Case Report: Molecular autopsy underlie COVID-19-associated sudden, unexplained child mortality

Front Immunol. 2023 Apr 18:14:1121059. doi: 10.3389/fimmu.2023.1121059. eCollection 2023.

Abstract

Herein, we report a child with COVID-19 and seemingly no underlying disease, who died suddenly. The autopsy revealed severe anemia and thrombocytopenia, splenomegaly, hypercytokinemia, and a rare ectopic congenital coronary origin. Immunohistochemical analysis demonstrated that the patient had acute lymphoblastic leukemia of the B-cell precursor phenotype (BCP-ALL). The complex cardiac and hematological abnormalities suggested the presence of an underlying disease; therefore, we performed whole-exome sequencing (WES). WES revealed a leucine-zipper-like transcription regulator 1 (LZTR1) variant, indicating Noonan syndrome (NS). Therefore, we concluded that the patient had underlying NS along with coronary artery malformation and that COVID-19 infection may have triggered the sudden cardiac death due to increased cardiac load caused by high fever and dehydration. In addition, multiple organ failure due to hypercytokinemia probably contributed to the patient's death. This case would be of interest to pathologists and pediatricians because of the limited number of NS patients with LZTR1 variants; the complex combination of an LZTR1 variant, BCP-ALL, and COVID-19; and a rare pattern of the anomalous origin of the coronary artery. Thus, we highlight the significance of molecular autopsy and the application of WES with conventional diagnostic methods.

Keywords: BCP-ALL; SARS-CoV-2; autopsy; noonan syndrome (NS); sudden child death.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Autopsy
  • COVID-19*
  • Child Mortality
  • Cytokine Release Syndrome
  • Humans
  • Noonan Syndrome* / genetics
  • Phenotype
  • Transcription Factors / genetics

Substances

  • LZTR1 protein, human
  • Transcription Factors

Grants and funding

This work was supported by Japan Agency for Medical Research and Development (Grant Number: JP21fk0108436) to SO. The funding body played no role in 1) the design of the study; 2) the collection, analysis, and interpretation of data; 3) the writing of the report; and 4) the decision to submit the manuscript for publication.