Literature DB >> 17455094

Electron microscopy of chorionic villus samples for prenatal diagnosis of lysosomal storage disorders.

D J Fowler1, G Anderson, A Vellodi, M Malone, N J Sebire.   

Abstract

Some lysosomal storage disorders cause progressive prenatal accumulation of undegradable metabolites that manifest as membrane-bound vacuoles in endothelial cells, fibroblasts, and trophoblast, identifiable by electron microscopic examination of chorionic villus samples (CVS). There were 111 CVS, which had ultrastructural examination for suspected storage disorders at Great Ormond Street Hospital (1988-2005). There were 31 positive diagnoses, including glycogen storage disease type II, gangliosidosis type 1, mucopolysaccharidosis type 1, MPS not specified, Niemann-Pick type A, sialidosis/mucolipidosis type 1, neuronal ceroid lipofuscinoses (including variant forms), Wolman disease, sialic acid storage disease, and storage disease not specified. In most of these cases the indication was a previously affected individual. Seventy-seven cases showed no evidence of storage disease; 3 samples were inadequate for ultrastructural diagnosis. In selected cases, one-third of CVS may demonstrate distinctive ultrastructural features allowing prenatal diagnosis of a range of storage diseases.

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Year:  2007        PMID: 17455094     DOI: 10.1080/01913120601169469

Source DB:  PubMed          Journal:  Ultrastruct Pathol        ISSN: 0191-3123            Impact factor:   1.094


  3 in total

1.  Antenatal diagnosis of pompe disease by fetal echocardiography: impact on outcome after early initiation of enzyme replacement therapy.

Authors:  Mohamed A Hamdan; Bushra A El-Zoabi; Muzibunnisa A Begam; Hisham M Mirghani; Mohamed H Almalik
Journal:  J Inherit Metab Dis       Date:  2010-09-04       Impact factor: 4.982

2.  Early administration of enzyme replacement therapy for Pompe disease: short-term follow-up results.

Authors:  M A Hamdan; M H Almalik; H M Mirghani
Journal:  J Inherit Metab Dis       Date:  2008-12-12       Impact factor: 4.982

3.  Challenges in Diagnosing Rare Genetic Causes of Common In Utero Presentations: Report of Two Patients with Mucolipidosis Type II (I-Cell Disease).

Authors:  Gregory Costain; Michal Inbar-Feigenberg; Maha Saleh; Shimrit Yaniv-Salem; Greg Ryan; Eric Morgen; Elaine S Goh; Gen Nishimura; David Chitayat
Journal:  J Pediatr Genet       Date:  2018-03-09
  3 in total

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