Literature DB >> 3422850

Familial Ehlers-Danlos syndrome type II: abnormal fibrillogenesis of dermal collagen.

R Rizzo1, M B Contri, G Micali, D Quaglino, L Pavone, I P Ronchetti.   

Abstract

We examined a father and son affected by Ehlers-Danlos syndrome type II. Both patients had micrognathia together with ligament and skin hyperlaxity. The son exhibited complete cleft palate. Ultrastructural studies revealed abnormal collagen fibrils in the dermis of both patients. In the child the most striking alterations consisted of lateral fusion of an enormous number of collagen fibrils giving rise to huge polymorphic collagen masses. In the father's dermis the great majority of collagen fibrils appeared normal; however, lateral fusion of fibrils together with local abnormal collagen aggregation were occasionally seen. In both patients the dermal elastic network was well developed and elastic fibers appeared normal.

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Year:  1987        PMID: 3422850     DOI: 10.1111/j.1525-1470.1987.tb00778.x

Source DB:  PubMed          Journal:  Pediatr Dermatol        ISSN: 0736-8046            Impact factor:   1.588


  2 in total

1.  The phenotypic spectrum of contiguous deletion of CYP21A2 and tenascin XB: quadricuspid aortic valve and other midline defects.

Authors:  Wuyan Chen; Mimi S Kim; Sujata Shanbhag; Andrew Arai; Carol VanRyzin; Nazli B McDonnell; Deborah P Merke
Journal:  Am J Med Genet A       Date:  2009-12       Impact factor: 2.802

2.  A cohort of 17 patients with kyphoscoliotic Ehlers-Danlos syndrome caused by biallelic mutations in FKBP14: expansion of the clinical and mutational spectrum and description of the natural history.

Authors:  Cecilia Giunta; Matthias Baumann; Christine Fauth; Uschi Lindert; Ebtesam M Abdalla; Angela F Brady; James Collins; Jahannaz Dastgir; Sandra Donkervoort; Neeti Ghali; Diana S Johnson; Ariana Kariminejad; Johannes Koch; Marius Kraenzlin; Nayana Lahiri; Bernarda Lozic; Adnan Y Manzur; Jenny E V Morton; Jacek Pilch; Rebecca C Pollitt; Gudrun Schreiber; Nora L Shannon; Glenda Sobey; Anthony Vandersteen; Fleur S van Dijk; Martina Witsch-Baumgartner; Johannes Zschocke; F Michael Pope; Carsten G Bönnemann; Marianne Rohrbach
Journal:  Genet Med       Date:  2017-06-15       Impact factor: 8.822

  2 in total

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