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Eculizumab in a pregnant patient with laboratory onset of catastrophic antiphospholipid syndrome: A case report
RATIONALE: Hypercoagulability and pregnancy morbidity are hallmarks of the antiphospholipid syndrome (APS). Catastrophic antiphospholipid syndrome (CAPS) is a potentially life-threatening omplication of APS, with widespread acute thrombotic microangiopathy (TMA) that can be precipitated by pregnancy...
Autores principales: | , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
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Wolters Kluwer Health
2018
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6200483/ https://www.ncbi.nlm.nih.gov/pubmed/30290624 http://dx.doi.org/10.1097/MD.0000000000012584 |
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author | Rovere-Querini, Patrizia Canti, Valentina Erra, Roberta Bianchi, Esperia Slaviero, Giorgio D’Angelo, Armando Rosa, Susanna Candiani, Massimo Castiglioni, Maria Teresa |
author_facet | Rovere-Querini, Patrizia Canti, Valentina Erra, Roberta Bianchi, Esperia Slaviero, Giorgio D’Angelo, Armando Rosa, Susanna Candiani, Massimo Castiglioni, Maria Teresa |
author_sort | Rovere-Querini, Patrizia |
collection | PubMed |
description | RATIONALE: Hypercoagulability and pregnancy morbidity are hallmarks of the antiphospholipid syndrome (APS). Catastrophic antiphospholipid syndrome (CAPS) is a potentially life-threatening omplication of APS, with widespread acute thrombotic microangiopathy (TMA) that can be precipitated by pregnancy and delivery and result in multiorgan damage. Unrestrained activation of the complement cascade is involved, favoring endothelial activation, tissue factor expression by leukocytes, and platelet aggregation. The complement block, which interrupts this amplification cycle, could prevent CAPS in patients with early TMA who face precipitating events. PATIENT CONCERNS: We present a nulliparous pregnant woman with APS at the 30(+6) week of gestation who has developed thrombocytopenia, intravascular hemolysis, elevated creatinine, proteinuria, and hematuria. DIAGNOSES: These featurs were compatible with the diagnosis of CAPS. Consensually, serum C3 protein levels were rapidly decreasing, reflecting complement consumption. INTERVENTIONS: She was treated with eculizumab, a humanized monoclonal antibody against C5 that prevents the formation of the complement membrane attack complex. OUTCOMES: Laboratory parameters improved and the patient did not develop thrombosis or detectable organ/tissue damage. The patient safely delivered by cesarean section at week 32 of gestation a healthy 1640 g male infant. After 5 days, she received additional eculizumab, with complete resolution of the clinical condition. Low complement activity was detectable in the infant blood for a week after delivery. No infectious complication occurred. LESSONS: Inhibition of the terminal complement activation is safe and might be effective in patients with APS developing early TMA, enabling safe delivery and preventing thrombotic events both in the mother and in the newborn. |
format | Online Article Text |
id | pubmed-6200483 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2018 |
publisher | Wolters Kluwer Health |
record_format | MEDLINE/PubMed |
spelling | pubmed-62004832018-11-07 Eculizumab in a pregnant patient with laboratory onset of catastrophic antiphospholipid syndrome: A case report Rovere-Querini, Patrizia Canti, Valentina Erra, Roberta Bianchi, Esperia Slaviero, Giorgio D’Angelo, Armando Rosa, Susanna Candiani, Massimo Castiglioni, Maria Teresa Medicine (Baltimore) Research Article RATIONALE: Hypercoagulability and pregnancy morbidity are hallmarks of the antiphospholipid syndrome (APS). Catastrophic antiphospholipid syndrome (CAPS) is a potentially life-threatening omplication of APS, with widespread acute thrombotic microangiopathy (TMA) that can be precipitated by pregnancy and delivery and result in multiorgan damage. Unrestrained activation of the complement cascade is involved, favoring endothelial activation, tissue factor expression by leukocytes, and platelet aggregation. The complement block, which interrupts this amplification cycle, could prevent CAPS in patients with early TMA who face precipitating events. PATIENT CONCERNS: We present a nulliparous pregnant woman with APS at the 30(+6) week of gestation who has developed thrombocytopenia, intravascular hemolysis, elevated creatinine, proteinuria, and hematuria. DIAGNOSES: These featurs were compatible with the diagnosis of CAPS. Consensually, serum C3 protein levels were rapidly decreasing, reflecting complement consumption. INTERVENTIONS: She was treated with eculizumab, a humanized monoclonal antibody against C5 that prevents the formation of the complement membrane attack complex. OUTCOMES: Laboratory parameters improved and the patient did not develop thrombosis or detectable organ/tissue damage. The patient safely delivered by cesarean section at week 32 of gestation a healthy 1640 g male infant. After 5 days, she received additional eculizumab, with complete resolution of the clinical condition. Low complement activity was detectable in the infant blood for a week after delivery. No infectious complication occurred. LESSONS: Inhibition of the terminal complement activation is safe and might be effective in patients with APS developing early TMA, enabling safe delivery and preventing thrombotic events both in the mother and in the newborn. Wolters Kluwer Health 2018-10-05 /pmc/articles/PMC6200483/ /pubmed/30290624 http://dx.doi.org/10.1097/MD.0000000000012584 Text en Copyright © 2018 the Author(s). Published by Wolters Kluwer Health, Inc. http://creativecommons.org/licenses/by-nc/4.0 This is an open access article distributed under the terms of the Creative Commons Attribution-Non Commercial License 4.0 (CCBY-NC), where it is permissible to download, share, remix, transform, and buildup the work provided it is properly cited. The work cannot be used commercially without permission from the journal. http://creativecommons.org/licenses/by-nc/4.0 |
spellingShingle | Research Article Rovere-Querini, Patrizia Canti, Valentina Erra, Roberta Bianchi, Esperia Slaviero, Giorgio D’Angelo, Armando Rosa, Susanna Candiani, Massimo Castiglioni, Maria Teresa Eculizumab in a pregnant patient with laboratory onset of catastrophic antiphospholipid syndrome: A case report |
title | Eculizumab in a pregnant patient with laboratory onset of catastrophic antiphospholipid syndrome: A case report |
title_full | Eculizumab in a pregnant patient with laboratory onset of catastrophic antiphospholipid syndrome: A case report |
title_fullStr | Eculizumab in a pregnant patient with laboratory onset of catastrophic antiphospholipid syndrome: A case report |
title_full_unstemmed | Eculizumab in a pregnant patient with laboratory onset of catastrophic antiphospholipid syndrome: A case report |
title_short | Eculizumab in a pregnant patient with laboratory onset of catastrophic antiphospholipid syndrome: A case report |
title_sort | eculizumab in a pregnant patient with laboratory onset of catastrophic antiphospholipid syndrome: a case report |
topic | Research Article |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6200483/ https://www.ncbi.nlm.nih.gov/pubmed/30290624 http://dx.doi.org/10.1097/MD.0000000000012584 |
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