Document Detail


Fetal hydrops and familial pulmonary lymphatic hypoplasia.
MedLine Citation:
PMID:  12357424     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Two siblings were born with pleural effusions and hydrops. The first infant was a 26-week-old gestation male and died at 8 hours of life with radiographically small lungs and pulmonary insufficiency. No lung tissue was obtainable. This pregnancy was followed by two normal term infants, a male and female. The fourth pregnancy resulted in a female born at 35 weeks' gestation with pleural effusion and hydrops who died at 32 months of age. This infant was discharged from hospital at 32 days of age with small pleural effusions, but needed supplemental oxygen and daily diuretics to control edema. There were 14 additional admissions until death-all for respiratory distress or infections. An open lung biopsy at 2 months, showed dilated pleural lymphatics, with hypoplasia of the acinar and terminal bronchiolar lymphatics. At postmortem examination there was a markedly thickened pleura, and slit-like hypoplastic lymphatics of the acinar and terminal bronchioles and interlobular septal regions. This is the second family reported with these distinctive pulmonary intra-acinar and peri-acinar hypoplastic lymphatics. This disease is compatible with an autosomal recessive mode of inheritance.
Authors:
Donald W Thibeault; Philip Black; Eugenio Taboada
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  American journal of perinatology     Volume:  19     ISSN:  0735-1631     ISO Abbreviation:  Am J Perinatol     Publication Date:  2002 Aug 
Date Detail:
Created Date:  2002-10-01     Completed Date:  2002-12-13     Revised Date:  2004-11-17    
Medline Journal Info:
Nlm Unique ID:  8405212     Medline TA:  Am J Perinatol     Country:  United States    
Other Details:
Languages:  eng     Pagination:  323-31     Citation Subset:  IM    
Affiliation:
Department of Pediatrics, Children's Mercy Hospital, Kansas City, Missouri 64108, USA.
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MeSH Terms
Descriptor/Qualifier:
Adult
Autopsy
Bronchopulmonary Dysplasia / complications*,  genetics,  pathology
Fatal Outcome
Female
Humans
Hydrops Fetalis / complications*,  genetics,  pathology
Infant, Newborn
Lymphatic Diseases / complications*,  genetics
Lymphatic System / abnormalities*
Male
Pleural Effusion / etiology
Pregnancy
Pulmonary Edema / etiology
Respiratory Distress Syndrome, Newborn / etiology

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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