Literature DB >> 29620587

Mucolipidosis Type II Affecting 1 Fetus and Placental Disk of a Dichorionic-Diamnionic Twin Gestation: A Case Report and Review of the Literature.

David B Chapel1, Bonnie Choy, Peter Pytel, Aliya N Husain, Ricardo R Lastra.   

Abstract

Mucolipidosis type II, also known as I-cell disease, is an autosomal recessive inborn error of metabolism, resulting from loss-of-function mutations in GNPTAB. Affected infants exhibit multiple physical anomalies and developmental delay, and death from disease follows in early childhood. Here we present an instructive case of mucolipidosis type II affecting 1 fetus and placental disk in a dichorionic-diamnionic twin pregnancy delivered at 36-wk gestation. The second twin and placental disk showed no abnormality. On microscopic examination, the affected placenta displayed marked vacuolization of the syncytiotrophoblast and Hofbauer cells, which was confirmed on ultrastructural examination. To our knowledge, this is the first description of placental findings in a twin pregnancy, wherein only 1 twin is affected by an inborn error of metabolism. This provides an opportunity to highlight the placental abnormalities seen in this group of diseases, and to emphasize the role of pathologic examination in early detection of otherwise unsuspected inborn errors of metabolism.

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Year:  2019        PMID: 29620587     DOI: 10.1097/PGP.0000000000000506

Source DB:  PubMed          Journal:  Int J Gynecol Pathol        ISSN: 0277-1691            Impact factor:   2.762


  2 in total

1.  Placental pathology in an unsuspected case of mucolipidosis type II with secondary hyperparathyroidism in a premature infant.

Authors:  Parith Wongkittichote; Garland Michael Upchurch; Louis P Dehner; Timothy Wood; Jorge L Granadillo
Journal:  Mol Genet Metab Rep       Date:  2021-03-25

2.  Case Report: Mucolipidosis II and III Alpha/Beta Caused by Pathogenic Variants in the GNPTAB Gene (Mucolipidosis).

Authors:  Shao-Jia Mao; Yu-Mei Zu; Yang-Li Dai; Chao-Chun Zou
Journal:  Front Pediatr       Date:  2022-04-08       Impact factor: 3.418

  2 in total

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