University named after I.I. Mechnikov, St. Petersburg 191015, Russia. drsitkin@gmail.com.

Abstract

Background: Necrotizing enterocolitis (NEC) is a multifactorial disease that predominantly affects premature neonates. Intestinal dysbiosis plays a critical role in NEC pathogenesis in premature neonates. The main risk factor for NEC in term infants is mesenteric hypoperfusion associated with ductal-dependent congenital heart disease (CHD) that eventually leads to intestinal ischemia. The incidence of NEC in neonates with critical CHD is 6.8%-13%. However, the role of the intestinal microbiome in NEC pathogenesis in infants with ductal-dependent CHD remains unclear.

Case summary: A male term neonate with right atrial isomerism underwent modified Blalock-Taussig shunt placement on the 14^th day of life and had persistent mesenteric hypoperfusion after surgery. The patient had episodes of NEC stage IIA on the 1^st and 28^th days after cardiac surgery. Fecal microbial composition was analyzed before and after cardiac surgery by sequencing region V4 of the 16S rRNA gene. Before surgery, species belonging to genera Veillonella and Clostridia and class Gammaproteobacteria were detected, Bifidobacteriaceae showed a low abundance. The first NEC episode was associated with postoperative hemodynamic instability, intestinal ischemia-reperfusion injury during cardiopulmonary bypass, and a high abundance of Clostridium paraputrificum (Clostridium sensu stricto I) (56.1%). Antibacterial therapy after the first NEC episode resulted in increased abundance of Gammaproteobacteria, decreased abundance of Firmicutes, and low alpha diversity. These changes in the microbial composition promoted the growth of Clostridium sensu stricto I (72.0%) before the second NEC episode.

Conclusion: A high abundance of Clostridium sensu stricto I and mesenteric hypoperfusion may have contributed to NEC in the present case.

Keywords: Case report; Congenital heart disease; Heterotaxy; Intestinal microbiome; Ivemark syndrome; Necrotizing enterocolitis; Right atrial isomerism.

Publication types

Case Reports