Document Detail


Neonatal nemaline myopathy presenting with multiple joint contractures.
MedLine Citation:
PMID:  2414108     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
A sporadic case of the rare and most severe neonatal form of nemaline myopathy is reported. Intrauterine manifestation included reduced fetal movements and breech position with a normal amount of amniotic fluid. After delivery by Caesarian section at 34 weeks of gestation the infant boy, who was not asphyctic, failed to establish spontaneous breathing and required immediate intubation and ventilation. Marked muscular hypotonia and weakness persisted and reflexes remained absent. Hip dislocation, joint contractures, absent palmar creases, prominent lateral palatal ridges and cryptorchidism were interpreted as consequent to prenatal paralysis. The boy died after 5 h due to hyaline membrane disease and meconium aspiration. At autopsy the skeletal muscles were found to be hypoplastic. The muscle fibres contained numerous rods, a typical finding of nemaline myopathy. Nemaline myopathy should be considered in fetuses and newborns with multiple joint contractures, severe muscular weakness and respiratory insufficiency.
Authors:
H U Bucher; E Boltshauser; J Briner
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  European journal of pediatrics     Volume:  144     ISSN:  0340-6199     ISO Abbreviation:  Eur. J. Pediatr.     Publication Date:  1985 Sep 
Date Detail:
Created Date:  1985-12-18     Completed Date:  1985-12-18     Revised Date:  2004-11-17    
Medline Journal Info:
Nlm Unique ID:  7603873     Medline TA:  Eur J Pediatr     Country:  GERMANY, WEST    
Other Details:
Languages:  eng     Pagination:  288-90     Citation Subset:  IM    
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MeSH Terms
Descriptor/Qualifier:
Arthrogryposis / pathology*
Humans
Inclusion Bodies / ultrastructure
Infant, Newborn
Male
Microscopy, Electron
Muscles / pathology*
Muscular Diseases / congenital*,  pathology
Staining and Labeling

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


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