Chorionic villi ultrastructure in the prenatal diagnosis of glycogenosis type II

J Inherit Metab Dis. 2010 Dec:33 Suppl 3:S105-11. doi: 10.1007/s10545-009-9033-6. Epub 2010 Feb 16.

Abstract

Objective: To perform the ultrastructural examination of a chorionic villi biopsy as a predictor of foetal involvement in the infantile form of glycogenosis type II (Pompe disease).

Methods: Ultrastructural, biochemical and genetic analyses were performed on chorionic villi biopsies of three consecutive pregnancies in a woman with a previous child affected by Pompe disease.

Results: In the only affected foetus, glycogen storage was observed in fibrocytes and endothelial cells of a chorionic villi sample at 11 week's gestation. Severe multi-organ involvement was demonstrated in the tissues of the aborted foetus. No abnormal material was found in the chorionic samples of two subsequent pregnancies, and a healthy boy and girl were born at term and remain unaffected. Both exhibited a partial reduction in acid maltase and were carriers of the maternal mutation.

Conclusions: Ultrastructural findings correlated with biochemical and genetic results, providing a clear and early indicator of the definite diagnosis for future pregnancy management or an early therapeutic approach.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Cells, Cultured
  • Chorionic Villi / enzymology
  • Chorionic Villi / ultrastructure*
  • Chorionic Villi Sampling*
  • DNA Mutational Analysis
  • Endothelial Cells / enzymology
  • Fatal Outcome
  • Female
  • Fibroblasts / enzymology
  • Genetic Predisposition to Disease
  • Genetic Testing
  • Gestational Age
  • Glycogen / metabolism
  • Glycogen Storage Disease Type II / enzymology
  • Glycogen Storage Disease Type II / genetics
  • Glycogen Storage Disease Type II / pathology*
  • Heterozygote
  • Humans
  • Infant, Newborn
  • Male
  • Microscopy, Electron, Transmission*
  • Mutation
  • Pedigree
  • Phenotype
  • Predictive Value of Tests
  • Pregnancy
  • Prenatal Diagnosis / methods*
  • alpha-Glucosidases / deficiency
  • alpha-Glucosidases / genetics

Substances

  • Glycogen
  • GAA protein, human
  • alpha-Glucosidases

Associated data

  • OMIM/OMIM232300