Literature DB >> 7566313

Hereditary osteo-onycho-renal dysplasia with excess urinary pyridinoline cross-links and abnormal kidney collagen cross-linking.

B Lubec1, K Arbeiter, W Ulrich, G Frauscher.   

Abstract

HOOD syndrome is a rare genetic disorder also known as nail patella syndrome. Biochemical and molecular biological data are rare and not conclusive. Preliminary data suggest the involvement of collagen type IV. This would, however, not be enough to explain nail or bone involvement. As kidney pathology would be consistent with a cross-linking disorder, we tested collagen IV and kidney collagen cross-linking on SDS-PAGE. This method showed a remarkable reduction in high-molecular-weight collagen polymers. The patient's and his mother's urinary pyridinoline cross-link excretion was manifold increased. Disturbed cross-linking of connective tissue proteins would help to explain the multiorgan involvement.

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Year:  1995        PMID: 7566313     DOI: 10.1159/000188593

Source DB:  PubMed          Journal:  Nephron        ISSN: 1660-8151            Impact factor:   2.847


  2 in total

1.  The LIM-homeodomain transcription factor Lmx1b plays a crucial role in podocytes.

Authors:  Claudia Rohr; Jürgen Prestel; Laurence Heidet; Hiltraud Hosser; Wilhelm Kriz; Randy L Johnson; Corinne Antignac; Ralph Witzgall
Journal:  J Clin Invest       Date:  2002-04       Impact factor: 14.808

2.  A synonymous genetic alteration of LMX1B in a family with nail-patella syndrome.

Authors:  Joo Ho Ham; Seok Joon Shin; Kyu Re Joo; Sung Min Park; Hye Young Sung; Joong Seok Kim; Jin Soo Choi; Yeong Jin Choi; Ho Cheol Song; Eui Jin Choi
Journal:  Korean J Intern Med       Date:  2009-08-26       Impact factor: 3.165

  2 in total

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