Am J Perinatol 2022; 29(14): 1514-1518
DOI: 10.1055/a-1733-4163
Short Communication

COVID-19-Associated Multisystem Inflammatory Syndrome in a Neonate with Atypical Coronary Artery Involvement

Simonetta Costa*
1   Dipartimento di Scienze della Salute della Donna, del Bambino e di Sanità Pubblica, Fondazione Policlinico Universitario A. Gemelli Istituto Di Ricovero E Cura A Carattere Scientifico, Rome, Italy
,
Angelica B. Delogu*
1   Dipartimento di Scienze della Salute della Donna, del Bambino e di Sanità Pubblica, Fondazione Policlinico Universitario A. Gemelli Istituto Di Ricovero E Cura A Carattere Scientifico, Rome, Italy
2   Dipartimento di Scienze della Vita e Sanità Pubblica, Università Cattolica del Sacro Cuore, Rome, Italy
,
Anthea Bottoni
1   Dipartimento di Scienze della Salute della Donna, del Bambino e di Sanità Pubblica, Fondazione Policlinico Universitario A. Gemelli Istituto Di Ricovero E Cura A Carattere Scientifico, Rome, Italy
,
Velia Purcaro
1   Dipartimento di Scienze della Salute della Donna, del Bambino e di Sanità Pubblica, Fondazione Policlinico Universitario A. Gemelli Istituto Di Ricovero E Cura A Carattere Scientifico, Rome, Italy
,
Vito D'Andrea
1   Dipartimento di Scienze della Salute della Donna, del Bambino e di Sanità Pubblica, Fondazione Policlinico Universitario A. Gemelli Istituto Di Ricovero E Cura A Carattere Scientifico, Rome, Italy
,
Angela Paladini
1   Dipartimento di Scienze della Salute della Donna, del Bambino e di Sanità Pubblica, Fondazione Policlinico Universitario A. Gemelli Istituto Di Ricovero E Cura A Carattere Scientifico, Rome, Italy
,
Sara Muto
2   Dipartimento di Scienze della Vita e Sanità Pubblica, Università Cattolica del Sacro Cuore, Rome, Italy
,
Riccardo Marano
3   Dipartimento di Diagnostica per Immagini, Radioterapia Oncologica ed Ematologia, Fondazione Policlinico Universitario A. Gemelli Istituto Di Ricovero E Cura A Carattere Scientifico, Rome, Italy
4   Dipartimento di Scienze radiologiche ed ematologiche, Università Cattolica del Sacro Cuore, Rome, Italy
,
Giancarlo Savino
3   Dipartimento di Diagnostica per Immagini, Radioterapia Oncologica ed Ematologia, Fondazione Policlinico Universitario A. Gemelli Istituto Di Ricovero E Cura A Carattere Scientifico, Rome, Italy
,
Aurelio Secinaro
5   Department of Imaging, Ospedale Pediatrico Bambino Gesù, Istituto Di Ricovero E Cura A Carattere Scientifico, Rome, Italy
,
Fabrizio De Benedetti
6   Division of Rheumatology and Laboratory of Immuno-Rheumatology, Ospedale Pediatrico Bambino Gesù, Istituto Di Ricovero E Cura A Carattere Scientifico, Rome, Italy
,
Rita Carsetti
7   Diagnostic Immunology Research Unit, Multimodal Medicine Research Area, Ospedale Pediatrico Bambino Gesù, Istituto Di Ricovero E Cura A Carattere Scientifico, Rome, Italy
8   Microbiology and Diagnostic Immunology Unit, Ospedale Pediatrico Bambino Gesù, Istituto Di Ricovero E Cura A Carattere Scientifico, Rome, Italy
,
Eva Piano Mortari
7   Diagnostic Immunology Research Unit, Multimodal Medicine Research Area, Ospedale Pediatrico Bambino Gesù, Istituto Di Ricovero E Cura A Carattere Scientifico, Rome, Italy
,
Teresa Spanu
9   Dipartimento di Scienze di Laboratorio e Infettivologiche, Fondazione Policlinico Universitario A. Gemelli Istituto Di Ricovero E Cura A Carattere Scientifico, Rome, Italy
10   Dipartimento di Scienze Biotecnologiche di Base, Cliniche Intensivologiche e Perioperatorie, Università Cattolica del Sacro Cuore, Rome, Italy
,
Giovanni Vento
1   Dipartimento di Scienze della Salute della Donna, del Bambino e di Sanità Pubblica, Fondazione Policlinico Universitario A. Gemelli Istituto Di Ricovero E Cura A Carattere Scientifico, Rome, Italy
2   Dipartimento di Scienze della Vita e Sanità Pubblica, Università Cattolica del Sacro Cuore, Rome, Italy
› Author Affiliations
Funding None.

Abstract

Objective The study aimed to report a novel coronavirus disease 2019 (COVID-19)-associated multisystem inflammatory syndrome in children (MIS-C) in a neonate found to have an atypical diffuse thickening in coronary artery walls whose diagnosis required a multi-imaging approach.

Study Design A neonate presented at birth with multiple organ involvement and coronary artery anomalies. A diagnosis of MIS-C associated with COVID-19 was supported by maternal severe acute respiratory syndrome-coronavirus-2 (SARS-CoV-2) infection during pregnancy, and by the presence of both immunoglobulin (Ig)-G against SARS-CoV-2 and spike-specific memory B-cells response in the neonatal blood. Other plausible causes of the multiple organ involvement were excluded.

Result At admission, a severe coronary artery dilatation was identified on echocardiography, supporting the diagnosis of the MIS-C Kawasaki-like disease; however, coronary artery internal diameters were found to be normal using cardiac computed tomography angiography. At discharge, comparing the two imaging techniques each other, the correct diagnosis resulted to be an abnormal thickening in coronary arterial walls. These findings suggest that the inflammatory process affecting the coronary arterial wall in MIS-C could result not only in typical coronary artery lesions such as dilatation of the lumen or aneurysms development but also in abnormal thickening of the coronary artery wall.

Conclusion Our case provides an alert for pediatric cardiologists about the complexity to assess coronary artery involvement in MIS-C and raises the question that whether an abnormal vascular remodeling, with normal inner diameters, is to be considered like coronary artery dilatation for risk stratification.

Key Points

  • COVID-19 associated MIS-C can present in neonates with multiple organ involvement.

  • Coronary artery assessment in neonatal MIS-C could be complex, and a multi-imaging approach could be required.

  • Beside the typical coronary artery lesions, such as dilatation of the lumen or aneurysms, also abnormal thickening of the coronary artery wall can occur.

* These authors contributed equally to this work.




Publication History

Received: 22 November 2021

Accepted: 05 January 2022

Accepted Manuscript online:
07 January 2022

Article published online:
31 January 2022

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  • References

  • 1 World Health Organization. Multisystem inflammatory syndrome in children and adolescents with COVID-19. Accessed January 17, 2022 at: https://www.who.int/publications/i/item/multisystem-inflammatory-syndrome-in-children-and-adolescents-with-covid-19
  • 2 CDC Health Alert Network. Multisystem inflammatory syndrome in children (MIS-C) associated with coronavirus disease. Accessed January 2, 2021 at: https://emergency. cdc.gov/han/2020/han00432.asp
  • 3 Royal College of Paediatrics and Child Health. Paediatric multisystem inflammatory syndrome temporally associated with COVID-19 (PIMS): guidance for clinicians. Accessed January 17, 2022 at: https://www.rcpch.ac.uk/resources/guidance-paediatric-multisystem-inflammatory-syndrome-temporally-associated-Covid-19-pims
  • 4 Henderson LA, Canna SW, Friedman KG. et al. American College of Rheumatology Clinical Guidance for multisystem inflammatory syndrome in children associated with SARS-CoV-2 and hyperinflammation in pediatric COVID-19: version 2. Arthritis Rheumatol 2021; 73 (04) e13-e29
  • 5 Divekar AA, Patamasucon P, Benjamin JS. Presumptive neonatal multisystem inflammatory syndrome in children associated with coronavirus disease 2019. Am J Perinatol 2021; 38 (06) 632-636
  • 6 Khaund Borkotoky R, Banerjee Barua P, Paul SP, Heaton PA. COVID-19-related potential multisystem inflammatory syndrome in childhood in a neonate presenting as persistent pulmonary hypertension of the newborn. Pediatr Infect Dis J 2021; 40 (04) e162-e164
  • 7 Kappanayil M, Balan S, Alawani S. et al. Multisystem inflammatory syndrome in a neonate, temporally associated with prenatal exposure to SARS-CoV-2: a case report. Lancet Child Adolesc Health 2021; 5 (04) 304-308
  • 8 Lima ARO, Cardoso CC, Bentim PRB. et al. Maternal SARS-CoV-2 infection associated to systemic inflammatory response and pericardial effusion in the newborn: a case report. J Pediatric Infect Dis Soc 2021; 10 (04) 536-539
  • 9 Shaiba LA, Hadid A, Altirkawi KA. et al. Case report: neonatal multi-system inflammatory syndrome associated with SARS-CoV-2 exposure in two cases from Saudi Arabia. Front Pediatr 2021; 9: 652857
  • 10 Pawar R, Gavade V, Patil N. et al. Neonatal multisystem inflammatory syndrome (MIS-N) associated with prenatal maternal SARS-CoV-2: a case series. Children (Basel) 2021; 8 (07) 572
  • 11 Diggikar S, Nanjegowda R, Kumar A, Kumar V, Kulkarni S, Venkatagiri P. Neonatal multisystem inflammatory syndrome secondary to SARS-CoV-2 infection. J Paediatr Child Health 2021; 13: x
  • 12 Diwakar K, Gupta BK, Uddin MW, Sharma A, Jhajra S. Multisystem inflammatory syndrome with persistent neutropenia in neonate exposed to SARS-CoV-2 virus: a case report and review of literature. J Neonatal Perinatal Med 2021; (e-pub ahead of print) DOI: 10.3233/NPM-210839.
  • 13 Tsuchihashi T, Kakimoto N, Takeuchi T. et al. Intimal thickening and disruption of the media occur in the arterial walls of coronary arteries not associated with coronary arterial aneurysms in patients with Kawasaki disease. BMC Cardiovasc Disord 2021; 21 (01) 278