Larsen-like phenotype associated with partial trisomy 3p and monosomy 5p

Prenat Diagn. 2008 Feb;28(2):131-4. doi: 10.1002/pd.1928.

Abstract

Background: We report on a fetus with radiographic features of Larsen Syndrome (LS) and unbalanced 3;5 translocation. Recently LS was shown to be caused by mutations in FLNB gene which maps on 3p14.3.

Methods: Comparative genomic hybridization (CGH) was performed to search for genomic imbalances. Fluorescence in situ analysis (FISH) was done with BAC clone RP11-754F19 probe from the FLNB gene region (3p14.3).

Results: CGH showed a large loss of the chromosome 5 short arm and a gain of half of the short arm of chromosome 3 resulting from a derivative chromosome 5. FISH analysis with FLNB probe demonstrated that it was not triplicated. Thus, we excluded the role of a gene dosage effect of FLNB in abnormal craniofacial development in this fetus.

Conclusions: To our knowledge, this is the first report of Larsen-like phenotype associated with unbalanced translocation resulting in partial trisomy 3p and monosomy 5p.

Publication types

  • Case Reports

MeSH terms

  • Abnormalities, Multiple / genetics*
  • Abortion, Therapeutic
  • Adult
  • Amniocentesis
  • Chromosomes, Human, Pair 3 / genetics*
  • Chromosomes, Human, Pair 5 / genetics*
  • Congenital Abnormalities / genetics*
  • Female
  • Humans
  • Infant, Newborn
  • Karyotyping
  • Nucleic Acid Hybridization
  • Phenotype
  • Pregnancy
  • Syndrome
  • Translocation, Genetic / genetics*
  • Trisomy*
  • Ultrasonography, Prenatal