Literature DB >> 7458068

Lung collagen in type IV Ehlers-Danlos syndrome: ultrastructural and biochemical studies.

J G Clark, C Kuhn, J Uitto.   

Abstract

A patient with type IV Ehlers-Danlos syndrome developed recurrent pneumothoraces. At operation for poudrage of the pleura, an apical bulla was resected. Biochemical analysis of the lung tissue revealed increased solubilization of collagen by acetic acid extraction and by pepsin proteolysis. The relative proportion of type III collagen in the pepsin-solubilized collagen fraction was markedly decreased. Fibroblasts cultured from the patient's lung produced less type III procollagen relative to type I procollagen than control lung fibroblasts. Electron microscopic examination of the lung tissue showed dilated endoplasmic reticulum of the fibroblasts, and collagen fibers were normal. These observations suggested that the structural abnormalities in the lung that led to pneumothorax in this patient were due to deficiency of type III collagen.

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Year:  1980        PMID: 7458068     DOI: 10.1164/arrd.1980.122.6.971

Source DB:  PubMed          Journal:  Am Rev Respir Dis        ISSN: 0003-0805


  8 in total

Review 1.  The Genetics of Pneumothorax.

Authors:  Philip M Boone; Rachel M Scott; Stefan J Marciniak; Elizabeth P Henske; Benjamin A Raby
Journal:  Am J Respir Crit Care Med       Date:  2019-06-01       Impact factor: 21.405

2.  Type II achondrogenesis-hypochondrogenesis: identification of abnormal type II collagen.

Authors:  M Godfrey; D W Hollister
Journal:  Am J Hum Genet       Date:  1988-12       Impact factor: 11.025

3.  Spontaneous hemo-pneumothorax in a patient with Ehlers-Danlos syndrome.

Authors:  Rayees Ahmad Dar; Sabiya Hamid Wani; Majid Mushtaque; Reyaz Ahmad Kasana
Journal:  Gen Thorac Cardiovasc Surg       Date:  2012-05-19

4.  Cavitary pulmonary lesions in type IV Ehlers-Danlos syndrome.

Authors:  T E Herman; W H McAlister
Journal:  Pediatr Radiol       Date:  1994

5.  High prevalence of eosinophilic esophagitis in patients with inherited connective tissue disorders.

Authors:  J Pablo Abonia; Ting Wen; Emily M Stucke; Tommie Grotjan; Molly S Griffith; Katherine A Kemme; Margaret H Collins; Philip E Putnam; James P Franciosi; Karl F von Tiehl; Brad T Tinkle; Keith A Marsolo; Lisa J Martin; Stephanie M Ware; Marc E Rothenberg
Journal:  J Allergy Clin Immunol       Date:  2013-04-19       Impact factor: 10.793

6.  Ehlers-Danlos syndrome type IV: phenotypic consequences of a splicing mutation in one COL3A1 allele.

Authors:  D O Sillence; A A Chiodo; P E Campbell; W G Cole
Journal:  J Med Genet       Date:  1991-12       Impact factor: 6.318

7.  Abnormalities of the lungs and thoracic cage in the Ehlers-Danlos syndrome.

Authors:  J G Ayres; F M Pope; J F Reidy; T J Clark
Journal:  Thorax       Date:  1985-04       Impact factor: 9.139

Review 8.  A review of respiratory manifestations and their management in Ehlers-Danlos syndromes and hypermobility spectrum disorders.

Authors:  Karan Chohan; Nimish Mittal; Laura McGillis; Laura Lopez-Hernandez; Encarna Camacho; Maxim Rachinsky; Daniel Santa Mina; W Darlene Reid; Clodagh Mai Ryan; Kateri Agnes Champagne; Ani Orchanian-Cheff; Hance Clarke; Dmitry Rozenberg
Journal:  Chron Respir Dis       Date:  2021 Jan-Dec       Impact factor: 2.444

  8 in total

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