| Literature DB >> 9061768 |
S L van Zelderen-Bhola1, E J Breslau-Siderius, G C Beverstock, I Stolte-Dijkstra, L S de Vries, P Stoutenbeek, J M de Pater.
Abstract
We present here a case report of a fetus with a kidney anomaly and dilated occipital horns, detected initially by echoscopy at 29 weeks' amenorrhoea. After 31 weeks of gestation, the proband was born with clinical symptoms of Miller-Dieker syndrome. This was subsequently confirmed by fluorescence in situ hybridization (FISH), but not by conventional cytogenetic analysis. FISH using a cocktail of cosmids (c197-2, c197-4, c197-9) from the Miller-Dieker critical region showed a deletion of 17p13.3 in one homologue of chromosome 17. Additional FISH studies revealed a subtle 17p;20q translocation in the father, his sister, and the paternal grandmother. Hence, our patient is a carrier of an unbalanced 17;20 translocation resulting in a partial deletion of 17p and a partial trisomy 20q. Whenever kidney anomalies and dilated occipital horns are observed together with polyhydramnios during prenatal ultrasound examination, the possibility of Miller-Dieker syndrome should be suspected. In such cases, prenatal and/or postnatal chromosome studies should also include FISH analysis with the appropriate probes.Entities:
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Year: 1997 PMID: 9061768 DOI: 10.1002/(sici)1097-0223(199702)17:2<173::aid-pd30>3.0.co;2-v
Source DB: PubMed Journal: Prenat Diagn ISSN: 0197-3851 Impact factor: 3.050