Cargando…
Case Report: Coexistence of generalized arterial calcification of infancy (GACI) and maternal infections with cytomegalovirus and Toxoplasma gondii-unexpected fatal complication in a newborn
Generalized arterial calcification of infancy (GACI) is an extremely rare autosomal recessive condition characterized by the storage of calcium at the level of internal elastic membrane of arteries. The main consequences are intimal fibrous thickening and arterial occlusion. We present the case of a...
Autores principales: | , , , , , |
---|---|
Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Frontiers Media S.A.
2022
|
Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9434010/ https://www.ncbi.nlm.nih.gov/pubmed/36061401 http://dx.doi.org/10.3389/fped.2022.922379 |
_version_ | 1784780766955175936 |
---|---|
author | Gurzu, Simona Burlacu, Diana Sánta, Réka Jung, Ioan Slevin, Mark Fulop, Emöke |
author_facet | Gurzu, Simona Burlacu, Diana Sánta, Réka Jung, Ioan Slevin, Mark Fulop, Emöke |
author_sort | Gurzu, Simona |
collection | PubMed |
description | Generalized arterial calcification of infancy (GACI) is an extremely rare autosomal recessive condition characterized by the storage of calcium at the level of internal elastic membrane of arteries. The main consequences are intimal fibrous thickening and arterial occlusion. We present the case of a preterm male infant, born from an improperly dispensed pregnancy. At birth, the newborn presented generalized edema and hypotonia, and abolished heart sounds, without response to stimulation. Despite the mechanical ventilation, the infant died 2 h after birth. The death was clinically presumed to be related to the maternal infection with cytomegalovirus (CMV) and Toxoplasma gondii. The infant's mother affirmed the history of 6 previous miscarriages and a non-consanguineous marriage. At autopsy, microscopic examination showed generalized vasculitis secondary to minimal calcification of the large and medium-sized vessels of the lungs, liver, and tongue. These findings supported the diagnosis of GACI. Hydrothorax, non-infective ascites, and necrosis of the brain parenchyma were also associated. The premature infant died due to tonsillar herniation associated with decreased vessel compliance and refractory pulmonary hypertension thus leading to congestive cardiac failure. CMV was not detected on histopathological assessment nor were signs of any other infections. To the best of our knowledge, this is the first case of GACI occurring in a baby from a mother co-infected with CMV and T. gondii. |
format | Online Article Text |
id | pubmed-9434010 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2022 |
publisher | Frontiers Media S.A. |
record_format | MEDLINE/PubMed |
spelling | pubmed-94340102022-09-02 Case Report: Coexistence of generalized arterial calcification of infancy (GACI) and maternal infections with cytomegalovirus and Toxoplasma gondii-unexpected fatal complication in a newborn Gurzu, Simona Burlacu, Diana Sánta, Réka Jung, Ioan Slevin, Mark Fulop, Emöke Front Pediatr Pediatrics Generalized arterial calcification of infancy (GACI) is an extremely rare autosomal recessive condition characterized by the storage of calcium at the level of internal elastic membrane of arteries. The main consequences are intimal fibrous thickening and arterial occlusion. We present the case of a preterm male infant, born from an improperly dispensed pregnancy. At birth, the newborn presented generalized edema and hypotonia, and abolished heart sounds, without response to stimulation. Despite the mechanical ventilation, the infant died 2 h after birth. The death was clinically presumed to be related to the maternal infection with cytomegalovirus (CMV) and Toxoplasma gondii. The infant's mother affirmed the history of 6 previous miscarriages and a non-consanguineous marriage. At autopsy, microscopic examination showed generalized vasculitis secondary to minimal calcification of the large and medium-sized vessels of the lungs, liver, and tongue. These findings supported the diagnosis of GACI. Hydrothorax, non-infective ascites, and necrosis of the brain parenchyma were also associated. The premature infant died due to tonsillar herniation associated with decreased vessel compliance and refractory pulmonary hypertension thus leading to congestive cardiac failure. CMV was not detected on histopathological assessment nor were signs of any other infections. To the best of our knowledge, this is the first case of GACI occurring in a baby from a mother co-infected with CMV and T. gondii. Frontiers Media S.A. 2022-08-18 /pmc/articles/PMC9434010/ /pubmed/36061401 http://dx.doi.org/10.3389/fped.2022.922379 Text en Copyright © 2022 Gurzu, Burlacu, Sánta, Jung, Slevin and Fulop. https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms. |
spellingShingle | Pediatrics Gurzu, Simona Burlacu, Diana Sánta, Réka Jung, Ioan Slevin, Mark Fulop, Emöke Case Report: Coexistence of generalized arterial calcification of infancy (GACI) and maternal infections with cytomegalovirus and Toxoplasma gondii-unexpected fatal complication in a newborn |
title | Case Report: Coexistence of generalized arterial calcification of infancy (GACI) and maternal infections with cytomegalovirus and Toxoplasma gondii-unexpected fatal complication in a newborn |
title_full | Case Report: Coexistence of generalized arterial calcification of infancy (GACI) and maternal infections with cytomegalovirus and Toxoplasma gondii-unexpected fatal complication in a newborn |
title_fullStr | Case Report: Coexistence of generalized arterial calcification of infancy (GACI) and maternal infections with cytomegalovirus and Toxoplasma gondii-unexpected fatal complication in a newborn |
title_full_unstemmed | Case Report: Coexistence of generalized arterial calcification of infancy (GACI) and maternal infections with cytomegalovirus and Toxoplasma gondii-unexpected fatal complication in a newborn |
title_short | Case Report: Coexistence of generalized arterial calcification of infancy (GACI) and maternal infections with cytomegalovirus and Toxoplasma gondii-unexpected fatal complication in a newborn |
title_sort | case report: coexistence of generalized arterial calcification of infancy (gaci) and maternal infections with cytomegalovirus and toxoplasma gondii-unexpected fatal complication in a newborn |
topic | Pediatrics |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9434010/ https://www.ncbi.nlm.nih.gov/pubmed/36061401 http://dx.doi.org/10.3389/fped.2022.922379 |
work_keys_str_mv | AT gurzusimona casereportcoexistenceofgeneralizedarterialcalcificationofinfancygaciandmaternalinfectionswithcytomegalovirusandtoxoplasmagondiiunexpectedfatalcomplicationinanewborn AT burlacudiana casereportcoexistenceofgeneralizedarterialcalcificationofinfancygaciandmaternalinfectionswithcytomegalovirusandtoxoplasmagondiiunexpectedfatalcomplicationinanewborn AT santareka casereportcoexistenceofgeneralizedarterialcalcificationofinfancygaciandmaternalinfectionswithcytomegalovirusandtoxoplasmagondiiunexpectedfatalcomplicationinanewborn AT jungioan casereportcoexistenceofgeneralizedarterialcalcificationofinfancygaciandmaternalinfectionswithcytomegalovirusandtoxoplasmagondiiunexpectedfatalcomplicationinanewborn AT slevinmark casereportcoexistenceofgeneralizedarterialcalcificationofinfancygaciandmaternalinfectionswithcytomegalovirusandtoxoplasmagondiiunexpectedfatalcomplicationinanewborn AT fulopemoke casereportcoexistenceofgeneralizedarterialcalcificationofinfancygaciandmaternalinfectionswithcytomegalovirusandtoxoplasmagondiiunexpectedfatalcomplicationinanewborn |