Document Detail


Pulmonary involvement in a child with ligneous conjunctivitis and homozygous type I plasminogen deficiency.
MedLine Citation:
PMID:  11477736     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Ligneous conjunctivitis (LC) is a rare disorder characterized by a chronic course of recurrent membranous lesions at conjunctivae. Pseudomembranes of other mucous membranes have been reported in patients with LC, but to the best of our knowledge, no case with alveolar involvement has been described. Here, we report a 2.5-year-old girl with LC who had tracheoaveolar involvement and homozygous type I plasminogen deficiency. Tracheal involvement was diagnosed by bronchoscopic biopsy and alveolar involvement with postmortem biopsy. She was shown to be homozygous for frameshift mutation in plasminogen exon 14 (Gly565ins-G) with molecular genetic examination of DNA which was obtained from parafin embedded postmortem lung tissue. Ligneous inflammation of the upper and lower respiratory tract must be considered in children with LC and recurrent respiratory tract problems.
Authors:
U Ozçelik; Z Akçören; D Anadol; N Kiper; M Orhon; A Göçmen; M Irkeç; V Schuster
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Pediatric pulmonology     Volume:  32     ISSN:  8755-6863     ISO Abbreviation:  Pediatr. Pulmonol.     Publication Date:  2001 Aug 
Date Detail:
Created Date:  2001-07-30     Completed Date:  2001-09-20     Revised Date:  2006-03-28    
Medline Journal Info:
Nlm Unique ID:  8510590     Medline TA:  Pediatr Pulmonol     Country:  United States    
Other Details:
Languages:  eng     Pagination:  179-83     Citation Subset:  IM    
Copyright Information:
Copyright 2001 Wiley-Liss, Inc.
Affiliation:
Division of Chest Disease, Department of Pediatrics, Faculty of Medicine, Hacettepe University, Ankara, Turkey. uozcelik@gen.hum.edu.tr
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MeSH Terms
Descriptor/Qualifier:
Conjunctivitis / complications*,  genetics
DNA / genetics
Female
Humans
Infant
Inflammation
Lung Diseases / etiology*,  pathology
Mucous Membrane / pathology*
Plasminogen / deficiency*
Pulmonary Alveoli / pathology*
Trachea / pathology*
Chemical
Reg. No./Substance:
9001-91-6/Plasminogen; 9007-49-2/DNA

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


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