Case Report
Copyright ©The Author(s) 2022. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Oct 16, 2022; 10(29): 10583-10599
Published online Oct 16, 2022. 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
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
Aleksandra Kaplina, Tatiana Fedoseeva, Natalia Petrova, Research Laboratory of Physiology and Diseases of Newborns, Almazov National Medical Research Centre, St. Petersburg 197341, Russia
Ekaterina Zaikova, Olga Kalinina, Research Laboratory of Autoimmune and Autoinflammatory Diseases, Almazov National Medical Research Centre, St. Petersburg 197341, Russia
Artem Ivanov, Vladimir Ulyantsev, International Laboratory of Computer Technologies, ITMO University, St. Petersburg 197101, Russia
Yulia Volkova, Alexander Latypov, Department of Cardiovascular Surgery for Children, Almazov National Medical Research Centre, St. Petersburg 197341, Russia
Tatiana Alkhova, Department of Neonatal Physiology with an ICU Ward, Almazov National Medical Research Centre, St. Petersburg 197341, Russia
Vladimir Nikiforov, Pediatric Cardiac Intensive Care Unit, Almazov National Medical Research Centre, St. Petersburg 197341, Russia
Marina Khavkina, Neonatal and Preterm Special Care Unit (2nd Stage Care), Almazov National Medical Research Centre, St. Petersburg 197341, Russia
Tatiana Pervunina, Institute of Perinatology and Pediatrics, Almazov National Medical Research Centre, St. Petersburg 197341, Russia
Yulia Skorobogatova, Express Laboratory of Perinatal Centre, Almazov National Medical Research Centre, St. Petersburg 197341, Russia
Svetlana Volkova, Clinical Diagnostic Laboratory, Almazov National Medical Research Centre, St. Petersburg 197341, Russia
Stanislav Sitkin, Epigenetics and Metagenomics Group, Institute of Perinatology and Pediatrics, Almazov National Medical Research Centre, St. Petersburg 197341, Russia
Stanislav Sitkin, Department of Internal Diseases, Gastroenterology and Dietetics, North-Western State Medical University named after I.I. Mechnikov, St. Petersburg 191015, Russia
Author contributions: Petrova N, Sitkin S, Kaplina A and Kalinina O conceptualized the idea of the article; Kaplina A performed sample collection, contributed to manuscript drafting, and reviewed the literature; Petrova N, Sitkin S and Kalinina O contributed to manuscript drafting; Zaikova E, Ivanov A and Ulyantsev V performed 16S gene-based microbiota profiling; Volkova Y was the patient’s cardiologist and analyzed the echocardiography and computed tomography images; Alkhova T, Khavkina M and Fedoseeva T were patient’s neonatologists, contributed to clinical data analysis; Latypov A was the patient’s cardiac surgeon and contributed to operative data analysis; Nikiforov V was the patient’s anesthesiologist and intensive care specialist and contributed to early postoperative clinical data analysis; Skorobogatova Y performed laboratory testing; Volkova S performed microbiological examinations; Petrova N, Sitkin S, Pervunina T and Kalinina O interpreted clinical data and supervised the manuscript; all authors have read and agreed to the published version of the manuscript.
Supported by the Russian Science Foundation, No. 22-25-00484.
Informed consent statement: Informed consent was obtained from the patient’s parent for publication of this report.
Conflict-of-interest statement: The authors report having no potential conflicts of interest.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016) and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Stanislav Sitkin, MD, PhD, Associate Professor, Senior Researcher, Epigenetics and Metagenomics Group, Institute of Perinatology and Pediatrics, Almazov National Medical Research Centre, Akkuratova street, 2, St. Petersburg 197341, Russia. drsitkin@gmail.com
Received: February 16, 2022
Peer-review started: February 16, 2022
First decision: April 10, 2022
Revised: April 20, 2022
Accepted: August 14, 2022
Article in press: August 14, 2022
Published online: October 16, 2022
Processing time: 224 Days and 23.6 Hours
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 14th day of life and had persistent mesenteric hypoperfusion after surgery. The patient had episodes of NEC stage IIA on the 1st and 28th 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: Necrotizing enterocolitis; Congenital heart disease; Ivemark syndrome; Heterotaxy; Right atrial isomerism; Intestinal microbiome; Case report

Core Tip: Right atrial isomerism is associated with severe congenital heart disease, abnormal arrangement of internal organs, and asplenia/spleen hypoplasia. Infants with right atrial isomerism palliated with the systemic-to-pulmonary shunt have a “diastolic steal” of mesenteric blood flow, which increases the risk of necrotizing enterocolitis (NEC). The role of the microbiome in cardiogenic NEC remains unclear. This case report highlights possible interactions between dynamic changes in the fecal microbiome before and after cardiac surgery and development of recurrent NEC, based on hemodynamic changes, mesenterial perfusion, and antibacterial treatment in an asplenic infant with right atrial isomerism who underwent palliative cardiac surgery.