Literature DB >> 24411048

Detection of a de novo Y278C mutation in FGFR3 in a pregnancy with severe fetal hypochondroplasia: prenatal diagnosis and literature review.

Chih-Ping Chen1, Yi-Ning Su2, Tzu-Hung Lin3, Tung-Yao Chang4, Jun-Wei Su5, Wayseen Wang6.   

Abstract

OBJECTIVE: We describe a prenatal molecular diagnosis of hypochondroplasia (HCH) in a pregnancy not at risk of HCH and review the literature on prenatal diagnosis of HCH. CASE REPORT: A 28-year-old primigravid woman was referred for genetic counseling at 30 weeks of gestation because of short-limbed dwarfism in the fetus. The woman had a body height of 152 cm. Her husband had a body height of 180 cm. Level II ultrasound showed a normal amount of amniotic fluid and a singleton fetus with fetal biometry equivalent to 30 weeks except for short limbs. Fetal biometry measurements were as follows: biparietal diameter = 7.38 cm (30 weeks); head circumference = 28.14 cm (30 weeks); abdominal circumference (AC) = 24.64 cm (30 weeks); femur length (FL) = 3.97 cm (<5th centile); FL/AC ratio = 0.161 (normal > 0.18); humerus = 3.64 cm (<5th centile); radius = 3.49 cm (30 weeks); ulna = 3.76 cm (<5(th) centile); tibia = 3.67 cm (<5th centile); and fibula = 3.72 cm (<5th centile). The digits and craniofacial appearance were normal. A tentative diagnosis of achondroplasia (ACH) was made. DNA testing for the FGFR3 gene and whole-genome array comparative genomic hybridization (aCGH) analysis were performed using cord blood DNA obtained by cordocentesis. FGFR3 mutation analysis revealed a de novo heterozygous c.833A > G, TAC > TGC transversion in exon 7 leading to a p.Tyr278Cys (Y278C) mutation in the FGFR3 protein. aCGH analysis revealed no genomic imbalance in cord blood. After delivery, the fetus had short limbs, a narrow thorax, brachydactyly, and relative macrocephaly. Cytogenetic analysis of cultured placental cells revealed a karyotype of 46,XX.
CONCLUSION: Prenatal diagnosis of abnormal ultrasound findings suspicious of ACH should include a differential diagnosis of HCH by molecular analysis of FGFR3.
Copyright © 2013. Published by Elsevier B.V.

Entities:  

Keywords:  FGFR3; hypochondroplasia; prenatal diagnosis

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Year:  2013        PMID: 24411048     DOI: 10.1016/j.tjog.2013.10.023

Source DB:  PubMed          Journal:  Taiwan J Obstet Gynecol        ISSN: 1028-4559            Impact factor:   1.705


  2 in total

1.  Whole-exome sequencing and whole genome re-sequencing for prenatal diagnosis of achondroplasia.

Authors:  Rong Zhao; Yan Ruan; Xin Wang
Journal:  Int J Clin Exp Med       Date:  2015-10-15

2.  Non-Invasive Prenatal Diagnosis of Lethal Skeletal Dysplasia by Targeted Capture Sequencing of Maternal Plasma.

Authors:  Shan Dan; Yuan Yuan; Yaoshen Wang; Chao Chen; Changxin Gao; Song Yu; Yan Liu; Wei Song; Hongmei Zhu; Ling Yang; Hongmei Deng; Yue Su; Xin Yi
Journal:  PLoS One       Date:  2016-07-19       Impact factor: 3.240

  2 in total

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