Literature DB >> 1946081

Diagnosis of unsuspected fetal metabolic storage disease by routine placental examination.

D J Roberts1, M G Ampola, J M Lage.   

Abstract

GM1 gangliosidosis (type 1) is a rare hereditary, autosomal recessive, lysosomal storage disease characterized by a marked deficiency of active acid beta-galactosidase resulting in accumulation of gangliosides and mucopolysaccharides in tissues. Disease status of newborns from affected kindreds may be diagnosed by placental examination. Typical findings include a characteristic vacuolar distension of the cytoplasm of syncytiotrophoblast and stromal Hofbauer cells. We report a case of unsuspected fetal storage disorder initially diagnosed by routine placental examination of a normal-appearing infant born to a previously unaffected family. Progressive, third-trimester oligohydramnios and fetal growth retardation had been documented by ultrasonography. Placental findings included vacuolization of syncytiotrophoblast, intermediate trophoblast, and stromal Hofbauer cells. Subsequent enzyme analysis confirmed the placental findings of storage disorder and diagnosed GM1 gangliosidosis.

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Year:  1991        PMID: 1946081     DOI: 10.3109/15513819109064796

Source DB:  PubMed          Journal:  Pediatr Pathol        ISSN: 0277-0938


  4 in total

Review 1.  Lysosomal storage disorders in the newborn.

Authors:  Orna Staretz-Chacham; Tess C Lang; Mary E LaMarca; Donna Krasnewich; Ellen Sidransky
Journal:  Pediatrics       Date:  2009-04       Impact factor: 7.124

Review 2.  Placental Hofbauer cells and complications of pregnancy.

Authors:  Zhonghua Tang; Vikki M Abrahams; Gil Mor; Seth Guller
Journal:  Ann N Y Acad Sci       Date:  2011-03       Impact factor: 5.691

3.  Retarded bone formation in GM1-gangliosidosis: a study of the infantile form and comparison with two canine models.

Authors:  J Alroy; K Knowles; S H Schelling; E M Kaye; A E Rosenberg
Journal:  Virchows Arch       Date:  1995       Impact factor: 4.064

4.  Protein modeling and clinical description of a novel in-frame GLB1 deletion causing GM1 gangliosidosis type II.

Authors:  John E Richter; Michael T Zimmermann; Patrick R Blackburn; Ahmed N Mohammad; Eric W Klee; Laura M Pollard; Colleen F Macmurdo; Paldeep S Atwal; Thomas R Caulfield
Journal:  Mol Genet Genomic Med       Date:  2018-09-05       Impact factor: 2.183

  4 in total

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