Hemolytic uremic syndrome due to Shiga toxin–producing Escherichia coli and hypocomplementemia with favorable response to eculizumab: a case report

Introduction: Neurologic involvement in hemolytic uremic syndrome related to Shiga toxin–producing Escherichia coli (STEC-HUS) is the main cause of death. In last years has been demonstrated that activation of complement alternative pathway also contributes to organ damage. This finding led to the r...

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Autores principales: Balestracci, Alejandro, Meni Battaglia, Luciana, Martin, Sandra Mariel, Toledo, Ismael, Puyol, Iris, Beaudoin, Laura, Robledo, Natalia Luján
Formato: Artículo revista
Lenguaje:Español
Publicado: Universidad Nacional Córdoba. Facultad de Ciencias Médicas. Secretaria de Ciencia y Tecnología 2021
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Acceso en línea:https://revistas.unc.edu.ar/index.php/med/article/view/29934
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id I10-R327-article-29934
record_format ojs
institution Universidad Nacional de Córdoba
institution_str I-10
repository_str R-327
container_title_str Revista de la Facultad de Ciencias Médicas de Córdoba
language Español
format Artículo revista
topic hemolytic-uremic syndrome
escherichia coli
complement C3
seizures
síndrome hemolítico-urémico
escherichia coli
complemento C3
convulsiones
síndrome hemolítico-urêmica
escherichia coli
complemento C3
convulsões
spellingShingle hemolytic-uremic syndrome
escherichia coli
complement C3
seizures
síndrome hemolítico-urémico
escherichia coli
complemento C3
convulsiones
síndrome hemolítico-urêmica
escherichia coli
complemento C3
convulsões
Balestracci, Alejandro
Meni Battaglia, Luciana
Martin, Sandra Mariel
Toledo, Ismael
Puyol, Iris
Beaudoin, Laura
Robledo, Natalia Luján
Hemolytic uremic syndrome due to Shiga toxin–producing Escherichia coli and hypocomplementemia with favorable response to eculizumab: a case report
topic_facet hemolytic-uremic syndrome
escherichia coli
complement C3
seizures
síndrome hemolítico-urémico
escherichia coli
complemento C3
convulsiones
síndrome hemolítico-urêmica
escherichia coli
complemento C3
convulsões
author Balestracci, Alejandro
Meni Battaglia, Luciana
Martin, Sandra Mariel
Toledo, Ismael
Puyol, Iris
Beaudoin, Laura
Robledo, Natalia Luján
author_facet Balestracci, Alejandro
Meni Battaglia, Luciana
Martin, Sandra Mariel
Toledo, Ismael
Puyol, Iris
Beaudoin, Laura
Robledo, Natalia Luján
author_sort Balestracci, Alejandro
title Hemolytic uremic syndrome due to Shiga toxin–producing Escherichia coli and hypocomplementemia with favorable response to eculizumab: a case report
title_short Hemolytic uremic syndrome due to Shiga toxin–producing Escherichia coli and hypocomplementemia with favorable response to eculizumab: a case report
title_full Hemolytic uremic syndrome due to Shiga toxin–producing Escherichia coli and hypocomplementemia with favorable response to eculizumab: a case report
title_fullStr Hemolytic uremic syndrome due to Shiga toxin–producing Escherichia coli and hypocomplementemia with favorable response to eculizumab: a case report
title_full_unstemmed Hemolytic uremic syndrome due to Shiga toxin–producing Escherichia coli and hypocomplementemia with favorable response to eculizumab: a case report
title_sort hemolytic uremic syndrome due to shiga toxin–producing escherichia coli and hypocomplementemia with favorable response to eculizumab: a case report
description Introduction: Neurologic involvement in hemolytic uremic syndrome related to Shiga toxin–producing Escherichia coli (STEC-HUS) is the main cause of death. In last years has been demonstrated that activation of complement alternative pathway also contributes to organ damage. This finding led to the recognition of decreased C3 levels at admission as a marker of poor prognosis as well as the evaluation of the use of eculizumab in cases with neurologic compromise. Objective: to report a patient with STEC-HUS and hypocomplementemia with neurological involvement treated with eculizumab. Clinical case: A 17-month-old male was admitted due to seizures and anuria for last 24 h with a history of 48 h of bloody diarrhea. He presented a laboratory profile compatible with STEC-HUS and severe hyponatremia, results of brain tomography were normal. Also there was complement activation: C3 73 mg/dl (normal > 90 mg/dL) and C5b-9 778.9 ng/ml (normal 135.8-385.3 ng/ml). Initial treatment includes normal saline solution and anticonvulsants drugs, sodium correction and peritoneal dialysis. On third day of hospitalization, because of progression of the neurologic involvement a dose of eculizumab (300 mg) was given, showing at 24 h a markedly neurologic improvement along with and increasing platelet count and a descending lactic dehydrogenase levels. He was discharged after 14 days in a good condition. Later a STEC O157:H7 infection was confirmed and he also normalized the C3 level.    Conclusion: This case shows that decreased C3 level at admission was associated to neurologic involvement and suggests that eculizumab might be a favorable therapeutic option.
publisher Universidad Nacional Córdoba. Facultad de Ciencias Médicas. Secretaria de Ciencia y Tecnología
publishDate 2021
url https://revistas.unc.edu.ar/index.php/med/article/view/29934
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spelling I10-R327-article-299342021-12-29T20:15:49Z Hemolytic uremic syndrome due to Shiga toxin–producing Escherichia coli and hypocomplementemia with favorable response to eculizumab: a case report Síndrome urémico hemolítico por Escherichia Coli e hipocomplementemia con respuesta favorable a eculizumab: comunicación de un caso Hemolytic uremic syndrome due to Shiga toxin–producing Escherichia coli and hypocomplementemia with favorable response to eculizumab: a case report Balestracci, Alejandro Meni Battaglia, Luciana Martin, Sandra Mariel Toledo, Ismael Puyol, Iris Beaudoin, Laura Robledo, Natalia Luján hemolytic-uremic syndrome escherichia coli complement C3 seizures síndrome hemolítico-urémico escherichia coli complemento C3 convulsiones síndrome hemolítico-urêmica escherichia coli complemento C3 convulsões Introduction: Neurologic involvement in hemolytic uremic syndrome related to Shiga toxin–producing Escherichia coli (STEC-HUS) is the main cause of death. In last years has been demonstrated that activation of complement alternative pathway also contributes to organ damage. This finding led to the recognition of decreased C3 levels at admission as a marker of poor prognosis as well as the evaluation of the use of eculizumab in cases with neurologic compromise. Objective: to report a patient with STEC-HUS and hypocomplementemia with neurological involvement treated with eculizumab. Clinical case: A 17-month-old male was admitted due to seizures and anuria for last 24 h with a history of 48 h of bloody diarrhea. He presented a laboratory profile compatible with STEC-HUS and severe hyponatremia, results of brain tomography were normal. Also there was complement activation: C3 73 mg/dl (normal > 90 mg/dL) and C5b-9 778.9 ng/ml (normal 135.8-385.3 ng/ml). Initial treatment includes normal saline solution and anticonvulsants drugs, sodium correction and peritoneal dialysis. On third day of hospitalization, because of progression of the neurologic involvement a dose of eculizumab (300 mg) was given, showing at 24 h a markedly neurologic improvement along with and increasing platelet count and a descending lactic dehydrogenase levels. He was discharged after 14 days in a good condition. Later a STEC O157:H7 infection was confirmed and he also normalized the C3 level.    Conclusion: This case shows that decreased C3 level at admission was associated to neurologic involvement and suggests that eculizumab might be a favorable therapeutic option. Introducción: En compromiso neurológico en el síndrome urémico hemolítico producido por Eschericha coli productor de Shiga toxina (STEC-SUH) es la primera causa de mortalidad. En los últimos años se ha demostrado que también contribuye al daño de órgano la activación de la vía alterna del complemento. Este hallazgo dio lugar al reconocimiento del descenso de C3 como marcador de mal pronóstico así como a la evaluación del uso de eculizumab ante compromiso neurológico severo. Objetivo: Comunicar un paciente con STEC-SUH e hipocomplementemia con compromiso neurológico tratado con eculizumab. Caso clínico: Varón de 17 meses que ingresa por síndrome convulsivo y 24 horas de anuria con antecedente de diarrea con sangre de 48 horas de evolución. Presentaba al ingreso laboratorio compatible con STEC-HUS e hiponatremia severa, con tomografía de cerebro normal. Además presentaba activación del complemento: C3 73 mg/dl (normal > 90 mg/dL) y C5b-9 778,9 ng/ml (normal 135,8-385,3 ng/ml). Se administró solución fisiológica y anticonvulsivantes, se corrigió la natremia y comenzó diálisis peritoneal. Al tercer día, por progresión del compromiso neurológico, se administró eculizumab (300 mg) experimentando una notable recuperación neurológica a las 24 horas junto a aumento de plaquetas y descenso de láctico deshidrogenasa. El paciente fue dado de alta luego de 14 días en buen estado general. Posteriormente se confirmó aislamiento de STEC O157:H7 y normalización de C3. Conclusión: este caso demuestra que el descenso de C3 al ingreso se asoció con daño neurológico y sugiere que la administración de eculizumab podría ser una alternativa terapéutica favorable. Introdução: No comprometimento neurológico na síndrome hemolítico-urêmica produzida por Eschericha coli produtora de toxina Shiga (STEC-SHU) é a primeira causa de mortalidade. Nos últimos anos, foi demonstrado que a ativação da via alternativa do complemento também contribui para danos aos órgãos. Esse achado levou ao reconhecimento da diminuição de C3 como marcador de mau prognóstico, bem como à avaliação do uso de eculizumabe em caso de comprometimento neurológico grave. Objetivo: Relatar um paciente com STEC-SHU e hipocomplementemia com comprometimento neurológico tratado com eculizumabe. Caso clínico: Homem de 17 meses admitido com síndrome convulsiva e anúria 24 horas com história de diarreia hemorrágica com 48 horas de evolução. Na admissão, apresentou laboratório compatível com STEC-SUH e hiponatremia grave, com tomografia cerebral normal. Além disso, apresentava ativação do complemento: C3 73 mg/dl (normal >90 mg/dL) e C5b-9 778,9 ng/ml (normal 135,8-385,3 ng/ml). Foi administrada solução fisiológica e anticonvulsivante, corrigida a natremia e iniciada diálise peritoneal. No terceiro dia, devido à progressão do comprometimento neurológico, foi administrado eculizumabe (300 mg), apresentando notável recuperação neurológica em 24 horas, com aumento das plaquetas e diminuição da desidrogenase láctica. O paciente recebeu alta após 14 dias em bom estado geral. Posteriormente, o isolamento de STEC O157:H7 e a normalização de C3 foram confirmados. Conclusão: este caso mostra que a diminuição de C3 na admissão foi associada a danos neurológicos e sugere que a administração de eculizumabe pode ser uma alternativa terapêutica favorável. Universidad Nacional Córdoba. Facultad de Ciencias Médicas. Secretaria de Ciencia y Tecnología 2021-06-28 info:eu-repo/semantics/article info:eu-repo/semantics/publishedVersion application/pdf text/html https://revistas.unc.edu.ar/index.php/med/article/view/29934 10.31053/1853.0605.v78.n2.29934 Revista de la Facultad de Ciencias Médicas de Córdoba.; Vol. 78 No. 2 (2021); 188-192 Revista de la Facultad de Ciencias Médicas de Córdoba; Vol. 78 Núm. 2 (2021); 188-192 Revista da Faculdade de Ciências Médicas de Córdoba; v. 78 n. 2 (2021); 188-192 1853-0605 0014-6722 10.31053/1853.0605.v78.n2 spa https://revistas.unc.edu.ar/index.php/med/article/view/29934/33915 https://revistas.unc.edu.ar/index.php/med/article/view/29934/33523 Derechos de autor 2021 Universidad Nacional de Córdoba http://creativecommons.org/licenses/by-nc/4.0