Document Detail

Congenital hypothyroidism and nonimmune hydrops fetalis: associated?
MedLine Citation:
PMID:  9917489     Owner:  NLM     Status:  MEDLINE    
Hydrops fetalis (HF) consists of an abnormal accumulation of fluid in two or more fetal compartments, including ascites, pleural effusion, pericardial effusion, and skin edema. Almost all observed cases of HF are of the nonimmune type, the causes of which remain undetermined in 15% of patients. We report a newborn infant with nonimmune hydrops fetalis (NIHF) and congenital hypothyroidism. The infant's mother was healthy and there were no malformations of the placenta or umbilical cord. The infant did not show any structural abnormalities of his central nervous, cardiovascular, gastrointestinal, or urinary tract systems, and there was no evidence of anemia, infectious disease, or inborn error of metabolism. An immune-based process was unlikely, because the blood group of the mother and infant was A-positive and results of an indirect Coombs test in the mother and a direct Coombs test in the infant were negative. The patient's condition gradually improved with mechanical ventilation, repeated thoracocentesis, and total parenteral nutrition. By day 5 of age the skin edema, pericardial effusion, and ascites disappeared, but accumulation of significant amounts of chylous pleural fluid persisted. Because of lethargy, FT4 and thyroid-stimulating hormone levels were obtained and showed hypothyroidism. Thyroid hormone supplementation was then started, and within 4 days the infant became more vigorous and was weaned from mechanical ventilation. After 7 days, the chylothorax resolved completely as the serum thyroxine level normalized. No reaccumulation of pleural effusion was noticed. The infant started to gain weight and was discharged from the hospital at 35 days of age. A possible pathophysiologic association between congenital hypothyroidism and NIHF is discussed. NIHF may be caused by lymphatic congestion attributable to an impairment of lymphatic flow and a delayed return of lymph to the vascular compartment. There could be a possibility that because of thyroid hormone deficiency in this patient, there was reduced adrenergic stimulation of the lymphatic system. This could result in a sluggish flow of the lymph with engorgement of the lymphatic system, leakage of lymph into the pleura and the interstitial spaces, and the production of chylothorax with NIHF. Animal studies demonstrate a direct relationship between lymph flow rate or lung liquid clearance and adrenergic receptor activity in the lymphatic system. These observations support our hypothesis that deficient adrenergic activity in congenital hypothyroidism might lead to chylothorax with NIHF in the fetus. We speculate that thyroid hormone may play a role in the regulation of adrenergic receptors in the lymphatic system and lungs, thus modulating both the lymphatic flow rate and lung liquid clearance, and facilitating the resolution of chylothorax. Examination of thyroid functions should be included in the investigation of fetuses and neonates with NIHF of an obscure origin.
I Kessel; I R Makhoul; P Sujov
Related Documents :
10447019 - Hypothyroxinemia in premature infants: is thyroxine treatment necessary?
24613039 - 40-hz multiple auditory steady-state responses to narrow-band chirps in sedated and ana...
2894819 - Transient neonatal hyperthyroidism and maternal thyroid stimulating immunoglobulins.
8435899 - Congenital hypothyroidism: the riyadh military hospital experience.
7044179 - Paralysis of the right hemidiaphragm.
22319539 - How can the microbiologist help in diagnosing neonatal sepsis?
16202159 - Birth outcomes in colorado's undocumented immigrant population.
1313609 - Prevention of transfusion-associated cytomegalovirus infection in neonatal patients by ...
642439 - The electrocardiogram of macaca fascicularis.
Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Pediatrics     Volume:  103     ISSN:  1098-4275     ISO Abbreviation:  Pediatrics     Publication Date:  1999 Jan 
Date Detail:
Created Date:  1999-02-02     Completed Date:  1999-02-02     Revised Date:  2005-11-17    
Medline Journal Info:
Nlm Unique ID:  0376422     Medline TA:  Pediatrics     Country:  UNITED STATES    
Other Details:
Languages:  eng     Pagination:  E9     Citation Subset:  IM    
Department of Neonatology, Rambam Medical Center, Rappaport Faculty of Medicine Technion, Israel Institute of Technology, Haifa 31096, Israel.
Export Citation:
APA/MLA Format     Download EndNote     Download BibTex
MeSH Terms
Congenital Hypothyroidism*
Hydrops Fetalis / etiology*
Hypothyroidism / complications*
Infant, Newborn
Comment In:
Pediatrics. 1999 Dec;104(6):1416-7   [PMID:  10610498 ]

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

Previous Document:  Teaching pediatric procedures: the Vancouver model for instructing Seldinger's technique of central ...
Next Document:  Intractable diarrhea from cytomegalovirus enterocolitis in an immunocompetent infant.