Tonni Gabriele, Azzoni Daniela, Ventura Alessandro, Ferrari Bruno, Felice Claudio De, Baldi Maurizia
Guastalla Civil Hospital, Department of Gynecology & Obstetrics, Guastalla, Italy.
Fetal Pediatr Pathol. 2010;29(5):314-22. doi: 10.3109/15513811003796938.
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.
报告了一例I型致死性骨发育不全(TD),其与孕早期唐氏综合征筛查时颈部半透明带严重增厚相关。一名38岁女性,孕2产1,既往妊娠顺利,因孕早期综合筛查结果阳性,于孕16周转诊行羊膜腔穿刺术。羊膜腔穿刺术显示胎儿核型为46,XX。孕16周时,胎儿超声检查显示胸部狭窄、肋骨短小以及长骨普遍严重缩短。患者在孕19周时接受了随访超声检查,结果显示与严重的近段短小肢端侏儒症相符。对一系列与骨骼发育异常相关的基因突变进行了广泛的产前检测。采用QF-PCR对成纤维细胞生长因子受体3(FGFR3)基因的第7、10、15、19外显子进行核苷酸序列分析,结果显示存在742 C>T(R248C)突变,该突变导致杂合状态下的精氨酸248被半胱氨酸替代,从而产前诊断为I型致死性骨发育不全。这种致死性骨骼发育异常的早期诊断为产前咨询提供了指导,并有助于进行适当的产科管理。尸体解剖、死后X线检查以及生长板的组织学分析可能有助于I型TD的诊断。