Literature DB >> 20704477

Thanatophoric dysplasia type I associated with increased nuchal translucency in the first trimester: Early prenatal diagnosis using combined ultrasonography and molecular biology.

Gabriele Tonni1, Daniela Azzoni, Alessandro Ventura, Bruno Ferrari, Claudio De Felice, Maurizia Baldi.   

Abstract

A case of thanatophoric dysplasia (TD) type I associated with severely increased nuchal translucency at first trimester screening for Down syndrome is reported. A 38-year-old woman, G2P1, with previous uneventful pregnancy, was referred for amniocentesis at 16 weeks due to positive first trimester integrated test. Amniocentesis revealed a 46,XX fetus. At 16 weeks gestation, the ultrasound examination of the fetus revealed a narrow chest, short ribs, and a generalized severe shortening of the long bones. The patient underwent a follow-up scan at 19 weeks which demonstrated ultrasound findings consistent with severe rhizomelic micromelia. A wide prenatal panel of gene mutations related with skeletal dysplasia was performed. Nucleotidic sequence using QF-PCR on exons 7,10, 15, 19 of the fibroblast growth factor receptor 3 (FGFR3) demonstrated a 742 C>T (R248C) mutation, which resulted in an Arg248Cys substitution in heterozygous state, leading to a prenatal diagnosis of thanatophoric dysplasia type I. The early diagnosis of this lethal form of skeletal dysplasia directed the prenatal counseling and allowed appropriate obstetric management. Necropsy, post-mortem x-ray, and histologic analysis of the growth plate might aid the diagnosis of TD type I.

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Year:  2010        PMID: 20704477     DOI: 10.3109/15513811003796938

Source DB:  PubMed          Journal:  Fetal Pediatr Pathol        ISSN: 1551-3815            Impact factor:   0.958


  3 in total

1.  Comparative X-ray morphometry of prenatal osteogenesis imperfecta type 2 and thanatophoric dysplasia: a contribution to prenatal differential diagnosis.

Authors:  Maria Pia Bondioni; Ugo Ernesto Pazzaglia; Claudia Izzi; Giuseppe Di Gaetano; Francesco Laffranchi; Maurizia Baldi; Federico Prefumo
Journal:  Radiol Med       Date:  2017-07-03       Impact factor: 3.469

2.  Thanatophoric dysplasia type 1 with tectal plate dysplasia and aqueductal stenosis.

Authors:  Ai Peng Tan; Gema Priego
Journal:  Childs Nerv Syst       Date:  2019-01-04       Impact factor: 1.475

3.  Prenatal Diagnosis of Skeletal Dysplasia and Review of the Literature.

Authors:  Bashiru Babatunde Jimah; Teresa Aba Mensah; Kofi Ulzen-Appiah; Benjamin Dabo Sarkodie; Dorothea Akosua Anim; Emmanuella Amoako; Evelyn Antwiwaa Gyamfi
Journal:  Case Rep Obstet Gynecol       Date:  2021-04-13
  3 in total

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