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

Prenat Diagn. 2006 Nov;26(11):1058-61. doi: 10.1002/pd.1555.

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.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Adult
  • Chylothorax / congenital*
  • Diabetes Mellitus, Type 1 / complications
  • Fatal Outcome
  • Female
  • Fetal Diseases / therapy
  • Humans
  • Infant, Newborn
  • Infant, Premature
  • Infant, Premature, Diseases / pathology*
  • Lung Diseases / congenital*
  • Lung Diseases / pathology
  • Lymphangiectasis / congenital*
  • Lymphangiectasis / pathology
  • Male
  • Pleural Effusion / congenital*
  • Pleural Effusion / embryology
  • Pleural Effusion / therapy
  • Pregnancy
  • Pregnancy in Diabetics