Massive fetal chylothorax successfully treated with postnatal talc pleurodesis: A case report and review of the literature

Despite the rapid advances in fetal medicine and pediatric surgery, congenital chylothoraces have an associated mortality of 22–65% and an increased morbidity resulting from pulmonary hypoplasia, severe infections secondary to immune globulin deficiencies, protein malnutrition, and coagulopathy. Whi...

Full description

Bibliographic Details
Main Authors: Maggie M. Hodges, Timothy M. Crombleholme, Mariana Meyers, Ann Kulungowski, Ahmed I. Marwan, Taizo Nakano, Nicholas Behrendt, Kenneth W. Liechty
Format: Article
Language:English
Published: Elsevier 2016-06-01
Series:Journal of Pediatric Surgery Case Reports
Subjects:
Online Access:http://www.sciencedirect.com/science/article/pii/S2213576615300671
_version_ 1818511406295678976
author Maggie M. Hodges
Timothy M. Crombleholme
Mariana Meyers
Ann Kulungowski
Ahmed I. Marwan
Taizo Nakano
Nicholas Behrendt
Kenneth W. Liechty
author_facet Maggie M. Hodges
Timothy M. Crombleholme
Mariana Meyers
Ann Kulungowski
Ahmed I. Marwan
Taizo Nakano
Nicholas Behrendt
Kenneth W. Liechty
author_sort Maggie M. Hodges
collection DOAJ
description Despite the rapid advances in fetal medicine and pediatric surgery, congenital chylothoraces have an associated mortality of 22–65% and an increased morbidity resulting from pulmonary hypoplasia, severe infections secondary to immune globulin deficiencies, protein malnutrition, and coagulopathy. While the mainstay of therapy is medical management, large volume chylothoraces often require surgical management. In both the prenatal and postnatal periods, the recommended management of congenital chylothoraces is still controversial. We present a case of a prenatally diagnosed large chylothorax associated with a cervical lymphatic malformation. In our patient, the chylothorax persisted despite optimal postnatal medical management with drainage by tube thoracostomy, TPN, and octreotide. Adjuvant therapies included sirolimus and sclerotherapy directed toward the treatment of the macrocystic lymphatic malformation. We report the first case of a persistent congenital chylothorax associated with a lymphatic malformation successfully treated with thoracoscopic talc pleurodesis and sclerotherapy.
first_indexed 2024-12-10T23:32:56Z
format Article
id doaj.art-a180994b386f45518f9be2b948529455
institution Directory Open Access Journal
issn 2213-5766
language English
last_indexed 2024-12-10T23:32:56Z
publishDate 2016-06-01
publisher Elsevier
record_format Article
series Journal of Pediatric Surgery Case Reports
spelling doaj.art-a180994b386f45518f9be2b9485294552022-12-22T01:29:18ZengElsevierJournal of Pediatric Surgery Case Reports2213-57662016-06-019C1410.1016/j.epsc.2016.03.014Massive fetal chylothorax successfully treated with postnatal talc pleurodesis: A case report and review of the literatureMaggie M. Hodges0Timothy M. Crombleholme1Mariana Meyers2Ann Kulungowski3Ahmed I. Marwan4Taizo Nakano5Nicholas Behrendt6Kenneth W. Liechty7Colorado Fetal Care Center, Colorado Institute for Fetal & Maternal Health, Children's Hospital Colorado, Aurora, CO, USAColorado Fetal Care Center, Colorado Institute for Fetal & Maternal Health, Children's Hospital Colorado, Aurora, CO, USAColorado Fetal Care Center, Colorado Institute for Fetal & Maternal Health, Children's Hospital Colorado, Aurora, CO, USADivision of Pediatric Surgery, Children's Hospital Colorado, Aurora, CO, USAColorado Fetal Care Center, Colorado Institute for Fetal & Maternal Health, Children's Hospital Colorado, Aurora, CO, USACenter for Cancer and Blood Disorders, Children's Hospital Colorado, Aurora, CO, USAColorado Fetal Care Center, Colorado Institute for Fetal & Maternal Health, Children's Hospital Colorado, Aurora, CO, USAColorado Fetal Care Center, Colorado Institute for Fetal & Maternal Health, Children's Hospital Colorado, Aurora, CO, USADespite the rapid advances in fetal medicine and pediatric surgery, congenital chylothoraces have an associated mortality of 22–65% and an increased morbidity resulting from pulmonary hypoplasia, severe infections secondary to immune globulin deficiencies, protein malnutrition, and coagulopathy. While the mainstay of therapy is medical management, large volume chylothoraces often require surgical management. In both the prenatal and postnatal periods, the recommended management of congenital chylothoraces is still controversial. We present a case of a prenatally diagnosed large chylothorax associated with a cervical lymphatic malformation. In our patient, the chylothorax persisted despite optimal postnatal medical management with drainage by tube thoracostomy, TPN, and octreotide. Adjuvant therapies included sirolimus and sclerotherapy directed toward the treatment of the macrocystic lymphatic malformation. We report the first case of a persistent congenital chylothorax associated with a lymphatic malformation successfully treated with thoracoscopic talc pleurodesis and sclerotherapy.http://www.sciencedirect.com/science/article/pii/S2213576615300671Fetal pleural effusionCongenital chylothoraxTalc pleurodesisSurgical management
spellingShingle Maggie M. Hodges
Timothy M. Crombleholme
Mariana Meyers
Ann Kulungowski
Ahmed I. Marwan
Taizo Nakano
Nicholas Behrendt
Kenneth W. Liechty
Massive fetal chylothorax successfully treated with postnatal talc pleurodesis: A case report and review of the literature
Journal of Pediatric Surgery Case Reports
Fetal pleural effusion
Congenital chylothorax
Talc pleurodesis
Surgical management
title Massive fetal chylothorax successfully treated with postnatal talc pleurodesis: A case report and review of the literature
title_full Massive fetal chylothorax successfully treated with postnatal talc pleurodesis: A case report and review of the literature
title_fullStr Massive fetal chylothorax successfully treated with postnatal talc pleurodesis: A case report and review of the literature
title_full_unstemmed Massive fetal chylothorax successfully treated with postnatal talc pleurodesis: A case report and review of the literature
title_short Massive fetal chylothorax successfully treated with postnatal talc pleurodesis: A case report and review of the literature
title_sort massive fetal chylothorax successfully treated with postnatal talc pleurodesis a case report and review of the literature
topic Fetal pleural effusion
Congenital chylothorax
Talc pleurodesis
Surgical management
url http://www.sciencedirect.com/science/article/pii/S2213576615300671
work_keys_str_mv AT maggiemhodges massivefetalchylothoraxsuccessfullytreatedwithpostnataltalcpleurodesisacasereportandreviewoftheliterature
AT timothymcrombleholme massivefetalchylothoraxsuccessfullytreatedwithpostnataltalcpleurodesisacasereportandreviewoftheliterature
AT marianameyers massivefetalchylothoraxsuccessfullytreatedwithpostnataltalcpleurodesisacasereportandreviewoftheliterature
AT annkulungowski massivefetalchylothoraxsuccessfullytreatedwithpostnataltalcpleurodesisacasereportandreviewoftheliterature
AT ahmedimarwan massivefetalchylothoraxsuccessfullytreatedwithpostnataltalcpleurodesisacasereportandreviewoftheliterature
AT taizonakano massivefetalchylothoraxsuccessfullytreatedwithpostnataltalcpleurodesisacasereportandreviewoftheliterature
AT nicholasbehrendt massivefetalchylothoraxsuccessfullytreatedwithpostnataltalcpleurodesisacasereportandreviewoftheliterature
AT kennethwliechty massivefetalchylothoraxsuccessfullytreatedwithpostnataltalcpleurodesisacasereportandreviewoftheliterature