Literature DB >> 1621759

Nail-patella syndrome in a spontaneously aborted 18-week fetus: ultrastructural and immunofluorescent study of the kidneys.

R M Drut1, S Chandra, R Latorraca, E Gilbert-Barness.   

Abstract

Nail-patella syndrome (NPS), hereditary onycho-osteodysplasia, is an autosomal dominant disorder of nail dystrophy, patellar absence or hypoplasia, incomplete elbow extension, conical posterior iliac horns, and nephropathy. We studied the kidneys of an 18-week spontaneously aborted fetus of a mother with the NPS. Ultrastructural examination of the kidney showed thickening of the capillary walls of the glomeruli and mesangium. There was irregular thickening of basement membranes with subendothelial fibrillar electron-dense deposits. Immunofluorescence showed fibrinogen deposition in glomerular basement membranes. Fibrinogen deposition in utero may ultimately lead to glomerular fibrosis and intrabasement membrane collagen deposition as seen in the adult renal lesion of this syndrome. This is the first report of the NPS in which the renal abnormalities have been studied in a fetus. These findings provide support for possible prenatal diagnosis of NPS by intra-uterine kidney biopsy.

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Year:  1992        PMID: 1621759     DOI: 10.1002/ajmg.1320430408

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  3 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.  In vivo expression of putative LMX1B targets in nail-patella syndrome kidneys.

Authors:  Laurence Heidet; Ernie M H F Bongers; Mireille Sich; Shao-Yu Zhang; Chantal Loirat; Alain Meyrier; Michel Broyer; Gérard Landthaler; Bernadette Faller; Yoshikazu Sado; Nine V A M Knoers; Marie-Claire Gubler
Journal:  Am J Pathol       Date:  2003-07       Impact factor: 4.307

3.  Prenatal diagnosis of nail patella syndrome: A case report.

Authors:  L D Padmanabhan; D Yesodharan; S Nampoothiri
Journal:  Indian J Radiol Imaging       Date:  2017 Jul-Sep
  3 in total

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