Document Detail


Combination of cor triatriatum sinistrum and hypoplastic left heart syndrome in Meckel-Gruber syndrome: a case report.
MedLine Citation:
PMID:  19358626     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Meckel-Gruber syndrome (MKS) is a fatal, autosomal recessive disorder characterized by malformation of central nervous system, particularly occipital encephalocele, bilateral renal dysplasia, and polydactyly. However, the clinical findings of this syndrome encompass various organ abnormalities as a result of genetic heterogeneity. The associated heart anomaly in MKS is inconstant. Its prevalence is rare and no striking or specific cardiac defects have been documented. We present a case of MKS with combined cor triatriatum sinistrum (left atrium divided into upper and lower compartment by a thin membrane) and hypoplastic left heart syndrome (underdeveloped mitral valve, left ventricle, and aorta) in a 33-week male fetus that was ultrasonographically detected and confirmed by autopsy. In addition to the cardiac defects, the patient was found to have postaxial polydactyly of 4 extremities, Dandy-Walker malformation, bilateral renal cystic dysplasia, and hepatic plate malformation. To the best of our knowledge, this is the first time that a combination of cor triatriatum sinistrum and hypoplastic left heart syndrome in MKS has been reported in the literature.
Authors:
Mana Taweevisit; Jitsupa Treetipsatit; Patou Tantbirojn; Paul Scott Thorner
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Pediatric and developmental pathology : the official journal of the Society for Pediatric Pathology and the Paediatric Pathology Society     Volume:  12     ISSN:  1093-5266     ISO Abbreviation:  Pediatr. Dev. Pathol.     Publication Date:    2009 Sep-Oct
Date Detail:
Created Date:  2009-11-03     Completed Date:  2009-12-23     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  9809673     Medline TA:  Pediatr Dev Pathol     Country:  United States    
Other Details:
Languages:  eng     Pagination:  404-9     Citation Subset:  IM    
Affiliation:
Department of Pathology, Faculty of Medicine, Chulalongkorn University, 1873 King Rama IV Street, Pathumwan, Bangkok, Thailand. dr.mana4@gmail.com
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MeSH Terms
Descriptor/Qualifier:
Abnormalities, Multiple / pathology*
Cor Triatriatum / complications*,  pathology*
Dandy-Walker Syndrome / complications,  pathology
Humans
Hypoplastic Left Heart Syndrome / complications*,  pathology*
Kidney Diseases, Cystic / complications,  congenital,  pathology
Male
Polydactyly / complications
Syndrome

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


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