Acute Myocarditis and Eculizumab Caused Severe Cholestasis in a 17-Month-Old Child Who Has Hemolytic Uremic Syndrome Associated with Shiga Toxin-Producing Escherichia coli

Osman Yesilbas; Can Yilmaz Yozgat; Nurver Akinci; Sirin Sonmez; Eser Tekin; Faraz Talebazadeh; Uzeyir Jafarov; Hafize Otcu Temur; Yilmaz Yozgat

doi:10.1055/s-0040-1713111

Journal of Pediatric Intensive Care, Inhaltsverzeichnis

J Pediatr Intensive Care 2021; 10(03): 216-220
DOI: 10.1055/s-0040-1713111

Case Report

Acute Myocarditis and Eculizumab Caused Severe Cholestasis in a 17-Month-Old Child Who Has Hemolytic Uremic Syndrome Associated with Shiga Toxin-Producing Escherichia coli

Osman Yesilbas

¹Department of Pediatric Critical Care Medicine, Bezmialem Vakif University, Istanbul, Turkey

,

Can Yilmaz Yozgat

²Faculty of Medicine, Bezmialem Vakif University, Istanbul, Turkey

,

Nurver Akinci

³Department of Pediatric Nephrology, Bezmialem Vakif University, Istanbul, Turkey

,

Sirin Sonmez

⁴Department of Pediatrics, Bezmialem Vakif University, Istanbul, Turkey

,

Eser Tekin

⁴Department of Pediatrics, Bezmialem Vakif University, Istanbul, Turkey

,

Faraz Talebazadeh

⁴Department of Pediatrics, Bezmialem Vakif University, Istanbul, Turkey

,

Uzeyir Jafarov

⁴Department of Pediatrics, Bezmialem Vakif University, Istanbul, Turkey

,

Hafize Otcu Temur

⁵Department of Radiology, Bezmialem Vakif University, Istanbul, Turkey

,

Yilmaz Yozgat

⁶Department of Pediatric Cardiology, Bezmialem Vakif University, Istanbul, Turkey

› Institutsangaben

Abstract

Cardiovascular involvement is uncommon in pediatric patients with hemolytic uremic syndrome associated with Shiga toxin-producing Escherichia coli (STEC-HUS). In this case report we presented a case of 17-month-old toddler who had a sporadic type of STEC-HUS complicated by acute myocarditis. The patient was successfully treated by a single dose of eculizumab after six doses of therapeutic plasma exchange (TPE) were inefficient to prevent the cardiac complication. Hepatotoxicity was observed after a single dose of eculizumab. Hepatic and cholestatic enzyme levels slowly returned to normal within 6 months. To the best of our knowledge, this is the first case of myocarditis/cardiomyopathy treated with eculizumab in STEC-HUS. This case illustrates the need for vigilance regarding myocardial involvement and eculizumab-induced hepatotoxicity in STEC-HUS.

Keywords

hemolytic uremic syndrome - myocarditis - eculizumab - therapeutic plasma exchange

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Referenzen

References
1 Canpolat N. Hemolytic uremic syndrome. Turk Pediatri Ars 2015; 50 (02) 73-82
2 Khalid M, Andreoli S. Extrarenal manifestations of the hemolytic uremic syndrome associated with Shiga toxin-producing Escherichia coli (STEC HUS). Pediatr Nephrol 2019; 34 (12) 2495-2507
3 Thayu M, Chandler WL, Jelacic S, Gordon CA, Rosenthal GL, Tarr PI. Cardiac ischemia during hemolytic uremic syndrome. Pediatr Nephrol 2003; 18 (03) 286-289
4 F Andersen R, V Bjerre J, V Povlsen J, Veien M, Kamperis K, Rittig S. HUS-induced cardiac and circulatory failure is reversible using cardiopulmonary bypass as rescue. Pediatr Nephrol 2017; 32 (11) 2155-2158
5 Palanca Arias D, López Ramón M, Jiménez Montañés L. Biomarkers detect involvement of acute myocardial injury in a paediatric haemolytic-uraemic syndrome patient. Cardiol Young 2016; 26 (05) 983-986
6 Abu-Arafeh I, Gray E, Youngson G, Auchterlonie I, Russell G. Myocarditis and haemolytic uraemic syndrome. Arch Dis Child 1995; 72 (01) 46-47
7 Ray CG, Portman JN, Stamm SJ, Hickman RO. Hemolytic-uremic syndrome and myocarditis. Association with coxsackievirus B infection. Am J Dis Child 1971; 122 (05) 418-420
8 Buelli S, Zoja C, Remuzzi G, Morigi M. Complement activation contributes to the pathophysiology of shiga toxin-associated hemolytic uremic syndrome. Microorganisms 2019; 7 (01) E15
9 Loos S, Oh J, Kemper MJ. Eculizumab in STEC-HUS: need for a proper randomized controlled trial. Pediatr Nephrol 2018; 33 (08) 1277-1281
10 Mahat U, Matar RB, Rotz SJ. Use of complement monoclonal antibody eculizumab in Shiga toxin producing Escherichia coli associated hemolytic uremic syndrome: a review of current evidence. Pediatr Blood Cancer 2019; 66 (11) e27913
11 Percheron L, Gramada R, Tellier S. et al. Eculizumab treatment in severe pediatric STEC-HUS: a multicenter retrospective study. Pediatr Nephrol 2018; 33 (08) 1385-1394
12 Hayes W, Tschumi S, Ling SC, Feber J, Kirschfink M, Licht C. Eculizumab hepatotoxicity in pediatric aHUS. Pediatr Nephrol 2015; 30 (05) 775-781
13 Oruc A, Ayar Y, Vuruskan BA. et al. Hepatotoxicity associated with eculizumab in a patient with atypical hemolytic uremic syndrome. Nefrologia 2018; 38 (04) 448-450
14 Yesilbas O, Sevketoglu E, Petmezci MT. et al. Infant onset severe complement-mediated hemolytic uremic syndrome complicated by secondary sclerosing cholangitis. J Clin Apher 2018; 33 (05) 619-623
15 Mauras M, Bacchetta J, Duncan A. et al. Escherichia coli-associated hemolytic uremic syndrome and severe chronic hepatocellular cholestasis: complication or side effect of eculizumab?. Pediatr Nephrol 2019; 34 (07) 1289-1293