Literature DB >> 16941717

Congenital pulmonary lymphangiectasis sequence: a rare, heterogeneous, and lethal etiology for prenatal pleural effusion.

R Douglas Wilson1, Bruce Pawel, Michael Bebbington, Mark P Johnson, Foong-Yen Lim, David Stamilio, Angela Silber, Elaine Zakii, Alan W Flake.   

Abstract

OBJECTIVE: Case report and literature review for congenital pulmonary lymphangiectasis (CPL) CASE REPORT: Male fetus with bilateral pleural effusion, thoracoamniotic shunt, preterm delivery, and prolonged neonatal course with neonatal death at 3 months. Autopsy-identified CPL. DISCUSSION: Review of pathology, clinical course, and genetics of CPL.
CONCLUSION: This postnatal diagnosis of CPL/Hennekam syndrome must be considered with prenatal counseling regarding a fetus with bilateral pleural effusions. This pathological entity is autosomal recessive and has a significant risk of lethality.

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Mesh:

Year:  2006        PMID: 16941717     DOI: 10.1002/pd.1555

Source DB:  PubMed          Journal:  Prenat Diagn        ISSN: 0197-3851            Impact factor:   3.050


  6 in total

Review 1.  Fetal pleural effusion and Down syndrome.

Authors:  Li Cao; Yan Du; Ling Wang
Journal:  Intractable Rare Dis Res       Date:  2017-08

2.  The fetal MR appearance of 'nutmeg lung': findings in 8 cases linked to pulmonary lymphangiectasia.

Authors:  Teresa Victoria; Savvas Andronikou
Journal:  Pediatr Radiol       Date:  2014-05-01

3.  The utility of exome sequencing for fetal pleural effusions.

Authors:  Angie C Jelin; Nara Sobreira; Elizabeth Wohler; Benjamin Solomon; Teresa Sparks; Katelynn G Sagaser; Katherine R Forster; Jena Miller; P Dane Witmer; Ada Hamosh; David Valle; Karin Blakemore
Journal:  Prenat Diagn       Date:  2020-02-17       Impact factor: 3.050

4.  Congenital Chylothorax of the Newborn: A Systematic Analysis of Published Cases between 1990 and 2018.

Authors:  Bernhard Resch; Gülsen Sever Yildiz; Friedrich Reiterer
Journal:  Respiration       Date:  2021-09-01       Impact factor: 3.580

5.  Lymphangiectasis of lower limb: A rare challenging case.

Authors:  Visweswar Bhattacharya; Biswajit Mishra; Partha Sarathi Barooah; Gaurab Ranjan Chaudhuri; Siddhartha Bhattacharya
Journal:  Indian J Plast Surg       Date:  2009-07

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

Authors:  Cristiane Rúbia Ferreira; Verônica Sibre; Regina Schultz; Ana Maria Andrello Gonçalves Pereira de Melo; Silvia Maria Ibidi; Jackeline Della Torre
Journal:  Autops Case Rep       Date:  2015-12-30
  6 in total

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