Literature DB >> 9643297

New lethal disease involving type I and III collagen defect resembling geroderma osteodysplastica, De Barsy syndrome, and Ehlers-Danlos syndrome IV.

A Jukkola1, S Kauppila, L Risteli, K Vuopala, J Risteli, J Leisti, L Pajunen.   

Abstract

We describe the clinical findings and biochemical features of a male child suffering from a so far undescribed lethal connective tissue disorder characterised by extreme hypermobility of the joints, lax skin, cataracts, severe growth retardation, and insufficient production of type I and type III procollagens. His features are compared with Ehlers-Danlos type IV, De Barsy syndrome, and geroderma osteodysplastica, as these disorders show some symptoms and signs shared with our patient. The child died because of failure of the connective tissue structures joining the skull and the spine, leading to progressive spinal stenosis. The aortic valve was translucent and insufficient. The clinical symptoms and signs, together with histological findings, suggested a collagen defect. Studies on both skin fibroblast cultures and the patient's serum showed reduced synthesis of collagen types I and III at the protein and RNA levels. The sizes of the mRNAs and newly synthesised proteins were normal, excluding gross structural abnormalities. These findings are not in accordance with any other collagen defect characterised so far.

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Year:  1998        PMID: 9643297      PMCID: PMC1051350          DOI: 10.1136/jmg.35.6.513

Source DB:  PubMed          Journal:  J Med Genet        ISSN: 0022-2593            Impact factor:   6.318


  16 in total

Review 1.  The family of collagen genes.

Authors:  E Vuorio; B de Crombrugghe
Journal:  Annu Rev Biochem       Date:  1990       Impact factor: 23.643

Review 2.  Type III collagen mutations in Ehlers Danlos syndrome type IV and other related disorders.

Authors:  F M Pope; A C Nicholls; P Narcisi; A Temple; Y Chia; P Fryer; A De Paepe; W P De Groote; J R McEwan; D A Compston
Journal:  Clin Exp Dermatol       Date:  1988-09       Impact factor: 3.470

3.  Dwarfism, oligophrenia and degeneration of the elastic tissue in skin and cornea. A new syndrome?

Authors:  A M de Barsy; E Moens; L Dierckx
Journal:  Helv Paediatr Acta       Date:  1968-06

4.  [A sex-linked disorder: hereditary osteodysplastic geroderma (20 years of observation)].

Authors:  D Klein; F Bamatter; A Franceschetti; G Boreux; J E Brocher; P Holenstein
Journal:  Rev Otoneuroophtalmol       Date:  1968-11

5.  Incorporation of sulphate into type III procollagen by cultured human fibroblasts. Identification of tyrosine O-sulphate.

Authors:  A Jukkola; J Risteli; O Niemelä; L Risteli
Journal:  Eur J Biochem       Date:  1986-01-02

6.  Coordinated regulation of type I and type III collagen production and mRNA levels of pro alpha 1(I) and pro alpha 2(I) collagen in cultured morphea fibroblasts.

Authors:  T Vuorio; J K Mäkelä; V M Kähäri; E Vuorio
Journal:  Arch Dermatol Res       Date:  1987       Impact factor: 3.017

7.  Cloning of full-length elastin cDNAs from a human skin fibroblast recombinant cDNA library: further elucidation of alternative splicing utilizing exon-specific oligonucleotides.

Authors:  M J Fazio; D R Olsen; E A Kauh; C T Baldwin; Z Indik; N Ornstein-Goldstein; H Yeh; J Rosenbloom; J Uitto
Journal:  J Invest Dermatol       Date:  1988-11       Impact factor: 8.551

8.  Gerodermia osteodysplastica hereditaria: report of three affected brothers and literature review.

Authors:  R Lisker; A Hernández; M Martínez-Lavin; O Mutchinick; C Armas; P Reyes; J Robles-Gil
Journal:  Am J Med Genet       Date:  1979

9.  Radioimmunoassay of the carboxyterminal propeptide of human type I procollagen.

Authors:  J Melkko; S Niemi; L Risteli; J Risteli
Journal:  Clin Chem       Date:  1990-07       Impact factor: 8.327

10.  Geroderma osteodysplastica. A report of two affected families.

Authors:  A G Hunter; J T Martsolf; C G Baker; M H Reed
Journal:  Hum Genet       Date:  1978-02-16       Impact factor: 4.132

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  1 in total

1.  Recurrent De Novo Mutations Affecting Residue Arg138 of Pyrroline-5-Carboxylate Synthase Cause a Progeroid Form of Autosomal-Dominant Cutis Laxa.

Authors:  Björn Fischer-Zirnsak; Nathalie Escande-Beillard; Jaya Ganesh; Yu Xuan Tan; Mohammed Al Bughaili; Angela E Lin; Inderneel Sahai; Paulina Bahena; Sara L Reichert; Abigail Loh; Graham D Wright; Jaron Liu; Elisa Rahikkala; Eniko K Pivnick; Asim F Choudhri; Ulrike Krüger; Tomasz Zemojtel; Conny van Ravenswaaij-Arts; Roya Mostafavi; Irene Stolte-Dijkstra; Sofie Symoens; Leila Pajunen; Lihadh Al-Gazali; David Meierhofer; Peter N Robinson; Stefan Mundlos; Camilo E Villarroel; Peter Byers; Amira Masri; Stephen P Robertson; Ulrike Schwarze; Bert Callewaert; Bruno Reversade; Uwe Kornak
Journal:  Am J Hum Genet       Date:  2015-08-27       Impact factor: 11.025

  1 in total

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