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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 
1   Department of Pediatric Critical Care Medicine, Bezmialem Vakif University, Istanbul, Turkey
,
Can Yilmaz Yozgat 
2   Faculty of Medicine, Bezmialem Vakif University, Istanbul, Turkey
,
Nurver Akinci 
3   Department of Pediatric Nephrology, Bezmialem Vakif University, Istanbul, Turkey
,
Sirin Sonmez
4   Department of Pediatrics, Bezmialem Vakif University, Istanbul, Turkey
,
Eser Tekin
4   Department of Pediatrics, Bezmialem Vakif University, Istanbul, Turkey
,
Faraz Talebazadeh 
4   Department of Pediatrics, Bezmialem Vakif University, Istanbul, Turkey
,
Uzeyir Jafarov
4   Department of Pediatrics, Bezmialem Vakif University, Istanbul, Turkey
,
Hafize Otcu Temur 
5   Department of Radiology, Bezmialem Vakif University, Istanbul, Turkey
,
Yilmaz Yozgat 
6   Department of Pediatric Cardiology, Bezmialem Vakif University, Istanbul, Turkey
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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

Authors' Contributions

All authors participated in creating content for the manuscript, editing, and provided final approval for submission. No undisclosed authors contributed to the manuscript.


Ethical Approval

Permission was granted by the parents and patient to publish the case report.




Publication History

Received: 14 February 2020

Accepted: 02 May 2020

Article published online:
05 August 2020

© 2020. Thieme. All rights reserved.

Georg Thieme Verlag KG
Rüdigerstraße 14, 70469 Stuttgart, Germany

 

  • References

  • 1 Canpolat N. Hemolytic uremic syndrome. Turk Pediatri Ars 2015; 50 (02) 73-82
    CrossrefPubMedGoogle Scholar
  • 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
    CrossrefPubMedGoogle Scholar
  • 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
    CrossrefPubMedGoogle Scholar
  • 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
    CrossrefPubMedGoogle Scholar
  • 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
    CrossrefPubMedGoogle Scholar
  • 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
    CrossrefPubMedGoogle Scholar
  • 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
    CrossrefPubMedGoogle Scholar
  • 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
    CrossrefPubMedGoogle Scholar
  • 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
    CrossrefPubMedGoogle Scholar
  • 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
    CrossrefPubMedGoogle Scholar
  • 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
    CrossrefPubMedGoogle Scholar
  • 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
    CrossrefPubMedGoogle Scholar
  • 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
    CrossrefPubMedGoogle Scholar
  • 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
    CrossrefPubMedGoogle Scholar
  • 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
    CrossrefPubMedGoogle Scholar