Literature DB >> 7671971

Effects of galactosemia in utero.

J B Holton1.   

Abstract

There is direct evidence that in galactosemia, due to galactose-1-phosphate uridyltransferase deficiency, galactose, galactose-1-phosphate and galactitol accumulate in the fetus by week 20 of gestation. The metabolic abnormality may develop earlier than this, however, since the key enzymes in galactose metabolism have been shown to be present in normal fetal liver from the 10th week of gestation. There has been a single report of increased galactitol in amniotic fluid obtained at 10 weeks gestation, the outcome being a baby affected with galactosemia. Cataract formation in the fetus is rare and the only direct evidence that galactosemia may have harmful effects in utero. However, it has been concluded that the liver pathology seen in some patients who died in the neonatal period originated prenatally, and some studies have found that galactosemia is associated with reduced birth weight. Reports of two patients with histologically normal ovaries very soon after birth have been cited as evidence against gonadal dysfunction arising during fetal life, but it should be noted that this is not a constant feature in female galactosemics. Other observations, particularly those made from animal models, would suggest that ovarian dysfunction is most likely to have been caused in utero.

Entities:  

Mesh:

Substances:

Year:  1995        PMID: 7671971     DOI: 10.1007/bf02143809

Source DB:  PubMed          Journal:  Eur J Pediatr        ISSN: 0340-6199            Impact factor:   3.183


  24 in total

1.  The value of galactose phosphate determinations in the treatment of galactosaemia.

Authors:  V SCHWARZ
Journal:  Arch Dis Child       Date:  1960-10       Impact factor: 3.791

2.  The enzymatic transformation of uridine diphosphate glucose into a galactose derivative.

Authors:  L F LELOIR
Journal:  Arch Biochem Biophys       Date:  1951-09       Impact factor: 4.013

3.  Cerebral edema and galactosemia.

Authors:  R J Welch; D W Milligan
Journal:  Pediatrics       Date:  1987-10       Impact factor: 7.124

4.  Dietary restriction of maternal lactose intake does not prevent accumulation of galactitol in the amniotic fluid of fetuses affected with galactosaemia.

Authors:  C Jakobs; W J Kleijer; H D Bakker; A H van Gennip; H Przyrembel; M F Niermeijer
Journal:  Prenat Diagn       Date:  1988-11       Impact factor: 3.050

5.  Human hepatic uridine diphosphate galactose pyrophosphorylase. Its characterization and activity during development.

Authors:  H D Abraham; R R Howell
Journal:  J Biol Chem       Date:  1969-02-25       Impact factor: 5.157

6.  Assay of UDP-galactose 4-epimerase.

Authors:  Y S Shin; A von Rucker; M Rieth; W Endres
Journal:  Clin Chem       Date:  1982-11       Impact factor: 8.327

7.  Accumulation of galactose-1-phosphate in the galactosemic fetus despite maternal milk avoidance.

Authors:  M Irons; H L Levy; S Pueschel; K Castree
Journal:  J Pediatr       Date:  1985-08       Impact factor: 4.406

8.  Galactitol in the tissues of a galactosemic child.

Authors:  R Quan-Ma; H J Wells; W W Wells; F E Sherman; T J Egan
Journal:  Am J Dis Child       Date:  1966-11

9.  The activity of galactose-1-phosphate uridyltransferase and galactokinase in human fetal organs.

Authors:  Y S Shin-Buehring; T Beier; A Tan; M Osang; J Schaub
Journal:  Pediatr Res       Date:  1977-10       Impact factor: 3.756

Review 10.  Galactose-1-phosphate in the pathophysiology of galactosemia.

Authors:  R Gitzelmann
Journal:  Eur J Pediatr       Date:  1995       Impact factor: 3.183

View more
  9 in total

Review 1.  Galactosaemia: pathogenesis and treatment.

Authors:  J B Holton
Journal:  J Inherit Metab Dis       Date:  1996       Impact factor: 4.982

Review 2.  Classical galactosaemia revisited.

Authors:  Annet M Bosch
Journal:  J Inherit Metab Dis       Date:  2006-07-11       Impact factor: 4.982

3.  Correlates of language impairment in children with galactosaemia.

Authors:  N L Potter; J-A C Lazarus; J M Johnson; R D Steiner; L D Shriberg
Journal:  J Inherit Metab Dis       Date:  2008-07-12       Impact factor: 4.982

Review 4.  In utero galactose intoxication in animals.

Authors:  S Segal
Journal:  Eur J Pediatr       Date:  1995       Impact factor: 3.183

Review 5.  Galactosemia unsolved.

Authors:  S Segal
Journal:  Eur J Pediatr       Date:  1995       Impact factor: 3.183

6.  Galactitol in galactosemia.

Authors:  C Jakobs; S Schweitzer; B Dorland
Journal:  Eur J Pediatr       Date:  1995       Impact factor: 3.183

Review 7.  Ovarian function in girls and women with GALT-deficiency galactosemia.

Authors:  Judith L Fridovich-Keil; Cynthia S Gubbels; Jessica B Spencer; Rebecca D Sanders; Jolande A Land; Estela Rubio-Gozalbo
Journal:  J Inherit Metab Dis       Date:  2010-10-27       Impact factor: 4.982

8.  Deficits of facial emotion recognition and visual information processing in adult patients with classical galactosemia.

Authors:  Mirjam Korner; Sonja Kälin; Antoinette Zweifel-Zehnder; Niklaus Fankhauser; Jean-Marc Nuoffer; Matthias Gautschi
Journal:  Orphanet J Rare Dis       Date:  2019-02-26       Impact factor: 4.123

Review 9.  Galactosemia: Towards Pharmacological Chaperones.

Authors:  Samantha Banford; Thomas J McCorvie; Angel L Pey; David J Timson
Journal:  J Pers Med       Date:  2021-02-07
  9 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.