Case Report
Treatment of Shiga-Toxin Hus with Severe Neurologic
Features with Eculizumab
Jacob H. Umscheid ,
1
Collin Nevil,
1
Rhythm Vasudeva,
1
Mohammed Farhan Ali,
1,2
and Nisha Agasthya
1
1
University of Kansas, School of Medicine, Wichita, USA
2
Children’s Mercy Hospital, Department of Nephrology, Kansas City, USA
Correspondence should be addressed to Jacob H. Umscheid; jumscheid3@kumc.edu
Received 16 August 2021; Accepted 5 November 2021; Published 13 November 2021
Academic Editor: Junji Takaya
Copyright © 2021 Jacob H. Umscheid et al. is is an open access article distributed under the Creative Commons Attribution
License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is
properly cited.
Hemolytic Uremic Syndrome (HUS) is a constellation of microangiopathic hemolytic anemia, thrombocytopenia, and acute renal
failure. Shiga toxin-producing Escherichia coli- (STEC-) mediated HUS is a common cause of acute renal failure in children and
can rarely result in severe neurological complications such as encephalopathy, seizures, cerebrovascular accidents, and coma.
Current literature supports use of eculizumab, a monoclonal antibody that blocks complement activation, in atypical HUS
(aHUS). However, those with neurologic complications from STEC-HUS have complement activation and deposition of ag-
gregates in microvasculature and may be treated with eculizumab. In this case report, we describe a 3-year-old boy with diarrhea-
positive STEC-HUS who developed severe neurologic involvement in addition to acute renal failure requiring renal replacement
therapy. He was initiated on eculizumab therapy, with clinical improvement and organ recovery. is case highlights systemic
complications of STEC-HUS in a pediatric patient. e current literature is limited but has suggested a role for complement
mediation in cases with severe complications. We review the importance of early recognition of complications, use of eculizumab,
and current data available.
1. Introduction
Shiga toxin-producing Escherichia coli (STEC) is a well-
known etiology for gastroenteritis and colitis; however, the
toxin can traverse through the gastrointestinal system into
the bloodstream causing injury to vascular endothelial cells
[1, 2]. is results in a microangiopathic hemolytic anemia,
thrombocytopenia, and renal failure, also known as he-
molytic uremic syndrome (HUS) [1, 3]. HUS is a common
cause of acute kidney injury in children, accounting for
around 17% of cases and usually between the ages of 1 to 5
years (74%) [4]. is is secondary to toxin binding to the
renal endothelial cells, but may extend to extrarenal tissue
resulting in severe neurological complications of altered
mental status, seizures, cerebrovascular accidents, and coma
[5]. When neurologic complications of HUS are present, the
risk of severe sequelae and mortality significantly increases
[6, 7]. In a large retrospective cohort study of 3915 pediatric
patients, 10.4% developed acute neurologic manifestations
and had significantly worse mortality (13.9%) when com-
pared to HUS patients without neurologic involvement
(1.8%) [6].
Endothelial injury to small vessels through Shiga-like
toxin binding to globotriaosylceramide (Gb3) receptors and
microvascular thrombosis results in the majority of damage
to organs in HUS [8]. e Gb3 receptor is present not only
throughout renal and cerebral microvascular endothelial
cells but also on neuronal cells, placing the entire central
nervous system (CNS) at risk for injury [8]. Also seen is an
increase in cytokine production in response to Shiga-like
toxin at the blood-brain barrier resulting in cytotoxic effects
to the CNS parenchyma [7, 9–11]. is increased inflam-
matory response may suggest why immunomodulators, such
as eculizumab, may have a role in management.
Hindawi
Case Reports in Pediatrics
Volume 2021, Article ID 8053246, 6 pages
https://doi.org/10.1155/2021/8053246