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The first case of pediatric inflammatory multisystem syndrome temporally associated with SARS-CoV-2 infection (PIMS-TS) in Poland, complicated by giant coronary artery aneurysms

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KARDIOLOGIA POLSKA 2020; 78 (10) 1064

5.67) (FIGURE 1), without any myocardial contrac‑

tility disorders or pericardial effusion.

Except for prolonged fever, none of the typ‑

ical Kawasaki disease symptoms were present.

However, because of laboratory findings and cor‑

onary artery aneurysms (CAAs), Kawasaki dis‑

ease–specific treatment was administered. Fe‑

ver resolved after intravenous immunoglobulin administration (2 g/kg of body weight). In addi‑

tion, an antiplatelet agent (acetylsalicylic acid) and warfarin were introduced. The patient was discharged home in good condition. The coro‑

nary artery aneurysms persisted on follow ‑up echocardiography performed after 3 months.

The presented patient was, to our knowledge, the first case of pediatric inflammatory multi‑

system syndrome temporally associated with SARS‑CoV‑2 infection (PIMS ‑TS) in Poland.

At that time, except for the Royal College of Pe‑

diatrics and Child Health recommendations, no information on this disease was available in the medical literature. Since then, several defi‑

nitions of PIMS (called multisystem inflamma‑

tory syndrome in children [MIS ‑C] in the Unit‑

ed States) were developed.1‑3

Reports from various countries1,4,5 have pro‑

vided a growing set of data on children present‑

ing with signs and symptoms overlapping with Kawasaki disease and toxic shock syndrome, with frequent cardiac involvement and pro‑

gression to shock. Compared with those data, our patient was at younger age than the medi‑

an age of patients with Kawasaki disease–like On May 3, 2020, a 2‑year ‑old boy in good con‑

dition, with no significant medical history, was admitted to our hospital on the 10th day of fever (body temperature of up to 40 °C) af‑

ter ineffective oral empiric antibiotic therapy with amoxicillin with clavulanic acid. During the first 2 days of the disease, he complained of joint pain and mild diarrhea. No other signs or symptoms were reported. About 5 weeks earli‑

er (at the beginning of the coronavirus disease 2019 [COVID‑19] pandemic in Poland), he and his family suffered from mild upper respirato‑

ry tract infection accompanied by low ‑grade fever and rhinosinusitis. No tests were per‑

formed at that time.

On admission, differential diagnosis did not indicate any typical cause of fever of unknown origin. The polymerase chain reaction (PCR) na‑

sopharyngeal swab test for severe acute respi‑

ratory syndrome coronavirus 2 (SARS ‑CoV‑2) yielded negative results twice. The result of the Anti ‑SARS ‑CoV‑2 ELISA test (Euroimmun, Lübeck, Germany) was positive for immuno‑

globulin G, and negative for immunoglobulin M.

Laboratory test results are presented in Sup‑

plementary material, Table S1. No abnormali‑

ties were found on chest X ‑ray and abdominal ultrasound. Transthoracic echocardiography showed a left main coronary artery aneurysm of 4.3 mm in diameter (z score, 5.67), a left an‑

terior descending artery aneurysm of 6.3 mm in diameter (z score, 13.17), and a right coronary artery aneurysm of 5.4 mm in diameter (z score,

Correspondence to:

Jacek Kusa, PhD, Department of  Pediatric Cardiology, Regional  Specialist Hospital in Wrocław,  Research and Development  Center, ul. Kamieńskiego 73A,  51‑124 Wrocław, Poland,  phone: +48 71 327 01 40,  email: jkusa@poczta.onet.pl Received: July 3, 2020.

Revision accepted:

September 20, 2020.

Published online:

September 25, 2020.

Kardiol Pol. 2020; 78 (10): 1064‑1065 doi:10.33963/KP.15623 Copyright by the Author(s), 2020

C L I N I C A L V I G N E T T E

The first case of pediatric inflammatory

multisystem syndrome temporally associated with SARS‑CoV‑2 infection (PIMS ‑TS) in Poland,

complicated by giant coronary artery aneurysms

Paweł Tracewski1, Kamila M. Ludwikowska2, Leszek Szenborn2, Jacek Kusa1

1  Department of Pediatric Cardiology, Regional Specialist Hospital in Wrocław, Research and Development Center, Wrocław, Poland 2  Department of Pediatric Infectious Diseases, Wrocław Medical University, Wrocław, Poland

(2)

C L I N I C A L V I G N E T T E Giant coronary artery aneurysms in PIMS ‑TS 1065 natriuretic peptide levels in our patient were within the reference range.

Coronary artery aneurysms can complicate all phenotypes of PIMS ‑TS, not only the Kawasa‑

ki disease–like presentation. Our case supports the conclusion derived from the current data, namely, indicating that all children in whom PIMS ‑TS is suspected should undergo cardiac evaluation. Further research is needed to estab‑

lish best therapeutic options for these patients, as they may differ from those known in Kawa‑

saki disease before the COVID‑19 pandemic.5 SUPPLEMENTARY MATERIAL

Supplementary material is available at www.mp.pl/kardiologiapolska.

ARTICLE INFORMATION

CONFLICT OF INTEREST None declared.

OPEN ACCESS This is an Open Access article distributed under the terms  of  the  Creative  Commons  Attribution ‑NonCommercial ‑NoDerivatives  4.0  In‑

ternational License (CC BY ‑NC ‑ND 4.0), allowing third parties to download ar‑

ticles and share them with others, provided the original work is properly cited,  not changed in any way, distributed under the same license, and used for non‑

commercial purposes only. For commercial use, please contact the journal office  at kardiologiapolska@ptkardio.pl.

HOW TO CITE Tracewski P, Ludwikowska KM, Szenborn L, Kusa J. The first  case of pediatric inflammatory multisystem syndrome temporally associated with  SARS‑CoV‑2 infection (PIMS ‑TS) in Poland, complicated by giant coronary artery  aneurysms. Kardiol Pol. 2020; 78: 1064‑1065. doi:10.33963/KP.15623

REFERENCES

1  European Centre for Disease Prevention and Control. Paediatric inflammato‑

ry multisystem syndrome and SARS ‑CoV‑2 infection in children. https://www.ecdc.

europa.eu/sites/default/files/documents/covid‑19‑risk‑assessment‑paediatric‑

inflammatory‑multisystem‑syndrome‑15‑May‑2020.pdf. Accessed May 15, 2020.

2  World  Health  Organization.  Multisystem  inflammatory  syndrome  in  chil‑

dren  and  adolescents  with  COVID‑19:  scientific  brief.  https://www.who.int/

publications ‑detail/multisystem ‑inflammatory ‑syndrome ‑in ‑children ‑and‑

‑adolescents ‑with ‑covid‑19. Accessed May 15, 2020.

3  CDC Centers for Disease Control and Prevention. Multisystem inflammato‑

ry syndrome in children (MIS ‑C) associated with coronavirus disease 2019 (CO‑

VID‑19).  https://emergency.cdc.gov/han/2020/han00432.asp.  Distributed  via  the CDC Health Alert Network on May 14, 2020.

4  Belhadjer Z, Méot M, Bajolle F, et al. Acute heart failure in multisystem inflam‑

matory syndrome in children (MIS ‑C) in the context of global SARS ‑CoV‑2 pandem‑

ic. Circulation. 2020 May 17. [Epub ahead of print].

5  Whittaker E, Bamford A, Kenny J, et al. Clinical characteristics of 58 children  with a pediatric inflammatory multisystem syndrome temporally associated with  SARS ‑CoV‑2. JAMA. 2020; 324: 259‑269.

PIMS ‑TS and the presentation was more typi‑

cal of Kawasaki disease. His disease course was not affected by most commonly described car‑

diac complications, ie, myocarditis and acute heart failure, but he developed CAAs—anoth‑

er common complication of PIMS. The size of CAAs with a z score greater than 10 was observed in the minority of PIMS ‑TS cases.5 His clinical symptoms did not correspond with the severi‑

ty of CAAs, but no clinical or laboratory CAA risk factors have been found so far.5 Laboratory test results (Supplementary material, Table S1) shared a few features with those most common‑

ly observed in PIMS: high levels of inflammato‑

ry markers, anemia, lymphopenia, and low sodi‑

um concentration. Interleukin 6 levels were not tested, but other biomarkers related to inflam‑

matory response—C ‑reactive protein concentra‑

tion and erythrocyte sedimentation rates—were significantly increased.

Unlike most PIMS cases, ferritin and N ‑ter‑

minal fragment of the  prohormone brain FIGURE 1  Echocardiography in a 2‑year ‑old boy with pediatric inflammatory multisystem  syndrome accompanied by giant coronary artery aneurysms: A – right coronary artery aneurysm (arrow); B – left coronary artery aneurysm (arrow)

A

B

Cytaty

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