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Congenital generalized lymphangiectasia: a rare developmental disorder for non-immune fetal hydrops

Firstly described by Rudolf Virchow in the 19th century, congenital generalized lymphangiectasia is a rare entity characterized by dilation of lymphatic vessels, and was recently classified in primary or secondary lymphangiectasia. Generalized forms may be diagnosed during pre-natal follow-up with u...

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Autores principales: Ferreira, Cristiane Rúbia, Sibre, Verônica, Schultz, Regina, de Melo, Ana Maria Andrello Gonçalves Pereira, Ibidi, Silvia Maria, Torre, Jackeline Della
Formato: Online Artículo Texto
Lenguaje:English
Publicado: São Paulo, SP: Universidade de São Paulo, Hospital Universitário 2015
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4757917/
https://www.ncbi.nlm.nih.gov/pubmed/26894043
http://dx.doi.org/10.4322/acr.2015.027
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author Ferreira, Cristiane Rúbia
Sibre, Verônica
Schultz, Regina
de Melo, Ana Maria Andrello Gonçalves Pereira
Ibidi, Silvia Maria
Torre, Jackeline Della
author_facet Ferreira, Cristiane Rúbia
Sibre, Verônica
Schultz, Regina
de Melo, Ana Maria Andrello Gonçalves Pereira
Ibidi, Silvia Maria
Torre, Jackeline Della
author_sort Ferreira, Cristiane Rúbia
collection PubMed
description Firstly described by Rudolf Virchow in the 19th century, congenital generalized lymphangiectasia is a rare entity characterized by dilation of lymphatic vessels, and was recently classified in primary or secondary lymphangiectasia. Generalized forms may be diagnosed during pre-natal follow-up with ultrasound examination, and, depending on its severity, the newborn outcome is very poor. The authors report the case of a female newborn with a previous diagnosis of fetal hydropsy who was born after a full-term gestation with respiratory failure due to bilateral voluminous pleural effusion and ascites. Physical examination also disclosed syndromic facies. Despite all efforts of the intensive supportive care, the patient died after 24 days of life. The autopsy findings were consistent with the diagnosis of congenital pulmonary lymphangiectasia. The authors call attention to this rare diagnosis in patients with cavitary effusion and respiratory insufficiency at birth.
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spelling pubmed-47579172016-02-18 Congenital generalized lymphangiectasia: a rare developmental disorder for non-immune fetal hydrops Ferreira, Cristiane Rúbia Sibre, Verônica Schultz, Regina de Melo, Ana Maria Andrello Gonçalves Pereira Ibidi, Silvia Maria Torre, Jackeline Della Autops Case Rep Article / Autopsy Case Report Firstly described by Rudolf Virchow in the 19th century, congenital generalized lymphangiectasia is a rare entity characterized by dilation of lymphatic vessels, and was recently classified in primary or secondary lymphangiectasia. Generalized forms may be diagnosed during pre-natal follow-up with ultrasound examination, and, depending on its severity, the newborn outcome is very poor. The authors report the case of a female newborn with a previous diagnosis of fetal hydropsy who was born after a full-term gestation with respiratory failure due to bilateral voluminous pleural effusion and ascites. Physical examination also disclosed syndromic facies. Despite all efforts of the intensive supportive care, the patient died after 24 days of life. The autopsy findings were consistent with the diagnosis of congenital pulmonary lymphangiectasia. The authors call attention to this rare diagnosis in patients with cavitary effusion and respiratory insufficiency at birth. São Paulo, SP: Universidade de São Paulo, Hospital Universitário 2015-12-30 /pmc/articles/PMC4757917/ /pubmed/26894043 http://dx.doi.org/10.4322/acr.2015.027 Text en Autopsy and Case Reports. ISSN 2236-1960. Copyright © 2015. http://creativecommons.org/licenses/by-nc/3.0/ This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License which permits unrestricted non-commercial use, distribution, and reproduction in any medium provided article is properly cited.
spellingShingle Article / Autopsy Case Report
Ferreira, Cristiane Rúbia
Sibre, Verônica
Schultz, Regina
de Melo, Ana Maria Andrello Gonçalves Pereira
Ibidi, Silvia Maria
Torre, Jackeline Della
Congenital generalized lymphangiectasia: a rare developmental disorder for non-immune fetal hydrops
title Congenital generalized lymphangiectasia: a rare developmental disorder for non-immune fetal hydrops
title_full Congenital generalized lymphangiectasia: a rare developmental disorder for non-immune fetal hydrops
title_fullStr Congenital generalized lymphangiectasia: a rare developmental disorder for non-immune fetal hydrops
title_full_unstemmed Congenital generalized lymphangiectasia: a rare developmental disorder for non-immune fetal hydrops
title_short Congenital generalized lymphangiectasia: a rare developmental disorder for non-immune fetal hydrops
title_sort congenital generalized lymphangiectasia: a rare developmental disorder for non-immune fetal hydrops
topic Article / Autopsy Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4757917/
https://www.ncbi.nlm.nih.gov/pubmed/26894043
http://dx.doi.org/10.4322/acr.2015.027
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