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伴有尿吡啶啉交联物过多及肾脏胶原交联异常的遗传性骨-甲-肾发育不良。

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

作者信息

Lubec B, Arbeiter K, Ulrich W, Frauscher G

机构信息

Department of Pediatrics, University of Vienna, Austria.

出版信息

Nephron. 1995;70(2):255-9. doi: 10.1159/000188593.

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.

摘要

胡德综合征是一种罕见的遗传性疾病,也被称为指甲髌骨综合征。生化和分子生物学数据稀少且尚无定论。初步数据表明IV型胶原可能参与其中。然而,这并不足以解释指甲或骨骼受累的情况。由于肾脏病理与交联障碍相符,我们在十二烷基硫酸钠-聚丙烯酰胺凝胶电泳(SDS-PAGE)上检测了IV型胶原和肾脏胶原交联情况。该方法显示高分子量胶原聚合物显著减少。患者及其母亲尿吡啶啉交联物排泄量大幅增加。结缔组织蛋白交联紊乱有助于解释多器官受累的情况。

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