Literature DB >> 34187405

Prenatal diagnosis of a novel pathogenic variation in the ACAN gene presenting with isolated shortening of fetal long bones in the second trimester of gestation: a case report.

Paolo Toscano1,2, Lavinia Di Meglio3,4, Fortunato Lonardo5, Letizia Di Meglio2, Laura Letizia Mazzarelli1,2, Carmine Sica2, Aniello Di Meglio2.   

Abstract

BACKGROUND: Heterozygous mutations of the ACAN gene are a major cause of different evolutive growth defects in the pediatric population, but were never described as a cause of fetal skeletal dysplasia. CASE
PRESENTATION: A G1 at 21w + 3d came to our institution for the second-trimester ultrasound and a skeletal dysplasia with prevalent involvement of limb's rhizomelic tracts was suspected. Amniocentesis followed by CGH-array was performed, with normal results. An examination by NGS of some genes associated with skeletal dysplasias showed a novel pathogenic variant of the ACAN gene: c.2677delG.
CONCLUSION: Sequence variations of ACAN were never described as a possible cause of fetal skeletal anomalies to date. In this case report, we describe the first prenatal diagnosis of skeletal dysplasia associated with a pathogenic variant of ACAN.

Entities:  

Keywords:  ACAN; Aggrecan; Case report; Prenatal diagnosis; Skeletal dysplasia

Year:  2021        PMID: 34187405     DOI: 10.1186/s12884-021-03952-w

Source DB:  PubMed          Journal:  BMC Pregnancy Childbirth        ISSN: 1471-2393            Impact factor:   3.007


  2 in total

Review 1.  The aggrecanopathies; an evolving phenotypic spectrum of human genetic skeletal diseases.

Authors:  Beth G Gibson; Michael D Briggs
Journal:  Orphanet J Rare Dis       Date:  2016-06-28       Impact factor: 4.123

2.  The role of aggrecan in normal and osteoarthritic cartilage.

Authors:  Peter J Roughley; John S Mort
Journal:  J Exp Orthop       Date:  2014-07-16
  2 in total

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