Literature DB >> 17596732

Right-sided diaphragmatic eventration: a rare cause of non-immune hydrops fetalis.

A Zankl1, M-C Osterheld, Y Vial, N Beurret, R Meuli, K Meagher-Villemure, M Roth-Kleiner.   

Abstract

We report 2 cases of non-immune hydrops fetalis (NIHF) in which autopsy findings revealed an association with right-sided congenital diaphragmatic eventration (CDE). Both patients born at 30 weeks of gestation presented with severe generalized skin oedema, pleural effusions and ascites. They both died shortly after birth of cardiorespiratory insufficiency due to lung hypoplasia and low output heart failure. The right thoracic cavity was filled with the membranous but intact right diaphragm covering the herniated visceral organs including parts of the liver, small bowel and colon surrounded by voluminous ascites. In similar fashion to the situation seen in congenital diaphragmatic hernia (CDH), the displaced visceral organs led to impaired lung growth resulting in important lung hypoplasia and obstructed venous return. Extravascular liquid accumulation was probably further aggravated by hypoproteinaemia secondary to liver dysfunction resulting from the venous congestion and cardiac failure. In summary, CDE is a rare condition resulting from impaired ingrowth of muscle fibres into the diaphragm during the first trimester. Prenatal differentiation of CDE from CDH is a real challenge. The association of CDE and NIHF has not been previously described.

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Year:  2007        PMID: 17596732     DOI: 10.1159/000098412

Source DB:  PubMed          Journal:  Neonatology        ISSN: 1661-7800            Impact factor:   4.035


  3 in total

Review 1.  Imaging findings in fetal diaphragmatic abnormalities.

Authors:  Leonor Alamo; François Gudinchet; Reto Meuli
Journal:  Pediatr Radiol       Date:  2015-08-09

2.  Right sided congenital diaphragmatic hernia: A rare neonatal emergency.

Authors:  Leena Harshad Parate; Chamanahalli Rajappa Geetha; Saurabh Vig
Journal:  Saudi J Anaesth       Date:  2015 Apr-Jun

3.  Interstitial deletion of chromosome 1 (1p21.1p12) in an infant with congenital diaphragmatic hernia, hydrops fetalis, and interrupted aortic arch.

Authors:  Masitah Ibrahim; Matthew Hunter; Lucy Gugasyan; Yuen Chan; Atul Malhotra; Arvind Sehgal; Kenneth Tan
Journal:  Clin Case Rep       Date:  2017-01-23
  3 in total

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