Hemolytic uremic syndrome in the setting of COVID-19 successfully treated with complement inhibition therapy: An instructive case report of a previously healthy toddler and review of literature

IntroductionAs the global pandemic continues, new complications of COVID-19 in pediatric population have turned up, one of them being hemolytic uremic syndrome (HUS), a complement-mediated thrombotic microangiopathy (CM-TMA) characterized by triad of thrombocytopenia, microangiopathic hemolytic anem...

Full description

Bibliographic Details
Main Authors: Matija Matošević, Ivanka Kos, Maša Davidović, Maja Ban, Hana Matković, Ivan Jakopčić, Ivana Vuković Brinar, Ágnes Szilágyi, Dorottya Csuka, György Sinkovits, Zoltán Prohászka, Kristina Vrljičak, Lovro Lamot
Format: Article
Language:English
Published: Frontiers Media S.A. 2023-02-01
Series:Frontiers in Pediatrics
Subjects:
Online Access:https://www.frontiersin.org/articles/10.3389/fped.2023.1092860/full
_version_ 1797924610488926208
author Matija Matošević
Ivanka Kos
Maša Davidović
Maja Ban
Hana Matković
Ivan Jakopčić
Ivana Vuković Brinar
Ivana Vuković Brinar
Ágnes Szilágyi
Dorottya Csuka
György Sinkovits
Zoltán Prohászka
Zoltán Prohászka
Kristina Vrljičak
Lovro Lamot
Lovro Lamot
author_facet Matija Matošević
Ivanka Kos
Maša Davidović
Maja Ban
Hana Matković
Ivan Jakopčić
Ivana Vuković Brinar
Ivana Vuković Brinar
Ágnes Szilágyi
Dorottya Csuka
György Sinkovits
Zoltán Prohászka
Zoltán Prohászka
Kristina Vrljičak
Lovro Lamot
Lovro Lamot
author_sort Matija Matošević
collection DOAJ
description IntroductionAs the global pandemic continues, new complications of COVID-19 in pediatric population have turned up, one of them being hemolytic uremic syndrome (HUS), a complement-mediated thrombotic microangiopathy (CM-TMA) characterized by triad of thrombocytopenia, microangiopathic hemolytic anemia and acute kidney injury (AKI). With both multisystem inflammatory syndrome in children (MIS-C) and HUS sharing complement dysregulation as one of the key factors, the aim of this case report is to highlight differences between these two conditions and also emphasize the importance of complement blockade as a treatment modality.Case reportWe describe a 21-month-old toddler who initially presented with fever and confirmed COVID-19. His condition quickly deteriorated and he developed oliguria, accompanied with diarrhea, vomiting and oral intake intolerance. HUS was suspected, supported with compelling laboratory findings, including decreased platelets count and C3 levels, elevated LDH, urea, serum creatinine and sC5b-9 and presence of schistocytes in peripheral blood, negative fecal Shiga toxin and normal ADAMTS13 metalloprotease activity. The patient was given C5 complement blocker Ravulizumab and started to display rapid improvement.ConclusionAlthough reports of HUS in the setting of COVID-19 continue to pour in, the questions of exact mechanism and similarities to MIS-C remain. Our case for the first time accentuates the use of complement blockade as a valuable treatment option in this scenario. We sincerely believe that reporting on HUS as a complication of COVID-19 in children will give rise to improved diagnosis and treatment, as well as better understanding of both of these intricating diseases.
first_indexed 2024-04-10T15:04:44Z
format Article
id doaj.art-e7efc578371b4f5eb05cbe2fb4098080
institution Directory Open Access Journal
issn 2296-2360
language English
last_indexed 2024-04-10T15:04:44Z
publishDate 2023-02-01
publisher Frontiers Media S.A.
record_format Article
series Frontiers in Pediatrics
spelling doaj.art-e7efc578371b4f5eb05cbe2fb40980802023-02-15T08:41:39ZengFrontiers Media S.A.Frontiers in Pediatrics2296-23602023-02-011110.3389/fped.2023.10928601092860Hemolytic uremic syndrome in the setting of COVID-19 successfully treated with complement inhibition therapy: An instructive case report of a previously healthy toddler and review of literatureMatija Matošević0Ivanka Kos1Maša Davidović2Maja Ban3Hana Matković4Ivan Jakopčić5Ivana Vuković Brinar6Ivana Vuković Brinar7Ágnes Szilágyi8Dorottya Csuka9György Sinkovits10Zoltán Prohászka11Zoltán Prohászka12Kristina Vrljičak13Lovro Lamot14Lovro Lamot15Department of Pediatrics, University of Zagreb School of Medicine, Zagreb, CroatiaDivision of Nephrology, Dialysis and Transplantation, Department of Pediatrics, University Hospital Center Zagreb, Zagreb, CroatiaDivision of Nephrology, Dialysis and Transplantation, Department of Pediatrics, University Hospital Center Zagreb, Zagreb, CroatiaDivision of Nephrology, Dialysis and Transplantation, Department of Pediatrics, University Hospital Center Zagreb, Zagreb, CroatiaDivision of Nephrology, Dialysis and Transplantation, Department of Pediatrics, University Hospital Center Zagreb, Zagreb, CroatiaDivision of Nephrology, Dialysis and Transplantation, Department of Pediatrics, University Hospital Center Zagreb, Zagreb, CroatiaDepartment of Nephrology, Hypertension, Dialysis and Transplantation, University Hospital Center Zagreb, Zagreb, CroatiaDepartment of Internal Medicine and Haematology, Semmelweis University, Budapest, HungaryDepartment of Internal Medicine and Haematology, Semmelweis University, Budapest, HungaryDepartment of Internal Medicine and Haematology, Semmelweis University, Budapest, HungaryDepartment of Internal Medicine and Haematology, Semmelweis University, Budapest, HungaryDepartment of Internal Medicine and Haematology, Semmelweis University, Budapest, HungaryResearch Group for Immunology and Haematology, Semmelweis University- Eötvös Loránd Research Network (Office for Supported Research Groups), Budapest, HungaryDivision of Nephrology, Dialysis and Transplantation, Department of Pediatrics, University Hospital Center Zagreb, Zagreb, CroatiaDepartment of Pediatrics, University of Zagreb School of Medicine, Zagreb, CroatiaDivision of Nephrology, Dialysis and Transplantation, Department of Pediatrics, University Hospital Center Zagreb, Zagreb, CroatiaIntroductionAs the global pandemic continues, new complications of COVID-19 in pediatric population have turned up, one of them being hemolytic uremic syndrome (HUS), a complement-mediated thrombotic microangiopathy (CM-TMA) characterized by triad of thrombocytopenia, microangiopathic hemolytic anemia and acute kidney injury (AKI). With both multisystem inflammatory syndrome in children (MIS-C) and HUS sharing complement dysregulation as one of the key factors, the aim of this case report is to highlight differences between these two conditions and also emphasize the importance of complement blockade as a treatment modality.Case reportWe describe a 21-month-old toddler who initially presented with fever and confirmed COVID-19. His condition quickly deteriorated and he developed oliguria, accompanied with diarrhea, vomiting and oral intake intolerance. HUS was suspected, supported with compelling laboratory findings, including decreased platelets count and C3 levels, elevated LDH, urea, serum creatinine and sC5b-9 and presence of schistocytes in peripheral blood, negative fecal Shiga toxin and normal ADAMTS13 metalloprotease activity. The patient was given C5 complement blocker Ravulizumab and started to display rapid improvement.ConclusionAlthough reports of HUS in the setting of COVID-19 continue to pour in, the questions of exact mechanism and similarities to MIS-C remain. Our case for the first time accentuates the use of complement blockade as a valuable treatment option in this scenario. We sincerely believe that reporting on HUS as a complication of COVID-19 in children will give rise to improved diagnosis and treatment, as well as better understanding of both of these intricating diseases.https://www.frontiersin.org/articles/10.3389/fped.2023.1092860/fullhemolytic uremic syndromeHUSthrombotic microangiopathyTMACOVID-19complement blockade
spellingShingle Matija Matošević
Ivanka Kos
Maša Davidović
Maja Ban
Hana Matković
Ivan Jakopčić
Ivana Vuković Brinar
Ivana Vuković Brinar
Ágnes Szilágyi
Dorottya Csuka
György Sinkovits
Zoltán Prohászka
Zoltán Prohászka
Kristina Vrljičak
Lovro Lamot
Lovro Lamot
Hemolytic uremic syndrome in the setting of COVID-19 successfully treated with complement inhibition therapy: An instructive case report of a previously healthy toddler and review of literature
Frontiers in Pediatrics
hemolytic uremic syndrome
HUS
thrombotic microangiopathy
TMA
COVID-19
complement blockade
title Hemolytic uremic syndrome in the setting of COVID-19 successfully treated with complement inhibition therapy: An instructive case report of a previously healthy toddler and review of literature
title_full Hemolytic uremic syndrome in the setting of COVID-19 successfully treated with complement inhibition therapy: An instructive case report of a previously healthy toddler and review of literature
title_fullStr Hemolytic uremic syndrome in the setting of COVID-19 successfully treated with complement inhibition therapy: An instructive case report of a previously healthy toddler and review of literature
title_full_unstemmed Hemolytic uremic syndrome in the setting of COVID-19 successfully treated with complement inhibition therapy: An instructive case report of a previously healthy toddler and review of literature
title_short Hemolytic uremic syndrome in the setting of COVID-19 successfully treated with complement inhibition therapy: An instructive case report of a previously healthy toddler and review of literature
title_sort hemolytic uremic syndrome in the setting of covid 19 successfully treated with complement inhibition therapy an instructive case report of a previously healthy toddler and review of literature
topic hemolytic uremic syndrome
HUS
thrombotic microangiopathy
TMA
COVID-19
complement blockade
url https://www.frontiersin.org/articles/10.3389/fped.2023.1092860/full
work_keys_str_mv AT matijamatosevic hemolyticuremicsyndromeinthesettingofcovid19successfullytreatedwithcomplementinhibitiontherapyaninstructivecasereportofapreviouslyhealthytoddlerandreviewofliterature
AT ivankakos hemolyticuremicsyndromeinthesettingofcovid19successfullytreatedwithcomplementinhibitiontherapyaninstructivecasereportofapreviouslyhealthytoddlerandreviewofliterature
AT masadavidovic hemolyticuremicsyndromeinthesettingofcovid19successfullytreatedwithcomplementinhibitiontherapyaninstructivecasereportofapreviouslyhealthytoddlerandreviewofliterature
AT majaban hemolyticuremicsyndromeinthesettingofcovid19successfullytreatedwithcomplementinhibitiontherapyaninstructivecasereportofapreviouslyhealthytoddlerandreviewofliterature
AT hanamatkovic hemolyticuremicsyndromeinthesettingofcovid19successfullytreatedwithcomplementinhibitiontherapyaninstructivecasereportofapreviouslyhealthytoddlerandreviewofliterature
AT ivanjakopcic hemolyticuremicsyndromeinthesettingofcovid19successfullytreatedwithcomplementinhibitiontherapyaninstructivecasereportofapreviouslyhealthytoddlerandreviewofliterature
AT ivanavukovicbrinar hemolyticuremicsyndromeinthesettingofcovid19successfullytreatedwithcomplementinhibitiontherapyaninstructivecasereportofapreviouslyhealthytoddlerandreviewofliterature
AT ivanavukovicbrinar hemolyticuremicsyndromeinthesettingofcovid19successfullytreatedwithcomplementinhibitiontherapyaninstructivecasereportofapreviouslyhealthytoddlerandreviewofliterature
AT agnesszilagyi hemolyticuremicsyndromeinthesettingofcovid19successfullytreatedwithcomplementinhibitiontherapyaninstructivecasereportofapreviouslyhealthytoddlerandreviewofliterature
AT dorottyacsuka hemolyticuremicsyndromeinthesettingofcovid19successfullytreatedwithcomplementinhibitiontherapyaninstructivecasereportofapreviouslyhealthytoddlerandreviewofliterature
AT gyorgysinkovits hemolyticuremicsyndromeinthesettingofcovid19successfullytreatedwithcomplementinhibitiontherapyaninstructivecasereportofapreviouslyhealthytoddlerandreviewofliterature
AT zoltanprohaszka hemolyticuremicsyndromeinthesettingofcovid19successfullytreatedwithcomplementinhibitiontherapyaninstructivecasereportofapreviouslyhealthytoddlerandreviewofliterature
AT zoltanprohaszka hemolyticuremicsyndromeinthesettingofcovid19successfullytreatedwithcomplementinhibitiontherapyaninstructivecasereportofapreviouslyhealthytoddlerandreviewofliterature
AT kristinavrljicak hemolyticuremicsyndromeinthesettingofcovid19successfullytreatedwithcomplementinhibitiontherapyaninstructivecasereportofapreviouslyhealthytoddlerandreviewofliterature
AT lovrolamot hemolyticuremicsyndromeinthesettingofcovid19successfullytreatedwithcomplementinhibitiontherapyaninstructivecasereportofapreviouslyhealthytoddlerandreviewofliterature
AT lovrolamot hemolyticuremicsyndromeinthesettingofcovid19successfullytreatedwithcomplementinhibitiontherapyaninstructivecasereportofapreviouslyhealthytoddlerandreviewofliterature